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AISC Team B1 5

3968549-1

noncomm/PMC003xxxxxx/PMC3968549.xml

Revision Total Hip Arthroplasty Combined with Subtrochanteric Shortening Osteotomy: Case Report

A 66-year old female was referred to our hospital with a chief complaint of pain in her bilateral buttocks and the need to use two crutches to walk.\nShe had a history of acetabular dysplasia, however, the treatment was unspecified. When she was 44 years old, she underwent bipolar head arthroplasty (BHA) with a bone graft.\nNo clinical signs and laboratory findings of infection were observed prior to the revision surgery.\nRoentgenograms showed that the implant of her right BHA had migrated proximally, and sapphire pins were placed at the new acetabulum. We concluded that she underwent BHA with a bone graft to create a new acetabulum in the high hip center, because her right true acetabulum was intact in spite of the hypoplasia. In addition to the right implant migration, her left hip joint was congenitally completely dislocated (Fig. ).\nThe patient then underwent revision THA combined with V-shaped subtrochanteric shortening osteotomy and allogenic bone grafting. A posterolateral approach was used under spinal anesthesia.\nAround the proximal femur, there was massive metallosis which had been created from the friction between the outer head and sapphire pins. In addition, there was a marked bone defect in the proximal femur, and stem loosening was also observed.\nFirst, the prosthesis of the BHA and sapphire pins were removed, and sufficient irrigation and debridement were performed. Placing a shell at the new acetabulum was impossibile, because there was a severe bone defect. Therefore, a V-shaped subtrochanteric shortening osteotomy was performed in order to place the shell at the true acetabulum. The V-shaped osteotomy is considered to be an appropriate surgical procedure because the osteotomy site is torsionally more stable than other types of osteotomy and the osteotomy site can be stabilized with a shorter stem. We performed the V-shaped osteotomy using a special device that was developed by Hotokebuchi et al. [] (Fig. ).\nThen, an AMS-HA acetabular shell with an AMS (polyethylene liner) liner was placed with six screws at the true acetabulum, and a cementless full hydroxyapatite-coated femoral component (a PerFix-HA Mid Size Stem) with a 28-mm zirconia ball (Kyocera, Kyoto, Japan) was used.\nMorselized allogenic bone was grafted at the superior site of the shell and defect site of the proximal femur, and bulk allogenic bone was grafted at the new acetabulum (Fig. ). The allogenic bone was frozen, and the grafts were processed aseptically. And they were not irradiated. The total length of the operation was 125 minutes, and the total blood loss was 1151 ml.\nUnfortunately, the patient had to undergo stem revision 26 days after the first revision surgery for a recurrent dislocation. Moreover, two months after the first revision surgery, she underwent irrigation and debridement for an infected right revisionTHA due to Staphylococcus caprae. The infection was not controlled, and two-stage revision surgery using a cement mold including antibiotics was performed to treat the infected revision THA. The femoral component was changed from a PerFix-HA Mid Size Stem to a Per-Fix HA Long Size Stem (Kyocera, Kyoto, Japan).\nThereafter, we performed THA with a subtrochanteric shortening osteotomy for the left hip. The last follow-up occurred approximately four years after the first revision surgery, at which time all laboratory findings indicative of infection remained well within the normal limits, and no clinical signs of infection were noted. The patient is currently able to walk without a cane.\nRoentgenograms showed marked bone absorption at the site of the stem shaft, however, there was no evidence of instability (Fig. ).

[[66.0, 'year']]

F

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{}

3968565-1

noncomm/PMC003xxxxxx/PMC3968565.xml

Unveiling The Hidden Eagle: Acute Parotitis-Induced Eagle Syndrome

A 65-year-old woman with past medical history of diabetes mellitus, chronic kidney disease presented with a five day history of right facial pain and swelling that started after a right upper first premolar root canal procedure. The pain and swelling progressed despite treatment with amoxicillin/clavulanate to the point that she could not open her mouth due to severe right facial pain. She denied fever, sinus pain, ear pain, nasal discharge, tongue swelling, breathing difficulty, voice change or drooling but did have chills. Physical examination demonstrated erythema, painful swelling in the preauricular area extending to the right mandibular angle. There was no purulent discharge from the orifice of Stensen's duct on the right. Temporary amalgam was seen on the right upper first premolar without swelling or tenderness at the dental root. Otoscopic examination was within normal limits bilaterally. Pertinent laboratory investigation revealed a white blood cell 7.7 (neutrophil 73.6%, lymphocyte 15.7%, monocyte 9.5%), amylase 81 U/l, and negative blood cultures. Neck and sinus computed tomography (CT) scan revealed asymmetric enhancement of the right parotid gland compatible with acute parotitis []. No calculus, mass, or abnormal collection was identified. The sub-mandibular glands were unremarkable, and para-pharyngeal fat was preserved. The patient was treated with intravenous ceftriaxone and clindamycin. The inflammation almost completely subsided to the point that patient could open her mouth. However, patient started to complain of pain in right mandibular, ear, and neck region with foreign sensation in her throat. The pain was felt to be out of proportion to the inflammation by examination. The patient denied a history of recurrent head and neck pain, syncope, or stroke in the past. CT was reviewed with the radiologist and found to have an asymmetrically long right styloid process measures 5.3 cm in length []. The patient was diagnosed with acute parotitis-induced Eagle syndrome and was referred to maxillofacial surgeon for further treatment.

[[65.0, 'year']]

F

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{}

3968566-1

noncomm/PMC003xxxxxx/PMC3968566.xml

Scalp Squamous Cell Carcinoma in Xeroderma Pigmentosum

A three-year-old boy, known case of XP, presented to our clinic with an ulcerating mass in the scalp []. The ulcer was round in shape, measured about 10 × 15 cm, with punched out edges. There was no discharge or bleeding. At presentation, he was irritable and photophobic. There were diffuse hyperpigmented lesions over the face, trunk, and extensor surface of the upper extremities. The skin was unnaturally dry and rough all over the previously mentioned areas. Hyperpigmented hyperkeratotic changes were observed over some lesions. There was a congestion of conjunctivae of both eyes. Neurological examination was normal. He has a family history of thalassemia major in two siblings, but no similar case. There is a positive history of consanguinity. Biopsy showed moderately differentiated squamous cell carcinoma. The patient presented with a late diagnosis of SCC that has been progressing for 2 years. At age of three months, he started to develop rashes and hyperpigmentation (freckles) on the face and extremities. Previously, he sought medical advice that has poor diagnostic facilities near his town. Computerized Tomography (CT) scan of the brain demonstrates evidence of erosion of the outer and inner plate of scalp exposing the dura with no intracranial extension. Excision of the lesion with 1 cm safety margin circumferentially was done. Scalp reconstruction was performed using local flaps and skin graft. A written informed consent was obtained from the parents for publication of this case report and any supplementary image.

[[3.0, 'year']]

M

{'33996357': 1, '16970698': 1, '18329345': 1, '22044607': 1, '18937855': 2, '21097776': 1, '24695441': 2}

{'2577106-1': 1}

3968597-1

noncomm/PMC003xxxxxx/PMC3968597.xml

Tenofovir induced Fanconi syndrome: A rare cause of hypokalemic paralysis

A 55-year-old female presented with acute onset weakness of all four limbs of one day duration. She also noted increased fatigue and polyuria over the past three days. There was no past history of diarrhea or upper respiratory infection in the recent past or any past history of neurological or renal disorders. She was diagnosed to have HIV infection 7 years back and was started on tenofovir, emtricitabine and atazanavir 10 months ago. Physical examination revealed cachexia and muscle power of 2/5 in all four limbs. Deep tendon reflexes were sluggish and pin prick sensation was normal. There was no respiratory distress and her vital parameters were also normal.\nIn the emergency room her potassium was 1.66 meq/l, serum glucose 112 mg/dl, serum sodium 140 meq/l, serum chloride 118 meq/l, serum bicarbonate 9.4 meq/l, serum phosphorus 2.2 mg/dl and calcium 8.4 mg/dl. Patient had normal anion gap metabolic acidosis and hence renal tubular acidosis was suspected. Urine anion gap was positive (15). Her serum creatinine was 2.2 mg/dl, urea 41 mg/dl, urine analysis showed 2 + albuminuria, 3 + glucosuria, 2 + myoglobinuria, pH 5.5, spot potassium 32 meq/l, spot sodium 75 meq/l and spot chloride 92 meq/l. Arterial blood gas values were pH 7.194, pCO2 23.4, pO2 107, bicarbonate 8.8, lactate 2.5. Complete blood count showed high erythrocyte sedimentation rate of 62 and creatinine phosphokinase was 616, rest were within normal limit.\nFS leading to hypokalemic paralysis was suspected given the presence of a hyperchloremic metabolic acidosis, hypokalemia, hypophosphatemia, glucosuria and proteinuria. The patient was aggressively rehydrated with intravenous fluids. She required replacement with intravenous potassium chloride for 2 days to keep potassium above 3 meq/l. Patients muscle power dramatically improved within 12 h of hospital admission with potassium correction. Other renal parameters, also improved slowly with treatment. Tenofovir was stopped as we suspected it to be the cause for FS.

[[55.0, 'year']]

F

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{'4660566-1': 1}

3968598-1

noncomm/PMC003xxxxxx/PMC3968598.xml

Atypical presentation of post infectious glomerulonephritis as malignant hypertension and thrombotic microangiopathy

A 28-year-old male was healthy until one month before admission, when he experienced increasing tiredness and lethargy. Two days before hospitalization, he developed fever, headache with blurring of vision. On the day of admission he had one episode of convulsions and developedaltered sensorium. Clinical examination revealed pallor bilateral pitting pedal edema. Blood pressure was 180/100 mmHg in supine position; heart rate, 96 beats/min; temperature, 101° F; respiratory rate, 32 breaths/min. Neurologic examination suggested postictal confusion with papilloedema, bilateral flexor plantar reflex and no signs of neck stiffness. Lungs showed bibasal rales. Rest of the physical examination was unremarkable. Initial laboratory investigation showed serum creatinine, 3.89 mg/dl; blood urea nitrogen, 84 mg/dl; serum potassium, 4.5 mEq/l; chloride, 99 mmol/l; sodium, 134 mmol/l; white blood cell count, 13600 cells/cumm; hemoglobin, 7.8 gm/dl; platelet count, 90000/cumm; calcium, 8.6 mg/dl; phosphorus, 3.5 mg/dl; albumin, 3.6 gm/dl; bilirubin 0.9 mg/dl; serum iron, 20 μmol/l and lactate dehydrogenase(LDH), 1280 U/l. Urinanalysis showed specific gravity 1.025, pH 6.0, 4+ protein, 15-20 pus cells/hpf, 25-30 red blood cells (RBCs)/hpf and 1-2 granular casts. 24 h urine protein leak was 1.9 g/d. Liver function test, lipid profile and thyroid function tests were within normal limits. On ultrasonography - both kidneys were of normal size, but with increased parenchymal echogenicity. Computed tomography scan of head revealed ill-defined hypodensities at bilateral parieto-occipital region consistent with posterior reversible encephalopathy syndrome. Because of the complex clinical syndrome (hypertensive encephalopathy syndrome coupled with proteinuria and active urine sediments), a percutaneous renal biopsy was performed. Serologic tests were negative for anti-streptolysin-O antibody titre, antinuclear antibody, anti-double stranded deoxyribonucleic acid antibody, anti-neutrophil cytoplasmic antibodies profile by ELISA and anti-glomerular basement membrane antibody. Serum complement levels (C3, C4) were normal. Screening for hepatitis B, hepatitis C and human immunodeficiency virus was negative. On light microscopy, there were twenty two viable glomeruli displaying two pathologies simultaneously. Majority of them appeared solid and proliferative comprising neutrophilic infiltration in the capillary tufts obliterating capillary lumen secondary to proliferation of endothelial cells, neutrophils and sparse mesangial cells; with single contoured basement membranes []. Two of these glomeruli (9%) possessed circumferential oriented active cellular crescents, without necrotizing lesions []. Lobular accentuation was absent. In addition, five glomeruli (devoid of neutrophilic infiltration) showed changes of TMS in the form of blood filled, engorged capillaries [], and fibrin thrombus in the hilar arteriole. Mesangiolysis was noted. Moderate degree of acute tubular injury was noticed with fresh RBC's in the lumen. There was no focus of tubular atrophy/interstitial fibrosis. Interstitium lacked inflammation or granuloma. Artery and arterioles showed hypertensive changes to a moderate extent in the form of hypertrophic muscular wall and intimal thickening. Marked intimal hyalinosis was present in smaller arterioles. Fibrinoid necrosis of vessel wall, luminal thrombosis or vasculitis was not evident. Immunofluorescence revealed diffuse and global, coarse granular deposits along the capillary walls and mesangium with C3 (3+) and IgG (2+) in five available glomeruli []. Kappa and lambda light chain stains showed 2+ positivity in a similar pattern. C3 deposits were also seen in vessels. IgA, IgM and C1q were negative. A diagnosis of coexistent TMA and APIGN with focal (9%) crescents was established. Upon probing, patient denied any symptom of previous infective episode.\nPatient was treated with intravenous antibiotics, intravenous pulse methyl prednisolone (1 g for 3 days) considering the presence of active crescents histologically, followed by tapering dose of oral prednisolone; along with strict control of blood pressure. He was treated with intravenous nitroglycerine infusion during the early phase of the disease, followed by oral ramipril 5 mg twice daily, clonidine 0.1 mg thrice daily, frusemide 40 mg thrice daily, metalozone 5 mg twice daily and prazocin 5 mg twice daily. There was a dramatic improvement in his renal function with decrease in serum creatinine (1.5 mg/dl) as well as trace proteinuria. TMA was also resolved as evidenced by normalization of serum LDH level, hemoglobin and platelet count.

[[28.0, 'year']]

M

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{'4676346-1': 1}

3968599-1

noncomm/PMC003xxxxxx/PMC3968599.xml

Mesangial proliferative glomerulonephritis with acute tubule interstitial nephritis leading to acute kidney injury in influenza A (H1N1) infection

A 52-year-old female patient was admitted in March 2012 with fever, cough, breathlessness and progressive dyspnea with respiratory distress and decrease urine output of 10 days duration. There was no history of abdominal pain, dysuria or trauma and no recent use of non-steroidal anti-inflammatory medication.\nOn examination, she was breathless with blood pressure of 150/90 mmHg, temperature 40°C, respiratory rate of 30 breaths/min, heart rate of 100 bpm. Laboratory investigations revealed hemoglobin, 10.3 g/l; total white cell count, 9.88 × 103/μl (differential count: 74% neutrophils, 20% lymphocytes, 4% monocytes and 2% eosinophils); platelet count, 211 × 103/μl; serum creatinine (SCr), 10.8 mg/dl sodium, 138 mEq/l; potassium, 5.3 mEq/l; blood urea, 76 mg/dl; and blood glucose, 72 mg/dl.\nRenal ultrasound showed normal sized kidneys with hyper reflective cortex. Serological tests for malaria, leptospirosis, dengue and viral hepatitis were negative. The initial blood, urine and sputum cultures were sterile. Urinalysis showed proteinuria, hematuria and two to five fine granular casts. SCr phosphokinase levels and liver function test chest radiograph were unremarkable while tests for antinuclear antibody, anti-double-stranded deoxyribonucleic acid, anti-neutrophil cytoplasmic antibody, anti-glomerular basement membrane antibody, coombs’ test, cryoglobulins and C-reactive protein were all negative and complement components (C3 and C4) were low. Enzyme-linked immunosorbent assays test for human immunodeficiency virus (HIV), hepatitis B surface antigen and hepatitis C virus were negative. Patients had influenza H1N1 virus subtype specific ribonucleic acid detected in a nasopharyngeal swab using reverse transcriptase-polymerase chain reaction. She had a history of exposure to swine flu positive patient and was managed using oseltamivir, antibiotic, fluid replacement and dialysis. Patients received oseltamivir, 75 mg within 48 h of presumed diagnosis for 5 days and supportive measures as needed. Initially, she was treated with peritoneal dialysis and she remained oliguric and required intermittent hemodialysis for a total of 5 times through temporary jugular dialysis catheter for 4 h each.\nRenal biopsy [] revealed two fragment of renal tissue containing 11 glomeruli with associated tubules and vessels. All the glomeruli show uniform mesangial prominence. Capillary lumina were open, and membranes were of normal thickness. Tubules showed mild to moderate degeneration. Interstitium was mildly prominent for uniform edema and diffuse infiltration with leucocytes. Immunofluorescence microscopy did not reveal staining with anti-human sera tested (IgG, IgM, IgA and C1q). Influenza nucleoprotein (InfA-NP) antigen-positive cells were not detected in kidney tissue. A diagnosis of mesangial proliferative glomerulonephritis with acute tubule interstitial nephritis was made. The patient was treated with methyl prednisolone 500 mg for 3 days followed by oral prednisolone 1 mg/kg/day. Her condition also improved rapidly and she was discharged with SCr of 1.5 mg/dl. Her SCr was 1 mg/dl at 1 month follow-up.

[[52.0, 'year']]

F

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{}

3968600-1

noncomm/PMC003xxxxxx/PMC3968600.xml

Karyomegalic interstitial nephritis with focal segmental glomerulosclerosis: A rare association

An 8-year-old male child presented with a history of facial puffiness and edema of one year duration. He was diagnosed to have nephrotic syndrome and was treated by a local physician with steroids. He did not improve, however, and was referred to this hospital for further management.\nThere was no family history of kidney disease. He is the first child of non-consanguineous marriage. There was no history of drug ingestion including exposure to mycotoxins or other herbal medicines. No history of recurrent respiratory symptoms. On examination, patient had stunted growth with moon facies, secondary to steroids and had clinical features of rickets.\nHis investigations revealed hemoglobin of 10 g/dl, blood urea 33 mg/dl and serum creatinine 0.7 mg/dl. His liver functions revealed hypoalbuminemia of 2.6 g/dl. His liver enzymes were within normal limits. He was human immunodeficiency virus non-reactive and hepatitis B surface antigen and hepatitis C virus and 24 h urine protein was 3565 mg/dl. Complete urine examination showed 2+ albumin, 4-6 red blood cells and granular casts. Ultrasound revealed normal sized kidneys with normal echotexture.\nHe was started on cyclophosphamide. However, there was no response even after 6 months. Hence a kidney biopsy was done. Biopsy showed 23 glomeruli, four of whom showed segmental sclerosis []. Basement membrane and cellularity were normal in the uninvolved glomeruli. Tubular epithelial cells revealed changes diffusely. The lining cells in many cells revealed enlarged nuclei with lobulated appearance. These cells are larger than the normal tubular epithelial cells []. There was chromatin clumping giving a hyperchromatic appearance. Mitoses were sparse. There is atrophy of the tubules with thickening of tubular basement membrane. There is interstitial fibrosis and interstitial mild lymphomononuclear infiltration [].\nImmunohistochemical stains for BK virus and cytomegalovirus were negative. Ki-67 index was low. Immunofluorescence did not reveal any significant immune deposits.\nHe is under regular follow-up and his last creatinine has increased to 1.2 mg/dl.

[[8.0, 'year']]

M

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{'7699658-1': 1, '8597774-1': 1, '8597774-2': 1, '4964693-1': 1}

3968601-1

noncomm/PMC003xxxxxx/PMC3968601.xml

Pulmonary histoplasmosis in a renal allograft recipient

A 55-year-old lady, on dialysis since February 2011, underwent a renal transplantation on 23rd February 2012 with an allograft kidney from her daughter. She had been diagnosed with tuberculous lymphadenopathy in September 2011 and received isoniazid, rifampicin, pyrazinamide and ethambutol from September to December 2011. Pyrazinamide was stopped in December 2011 and rifampicin replaced with ciprofloxacin in February 2012. There was 1 B and 1 DR mismatch and the lymphocyte cross match by complement dependent cytotoxicity was negative at 4%.\nImmunosuppression consisted of intraoperative methylprednisolone, followed by tapering doses of oral prednisolone until 10 mg daily, tacrolimus 0.15 mg/kg/day adjusted according to trough levels and azathioprine 2 mg/kg/day.\nShe was admitted on 18th July 2012 with daily evening fever up to 100°F for 10 days, dry cough for 7 days and left sided pleuritic chest pain for 5 days. She had received oral amoxicillin clavulunate and paracetamol with temporary relief, but symptoms had worsened on the day of admission. Examination revealed a temperature of 100.4°F, pulse of 96/min and respiratory rate of 20/min. Blood pressure was 130/85 mm of Hg. Auscultation of the chest revealed decreased breath sounds at the left infraaxillary and infrascapular regions, without any bronchial breathing, rub or rales. A chest roentgenogram showed a homogeneous opacity without an air bronchogram in the left lower zone with obscured left costophrenic angle [].\nOther investigations are shown in .\nShe received empirical cefotaxime 2 g q 12 h, azithromycin 0.5 g daily and nebulization to induce sputum production. She continued to have chest pain and fever despite negative blood cultures and a complete absence of sputum. A left lateral roentgenogram confirmed the opacity and showed no evidence of pleural fluid. A high resolution computed tomography scan of the thorax on the 25th of July 2012, revealed a heterogeneously enhancing necrotic mass lesion in the left lower lateral basal segment reaching up to the pleural surface, multiple discrete lower lobe nodules bilaterally, a discrete 1 cm nodular density in the inferior and paracardiac region of the right upper lobe and enlarged pre-tracheal, paratracheal, precarinal and subcarinal lymph nodes [].\nA fine-needle aspiration and biopsy of the lesion was performed on 26th July 2012, under computed tomographic guidance. The aspirate showed a necrotic background with few polymorphs, lymphocytes, eosinophils and budding yeast like fungal elements and was negative for acid fast bacilli, nocardia and Pneumocystis jiroveci. The biopsy showed widespread areas of necrosis, infiltration with polymorhonuclear leukocytes and lymphocytes and histiocytic proliferation and fibrosis. The histiocytes contained multiple tiny, budding yeast like structures, which on Periodic acid Schiff (PAS), showed a distinct positively stained capsule []. The lesion was suggestive of histoplasmosis, which was confirmed on culture after 23 days of incubation. Patient was treated with liposomal amphotericin B 200 mg daily, (4 mg/kg/day) and discontinuation of azathioprine. After 20 days, her chest roentgenogram showed significant improvement and she was discharged on oral itraconazole 200 mg bid and her chest roentgenogram after 5 months of treatment [], showed complete resolution of the lesion. In October 2012, she was found to have developed new onset diabetes after transplantation, which was controlled with oral hypoglycemic agents.

[[55.0, 'year']]

F

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{'3507415-1': 1}

3968620-1

noncomm/PMC003xxxxxx/PMC3968620.xml

Evidence Based Solving Approach in Diagnosis of Primary Hyperparathyroidism with Oral Manifestations: Report of Three Unusual Cases

A 32-year-old female patient reported with complaint of swelling over the front right side of the lower jaw since 2 months with history of a small painful swelling over the chin region which gradually increased to the present size []. Patient also gave history of joint pain, backache, and weakness and renal stones 2 years back.\nExtraoral examination revealed a well-defined, soft to firm and tender swelling over the mental and right body of mandible of 6 × 4 cm in size which extended anteroposteriorly from left parasymphysis to the right mandibular body and superoinferiorly from the level of vermilion border of lower lip to the submental region [; inset]. Detailed intraoral examination revealed obliteration of labial vestibule i.r.t 41-45 region with Grade 1 mobility of involved teeth. Radiographic examination revealed irregular radiolucency with ill-defined margins seen in the mandible extending from 35-45 region. Generalized rarefaction of the trabeculae and altered trabecular pattern was also seen along with generalized loss of lamina dura []. Full mouth intraoral periapical (IOPA) survey revealed generalized loss of lamina dura. Computed tomography (CT) scan examination revealed gross bone destruction with soft tissue mass.

[[32.0, 'year']]

F

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{'3968620-2': 2, '3968620-3': 2, '3125657-1': 1}

3968620-2

noncomm/PMC003xxxxxx/PMC3968620.xml

Evidence Based Solving Approach in Diagnosis of Primary Hyperparathyroidism with Oral Manifestations: Report of Three Unusual Cases

A 40-year-old female patient presented with swelling in the right maxillary region since 6 months with history of swelling in the same region 5 years back and thereafter swelling was excised. A small sized swelling recurred in the same region which gradually increased to the present size. Patient also gave the history of 5-6 kg weight loss in the past few months and felt anorexic and depressed.\nExtraoral examination revealed a firm, nontender round swelling, 6 cm in diameter in right maxillary region which extended anteroposteriorly from ala of nose to 2 cm medial to tragus of ear and superoinferiorly 2 cm below infraorbital margin to 1 cm below corner of the mouth.\nIntraoral examination revealed a firm, nontender, nonpulsatile reddish brown growth of size 5 × 5 cm extending buccally and palatally i.r.t 12-18 region. Displacement and mobility of 13,14, and 15 pus discharge with 12, 13, and 14 was noticed [; inset].\nIOPA radiograph revealed loss of lamina dura and cyst-like lesions i.r.t 45, 46, 47. Orhtopantomogram (OPG) revealed a soft tissue mass causing resorption of right maxillary alveolar region with displacement of regional teeth. Multiple cyst-like radiolucent areas in right side of body of the mandible and thinning of inferior border of mandible was observed. Generalized loss of lamina dura was also observed []. CT scan was also carried out which revealed tumor causing destruction of the alveolar bone in right maxilla and lytic lesion involving the left body of mandible [].

[[40.0, 'year']]

F

{'16798410': 1, '21321169': 1, '19193909': 1, '21747854': 1, '23059578': 1, '21897897': 1, '32911586': 1, '28616750': 1, '15262510': 1, '10934983': 1, '23230541': 1, '21879476': 1, '22431655': 1, '21731279': 2, '7350459': 1, '19114425': 1, '24701104': 2}

{'3968620-1': 2, '3968620-3': 2, '3125657-1': 1}

3968620-3

noncomm/PMC003xxxxxx/PMC3968620.xml

Evidence Based Solving Approach in Diagnosis of Primary Hyperparathyroidism with Oral Manifestations: Report of Three Unusual Cases

A 38-year-old female complained of swelling in anterior maxillary region since 2 months. She gave history of swelling in mandibular right posterior region 3 years back which was diagnosed as central giant cell granuloma and removed surgically. Two months later left posterior teeth were extracted due to mobility. No significant contributory medical history was there. Intraoral examination revealed a firm, nontender, nonpulsatile swelling extending from 23 to 26 with displacement of 23, 24, and 25 with grade 1 mobility [; inset]. Patient was known case of hypertension since 2 years.\nRadiographic examination revealed a soft tissue mass causing resorption of left posterior maxillary alveolar region with displacement of 23 and 24. Multiple, radiolucent, cyst-like lesions involving whole of the body of mandible and thinning of inferior border of mandible was observed as incidental finding []. CT scan was also performed which revealed osteolytic lesions in the whole body of mandible.\nThese clinicoradiographic findings raised the suspicion of HPT. So further investigations in the form of radiographic skeletal survey, biochemical tests [], fine needle aspiration cytology histopathology, and nuclear imaging was carried out in all the three cases.\nThe patients were thereafter subjected to incisional biopsy and histopathologic examination revealed numerous capillaries and yellowish brown pigmentation due to hemosiderin deposition along with innumerable multinucleated giant cells []. Based on these clinicoradiographic, histopathological and biochemical evidences confirmative diagnosis of HPT was given. All patients were treated surgically followed by medicinal therapy. Regular follow up of patients were done with significant improvement without any reported recurrence.

[[38.0, 'year']]

F

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{'3968620-1': 2, '3968620-2': 2, '3125657-1': 1}

3968621-1

noncomm/PMC003xxxxxx/PMC3968621.xml

A Rare Case of Primary Lymphoma of the Caecum Presenting as Intussusception

A 35-year-old woman presented with a pain in the right iliac fossa of one month duration with a sudden increase in the severity of the pain. On examination, a tender mass was palpable in the right iliac fossa. Ultrasound and CT scan of the abdomen revealed a ileocolic intussusception with homogenous enhancing mass lesion approximately 5.6 × 5.3 × 3.5 cms seen at the lead point. The ileocecal junction, cecum and ascending colon pulled up by intussusceptum. Multiple mesenteric nodes were seen. The differential diagnosis offered were adenoma/lymphoma. A right hemicolectomy was done with an uneventful postoperative course.\nOn gross examination, a fleshy circumscribed polypoid mass was seen at the caecum measuring 5.5 × 4.5 × 3.5 cm, with the cut surface showing areas of hemorrhage [].\nMicroscopy of the lesion with adjacent intestinal mucosa revealed necrosis of the mucosa. The lamina propria was infiltrated by a monotonous population of lymphoid cells that showed destruction of the glands and were seen extending into the muscularis propria and the serosa. The cells were small with irregular nuclear membrane, having hyperchromatic nucleus and scant cytoplasm. The tumor cells were characteristically arranged around the epithelium (lymphoepithelial lesion) []. Seventeen lymph nodes isolated showed reactive hyperplasia. A histological diagnosis of MALT lymphoma was offered. Immunohistochemistry was done which showed strong positivity of the tumor cells for CD-20 [] and negative for CD 10, thus confirming the diagnosis. The patient was referred to oncology for chemotherapy.

[[35.0, 'year']]

F

{'27217691': 1, '30664435': 1, '10195723': 1, '11559658': 1, '9234576': 1, '6193858': 1, '21798075': 1, '18647965': 1, '17233195': 1, '17214096': 1, '8470758': 1, '18409078': 1, '21390139': 1, '22803021': 1, '9296505': 1, '27190819': 1, '19760945': 1, '24701105': 2}

{}

3968622-1

noncomm/PMC003xxxxxx/PMC3968622.xml

Can a Postmortem Skin Biopsy Predict Cause of Death?

A 28-year-old male, plumber by occupation, was brought dead to casualty by an attendant. He waslast seen alive working on a motor that pumps water out of a well. On postmortem cause of death could not be identified except an electric mark on palm. It was a circular raised pale lesion measuring 2 × 1cm on the left hand palmar aspect, firm in consistency without surroundinginflammation. Inquest also ruled out foul play. Viscera were sent for chemical analysis which ruled out poisoning and intoxication. The biopsy of the skin lesion was sent for histopathology to find out whether the death was due to electrocution. Formalin-fixed tissue comprising of skin and subcutaneous tissue measuring 2 × 1 × 1 cm was received and was processed entirely. Care was taken to avoid autolysis in microscopic sections and to include the epidermis in the sections. Routine hematoxylin and eosin stain and Von Gieson stain was done. On microscopy, intraepidermal separation and focal necrosis within epidermis was seen []. There were nuclear elongation, pyknosis, and palisading (spindle-shaped nuclei in the epidermis arranged parallel to each other) seen in the basal layers epidermis indicative of electrocutive changes [].

[[28.0, 'year']]

M

{'8340587': 1, '18472235': 1, '16257069': 1, '14655966': 1, '7632603': 1, '19369109': 1, '12762539': 1, '24701106': 2}

{}

3968624-1

noncomm/PMC003xxxxxx/PMC3968624.xml

Isolated Plexiform Neurofibroma of the Tongue

An 11-year-old girl was admitted to the department of E.N.T. of our institute, with the complaint of inability to close the mouth because of a large, asymmetrical, protruding tongue. The girl had difficulty in swallowing, breathing, and clear speaking since early months of her life. On palpation, the right side of the tongue was hypertrophied and the movements of the tongue were normal, but she was unable to hold the tongue completely in the mouth because of its enormous size. The swelling was soft, nontender, nonreducible, and nonpulsatile on palpation. There was also slight generalized swelling of the right side of the base of the tongue. No neck glands were palpable and functions of V, VII, X, and XII cranial nerves were intact. A magnetic resonance image was obtained to better assess the base of tongue, which revealed a 3 × 3 × 2.5 cm heterogeneously enhancing mass with infiltration in the tongue and tongue base, this lesion was hypervascular and well-circumscribed. Under general anesthesia, a deep biopsy was taken from the tongue. Gross examination of the specimen revealed irregular surface which was soft in consistency []. Histopathological examination revealed well circumscribed multiple nodular lesions composed of elongated spindle wavy cells with muscle fibers in between, these wavy cells were arranged against a myxoid background. No atypia or increase in mitosis was seen [Figures and ]. An ophthalmologic examination was carried out which showed no eye lesions of neurofibromatosis (such as Lisch nodules in the iris, ephelides on eyelids, or ectopia lentis). The skin of the whole body was examined and no café au lait spots or frecklings of the axillary and inguinal region were observed. On plain radiograph, no bone pathology was found. After the diagnostic work-up, surgical treatment was planned. Under general anesthesia, the tumor was approached intraorally, and the biopsy report was consistent with plexiform neurofibroma. Postoperatively, a remarkable reduction in the size of the tongue was achieved. The movements of the tongue were preserved, and, in addition, a significant improvement in her speech was observed. The patient has been on follow-up for 10 months and no additional treatment has been necessary.

[[11.0, 'year']]

F

{'16481830': 1, '16848118': 1, '27525129': 2, '21571578': 1, '8961022': 1, '25478247': 2, '16476032': 1, '33786168': 2, '17023749': 1, '24701108': 2}

{'4971312-1': 1, '7988485-1': 1, '4244939-1': 1}

3968625-1

noncomm/PMC003xxxxxx/PMC3968625.xml

Bartholin's Gland Abscesses Caused by Streptococcus pneumoniae in a Primigravida

A 25-year-old non-diabetic primigravida of 31 weeks gestation presented to the outpatient obstetrics and gynecology clinic with a painful lump on the right side of the vaginal opening and pain during the sexual intercourse of 1 week duration. She had a history of urinary tract infection 2 weeks before the presentation. On examination, there were a body temperature of 38°C and swollen and inflamed right labia majora. Pelvic examination revealed an enlarged, firm and tender 5 by 4 cm swelling of Bartholin's gland on the right side. Her laboratory work-up revealed low hemoglobin of 10.3 g%, white blood cell count of 6300/dl with differential count showing polymorphs of 73% and lymphocytes of 20%, eosinophil 1% and monocytes 6%. She has also reduced glucose-phosphate dehydrogenase (G6PD) activity. Her glucose tolerance test for pregnancy was 5.7 mmol/L (normal is <7.8 mmol/L). Here, hepatitis B and C and venereal disease research laboratory tests were negative. Her urine analysis showed pus cells 50-99 cell/HPF, with positive leukocyte esterase of 3+ and urine nitrite was negative; but her urine culture was sterile.\nComplete drainage of the abscess was done through a small surgical cut under local anesthesia in the out-patient clinic. Pus drained and sent immediately to the laboratory. Gram stain of pus revealed the presence of plenty of pus cells with Gram-positive cocci in pairs and short chains with its characteristic lancet shaped appearance and no Gram-negative diplococci were seen.\nCulture of the abscess material on blood and chocolate agar in 5% CO2 atmosphere revealed alpha hemolytic slightly mucoid colonies with “punched-out” center as the colonies age. No growth on MacConkey agar and anaerobic culture media was observed. Identification was done using catalase, bile solubility and optochin susceptibility tests. Catalase test was negative. Bile solubility test induced lysis of the organism using the tube method while the optochin susceptibility test showed a zone of inhibition of 16 mm with a 6 mm of 0.5 μg optochin disk. Antibiotic susceptibility testing done by disk diffusion method (using Mueller Hinton agar supplemented with 5% defibrinated sheep blood and the plate was incubated at 35°C in 5% CO2 atmosphere for 24 h) revealed resistance of the organism (no zone) to 1 μg oxacillin disk from oxoid (Unipath, Ontario, Canada). Hence, minimal inhibitory concentration (MIC) was done according to clinical and laboratory standards institute (2012)[] using Phoenix automated microbiology system (BD Diagnostics, Sparks, MD), which revealed penicillin MIC of 2 μg/ml that indicates susceptibility to parentral penicillin, amoxicillin, amoxicillin-clavulanic acid, cefepime, cefotaxime, ceftriaxone and meropenem.[] The organism was sensitive to amoxicillin (MIC 1 μg/ml), cefuroxime (MIC ≤ 0.5 μg/ml), cefotaxime (MIC ≤ 0.5 μg/ml), cefepime (MIC ≤ 0.5 μg/ml), meropenem (MIC 0.5 μg/ml), erythromycin (MIC ≤ 0.0625 μg/ml), tetracycline (MIC ≤ 0.5 μg/ml), levofloxacin (MIC ≤ 0.5 μg/ml) and vancomycin (MIC ≤ 0.5 μg/ml). The patient was treated with oral cefuroxime 500 mg twice-a-day for 1 week and repeat culture from the wound site on follow-up was found to be sterile.

[[25.0, 'year']]

F

{'16081994': 1, '2108028': 1, '12962350': 1, '18475349': 1, '8070113': 1, '12887119': 1, '19332936': 1, '31023734': 1, '16925067': 1, '16276738': 1, '900177': 1, '28875758': 1, '14978696': 1, '19445813': 1, '24701109': 2}

{}

3968626-1

noncomm/PMC003xxxxxx/PMC3968626.xml

Urinary Tract Infection due to Paenibacillus alvei in a Chronic Kidney Disease: A Rare Case Report

The patient is a 60-year-old male, farmer by occupation, residing in a village complained of dysuria and fever for which he consulted the local physician. He was advised to do the minimum available investigations, which showed fasting blood sugar 81 mg/dL; hemoglobin (Hb) 9.6 gm/dL; and total leukocyte count 11,200 cells/mm3. His differential count revealed increased percentage of neutrophil (85%). Routine and microscopic examination of urine showed presence of albumin (1+), plenty of pus cells, and 2-3 epithelial cells per high power field. Based on this report, the patient was advised oral administration of Norfloxacin 400 mg twice daily for 7 days. But 12 days later, patient complained of right side flank pain and increased intensity of fever for last 8 days. So he was referred to the tertiary care hospital for consultation, where different investigations were carried out. Ultrasonography of abdomen revealed hypoplastic right kidney []. Serum urea and creatinine levels were 56 and 1.3 mg/dL, respectively. The patient was diagnosed as a case of chronic kidney disease and his urine sample was sent to the Department of Microbiology for different investigations. Routine and microscopic examination of urine revealed presence of albumin (1+), plenty of pus cells, 1-2 epithelial cells per high power field. Bacteriological culture of urine showed significant bacteriuria (105 cfu/ml) and the colonies [] were moist, glossy with an unpleasant odor. Gram staining of colonies detected presence of Gram-positive bacilli bearing ellipsoidal, subterminal spores []. By standard biochemical reactions, the isolate was identified as Paenibacillus alvei.[] A repeat culture of the urine sample was performed to confirm its isolation. The isolate was sensitive to ampicillin, amikacin, nitrofurantoin, cefotaxime, cefixime, and resistant to norfloxacin. Hence, the patient was administered with intravenous injection of 1 g of cefotaxime 8-hourly for 7 days. Then a repeat urine culture was performed which was found to be bacteriologically sterile.

[[60.0, 'year']]

M

{'31832844': 1, '2754007': 1, '2801870': 1, '32995508': 1, '26272571': 1, '7665681': 1, '9053012': 1, '1774295': 1, '27031639': 1, '14608918': 1, '27905924': 1, '26900405': 1, '34841315': 1, '8085788': 1, '24701110': 2}

{}

3968627-1

noncomm/PMC003xxxxxx/PMC3968627.xml

Molecular Characterization of Methicillin-Resistant Staphylococcus aureus Causing Fatal Purulent Pericarditis

A 78-year-old male who was a known case of squamous cell carcinoma of buccal mucosa, which was excised with cervical lymph node dissection two years ago along with systemic hypertension and renal failure (Urea 179 mg/dl, Creatinine 5.28 mg/dl) was hospitalized with complaints of fever since 10 days and dyspnoea on exertion class III of two days duration. A chest X-ray [Figures and ] showed massive pericardial effusion and an electrocardiogram (ECG) [] revealed low voltage waves, ST elevation, atrial fibrillation and a fast ventricular rate. The white cell count was 21 K/μl with 88% neutrophils and liver enzymes were also elevated (SGOT 103 IU/L, SGPT 80 IU/L). The patient developed cardiac tamponade. Emergency pericardiocentesis was done and 800ml of fluid was drained and a pigtail catheter was introduced through a subxiphoid incision and placed under fluoroscopic guidance in the pericardial cavity. Pericardial fluid analysis showed ADA 15.2 U/L, glucose 97.8 g/dl, protein 5.84 g/dl.\nBlood and pericardial fluid cultures grew Gram positive cocci which were identified as MRSA with the Vitek 2 (BioMerieux, Inc., Durham, NC). Both the MRSA isolates (from blood and pericardial fluid) were resistant to multiple classes of antibiotics namely oxacillin, penicillin, trimethoprim-sulfamethoxazole, gentamicin and ciprofloxacin while susceptible to vancomycin, clindamycin, tertracyline, linezolid, quinupristin/dalfopristin, nitrofurantoin, and rifampicin as determined by Vitek 2 system. Vancomycin (1 μg/ml) and daptomycin (0.94 μg/ml) susceptibility was confirmed by Etest (BioMerieux, Inc., Durham, NC). The isolate was intermediately sensitive to erythromycin (1 μg/ml) and a D-test for inducible clindamycin resistance was found to be negative. The MRSA isolate from blood and pericardial fluid were identical and identification was confirmed by amplifying the 16S rDNA gene of the genus Staphylococcus and sau gene specific for S. aureus.[] A multiplex polymerase chain reaction (PCR) was done to simultaneously detect genes responsible for resistance to four classes of antibiotics and the isolate was positive for aac A-aph D, erm (C) and mecA encoding for resistance to aminoglycoside, erythromycin and oxacillin respectively.[] The organism was susceptible to tetracycline and, therefore, tetK and tetM genes were not amplified.[] All the primers were synthesized at Sigma Genosys, India.\nThe patient who was empirically put on piperacillin/tazobactum was shifted to vancomycin when cultures identified the isolate as MRSA on the third day. His renal function worsened and he was put on hemodialysis and mechanical ventilatory support. Subsequently he developed septic shock and metabolic acidosis and expired on the fourth day of admission due to heart failure.

[[78.0, 'year']]

M

{'6601211': 1, '15548780': 1, '8227835': 1, '930941': 1, '18392307': 1, '7811862': 1, '12958230': 1, '19352300': 1, '30079377': 1, '24701111': 2}

{}

3968653-1

noncomm/PMC003xxxxxx/PMC3968653.xml

Unexpected refractory intra-operative hypotension during non-cardiac surgery: Diagnosis and management guided by trans-oesophageal echocardiography

A 48-year-old, dialysis dependent male (ASA physical status III, body weight 82 kg) with polycystic kidney and liver disease and post bilateral nephrectomies status presented with persistent massive ascites, mild pericardial and bilateral pleural effusions. His ascites and effusions were secondary to a combination of obstruction of hepatic venous outflow and compression of the right heart by cysts in the liver and chronic renal failure. He was scheduled for a debulking liver resection and deroofing of cysts to relieve compression. Other pertinent medical history included diabetes and hypertension. He was also on the waiting list for a combined liver and kidney transplant. His pre-transplant cardiac workup included a trans-thoracic echocardiography (TTE) that showed mild left ventricular dysfunction (EF 45-50%), normal right ventricular function, mild pericardial effusion, no LVOT gradient and no valvular or wall motion abnormalities. Nuclear myocardial perfusion scan demonstrated a small reversible septal defect consistent with myocardial ischemia and a follow-up coronary angiography showed normal coronary circulation.\nGeneral anaesthesia was induced after establishing standard ASA monitors and central venous pressure (CVP) was monitored from a pre-existing right internal jugular dialysis catheter. At baseline his blood pressure (BP) was 107/56 mmHg, heart rate (HR) 102 bpm, CVP 16. Intravenous bolus induction drugs include fentanyl 150 mcg, midazolam 2 mg, propofol 150 mg, and rocuronium 40 mg. Anaesthesia was maintained on O2/N2O (50:50), sevoflurane at 0.5-1 MAC and fentanyl boluses as required. His ventilation parameters included a tidal volume of 8 ml/kg, respiratory rate of 10/min and PEEP of 8 cm H2O. A 20-gauge radial arterial line and a 14-gauge intravenous cannula were inserted in the left arm post induction. He became progressively hypotensive, prompting volume replacement, several bolus doses of phenylephrine (100-200 μg aliquots) and initiation of IV norepinephrine infusion. Hypotension and tachycardia worsened following drainage of a large ascites, despite aggressive volume replacement with colloid and crystalloid guided by trends in CVP (range 9-14 cm H2O), systolic pressure variation (SPV) on the arterial line tracing (7-14 mmHg) and increasing norepinephrine infusion (0.02-0.15 mcg/kg/min). Given his continued hypotension, IV epinephrine infusion (0.02-0.07 mcg/kg/min) was initiated, which resulted in further deterioration of haemodynamics. (Please see for haemodynamic trends).\nEmergent TOE demonstrated an under filled and hyper dynamic LV with SAM of the AML leading to functional LVOT obstruction and moderate mitral regurgitation (MR) [Figures and ]. Hypotension resolved after cessation of epinephrine, further volume replacement, increasing norepinephrine infusion (0.3 mcg/kg/min), addition of IV vasopressin infusion (0.04 units/min) and reduction in heart rate with beta blocker (10 mg of IV metoprolol in increments).\nThe procedure lasted 4.5 hours and was remarkable for a large blood loss of 1500 cc requiring blood and blood product transfusion. He was transferred to ICU postoperatively, extubated on day 3 and weaned off pressors on day 5. He was discharged from ICU on day 6 and from hospital on day 17. He returned back after 6 months to receive a combined liver and kidney transplantation successfully without any complications.

[[48.0, 'year']]

M

{'26310923': 1, '28523177': 1, '18821909': 1, '11028493': 1, '11886323': 1, '18490319': 1, '24700891': 1, '25927407': 2, '21636935': 1, '16290226': 1, '20592665': 1, '24700900': 2}

{'4424508-1': 1}

3968654-1

noncomm/PMC003xxxxxx/PMC3968654.xml

Emergency caesarean delivery in a patient with cerebral malaria-leptospira co infection: Anaesthetic and critical care considerations

A 24-year-old primigravida, at 37 weeks of gestation was referred to our institute for further management on supplemental oxygen (Venturi mask) after Rapid test diagnosis of Plasmodium falciparum (Pf) malaria (ParaSight™-F, Becton Dickinson, Sparks, MD) associated with acute icteric leptospirosis (microscopic agglutination test (MAT) titers for Leptospira ictero-haemorrhagiae of 1:400). Her history revealed high grade fever, decreased urine output with yellowish discolouration and altered sensorium. She was disoriented, febrile and icteric and had sub-conjunctival haemorrhages and fine basal crepitations on auscultation. She was receiving intravenous (i.v.) artesunate and ceftriaxone and doxycycline capsules. Her pre-operative investigations in referral card revealed anaemia, jaundice with deranged liver function, coagulopathy, thrombocytopenia, increased total leucocyte count, elevated blood urea nitrogen and serum creatinine. Abdominal ultrasonography showed a singleton pregnancy, adequate liquor, umbilical artery systolic-diastolic ratio of 2:1 and mild hepato-splenomegaly. Within 1 h of arrival she was posted for emergency caesarean delivery because of non-reassuring fetal heart rate [].\nAfter administering aspiration prophylaxis and taking high-risk consent she was shifted to operation theatre. Basic monitors were attached and rapid sequence intubation with cricoid pressure was carried out using thiopental and succinylcholine. Right internal jugular vein and left radial artery were cannulated under ultrasonographic guidance simultaneously by two anaesthetists. Her baseline serum glucose level was normal. Anaesthesia was maintained with isoflurane in N2O-O2 mixture and atracurium until delivery of a 2.25 kg baby. After cord separation, fentanyl was administered. Oxytocin (25 IU) infusion was started in titrated doses. Neonatal data recorded were an Apgar score of 6 and 8 at 1 and 5 min respectively, with cord blood pH of 7.28, base deficit of 5.4 and blood sugar level of 50 mg/dl. Hypotension started after placental separation. Blood loss was managed with packed red blood cells, fresh frozen plasma and platelets transfusion followed by noradrenaline infusion (titrated doses). At the end of the surgery, blood gases showed mild acidosis, but normal electrolytes and serum glucose. She was shifted to intensive care unit (ICU) in intubated state and on noradrenaline infusion. Post-operative analgesia was maintained by ultrasound guided bilateral transverse abdominis plane block. In ICU, on the second postoperative day, she developed pulmonary haemorrhage []. Considering coagulopathy, blood products were transfused, but it did not benefit. Bed side fibre-optic bronchoscopy was carried out, which revealed erythematous mucosa, blood clots, active oozing and diffuse bleeding from distal airway. Broncho alveolar lavage was performed and Factor VIIa (AryoSeven, AryoGen Biopharma Co, Tehran, Iran) was administered (50 μg/Kg of rFVIIa in 50 ml distrilled water through the fibre-optic bronchoscope (25 ml in each main bronchus after lavage) because of its thrombogenic effect. Following this immediate bleeding cessation and significant reduction of inspired oxygen fraction (FiO2) was observed in the next 12 h. Her chest radiograph, which showed bilateral non-homogeneous opacities (lower and middle zones), the previous day became almost clear the next day. In view of acute kidney injury, severe hyperbilirubinemia and blood picture indicating haemolytic uremic syndrome (high serum lactate dehydrogenase, schistocytes in peripheral blood), nephrology opinion was obtained and based on their suggestion, plasmapheresis followed by dialysis was done (seven cycles each). Because of the altered mental status, computed tomography of the brain was carried out, which showed diffuse cerebral oedema. for which cerebroprotective measures (head elevation, not rotating neck, maintain serum sodium 145-155 Meq/L, keeping blood partial pressure of carbondioxide 32-40 mmHg) were undertaken. Other investigations such as blood cultures, urine cultures, Weil-Felix test, typhi dot, dengue, and hepatitis viruses serology yielded negative results. Supportive therapies like stress ulcer prevention, deep vein thrombosis prophylaxis (pneumatic compression) and glycemic control were provided. By the fourth post-operative day, the intensity of fever started reducing, consciousness improved, inotropic support tapered, urine output improved and liver function test with coagulation parameters showed improving trends. Her Pao2/Fio2 ratio also gradually improved. By the sixth post-operative day, she could be extubated. Subsequently, she was shifted to wards and safely discharged from the hospital.

[[24.0, 'year']]

F

{'28355302': 1, '23327017': 1, '19547702': 1, '34502012': 1, '8203710': 1, '27066212': 2, '12092039': 1, '21840208': 1, '17184515': 2, '12812350': 1, '11874897': 1, '24700901': 2}

{'4823419-1': 1, '1794493-1': 1, '1794493-2': 1, '1794493-3': 1, '1794493-4': 1, '1794493-5': 1, '1794493-6': 1}

3968655-1

noncomm/PMC003xxxxxx/PMC3968655.xml

Cytoreduction with hyperthermic intraperitoneal chemotherapy: An anaesthetic challenge

A 52-year-old postmenopausal lady weighing 74 kg, diagnosed to have pseudomyxoma peritonei was planned for cytoreductive surgery (CRS) with HIPEC presented to the pre-anaesthesia clinic. She had gross abdominal distension [] and bilateral pitting pedal oedema. Vital signs were within the normal range. Her haemoglobin (Hb) was 7.6 g% and blood group A negative. Echocardiography was normal. Pre-operatively, she was prepared with incentive spirometry and packed cell transfusion to bring her Hb to the optimal level. Our hospital has a policy of improving the Hb to at least 9 g% when major surgery with massive blood loss is anticipated. Prior to surgery anti-aspiration prophylaxis was administered and nasogastric tube was inserted. Intraoperative monitoring included non-invasive blood pressure, electrocardiogram, pulse oximetry, invasive arterial pressure, central venous pressure (CVP), capnography and temperature monitoring. Due to the grossly distended abdomen modified rapid sequence induction was conducted using injection propofol and injection scoline. Following intubation ventilation was difficult with airway pressure reaching 40-42 cm of H2O. We could achieve a tidal volume of only 150 ml, at this point the end tidal carbon dioxide (ETCO2) ranged from 50 to 56 mm of Hg and saturation fell to 76% with a fraction of inspired oxygen (FiO2) 100%. However, bilateral air entry was equal. The surgeons were requested to open the abdomen and do decompression. Over a period of time slowly ventilation improved. Anaesthesia was maintained with air, oxygen, sevoflurane, atracurium and morphine.\nAfter partial removal of mucinous material, airway pressure improved to less than 20 cm H2O and we were able to come down on the FiO2-40% and saturation came up to 100%.\nMucinous material weighing 21 kg was removed from the abdomen. Pan hysterectomy and anterior abdominal peritonectomy was done. After 10 h of surgery with blood loss of around 5500 ml as more than 50% of the procedure was still pending, decision was taken by the team to do the rest of the surgical procedure and HIPEC as a staged procedure. Patient received 8 units of packed cells along with blood products, adequate crystalloids and colloids. Intraoperative arterial blood gas (ABG) was normal with Hb 10 g%. Patient was shifted to post-operative intensive care unit (ICU) for elective ventilation on intravenous morphine analgesia. She was extubated the following day.\nThe second stage was planned after 2 weeks and meanwhile patient was given high protein parentral nutrition, deep vein thrombosis prophylaxis and was advised spirometry. On the day of surgery following an uneventful anaesthesia, total peritonectomy with complete omentectomy was done. HIPEC was initiated by a CLOSED technique [], five drainage tubes (two inlet and three outlet tubes) were inserted into the abdomen and skin closed. Perfusate was peritoneal dialysis fluid along with chemotherapy agent mitomycin C at a temperature of 42°C. The solution was circulated for 70 min in the abdominal cavity by tilting the operation table in different directions and by shaking the patient to facilitate the circulation of the solution. During the HIPEC period, a raise in body temperature above 42 should be alerted to the perfusionist so temperature should be recorded at 15 min interval, similarly, urine output should also be measured at 15 min interval as most of the chemotherapeutic agents are nephrotoxic, blood sugar and lactate were monitored every 15 min.[] ABG was checked at 30 min interval. Throughout, the procedure patient was haemodynamically stable with adequate urine output. The surgery lasted for 7 h with blood loss of 3500 ml. Six units of blood and other blood products along with adequate crystalloids and colloids were replaced. At the end of the procedure, cooling measures were adopted by switching our warming blanket (Geratherm Operation System) to the cooling mode at 34°C, we used cooled saline to replace the lost volume and icepacks wrapped in sterile sheets were placed in the groin and axilla by the surgeons. Our observation was that temperature gradually came down to normal within 30 min. Patient was shifted to ICU for elective ventilation, the next day she was extubated without complications.

[[52.0, 'year']]

F

{'15685704': 1, '8160994': 1, '2118420': 1, '19453319': 1, '23021714': 1, '10819370': 1, '6766084': 1, '15650630': 1, '10655918': 1, '18336490': 1, '29643552': 1, '30618002': 1, '24700902': 2}

{}

3968656-1

noncomm/PMC003xxxxxx/PMC3968656.xml

Anaesthesia for a child with adrenoleukodystrophy: A case report and review of the literature

Our patient was a 9-year-old Indian male with X-linked cerebral ALD who required general anaesthesia for a permanent catheter insertion via left internal jugular vein for plasmapheresis. The patient was diagnosed in June 2011 at 6 years of age when he presented with partial seizures. He underwent an unrelated donor umbilical cord blood transplant (minor B->O incompatibility) and maintained stable donor chimerism. Post-operative recovery was uneventful and he led a relatively normal seizure free life.\nHe presented on August 2012 with blood-stained urine and chest pain. Investigations determined that he was suffering from severe autoimmune haemolytic anaemia, liver dysfunction and steroid related side effects.\nPhysical examination revealed a 46 kg male with pallor, yellow sclera and cushingoid facies. He was confined to the wheelchair and demonstrated hypotonia. The child showed limited interaction with his environment. Pre-operative medications included levitiracetam, fludrocortisone and famotidine. His pre-operative investigations showed a haemoglobin concentration of 3 g/dL, which eventually improved to 6.9 g/dL after several cycles of plasmapheresis and blood transfusion, and a raised total bilirubin of 97 μmol/L.\nStandard monitoring was attached, and intravenous hydrocortisone 100 mg was administered. The patient was tilted 30 degrees head-up, pre-oxygenated and cricoid pressure was applied. However, ventilation could not be achieved, and decision was made to release cricoid pressure. Anaesthesia was induced with a target propofol concentration of 4 μg/mL and remifentanil infusion rate of 4 ng/mL. Muscle relaxation was provided with rocuronium 0.7 mg/kg. The trachea was intubated orally with a size 6 cuffed tracheal tube. Maintenance of anaesthesia was achieved with a target propofol concentration of 2.5 μg/mL and remifentanil infusion rate of 3 ng/mL. Post-operative analgesia was obtained with 10 mL of lignocaine 1% infiltrated over the surgical site.\nFollowing the procedure that lasted 105 minutes, residual neuromuscular blockade was reversed with neostigmine 2.5 mg and glycopyrrolate 0.4 mg. Neuromuscular function was assessed with electromyography. After the patient demonstrated airway protective reflexes, spontaneous eye opening and a regular respiratory pattern, the trachea was extubated. Recovery was uneventful, and he was subsequently discharged to the general ward.

[[9.0, 'year']]

M

{'8218070': 1, '17573390': 1, '16430428': 1, '8111945': 1, '14768930': 1, '17042840': 1, '10903016': 1, '29119207': 1, '5468914': 1, '19453585': 1, '19457894': 1, '1563064': 1, '24700903': 2}

{}

3968657-1

noncomm/PMC003xxxxxx/PMC3968657.xml

Anaesthesia for autotransplantation after extracorporeal nephron sparing surgery for bilateral giant renal angiomyolipoma

A 28-year-old unmarried female weighing 55 kg presented with lower abdominal discomfort and vague lump in the abdomen of 5 years duration. She was diagnosed to have giant tumour in both kidneys. She sought medical advice at different hospitals and the tumour was deemed inoperable. Examination showed huge flank masses. Blood investigations were normal and contrast enhanced abdominal CT with angiogram showed bilateral highly vascular huge renal masses suggestive of angiomyolipoma []. She was taken up for right radical nephrectomy, bench surgery and autotransplantation after adequate pre-operative preparation.\nWe instituted general anaesthesia supplemented with low thoracic epidural at T11-12 interspace and epidural analgesia was maintained with 0.2% ropivacaine and fentanyl 2 mcg/ml at the rate of 4-6 ml/h. Anaesthesia was induced with glycopyrrolate 0.2 mg, midazolam 1mg, ramosetron 0.3 mg, fentanyl 100 mcg and propofol 100 mg. Intubation was facilitated with 100 mg succinylcholine and anaesthesia was maintained with N2O, O2, isoflurane and atracurium.\nElectrocardiogram, pulse oximetry, invasive blood pressure, end-tidal carbon dioxide, central venous pressure (CVP), core temperature and hourly urine output were monitored. Abdomen was entered through a right thoracoabdominal incision [], excising the tenth rib and splitting the diaphragm. Right kidney with the mass (30 cm × 15 cm) was removed and cannulated for cooling with the histidine tryptophan ketoglutarate solution. The warm ischemia time was 1 min and 45 s. Bench dissection was carried out and kidney was autotransplanted. Cold ischemia time was 4 h. During this period, the anaesthetic goal was to maintain mild hypertension. Mannitol was given for renoprotection (0.5 g/kg). Patient had massive blood loss approximately 5 l, which was corrected with five units of packed cells, four units of fresh frozen plasma, 9 l of crystalloids and 1 l of colloid. Dopamine and subsequently dobutamine infusion were administered to maintain her haemodynamic parameters. Urine output was sluggish after the release of cross clamps. Patient was electively ventilated. She had continuous oozing from the wound site suggestive of coagulopathy due to massive blood loss and transfusion which was corrected with further transfusion of blood components (two units of packed cells, three units of fresh frozen plasma, four units of platelets and four units of cryoprecipitate). She was haemodynamically stable and coagulopathy was corrected by the second post-operative day and she was weaned off inotropic and ventilatory support.\nAfter 4 weeks, she was posted for the left nephrectomy with bench surgery and autotransplantation as she had a large tumour (26 cm × 18 cm) in the left kidney with high risk of bleeding. Renal Doppler showed suboptimal function of right transplanted kidney. Only a single arcuate artery in the lower pole cortex picked up Doppler flow with increased resistance index. Anaesthetic plan was to restrict fluids intraoperatively and to maintain haemodynamic status. The surgical technique and induction of anaesthesia were similar to what was used for the right nephrectomy. Patient was adequately hydrated before nephrectomy with 3 l of crystalloids to maintain adequate urine output. Warm ischemia time was 2 min 13 s. Cold ischaemia time was 2 h 48 min. After the nephrectomy, fluids were restricted with a goal to maintain CVP of 6-7 cm of water. Before the release of the cross clamp after transplantation, CVP was increased to 16-18 cm of H2O. The target blood pressures were systolic more than 140 mm Hg, mean arterial pressures more than 95 mm Hg and the diastolic pressure more than 85 mm Hg as was practiced in renal allotransplantation. Frusemide 200 mg and sodium bicarbonate 25 ml were administered and urine output was adequate after reperfusion. Blood loss was around 1.5-2 l. She was infused nine litres of crystalloids, 500 ml colloid and two units of packed cells. The surgery lasted for 6 h. She was ventilated overnight and extubated next day. The urine output was 3.6 l. At the time of discharge her creatinine was 1.4 mg/dl and it was 1.5 mg/dl on day 30. The follow-up angiogram after 1 month showed a well-perfused kidney on the left side.

[[28.0, 'year']]

F

{'20640215': 2, '8614094': 1, '9834595': 1, '20418304': 1, '22982456': 1, '17240527': 1, '24700904': 2}

{'2894490-1': 1}

3968658-1

noncomm/PMC003xxxxxx/PMC3968658.xml

Radiographic mislead: apparent arterial placement of subclavian central venous catheter due to mediastinal shift

A 32-year-old lady with leiomyosarcoma of hand, underwent amputation of right forearm. On second postoperative day, her general condition gradually deteriorated. She was shifted to intensive care unit (ICU) in view of tachypnoea and desaturation to 84% on oxygen (60% byVenturi) support. She was haemodynamically unstable with a heart rate of 120/min and blood pressure (BP) of about 85/45 mmHg. Auscultation of chest revealed decreased air entry on the right side of chest. Patient did not co-operate for the noninvasive ventilation. She was intubated with a 7.0 mm internal diameter cuffed Portex© endotracheal tube (ETT) and started on dopamine infusion at 10 μg/kg/min through 18 gauge peripheral intravenous (IV) line. A 15 cm seven French triple lumen CVC was secured in the right subclavian vein in first attempt using the Seldinger technique, and backflow of blood was confirmed in all three lumens. We planned to administer ionotropes through CVC once its placement was confirmed by a chest radiograph. Urgent portable chest radiograph was sought for and it showed CVC crossing the midline with gradual turn leftwards and downwards []. The position of the CVC tip was above and on the right side of carina, but it never in its course crossed the trachea []. The radiograph also revealed right-sided massive pleural effusion and tracheal shift to the left side []. Pressure transducer was not available at that point of time. So, to confirm correct placement of CVC tip, two separate samples for blood gas analysis were sent, one from CVC and another from radial artery, which confirmed that CVC was in subclavian vein. Ionotropes were then administered through the CVC and the patient improved haemodynamically. Malignant pleural effusion was diagnosed provisionally. Right-sided intercostal drain (ICD) was inserted. Plain and contrast computerized tomography (CT) of thorax revealed loculated right-sided pleural effusion with right-sided lung collapse and marked mediastinal shift of the cardia and great vessels to the left. Patient's condition gradually improved in the next three days. She was extubated and finally discharged from ICU on oxygen (35%) by Venturi.

[[32.0, 'year']]

F

{'23716794': 1, '21633555': 1, '7653766': 1, '20972824': 1, '3665610': 1, '20606809': 1, '16415318': 1, '111506': 1, '18703308': 1, '18326129': 1, '26637319': 1, '20859495': 1, '24700905': 2}

{}

3968661-1

noncomm/PMC003xxxxxx/PMC3968661.xml

Acute fatty liver of pregnancy complicating a twin pregnancy

A 37-year-old primigravida with twin gestation at 34-week was admitted with epigastric discomfort, abdominal pain, malaise and vomiting. Diagnosed with hypertension in third trimester, she was prescribed alpha methyldopa. Heart rate was 102/min and blood pressure was 170/90 mm Hg. Icterus was noticed later on the day. Foetal heart sounds were heard. Ultra sonography of abdomen showed fatty liver and twin pregnancy []. Investigations: haemoglobin - 10.7 g%, total count - 10,700/mm3, platelets - 2.6 lakhs, urea - 11 mg/dl, creatinine - 1.4 mg/dl, uric acid - 5.3 mg/dl, lactate dehydrogenase - 442 U/L, peripheral smear negative for haemolysis. Hepatitis B surface antigen, hepatitis C virus and human immunodeficiency virus negative, prothrombin time test 48 s control - 14 s, International Normalised Ratio - 4.0, total bilirubin - 7.8 mg/dl (0.3-1.0 mg/dL), direct bilirubin - 4.0 mg/dl (0z, serum glutamic oxaloacetic transaminase - 316 U/L (0-35 U/L), serum glutamic-pyruvic transaminase - 313 U/L (0-35 U/L), alkaline phosphatase 974 (30-300 IU/L), total protein - 5.3 U/L, albumin - 2.8 U/L, globulin - 2.5 U/L, plasma ammonia - 93 meq/d, urine albumin++, cardiotocography showed evidence of foetal distress. A presumptive diagnosis of pre-eclampsia and/or acute fatty liver of pregnancy (AFLP) was made. Emergency caesarean section was planned under general anesthesia. She was given vitamin K and fresh frozen plasma (FFP). Induction by rapid sequence using thiopentone, followed by suxamethonium, trachea intubated using size seven endotracheal tube. Anaesthesia maintained with oxygen: Nitrous oxide, sevoflurane, fentanyl and atracurium. Mannitol and albumin were transfused to prevent hepatorenal syndrome. Twin babies delivered were healthy. Extubation and recovery were uneventful. Cryoprecipitate and FFP were transfused in post-anaesthesia care unit. Broad spectrum antibiotics started. Magnesium sulphate was continued at 2 g/h. Serum magnesium levels monitored 6th hourly were normal. On the second post-operative day, liver function deteriorated further. On the 3rd post-operative day, she had two episodes of tonic clonic convulsions and respiratory distress. Chest X-ray was suggestive of acute respiratory distress syndrome. Mechanical ventilation initiated in synchronized intermittent mandatory ventilation mode with pressure support. Antibiotic was changed to tobramycin based on culture and sensitivity report. Computed tomography scan brain and electroencephalography were normal. Repeat ultra sound scan of abdomen showed ascites with hyperechoic features suggestive of haemorrhagic ascites as the patient already had coagulopathy. There were no gall stones or dilatation of biliary tract. Central venous pressure was maintained between 8 and 12 mm Hg. Weaned off on the 7th day but non-invasive ventilation was continued for three more days. Hepatic function gradually improved. Viral markers were negative on repeated tests. Coagulopathy was corrected with a total of 32 units of FFP, 4 units of platelets and 12 units of cryoprecipitate. Patient gradually improved with supports and was shifted out from post-operative intensive care unit on the 20th day.

[[37.0, 'year']]

F

{'16215356': 1, '16394048': 1, '19881179': 2, '12427793': 1, '12037242': 1, '8055222': 1, '7977544': 1, '3892299': 1, '18332072': 1, '10406228': 1, '24700907': 2}

{'2772253-1': 1}

3968662-1

noncomm/PMC003xxxxxx/PMC3968662.xml

Intraoperative interfascicular ventricular tachycardia: A rare occurrence

A 66-year-old, 70 kg male patient with a BMI of 33 presented for laparoscopic cholecystectomy. He was a chronic smoker with chronic obstructive pulmonary disease which required salbutamol MDI. He was a hypertensive on treatment with oral ramipril and losartan. He had one episode of acute coronary syndrome 18 months back which required percutaneous intervention with stenting of right coronary artery. Subsequently, the patient was advised to take aspirin and clopidogrel orally which had been stopped 5 days prior to the surgery. History pertaining to other organ systems was unremarkable. On preoperative evaluation he was NYHA class II with decreased effort tolerance (<4 METs). His ECG showed normal sinus rhythm with left anterior fascicular block [] while his resting echocardiography showed regional wall motion abnormalities in the right coronary artery territory with an ejection fraction of 40%.\nIn the operation theatre ECG, non-invasive blood pressure and SpO2 monitor were attached. After intravenous access and radial artery cannulation, anaesthesia was induced with 150 mcg of fentanyl and 100 mg of propofol. 35 mg of atracurium was given to attain muscle relaxation to facilitate tracheal intubation. There was a fall in blood pressure to 70/40 mmHg after induction which was managed with 6 mg of ephedrine. The haemodynamics recovered to normal limits after endotracheal intubation. However, immediately after creation of pneumoperitoneum, the patient developed regular WCT with a ventricular rate of 150/min and a mean blood pressure of 80-90 mmHg. The abnormal heart rhythm immediately reverted back to normal on deflation of pneumoperitoneum. An open cholecystectomy was planned in consultation with the surgeons. Following skin incision the same pattern of WCT recurred. The arrhythmia reverted spontaneously within 3 to 4 min of stopping the surgery. The surgery was abandoned and the patient immediately shifted to ICU without reversal of anaesthesia. In the ICU, the patient had multiple episodes of WCT with severe hypotension which required synchronised DC cardioversion with 100-150 joules biphasic energy. An infusion of dopamine was also started at the rate of 10 mcg/kg/min. Cardiologist was urgently summoned who diagnosed the WCT as interfascicular VT on 12 lead ECG [Figure , ] and thus a loading dose of 150 mg of amiodarone was administered over 10 min intravenously followed by infusion at 1 mg/min for 6 hours and 0.5 mg/min for subsequent 18 hours. Despite the treatment the patient had multiple episodes of WCT which required cardioversion again with 150 joules of biphasic energy. All his biochemical parameters like serum electrolytes, renal and liver function tests were normal, and no increase in cardiac enzymes occurred thus ruling out possible myocardial ischemia. An echocardiography could not be done at as facilities of in-bed echocardiography were not available with us. The patient was weaned off inotropes over a period of 6 hours, the tracheal tube was subsequently removed, and he could be discharged from the hospital after 3 days.

[[66.0, 'year']]

M

{'2914345': 1, '21410719': 1, '15965569': 1, '21343901': 1, '18471458': 1, '2022022': 1, '20956224': 1, '21131372': 1, '17272358': 1, '24700908': 2}

{}

3968663-1

noncomm/PMC003xxxxxx/PMC3968663.xml

Successful anaesthetic management of a patient with prior history of malignant hyperthermia for corrective scoliosis surgery

A 14-year-old girl was scheduled for correction of idiopathic infantile right thoracolumbar scoliosis. The surgery was previously attempted at a different hospital 2 years before. But, because she developed features suggestive of malignant hyperthermia, the procedure was abandoned. She had apparently developed some features during the 3 h of intra-operative period; tachycardia, muscle rigidity, hypercarbia, dark-coloured urine and hyperthermia (1080 F). On laboratory evaluation, she was found to have respiratory acidosis and elevated CK levels, which settled over the next 3 days, for which she was treated symptomatically and discharged. Muscle biopsy performed at a later date showed congenital myopathy with type 1 fibre predominance.\nHer past history revealed delayed gross motor milestones, recurrent respiratory tract infections and progressive spine deformity with back pain. With a history of easy fatigability and dyspnoea on mild exertion and a breath holding time of 8-10 s, she presented a picture of gross respiratory compromise, which was confirmed by pulmonary function tests that revealed severe restrictive defect [forced expiratory volume in 1 second (FEV1) - 25% predicted; forced vital capacity (FVC) - 28% predicted, FEV1/FVC - 83% predicted] and not responding to bronchodilators. Other than being underweight, her general physical and airway examinations were normal. All laboratory investigations (complete blood picture, blood urea, serum creatinine, serum electrolytes, coagulation profile, liver function tests) and two-dimensional echocardiogram were normal. X-ray chest showed right thoracic scoliosis with left lumbar compensation, with a Cobb's angle of 90 degrees and normal lung fields. Baseline arterial blood gases (ABG) and CK were normal and urine tested negative for myoglobinuria.\nInformed consent was taken after counselling the family members regarding all the potential complications – recurrence of malignant hyperthermia, rhabdomyolysis, increased blood loss, haemodynamic fluctuations, delayed recovery, post-operative ventilator support and intensive care unit (ICU) stay.\nOn the day of the surgery, the anaesthesia workstation was flushed with 100% oxygen after removing the vaporizers. Fresh soda lime and anaesthetic circuit were used. Dantrolene, all other resuscitation drugs and ice packs were kept ready. Pre-operative vitals and arterial blood sample on room air were normal. She was wheeled into the operating theatre and anaesthesia was induced with inj fentanyl 2 mcg/kg and inj propofol 2 mg/kg. After confirming adequate ventilation, inj atracurium 0.5 mg/kg was administered and intubated with an oral cuffed endotracheal tube 6.5 and connected to the ventilator. The right internal jugular vein and the left radial artery were cannulated. She was then turned prone and the pressure points were adequately padded. Oral airway was placed to prevent tongue bite during monitored evoked potential (MEP) stimulus.\nApart from standard monitoring, neuromuscular monitoring was also performed using MEP. Anaesthesia was maintained using oxygen – air mixture 40:60 and propofol (125-150 mcg/kg/min) and fentanyl (1-1.5 mcg/kg/h). No further muscle relaxants were administered. The procedure lasted for nearly 7 h and was uneventful. MEPs were adequate during the procedure. Intra-operative blood loss was approximately 1500 mL and four units of whole blood were transfused. Intra-operative urine output was 1 mL/kg/h. At the end of surgery, the patient was turned supine and muscle relaxation was reversed. Extubation was performed as she was fully conscious, obeying commands and had stable respiratory parameters.\nPost-operatively, her ABG revealed respiratory acidosis. As she was conscious and co-operative, intermittent bilevel positive airway pressure (BiPAP) support was given for 2 days post-operatively. She was shifted out of the ICU on the 4th post-operative day and discharged on the 6th post-operative day.

[[14.0, 'year']]

F

{'10790202': 1, '7676331': 1, '10928000': 1, '22013246': 1, '14732615': 1, '19762745': 1, '14504148': 1, '24700909': 2}

{}

3968665-1

noncomm/PMC003xxxxxx/PMC3968665.xml

An adult non-obese male, a case of obstructive sleep apnoea posted for tonsillectomy and septoplasty - challenges faced

A 30-year-old male patient with history suggestive of OSA was posted for tonsillectomy and septoplasty under general anaesthesia. He had history of recurrent attacks of rhinitis on and off over a year. Besides, he gave history of snoring severe enough to disturb the person next door, episodes of airway obstruction, probable hypopnoea during sleep and daytime somnolence. He was thus investigated and polysomnography done. The study showed minimum SpO2 values of 51% lasting up to 10 seconds, seven episodes of SpO2 <88%, total snoring episodes of 205 and mean duration of snoring episodes of 6 seconds. The average minimum O2 saturation was 83.9%, maximum heart rate 141/min with an average of 96.4/min and desaturation index of 81.1. The AHI (Apnoea hypopnoea index) was significantly high at 80.0/hour. He had enlarged tonsils and left deviated nasal septum. On admission, a thorough preoperative evaluation was done. General and systemic examination did not suggest any gross abnormality. The blood pressure was 130/80 mmHg. The calculated BMI (body mass index) was 26.1 kg/m2 and neck circumference 41cm. He had a high arched palate, large tongue [] and an MPC (Mallampati classification) grading of 3. The thyromental distance was 6cm and sternomental distance was 11.7 cm. The routine investigations including the PCV (Packed cell volume), ECG (Electrocardiogram), CXR (Chest X-ray), thyroid function and coagulation studies were normal. After a thorough preoperative assessment, confirmation of NBM (Nil by mouth) status and written informed consent, the patient was taken up for tonsillectomy under general anaesthesia. He was premedicated with inj. atropine 0.6mg IM and inj. diclofenac sodium 75 mg IM half an hour prior to surgery. After securing the intravenous line, inj. ranitidine 50mg. and inj. ondensetron 4 mg were given. The patient was preoxygenated with 100% oxygen for five minutes and induced with gradually increasing concentration of sevoflurane up to 8% with 100% oxygen. Obstruction during mask ventilation was noted and an oral airway was thus inserted. After good jaw relaxation, decrease in muscle tone and depth of respiration, laryngoscopy was attempted. Laryngoscope was difficult to negotiate due to large tongue and only the epiglottis was visualised (Cormack and Lehane grade III), suggesting difficult intubation. Blind nasal intubation was done using a no. 7.5 cuffed endotracheal tube. The tube was secured and throat pack inserted. Maintenance of anaesthesia was done using o2, sevoflurane and vecuronium as muscle relaxant. Enlarged tonsils were removed. After achieving haemostasis, checking adequacy of reflexes, tone, power and awake status, extubation was done after thorough suctioning under vision and pack removal. Inj. paracetamol 1gm IV was given postoperatively every 6 hourly over 24 hours. Intraoperatively, pulse rate, non-invasive blood pressure, SpO2 (oxygen saturation) and EtCO2 (end tidal carbon dioxide) were monitored. Postoperatively, the patient was kept in right lateral position with oxygen by ventimask and nasopharyngeal airway in situ.\nWe monitored the pulse rate, blood pressure and oxygen saturation throughout the first postoperative night, off O2 mask. Clinically, we found that the number of hypoapnoeic episodes had dropped, the minimum drop in O2 saturation was 80% and the daytime somnolence was considerably lower the next day.\nSeptoplasty was deferred for the present as it would involve plastering the nose and further jeopardize respiration.

[[30.0, 'year']]

M

{'29492464': 1, '20956751': 1, '17721406': 1, '12512263': 1, '16645462': 1, '16840395': 1, '11331179': 1, '14604837': 1, '18931212': 1, '19742249': 1, '24700911': 2}

{}

3968716-1

noncomm/PMC003xxxxxx/PMC3968716.xml

An unusual presentation of Hashimoto's encephalopathy

An 18-year-old male patient was diagnosed to have primary hypothyroidism 6 months back following weight gain, edema and facial puffiness of 3 years duration. His initial thyroid function test (TFT) was T4: 2.3 μg/dl (5.01-12.4) T3: 0.35 ng/ml (0.6-1.81) and thyroid stimulating hormone (TSH): 180 μIU/ml (0.35-5.50) and was started on L thyroxine, with a significant improvement of symptoms. He started developing myoclonus for the past 1 month, for which he was referred to our hospital.\nOn examination, he was conscious, with a cognitive impairment and had memory loss for recent events, with the poor attention span, difficulty in finding words, dyscalculia and dysgraphia. His mini mental status examination (MMSE) was 11 suggestive of moderate cognitive impairment. He also had myoclonus, involving both upper limbs and lower limbs.\nOn investigations, blood routine examination and erythrocyte sedimentation rate was normal and liver function test, kidney function test, sodium, potassium, calcium and magnesium were normal and so were the arterial blood gas analysis. His latest TFTs show normal T3 and T4 with mild elevation of TSH (TSH-9.1 μIU/ml). Anti-thyroid peroxidase antibody was positive (>1300 IU ml). His electroencephalogram (EEG) showed a diffuse slow wave activity [] and magnetic resonance images (MRI) showed a pituitary hyperplasia [] with non-specific White matter changes []. Cerebrospinal fluid (CSF) study had normal cytology, but slightly increased protein.\nAntinuclear antibody titer, anti-double-stranded deoxyribonucleic acid, hepatitis B surface antigen, anti-hepatitis C virus, lupus anticoagulant and Venereal Disease Research Laboratory (VDRL) test were carried out to evaluate the cause of decreased cognitive function, myoclonus and seizures, which were all negative. CSF electrophoresis was normal.\nIn view of these neurologic symptoms, associated with high titers of anti-thyroid antibodies and the exclusion of other possible causes of encephalopathy, patient was diagnosed with Hashimoto's encephalopathy. He was started on methylprednisolone 1 g/day for 3 days and was shifted over to prednisone 1 mg/kg/day. There was a marked improvement in the symptoms with no further episodes of myoclonus or seizures. One month after discharge, the patient reported a sustained improvement on all parameters, including memory and cognition, making it possible for his to return to an almost normal routine. His MMSE had improved to 26 and he is seizure free. Prednisone dose was tapped over the period of 3 months without the recurrence of symptoms.

[[18.0, 'year']]

M

{'7702695': 1, '16476807': 1, '33442088': 2, '30355921': 1, '4161638': 1, '26649860': 1, '32775033': 2, '8865025': 1, '11085193': 1, '12580699': 1, '22231308': 1, '17850170': 1, '28904921': 2, '24701441': 2}

{'5590384-1': 1, '7394228-1': 1, '7784182-1': 1, '7784182-2': 1, '7784182-3': 1, '7784182-4': 1, '7784182-5': 1, '7784182-6': 1, '7784182-7': 1}

3969343-1

noncomm/PMC003xxxxxx/PMC3969343.xml

Indirect cyclopexy for treatment of a chronic traumatic cyclodialysis cleft with hypotony

A 70-year-old Caucasian male followed at the Martinsburg Veterans Affair Hospital Ophthalmology Clinic, Martinsburg, West Virginia, USA, since 2002 was found consistently to have intraocular pressure (IOP) asymmetry, with the right eye IOP ranging from 6–15 mmHg and usually having an IOP 04 mmHg lower than the left eye. He was monitored for primary open-angle glaucoma and was on latanoprost eye drops in the left eye for field defects in that eye. In April 2008, his right eye IOP dropped to 04 mmHg. On further evaluation, gonioscopy revealed a cyclodialysis cleft from 11–4 o’clock. The patient’s history was significant for trauma to the right eye with a piece of wood over 30 years ago, which was thought to be the cause of the cleft.\nWhile the patient had hypotony, he maintained good visual acuity, ranging from 20/25 to 20/40, and was therefore observed for approximately 3 years until December 2011. At this visit, his visual acuity dropped to 20/150 and he clinically developed Descemet’s folds with a shallow anterior chamber and an IOP of 02 mmHg. On dilated fundus exam, he had a shallow choroidal hemorrhage and effusion in the far nasal periphery. The patient was initially treated medically with atropine and prednisolone acetate 1% ophthalmic drops for approximately 2 months, without improvement in IOP and with persistence of choroidal hemorrhage and effusion. Vision also remained poor. Given the chronicity and size of this cyclodialysis cleft, surgical repair was planned.\nAn indirect method of closure was performed and the technique is described below. Postoperatively, the choroidal effusion resolved, but the patient’s IOP did not improve. On repeat gonioscopy, it was determined that there were 2 clock hours of remaining cleft. Therefore, the patient was taken for additional surgical repair of the persistent cleft by the same technique, this time with successful closure.\nOn postoperative day 1 following the second surgery, the patient complained of severe eye pain and nausea, and was found to have an lOP of 58 mmHg. Elevated intraocular pressure was anticipated with successful closure of cyclodialysis cleft. The patient was treated with topical anti-glaucoma drops and oral acetazolamide, with reduction in the IOP to the mid-teens and resolution of his pain. On exam, there was no distortion of the pupil by sutures and the anterior chamber appeared deep (). Repeat gonioscopy also confirmed cleft closure (). The patient was subsequently tapered off the oral acetazolamide and topical drops, with stabilization of the pressure in the mid-teens and maintenance of 20/70 visual acuity at 1 year postoperatively. Final visual acuity was limited by subsequent progression of ocular comorbidities in that eye, including cataract and proliferative diabetic retinopathy.\nA nasal conjunctival peritomy was performed, and dissection down to bare sclera was carried out and continued superotemporally to encompass the region of the cyclodialysis cleft. A 10-0 nylon suture on CS175-6 needle (Ethicon), which is a 7.0 mm ½-circle needle, was selected for cyclopexy. Interrupted 10-0 nylon sutures were radially passed through the corneal side of the limbus, then through the iris root and ciliary body, and carried out through the sclera on the opposite side of the limbus (). To ensure that ciliary body was incorporated into each pass, the sutures were tugged on intraoperatively and the iris root was evaluated for movement with mechanical pull. Additionally, as more sutures were placed, the eye appeared to become more firm. The sutures were placed approximately 1 mm apart. We found the needle type to have a curvature and size that was very helpful for the angle and length of the pass required.

[[70.0, 'year']]

M

{'11706382': 1, '15006862': 1, '778720': 1, '7189850': 1, '29780959': 1, '13497654': 1, '28723783': 1, '7862421': 1, '20051856': 1, '11730666': 1, '31878971': 1, '30267206': 1, '27392912': 1, '24711691': 2}

{}

3969602-1

noncomm/PMC003xxxxxx/PMC3969602.xml

Lung physiology at play: Hemoptysis due to underwater hockey

A 34 year-old male presented to the Emergency Department with hemoptysis following a strenuous game of underwater hockey. Underwater hockey is played in a swimming pool at a depth of 2–4 m. Wearing a snorkel mask and fins, players pass a weighted puck from the bottom of the pool, during repeated apneic dives. The patient reported a cough productive of approximately four teaspoons of bright red blood. Similar episodes of hemoptysis had occurred twice previously, each time following a game of underwater hockey. He denied ever experiencing shortness of breath or chest pain with exercise. Review of systems was negative. There was no history of respiratory disease, and he was a non-smoker.\nOn presentation to hospital, vital signs were normal and physical examination was unremarkable.\nBlood work was normal and included: hemoglobin 155 g/L, platelet count 182 × 109/L, INR 1.0, PTT 26.1 s, anti-nuclear antibody 1:40 (homogenous), rheumatoid factor <8 (negative), negative anti-neutrophil cytoplasmic antibody (ANCA) and anti-glomerular basement membrane antibody levels. Chest x-ray was normal. CT angiogram of the chest revealed normal lung parenchyma, no vascular abnormalities, and no evidence of pulmonary emboli. Trans-thoracic echocardiogram showed borderline concentric left ventricular hypertrophy but normal left ventricular diastolic function. The right ventricular systolic pressure (RVSP) was at the upper limit of normal at 35 mmHg. Stress echocardiogram was normal after 14 min of exercise, with an increase in RVSP from 40 to 50 mmHg. Bronchoscopy revealed slightly erythematous mucosa but no frank bleeding or endobronchial lesions. Bronchoalveolar lavage demonstrated no abnormalities.\nThe patient was discharged from our clinic with no clear diagnosis. Interestingly, he was referred back seventeen years later for an incidental finding of a pulmonary nodule. He reported that he had ultimately stopped playing underwater hockey and experienced no further episodes of hemoptysis. In retrospect, recent literature, reviewed below, quite clearly characterizes the cause of his previous hemoptysis.

[[34.0, 'year']]

M

{'5035546': 1, '15302723': 1, '11273969': 1, '15900938': 1, '15730335': 1, '34960366': 1, '931839': 1, '11742946': 1, '27512564': 1, '10232449': 1, '5725383': 1, '7656948': 1, '9116992': 1, '2055852': 1, '17021008': 1, '26029522': 2}

{}

3969607-1

noncomm/PMC003xxxxxx/PMC3969607.xml

Granulomatous PJP presenting as a solitary lung nodule in an immune competent female

A previously well, 49 year old Caucasian female presented to her family physician with increasingly persistent cough and night sweats. She reported a several year history of ongoing morning cough with dark sputum production. Her medical history was unremarkable with the exception of a 20-pack year smoking history and she had no significant risk for endemic mycoses or tuberculosis exposures. Physical exam was non-contributory. Chest imaging revealed a 1.5 cm2 smoothly marginated, non-calcified, non-cavitary left upper lobe pulmonary nodule (). A surveillance bronchoscopy with brushing was negative for malignancy. Despite this, a repeat computed tomography (CT) scan six months after the bronchoscopy identified the lesion to be increasing to 2.3 cm2. Other than a mildly appreciable left superhilar lymph node, the mediastinal structures were unremarkable. This, concurrent with a 30 lb weight loss over the same period, prompted the patient to undergo a left thoracic wedge resection of the left lung. Areas of necrotizing granulomatous inflammation with PJP cysts were identified on histopathology (). No other pathogens, including aerobic organisms, acid-fast bacilli or other fungal species were in either the bronchoalveolar lavage or biopsy. No other pathologic features were apparent. PJP cysts were not present on an expectorated sputum sample collected two days later.\nAn extensive workup was initiated to elicit obvious immunodeficiencies in the patient given that PJP is not expected in an immunocompetent host. She tested negative for HIV-1&2 (by enzyme EIA assay and P24 antigen), HTLV-1&2, hepatitis B and C. Serum immunoglobulins (IgG, IgM, IgA, IgE), lymphocyte subpopulation count (CD3, CD4, CD8), collagen vascular disease markers (ANA, Rheumatoid factor, GBM antibody, ANCA, anti-MPO, PR3) and malignancy investigation (CT of chest and abdomen) were all within normal limits. She had an ability to mount an appropriate immune response, as evidenced by the presence of IgG antibodies for mumps and rubella, diphtheria and tetanus, suggesting appropriate vaccination against such diseases. Laboratory test for complement C3, complement C4, alpha-1 antitrypsin, anti-actin were normal.\nWhile initially no treatment was provided, a lingering cough prompted a course of trimethoprim-sulfamethoxazole (TMP/SMZ) DS 800/160 mg two tablets by mouth thrice daily for three weeks. Symptoms resolved and did not recur through eighteen months of observation nor did any new syndrome to suggest undiagnosed immunodeficiency. Follow up radiographic imaging did not show evidence of recurrence.

[[49.0, 'year']]

F

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{}

3969627-1

noncomm/PMC003xxxxxx/PMC3969627.xml

A Rare Case of Transient Inferior ST Segment Elevation

The patient is an 86-year-old white male with a past medical history of hypertension and a 12-year history of a hiatal hernia. He does not have a history of diabetes, hyperlipidemia, or coronary artery disease. He is a non-smoker, active for his age, and has no recent history of chest pain. The patient takes 25 mg of metoprolol twice a day for his hypertension and 40 mg of omeprazole daily for GERD.\nThe patient presented to the emergency department with a chief complaint of persistent nausea. He received an abdominal film during his work-up which revealed a large hiatal hernia, which along with his persistent nausea, the possibility of gastric volvulous was entertained []. The patient subsequently underwent an abdominal CT scan that ruled out gastric volvulous and demonstrated a large hiatal hernia in which the stomach was almost entirely in the thorax []. An electrocardiogram while patient was in Emergency Department revealed new ST segment elevation in the inferior leads without reciprocal changes []. The patient continued to have persistent nausea and started to develop pleuritic left sided chest pain. His vital signs remained stable with a temperature of 98.4, 16 respirations a minute, heart rate of 70, blood pressure of 156/63, and an oxygen saturation of 98% on room air. He was admitted for 24-h observation. His cardiac markers remained within normal limits. An echocardiogram revealed no wall motion abnormalities and a preserved ejection fraction.\nDuring the hospitalization, the patient had a number of episodes of large volume emesis, resulting in dramatic improvement of his symptoms. Repeat electrocardiogram revealed resolution of the ST segment elevation []. The patient received supportive care during his hospitalization and was referred to cardiothoracic surgery at discharge. Two months later, the patient received a laparoscopic Nissen fundoplication. The procedure and post-operative course were without complications and the patient has been in good health since the surgery.\nArrhythmias such as atrial tachycardia, atrial fibrillation, supraventricular tachycardia, paroxysmal atrial flutter as well as electrocardiographic changes such as T wave inversion have been reported with large hiatal hernias in previous case reports. The exact mechanism of these electrocardiographic changes is not well understood. Kounis and colleagues hypothesized that an increase in direct or indirect pressure to the global surface of the heart caused electrical alternation seen on electocardiography.[] Schilling and colleagues hypothesized two theories in their case of paroxysmal atrial flutter. First, that compression of the heart caused either ischemic changes or an anatomic conduction block causing the reentry. Second, that the hiatal hernia may cause compression of the vagal innervation to the heart causing electrocardiographic changes.[]\nPatients with large hiatal hernias have been reported to have cardio-pulmonary symptoms as well as electrocardiographic and echocardiographic manifestations in several case reports. These findings are reviewed in []. Hokamaki and colleagues described an interesting case of a 79-year-old woman who developed diffuse ST segment elevation after decompression of a large hiatal hernia.[] In their report, they hypothesize that rapid decompression of the hiatal hernia may have caused pericardial inflammation resulting in pericarditis. Tursi and colleagues also hypothesized that their finding of a supraventricular arrhythmia may have been caused by pericardial irritation.[]\nOur case represents a patient with focal ST segment elevation of the inferior leads, a new electrocardiographic finding associated with large hiatal hernias. The exact mechanism of these electrocardiographic changes is not known. We hypothesize that the focal ST segment elevation in the inferior leads could be related to torsion or compression of the epicardial artery from direct pressure from the hiatal hernia. These electrocardiographic changes could also be related to rotational changes of the heart associated with compression from the hiatal hernia. This may also explain the changing depth and duration of the inferior Q-waves. Regardless of the exact cause of these electrocardiographic changes, once the patient's had repeated episodes of large volume emesis, his hiatal hernia decompressed leading to resolution of his electrocardiographic findings.

[[86.0, 'year']]

M

{'12564111': 1, '15811895': 1, '21382898': 1, '2616418': 1, '15750274': 1, '19918411': 2, '1732116': 1, '18400640': 1, '11847171': 1, '3394624': 1, '15379832': 1, '11197272': 1, '31092493': 1, '8624980': 1, '85405': 1, '31425572': 2, '34336300': 2, '3266413': 1, '11775629': 1, '9633074': 1, '7917355': 1, '17354243': 1, '5640901': 1, '8625669': 1, '24696756': 2}

{'6764569-1': 1, '8286180-1': 1, '2769421-1': 1}

3969642-1

noncomm/PMC003xxxxxx/PMC3969642.xml

A Unique Case of Diffuse Histiocytic Proliferations Mimicking Metastatic Clear Cell Carcinoma in the Hydrocele Sac

A 45-year-old man came with a complaint of scrotal swelling for 1 month and dull dragging pain for 7 days. He gave a history of minor trauma on the scrotum 15 days back. There was no other significant medical history. On local examination, the right hemiscrotum was tender (7 × 6 cm) and did not transilluminate. The left hemiscrotum (8.5 × 7cm) was nontender and transilluminated. There was no erythema or ulcer of the scrotal skin. There was no inguinal lymphadenopathy. Provisional clinical diagnosis of the right hemiscrotal mass with left hydrocele was made. Ultrasonography of the scrotum diagnosed right scrotal pyocele and left scrotal hydrocele. Both the testis and epididymis were normal. Left-sided Jaboulay repair and right scrotal incision and drainage was performed. Preoperative and postoperative intravenous antibiotics were given.\nPartially excised left hydrocele sac was received in multiple bits for histopathological examination []. Histopathological examination showed fibrocollagenous wall of hydrocele sac that was diffusely infiltrated by large cells having clear to multivacuolated cytoplasm []. Clear cells did not infiltrate outside the confines of the hydrocele sac wall. Nuclei were round to oval, showing moderate pleomorphism. Nuclei in few cells were pushed to the periphery with little distortion []. Scant mitotic activity was seen. Few small foci of hemorrhages and lymphocytes were seen. There was no necrosis, significant inflammation, epitheloid granulomas or giant cells. Metastasis of clear cell carcinoma was suspected.\nCytoplasm of the cells did not stain with Mucicarmin stain [Figure ]. In immunohistochemistry (IHC) staining, cells were negative with epithelial membrane antigen (EMA), pan-Cytokeratin and calretinin [Figure –]. Cells showed strong diffuse cytoplasmic positivity with CD68 []. Carcinoembryonic antigen (CEA) showed nonspecific staining [] and cytokeratin 7 (CK7), cytokeratin 20 (CK20) and prostate-specific antigen (PSA) were negative [–]. Absence of significant inflammation, epitheloid granulomas and Michaelis-Gutman bodies in the histology sections ruled out xanthogranulomatous inflammation and malakoplakia. A computed tomography scan of the abdomen and thorax ruled out primary neoplastic lesions in the thorax and abdomen. Final histological diagnosis of diffuse histiocytic proliferation (reactive) in the hydrocele was made.

[[45.0, 'year']]

M

{'17805564': 1, '17882900': 1, '9199643': 1, '11211614': 1, '11917223': 1, '15335251': 1, '22499325': 1, '15185256': 1, '9580050': 1, '28559772': 1, '1109769': 1, '24696560': 2}

{}

3969643-1

noncomm/PMC003xxxxxx/PMC3969643.xml

Necrotizing Soft-Tissue Infection: Laboratory Risk Indicator for Necrotizing Soft Tissue Infections Score

A 64-year-old male patient was brought to the surgical OPD of JSS Medical College, Mysore, Karnataka, on the 29th of February, 2013, with the complaints of fever and a painful blackish lesion on the right lower abdomen. Patient was a diabetic, on dietary modification, under glycemic control. He was also a known case of bronchiectasis on treatment with steroids and had visited a local doctor a week back for neuritic type of pain in the right lower abdomen, for which a Diclofenac patch, had been applied by the doctor. Patient noticed pain and swelling in the area 2 days later. When the lesion started spreading and he also began to have spikes of fever, patient was brought to our hospital.\nOn examination, there was a patch of gangrene on the anterior abdominal wall in the iliac region measuring 5 cm × 5 cm. There was pus oozing from a small area of the wound. Patient was febrile; there were no other significant findings except bilateral basal crepitations.\nIn view of the immunocompromised state, the toxicity and the gangrene, possibility of a necrotizing infection was considered and biochemical and hematological workup was ordered. The LRINEC score at the time of admission was 7 as indicated []. Pus was collected and subjected to Gram stain, acid fast stain and bacterial, mycobacterial and fungal cultures were put up. Gram stained smear revealed the presence of plenty of inflammatory cells, Gram positive cocci in chains and Gram negative coccobacilli. There were also a large number of broad aseptate fungal hyphae, suggestive of Zygomycosis. Blood culture (BacT Alert) did not yield any bacterial or fungal growth.\nIn view of the clinical and laboratory findings, the patient was posted for wide excision and debridement. He was started on treatment with Imipenem, Linezolid and also low dose amphotericin B.\nThere was soakage of the dressing on the 2nd day after surgery, there was sloughing of the skin and subcutaneous tissue and pain persisted. Repeat Gram stain showed the same findings. Aerobic bacterial culture yielded the growth of Enterococci sensitive to Linezolid and Acinetobacter sensitive to piperacillin-tazobactam, imipenem, meropenem and amikacin. The bacterial isolates were identified by using Vitek 2 automated system. Anaerobic cultures (Gaspak) did not yield any bacterial growth.\nFungal culture showed a fluffy wooly white colony with no sporulation after 3 days. On slide culture at 25°C and 40°C in biological oxygen demand incubator, sporulation was observed after 10 days. Broad aseptate hyphae with unbranched sporangiophores, prominent funnel shaped apophyses and pyriform sporangia were observed []. Based on thermotolerance (up to 40°C) and morphological features the fungus was presumptively identified as Apophysomyces elegans.\nHistopathological examination (H and E stain) revealed broad aseptate hyphae suggestive of Zygomycosis. The hyphae were periodic acid schiff (PAS) positive. Vascular invasion was observed.\nAntibiotic treatment was continued, repeat LRINEC scores were validated [] and a repeat wider debridement was done. However, the lesion continued to spread relentlessly, the edge of the lesion was black and necrotic muscle tissue was seen in the wound []. Meanwhile patient's respiratory distress also worsened. He was put on oxygen support initially, ventilated later for respiratory distress. Patient succumbed to his illness on the 10th day after admission.

[[64.0, 'year']]

M

{'21410101': 1, '8681631': 1, '10621873': 1, '20571164': 1, '8501244': 1, '28270166': 1, '15241098': 1, '12574283': 1, '8916882': 1, '9061588': 1, '17278065': 1, '24696561': 2}

{}

3969645-1

noncomm/PMC003xxxxxx/PMC3969645.xml

Angioleiomyoma of the Forearm

A 37-years-old female presented with a slowly enlarging mass in the right forearm since two years. She occasionally experienced pain and tenderness over the lesion. There was no history of trauma, and the patient was otherwise healthy.\nOn physical examination, an ovoid swelling measuring 1.5 × 1.5 × 1 cm was present on the right forearm. It was subcutaneous in plane and free from underlying bone. The lesion was smooth and firm. There were no skin changes and no signs of infection. There were no other masses and lymphadenopathy.\nAn excisional biopsy was done and sent for histopathological examination. The specimen, measuring 1.5 × 1.5 × 1 cm, was well encapsulated. On cut section it was gray white and solid. On examining the histopathological sections, many slits like vessels interspersed among spindled shaped tumor cells arranged in whorls and interlacing fascicles were revealed. The vessels were surrounded by an inner layer of muscle arranged circumferentially and outer layer blending with the less ordered tumor cells []. Islands showing lipometaplasia were also observed among tumor cells []. There was no evidence of nuclear atypia, mitosis, or necrosis. On immunohistochemistry, these tumor cells were diffusely positivity for Smooth Muscle Actin (SMA) [] and endothelial cells lining the vascular spaces were positive for CD34 []. In view of the above histopathological and immunohistochemical findings, a diagnosis of solid variant of angioleiomyoma was made.\nPost-operative period was uneventful with no evidence of recurrence.

[[37.0, 'year']]

F

{'6707425': 1, '28455461': 1, '27200167': 1, '32567422': 1, '22442524': 2, '6722737': 1, '1446202': 1, '15311559': 1, '7594987': 1, '15064620': 1, '24696034': 2}

{'3304275-1': 1}

3969646-1

noncomm/PMC003xxxxxx/PMC3969646.xml

Subcutaneous Mercury Injection by a Child: A Histopathology Case Report

A 15-year-old male child presented to our tertiary care trauma center with multiple non-healing ulcers on the left forearm since 2 months following trauma by a sharp object []. His general physical examination was unremarkable with stable vital signs. On local examination, there were multiple ulcers, the largest of size 2 × 2 cm over the distal forearm and nodular lesions over the volar aspect of the forearm and lower arm. The nodules were firm and fixed. There was no local lymphadenopathy. His systemic examination was normal. He was under treatment by a surgeon for non-healing ulcer in a peripheral hospital for the last 1.5 months. Discrepancy between history and physical examination prompted surgeons to suspect substance abuse; therefore, a psychiatric referral was performed. This revealed intentional subcutaneous injection of mercury at least thrice, obtained after breaking a thermometer and a sphygmomanometer. The patient was inspired by the movie X-Man Wolverine and wanted to simulate a character called “Mercury.” Interestingly, he had a past history of multiple bites by spiders to simulate Spiderman. Surprisingly, he had no other psychiatric problems and had a normal IQ. Complete biochemical and toxicology evaluations, including elemental mercury level in blood, urine and nails, were performed. X-ray and non-contrast computed tomography scan of the left upper limb was performed, which showed a diffuse area of multiple, subcutaneous radioopaque deposits up to the lower arm []. A Doppler study was performed to rule out any intravascular deposit, but all major vessels were patent, which led to the conclusion that the patient had injected mercury subcutaneously. Mercury was not detected in the serum and nails. However, the concentration of mercury in the urine was 0.139 mg/L (normal range = 0.020 mg/L). The chest X-ray was normal. Excision of the skin and subcutaneous tissue containing granuloma was carried out under X-ray guidance. The skin was left open and a split-thickness skin graft was applied later and the tissue was submitted for biopsy. The surgical wounds healed uneventfully.\nThe pathologic specimen consisted of three tissue pieces measuring 2 × 3 cm, 1 × 1 cm and 1 × 0.5 cm, comprising skin and subcutaneous tissue. The cut-section revealed hemorrhagic and necrotic areas with exuding shiny droplets of mercury. The tissues were processed and microscopic sections were stained with hematoxylin and eosin stain.\nThe microscopy showed epidermis along with dermis showing multiple cystic spaces containing concentric droplets of mercury, which appeared purplish-black in color. Surrounding these spaces were foreign body-type giant cells, histiocytes, lymphocytes and plasma cells [].\nSerum and urinary mercury levels were repeated 1 month after follow-up. The patient is on regular follow-up and there are no signs of systemic absorption until now.

[[15.0, 'year']]

M

{'20808663': 2, '10863229': 1, '9347816': 1, '27012667': 2, '3243178': 1, '3733121': 1, '1761770': 1, '22014404': 1, '4590643': 1, '13588955': 1, '3420455': 1, '3882785': 1, '14669908': 1, '24696563': 2}

{'2923418-1': 1, '4807590-1': 1}

3969647-1

noncomm/PMC003xxxxxx/PMC3969647.xml

Hymenolepis diminuta Infection in a Child from a Rural Area: A Rare Case Report

A 10-year-old female belonging to a rural area of Kendrapada district of Odisha came to our pediatrics outpatient department with a chief complaint of intermittent abdominal pain, anal pruritus and nocturnal restlessness since the last 2-3 weeks. On physical examination, there were no positive clinical findings. No abnormal readings were found in the blood and urine analyses. Her height was 130 cm and weight was 29 kg. Examination of the stool samples revealed spherical eggs 70 µm in diameter, yellow-colored, thick-shelled eggs that contained six central hooklets but no polar filaments []. These were identified as H. diminuta eggs and were differentiated from H. nana eggs, which have a similar appearance but are smaller in size and have two evident polar thickenings, from each of which four to eight polar filaments arise.[] Further confirmation was done by examination of another stool sample by the concentration method. Again, the same findings were noted. The patient was successfully treated with praziquantel (10 mg/kg body weight) for 7 days.

[[10.0, 'year']]

F

{'27473838': 1, '3494805': 1, '30693221': 2, '29157281': 1, '5610041': 1, '34819766': 1, '7444572': 1, '29113595': 1, '32292738': 2, '18417890': 1, '31517102': 1, '9666029': 1, '5173451': 1, '27047007': 1, '25324904': 1, '6537409': 1, '34083936': 1, '27790438': 1, '2703751': 1, '29062858': 1, '12904439': 1, '24696564': 2}

{'6329270-1': 1, '7145915-1': 1}

3969648-1

noncomm/PMC003xxxxxx/PMC3969648.xml

Cytodiagnosis of Epidermoid Cyst of the Upper Lip: A Common Lesion in an Uncommon Site

A 52-year-old male attended FNAC clinic of our hospital with a globular swelling in the upper lip []. The swelling was present for last 5 years and gradually increasing in size. It was not associated with pain. On physical examination, the size of the swelling was approximately 2.5 cm × 1 cm, cystic, non-tender and soft in consistency. No associated cervical lymphadenopathy was detected.\nWith a clinical diagnosis of mucocele, fine needle aspiration was performed using a 22 gauge needle. Aspiration yielded pultaceous material and the smears were stained with May-Grünwald Giemsa stain. Cytological smears showed many anucleated squames and few benign mature squamous cells in a dirty background []. A cytological diagnosis of epidermoid cyst was made with a recommendation for excision of the swelling and histopathological examination.\nExcision biopsy and histopathological examination of the resected specimen showed a cyst lined by stratified squamous epithelium filled with lamellated keratin []. No skin appendages seen. Thus, hispathological diagnosis of epidermoid cyst was confirmed.

[[52.0, 'year']]

M

{'28450777': 1, '2919062': 1, '32827919': 1, '10927691': 1, '6092589': 1, '23798854': 2, '17625630': 1, '3463715': 1, '23248482': 2, '17875211': 2, '15132815': 1, '7735754': 1, '17351686': 1, '3859819': 1, '14578785': 1, '24696565': 2}

{'3687176-1': 1, '3519225-1': 1, '2020471-1': 1}

3969677-1

noncomm/PMC003xxxxxx/PMC3969677.xml

Hypnotherapy: A Useful Adjunctive Therapeutic Modality in Hansen's Disease

A 22-year-old male suffering from Hansen's disease-borderline tuberculoid presented with type 1 reaction. His main symptom was severe neuritic pain of right lower limb and the skin lesions present over the right leg and trunk. The pain was increasing steadily day by day and after a few days, the patient was screaming with pain (visual analog scale [VAS][] rating showed the worst pain-10). This man was a very active karate black belt holder. After knowing the diagnosis, he thought that his nerves are completely damaged by the disease and there was no more hope in future. This thought lead him to severe depression (hospital anxiety and depression scale.\nThe patient was on high dose of systemic steroid (inj. dexamethasone. 2cc once daily for 2 weeks initially and then twice daily for 4 weeks), aspirin (325 mg thrice daily for 2 months), clofazimine (100 mg, initial 1 week, 200 mg next 2 weeks and 100 mg thrice daily for next 5 weeks), and diclofenac sodium (50 mg thrice daily for 2 weeks) daily. As he was not improving, we subjected the patient to hypnotherapy after informed consent. During hypnosis, the patient was asked to visualize his skin, brain, and peripheral nerves according to his mental concept and to imagine that the skin and the nervous system has completely recovered to the pre-diseased state and he is leading a healthy life. After the first session, the patient had a subjective decrease of pain around 70% (VAS scale-3). Two more sessions were given at 3 days interval. The pain markedly decreased after third sitting (VAS-scale-1) and showed excellent improvement psychologically (HADS-4, DLQI-4) and we reduced the drugs very quickly (inj. dexamethasone 2 cc- 1 week, 1 cc- 1 week, tab. Prednisolone 20 mg- 1 week, 15 mg- 1 week, 10 mg 1 week, 5 mg 1 week and stopped. Aspirin 375 mg twice daily-1 week, once daily- 10 days and stopped. Clofazimine 200 mg daily- 2 weeks, 100 mg daily- 2 weeks and stopped and diclofenac sodium was completely stopped) and continued MDT up to 6 months. We followed the patient for 2 years and he had no features of reaction or neuralgia thereafter.

[[22.0, 'year']]

M

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{'3969677-2': 2}

3969677-2

noncomm/PMC003xxxxxx/PMC3969677.xml

Hypnotherapy: A Useful Adjunctive Therapeutic Modality in Hansen's Disease

A 35-year-old female presented with Hansen's disease-lepromatous lepromatous with recurrent ENL lesions. The patient was on MDT for 8 months, systemic corticosteroids (prednisolone 30 mg oral daily for 6 weeks, then inj. dexamethasone 2 ml daily for 4 weeks) and thalidomide (100 mg twice daily for 4 weeks initially followed by thrice daily for 2 months). The course of the reaction was characterized by repeated remissions and exacerbations necessitating five admissions to the hospital during a span of 3 months. On psychological evaluation, she was observed to be depressed (HAD scale-19, DLQI-29). After obtaining informed written consent, we subjected her to hypno analysis and we could realize that the fear of spreading the disease to her family members was the reason for her stress. We asked her to visualize that her body infected with the lepra bacilli and after taking the MDT all the bacteria were killed completely and not even a single bacterium was left behind to infect her family members. After the first session, her fear was markedly decreased (HAD scale-11, DLQI-18). The ENL lesions stared subsiding. We repeated two more sessions at 3 days intervals. Psychologically she became stable (HAD scale-5, DLQI-6) and the ENL lesions subsided completely by 3 weeks. We could reduce the dose of corticosteroids (inj. dexamethasone 1.5 cc for 1 week, 1 cc for 1 week, Tab. Prednisolone 20 mg 1 week, 15 mg 1 week, 10 mg 1 week, 5 mg 1 week and then stopped) and thalidomide (100 mg thrice daily- 1 week, 100 mg twice daily- 1 week, 100 mg once daily - 2 weeks and stopped by 1 month) quickly. We continued the MDT up to 1 year and followed-up the patient for 2 years. She had no more ENL recurrences and admissions thereafter.

[[35.0, 'year']]

F

{'6880820': 1, '8033378': 1, '10769981': 1, '32742673': 1, '32874007': 1, '16844504': 1, '7772485': 1, '20921635': 1, '24700936': 2}

{'3969677-1': 2}

3969679-1

noncomm/PMC003xxxxxx/PMC3969679.xml

Radiation Port Cutaneous Metastases: Reports of Two Patients Whose Recurrent Visceral Cancers Presented as Skin Lesions at the Site of Previous Radiation and Literature Review

A 65-year-old man with a history of squamous cell carcinoma of the anal canal presented in February 2011 with newly formed nodules on his scrotum. The patient had been diagnosed with cancer of the anal canal in September 2010 and was treated with chemotherapy (5-fluorouracil and cisplatin) and concurrent radiation. His radiation therapy was delivered in 29 2 Gy fractions over 38 days for a total of 58 Gy. His treatment was completed on November 4, 2010. During his therapy, he developed severe radiodermatitis which subsequently resolved completely.\nIn January 2011, he discovered several new skin lesions on his scrotum [Figures and ]. Cutaneous examination revealed eight painless nodules on his right scrotum, and two additional nodules on his right inguinal fold. All of the new lesions were restricted to the area within his radiation port. They were mildly pruritic; one had ruptured and extruded foul-smelling white material. Biopsies from both sites showed metastatic squamous cell carcinoma, indicating loco-regional failure of his chemoradiation therapy.\nRestaging chest roentgenogram and computerized axial tomography scan also discovered lung metastases. The patient was sequentially treated with several different antineoplastic agents either as single or combination drug therapy. However, there was not only increased size of previous scrotal nodules but also continued appearance of new cutaneous metastases. He was referred to the Phase 1 department to be considered for a trial of an investigational drug treatment.\nCase 2\nA 52-year-old woman was diagnosed in February 2009 with invasive ductal carcinoma in her left breast and poorly differentiated carcinoma in her right breast. She was subsequently treated with neoadjuvant chemotherapy consisting of four cycles of doxorubicin and cyclophosphamide. Following her initial chemotherapy, she received weekly paclitaxel for 12 weeks and trastuzumab every 3 weeks for 1 year.\nIn October 2009, the patient underwent modified radical mastectomy of the left breast and right lumpectomy with axillary lymph node dissection. She began radiation therapy in December 2009. This included bilateral radiation of the chest wall, internal mammary nodes, supraclavicular fossa, and axillary apex totaling 50 Gy in 25 fractions. In addition, her left infraclavicular fossa received 10 Gray in 5 fractions, and her mastectomy and lumpectomy scars were boosted with an additional 14 Gray in 7 fractions. In February 2010, she was restaged and considered to be free of cancer.\nCutaneous examination in August 2011 revealed macular erythema with pruritic, erythematous macules, superficial plaques, and small papules within the radiation port on the left chest wall [Figures and ]. She also had a nonpruritic, hyperpigmented superficial plaque with surrounding areas of erythema within the radiation port on the right breast [Figures and ]. The skin lesions had appeared 1 month earlier and had not improved with over-the-counter topical preparations applied by the patient. Biopsy of both areas revealed metastatic breast carcinoma.

[[65.0, 'year']]

M

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{'5409821-1': 1}

3969681-1

noncomm/PMC003xxxxxx/PMC3969681.xml

Unusual Presentation of Idiopathic Sweet's Syndrome in a Photodistributed Pattern

A 40-year-old lady with unknown co-morbidities presented with a 10 days history of multiple red raised painful lesions over body. Lesions were initially noted over forearms and in the next 2-3 days, new lesions appeared over arms and back. Patient complained of increased pain and burning sensation in the lesions while working in sunlight. Associated mild to moderate grade fever, intermittent in nature was present for the same duration. There was a history of malaise, pain over large joints, decreased appetite and redness of eyes. No history of oral ulcers, weight loss or any drug intake prior to her symptoms was present.\nGeneral examination revealed that she was febrile with an axillary temperature of 102°F. She also had pallor. Her systemic examination was essentially within normal limits. Dermatological examination revealed erythematous papules coalescing to form plaques with a pseudovesicular appearance over the extensor aspect of forearms and photo-exposed areas on back [Figures and ]. There was a sharp cut-off between the lesions and the photo-protected areas, where there were absolutely no lesions []. The plaques were tender. Conjunctival congestion was also present.\nInvestigations revealed anemia with haemoglobin of 9.5 gms/dl, and neutrophilic leukocytosis with 86% neutrophils out of a total of 13,000/mm3. She had an ESR of 36 mm fall in the first hour by the Westergren's technique. C reactive protein was positive. Antinuclear antibodies ANA and ELISA for HIV were negative. Extensive radiological, biochemical and hematological investigations did not reveal any underlying malignancy or any systemic association. Skin biopsy revealed edema of the papillary dermis along with a heavy neutrophilic infiltrate in the upper dermis. Furthermore, coalescing infiltrate of neutrophils in the lower dermis especially around blood vessels with absence of vasculitis was seen []. There was evidence of endothelial swelling with few fragmented neutrophilic nuclei but no fibrinoid necrosis []. She responded dramatically to a course of oral steroids in the form of Prednisolone 40 mg/day and topical broad spectrum photo-protection. Within seven days, her fever had subsided and within the next two weeks most of her skin lesions regressed. The oral steroids were tapered off in another 4 weeks. There was no recurrence of lesions after three months of follow-up.

[[40.0, 'year']]

F

{'3897327': 1, '26677298': 1, '3300306': 1, '10856659': 1, '14521689': 1, '30930894': 1, '23248368': 2, '14511020': 1, '11493115': 1, '7504506': 1, '9129230': 1, '8621829': 1, '7532508': 1, '32995191': 1, '24700940': 2}

{'3519257-1': 1}

3969682-1

noncomm/PMC003xxxxxx/PMC3969682.xml

Hailey-Hailey Disease Responding to Thalidomide

A 30-year-old unmarried male presented with a history of recurrent painful fluid and pus filled lesions followed by painful erosions over the neck, axillae, trunk and groins of 15 years duration exacerbated over the past 10-15 days. The lesions were mildly pruritic and the discharge was foul-smelling. The lesions aggravated on sun exposure with exacerbation in summer season. He gave family history of similar disease in his father, paternal grandmother and paternal uncles. Before coming to us, he was diagnosed as a case of HHD, which was confirmed histopathologically. He had been treated with dapsone following which he developed agranulocytosis and dapsone was discontinued. Several other treatment modalities including oral and topical antibiotics, corticosteroids were tried but resulted in only partial or temporary improvement. His condition had been steadily deteriorating.\nOn examination, he was febrile. Large macerated plaques studded with foul-smelling pus along with erosions, fissures, and crusts were present on the anterior and the lateral aspect of the neck, axillae, back, lower abdomen, pubic area, gluteal cleft, groins, upper medial thighs and scrotum []. Surrounding skin was friable and erythematous. Nikolsky's sign was positive perilesionally. Few types of erosion with slough were seen on the tongue dorsum and throat. Erosions and crusts were seen on the glans with purulent foul smelling discharge in the prepucial sac, sparing rest of the penis. Nails showed longitudinal ridging. The skin biopsy specimen for histopathological reconfirmation of the diagnosis revealed focal suprabasilar clefting, few acantholytic cells, intercellular edema with dilapidated brick wall appearance which confirmed our diagnosis [Figures and ].\nPatient was started on intravenous (IV) betamethasone 4 mg, 8th h which was tapered off with the introduction of 40 mg of oral prednisolone, IV ceftriaxone 1 mg twice daily and later IV piperacillin-tazobactam based on pus culture and sensitivity reports along with a combination of fusidic acid and betamethasone for topical application.\nHe continued to develop new lesions with the deterioration of his general condition and was febrile throughout. In spite of controlling the secondary infection, the vesicular eruptions persisted. Therefore, he was started on oral thalidomide 100 mg three times a day and a combination of betamethasone and gentamycin for topical application. Marked improvement was seen within a week of starting thalidomide [] and hence we discontinued corticosteroid after rapid tapering of its dose and continued treating him with oral thalidomide 100 mg three times a day for one more week, which was then tapered off in 3-4 weeks and maintained on 50 mg daily. After 6 months of receiving 50 mg thalidomide, the drug was stopped in view of no recurrence of the lesions. He has improved well and comes to us for regular follow-up.

[[30.0, 'year']]

M

{'20169303': 1, '30304341': 1, '16045714': 1, '16710588': 1, '16417242': 1, '27688434': 1, '1554604': 1, '10206049': 1, '20161849': 1, '22293917': 1, '3065260': 1, '24700941': 2}

{}

3969683-1

noncomm/PMC003xxxxxx/PMC3969683.xml

Squamous Cell Carcinoma in a Patient with Vitiligo of Photo-covered Skin

A 65-year-old woman with Fitzpatrick type V skin presented with an asymptomatic keratotic papule in the right knee. She had a single focal vitiligo patch in the right knee for 10 years. There was no family history of vitiligo. As the patch was in a covered area, she did not take any specific treatment for it. She had no history of excessive sun exposure, x-irradiation, or chronic arsenic exposure. The keratotic papule was first noticed 6 months before presentation to us. It grew insidiously to attain the current size in 6 months. Physical examination revealed a round keratotic papule of 1.5 cm in diameter [], with irregular surface and firm to hard consistency. The papule was non-tender with no sign of ulceration. The lesion was not fixed to the underlying tissue. There was no significant regional lymphadenopathy. Routine blood investigations, X-ray of chest and right knee were normal. A provisional diagnosis of SCC was made with seborrheic keratosis and keratoacanthoma as differential diagnosis. Excisional biopsy with 5 mm margin on all sides was performed. Histopathological examination of the specimen revealed hyperplastic epidermis with well-differentiated horny pearls in the dermis [Figure and ]. The diagnosis of SCC was made and the patient was referred to an oncologist, who suggested no further treatment. Follow-up examination done 6 months after the biopsy revealed that the area had healed uneventfully, with no signs of recurrences or new lesions.

[[65.0, 'year']]

F

{'19067714': 1, '12037447': 1, '11712068': 1, '12823440': 1, '31534749': 2, '8689768': 1, '10792246': 1, '8982403': 1, '24700942': 2}

{'6745442-1': 1}

3969684-1

noncomm/PMC003xxxxxx/PMC3969684.xml

Digital Gangrene Associated with Anticentromere Antibodies

A 75 year old lady presented with sudden onset of severe pain and blackish discoloration of the index finger of her left hand. She did not give any history suggestive of Raynaud's phenomenon, or arthritis. In fact she did not have any significant illness in the past, including diabetes, hypertension, or coronary artery disease. She did not have any addictions and she was not on any regular medications. On examination, there was digital necrosis of the distal finger with an adjacent area of pale swollen tissue with ulceration. No sclerodactyly was evident. There was no evidence of peripheral vascular disease. All her peripheral pulses were felt equally on both sides. There was no audible bruit. Her BP was 130/80 mm of Hg. All system examinations were within normal limits. Investigations revealed normal hemoglobin, total leukocyte count, platelet count and ESR. Her blood sugar, renal and liver function tests were within normal limits. ECG, X-ray chest and USG abdomen were also normal. Her ANA titre was elevated and ANA profile showed a strongly positive anticentromere antibody. Anti-double stranded DNA, anti-Sjogren's Syndrome A, anti-Sjogren's Syndrome B and anti-ribonucleoprotein antibodies (anti-SSA, anti-SSB, anti-RNP), anti-Sm, anti-Scl-70 were negative.

[[75.0, 'year']]

F

{'1599523': 1, '33363819': 2, '9291859': 1, '2730983': 1, '8215621': 1, '20569489': 2, '8461923': 1, '16802364': 1, '24700943': 2}

{'2901265-1': 1, '7752590-1': 1, '7752590-2': 1}

3969698-1

noncomm/PMC003xxxxxx/PMC3969698.xml

Alopecia Areata and Vitiligo as Primary Presentations in a Young Male with Human Immunodeficiency Virus

A 26-year-old Chinese male presented with hair loss and depigmented macules. Two years ago, several dispersed hair-loss patches appeared on the patient's scalp without any subjective symptom. Six months ago, several depigmented macules appeared on his face and neck. He previously visited a dermatologist and was diagnosed with alopecia areata as well as vitiligo. However, the treatment was ineffective. The patient, a homosexual man, denied drug abuse and blood transfusion. No member of his family had vitiligo or alopecia.\nDermatological examination revealed several depigmented macules measuring 2 cm to 5 cm with clear boundaries distributed on the face and neck. Several smooth hair-loss patches measuring 1-2 cm were also distributed on the top of the head, and diffuse alopecia was observed around the head [Figure , , and ].\nEnzyme-linked immunosorbent assay and Western blot revealed a positive result for serum anti-HIV antibody. Flow cytometry revealed that the patient's CD4 lymphocyte count and CD4/CD8 ratio were 20 cells/μL (3.7%) and 0.04, respectively, which are considerably low. The thyrotropic-stimulating hormone and free thyroxine levels were 7.69 μIU/mL and 10.06 pmol/L, respectively. Serological test of syphilis showed a positive T. pallidum particle agglutination, but the toluidine red unheated serum test was negative.\nThe patient did not accept any therapy for 1 month until his second visit to the clinic. His alopecia areata worsened, presented ophiasis pattern with 80% hair loss on his head, and even his brows []. The patient's general state of health was still good, and manifestations of opportunistic infection and malignant tumor were not observed.\nOne month after antiretroviral therapy (ART), the patient's alopecia areata dramatically improved, but no evident improvement in his vitiligo was found []. Laboratory results revealed that his CD4 lymphocyte count increased to 40 cells/μL. His CD8 lymphocyte count had a more considerable increase, resulting in a CD4/CD8 ratio of 0.03. Although, his CD4 lymphocyte count was still very low, no other complaints were reported. The patient received benzathine penicillin injection for 3 times, but his vitiligo was not still improved.

[[26.0, 'year']]

M

{'16325065': 1, '14870989': 1, '22512484': 1, '29387768': 1, '30425865': 2, '31885599': 2, '27738425': 1, '8881662': 1, '19532080': 1, '23130209': 2, '21334464': 1, '18064031': 1, '18026114': 1, '2091731': 1, '24700956': 2}

{'6217883-1': 1, '3481795-1': 1, '6927064-1': 1}

3969699-1

noncomm/PMC003xxxxxx/PMC3969699.xml

Syphilitic Hepatitis: An Uncommon Manifestation of a Common Disease

Thirty nine years old male patient presented to medicine out patient department (OPD) with complaints of low grade fever, anorexia, malaise and pain in upper abdomen of one month duration along with dark coloured urine of 10 days duration. Patient gave history of consuming 40-50 grams of alcohol daily for past 3 years. There was no history of drug abuse or blood transfusion.\nExamination disclosed presence of icterus with normal vital signs. Per abdomen examination revealed 2 cm, firm, non tender hepatomegaly without splenomegaly or ascites.\nBiochemical analysis revealed increased values of serum bilirubin 3.5 mg/dl (normal 0.0-1.5 mg/dl), serum alanine transaminase (ALT) 357 IU/ml (normal < 50), serum aspartate transaminase (AST) 175 IU/ml (normal < 45) and serum alkaline phosphatase 536 IU/ml (normal 20-135). Screening for Hepatitis A, B, C and E were negative. Ultrasound abdomen revealed hepatomegaly with liver span of 16.4 cm with normal echotexture. Magnetic resonance cholangio pancreatography (MRCP) confirmed hepatomegaly with no space occupying lesion, lymphadenopathy or extra hepatic cholestasis.\nThe patient was thought to have acute hepatitis probably due to alcohol or non-hepatotropic viruses. Since the patient was showing clinical recovery with regression in serum bilirubin (1 mg/dl), AST (90 IU/ml) and ALT (152 IU/ml) though with rising alkaline phosphatase (711 IU/ml), Patient was asked to review after 1 month with the plan of performing liver biopsy after the resolution of acute phase.\nOn reporting back he presented to dermatology OPD with intensely itchy rash. Examination revealed multiple erythematous papules and plaques on neck, trunk, palms and soles [Figures –]. Buschke Olendroff sign was positive. There was bilateral, non tender, shotty inguinal lymphadenopathy, congestion of soft palate and posterior pharyngeal wall. Genital, per rectal, ophthalmic and auditory examination was normal.\nPatient gave history of repeated unprotected heterosexual intercourse with multiple partners during past two years. There were no symptoms suggestive of any sexually transmitted disease.\nA provisional diagnosis of secondary syphilis with syphilitic hepatitis was made. VDRL titre was 1:16 and TPHA was positive. Liver biopsy was suggestive of syphilitic hepatitis showing maintained hepatic architecture with Kupffer cell hyperplasia, lymphocytic and neutrophillic infiltration of portal tracts with mild neutrophillic infiltrate in hepatic lobules and no necrosis, granuloma, gumma or Treponema pallidum. Reticulin stain revealed early fibrosis around the portal tracts []. ELISA for HIV was negative. Positron emission tomography (PET) scan revealed hepatomegaly and presence of retroperitoneal, hilar, mediastinal, bilateral axillary, inguinal and external iliac lymph nodes. Patient was treated with 3 doses of intramuscular Benzathine Penicillin 2.4 million units, given weekly. He became asymptomatic with resolution of pruritus and mucocutaneous findings. There was normalization of serum bilirubin, ALT, AST values and reduction in alkaline phosphatase (383 IU/ml).

[[39.0, 'year']]

M

{'33868613': 2, '18275654': 1, '30271743': 1, '15546070': 1, '30425865': 2, '29301812': 1, '3578236': 1, '31885599': 2, '28469107': 1, '53374': 1, '7123342': 1, '426583': 1, '7377174': 1, '24700957': 2}

{'6217883-1': 1, '8035539-1': 1, '6927064-1': 1}

3969700-1

noncomm/PMC003xxxxxx/PMC3969700.xml

Localized Pemphigus Vegetans without Mucosal Involvement

A 62-year-old healthy woman presented to our dermatology out-patient department with single painful red colored elevated lesion covered with pustules on the left breast for 6 months. There was no history suggestive of any systemic involvement. There was no history of fever, any drug intake, eye or mucosal complaints, no past or family history of similar illness, no history of any topical application. On examination, a single well-defined polycyclic erythematous plaque of size 6 × 7 cm with pustules concentrated mainly at the periphery of the plaque was present on left breast. KOH mount for fungus was negative, but based on clinical appearance probable diagnosis of tinea corporis was kept, and she was started on oral Fluconazole 150 mg once a week and terbinafine cream local application twice daily for 2 weeks. On subsequent follow-up after 2 weeks, there was no improvement, but the lesion in the left breast had progressed and involved the entire left breast to form a single well-defined moist, verrucous vegetating plaque of size 8×10 cm with multiple pustules, yellow to brown colored crusts at places, and some raw oozy areas. Plaque was surrounded by a broad rim of erythema []. She also developed similar verrucous lesion in the left groin on subsequent 2 weeks follow-up []. Rest of the mucocutaneous examination was within normal limits. Nikolsky's sign was negative and Tzanck smear showed eosonophils, but no acantholytic cells. Gram stain for bacteria showed only mixed inflammatory infiltrate with eosinophil predominance and acid fast bacteria staining was negative. Pus culture from the pustule was sterile. Routine laboratory studies including complete blood count, C reactive protein, erythrocyte sedimentation rate, liver and renal function tests, and chest X-ray were all within normal limits. Human immunodeficiency virus serology was negative. There was no evidence of inflammatory bowel disease. Biopsy specimens were obtained from the right breast with the differential diagnosis of pemphigus vegetans, pyoderma gangrenosum.\nPatient was started on oral prednisolone 40 mg once daily with which she showed dramatic response within a week. Plaque healed completely with hyperpigmentation at 2 weeks [Figures and ].\nHistopathological examination [Figures –] showed suprabasal cleft with acantholytic cells, numerous eosinophils, intraepidermal eosinophilic abscesses, and dense inflammatory infiltrate in the dermis. As the biopsy was taken from an early pustule, that might be an explanation for the absence of hyperkeratosis and papillomatosis in the present case. Direct immunofluorescence (DIF) revealed intercellular Immunoglobulin G (IgG) deposits in the epidermis [].

[[62.0, 'year']]

F

{'6365813': 1, '34630689': 1, '27057064': 1, '29560011': 2, '17951135': 1, '32055517': 1, '9810918': 1, '31922111': 2, '9810919': 1, '2249360': 1, '2753595': 1, '15762223': 1, '24700958': 2}

{'5821983-1': 1, '6896381-1': 1}

3969702-1

noncomm/PMC003xxxxxx/PMC3969702.xml

Rare case of phenytoin induced acute generalized exanthematous pustulosis with cerebellar syndrome

An 18-year-old male was admitted to infectious disease ward for fever and generalized erythematous rash since one day. A diagnosis of viral exanthem was made. On detailed history taking patient was found to be a known case of seizure disorder on regular levetiracetam since three years. Patient gave past history of an allergic rash to phenytoin three years ago. Patient had received loading intravenous dose of phenytoin (1000 mg) one day prior to admission. On examination patient was febrile and was having facial edema [] with predominant involvement of lips. There was total sparing of oral mucosa. Patient had generalized erythematous macular rash along with multiple non follicular pustules more pronounced on chest [] and back [] but also involved the axilla [] and neck. There was no involvement of palm and soles. In addition, patient had cerebellar signs in the form of horizontal, up beating and down beating nystagmus along with bilateral swaying while walking suggestive of signs of acute phenytoin toxicity. Rest of general and systemic examination was unremarkable. In view of clinical presentation and history, diagnosis of acute drug reaction was kept and dermatologist opinion was taken. A provisional diagnosis of acute generalized exanthematouspustulosis was made and skin biopsy and gram stain of the pustule was done. His investigations revealed leucocytosis (TLC-11,500) and deranged kidney function test (Urea-55 mg/dl; Creatinine-2.2 mg/dl) with normal liver function test. Skin biopsy revealed spongiosis with dermal neutrophilic and eosinophilic infiltrate [] which are features of acute generalized exanthematous pustulosis. Gram staining of smear from the pustule showed no organism. Patient was treated with tapering doses 40 mg of oral corticosteroids along with antihistaminics. Patient responded dramatically with total subsidence of rash with desquamation [Figures and ]. Facial edema subsided completely. His cerebellar signs disappeared and kidney function test was normal within 15 days and patient was discharged with no sequelae.

[[18.0, 'year']]

M

{'1832534': 1, '20227592': 1, '27472323': 1, '19261012': 1, '21965865': 1, '11168761': 1, '22707770': 1, '6989310': 1, '20542841': 1, '24700960': 2}

{}

3969703-1

noncomm/PMC003xxxxxx/PMC3969703.xml

Tumid Lupus Erythematosus: An Intriguing Dermatopathological Connotation Treated Successfully with Topical Tacrolimus and Hydroxyxhloroquine Combination

A 27-year-old married woman, mother of two, accessed our dermatology outpatient department with an asymptomatic pink-red plaque over the right cheek for the last 5 months. She denied photosensitivity, oral ulcerations, fever, and joint pains. Rest of the systemic examination was insignificant. Physical examination revealed single erythematous, indurated, 3 cm-sized plaque over the right cheek []. Regional lymph nodes were not enlarged. Rest of the muco-cutaneous and systemic examination was normal. A clinical differential diagnosis of tumid lupus erythematosus (tumid LE), Jessner's lymphocytic infiltrate, and pseudolymphoma was made. Microscopic examination of the sections from the lesion revealed slightly thinned epidermis, subepidermal edema, superficial and deep, largely perivascular and some periappendageal lymphocytic infiltrate []. Dermo-epidermal junction was normal. Increased amounts of mucin were identified on staining with Alcian blue. The biopsy specimen from the lesion was subjected to direct immunofluorescence, which showed moderate IgG, and weak IgA, IgM, C3, and fibrin deposits along the basement membrane zone in a linear fashion []. Facility for CD4 and CD8 staining was not available in our institution. Further instrumental investigations including anti-nuclear antibody (ANA) titers (by indirect immunofluorescence on Hep-2 cell line), complete blood counts, liver and renal functions, and blood sugar levels were normal. LE cell test was not done. Urine routine microscopy and 24-hour urine protein were normal too. In addition, imaging studies including X-ray chest and an ultrasound of abdomen were unremarkable. Accordingly, a diagnosis of tumid lupus erythematosus was entertained. After obtaining an ophthalmology clearance, the patient was administered hydroxychloroquine sulphate in a dose of 200 mg daily and was asked to take adequate photoprotection including broad-spectrum sunscreen. The lesion had regressed after a period of 4 months. However, a month later, she had a flare up in the lesion while on hydroxychloroquine and despite adequate photoprotection. Considering the potential side effects of potent topical corticosteroids on face and the very fact that topical tacrolimus has been shown to be effective in non-hyperkeratotic LE lesions, it was decided to add topical tacrolimus 0.1% ointment to HCQS. The lesion resolved completely over a period of 3 weeks []. Subsequently, HCQS was stopped, and she is now on oral antioxidants and photoprotection. The patient is on follow-up for 4 months post-treatment and has not shown recurrence.

[[27.0, 'year']]

F

{'18083273': 1, '34671535': 2, '32083062': 2, '9404852': 1, '16700782': 1, '11251561': 1, '19712116': 1, '12789183': 1, '12910555': 1, '21957901': 1, '10926740': 1, '24700961': 2}

{'7011746-1': 1, '8520756-1': 1}

3970270-1

noncomm/PMC003xxxxxx/PMC3970270.xml

Pheochromocytoma with Markedly Abnormal Liver Function Tests and Severe Leukocytosis

A 49-year-old male presented to our hospital complaining of chest pain. For the past few years the patient reported experiencing intermittent chest pains at night. In addition to the chest pain, the patient developed pyrexia and perspiration beginning a week before admission. He initially visited another hospital for worsening symptoms where examination found abnormal electrocardiography results (the ST elevation in leads aVL and V1 to V6), leukocytosis, and abnormal liver function tests. Echocardiography revealed a decreased cardiac output with regional wall motion abnormality. Abdominal computed tomography (CT) results showed hepatic parenchymal abnormality and a tumor on the left adrenal gland. The patient was transferred to the reporting hospital with these results.\nSeven years prior to presentation the patient had been diagnosed with hypertension and was put on antihypertensive medication, but remained poorly controlled over the 3 years prior to presentation. He was also diagnosed with diabetes mellitus 8 years ago and has been treated with hypoglycemic agents, but his blood sugar was poorly controlled. There was no other medical history.\nOn admission, his blood pressure was 140/90 mm Hg with a pulse rate of 66 beats per minute, was breathing at a rate of 20 times per minute, and had a body temperature of 36.8℃. He had clear consciousness without signs of conjunctival pallor, scleral jaundice, dehydration, and absent dilatation of the jugular vein. On chest auscultation breath sounds were clear without wheezing or crackles detected. Cardiac auscultation revealed a heartbeat that was regular without murmur. On examining the abdomen, bowel sounds seemed reduced and the right upper abdomen had slight but not significant tenderness. No masses were detected on abdomen. No limb edema was seen.\nTwo days prior to admission, the patient had been tested in another hospital. Blood tests resulted as follows: aspartate aminotransferase (AST) 444 IU/L, alanine aminotransferase (ALT) 469 IU/L, alkaline phosphatase (ALP) 1,153 IU/L, white blood cell count 50,000/µL, hepatitis B surface antigen (HBsAg) negative, anti-HBsAb positive, antihepatitis C virus (anti-HCV) antibody (Ab) negative, antihepatitis A virus (anti-HAV) immunoglobulin M (IgM) Ab negative, and anti-HAV IgG Ab positive. On the day of admission to our hospital, the blood test results were as follows: hemoglobin 15.8 g/dL, white blood cell count 59,360/µL, platelet count 244,000/µL, erythrocyte sedimentation rate 4 mm/hr, and C-reactive protein 3.446 mg/L, blood urea nitrogen 22.7 mg/dL, serum creatinine 0.76 mg/dL, serum calcium 7.2 mg/dL, inorganic phosphorous 2.0 mg/dL, sodium 134 mmol/L, and potassium 4.4 mmol/L. The results also showed that total protein was 5.5 g/dL, albumin was 2.7 g/dL, AST 156 IU/L and ALT 317 IU/L, ALP 289 IU/L, and total bilirubin 1.05 mg/dL. Hemoglobin A1c was 10.7%. At fasting, C-peptide was 0.40 ng/mL (range, 0.48 to 3.3), and 2 hours after a meal it was 0.66 ng/mL. Lactate dehydrogenase was 1,240 IU/L. Creatine kinase MB and troponin-T were found to be 5.99 ng/mL (range, 0.6 to 6.3) and 0.073 ng/mL (range, 0.0 to 0.1), respectively.\nChest X-ray found no abnormal finding. Abdominal CT at another hospital detected several small-sized and low attenuation lesions on both lobes of the liver, and a 5×5-cm-sized tumor enhanced unevenly on the left adrenal gland ().\nElectrocardiography revealed sinus rhythm with T wave inversion and QT interval prolongation observed in leads II and V1 to V6 (). Although the cardiac ejection fraction was 69%, echocardiography detected severely hypokinetic wall motion of mid-left ventricle sparing basal and apical wall motions.\nThe tumor on the left adrenal gland was tested for hormones. The results of 24 hours urinalysis were as follows: vanillylmandelic acid (VMA) 33.93 mg/day (range, 0 to 8.0), metanephrines 20.45 mg/day (range, 0 to 1.3), epinephrine 2,034 µg/day (range, 0 to 40), norepinephrine 978 µg/day (range, 0 to 80), serum epinephrine 1,221 pg/mL (range, 0 to 120), and serum norepinephrine 778 pg/mL (range, 100 to 410). Other tests including aldosterone, plasma renin activity, and cortisol were all normal. On 131I-metaiodobenzylguanidine scan examination, uptake of the radioactive isotope was increased in the left adrenal gland, but no other lesions were found ().\nBased on the examination results above, the patient was diagnosed with a pheochromocytoma on the left adrenal gland. On the 12th day of admission oral phenoxybenzamine administration was started, and blood pressure was followed after having ceased all previous antihypertensives. The dosage of phenoxybenzamine was increased to 35 mg/day. After 19 days of administration, resection of the left adrenal gland was performed under laparoscope. The size of the resected tumor was 5×5 cm, and the surface was a mixture of a hemorrhagic and a cystic lesion. The tissue pathological examination showed the tumor cells with eosinophillic cytoplasm and polygonal shape surrounded by mesenchyma of vasculature on H&E staining, while immunohistochemical stains of chromogranin, synaptophysin, and S-100 immunoperoxidase staining revealed the proper findings to diagnose pheochromocytoma as positive (). Thirteen days after tumor resection a panel of endocrine tests was performed to follow-up on the pheochromocytoma. A 24 hours urinalysis showed a VMA of 6.51 mg/day with metanephrines at 0.61 mg/day, epinephrine at <5 µg/day, norepinephrine at 101.48 µg/day, serum epinephrine at <15 pg/mL, and serum norepinephrine at 536.60 pg/mL (). When first admitted, the patient had uncontrolled blood pressure even with amlodipine 10 mg and irbesartan 150 mg plus 12.5 mg of hydrochlorothiazide. However, when discharged, the patient had controlled blood pressure with a reduced dosage of antihypertensives consisting of amlodipine 10 mg and irbesartan 150 mg.\nPrior to his admission to the outside hospital, the patient had no history of new drug administration that might cause liver function failure. He also had no history of drinking and had a normal body weight (body mass index 23 kg/m2). A panel for hepatic viral disease (hepatitis B virus, HCV, HAV) was negative, and no abnormality was found through immunological testing (antismooth muscle, antinuclear, antimitochondria antibody), or screening for genetic diseases (plasma iron parameters, ceruloplasmin, Cu). This led to the challenge of finding a clear cause for the liver function failure. The liver function improved with conservative treatment, and had resolved by the time phenoxybenzamine was administered (). On the 22nd day after left adrenal gland resection, AST and ALT had normalized at 23 and 25 IU/L, respectively (), and normal liver function test remained within normal limits at 80 days after surgery.\nAfter admission to our hospital, the patient's leukocytosis continued, and follow-up tests were performed to examine for secondary causes such as blood diseases, infection, and pheochromocytoma. On the fourth day of phenoxybenzamine administration the white blood cell count had recovered to 5,900/µL and continued to be within the normal range even after surgery ().\nEchocardiography indicated cardiac ejection fraction of 78% on the 15th day after admission (fourth day of phenoxybenzamine administration), and no left ventricle wall motion abnormalities were observed.\nOn admission the patient was administered insulin and metformin, but the range of blood glucose fluctuation was wide. However, after surgery blood glucose levels returned to the normal range without the previously administered medications. Previously suppressed C-peptide secretion was recovered 3 days after surgery. On the 80th day after surgery, the hemoglobin A1c was measured found to be 5.7% ().

[[49.0, 'year']]

M

{'6243675': 1, '12766539': 1, '16112304': 1, '7859594': 1, '11041903': 1, '25827457': 1, '16600345': 1, '18094670': 1, '12915647': 1, '13701725': 1, '14557417': 1, '34430009': 1, '6303636': 1, '29923996': 1, '11903030': 1, '17635948': 1, '8811932': 1, '15863947': 1, '7607842': 1, '3348424': 1, '11113629': 1, '24741459': 2}

{}

3970271-1

noncomm/PMC003xxxxxx/PMC3970271.xml

Hypoparathyroidism and Subclinical Hypothyroidism with Secondary Hemochromatosis

A 29-year-old female admitted to our hospital for evaluation of asymptomatic hypocalcemia. Fifteen years ago she was diagnosed with aplastic anemia, treated with antilymphocyte globulin and antithymocyte globulin, and had routine transfusions of RBCs of 2 units per week (about 1,400 units over 15 years). In addition, starting 10 years ago, selective iron-chelating agent (deferasirox 1,500 mg/day) was administered due to increased serum ferritin levels. Continued hypocalcaemia was detected in blood tests at regular follow-up visits and the patient visited the endocrinologic department as an outpatient for additional examinations and treatment. She has no histories of cervical irradiation, thyroid disease or related family history. She had a blood pressure of 120/80 mm Hg, pulse rate of 78 beats per minute, temperature of 36.8℃, and a respiratory rate of 20 breaths per minute. Her skin was unusually dark in color with conjunctiva pallor. Chest examination showed that breathing was clear, heartbeat was regular and no unusual sounds were detected. Her abdomen was flat and soft with no unusual sounds, and the liver and spleen were not palpated. Neither leg had pitting edema. Neurological tests showed no local neurological symptoms, Chvostek sign or Trousseau sign.\nIn the laboratory findings, peripheral blood test indicated a white blood cell count of 700/mm3 (neutrophil 150), hemoglobin level of 8.0 g/dL, and platelet count of 37,000/mm3. A serum biochemical test reported blood urea nitrogen levels of 10.1 mg/dL, serum creatinine of 0.7 mg/dL, albumin levels of 3.9 g/dL, aspartate aminotransferase levels of 53 IU/L, alanine aminotransferase levels of 60 IU/L, alkaline phosphatase of 94 IU/L, and total bilirubin of 0.5 mg/dL. In a serum electrolyte test, sodium level was 140 mEq/L and potassium level was 4.3 mEq/L, both of which are in the normal range. However, total calcium was 6.7 mEq/dL (albumin corrected calcium 6.78 mEq/dL; normal range, 8.4 to 10.2), and ionized calcium was 1.78 mEq/L (normal range, 2.2 to 2.6). Serum ferritin levels were 27,583.03 ng/mL (normal range, 4.63 to 274.6), iron levels were 291 µg/dL (normal range, 65 to 157), and total iron binding capacity was 389 µg/dL (normal range, 256 to 426) which showed an increase. The transferrin saturation level was high to 74.8% (normal range, 22% to 46%).\nShe was diagnosed with hypocalcaemia induced by hypoparathyroidism based on test results showing intact parathyroid hormone (iPTH) levels of 31 pg/mL (normal range, 9 to 55), 25(OH)VitD of 7.03 ng/mL (normal range, 4.8 to 52.8), and 1,25(OH)2VitD levels of 36.2 pg/mL (normal range, 25.1 to 66.1). Although 1,000 mg of calcium carbonate and 0.5 µg of alfacalcidol were given twice a day, hypocalcaemia continued with increased levels of 24 hours urine calcium (700 mg/day) and 24 hours urine creatinine (0.99 g/day); thiazide 12.5 mg was also administered to maintain the total calcium at 7.5 to 8.0 mEq/dL and ionized calcium at 1.9 to 2.2 mEq/L. Tests were performed to determine if the hemochromatosis had spread to other organs; chest X-rays showed no signs of cardiomegaly, but computed tomography (CT) before contrast enhancement showed thyroid (Hounsfield unit 157.0) and liver (Hounsfield unit 136.0), as well as a increased signal intensity associated with the cardiac muscle and enlarged inferior vena cava and hepatic veins (). The T2-weighted magnetic resonance imaging (MRI) of the abdomen revealed a dark signal intensity in the liver, spleen, bone marrow, and pancreas, leading to the secondary diagnosis of hemochromatosis (). Organs showing signs of iron impregnation were tested for function; the thyroid function test showed elevated levels of thyroid stimulating hormone (TSH, 17.18 µIU/mL; normal range, 0.4 to 4.8) with free thyroxine levels of 1.28 ng/dL (normal range, 0.8 to 1.71), and triiodothyronine levels of 1.43 ng/mL (normal range, 0.6 to 1.6). Thyroid peroxidase antibody and thyroglobulin antibody were both negative which resulted in apparent hypothyroidism caused by hemochromatosis. The patient was receiving low-dose of thyroid hormone (levothyroxine 50 µg/day), but her TSH levels were still over 10 µIU/mL at follow-up. Therefore the dose of the hormone was gradually increased. The most recent test result showed TSH levels of 8.12 µIU/mL, and the dose of thyroid hormone was increased to 150 µg/day for follow-up observation. Echocardiography revealed dilated left ventricular chamber size (left ventricular end diastolic diameter 54 mm) with global hypokinesia, and a moderate to severe left ventricular systolic dysfunction (ejection fraction 23%), which suggests congestive heart failure with dilated cardiomyopathy and restrictive diastolic dysfunction induced by hemochromatosis. The patient is medicated with digoxin 0.1875 mg, candesartan 8 mg, bisoprolol 2.5 mg, and spiractone 25 mg without any significant symptoms and is on follow-up observation as an outpatient.

[[29.0, 'year']]

F

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{'7261529-1': 1, '7261529-2': 1}

3970275-1

noncomm/PMC003xxxxxx/PMC3970275.xml

Medullary Thyroid Carcinoma with Ectopic Adrenocorticotropic Hormone Syndrome

A 48-year-old male was referred to the endocrinology clinic complaining of generalized edema, increased blood pressure and blood glucose levels. The patient was on medication due to hypertension that was diagnosed 1 year ago. Recently, the blood pressure was not well-controlled, the blood glucose level was high and generalized edema developed.\nThe patient was diagnosed with MTC 14 years ago; total thyroidectomy with right cervical lymph node dissection was performed and I-131 metaiodobenzylguanidine (MIBG) 200 mCi was administered. He was treated with tumor resection for recurred MTC on a mediastinal lymph node 13 years ago. Nine years ago, MTC recurred on the left cervical and left parahilar lymph nodes and was treated with I-131 MIBG 200 mCi after surgery. Two years ago, MTC recurred thirdly on the cervical and mediastinal lymph nodes and lung and was treated with cervical and mediastinal lymph node dissection and the left upper lobe wedge resection of the lung. Since then, the patient has not shown any signs or symptoms of recurrence.\nThe patient's height was 172 cm and weight was 84 kg, 2 kg over his usual weight. His blood pressure was 185/110 mm Hg with a pulse rate of 90 beats per minute and a breathing rate of 18 breaths per minute. The patient's body temperature was 37℃. Moon face and facial flushing were observed. Buffalo hump and purple striae were absent. Mild pitting edema was observed on the anterior tibia and slight skin pigmentation was present.\nPeripheral blood examination showed a white blood cell count of 14,450 cells/µL, hemoglobin of 15.3 g/dL, and platelet count of 358,000 cells/µL. On serum biochemical examination, total cholesterol was 252 mg/dL with total protein of 6.5 g/dL, albumin of 4.1 g/dL, total bilirubin of 1.1 mg/dL, alkaline phosphatase of 101 U/L, aspartate aminotransferase of 17 U/L, alanine aminotransferase of 62 U/L, blood urea nitrogen of 10.3 mg/dL, and creatinine of 0.6 mg/dL. On serum electrolyte examination, sodium was 145 mmol/L with potassium of 2.8 mmol/L, chloride of 94 mmol/L, and bicarbonate of 40.4 mmol/L. Fasting plasma glucose level was 168 mg/dL with glycated hemoglobin of 8.6%. Hormone examination showed total T4 of 12.75 µg/dL (range, 5 to 13) and thyroid stimulating hormone of 0.07 µIU/mL (range, 0.15 to 5.0). Serum calcitonin was 127.86 pg/mL (<10.0) and carcinoembryonic antigen was 29.46 ng/mL (<5). Plasma ACTH concentration was measured with immunoradiometric assay (ELSA-ACTH kit, CisBio, Belford, MA, USA), and was 203.9 pg/mL (range, 6.0 to 60.0). Serum cortisol concentration was 36.0 µg/dL (range, 5 to 25.0) with a 24-hour urine cortisol of 2,237.1 µg/day (range, 20 to 90). On the low dose dexamethasone suppression test, inhibited serum cortisol was 32.50 µg/dL. The 24-hour urine examination results were as follows: metanephrine 14.46 µg/day (range, 52 to 341), normetanephrine 28.21 µg/day (range, 88 to 444), epinephrine 6.57 µg/day (<40), norepinephrine 26.43 µg/day (<80), and vanillylmandelic acid 3.51 mg/day (<8). To validate the RET gene mutation, peripheral blood was collected from the patient and white blood cells were separated by centrifugation to extract genomic DNA. For polymerase chain reaction, BIGDYE version 3.1 cycles sequencing kit (Applied Biosystems, Foster, CA, USA) reagent was utilized and 3,730 automated sequencer (Applied Biosystems) was used to analyze DNA sequence. The DNA sequencing showed no RET gene mutation.\nChest and abdomen computed tomography revealed recurred lesions on the mediastinal lymph nodes, lungs, and liver (), but no abnormal findings in adrenal glands (). Sellar magnetic resonance imaging showed no abnormality in the pituitary gland (). Gastroscopy, colonoscopy, and positron emission tomography did not identify suspicious primary lesions in other regions.\nCalcitonin and chromogranin A staining was positive on resected tissues on the third recurrence 2 years ago, which is indicative of MTC. ACTH immunohistochemistry staining was negative ().\nBecause curative resection of the MTC lesions was difficult on the fourth recurrence, close follow-up with further chemotherapy was considered. To control the ectopic ACTH syndrome, the patient was administered ketoconazole. The dosage was gradually increased over 3 months up to the maximum dosage (1,200 mg/day), but the serum cortisol concentration level did not decrease. Therefore, bilateral adrenalectomy was performed. After surgery, ACTH remained above 200 pg/mL, but plasma glucose and blood pressure levels were improved sufficiently and the oral antidiabetic agent and antihypertensive medication were discontinued. The patient has been living in good condition until 6 months after the surgery without any medication other than glucocorticoid and mineralocorticoid agents.

[[48.0, 'year']]

M

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{'1920524-1': 1}

3970286-1

noncomm/PMC003xxxxxx/PMC3970286.xml

Hyponatremia after Thyroid Hormone Withdrawal in a Patient with Papillary Thyroid Carcinoma

A 76-year-old female visited the emergency room with symptoms of communication disability, mumbling, and impatience. She was treated with total thyroidectomy and radioiodine therapy for papillary thyroid carcinoma in October 2001. In April 2011, she was treated with cervical lymphadenectomy for left cervical lymph node metastasis of papillary thyroid carcinoma, radical nephrectomy for left renal tubular carcinoma, and finally radioiodine therapy in July 2011. She was initially administered levothyroxine 0.1 mg daily and in April 2012 blood examination showed a high level of thyroglobulin at 14.67 ng/mL. The thyroid hormone was replaced by liothyronin sodium, and after 2 weeks of complete thyroid hormone withdrawal an iodine whole-body scan was conducted 5 days before admission. The level of free T4 was decreased to 0.25 ng/dL (normal range, 0.93 to 1.7) and thyrotropin (TSH) was increased to 106 µIU/mL (normal range, 0.27 to 4.2). After examination, levothyroxine 0.1 mg daily was readministered. Because cervical node metastasis was suspected, a fine needle aspiration test was reserved before release. Dizziness and weakness were present before the iodine whole body scan, but no particular treatment was given due to its low level of severity. However, 2 days after the iodine whole body scan and 3 days before admission, she visited another hospital for pain in her heels where drugs including nonsteroidal anti-inflammatory were given twice. Her dizziness and nausea worsened after which she reported vomiting that occurred more than 10 times a day. A day before admission, the patient visited the emergency room and was examined for a suspicious gastrointestinal tract reaction induced by nonsteroidal anti-inflammatory drugs. She was administered antiemetic drugs, and sent home with a serum sodium level of 121 mEq/L. After getting home, her nausea continued despite the antiemetics and she was hospitalized in the same night from the emergency room with symptoms of communication disability, mumbling, and impatience.\nThe patient was diagnosed with diabetes and hypertension about 20 years ago and was on medication including oral hypoglycemic agents (pioglitazone 15 mg daily, metformin 250 mg daily) and oral antihypertensive drugs (perindopril tert-butylamine 8 mg daily, amlodipine besylate 5 mg daily). In 1999, she underwent hip bipolar hemiarthroplasty and right total knee arthroplasty (TKA) and, in 2000, left TKA.\nAt admission, she presented with signs of delirium and disorientation and her vital signs were as follows: blood pressure 160/80 mm Hg, pulse rate 66 times per minute, breathing rate 20 times per minute, body temperature 36.8℃, and oxygen saturation on room air was 95%. There were no reported changes in body weight.\nSkin tension was not decreased, there was no conjunctival pallor or sclera jaundice, and the papillary reflex was normal. Cervical lymphadenopathy was not diagnosed, and both cardiac and breathing sounds were normal on chest examination. On abdominal examination, bowel sound was normal, and there were no masses or pain. There was no limb edema on examination of the peripheral extremities.\nPeripheral blood examination showed a white blood cell count, hemoglobin concentration, and platelet count as 7,700/mm3, 9.7 g/dL, and 144,000/mm3, respectively. Electrolyte tests showed a sodium of 110 mEq/L, a potassium of 3.9 mEq/L, and a chloride of 82 mEq/L which suggested hyponatremia. Serum osmolality was 233 mOsm/kg and a urine osmolality was 418 mOsm/kg, indicating a severe hypotonic hyponatremia. Biochemical blood tests showed a blood glucose of 133 mg/dL, a blood urea nitrogen of 11 mg/dL, a creatinine of 1.2 mg/dL, a total protein of 6.7 g/dL, albumin of 4.0 g/dL, total bilirubin of 0.71 mg/dL, aspartate aminotransferase 61 IU/L, alanine aminotransferase 25 IU/L, alkaline phosphatase 175 IU/L, r-guanosine triphosphate 11 IU/L, creatine kinase 1,380 IU/L, lactic dehydrogenase 779 IU/L, and total cholesterol was 206 mg/dL. Serum C-reactive protein, PT, and aPTT was 0.1 mg/dL, 11.5 seconds (INR 1.06), 28.1 seconds, respectively.\nHepatitis testing was hepatitis B surface antigen negative, antihepatitis B surface antibody negative, and antihepatitis C virus negative. Chemical urinalysis showed sodium, potassium, chloride, and creatinine to be 50, 41.6, 58.1 mEq/L, and 63.65 mg/dL respectively. Electrocardiogram showed normal sinus rhythm (66 times per minute).\nA hormone test was conducted to identify the cause of hypoosmolar hyponatremia, and the result showed that the TSH was 90.78 µIU/mL with a free T4 of 0.61 ng/dL, an ACTH of 33.3 pg/mL, and a cortisol of 341.84 ng/mL ().\nBrain magnetic resonance imaging and computed tomographic features showed no signs of acute lesions, and chest and abdominal X-ray, and echocardiography results gave no significant diagnosis.\nThe patient showed signs of severe hyponatremia with continuous nausea and disorientation and had a serum sodium level of 110 mEq/L, so 3% normal saline was injected. On the second day after admission, levothyroxine 0.2 mg was administered for 3 days at 6 hours intervals, then reduced to 0.1 mg every 12 hours for 8 days, and finally maintained at 0.1 mg every 24 hours. On the third day of admission, consciousness and nausea had improved. The electrolyte levels gradually improved, and normalized by the 12th day. On the fifth day of admission, external tube feeding began. On the sixth day, full consciousness returned enabling oral feeding. On the 13th day, after the volume of food had increased and the serum sodium level normalized, fluid infusion was stopped, and the serum sodium level was maintained without fluid infusion.

[[76.0, 'year']]

F

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{}

3970306-1

noncomm/PMC003xxxxxx/PMC3970306.xml

Mycotic Abdominal Aortic Aneurysm Caused by Bacteroides Thetaiotaomicron and Acinetobacter Lwoffii: The First Case in Korea

A 60-year-old man was admitted to a hospital because of a 10-day history of lower abdominal pain and fever. His past medical history included diabetes mellitus, which was poorly controlled (HbA1c level of 11.2%). He had no history of recent infection, acupuncture, or intravenous injection.\nOn admission, he was febrile and complained of lower abdominal pain, but no abdominal tenderness or rebound tenderness was elicited. Other meaningful symptoms and signs suggesting metastatic complications (endophthalmitis, central nervous system infection, etc.) were not present. His initial vital signs included high fever (39.0℃), blood pressure of 140/80 mmHg, a heart rate of 92 beats/min, and a respiration rate of 20 breaths/min.\nLaboratory investigations showed a white blood cell (WBC) count of 8,610/mm3 (neutrophils, 83.9%; lymphocytes, 7.3%; monocytes, 6.3%; basophils 0.3%), a hemoglobin level of 13.6 g/dL, and a platelet count of 122,000/mm3. The C-reactive protein (CRP) level was 16.56 mg/dL.\nEnhanced computed tomography (CT) revealed a mycotic aneurysm originating from the abdominal aorta (), and no other infectious focus was observed on abdominal CT. A transthoracic echocardiogram did not reveal any evidence of infective endocarditis.\nThe patient was diagnosed with an abdominal aortic mycotic aneurysm. Because the patient had poorly controlled diabetes mellitus and mycotic aneurysm has a high mortality rate and can be caused by various organisms, strong broad-spectrum intravenous antibiotic therapy was immediately initiated (intravenous 1g q8hr of meropenem plus 200 mg q24hr of teicoplanin). The fever was controlled and the CRP level decreased during eight days of antibiotic therapy. However, even with intravenous antibiotics, he continued to complain of abdominal pain.\nThe patient's vital signs were stable without fever and laboratory-tested parameters did not worsen. However, we conducted a follow-up enhanced CT to assess the need for surgical management (8 hospital days). Enhanced CT showed an enlarged aneurysm in a state of impending rupture (). Surgery was immediately performed to address the infection. Massive atherosclerotic changes and plaque formation were observed in the intima of the abdominal aorta, and inflammatory change was noted from 1 cm below the renal artery bifurcation to the iliac artery bifurcation. The infected aortic wall was debrided and an aorto-bi-external iliac artery bypass was performed.\nLater, two out of three anaerobic blood cultures were positive for unidentified bacilli. To identify the isolate, 16S rRNA was sequenced (Macrogen, Daejeon, Korea). In brief, DNA was extracted and PCR was performed using primers targeting the 16S rRNA region, followed by purification, the sequencing reaction, and analysis. The resulting sequence was compared with sequences stored in the GenBank () and EzTaxon () databases, and one isolate was identified as B. thetaiotaomicron, with a percentage identity of 98.08%. Tissue biopsied from the wall of the aortic aneurysm was also positive for A. lwoffii, which was detected using standard microbiological methods. The following minimum inhibitory concentrations (MIC via VITEK II) were recorded: ampicillin/sulbactam 2; amikacin 2; ciprofloxacin 0.25; colistin 0.5; cefepime 1; imipenem 0.25; meropenem 0.25; ceftazidime 1; cefotaxime 8; and piperacillin/tazobactam 4. According to the microbiological results, the antibiotic therapy was changed from meropenem plus teicoplanin to ciprofloxacin plus metronidazole (intravenous 500 mg q12hr of ciprofloxacin and 500 mg q8hr of metronidazole).\nThe day after surgery, the patient had no fever and laboratory data indicated an improvement. Enhanced CT performed on day 7 after surgery () revealed no serious complications or re-infection. After 12 days of intravenous antibiotic therapy, the WBC count and CRP level had decreased to within the normal range. The patient recovered and was discharged. He remained in good condition and continued to take oral ciprofloxacin and metronidazole for 13 days after surgery. He is being followed-up at the outpatient clinic and is reported to be in good condition.

[[60.0, 'year']]

M

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{'2430200-1': 1}

3970310-1

noncomm/PMC003xxxxxx/PMC3970310.xml

Simultaneous Chylothorax and Chylous Ascites Due to Tuberculosis

A 17-year-old girl was admitted to the Korean University Ansan Hospital with a one-month history of fever, abdominal distention and dyspnea. The patient had been treated in another hospital for 10 days but the cause of ascites and pleural effusion had not been discovered. She was transferred to our hospital for further care. On admission, body temperature was 38.6℃, heart rate was 146/min and body weight was 56 kg. On chest auscultation, breathing sound was decreased on both lower lung fields. Abdominal examination revealed shifting dullness and the lower extremities were severely edematous. A chest radiograph taken on admission showed diffuse fine nodular opacities in both lungs and bilateral pleural effusion. Chest computed tomography (CT) showed pleural effusion of both sides, more serious on the left side, and pericardial effusion and multifocal nodular consolidation on both lung fields (). Abdominal CT showed a large amount of ascites with diffuse peritoneal thickening and multiple necrotic lymphadenopathy in the abdominal cavity, and enhanced wall thickening in the distal ileum and ascending colon (). Initial laboratory results were as follows: white blood cell count 6,670/mm3, hemoglobin 6.7 g/dL, serum total protein 4.6 g/dL and albumin 2.2 g/dL. Thoracentesis and paracentesis were performed to verify the nature of fluid apparent in the chest and abdominal CT images.\nPleural fluid () was turbid and whitish-colored, with pH 8.0, protein level 3.1 g/dL, lactate dehydrogenase of 5,855 IU/L, triglyceride 133 mg/dL and adenosine deaminase 121.6 IU/L. The lactate dehydrogenase ratio of pleural fluid versus serum was 15, consistent with exudate. Ascites () was milk-like and turbid in appearance, pH 8.0, albumin level 0.5 g/dL, triglyceride 864 mg/dL and adenosine deaminase 14.8 IU/L. We diagnosed chylothorax and chylous ascites because the triglyceride level of pleural fluid and ascites were higher than 110 mg/dL and 200 mg/dL, respectively. Direct smear of pleural fluid and ascites for acid fast bacilli (AFB) was positive only in pleural fluid. So, finally we diagnosed the patient as having TB associated with chylothrax and chylous ascites on the basis of positive AFB stain and abdomen CT. Initially, the patient received anti-TB treatment with quadritherapy including isoniazid 300 mg/day, rifampin 600 mg/day, pirazinamide 1,500 mg/day and etambutol 1,200 mg/day. The patient also was put on a high protein and low fat diet with medium chain triglyceride. Mycobacterium tuberculosis was cultured from sputum and pleural fluid on 28th day from admission and the isolate was susceptible to all anti-TB medications. The patient improved continuously after anti-tuberculosis medication, and pleural effusion and ascites were slightly decreased on follow-up chest and abdomen X-ray. The patient was discharged on day 60 of hospitalization with ongoing anti-tuberculosis treatment.

[[17.0, 'year']]

F

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{'5336629-1': 1, '5351360-1': 1, '8278846-1': 1, '5042527-1': 1}

3970313-1

noncomm/PMC003xxxxxx/PMC3970313.xml

Nocardia Brain Abscess in an Immunocompetent Patient

A 71-year-old male was admitted due to headache, dizziness, and nausea for three days. He was healthy, except that he had been diagnosed with hypertension two years before. His past medical history was unremarkable. His social history included intermittent alcohol consumption without smoking. The initial vital signs revealed blood pressure of 120/80 mmHg, heart rate of 76 beats per minute, respiration rate of 18 breaths per minute and body temperature of 36.9℃. He was alert upon admission. Physical examination revealed clear lung sound without rale or wheezing. The heart beat was regular without any murmur. There was no tenderness or rebound tenderness in the abdomen. Findings from other physical and neurological examinations were normal. There were no other symptoms such as fever, neck stiffness, photophobia, papilledema or other abnormalities. The initial peripheral blood count showed a white blood cell count of 11,200/mm3 (neutrophils 64.6%, lymphocytes 24.5%, monocytes 6.8%), hemoglobin level of 13.0 g/dL, platelet count of 211,000/mm3, erythrocyte sedimentation rate of 38 mm/hr, and C-reactive protein level of 1.11 mg/dL. Blood chemistry showed a total protein level of 7.6 g/dL, albumin level of 4.3 g/dL, total bilirubin level of 0.9 mg/dL, aspartate aminotransferase/alanine aminotransferase 27/16 IU/L, alkaline phosphatase 77 IU/L, BUN/Cr 9.2/0.8 mg/dL, Na/K 136/3.8 mEq/L, and Ca/P 9.0/4.7 mg/dL. Result of urinalysis was normal. Human immunodeficiency virus (HIV) antibody, hepatitis B surface antigen and hepatitis C antibody were all negative. Chest and abdomen radiographs were normal. Brain CT scan and magnetic resonance (MR) imaging showed multiple rim enhancing lesions with surrounding edema, which was presumed to be a brain abscess ().\nAfter admission, the patient underwent a craniotomy with stereotactic aspiration of the abscess. During the operation, yellowish and turbid pus was aspirated and a cerebral abscess was diagnosed. Empirical parenteral antibiotics were started with ceftriaxone 2 g bid and metronidazole 500 mg tid. Gram stain of the pus showed beaded branching gram-positive bacilli. Urease test, esculin hydrolysis test, citrate test, and modified acid-fast stain using 1% acid alcohol were all positive and the bacteria grew at 42℃. Nocardia species were suggested by colonial morphology, Gram-stain, and modified acid fast bacillus (AFB) stain (). By manual, culture was identified as Nocardia species. For the accurate identification of Nocardia species, 16S ribosomal RNA (rRNA) gene-based polymerase chain reaction (PCR) and sequencing was carried out. Primers of 27F (5'-AGA GTT TGA TCM TGG CTC AG-3') and 1492R (5'-TAC GGY TAC CTT GTT ACG ACT T-3') were used for 16S rRNA gene amplification. Size of 1466 bp 16S rRNA gene amplification sequencing was carried out by Solgent (Solgent Co, Daejeon, Korea) company which uses Big Dye Terminator Cycle Sequencing Kit and ABI PRISM 3730 DNA analyzer (PE Applied Biosystem, Foster, CA, USA). It showed 100% concordance to N. farcinica (GenBank accession number: KC478309) by BLAST Similarity Searching in GenBank. The organism was susceptible to amikacin, imipenem, trimethoprim-sulfamethoxazole (TMP-SMX), ciprofloxacin, and resistant to erythromycin, kanamycin, ampicillin, gentamicin. After identifying the result of pus culture, the antibiotic regimen was changed to TMP-SMX (15 mg/kg of the TMP component per day) and imipenem 500 mg qid. Due to the persistence of the patient's symptoms, re-imaging was performed two weeks later. On day 24 of admission, the patient underwent a repeated craniotomy with stereotactic aspiration of the abscess because the abscess and surrounding edema have increased than before. Ciprofloxacin (400 mg every 12 h, intravenous) was added to the TMP-SMX treatment and changed from imipenem to meropenem. After performance of repeated surgery and administration of additional antibiotics, we observed improvement of the patient's symptoms. Contrast-enhanced brain CT scan performed three weeks after reoperation, showed substantial resolution of the brain lesion. Meropenem and ciprofloxacin were continued for six weeks. On hospital day 67, he was discharged to go home to complete four months of oral TMP-SMX therapy. On three-month follow-up brain CT after discharge, resolution of the brain abscess was confirmed.

[[71.0, 'year']]

M

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{'8476204-1': 1, '8335456-1': 1, '6416759-1': 1, '8105746-1': 1, '7744209-1': 1, '7869307-1': 1, '7206790-1': 1, '5682745-1': 1}

3970374-1

noncomm/PMC003xxxxxx/PMC3970374.xml

Fatal Acute Right Heart Failure in Gastric Cancer Patients

A 46-year-old woman with gastric cancer presented with a 2 week period of progressively worsening shortness of breath. Six months earlier, she had a total gastrectomy. However, the adjuvant chemotherapy was not done because of her refusal. On admission, the physical examination including auscultation was unremarkable. The patient was afebrile with tachycardia of 116/min, respiratory rate of 28/min, and blood pressure of 100 /60 mmHg. Laboratory findings were remarkable for microcytic anemia and a strong positive D-dimer (11.8 μg/mL, normal <0.55). Electrocardiography showed sinus tachycardia. Echocardiography revealed feature of pulmonary artery hypertension, namely a severely dilated right ventricle with grossly impaired systolic function and an estimated pulmonary artery pressure of 52 mmHg []. Chest computer tomography (CT) presented no evidence of pulmonary emboli []. She rapidly developed hypoxemic respiratory failure and desaturated 80% on 10 liters of oxygen. The patient's condition progressively worsened and took a rapid downhill course, despite aggressive hemodynamic support. Finally, the patient developed an intractable respiratory failure and died 14 hours after hospitalization.

[[46.0, 'year']]

F

{'31273537': 1, '15660700': 1, '7581935': 1, '17349704': 1, '31032740': 1, '17539970': 1, '1175142': 1, '12935829': 1, '17290069': 1, '2163747': 1, '19461409': 1, '24696760': 2}

{'3970374-2': 2}

3970374-2

noncomm/PMC003xxxxxx/PMC3970374.xml

Fatal Acute Right Heart Failure in Gastric Cancer Patients

A 48-year-old man presented with a 2 week history of rapid progressive exertional dyspnea. His gastric cancer had been diagnosed 1 year previously and was treated with gastrectomy and chemotherapy. On physical examination, the patient was afebrile with respiratory rate of 25/min, and blood pressure of 100/60 mmHg. Electrocardiography showed sinus tachycardia. Echocardiography revealed a right ventricular overloading sign with an elevated pulmonary artery pressure of 70 mmHg. Laboratory results showed microcytic anemia and an elevated D-dimer serum level (19.1 μg/mL, normal <0.55). Chest CT presented no evidence of pulmonary thromboembolism. Ten hours after hospital admission, the patient's state rapidly deteriorated, with increasing dyspnea, peripheral cyanosis. He showed poor response to the initial management with oxygen and continuous positive airway pressure. He progressed to cardiogenic shock and had no improvement with a vasoactive drug. He had persistent hypoxemia. He died from refractory right heart failure caused by pulmonary artery hypertension.

[[48.0, 'year']]

M

{'31273537': 1, '15660700': 1, '7581935': 1, '17349704': 1, '31032740': 1, '17539970': 1, '1175142': 1, '12935829': 1, '17290069': 1, '2163747': 1, '19461409': 1, '24696760': 2}

{'3970374-1': 2}

3970375-1

noncomm/PMC003xxxxxx/PMC3970375.xml

Inadvertent Temporary Pacemaker Lead Placement in Aortic Sinus

A 50-year-old female was referred to our emergency with history of recurrent syncope followed by cardiac arrest for which she underwent temporary pacemaker insertion at a peripheral primary care center. The pacemaker was inserted through right femoral access without fluoroscopic guidance. After haemodynamic stabilization she was referred to our tertiary care center for further management. On examination, pulse rate was 60/min and regular. Blood pressure was 130/70 mm Hg. Cardiovascular system examination was unremarkable. Electrocardiogram showed paced rhythm mimicking atrial pacing with short PR interval followed by narrow QRS complexes []. The QRS axis was normal (+45°). Echocardiography showed looped pacing lead in the ascending aorta. There was no pericardial effusion.\nFluoroscopy showed temporary pacing lead, which traversed through the aorta and forming abnormal loops within ascending aorta with the tip facing downwards and engaged in the non-coronary sinus [Figure and ; Videos and ]. Vascular access sheath guide wire was also seen within the right atrium extending into superior venacava and inferior venacava. The pacing threshold was 5 Volts and she was pacemaker dependent.\nThe patient underwent successful percutaneous retrieval of the guide wire using gooseneck snare [Figure and , Videos and ] followed by placement of trans- venous temporary pacemaker lead inside RV. Later the pacemaker lead inside ascending aorta was removed. Finally she underwent permanent pacemaker implantation through right subclavian vein access.

[[50.0, 'year']]

F

{'1604926': 1, '6382429': 1, '17600676': 1, '3734207': 1, '24696761': 2}

{}

3970376-1

noncomm/PMC003xxxxxx/PMC3970376.xml

Recurrent Transient Apical Cardiomyopathy (Tako-Tsubo-Like Left Ventricular Dysfunction) in a Postmenopausal Female with Diffuse Esophageal Spasms

A 66-year-old woman with a history of AV nodal ablation for atrial fibrillation and severe, diffuse esophageal spasms which required multiple dilations and surgical myectomy had been hospitalized multiple times at a nearby institution with shortness of breath and chest pain. Most of her symptoms had been attributed to esophageal spasms confirmed by manometry. During her first admission, an echocardiogram demonstrated an ejection fraction of 25-30% with apical anterior, inferior and lateral wall akinesis, compatible with Tako-tsubo-like cardiomyopathy. She underwent a coronary angiogram 48 h later with no evidence of obstructive disease and normalization of left ventricular function. Coronary vasospasm was suspected but no provocative testing was performed.\nShe presented six months later to our hospital complaining of severe chest pressure that started abruptly after she became furious at her driver for being late. Her ECG on admission [] displayed a ventricular paced rhythm, with hyperacute T waves and new ST segment elevation in anterior precordial leads; she was given aspirin and started on heparin infusion. Once intravenous infusion of nitroglycerin was initiated, she had resolution of her index symptoms, as well as most of her ECG changes []. She had a minor elevation in her troponin levels. Echocardiogram performed the next morning [] revealed an ejection fraction of 15% with apical ballooning, consistent with Tako-tsubo-like cardiomyopathy. She received supportive management and was started on long acting nitrates and calcium channel blockers with no recurrence of symptoms. On admission day # 5, her left ventricular function was reassessed by echocardiogram and noted to be within normal limits.

[[66.0, 'year']]

F

{'20200634': 1, '16387792': 1, '20215125': 1, '18288755': 1, '18687244': 1, '16325287': 1, '11258574': 1, '2868204': 1, '12628716': 1, '17403953': 1, '24695751': 2}

{}

3970922-1

noncomm/PMC003xxxxxx/PMC3970922.xml

A challenging diagnosis: case report of extensive pyoderma gangrenosum at multiple sites

A 51-year-old female initially presented with infected abdominal, thigh, and sacral ulcers. Her ulcers developed as a result of poor self-care and hygiene. Her past medical history included type 2 diabetes, hypertension, hypercholesterolemia, and back pain. Physical examination revealed an abdominal ulcer measuring 4 cm × 2 cm in the right iliac fossa with surrounding erythema, a small ulcer on her left thigh (1 cm × 1 cm), and two sacral ulcers (7 cm × 3 cm and 4.5 cm × 2 cm, respectively). Ulcer cultures revealed skin flora. Although she did not have any systemic features of infection, blood investigations on admission showed an elevated white cell count of 14.9×109/L (reference interval, 4.0×09−11.0×109/L) and C-reactive protein of 120 mg/L (reference interval, 0–10 mg/L). She was subsequently commenced on 2 g of empirical intravenous flucloxacillin four times daily.\nIn the next few weeks, all her wounds worsened despite wound dressings and antibiotics. Her abdominal and thigh ulcers had increased in size. The size of her two sacral ulcers remained the same but had increased wound necrosis. One month after admission, her abdominal ulcer measured 21 cm × 7 cm, and the thigh ulcer was 4 cm × 1 cm. In view of this, surgical excisions of the necrotic areas were performed. Vacuum-assisted closure dressings were applied to the anterior abdomen and sacrum.\nThree days later, the patient underwent a routine vacuum-assisted closure dressing change. It was noted that her wounds had worsened rapidly since the last debridement. The once healthy wound edges had become necrotic. Further debridement was performed. Excised tissues sent off for microscopy and culture grew methicillin-sensitive Staphylococcus aureus and Enterobacter aerogenes. Intravenous tazocin (4.5g 8 hourly) was added for suspected postoperative wound infection.\nThe patient’s wounds continued to deteriorate with antibiotics and further debridements. After the third debridement, the abdominal ulcer had increased to a size of 32 cm × 11 cm, and the two sacral ulcers became a huge sacral defect extending into the perianal region ( and ). Cultures of subsequently debrided tissue were negative for bacteria and fungi. Repeated histological sections of sacral and abdominal tissues revealed neutrophilic infiltrates extending into deep dermis and no evidence of malignancy ( and ). Antinuclear antibodies, extractable nuclear antigens, rheumatoid factor, and anti-neutrophil cytoplasmic antibodies were all negative. A diagnosis of PG was made, and our patient was commenced on 50 mg of prednisolone daily. After 1 week, her wounds showed slow improvement, with resolution of necrotic wound edges and formation of granulation tissue. Two weeks later, she developed steroid-induced hyperglycemia, and 500 mg twice daily of mycophenolate mofetil was added as a steroid sparing agent. Her prednisolone dose was gradually tapered by 5 mg every 2 weeks, and the dose of mycophenolate mofetil increased to 1 g twice daily. Four months after admission, the patient underwent successful split skin grafts and primary closure to her abdominal and sacral ulcers while on immunosuppressive therapy ( and ). At 3-month follow-up, there was no recurrence of the disease.

[[51.0, 'year']]

F

{'3351014': 1, '957999': 1, '15533059': 1, '28944125': 2, '25374597': 1, '22273223': 1, '26782091': 1, '19903010': 1, '12907338': 1, '3876359': 1, '21745697': 1, '10428135': 1, '15888172': 1, '20666672': 1, '11978136': 1, '686728': 1, '4508849': 1, '12510450': 1, '10670408': 1, '786171': 1, '11971877': 1, '24741322': 2}

{'5602483-1': 1}

3970940-1

noncomm/PMC003xxxxxx/PMC3970940.xml

Never neglect the atmospheric pressure effect on a brain with a skull defect

A 65-year-old male patient was transferred to our hospital with a large, right-sided skull defect and severe scalp depression. He had suffered a traumatic brain injury 5 months earlier and undergone decompressive craniectomy and removal of an intracranial hemorrhage at that time. Hydrocephalus developed one month after the operation and he received a ventriculoperitoneal shunt. However, his level of consciousness progressively deteriorated thereafter. On neurological examination, he was drowsy with a Glasgow Coma Scale score of E1V1M4 and profound left hemiplegia. A computed tomography (CT) brain scan revealed marked concavity of the brain at the craniectomy site associated with midline shift to the left (). Transcranial Doppler sonography (Multi-Dop® X2, DWL; Elektronische Systeme GmbH, Schlotheim, Germany) was unable to detect the wave pattern of the blood flow in the middle cerebral artery of the concave brain. The impression was of sinking skin flap syndrome, so cranioplasty with bone cement was performed. Postoperatively, the patient was treated with hydration and bed rest for 3 days. Four days after his cranioplasty, follow-up CT images showed reversal of the midline shift with no significant complications in the underlying brain (). Follow-up transcranial Doppler revealed a mean velocity of 30 cm per second in the right middle cerebral artery. After the surgery, the patient’s mentality gradually improved to an alert state and he was discharged with a Glasgow Coma Scale score of E34V1M5–6 one month after cranioplasty.

[[65.0, 'year']]

M

{'558536': 1, '12383414': 1, '27810603': 1, '26889273': 1, '12915237': 1, '11426120': 1, '8008174': 1, '16551430': 1, '15177389': 1, '14718728': 1, '19096548': 1, '16394936': 1, '33628544': 2, '14639563': 1, '24741332': 2}

{'7886569-1': 1, '7886569-2': 1, '7886569-3': 1}

3971769-1

noncomm/PMC003xxxxxx/PMC3971769.xml

Distal ‘buddy-in-jail’ technique: a complementary ‘Jail with stent’ method for stent delivery

A 60-year-old male, heavy smoker and hypercholesterolemic was admitted for NSTEMI. Performed by 5F right transradial access, the coronary angiogram showed severe multifocal RCA disease, with a highly calcified and tortuous vessel. The mid to distal segment was the most severely diseased. Ad hoc PCI was decided, starting with a 5F RRAD Launcher from Medtronic as guiding catheter. Buddy wiring was planned from the beginning with one SION (Asahi) as main wire and one Balance Middle Weight® (BMW) (Abbott Vascular) as the buddy wire. Starting from the distal lesion, we then proceeded with balloon debulking: 2–10, 2.75–10 and finally 3–10 balloons (Sapphire II, OrbusNeich™) were successfully advanced and inflated through all lesions. Despite this debulking and the presence of the BMW buddy wire, a bare metal stent (BMS) 3–13 (Azule™SDS, OrbusNeich™) failed to reach the target distal lesion: the stent was unable to cross the bend at the level of the residual mid RCA lesion. We then changed to a shorter and smaller BMS (2.75–09 PRO-kinetic Energy®, Biotronik), which fortunately reached the distal lesion. It was deployed at 18 atmospheres, jailing the BMW wire. This BMW wire, now in a jailed position, was used subsequently to load the same 3–13 Azule™ stent which had previously failed. We left the Sion wire as the buddy wire. The stent was successfully pushed forward and positioned at the mid RCA lesion. Before delivery, the Sion wire was removed. Before un-jailing the BMW wire, we further expanded the Azule stent with a non-compliant balloon. The final angiographic result was good.

[[60.0, 'year']]

M

{'15867450': 1, '12822150': 1, '19472348': 1, '10525229': 1, '17404407': 1, '24552227': 2}

{}

3971912-1

noncomm/PMC003xxxxxx/PMC3971912.xml

Refractory headaches treated with bilateral occipital and temporal region stimulation

A 31-year-old female had a 10-year history of daily severe occipital and temporal region headaches that were refractory to numerous conservative therapies. She had initially presented with new daily persistent headache (NPDH) in the absence of a prior history of migraine. However, the phenotype of the headache disorder did not meet ICHD-II (International Headache Classification – 2nd edition) criteria for NDPH due to the daily presence of migraine features including nausea, photophobia, and phonophobia. The phenotype did meet the ICHD-IIR (revised) criteria for chronic migraine, with the exception of not having 5 prior attacks. A complete, thorough, and exhaustive search for secondary causes was unrevealing. The patient developed a daily and persistent headache in the absence of any analgesic use; ie, she did not have a medication overuse headache. She did begin to overuse analgesic medications as a consequence of her new and persistent daily pain. The patient was withdrawn from all analgesics after a rigorous withdrawal schedule. Unfortunately, despite being off analgesics for >3 months, there was no change in her headache pattern in terms of severity, disability, or its continuous nature.\nThe patient was ultimately diagnosed with chronic migraine (>15 headache days per month for at least 3 months) and treated with numerous medications including divalproex sodium, amitriptyline, gabapentin, topiramate, botulinum toxin, opiates, and metoclopramide. Lithium and verapamil were also tried after all other conventional and evidence-based options had been exhausted.\nThe patient was referred to the pain clinic and after a 5 day successful trial (>50% reduction in pain severity) of bilateral ONS with Advanced Neuromodulation Systems leads (now St Jude Medical Neuromodulation; St Paul, MN, USA) the patient underwent permanent implant of an ONS system that included bilateral quadripolar leads and placement of an internal pulse generator (IPG) in her lower lateral abdomen. This resulted in sustained, compete resolution of her occipital pain. However, she continued to report significant pain in her temporal regions bilaterally that was unfortunately not diminished with ONS. Specifically, she identified the areas lateral to the eyebrows and superior to her zygomatic arches as the regions most affected.\nDespite medical management, the patient experienced daily pain in these areas. She reported to her headache neurologist that her bilateral temporal pain was 10/10 in intensity, and that four to five days per week she was bedbound. Her primary care physician prescribed local anesthetics that she self-injected subcutaneously up to eight times per day, providing mild relief from her intractable temporal headaches. After consultation with neurology headache, pain management, and neurosurgical specialists at our institution, the interdisciplinary team felt she would benefit from placement of bilateral temporal region stimulators.\nUnder intravenous sedation and local anesthesia, a St Jude Neuromodulation quadripolar lead (St Jude Medical Neuromodulation) was inserted subcutaneously from a point approximately 6 cm above the temporal region. This was carried out bilaterally through a 14-gauge Tuohy needle, with the goal of maintaining the needle in the subcutaneous fat, above the fascial layer. Intraoperative testing confirmed appropriate patterns of stimulation in the region of the patient’s temporal pain. The stimulation patterns were a result of various electrode combinations, rates, and pulse widths that provided a non-painful sensation of stimulation.\nNext, under general anesthesia, the left electrode was tunneled to a point near the vertex in the subcutaneous channel. Both leads were then tunneled down the right side of her neck and between her breasts to reach the IPG (). Therefore, the patient had tunneled extensions both anteriorly for her temporal stimulator and posteriorly for her ONS ().\nThe patient tolerated the procedure well. At 6-month and 12-month follow up, the patient noted considerable improvement in her daily headaches. Stimulation settings were adjusted at each follow-up visit to patient comfort and effect, and varied somewhat over time. Her stimulator settings fell into ranges typical for this patient population: rate of 30–60 Hz; amplitude 1–5 volts; pulse width 250–450 milliseconds.\nThe patient essentially began to experience improvement immediately after implantation. Prior to implantation, she reported daily 10/10 pain; following the operation, she was reporting 10/10 temporal pain just one day per week and on average, 8/10 on the numeric pain scale, which she considered a significant improvement although she continued to have daily headaches. She was using both the occipital and temporal stimulators continuously and her only pain medications were over-the-counter analgesics on a pro re nata basis. Overall, she noted a 50% improvement in her functional status. She was no longer bedbound the majority of the week; she was able to participate in activities of daily living including caring for her three-year-old child and attending yoga classes. Her quality of life was markedly improved and during the intervening year she delivered a second child.\nUnfortunately, an infection developed over the anchor in her right parietal region. Initially, antibiotic treatment was attempted but when component erosion occurred, the bi-temporal stimulator components were removed. The patient has indicated she strongly wants the temporal stimulator replaced as soon as possible.

[[31.0, 'year']]

F

{'22151211': 1, '17341049': 1, '23034698': 1, '23707274': 1, '19614702': 1, '26177612': 1, '17381554': 1, '22882274': 1, '21992787': 1, '24338699': 1, '21854497': 1, '22150992': 1, '25688595': 1, '21422775': 1, '21267040': 1, '26049769': 1, '24348076': 1, '26558174': 2, '18279430': 1, '17635606': 1, '19732075': 1, '24707189': 2}

{'4633430-1': 1}

3971937-1

noncomm/PMC003xxxxxx/PMC3971937.xml

Bilateral papillopathy as a presenting sign of pheochromocytoma associated with von Hippel–Lindau disease

An otherwise healthy 7-year-old girl was evaluated for 1 month of bilateral vision loss. She reported no history of headaches, eye pain, neurological deficits, or other associated symptoms. Her past medical history and family history were negative for eye disease, renal cell carcinoma, or neurologic syndromes. She was on no medications and had no allergies.\nThe patient was in no distress, and her screening vital signs were normal with a blood pressure of 110/70 mmHg and a heart rate of 95 bpm. Best-corrected visual acuity was 20/50 in the right eye and 20/60 in the left eye, with a normal pupillary examination and intact color vision. Ocular motility and intraocular pressures were normal. Automated visual field testing (Humphrey 30-2 threshold, Swedish Interactive Threshold Algorithm-standard strategy; Carl Zeiss Meditec, Dublin CA, USA) demonstrated bilateral enlarged blind spots and cecocentral scotomas (). Fundoscopy showed diffuse bilateral optic nerve head edema with extensive circumferential peripapillary and macular exudation. The optic nerve heads had no spontaneous venous pulsations (). The neurologic examination was unremarkable.\nThe brain magnetic resonance imaging (MRI) with and without contrast revealed multiple high T2 and fluid-attenuated inversion–recovery (FLAIR) signal lesions found diffusely in the cortical and subcortical white matter region of the frontal and parietal lobes (). One lesion in the left frontal posterior frontal lobe had higher T1 signaling, and a computed tomography (CT) scan confirmed calcifications of this single lesion ( and ). A lumbar puncture revealed an opening pressure of 150 mm water with normal cerebrospinal fluid cytology and analysis: red blood cells =1; white blood cell =0; protein =20 mg/dL; glucose =71 mg/dL; immunoglobulin G =1.76 mg/dL; no oligoclonal bands; and no growth in cultures.\nThree days later on follow-up examination, the patient was found to have paroxysmal tachycardia with a heart rate of 180 bpm and blood pressure of 120/75 mmHg. Repeat blood pressure measurements showed episodic elevation in blood pressure reaching as high as 240/160 mmHg. The patient was treated medically for hypertension. In an extensive workup, an abdominal CT scan showed a 3.5×3.0×3.0 cm enhancing noncalcified mass in the right adrenal gland ().\nLaboratory analysis of the patient’s 24-hour urine collection showed elevated vanillylmandelic acid of 15.1 mg (normal <2.3 mg) and random total metanephrines of 22,295 μg/g of creatinine (normal 202–750 ug/g). A scintigraphic distribution of iodine-131-meta-iodobenzylguanidine (I-131 MIBG), an adrenal medullary imaging agent, showed increased radioactive I-MIBG uptake by the right adrenal mass but the absence of pathologic radioactivity elsewhere, and the patient underwent laparoscopic resection of the mass. The pathological specimen showed whorled nests of chromaffin tumor cells () embedded in a fibrovascular stroma. These histopathological findings were consistent with pheochromocytoma.\nPostoperatively, the episodic hypertensive episodes resolved and vanillylmandelic acid/metanephrines normalized. After 5 months, the patient’s visual acuity improved to 20/30 in both eyes. Fundoscopically, the optic disc edema and exudates improved, and the postoperative MRI showed a subsequent decrease in white matter lesions ().\nThe patient was lost to follow-up for many years and returned at the age of 18 when she developed shortness of breath over 4 days. She was found to have multiple pulmonary lesions bilaterally; a biopsy showed metastatic pheochromocytoma. Blood pressure at that time was well-controlled on atenolol; however, serum metanephrines were elevated. She underwent serial thoracotomies for resection of the lesions, and she then started on chemotherapy with sunitinib for 12 months. Multiple follow-up metaiodobenzylguanidine (MIBG) scans and urine collections have been negative for metastatic pheochromocytoma. Subsequent genetic testing identified a 3p25-26 (c.482 G>A) chromosomal mutation consistent with a mutation of the VHL gene. RET proto-oncogene sequencing detected no mutation in exons 10, 11, 13, 14, 15, and 16.\nThe patient’s family history was negative for neuroendocrine disorders, including multiple endocrine neoplasia 2A and 2B, VHL disease, von Recklinghausen disease, tuberous sclerosis, and Sturge–Weber syndrome. Recommendations were made to have family members tested; however, the patient declined as the family lived out of the country. Ophthalmic evaluation at this time showed multiple peripheral vascular dilations and small retinal capillary hemangioblastomas (). Repeat imaging and follow-up over a year showed her to be stable without evidence of brain or spinal cord hemangioblastomas, or abdominal pathology, which is concerning for renal cell carcinoma.

[[7.0, 'year']]

F

{'33531956': 2, '11578646': 1, '9862313': 1, '18769700': 1, '12000821': 1, '9220570': 1, '1967325': 1, '12814730': 1, '2274658': 1, '22245022': 1, '26268347': 2, '4026651': 1, '9267873': 1, '3951818': 1, '29260095': 1, '8898786': 1, '11801269': 1, '17694830': 1, '3235439': 1, '11668103': 1, '10768593': 1, '9881533': 1, '24707167': 2}

{'7847098-1': 1, '4535675-1': 1}

3971947-1

noncomm/PMC003xxxxxx/PMC3971947.xml

Near-infrared spectroscopy and plasma homovanillic acid levels in bipolar disorder: a case report

The patient was a 22-year-old man who had no history of psychiatric illness. He was admitted to our hospital due to major depression that was diagnosed according to the Diagnostic and Statistical Manual for Mental Disorders, Fourth Edition, with moderate sadness, agitation, retardation, and difficulty in concentrating. The clinical course after admission is summarized in . We treated the patient with paroxetine and lorazepam for about 4 weeks, but treatment was not successful. On day 4, his Montgomery Åsberg Depression Rating Scale (MADRS) score was 44, and his plasma levels of HVA and MHPG were 4.6 ng/mL and 1.9 ng/mL, respectively. shows the waveform of [oxyHb] during the cognitive activation of the word fluency task. NIRS signal changes were measured during a 10 second pretask baseline period, a 60 second task period, and a 55 second posttask baseline period. The initial syllables assigned were changed every 20 seconds (/a/, /ka/, and /sa/, respectively) during the 60 second task period. During the task period, the patient was instructed to generate as many Japanese words beginning with the designated syllable as possible. In the pretask and the posttask baseline periods, the patient was instructed to repeat a train of syllables (/a/, /i/, /u/, /e/, and /o/). He sat on a chair in a room with daylight with his eyes open throughout the measurements. We focused on increases in [oxyHb], because this appears to reflect task-related cortical activation more directly than decreases in [deoxyHb]. We defined the patterns of NIRS waveforms using the grand average waveforms in the frontal region (CH 25–28, CH 36–38, and CH 46–49; ) and two simple visual indices, integral value, and centroid value. The integral value describes the size of the hemodynamic response during the 60 second activation task period, whereas the centroid value serves as an index of time course changes throughout the task, with periods representing the timing of the hemodynamic response. A recent study reported that the optimal threshold of the centroid value of the frontal region between major depressive disorder and two other disorders (schizophrenia and bipolar disorder) is 54 seconds (major depressive disorder < other 2 disorders). On day 4, the centroid value of the grand average in the central region was 68.6 seconds (), and the NIRS waveforms showed an [oxyHb] reincrease in the posttask period (CH 34, 35, 36, 39, 45, 46, 47, and 49; ), which is characteristic of schizophrenia.,,– On day 25, his MADRS score was 40, his plasma HVA level was markedly higher at 49.3 ng/mL, and his plasma MHPG level was 1.6 ng/mL. On day 27, he was voluntarily discharged from the hospital, but was still in a depressive state. On day 30, 3 days after leaving the hospital, he became manic and was readmitted to our hospital. He was in a state of psychotic mania with elevated mood, grandiosity, aggression, auditory hallucination, and thought broadcast, with a Young Mania Rating Scale (YMRS) score of 48. Paroxetine and lorazepam were discontinued. We subsequently treated him with lithium, risperidone, and zotepine for approximately 3 weeks, and his manic state improved. On day 46, his YMRS score was 29, and plasma levels of HVA and MHPG were 17 ng/mL and 1.3 ng/mL, respectively. However, he refused drugs because of extrapyramidal symptoms and became manic with psychotic symptoms again. Therefore, we tried a combination of lithium, olanzapine, and zotepine, and the treatment was successful. On day 77, the NIRS waveforms turned to a negative pattern (), which was defined by an integral value of the grand average in the central region <0, whereas there were [oxyHb] reincreases in the posttask period (CH 33, 41, 42, 43, 44, 51, and 52). On day 99, his YMRS score decreased to 3, and his plasma levels of HVA and MHPG were 8.7 ng/mL and 1.9 ng/mL, respectively. He was discharged from the hospital on day 106. Nine months later, he was off medication, became manic again, and was admitted to another psychiatric hospital.

[[22.0, 'year']]

M

{'11078046': 1, '9495606': 1, '6155678': 1, '21600294': 1, '15023578': 1, '16125979': 1, '21068827': 1, '24016725': 1, '23437963': 1, '11926074': 1, '18063344': 1, '23764293': 1, '17029305': 1, '22572569': 1, '12063160': 1, '10357026': 1, '15820269': 1, '22198450': 1, '8746704': 1, '10327429': 1, '31973606': 1, '22935269': 1, '22330179': 1, '24707175': 2}

{}

3972028-1

noncomm/PMC003xxxxxx/PMC3972028.xml

“Denervation” of autonomous nervous system in idiopathic pulmonary arterial hypertension by low-dose radiation: a case report with an unexpected outcome

A 58-year-old woman presented to the Coburg Clinic in February 2013 with dyspnea, loss of energy, slight edema of the lower extremities, and a 4 kg weight loss in the 4 weeks before presentation. A 3/6 systolic murmur with a left apical maximum point was noted on physical examination. Except for an appendectomy more than 30 years earlier, the patient had never been hospitalized or on any medication. She had never smoked. Family history was unremarkable, except for a sister who had experienced uncomplicated tuberculosis. She had worked as a seamstress until giving birth to a healthy daughter in 1973. From 1976 to 1980 and then again from 1985 to1997 she reported having worked in the ceramics industry, but her job was not associated with significant exposure to ceramic dust. During that time, living in the former German Democratic Republic, she had had annual chest X-rays without any abnormalities. During a second examination, she reported having felt dyspnea on exercise since 2003, but did not complain of loss of energy, and had been able to perform regular work. The electrocardiogram performed on admission showed sinus tachycardia at a rate of 109 beats per minute, incomplete right bundle branch block, and preterminal inverted T-waves in II, III, aVF, and V1–V6.\nBody plethysmography showed slight restriction with forced expiratory volume in one second being 68% predicted, inspiratory vital capacity 70% predicted, and a Tiffeneau index of 101%. Her diffusion capacity was reduced at 60%. Her respiratory insufficiency was managed with oxygen substitution therapy (pO2 60 mmHg and pCO2 33 mmHg under oxygen 3 L per minute).\nAn initial transthoracic echocardiogram on February 1, 2013 demonstrated dilatation of the right ventricle to 38 mm and dilatation of the left atrium to 43 mm. Sclerosis of the aortic valve, minimal aortic insufficiency, first-degree mitral insufficiency, obvious tricuspid valve insufficiency with a diastolic gradient of 67 mmHg, and diastolic dysfunction of the left ventricle with a slightly pathological E/A ratio could also be visualized. Left ventricular ejection fraction was normal at 65%.\nThe woman presented for the first time to the pulmonary consultant late on a Friday afternoon with dyspnea and spontaneous oxygen saturation of 83% on the bronchoscopy table (pO2 38 mmHg, pCO2 31 mmHg). She complained of rapid deterioration in the previous 2 days in comparison with her status on admission. Her weight was 58 kg, but she complained a 19 kg weight loss in the previous 2 months. Her height was 158 cm (body mass index 23.1 kg/m2). At that time, only a standard chest X-ray taken on admission was available, and showed a prominent bihilar region with a suspicious mass in the left apex of the lung (). Standard serum analysis showed slightly elevated lactate dehydrogenase and uric acid levels, with a mild anemia. On fluoroscopy, the right hilum appeared to be enlarged compared with the X-ray taken on admission. No result suspicious for a central tumor or other infiltrative disease was seen on bronchoscopy. Endobronchial ultrasound revealed enlarged pulmonary arteries containing multiple thrombi, a few enlarged lymph nodes, and abnormal mediastinal tissue anatomy suspicious for mediastinal infiltration of a malignant process. We did not perform transbronchial needle aspiration before the weekend due to her dyspnea. She was put on low-dose low molecular weight heparin. The decision was taken to start her on radiotherapy over the weekend due to the suspicion of rapid progression of small cell lung cancer or lymphoma in the mediastinum. Computed tomography planning was done in the radiotherapy department on the following day and she received radiation with 4 Gy to the mediastinum including both hila over the weekend (2 Gy per day).\nAfter the weekend, we observed a mediastinal mass, abnormally enlarged central pulmonary arteries with tortuous peripheral branches, and a consolidation in the left upper lobe on the patient’s computed tomography scan ( and ). At that point we stopped radiation therapy because it was clear that we were not dealing with a typical case of suspected small cell lung cancer or lymphoma. Repeat bronchoscopy and endobronchial ultrasound demonstrated partial compression of the central vessels by a mass located in the left mediastinum, and partial infiltration of the walls by this mass was suspected. Given that the lymph nodes in position 10 R (right) and 7 had a diameter of 15–20 mm, we took several biopsies from these sites in addition to biopsies of the mediastinal mass. Histologically, there was no suggestion of malignancy, but a strong suspicion for fibrosis was raised on evaluation of the magnetic resonance imaging (MRI) scans () but could not be proven.\nTwo weeks later, an endoesophageal ultrasound was performed with a thicker needle in lymph node position 7. Again there was only a hint of fibrosis. A transthoracic lung puncture of the mass in the left upper lobe revealed thickened pulmonary arterial walls, activated pneumocytes, and fibrosis (–). Several endoscopic ultrasound consultants and endobronchial ultrasound examinations reported massive “smoke” (indicating prethrombotic formations) in the central pulmonary arterial vessels. Tuberculosis was ruled out by a negative interferon gamma release assay, several negative bronchial specimens examined by microscopy, polymerase chain reaction, and cultures for tuberculosis collected by bronchoscopy. Nontuberculous mycobacteria was ruled out by negative polymerase chain reaction, microscopy, and culture results, as was sarcoidosis, given the patient’s normal serum soluble interleukin-2 receptor and angiotensin-converting enzyme levels. We could not find any histological suggestion of histoplasmosis, but this diagnosis was not ruled out by serum analysis as the patient appeared to be immunocompetent with normal immunoelectrophoresis and a negative human immunodeficiency virus test result.\nCardiac MRI showed reduced right ventricular function, dilatation of the right ventricle and a hypertrophic wall, a flattened septum with paradoxical movement (), and dilated pulmonary artery trunks. There was no suggestion of intravascular thrombus on pulmonary angiography during cardiac MRI. A sudden change in diameter of the pulmonary arteries could be seen in the peripheral regions. Small tortuous peripheral pulmonary arteries were seen on cardiac MRI and computed tomography (). The aforementioned abnormal mediastinal mass and left upper lobe mass were also observed.\nLung embolism was excluded using ventilation and perfusion scintigraphy. Combined ventilation and perfusion defects in the left upper and middle lobes were associated with the mediastinal mass.\nPrecapillary PAH with a PAR of 1,360 dyn · s · cm−5 and completely normal wedge pressure was seen on right heart Swan-Ganz catheterization, coronary angiography, and dextrography (). There were no definite criteria for reactive PAH on inhalation of iloprost. The right ventricle was hypokinetic especially in its anterior portion ( and ) with an enlarged wall which, on echocardiography, was 8 mm thick (ie, thickness of right ventricular anterior wall in end diastole). The median right atrial pressure was 8 mmHg and borderline for right heart decompensation. The pulmonary arteries were shown to be dilated on angiography, but there was no indication of peripheral obstruction or chronic thromboembolic pulmonary hypertension. The left marginal artery and diagonal branch showed an angiographic stenosis of 70% ( and ), and left ventricular ejection fraction was 70%. There was endothelial dysfunction with low flow phenomenon in different coronary arteries, besides the partly abnormal coronary anatomy with dilated cardiomyopathy ( and ). There was no suggestion of vasculitis, coagulopathy, a thyroid or hepatic disorder, or collagenosis on serum analysis. No other predominant cardiopulmonary disease directly involved in the initially highly elevated PAH hemodynamics was found after working through the algorithm for PAH recommended by the European Society of Cardiology.\nA dobutamine MRI stress test was performed a week later without any suspicion for ischemia, so stenting of the abovementioned stenosis observed on angiography was not performed. Because thrombus was expected in the pulmonary arterial walls, transesophageal echocardiography was performed and supported the presumption of thrombus in the pulmonary arterial walls, including prethrombotic “smoke” in the central vessels. No shunt was observed. Phenprocoumon was started as oral anticoagulation therapy. Thoracic surgeons at the nearby Kutzenberg Hospital refused to do mediastinoscopy because of the diagnosis of PAH.\nPositron emission tomography at the University of Erlangen showed no suspicion of tumor or lymphoma in the mediastinum or elsewhere, and mediastinal fibrosis was again diagnosed. Lymphoma was ruled out in an oncologic ambulatory setting via peripheral lymph node biopsies from the groin, serum electrophoresis, urinalysis, and bone marrow puncture. Serum beta2-microglobulin was negative.\nAt the end of April, the patient was presented for the first time at the National Pulmonary Hypertension Center of Excellence in Bad Nauheim/Giessen and a second time in May 2013 () with a differential diagnosis of idiopathic PAH. This was done because the referring cardiopulmonary consultant at Coburg Clinic, who is also an expert in the treatment of PAH and a member of the national guideline group, was surprised by this patient’s abnormal anatomy and the massive extent of central smoke without any peripheral suggestion of embolism. Repeat pulmonary angiography showed no thrombus (), and chronic thromboembolic pulmonary hypertension was excluded on ventilation/perfusion (V/Q) scintigraphy. Right heart catheterization revealed precapillary pulmonary hypertension with a PAR of 600 dyn · s · cm−5 and no reactive properties, which was quite striking given that our patient had not received any specific treatment in the preceding weeks. Cardiac MRI revealed the same pathologies in the pulmonary arterial walls as before, and functional and anatomic abnormalities of the right ventricle and mediastinal fibrosis were again reported. The diagnosis of idiopathic PAH was confirmed at several conferences with the team led by Ghofrani and Mayer (Professor Hossein A Ghofrani, Pulmonary Department, University Hospital Giessen, Giessen, Germany; and Professor Eckhard Mayer, Thoracic Surgery Department, Bad Nauheim, Germany). During discussion between the experts at both institutions, the question was raised as to whether a spontaneous dissection of the pulmonary arterial walls with secondary thrombus formation in the walls was possible, and a diagnosis of mediastinal fibrosis was accepted.\nFour months after her first admission, the patient was electively rehospitalized to initiate specific treatment in June 2013. In view of her coronary artery disease without evidence of ischemia, the decision was made to start treatment with bosentan instead of sildenafil. On this admission, an echocardiogram showed no signs of pulmonary hypertension, and no enlargement of the right ventricle or paradoxical septal shift was detectable by an experienced examiner. The patient had not had any complaints for at least 2 months, and was started on bosentan without any problems.\nThe patient was evaluated 2 months later by Swan-Ganz catheterization of the right heart on full-dose bosentan 125 mg twice daily for 4 weeks. Again, precapillary pulmonary hypertension was reported and her PAR had dropped to 450 dyn · s · cm−5. Meanwhile, her spontaneous pO2 at rest had increased to 76 mmHg and her 6-minute walking distance was 360 m ().\nAs a proof-of-concept of our hypotheses, we interrupted her full-dose bosentan therapy for 2 weeks and again obtained invasive hemodynamic measurements via Swan-Ganz catheterization of the right heart. The results seemed to be only slightly different and obvious precapillary pulmonary hypertension was again demonstrated but, interestingly, her PAR was 400 dyn · s · cm−5, and therefore not elevated compared with the last measurement taken when the patient was on bosentan 125 mg twice daily.\nSeven and a half months after the initial diagnosis of idiopathic PAH with a PAR of 1,360 dyn · s · cm−5, we observed a reduction in PAR of at least two thirds without having established any specific treatment, except for low-dose radiation to the main stems of the pulmonary arteries ().

[[58.0, 'year']]

F

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{}

3972408-1

noncomm/PMC003xxxxxx/PMC3972408.xml

Diverse imaging characteristics of a mandibular intraosseous vascular lesion

A 30-year-old female patient presented with a painful swelling in the left mandibular anterior region. The swelling had been enlarging gradually and had been symptomatic for 2 months. No history of trauma preceded the swelling. The patient's physical examination revealed no abnormality, and the vital signs were within the normal range. Facial asymmetry due to a solitary diffuse swelling measuring about 3 cm×2 cm on the left side of the mandible was evident. Tenderness and a local rise in temperature were noted, and the swelling was soft in consistency. Intraorally, the oval-shaped swelling was 3 cm×3 cm in size, extending from the left mandibular canine to the distal side of the left mandibular second premolar. The overlying mucosa appeared to be slightly bluish with a smooth and shiny surface (). On palpation, the swelling was soft and fluctuant in the buccal aspect. The adjacent left mandibular canine and the first premolar revealed grade I mobility. The clinical differential diagnoses considered were dentigerous cyst, unicystic ameloblastoma, adenomatoid odontogenic tumour, central giant-cell granuloma, aneurysmal bone cyst, and intraosseous vascular malformation and/or intraosseous hemangioma.\nA routine radiographic examination was performed. The resulting panoramic radiograph revealed a well-defined multilocular radiolucency extending from the left mandibular canine to the left mandibular second premolar with the presence of small loculations and fine trabeculae (). The roots of the left mandibular premolars were displaced laterally and showed no resorption. The mandibular occlusal radiograph showed bone spicules radiating from the buccal margins of the lesion, resembling a "sunburst" appearance (). On the intraoral periapical radiograph, a radiolucent lesion with small loculations and a fine trabecular pattern, and having a honeycomb appearance, was clearly appreciable (). The radiographic differential diagnosis of a multilocular lesion of the jawbone with a honeycomb pattern includes ameloblastoma, odontogenic myxoma, central giant-cell granuloma, multiple myeloma, aneurysmal bone cyst, and fibrous dysplasia. The multicystic (solid) variant of ameloblastoma typically appears multiloculated with internal septations manifested as a honeycomb or soap-bubble appearance. Odontogenic myxoma might present itself as an expansile, multilocular lesion with a tennis-racket or honeycomb-like pattern. Central giant-cell granuloma might appear as a poorly defined unilocular radiolucency or multilocular radiolucency with scalloped borders and is characterized by wispy ill-defined trabeculation. An aneurysmal bone cyst presents itself as a ballooned-out multilocular radiolucency with a honeycomb or soap-bubble appearance. These lesions show tooth displacement more commonly than root resorption. In our case, the "sunburst" appearance helped differentiate among the abovementioned lesions, all of which had a multilocular honeycomb appearance. Osteosarcoma also produces a sunburst appearance in addition to many different periosteal reactions, including an onion-skin appearance and asymmetric broadening of the periodontal ligament space. Fibrous dysplasia might also present itself with indistinct borders and a ground-glass appearance, while multiple myeloma might present itself with multiple lesions rather than multilocular lesions restricted to one jawbone.\nNeedle aspiration was performed on the buccal aspect of the lesion in the second premolar region, and the process yielded 3 mL of frank blood (). The presence of blood, demonstrated by the large number of erythrocytes, was confirmed through cytology, and a strong likelihood of the existence of a vascular lesion was established. Aneurysmal bone cyst, intraosseous vascular malformation, or intraosseous hemangioma was considered in the differential diagnosis following needle aspiration.\nMagnetic resonance imaging (MRI) and computed tomographic angiography (CTA) with three-dimensional (3D) reconstruction were performed. The computed tomography (CT) images showed an expansile multiocular lesion with an indistinct periosteal reaction on the buccal cortex. Internal loculations were not clearly evident in the CT scan. The axial CT scan images showed a trabeculated lesion in the left hemi-mandible, with feeder vessels supplying the vascular lesion. A CT angiogram was obtained; it showed that the branches of the right external carotid and the facial artery were in close proximity to the lesion. However, the peripheral feeder vessel was not determined, and therefore, we considered the left inferior alveolar artery to be the feeder (). Three-dimensional CT angiogram images showed a vascular lesion and the peripheral feeder vessel. The CT angiogram did not reveal any areas of nidus formation or entangled focus of blood vessels, as normally noted in the case of vascular malformations ().\nThe T1-weighted MR images (T1WI) obtained using fat suppression (FS) showed a lesion with an iso-intense signal. Signal voids were not detected, suggesting a slow flow lesion. Internal loculations were noted in the coronal plane and the axial plane, suggesting multilocularity. The coronal T1WI () and the axial T1WI () showed an expansile low-intensity lesion in the marrow of the left hemi-mandible with a cortical break. The T2-weighted images (T2WI) showed hyperintense signals, thus confirming a vascular lesion (). Axial T2WI showed high signal intensity and an expansile lesion in the marrow of the left hemi-mandible with a cortical break. MRI revealed the fluid-fluid levels in the aneurysmal bone cyst in T1WI, thus helping to differentiate it from vascular malformation. Further, Gd-T1WI showed heterogeneous signal intensity in the osteosarcoma, which was absent in vascular malformations. A slow flow malformation in the Gd-T1WI might sometimes present a "bubbly" appearance, which was seen in aneurysmal bone cyst (ABC) as well.,\nThe lesion was excised, and on microscopic examination (), numerous small and a few dilated capillaries lined with plump endothelial cells were noted; a few areas showed active endothelial proliferation forming an indistinct vascular lamina. The final diagnosis was given as intraosseous vascular malformation of the left mandibular region based on the clinical, imaging and histological features. The 12-month follow-up did not reveal any recurrence.

[[30.0, 'year']]

F

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{'5268109-1': 1}

3972410-1

noncomm/PMC003xxxxxx/PMC3972410.xml

Glandular odontogenic cyst: A case report

A 17-year-old female patient was referred to the Outpatient Department of Oral Medicine and Radiology at Nair Hospital Dental College, Mumbai, by a local orthodontist after discovering a well-defined radiolucent pathology in the right ramal region of the mandible on a routine panoramic view.\nThe patient reported mild dull pain over the right posterior mandibular region for a period of 15 days with no aggravating or relieving factors. An extraoral examination revealed no significant clinical findings. Intraorally, there was mild obliteration of the buccal vestibule in the right lower second molar region with no other relevant clinical finding. The patient's medical history revealed that she had suffered from allergic asthma since childhood, she was not on any medication for the condition.\nA panoramic radiograph was obtained using a Planmeca Proline CC machine (Planmeca OY, Helsinki, Finland) with exposure parameters of 73 kVp, 10 mA, and 15 s exposure time. A well-defined multiloculated radiolucent lesion was observed extending from the distal border and apex of the lower right second molar into the ramus to approximately 3 mm away from the sigmoid notch enclosing the developing tooth bud of the right lower third molar (). There was no expansion of the posterior or inferior borders of the ramus, as seen on the panoramic radiograph.\nA cone-beam computed tomographic (CBCT) scan was obtained using a Kodak 9000 3D machine (Carestream Health Inc., Rochester, NY, USA), with a field-of-view (FOV) of 50 mm×37 mm, voxel size of 76.5 µm×76.5 µm×76.5 µm, and exposure time of 10.8 s. The captured images were reconstructed using a high-spatial-frequency reconstruction algorithm, and these images revealed a well-defined multiloculated radiolucent lesion in the right mandibular ramal region (). This lesion extended from the right lower second molar to the upper third of the ramus, with resorption of the apex of the right mandibular second molar having well-defined, sharp right-angled septa (). The radiolucency had scalloped borders and surrounded the developing third molar tooth bud (). There was perforation of the lingual cortical plate and thinning of the buccal cortical plate, as seen on the CBCT coronal section of the lesion ().\nBased on the clinical and radiographic findings, a provisional diagnosis of keratocystic odontogenic tumor due to minimal expansion with considerable bony involvement was proposed with a differential diagnosis of odontogenic myxoma due to the presence of right-angled straight septa, as seen on the reconstructed panoramic section in the CBCT image.\nThereafter, an incisional biopsy of the lesion along with enucleation of the third molar tooth bud was performed under antibiotic coverage and local anesthesia. The specimen was submitted for histopathological evaluation, which revealed a pseudo-stratified, ciliated columnar epithelial cystic lining covering mature fibrous connective tissue. Thus, the overall histopathological findings were suggestive of a GOC. Following these investigations, surgical exploration with curettage of the GOC of the hemimandible was performed under general anesthesia, and the specimen was sent for histopathological confirmation. The hematoxylin and eosin stained section showed cystic lumen lined by pseudo-stratified columnar epithelium with filiform extensions of the cytoplasm and mucous-secreting cells with intra-epithelial spherule formation, which is characteristic of GOCs, thus confirming the previous histopathological diagnosis of GOC ().\nAfter a 3-month follow-up of the patient, no complication or recurrence was reported ().

[[17.0, 'year']]

F

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{'2996005-1': 1, '4182361-1': 1, '7044353-1': 1, '4358051-1': 1}

3972411-1

noncomm/PMC003xxxxxx/PMC3972411.xml

Gold-wire artifacts on diagnostic radiographs: A case report

A 76-year-old woman was referred to the Department of Periodontology at the University Hospital, Catholic University Leuven, Belgium. The patient consulted the department for a preoperative implant rehabilitation follow-up. During the consultation, she reported a vague toothache in her right maxilla. The medical history of the patient revealed hypertension and hypothyroidism, two chronic conditions that were being controlled by medication. The patient could not remember any further surgical intervention, apart from oral implant placement.\nAn extra-oral examination of the head and neck showed no obvious abnormalities (). An intra-oral examination revealed the presence of several implants in the posterior areas of the mandible and the maxilla, and natural teeth in the front. Neither periodontitis nor peri-implantitis symptoms were observed. Digital intra-oral radiographs (Digora® PSP imaged with Minray®, Soredex, Tuusula, Finland) identified the subgingival caries on the canine and the first premolar. The intra-oral images showed irregular strings and curved lines of a dense radiopaque material (). These foreign bodies demonstrated typical dense metallic characteristics.\nAs part of the implant therapy, a panoramic image (Veraview 2D, J. Morita Corp., Kyoto, Japan) was taken to verify the health of the maxillary sinuses, the absence of bone pathology, and the presence of sufficient bone height. On the panoramic image, irregular strings and curved lines of a dense radiopaque material were observed. Lines were observed bilaterally on the posterior part of the mandible, maxilla, and ramus (). In order to plan the implant surgery, a three-dimensional (3D) Acuitomo® 170 CBCT (Morita Corp., Kyoto, Japan) was taken. This radiographic image confirmed the irregular strings of dense radiopaque lines, but now, they were clearly located in the soft tissues of the cheek, far away from the alveolar bone ().\nIn the differential diagnosis, various options were considered. A MEDLINE-PubMed search gave the following possible explanations for radiopaque marks in the lateral areas of the craniofacial area: synthetic hair braid extension artifacts,, multiple miliary osteoma cutis,,, tonsillolith, acupuncture needles, surgical clips, plastic surgery,,, traumatic insertions (accidental insertions), and calcified phleboliths associated with soft tissue haemangioma.,,\nThe medical history of the patient was re-explored. Now, the patient confirmed that she had undergone a kind of plastic surgery approximately 25 years ago. The physician could confirm that the patient had indeed undergone a face lifting procedure with gold-wire (thickness: 0.1 mm) in 1986.

[[76.0, 'year']]

F

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{'8589502-1': 1, '5468557-1': 1, '8497133-1': 1, '8497133-2': 1, '6145801-1': 1}

3972412-1

noncomm/PMC003xxxxxx/PMC3972412.xml

Osteosarcoma of the mandible: A case report with an early radiographic manifestation

A 40-year-old female reported with the chief complaint of swelling in the right side of the lower jaw for the previous 10 days. The patient's medical and dental history was unremarkable, and the patient did not seem to have any harmful habits.\nOn extraoral examination, there was a swelling in the right mandible with a normal color and texture of the overlying skin. Mouth opening was found to be normal. Paraesthesia was detected in the skin over the swelling. The right submandibular lymph node was of 2 cm in size, palpable, tender, and non-fixed.\nIntraoral examination revealed a nodular swelling in the right mandible extending from the mandibular canine to the mandibular first molar. It measured approximately 3 cm×5 cm. The overlying mucosa was eyrthematous in appearance and the borders were well defined, with a consistency varying from firm to hard. Tenderness was present on palpation. The teeth near the swelling were not mobile ().\nA panoramic radiograph revealed a periradicular widening of the periodontal ligament space and loss of lamina dura involving the right mandibular second premolar and the mesial and distal roots of the first molar with mild rarefaction of the inter-dental alveolar bone between the roots of the mandibular right first molar and second molar ().\nAn intraoral periapical radiograph revealed a loss of the lamina dura and periradicular bone loss involving the interdental bone between the mandibular right second premolar and first molar (). A shadow of the soft tissue mass on the right side and mild peripheral cortical bone reactive bone formation in the molar and premolar region were visible in the right mandibular lateral occlusal radiograph (). Correlating the clinical and radiographic features, a provisional diagnosis of osteosarcoma involving the jaw bone was made.\nIncisional biopsy of the lesion was performed, and the hematoxylin-eosin stained section showed highly cellular connective tissue stroma, predominantly composed of neoplastic spindle cells that exhibited pleomorphism and hyperchromatism. Abundant neoplastic osteoid formation with varying degrees of calcification was seen within the stroma. The neoplastic bone showed typical peripheral rim of malignant osteoblasts as well as entrapped pleomorphic, hyperchromatic cells. Mitotic figures were also seen in the stroma (). A histopathological diagnosis of osteosarcoma was made.\nBased on the report, segmental resection of the right body of the mandible was performed with wide surgical resection margins. The resected tumor specimen also exhibited similar histopathological findings consistent with the incisional biopsy report of osteosarcoma.

[[40.0, 'year']]

F

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{'6463331-1': 1, '3517821-1': 1}

3974696-1

noncomm/PMC003xxxxxx/PMC3974696.xml

Lupus nephritis flare in young patients: relapse or nonadherence to treatment?

A 23-year-old Spanish woman was diagnosed with SLE and LN in July 2004 when she was 15. Her disease manifested as: nephrotic syndrome (proteinuria 8 g/24 hours, serum albumin 1.5 g/dL); normal renal function (serum creatinine 0.7 mg/dL); microhematuria; high titers of antinuclear antibodies; and presence of lupus anticoagulant. Renal biopsy revealed class V LN (membranous, the Society of Nephrology/Renal Pathology Society Classification 2003) with neither vascular nor interstitial lesions. She was treated with oral prednisone (1 mg/kg/24 hours) and cyclosporine A (200 mg/24 hours) for 8 weeks, followed by tapering the doses of prednisone. Although partial remission was achieved, she developed several disturbing side effects, such as Cushingoid face and gingival hypertrophy, which affected adherence. She subsequently presented with symptomatic thrombocytopenia that resolved after resumption of prednisone and cyclosporine A at lower doses.\nOne year later, after she had achieved complete renal remission, mycophenolate mofetil (1,500 mg/24 hours) was begun to treat proteinuria (3 g/24 hours). The patient soon developed severe renal damage (relapse of nephrotic syndrome and increase in serum creatinine to 4 mg/dL) associated with anemia and thrombocytopenia. A second renal biopsy revealed a transformation to class IV LN (diffuse proliferative glomerulonephritis) with a high activity index (Austin Index, 18/24) and a low chronicity index (Austin Index, 4/12). Therefore, she received daily pulses of 6-methylprednisolone (500 mg intravenous ×3), prednisone (1 mg/kg/24 hours by mouth), and monthly pulses of cyclophosphamide (500 mg intravenous ×3). She also required hemodialysis, owing to the presence of anasarca and poor recovery of the renal function. Given the possibility of a refractory form of LN, she received four weekly doses of rituximab (375 mg/m2). In the following weeks, renal function improved, and dialysis was stopped. Prednisone was tapered, and mycophenolate mofetil was begun (1,500 mg/24 hours). After several months with a stable clinical picture and renal function (serum creatinine 1.4–1.6 mg/dL and proteinuria 1–1.5 g/24 hours), she experienced a new worsening of renal function with nephrotic proteinuria and was readmitted for treatment. On day 5 of admission, the blood levels of mycophenolic acid, the active metabolite of mycophenolate mofetil, were undetectable (<0.8 μg/mL). Although the patient (and her family) initially denied poor adherence, she eventually admitted that she had not taken her treatment correctly, since she was worried about her physical appearance and other troublesome gastrointestinal side effects. After resumption of prednisone and mycophenolate, renal function improved, and proteinuria decreased to previous levels. She was discharged after starting hydroxychloroquine and sent to a psychologist to improve adherence. Four years after the event, she has not had any further flares of SLE-related LN.

[[23.0, 'year']]

F

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{'3974696-2': 2}

3974696-2

noncomm/PMC003xxxxxx/PMC3974696.xml

Lupus nephritis flare in young patients: relapse or nonadherence to treatment?

A 19-year-old Spanish woman was diagnosed with SLE in January 2008 when she was 17. The disease manifested with pericarditis, hemolytic anemia, and high titers of antinuclear and anti-DNA antibodies. Four months later, she developed nephrotic syndrome with microhematuria and normal renal function. A renal biopsy revealed class IV LN. She was treated with daily pulses of 6-methylprednisolone (1,000 mg intravenous ×3), prednisone (1 mg/kg/24 hours by mouth), and monthly pulses of cyclophosphamide (500 mg intravenous ×6). She quickly achieved complete renal remission, and, after receiving the sixth pulse of cyclophosphamide, maintenance treatment was begun with mycophenolate mofetil (1,000 mg/24 hours). She subsequently complained that the mycophenolate produced gastrointestinal discomfort, so she reduced the dose on her own initiative. In fact, levels of serum mycophenolic acid were undetectable. She agreed to switch to mycophenolic acid (Myfortic®, Novartis, Basel, Switzerland), which resulted in better tolerance. The renal response was complete for 2 years, after which time she was admitted with fever, polyserositis, pancytopenia, and increasing proteinuria. She received three intravenous pulses of 6-methylprednisolone. Given her severe leukopenia (<200 neutrophils/μL), she received several doses of a leukocyte-stimulating agent (filgrastim, Neupogen®, Amgen Inc., Thousand Oaks, CA, USA) with a good response. After her neutrophil count recovered, pulses of cyclophosphamide were administered (six monthly doses), followed by mycophenolic acid. Three months later, she was hospitalized with abdominal vasculitis, increased proteinuria (2 g/24 hours), and impaired renal function (serum creatinine, 2.6 mg/dL). She received a further three pulses of 6-methylprednisolone and four weekly doses of rituximab (375 mg/m2). At first, we considered the possibility of refractory LN, and both she and her family claimed that she had taken all the medication correctly. However, after a detailed interview, the patient admitted that she had often missed taking medication because she was concerned about her physical appearance and weight gain. Indeed, she confessed to having lied as a result of family pressure and had decided to reduce her medication on her own initiative. After resuming her previous medications, starting hydroxychloroquine, and receiving psychological counseling, she was discharged and has been in clinical and analytical remission for 2 years.

[[19.0, 'year']]

F

{'28904681': 1, '17307754': 1, '19177526': 1, '12387811': 1, '18185905': 1, '18636020': 1, '22805617': 1, '16546058': 1, '17324970': 1, '22588588': 1, '22089460': 1, '20676388': 1, '20869188': 1, '20010545': 1, '30426342': 1, '17665465': 1, '23554031': 1, '24098003': 1, '26246906': 1, '15077269': 1, '16079372': 1, '20837569': 1, '20210680': 1, '15462484': 1, '24729723': 2}

{'3974696-1': 2}

3974961-1

noncomm/PMC003xxxxxx/PMC3974961.xml

A case of oxaliplatin-induced immune-mediated thrombocytopenia

A 53-year-old man with type 2 diabetes mellitus and hypertension was diagnosed with stage IIIB transverse colon cancer in February 2011. He underwent a right hemicolectomy and subsequently received 10 cycles of adjuvant chemotherapy with oxaliplatin and 24-hour infusions of fluorouracil (5FU) and leucovorin (FOLFOX). Before starting the 11th FOLFOX cycle, a complete blood count (CBC) showed the following values: neutrophils, 3.38×109/L, platelets, 113×109/L, and hemoglobin, 14.8 g/dL. During the 11th FOLFOX cycle, he experienced mild fever (37.6℃) and pain in his injected arm. Gum bleeding and petechiae on his legs also developed. The CBC showed: platelets, 3×109/L, neutrophils, 4.18×109/L, and hemoglobin, 13.8 g/dL. The patient's prothrombin time (PT, 10.5 seconds) and activated partial thromboplastin time (APTT, 25.5 seconds) were within normal range. There were no relevant abnormalities in total bilirubin, aminotransferase, blood urea nitrogen (BUN), creatinine and complement factors (C3 and C4). He was hospitalized and treated with platelet transfusion, immunoglobulin 55 g/day for 2 days and corticosteroids. His platelet count recovered to 83×109/L, and his gum bleeding and petechiae resolved (). The liver and spleen were not palpable. Human immunodeficiency virus testing, hepatitis B and C screening, viral marker of Epstein-Barr virus early antigen (EBV-EA) IgM and cytomegalovirus (CMV) antigen (Ag), antinuclear antibody analysis and thyroid function tests were all negative. The results of bone marrow aspiration and biopsy were unremarkable. Because all other causes of thrombocytopenia were excluded, we concluded that oxaliplatin was the only cause of his thrombocytopenia. He was discharged with oral prednisone, and his platelet count recovered completely without further treatment in 2 months.

[[53.0, 'year']]

M

{'16493620': 1, '15847658': 1, '11161414': 1, '17303980': 1, '16968538': 2, '32583132': 1, '20346128': 2, '30687065': 2, '22534771': 1, '31781443': 2, '20406923': 1, '10513718': 1, '24724069': 2}

{'2859393-1': 1, '2859393-2': 1, '1574347-1': 1, '6341312-1': 1, '6875012-1': 1}

3974963-1

noncomm/PMC003xxxxxx/PMC3974963.xml

A rare case of isolated myeloid sarcoma of the small gut with inv(16)(p13;q22) without bone marrow involvement

A 27-year-old woman presented with recurrent, colicky abdominal pain associated with occasional vomiting for 2 months. There was no history of fever, jaundice, hematemesis, or melena. She had been hospitalized several times and treated conservatively. The patient was conscious, alert, oriented, and afebrile, and her vitals were within normal limits. No pallor, edema, jaundice, clubbing, or superficial lymph node enlargement were present. Her abdomen was soft, non-tender, and not distended. Other systemic examinations revealed no abnormality. Contrast enhanced computerized tomography of the abdomen showed a soft tissue mass lesion () in the left side of the mesentery with adherent small gut loops.\nExploratory laparotomy was performed. There was moderate ascites without liver nodules or peritoneal deposits. There was an 8.0×7.0 cm globular mass with serosal involvement in the ileum, located 40 cm proximal to the ileo-cecal junction causing luminal obstruction and proximal dilatation, with no apparent mucosal involvement and thickening of the adjacent small bowel. The involved Ileal segment was resected with a 10 cm proximal and a 5 cm distal margin, and a side-to-side ileo-ileal anastomosis was performed. On gross examination of the specimen, there was a tumor, 6 cm in diameter, extending outwards into the mesentery and protruding into the intestinal lumen. The cut surface was grayish and fleshy. A total of 10 tiny lymph nodes were seen in the mesentery. Microscopically, the small intestine showed a tumor composed of sheets of atypical large cells with moderate to scanty cytoplasm, a high nucleo-cytoplasmic ratio, and occasional prominent nucleoli. A fair number of eosinophils were also observed (, ). The tumor cells infiltrated the muscle coat of the intestine and extended up to the serosa. A morphological diagnosis of high grade NHL, large cell type vs. granulocytic sarcoma were differential diagnoses. The surgical resection margins or the lymph nodes were not involved. On IHC analysis, the tumor cells were positive for the leukocyte common antigen, CD117, CD34, myeloperoxidase (MPO), and CD43 and negative for CD20, CD3, CD5, CD10, and CD23. Thus, a final diagnosis of GS (MS) of the small gut was made. A peripheral blood smear showed apparently normal WBC counts with normal differential and platelet counts. Bone marrow aspiration showed normocellular marrow with tri-lineage differentiation. Blast counts were not increased. Conventional cytogenetics showed a normal 46, XX chromosomal pattern. Subsequently, the marrow material was assessed for the presence of common translocations by multiplex RT-PCR and inv(16)(p13;q22); CBFB-MYH11 was detected.\nIn this case, the bone marrow did not show any morphological involvement; however, the detection of the molecular marker of inv(16)(p13;q22) fulfills the criteria of AML diagnosis as defined by the World Health Organization in 2008 []. Routine biochemical parameters including serum potassium, calcium, phosphorus, and magnesium were within the reference ranges. Viral markers were negative. After proper counseling, the patient was treated with standard "3+7" daunomycin and cytarabine as remission induction followed by 3 cycles of consolidation therapy with high dose cytarabine []. The clinical course of the patient was uneventful during chemotherapy except for 2 occasions of febrile neutropenia, which were managed successfully by antimicrobials. Post consolidation, inv(16)(p13;q22) was negative from bone marrow. Post chemotherapy, she is on regular follow up and doing well.

[[27.0, 'year']]

F

{'17170724': 1, '17090205': 1, '11920536': 1, '9737707': 1, '15119007': 1, '7602369': 1, '2423654': 1, '10231412': 1, '17169797': 1, '3465429': 1, '14615822': 1, '17446522': 1, '21795742': 1, '19433489': 1, '24724071': 2}

{}

3975172-1

noncomm/PMC003xxxxxx/PMC3975172.xml

The Expansion of RPE Atrophy after the Inverted ILM Flap Technique for a Chronic Large Macular Hole

A 79-year-old woman presented with a chronic large MH which remained open despite pars plana vitrectomy (PPV). The surgery for the MH closure was performed twice 14 years earlier. ILM peeling was not done during previous surgeries. The best-corrected visual acuity (BCVA) with the Landolt ring chart was 0.08 at her visit. The minimum MH diameter was 1,240 μm. Inverted ILM flap technique with 20% SF6 gas tamponade was performed for the MH closure. For the inverted ILM flap technique, 25-gauge PPV and ILM staining with indocyanine green (ICG) (0.25%) were used. The ILM was peeled off for 2 disc diameters around the MH, but it was not completely removed. The ILM was then inverted and covered the MH. One week after surgery, the MH was closed, accompanied by glial cell proliferation, spreading from the inverted ILM flap (as reported before) (fig. ) []. On the other hand, the area of the submacular RPE atrophy and gliosis on the surface of the retina, which were already observed 1 week after surgery, gradually increased in size (fig. ). BCVA improved to 0.3 six months after surgery.

[[79.0, 'year']]

F

{'28161828': 1, '31061725': 1, '9339035': 1, '11054324': 1, '19052655': 1, '24263468': 1, '19682624': 1, '26208719': 2, '15978253': 1, '32385224': 1, '32420232': 1, '32626601': 1, '29532170': 1, '27047686': 1, '23622567': 1, '15076960': 1, '31073163': 1, '33512612': 1, '32943839': 1, '29643791': 2, '31275632': 1, '31956583': 1, '30809480': 1, '20541263': 1, '24212316': 1, '24707278': 2}

{'4515012-1': 1, '5892329-1': 1}

3975174-1

noncomm/PMC003xxxxxx/PMC3975174.xml

Gastrointestinal and Extragastrointestinal Stromal Tumors: Report of Two Cases and Review of the Literature

An 82-year-old male patient diagnosed with pulmonary carcinoma underwent a lung and abdominal CT examination for preoperative staging, which showed a round homogeneous soft tissue mass of 4.8 × 5.5 cm in the stomach as an incidental finding (fig. ). Gastroscopy confirmed the presence of the mass. An endoscopic biopsy was taken and histology showed positive immunohistochemical staining for CD117, a defining feature of GIST in the stomach. The patient was scheduled for lung cancer resection, however he refused any medical treatment of the gastric lesion. For the next 4 years he had CT follow-up examinations for both malignancies, which demonstrated that the gastric mass remained unchanged, without any significant differences in the morphologic characteristics.

[[82.0, 'year']]

M

{'12640147': 1, '21830406': 1, '22355467': 1, '16549611': 1, '26021500': 1, '1540870': 1, '22783003': 1, '26998117': 1, '20137358': 1, '16293410': 1, '14753595': 1, '18235121': 1, '31649795': 1, '14605627': 1, '28185152': 1, '11213830': 1, '10636102': 1, '16402273': 1, '24707244': 2}

{'3975174-2': 2}

3975174-2

noncomm/PMC003xxxxxx/PMC3975174.xml

Gastrointestinal and Extragastrointestinal Stromal Tumors: Report of Two Cases and Review of the Literature

A 58-year-old female patient with a history of hypothyroidism, arterial hypertension and mitral regurgitation presented with left thoracic pain and fever up to 38.5°C during the last 3 days. Laboratory examination showed: WBC at the highest normal level (10,100) with elevated neutrophils count (81%), increased erythrocyte sedimentation rate of 53 and elevated cancer antigen 125 of 8,300. A lung and abdominal CT scan was performed and showed an inhomogeneous mass sized 5.8 cm located in the hepatogastric ligament, in contact with the left liver lobe (fig. ). The lesion demonstrated heterogeneity after intravenous contrast medium administration. Two small lesions of the liver compatible with hepatic metastases were also depicted. Moreover, the CT showed an extensive left pleural effusion and a myelolipoma of the left adrenal gland 3 cm in size. The patient underwent endoscopy, bronchoscopy and video-assisted thoracic surgery, which did not reveal any pathological findings. Fine needle aspiration and biopsy of the mass proved immunohistochemical features of a CD117-positive mesenchymal tumor of the hepatogastric ligament without connection to the gastrointestinal tract, compatible with an extragastrointestinal tumor. Because of the presence of hepatic metastases, conservative therapy with Gleevec was initially decided.

[[58.0, 'year']]

F

{'12640147': 1, '21830406': 1, '22355467': 1, '16549611': 1, '26021500': 1, '1540870': 1, '22783003': 1, '26998117': 1, '20137358': 1, '16293410': 1, '14753595': 1, '18235121': 1, '31649795': 1, '14605627': 1, '28185152': 1, '11213830': 1, '10636102': 1, '16402273': 1, '24707244': 2}

{'3975174-1': 2}

3975192-1

noncomm/PMC003xxxxxx/PMC3975192.xml

A Case of Acute Generalised Exanthematous Pustulosis after Unprotected Sexual Intercourse on Holidays in Bali

A 43-year-old man presented to a rural hospital in Australia for lethargy, fever, arthralgia, chills, peri-orbital oedema and an erythematous pustular rash increasing in size over his hands, back, chest and face for approximately 48 h. He was a fly-in fly-out worker in the catchment area of the hospital. He mentioned holidaying in Bali, Indonesia, for 9 days to visit a female partner with whom he had unprotected sexual intercourse. The symptoms started promptly upon his return. All in all, he had visited his partner five times this calendar year. He reported to have contracted genital herpes from this partner on his first visit and had sought treatment with antivirals (acyclovir) upon his return to Australia and had remained on them ever since. He also mentioned that both he and his partner had been monogamous since the day they met each other. Furthermore, he described some recent coryzal symptoms towards the end of his holiday, for which he bought some local cold and flu medication, the name and ingredients of which being unknown but being somewhat ‘like Sudafed®’. He also took some Nurofen® tablets (ibuprofen) in Bali and upon returning to Australia. Apart from cystic acne 25 years ago and intermittently taking ibuprofen, the patient reported no significant past medical history.\nAt this rural hospital, he continued to decline systemically (fever >39.5°C, heart rate >125 bpm) over 36 h and was flown to a tertiary referral hospital in Brisbane. On arrival, he had widespread pustules on his face, neck, back, chest, upper and lower limbs (fig. , fig. , fig. ). It was noted that he was also erythematous and had an oedematous face. He described only a mild itch and denied any genitourinary symptoms. Some inguinal and cervical lymphadenopathy and minimal oral involvement were observed. He had a raised white blood cell count (13.2), neutrophilia (12.17), a mild lymphocytosis (0.56), a raised C-reactive protein level (273), and was started immediately on intravenous piperacillin/tazobactam three times daily, intravenous vancomycin twice daily and intravenous acyclovir three times daily with a suspected but yet unknown infection.\nHe had negative serologies over the initial week for the following infections: streptococcus, syphilis, Ross River virus, Barmah Forest virus, alphavirus, Rickettsia rickettsii, measles, mumps, leptospirosis, parvovirus B19, enterovirus, varicella and HIV.\nHistological examination of biopsies revealed conflicting features. A biopsy of the hand showed evidence of spongiotic dermatitis with eosinophilic dermal infiltrates and occasional intra-epidermal vesicles harbouring Langerhans cells. A second specimen of the abdomen revealed acute neutrophilic suppurative folliculitis. Direct immunofluorescence of the abdomen biopsy was negative. These results were considered inconsistent with the clinical impression of AGEP held at that time. Further biopsies were then taken from his chest and left thigh, which showed spongiform subcorneal pustules accompanied by dermal inflammatory infiltrates and perivascular infiltrates with occasional eosinophils reported as ‘acute neutrophilic folliculitis’. Acanthosis and papillomatosis were not observed. A biopsy of the right foot favoured a spongiotic hypersensitivity reaction with a vasculitic component.\nClinically, his symptoms appeared to indicate AGEP at this stage, and given that serologies up until this point were negative, he was started on oral prednisone 0.5 mg/kg and topical betamethasone dipropionate 0.05% ointment twice daily, with a very good effect over the proceeding 5 days. It was only on day 6 of this treatment that, with the help of the infectious diseases team, a positive scrub typhus (Orientia tsutsugamushi) Igtotal serology was confirmed, with a titre of 1:128. This was not considered a primary infection, and paired tests 1 and 4 weeks later showed that the titre was stable and that this was likely an old rickettsial infection or a cross-reaction. This was also paired with scrub typhus and spotted fever PCR testing, which was also negative. Prednisone was weaned down to half the dose over the following 5 days, and he was discharged home on day 13 of admission (significantly improved on emollients and weaning dose of prednisone). Post-pustular desquamation was observed around this time. An outpatient review 12 days following discharge revealed a slight flare-up again at home after taking ibuprofen tablets for 48 h for aches and pain. However, this resolved promptly with the cessation of the non-steroidal anti-inflammatory drug.

[[43.0, 'year']]

M

{'1832534': 1, '13679697': 1, '11168761': 1, '9069592': 1, '8354488': 1, '20542841': 1, '24707246': 2}

{}

3975196-1

noncomm/PMC003xxxxxx/PMC3975196.xml

Keratitis with Kocuria palustris and Rothia mucilaginosa in Vitamin A Deficiency

A 58-year-old African-American female presented to a hospital emergency room with a superotemporal marginal corneal ulcer of the left eye two clock hours in length, with mild corneal thinning. Uncorrected visual acuity on the initial exam was 20/30 in the right eye and 20/50 in the left. Both eyes showed diffuse punctate corneal epitheliopathy in the palpebral fissure. Her past medical history included alcohol use since the age of 14, serious alcoholism since the age of 34, and chronic pancreatitis and diarrhea since the age of 52. She reported that she drank 6–12 beers per day. She had a smoking history of >50 pack-years as well as a history of gastrointestinal bleed, liver cirrhosis, cholecystectomy, chronic obstructive pulmonary disease, and anaphylactic reaction to peanuts. She had lived in urban USA since birth.\nAfter corneal culture, the patient was started on hourly topical moxifloxacin 0.5% eyedrops in the left eye as well as artificial tears in both eyes. Culture was positive for alpha-hemolytic streptococcus and catalase-positive Gram-positive cocci in groups with preliminary identification as coagulase-negative Staphylococcus. She returned 3 days later, with an unchanged exam except for mild injection of the right eye, and was maintained on the same antibiotic drops but with an increase in artificial tears in the right eye. Three days after that (6 days after the initial visit), she presented to an outpatient office with bilateral marginal corneal ulcers (fig. , fig. , fig. ), both with a thinning >50% and parallel to the limbus. The left eye also had a small hypopyon. Uncorrected visual acuity was 20/40 right, 20/80 left. She did not complete workup or treatment and was lost to follow up for over 1 week. She returned to the emergency room 17 days after initial presentation, with a complaint of bilateral severe vision decrease and painful swelling of the left lower lid of 2 days duration. She was hospitalized at that point.\nOn admission, vision was hand motions in both eyes. The right eye had deep marginal thinning for 6 clock hours inferiorly. The left cornea had a marginal area of deep stromal thinning temporally for 5 clock hours, and the inferior lid showed tender erythema and edema. Diffuse severe bilateral corneal edema and hypopyon (1.5 mm in the right eye and 1.0 mm in the left eye) were present. B-scan ultrasound of the fundus of both eyes was unremarkable.\nAfter corneal culture, the patient was placed on bilateral topical ocular application of fortified vancomycin eyedrops (25 mg/ml) every hour (for Gram-positive coverage) and moxifloxacin 0.5% eyedrops every hour (for Gram-negative coverage). Cultures from admission grew Pseudomonas aeruginosa and Staphylococcus hominis from the right eye and Staphylococcus warneri and Staphylococcus lugdunensis from the left eye. On the third day of hospitalization, when culture results were available, the eyedrops were changed to fortified vancomycin every 2 h and fortified tobramycin (14 mg/ml) every 2 h. Tobramycin was started because of its potency against Pseudomonas. The presence of three Staphylococcus species in the cultures warranted continuation of the vancomycin eyedrops, but we decreased the frequency to every 2 h around the clock to reduce epithelial toxicity and patient stress. The left lower lid cellulitis did not yield any material for culture, but the patient received IV cefazolin (1 g every 8 h) for 2 weeks with resolution of the cellulitis. Due to the severe corneal ulceration, bilateral amniotic membrane grafts (AmbioDisk; IOP Ophthalmics, Costa Mesa, Calif., USA) with an overlying 16.0-mm Kontur bandage contact lens (Kontur Kontacts, Hercules, Calif., USA) were placed on the fourth day of hospitalization. A urinary tract infection found during hospitalization was culture-positive for vancomycin-resistant Enterococcus and was treated with linezolid.\nTesting revealed a marked vitamin A deficiency, with a level of 0 μg/dl (reference range 18–77). Vitamin A analysis was performed by LabCorp using high performance liquid chromatography. The patient had mild protein malnutrition with total protein 6.5 g/dl (reference range 6.6–8.7). Her BMI was 21.4 (normal range 18.5–25). Laboratory analysis showed hypokalemia, hyponatremia, hypomagnesemia, and hypocalcemia. Testing ruled out HIV, tuberculosis, syphilis, sarcoidosis, rheumatoid arthritis, hepatitis A, B, C, and Wegener granulomatosis. The ANA titer was elevated (40) with a fine speckled pattern, but not to the level of significance (80). Sjogren syndrome testing for SS-A/Ro, SS-B/La, Smith antibody, and ribonucleoprotein antibodies were all negative by microparticle multiple immunoassay. Erythrocyte sedimentation rate and C-reactive protein were normal.\nIntramuscular vitamin A was not available, so it was repleted with 100,000 IU orally for 3 days, followed by 50,000 IU orally every day for 2 weeks. After initial treatment of the infections and after ruling out systemic tuberculosis, systemic steroids were given (IV methylprednisone starting at 50 mg daily and tapered over 2 weeks). The patient also received thiamine, folate, ascorbate, calcium, vitamin D, multivitamins, topical cyclosporine 0.05% eyedrops, and intensive eye lubrication. The patient's diarrhea, pancreatic enzyme deficiencies, electrolyte abnormalities, and protein malnutrition were treated, and she received extensive nutritional and alcoholism counseling.

[[58.0, 'year']]

F

{'21295817': 1, '27630804': 1, '33927037': 1, '27103973': 2, '9520204': 1, '670378': 1, '27125478': 1, '29063496': 1, '21153041': 1, '21566368': 1, '8011854': 1, '28138334': 2, '16045134': 1, '28914002': 1, '10028258': 1, '22525781': 1, '19602923': 1, '18614658': 1, '22087104': 1, '9827277': 1, '26472837': 1, '23380989': 1, '2310332': 1, '24707276': 2}

{'5264429-1': 1, '4815942-1': 1}

3975198-1

noncomm/PMC003xxxxxx/PMC3975198.xml

Blue Nevus with a Dermoscopic Appearance of Peripheral Streaks with Branches

A 46-year-old Japanese woman came to us with an asymptomatic pigmented plaque on her left arm. The patient did not remember its onset. She was otherwise healthy. A physical examination revealed an asymptomatic blue-blackish plaque 2 mm in size on the left upper extremity (fig. ). Dermoscopy showed a central homogeneous dark blue-black pigmentation and peripheral blue-brownish streaks with branches (arrow) (fig. ). We performed a whole excision as biopsy, because we suspected the plaque to be a malignant melanoma. The specimen from the central lesion showed spindle cells with melanin pigments in the upper dermis, but not in the epidermis (fig. ). A deep section of the peripheral lesion identified focally aggregated and isolated spindle cells with melanin pigments in the upper dermis (fig. ).\nWith the deep sections, we confirmed that the dermal dendritic melanocytes were distributed in the direction of the streaks. Using dermoscopy, we confirmed peripheral branching streaks and diagnosed the blue-blackish plaque as blue nevus with histopathologic assessment.

[[46.0, 'year']]

F

{'18462445': 1, '24163656': 2, '22030018': 1, '23785621': 1, '16301385': 1, '21175749': 1, '31839774': 1, '24707253': 2}

{'3806697-1': 1}

3975199-1

noncomm/PMC003xxxxxx/PMC3975199.xml

Traumatic Maculopathy 6 Months after Injury: A Clinical Case Report

A previously healthy, 12-year-old male presented for a routine visit with complaints of a 2-month history of decreased visual acuity in his left eye (LE). Six months before the visit, he suffered blunt trauma to the LE during a struggle, with no medical observation. At the initial visit, BCVA in the LE was counting fingers and in the right eye, it was 20/20. He had neither restricted eye movements nor afferent pupillary defects. Biomicroscopic examination of the anterior segment was normal. Fundus examination of the LE showed a central macular lesion of 1 disc diameter with fibrosis, increased retinal thickness and intraretinal hemorrhage (fig. , left). Peripheral retinal examination was normal. Spectralis OCT (Heidelberg Engineering Inc., Heidelberg, Germany) showed disruption of the IS/OS junction with corresponding increased reflectivity, loss of the outer nuclear layer, cell infiltration of the retinal wall and subretinal fibrosis. Retinal thickness was 289 µm at the site of the lesion (fig. , right). A fluorescein angiography (FA) revealed early impregnation with no diffusion, increasing with time (fig. ). With support from Pediatrics, off-label, high-steroid pulse therapy (intravenous methylprednisolone 500 mg for 3 days and oral prednisolone 30 mg, tapering for 10 days) was done, resulting in an increase of LE BCVA to 20/200 and a decrease of retinal thickness by 71 µm to 218 µm 1 week after treatment. In an attempt to achieve additional improvement without undesired systemic effects, off-label intravitreal triamcinolone acetonide (IVTA; 0.05 ml/2 mg) was administered 2 weeks after oral treatment. LE BCVA improved to 20/150, and retinal thickness decreased by 10 µm 3 weeks after IVTA. Three months after initial presentation, LE BCVA was 20/125 and retinal thickness was 208 µm. However, the alterations in fundoscopy and OCT remained (fig. ).

[[12.0, 'year']]

M

{'23618228': 1, '19041478': 1, '23972302': 1, '26060831': 1, '24019789': 2, '286456': 1, '22105503': 1, '33976675': 2, '4820984': 1, '21642901': 1, '24707277': 2}

{'8077660-1': 1, '3764958-1': 1}

3975200-1

noncomm/PMC003xxxxxx/PMC3975200.xml

Vitreous Hemorrhage Caused by Ruptured Retinal Macroaneurysm

A 58-year-old woman had a sudden appearance of floaters in her right eye. She was neither hypertensive nor diabetic. At the initial visit, her BCVA was −0.18 logMAR units in the right eye and −0.20 logMAR units in the left eye. The slit-lamp findings were unremarkable in both eyes. Fundus examination revealed a ruptured retinal arterial macroaneurysm at the optic disc and a small VH (fig. ). The OCT image did not show a subretinal hemorrhage and edema around the macroaneurysm. Only hemostatics (kalliginogenase) and circulatory agents (carbazochrome sodium sulfonate hydrate) were prescribed because no retinal detachment was observed by B-scan ultrasonography. Three months later, her BCVA remained at −0.18 logMAR units, and the size of the ruptured macroaneurysm decreased. Six months later, she had no recurrences and her BCVA remained at −0.18 logMAR units.

[[58.0, 'year']]

F

{'18562843': 1, '7862424': 1, '16882398': 1, '7781479': 1, '3055391': 1, '12831156': 1, '7182954': 1, '19516118': 1, '3689733': 1, '20865922': 1, '18071732': 1, '16187050': 1, '16138114': 1, '15580007': 1, '24707271': 2}

{'3975200-2': 2}

3975200-2

noncomm/PMC003xxxxxx/PMC3975200.xml

Vitreous Hemorrhage Caused by Ruptured Retinal Macroaneurysm

An 82-year-old woman had a sudden decrease of vision in her left eye. She had hypertension and was taking antihypertensive medication. At the initial examination, her BCVA was 2.0 logMAR units in the left eye. The results of the slit-lamp examination were unremarkable in both eyes. Her fundus could not be observed because of the massive VH; however, B-scan ultrasonography did not show a retinal detachment. One month after medicinal treatment, the VH was not resorbed and vitrectomy was performed. After the hemorrhage was removed, a ruptured macroaneurysm with subretinal hemorrhage was detected within the vascular arcade (fig. ). After the surgery, the macroaneurysm became atrophic, and the subretinal hemorrhage gradually decreased. The OCT images showed a subretinal hemorrhage around the ruptured macroaneurysm. Three months later, the BCVA improved to −0.18 logMAR units and the macroaneurysm was atrophic. The size of the subretinal hemorrhage near the macroaneurysm was reduced. Six months later, she has had no recurrences, and the BCVA remained at −0.18 logMAR units.

[[82.0, 'year']]

F

{'18562843': 1, '7862424': 1, '16882398': 1, '7781479': 1, '3055391': 1, '12831156': 1, '7182954': 1, '19516118': 1, '3689733': 1, '20865922': 1, '18071732': 1, '16187050': 1, '16138114': 1, '15580007': 1, '24707271': 2}

{'3975200-1': 2}

3975201-1

noncomm/PMC003xxxxxx/PMC3975201.xml

Purpura of the Face and Neck: An Atypical Clinical Presentation Revealing a Hepatosplenic T Cell Lymphoma

A 62-year-old woman presented to our department with an asymptomatic cutaneous eruption of the face extending to the neck that had appeared 2 weeks earlier. She did not report any infectious symptoms in the past weeks. Fever, night sweats or weight loss were denied. There was neither anamnestical atopy nor known allergies. Medication was limited to estradiol (substitution therapy) and occasional ibuprofen intake. Physical examination revealed non-palpable petechial purpura distributed on the face and extending to the neck (fig. ). Peripheral white blood cell count, repartition and hemoglobin concentration were in the normal range, while platelet count was decreased to 90 G/l. Coagulation tests (aPTT, prothrombin time, fibrinogen) showed no anomaly. Lactate dehydrogenase, aspartate aminotransferase, and alanine aminotransferase were slightly elevated (LDH 316 U/l, ASAT 58 U/l, ALAT 52 U/l); alkaline phosphatase, gamma-glutamyl transferase, total bilirubin and serum creatinine concentrations were all within normal limits. Protein electrophoresis and immune subtraction allowed ruling out a monoclonal gammopathy. Erythrocyte sedimentation rate and serum complement concentrations were within normal limits; antinuclear antibodies, ANCAs (c-ANCA, p-ANCA and x-ANCA) as well as serologies for HIV, HBV, HCV, CMV, EBV and PVB19 were all negative. Histopathological examination of the purpuric macules showed extravasation of erythrocytes and a slight lymphohistiocytic perivascular inflammation in the superficial and mid dermis, without evidence of vasculitis (fig. ). Amplification of the TCR in the skin biopsy showed no monoclonality. Direct immunofluorescence was negative. Given the particular distribution of the purpura in the upper part of the body, a cervico-thoracoabdominal CT scan was performed, which allowed to rule out a compressive component on the venous flow. It showed a multinodular goiter as well as a slight splenomegaly (12 cm). At that time, the patient was released with a diagnosis of idiopathic thrombocytopenic purpura.\nTwo months later, the patient complained of night sweats and the purpuric eruption had extended to the whole body (fig. ). Physical examination revealed a hepatosplenomegaly without palpable adenopathy. Thrombocytopenia had progressed to 30 G/l, LDH was 921 U/l, ALAT 179 U/l, ASAT 236 U/l, alkaline phosphatase 156 U/l, gamma-glutamyl transferase 96 U/l and total bilirubin was 31 μmol/l. Other laboratory examinations were within normal limits. A thoracoabdominal CT scan showed progression of the splenomegaly (from 12 to 16 cm). The examination of the bone marrow biopsy and aspirate showed an atypical lymphoid infiltrate composed of medium-sized cells with oval to irregular nuclei, moderately abundant cytoplasms, featuring a predominantly sinusoidal distribution. Immunophenotyping (immunohistochemistry and flow cytometry) revealed atypical lymphoid cells which were CD2+/CD3+/CD4–/CD5–/CD7–/CD8–/CD30– and focal expression of the T cell-restricted intracellular antigen-1 (TIA1). T cells strongly expressed the gamma/delta TCR and were negative for TCR beta F1. A monoclonal rearrangement of the TCR-gamma gene was demonstrated by PCR. We retained a diagnosis of HSTL and CHOP chemotherapy was administered together with a prophylactic intrathecal mixed chemotherapy (cytarabine, methotrexate, hydrocortisone). After two courses of treatment, the patient reported a diminution of night sweats, although the thoracoabdominal CT scan showed no modification, in particular no change in hepato- and splenomegaly; clinically, the purpuric rash showed a dramatic regression paralleled by a normalization of the platelet count (platelet count 157 G/l). After six courses of chemotherapy, as the infiltration of the bone marrow by neoplastic T cells persisted, the patient underwent a well-tolerated autologous bone marrow graft. Two months later, no recurrence of the purpura was observed. A close clinical, biological and radiological follow-up for early detection of a potential HSTL recurrence is currently ongoing at the oncological unit.

[[62.0, 'year']]

F

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{}

3975202-1

noncomm/PMC003xxxxxx/PMC3975202.xml

Combination of Nasolabial V-Y Advancement Flap and Glabellar Subcutaneous Pedicled Flap for Reconstruction of Medial Canthal Defect

A 77-year-old woman presented with a 1-year history of a black tumor in the right medial canthal region. The tumor measured 3 × 5 mm at the first examination. An incisional biopsy revealed that the tumor was a basal cell carcinoma.\nWe excised the tumor with a 4-mm safety margin (fig. ). The defect included the areas superior and inferior to the MCT and was adjacent to the medial eyelid commissure (fig. ). We planned to separately reconstruct the upper and lower parts of the defect using a nasolabial V-Y advancement flap and a glabellar subcutaneous pedicled flap (fig. ). We first advanced the nasolabial V-Y flap from the inferior aspect to create an elliptic defect in the upper part and fixed it to the dermis using 6-0 PDS® II sutures (Johnson & Johnson K.K., Tokyo, Japan) (fig. ). We then created a glabellar subcutaneous pedicled flap to match the residual upper elliptic defect with the major axis set along a relaxed skin tension line (fig. ). A subcutaneous tunnel was made between the flap and the defect, and the flap was passed through the tunnel to the upper residual defect (fig. ). The resultant defect in the glabella was closed in a dermostitch fashion with 6-0 PDS® II (fig. ). Each flap was anchored to the MCT to create a concavity in the medial canthal region. The skin was sutured with interrupted 6-0 polyvinylidene fluoride sutures (Asflex®; Kono Seisakusho Co., Ltd., Tokyo, Japan) (fig. ).\nThe excised tissue margin was histopathologically free of tumor cells. At 6 months postoperatively, no tumor recurrence or deformity was evident, and only an inconspicuous scar was present (fig. ).

[[77.0, 'year']]

F

{'16401793': 1, '20348864': 1, '11316716': 1, '27942380': 1, '9783288': 1, '24707272': 2}

{}

3975203-1

noncomm/PMC003xxxxxx/PMC3975203.xml

Certolizumab-Induced Uveitis: A Case Report and Review of the Literature

A 64-year-old woman was referred to our clinic due to bilateral reduced visual acuity (VA), mainly in her left eye. The patient's medical history was significant for seropositive RA, which had been well controlled for the previous 3 years under treatment with methotrexate and certolizumab (Cimzia). The patient had no previous ocular history.\nThe patient complained of a mild reduction in VA in both eyes during the previous year and noticed a significant reduction in her left eye (which occurred 1 week prior to her presentation). Initial examination revealed a VA of 20/32 in the right eye and 20/100 in the left eye. Anterior segment examination revealed no corneal keratic precipitates, mild (+1) flare in the anterior chambers and posterior synechiae bilaterally (fig. ). Dilated fundus examination revealed vitreal cells and haze, which were present bilaterally, but more distinct in the left eye. Macular edema (fig. ) and peripheral retinal punched-out lesions were visible (fig. ). Intraocular pressure (IOP) was 21 mm Hg in the right eye and 30 mm Hg in the left eye.\nThe integration of the clinical signs at presentation led to a working diagnosis of ocular sarcoidosis, possibly associated with anti-TNFα treatment. Workup included normal complete blood count, chemistry and C-reactive protein. Angiotensin-converting enzyme (ACE) levels were mildly elevated (61 U/l, normal range 10–51 U/l). Physical examinations and chest X-rays were normal, and so was the pulmonary function test. The patient was also examined by an expert pulmonologist who ruled out any lung disease.\nShe was treated with topical steroid drops as well as anti-glaucoma medication. By doing so, an improvement of the anterior uveitis and a normalization of the IOP were achieved. In coordination with the rheumatologist, the certolizumab was discontinued and the methotrexate dosage was increased. The topical steroidal treatment was tapered down and discontinued. Two months after the discontinuation of certolizumab, the uveitis had decreased and VA had improved to 20/32 in both eyes. Nevertheless, the macular edema persisted and the patient underwent periocular injections of 40 mg triamcinolone acetonide (Triesence; Alcon Inc.) in both eyes, with a partial resolution of the macular edema (fig. ).\nOne year after her initial presentation, the patient's RA is well controlled, and the change in systemic treatment did not result in any exacerbation. Her VA remains 20/32 in both eyes, and IOP is 17 mm Hg in both eyes (under topical treatment). The macular edema has improved, but still remains significant and is likely the cause of her impaired VA (fig. ).

[[64.0, 'year']]

F

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{'8365578-1': 1}

3975204-1

noncomm/PMC003xxxxxx/PMC3975204.xml

Transcatheter Arterial Embolization for Shock Caused by Intratumoral Hemorrhaging in Neurofibromatosis Type 1: A Report of Two Cases

A 77-year-old man fell off his bicycle and bruised the back of his head. He was examined at a local hospital. A computed tomography (CT) scan revealed a hemorrhage in the occipital region so he was admitted to the hospital, where he went into shock and was transferred to our hospital 9 h after the injury.\nOn examination, he was found to be alert, his blood pressure was 91/54 mm Hg, his pulse rate was 126 beats/min, and his oxygen saturation level was 100% whilst breathing through a face mask (5 liter/min). Anemia of the palpebral conjunctiva subsequently developed. Severe swelling and tenderness were observed from the left occipital region to the left side of the neck (fig. ). Laboratory analysis revealed a blood hemoglobin level of 6.2 g/dl, but this recovered to 10.0 g/dl after the transfusion of 14 units of erythrocytes. We immediately performed a contrast-enhanced CT scan. However, during the scan a mass in the patient's neck suddenly began to swell and obstruct his airway. He underwent emergency intubation. A second contrast-enhanced CT scan revealed an intratumoral hemorrhage, extending from the lower jaw to the left side of the neck and the displacement of the trachea to the right (fig. ). Thus, he immediately underwent transcatheter arterial embolization (TAE). A left vertebral artery angiogram demonstrated that the branches of the vertebral artery extending into the rear of the neck had expanded and proliferated markedly, and extravasation of contrast material was detected in the peripheral branches (fig. ). We superselectively embolized the artery using an infusion catheter before injecting gelatin sponge. A subsequent left subclavian artery angiogram depicted extravasation of contrast material in the peripheral branches of the transverse cervical artery. We embolized the artery by injecting an emulsion of n-butyl-2-cyanoacrylate and iodized oil at a ratio of 1:4 (Histoacryl/Lipiodol) (fig. ).\nThe TAE was successful, and the hemorrhage gradually reduced in size. Since the narrowing of the patient's airway improved, the patient was extubated at 8 days after admission. Subsequently, the hemorrhagic region exhibited autolysis, and the ulcers on the back of the patient's head and left shoulder were drained. He underwent a free skin grafting at 21 days after admission. The patient's wounds completely healed, and he was discharged at 41 days after admission (fig. ).

[[77.0, 'year']]

M

{'11283797': 1, '4212953': 1, '23323035': 2, '22531095': 2, '9227730': 1, '4621893': 1, '20160346': 1, '17551167': 1, '17681709': 1, '20566100': 1, '24707252': 2}

{'3975204-2': 2, '3423040-1': 1, '3542307-1': 1}

3975204-2

noncomm/PMC003xxxxxx/PMC3975204.xml

Transcatheter Arterial Embolization for Shock Caused by Intratumoral Hemorrhaging in Neurofibromatosis Type 1: A Report of Two Cases

A 33-year-old woman developed a huge pseudoneuroma in her right thigh; she had undergone six tumor resection operations and an above-the-knee amputation. Suddenly, she complained of pain in her right thigh. Her blood pressure was 89/77 mm Hg, and her pulse rate was 128 beats/min. Severe pain and swelling developed in her right thigh (fig. ). Her blood pressure was immediately increased by fluid administration. A contrast-enhanced CT imaging showed an aneurysm, which had ruptured, resulting in a huge intratumoral hemorrhage (fig. ). We immediately performed TAE, and extravasation of contrast material was detected in the peripheral branches of the lateral circumflex femoral artery (fig. ). We embolized the contrast material leakage sites by injecting an emulsion of n-butyl-2-cyanoacrylate and iodized oil at a ratio of 1:4 (Histoacryl/Lipiodol). After the TAE had been completed, the patient's blood hemoglobin level fell to 6.2 g/dl, but it was successfully restored via the transfusion of two units of erythrocytes. The patient was discharged without complications.

[[33.0, 'year']]

F

{'11283797': 1, '4212953': 1, '23323035': 2, '22531095': 2, '9227730': 1, '4621893': 1, '20160346': 1, '17551167': 1, '17681709': 1, '20566100': 1, '24707252': 2}

{'3975204-1': 2, '3423040-1': 1, '3542307-1': 1}

3975205-1

noncomm/PMC003xxxxxx/PMC3975205.xml

Abdominal Inflammatory Myofibroblastic Tumor

A 28-year-old woman was referred to our hospital because of abdominal pain and weight loss. During 4 weeks she had lost 15 kg in weight because of postprandial pain. She had pre-existent constipation, but no rectal bleeding, nor did she experience any night sweating. Physical examination did not show any anomalies except a left paraumbilical palpable mass. Ultrasound revealed a 6-cm hypoechogenic tumorous mass with sharp margins. A CT scan of the chest and abdomen showed an inhomogeneous round tumor of 73 × 61 mm with sharp margins and with hypodense fatty components in combination with enhancing soft tissue. The mass was located mesenterially, ventrally of the musculus iliopsoas with surrounding enlarged lymph nodes (maximum diameter 12 mm) (fig. ). Differential diagnoses like teratoma, liposarcoma or IMT were suggested. An additional PET scan revealed paratracheal lymph nodes which were interpreted as reactive, and the known left paraumbilical mass with only capsular FDG uptake. Ultrasound-guided biopsies were taken which revealed spindle cell proliferation with a mixed inflammatory cell infiltrate, in which immunohistochemical analysis showed positivity for vimentin, pankeratin (not uniform), and focally smooth muscle actin and calponin, suggesting an IMT.\nGiven the clinical presentation and the pathological findings, it was decided to perform an explorative laparotomy with total resection of the tumor. Macroscopic inspection showed a tumor with a diameter of 10 cm with a smooth capsular outer surface (fig. ). Due to locally aggressive growth, both a section of the colon and the jejunum had to be resected. Microscopy showed a mesenteric tumor comprising diffuse, not sharply delineated and unencapsulated spindle cell proliferation with lymph node involvement per continuitatem but without involvement of the colon or jejunum and without atypia. This spindle cell proliferation was accompanied by a cell-rich mixed inflammatory infiltrate with for instance many plasma cells, lymphocytes and eosinophilic granulocytes. The tumor contained centrally a large area of necrotic debris, maybe fat necrosis. Immunohistochemical analysis did not reveal signs of a specific spindle cell tumor, carcinoma or malignant lymphoma (fig. ). The diagnosis was IMT (differential diagnosis: primary fat necrosis surrounded by a pseudocapsule of extensive reactive fibrosis with organization). The postoperative period was uncomplicated and the patient could be discharged from the hospital after 4 days.

[[28.0, 'year']]

F

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{'2915987-1': 1}

3975206-1

noncomm/PMC003xxxxxx/PMC3975206.xml

A Case of Malignant Melanoma with In-Transit Metastasis That Responded to Intravenous Infusion of Interferon-β

The patient was a 77-year-old male with rash on the fifth toe of the left foot and lower left leg. His medical history included hypertension, diabetes, and malignant melanoma that was surgically removed (fifth toe amputation) at our department 14 years ago (fig. ). Otherwise, his family history was unremarkable. Three months before presentation, a skin ulcer appeared at the same site, and red nodules developed on the lower left leg. Subsequently, the patient consulted the Kariya Toyota General Hospital. Malignant melanoma was suspected, and the patient was referred to our department.\nAt the amputation site on the left foot, there was a skin ulcer measuring 25 × 20 mm with several red nodules, primarily on the anterior surface of the lower left leg (fig. ). Skin biopsies were obtained from the red nodules on the lower leg and the skin ulcer on the left foot.\nHistopathological examination of both lesion samples revealed tumor cells with eosinophilic cytoplasm and medium-to-large nuclei with distinct nucleoli extending through all layers of the dermis and into some fatty tissue. The tumor comprised an alveolar structure and had a solid proliferative area (fig. ). Immunostaining was positive for vimentin, S-100, HMB-45, melanoma, and Melan-A. The MIB-1 index was 38.3%. Based on these results, a diagnosis of recurrent malignant melanoma with in-transit metastasis was made.\nSubtotal integumentectomy was scheduled; however, 2 weeks later, the patient developed acute myocardial infarction and was hospitalized at Kariya Toyota General Hospital. Treatment for acute myocardial infarction continued for 1 month; thereafter, the patient was transferred to our department. As the myocardial infarction decreased the patient's tolerance to surgery, intravenous infusion of IFN-β was decided as the treatment of choice. Initially, IFN-β was administered at 3 million U/day for 10 consecutive days. As a countermeasure against fever, which is a side effect of IFN-β, Loxonin® was orally administered three times per day on the days of infusion. Thereafter, the patient was treated as an outpatient, with intravenous infusion of IFN-β 3 million U/day once a week. Twenty-six weeks later, the skin ulcer on the fifth toe of the left foot was cured, the nodes on the left leg had disappeared, and only pigmentation was observed (fig. ). After 1 year, the regimen was changed to intravenous infusion of IFN-β 3 million U/day once every 2 weeks. The patient's progress has been satisfactory, with no evidence of recurrence or metastasis at 1 year and 9 months after treatment (fig. ). The patient continues to receive IFN-β 3 million U/day every 2 weeks.

[[77.0, 'year']]

M

{'19617295': 1, '34462667': 1, '16899608': 1, '20535130': 1, '24707255': 2}

{}

3975207-1

noncomm/PMC003xxxxxx/PMC3975207.xml

Perforated Sigmoid Diverticulitis in the Presence of Toxic Epidermal Necrolysis

A 91-year-old female was admitted to our Emergency Department shortly after having been found lying in the shower at home. The patient had lived by herself up to that point and presented with a medical history of arterial hypertension and spinal arthrosis. She had been taking mefenamic acid and paracetamol, but the latter had been changed to metamizole – a pyrazolone derivative – approximately 3 weeks prior to admission. Taking the patient's history was complicated by her age and dementia, and retrospectively, possibly due to a delirious state. On admission, the patient reported having suffered burns from a stove. She then took a shower where she slipped and fell, and was burnt additionally by the hot water. She denied any other respiratory, gastrointestinal and musculoskeletal complaints. Clinically, she presented with epidermal lysis on both thighs and proximal upper extremities, totaling approximately 7% of the TBSA, which in the context of the reported burning, were interpreted as second-degree burns (fig. ). A brief debridement and wound dressing was performed. The skin showed no adherence to the underlying tissue and could be stripped off without effort. A slight reddish rash was noted on both breasts and the lower abdomen and was interpreted as a reaction to the bandages. In addition, a contusion on the left hip was diagnosed. The patient denied any abdominal pain. She presented with a distended abdomen, tender to the touch in the left lower quadrant, lacking rebound tenderness.\nLaboratory studies showed elevated C-reactive protein of 169 mg/l and leucocytes of 33,100/μl. Moreover, she presented with lymphocytopenia (lymphocyte count 500/μl), renal insufficiency (creatinine 121 μmol/l) and a hypokalemia level of 2.7 mmol/l. She tested positive for a nitrite-positive urinary tract infection. An abdominal CT scan was performed. Free air in the abdominal cavity and signs suggestive of perforated sigmoid diverticulitis were observed (fig. ). The condition was managed by emergent sigmoid colectomy with colostomy (Hartman's procedure). In the course of intubation and surgery, a rapid progression of the epidermal detachment was noted on the abdomen, spreading to 30–40% of her TBSA. A direct Nikolsky sign could be elicited, and an attempt at an indirect sign was negative. Following surgery, the patient was treated with piperacillin/tazobactam (blood cultures remained negative). Renal function deteriorated progressively. During the following 2 days, further cutaneous vesicles developed, the epidermolysis generalized and the patient became increasingly delirious. After 3 days, she showed signs of apneic breathing, after which we decided on palliative treatment. The patient passed away 3 days after admission.

[[91.0, 'year']]

F

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{'7533238-1': 1}

3975208-1

noncomm/PMC003xxxxxx/PMC3975208.xml

Topical Treatment for Orbital Capillary Hemangioma in an Adult Using a β-Blocker Solution

A 43-year-old female was admitted to hospital with a complaint of bleeding from the right medial ocular angle. She had had a long history of capillary hemangiomas, as described below.\nShe had developed capillary hemangiomas of the right upper eyelid at the age of 4 and of the right buccal region at the age of 9. Both of those lesions were surgically excised at the time of development. She had also developed a right orbital tumor at the age of 13, which was monitored without any treatment. She presented at the age of 34 with a complaint of right proptosis with ocular pain due to the right orbital tumor, which was confirmed to be an orbital capillary hemangioma after being pathologically diagnosed from an endoscopic biopsy at that time. In the biopsy sample, a lot of dilated capillaries were observed in the mucosa (fig. , arrows), indicating that the lesion was a capillary hemangioma/malformation, but not infantile hemangioma. Radiation therapy and surgical excision were considered but rejected due to a risk of visual loss since the lesion was too close to the optic nerve. Fortunately, the right proptosis with ocular pain had almost disappeared 2 months later with no treatment other than the biopsy. She was then observed without any treatment for years.\nHer best-corrected visual acuity was 1.2 OD and 1.5 OS, and the intraocular pressure was 12 mm Hg in both eyes. Slit-lamp examination revealed no abnormalities except the superficial lesions of the capillary hemangioma of her right eye. No abnormalities were found in the retinas of either eye.\nThe hemangioma presented both superficial and deep orbital components. The superficial lesion was seen at the right medial ocular angle, that is, on the upper/lower eyelids, conjunctiva and expanding into the subconjunctival space. It appeared as superficial tortuous blood vessels, and the conjunctiva appeared violet/blue in color (fig. ). The deeper lesion lay posterior to the orbital septum and was detected using MRI with low signal intensity on T1-weighted images versus high signal intensity on T2-weighted images with internal signal void (fig. , arrows) and gadolinium enhancement.\nThe patient was instructed to apply an ophthalmic solution of timolol maleate 0.5% twice daily. The superficial lesion gradually regressed, as shown in fig. , and had almost disappeared after 1 year of treatment (fig. ). The deeper lesion had also reduced in size, with maximal MRI axial dimensions of 16 × 11 mm (fig. ), decreasing to 12 × 8 mm (fig. , arrow) after 1 year of treatment. No apparent recurrence of bleeding in her eye was observed. No local or systemic adverse effects were noted.

[[43.0, 'year']]

F

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{}

3975209-1

noncomm/PMC003xxxxxx/PMC3975209.xml

Spontaneous Resolution of Vitreomacular Traction in Two Patients with Diabetic Macular Edema

Case 1 is a 63-year-old white male, previously treated for proliferative diabetic retinopathy with panretinal photocoagulation. Additionally, the patient had persistent DME OU with stable vision after focal laser photocoagulation and anti-vascular endothelial growth factor. His visual acuity in OS was 6/9, and he had macular thickening diagnosed as mild diffuse DME [central macular thickness (CMT) 344 μm]. The OCT also demonstrated inner retinal tenting, confirming VMT OS (fig. ). Because his edema and visual acuity were stable, observation was recommended. Six months later, the patient returned and had developed spontaneous resolution of VMT and an improved visual acuity of 6/6 OS with no interval interventions. Of note, the macula remained thickened (CMT 325 μm) and cystic (fig. ) OS.

[[63.0, 'year']]

M

{'20138368': 1, '23591535': 1, '22929316': 1, '23714857': 1, '19447497': 1, '20616687': 1, '28099316': 1, '22894573': 1, '33989683': 1, '23068973': 1, '29090053': 2, '21416651': 1, '19430388': 1, '11709016': 1, '24707275': 2}

{'3975209-2': 2, '5644410-1': 1, '5644410-2': 1}

3975209-2

noncomm/PMC003xxxxxx/PMC3975209.xml

Spontaneous Resolution of Vitreomacular Traction in Two Patients with Diabetic Macular Edema

Case 2 is a 68-year-old white male with non-proliferative diabetic retinopathy OU and DME OD. In OD, he had a visual acuity of 6/12, mild DME (CMT 314 μm), and OCT evidence of VMT (fig. ). Similar to Case 1, the acuity and retinal thickness were stable and observation was recommended. Six months later, VMT had spontaneously resolved with no interval treatments during the follow-up period. The visual acuity improved to 6/7.5 in OD, but the mild cystic DME was unchanged (CMT 314 μm) on OCT.

[[68.0, 'year']]

M

{'20138368': 1, '23591535': 1, '22929316': 1, '23714857': 1, '19447497': 1, '20616687': 1, '28099316': 1, '22894573': 1, '33989683': 1, '23068973': 1, '29090053': 2, '21416651': 1, '19430388': 1, '11709016': 1, '24707275': 2}

{'3975209-1': 2, '5644410-1': 1, '5644410-2': 1}

3975211-1

noncomm/PMC003xxxxxx/PMC3975211.xml

Clinical Presentation of a Patient with Congenital Cutis Laxa and Abnormal Thyroid Hormone Levels

A 7-year-old female patient was admitted to our hospital due to developmental delay, growth retardation, and loose facial skin since birth. At 2 months of age, she was found to have a hoarse voice. At the age of 3 years, her height was significantly lower than that of children of the same age. However, she had no other symptoms, had normal intelligence, and was able to perform normal activities. She underwent surgery to repair an inguinal hernia at the age of 3 years. Her father had a similar skin disease, but his height was normal with about 5.6 feet (1.7 m). Physical examination revealed that the patient's height was 100 cm, weight 16 kg, and she had loose inelastic skin hanging in folds. Her face appeared like that of an elderly woman (fig. ). Loose and sagging skin folded over the neck, arm, and trunk, which gave her a prematurely senile appearance with her skin displaying obvious relaxation and elasticity (fig. ). The thyroid was not enlarged. Her father also had skin hanging in folds over his abdomen (fig. ). Further examination after admission revealed a normal electrocardiogram and chest X-ray. Radiological investigation of the hand wrist bone showed a markedly delayed bone age. Laboratory values revealed normal values for complete blood count, erythrocyte sedimentation rate, routine urine analysis, blood biochemistry, hepatic enzymes, and serum electrophoresis. Serologic tests for syphilis (RPR, TPPA) were negative. Skin biopsy specimens taken from the involved area were stained with hematoxylin-eosin and Weigert stains. Histological findings from the skin biopsy revealed a dermal collagen fiber glass-like swelling and perivascular lymphocyte infiltration. Elastic fiber staining showed that the elastic fibers present were fragmented, arranged in disorder, and had a granular appearance (fig. ). By contrast, the collagen structure, content, and distribution were normal (fig. ). Because of the growth delay, the thyroid functions were tested and revealed FT3 7.71 mmol/l (normal range: 3.8–6.0), FT4 24.80 mmol/l (normal range: 7.9–14.4), TSH 2.29 mIU/l (normal range: 0.34–5.0 mIU/l), thyroid microsomal antibody 4.16% (normal range: <15), and thyroglobulin antibody 9.23% (normal range: <30). Growth hormones and adrenocorticotropic hormones were all within normal ranges, as were skull radiographs. Based on these clinical manifestations and laboratory examinations, the most likely diagnosis in our patient was CL syndrome with abnormal thyroid hormone levels.

[[7.0, 'year']]

F

{'21431621': 1, '22387031': 1, '17958802': 1, '19401719': 1, '20585600': 2, '23401092': 1, '23154624': 1, '22110782': 1, '5046633': 1, '11041445': 1, '24707249': 2}

{'2879976-1': 1}

3975634-1

noncomm/PMC003xxxxxx/PMC3975634.xml

Type F Congenital Quadricuspid Aortic Valve: A Very Rare Case Diagnosed by 3-dimenional Transoesophageal Echocardiography

A 52-year-old lady presented to our centre for echocardiogram from her primary-care-physician because he elicited a cardiac murmur. She had history of worsening breathlessness over a period of 7 years. She also had a history of pre-syncope.\nShe had a standard transthoracic echocardiogram (TTE), which revealed that her left ventricle (LV) is dilated with evidence of significant systolic dilatation (LV internal diameter systolic 55 mm) associated with severe systolic impairment with an ejection fraction of 33% (Simpson’s Bi-plane method). The aortic valve appeared to be bicuspid on sub-costal views. The parasternal views of the aortic valve were somewhat limited. There was evidence of severe aortic regurgitation (AR) with a central regurgitant jet, which occupied greater than 65% of the LV outflow tract and there was clear flow reversal in the aortic arch and descending aorta. In addition to these findings there is also mild mitral regurgitation (MR) and preserved right ventricular (RV) systolic function. She also informed us that mother had aortic root pathology and had to have aortic root replacement surgery. This hinted to hereditary congenital aortic or aortic valve pathology. She was admitted to our unit and received heart failure treatment and consequently had transoesopha-geal echocardiography (TOE) (Philips IE33 using 3D-TOE probe).

[[52.0, 'year']]

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{'4860453-1': 1}

3975721-1

noncomm/PMC003xxxxxx/PMC3975721.xml

Hemodialysis without Anticoagulation for a Patient with Chronic Disseminated Intravascular Coagulation

In November 2008, a 68-year-old Japanese man was admitted to our hospital for the construction of an arteriovenous fistula (AVF). Proteinuria and hematuria had been noted at the age of 18 years. Hypertension was diagnosed when he was 40 years old and a calcium channel blocker was prescribed. In 1990, chest pain occurred suddenly without any precipitating cause, and dissection of the ascending thoracic aorta (Stanford A type) was diagnosed. Surgical treatment was performed using a synthetic graft. In 2002, dissection also occurred in the descending thoracic aorta (Stanford B type) and surgical repair was done. Since then, mild thrombocytopenia with a platelet count of around 100,000/ml had been noted, but he was asymptomatic. Computed tomography (CT) was done every 6 months and revealed a new aneurysm with thrombosis in the residual aorta, adjacent to the anastomosis, in 2005 (fig. ). No further surgical treatment was performed. In August 2008, an AVF was constructed to allow dialysis for end-stage renal failure, but a transfusion was required (400 ml of red blood cells and 1,600 ml of fresh-frozen plasma) because of the massive bleeding. Thereafter, his platelet count decreased to 52,000/μl while his prothrombin time (PT) was 80.5% (normal: >75), his activated partial thromboplastin time (APTT) was 31.9 s (normal range: 27.0–40.0), fibrinogen was 128.4 mg/dl (normal range: 120–300), fibrinogen degradation products (FDP) were 119.5 μg/ml (normal range: <5), and D-dimer was 79.8 μg/l (normal range: <1). In addition, the thrombin-antithrombin complex level was 46.4 ng/ml (normal range: 3–4), the plasmin-α2 plasmin inhibitor complex was 4.9 mg/ml (normal range: <0.8), and antithrombin-3 was 69.6% (normal range: 84–122) (table ). Chronic DIC, a less active condition than acute DIC, was diagnosed based on the following findings: an underlying disease associated with DIC (aortic aneurysm), elevation of FDP, APTT, PT, and fibrinogen within the normal range and mild thrombocytopenia [, ]. CT showed no change of the aortic aneurysm. There was no evidence of other risk factors for chronic DIC, including malignancy. Therefore, his chronic DIC was considered to be secondary to the aortic aneurysm with thrombosis. Initiation of hemodialysis was required in December 2008 due to a urea nitrogen level of 120 mg/dl and a creatinine level of 7.1 mg/dl. It was considered that administration of an anticoagulant for dialysis sessions was associated with a high risk of worsening his aortic condition. Therefore, because his ACT was prolonged to 150–180 s (normal range: 90–140), hemodialysis was started without the administration of an anticoagulant and was therefore performed successfully without any complications (such as clotting inside the dialysis circuit or dialyzer). Thereafter, hemodialysis (4 h, 3 times weekly) was continued without an anticoagulant, and he is doing well as of February 2012.

[[68.0, 'year']]

M

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3975722-1

noncomm/PMC003xxxxxx/PMC3975722.xml

Reversal of Cognitive Dysfunction by Total Removal of a Large Lateral Ventricle Meningioma: A Case Report with Neuropsychological Assessments

A 66-year-old, right-handed man earned his living by cultivating agricultural products and cooking. His highest level of educational was high school graduation. He had no past medical history and had been incidentally diagnosed with a right ventricular meningioma 20 years earlier. He had no neurological symptoms and was conservatively followed up because the tumor diameter was about 1 cm. However, the tumor had gradually increased in size on MRI during these 20 years. Finally, over the past year, his working performance had started to deteriorate. He experienced increasing difficulty in concentrating on various tasks (making errors while cooking, miscalculations, and aprosexia) and gradually exhibited inappropriate social behaviors (abusive words, violent manner, and juvenile behavior). His short-term memory also declined. Therefore, he was referred to our hospital for further examination. A standard neurological examination performed on admission revealed no definite abnormalities such as auditory agnosia, left hemispatial neglect, 2-point discrimination, callosal signs, optic ataxia, and astereognosis. Callosal signs such as auditory disconnection syndrome, extinction, and left-hand apraxia were absent. His visual field was kept intact on Goldmann visual field testing. MRI showed a large round mass in the trigone of the right lateral ventricle with a diameter of 5.2 cm and large perifocal edema around the ventricle limbic structure. The signal of the mass was low and high on T1- and T2-weighted images, respectively. The entire mass was strongly enhanced with Gd-DTPA (fig. ). Cystic formation was observed lateral to the tumor. The midline structures were shifted to the left, and cerebral edema was observed in the white matter around the tumor. MR tractography could clearly localize the optic radiations around the tumor (fig. ). Cerebral angiography showed that the posterior lateral choroidal artery was the main feeder of the tumor. Based on these findings, the tumor was diagnosed as meningioma. Then, cognitive function was precisely evaluated, using the Mini-Mental State Examination (MMSE), Standard Language Test of Aphasia (SLTA), and Repeatable Battery for the Assessment of Neuropsychological Status (RBANS). The scores on the MMSE and SLTA were within normal limits: 27 points and normal, respectively. On the other hand, RBANS revealed that the patient had marked impairment of attention, language, visuospatial/constructional abilities, and delayed memory function (fig. ).\nPrior to surgery, the lateral posterior choroidal artery was embolized via a microcatheter, using n-butyl-2-cyanoacrylate. MRI following embolization showed that the major part of the tumor was not further enhanced. Two days later, the patient underwent total tumor removal. A large temporoparietal craniotomy was performed, and the dura mater was opened. Under intraoperative navigation, the tumor was exposed through a 2-cm corticotomy on the superior temporal gyrus. The tumor was totally removed without any significant bleeding. Transfusion was not performed.\nThe postoperative course was uneventful, and the patient's motor and visual functions were intact. Moreover, a standard neurological examination performed after tumor removal revealed no abnormalities such as auditory agnosia, hemispatial neglect, 2-point discrimination, callosal signs, optic ataxia, and astereognosis. Callosal signs such as disconnection syndrome, left-hand apraxia, and auditory extinction were also absent. His visual field was kept intact on Goldmann visual field testing. Total tumor removal was confirmed on follow-up MRI. Neuropsychological examinations were repeated 13 days after surgery. Results showed that all scores on RBANS significantly improved back to the normal range (fig. ). Difficulty in concentrating on various tasks and the exhibiting of inappropriate social behaviors were completely resolved. He could perform various tasks (without making errors while cooking, miscalculation, and aprosexia) in his daily living after tumor removal. He was discharged and returned to his former position. A histopathological examination confirmed the diagnosis of fibrous meningioma. The mib-1 index was 2.3%.

[[66.0, 'year']]

M

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