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AISC Team B1 5

3935219-1

noncomm/PMC003xxxxxx/PMC3935219.xml

Histopathological effects of radiosurgery on a human trigeminal nerve

A 43-year-old female not known to have any concomitant medical problems was diagnosed to have classical trigeminal neuralgia 4 years ago when she presented with paroxysmal electric-shock-type pain involving the dermatomes of the left maxillary and mandibular trigeminal branches. Her symptoms were aggravated by eating, brushing her teeth, and drinking cold water. The maximal intensity of pain was at the left cheek and left side of the upper lip and radiating to the side of the nose. There was no history of any associated sustained background pain or sensory symptoms. Her pain responded well to carbamazepine initially; however, the dose was escalated after few months without complete control of her symptoms. Subsequently, she was treated with carbamazepine, pregabalin, and baclofen without complete resolution of her severe pain. The pain remained intractable to medical therapy with a visual analogue score (VAS) for pain of 10 points prior to medical therapy and 8 points after.\nOn clinical examination, there were no sensory deficits or dermatological changes at trigeminal nerve distribution and no abnormal neurological signs. Magnetic resonance imaging (MRI) of the brain did not reveal any signs of demyelinating disease, tumor, vascular malformation, or any other structural lesion. A small vessel was seen above the trigeminal nerve juxtpontine segment by MRI constructive interference in steady state (CISS) sequence []. Management options were offered for the patient, which included percutaneous procedures, radiosurgery, and MVD. After discussing the pros and cons of each procedure, the patient decided to go for radiosurgery.\nSubsequently, she underwent stereotactic image-guided radiosurgery using CyberKnife® (Accuray, Inc., Sunnyvale, CA, USA). A total dose of 60 Gy prescribed to 76% Isodose line covering 100% of the target, which consisted of a 6-mm length of the left trigeminal nerve cisternal segment []. Maximum dose to the target was 7894.74 cGy. This was delivered by a single fixed 5-mm cone with a total of 100 beams. The conformality index was 1.61, and the homogeneity index was 1.32. The stereotactic planning was carried out with fusing 1 mm cuts computed tomography (CT) scan with T1 3D MRI and 3D CISS sequence. Of note, the identified length of trigeminal nerve by MRI CISS sequence was 11.5 mm. The pain relief benefit started approximately 10 days after the procedure, and the patient continued to have 80% reduction of the lancinating trigeminal neuralgia pain (Barrow Neurological Institute (BNI) grade III).[] However, 2 months later, she started complaining of dull background pain with an intensity of 3 VAS and intermittent mild facial numbness at V2 distribution (BNI grade II for facial numbness). There was no change in the lancinating shooting pain pattern. The medications remained unchanged. The patient had a recurrence of the pain 8 months later with more frequent attacks than before the procedure and remained intractable to medical therapy. At that time, clinical examination showed reduced sensation at V2 and V3 and relative reduction in the corneal reflex on the left eye. No corneal ulceration was noted, and the patient had with normal visual acuity. MRI of the brain did not demonstrate any enhancement or signal changes at the trigeminal nerve and brainstem.\nWe elected to perform MVD to explore the nerve with possible partial sensory sectioning. The procedure was done under neurophysiology monitoring of the facial, vestibulocochlear, and trigeminal nerves on the left side in addition to motor evoked potential (MEP) and somatosensory evoked potential (SSEP). Electromyography (EMG) was used for trigeminal nerve monitoring. Intraoperatively, the trigeminal nerve appeared atrophied and flattened with grayish discoloration of the nerve rootlets []. An arterial loop was identified at the superior surface of the juxtpontine trigeminal nerve segment; however, the artery was not causing obvious compression []. This vessel was separated from the nerve by a Teflon patch. Partial sensory sectioning was done and a small nerve rootlets biopsy from the midcisternal nerve segment was taken and submitted for histopathological examination []. The interval between histological examination and radiosurgery was 13 months.\nThe submitted specimen was too small to be utilized for full staining methods. Therefore, the biopsy was completely processed for electron microscopy (EM). The toluidine blue-stained, epoxy-embedded section revealed fibrous tissue (fibrosis) and a few haphazardly arranged and distorted myelinated nerve fibers []. Some were thinly myelinated and many had empty or dense axoplasm. A regenerative axonal cluster was noted as well. Transmission EM revealed variably sized myelinated axons in a marked collagenous background []. Most axons had empty axoplasm with markedly degraded myelin sheaths. Few had filamentous axoplasmic inclusions. There were many degenerated myelin/myelinoid profiles in the background. No active cellular component was noted. The changes were suggestive of advanced traumatic/destructive process.

[[43.0, 'year']]

F

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{}

3935259-1

noncomm/PMC003xxxxxx/PMC3935259.xml

Imaging Diagnosis of Neonatal Anemia: Report of Two Unusual Etiologies

A 3-month-old male child presented with history of pallor, abdominal distension, petechial rashes, recurrent respiratory infections, and repeated hospitalization since birth. Records of the previous hospitalization revealed persistent anemia and thrombocytopenia. Routine hemogram at our hospital revealed anemia (Hb 6.3 g/dl), leukocytosis (total leukocyte count (TLC)-83,000 cells/mm3), and thrombocytopenia (platelet count-35,000 cells/mm3). Skeletal survey (X-ray) requisitioned to rule out congenital infections revealed diffuse osteopenia and prominent calcific zone at metaphyseal ends of the long bones []. Radiograph of the dorsolumbar spine showed osteopenia with prominent vertebral end plates []. Abdominal sonography revealed hepatomegaly and increased hepatic echogenicity []. The infant showed no facial dysmorphisms suggestive of chromosomal disorder and karyotyping was negative for chromosomal anomalies. The serum biochemical parameters including the liver and renal function tests were within normal limits. The TORCH serology test for toxoplasmosis, rubella, cytomegalovirus, and herpes virus that includes enzyme-linked immunosorbent assay for detection of immunoglobulin IgG antibodies was found to be negative. The radiological and sonographic abnormalities seen in an anemic neonate with negative TORCH serology, and normal serum biochemical parameters suggested leukemic infiltration. Magnetic resonance imaging (MRI) of the lumbosacral spine and pelvis, revealed abnormal marrow signal intensities in bilateral iliac bones []. Bone marrow biopsy from the iliac bones revealed marrow infiltration with immature lymphoblasts and diagnosis of acute lymphoblastic leukemia was confirmed []. The infant was started on induction chemotherapy and is on regular follow-up.

[[3.0, 'month']]

M

{'15531837': 1, '12539590': 1, '19232111': 1, '19679962': 1, '14703230': 1, '24605254': 2}

{'3935259-2': 2}

3935259-2

noncomm/PMC003xxxxxx/PMC3935259.xml

Imaging Diagnosis of Neonatal Anemia: Report of Two Unusual Etiologies

A 3-month-old male infant was also brought with features of pallor, recurrent respiratory infections, and abdominal distension since birth. Records of previous hospitalization revealed persistent anemia (hemoglobin (Hb) 8.1 g/dl) and thrombocytopenia (platelet count-23,000 cells/mm3). Skeletal survey was requisitioned for this infant at our hospital to exclude congenital infections. Radiographs in this patient revealed diffusely increased bone density with loss of medullary cavity in the long bones [Figures and ]. A ‘bone within bone’ appearance was also noted in the ends of long bones []. Radiograph of dorsolumbar spine showed increased density of the vertebral body with central linear lucency giving the appearance of ‘sandwich vertebra’ []. Radiograph of the skull revealed marked sclerosis of the skull base []. Computed tomography (CT) of the brain done with adequate body lead shielding and using minimal dose, did not reveal any hydrocephalus or cranial nerve compression []. The radiological features were characteristic for OP. Attempted bone marrow aspiration revealed a dry tap. Bone biopsy revealed disorganized bony trabaculae with obliteration of marrow spaces, which confirmed the diagnosis of OP [].

[[3.0, 'month']]

M

{'15531837': 1, '12539590': 1, '19232111': 1, '19679962': 1, '14703230': 1, '24605254': 2}

{'3935259-1': 2}

3935262-1

noncomm/PMC003xxxxxx/PMC3935262.xml

A Rare Case of Pericallosal Lipoma Associated with Bilaterally Symmetrical Lateral Ventricular Choroid Plexus Lipomas without Corpus Callosal Anomalies

A 58-year-old Arab man presented to our hospital with a 12-month history of recurrent headache. Clinical and neurological examinations were unremarkable. Radiological evaluation included computed tomography (CT) and Magnetic resonance imaging (MRI) scanning.\nThe CT and MRI images revealed a PCLp cephalad to the entire antero-posterior extent of the corpus callosum; continuing as bilaterally symmetrical lateral ventricular CPLps [Figures –]. The PCLp continued through the choroidal fissure as bilaterally symmetrical lateral ventricular CPLps [Figures and ]. Peripheral calcifications were noted within the PCLp [], and the corpus callosum was unremarkable []. The fat appeared more hypodense than CSF on CT scans []. PCLp measured approximately 11.2 × 2.1 cm. CPLps measured approximately 1.3 cm in maximum diameter. Discrete areas of fatty tissue were noted in the subarachnoid space along the anterior interhemispheric fissure [] and also in the medial sulcus of the right frontal lobe []. Incidentally, unusually thick dural calcifications and cavum septum pellucidum were also noted with bilateral supraventricular white matter ischemic lesions. No other central nervous system abnormalities were noted. Sagittal unenhanced T1 weighted MRI scans of a normal brain with superimposed black shading (black arrows) show morphological features that classify PCLps into anterior [] and posterior [] groups and is compared to features seen in our case [].

[[58.0, 'year']]

M

{'1388423': 1, '20224717': 1, '1565215': 1, '18425000': 1, '2105607': 1, '2119122': 1, '27236781': 1, '33354476': 2, '23466841': 1, '1885799': 1, '24605264': 2}

{'7746315-1': 1}

3935299-1

noncomm/PMC003xxxxxx/PMC3935299.xml

Malignant rhabdoid tumor of liver

A 6-month-old boy presented with a 15 days history of abdominal distension, irritability and reluctance to feed. There was no vomiting, constipation or jaundice. On physical examination, the child was febrile with normal vitals; abdomen was distended with hepatomegaly, no lump was felt. Ultrasound of the abdomen revealed the presence of hyperechoic lesions in the liver. Contrast enhanced computed tomography (CT) scan of the abdomen revealed multiple lesions in the liver (both lobes), which were homogeneously hypodense; had no evidence of calcification [Figure and ]. There was no change in the caliber of the infraceliac abdominal aorta or enlargement of the hepatic artery. Magnetic resonance imaging (MRI) showed that the lesions were hypointense on T1-Weighted (W) and hyperintense on T2-W images [Figure and ]. MIBG (meta-iodobenzylguanadine) scan did not reveal any uptake. A 24 h urinary vanillylmandelic acid was with in normal limits (0.46 mg/g of creatinine); serum homovanillic acid was mildly raised (28.18 mg/g of creatinine); and serum alpha-fetoprotein (αFP) was within normal limits. Fine needle aspiration cytology of the lesion was inconclusive; hence, a wedge biopsy was performed through a minilaparotomy. Macroscopic examination of the wedge showed a stretched out capsule on the exterior aspect with a tan colored tumor within showing areas of necrosis. On microscopic examination of H and E stained sections [] a tumor was seen adjacent to normal liver tissue. The tumor was arranged in nodules with the absence of trabeculae and sinusoids. The tumor cells had large nucleus having prominent nucleoli and moderate to abundant cytoplasm with pink inclusion bodies. The immunohistochemistry revealed positivity for cytokeratin and vimentin while it was negative for glycogen, desmin and chromogranin. Immunocytochemistry for INI-1 was not available. The histology was consistent with MRT of the liver. Patient was started on chemotherapy (carboplatin, etoposide and cyclophosphamide), but died soon after of progressive tumor and massive unrelenting ascites.

[[6.0, 'month']]

M

{'7200394': 1, '1700599': 1, '12378463': 1, '15260854': 1, '9100795': 1, '25918621': 2, '8453588': 1, '19305156': 1, '206343': 1, '21744471': 1, '17551403': 1, '19452146': 1, '33670452': 1, '20048688': 1, '24604983': 2}

{'4387326-1': 1}

3935300-1

noncomm/PMC003xxxxxx/PMC3935300.xml

Segmental dilatation of ureter: Report of two cases

A 14-month-old male child presented with abdominal distension and pain of 2 months duration. USG revealed moderate hydronephrosis on right side; ureters were not seen. A thick-walled cystic structure with thick fluid inside (4 cm × 3 cm) was noted on right side just above the bladder. IVP showed right-sided hydronephrosis, as the radiologist missed the non-functioning lower moiety. Left kidney was normal. MCUG showed normal urinary bladder and no reflux. Renal biochemical parameters were within normal limits. At operation, right-sided duplex kidney was noted. Normal size upper moiety ureter (8 cm long) drains into the dilated segment (3 cm × 2 cm). The small lower moiety and dilated upper ureter (4 cm long) drains into a markedly dilated, thickened ureteral segment (6 cm × 4 cm). The dilated segments were densely adherent to the bladder wall and extended up to the recto-vesical pouch. Normal size lower ureters of both moieties opened into the bladder separately. Partial nephroureterectomy (lower moiety) was done with excision of the dilated segment of both upper and lower moiety ureters. Normal upper moiety ureter was re-implanted into the bladder by Leadbetter-Politano technique. Histology of the lower moiety showed features of XGP []. The patient had uneventful recovery and was doing well till last follow up at 18 months.

[[14.0, 'month']]

M

{'1942352': 1, '15105004': 1, '7722817': 1, '9224363': 1, '24604984': 2}

{}

3935301-1

noncomm/PMC003xxxxxx/PMC3935301.xml

Benign papillomatosis of common bile duct in children: A rare case report

Our case was a 3-year-old female child who presented to our pediatric surgical outpatient department with complaints of pain in the upper part of the abdomen along with jaundice and fever for last 6 months. On per abdominal examination no lump or hepatosplenomegaly was found. With the clinical possibility of a choledochal cyst, ultrasonography (USG) along with liver function test and routine hematological investigations were advised. USG showed impacted worm-like appearance in the lower common bile duct (CBD) with proximal dilatation []. Choledochal cyst was ruled out. In view of the deepening jaundice, child was advised endoscopic retrograde cholangiopancreatography (ERCP) to clear the worm load in the lower end of CBD and to place the stent for establishing the bile flow. Thus, endoscopic sphincterotomy with placement of 10 Fr stent was done []. There were no worms in the CBD in this patient. The child also received anthelmintics. Following stent placement child recovered from jaundice within 2 weeks. As there was a doubt of persistent worm in the distal CBD, exploration was planned; and after obtaining informed written consent, the child was explored under general anesthesia. Right upper transverse incision was given, abdominal cavity was opened, liver mobilized, gall bladder identified, and CBD traced using stent as a guide. On opening the CBD multiple polyps were found with stent in situ [] without stone or worm. In view of possible malignancy a frozen section was sent and when confirmed to be benign, polyps were removed completely along with stent and CBD was repaired over a T-tube. Postoperatively T-tube was removed after 3 weeks. The child recovered uneventfully and presently she is in close follow-up and doing well. Specimen sent for histopathological examination showed adenomatous polyps with colonic metaplasia.

[[3.0, 'year']]

F

{'10198612': 1, '17026772': 2, '11375588': 1, '10849979': 1, '14598142': 1, '14770435': 1, '6773387': 1, '24604985': 2}

{'1618388-1': 1}

3935302-1

noncomm/PMC003xxxxxx/PMC3935302.xml

Esophageal exclusion and bypass for corrosive injury: The lessons learnt

A 3-year-old boy underwent emergency end cervical esophagostomy, decompressive gastrostomy and feeding jejunostomy following esophageal perforation sustained during esophageal dilatation for corrosive stricture. Three months later, a substernal gastric pull up was performed, the native oesophagus being retained in situ with both ends divided and suture closed. Excision of the diseased native oesophagus was not attempted on account of prior mediastinitis in the setting of corrosive esophageal stricture. One year following the substernal gastric pull up, the boy presented with a 4-weeks history of repeated episodes of fever, tachypnea and an expiratory stridor on physical exercise or crying. An upper gastrointestinal endoscopy and barium swallow did not reveal any anastomotic stricture or delayed gastric emptying which could predispose to recurrent pulmonary aspiration. Subsequent contrast-enhanced computed tomography (CECT) scan of the chest revealed a size 3 × 11 cms. posterior mediastinal cystic structure causing anterior and lateral displacement of the trachea with compression of its lumen [Figures and ]. A diagnosis of mediastinal mucocele of the retained native oesophagus was made. The mucocele was excised by a right posterolateral thoracotomy. Operative findings revealed a thick walled, tense, mucocele containing frankly purulent fluid and an ulcerated mucosa []. There were significant adhesions to the mediastinal structures. The early postoperative issues were related to upper airway edema which gradually resolved. At 15-months post-surgery, the patient is free of dysphagia, regurgitation, or respiratory symptoms with an appropriate weight gain.

[[3.0, 'year']]

M

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{'7837637-1': 1}

3935303-1

noncomm/PMC003xxxxxx/PMC3935303.xml

Gastric volvulus with partial and complete gastric necrosis

A 10-month-old male child admitted with features of acute intestinal obstruction with bilious vomiting and occasional hematemesis for last two days. There was difficulty in placing the Ryle's tube even on repeated attempts. The patient was having epigastric fullness. There was history of repeated chest infections in the past for which the patient was treated with antibiotics by a local physician. A babygram showed diaphragmatic defect with a large single air-fluid level []. On exploration, organo-axial gastric volvulus with total necrosis of the stomach was detected [] along with eventration of the left hemidiaphragm. The spleen and small bowel were lying high up because of eventration of diaphragm. Total gastrectomy with closure of duodenal stump and esophagojejunostomy (Roux loop) [] and jejunojejunostomy was done and diaphragmatic eventration was repaired. The resected specimen was sent for histopathologic analysis. The affected hemidiaphragm was plicated with 3-0 polypropylene stitches in interrupted fashion.\nA postoperative contrast study done on day 5 revealed a smooth passage of dye without leakage or retention. From the same day, feeds were started per oral and the patient tolerated the feeds well. The patient was discharged on the eighth postoperative day. The histopathology report on Haematoxylin and Eosin stain in high power (i.e. 400X magnification) showed necrosis along with infiltration of inflammatory cells.\nOn follow-up after 15 days, 3 months, and 12 months, the patient is doing well and gaining appropriate weight.

[[10.0, 'month']]

M

{'30900131': 1, '27988456': 1, '31258281': 1, '8792721': 1, '18167432': 1, '11101746': 1, '34691417': 1, '5558165': 1, '34093935': 2, '11673711': 1, '28954749': 1, '22856443': 1, '29764844': 1, '2276024': 1, '1929847': 1, '15531850': 1, '24604987': 2}

{'3935303-2': 2, '8167805-1': 1}

3935303-2

noncomm/PMC003xxxxxx/PMC3935303.xml

Gastric volvulus with partial and complete gastric necrosis

A 6-month-old girl was brought to the emergency with sudden abdominal distention, nonbilious vomiting, and restlessness for three days. She did not have any similar history of vomiting, respiratory complaints, or abdominal distension in the past. No similar illness ran in her family. On admission, dehydration was present with a pulse rate of 120/min. Epigastrium was full. Per rectal examination was normal. Straight X-ray revealed left-sided eventration of the diaphragm with prominent gastric bubble with air-fluid levels.\nRyle's tube aspirate was blood-stained and scanty in amount. The patient was resuscitated with intravenous fluids and antibiotics. On exploration, mesenteroaxial volvulus of the stomach was noted. The whole of stomach, sparing the pyloric end, was necrotic. The necrotic stomach was excised and sent for histopathology. Duodenum was mobilized. A pyloric tube was made after pyloromyotomy and oesophago-pylorostomy was performed in a single-layer interrupted fashion with polyglactin 4-0 stitches. Plication of the diaphragm was done with 3-0 polypropylene stitches in an interrupted fashion. Feeding jejunostomy was additionally performed.\nFeeding was started from postoperative day 3 through the jejunostomy tube. A contrast study performed after two weeks showed no anastomotic leak Subsequently, the patient was allowed oral diet and discharged. On follow-up 15 days and 6 months later, the patient is doing well.

[[6.0, 'month']]

F

{'30900131': 1, '27988456': 1, '31258281': 1, '8792721': 1, '18167432': 1, '11101746': 1, '34691417': 1, '5558165': 1, '34093935': 2, '11673711': 1, '28954749': 1, '22856443': 1, '29764844': 1, '2276024': 1, '1929847': 1, '15531850': 1, '24604987': 2}

{'3935303-1': 2, '8167805-1': 1}

3935506-1

noncomm/PMC003xxxxxx/PMC3935506.xml

Response of serous retinal pigment epithelial detachments to intravitreal aflibercept in polypoidal choroidal vasculopathy refractory to ranibizumab

A 79-year-old man presented with a large serous PED. Fluorescein angiography (FA) displayed an occult CNV, and indocyanine green (IA) images showed a PCV. At the initial visit, his best-corrected visual acuity (BCVA) was 2/20 oculus sinister (OS). Three monthly injections of ranibizumab slightly flattened the PED and reduced the subretinal fluid (SRF), but a recurrence developed. Three additional monthly injections of ranibizumab (total of six injections) did not improve the PED, and there was an increase of the highly reflective material beneath the outer surface of the RPE in the SD-OCT images.\nWe switched to aflibercept and, after one intravitreal injection, the PED was slightly flattened; two additional injections of aflibercept were given, which flattened the PED over the hyper-reflective material observed by SD-OCT. The BCVA remained at 2/20 OS ().

[[79.0, 'year']]

M

{'20463807': 1, '27848983': 1, '27478354': 1, '23706500': 1, '30834360': 1, '4663679': 1, '28428893': 1, '2408659': 1, '26719668': 1, '26289356': 1, '23558214': 1, '17965108': 1, '18327139': 1, '19152869': 1, '28966847': 1, '30662840': 1, '2483142': 1, '3310342': 1, '23766432': 1, '27779169': 1, '24591809': 2}

{'3935506-2': 2, '3935506-3': 2}

3935506-2

noncomm/PMC003xxxxxx/PMC3935506.xml

Response of serous retinal pigment epithelial detachments to intravitreal aflibercept in polypoidal choroidal vasculopathy refractory to ranibizumab

A 72-year-old man presented with a large serous PED in his right eye associated with a PCV. His BCVA was 14/20 OD. He had received six ranibizumab injections over a 10-month period, but the PED worsened and the SRF recurred. Hyper-reflective materials were seen beneath the outer surface of the RPE in the SD-OCT images. The BCVA decreased to 8/20 OD.\nTwo monthly injections of aflibercept resulted in a complete resolution of the serous PED; however, the flattened PED contained fibrovascular material beneath the RPE layer in the SD-OCT images.

[[72.0, 'year']]

M

{'20463807': 1, '27848983': 1, '27478354': 1, '23706500': 1, '30834360': 1, '4663679': 1, '28428893': 1, '2408659': 1, '26719668': 1, '26289356': 1, '23558214': 1, '17965108': 1, '18327139': 1, '19152869': 1, '28966847': 1, '30662840': 1, '2483142': 1, '3310342': 1, '23766432': 1, '27779169': 1, '24591809': 2}

{'3935506-1': 2, '3935506-3': 2}

3935506-3

noncomm/PMC003xxxxxx/PMC3935506.xml

Response of serous retinal pigment epithelial detachments to intravitreal aflibercept in polypoidal choroidal vasculopathy refractory to ranibizumab

An 80-year-old man presented with a large serous PED. FA displayed an occult CNV, and IA images showed a PCV. His BCVA was 10/20 OS. Three monthly injections of ranibizumab slightly flattened the PED, but a recurrence was observed after 3 months. Three additional monthly injections of ranibizumab (total of six injections) failed to reduce the PED, and hyper-reflective materials were seen beneath the outer surface of the RPE in the SD-OCT images. We switched to aflibercept, and after the two additional monthly injections, an exudative CNV lesion was observed instead of the serous PED. OCT showed a loss of optical free space and highly reflective materials, suggesting the formation of fibrovascular PED. One day after the third monthly injection of aflibercept, a subretinal hemorrhage and recurrent PED were observed. BCVA was reduced to 2/20 ().

[[80.0, 'year']]

M

{'20463807': 1, '27848983': 1, '27478354': 1, '23706500': 1, '30834360': 1, '4663679': 1, '28428893': 1, '2408659': 1, '26719668': 1, '26289356': 1, '23558214': 1, '17965108': 1, '18327139': 1, '19152869': 1, '28966847': 1, '30662840': 1, '2483142': 1, '3310342': 1, '23766432': 1, '27779169': 1, '24591809': 2}

{'3935506-1': 2, '3935506-2': 2}

3935782-1

noncomm/PMC003xxxxxx/PMC3935782.xml

Diagnosis of Necrotizing Faciitis with Bedside Ultrasound: the STAFF Exam

A 44-year-old female presented to the emergency department (ED) with a 3-day history of left groin and inner thigh redness, pain, and swelling; associated with fever, chills, and vomiting. The patient was seen the prior day in a local urgent care at which time she was treated with intravenous vancomycin for cellulitis and discharged on oral antibiotics. The patient presented to the ED within 24 hours of discharge from the urgent care. The initial ED vitals revealed an afebrile (36.9ºC), normotensive (126/63) patient with tachycardia (124) and mild tachypnea (22). On physical exam, the patient had left inner thigh and groin induration measuring 12 cm × 12 cm. The patient was morbidly obese with a BMI>45 kg/m2, making crepitus difficult to appreciate on physical exam. Her initial labs were as follows: lactate 3.5 mmol/L, WBC 18.2 × 103 per mm3, hemoglobin 12.3 g/dL, sodium 136 mmol/dL, glucose 225 mg/dL (12.5 mmol/L), and creatinine 1.8 mg/dL (159 μmol/L). This gave her a laboratory risk indicator for necrotizing fasciitis (LRINEC) score of 6, C-reactive protein (CRP) excluded. (CRP was not ordered in the ED). A bedside ultrasound was performed, which showed positive subcutaneous thickening, air, and fascial fluid (STAFF) concerning for necrotizing fasciitis (). The patient’s soft tissue ultrasound findings are significantly different when compared to normal soft tissue ultrasound (). The patient was started on intravenous vancomycin and piperacillin/tazobactam empirically, and surgery was consulted.\nBased on the LRINEC score, ultrasound findings, and physical exam, the patient was taken immediately to the operating room for presumed necrotizing fasciitis and forwent either CT or magnetic resonance imaging (MRI). In the operating room she underwent operative debridement of the left groin and perineum, resulting in excision of 15 cm × 23 cm of tissue with extensive washout. At the close of the surgery the patient was admitted to the surgical intensive care unit post-operatively for septic shock requiring vasopressors and ventilator dependence. The patient underwent repeat washouts with minor debridements daily for 3 days, with lactate normalization and a down-trending WBC to 13.5 × 103 per mm3 on post-operative day 3. She was extubated and transferred to a step-down unit on post-operative day 5, after which plastic surgery was consulted to evaluate for possible skin graft. Over the course of 9 days and 4 additional operative washouts, the patient was deemed to be a poor graft candidate. A wound vacuum-assisted closure (V.A.C.) device was placed, and the patient was transferred to the plastic surgery service on day 9. The decision was made to forgo skin graft during her immediate hospital stay. The patient was fully ambulatory and discharged home with a wound V.A.C. on post-operative day 28. The patient subsequently received a skin graft, and has been recovering well since.

[[44.0, 'year']]

F

{'12925624': 1, '29784062': 2, '19197096': 1, '31970543': 2, '19732035': 1, '26176579': 1, '25349748': 2, '10938701': 1, '24992977': 1, '33204184': 1, '32323044': 2, '15241098': 1, '2031360': 1, '20479343': 1, '10206215': 1, '18349455': 1, '22511053': 1, '32181091': 2, '29097860': 1, '29541493': 2, '12460854': 1, '27529031': 1, '31750343': 2, '24578776': 2}

{'7053670-1': 1, '4202280-1': 1, '7176767-1': 1, '6854421-1': 1, '6977785-1': 1, '5963038-1': 1, '5843965-1': 1}

3935783-1

noncomm/PMC003xxxxxx/PMC3935783.xml

Recurrent Priapism from Therapeutic Quetiapine

A 43-year-old African American man with a history of schizoaffective disorder and ulcerative colitis presented to the emergency department (ED) for a painful erection lasting 15 hours. He reported noncompliance with sulfasalazine for ulcerative colitis but intermittent use of quetiapine, 100 mg every morning and 200 mg every evening, for his schizoaffective disorder. Prior to the ED visit, previous doses of quetiapine resulted in erections lasting 3 hours every morning that resolved spontaneously. He denied any history of trauma, sickle cell disease or trait, illicit drug use, vasoactive agents, including nitrates, recent intercourse, or use of any medications or devices for sexual enhancement. Examination demonstrated a tender and erect phallus without evidence of injury, fibrosis, angulation, lesions, or discharge. Testicles were normal, and no hernias were present.\nSubcutaneous terbutaline and oral pseudoephedrine were administered with no effect. Aspiration of 10 cc of intracavernosal blood followed by intracavernosal phenylephrine injection led to successful detumescence. The patient was advised to discontinue use of quetiapine and arrange follow-up with his primary care physician.\nThe patient returned to the ED 4 times over the course of the next year. Each time the patient had a morning erection lasting 6 to 9 hours following 200 mg of quetiapine ingestion the prior evening. Detumescence was achieved successfully with aspiration and intracavernosal phenylephrine injection. The patient was ultimately transitioned from quetiapine to ziprasidone and amitriptyline with resolution of his recurrent priapism.\nThree years after his initial presentation, the patient returned to the ED with yet another episode of priapism. He reported accidentally taking 100 mg of quetiapine instead of his normal dose of amitriptyline the evening prior to presentation. He awoke with an erection and came to the ED after the erection failed to resolve spontaneously after 8 hours. Detumescence was again achieved with intracavernosal aspiration and injection of phenylephrine. Intracavernosal blood was found to contain quetiapine with a level of 502 ng/ml. The patient was discharged home and advised to dispose of any excess quetiapine.

[[43.0, 'year']]

M

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{'7676772-1': 1}

3935786-1

noncomm/PMC003xxxxxx/PMC3935786.xml

Betrayed Mood in Public View: Taking a MySpace History

A 19-year-old male was brought to the emergency department (ED) by the fire department after he was found wandering in the bus terminal, combative and agitated. He provided us with 2 different names and his city of origin but would not divulge any details such as family history, past medical or surgical history, current medications, social history or whether he had ingested any medications or taken any drugs prior to arrival. Among his belongings was an empty pill bottle labeled cyclobenzaprine, which had been prescribed to someone other than the name provided. He had 3 different identification cards in his possession, yet none of them had any photographs resembling him.\nOn examination, he was well-appearing, disoriented, agitated and mumbling anxiously. His heart rate was 130 beats per minute, blood pressure was 96/60 mmHg, temperature was 35.9°C (96.7°F), respiratory rate was 24 breaths per minute with an oxygen saturation of 96% on room air. His skin was flushed, warm and dry without obvious trauma or any other abnormalities. The pupils were mid-range and sluggish to light. Cardiac examination revealed tachycardia without murmurs, and the lung examination was clear to auscultation bilaterally. He had no abdominal tenderness. He displayed no focal neurologic deficits and moved all extremities spontaneously with 5/5 strength. Reflexes were +2/4 bilaterally. He was disoriented to place and time and after persistent requests to state his name, he gave the same name consistently as time progressed.\nA chest radiograph showed clear lung fields, and an ECG showed sinus tachycardia without QRS widening or QT prolongation. The patient became increasingly anxious, attempting to leave the room and becoming more hostile with medical personnel even after multiple trials of reorientation, reassurance and calming techniques. He would not provide us with any family contacts for further information. He was restrained for his own safety. A basic metabolic panel, a complete blood count, acetaminophen and aspirin levels, and a urine drug screen were ordered.\nAs we were unable to determine his true identity, we used the names he had given us to search the Internet. Upon entering one of the names on , an account that displayed a picture of our patient appeared. Positive identity was made after reviewing several photographs while matching his hometown to what was included as “location” on his profile. On his public profile, 3 days prior to presentation, he stated that he was “chillin in [a] motel room,” and his mood was “betrayed.” This enabled us to determine that he might have suffered a life stressor recently that eventually led to his visit to the ED that night.

[[19.0, 'year']]

M

{'2018200': 1, '9489272': 1, '19734511': 1, '19619037': 1, '18275317': 1, '9364758': 1, '19366321': 1, '17604833': 1, '20164062': 1, '19124701': 1, '18166570': 1, '19788375': 1, '24578766': 2}

{}

3936049-1

noncomm/PMC003xxxxxx/PMC3936049.xml

A Case of Eosinophilic Fasciitis Presenting as Pitting Edema of the Lower Extremities

A 19-year-old female visited our hospital with a 2 wk history of progressive pitting edema in both lower legs associated with a 2 kg weight gain. Symptom onset began shortly after intense physical exertion (dance practice). Her medical history included allergic rhinitis with sensitization to house dust mite and pollen, and she had visited the pediatric allergy clinic for about 10 years. None of her family members had a history of similar symptoms or specific diseases. Additionally, she noted mild pain in both knees and ankles, but tenderness was not remarkable on physical examination. She denied fever, dyspnea, chest pain, and urinary difficulty. She was not taking any medications. Physical examination revealed moderate bilateral pretibial pitting edema. No redness, induration, or thickened skin was observed. No contractures of joints were found, and muscle strength was normal in the upper and lower extremities.\nAt first, she visited the cardiovascular surgery clinic and was admitted for evaluation and treatment of deep vein thrombosis. However, computed tomography (CT) angiography of her lower extremities revealed no evidence of venous disturbance. Treatment with heparin and application of compression stockings did not alleviate her symptoms. Initial laboratory findings included a white blood cell count of 8,090 cells/µL with 28.9% eosinophils. Her eosinophil count was 2,340 cells/µL. Her hemoglobin level and platelet count were normal and her serum total IgE level was 786 kIU/L, but no remarkable change was observed compared to the previous year. The erythrocyte sedimentation rate and C-reactive protein level were not elevated. Serum urea nitrogen and creatinine were normal and urinalysis revealed no proteinuria or hematuria. Serum albumin was unremarkable at 4.2 g/dL. Serum protein electrophoresis results were within the normal range. Other laboratory data, including serum aminotransferase, alkaline phosphatase, cholesterol, and electrolyte levels, were normal. Further evaluations to determine the cause of her symptoms and peripheral eosinophilia were performed. There was no evidence of parasitic infection. A thyroid function test revealed normal thyroid function. The results of electrocardiogram and pulmonary function tests were normal. The patient was negative for rheumatoid factor and antinuclear antibody. An abdominal ultrasound showed no pathological changes. However, during the time of evaluation, the patient's blood eosinophil count increased rapidly to 4,880 cells/µL.\nThe patient was referred to our division of pediatric allergy considering the findings of peripheral eosinophilia and a past history of allergy, and a magnetic resonance imaging (MRI) was performed in her lower thigh. As shown in , the signal intensity of subcutaneous lesions was increased in fat-saturated T2-weighted images, suggesting inflammation of the subcutaneous fat layer and superficial fascia. Mild synovitis of both knees was also suggested. No evidence of thickening or fibrosis of the deep fascia was present. A biopsy of the right dorsal foot was performed, and the biopsy specimen included skin and underlying subcutaneous tissue. Biopsy examination revealed dermal edema and perivenular lymphoplasmacytic and eosinophilic infiltration in the subcutaneous fat tissue (). Based on the MRI and biopsy results, we diagnosed EF. We started intravenous corticosteroid therapy: dexamethasone 15 mg/day on the day of diagnosis. After 2 days of dexamethasone therapy, the symmetric pitting edema improved dramatically. The eosinophil count decreased rapidly from 4,880 to 140 cells/µL. We changed the intravenous dexamethasone to 30 mg/day of oral prednisolone, and the patient received an additional day of therapy with oral steroids. After a total of 3 days of intravenous plus oral steroid therapy, the patient recovered fully. She was discharged from the hospital without medication. Mild edematous changes in both legs developed 2 weeks later, but the patient's symptoms immediately relieved after administration of 30 mg/day prednisolone for 1day. She was monitored in an outpatient clinic for 4 months after cessation of therapy. The most recent eosinophil count was 320 cells/µL, and no relapses were observed.

[[19.0, 'year']]

F

{'15196276': 1, '17503330': 1, '9143512': 1, '9034156': 1, '11254454': 1, '9005985': 1, '20875077': 1, '8597956': 1, '2215562': 1, '15647285': 1, '15164069': 1, '28300895': 2, '9464841': 1, '3605835': 1, '11404819': 1, '11877470': 1, '20066174': 1, '1342912': 1, '3666445': 1, '12748907': 1, '10619836': 1, '17983872': 1, '31130746': 1, '10934091': 1, '12761043': 1, '16210640': 1, '24587957': 2}

{'5324994-1': 1}

3936050-1

noncomm/PMC003xxxxxx/PMC3936050.xml

A Case of Serum Sickness-Like Reaction and Anaphylaxis - Induced Simultaneously by Rifampin

A 25-year-old male was diagnosed with tuberculous meningitis at Chonnam National University Hospital, and was started on a treatment regime of 300 mg isoniazid (Yuhanizid®, Yu Han Pharm., Seoul, Korea), 800 mg ethambutol (Tambutol®, Chong Kun Dang Pharm., Seoul, Korea), 600 mg rifampin (Rifodex®, Chong Kun Dang Pharm.) and 1,500 mg pyrazinamide (Yu Han Pharm.) daily. He became nauseous, vomited, and had generalized abdominal discomfort 2 weeks later. He stopped taking the anti-tuberculosis drugs for 2 days, and the symptoms disappeared. He restarted taking the anti-tuberculosis drugs 2 days later but complained of pain and numbness in his extremities 30 minutes later. He was found to have conjunctival hemorrhage in both eyes, a maculopapular rash on the neck and anterior chest, and erythematous papules and vesicles on both hands.\nHe was admitted to the Department of Neurology. Laboratory tests revealed C-reactive protein, 10.9 mg/dL; erythrocyte sedimentation rate, 22 mm/h; white blood cells, 7,800/µL; eosinophils, 100/µL; hemoglobin, 15.8 g/dL; platelets, 230,000/µL; aspartate aminotransferase, 645 U/L; and alanine aminotransferase, 172 U/L. Levels of both C3 (62.9 mg/dL) and C4 (7.9 mg/dL) were low. Anti-nuclear antibody and anti-neutrophil cytoplasmic antibody were negative.\nThe anti-tuberculosis drugs were administered again. However, he developed a high fever of 39.1℃ 24 hours later, and his blood pressure decreased to 80/50 mm Hg. Pulse and respiratory rates increased to 120/min and 24/min, respectively. A maculopapular rash appeared on the neck and anterior chest and numbness and pain occurred in his extremities. A nerve conduction velocity test and electromyography showed bilateral, diffuse, axonal-type, and sensorimotor polyneuropathy. The first-line drugs were changed to second-line drugs, including 250 mg cycloserine (Closerin®, Dong-A Pharm., Seoul, Korea), 500 mg levofloxacin (Levofexin®, Il Dong-A Pharm.), and 1 g streptomycin (Chong Kun Dang Pharm.). Daily 50 mg prednisolone (Solondo®, Yuhan Pharm.) was added, and the symptoms and signs disappeared.\nBecause the first-line drugs were effective for treating the tuberculous meningitis, they were administered one at a time. Oral prednisolone was given continuously and then tapered off. Oral administration of 600 mg rifampin produced dyspnea, chest discomfort, and mild fever of 37.6℃ after 30 minutes. Blood pressure was 110/80 mm Hg, pulse rate was 132/min, respiration rate was 22/min, and oxygen saturation was 95%. The dyspnea and chest discomfort disappeared after 3 hours. Administration of 300 mg isoniazid or 800 mg ethambutol produced no symptoms. Administration of 1,500 mg pyrazinamide induced a fever of 38.3℃ after 8 hours. It was unclear whether the fever was due to the pyrazinamide or other causes related to the discontinuation of the oral steroid, because prednisolone was tapered off 1 day before administration of pyrazinamide. The drug responsible was not determined, and the patient was referred to the Department of Allergies for further evaluation.\nDrug provocation tests with pyrazinamide and rifampin were repeated. Pyrazinamide was given orally at 1 hour intervals at doses of 5, 15, 25, 100, and 355 mg, but no reactions were observed. Thereafter, rifampin was given at doses of 6, 30, 90, and 270 mg. Gastrointestinal symptoms, such as diarrhea and epigastric discomfort, developed 30 minutes after the administration of 270 mg rifampin and lasted for 24 hours. A fever of 37.5℃ occurred 3 hours after the administration and was maintained for 3 hours. Blood pressure decreased to 90/60 mm Hg 5 hours after the administration and to 70/40 mmHg 3 hours later, but returned to the normal range 15 hours later. These results confirmed that rifampin was responsible for the adverse symptoms (). The levels of both C3 (76.0 mg/dL) and C4 (9.5 mg/dL) were below the normal range 5 days after the rifampin challenge. Unfortunately, the complement levels were not measured again after the symptoms disappeared.\nRifampin skin tests were performed. Rifampin (600 mg) was dissolved in saline. The skin prick test was negative to 3 mg/mL, but the intradermal test showed an immediate positive wheal (8.0×7.6 mm) and flare reaction (22.2×22.0 mm) to 0.3 mg/mL. Saline and 1 mg/mL histamine were used as negative and positive controls, respectively. Intradermal tests to identify a non-irritating concentration (NIC) of rifampin were performed in nine normal controls. NIC was determined using the method described by Broz et al., with some modifications. NIC was considered if the ratio of rifampin-induced mean wheal diameter to histamine-induced mean wheal diameter was less than 0.4. All test subjects were male, aged 22±1.58 (mean±SD) years. NIC was 0.3 mg/mL ().\nIsoniazid, levofloxacin, ethambutol, and streptomycin were prescribed, but rifampin was not. No adverse drug reactions were observed. The tuberculous meningitis was finally cured without sequelae.

[[25.0, 'year']]

M

{'2810930': 1, '3877081': 1, '8067595': 1, '33539388': 1, '17715477': 1, '6450290': 1, '1433700': 1, '30915953': 1, '7897405': 1, '4126802': 1, '22435670': 1, '31049868': 1, '6387492': 1, '11221090': 1, '33546741': 2, '1580312': 1, '24587958': 2}

{'7866636-1': 1}

3936517-1

noncomm/PMC003xxxxxx/PMC3936517.xml

Wegener's Granulomatosis Presenting as Bilateral Otalgia with Facial Palsy: A Case Report

A 59-year old woman visited our hospital for otalgia, hearing loss of both ears beginning a month ago. On examination, otoendoscopic examination revealed thickened and hyperemic tympanic membrane with granulation of the both ear (). In initial blood tests, serum white blood cells was within normal limit (6470), erythrocyte sedimentation rate (ESR) and C-reactive protein (CRP) were elevated (33 mm/hr, 1.10 mg/dL)(). The rest of the routine hematological, biochemical tests and chest posteroanterior (PA) were normal.\nThe pure tone audiometry showed bilateral mixed hearing loss with bone conduction threshold 30-35 dB. Computed tomography scanning of the temporal bone showed abnormal soft tissue densities in both mastoid, middle ear, epitympanum with left side worse. The canal of facial nerve was intact throughout its course.\nIntravenous antibiotics treatment was started with impression of bilateral acute middle ear infection. On 6th day in hospital, we did myringotomy in left tympanic membrane and found yellowish granulation and scanty mucopus from middle ear. On 7th day, she expressed more otalgia and left facial nerve palsy (House-Brackmann grade III) was noticed. After considering aggravation of lest facial palsy (H-B grade V), hearing deterioration, and continuous elevation of ESR and CRP, we decided to perform left mastoidectomy on 11th day (). Intraoperative finding showed loose granulation tissues filling entire mastoid and middle ear, which were easily sucked out with suction tip. Tissues were collected for bacterial, fungus, tuberculosis culture and pathologic evaluation. After left mastoidectomy, her left hearing seemed to improve slightly because of reduced air-bone gap. But otalgia, facial expression, ESR and CRP didn't show any improvement. Eight days after left ear surgery, right otalgia was dramatic aggravated with development of right facial palsy (House-Brackmann grade V). Wide myringotomy was done. At this time point, pathologic report showed necrotizing granulomatosis with negative Acid Fast Bacillus, tuberculosis-polymerase chain reaction results (). We started high dose steroid treatment with suspicion of WG and checked serologic test including pancytoplastmic granular staining pattern anti-neutrophil cytoplasmic antibodies (c-ANCA) and rechecked chest PA. Follow-up chest PA showed enlarged both hilar mass lesions, which were not showed on initial chest PA (). Subsequent chest CT showed multiple consolidations in both lungs with narrowing of left bronchus (). Ophthalmological evaluation of her left eye revealed conjunctivitis and marginal keratitis ().\nSerology test showed elevated c-ANCA, antineutrophil antibodies, RA factor. Left facial expression started to recover. We transferred her to rheumatology department for further treatment. Her facial expression came back to normal function during follow-up, but both tympanic membrane perforation and intermittent ear discharge and conductive hearing loss have remained until last follow-up (up to 1 year from first visit).

[[59.0, 'year']]

F

{'34395012': 2, '18503861': 1, '27276989': 1, '7443265': 1, '8489162': 1, '7271127': 1, '1762779': 1, '11407453': 1, '14567060': 1, '27493820': 2, '24653903': 2}

{'8360741-1': 1, '4963591-1': 1}

3936520-1

noncomm/PMC003xxxxxx/PMC3936520.xml

A Case of Subcutaneous Hemangioma Presenting as a Preauricular Sinus

A 44-year-old female came to our department with a 5-month history of intermittent right preauricular swelling and febrile sensation. She denied having had hearing loss, aural fullness, otaglia, tinnitus, and painful sensation. Her medical, surgical and family history were unremarkable. Upon physical examination, an ovoid shaped mass was softly palpable on 1 cm anterior from the tragus of right ear. The other findings of the head and neck were not significant but particularly, there was no pit around the softly palpable mass. Also, there was no secretion and painful sensation with pressure. Computed tomography (CT) demonstrated a 0.9 cm sized soft tissue mass in anterolateral to the right external auditory canal, which enhanced with contrast. The isolated mass was ovoid shaped and well-marginated without evidence of bony erosion of outer ear (). Based on the physical examination and computed tomography, preauricular fistula, salivary gland tumor, and enchondroma were possible diagnosis.\nUnder general anaesthesia, the mass was smoothly excised via the preauricular approach. The standard technique for preauricular sinus excision was used with a careful elliptical incision anterior to the crus and tragus on the superior portion of the mass and dissection without epithelial remnants. The size of the surgical specimen was 1.0×0.8 cm, reddish and soft (). Pathologic evaluation revealed well circumscribed, exophytic mass, delineated by squamous epithelium and proliferative small vessels (). On the basis of clinical examination and histopathology, a diagnosis of capillary haemangioma was concluded. The patient visited our clinic after a week with normal healing. She was also reviewed 1 and 3 months after surgical excision without the leision recurred.

[[44.0, 'year']]

F

{'22627434': 1, '22500069': 1, '18940514': 1, '7710791': 1, '5061701': 1, '2013460': 1, '8486111': 1, '3290513': 1, '25426340': 1, '15165194': 1, '9269015': 1, '24653902': 2}

{}

3936521-1

noncomm/PMC003xxxxxx/PMC3936521.xml

Compound Nevus Occurring Near External Auditory Canal: Successful Treatment by CO2 Laser Abrasion

A 47-year-old Korean man presented with a 1-year history of a well-demarcated 1×1 cm, dark-brown to black papillomatous plaque in his right cavum conchae (). He didn't complain of any pain, itching, intermittent oozing, or hearing disturbance. On otoscopic examination, his right tympanic membrane and external auditory canal were normal. He was healthy and had no history of skin cancer or other skin problem. A skin biopsy specimen showed numerous nests and clumps of melanocytic nevus cells in the upper dermis and dermoepidermal junction (). The lesion was treated with CO2 laser (Model 1020C 20WA, Sharplan Lasers Inc., Migdal Haemek, Israel) abrasion at 1.0 watt in the super-pulsed mode. Only topical antibiotic ointment without coverage was applied for post-laser wound care for 1 week. The patient showed no recurrence during the 6-month follow-up period ().

[[47.0, 'year']]

M

{'15798434': 1, '16012861': 1, '16446902': 1, '33833888': 2, '24653901': 2}

{'8012142-1': 1}

3936523-1

noncomm/PMC003xxxxxx/PMC3936523.xml

Tuberculous Otitis Media with Facial Paralysis Combined with Labyrinthitis

A 44-year-old woman visited our department for evaluation of progressive left facial paralysis, dizziness, and hearing loss for 7 days with a 3-month history of an intermittent discharge from the left ear. On physical examination, facial paralysis was identified as Grade V using the House-Brackmann grading system. Endoscopic exam revealed total perforation of the tympanic membrane, edema of the middle ear mucosa, and profound discharge from her left ear (). Initial ear culture studies did not identified pathogens. On electronystagmography, spontaneous nystagmus toward the right side was identified and left-sided caloric weakness of 71% was found (). She denied a history of diabetes mellitus, hypertension, or pulmonary tuberculosis. Pure-tone audiometry showed deafness of the left ear. A temporal bone CT scan revealed that a soft tissue density filling the middle ear cavity and mastoid, and dehiscence of the tympanic segment of the facial nerve was suspected (). She was initially treated with an intravenous 3rd generation cephalosporin and a 4 day course of oral prednisone (60 mg/d) was started on the day of presentation and tapered off between days 10 and 12. Preoperative eletroneurography demonstrated 75% degeneration of the facial nerve. An emergent surgical procedure was scheduled to improve facial nerve paralysis and resolve dizziness. Under general anesthesia, retroauricular incision was performed. We removed a significant amount of granulation tissue with the consistency of cheese from the middle ear and mastoid by canal wall down mastoidectomy. During the operation, dehiscence of the facial fallopian canal and a swollen facial nerve were identified and facial nerve decompression was performed to resolve the facial nerve paralysis (). Intraoperative finding, all ossicles were surrounded by granulation tissues and incus was partially eroded. After malleus and incus were removed, tympanoplasty was performed using temporalis fascia. The most prominent finding on histopathologic examination was chronic caseating granulomatous inflammation and many bacilli were identified on AFB stain (). The chest X-ray was normal, but polymerase chain reaction testing for middle ear mucosa was positive for Mycobacterium tuberculosis. The patient was started on a 12-month course of empirical four-drug anti-tuberculosis treatment with oral rifampin, isoniazid, ethambutol, and pyrazinamide. The post-operative course was unremarkable and dizziness resolved by post-op day 7. The patient was discharged by post-op day 14. Nystagmus was un-remarkable on electronystagmography at a 3 month follow-up visit. The facial paralysis slowly improved until it had com-pletely resolved by 4 months post-op. However, there was no change in hearing threshold by 6 months post-op.

[[44.0, 'year']]

F

{'12472035': 1, '11489368': 1, '16220854': 1, '5115193': 1, '18852993': 1, '17011437': 1, '33786147': 1, '15195064': 1, '11866208': 1, '24653900': 2}

{}

3936524-1

noncomm/PMC003xxxxxx/PMC3936524.xml

A Case of Bilateral Sudden Hearing Loss and Tinnitus after Salicylate Intoxication

A 46-year-old female (42 kg) was taken to the emergency unit with her husband. Her mental status was drowsy and she was reported to have taken 200 tablets of aspirin (325 mg×200 tablets=65000 mg) with an alcoholic beverage (soju, alcohol content 19%, 200 mL). A blood sample was taken immediately and showed no abnormality of liver or renal function. The electrolyte profile was also within normal values but arterial blood gas analysis showed metabolic acidosis (pH 7.39, pCO2 33 mm Hg, and bicarbonate 21.7 mmEq/L). Urine chemistry was normal. A nasogastric tube was inserted and activated charcoal and intravenous fluids administered to remove the remaining salicylate in her body. Sodium bicarbonate was used for the correction of metabolic acidosis. She found that her hearing deteriorated and tinnitus developed on the second hospital day, and she was subsequently referred to our department. She was stable in terms of mental status, and her metabolic acidosis was corrected at the time of otologic assessment. Both middle ears were clear in otoscopic views. Neurological examination also revealed no abnormal findings. Pure tone audiometry (PTA) showed bilateral symmetric sensorineural hearing loss with a threshold of 50 dB HL in all frequencies, and tinnitus was equivalent to the narrow-band noise of 4 kHz and 5-10 dB SL bilaterally (). A high-dose oral steroid (prednisolone 60 mg/day) regimen was initiated following medical consultation. Hearing thresholds were unchanged on follow-up PTA and were 60 dB nHL by auditory brainstem response (). Distortion product otoacoustic emission showed no response on both ears (). On the fourth hospital day, PTA thresholds improved to the level of 36 dB HL on both ears () but she refused further admission therapy. She received a daily dose of 60-mg oral prednisolone during the 4 days of admission, which was tapered as scheduled. She revisited the clinic 3 days later and PTA thresholds were normalized to 18 dB HL (). However, she complained that her hearing had not fully recovered and tinnitus remained in the right ear. The tinnitus was equivalent to a pure tone of 125 Hz and 5-10 dB SL in the right ear. Monthly follow-up was scheduled without further medication; she last visited the clinic 4 months after intoxication. PTA thresholds were unchanged and she did not complain of hearing loss or tinnitus.

[[46.0, 'year']]

F

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{'4782479-1': 1}

3936526-1

noncomm/PMC003xxxxxx/PMC3936526.xml

Otogenic Brain Abscess Presenting with Gait Ataxia

A 67 year old male patient was admitted to the emergency room with dizziness and gait ataxia that had suddenly developed two days previously. The patient was receiving pharmacological treatment for hypertension and diabetes and there was no relevant family history. Other than the dizziness and gait ataxia, the patient complaining of headache, hearing loss and aural pressure in the left ear, and aural discharge but these did not accompany otalgia or tinnitus. The patient displayed a clear state of consciousness in neurological examination, his papillary reflex was normal, and nystagmus was not observed. The patient fell toward left even with his eyes open in the Romberg test and displayed dysmetria on the left side in the cerebellar function finger to nose test. Brain CT taken in the emergency room displayed the formation of a common wall, including the wall that enhanced the contrast in a circle of 5×5 cm toward the left cerebellum and a manifestation in which it pressed the brain stem and the fourth ventricle (). With the suspicion of brain abscess in the left cerebellum, the patient was admitted to the neurosurgical ward and received antibiotic treatment. Two days after he was admitted to hospital, his state of consciousness suddenly altered to drowsy and craniotomy and drainage of abscess was performed in response. As a result of requesting department of otorhinolaryngology for phy-sical examination in order to assess the causative diseases after surgery, it was found that the external auditory canal was narrow and aural discharge and granulation tissue were observed in the left ear. The right ear was normal. Tympanic mem-brane retraction and the aural discharge that accompanies keratin were also observed ().\nOn temporal bone CT taken with the suspicion of left otitis media cholesteatomatica, an overall destruction of the cochlea and vestibular organ in the left ear caused by cholesteatoma was indicated (). Air conduction hearing and bone conduction hearing of 36 dB respectively was presented for the right ear and hearing loss was found in the left ear when performing pure tone audiometry. The degree of paralysis in the left semicircular duct was 100% on the results of a caloric test using air. Under diagnosis of otitis media cholesteatomatica, which causes cerebellar abscess with overall invasion of labyrinth and intracranial expansion, cholesteatoma and granulation tissue were removed through transotic approach (). The surgery was performed a month after performing drainage of the abscess in neurosurgery. As the granulation tissue including keratin had infiltrated the mastoid antrum, auris media, and vestibular labyrinth and had destructed the bone structure, it was difficult to pinpoint the landmark that is seen during tympanomastoidectomy. Although the abscess was discharged within the cochlea, there was invasion into the internal acoustic canal. The junction between the mastoid antrum and posterior fossa was cut after connecting with fibrous tissue and there was no outflow of cerebrospinal fluid. As partial outflow of cerebrospinal fluid was found in the internal acoustic canal, this was stopped with soft tissue and the surgery was finished after filling the deletion by sampling fatty tissue from the stomach. As a result of assessing the bacterial culture from the abscess that was displayed in the mastoid antrum during surgery, group D enterococcus was cultivated and the result for acid-fast bacilli culture was negative. The patient was discharged from the hospital after recovery without neurological sequela and is currently under observation through follow-up CT without relapse ().

[[67.0, 'year']]

M

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{}

3936528-1

noncomm/PMC003xxxxxx/PMC3936528.xml

A Case of Middle Ear Adenoma

A 17-year old male presented with a two-year history of tinnitus and intermittent bloody otorrhea in the left ear. He had no definitive abnormal findings based on his past medical history. He had no accompanying symptoms such as otalgia, dizziness or facial paralysis.\nThe right tympanic membrane was normal. The left tympanic membrane was not perforated but was observed as having a milky-white, middle ear polyp which has approximately 3/4 of the eardrum protruded ().\nPure-tone audiometry showed normal hearing in the right ear and mixed hearing loss in the left ear with a 32-dB air-bone gap and a 37 dBHL air conduction threshold. Temporal bone CT findings showed a pneumatized mastoid cavity, although there was a soft tissue density which caused the eardrum to protrude outwardly, and filling of the inside of the mastoid antum, middle ear, and the epitympanum was observed. No structural damage of the ossicle, facial canal or lateral semicircular canal was identified ().\nAs left middle ear tumor or congenital cholesteatoma was suspected, we performed intact canal wall mastoidectomy with tympanization. According to the operative findings, a grayish-white multilobular mass and granulation tissue fully filling the mesotympanum, epitympanum, and mastoid cavity were detected (). The multilobular mass and granulation tissue were adjacent to the ossicle, although there was no ossicle erosion, and the hypotympanum and orifice of the Eustachian tube were preserved. No mass invasion into the sinus tympani was observed. Intact canal wall mastoidectomy was then performed, and the mass inside the epitympanium was excised by removing incus.\nTissue biopsy was performed to confirm the safety margin at the outer surface of the tympanic membrane which protruded to the external auditory canal because of the mass, mucosa of hypotympanum and epitympanum.\nIn these tissues, no tumor cells were observed. Ossiculoplasty to restore hearing was not performed. Rather, a temporalis muscle facial graft was performed.\nPostoperative histopathologic results revealed a small, round-cell tumor composed of even-sized, globular cells with plenty of eosinophilic cytoplasm arranged in a platy structure. There were no findings of mitosis, necrosis or dysplastic were.\nTo obtain the differentiation diagnosis of small, round-cell tumor, immunohistochemistry was performed. Based on the findings which were negative to the Ki-67 antigen, positive to synaptophysin, mildly positive to CD56, negative to chromogranin, and negative to S-100 protein, middle ear adenoma was diagnosed ().\nIn the three weeks following surgery, all of the packing material in the external auricular canal was removed, and two months after the surgery, the eardrum was well-grafted and stabilized. Two and seven months after surgery, temporal bone CT and pure tone audiometry were both performed. Pure-tone audiometry showed 8 dBHL of the left bone conduction threshold and 32 dB of the air conduction threshold, both of which were similar to the patient's preoperative hearing level, however, the patient's tinnitus had improved. According to the temporal bone CT performed seven months after the surgery and otoscopy which was performed two years following the surgery, there was no evidence of recurrence.

[[17.0, 'year']]

M

{'16148713': 1, '2782809': 1, '20345079': 1, '6156597': 1, '1031337': 1, '2782545': 1, '16831038': 1, '8247568': 1, '19476759': 1, '11214650': 1, '20111612': 2, '4036884': 1, '24653866': 2}

{'2812983-1': 1}

3936529-1

noncomm/PMC003xxxxxx/PMC3936529.xml

A Case of Jugular Bulb Diverticulum Invading the Internal Auditory Canal

A 54-year-old female presented with acute onset of severe whirling-type vertigo. The vertigo developed abruptly when the patient got out of the car and aggravated with positional change. The patient also complained of left pulsatile tinnitus like a "snoring" sound. Nausea and vomiting were accompanied initially, however, disappeared within 3 days. There were 5 episodes of vertigo, during the last 5 years. The vertigo was associated with some mental/physical exhaustion and the patient recovered in several days with no medical treatment. The patient denied otalgia, otorrhea and hearing loss.\nThe patient did not report any specific medical or surgical history but hyperlipidemia. The otoscopic and neurologic exams were unremarkable.\nSpontaneous horizontal nystagmus was observed toward the left side at first and changed toward the right side on the fifth hospital day. Pure-tone audiometry on the third hospital day showed SNHL of about 60 dBHL with descending type over 1 kHz and speech discrimination was 28% on the left side (). Vestibular evoked myogenic potential (VEMP) testing was performed on the fifth hospital day and showed to be normal. Saccade test, pursuit test and optokinetic test presented normal ranges, but caloric test showed the left canal paresis of 21% on the third hospital day and 73% on the eighth hospital day.\nMagnetic resonance imaging (MRI) was ordered to rule out the possibility of the left cerebellopontine angle (CPA) tumor. The official report did not identify any abnormal finding. However, we suspected high jugular bulb encroaching the IAC on the left side (). Computed tomography (CT) of the temporal bone revealed high jugular bulb and jugular bulb diverticulum with the eroded IAC on the left side ().\nWhen the left high-frequency SNHL was found on the third hospital day, we started to administer 5 mg dexamethasone, intravenously, three times a day, in a regimen of management for sudden sensorineural hearing loss (SSNHL). Even after JBD was diagnosed by radiologic evaluations, the patient received high-dose steroids with the forementioned method for 9 days and underwent a 5-day oral taper, expecting the anti-inflammatory effect of corticosteroids for the cochlear/vestibular nerve.\nOn the twenty-fifth hospital day, pure-tone audiometry showed high-frequency SNHL with partially recovered threshold of 40 dBHL and speech discrimination score was 72% on the left side. In a month after initial presentation, the patient still had positional vertigo and nystagmus, with newly developed benign positional paroxysmal vertigo and recovered completely in several days with repositioning maneuvers. Over 21 months after the onset, there has been no recurrence of vertigo, tinnitus or aggravated hearing deterioration.

[[54.0, 'year']]

F

{'3947273': 1, '19924772': 1, '17847732': 1, '15899333': 1, '12646841': 1, '34584695': 1, '19358118': 1, '6865626': 1, '31583209': 2, '3101406': 1, '3096082': 1, '10740182': 1, '7556255': 1, '33408959': 1, '7863937': 1, '24653869': 2}

{'6761058-1': 1, '6761058-2': 1, '6761058-3': 1}

3936541-1

noncomm/PMC003xxxxxx/PMC3936541.xml

Two Cases of Acute Mastoiditis with Subperiosteal Abscess

A 2 year old female admitted to the emergency department due to 2 days of right postauricular area swelling. The patient was previously treated with antibiotics for a week at a local clinic for acute otitis media. Despite antibiotic treatment, the patient suffered from right otalgia and fever upon admission. Through physical examination, bulging of the tympanic membrane and erythematous change and swelling of the right postauricular area with tenderness were noticed. Further evaluation by temporal bone CT revealed soft tissue density filling the right middle ear and mastoid cavity with erosion of the cortical bone along with formation of subperiosteal abscess (). Under the impression of acute mastoiditis with subperiosteal abscess, percutaneous aspiration was performed on the postauricular area resulting in 3 cc of purulent fluid. The procedure was followed by incision and drainage (I & D) and oral antibiotics were prescribed to the patient. On third day after I & D, the patient showed persistent redness, tenderness, and swelling of the postauricular area () and injected bulging tympanic membrane, regardless of the treatment. Therefore, it was postulated that I & D and oral antibiotics were inadequate and the patient was admitted to our department for intravenous antibiotics therapy.\nOn the second day of hospitalization, the patient underwent surgical treatment of right simple mastoidectomy and ventilation tube insertion because of consistent symptoms and physical findings in spite of 2 days of intravenous antibiotic treatment. Intraoperative finding showed massive granuloma beneath the postauricular area and in the mastoid cavity (). Erosion of cortical bone was also noticed after Palva flap elevation. Intraoperative culture study resulted in Streptococcus pneumoniae which was only susceptible to vancomycin. Intravenous antibiotic treatment with vancomycin was immediately initiated and the child was discharged after 13 days of surgery without any postoperative complications. During the 5 month follow up, the patient showed no signs or symptoms of recurrence.

[[2.0, 'year']]

F

{'8630206': 1, '15895789': 1, '15908017': 1, '31750180': 1, '4038726': 1, '17493691': 1, '10576611': 1, '11165635': 1, '11997153': 1, '16413617': 1, '19620582': 1, '9018253': 1, '16402975': 1, '24653915': 2}

{'3936541-2': 2}

3936541-2

noncomm/PMC003xxxxxx/PMC3936541.xml

Two Cases of Acute Mastoiditis with Subperiosteal Abscess

A 41 year old male visited the emergency department suffering from right otalgia for 1 month. The patient had no known medical history and complained of hearing impairment and otalgia on the right side. Physical examination showed injected bulging tympanic membrane associated with right postauricular swelling, redness and tenderness (). Further evaluation through pure tone audiometry revealed a mixed hearing loss on the right side with 60 dB air conduction level and 30 dB air-bone gap. Radiologic examinations of temporal bone CT and MRI showed soft tissue density filling the right middle ear and mastoid cavity with cortical bone erosion. Subperiosteal abscess was also detected over the cortical bone (). The patient was admitted and treated with I & D and antibiotics. Culture study that was processed during I & D revealed penicillin-sensitive Streptococcus pneumoniae. Even after 7 days of hospitalization, there were no signs of improvement. Thus, on the 8th day of hospitalization, right canal wall up mastoidectomy and tympanoplasty were performed. During the operation, granulation tissue in the mastoid cavity and middle ear cavity was detected and removed completely (). The patient was discharged after a week of surgery with significant improvement of previous symptoms and physical findings. No suspicious signs of recurrence were encountered during the 1 year follow up.

[[41.0, 'year']]

M

{'8630206': 1, '15895789': 1, '15908017': 1, '31750180': 1, '4038726': 1, '17493691': 1, '10576611': 1, '11165635': 1, '11997153': 1, '16413617': 1, '19620582': 1, '9018253': 1, '16402975': 1, '24653915': 2}

{'3936541-1': 2}

3936545-1

noncomm/PMC003xxxxxx/PMC3936545.xml

A Case of Spontaneous Temporomandibular Joint Herniation into the External Auditory Canal with Clicking Sound

A 53-year-old man came to the Otorhinolaryngology department as an outpatient in September 2011 for the chief complaint of clicking tinnitus on the left. The patient had a past medical history of arrhythmia and heart valve disease but there was no history of trauma or surgery which could have led to tinnitus. Since July 2008, he had received rehabilitation treatment due to cerebral infarction in the middle cerebral artery zone. The clicking tinnitus started suddenly 3 weeks before he visited the hospital, mostly during mastication and when opening and closing his mouth. During the 3 weeks, the clicking tinnitus happened without strength change and without other otologic symptoms such as otalgia and otorrhea. In the otoscopic examination, the tympanic membrane was normal but there was slight protrusion of the soft tissue, originating from the anterior wall of the left EAC. This protrusion was observed when the mouth was closed and if it was open, the protrusion retracted (). The clicking sound that was heard in the left ear of the patient was also heard by the observer when he put a stethoscope on the EAC of the patient when the mouth was opened and closed.\nThe patient did not feel uncomfortable in his daily life from the clicking tinnitus, and the Tinnitus Handicap Inventory (THI) score was 18. According to the pure tone audiometry, the air conduction average threshold value (4 division method) was 23 dB on the right and 22 dB on the left while the average threshold value from bone conduction audiometry was 21 dB and 20 dB respectively. On the impedance test, both sides were A type. We conducted a tinnitogram but the patient complained of just clicking and we failed to find the exact strength and frequency.\nAccording to his otologic history, he had clicking at the time of mastication. Pure tone audiometry also failed to detect sensorineural or conductive hearing loss while the otoscopy found an anterior wall protrusion at the left EAC, and so TMJ herniation was suspected. Therefore, we conducted TMJ X-ray and temporal computed tomography. From the TMJ X-ray, we could see a left temporomandibular joint herniation at the time of mastication (), and the temporal bone computed tomography detected a 6 mm bony defect in the left EAC anterior wall and subsequent soft tissue herniation (). At the right EAC, there was no bony defect or other unusual findings. The left clicking tinnitus complained by the patient was revealed to occur from TMJ tissue herniation into the EAC at the time of mastication. Therefore, we considered surgical treatment for the left EAC anterior wall defect but the patient refused surgery because he felt uncomfortable with surgical treatment as he was receiving rehabilitation for cerebral infarction, and furthermore, he did not suffer much inconvenience from the clicking tinnitus. Thus, the authors treated him with an anxiolytic and muscle relaxant for 2 weeks, after which we could not see any change in loudness of the tinnitus (according to the patient) and emotional reaction. Now, the authors are observing the progress of the clicking tinnitus.

[[53.0, 'year']]

M

{'20939076': 1, '1437197': 1, '10845043': 1, '8572137': 1, '2685214': 1, '1920577': 1, '16369162': 1, '28560034': 1, '27942877': 1, '19545461': 1, '15956489': 1, '28127148': 1, '27504269': 1, '31395661': 1, '24653913': 2}

{}

3936547-1

noncomm/PMC003xxxxxx/PMC3936547.xml

Bilateral Ossiculoplasty in 1 Case of Achondroplasia

A 12 year-old female patient suffering from persistent hearing loss after birth visited the outpatient clinic for evaluation and treatment of the hearing loss. The patient showed typical features of achondroplasia, such as short limbs, especially the proximal segment, with a long trunk, narrow thorax, large head with frontal bossing, and midfacial hypoplasia at birth. Although there was no one with achondroplasia in the family, and a genetic study of FGFR3 was not performed, the typical features of achondroplasia in the patient and the short statue were enough to diagnose the achondroplasia. The patient had suffered from frequent otitis media since childhood, and had a history of bilateral tympanostomy tube insertion 7 years ago. Both tympanostomy tubes were extruded 2 years after insertion. Because she showed a feature of sensorineural hearing loss, she was referred to our hospital for consultation for hearing rehabilitation, such as the use of a hearing aid.\nThe right tympanic membrane was intact and there was a large perforation in the left tympanic membrane. Pure tone audiometry (PTA) was performed and the hearing threshold averaged from 0.5 kHz, 1 kHz, 2 kHz, and 4 kHz. The air conduction hearing threshold was 82 dB HL on the right and 52 dB HL on the right, and the bone conduction hearing threshold was 46 dB HL on the left, and 44 dB HL on the left. The type of hearing loss was mixed hearing loss (). Speech recognition threshold and the speech discrimination score were 70 dB HL and 96% on the right, and 50 dB HL and 100% on the left, respectively. On auditory brainstem response testing, there was prolonged interlatency of the right side, while the left side was normal (). The patient was recommended to use a hearing aid on the right side and have tympanoplasty for the left side. But the patient denied further treatment and was lost.\nThe patient re-visited the outpatient clinic 5 years later after the first visit. The right ear had been operated at another hospital but she still complained of hearing loss on the right side, and she suffered from intermittent otorrhea and otalgia of the left ear. There were no specific findings of the right ear. The tympanic membrane of the left ear was perforated and the margin of the perforation was attached to the mucosa of the middle ear. Air conduction and bone conduction hearing threshold were 65 dB HL and 17 dB HL for the right ear, and 30 dB HL and 12 dB HL for the left ear, respectively (). Temporal bone computed tomography was performed. The right ear was well pneumatized, and was in a canal-wall-up mastoidectomized state. There was no incus and malleus head at the epitympanum, and foreign material, estimated to be material of the implanted prosthesis, was visible in the mesotympanum. There was no abnormal lesion in the middle ear cavity. Although the left ear was well pneumatized, a lesion with soft tissue attenuation partially filled the mastoid air cells. The incus and malleus were normal in appearance ().\nRight exploratory tympanotomy was planned first because the air-bone gap was larger and the state of the tympanic membrane was better than in the left ear. A previously inserted total ossicular replacement prosthesis (TORP) was found during the operation, and the TORP was displaced from the stapes footplate. Curetting of the posterior-superior portion of the bony tympanic ring was done and a stapedial superstructure was observed. All stapedial superstructures were intact and located at the posterior-superior side of the middle ear cavity. A round window reflex during stapes palpation was observed. A partial ossicular replacement prosthesis (PORP) made of titanium was inserted on the stapes head and the conchal cartilage was interpositioned between the tympanic membrane and prosthesis head (). Gelfoam was packed into the external auditory canal, but not in the middle ear cavity. The whole of the gelfoam was removed two weeks after the operation, and postoperative PTA was performed four weeks after the operation: the hearing threshold was 22 dB HL on the right, and 38 dB HL on the left (). A second PTA was performed three months after the operation, and the hearing threshold was 21 dB HL on the right, and 36 dB HL on the left ().\nAfter confirmation of correction of hearing in the right ear, an operation on the left ear was planned. During the operation on the left ear, a large sized central perforation with adhesion of the perforation margin to the middle ear mucosa was observed. Although all ossicles were normal and their continuity was intact, the mobility of the ossicles was decreased during palpation. The incudostapedial joint was separated and the distal portion of the long process of the incus was removed because of decreased motility. The PORP was interpositioned between the stapes head and the handle of the malleus, and the conchal cartilage was also interpositioned on the prosthesis head to avoid contact of the prosthesis with the tympanic membrane. The temporalis muscle fascia was inserted via the underlay method, and gelfoam was packed into the middle ear cavity and external auditory canal. The gelfoam was removed on the postoperative 14th day. PTA was performed three months after the second operation, and the hearing threshold was 16 dB HL on the right, and 26 dB HL on the left (). The patient was observed at the outpatient clinic.

[[12.0, 'year']]

F

{'17372080': 1, '8456822': 1, '3559799': 1, '8880574': 1, '3143244': 1, '458831': 1, '7287315': 1, '5504223': 1, '3877092': 1, '3877108': 1, '1914954': 1, '21324899': 1, '3240252': 1, '24653923': 2}

{}

3936549-1

noncomm/PMC003xxxxxx/PMC3936549.xml

A Case of Auricular Chondroma

52-year old male patient visited our clinic presenting with an unilateral mass of the auricle for several years. Physical examination revealed a 1.5×1.5 cm sized painless, hard mass accompanying a nodular irregularity, located on the superior portion of the helix (). Surgical removal was performed under a local anesthesia. A 1.0×0.8 cm sized firm, round, and well-encapsulated mass was completely removed by wedge excision (), and the remaining defect was approximated with a creation of Burow's triangle in the scapha (). Histopathologic examination revealed a mass consisted of an acidophilic hyalinized matrix with cartilage cells scattered among them, and in the center, local calcification was observ-ed. Tumor cells had cytoplasms with a distinct border, and the nuclei were of a circular or an oval form; atypical or abnormal mitosis was not observed (). There was no recurrence at the 12-month follow-up.

[[52.0, 'year']]

M

{'8356273': 1, '29966951': 1, '76505': 1, '21119479': 1, '25117074': 1, '3351074': 1, '22844293': 2, '18593455': 1, '24653926': 2}

{'3400396-1': 1}

3936551-1

noncomm/PMC003xxxxxx/PMC3936551.xml

Two Cases of Malleostapedotomy in Congenital Oval Window Atresia

A 40-year-old male visited our outpatient clinic with right-sided hearing loss since childhood. The patient had no previous history of trauma, and showed conductive hearing loss with an air-bone gap (ABG) of 50 dB (60/10 dB; ). Temporal bone computed tomography (TBCT) showed normal images of the malleus, but revealed atresia of the right-side oval window, with abnormal placement of the facial nerve (). First, ossiculoplasty using a total ossicular replacement prosthesis (TORP) was performed, with no hearing improvement. We then planned a second operation, a malleostapedotomy. The tympanic cavity was exposed through an endaural approach and the posterior wall of the external auditory canal was leveled down with a mallet and gauge to expose the stapes. During the operation, a normal structure of the malleus was noted, but the long process of the incus was short. Additionally, the stapes was missing, and the facial nerve was covering the oval window. The facial nerve was displaced towards the cephalic side using a fine swab, and a 0.6-mm window was drilled in the obstructed oval window using a 0.4-mm Skeeter drill. The facial nerve was then put back in the original position. Because the malleus was placed anterior to the stapes footplate, the piston wire prosthesis was bent 20°, modified to a 7-mm length, and fixed on the normal malleus (). At 1 year following the operation, pure tone audiometry showed improvement of the ABG, from 50 to 30 dB (), with no complication of facial nerve palsy or dizziness.

[[40.0, 'year']]

M

{'1589221': 1, '18898217': 1, '9051082': 1, '18199954': 1, '8352484': 1, '12825026': 1, '11698795': 1, '3260444': 1, '22664905': 1, '2094227': 1, '6967577': 1, '8065761': 1, '24653925': 2}

{'3936551-2': 2}

3936551-2

noncomm/PMC003xxxxxx/PMC3936551.xml

Two Cases of Malleostapedotomy in Congenital Oval Window Atresia

A 10-year-old male visited our outpatient clinic with bilateral hearing loss for several years. Audiometric evaluation showed bilateral conductive hearing loss (), and TBCT revealed bilateral atresia of the oval window, with an abnormal array of the facial nerve (). Explo-tympanoplasty was performed primarily on the left ear with an ossiculoplasty using a TORP; there was no hearing improvement. A stapedotomy was planned using a retroauricular approach. During the operation, the malleus showed normal images, but the long process of the incus was short. In addition, the stapes were missing, and the facial nerve was lying over the obstructed oval window. The facial nerve was cephalically rotated using the "facial roll" method, and a 0.6-mm window was created at the center of the stapes footplate using a 0.4-mm Skeeter drill. A 7-mm-long piston wire was fixed on the malleus and tightened with piston forceps (). Immediately postoperatively, the patient showed no dizziness or loss of taste, but mild facial palsy, House-Brackmann Grade III, was noted. By 1 month later, the facial palsy had recovered, to Grade I, and the ABG was reduced, from 40 dB (pre-operative) to 15 dB (6 month post-operative) on pure tone audiometry ().

[[10.0, 'year']]

M

{'1589221': 1, '18898217': 1, '9051082': 1, '18199954': 1, '8352484': 1, '12825026': 1, '11698795': 1, '3260444': 1, '22664905': 1, '2094227': 1, '6967577': 1, '8065761': 1, '24653925': 2}

{'3936551-1': 2}

3936553-1

noncomm/PMC003xxxxxx/PMC3936553.xml

Repair of Spontaneous Cerebrospinal Fluid Otorrhea from Defect of Middle Cranial Fossa

A 49-year-old woman was referred to our hospital because of right pulsatile otorrhea after myringotomy. Initially, she had experienced right ear fullness and hearing disturbance for 2 weeks. When a local otolaryngologist examined her ear, there was visible fluid through the tympanic membrane. Thus, she was diagnosed as right serous otitis media and treated with antibiotics. However, the fluid remained despite a 2-week treatment. Myringotomy was attempted but failed due to profuse pulsatile clear discharge from the myringotomy site.\nThe pure tone audiogram of the right ear revealed conductive hearing loss of 40 dB (air-bone gap 32 dB). Her body weight was 70 kg and body mass index was 27.7 kg/m2. She had no specific past medical history and family history. High resolution computed tomography (HRCT) scan and magnetic resonance imaging (MRI) of temporal bone were performed for further evaluation of the otorrhea. The HRCT scan revealed a large, bony, anteromedial defect from the anterior wall to the roof of the right epitympanum (). The right middle ear cavity and mastoid cavity were filled with soft tissue density. T2 weighted MRI demonstrated high signal intensity in right middle ear and mastoid cavity, consistent with CSF ().\nRepair of CSF leak was planned through a transmastoid approach. First, lumbar drainage was done after general anesthesia. Temporalis muscle fascia was harvested. Several pieces of bone chips were taken from mastoid cortex. A cortical mastoidectomy was performed. On entering the antrum, a profuse clear discharge from epitympanum was noted. To further evaluate the tegmen tympani, the incus and head of malleus were removed. An approximately 8 mm bony defect was apparent from the anterior roof of the epitympanum extending to the anterior wall. There was no identifiable any healthy dura or brain tissue herniation along with the CSF leak site.\nCovering the defect began with insertion of prepared temporalis fascia into the bony defect. After that, large piece of bone chip was inserted into the defect to cover the fascia. The bony defect was still visible because of the large defect size. Other bone chips were placed in the defect, unless there was no evidence of the CSF leak. Surgical glue was applied around bone chips and gelfoams were packed in the cavity.\nA postoperative CT scan demonstrated that the defect had sealed and revealed a pneumocephalus in the right temporal lobe (). The patient did not complain of headache or any neurologic deficits. Lumbar drainage was maintained for 6 days. Two weeks later, the pneumocephalus had disappeared and the patient was discharged. One year later, CT scan showed a well-healed defect (). There has been no evidence of recurrent CSF leak up to date.

[[49.0, 'year']]

F

{'2241009': 1, '15224872': 1, '1449180': 1, '19029865': 1, '7213189': 1, '16900803': 1, '3520197': 1, '17921911': 1, '20684056': 1, '18000466': 1, '24653924': 2}

{}

3936554-1

noncomm/PMC003xxxxxx/PMC3936554.xml

Pulsatile Tinnitus with a Dural Arterio-Venous Fistula Diagnosed by Computed Tomography-Angiography

A 43 year-old woman was admitted to hospital with headache of two months' duration and left-sided pulsatile tinnitus that occurred suddenly one month ago and had become severe. She complained of a constant rhythmic sound that beat in synchrony with her heartbeat. The sound became louder in her ear, making it difficult to sleep. On admission, the patient did not have any otologic symptoms such as otorrhea, dizziness and hearing loss except ear-fullness in the affected ear. She had no previous history of surgery, head and neck trauma, or systemic diseases. Based on the results of a questionnaire in which the answers were measured on a 10-point visual analog scale (VAS), her intensity of tinnitus was 7, tinnitus-induced annoyance was 8 and the effects of the tinnitus on her life was 6, showing a high level of discomfort. The tinnitus handicap inventory (THI) was also high, as much as 76. The physical examination revealed no mass in the head and neck, and the otoscopy exhibited no abnormal characteristics of the tympanic membranes. No sound waves were detected by auscultation. The tinnitus sound became weaker when the patient turned her head to the left and when her neck was pressed with the hand. The result of the pure tone audiometric evaluation was 11 dB in the right ear and 18 dB in the left ear. The impedance audiometry produced a Type A curve for both ears. In the tinnitogram, the patient showed pulsatile tinnitus in the left ear. And the sound pressure was 24 dB over the frequency of 1000 Hz. The results of the auditory brainstem response test and distortion product otoacoustic emission test were normal.\nBased on the patient's condition and test results, pulsatile tinnitus was suspected, and CTA was performed as a screening test. In the CTA, a prominent vascular structure was observed along the left tentorial margin, and focal stenosis at the transverse-sigmoid junction was present. Distension of the left occipital artery was also observed (). The CTA findings suggested the possibility of a DAVF, and a 4-vessel CA was performed for the diagnosis.\nAngiography of the left internal carotid artery revealed no abnormal findings in the arterial phase. However, the left external carotid angiogram revealed an enlarged occipital artery. Also, the blood drained to the left transverse sinus and the meningeal vein passing through the transcranial route, extending from the left occipital artery, middle meningeal artery, posterior auricular artery, and superficial temporal artery. The diagnosis of DAVF, fed by the left external carotid artery (ECA), and draining into the left transverse sinus, was therefore made (). Although excessive drainage into the cerebral cortex or brainstem was not observed, transarterial embolization was performed due to the severity of the tinnitus symptoms and the high level of discomfort. First, a 6-Fr guiding catheter was placed through the left ECA to identify the enlarged occipital artery for catheterization. Embolizations with ethylene-vinyl alcohol copolymers (ONYX) were performed to embolize the four feeding arteries. Embolization of the middle meningeal artery was performed to embolize the fifth feeder, followed by transcranial arterial embolization in the proximal portion of the superficial temporal artery. A total of six feeding arteries were embolized. Post-embolization angiography revealed blood flow in fine arteries branching off from the posterior auricular artery and occipital artery. However, the feeders were too small and numerous to perform embolization on. During embolization, the pulsatile tinnitus disappeared, and no post-embolization complications were found. During the outpatient follow-up, the patient did not show tinnitus symptoms but claimed minor fullness in the ear. A THI questionnaire was conducted 3 months postoperatively, and the score for tinnitus-induced discomfort was 0. However, the VAS score for annoyance associated with ear-fullness on the affected side was 2.

[[43.0, 'year']]

F

{'8059655': 1, '12105359': 1, '9661757': 1, '11922702': 1, '1590181': 1, '9553842': 1, '28942629': 2, '18842759': 1, '20950972': 1, '21402614': 1, '19185662': 1, '10565875': 1, '17320776': 1, '20152277': 1, '7666234': 1, '24653921': 2}

{'5621796-1': 1}

3936555-1

noncomm/PMC003xxxxxx/PMC3936555.xml

A Case of Auditory Neuropathy with Recovery of Normal Hearing

A six-year-old boy presented to the outpatient clinic with abnormalities of hearing and language development. The patient was born vaginally at 40 weeks. No congenital anomalies were evident at birth. However, he was not able to speak until 16 months of age and his response to external sound stimuli was slow. He showed no ABR-response to 65 dB nHL click stimuli at 16 months age. Since then he had worn a hearing aid in the right ear (). At 25 months of age, aided hearing threshold was about 30 dB HL in sound field pure tone audiometry (). He wore the hearing aid for another 2 years.\nAt 5 years, 10 months of age, he visited our hospital for a hearing evaluation. His parents related that his hearing function seemed to have improved with time, because he had no difficulty communicating with others without a hearing aid. Physical examination revealed that both tympanic membranes were normal. ABR test showed no click sound response in both ears at 90 dB nHL (). Transient evoked otoacoustic emission showed response reproducibility of 88% (right ear) and 95% (left ear), and normal distortion product otoacoustic emissions responses were reported (). These results were compatible with auditory neuropathy. However, normal pure tone threshold was evident at both ears; speech reception threshold was 22 dB (right ear) and 26 dB (left ear) and speech discrimination score was 96% (right ear) and 88% (left ear) (). He was recommended to stop wearing the hearing aid. A language development test conducted at 7 years, 4 months of age revealed an age equivalent of 6 years on expressive vocabulary test and 5 years on receptive vocabulary test. Language developmental delay and mild phonological disorder were diagnosed. He is still participating in language therapy

[[6.0, 'year']]

M

{'21466261': 1, '20593543': 1, '12220206': 1, '21940978': 1, '20053155': 1, '12495954': 1, '16769129': 1, '21493430': 1, '8673487': 1, '10890762': 1, '24653922': 2}

{}

3936559-1

noncomm/PMC003xxxxxx/PMC3936559.xml

A Case of Basal Cell Carcinoma of External Auditory Canal

A 55-year-old female in otherwise good health presented with a 10-year history of an intermittent otorrhea in the right ear.\nA physical examination revealed a small nodular mass arising from the posterior inferior aspect of the right cartilaginous portion of the EAC (). Right tympanic membrane was intact.\nOther clinical examinations including the head and neck region, laboratory findings, and audiologic findings were within normal limits. It appeared to be a benign lesion of EAC, an excisional biopsy was performed under local anesthesia. Contrary to expectation, histologic examination was interpreted as a basal cell carcinoma (). Fortunately, surgical margin was negative for malignancy. Further evaluation of the temporal bone by computed tomography (CT) and a magnetic resonance imaging () of the head and neck showed no involvement of any other temporal bone or of head and neck structures.\nAlthough less probable, because distant metastasis cannot be ruled out completely, a full metastatic workup was done. Bone scan, hepatobilliary sonography, duodenoscopy, chest X-ray, and CT of the chest, PET-CT were all negative for signs of nodal involvement or metastases to other organs.\nAccording to the Pittsburgh staging system, the patient had tumor limited to the EAC without bony erosion or evidence of soft tissue involvement (T1), no lymph node metastases (N0), and no distant metastases (M0).\nThe patient underwent sleeve resection of the EAC reconstructed with a coverage of the EAC using a left thigh split-thickness skin graft (). Pathologic findings confirmed a basal cell carcinoma in situ not invading cartilage, all surgical margins being free of invasion (). No adjuvant chemotherapy or radiation was administered.\nAfter 1 year of surgery, the patient is in good health with no evidence of complication or recurrence ().

[[55.0, 'year']]

F

{'14515088': 1, '7917206': 1, '30039068': 2, '15925819': 1, '10912706': 1, '13103895': 1, '19761431': 1, '2396807': 1, '8134137': 1, '1909009': 1, '8893219': 1, '12117346': 1, '9236591': 1, '24653879': 2}

{'6032558-1': 1}

3936561-1

noncomm/PMC003xxxxxx/PMC3936561.xml

A Case of Bilateral Postauricular Sinuses

A 27 year old male visited the department of Otorhinolaryngology-Head and Neck Surgery, Chung-Ang University hospital, with complaints of painful swelling, erythema, and tenderness on the left retroauricular area. He had a history of incision and drainage twice in the right ear, and once in the left ear due to similar symptoms in the past. On physical examination, there was swelling and tenderness in the left retroauricular area. Further, in the center of the crus of helix, there was a small opening showing pus drainage. There was no swelling or tenderness on the right auricle, but there was a small opening in the center of the crus of helix.\nSince there were sinus openings in both crus of helix (), manifestations of retroauricular area swelling, and history of recurrent infection, the patient was prediagnosed as the bilateral variant type of preauricular sinus, called 'postauricular sinus'. Surgery was performed after one week of antibiotics treatment and acute inflammation was relieved. Fistulography was performed in order to identify the location of the sinus and the length of the sinus tract, and there were a 0.5 cm, 1.0 cm sized sinus tract for each right and left side, heading towards the posterior-middle portion of the auricles ().\nDuring the surgery, local anesthesia was performed around the left sinus opening and retroauricular area. Then, gentian violet was injected into the sinus tract for dyeing. Perpendicular-oval shaped incision, including the opening of the sinus, was done and excision was preceded by identifying the dyed sinus. The direction of the sinus tract could be indentified using a lacrimal probe. The sinus was observed to penetrate the cartilage of the crus of helix and directed toward posterior part of the auricle as in the sinus form. Retroauricular incision was performed for complete removal of the sinus, and cartilage of the crus of helix was also removed (). Then, a drain was inserted and primary closure was performed. Same procedure was performed for the right side, but upon the insertion of the probe, the length of the sinus was small, and it was observed that it invaded to invade only a portion of cartilage of the crus of helix. Therefore, only an incision around the sinus opening was used completely to remove the sinus (). The patient was discharged without complications and total stitch off was done 7 days after the surgery. Histopathologic studies diagnosis was confirmed it as preauricular sinus, since the sinus tract lined with chronic inflammatory squamous epithelium (, ), and 12 months after the surgery, the patient is on follow up without complications and signs of recurrence.

[[27.0, 'year']]

M

{'27790479': 1, '15857338': 1, '27069313': 1, '2308458': 1, '29349057': 2, '17713452': 1, '32742852': 1, '17084223': 1, '20999293': 1, '15165194': 1, '24653881': 2}

{'5759660-1': 1}

3936562-1

noncomm/PMC003xxxxxx/PMC3936562.xml

A Case of Delayed Spontaneous Aneurismal Rupture after Ear Surgery Under General Anesthesia

A 54-year-old woman visited our department due to bilateral hearing impairment and intermittent otorrhea without dizziness of protracted duration. She did not have tinnitus or otalgia, but complained of recent symptom aggravation. Furthermore, she had no known history of medical or surgical problems, except that she was a chronic hepatitis B carrier.\nDuring physical and radiological examinations, a yellowish purulent discharge was noted in the right ear canal and attic destruction was identified. The left ear drum was thinned inferiorly and in a retracted state (), and the Weber test showed left deviation. Pure tone audiometry showed air conduction threshold of 72 dB HL and bone conduction threshold of 28 dB HL for the right ear and air conduction threshold of 50 dB HL and bone conduction threshold of 20 dB HL for the left. Auditory brainstem response testing showed peak waves at 70 dBnHL on the right and at 50 dBnHL on the left. A temporal bone computed tomographic (CT) scan showed sclerosis of mastoid bone and soft tissue densities in the middle ear, attic area, and mastoid cavity on the right side (), but ossicles and other inner ear structures, and the external auditory canal were relatively healthy. Other routine laboratory tests were within normal ranges. However, no evaluation of cerebrovascular structures was performed.\nTwo days after admission, right side canal wall up mastoidectomy with type I tympanoplasty was performed under general anesthesia. The dura and sigmoid sinus were not exposed during surgery. At about 2 hours after surgery, the patient complained of a headache and a rigid sensation in upper and lower extremities. At this time, her blood pressure was elevated at 170/100. Mentally, she was mildly drowsy and oxygen saturation had fallen to 80%. Accordingly, she was placed on mask ventilation, and our department of internal medicine was consulted regarding management. Endo-tracheal intubation was decided upon but not instituted because her condition recovered to near normal within an hour. Department of neurosurgery was then asked to undertake a proper evaluation. However, brain CT returned non-specific findings. Subsequently, the patient was followed a normal recovery course and pattern. Nevertheless, we recommended further evaluation, particularly brain magnetic resonance image, but the patient and her family rejected the idea. Accordingly, regular discharge was ordered 1 week after surgery.\nTwo weeks after discharge, the patient visited our emergency room with a severe headache and a mental status of stupor. Emergent brain CT and angiography was performed and a subarachnoid hemorrhage (SAH) from a ruptured anterior intercommunicating artery was visualized (). Emergent trephination was performed, and subsequently, she was admitted to our neurologic intensive care unit. Nevertheless, intracranial bleeding persisted at follow-up brain CT 2 days later (). The patient expired 1 week after this second admission without recovering consciousness.

[[54.0, 'year']]

F

{'2728035': 1, '3484561': 1, '15819296': 1, '11850607': 1, '7792692': 1, '12383354': 1, '1633478': 1, '7746399': 1, '6784546': 1, '24653877': 2}

{}

3936565-1

noncomm/PMC003xxxxxx/PMC3936565.xml

A Case of Giant Osteoma Developed from the Mastoid Cortical Bone

A 42-year-old woman visited out-patient clinic complaining of right retroauricular mass. The lesion of the right mastoid area developing over a 20-year period showed a severe cosmetic deformity (). On physical examination, both tympanic membranes were intact and huge, firm, painless, mass-like lesion was found on the right retroauricular area. Temporal bone computed tomography scan (TBCT) demonstrated 2.3×2.3×4.3 cm sized bony mass with a wide base on the surface of the mastoid cortical bone and squama without abnormalities of the middle and inner ear structures ().\nThe resection of tumor was performed using retroauricular approach (). The mass was widely exposed after elevation of mastoid periosteal flap and the margin of the mass was drilled down until the normal air cells were encountered. The mass attached to the lower part of the squama was drilled out. Remaining main mass was totally removed and the defect of mastoid area was reinforced by covering with the titanium mesh. The final histopathologic examination confirmed the osteoma with a compact subtype (). Four months after surgery, the surgical wound was intact, and the evidence of complication or recurrence was not found.

[[42.0, 'year']]

F

{'16371844': 1, '9441563': 1, '508196': 1, '15224871': 1, '34593383': 1, '29754138': 1, '33409144': 2, '702417': 1, '27051540': 2, '8464644': 1, '15021765': 1, '8959933': 1, '1870173': 1, '10190811': 1, '27407714': 1, '30069840': 1, '32566344': 2, '16864503': 1, '31750158': 1, '24653880': 2}

{'7298327-1': 1, '7298327-2': 1, '4804070-1': 1, '7758266-1': 1}

3936567-1

noncomm/PMC003xxxxxx/PMC3936567.xml

A Case of Linear Scleroderma Involving Cerebellum with Vertigo

A 17-year-old female patient visited our hospital for continuous spinning sense, which had been occurring for the past three days. The dizziness was continuous at rest, and aggravated with the moving head, accompanied by nausea and vomiting. The patient complained of left hearing loss that began approximately 1 year prior. There were no other otologicsymptoms, such as otalgia, ear fullness or otorrhea, etc. She had been taking Methotrexate (MTX) for linear scleroderma of the scalp since 2001, and had a history of undergoing resection and primary closure for progressive depression of the left forehead skin in 2010. Otoscopic examination of the ear showed that the external auditory canals and tympanic membranes in both ears were normal. Pure-tone audiometry showed sensorineural hearing loss of about 53 dB HL on the left side and normal hearing on the right side (). Tympanogram showed type A on both sides and stapedial reflex test was normal. In videonystagmography, spontaneous nystagmus toward the right side was observed (). The patient was admitted under suspicion for vestibular neuronitis and routine blood tests, electrocardiogram, and chest X-ray after admission were normal.\nIn positional nystagmus test, on the second hospital day, DCPN was observed. The nystagmus to the right side (25 deg/s) first appeared by rolling the head to the right in supine position, but when rolling the head to left, after about 58 seconds of right beating nystagmus (25 deg/s), the direction of nystagmuswas changed to the left side (20 deg/s), without changing the head position (). Saccade test, pursuit test and optokinetic test presented normal ranges, andbithermal caloric test showed the left canal paresis of 19% (). The neurologic and cerebral function exams were unremarkable. In cerebellar function tests, finger-to-nose test and heel-to-shin test were normal, but she showed instability in tandem gait test. Videonystagmography was performed again, and inhibition by visual fixation was not presented. With suspicion of central lesion, brain MRI and computed tomography were carried out. Multiple calcified lesions at the left cerebellum were observed. Compared with brain MRI in 2004, progressed lesions of the cerebellum were observed (). The cerebospinal fluid (CSF) study and eosinophil count study undergone for suspecting parasite infection were normal.\nThe patient was given a prescription for administration of antiemeticagent and antivertigo medication. Sixth day of admission, the patient was discharged because dizziness was improved. After two weeks, she still had the symptoms of dizziness, but it became mild and spontaneous nystagmus was not observed. At present, conservative treatment and observation is maintained.

[[17.0, 'year']]

F

{'4068089': 1, '16326691': 1, '3489418': 1, '28480106': 2, '21748654': 1, '24653878': 2}

{'5402329-1': 1}

3936570-1

noncomm/PMC003xxxxxx/PMC3936570.xml

A Case of Ramsay Hunt Syndrome with Cranial Polyneuropathy

A 66-year-old female was referred to our clinic with left facial palsy, diplopia, and vertigo six days after the onset of left ear pain and headache. The patient underwent a severe emotional stress due to her sister's death at the time of onset of left ear pain. The clinical manifestations developed gradually over a few days. Physical examinations revealed vesicular eruptions with crusts on left auricle, left facial palsy, left abducens nerve palsy, and mild right beating spontaneous nystagmus. The degree of FNP was House-Brackmann (HB) grade V (). Diplopia was present in the primary position and was more prominent on attempted abduction of the left eye (). Audiogram showed sensorineural hearing loss on her left ear (). Vestibular function test was performed on the fourth hospital day when the patient no longer complained of dizziness, and videonystagmography showed no evidence of spontaneous nystagmus. The size and response to light were normal on both pupils. Except for cranial nerves VI, VII, and VIII, examination for other cranial nerves revealed intact function. Magnetic resonance imaging showed no sign of tumor, infection or other demyelinating diseases of the brain. Serology results for VZV IgG antibodies were positive, but negative for VZV IgM antibodies. The patient had no associated immunocompromised condition including immunosuppressant drug use or immunodeficiency syndrome. A diagnosis of RHS with CP was made based on the patient's characteristic presentation, and from a clinical point of view cranial nerves VI, VII, and VIII were found to be involved. Systemic administration of steroid and antiviral agents was started. Sixty mg of prednisolone was initially given to the patient and tapered orally for 14 days, and 3000 mg of acyclovir was administered intravenously for 7 days. No immediate or delayed side effects were observed during and after the treatment. Among symptoms, dizziness improved most rapidly within several days. During 3 weeks from the start of treatment a gradual improvement of the facial weakness and diplopia was observed. Four months after the treatment, FNP recovered to HB grade I and the abducens nerve palsy completely improved without diplopia ().

[[66.0, 'year']]

F

{'26308524': 1, '12482166': 1, '15517838': 1, '8335601': 1, '34687339': 1, '20824789': 1, '6981355': 1, '2543853': 1, '31177200': 1, '28839425': 2, '11459884': 1, '34788266': 1, '28784901': 1, '9084199': 1, '24653876': 2}

{'5551344-1': 1}

3936642-1

noncomm/PMC003xxxxxx/PMC3936642.xml

Atrial Tachycardia Originating from the Aortomitral Junction

A 37-year-old woman with no relevant medical history was admitted to our center after 2 years of recurrent disabling episodes of palpitation and dizziness. She presented with abrupt onset and offset clinical tachycardia. The baseline electrocardiogram (ECG), chest radiograph, and echocardiogram were normal. The ECG recorded during an episode of palpitation revealed a narrow QRS complex tachycardia with a cycle length of 350 ms. The P-wave polarity was isoelectric in lead I and biphasic (initially negative with a late positive component) in leads V1 and V3. In addition, the P-wave polarity was positive in the inferior leads (II, III, and aVF) and negative in lead aVL ().\nAfter obtaining informed consent, we performed an electrophysiological study without sedation. Two quadripolar catheters were introduced into the right ventricle and His bundle region via the femoral vein. A 20-pole catheter with an interelectrode spacing of 2-5-2 mm was positioned at the lateral right atrium (RA), with its distal 10 electrodes in the coronary sinus. Ventriculoatrial conduction was absent at baseline and during isoproterenol infusion. Tachycardia was induced by burst pacing from the RA during isoproterenol infusion without a "jump-up" in the atrial-His (A-H) interval. The tachycardia was perpetuated regardless of the spontaneous AV block. The entrainment phenomenon was not observed during atrial pacing. An intravenous bolus injection of 2-mg adenosine 5'-triphosphate during tachycardia reproducibly terminated the tachycardia without any development of AV block. Ventricular overdrive pacing during tachycardia resulted in AV dissociation without tachycardia entrainment.\nActivation mapping in the RA during tachycardia revealed the earliest atrial activation at the His bundle region, which preceded the onset of the P wave by 30 ms on the surface ECG. Radiofrequency energy was delivered to the superoseptal RA, where the earliest RA activation was recorded, using a 7 F, 4-mm-tipped ablation catheter (Blazer II, Boston Scientific, Natick, MA, USA), with a maximal temperature of 55℃ and maximal power output of 35 W. The His bundle potential was not recorded from the ablation sites at the right superoseptum. The tachycardia was terminated with junctional beats. However, the tachycardia was still inducible and sustainable after the ablation at these sites. To find the other earliest atrial activation site, mapping was then performed around the left atrial septum. The activation at the left atrial septum close to the His-bundle region during the tachycardia was later than that at the right His-bundle region. Subsequently, detailed activation mapping was performed at the aortic sinus of Valsalva, using a transaortic approach, after the aortography. The local atrial electrogram inside the noncoronary aortic and left coronary sinuses preceded the onset of the surface P wave by 40 and 50 ms, respectively. Radiofrequency energy was delivered in the left coronary sinus. Although the tachycardia was terminated by the ablation, it was still inducible. Finally, the earliest site of atrial activation was found at the aortomitral junction where the local atrial electrogram preceded the onset of the surface P wave by 70 ms (). A single radiofrequency energy application at this site using the same ablation catheter with maximal temperature and power output of 50℃ and 30 W, respectively, terminated the tachycardia in 12 s. Thereafter, atrial tachycardia was no longer inducible regardless of isoproterenol infusion (). Neither junctional beats nor prolongation of the A-H interval occurred during or after the energy application. During a 12-month of postablation follow-up period, the patient has been free from recurrence of atrial tachycardia without any antiarrhythmic medications.

[[37.0, 'year']]

F

{'812612': 1, '12354440': 1, '11405398': 1, '18031512': 1, '14557361': 1, '8087935': 1, '16814658': 1, '12108509': 1, '9462592': 1, '18233981': 1, '19131496': 1, '17394965': 1, '11843486': 1, '20230911': 1, '21985795': 1, '8450158': 1, '18403384': 1, '24532528': 2}

{}

3936656-1

noncomm/PMC003xxxxxx/PMC3936656.xml

A Case of Pulsatile Tinnitus from the Atherosclerosis and Atheroma in Superior Labial Artery and Facial Artery

A 46 year old male patient experienced vibratory tinnitus on the right side for 10 years. He had pulsatile mass on the right side of his lip and did not undergo any treatment. Since 1 month before diagnosis, the level of tinnitus increased along with the size of the mass. Past medical history shows both lower limbs amputation 20 years ago and hypertension. Upon physical examination, there were no symptoms of tenderness, but 2 pulsatile masses were found on the right upper and right lateral side lip. The size of the former mass was 1×1 cm and the latter was 1.5×1.0 cm (). There were no other special abnormalities in the oral cavity, oropharynx and the other head and neck area. The tinnitus decreased when pressure was applied to the left tympanic membrane area and increased when leaning forward. The tympanic membrane was in sound condition and hearing tests revealed both ears to be normal. Similar observations were made during two times of tinnitogram. The frequency was 2 kHz, and loudness was between 60 and 70 decibels and there were no residual inhibitions. Arteriovenous malformation from the right facial artery was found resulting from computed tomography of the facial bone (). CT with angiography was not conducted due to hypotension and tachycardia after the injection of the contrast media. It was diagnosed as resulting from an abnormality in vessel and excisional biopsy was conducted through oral access after general anesthesia (). Operation revealed the superior labial artery and facial artery after incising the oral mucosa in the mass and the area showing evidence of vascular malformation was ligated. The area was irrigated and operation was concluded. Patient was allowed to return home 3 days after the operation. Pathology diagnosed the condition as atherosclerosis (). The patient did not complain of any tinnitus symptoms after the operation and shows no sign of recurrence up until now, 1 year after the operation.

[[46.0, 'year']]

M

{'1901141': 1, '27861380': 1, '9337936': 1, '1919691': 1, '1188624': 1, '24653894': 2}

{}

3936658-1

noncomm/PMC003xxxxxx/PMC3936658.xml

A Case of Malignant Small Round Cell Tumor of Temporal Bone with Facial Paralysis

A 5-year old boy visited outpatient clinics for left idiopathic acute facial paralysis (House-Brackmann grade IV). Pure tone audiometry showed normal healing level. Oral steroid medication was prescribed for 10 days as scheduled during admission. On imaging study, temporal magnetic resonance imaging showed a partial fluid signal at the left mastoid air cells and abnormal enhancement at the left facial nerve around the genu area (). He was diagnosed with Bell's palsy. However there was no improvement until a month after discharge. His parents wanted to transfer to another hospital and returned six months later. He showed marked improvement without any treatment and there were no sequelae (House-Brackmann grade I). Three months later, he visited again for left intractable otalgia without facial palsy. He had a medication history of local clinics for the last month. On physical examination, the left tympanic membrane could not be observed due to severe narrowing of left external auditory canal (). Pure tone audiometry showed sensorineuronal hearing loss of 60 dB threshold. Temporal bone computed tomography revealed expansile soft tissue densities and diffuse erosive destruction on left temporal bone (). On the basis of these findings, we suggested aggressive infectious disease or destructive cholesteatoma.\nWe performed an emergency modified radical mastoidectomy under general anesthesia. During the operation, we found a pale, grayish tumor occupying the mastoid and middle ear cavity (). On frozen biopsy, it was suspected to be a MSRCT.\nPathologic examination of the resected tumor showed an infiltrating tumor consisting of small round cells (). The results of immunohistochemical stainings for vimentin, neuron specific enolase (NSE), S-100 protein, pancytokeratin, synaptophysin, chromogranin A, smooth muscle actin, Sarcomeric actin, glial fibrillary acidic protein (GFAP), leuko-cyte common antigen were all negative, so we couldn't categorize the tumor.\nAfter the pathologic confirmation, the patient transferred to hematooncologic department for chemotherapy and radiotherapy.

[[5.0, 'year']]

M

{'12383375': 1, '17505616': 1, '7488369': 1, '12151977': 1, '21938141': 1, '22052164': 1, '15100645': 1, '12784241': 1, '24653891': 2}

{}

3936666-1

noncomm/PMC003xxxxxx/PMC3936666.xml

Preauricular Angioleiomyoma

An 85-year-old man presented to the Department of Otolaryngology-Head and Neck Surgery at our hospital with hearing difficulty in his left ear. Pure tone audiometry showed left sensorineural hearing loss. During the physical examination of the ear, a small round cystic mass was found on the left preauricular area (). It did not hurt and was not tender. The patient had noticed the mass about 6 months previously and it had grown slowly. No similar lesion was found on his body. His medical and family histories were unremarkable.\nAfter informed consent was obtained, the lesion was completely excised under local anesthesia in the operation room for differential diagnosis and cosmetic purposes. Grossly, the specimen was a 1.4×1.0×0.7 cm firm, spherical mass with a white-gray, solid cut surface. Microscopic examination revealed that the tumor was a well-defined nodule of smooth muscle bundles containing thick-walled blood vessels (). No significant cellular atypia or mitotic activity was observed. Based on histological findings, the pathologic diagnosis of venous type angioleiomyoma was made. No evidence of a recurrence was observed 6 months after surgery.

[[85.0, 'year']]

M

{'15817400': 1, '1951988': 1, '11458228': 1, '15672722': 1, '17606037': 1, '11032299': 1, '6722737': 1, '15311559': 1, '21935882': 1, '11006451': 1, '27508144': 1, '21682171': 1, '9030948': 1, '15064620': 1, '7594988': 1, '17580807': 1, '24653889': 2}

{}

3936668-1

noncomm/PMC003xxxxxx/PMC3936668.xml

Cerebrospinal Fluid Otorrhea Caused by Arachnoid Granulation

A 45 year-old female patient was referred to our department with right ear fullness which was precipitated by changes in position. Her symptom was initiated about 3 months ago. She has been well controlled hypertension. In physical examination, air fluid level was found in her right tympanic membrane, and the level increased when she hung her head down. Pure tone audiometry examination showed the right conductive hearing loss. Impedance test showed that the right was C type and the left was A type. In order to distinguish CSF otorrhea, her middle ear fluid was aspirated, and its glucose level noted 88 mg/dL. Computed tomogram (CT) of temporal bone demonstrated a bony defect adjacent to the soft tissue lesion of posterior wall of the temporal bone with an effusion showing high attenuation in the right mastoid cavity (). Thus, we presumed that CSF otorrhea might be caused by arachnoid granulation. She underwent mastoidectomy under general anesthesia. During a procedure for canal wall-up mastoidectomy, in accordance with the findings of the CT scan, the tissue which was suspected to be arachnoid granulation was observed with the bony defect of the posterior wall. The surrounding bone was removed by a diamond drill. The granulation tissue was placed at the defect of meninx (). The tissue was obtained for a biopsy. The remained granulation tissue was shrunk by bipolar cauterization. The bony defect was reconstructed by the auricular cartilage and perichondrium complex graft. Subsequently, fibrin glue was placed over the graft. The biopsy demonstrated a fibrillary tissue (). Moreover, glial fibrillary acidic protein stain indicated a positive reaction, and we confirmed arachnoid granulation which contained a portion of parenchyma of the cerebrum (). The patient has not had any symptoms of the disorder. In addition, 1 year and 3 months after the surgery, she's been continuing follow up in the outpatient clinics without any complications or recurrence.

[[45.0, 'year']]

F

{'9159387': 1, '2241009': 1, '834881': 1, '4197445': 1, '3932931': 1, '534052': 1, '10319986': 1, '10565723': 1, '5079659': 1, '34026411': 1, '10748823': 1, '21436752': 1, '18490573': 1, '3591928': 1, '15126733': 1, '8283502': 1, '3520197': 1, '24653893': 2}

{}

3937477-1

noncomm/PMC003xxxxxx/PMC3937477.xml

Classical Sweet's syndrome

A 38-year-old female presented with red raised, painful lesions on right forearm with fever since 10 days, which gradually coalesced to form larger lesions []. Patient had similar episodes 3 years back which had subsided on taking some medications. There was a prior history of upper respiratory tract infection. On examination, patient was febrile. A large erythematous plaque of size 8 × 6 cm was present on right upper extremity. Small erythematous plaques were also present on both upper limbs, right side of neck, and chest. Few lesions showed typical “relief of mountain range appearance”. Local temperature was raised in all the lesions. Superficial nerves were not thickened and sensations on the plaques were normal. All other investigations were within normal limits apart from elevated ESR. Histopathology showed a diffuse infiltrate consisting predominantly of mature neutrophils located in the upper dermis without evidence of primary leukocytoclastic vasculitis []. Our patient was initially treated with tablet dapsone 100 mg HS with dramatic early improvement and relapse on discontinuation. The second episode was treated with a combination of dapsone and oral prednisolone started at 30 mg/day and gradually tapered over 4 weeks. The patient is now in remission [].

[[38.0, 'year']]

F

{'6696481': 1, '2212153': 1, '8339190': 1, '14201182': 1, '27587257': 1, '27294187': 1, '24616879': 2}

{}

3937482-1

noncomm/PMC003xxxxxx/PMC3937482.xml

Aquagenic keratoderma. Two new case reports and a new hypothesis

A 12-year old girl presented with three months history of burning of palms and loss of skin dermatoglyphics after exposure to water []. Symptoms were temporary; changes faded away within 20 min. The patient did not have concomitant diseases and was not on any medication. Clinical examination revealed hyper-wrinkling of the palms after immersion in water without any other skin and/or mucous changes. Routine laboratory examination was unremarkable.\nTherapy was initiated with 20% alcohol solution of aluminum chloride hexahydrate once daily at night. The treatment resulted in significant clinical improvement with reduction of the frequency and duration of AK episodes.

[[12.0, 'year']]

F

{'16336521': 1, '21779403': 1, '18625374': 1, '11834842': 1, '20673535': 1, '16476292': 1, '33484064': 1, '31799354': 1, '31544086': 1, '11260552': 1, '17671517': 1, '20302572': 1, '21733844': 1, '20055864': 1, '17179009': 1, '18852901': 1, '22471628': 1, '20015211': 1, '18503605': 1, '22750231': 1, '22044449': 1, '10954989': 1, '17168867': 1, '15727674': 1, '16612557': 1, '16648884': 1, '23067094': 1, '8601664': 1, '17943189': 1, '22512866': 1, '16872579': 1, '20402952': 1, '14576620': 1, '29723369': 1, '21995840': 1, '18973409': 1, '20560957': 1, '33403185': 2, '17614805': 1, '18182813': 1, '34136618': 1, '12007216': 1, '16445410': 1, '24616851': 2}

{'3937482-2': 2, '7773311-1': 1}

3937482-2

noncomm/PMC003xxxxxx/PMC3937482.xml

Aquagenic keratoderma. Two new case reports and a new hypothesis

The second patient was a 27-year-old male suffering from cystic fibrosis (CF) and focal palmar hyperhidrosis. He was tested positive for homozygosity for the ΔF508 mutation of the CF transmembrane conductance regulator (CFTR) gene. His medication consisted of macrolide antibiotic prophylaxis and inhaled corticosteroids. Without immersion in the water, he showed whitish translucent papules and increased wrinkling of the palms []. Symptoms worsened after water immersion. He did not report pruritus or pain sensation.\nWe treated him twice daily with a topical aluminum chlorohydrate emulsion (Ansudor N, Galderma). Palmar symptoms improved markedly.

[[27.0, 'year']]

M

{'16336521': 1, '21779403': 1, '18625374': 1, '11834842': 1, '20673535': 1, '16476292': 1, '33484064': 1, '31799354': 1, '31544086': 1, '11260552': 1, '17671517': 1, '20302572': 1, '21733844': 1, '20055864': 1, '17179009': 1, '18852901': 1, '22471628': 1, '20015211': 1, '18503605': 1, '22750231': 1, '22044449': 1, '10954989': 1, '17168867': 1, '15727674': 1, '16612557': 1, '16648884': 1, '23067094': 1, '8601664': 1, '17943189': 1, '22512866': 1, '16872579': 1, '20402952': 1, '14576620': 1, '29723369': 1, '21995840': 1, '18973409': 1, '20560957': 1, '33403185': 2, '17614805': 1, '18182813': 1, '34136618': 1, '12007216': 1, '16445410': 1, '24616851': 2}

{'3937482-1': 2, '7773311-1': 1}

3937483-1

noncomm/PMC003xxxxxx/PMC3937483.xml

Cerebriform intradermal nevus presenting as cutis verticis gyrata with multiple cellular blue nevus over the body: A rare occurrence

A 28-year-old male patient presented to the skin out-patient department with history of a huge progressively increasing swelling over the back of the scalp [] and multiple black nodules at various parts of the body. According to patient same kind of lesions were present over the trunk at birth, which were surgically removed when he was 4-year-old. The lesion over the scalp started at the age of 6-7 years and the truncal lesions developed gradually since then. On examination, there was a single bosselated irregular non-tender skin colored swelling of 15 cm × 20 cm with deep folds over the occipital region extending toward the nape of the neck. There were certain areas of depigmentation [ along with blue streaks over the swelling. The surface of the skin was hairless, the skin over the swelling was non-pinchable and the swelling was immobile. There was no skin erosion, dermatitis, infection, odor or tenderness.\nThere were multiple hyperpigmented nodules distributed over different areas of the body such as mandible, flanks, abdomen [], front and back of both thighs, right knee joint, lower leg [], right sole and over volar aspect of first and second finger of the left hand []. The size of the nodules ranged from 0.5 cm × 1 cm to 1.5 cm × 2.5 cm. The nodules were blackish brown in color surface was smooth and hairless. They were asymptomatic and non-tender on palpation. Nails and mucous membrane examination were within normal limits. He had normal intelligence (the full-scale intelligence quotient was 97) and he had no psychiatric problem. Examination of the neurological, ophthalmological and other systems was normal. Laboratory results, including venereal disease research laboratory test, serology for human immunodeficiency virus, growth hormone and thyroid function, were all negative or normal. A punch biopsy from the scalp lesion and an excision biopsy from the nodule over the body was performed. Section from the specimen showed multiple nests and cords of nevus cells in the upper dermis, which contained a moderate amount of melanin []. There was no cellular atypia. Section from the body lesion showed deeply pigmented dendritic melanocytes in addition to nests and fascicles of spindle shaped cells with abundant pale cytoplasm containing little melanin [Figures and ]. The spindle cells were arranged in a storiform pattern. Based on the clinical feature and histology it was diagnosed as a case of congenital melanocytic nevus with cellular blue nevi.

[[28.0, 'year']]

M

{'10721658': 1, '18211488': 1, '8513202': 1, '33947392': 2, '21199640': 1, '21083380': 1, '8482606': 1, '18304152': 1, '26955158': 1, '27730054': 1, '6703738': 1, '17461810': 1, '19793094': 1, '24616852': 2}

{'8094459-1': 1}

3937484-1

noncomm/PMC003xxxxxx/PMC3937484.xml

Progressive cribriform and zosteriform hyperpigmentation

A healthy 28-year-old male presented with progressively increasing brown to black macules and patches arranged along the lines of Blaschko on the right side of abdomen and back. The onset of lesions was first from back and the spread towards abdomen. The lesion appeared at the age of 20 and has been progressive since then. He had no history of any preceding eruption, drug intake or trauma to the area. There was no evidence of internal diseases and no family history of skin abnormalities.\nCutaneous examination revealed linear, cribriform, brown to black pigmentation along lines of Blaschko extending from back to abdomen till pubic area on right side []. Multiple macules coalescing at places to form patches were seen and at some places lesions were slightly palpable with velvety appearance. Laboratory studies, including complete blood cell count, liver and renal function tests and serum electrolyte, were within normal limits.\nA diagnosis of zosteriform lichen planus, non-hypertrichotic variant of Becker's nevus and nevoid acanthosis nigricans was thought. A skin biopsy specimen was obtained from most recent lesion which revealed uniformly increased pigmentation within the basal keratinocytes [Figures and ]. There were few dermal melanophages with mild pigment incontinence without much inflammatory infiltrate in the dermis []. Nevus cells were absent. Absences of lichenoid infiltrate and interface change ruled out lichen planus. Lack of an increase in smooth muscle fiber and hair follicles ruled out Becker's nevus. Lack of church-spire hyperkeratosis, presence of heavy basal layer pigmentation with mild pigment incontinence ruled out epidermal nevus and acanthosis nigricans. Also clinically there were many skip areas giving a cribriform appearance. Thus, the diagnosis of PCZH was made on clinicopathological correlation.

[[28.0, 'year']]

M

{'33447068': 1, '619793': 1, '22435426': 1, '17542865': 1, '3204178': 1, '16394371': 1, '16164727': 1, '8239703': 1, '24616853': 2}

{}

3937486-1

noncomm/PMC003xxxxxx/PMC3937486.xml

Metastatic Crohn's disease

A 20-year-old woman, mother of a 1.5-year-old child, presented with a swelling in the genitalia of 6 months duration. Cutaneous examination revealed a single, non-tender, pedunculated, polypoid growth, about 6 × 7 cm, hanging from the left labium minus []. She had minimal swelling of the left labium majus. There was a tiny ulcer near the superior commissure of the labia majora. However, many scars were seen on the medial aspect of both the thighs, which were attributed to pruritic ulcers in childhood, details of which were unavailable. Her vagina, cervix, and uterus were normal. Systemic examination, including the eyes and oral mucosa, did not reveal any abnormality. Filariasis being endemic in Cuddalore district of Tamil Nadu, elephantiasis of external genitalia due to filariasis, besides, lymphogranuloma venereum and granuloma inguinale were also considered.\nA clinical diagnosis of soft fibroma was entertained and an excision biopsy of the nodule was undertaken. Histolopathological examintion (HPE) revealed multiple non-caseating granulomas, edema, and dense lymphocytic infiltration in the dermis. Ziehl–Neelsen staining for Mycobacterium tuberculosis was negative []. Special stains for fungal organisms were not performed.\nA week later, at the time of suture removal, the patient presented with multiple typical “knife-cut” ulcers on the external genitalia, in the inguino-crural fold, interlabial creases, and natal cleft, leaving no doubt in the diagnosis of CCD []. Biopsy from knife cut ulcers could not be carried out since the patient was not willing to undergo the procedure. She did not have any intestinal symptoms or pain abdomen at any time in the past or present.\nOn investigation, she was found to have hypochromic microcytic anemia (Hb: 10.5 gm/dl) and mild neutrophilic leucocytosis. Other relevant investigations including Mantoux test, ultrasonogram of the abdomen, and chest radiograph were not contributory. Both Enzyme-Linked ImmunoAssay (ELISA) test for HIV and VDRL test were negative.\nThe patient was referred to a gastroenterologist for further work up, and no gastrointestinal involvement was reported. With this background, a diagnosis of CCD was made.\nThe patient was managed with metronidazole 400 mg t.i.d. for 1 week and ciprofloxacin 500 mg bid. for 10 days in addition to oral prednisolone at 30 mg/day, which was gradually tapered over a period of 6 weeks.\nShe reported after 2 weeks with ulcers showing signs of healing, and by 6 weeks, they resolved. Later, the patient was lost on follow-up.\nHowever, she reappeared 2 years later with massive swellings of both the labia majora, more marked on the left labium majus. She gave a history of having had a full-term normal child, 3 months earlier, delivered by an elective lower segment caesarean section (LSCS) in view of the edema of the labia majora [].

[[20.0, 'year']]

F

{'1469154': 1, '7172746': 1, '26500880': 1, '3412990': 1, '14756892': 1, '19119511': 1, '20883918': 1, '21508582': 1, '30078880': 1, '32467818': 2, '24616854': 2}

{'7250390-1': 1}

3937487-1

noncomm/PMC003xxxxxx/PMC3937487.xml

Epidermolysis bullosa pruriginosa: A report of two cases

A 52-year-old lady presented to our outpatient department with complaints of itching and blackish discoloration of skin of both the lower limbs for more than 35 years and fluid filled lesions over the lower limbs since two years. Lesions were extremely pruritic without any diurnal variation. There was no history of drug intake before the onset of lesions nor any seasonal exacerbation. There was no history of similar complaints in other family members. Patient was a known case of diabetes since five years and on regular treatment. On examination, multiple lichenified papules to nodules were present over the lower limbs extending from the knee to the ankle joint. Depigmentation at the center with scarring was seen over the larger nodules. Few pus filled lesions were also seen []. There was no associated nail involvement. Routine investigations were normal. Based on the clinical picture, EB pruriginosa was suspected. Skin biopsy and immunohistochemistry was done to confirm the diagnosis.\nOn histopathological examination, epidermis showed subepidermal and intraepidermal bulla with degeneration of keratinocytes. Foci of cellular infiltrate consisting of fragmented neutrophils with occasional mononuclear cells and hemorrhage [Figures and ]. Direct immunofluorescence was negative. Patient was admitted for four days and was given parenteral steroids and antibiotics. Itching subsided and lesions began to heal. On discharge patient was started on tab dapsone 100 mg once a day with good response. Hematological parameters and lipid profile were monitored.

[[52.0, 'year']]

F

{'4788473': 1, '15262686': 1, '20877091': 1, '942257': 1, '8204470': 1, '15949003': 1, '9182828': 1, '1155959': 1, '31561504': 1, '9292092': 1, '24616855': 2}

{'3937487-2': 2}

3937487-2

noncomm/PMC003xxxxxx/PMC3937487.xml

Epidermolysis bullosa pruriginosa: A report of two cases

A 34-year-old female patient came to the outpatient department with complaints of skin lesion over both the lower limbs associated with intense itching was noted since 15 years. No seasonal variation in lesions. There were no lesions in the oral cavity. None of the family members had similar lesions. The patient was non-diabetic and normotensive. Cutaneous examination revealed lichenoid papules over both shins extending on to the knee [Figures and ]. Papules were non tender associated with mild scaling. There was no associated nail or mucosal involvement. Lab investigations were normal. Skin biopsy showed subepidermal bulla filled with fibrin and RBCs. Dermis showed perivascular mixed inflammatory cell infiltrate and cyst lined by stratified squamous epithelium [Figures and ]. Direct immunofluorescence was negative.\nThe patient was started on topical steroids with systemic antihistamines with minimal response after one month.

[[34.0, 'year']]

F

{'4788473': 1, '15262686': 1, '20877091': 1, '942257': 1, '8204470': 1, '15949003': 1, '9182828': 1, '1155959': 1, '31561504': 1, '9292092': 1, '24616855': 2}

{'3937487-1': 2}

3937488-1

noncomm/PMC003xxxxxx/PMC3937488.xml

Erythema elevatum diutinum in association with dermatitis herpetiformis

A 50-year-old male presented with firm skin-colored and erythematous nodules over the knuckles, elbows, buttocks, knees, and lower legs which gradually increased in size in since 3 years duration []. Complete blood picture, liver function tests, urine routine, blood sugar were within normal limits. Biopsy of the nodules of the knuckles showed focal parakeratosis and mild spongiosis in the epidermis, perivascular neutrophilic infiltrate in the dermis, neutrophils in the vessel wall and leukocytoclasia []. Immunofluorescence showed linear IgA deposits in the basement membrane zone and weak granular perivascular C3 and fibrinogen deposits []. Four years ago itchy vesicles over the back and arms were detected in the patient. Biopsy of the vesicular lesions were consistent with dermatitis herpetiformis []. He was administered dapsone which leads to disappearance of lesions with recurrence after stopping treatment. With the clinical presentation, biopsy and immunofluorescence results, a diagnosis of erythema elevatum diutinum on a background of dermatitis herpetiformis was made.

[[50.0, 'year']]

M

{'16983903': 1, '9185905': 1, '8915315': 1, '28971130': 1, '28050587': 1, '12477369': 1, '34934715': 1, '9173057': 1, '24616856': 2}

{}

3937489-1

noncomm/PMC003xxxxxx/PMC3937489.xml

A case of subcutaneous zygomycosis

A 6 month-old female child presented with the chief complaints of multiple painless swellings over her left knee for the past 4 months, which was insidious in onset and gradually progressive. The present illness was preceded by an insect bite 4 months ago following which she developed erythematous nodules, which have gradually progressed with ulceration. It was associated with low -grade fever for the first 10 days following the insect bite. Physical Examination revealed multiple nodules over the extensor aspect of left thigh and a single well- defined erythematous indurated non- tender nodule of size 3 cm × 4 cm with central ulceration with eschar formation present over the left knee. The swelling was firm in consistency with smooth and rounded edges. It was freely mobile over the underlying structure [Figures and ]. Bilateral inguinal lymphadenopathy was noted.\nRoutine investigations were carried out; complete blood picture-Haemoglobin -13.7 g%, Total leucocyte count -9,900 cell\cu.mm, Differential Count –Neutrophils -55%, Lymphocytes- 40%, Eosinophils - 4%, Monocytes - 1%, and Platelets-1.76 lakhs/cu.mm. Complete urine examination - urine was clear with 2-3 pus cells, 3-4 epithelial cells with no albumin and sugar. Human immuno-deficiency virus 1 and 2- non- reactive; and Liver Function Test -normal; X-ray left thigh– showed soft- tissue swelling. Microbiological investigations: On direct examination of the tissue specimen with 10% KOH showed fungal elements with broad aseptate hyphae. Growth on Sabourauds Dextrose Agar at 30°C showed flat creamy brown, furrowed colonies with pale reverse by 3 days []. After about a week, satellite colonies have developed and the colonies have turned brown in colour []. Lactophenol cotton blue wet mount demonstrated aseptate hyphae and smooth walled zygospores with characteristic conjugation beaks, which confirmed the fungus to be B. ranarum []. Histopathological report says sections of normal skin with subcutaneous tissue showing inflammatory granulomatous reaction with diffuse and dense infiltration by eosinophils, polymorphs, histiocytes, plasma cells, lymphocytes, and few multinucleated giant cells. The central fragments showed fragments of aseptate hyphae.\nFollowing the confirmation of the diagnosis as basidiobolomycosis by culture and microscopy, the patient was put on saturated solution of oral potassium iodide (KI) in milk. The initial dose was 5 drops (approximately 1 ml) thrice a day after meals followed by increase in dosage by 1 drop/dose until 40 drops/dose. Therapy was continued untill the signs of active disease have disappeared. Subsequently, the dosage has been decreased by 1 drop/dose until 5 drops were given, followed by discontinuation. Complete resolution of the lesions was seen by 8 weeks after initiation of treatment [].

[[6.0, 'month']]

F

{'14748801': 1, '33959531': 2, '10669127': 1, '28959073': 1, '30022946': 2, '19961248': 1, '25821287': 1, '30158298': 1, '30859059': 1, '10756000': 1, '24616857': 2}

{'8088161-1': 1, '6047539-1': 1}

3937490-1

noncomm/PMC003xxxxxx/PMC3937490.xml

Efficacy of oral zinc therapy in epidermodysplasia verruciformis with squamous cell carcinoma

A 24-year-old male presented in our out-patient department with complaints of multiple asymptomatic raised warty lesions on the face and extremities since 4 years of age and multiple asymptomatic light colored flat lesions on the trunk and axilla since 4 years of age. These lesions had appeared over a period of time and were progressively increasing in number, size and area of involvement.\nHe also had an asymptomatic non-healing ulcer in the right first web space for the last 1 year. Initially, it was small but over past 3 months it progressively increased in size. Mucocutaneous examination revealed multiple dark colored warty papules and plaques over the face, neck and extremities [Figures -]. Multiple hypopigmented macules like those of pityriasis versicolor were present predominantly in bilateral axilla, upper back and shoulders. An ulcer of size 6-7 cm was present in first web space of the right hand []. It was non-tender, firm in consistency and the surface showed hemorrhagic crusting with rolled out margins.\nHematological, renal, hepatic biochemical parameters and chest X-ray were normal. X-ray of the right hand showed soft-tissue shadows in first web space with no bony involvement. MRI of right hand showed ill-defined heterogeneous lesion in the first web space of right hand (2.4 × 2.8 × 3.3), involving the lumbricals and palmar interossei on the lateral aspect and closely averting flexor digitorum superficialis tendon.\nSkin biopsy from the warty lesion present on the extensor surface of right forearm revealed hyperkeratosis, irregular acanthosis, and an enlarged vacuolated cells suggestive of koilocytes. These histopathological features were consistent with EV []. Histopathological examination from the margin of ulcer present on the right first web space showed multiple dysplastic cells and was consistent with well differentiated squamous cell carcinoma. Rest of the metastatic work up was within normal limit.\nWith all above findings the diagnosis of EV with squamous cell carcinoma of right hand was made. The patient was started on oral zinc sulphate 550 mg/day (10 mg/kg) for twelve weeks and followed-up for the next 6 months. Serum zinc level prior to starting zinc therapy was 58.09 μg/100 mL [] and on completion of treatment it was 168 μg/100 mL (difference 168 - 58.09 = 109.91) []. The main outcome measured was complete clearance of verrucae at 12 weeks as observed on digital photographs taken by the physician delivering the treatment at baseline and 12 weeks. Complete clearance of verrucae was defined as the restoration of normal skin upon close inspection, as assessed by the physician [Figures -].\nThe squamous cell carcinoma of right hand was removed after surgical consultation. No side-effects were reported by patient during the therapy and in the follow-up period.

[[24.0, 'year']]

M

{'19706093': 1, '17367237': 1, '11952542': 1, '17139267': 1, '19293025': 1, '21352302': 1, '20824263': 1, '19377755': 1, '25120566': 1, '16045695': 1, '32782865': 2, '12582373': 1, '16892068': 1, '2990354': 1, '29872682': 1, '18158319': 1, '24616858': 2}

{'7410503-1': 1}

3937491-1

noncomm/PMC003xxxxxx/PMC3937491.xml

Hansen's disease associated with erythromelalgia mimicking Lupus erythematosus

A 39-year-old male teacher presented with reddish skin lesions over the face with history of increase in redness and burning sensation on sun exposure of 25-day duration. He had also noticed redness and burning over the finger tips which was relieved by immersion of hands in cold water. In addition, he had intermittent joint pains involving knee and interphalangeal joints of 4-year duration. There were no systemic complaints such as hematuria, seizures, dyspnea or palpitation, although there was one episode of oral ulcers 10 days ago.\nExamination revealed multiple well-defined, irregular erythematous mildly scaly plaques over the forehead, cheeks, ears, V area of neck []. There was erythema, warmth, and mild tenderness over the bilateral finger tips []. There was no sensory loss over the skin lesions or thickening of peripheral nerves. Sensory and motor examination of the extremities was normal. Hot and cold water immersion tests for erythromelalgia and Raynaud's phenomenon were negative with no worsening of redness or bluish discoloration of fingers, respectively.\nIn view of arthritis, photosensitivity, history of oral ulcers, a clinical diagnosis of acute cutaneous lupus erythematosus with erythromelalgia was considered and patient investigated. The hemogram, chest X-ray and renal function tests, serum C3 and C4 levels were normal. Liver function tests were normal except for mildly elevated ALT. ANA was negative. USG abdomen showed grade 2 fatty infiltration of liver. Blood sugars were elevated.\nSlit smear examination for lepra bacilli and direct immunofluorescence test of skin lesions were negative. Treatment was commenced with 32 mg of oral methylprednisolone per day along with subcutaneous insulin. H and E examination of skin biopsy specimen showed thinning and follicular plugging of epidermis, a grenz zone with ill-defined collections of lymphocytes, macrophages, and epitheloid cells in a perineural, periadnexal, and perivascular location [Figures -]. Fite stain was positive for lepra bacilli.\nA revised diagnosis of borderline tuberculoid leprosy with type 1 reaction was made and antileprosy treatment as for multibacillary regime with 3 drugs (oral pulse of Rifampicin 600 mg and clofazimine 300 mg once in 28 days, daily oral clofazimine 50 mg and dapsone 100 mg) was started along with oral steroids in tapering doses. The facial lesions and finger tip erythema resolved in 2-month time [] and patient is on regular follow-up.

[[39.0, 'year']]

M

{'16178886': 1, '1800668': 1, '15955112': 1, '18380211': 1, '14568838': 1, '20186674': 1, '33954015': 1, '29641704': 1, '1322958': 1, '28405562': 1, '24616859': 2}

{}

3937492-1

noncomm/PMC003xxxxxx/PMC3937492.xml

Follicular psoriasis causing erythroderma in a child: A rare presentation

A 13-year-old boy presented with scaling and erythema of skin since six months and difficulty in closing the eyes completely since four months. His skin lesions started as dark, rough, horny papules over the knees and elbows, which became erythematous and scaly after a few months and gradually spread to involve the whole body. Small areas of skin (spared of erythema and scaling) with these horny papules were still visible over the shoulders and flanks at the time of admission []. The face showed tightening of skin, bilateral ectropion of lower eyelids, furrowing of skin of the nasolabial area, and fissuring at the angle of the mouth []. Scalp was covered with thick scales. Palmoplantar hyperkeratosis was present and associated with thick, lusterless nails with subungual hyperkeratosis, and longitudinal ridging [].\nThe joints of both the knees were widened with bilateral genu valgum []. The range of movement was normal, but they had recently become painful. Radiographic findings suggested changes similar to rickets. Family history was negative and there was no history of trauma, drugs, infections or any other triggers for his condition.\nBased on the above findings and after careful exclusion of other causes of erythroderma in children, a clinical diagnosis of erythroderma due to follicular psoriasis was made. This was confirmed by histopathology [] which showed parakeratosis, hypogranulosis, elongated rete ridges with corresponding dermal papillary hyperplasia, suprapapillary thinning of epidermis, and neutrophilic infiltrate in the dermis. The follicles also showed plugging and ostial parakeratosis.\nThe patient was managed conservatively using emollients and a well-controlled fluid and food intake. Methotrexate was started in the dose of 7.5 mg/week after ensuring that the routine investigations were normal. A single injection of vitamin D (6 lakhs IU) was given for the joint deformities and braces were advised for the same. The erythroderma showed significant improvement in 3 weeks and the follicular lesions resolved completely by 8 weeks.

[[13.0, 'year']]

M

{'10487005': 1, '28856115': 1, '13544619': 1, '9470921': 1, '19222515': 1, '7213548': 1, '19898649': 1, '20657113': 1, '19922529': 1, '24616860': 2}

{}

3937493-1

noncomm/PMC003xxxxxx/PMC3937493.xml

Disseminated extragenital bullous lichen sclerosus

A 61-year-old Caucasian female was seen with a several year history of large pruritic plaques on the trunk and proximal extremities that proved to be lichen sclerosus et atrophicus on biopsy. Over the preceding months many of the truncal lesions developed painful bullae. Multiple therapies including ultra-potent topical steroids, topical vitamin D derivative (Dovonex), narrowband ultraviolet (UV) B, and hydroxychloroquine had failed to alleviate her symptoms or halt the progression of her dermatitis in the past.\nPhysical examination revealed several hypopigmented atrophic plaques, ranging in size from 5 cm to 10 cm, consistent with LS lesions that were widely distributed over the trunk, arms, forearms, and medial thighs. Hemorrhagic bullae were present within pre-existing atrophic lesions involving her abdomen, left flank, bilateral medial arms and antecubital fossae []. No oral or genital lesions were noted. The remainder of the physical examination was unremarkable.\nBiopsies of the bullous lesions on the left flank were obtained for routine histological examination and immunofluorescence staining. Hematoxylin and eosin staining demonstrated features consistent with bullous LS including thinning of the epidermis with compact hyperkeratosis, degeneration of the basal layer, edema with the early stages of a subepidermal blister, and a band of homogenized collagen in the dermis []. Direct immunofluorescence staining was negative.\nThe patient was treated with topical calcitriol 3 mcg/g ointment and topical triamcinolone 0.1% cream daily in combination with oral acitretin 25 mg daily and responded with dramatic improvement of her lesions and level of pain. Future therapy considerations include high intensity ultraviolet A1 and immunosuppressive therapy with prednisone or methotrexate.

[[61.0, 'year']]

F

{'17367620': 1, '3342175': 1, '10348006': 1, '22044240': 1, '19917961': 1, '11807437': 1, '20854400': 1, '21353334': 1, '17291303': 1, '15024189': 1, '12366407': 1, '19281711': 1, '17224537': 1, '22533994': 1, '7868709': 1, '24616861': 2}

{}

3937494-1

noncomm/PMC003xxxxxx/PMC3937494.xml

Hyper IgE syndrome

An 18-year-old boy was referred to us because of widely disseminated erythematous plaque with scaling and lichenification since the age of one year. Growth retardation and mild psychomotor delay were observed during his development. At the six year of age, a history of intermittent epilepsy was recorded. The seizures consisted of loss of consciousness, deviation of eyes and head along with tonic posturing of arm or leg of either side or associated flushing. The seizures usually lasted for 10 minutes. The initial frequency was 5-6 times per year. He had been treated with sodium valproate. Improvement was noted after six years with frequency decreased to once per year. He was born healthy (weight = 3.59 kg, height = 49 cm) and followed the recommended schedule for vaccination. There was no history of asthma, epilepsy, trauma or atopic dermatitis among family members.\nPhysical examination revealed growth retardation for his age with height 150 cm and weight 40 kg. His facial abnormalities was obvious including a prominent forehead, a broad nasal bridge, deep set eyes, broad outer canthal distance []. Chronic eczematous dermatitis including a diffuse erythematous with scaling and lichenification spread through his face, entire trunk and extremities [Figure , ]. No obvious abscesses were noted on skin but seldom atrophic scars. Hyperextensible joints including scoliosis and talipes cavus [] were also seen. There was no abnormality of the hair or nails. Neurological examination showed limb muscle strength of grade V, normal muscular tension of upper limb, increasing active achilles tendon reflex of lower limb. The findings of the rest of the physical examination were unremarkable.\nHis laboratory work up included excessively elevated serum level of IgE 20800 IU/ml (normal range 0-100 IU/ml), IgA 441 mg/dl (normal range 69-382 mg/dl) and eosinophils 8.6% with absolute eosinophils\n0.8×109/L (normal range). Levels of IgM, IgG, and number of CD4+ and CD8+ T lymphocyte in serum were in normal range. The skin biopsy demonstrated mild epidermal hyperkeratosis with intermittent parakeratosis, exocytosis of neutrophils and intercellular edema. In the dermis, papillary edema, infiltration of lymphocytes and dilated capillaries in superficial dermis were prominent []. Maxillofacial X-ray showed primary dentition of the left maxillary first molar was retained [] and X-ray of feet showed features of talipes cavus. EEG, CT of the brain, and chest X-ray was normal. Intelligence assessment showed decreased calculation ability.\nBased on the typical clinical features, excessively increased IgE levels and the histopathology findings, the diagnosis of HIES was confirmed. Moreover, neurological complication was prominent in this case and other related neurological diseases were differentiated. The patient was treated with moisturizer for the rash and oral antihistamine. Improvement was noted at week three post-treatment and the treatment was well tolerated.

[[18.0, 'year']]

M

{'4161105': 1, '20004785': 1, '19190525': 1, '17438082': 1, '15043676': 1, '21855090': 1, '17881745': 1, '15081523': 1, '14722525': 1, '28197791': 1, '19483664': 1, '28939137': 1, '24616862': 2}

{}

3937496-1

noncomm/PMC003xxxxxx/PMC3937496.xml

Papulonecrotic tuberculid in a patient with pulmonary tuberculosis

A 55-year-old male presented with recurrent crops of crusted papular lesions on extensor surface of upper limbs and back for 1month and boils over buttocks for 20 days. He also gave history of productive cough for 15 days and occasionally he coughed out blood-tinged sputum. On examination, multiple papules with crusting and central necrosis were seen on bilateral extensor surface of upper limbs with clustering of lesions around elbows and the back [Figures and ]. Few nodules with central necrosis were seen on buttocks and scalp. One ulcer of size 4×4cm with necrotic base and surrounding erythema were seen on left buttock [].\nInvestigations revealed raised ESR 55 mm/h (Westergren), with evidence of tuberculosis on chest X-ray in bilateral lung fields. His Mantoux was strongly positive with induration of 20 mm. Sputum for AFB was positive. Biopsy from the lesion on the right elbow revealed a single focus of leucocytoclastic vasculitis affecting a small- to medium-sized vessel in mid-dermis. Surrounding this focus was dense neutrophilic inflammation. Upper dermis showed large subepidermal pustule with marked edema. There was no granuloma. These features were consistent with PNT [Figures and ]. Rest of the investigations like complete blood count, liver function tests and renal function tests were within normal limits.\nPatient was started on multidrug therapy (MDT) consisting of rifampicin 600 mg, isoniazid 300 mg, pyrazinamide 1500 mg, and ethambutol 800 mg. On starting therapy, many lesions on back disappeared within 2weeks. His general condition has improved and at 4weeks of therapy, he has no cough. He is still on MDT and his skin lesions are healing with scarring.

[[55.0, 'year']]

M

{'18474159': 1, '7802163': 1, '31238468': 2, '7642380': 1, '13593873': 1, '20921803': 1, '3384430': 1, '4279670': 1, '11155777': 1, '1769771': 1, '24616863': 2}

{'6611300-1': 1}

3937497-1

noncomm/PMC003xxxxxx/PMC3937497.xml

Multiple cutaneous reticulohistiocytoma in middle aged female

A 35-year-old female presented with gradual onset of multiple skin colored, asymptomatic pea sized swellings, which started with face and gradually involved back, arms, and legs. There was no history of preceding local trauma, discharge, pain in these lesions. There were no constitutional symptoms like fever, joint pain, and complaints referable to other systems. The patient's main concern was regarding the cosmetic appearance as lesions were present over the face. Her family history was negative and she had no history of any other chronic illness.\nThe patient appeared in good general health with normal weight and height. Mucocutaneous examination revealed multiple (20 in number), discrete, skin colored nodules, 1–2 cm in size scattered over her face, back, abdomen, thighs, and lower legs []. They were firm in consistency, non tender with a smooth surface. Hair, nail, and mucosa were normal. Her systemic examination did not reveal any abnormality. Her complete blood count along with general blood picture, liver, and renal function tests, fasting lipid profile and urinalysis were normal. Abdominal and pelvis ultrasound were found to be normal. Radiographs of chest, knee, and bilateral hands were found to be normal. Rheumatoid factor and antinuclear antibodies were found to be within normal limits. On the basis of history and clinical examination, differential diagnosis of xanthoma, reticulohistiocytoma, sarcoidosis, and histoid leprosy were made.\nExcisional biopsy of a nodule over the trunk was done and histopathological examination revealed circumscribed large focus of diffuse dense infiltrate of large histiocytes and histiocytic giant cells with scattering of lymphocytes and a few neutrophils []. The histiocytes had rounded vesicular nuclei and abundant pink homogenous stained ground glass cytoplasm. Some of these also had foamy cytoplasm. Several histiocytic giant cells resembling the Touton giant cells were also seen []. Overlying epidermis showed flattening and thinning and was covered by parakeratosis containing collection of neutrophils. Based on clinical and histopathological findings a diagnosis of multiple cutaneous reticulohistocytoma was made. The nature of the disease was explained to the patient and was referred to the plastic surgeon for the cosmetic correction of the facial lesion. Patient was advised to review if any new lesions or any new complaints develop. Lesions remained stable with no significant disability even after 6 months of follow up.

[[35.0, 'year']]

F

{'16625100': 1, '7864294': 1, '17023812': 1, '15138655': 1, '1761777': 1, '15530301': 1, '33235866': 1, '24616864': 2}

{}

3937498-1

noncomm/PMC003xxxxxx/PMC3937498.xml

Post herpes-zoster scar sarcoidosis with pulmonary involvement

The present case report is about a 50-year-old man who presented with multiple, mildly tender skin colored lesions over left upper back of 18 months duration. Patient had a history of eruption of painful, grouped blisters in this region 2 years before, that resolved leaving behind scars. At 6 months later, patient noticed skin colored raised lesions appearing over these scars that gradually increased in extent. He also complained of low grade fever and progressive breathlessness on routine work for last 6 months. There was no history of hemoptysis. Cutaneous examination revealed multiple irregular, grouped skin colored papules and plaques of 3-10 mm in size arranged in a zosteriform pattern along right thoracic (T1) segment []. There was no evidence of excessive scarring or keloid formation at the site of previous healed injuries elsewhere in the body. Systemic examination including chest examination did not reveal any significant finding. Hematological investigations including serum calcium were within the normal limits. The level of serum angiotensin-converting enzyme was 82 μg/L (normal range 8-47 μg/L). Ophthalmologic examination and electrocardiogram were normal. Mantoux test performed with 1 IU of PPD was negative after 48 h. Sputum for acid fast bacilli (AFB) was negative on 3 consecutive occasions. Chest radiography showed hilar prominence on the right side. Contrast enhanced computed tomography – chest revealed bilateral lung fields showing miliary nodules along with subpleural involvement with interlobular peribroncho-vascular thickening and interspersed areas of consolidation involving bilateral upper lobes, lingula and apical segment of right upper lobe. Areas of fibrosis in right upper lobe with emphysematous changes were seen along with bilateral hilar lymph nodes []. The findings were suggestive of pulmonary sarcoidosis. Ultrasound abdomen was normal.\nHistopathological examination of lesional biopsy on hematoxylin-eosin stained sections revealed multiple, non-caseating epithelioid granulomas with Langhan's giant cells, focal fibrinoid necrosis and occasional lymphocytes at the periphery in superficial and deep dermis consistent with the diagnosis of scar sarcoidosis []. The evidence of scar was evident in the form of haphazard thick collagen bundles in the dermis extending up to the subcutaneous tissue. Polarizing microscopy and special stains for AFB and fungus were negative. A diagnosis of scar sarcoidosis with pulmonary involvement was made. Patient was prescribed prednisolone 40 mg/day and clobetasol propionate (0.05%) cream to be applied twice a day over the lesions. At 4 weeks treatment led to decreased breathlessness and a 50% reduction in size of the lesions.

[[50.0, 'year']]

M

{'31555445': 1, '28775813': 1, '11176663': 1, '6614968': 1, '15807680': 1, '34414097': 1, '10461661': 1, '8420479': 1, '15640432': 1, '10086879': 1, '26313335': 1, '9536241': 1, '3597868': 1, '30656383': 1, '24616865': 2}

{}

3937499-1

noncomm/PMC003xxxxxx/PMC3937499.xml

Cutis marmorata telangiectatica congenita

An eighty-day-old female child, first born to nonconsanguineous parents, presented with reduced growth of left upper and lower limbs. Birth and developmental history were normal. There was no family history of vascular malformations. Physical examination revealed a fixed, purple, reticulated skin lesion involving left upper and lower limbs [] with underlying atrophic changes seen over the left extremities []. Anthropometric measurements were within the normal limits except for the reduced left upper and lower limb girths. Systemic examination did not reveal any abnormality. With the above findings, clinically we made the diagnosis as cutis marmorata telangiectatica congenita. Parents were reassured about the benign nature of the skin lesion and the need for periodic follow up on outpatient basis.

[[80.0, 'day']]

F

{'15295919': 1, '10792796': 1, '20101343': 1, '19196300': 1, '21220441': 1, '19706101': 1, '24616866': 2}

{}

3938365-1

noncomm/PMC003xxxxxx/PMC3938365.xml

Fabry disease: clinical and genotypic aspects of three cases in first\ndegree relatives

Case 1: 16-year-old male, with asymptomatic periumbilical lesions since his\nfirst years of life, progressing in number and size for the last 3 years. He presented\nright hypoacusia, tingling in hands and feet and burning plantar pain, which worsened\nwith hot weather and physical activity.\nAt the dermatological exam he presented erythematous-violaceous papules of keratotic\nsurface, grouped on the upper limbs, paravertebral, paraumbilical, inguinal, scrotum and\npenile regions, right thigh and knees ().\nIn the ophthalmological investigation the diagnosis was cornea verticillata (). He presented hearing deficit in the right\near at audiometry. In complementary exams, he showed high 24hours proteinuria (0.14; VR\n< 0.05); remaining exams were normal (blood count, renal function, microalbuminuria,\nECG, spirometry and cranial magnetic resonance imaging-MRI). The deficiency of α-Gal\nactivity was confirmed in the plasma (0.0 umol/h of protein; > 2.5) and leukocytes\nthrough paper filter (0.16 nmol / h / mg of protein; > 1.4). The biopy of umbilical\nlesion resulted compatible with angiokeratoma ().

[[16.0, 'year']]

M

{'21552486': 1, '18940466': 1, '23413206': 1, '19765408': 1, '17371887': 1, '27579746': 1, '16498740': 1, '20097359': 1, '19851669': 1, '19318041': 1, '17179052': 1, '24626659': 2}

{'3938365-2': 2, '3938365-3': 2}

3938365-2

noncomm/PMC003xxxxxx/PMC3938365.xml

Fabry disease: clinical and genotypic aspects of three cases in first\ndegree relatives

Case 2: an 11-year-old boy presented burning pains in the lower limbs,\nwhich worsened with hot weather, although less intensively than in his brother's case\n(Case 1). During examination angiokeratomas were noticed, limited to the periumbilical\nregion. He presented cornea verticillata, while the audiometry and other laboratory,\nelectrocardiographic and imaging exams were normal. Enzyme α- Gal A deficiency was\nobserved in the plasma and leukocytes (plasma: 0.11 umol/h; leukocytes: 0.46\nnmol/mg/h).

[[11.0, 'year']]

M

{'21552486': 1, '18940466': 1, '23413206': 1, '19765408': 1, '17371887': 1, '27579746': 1, '16498740': 1, '20097359': 1, '19851669': 1, '19318041': 1, '17179052': 1, '24626659': 2}

{'3938365-1': 2, '3938365-3': 2}

3938365-3

noncomm/PMC003xxxxxx/PMC3938365.xml

Fabry disease: clinical and genotypic aspects of three cases in first\ndegree relatives

Case 3: a 39-year-old female, mother of patients 1 and 2, presented with\nmild acroparestesia and cornea verticillata, without skin lesions. Audiometry revealed\ndiminished hearing in the right ear and slight loss in the left, discrete proteinuria\n(0.09) and microalbuminuria (20.93 VR < 20 ). Normal blood count, renal function,\nESR, ECG and cranial MRI. Dosage of α-Gal A activity in the plasma was within normal\nrange (3.02 umol/h).\nGenotyping of the 3 patients revealed a similar mutation: Exon 7 Arg 342 Gln. In the\ntwo-year followup, patient 1 presented worsening of lower limb pains and microangiopathy\nwas detected in cranial MRI. Patient 2 started presenting proteinuria and increase of\nangiokeratomas in size, number and distribution. The mother remained stable. The three\npatients still have not started enzyme replacement due to family decision of postponing\nthe beginning of treatment.

[[39.0, 'year']]

F

{'21552486': 1, '18940466': 1, '23413206': 1, '19765408': 1, '17371887': 1, '27579746': 1, '16498740': 1, '20097359': 1, '19851669': 1, '19318041': 1, '17179052': 1, '24626659': 2}

{'3938365-1': 2, '3938365-2': 2}

3938366-1

noncomm/PMC003xxxxxx/PMC3938366.xml

Acquired epidermodysplasia verruciformis in a renal transplant recipient\n- Case report

A 24-year-old male patient, student, presented for two years asymptomatic lesions in\nareas exposed to light, which increased progressively in number. He used oral and\ntopical antifungals without improvement. He had history of systemic lupus erythematosus\nfor 11 years and kidney transplant due to renal failure secondary to lupus nephritis for\nthe last 6 years and, for this reason, he is undergoing immunosuppressant treatment with\nsirolimus 1mg 2x/day, mycophenolate sodium 360mg/day and prednisone 10mg/day. There was\nno history of consanguinity and affected relatives. On examination, he had numerous\nerythematous papules and macules (pityriasis versicolor-like) on his face, neck,\nV-shaped neckline, forearms and abdomen (). Histologic examination of the skin biopsy from abdomen revealed vacuolated\ncells in the upper epidermis with a bubbly, bluish cytoplasm, thickened granular layer\nand mild perivascular infiltrate in the superficial dermis, consistent with EV ().

[[24.0, 'year']]

M

{'2838553': 1, '33363803': 1, '12816165': 1, '22068772': 1, '1847068': 1, '19150275': 1, '6135911': 1, '26588337': 1, '11583488': 1, '6253573': 1, '21292228': 1, '24626660': 2}

{}

3938367-1

noncomm/PMC003xxxxxx/PMC3938367.xml

Superficial Acral Fibromyxoma involving the nail's apparatus. Case report\nand literature review

We present a 43-year-old woman, without any known allergies whose personal history\nreports beta thalassemia. She referred having had cutaneous changes not associated with\nany trauma for 8 years, consisting of swelling, partial nail loss and distal ulcerations\nwith occasional bleeding on the 5th toe of the right foot. When the patient\nwore open shoes it was painless; however, it hurt and bled when she wore closed shoes.\nUpon examination, the distal end of the 5th toe presented a central ulcer\nwith blood remains and partial onycholysis ().\nAntero-posterior and oblique X-rays were requested of both feet, which showed\nsubluxation of the distal phalanges of the 5th toes without signs of bone\ninfiltration and a diagnostic biopsy was performed.\nHistological results showed neoplastic dermal proliferation of fusiform cells without\nany relevant atypia, immersed in a myxoid stroma with collagenized areas and a prominent\nvascular weave (). Immunohistochemical\nstudies reported positive results for CD34 and negative for S100, AME and AML (). The proliferation index, valued with Ki67\nwas low (less than 1%). These findings led to the diagnosis of Superficial Acral\nFibromyxoma.\nThe subsequent therapeutic approach included complete removal of the tumor as well as\nthe nail in order to avoid recurrence.\nHistological examination of the surgical piece was similar to the previously described.\nThe tumor was extirpated with wide margins, including the nail matrix, respecting the\ndistal phalanx (). Resection margins were\nreported as tumor-free.\nAfter a 22-month follow-up there was no recurrence of the tumor.

[[43.0, 'year']]

F

{'33088818': 1, '15897044': 1, '23359849': 1, '22078141': 1, '20548898': 1, '31736517': 1, '23317489': 1, '31650981': 1, '11486169': 1, '18764846': 1, '6614310': 1, '21810002': 1, '18537858': 1, '24626661': 2}

{}

3938368-1

noncomm/PMC003xxxxxx/PMC3938368.xml

Extra-pulmonary manifestations of paracoccidioidomycosis associated with\nacquired immunodeficiency syndrome: a case report

A 28-year-old male from Santarém, Pará, Brazil, with a history of leisure activities in\na rural area, tractor driver, a known carrier of the HIV virus since 2010, however\nwithout previous follow-up, contacted us for the first time at Fundação de Medicina\nTropical Dr. Heitor Vieira Dourado, Manaus, Amazonas, Brazil, for investigation of left\nknee chronic monoarthritis associated with tegumentary lesions. He had been using\nzidovudine, lamivudine, lopinavir/ritonavir for three weeks, this being the first\nantiretroviral regimen used. He had never used prophylactic\ntrimethoprim-sulfamethoxazole. He denied being a smoker or alcohol user. He reported\nthat the left knee arthritis appeared after local trauma identified five months before,\npreceding the tegumentary lesions that appeared two months after the fact. During the\nclinic exam, polymorphism of skin lesions was noted with pustules of follicular\ndistribution, papules with central necrosis, erythematous-infiltrated plaques covered\nwith pustules and ulcerated plaques, located on the face, trunk, abdomen and limbs,\nassociated with left knee arthritis (,\n and ). Oral and genital mucosa were spared. Lymphadenopathy and hepatosplenomegaly\nwere not observed. Blood cultures for aerobes, mycobacteria, fungi and leukocyte cream\nwere negative. VDRL resulted non-reagent. The Leishmaniasis screening exam was negative.\nThe analysis of synovial fluid suggested infectious arthritis. Left knee x-ray showed\nreduction of joint space and osteolytic patellar lesion (). Patient underwent surgical cleansing of joint, biopsy of synovial\nmembrane and skin. Both exams identified round cells, birefringent, with multiple\nbuddings compatible with P. brasiliensis (). Culture of synovial tissue confirmed the finding.\nComplementary propedeutics suggested pulmonary involvement and the Xpert/MTB\nRIF® sputum test was negative. At the time of diagnosis T CD4+ count was\n44 cells/mm3 . Patient was treated with Amphotericin B deoxycholate 50mg/day\n(cumulative dose of 650mg) but despite the therapy, his clinical condition worsened,\nprogressing to death on the 20th day of hospitalization. Necropsy revealed\ninvolvement of lungs, liver, spleen, kidneys, adrenals and bone marrow, in addition to\nthe previously described sites ().

[[28.0, 'year']]

M

{'21738969': 1, '18404552': 1, '27895278': 1, '8699961': 1, '16230222': 1, '11049788': 1, '16906260': 1, '24626662': 2}

{}

3938369-1

noncomm/PMC003xxxxxx/PMC3938369.xml

Segmental vitiligo after infliximab use for rheumatoid arthritis - A case\nreport

A forty-six-year old female patient, of mixed race, diabetic, with rheumatoid arthritis\nfor seven years. Denies personal or familial history of vitiligo. She mentioned the\nonset of achromic macules six months ago, initially on the left hand, which progressed\nin the craniocaudal direction involving all the left hemithorax and upper left limb.\nShe was using infliximab every eight weeks for eight months.\nShe was also using propranolol 20mg/day, metformin 850mg/day and glibenclamide\n5mg/day.\nFor this cutaneous state she had used topical 10% phenylalanine, clobetasol 0.025%, 2%\nvitamin E and Polipodium leucotomos 240 mg/day orally for 30 days, at\nanother service, with no improvement.\nAt the dermatological examination achromic macules were observed on the left upper limb\nand left hemithorax, which under Wood's light presented nacreous white coloration ().\nThere were no abnormalities detected in the remainder of the physical exam.\nAmong the laboratory exams she presented hemogram, biochemistry and thyroid hormones\nwithout changes, serologies for HIV, hepatitis and syphilis negative and one ANF 1: 640\nof mixed pattern, nuclear dense fine speckled and cytoplasmic dotted reticular, with\nnative anti-DNA autoantibodies, anti Jo, anticardiolipin, anti-centromere, anti Sm, anti\nRo, anti La, anti TPO negative.

[[46.0, 'year']]

F

{'20499000': 1, '21962188': 1, '21603808': 1, '16389417': 1, '15604556': 1, '26149498': 1, '21362795': 1, '31579486': 1, '34768860': 1, '21091114': 1, '22766431': 1, '33193342': 1, '33437542': 1, '20110063': 1, '22329760': 1, '26787888': 1, '24626663': 2}

{}

3938371-1

noncomm/PMC003xxxxxx/PMC3938371.xml

Dysplastic nevus associated with seborrheic keratosis

A 38-year-old woman presented a dark coloured papule at physical examination measuring 9\nmm on the upper right back (). Dermoscopy\nrevealed asymmetrical shape, comedo-like openings, milia-like cysts, bluish veil, and\ngranulation (). Date of lesion appearance\nwas not known. The following hypotheses were formulated: Seborrheic keratosis, lichen\nplanus-like keratosis, melanoma, and atypical nevus. Exeresis with a 2mm margin was\nindicated and histological study revealed hyperkeratosis, horny pseudocysts, basaloid\ncells, presence of fusion of nests along the sides of rete ridges, concentric\nfibroplasia, atypical melanocytes, melanophages and discrete perivascular lymphocytic\ninfiltrate. From this description the conclusion was seborrheic keratosis associated\nwith dysplastic nevus ( e ).

[[38.0, 'year']]

F

{'12472342': 1, '10775864': 1, '16637805': 1, '12622648': 1, '18328596': 1, '15056152': 1, '12472343': 1, '8806962': 1, '16637806': 1, '21308311': 1, '24626665': 2}

{}

3938374-1

noncomm/PMC003xxxxxx/PMC3938374.xml

Case for diagnosis

A 27-year-old woman at 28-weeks gestation presented with a widespread, pruritic eruption\nof macular, confluent lesions with tense vesicles and some blisters in the arms and\nthighs (). The lesions initially presented\nat 26 weeks of gestation on the legs and spread to the abdomen, arms, and back. Past\nmedical history included one prior abortion due to sicklecell disease, without any\nhistory of similar symptoms. The patient had been previously treated with\nmethylprednisolone cream and oral cetirizine, with persistence of the skin lesions. A\ncutaneous biopsy was performed in lesional skin, showing the presence of multiple\nvesicles in the dermal-epidermal junction, filled by serosity and eosinophils. In the\nunderlying dermis, a marked edema outlined a dermal-epidermal detachment, with a dense\ninflammatory infiltrate (predominantly with eosinophils) extending to the dermis (). Direct immunofluorescence in perilesional\nnoninvolved skin showed linear deposists of C3 at the basement-membrane zone ().

[[27.0, 'year']]

F

{'10457123': 1, '12894083': 1, '16488288': 1, '21909194': 1, '19520487': 1, '19298272': 1, '24626668': 2}

{}

3938375-1

noncomm/PMC003xxxxxx/PMC3938375.xml

Case for diagnosis

A 55-year-old female, who had had a kidney transplant 3 years ago and from then on, had\nbeen taking prednisone, tacrolimus and azathioprine with initial doses of 60, 150, and 2\nmg/day, respectively. These doses had been reduced and she is currently taking 10 mg\nprednisone, 100 mg azathioprine, and 1 mg tacrolimus daily. Two months ago while using\nthe same drug doses she presented asymptomatic lesions on the dorsum of her left hand\nwithout a history of local trauma.\nDermatological examination revealed a violet erythematous nodule and 2 other\nerythematous exulcerated papules on the dorsum of the left hand ().\nAn excisional biopsy of the nodule was performed for diagnosis. Histopathology showed\nchronic granulomatous inflammation and the presence of dark wall yeast structures\ncompatible with dematiaceous fungus ().\nThe other two lesions were also removed and the wound was closed with a double\nadvancement flap ().

[[55.0, 'year']]

F

{'22031645': 1, '9146569': 1, '20944912': 1, '17505670': 1, '17097407': 1, '24626669': 2}

{}

3938376-1

noncomm/PMC003xxxxxx/PMC3938376.xml

Case for diagnosis

A 72-year-old man presented erythematous and scaled plaque on the right neck for three\nmonths (). The lesion grew progressively\nin size with no pain and pruritus. He had no symptom of fever, malaise or weight loss.\nPhysical examination revealed no sign of lymph node enlargement in the bilateral parts\nof neck, axillary fossa and groin. Laboratory investigations indicated that the routine\nhaematological and biochemical studies were normal. The skin biopsy specimen revealed\ndiffuse inflammatory infiltrate consisting of lymphocytes, histiocytes, sparse\npopulation of neutrophils and plasma cells in the dermis (). Some histiocytes contained lymphocytes, neutrophils, plasma cells\nand RBCs within their pale and foamy cytoplasm (). Immunohistochemical staining revealed that histiocytes were positive for\nS-100 protein and CD68, but negative for CD1a ( e ).

[[72.0, 'year']]

M

{'18321383': 2, '16505287': 1, '12357197': 1, '21697409': 1, '21152795': 1, '24626670': 2}

{'2270859-1': 1}

3938377-1

noncomm/PMC003xxxxxx/PMC3938377.xml

Case for diagnosis

A 72-year-old man in complete remission of an immunoglobulin G kappa multiple myeloma.\nTen months later he presented firm painful erythemato-violaceous or flesh color nodules\nand tumors on the trunk, axillae and face ( and ). The patient referred\nconcomitantly asthenia and fever for 3 days. An incisional biopsy showed a dense\ninfiltrate of plasmablasts in the dermis and hypodermis (). The immunohistochemistry evidenced a dense infiltrate of\nmonoclonal plasma cells expressing kappa light chains, strongly positive for CD138\n(). Serum immunoelectrophoresis\nidentified monoclonal IgG 2152 mg/dL (700-1600 mg/dL) and κ chains 702 mg/dL (170-370\nmg/dL). Urine immunoelectrophoresis showed monoclonal κ chains: 0,925 mg/24h (0-0,7 mg).\nBone marrow biopsy evidenced plasmocytosis with cellularity of almost 100%.

[[72.0, 'year']]

M

{'2461399': 1, '387825': 1, '12707095': 1, '33282589': 2, '28761299': 2, '26438677': 1, '22892782': 1, '24626671': 2}

{'5500716-1': 1, '7714738-1': 1}

3938378-1

noncomm/PMC003xxxxxx/PMC3938378.xml

Syndrome In Question

A 54-year-old man reported a painless retroauricular lesion, with progressive growth\nover 40 years, and lesions on the face and scalp for two years. He had hypertension\nwithout other comorbidities, and reported past surgery on the nasal crease with a\ndiagnosis of trichoepithelioma. There were no children or family history.\nAt examination he presented: frontal-parietal alopecia; (1) a 3.5 x 3 cm multilobular\nviolaceous nodule of elastic consistency in the left retroauricular region; (2) multiple\nerythematous papules and nodules on the scalp, with elastic consistency and\ntelangiectasias (turban aspect); (3) normochromic papules on the upper and lower\neyelids; (4) a 1 x 1 cm nodule with a pearlaceous border and telangiectasias in the left\nnasal region; (5) a 2 x 2 mm papule with a pearlaceous border and telangiectasias in the\nleft nasogenian groove; (6) multiple normochromic millimetre size papules on the back\n().\nIncision biopsies performed on these lesions revealed: cylindroma (1, 2, and 6);\ntrichoepithelioma (3 and 5), and trichoblastoma (4) ( e ).

[[54.0, 'year']]

M

{'21886698': 1, '18718192': 1, '30241087': 1, '15953067': 1, '15677973': 1, '11703297': 1, '20098863': 1, '24626672': 2}

{}

3938379-1

noncomm/PMC003xxxxxx/PMC3938379.xml

Syndrome In Question

A 40-year-old man had, since 1996, presented arthralgia of the knees, ankles, and\nwrists; recurrent oral and genital ulcers; papulopustular lesions on the upper limbs and\nupper front torso, and painful nodules on the lower limbs.\nA diagnosis of Behçet's disease was established from clinical criteria and\nhistopathological exam. Treatment was initiated with dapsone, deflazacort, calcium\ncarbonate, and Vitamin D with lesion remission.\nIn 2010 he began recurrent crises of oedema, erythema, and painful nodules on the ear\nauricles, more accentuated on the left side, which abated after increased corticosteroid\ntreatment ().\nA smoker and drinker, he presented femural neck osteopenia and hepatic steatosis of\nalcoholic aetiology. Annual ophthalmological exam was performed without ocular\ncompromise from Behçet's disease, although he presented hypertensive retinopathy.\nLaboratory exams had shown normal hepatic enzyme levels, serum negativity for hepatitis\nB & C, hemogram, and electrolytes. Inflammatory tests were altered: VHS 28 mm and\nPCR 36.0.

[[40.0, 'year']]

M

{'20414125': 1, '27293269': 2, '17067439': 1, '775252': 1, '32914210': 1, '4014306': 1, '2360844': 1, '9365335': 1, '1970380': 1, '9656217': 1, '21437518': 1, '24626673': 2}

{'4885202-1': 1}

3938440-1

noncomm/PMC003xxxxxx/PMC3938440.xml

Acquired factor V inhibitor in a patient with mantle cell lymphoma presenting with hematuria followed by thrombosis: a case report

A 64-year-old man initially presented to us with complaints of fatigue and joint discomfort for several months prior to December 2008. He denied experiencing any fever, night sweats, and weight loss, and did not notice any swelling, recurrent infection, easy bleeding, or bruising. Further, he did not have a family history of any hematological disorder. His past medical history included hernia repair, renal colic, gastroesophageal reflux, and osteoarthritis.\nUpon examination, one left axillary lymph node was swollen to approximately 1–2 cm, and was firm and slightly tender. His spleen was palpable on inspiration, and Castell’s sign was positive; however, his liver was not enlarged. A complete blood count showed normal platelet and hemoglobin levels of 206 × 10−9/L and 155 g/L, respectively, but a high white blood cell count of 18 × 10−9/L. A leukocyte differential indicated the following: lymphocytes 10.3; neutrophils 6.75; monocytes 0.58; eosinophils 0.23; and basophils 0.05. Peripheral blood flow cytometry revealed a CD19+, CD20+, CD5+, CD23−, CD10− clonal B-cell population. Additionally, 37% of gated lymphocytes were FMC7-positive. The proportion of this FMC7-positive population reduced to 17% during a second flow analysis one month later, suggesting the possibility of mantle cell lymphoma or CD23-negative chronic lymphoid leukemia. Given that the patient was asymptomatic and showed no signs of bone marrow failure, a strategy of watchful waiting was implemented.\nIn August 2009, 8 months after his initial visit, the patient was admitted to the emergency room with a 2-day history of hematuria. Evaluation of his blood plasma revealed a prolonged international normalized ratio (INR) of 6 and an activated partial thromboplastin time of 160 seconds. Except for some minor skin bruising on his face, the patient had no other bleeding or bruising, nor was any area abnormal on examination. Further, he had no past history of bleeding, with uneventful surgical procedures and tooth extractions in the past. On careful investigation of his coagulopathy, a prothrombin time mixing study was abnormal at −43 partially corrected to 30 seconds only (normal 12.5–15.7 seconds), suggesting the presence of an inhibitor. The thrombin time was 15.8 seconds (normal 15.5–18.3 seconds), serum fibrinogen level was 5.8 g/L (normal 2.00–4.30 g/L), factor V level was <0.01 U/mL (normal 0.5–1.5 U/mL), factor VIII level was 3.23 U/mL (normal 0.5–1.5 U/mL), factor X level was 1.08 U/mL (normal 0.5–1.5 U/mL), and factor IX level was 1.17 U/mL (normal 0.5–1.5 U/mL). Lupus anticoagulant testing could not be interpreted owing to the presence of the inhibitor. Serum protein electrophoresis did not indicate the presence of a monoclonal protein. Based on these results, a factor V inhibitor test was performed and indicated a factor V inhibitor titer of 80 Bethesda units. The patient was started on prednisone (1 mg/kg, 80 mg daily), and after 2 weeks of treatment, the bleeding had stopped and serial measurement of factor V levels showed a dramatic increase; moreover, within 7 weeks, factor V levels were normal and factor V inhibitor was undetectable.\nSurprisingly, 7 weeks after initiation of steroid treatment, the patient developed an unprovoked severe pain with swelling in his right leg. A week prior to this event, his prothrombin time was 17 seconds, activated partial thromboplastin time was 28 seconds, INR was 1.4, and the factor V assay value was 0.62 Bethesda units/mL, but factor V inhibitor was undetectable. Doppler ultrasound revealed deep venous thrombosis, and low-molecular-weight heparin (dalteparin, 12,500 U) and coumadin (7.5 mg) were administered orally once daily. The patient’s condition continued to improve with no further bleeding or thrombotic events, and anticoagulation was successfully discontinued after 8 months. The use of prednisone was gradually decreased over several weeks after confirmation of the disappearance of the inhibitor, and was discontinued completely in December 2009 (). Screening assays to rule out inherited thrombophilia were negative for antithrombin, protein C, protein S, factor V Leiden, and prothrombin 20210 gene mutations. Fluorescence in situ hybridization analysis of a peripheral blood sample showed IGH/CCND1 t(11;14), which confirmed the presence of a clonal B-cell population characteristic of mantle cell lymphoma.

[[64.0, 'year']]

M

{'9462552': 1, '19438479': 1, '7772528': 1, '12010798': 1, '11896308': 1, '7082847': 1, '21052780': 1, '19404538': 1, '21761437': 1, '23080366': 1, '14375712': 1, '34675735': 2, '22473049': 1, '15613918': 1, '8034625': 1, '8493990': 1, '7741151': 1, '22588327': 1, '30446942': 1, '24591851': 2}

{'8504934-1': 1}

3938441-1

noncomm/PMC003xxxxxx/PMC3938441.xml

A rare case of neuroleptic malignant syndrome without elevated serum creatine kinase

A 51-year-old man had experienced repetitive manic and delirious episodes since he was 17 years old and was previously diagnosed with schizoaffective disorder. He had to be psychiatrically hospitalized six times for recurrent psychotic episodes after discontinuing his medications. He did not have a history of drug allergies. In 1996, at the age of 34, he was admitted to a psychiatric hospital, where he remained until the time of the current report. At the time of admission, his height and weight was 172 cm and 63 kg, respectively. His body mass index (BMI) was 21.3 Kg/m2. Throughout the first year of admission, he suffered repeated manic episodes with excitement and delusion. During these episodes, he often had to be taken into a private room because of his violent behavior towards other patients. Since he often refused to take medications, he was given fluphenazine decanoate (25 mg), injected intramuscularly, every month. Additionally, he was given lithium carbonate (600 mg), zotepine (150 mg), haloperidol (9 mg), and biperiden (6 mg) daily. While being treated with lithium carbonate, the serum lithium levels were within the therapeutic range. Furthermore, during periods when he became manic and excited, his dietary intake decreased, leading to large decreases in body weight. As a result, his body weight ranged from 63 to 53 kg.\nHis body weight was about 55 kg in the first half of 2011 but was 63 kg in January of 2012. After this, his body weight decreased gradually along with the deterioration of his mental state, and by December of 2012, he weighed 49 kg. His changes in BMI are shown in . There were no complaints from the patient, but he was suspected of having malignant disease. However, blood tests showed no abnormalities: white blood cell (WBC) count 7,900/mm3, red blood cell (RBC) count 344 × 104/mm3, total protein 7.6 g/dL, total cholesterol 169 mg/dL, aspartate aminotransferase (AST) 12 mU/mL, alanine aminotransferase (ALT) 9 mU/mL, and CK 51 mU/mL (normal range 48–248).\nOn January 31, 2013, the patient weighed 48 kg. Upon evaluation, his blood pressure was 96/60 mmHg, his pulse rate was 58 bpm, and his body temperature was 35.5°C. At that time, valproic acid (600 mg), lithium carbonate (800 mg), zotepine (150 mg), and risperidone (6 mg) were administered. On February 11, he became excited and was physically violent towards other patients and was transferred to a private ward. On February 12, the patient received fluphenazine decanoate (25 mg) by intramuscular injection. On February 13, he was physically able to have a meal by himself. On February 14, when a nurse went for her rounds in the morning, she noticed that he could not raise himself up. He did not show headaches, meningeal signs, or focal neurological signs. Upon evaluation, his blood pressure was 106/72 mmHg, his pulse rate was 102 bpm, and his body temperature was 36.0°C but rose to 38.5°C in the afternoon (day 1 of NMS). Since he had difficulty swallowing, all medications were discontinued, and he received intravenous fluid replacement. At that time, an antibiotic was administered intravenously upon suspicion of an infection. On February 17, it was noted that he was unable to speak due to marked muscle rigidity, but he did not show diaphoresis. His chest X-rays were normal. On February 18, blood tests showed the following: WBC count 10,800/mm3 and CK 74 mU/mL. On February 20, diaphoresis was observed. Since fever, generalized “lead pipe rigidity”, and dysphagia were also found, NMS was suspected. Only supportive therapy, including intravenous fluid replacement and cooling, was sustained. Because his serum CK was within the normal range, we were hesitant to administer dantrolene; thus, dantrolene was not given. In addition, since the patient had been mute during the disease, the administration of benzodiazepines or electroconvulsive therapy (ECT) was not planned. Although the patient’s serum CK was measured two more times, the levels were within normal range. On February 24, the patient’s body temperature was 38.4°C, and hypersalivation and tachypnea (respiratory rate 34/min) became prominent. On February 25, his oxygen saturation was noted to be 89% with room air, and 100% O2 was immediately given via face mask. However, he suddenly died of respiratory failure on February 26 (day 13). An autopsy was not performed.\nThe profile of the clinical course, including body temperature, the degree of muscle rigidity, and laboratory findings consisting of the levels of serum CK and WBC, is shown in . Muscle rigidity was rated using the NMS evaluation scale proposed by Sachdev.

[[51.0, 'year']]

M

{'17541044': 1, '3290944': 1, '19898573': 1, '2863986': 1, '2886157': 1, '15996751': 1, '23818785': 1, '8093494': 1, '34294629': 2, '33520321': 2, '30567175': 1, '2876647': 1, '19698658': 1, '8731718': 1, '9590359': 1, '16095481': 1, '33272348': 2, '28144147': 1, '24591835': 2}

{'7817295-1': 1, '7791564-1': 1, '8316652-1': 1}

3938496-1

noncomm/PMC003xxxxxx/PMC3938496.xml

Ischemic retinal vasculitis in an 18-year-old man with chickenpox infection

A healthy 18-year-old man presented with a 2-week history of chickenpox, and was treated with oral acyclovir for 5 days. Ten days after onset of the cutaneous vesicular eruptions, he experienced blurring of vision in his right eye that descended to the level of hand motion (HM) in 6 hours. At presentation, 4 days after blurred vision, visual acuity in his right eye was hand movement. Examination of the anterior segments was unremarkable except for an obvious relative afferent pupillary defect. His extraocular muscle examination was normal. Fundus examinations revealed an edematous disc, retinal whitening and edema along the posterior pole, and the presence of a cherry red spot; no retinal infiltration or vitreous opacity was observed (). His left eye was normal with 20/20 vision. Optical coherence tomography showed increased thickness and hyperreflectivity in the inner retina layer, consistent with inner retina infarction. The early phase of fundus fluorescein angiography demonstrated delayed filling of the optic disc with minimal leakage from the small retinal arteriole, which was more prominent in the macular and optic disc regions. Minimal capillary dropout in the macular area was also noted. Staining of small vessels was observed in the late phase of the angiogram (–). A multifocal electroretinogram (ERG) showed diminished waveform in the entire macular area (). A full field ERG showed decreased b-wave amplitude of both rod and cone response and oscillatory potential wave. Electronegative ERG pattern was evidenced in maximal combined ERG that represented a generalized ischemic process, which involved mainly the inner retina layer (). The patient demonstrated decreased amplitude and increased latency in flash visual evoked potential (VEP), which is consistent with generalized involvement of the entire retina, not limited only to the posterior pole ().\nMinimal increased signal in T2-weighted magnetic resonance imaging and enhancing with contrast limited to the anterior part of the optic nerve could represent an inflammatory response to the ischemic process of the optic nerve head (). Serologic tests were positive for both immunoglobulin (Ig)M and IgG anti-VZV antibodies, confirming recent primary VZV infection. The diagnosis of ischemic vasculitis of the small retinal vessels associated with chickenpox was made. Since this process could either be caused by infection or an immunological response to VZV infection, and there is no consensus on the optimal treatment regimen for this VZV-associated vasculopathy, either treated with antiviral alone or combined with corticosteroid, we decided to treat the patient with antiviral agent according to the acute retinal necrosis treatment guideline published by the American Uveitis Society, ie, with intravenous Acyclovir 10 mg/kg every 8 hours for 2 weeks in conjunction with intravenous methylprednisolone 1 g daily for 3 days, then with oral prednisolone tapering over 1 month and oral Acyclovir 800 mg 5 times daily for 1 month. Despite the treatment, a pale optic disc and attenuated retinal artery resulted. His final best-corrected visual acuity was counting fingers at 1 foot at 6 months’ follow-up.

[[18.0, 'year']]

M

{'11589889': 1, '18657721': 1, '1666176': 1, '16123552': 1, '20622712': 1, '17675031': 1, '27896784': 2, '32823430': 1, '11914224': 1, '21482932': 1, '24591814': 2}

{'5126028-1': 1, '5126028-2': 1, '5126028-3': 1, '5126028-4': 1, '5126028-5': 1}

3939373-1

noncomm/PMC003xxxxxx/PMC3939373.xml

Cervical Symmetric Dumbbell Ganglioneuromas Causing Severe Paresis

A 72-year-old man came by ambulance to our hospital due to progressive incomplete paraplegia. One month previously, he had felt left upper extremity numbness. He could not stand alone or walk because of sudden onset lightheadedness and reduced muscular strength in both legs. Manual muscle test of the right side arm and leg (below C2) was graded as 4- and that of the left side arm and leg (below C2) was graded as 4. He showed bladder dysfunction. He had incomplete sensory loss below C3. Tonus was increased in both legs and deep tendon reflexes were hyperactive. In addition, he had been diagnosed with gastric cancer and had undergone total gastrectomy and splenectomy, resulting in peritoneal dissemination.\nMagnetic resonance imaging (MRI) demonstrated bilateral asymmetric masses at the C1/2 level. T1-weighted images exhibited hypointensity. T2-weighted images revealed heterogeneous hyperintensity (). Contrast-enhanced T1-weighted images showed enhancement in the tumor parenchyma (). Computed tomography myelogram showed spinal cord compression bilaterally at C1/2. Because the progression of the paralysis was rapid and the patient hoped for a palliative surgical procedure, we performed emergency operation to remove the intracanalar tumor and decompress the spinal cord.\nAfter the posterior arch at C1 and cranial portion of the C2 lamina was removed and the dura was exposed (), extradural bilateral tumor masses at C1/2 were identifiable (). Tumors surface were smooth with capsules. Firstly the extradural tumor was partially resected and the debulking effect obtained. Bilateral C2 dorsal roots were swollen, with cancerization and connected intradural portions. After durotomy, bilateral symmetric tumors originating from bilateral C2 dorsal root were identified (). These were partially adherent to the dura. The intradural portions of the tumors were totally resected piece by piece. Consequently, C2 bilateral dorsal roots were sacrificed. Subtotal resection for the bilateral extradural portions was then carried out. It was confirmed that that intracanalar portions were totally resected and the emergency operation was thus terminated, because radical resection for the extracanalar portions, which would have required dissection of the tumor from vertebral arteries, was not deemed appropriate given the patient's relatively poor general condition. Finally, we performed duraplasty with artificial dura mater to extend the intradural space.\nHistopathological examination of the surgical specimen indicated the tumor cells to be spindle cells with the presence of ganglion cells and no cellular pleomorphism. Ganglion cells were dissipated throughout the tumor (). The tumors were thus diagnosed as ganglioneuromas.\nAfter the operation, the patient's paraplegia improved gradually and he became able to walk. MRI after operation showed that bilateral foraminal portions were left intact and the intracanal tumor was completely resected, with sufficient spinal cord decompression (). He was followed up with MRI at three month intervals. Two years postoperatively, there was no recurrence intracanally and no increase in the foraminal portions. Taking into account his poor general condition, including cancerous ascites due to the peritoneal dissemination of past gastric cancer, we are not planning to perform an additional operation as long as his improvement in paresis is maintained.

[[72.0, 'year']]

M

{'15120227': 1, '30637260': 2, '11358598': 1, '13917633': 1, '15730843': 1, '20362242': 1, '16230822': 1, '13536319': 1, '18037807': 1, '28490166': 2, '27371333': 1, '8803848': 1, '24596609': 2}

{'6327123-1': 1, '5426442-1': 1}

3939384-1

noncomm/PMC003xxxxxx/PMC3939384.xml

Acute renal failure and normal blood count: A rare presentation of T-cell acute lymphoblastic leukemia

A 10-year-old previously healthy Caucasian boy presented with blurred vision, headache, nausea, and vomiting. The symptoms occurred intermittently without circadian variability. The patient had bilateral papilledema. He was pale but otherwise unaffected without enlarged lymph nodes or abdominal organs by palpation. Urinary production was normal. The blood pressure was elevated: 128/91 mmHg. Creatinine 101 μmol/l, urea 6.7 mmol/l, hemoglobin 9.5 g/l, platelets 150×109/l, white blood cell count 4.5×109 cells/l, absolute neutrophil count 1.75×109 cells/l, and lactate dehydrogenase 645 IU/l. Uric acid was 0.97 mmol/l and rasburicase was administrated twice, resulting in a rapid decrease (). Urine analysis showed marginal proteinuria of 0.2 g/day. An MRI scan of the cerebrum performed on day 3 showed no signs of malignancies or increased intracranial pressure.\nThe renal failure progressed reaching a peak creatinine level of 534 μmol/l. An ultrasound of the kidneys and urinary tract showed no signs of obstruction but bilateral diffuse enlargement of the kidneys (16 cm in craniocaudal length on both sides). A renal biopsy and a bone marrow aspiration and biopsy were performed on the fourth day from admission. The kidney biopsy showed massive infiltration of immature lymphoblasts (). The bone marrow biopsy showed 90% blasts of T-cell linage (CD3+, CD4+, CD10+, CD117+, CD7+, CD5+, and CD8−). Lumbar puncture showed CNS involvement with 12×106 cells/l; most of the cells were lymphoblasts.\nTreatment was initiated according to the NOPHO-ALL 2008 high-risk protocol. Due to high risk of tumor lysis, treatment was started with low-dose prednisolone alone (). Kidney function improved rapidly following initiation of therapy and dialysis did not become necessary.\nThe patient responded well to the leukemia treatment and remains in first complete remission three years after diagnosis. No further renal complications have occurred during the course of therapy and kidney size has returned to normal.

[[10.0, 'year']]

M

{'34113542': 1, '7626380': 1, '15160965': 1, '1395166': 1, '33489446': 1, '2355888': 1, '19437368': 1, '26376596': 1, '8846137': 1, '19998467': 1, '18239911': 1, '7484018': 1, '29901649': 2, '6940648': 1, '24596675': 2}

{'6024069-1': 1}

3941085-1

noncomm/PMC003xxxxxx/PMC3941085.xml

Inflammatory Bowel Disease, High-Dose Steroids, Osteoporosis, or an Oncological Etiology for a Pathological Femoral Neck Fracture in a Young\nAdult: A Case Report

A 29 year-old African-American female presented late one evening to a community hospital emergency department with severe right hip and groin pain. She stated the pain occurred during ambulation at a local store. She denied any trauma, but stated that the pain caused her to fall to the ground. She reported a three to four week history of groin and thigh pain for which she had been evaluated at a tertiary center emergency department three weeks earlier. At that time, radiographs were performed of the pelvis, hip, and sacroiliac joints (see Fig. ). She was diagnosed with a ‘hip strain’ and referred to outpatient physical therapy and primary care physician follow-up.\nShe had seen her primary care physician just prior to this current emergency department visit and admitted to receiving corticosteroid injection in the right trochanteric bursa for suspected trochanteric bursitis. Radiographs at the time of the current ED presentation are seen in Fig. ().

[[29.0, 'year']]

F

{'21061812': 1, '10505526': 1, '23283374': 1, '11254818': 1, '22962914': 1, '7608228': 1, '34668325': 1, '9807723': 1, '11804908': 1, '21605729': 1, '10796394': 1, '6725326': 1, '18829925': 1, '21701389': 1, '21586782': 1, '11433141': 1, '21649917': 2, '22294756': 1, '15269616': 1, '17805021': 1, '8422726': 1, '18757967': 1, '17664365': 1, '22971243': 1, '23010681': 1, '16343263': 1, '18676894': 1, '15614244': 1, '14251448': 1, '17948152': 1, '25673450': 1, '17169149': 2, '18832603': 1, '24596584': 2}

{'3118210-1': 1, '1716166-1': 1}

3941716-1

noncomm/PMC003xxxxxx/PMC3941716.xml

Steroid-induced Spinal Epidural Lipomatosis after Suprasella Tumor Resection

A 32-year-old woman was admitted in a wheelchair. She presented with leg weakness and radiating pain on both buttock, posterior thigh & calf for 1 year. Because pain and weakness in both legs was gradually aggravated during 4 months, she could not walk or stand when she was admitted. Prior to admission, she underwent an operation for a suprasellar tumor resection in our hospital when she was 14 years old. At that time, the pathologic diagnosis was pilocytic astrocytoma. After the operation, panhypopituitarism occurred, so steroid hormone replacement therapy was started in 1993. For a period of 19 years, exogenous steroid maintenance had been administrated (median dose of prednisolon 7.5mg). The neurologic examination revealed both legs weakness (motor grade IV/IV) and radiating pain on the L5 and S1 dermatomes. The straight leg raising test was negative, but the bilateral knee jerk and ankle jerk were slightly hyperreflexive. A magnetic resonance imaging (MRI) study showed severe compression of the lumbar spinal cord by epidural fat tissue. At the dorsum of the spinal cord from the L1 to L5 levels, high signal lesions on T1- and T2-weighted images were checked. The longest length from the dorsum of the spinal cord to the ligamentum flavum was 11.4mm on the L3 level and the median length was 9.2mm at the L1-L5 levels. Fat suppression MR imaging showed low signal intensity at the same lesion () as well. In the enhanced T1-weighted MRI, the epidural vessels were more developed than in a normal person's MRI. The engorged epidural vessels were positioned beneath the nerve root exit site, bilaterally ().\nConsidering the long-term exogenous steroid administration, her neurologic symptoms and MR images, we could make the diagnosis of spinal epidural lipomatosis. Because she complained of radiating pain on the bilateral L5 and S1 dermatomes, she had an operation to decompress the L5 and S1 nerve roots. In the operation field, fat tissue compressed the dorsum of the thecal sac and engorged epidural vessels were found on nerve root exit sites (). We removed the epidural fat tissue and coagulated the engorged epidural vessels for nerve root decompression. The postoperative course was good. The radiating pain in both legs was dramatically relieved and motor power was slightly improved. She was admitted in a wheelchair but discharged walking. After 1 month, we did a follow-up MRI. On the follow-up MRI, the epidural fat tissue did not compress the thecal sac and the courses of nerve roots were not interrupted.

[[32.0, 'year']]

F

{'20035646': 1, '18954986': 1, '19703892': 1, '11486570': 1, '3358807': 1, '21341972': 1, '15191340': 1, '20514010': 1, '1092979': 1, '19803112': 1, '11939947': 1, '16490695': 1, '15627168': 1, '21332929': 1, '15795966': 1, '17607308': 1, '29599885': 1, '24757466': 2}

{}

3941717-1

noncomm/PMC003xxxxxx/PMC3941717.xml

Anterior Screw Fixation of Anteriorly Displaced Type III Odontoid Fracture Corrected by Transoral Digital Manipulation

A 30-year-old man was transferred to the emergency department due to neck pain by falling down. There was no neurological deficit on admission. Cervical computed tomography (CT) showed the type III odontoid fracture accompanying partial fracture of left superior articular process of the axis () and a 5.4-mm anterior displacement of the dens (). Cervical magnetic resonance imaging showed an intact transverse atlantal ligament and no spinal cord injury. Gardner-Well tongs skull traction was performed. A reduction of displaced dens was failed. We decided to perform a surgery for intraoperative reduction and stabilization. Under general anesthesia with somatosensory evoked potential and motor evoked potential monitoring, closed reduction using distraction with flexion and extension was failed. A surgical assistant attempted to compress the posterior oropharynx via the patient's mouth under simultaneous fluoroscopic visualization. A fluoroscopic image showed a reduction of displaced bone fragment (). Anterior lag screw fixation was achieved under continuous transoral digital compression, but complete reduction was not achieved (). There was no neurological abnormality intraoperatively and postoperatively. After six months, he did not complain neck pain and show a limitation of cervical rotation. Radiographs showed an osseous union, although that the posterior margins of both dens and axis body were not matched after six months (). There was no instability in dynamic flexion-extension X-rays (). After one year, the patient was lost to follow-up.

[[30.0, 'year']]

M

{'4434035': 1, '16859271': 1, '20173530': 1, '10515475': 1, '25983835': 2, '9280020': 1, '19373433': 1, '28704908': 1, '20179556': 1, '4077905': 1, '16506472': 1, '24757470': 2}

{'4431022-1': 1}

3941718-1

noncomm/PMC003xxxxxx/PMC3941718.xml

Gelfoam-induced Swallowing Difficulty after Anterior Cervical Spine Surgery

A 77-year-old man was presented with neck pain for several months. Cervical lateral radiographics demonstrated a multisegmental anterior osteophyte formation, especially at C 3-4 (). However, he did notcomplain of any respiratory and swallowing difficulties. Therefore, we decided that conservative treatment may be better suited for this case. Five months later, he was admitted to our hospital with swallowing difficulty, which occurred ten days prior to presentation. He could only eat soft foods with frequent coughing. A computed tomography (CT) scan was performed and demonstrated anterior osteophyte with indentation on the esophagus (). Magnetic resonance (MR) images showed multisegmental anteriorly flowing osteophyte formation with esophageal indentation. The videofluoroscopic swallowing study (VFSS) showed abnormal coordination of peristaltic movement and reflux to the trachea on a pharyngeal phase. We recommended tube feeding.\nWe removed the DISH via anterolateral cervical approach and applied gelfoam on the osteophyte removal site, owing to the uneven surface of the remained osteophyte. After the operation, the patient was relieved from the swallowing difficulty and had normal diet, except solid foods. On POD 7, he complained of swallowing difficulty again, but the degree of dysphagia was less than the preoperative state. His vital signs, including respiration, were stable. Cervical lateral radiographics demonstrated air formation on osteophyte removal site (). CT scan demonstrated free air with low density fluid collection on the prevertebral and right paravertebral space and swelling (). On laryngoscopic examination, the perforation and fistula were not noted. Endoscopic examination showed no esophageal perforation. The repeated VFSS showed a decline of laryngeal elevation and disability of laryngeal closure, and recommended small amounts oral feeding. The symptom was not severe and respiratory difficulty was not noted. We closely observed the patient.\nFourteen days later, the swallowing difficulty was relieved and repeated CT scan showed lesser compression to the esophagus, in spite of the free air and swelling that still remain ().\nThree months later, the patient could eat all foods, including solid foods. The radiographic showed no swelling and free air ().

[[77.0, 'year']]

M

{'18495543': 1, '11598530': 1, '7644964': 1, '34734343': 1, '20099000': 1, '22084604': 1, '16803720': 1, '19478651': 1, '9950109': 1, '19714374': 1, '17906571': 1, '18646336': 1, '24757468': 2}

{}

3941719-1

noncomm/PMC003xxxxxx/PMC3941719.xml

Spontaneous Regression of Extruded Lumbar Disc Herniation: Three Cases Report

A 64-year-old male was admitted to our hospital with radiating pain and numbness in the right lower extremity. The symptom was aggravated one week ago. The motor weakness and sensory change was not noted. Magnetic resonance (MR) images showed extruded herniated nucleus pulposus (HNP) at the L 3-4 level (). He refused surgical treatment, so we performed transforaminal neural block. The symptom was relieved and he was discharged. On subsequent follow up, the symptom appeared to be gradually relieved. Follow-up MR study was done 3 months after and showed no HNP (). He did not complain of any symptom at that time.

[[64.0, 'year']]

M

{'17561749': 1, '33408591': 1, '30813213': 1, '34812080': 2, '19107686': 1, '8720409': 1, '21082061': 1, '10332782': 1, '15183452': 1, '30762791': 1, '11880844': 1, '10707394': 1, '25620984': 2, '8793781': 1, '11750011': 1, '16688039': 1, '15609071': 1, '18299107': 1, '11679814': 1, '25620990': 1, '8720408': 1, '9932124': 1, '25883664': 2, '24757463': 2}

{'3941719-2': 2, '3941719-3': 2, '8649452-1': 1, '4303279-1': 1, '4398824-1': 1}

3941719-2

noncomm/PMC003xxxxxx/PMC3941719.xml

Spontaneous Regression of Extruded Lumbar Disc Herniation: Three Cases Report

A 74-year-old male was admitted to our emergency room with pain that aggravated a day ago on the right anterior thigh and buttock. He had undertaken discectomy at the L 4-5 level 2 years ago. MR images showed ruptured HNP at the L 1-2 level (). He did not have motor weakness and bladder symptom. We started pain control by intravenous analgesics and muscle relaxant. Pain was subsided and he wanted to be discharged with oral medication. Three months later, he was admitted to our hospital for a follow-up checkup with no symptom. Ruptured HNP was not noted on MR image ().

[[74.0, 'year']]

M

{'17561749': 1, '33408591': 1, '30813213': 1, '34812080': 2, '19107686': 1, '8720409': 1, '21082061': 1, '10332782': 1, '15183452': 1, '30762791': 1, '11880844': 1, '10707394': 1, '25620984': 2, '8793781': 1, '11750011': 1, '16688039': 1, '15609071': 1, '18299107': 1, '11679814': 1, '25620990': 1, '8720408': 1, '9932124': 1, '25883664': 2, '24757463': 2}

{'3941719-1': 2, '3941719-3': 2, '8649452-1': 1, '4303279-1': 1, '4398824-1': 1}

3941719-3

noncomm/PMC003xxxxxx/PMC3941719.xml

Spontaneous Regression of Extruded Lumbar Disc Herniation: Three Cases Report

A 67-year-old male was admitted to our hospital with right lower extremity pain and hypoesthesia, especially on the left L5 dermatome. MR images showed extruded HNP at the L 4-5 level (). We recommended surgical treatment, but he refused. As such, we treated conservatively. The pain was relieved gradually and followed up as an outpatient. Nine months later, the pain disappeared and the hypoesthesia was weakly remained. Follow-up MR images showed no HNP ().

[[67.0, 'year']]

M

{'17561749': 1, '33408591': 1, '30813213': 1, '34812080': 2, '19107686': 1, '8720409': 1, '21082061': 1, '10332782': 1, '15183452': 1, '30762791': 1, '11880844': 1, '10707394': 1, '25620984': 2, '8793781': 1, '11750011': 1, '16688039': 1, '15609071': 1, '18299107': 1, '11679814': 1, '25620990': 1, '8720408': 1, '9932124': 1, '25883664': 2, '24757463': 2}

{'3941719-1': 2, '3941719-2': 2, '8649452-1': 1, '4303279-1': 1, '4398824-1': 1}

3941720-1

noncomm/PMC003xxxxxx/PMC3941720.xml

Triple Primary Origin Tumor: A Case Report

An abdominal CT scan was taken of a 57-year-old male patient who had sacral and right buttock and thigh pain, and revealed with a tumor-like lesion at the T12 vertebrae. Previously in 2007, the patient had been diagnosed with colon cancer, confirmed as adenocarcinoma by open biopsy. Targeting the cancer lesion, only endoscopic mucosal resection was executed at that time and was considered as a cure. After a period of 3 years, on a follow up large intestine endoscope, a recurred lesion was identified, thus abdominal low anterior tumor resection with colostomy was performed immediately. The patient was also diagnosed with liver cancer in 2009, which was confirmed as hepatocellular carcinoma. About this hepatic lesion, the patient was treated by radio-frequency ablation twice.\nThe T12 level tumor mass was first discovered in the abdominal CT which was taken to confirm the hepatic lesion, and in the follow-up abdominal CT executed 6 months later, the diameter of tumor mass was increased from 1.5 cm to 4 cm (, ). First, it was presumed that the lesion might be a metastatic lesion originated from the primary colon or liver cancer considering the previous cancer history of the patient. As a treatment for the vertebral lesion, total laminectomy, right hemi-vertebrectomy of T12 and tumor resection was performed, and pedicle screw fixation of T10, T11, L1, and L2 vertebral bodies were done. Observed intra-operatively, the tumor was found to be a brownish, hypervascular, and friable mass compressing the thecal sac on right side of T11-12 level, and additional erosion of the T12 lamina was also identified.\nOn the final pathologic report, however, it was confirmed as plasmacytoma (, ), which was completely different pathologically from previously diagnosed colon cancer or liver cancer. On additional immunostaining, the tumor cells also showed a monoclonality for lambda immunoglobulin in its light chain.\nIn conclusion, the patient was finally diagnosed to have triple primary-origin tumors of adenocarcinoma, hepatocellular carcinoma, and vertebral plasmacytoma. The patient underwent chemotherapy after the surgery.

[[57.0, 'year']]

M

{'17335683': 1, '10631690': 1, '19880681': 1, '12901544': 1, '21427182': 1, '20109225': 1, '24757467': 2}

{}

3941722-1

noncomm/PMC003xxxxxx/PMC3941722.xml

Desmoid Tumor of the Facet Joint: A Case Report

A 32-year-old man was admitted to our hospital for diagnosis and treatment of lower back pain, both buttock pain, and radiating pain in the left leg that were present for 1 month. He was a computer programmer who had experienced a traffic accident as a passenger 1 year ago, but he had no history of spine surgery. He had undergone nerve blocks several times for the treatment of pain. However, the treatment had had little effect.\nComputed tomography (CT) images of the lumbar spine showed a smooth, low-density area at the superior and inferior articular process of the left L3-4. Homogeneous enhancement was noted after contrast media injection, with pressure erosion of the facet joint and left lateral lamina (). On axial T1- and T2-weighted magnetic resonance imaging (MRI) scans, an intermediate-signal intensity mass of approximately 1.7×1.2×1.8 cm was seen in the left L3-4 paraspinal area. Strong enhancement was noted in the same region after contrast media injection ().\nThe patient underwent left L3-4 partial facetectomy without internal fixation and tumor removal under general anesthesia. In the operative field, a tumor, approximately 2.0×2.5 cm in size, with encapsulation was observed in the left L3-4 facet joint. The tumor was completely resected from its facet joint adhesions with a safety margin. The tumor was firm and yellowish-red in color ().\nHistology of the facet joint showed collagenous tissue infiltration and proliferation of apparently benign spindle cells separated by collagen that are characteristics of desmoid tumors. The histologic findings were typical of benign infiltrative fibromatosis, desmoid tumors ().\nFor 1 month, the patient performed rehabilitation exercises. Subsequently, his lower back pain, both buttock pain, and radiating pain in the left leg were significantly improved. Ten months after the operation, the patient had no neurologic deficit and no instability in following up radiography.

[[32.0, 'year']]

M

{'7939911': 1, '1749859': 1, '12385367': 1, '1831400': 1, '12663732': 1, '4717135': 1, '13706852': 1, '2546214': 1, '3962627': 1, '16739877': 1, '3616218': 1, '2756458': 1, '21534212': 1, '12467833': 1, '3824278': 1, '8488459': 1, '9336146': 1, '3240083': 1, '3946757': 1, '8905114': 1, '7889507': 1, '24757464': 2}

{}

3941723-1

noncomm/PMC003xxxxxx/PMC3941723.xml

Hemorrhagic Synovial Cyst Associated with Rheumatoid Atlantoaxial Subluxation

A 72-year-old woman presented with sudden severe headache in her left occipital area with dyspnea. The patient was alert and the function of cranial nerves was intact. Motor power and sensory were symmetric in all extremities. The dyspnea was aggravated on supine position. It was relieved on flexed head position. She had rheumatoid arthritis for 14-year, which was clinically and serologically proven. A laboratory examination of the patient's blood revealed no remarkable abnormality indicative of inflammation, but high-titer of rheumatoid factor (95.9 IU/mL). Seven months ago before visiting to our department, she checked brain MRI due to sudden attack of severe headache on left occipital area, which revealed non-specific lesion except rheumatoid pannus with small prevertebral cyst of C1-2 junction ().\nOn admission, there was a large hemorrhagic cystic mass around prevertebral space of the atlanto-axial joint on the left side, obstructing the nasopharyngeal cavity on cervical MRI (). The cystic mass was connected to atlanto-axial joint capsule on axial view of computed tomography (CT). Multiple bony erosion, rheumatoid atlanto-axial instability including left tilted C1-2 subluxation and cranial settling were demonstrated. And there were loss of lordosis and retrolisthesis in C 3-4, 4-5 ().\nIn the first operation stage, considering patient's dyspnea, aspiration of the cystic lesion was performed via transoral approach with otolaryngology surgeon () and airway obstruction was relieved. The volume of aspirated cystic fluid was about 5 cc. The color of specimen was xanthochromic, which suggested old hemorrhage. After 0.5 cm-sized mucosal incision on left side oropharyngeal wall, residual material of the cyst was squeezed by forceps and removed by suction tools.\nIn the second operation stage, there were rheumatoid atlanto-axial instability and retrolisthesis in C 3-4, 4-5, so it was followed by posterior occipito-cervical fusion that connected from occiput to C5, using vertex screw & rod system and iliac bone graft (). All the procedure was successfully done.\nThe patient was tolerable on her postoperative course and showed good respiration and relieved headache.

[[72.0, 'year']]

F

{'3561742': 1, '8893718': 1, '25593779': 2, '16925084': 1, '19564758': 1, '10752109': 1, '14652484': 1, '20872595': 1, '8862848': 1, '29443754': 2, '24757465': 2}

{'4287902-1': 1, '5839867-1': 1}

3941724-1

noncomm/PMC003xxxxxx/PMC3941724.xml

Tuberculous Spondylitis after Percutaneous Vertebroplasty: Misdiagnosis or Complication?

A 58-year-old woman, who had a history of complete recovery from pulmonary tuberculosis six years previously, was hospitalized for severe back pain after a fall. Plain radiographs and magnetic resonance imaging of the thoracic spine revealed a fresh compression fracture at the T12 thoracic vertebra (). Bone densitometry indicated that the spine density was below average; the mean spinal T-score was -3.98. Physical examination demonstrated marked tenderness on palpation of the T12 region. Except for the incapacitating pain, the vital signs were stable and there was no evidence of infection. The white blood cell (WBC) count was 6,900 cells/mm3 (normal range 3,000-10,500 cells/mm3). A hematological analysis demonstrated that erythrocyte sedimentation rate (ESR) of 12 mm/hr and C-reactive protein (CRP) of 0.5 mg/dL, which were all within normal range. The pain relief was refractory to medical treatment for 2 weeks; therefore, vertebroplasty was performed and there were no intraoperative complications. Antibiotic prophylaxis was not administered before the procedure. The back pain improved dramatically, and the patient was discharged two days after the procedure. However, three weeks after the vertebroplasty, the pain gradually worsened despite administration of analgesic agents; the patient was readmitted four weeks later. Moreover, the patient reported intermittent fever with cold sweats. At the time of re-admission, the patient had a low grade fever 37.5℃. Hematological investigation revealed a mild leukocytosis with a total WBC count of 11,000 cells/mm3, elevation of the erythrocyte sedimentation rate (ESR) to 42 mm/h (normal range <30 mm/h) and a C-reactive protein (CRP) concentration of 2.05 mg/dL (normal range <0.8 mg/dL). Radiography and computed tomographic scanning revealed severe kyphosis, and that the disc space at the T11-T12 level had collapsed (). MRI also showed heterogeneous signal intensity with the spondylitis (). Initially, the patient was impressed with a pyogenic spondylitis, and percutaneous needle biopsy from the T11 and T12 vertebrae was performed. The patient was treated with antibiotics empirically before the results of the culture were available. However, the culture of the biopsy specimens was negative and did not lead to the identification of the causative micro-organism, including acid fast bacilli. However, the polymerase chain reaction (PCR) test results, specific for Mycobacterium tuberculosis, were positive for three of the four specimens (one from T11 and two from T12). Medication for tuberculosis was started with the diagnosis of tuberculous spondylitis. After three weeks of tuberculosis drug therapy, the ESR and CRP decreased to nearly the normal range (30 and 0.38, respectively). However, the patient was not able to walk independently because of persistent pain and aggravated kyphosis. The patient underwent posterior fusion and instrumentation from T9 to L2 without neural decompression (). After surgery, pain relief was achieved. The medication for tuberculosis was continued for 14 months. By 20 months after the surgery, the patient could ambulate independently without difficulty and complained only of back stiffness when walking.

[[58.0, 'year']]

F

{'8235878': 1, '16985448': 1, '11905811': 1, '30419832': 1, '11846904': 1, '12394938': 1, '10934223': 1, '15131456': 1, '10219399': 1, '15626979': 1, '31627871': 1, '15912347': 1, '12653577': 1, '16584785': 1, '26986102': 1, '29239241': 2, '34221596': 2, '15149983': 1, '15636576': 1, '24757469': 2}

{'8247726-1': 1, '5971511-1': 1}

3941726-1

noncomm/PMC003xxxxxx/PMC3941726.xml

Extraforaminal Lumbar Interbody Fusion for Cage Migration after Screw Removal: A Case Report

A 55-year-old man initially presented with disabling pain in both legs with claudication of 5 months duration. Radiological studies, including simple radiographs and magnetic resonance imaging (MRI) studies, revealed right foraminal stenosis at the L4-L5 level. Preoperative laboratory tests and bone marrow densitometry (BMD) results were all within normal ranges. He underwent L4-L5 PLIF using Stryker titanium-threaded cages (length 25mm, height 9mm) with autogenous local bone chips following posterior instrumentation. Radiographs obtained immediately after surgery showed appropriate positioning of the cages, and his presenting symptoms were much improved. At his 12-month follow-up, the leg pain and claudication symptoms had resolved, but he complained of back pain, late-onset back discomfort, and a reduced range of motion (ROM). He requested implant removal. Plain radiographs showed that cage positions had been well-maintained (). Because he was believed to have solid arthrodesis and no other pain generator, such as, adjacent segment degeneration, flat back deformity, or pseudoarthrosis, we considered that spine implant removal would relieve the back pain and discomfort, which were responsive to injections of lidocaine. Pedicular screws were removed at 12 months after PLIF (). However, one month after screw removal, the pain in both legs severely worsened in spite of aggressive analgesic administration. Plain radiographs and MRI showed that posterior migration of the inserted into the disc space (). Patient was scheduled for extraforaminal lumbar inter-body fusion (ELIF) with percutaneous screw fixation, which enables facile exposure of the exiting nerve root for cage removal without violating previous posterior musculoligamentous structures or bony stabilizers of the spine. During revision surgery, after removing both offending cages, two peek cages (length 25mm, height 9mm) were inserted into the disc space, and percutaneous screw fixation with compression was applied to the disc space to prevent further cage migration. Dramatic improvements of severe back and radiating pain were achieved immediately after revision surgery (). At his 6-month follow-up, he was symptom free and plain radiographs and MR images showed that cage positions had not changed.

[[55.0, 'year']]

M

{'19096621': 1, '26175832': 1, '16205342': 1, '11337627': 1, '28816975': 1, '33371621': 1, '26834815': 1, '8793778': 1, '1833832': 1, '9546475': 1, '22647991': 1, '9122778': 1, '11013496': 1, '24757471': 2}

{}

3941728-1

noncomm/PMC003xxxxxx/PMC3941728.xml

Spinal Extradural Arachnoid Cyst: A Case Report

A 65-year-old male patient with history of fall-down injury from a cliff 50 years ago presented progressive paraparesis from 15 years following trauma. At the time of injury, the patient had transient back pain and returned to his normal daily life as a laborer after recovering from lumbago. After 15 years from the accident, mild motor weakness of bilateral legs developed, which progressed slowly, and the patient had been unable to walk since 35 years from the time of injury. Voiding difficulty and urinary incontinence developed two years ago. On admission, the patient presented motor power of grade two at both lower extremities, and ambulation was impaired. Muscle atrophy of both lower extremities was prominent. Knee jerk and ankle jerk were absent bilaterally. The patient had no significant underlying diseases.\nWhole spine MRI revealed long segmental cystic lesion of CSF signal intensity at dorsal extramedullary space of T11 to L3 level suggesting arachnoid cyst with diffuse cord compression (). The spinal cord was displaced anteriorly and flattened. Associated findings included nerve root sleeve cyst at T2 to T3 level located leftwardly. Electromyelography revealed bilateral lumbosacral polyradiculopathy consistent with cauda equina syndrome of chronic state.\nThe arachnoid cyst was exposed by total laminectomy of T12 and extended laminotomy to L1 level. Large sized epidural arachnoid cyst was located dorsal to the dural tube. On exposure, 7×5 mm sized ovoid shaped dural defect was identified at right sided dorsolateral aspect of the dura mater between nerve root sleeves at T11 and T12 level (). The cyst contained clear fluid suggestive of CSF. Microsurgical repair of the dural defect was performed using two sutures and application of collagen fleece (Tachocomb®) and fibrin glue (Tissucol®). Following repair of the dural defect, no further leakage of CSF was verified on Valsalva maneuver in head-up position.\nHistologic examination of the cyst wall revealed pathological diagnosis as arachnoid cyst. Postoperative MRI evaluation performed at 1 month following the surgery revealed decreased size of arachnoid cyst at T11 to L3 level along with decompression of the flattened spinal cord (). The neurological status including motor power of both lower extremities was stationary on follow-up examination at 5 months postoperatively. The patient is currently on supportive rehabilitation therapy.

[[65.0, 'year']]

M

{'33163450': 2, '16238081': 1, '33627910': 1, '17608349': 1, '10763684': 1, '6699707': 1, '17330546': 1, '9120625': 1, '19516954': 1, '16572626': 1, '17542516': 1, '10419356': 1, '31000984': 1, '1506883': 1, '3612266': 1, '10505507': 1, '24757455': 2}

{'7607013-1': 1}

3941731-1

noncomm/PMC003xxxxxx/PMC3941731.xml

Postoperative Cervical Cord Compression Induced by Hydrogel Dural Sealant (DuraSeal®)

A 59-year-old man presented to the outpatient department with a 2-year history of progressive leg numbness. MRI (Magnetic resonance image) had shown intradural mass broad-based on the left side of the dura that compresses the cord severely, displacing it to the right posterior aspect at C7 level (). Opening the lamina and dura at C7 level, the mass removal was performed. The mass was totally removed and the histopathologic findings of frozen biopsy were consistent with those of a meningioma. When repairing dura, DuraSeal® was applied to dural surface that was incompletely approximated with sutures. Eight hours after surgery, the patient developed left-side weakness to motor grade I. A MRI scan was urgently performed, which demonstrated a large extradural mass which was causing severe cord compression at the level of tumor removal (). The patient underwent an emergency wound exploration. On reoperating wound, a thick layer of gelatinous material consistent with DuraSeal® was initially encountered. The layer of hydrogel compressed the dural sac severely. The hydrogel layer was excised. Also the minimal amount of surgicel was removed, which was separate and not interfering with the DuraSeal®. After surgery the patient is showing slightly improvement with his power increasing to motor grade II.

[[59.0, 'year']]

M

{'34541383': 1, '31192094': 1, '17465365': 1, '26819683': 1, '31632835': 1, '22381303': 1, '21102284': 1, '28575349': 1, '32395458': 2, '26835216': 1, '19214084': 1, '31297382': 1, '21989779': 1, '16543872': 1, '17236487': 1, '27123027': 1, '20042944': 1, '22008746': 1, '30057825': 2, '15987581': 1, '21952743': 1, '24757459': 2}

{'7192801-1': 1, '6051269-1': 1}

3941732-1

noncomm/PMC003xxxxxx/PMC3941732.xml

Arnold Chiari I Malformation with Tip-Toe Gait: A Case Report

A 12-year-old male patient presented to our institute complaining of tip-toe gait for the previous ten years (). The patient was born via normal vaginal delivery with a weight of 3.2 kg and a gestational age of 36 weeks. The developmental course had been normal. The boy underwent surgery to correct a ventricular septal defect at four years of age, and his sister suffered from an atrial septal defect. His mother reported that the boy's maternal grandfather also had a tip-toe gait. The patient had not previously experienced abnormal motor or sensory deficits, pain or discomfort, nor did he have any problems walking or running. Neither he nor any of his family members have undergone ankle evaluation.\nOn physical examination, the motor and sensory abilities of both the upper and lower limbs were normal. No pathologic reflexes were observed, and the passive dorsiflexion and plantarflexion of both ankles were 0° and 50°, respectively. These results indicate reduced ranges of motion (ROM) of ankle dorsiflexion.\nOther than those of the ankles, the ROM of all joints were within the normal limits. No muscular atrophy was observed in the upper or lower limbs, and no valgus or varus deformities were observed in either ankle. And neurologic examination about cerebellar function was performed and showed normal findings. The biceps, triceps and knee jerk responses were normal, exhibiting the double-positive reaction; however, both ankle jerks were slightly hypertonic as triple-positive reactions. The patient's heels did not contact the floor during walking.\nAt admission, the blood test and ankle radiologic X-ray results were normal, and the electrodiagnostic study including nerve conduction test, electromyography and somatosensory-evoked potential studies were all normal. Therefore we thought the cause of his tip-toe gait was less likely to peripheral nerve or orthopedic problems. So tests for determining upper motor neuron disease and magnetic resonance images (MRI) of the brain and whole spinal cord were conducted. The MRI of the whole spinal cord was normal; however, a brain MRI revealed that the cerebellar tonsil was about 7mm lower than normal. This finding suggested the diagnosis of ACM I (). To differentiate from hereditary spastic paraplegia (SPG), SPG3A and SPG4 gene studies were performed with normal results.\nThe only symptoms demonstrated by the patient were tip-toe gait, hyperreflexic ankle tone and limitation of ankle dorsiflexion. Surgery was therefore not recommended, and the patient was observed and followed-up with the Neuro-surgery Department. He also underwent applied physical therapy for his ankles, resulting in improvements.

[[12.0, 'year']]

M

{'12900124': 1, '19519678': 1, '21113370': 1, '3096099': 1, '6073187': 1, '19433279': 1, '12456921': 1, '12131745': 1, '26512271': 2, '10619285': 1, '24757457': 2}

{'4623171-1': 1}