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3930962-2

noncomm/PMC003xxxxxx/PMC3930962.xml

Cryptococcal infection in non-HIV immunosuppressed patients – Three case reports in a nephrology setting

A 56 year-old caucasian male was admitted due to fever of unknown origin. He had end stage renal disease due to a rapidly progressive IgA nephropathy treated with cyclophosphamide for 6 months (cumulative dose 21.6 g) plus prednisone. Was on hemodialysis since April 2005 (day – 2355). He also had a prior history of Polycythemia Vera diagnosed in 2001 (day – 3566), arterial hypertension, rickettsiosis diagnosed and treated on May 2010 (day – 441), and pulmonary tuberculosis infection, with clinical response to treatment.\nPatient was submitted to deceased-donor renal transplant on 1st of September 2011 (day – 0). Induction immunosuppression was performed with basiliximab, tacrolimus, MMF, and prednisolone. Post-surgery was complicated with infected retroperitoneal hematoma (treated with meropenem and vancomycin), surgical wound infection by Pseudomonas aeruginosa (treated with ciprofloxacin plus ceftazidime), and Candida albicans oropharyngitis (resolved with fluconazole).\nAllograft presented delayed graft function and later, acute vascular rejection (Banff IIA) for which ten doses of Anti-thymocyte globulin (ATG) were administered (day – 35).\nDue to persistent fever and sepsis with negative blood cultures, and failure to recover renal function, patient was submitted to transplantectomy 3 months later (day – 88). Cultured fluid collected from the abdominal drain placed in the area of the removed allograft was positive for C. neoformans. Allograft histology also revealed the presence cryptococcus. Normal opening pressure was found on lumbar puncture, with negative India-ink CSF.\nOn day 89 patient was started on liposomal amphotericin B (1.0 mg/kg per day IV) for four weeks, with complete resolution of symptoms. He progressively recovered and was discharged under fluconazole (200 mg per day, oral).

[[56.0, 'year']]

M

{'16868977': 1, '17043051': 1, '11384512': 1, '15909263': 1, '24077953': 1, '33639642': 1, '8980772': 1, '21612758': 1, '27599471': 1, '20879857': 1, '20218876': 1, '20047480': 1, '33628263': 2, '16278598': 1, '15096556': 1, '31456952': 1, '24050410': 1, '22556106': 1, '12201357': 1, '24567894': 2}

{'3930962-1': 2, '3930962-3': 2, '7892246-1': 1}

3930962-3

noncomm/PMC003xxxxxx/PMC3930962.xml

Cryptococcal infection in non-HIV immunosuppressed patients – Three case reports in a nephrology setting

An 18 year-old male with unremarkable history until July 2011 (day – 173), was diagnosed with SLE due to anemia, thrombocytopenia, malar rash, and positive anti-nuclear (ANA) and double-strand DNA antibodies (dsDNA). He was prescribed oral prednisone, but failed to comply with. On December 2011 (day – 67) presented to the emergency department with nephritic syndrome. Kidney biopsy revealed a class IV lupus nephritis. He was prescribed high dose methylprednisolone (total 1.5 g) followed by oral prednisone (1 mg/kg), along with twice monthly administrations of intravenous (IV) 500 mg of cyclophosphamide (total 3 doses). Patient was discharged on the 08th of January 2012 (day – 42).\nSix weeks later patient presented to the emergency department (day – 0) due to fever (39 °C) and diarrhea. No other symptoms were reported. Physical examination was unremarkable. Analysis revealed normal leukocyte count, C-RP 70 mg/dL. Chest X-ray revealed no suspicious images. Empirical prescription with ciprofloxacin to an infectious diarrhea was performed and was admitted to the ward. Pneumonia was diagnosed on day 2 due to hypoxemic respiratory failure and a lower right lung condensation on the X-ray. Antibiotherapy spectrum was broadened to meropenem.\nC. neoformans was isolated on blood cultures retrieved at admission (day – 6). Lumbar puncture presented an opening pressure of 26 cm/H2O. Though clear and colorless, CSF fluid was India-ink and culture positive for C. neoformans. Immunosuppression was reduced and iposomal amphotericin B (1 mg/kg per day, IV) plus flucytosine (100 mg/Kg per day, IV) was prescribed. CSF fluid collected on day 28 was sterile and India-ink negative. Patient presented good clinical outcome and was discharged on day 41 prescribed with fluconazole (200 mg per day, oral) for 12 months.

[[18.0, 'year']]

M

{'16868977': 1, '17043051': 1, '11384512': 1, '15909263': 1, '24077953': 1, '33639642': 1, '8980772': 1, '21612758': 1, '27599471': 1, '20879857': 1, '20218876': 1, '20047480': 1, '33628263': 2, '16278598': 1, '15096556': 1, '31456952': 1, '24050410': 1, '22556106': 1, '12201357': 1, '24567894': 2}

{'3930962-1': 2, '3930962-2': 2, '7892246-1': 1}

3930963-1

noncomm/PMC003xxxxxx/PMC3930963.xml

High weekly doses of liposomal amphotericin B as secondary prophylaxis after cerebral aspergillosis in a paediatric patient

In September 2010 a 6-years-old male child was diagnosed standard risk acute lymphoblastic leukaemia and treated according to the protocol BFM-ALL-2000 . In April 2011, during the intensive re-induction phase, he developed an IFI with lung and cerebral localisation. The galactomannan test yielded a value of 0.95 and a CT scan revealed several, bilateral nodular solid lesions compatible with a mould infection. A cerebral MRI showed at least two intraparenchymal lesions with concomitant ventriculitis and a biventricular obstructive hydrocephalus (A).\nThe abscess was drained and A. fumigatus isolated from the purulent material. Voriconazole was given under therapeutic drug monitoring in the blood and cerebro-spinal fluid (CSF) through an external ventricular catheter, with constant dose adjustments ().\nCaspofungin (70 mg/m2 as a first dose and subsequently 50 mg/m2/day) was also added to the treatment, as proposed by some authors , even if its CNS penetration is considered to be generally low , to provide an additional systemic protection. Caspofungin was stopped after 4 weeks and voriconazole treatment (8 mg/kg/q12 h) continued. A cerebral MRI carried out 3 months after start of antifungal treatment showed a marked reduction of the abscesses but at the same time the appearance of new possible intracranial lesions (B), which, however, regressed in the following MRI controls. With the IFI under control, the antileukemic therapy (Phase IIB) could be resumed in August 2011 under secondary prophylaxis with voriconazole at drug blood levels higher than 1.2 mg/L. In February 2012 the patient developed a voriconazole-related Steven–Johnson syndrome. The drug was thus stopped and L-AmB⁎ started at an initial dosage of 5 mg/kg/week, increased to 7.5 mg/kg/week after 7 days and further to 10 mg/kg/week for the rest of the maintenance therapy. During high dose L-AmB all intracranial lesions resolved without any evidence of side effects. The patient ended secondary prophylaxis in October 2012 after 8 months of treatment in total with high dose L-Amb and since then he is in good physical conditions, with no evident loss of cognitive functions or neurological impairment and negative repeated cerebral MRI and cerebral fluid investigations (C).

[[6.0, 'year']]

M

{'18360617': 1, '19011743': 1, '10989471': 1, '18239620': 1, '12005089': 1, '20716206': 1, '23580827': 2, '21486728': 1, '19546359': 1, '18177225': 1, '10882607': 1, '15578352': 1, '17721739': 1, '22103347': 1, '27540288': 1, '17143808': 1, '17921665': 1, '16918062': 1, '11486459': 1, '17696807': 1, '24567890': 2}

{'3618648-1': 1}

3931139-1

noncomm/PMC003xxxxxx/PMC3931139.xml

Late recovery in cerebral fat embolism

A 20-year-old male presented 7 h following a Road Traffic Accident (RTA) with closed fracture of mid-shaft tibia and fibula involving both his legs. On arrival patient was conscious and oriented with a Glasgow Coma Scale (GCS) - 15/15. He was hemodynamically stable with oxygen saturation of 100% on room air. Blood investigations were normal except for raised serum lactate (5.7 mmol/L). Urine microscopy was negative for fat globules. Fractures in the legs were splinted by above knee slab application.\nTwo hours after admission (9 h since injury), patient became drowsy with a blood pressure of 116/74 mmHg, oxygen saturation – 84% at room air and pulse rate of 138/min. Oxygen supplementation through the face mask was given. Further 4 h later patient became lethargic and comatose with the GCS falling to 8/15. Electrolyte and metabolic imbalance was ruled out. Computed tomography (CT) brain was carried out which showed no abnormality. Patient was intubated and ventilated and was on supportive treatment with neuro-monitoring. Magnetic resonance imaging (MRI) of the brain revealed multiple well defined tiny hyperintense lesions on T2 and fluid-attenuated inversion-recovery sequences with restricted diffusion bilaterally scattered in the subcortical white matter, centrum semiovale, basal ganglia, thalami and cerebellum suggestive of multiple tiny infarcts due to fat embolism []. Chest X-ray which was initially clear, revealed minimal bilateral diffuse haziness only after 3 days []. Fractures of both bones of both legs were stabilized by external fixation on 4th day after injury. Patient regained consciousness only after 2 weeks, but had poor higher mental functions such as slurred speech, poor cognition and disturbed memory. GCS had improved to 13/15 and ventilator support was gradually weaned and withdrawn at 3 weeks. Later at 4 weeks (since injury), external fixator removal and interlocking nailing was carried out for fractures of both the tibiae. At 10 weeks postinjury, patient was conscious and oriented, but with poor cognition. At 5 months, patient had regained full neurological recovery and fractures of both bones of both the legs united []. Neurological recovery was evaluated by Glasgow outcome scale (GOS) and mini-mental state examination (MMSE), which showed good recovery and normal cognition respectively. MRI of the brain repeated at 10 months postinjury revealed resolution of previously visualized hyperintense lesions except in the periventricular region without any diffusion restriction pattern [].

[[20.0, 'year']]

M

{'30128324': 1, '34282643': 2, '21477362': 1, '46957': 1, '4547466': 1, '11740000': 1, '16552005': 1, '9108767': 1, '22001712': 1, '9205570': 1, '17473148': 1, '19292198': 1, '1202204': 1, '4191134': 1, '18971877': 1, '19092320': 1, '19561953': 1, '31421672': 2, '21957425': 2, '18370341': 1, '33712051': 1, '10718245': 1, '26019433': 2, '16983300': 1, '3703294': 1, '20169788': 1, '19394136': 1, '24600071': 2}

{'3931139-2': 2, '8295961-1': 1, '6698337-1': 1, '4445211-1': 1, '3168363-1': 1, '3168363-2': 1, '3168363-3': 1}

3931139-2

noncomm/PMC003xxxxxx/PMC3931139.xml

Late recovery in cerebral fat embolism

A 28 year old male presented with RTA and sustained closed fracture of the right femur. He was admitted in the evening at 6 pm and was planned for interlocking nailing the following day. He had no other injuries and was fully conscious at admission. Next day morning at 7 am, patient was found to be drowsy and CT of the brain was carried out, which was reported to be normal. The pulse rate was 114/min, blood pressure was 122/84 mmHg with an oxygen saturation of 86% at room air. Within 3 h, consciousness of patient rapidly deteriorated with the GCS falling to 7/15 and he required intubation and ventilation to maintain oxygen saturation. Clinically, a diagnosis of fat embolism syndrome was made after ruling out head injury, hypotension, electrolyte and metabolic disturbances. The fracture was then stabilized by an external fixator on the 2nd day after injury. Chest X-ray, was initially normal and showed bilateral diffuse haziness after 2 days []. Since consciousness did not improve even after 6 days, MRI of the brain was done, which confirmed cerebral fat embolism []. As there was continued requirement of mechanical ventilation with positive end expiratory pressure, tracheostomy was done. Mechanical ventilation was continued for 5 weeks until he regained consciousness with the GCS improving to 11/15. Gradual weaning was carried out with T-piece and ventilation support was discontinued at 6 weeks. External fixator removal and interlocking nailing was carried out for fracture femur at 8 weeks since injury. Neurologically, patient had poor cognition and spasticity initially, which gradually improved and the patient returned to his original job. At 6 months, postinjury the patient had good neurological recovery (GOS) and normal cognition (MMSE).

[[28.0, 'year']]

M

{'30128324': 1, '34282643': 2, '21477362': 1, '46957': 1, '4547466': 1, '11740000': 1, '16552005': 1, '9108767': 1, '22001712': 1, '9205570': 1, '17473148': 1, '19292198': 1, '1202204': 1, '4191134': 1, '18971877': 1, '19092320': 1, '19561953': 1, '31421672': 2, '21957425': 2, '18370341': 1, '33712051': 1, '10718245': 1, '26019433': 2, '16983300': 1, '3703294': 1, '20169788': 1, '19394136': 1, '24600071': 2}

{'3931139-1': 2, '8295961-1': 1, '6698337-1': 1, '4445211-1': 1, '3168363-1': 1, '3168363-2': 1, '3168363-3': 1}

3931140-1

noncomm/PMC003xxxxxx/PMC3931140.xml

Early detection of peroneal neuropathy by ultrasound

A 12 year old boy presented with left ankle swelling and pain. He sustained left ankle sprain while playing at school. On examination, active ankle sprain dorsiflexion toe dorsiflexion and eversion were normal and there was no sensory impairment as well. Only left toe extension impairment was noted. The ultrasound was done to evaluate the soft tissue injury of the ankle. Initially, we focused on the ankle area for a sprain. When comparing the bilateral peroneus longus muscles, we found the loss of normal muscular echogenic appearance over the entire left side. Tracing this back to the origin of peroneus muscle, we found a hypoechoic cyst with a well demarcated margin neighboring the left peroneal nerve just posterior to the fibula head []. Lower limb nerve conduction and electromyography study for peroneal nerve were done. The left common peroneal nerve injury over the fibular head area was found (conduction velocity: 37 ms, normal value: More than 41 ms; peak amplitude: 1.7 mV, normal value: More than 2.0 mV in nerve conduction velocity (NCV) study; only increased polyphasic motor unit action potential was found in electromyography study). In order to more thoroughly evaluate the origin of the mass lesion and adjacent structure, a knee MRI was done. The MRI showed a 33 mm synovial cyst as originating from the proximal tibiofibular articulation with neither rupture nor regional infiltration []. A left PTFJ synovial cyst with extraneural compression over the common peroneal nerve was diagnosed.\nSurgery was performed and the synovial cyst was exposed in the PTFJ area. Compression of the common peroneal nerve by the synovial cyst at fibular head and neck was found during surgical exploration. The compressed nerve was thin by about 60% in diameter when compared to the noncompressed area. Total excision of the synovial cyst was performed and a synovial cyst with a fibrous wall was confirmed on histopathology. After 3 months of the surgery, the physical examination revealed normal muscle power of the peroneal muscle and normal gait pattern was obtained. He returned to sports postoperatively. One year later, the subsequent conduction velocity study of the common peroneal nerve revealed the parameters had returned to normal range (NCV: 46 ms; peak amplitude: 3.7 mV in NCV study; no polyphasic motor unit action potential was found in electromyography study). There was no recurrence of the cyst at 3 years of followup.

[[12.0, 'year']]

M

{'11323778': 1, '20001941': 1, '9349893': 1, '22371152': 1, '16247119': 1, '3964937': 1, '15157299': 1, '15290172': 1, '15284460': 1, '22025446': 1, '18356462': 1, '14301788': 1, '12579147': 1, '24600072': 2}

{}

3931141-1

noncomm/PMC003xxxxxx/PMC3931141.xml

Conservative management of psoas haematoma following complex lumbar surgery

A 50-year-old female without any history of bleeding disorders presented to us with a 2 year old history of severe discogenic low back pain with left-sided lower limb radiculopathy in lumbar 4, 5 and S1 nerve root. Additional symptoms included difficulty in walking up to 15-20 m due to neurological claudication. A complete spinal assessment revealed restricted spinal movements and decreased sensation over left L3. Patient was on analgesics (Tramadol, Paracetamol), antihypertensives (Lisinopril) and inhalers for asthma. There was no history of ingestion of anticoagulant medications. The preoperative blood parameters including hematology, biochemistry and clotting profiles were within normal limits. Initial radiographs of lumbar spine revealed degenerative scoliosis with convexity to right from L1 to S1 with a sideway slip at L4/5 and rotation from the L3/4 level upward in the vertebrae [Figure and ]. Magnetic resonance imaging (MRI) scans preoperatively revealed severe stenosis at L4/5 and L5/S1 [Figure and ]. After failed conservative measures for 6 months, the patient was offered surgery. He underwent dynamic stabilization using Inlign™ multiaxial pedicle screws [Disc Motion Technologies (DMT), Yarm UK] at L3 and L4 levels and a posterior lumbar interbody fusion using 11 mm cages (SABER CFRP I/F cage system, DePuy spine, Warsaw, In, USA) using a less invasive approach at L4/5 and L5/S1 levels []. There were no intraoperative complications. She received two doses of perioperative antibiotic prophylaxis (Cefuroxime) and pneumatic calf compression device as mechanical thromboprophylaxis. In the immediate postoperative period, the radicular pain symptoms settled completely and no further neurological deficiency was observed. She was mobile independently without any aids on the second postoperative day and discharged with oral analgesics.\nSubsequently, on the fourth postoperative day, she was readmitted to the hospital with symptoms of left-sided radicular pain, similar to the preoperative symptoms. On clinical examination, she was apyrexial, tender over the left iliac fossa and percussion of the left flank produced left radicular symptoms again. A detailed neurological examination was performed which revealed decreased sensation over L4 and 5 dermatomes and motor power of Medical Research Council (MRC) grade 4 on left L4 and L5 myotomes. She was started on analgesic medications such as Tramadol, Paracetamol, Diclofenac, and oral morphine. In addition, Amitryptyline and Gabapentine were started to ease the radicular pain. Blood investigations such as full blood count, urea and electrolyte, C-reactive protein (CRP), coagulation profile, blood culture and erythrocyte sedimentation rate (ESR) were done. Initial postoperative radiographs of lumbar spine did not show any obvious abnormalities. A computed tomography (CT) scan of the abdomen was performed to rule out any retroperitoneal hematoma, deep infection, or screw malplacement. CT scan revealed a collection with gas behind the left psoas muscle at the level of L4/5 vertebral body. MRI [] of the lumbar spine revealed a large collection measuring 7.6 × 4.3 × 3.3 cm adjacent to the L4–S1 vertebral bodies. In addition, there was 3 × 3.5 cm collection containing gas, which was noted posteriorly at the L4 level, displacing the thecal sac anteriorly. This collection was communicating with the extradural hematoma through the left L4 exit foramen []. In addition to this, a decision was made for further image-guided drainage of the collection for the next day. However, as the patient reported substantial improvement in her symptoms within 12 h of starting conservative measures, it was decided to wait for further improvement. Surprisingly, within 2 days of starting the conservative therapy, her symptoms settled completely and she started mobilizing independently. She was discharged within 4 days of second admission. At discharge, her CRP was 6 mg/L and WBC 13 10 cells/L). Blood cultures did not grow any organisms. At 6 months followup, she was symptom free, without any residual neurological deficit, walking up to 2 miles, and had resumed her profession as a cook.

[[50.0, 'year']]

F

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{'7661274-1': 1, '6826083-1': 1, '6826083-2': 1, '7519345-1': 1}

3931160-1

noncomm/PMC003xxxxxx/PMC3931160.xml

Neglected surgically intervened bilateral congenital dislocation of knee in an adolescent

A 12-year-old male presented with inability to walk even a few steps due to buckling of knee and striking of both knees while taking a few steps, which was present since the child learnt walking after birth. The child preferred not to walk for these reasons. His mother noticed deformity immediately after birth. She, being illiterate, could not describe the deformity. There was no history of birth trauma and the child was born of normal vaginal delivery. He was the first sibling of three brothers. The parents were apparently healthy and there was no family history of congenital, metabolic, or neurological disorders. The child developed his milestones normally. There was no evidence of any other congenital anomaly and the child was advised about the need for surgery at the age of 5 or 6 years by a village doctor. At the age of 6 years, the patient underwent surgery at the level of lower thigh, presumably bilateral supracondylar femoral osteotomy. The child continued to have inability to walk even after surgery. He consulted this institute at the age of 12 years.\nGeneral physical examination revealed stunted growth with no joint laxity. There were no other associated congenital anomalies; local examination revealed a fixed flexion deformity of 30° with further range of flexion of 15° on the right side and a fixed flexion deformity of 45° on the left side with further range of flexion of 20°. There was prominence anteriorly confounding to tibial plateau bilaterally. There was a surgical scar of around 8 cm on the lateral aspect of both thighs at the level of the upper two-third and lower one-third junction. The popliteal area was full and the contours of femoral condyles were palpable in this area. There was wasting of quadriceps muscles.\nPlain radiograph showed hypoplastic femoral condyles displaced posteriorly in relation to upper end of tibial. The upper tibial surfaces were convex. The patellae were hypoplastic, located above and anterior to femoral condyles [Figure , ]. Magnetic resonance imaging (MRI) of both knees revealed medial bowed femur, trumpet shaped metaphysis and small epiphysis, posterior subluxation of knee, splayed capsule and attenuated ACL and posterior cruciate ligament (PCL) [Figure , ].\nAn informed consent was obtained from the parents of the patient for publication of the data. The surgical correction was planned in stages. First we applied Ilizarov ring fixators in order to distract the soft tissue around knee joints to avoid surgical shortening of femur. The construct consisted of one full and one half rings in femur and two full rings in tibia with hinges at knee joint. The distraction of 1 mm/day was performed till the overlap of tibia and femur was corrected which took 3 weeks. The deformity on the right side was corrected from 30° to 25°, whereas on the left side was corrected from 45° to 30°. The surgery was performed by the senior surgeon (AKJ) []. In the second stage, open reduction of both femoral and tibial condyles was performed. Both the tibial condyles were convex. The ACL and PCL were markedly attenuated and elongated. The menisci were attenuated, but shaped like tibial condyles. The open reduction could be achieved easily and was stabilized by two cross K-wires []. The limbs were immobilized with above knee plaster-of-Paris (POP) cast for 6 weeks. The K-wires were removed at 6 weeks and knee mobilization was started. The patient started walking at 6 months postoperatively with bilateral knee ankle foot orthosis (KAFO) and axillary crutches. He continued to walk for the next 1 year. At 1 year followup, the patient had 45° fixed flexion deformity on the right side with further range of motion till 100° and 25° fixed flexion deformity on the left side with further flexion range of motion till 90°. On followup X-rays at 1 year, the convex surfaces of upper end of tibia remolded to concavity []. The patient was upright, doing his activities of daily living with brace.\nA bilateral supracondylar femoral osteotomy (extension osteotomy) was done after 18 months of previous surgery to change the arc of motion (third stage) [Figure , ]. The arc of motion was improved from 5° to 100° on the right side and from 5° to 80° on the left side. At 4 years followup visit, the patient had range of motion of 5°–100° on the right and 5°–80° on the left. More or less, the knees were found to be stable [Figures and ]. The patient was able to walk with a KAFO. His activity of daily living had improved and was able to drive bicycle. He gained in height also.

[[12.0, 'year']]

M

{'7790480': 1, '6019376': 1, '5889980': 1, '20506946': 1, '5767318': 1, '10472826': 1, '10088699': 1, '8989703': 1, '8517894': 1, '27847857': 1, '2647786': 1, '2341209': 1, '3815940': 1, '3558805': 1, '24600070': 2}

{}

3931215-1

noncomm/PMC003xxxxxx/PMC3931215.xml

Painful cervical lymphadenopathy: An unusual presentation of chikungunya

A 41-year-old male presented to the surgery outpatient department in Goa with a 2 day history of painful swellings on the left side of his neck with low-grade fever, malaise, and muscle pain of one day duration. There was no history of accompanying chills and rigor or rash. There were no ear, nose, and throat (ENT) or tooth complaints. There was no history of night sweats, loss of weight and loss of appetite. His past medical and family history was unremarkable. He had no history of tuberculosis (TB) or TB contacts. There was no recent history of foreign travel, insect bites, exposure to cats or other animals.\nOn presentation, his general condition was good, with an axillary temperature of 38°C. On physical examination, he had multiple lymph nodes located in posterior triangle of the neck on left side; the largest being approximately 2 × 1.5 cm in diameter. The lymph nodes were tender, mobile and firm in consistency with a smooth surface. He did not have any other site of significantly enlarged lymph nodes. Thyroid gland was not palpable. ENT examination was normal. Physical examination revealed no other abnormalities. A cervical ultrasound scan confirmed the examination findings and also demonstrated few subcentimetric lymph nodes in the posterior triangle of the neck on right side. Abdominal ultrasound revealed no abnormalities. He was given a course of oral amoxicillin/clavulanate combination, antipyretics, analgesics and was reviewed at the end of a week. There was no change in the symptoms or size of the lymph nodal mass. Hence, a fine-needle aspiration cytology (FNAC) was performed. But the cytological features were inconclusive.\nThe patient was referred to the medicine department with fever increasing to around 40°C. He was advised certain investigations and prescribed another course of antibiotic for 5 days. With this treatment, fever subsided but joint pains appeared after 2 days, first in the wrists and fingers followed by shoulders on both sides. The joint swelling was more at the wrist and fingers. Laboratory investigations revealed normal blood cell counts. The peripheral smear was normal without evidence of atypical lymphocytes. His erythrocyte sedimentation rate (ESR) was 18 mm/hr. Urine analysis and serum uric acid was normal. Search for malaria parasites, antinuclear and double-stranded DNA antibodies, and rheumatoid factor was negative. Serologic tests for hepatitis A, B, and C, human immunodeficiency virus (HIV) and syphilis were negative. The serology was negative for both dengue IgM and IgG antibodies (immunochromatography). The chikungunya IgM antibody was not detected by serum IgM ELISA. The purified protein derivative (PPD) test was negative. There was no evidence of focal lesion on chest radiograph.\nAfter 5 days the lower extremity joints were involved with profound swelling and limitation of movement; the swelling being more at the ankle joint []. He also experienced severe degree of myalgia in the thigh region. Patient was started on non-steroidal anti-inflammatory drugs (NSAIDs) and local application of magnesium sulfate salt. With this treatment the swelling and pain significantly decreased. But the lymph node swelling on the left side persisted and one more lymph node swelling appeared on the right side []. Thus, an excision biopsy was performed, with removal of this enlarged cervical lymph node. Histopathological examination of biopsy revealed necrotizing lymphadenitis with reactive hyperplasia. There was no evidence of histiocytosis, tuberculoid granuloma, or malignancy. A diagnosis of chikungunya was made based on history elicited by the patient and clinical findings. The arthralgia and myalgia persisted for a total period of 3 months following acute infection.

[[41.0, 'year']]

M

{'23287142': 1, '23556021': 1, '22363413': 1, '17479933': 1, '10524970': 1, '29065182': 1, '29747416': 1, '20121621': 1, '19535840': 1, '6123291': 1, '24600579': 2}

{}

3931217-1

noncomm/PMC003xxxxxx/PMC3931217.xml

Primary squamous cell carcinoma of the breast

A 72-year-old postmenopausal lady presented with a lump in the right breast. The lump was gradually progressive in size, not associated with pain, nipple discharge and retraction or dimpling of the skin. There was no family history of breast malignancy. Physical examination revealed firm, rubbery, non-tender and non-mobile lesion of 8 × 7 cm in upper outer quadrant of the right breast. Axillary lymph nodes were not enlarged. Mammogram of the right breast demonstrated a spherical mass lesion with ill-defined speculated margins. The skin and the nipple-areola complex were not involved. Lumpectomy was performed. Grossly, the tumor was 8 × 6 × 4 cm in size. The cut section showed gray white appearance with few cystic areas. On histopathology, diagnosis of SqCC with involvement of surgical margins was given. An extensive work-up ruled out other primary site or metastatic disease. Right modified radical mastectomy with axillary lymph nodes dissection was performed. Microscopically, the tumor was composed of sheets and clusters of round to polyhedral cells with pleomorphic hyperchromatic nuclei and a moderate amount of eosinophilic cytoplasm. Individual cell keratinization and keratin pearls were also seen []. The tumor cells were positive for cytokeratin and negative for estrogen and progesterone receptors. Hence diagnosis of primary SqCC of the breast was given. All 12 axillary lymph nodes were free of tumor. Overlying skin and areola was uninvolved by tumor.

[[72.0, 'year']]

F

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{'6565815-1': 1}

3931218-1

noncomm/PMC003xxxxxx/PMC3931218.xml

Rare presentation of botulism with generalized fasciculations

A 35-year-old male, presented with complaints of sudden onset pain abdomen of 8 hours duration following consumption of stale food (fried gram flour in buttermilk; Dahi Vada (Indian snack) kept inside a container since 4 days). He had one episode of vomiting; however there was no complaint of diarrhea. On 2nd day, he developed acute onset bilateral symmetrical drooping of eyelids with blurring of vision and difficulty in swallowing along with hoarseness of voice and dryness of mouth. He gradually developed progressive weakness of both upper limbs followed by lower limbs, with associated involuntary generalized twitching movements involving entire body. There were no complaints of seizures, altered sensorium, sensory symptoms, and bladder involvement. There was no history of medication intake, insect bite, or snake bite.\nOn admission, he was conscious and oriented. Vitals were stable with pulse rate of 68/min and blood pressure was 130/80 mmHg without postural fall. He was afebrile with mild tachypnea. Single breath count was 26. Higher mental functions were normal. Cranial nerve examination revealed bilateral dilated sluggishly reactive pupils, bilateral symmetrical ptosis with complete ophthalmoplegia along with decreased gag reflex. Motor system examination showed normal tone with bilateral symmetrical quadriparesis (muscle power, Medical Research Council (MRC) grade 4-/5). Deep tendon reflexes were normally elicitable with bilateral flexor plantar response. Patient also had generalized fasciculations involving anterior chest wall and bilateral upper and lower limbs []. Sensory and cerebellar system examination was normal. There were no signs of meningeal irritation.\nIn view of history of symptom onset following stale food intake with presentation of dry mouth, bilateral ptosis with ophthalmoplegia, bulbar weakness, and lower motor neuron (LMN) quadriparesis, with normal deep tendon reflexes, possibility of neuromuscular junction affection; most likely food borne botulism was kept.\nHematological evaluation revealed hemoglobin (Hb)-14.8 gm%, total leukocyte count - 10,500/mm3, platelets-1.71 lakh/mm3, and erythrocyte sedimentation rate (ESR)-25 mm/1 h. Biochemical parameters showed fasting blood sugar-74 mg/dl, urea-35 mg/dl, serum (S.) creatinine-0.9 mg/dl, S. sodium-136 meq/l, S. potassium-4.5 meq/l, S. calcium-9.2 mg/dl, S. magnesium-2.0 mg/dl, and serum creatine phosphokinase (CPK)-793 IU/l. Liver function tests were mildly deranged with serum glutamic oxaloacetic transaminase (SGOT)-95 IU and serum glutamic-pyruvic transaminase (SGPT)-66 IU. Thyroid profile was normal. Electrocardiogram, chest X-ray, and ultrasonography (USG) abdomen were normal. HIV ELISA was negative. Stool culture was sent.\nNoncontrast computerized tomography of head with thin brainstem sections was normal. Motor and sensory nerve conduction study was suggestive of pure motor axonal affection of both upper and lower limbs with normal distal latency and conduction velocities. Three hertz repetitive nerve stimulation (RNS) test was negative for decremental response. High frequency (50 Hz) RNS also did not show any incremental response. Parenteral neostigmine test was negative.\nPatient was managed conservatively keeping in view the possibility of foodborne botulism presenting as features of neuromuscular blockade. He was treated with cathartics, intravenous antibiotics, and short course of intravenous steroids (hydrocortisone). Respiratory and hemodynamic parameters were closely monitored. Patient showed gradual improvement of ptosis, ophthalmoplegia, limb weakness, and dysphagia. Respiratory effort improved and fasciculations subsided []. Repeat biochemical and hematological parameters were normal. Stool culture was sterile. He was discharged after 2 weeks.

[[35.0, 'year']]

M

{'14218929': 1, '9585323': 1, '29293927': 1, '3514662': 1, '8815754': 1, '15257512': 1, '7191633': 1, '24600582': 2}

{}

3931242-1

noncomm/PMC003xxxxxx/PMC3931242.xml

Anaplastic Carcinoma of the Pancreas: Is There a Role for Palliative Surgical Procedure?

A 65-year-old male patient presented to our hospital with complaints of abdominal pain and bilious vomiting for 4 weeks. Abdominal pain was present in the left upper quadrant, radiating to the back. Vomiting was bilious, frequent, and profuse. Patient had no history of melena, jaundice, or any significant weight loss. Physical examination revealed an ill-defined, firm mass in the left hypochondrium. Blood investigations were normal except for hypokalemia from copious vomiting. CECT scan of the abdomen showed a 6 × 6 cm heterogenous mass arising from the body and tail region of the pancreas []. It had well defined borders and loss of fat plane with relation to the DJF, suggesting infiltration by the mass []. Peri-pancreatic nodes were seen. A diagnosis of pancreatic carcinoma with nodal metastasis was made. An exploratory laparotomy revealed a large, hard, and fixed mass arising from the distal body and tail of pancreas with infiltration of the DJF. There were large metastatic nodes at superior mesenteric and celiac axis. In view of these findings, the tumor was deemed inoperable. Patient underwent a palliative DJ (third part of duodenum and jejunum) in view of symptomatic obstruction to the DJF. A biopsy was taken from the mass and sent for histological study. The patient made an uneventful recovery. Histopathology revealed pleomorphic giant cell type of AC []. Postoperative palliative chemotherapy was not offered as the patient declined. Patient was well, tolerating both fluids and pureed diet at the clinic visit, 4 months after surgery. Later, he was lost to follow up.

[[65.0, 'year']]

M

{'11436166': 1, '11410776': 1, '29147182': 1, '21160673': 1, '20542529': 1, '19111699': 1, '18176976': 1, '17993730': 1, '20459551': 1, '24600183': 2}

{}

3931243-1

noncomm/PMC003xxxxxx/PMC3931243.xml

A Rare Case Report: Carcinoma Pancreas with Hepatocellular Carcinoma

The present case report is about a 64-year-old diabetic, non-alcoholic male patient who visited our hospital with the chief complaint of anorexia, malaise and icterus since 2 months for which he was evaluated outside with routine blood investigations showing raised serum bilirubin (6.5 mg/dl) and ultrasound reporting of hypo echoic mass (28.7 mm × 33 mm) in the head of pancreas.\nThere was no remarkable family history. On admission, patient was in good general health and vital signs were within the normal limits. On physical examination, the sclera was icterus. The abdomen was soft, but tender in the right upper quadrant. Routine blood examination showed the following: White blood cell, 10,200 cells/uL; hemoglobin, 12.1 g/dl; platelet, 218,000/mm3; blood glucose, 209 mg/dl; total bilirubin, 10.2 mg/dl; direct bilirubin, 6.2 mg/dl; alkaline phosphatize, 271 IU/l; serum glutamic-pyruvic transaminase, 101 IU/l; serum glutamic-oxaloacetic transaminase, 67 IU/l; and gamma-glutamyl transpeptidase, 343 IU/l. Viral markers were negative. Tumor marker assays showed raised alpha-fetoprotein 51.99 ng/ml (normal 0-8.5), carcinoembryonic antigen was 4.2 ng/ml (normal 0-5) and carbohydrate antigen 19-9 (CA 19-9) was markedly raised 65,919 U/ml (normal 0-37).\nA triple phase contrast enhanced computed tomography (CT) scan of the abdomen revealed 3.5 cm × 3.2 cm × 4.0 cm hypo-enhancing mass in the pancreatic head with upstream extrahepatic and bilobar intrahepatic biliary radicles dilatation [], multiple small peripancreatic and mesenteric nodes. Liver showed multiple nodular lesions (metastasis) of varying sizes with differential characteristics [], one of the large relatively defined mass in segment 7 of the liver is showing subtle arterial phase enhancement [] and washout in portal venous phase [] likely HCC. Upper gastrointestinal endoscopy was normal.\nThus, the provision diagnosis was carcinoma head of pancreas with multiple liver metastases synchronous with HCC? Ultrasonography guided fine-needle aspiration was taken from pancreatic mass, non-enhancing and enhancing liver SOL for confirmation.\nSmears from the enhancing liver lesion was hypercellular, comprise thickened trabeculae and groups lined by atypical hepatocytes with transgressing blood vessels and partial entrapment by endothelial cells at the periphery. There were few stripped nuclei seen in the background. The atypical hepatocytes show enlarged, hyperchromatic, markedly pleomorphic nuclei with irregular distribution of nuclear chromatin, prominent eosinophilic nucleoli, focal nuclear vacuolization and abundant granular eosinophilic cytoplasm with cholestasis [Figure –]. On immunohistochemistry-Tumor cells was positive for glypican 3 and hepatocyte specific antigen []. Histochemistry-trabeculare are reticulin poor. Above features are diagnostic of HCC Smears from the non-enhancing Liver lesion showed few small groups and acini of atypical epithelial cells, admixed with sheets and clusters of benign hepatocytes. These atypical epithelial cells show enlarged, hyperchromatic, markedly pleomorphic nuclei with high N:C ratio, irregular nuclear membrane, coarse chromatin and scanty cytoplasm suggestive of metastatic adenocarcinoma []. Smears from the pancreatic lesion showed few small groups and acini of atypical epithelial cells. These atypical epithelial cells show enlarged, hyperchromatic, markedly pleomorphic nuclei with high N:C ratio, irregular nuclear membrane, coarse chromatin and a fair amount of vacuolated cytoplasm suggestive of adenocarcinoma [].\nPatient was diagnosed as carcinoma pancreas with multiple liver metastases synchronous with solitary HCC. The conclusive stage of the pancreatic cancer was TNM stage IV {T2, N1, M1} whereas that of the HCC was Barcelona Clinic Liver Cancer (BCLC) stage A and TNM stage I {T2, N0, M0} according to Union for International Cancer Control classification. Percutaneous transhepatic biliary drainage followed by metallic stenting was done and planned for single agent gemcitabine based palliative chemotherapy once total serum bilirubin came down below 3 mg/dl.

[[64.0, 'year']]

M

{'12115351': 1, '12527496': 1, '10948656': 1, '12576929': 1, '11408798': 1, '16937043': 1, '15499648': 1, '10951336': 1, '28959430': 2, '7182564': 1, '14642630': 1, '10573522': 1, '10955735': 1, '16514137': 1, '14612894': 1, '24600184': 2}

{'5610581-1': 1}

3931244-1

noncomm/PMC003xxxxxx/PMC3931244.xml

Penile Metastasis Secondary to Bladder Cancer: A Report of Two Cases

An 85-year-old male, smoker presented with painless gross hematuria for last 1 year. His routine blood investigations and chest X-ray were within normal limit. Contrast-enhanced computed tomography (CECT) scan showed polypoidal mass in the right posterolateral wall of urinary bladder with bilateral pelvic lymphadenopathy. His metastatic work up (including chest X-ray, CECT chest, abdomen and pelvis, and bone scan) was normal. Patient underwent transurethral resection of bladder tumor and postoperative histopathology revealed a high grade papillary urothelial carcinoma infiltrating the lamina propria and deep muscle. In view of old age and poor general condition of the patient, he was not taken up for radical cystectomy or concurrent chemoradiation. He was planned for radical radiotherapy alone 50 Gy in 25 fractions over 5 weeks with conventional four field technique in 6 MV linear accelerator. After nine fractions, of radiation patient complained of swelling and pain in his penis. Clinical examination revealed a 3 × 2 cm indurated swelling in the shaft of the penis. Magnetic resonance imaging (MRI) showed an ill-defined area of altered signal intensity appearing isointense to muscle on T1 and of intermediate signal intensity on T2-weighed images is seen involving bilateral corpora cavernosa with probable extension to the corpus spongiosum as well []. Fine needle aspiration cytology (FNAC) of the penile swelling revealed metastatic transitional cell carcinoma []. After nine fractions, radiotherapy plan was revised and radiation field was enlarged along with special wax bolus to accommodate the penile lesion. He completed radical radiation uneventfully and was kept on follow-up. No chemotherapy was offered because of old age and poor general condition of the patient. After 2 months of follow-up, he underwent MRI pelvis and both the bladder as well as penile lesion showed partial response. Patient is relieved from intractable pain and is leading a good quality of life.

[[85.0, 'year']]

M

{'2767101': 1, '13330017': 1, '20075023': 1, '15575962': 2, '12837452': 1, '17032461': 2, '26818689': 1, '24600185': 2}

{'3931244-2': 2, '539302-1': 1, '1618838-1': 1}

3931244-2

noncomm/PMC003xxxxxx/PMC3931244.xml

Penile Metastasis Secondary to Bladder Cancer: A Report of Two Cases

A 58-year-old male presented with hematuria for 1 month. He smoked one pack of cigarettes per day for last 30 years. He was thoroughly investigated and CECT scan revealed a soft tissue density mass in posterior wall of urinary bladder. He underwent transurethral resection of bladder tumor and histopathology showed high grade urothelial carcinoma with involvement of deep muscle. He received four cycles of neoadjuvant chemotherapy based on injection cisplatin 70 mg/m2 intravenous day 1 and injection gemcitabine 1.2 g/m2 day 1 and day 15. After four cycles of neoadjuvant chemotherapy, he underwent radical cystectomy with pelvic lymphadenectomy and ileal conduit diversion. Patient was kept on follow-up and after 4 months of surgery patient complained of painful swelling on glans penis. Clinical examination revealed indurated swelling at base of penis and another small nodule present at the shaft of the penis. MRI pelvis showed evidence of well-defined heterogeneous soft tissue mass of size 5 × 2.8 × 2 cm seen in left crura of the penis near its base. It was hyperintense on T2W1 and isointense on T1W1and it showed thick irregular peripheral enhancement []. Similar lobulated soft tissue lesion of size 1.8 × 1.9 × 1 cm was seen just medial to right puborectalis muscle. FNAC of penile lesion revealed metastatic carcinoma []. Positron emission tomography scan showed intensely fluorodeoxyglucose (FDG) avid Standardized uptake value (SUV) maximum 17.6) heterogeneously enhancing soft tissue mass seen in the proximal two-third of the shaft of the penis extending to left crura with intensely FDG avid well-defined nodules of variable sizes were noted in bilateral lung fields [Figures and ]. He received palliative external beam radiotherapy to pelvis 20 Gy in five fractions delivered over a period of 1 week and he was registered in our palliative clinic. He did not receive any further chemotherapy in view of poor general condition and was expired within 2 months.

[[58.0, 'year']]

M

{'2767101': 1, '13330017': 1, '20075023': 1, '15575962': 2, '12837452': 1, '17032461': 2, '26818689': 1, '24600185': 2}

{'3931244-1': 2, '539302-1': 1, '1618838-1': 1}

3931632-1

noncomm/PMC003xxxxxx/PMC3931632.xml

Axitinib for preoperative downstaging of renal cell carcinoma with sarcomatoid differentiation and direct invasion of the duodenum and inferior vena cava: a case report

In November 2012, a 73-year-old man presented with a right renal mass and the chief complaints of anorexia, fatigue, and melena. Laboratory tests revealed anemia (hemoglobin, 6.2 g/dL) and hypercalcemia (corrected calcium, 11 mg/dL). The Karnofsky performance status was 50%–60%. Computed tomography (CT) showed a large right renal tumor with a diameter of 13 cm that had invaded the second part of the duodenum and had involved multiple regional lymph nodes without distant metastasis ().\nPositron emission tomography (PET) scans showed accumulation of fluorine-18-deoxyglucose in the tumor, with the maximum standardized uptake value being >40 (). Esophagogastroduodenoscopy detected direct invasion of the tumor into the second part of the duodenum with bleeding (). Biopsy of the protruding tumor revealed histologic features that were highly suggestive of sarcoma (). Accordingly, the clinical diagnosis was right RCC with sarcomatoid differentiation (cT4N1M0). It seemed to be difficult to safely perform radical nephrectomy, cavotomy with thrombectomy, and pancreatoduodenectomy. In comparison with the first-generation vascular endothelial growth factor receptor (VEGFR) tyrosine kinase inhibitors (TKIs) like sorafenib and sunitinib, axitinib is a more potent and selective second-generation VEGFR-TKI with better safety and tolerability, which has a higher affinity for the tyrosine kinase of VEGFR and a stronger inhibitory effect on its kinase activity with fewer serious adverse events, such as thrombocytopenia. At present, axitinib is generally recommended as a second-line therapy for advanced RCC, because the phase III axitinib versus sorafenib in advanced RCC (AXIS) trial demonstrated that axitinib treatment achieved longer progression-free survival than sorafenib in patients with metastatic RCC after failure of approved first-line systemic therapy, including sunitinib and cytokines.\nOur patient was unable to take food orally and had become emaciated. His classification was poor risk, according to the Memorial Sloan-Kettering Cancer Center (MSKCC) criteria. To decrease the tumor burden and improve the feasibility of surgery, we selected preoperative treatment with axitinib. If the patient showed a poor response to axitinib, we intended to perform palliative gastrojejunostomy to allow oral intake.\nThis study was conducted in accordance with the World Medical Association Inc’s Declaration of Helsinki and was approved by the Institutional Review Board of the Ethical Committee of Dokkyo Medical University Hospital. In addition, the patient signed a consent form that was approved by our institutional Committee on Human Rights in Research. Axitinib administration (10 mg/day) was started in December 2012; it was scheduled to continue for 4–6 weeks until the patient underwent radical surgery, comprising nephrectomy, cavotomy with thrombectomy, and pancreatoduodenectomy.\nDuring axitinib treatment, there were no adverse events > grade 3. After 2 weeks, the dose of axitinib was increased to 14 mg/day; then, it was raised to 20 mg/day after 2 more weeks. Attenuation of the tumor contrast uptake and enlargement of the duodenal lumen were observed on CT after 5 weeks of administration ().\nAfter 6 weeks of treatment, we decided to perform definitive surgery, and successfully carried out radical nephrectomy, cavotomy with thrombectomy, and pancreatoduodenectomy. The resected tumor was an invasive whitish and yellowish mass with partial necrosis. Pathological examination confirmed that the lesion was RCC with sarcomatoid differentiation with necrotic areas (pT4pN1M0, Fuhrman grade 4) (). The pathological treatment effect of axitinib was grade 2 (two-thirds necrosis). The patient has been doing well since the operation. Western blotting and immunohistochemistry of the resected specimen was performed to detect phosphorylated Akt (Ser-473), using a rabbit antihuman antibody (PhosphoPlus® Akt (Ser-473) Antibody Kit; #9270, Cell Signaling Technology, Inc., Danvers, MA, USA), according to the method described previously.,\nThese studies showed that expression of phosphorylated Akt (Ser-473) in the tumor was heterogeneous ( and ). Axitinib therapy (10 mg/day) was resumed because microscopic invasion was detected at the resection margin, and some of the tumor tissues showed viability and expression of phosphorylated Akt (Ser-473). The patient has been asymptomatic without apparent disease progression for 6 months after resuming treatment with axitinib.

[[73.0, 'year']]

M

{'17215529': 1, '22234634': 1, '22787405': 1, '28752352': 1, '27822939': 1, '26983443': 2, '18692951': 1, '22640774': 1, '22698625': 1, '22056247': 1, '12544348': 1, '23692504': 1, '25784113': 1, '14767273': 1, '19862815': 1, '21353796': 1, '24267160': 2, '19064974': 1, '21127411': 1, '24206640': 1, '21350947': 1, '24627641': 2}

{'4794818-1': 1, '4222084-1': 1}

3931697-1

noncomm/PMC003xxxxxx/PMC3931697.xml

Two cases of mild serotonin toxicity via 5-hydroxytryptamine 1A receptor stimulation

A 64-year-old woman who experienced auditory hallucinations and insomnia, was treated with hypnotic benzodiazepines and perospirone (8 mg/day; ). Perospirone had been administered for 4 months. She had developed chronic renal failure, which had continued for several years, with high serum levels of creatinine and blood urea nitrogen (). She also experienced depressed mood, diminished interest, and anhedonia; therefore, she was treated with paroxetine (10 mg/day). Eighteen hours after paroxetine was added to her ongoing perospirone treatment, she exhibited finger tremors, sweating, coarse shivering, hyperactive knee jerks, vomiting, diarrhea, tachycardia, and psychomotor agitation. After administrating paroxetine with perospirone twice, both paroxetine and perospirone were discontinued. Her symptoms gradually disappeared over a period of approximately 1 week.

[[64.0, 'year']]

F

{'15784664': 1, '19149529': 1, '7674802': 1, '17645916': 1, '8577805': 1, '17439416': 1, '18707769': 1, '20880320': 1, '20571976': 1, '21486038': 1, '8935801': 1, '11164765': 1, '7485652': 1, '15602109': 1, '12925718': 1, '30886699': 2, '2942947': 1, '16581169': 1, '17935833': 1, '21228393': 1, '30845376': 1, '17874986': 1, '10928399': 1, '11822997': 1, '16554526': 1, '15341484': 1, '24627634': 2}

{'3931697-2': 2, '6413434-1': 1}

3931697-2

noncomm/PMC003xxxxxx/PMC3931697.xml

Two cases of mild serotonin toxicity via 5-hydroxytryptamine 1A receptor stimulation

An 81-year-old woman who experienced persecutory delusions with mild dementia, was treated with neuroleptics (). She was admitted to a psychiatric unit, owing to increased hostility and aggression related to her delusions. The results of brain imaging tests revealed mild brain atrophy in the frontotemporal and hippocampal regions with no cerebrovascular lesions. After successful treatment of her paranoid state, the patient developed depressive symptoms, which were treated with 10 mg/day of paroxetine. The final neuroleptic dose was 8 mg/day of perospirone that had been administered for 3 weeks. Fourteen hours after the addition of paroxetine to the patient’s ongoing perospirone treatment, she exhibited tachycardia, finger tremors, prominent anxiety and agitation, and hyperactive knee jerks. The symptoms continued for days (ie, for as long as perospirone and paroxetine were coadministered), and they disappeared 3 days after the discontinuation of both drugs.

[[81.0, 'year']]

F

{'15784664': 1, '19149529': 1, '7674802': 1, '17645916': 1, '8577805': 1, '17439416': 1, '18707769': 1, '20880320': 1, '20571976': 1, '21486038': 1, '8935801': 1, '11164765': 1, '7485652': 1, '15602109': 1, '12925718': 1, '30886699': 2, '2942947': 1, '16581169': 1, '17935833': 1, '21228393': 1, '30845376': 1, '17874986': 1, '10928399': 1, '11822997': 1, '16554526': 1, '15341484': 1, '24627634': 2}

{'3931697-1': 2, '6413434-1': 1}

3932351-1

noncomm/PMC003xxxxxx/PMC3932351.xml

Petrositis With Bilateral Abducens Nerve Palsies complicated by Acute Otitis Media

A 8-year-old girl was admitted to our pediatric department with a 3-week history of headache and diplopia. She had visited a local clinic due to otorrhea of left side and taken medication under the diagnosis of left AOM. After a few days, otorrhea disappeared but headache and diplopia were worsened. Her past medical history was significant only for left convergent strabismus. At admission, physical examination showed restricted lateral gaze of both eyeballs and diplopia at central gaze, secondary to clearly apparent both abducens nerve palsies (). Facial motor examination was normal. The left tympanic membrane was amber and right tympanic membrane was intact. There was no fever, although she claimed that she had high fever up to 38.9℃ before she took medication at the local clinic. Her headache was located at left temporal area at the initial stage but migrated to frontal and then to whole head. The ophthalmologic examination showed bilateral optic disc swelling (). Laboratory tests showed a white blood cell count of 6, 290 mm3 with 67% neutrophils, and the C-reactive protein was 0.3 mg/L, and erythrocyte sedimentation rate (ESR) 6 mm/hour. Under the impression of meningitis at the pediatric department she took brain magnetic resonance imaging (MRI), spinal tapping, temporal bone computed tomography (CT), and bone scan. Her brain MRI showed a diffuse inflammatory process in the left mastoid air cells with invasion of the left petrous bone (). Temporal bone CT showed soft tissue densities at left mastoid, middle ear, and well-developed petrous apex cells (). The cerebrospinal fluid examination was normal, and bone scan showed increased uptake of the left mastoid and petrous bones. Diagnosis of left petrositis and bilateral abducens nerve palsies was made, and intravenous antibiotic treatment with ceftriaxone (1 g/day), clindamycin (300 mg/day) was initiated. After 9 days of intravenous antibiotics, consultation and transfer to otolaryngology department was done due to continuous headache and diplopia. Pure tone audiometry was done and showed air-bone gap of 10 dB on left ear. On physical examination, restricted lateral gaze on both sides due to abducens nerve palsies of both side was noted. Left tympanic membrane was amber and ventillation tube insertion was performed and serous profuse discharge in the middle ear was sent for culture. No bacteria was identified. Intravenous antibiotics (ceftriaxone, clindamycin) were continuously administered and topical ear drops were applied. On the 16th hospital day, diplopia was improved and the field of diplopic vision was shortened from 10 cm to 2 cm length (), but intermittent spiking fever up to 38.3℃ occurred, and intravenous amikacin was added. On the 19th hospital day, the fever continued and follow-up temporal bone CT with contrast enhance, H1N1 PCR for H1N1 flu which was common at the time, general laboratory studies and pediatric consultation was done. On temporal bone CT with contrast enhancement, remaining soft tissue densities at mastoid and petrous area with improving petrositis was noted (). And with negative H1N1 PCR and high AST/ALT of 617/365 IU/L, under the diagnosis of drug fever, she was transfered to the pediatric department, on the 21th hospital day when diplopia disappeared. Antibiotic was changed to intravenous vancomycin, and fever subsided with AST/ALT of 40/78 IU/L on the 30th hospital day. The strabismus returned to preadmission state, and the patient was finally discharged (). After two weeks at the out-patient department, follow-up temporal bone CT was taken and disappearance of opacification and improved aeration at left petrous apex area were noted (). There was also improvement of bilateral papilledema six weeks later of admission ().

[[8.0, 'year']]

F

{'9807067': 1, '12791460': 1, '933655': 1, '7441445': 1, '33194201': 2, '18472169': 1, '19990585': 1, '11165646': 1, '6660745': 1, '28589111': 2, '15942518': 1, '6876244': 1, '10527283': 1, '12359616': 1, '15949093': 1, '34214262': 1, '24587883': 2}

{'7594227-1': 1, '5448032-1': 1}

3932352-1

noncomm/PMC003xxxxxx/PMC3932352.xml

Aggravation of Relapsing Polychondritis due to the Infection and Its Manifestation on a Nasal Tip Graft

A 48-year-old woman initially presented in September 2009 with complaints of painful swelling and erythematous changes in the upper and lower lips, both ear auricles and the nasal tip for the previous three days (). She had undergone a filler injection procedure for cosmetic reasons on the both lips at an illegal cosmetic hair shop (not by a medical doctor) seven days previously. The cause of lip infection may be the filler materials which may be not sterilized and not qualified on Korea Food and Drug Administration. There were no other symptoms like fever, chills, arthralgias, dyspnea or voice changes and no specific lab findings (). She had a medical history of relapsing polychondritis and a surgical history of undergoing a surgical operation that was performed by a plastic surgeon for the tip of her nose. The nose-tip surgery had been done by using the right conchal cartilage for augmentation. After she underwent the operation, the nasal tip turned red whenever polychondritis symptoms-color changes of both ear auricles and arthralgia-developed. This case shows that grafted cartilage can also be affected like other cartilages. The symptoms improved a few days after she received methotraxate (2.5 mg, twice a day) and methylpredisolone (8 mg, twice a day) (). We used the medication for seven days. Four weeks after discharge, the swelling and erythematous changes of the nasal tip and the other cartilage resolved.

[[48.0, 'year']]

F

{'19144159': 1, '15529362': 1, '775252': 1, '714080': 1, '17437170': 1, '4223687': 1, '24587884': 2}

{}

3932353-1

noncomm/PMC003xxxxxx/PMC3932353.xml

Palatal Implants for Persistent Snoring and Mild Obstructive Sleep Apnea After Laser-Assisted Uvulopalatoplasty

A 56-year-old man visited our hospital with a 15-year history of persistent snoring and witnessed apnea after LAUP. His subjective snoring level was six on a seven-point Liker scale ranging from 0 (none of the time) to 6 (all of the time). He had no relevant medical history, including hypertension, diabetes mellitus, or stroke. Body mass index (BMI) was 24.8 kg/m2 (height was 162 cm and weight was 65 kg).\nOn direct examination of the oral cavity and oropharynx, the uvula was not observed because of the previous surgical procedure (). Palatine tonsils were small (hidden within the pillars), and the Friedman tongue position was presumed as grade 2 []. The soft palate length was longer than 2.5 cm, and nasopharyngeal stenosis was not found. Retropalatal obstruction was suspected based on physical examination and the Muller maneuver.\nHe was diagnosed with mild OSA based on diagnostic standard (overnight, attended, laboratory-based) polysomnographic results (AHI=7.6 events/hour [supine AHI=13.7 events/hour and non-supine AHI=0.6 events/hour], apnea index [AI]=0.8 events/hour, minimum SaO2=91%, arousal index=23.8 events/hour, and snoring=51.3%).\nContinuous positive airway pressure (CPAP) or oral appliance (OA) was recommended as a primary therapy. However, he was unwilling to accept CPAP or OA as a permanent solution and chose to undergo an alternative minimally invasive surgical treatment.\nA palatal stiffening procedure using the Pillar palatal implant procedure was performed under local anesthesia according to the following simple steps: (1) determine three palatal implant insertion sites and inject with local anesthetic agent on surgical fields; (2) insert the needle of the delivery system through the sub-mucosal tissue layer into the muscle and place the implants in the muscular layer of the palate (three implants were located at about 2 mm intervals); (3) withdraw the needle and inspect the palatal mucosal surface (oral and nasopharyngeal) to ensure the implants were properly placed. There were no specific perioperative complications except mild discomfort at the operative site.\nTwo months after the Pillar procedure, subjective snoring level decreased from 6 to 2 and follow-up standard polysomnographic results (AHI=1.3 events/hour [supine AHI=2.3 events/hour and non-supine AHI=0.3 events/hour], AI=0.0 events/hour, minimum SaO2=90%, arousal index=18.9 events/hour, and snoring=16.8%) were clearly improved compared to those of the preoperative sleep test (). BMI was 23.6 kg/m2 (body weight was 62 kg) at two months following the procedure. Mild discomfort at the operative site resolved without morbidity, and postoperative complications such as extrusion or infection were not observed.

[[56.0, 'year']]

M

{'7897766': 1, '12502473': 1, '21061864': 1, '11122588': 1, '2128762': 1, '18030486': 1, '8028940': 1, '19784401': 1, '17075397': 1, '12161725': 1, '1921639': 1, '24587885': 2}

{}

3932354-1

noncomm/PMC003xxxxxx/PMC3932354.xml

Coexistence of a Carotid Body Tumor and Thyroglossal Duct Carcinoma With Cervical Lymph Node Metastasis

A 78-year-old woman visited our clinic, presenting with a 3-year history of a midline neck mass in the region of the hyoid bone and bilateral cervical asymptomatic neck masses. There were no constitutional symptoms, including fever, night sweating, fatigue, dysphagia, dyspnea, and weight loss. She reported that the mass was progressively increasing in size. The rest of the history was unremarkable. Physical examination showed a 3×3-cm rubbery, non-tender, semi-fixed midline mass and several enlarged lymph nodes at level II and III bilaterally. There were no pulsations or bruits on auscultation of the lateral neck masses. The results of routine laboratory tests, including blood chemistry, complete blood cell count, coagulation study, serologic testing, and urine analysis, were unremarkable. Electrocardiogram and chest X-ray were within normal limits.\nComputed tomography (CT) with contrast showed an irregularly marginated, round, heterogeneously enhancing mass at the level of the hyoid bone and multiple enlarged lymph nodes bilaterally at level II to III (). Interestingly, the left-side neck mass was located at the carotid bifurcation, splaying the carotid bifurcation on a CT scan (). However, the CT scan did not indicate other instances of abnormal lymphadenopathy in the left neck. Furthermore, neck CT and sonography did not demonstrate any mass involving the thyroid (). Fine needle aspiration biopsy (FNAB) was done on the midline mass and right-side neck mass. The results of FNAB suggested the possibility of papillary carcinoma and lymph node metastasis from papillary carcinoma, respectively. From the above results, we made the tentative diagnosis of TGDC carcinoma with bilateral neck metastasis. We planned to resect the masses, including bilateral neck dissection, in accordance with TGDC with neck metastasis. Splaying of the carotid bifurcation, however, is usually indicative of a CBT. Through the head and neck tumor board, we decided that carotid arteriography should be preoperatively performed to differentiate a CBT from lymph node metastasis. The mass was confirmed as a highly vascular mass in the bifurcation of the carotid artery that was compressing the internal and external carotid arteries (). Ascending pharyngeal artery represented the major blood supply for this tumor. Our patient did not undergo carotid occlusion testing and embolization because of the potential complications. Results of screening for metanephrine and vanillymandelic acid in 24-hour urine samples were negative.\nSubsequently, the patient underwent wide excision of the midline neck mass, including resection of the hyoid bone, total thyroidectomy, central neck dissection and right-side selective neck dissection (levels II-V), involving resection of the sternocleidomastoid muscle, spinal accessory nerve, and internal jugular vein. Although there was mild adhesion between the midline mass and surrounding soft tissue, we obtained a generous resection margin (). During the exploration of left-side neck, we confirmed the CBT between external and internal carotid artery (). We did not try to remove the left-side neck mass (CBT) because it had a tendency to bleed when touched and the patient was elderly.\nHistological analysis revealed that a thyroglossal duct cyst was lined by a single row of columnar epithelium and the tumor was composed of small and large papillary structures with fibrovascular cores (). In addition, the tumor cells lining the papillary structures of the midline mass and cervical lymph nodes showed nuclear grooves and nuclear clearing, which are characteristic nuclear features of papillary carcinoma (). The final histopathologic diagnosis was consistent with TGDC papillary carcinoma with perithyroidal soft tissue extension and subsequent multiple lymph node metastasis (17 out of 55 lymph nodes). However, we could not demonstrate any evidence of papillary carcinoma in the thyroid gland.\nThe patient refused adjuvant therapy with 131I after surgery and was only treated with levothyroxine supplementation. At the 2-year follow-up, the patient remained free of disease.

[[78.0, 'year']]

F

{'11894786': 1, '15186613': 1, '1267333': 1, '7137085': 1, '4302028': 1, '15068626': 1, '11548172': 1, '837327': 1, '14495345': 1, '16538412': 1, '1998873': 1, '8060034': 1, '8544589': 1, '9704182': 1, '11003294': 1, '28293858': 1, '24587886': 2}

{}

3932356-1

noncomm/PMC003xxxxxx/PMC3932356.xml

Atypical Fibroxanthoma in Head and Neck

Our patient was a 70-year-old male who 3 months ago underwent an incision biopsy at another hospital for a 1.0×1.0 cm sized palpable and protruding reddish nodule in the left infra-auricular area. By result of a histological test, a malignant tumor was suspected and he was transferred to our hospital. In the physical examination, there was a round scar remaining from the previously performed biopsy and the size of the resected tissue was approximately 0.7×0.7 cm. Any other special findings were not detected. The specimen acquired at the other hospital was examined again at our hospital. In our histopathologic finding, we detected a numerously mitotic figure with atypical, bizarre, large and hyperchromatic nuclei (). After immunostaining, we had CD34, CD45, cytokeratin and desmin showing negative reaction with a positive reaction for Vimentin as well as CD68. Finally, we diagnosed it as an atypical fibroxanthoma [,]. In addition, the malignancy was confirmed in the previous incision margin, and there was no special findings in a metastatic work-up. Therefore, a wide excision was performed with 2 cm safety resection margin around the lesion which underwent the initial incision biopsy, and there was no malignancy detected by frozen biopsy (). The defect was then closed by simple suture (). Twelve months have passed after surgery and the patient is under the follow-up observation at our outpatient clinic without any local recurrence.

[[70.0, 'year']]

M

{'11260542': 1, '12820468': 1, '15798451': 1, '15606841': 1, '11406660': 1, '16972949': 1, '16197421': 1, '9107284': 1, '16965335': 1, '26618136': 1, '25392128': 1, '24587887': 2}

{}

3932357-1

noncomm/PMC003xxxxxx/PMC3932357.xml

A Hypopharyngeal Ductal Cyst Masquerading as a Laryngopharyngeal Reflux Disease

A 64-year-old man presented with a four-year history of globus sensation. He was referred for intractable symptoms despite having taken reflux medication for two months. Despite continued medication, his symptoms developed to dysphagia, even when swallowing liquid. He denied hoarseness or dyspnea. His medical history included a transurethral resection for bladder cancer 3 years previously. He had experienced weight loss of 3 kg over the previous 2 months. On his first presentation, flexible fiberoptic laryngoscopy showed pooling saliva, but no mass-like lesion in the hypopharynx. Vocal fold mobility was intact. The patient underwent a double-contrast pharyngoesophagogram due to persistent symptoms. An ovoid submucosal mass with a smooth contour in the left pyriform sinus was revealed (). Subsequently, computed tomography (CT) scans showed a 3×4 cm mass with central low attenuation in the left hypopharynx (). With the patient under general anesthesia, the lesion was removed via direct laryngoscopy using a microdissection electrode for endoscopic electrosurgery. The mass originated from the lateral wall of the pyriform sinus. It appeared as a pedunculated mass with a smooth, well-encapsulated surface. It was removed by means of submucosal dissection with the microelectrode. The resected specimen consisted of a 2×3×4 cm cystic mass with proteinaceous fluid (). Histologic examination showed a true epithelial-lined cyst consisting of cuboidal epithelium (). The final diagnosis, made by a pathologist, was a ductal cyst of the hypopharynx. Postoperative recovery of the patient was uneventful. The patient was free of symptoms after one month. Five years of follow-up showed no tumor recurrence and normal swallowing.

[[64.0, 'year']]

M

{'29110221': 1, '16719953': 1, '5411821': 1, '17146403': 1, '8522439': 1, '30927081': 1, '15728599': 1, '7980193': 1, '2722499': 1, '24587888': 2}

{}

3932379-1

noncomm/PMC003xxxxxx/PMC3932379.xml

Hepatoduodenal fistula formation following transcatheter arterial chemoembolization and radiotherapy for hepatocellular carcinoma: treatment with endoscopic Histoacryl injection

A 71-year-old male patient was readmitted to our hospital 1 month after discharge because of relapse of abdominal pain. One year prior, he had been diagnosed with HCC associated with chronic hepatitis B. He could not undergo surgical resection because of invasion of the hepatic and main portal veins. Therefore, he underwent TACE as a palliative treatment. Although he underwent five sessions of TACE over 10 months, newly viable HCC was found in the left lobe of the liver with thromboses of the portal vein, hepatic vein, and inferior vena cava. Palliative RT was administered to the HCC in the left lobe of the liver and area of left portal vein thrombosis using a dose of 33 Gy comprising daily doses of 3 Gy. He then underwent two additional TACE sessions. One month after the seventh TACE session, the patient presented to our institution with a complaint of persistent abdominal pain. He also had intermittent fever, chills, and weight loss. Abdominal computed tomography (CT) showed a gas-containing cavity suggestive of an abscess within a necrotic mass in the left lateral segment of the liver (). He was hospitalized, received intravenous empirical antibiotics, and underwent ultrasound-guided abscess aspiration, which produced 0.5 mL of turbid yellowish fluid with a foul odor. Percutaneous drainage catheter insertion was difficult because of the small size of the abscess pocket. Prevotella oralis and Clostridium bifermentans/sporogenes were isolated from the aspirated specimen. The patient's symptoms, including abdominal pain, fever, and chills, resolved completely after 2 weeks of intravenous antibiotic therapy, and he was discharged with oral antibiotics. Although he continued to take oral antibiotics, his abdominal pain recurred only 2 weeks after discharge, and the frequency of abdominal pain gradually increased. He was readmitted to our hospital 1 month after discharge. He complained of persistent abdominal pain; however, other generalized symptoms, including fever and chills, did not reappear. On physical examination, right upper quadrant tenderness was observed, and his serum C-reactive protein level was higher than that during the previous hospitalization. Follow-up CT was performed. Although the abscess had decreased in size, we detected a new communication between the remaining abscess and the duodenal bulb (). He received total parenteral nutrition and broad-spectrum antibiotics. Gastroduodenoscopy was performed to confirm the fistula through a 0.3-cm hole in the proximal portion of the duodenal bulb (). At that time, endoscopic clipping was attempted to close the fistula opening. Follow-up endoscopy was performed 2 days after clipping; however, there was still a fistula opening beside the clips (). The next approach to the fistula was by endoscopic retrograde cholangiopancreatography (ERCP). A ~1-cm fistulous tract was confirmed by guidewire insertion from the fistula hole of the duodenal lumen. A 1:3 mixture of Histoacryl and lipiodol solution (0.5 mL) was injected into the fistula. Follow-up endoscopy performed 2 days after this intervention revealed that the previous fistula orifice was filled with Histoacryl (). The patient was then discharged from the hospital because he had no abdominal pain after starting oral intake. One month after the intervention, a follow-up abdominal CT scan showed obliteration of the fistulous tract and a decrease in the extent of the abscess (). Eight months later, he underwent four sessions of additional TACE without abscess recurrence (). The most recent CT scan, taken 7 months after Histoacryl injection, showed advanced HCC without abscess or fistula formation. The patient is still alive and has been followed up at the outpatient clinic.

[[71.0, 'year']]

M

{'22113044': 1, '18451695': 1, '15256974': 1, '21415585': 2, '21296855': 1, '29467916': 1, '19042048': 1, '17103105': 1, '19451307': 1, '34146787': 1, '33833836': 1, '21960103': 1, '24574839': 2}

{'3304605-1': 1}

3932380-1

noncomm/PMC003xxxxxx/PMC3932380.xml

Spontaneous coronary artery dissection by intravascular ultrasound in a patient with myocardial infarction

A 60-year-old male presented with dizziness and chest discomfort. The patient had experienced severe crushing chest pain with sweating 15 years ago. However, the patient did not visit the hospital at that time because the chest pain completely resolved after several hours. He was a 20-pack-year smoker and had been taking a calcium channel blocker, an angiotensin receptor blocker, and a hydrochlorothiazide for hypertension.\nElectrocardiography showed the Q wave in the V1, V2, and V3 leads. Transthoracic echocardiography showed akinesia of the mid to apical left ventricular (LV) anteroseptal wall. Cardiac enzymes were within their normal ranges and remained normal throughout the follow-up.\nCoronary angiography revealed a dual lumen separated by a flap-like defect, indicative of intimal dissection in the proximal left anterior descending artery (LAD) (). Intravascular ultrasonography (IVUS) showed an entry intimal tear proximal to the dissection site in the LAD (). shows an intimal flap dividing the true lumen from the false one at the dissection site. The false lumen had a larger area than the true lumen. demonstrates a re-entry intimal tear distal to the dissection site.\nPercutaneous coronary angioplasty was performed, and a 4.0 × 22.0-mm everolimus-eluting stent (Xience Prime, Abbott Vascular, Santa Clara, CA, USA) was inserted at the proximal LAD lesion.

[[60.0, 'year']]

M

{'648228': 1, '19816020': 1, '2773790': 1, '10527054': 1, '25774346': 1, '8039214': 1, '15640544': 1, '20448134': 1, '8172069': 1, '8673763': 1, '32850088': 2, '24574840': 2}

{'3932380-2': 2, '7427437-1': 1}

3932380-2

noncomm/PMC003xxxxxx/PMC3932380.xml

Spontaneous coronary artery dissection by intravascular ultrasound in a patient with myocardial infarction

A 40-year-old male was admitted to our hospital because of frequent chest pain. The patient had experienced severe squeezing chest pain 3 months previously, and he had developed frequent chest discomfort with dyspnea since then.\nHe was a 20-pack-year smoker. He had been taking a calcium channel blocker, an angiotensin receptor blocker, and statin for hypertension and hyperlipidemia for the past 10 years.\nElectrocardiography showed ST elevation in the V1 to V4 leads and reciprocal ST depression in leads II, III, and aVF. Transthoracic echocardiography showed preserved LV global systolic function with no definite regional wall motion abnormality. Cardiac enzymes demonstrated mildly elevated troponin I, which was initially 0.2 ng/mL and peaked at 0.3 ng/mL during the follow-up.\nCoronary angiography showed a dual lumen defect, indicative of intimal dissection in the mid LAD (). The other coronary arteries were angiographically normal. IVUS showed an intimal flap dividing the true lumen and false lumen at the mid LAD site ().\nPercutaneous coronary angioplasty was performed, and a 4.0 × 22.0-mm zotarolimus-eluting stent (Resolute Integrity, Medtronic, Santa Rosa, CA, USA) was inserted at the mid LAD lesion.\nAfter stent insertion, resolution of the initial dissection area was confirmed angiographically ().

[[40.0, 'year']]

M

{'648228': 1, '19816020': 1, '2773790': 1, '10527054': 1, '25774346': 1, '8039214': 1, '15640544': 1, '20448134': 1, '8172069': 1, '8673763': 1, '32850088': 2, '24574840': 2}

{'3932380-1': 2, '7427437-1': 1}

3932381-1

noncomm/PMC003xxxxxx/PMC3932381.xml

Hypothermia-induced acute kidney injury in an elderly patient

A 78-year-old Korean male was admitted to our hospital in November for severe hypothermia. He had been diagnosed with hypertension at the age of 65 years, and had been treated with medications. He was found comatose on a mountaineering path 4 hours prior to admission. The temperature on the mountain, where he was lying, was reported to be 14℃. Upon admission, the patient was in deep stupor. His blood pressure, as measured in the supine position, was 80/50 mmHg, heart rate was 85 beats per minute, and respiratory rate was 19 per minute. The body temperature was below 34.0℃ (exceeding the measurement range of 34.0℃ to 44.0℃ of the thermometer, which had an error of measurement of ± 0.2℃). Electrocardiography showed J waves, prolonged QT, and occasional premature ventricular complexes (). Therefore, we estimated that his body temperature was indicative of moderate to severe hypothermia.\nChest radiography appeared normal, and ultrasonography of the kidneys showed normal echogenicity and intact corticomedullary differentiation without hydronephrosis. The patient's kidneys were within the normal range in size (the right kidney measured 9.3 × 5.4 cm, and the left kidney measured 9.9 × 4.6 cm). Renal scan (technetium-99m MAG3) indicated normal excretion and secretion functions. Urinalysis showed no proteinuria, and urine microscopic exam showed 10 to 19 red blood cells per high-power field with no casts. Blood urea nitrogen and serum creatinine concentrations were 25 mg/dL (normal range, 8 to 20) and 2.7 mg/dL (normal range, 0.6 to 1.2), respectively. In addition, fractional excretion of sodium was 4.5% and renal failure index was 2.25. The results of hormonal assays were as follow: plasma cortisol 14.6 µg/dL (normal range, 4.0 to 18.3), thyroid simulating hormone 1.43 µIU/mL (normal range, 0.17 to 4.05), free triiodothyronine 0.49 ng/dL (normal range, 0.78 to 1.82), and free thyroxine 1.25 ng/dL (normal range, 0.93 to 1.20). Anterior pituitary hormone levels were normal.\nThe results of arterial blood gas analysis were as follows: pH 7.260, pCO2 28.0 mmHg, pO2 151.0 mmHg, and HCO3- 12.6 mmol/L. Other laboratory studies produced the following results: serum calcium 8.8 mg/dL, serum phosphate 8.0 mg/dL, serum sodium 141 mEq/L, serum potassium 3.7 mEq/L, serum magnesium 3.1 mg/dL, hemoglobin A1c 5.4%, lactic acid 11.4 mmol/L, serum myoglobin 1,685.0 ng/mL, serum lactate dehydrogenase 159 IU/L, serum creatine phosphokinase 68 U/L, and serum amylase 582 U/L. Urinary concentrations of sodium, potassium, chloride were 95, 44.1, and 114 mEq/L, respectively. Urine osmolality was 537 mOsm/kg.\nUpon hospitalization, the patient was wrapped in a warm blanket. We provided him with a forced-air warming system (Warm Touch, Mallinckrodt Medical Inc., Dublin, Ireland) infused with warmed saline (42℃) by pressure infusion (Ranger, Alrizant Healthcare Inc., Eden Prairie, MN, USA). Five hours of warming resulted in an increase in body temperature to 37.3℃, and the patient regained consciousness. Subsequently, electrocardiography showed a normal sinus rhythm (). After supportive care, the serum creatinine levels decreased gradually from 2.7 to 1.1 mg/dL on the fourth day of the hospital stay. The patient was discharged after 12 days of hospitalization, and the serum creatinine and body temperature were 1.2 mg/dL and 36.5℃ at discharge ().

[[78.0, 'year']]

M

{'32778153': 1, '3544646': 1, '16565126': 1, '15617296': 1, '20075584': 1, '10097429': 1, '358883': 1, '31063411': 1, '11271094': 1, '7425418': 1, '27677616': 1, '3518093': 1, '24574841': 2}

{}

3932400-1

noncomm/PMC003xxxxxx/PMC3932400.xml

Duodenal adenocarcinoma following a neuroendocrine tumor in the duodenum

A 60-year-old male was admitted to the gastrointestinal medical clinic of Severance Hospital to undergo an operation for duodenal cancer.\nThe patient had developed renal tuberculosis 30 years previously, received medical treatment for several months, and was completely cured. In June 2009, he visited a local clinic after experiencing general weakness and melena over a period of 1 month, was diagnosed with duodenal cancer with multiple liver metastases (), and was transferred to our hospital.\nEsophagogastroduodenoscopy (EGD) () confirmed an ulcerofungating mass with poorly differentiated neuroendocrine carcinoma at the superior duodenal angle of the duodenum (). Initial computed tomography (CT) showed multiple liver metastases. From November 2009 to January 2010, six cycles of palliative chemotherapy comprising 5-fluorouracil (5-FU) at 1,000 mg/m2 (days 1 to 3), etoposide at 100 mg/m2 (days 1 to 3), and cisplatin at 70 mg/m2 (day 1) were performed. After these treatments, fludeoxyglucose uptake was no longer seen on positron emission tomography (PET)-CT. There was no remnant malignant lesion on EGD, and the patient had only pathologically confirmed chronic gastritis. Two more cycles of chemotherapy were performed. He was subsequently confirmed to have no evidence of disease in February 2010.\nIn June 2010, a polyp was detected in the former lesion site and was confirmed to be chronic inflammation by EGD biopsy. There were no abnormal findings on CT. In February 2011, a polyp was detected by EGD at the same site as that of the former polyp (). This was pathologically confirmed to be adenocarcinoma by immunohistochemical staining. Chromogranin A and synaptophysin, which are neuroendocrine markers, were negative. Carcinoembryonic antigen (CEA), which is usually detected in cases of adenocarcinoma, was positive (). The patient was admitted for an operation. Upon examination, his vital signs were normal. He reported no symptoms, and there was no palpable mass, organs, or tenderness in the abdomen. The results of a complete blood count were normal, as were the plasma levels of routine chemistry and tumor markers (CEA and CA 19-9). Magnetic resonance imaging (MRI) and PET-CT showed no abnormal findings at the site of the duodenal lesion, which was detected by EGD. However, there were suspicious metastatic lesions at segments 6/7 and segment 7 of the liver, so intraoperative radiofrequency ablation was planned and performed on 28 February 2011. The duodenal adenocarcinoma was not invading the mesentery. Pylorus-preserving pancreaticoduodenectomy was performed. A definite lesion was not found by intraoperative sonography, but a lesion was found attached to the right margin of the right hepatic vein on preoperative MRI. Thus, right posterior segmental curative resection of the liver was performed.\nThe surgical pathology results showed moderately differentiated duodenal adenocarcinoma at the site of the primary lesion, and mild nonspecific reactive hepatitis was confirmed in the liver via immunohistochemical staining (). At the site of the primary lesion, there were no neuroendocrine cells; only pure adenocarcinoma cells were present. Therefore, the final stage was T1N0M0, and the patient underwent no further adjuvant chemotherapy. The patient is undergoing follow-up in the outpatient department without recurrence.

[[60.0, 'year']]

M

{'8505174': 1, '17312381': 1, '20223786': 1, '10932598': 1, '17026561': 1, '27688667': 2, '20609724': 1, '21160582': 1, '15274064': 1, '8921448': 1, '24574838': 2}

{'5037094-1': 1}

3932568-1

noncomm/PMC003xxxxxx/PMC3932568.xml

Horse-shoe lung-rediscovered via volume rendered images

A 1-month-old male child initially presented with complaints of irritability and continuous crying. The child subsequently developed fever and chest infection. A chest radiograph done for the same revealed dextrocardia, and further evaluation by 2D echocardiography was done to look for congenital heart disease. The 2D echo findings showed dextroposed heart with lack of normal four pulmonary veins draining into the left atrium. A suspicion of scimitar syndrome was raised and further evaluation by CT scan was advised to assess the arterial supply and venous drainage of the right lung. CT imaging was preferred over MR angiography as there was a need to exclude lung sequestration besides delineating the pulmonary venous drainage.\nCT pulmonary angiography was performed on a 64 detector GE Lightspeed VCT scanner using non-ionic contrast media and bolus-tracking method. Contrast was injected through an intraven intervenues (IV) line placed in the right hand as lower limb venous access was small and would limit the flow rate. The study was performed under short acting general anesthesia wherein 1.5 cc/kg of contrast was injected through a dual head pressure injector with a flow rate of 0.4 cc/s. Images were acquired on 64-detector CT scanner without ECG gating, using a slice thickness of 0.7 mm and collimation 0.6 mm, followed by overlapping reconstruction. The acquired CT images were post-processed with focus on establishing the arterial supply to both lungs, the size and location of the main pulmonary artery (MPA), the right and left pulmonary arteries (RPA and LPA), and the venous drainage of both lungs. Volume-rendered (VR) reconstructions were done for depicting the pathology in 3D to aid understanding of the anomaly.\nA scout topogram of the chest taken for planning of CT imaging showed the heart shadow in the right hemithorax []. Evaluation of CT images showed that the right lung was small in size with compensatory emphysema of the left lung []. There was associated dextroposition of the heart with atrio-ventircular and ventriculo-arterial concordance.\nAll components of the pulmonary venolobar syndrome, also known as hypogentic lung or scimitar syndrome, were identified. These findings included systemic supply to the right lung base by two large collaterals arising from the upper abdominal aorta, one of which was a branch of the coeliac trunk []. These collaterals were seen supplying the basal segments of the right lower lobe. No collateral supply from the thoracic aorta was identified. There was no systemic collateral supply to the left lung. The right lung showed venous drainage to the suprahepatic inferior vena cava (IVC) through multiple small channels (at least three) []. In addition to these findings, there was also seen an anomalous tissue of lung extending in the retrocardiac region across the midline []. On standard axial images, both on mediastinal and lung windows, this lung tissue looked no different from the hyperinflated left lung tissue. However, on VR imaging with tracing of the main and branch pulmonary arteries and evaluation of the bronchi, peculiar findings of horseshoe lung were apparent [Figures and ]. These included a branch of the right pulmonary artery (RPA) extending across the midline to the left side. An associated bronchial supply also extending to the left was seen on VR imaging. These findings were reassessed on axial images and diagnosis of horseshoe lung was made in association with pulmonary venolobar syndrome. This highlights the importance of VR imaging in the evaluation of congenital anomalies. In this case, it helped in depicting the findings of a rare association of horseshoe lung with pulmonary venolobar or scimitar syndrome.

[[1.0, 'month']]

M

{'3484565': 1, '9486078': 1, '8469591': 1, '2962614': 1, '31420418': 1, '2251009': 1, '3484566': 1, '34269531': 1, '3952300': 1, '33604237': 1, '29970781': 1, '24604930': 2}

{}

3932569-1

noncomm/PMC003xxxxxx/PMC3932569.xml

A rare pleural mucinous cystadenocarcinoma mimicking loculated empyema initially: A case report

A 35-year-old male presented with upper respiratory symptoms and dyspnea for 3 days, along with right flank pain. He had been smoking for around 10 years. There was no significant past medical history. Laboratory tests showed an elevated white blood cell (WBC) count of 182,000/mm3 and C-reactive protein (CRP) level of 12.4 mg/dL. Chest radiograph revealed a large soft tissue opacity in the medial aspect of right lower hemithorax. It had smooth lobulated lateral margins whereas the medial margin merged with the mediastinal silhouette []. Contrast-enhanced chest CT showed a large, well-defined, ovoid, non-enhancing low attenuation lesion with mild peripheral rim-like enhancement in right posterior hemithorax (maximal diameter about 10 × 8 cm). Adjacent to the intrapleural low attenuation lesion, there was peribronchial consolidation in right posterior basal lung containing low attenuation area and fluid bronchogram with focal communication into adjacent pleural space [Figure and ]. Small amount of free pleural effusion was also detected. Neither mediastinal nor hilar lymphadenopathy was detectable. Initial CT diagnosis was a large loculated empyema with adjacent focal necrotizing pneumonia. Insertion of chest tube was performed for the drainage of pleural fluid and the resultant pleural cytology showed malignant cells. With the impression of intrapleural cystic malignancy, patient underwent open thoracotomy with decortication and right lower lobe (RLL) wedge resection. At surgery, a huge gelfoam like mass was found mainly occupying the pleural space in right posterior hemithorax. The specimen including pleural cystic mass and subpleural parenchyma, was of size 10 × 7 × 3 cm. Histologically, most of the tumor was composed of dissecting mucin material. The peripheral portion of the tumor showed mucinous epithelial cell nests with stratification and mild nuclear atypia. The mucinous epithelial cells had immunoreactivity for cytokeratin 7 (CK7) []. Photomicroscopic image revealed the tumor cells in fibrotic tissue. Mucinous material was attached to pleural fibrotic tissue []. Follow-up chest CT and [18]F-fluorodeoxyglucose (FDG) positron emission tomography (PET)-CT were performed a couple of weeks after the operation for the evaluation of residual lesion or extrathoracic primary site of metastasis. Imaging findings revealed remnant subpleural multiloculated cystic lesion in RLL, with postoperative changes along the resection site. There was no evidence of extrathoracic malignancy []. Patient received four cycles of gemcitabine and cisplatin chemotherapy. But the residual cystic mass in RLL increased on the follow-up chest CT scan after 7 months []. Reoperation with RLL wedge resection of the growing intrapulmonary cystic mass was done and pathological examination confirmed intrapulmonary mucinous cystadenocarcinoma. In 1 year follow-up after postoperative radiotherapy, the patient is stable.

[[35.0, 'year']]

M

{'28878858': 1, '17447515': 1, '746508': 1, '19830231': 2, '18618726': 1, '15898407': 1, '23483761': 2, '21252716': 1, '24604931': 2}

{'3590356-1': 1, '2737758-1': 1}

3932571-1

noncomm/PMC003xxxxxx/PMC3932571.xml

Isolated retroaortic left innominate vein in an adult without cardiac or aortic anomalies

A 68-year-old man was recovering from bacterial pneumonia involving both lungs. Patient had loss of weight and appetite. He did not have any heart disease. His clinical examination was unremarkable. No significant cardiac abnormality was noted on auscultation and in electrocardiography. However, his blood levels showed persistently high eosinophil count. Chest radiograph was unremarkable except for resolving pneumonia. Contrast-enhanced computed tomography (CT) scan of thorax, abdomen, and pelvis was done in dual-energy CT scanner (SOMATOM Definition Flash, Siemens Healthcare, Erlangen, Germany) to rule out any underlying neoplasm. No definite mass was noted in the CT. Incidentally, the left innominate vein had an abnormal course [] behind the ascending aorta before joining the right innominate vein to form the SVC which was normal in position (on the right side). The volume-rendered three-dimensional images obtained by dual-energy based bone removal clearly showed the relationship with the aortic arch []. The arch of aorta was left-sided. Heart was normal in CT scan. The anomalous course of the retroaortic innominate vein was highlighted in the report because of its clinical implications.

[[68.0, 'year']]

M

{'12910378': 1, '18716126': 1, '27186479': 2, '31088015': 1, '34805039': 2, '12768325': 1, '20300257': 1, '1430437': 1, '15788602': 1, '10549081': 1, '28828105': 1, '20830788': 1, '24604933': 2}

{'8602870-1': 1, '4844596-1': 1}

3932572-1

noncomm/PMC003xxxxxx/PMC3932572.xml

Acute pulmonary melioidosis in a child: A case report and review of literature

A 16-month-old girl child from North east district of India presented to the emergency department with multiple erythematous nodules and ulcers in the skin [Figure and ], difficulty in breathing, and fever since 5 days. The parents gave history of near drowning in drain water 7 days prior to the onset of illness. The birth history was unremarkable.\nOn general physical examination, the child was conscious, irritable, febrile with toxic look, had tachypnea and tachycardia, and weighed 8 kg (Grade I malnutrition for age). Multiple erythematous nodules and ulcers (ecthyma gangrenosum) were noted in the skin, with few eschars [Figure and ]. Her pulse was 100/min and BP 90/56 mm Hg. Routine blood examinations revealed raised leukocyte count (14,500/mm3), neutrophilia (89%), elevated C-reactive protein (CRP; 68 mg/l) and procalcitonin (2.56 ng/dl) levels. The liver enzymes were mildly elevated [aspartate transaminase (AST) 138 IU/l; alanine transaminase (ALT) 99 IU/l; alkaline phosphatase (ALP) 363 IU/l] and renal function tests were unremarkable. The blood sugar level was within normal limits. ELISA test for the qualitative determination of antibodies to human immunodeficiency virus (HIV) type 1 and 2 viruses was non reactive. On examining the respiratory system, coarse crepitations and rhonchi were noted bilaterally.\nInitial empirical antibiotic therapy was initiated with intravenous (IV) injections of Piperacillin + Tazobactum and Netilmicin. However, there was progressive deterioration of the patient's general condition with appearance of new skin lesions during the course of treatment.\nAn anterior–posterior (AP) view of chest radiograph [] revealed multiple patchy consolidations with air bronchogram in the left upper-mid zone and right mid and lower zones, with cavitations in few of them.\nHigh-resolution computed tomography (HRCT) and contrast-enhanced computed tomography (CECT) of the thorax was done on 64 slice MDCT scanner, (Aquilion 64, Toshiba Medical Systems Corporation, Japan) [Figures –] which revealed multiple variable-sized segmental and subsegmental air-space consolidations scattered in bilateral lung parenchyma, more on the right side where few of them showed areas of eccentrically located breakdown and cavitations. Multiple relatively larger nodular lesions were also noted in parenchyma of both the lungs. Most of the cavitary lesions showed dependant intracavitary low-density collections. No mediastinal lymphadenopathy was noted. Based on the imaging findings, an initial diagnosis of septic pyogenic pneumonic abscesses with differential diagnosis of pulmonary tuberculosis was given.\nHowever, a diagnosis of tuberculosis did not correlate well with the atypical skin lesions, the acute presentation, and the rapidly progressing clinical deterioration of the child. A fine-needle aspiration cytology (FNAC) from a nodule in the left forearm revealed frank pus [Figures and ]. A blood culture was done which showed growth of colonies of B. pseudomallei within 3 days, which established the diagnosis of melioidosis.\nBased on the culture and sensitivity reports, specific antibiotic therapy was started with IV Meropenam, along with nebulization. However, despite intensive care and antibiotic therapy unfortunately, the child died 2 days later due to septicemic pneumonia (1 week since admission).

[[16.0, 'month']]

F

{'16935773': 1, '21731307': 2, '7587451': 1, '19098086': 1, '3340766': 1, '29005374': 1, '25653950': 1, '10416320': 1, '12767750': 1, '22269609': 2, '20578524': 1, '18812053': 1, '31330088': 1, '24604934': 2}

{'3295726-1': 1, '3125033-1': 1}

3932575-1

noncomm/PMC003xxxxxx/PMC3932575.xml

Encephalocraniocutaneous lipomatosis with calvarial exostosis - Case report and review of literature

A 21-year-old female presented with skin-colored swelling over right frontotemporal region, present since birth. It was hard to touch with adjoining area of soft tissue. The overlying skin was devoid of hair. At birth, she was also noted to have a nodular lesion in the right upper eyelid and scleral mass in the right ocular globe. A few small skin nodules were also present over ipsilateral side of the face []. All these cutaneous and ocular lesions were increasing in size in proportion to patient's generalized body growth. The scleral mass was now interfering with her vision, so the patient wanted its removal. She also wanted surgical correction of the scalp swelling for cosmetic reasons. She had no history of seizures and neurological examination was unremarkable. Plain computed tomography (CT) and magnetic resonance imaging (MRI) brain were done to exclude intracranial and intraorbital extension of these lesions. The right frontotemporal scalp lesion was diagnosed as subcutaneous lipoma as it gave fat signal on T1W images and was suppressed on fat-saturated T1W [Figures and ]. Also, on plain CT, it had fat HU value of −60 to −80 HU. A large right temporal exostosis was seen within this scalp lipoma []. However, no intracranial extension of this lesion was seen. The soft tissue in the right upper eyelid gave fat attenuation and signal at places, hence possibility of dermolipoma was considered [Figures and ]. The scleral mass had no intraorbital extension. On MRI, a lipoma was found in the middle cranial fossa on the right side, adjacent to the temporal lobe and reaching up to right cerebellopontine cistern [Figures and ]. MRI of the whole spine was also done, but no abnormality was detected. In view of the imaging and constellation of clinical features, the final diagnosis of ECCL was given.\nThe original diagnostic criteria for ECCL were proposed by Hunter in 2006.[] The revised criteria were given by Ute Moog in 2009[] for definite/proven and possible ECCL cases [], excluding the third group of probable ECCL as given by Hunter. In the revised criteria for ECCL, there are one major and three minor criteria involving ocular system, three major and five minor criteria involving cutaneous system, and three major and six minor criteria for CNS involvement. Three major criteria are also seen involving the other systems.\nThree systems involved, major criteria in ≥2, or Three systems involved, proven NP or possible NP + ≥1 of the minor skin criteria 2-5, or Two systems involved with major criteria, one of which is proven NP or possible NP + ≥1 of the minor skin criteria 2-5.\nTwo systems involved, major criteria in both Two systems involved, proven or possible NP.\nOur case fulfilled the criteria for definite diagnosis for ECCL with three main systems’ involvement. The two major criteria in our case were eyelid choristoma and intracranial lipoma. The minor criteria were possible NP (in the absence of histopathologic confirmation), patchy non-scarring alopecia, subcutaneous lipoma in right frontotemporal region, and small nodular skin tags over ipsilateral face. Additionally, our case had a large temporal calvarial exostosis not reported earlier in the literature.

[[21.0, 'year']]

F

{'8549249': 1, '28707961': 1, '23197919': 1, '662483': 1, '18568254': 1, '16523517': 1, '17314447': 1, '27579462': 1, '27990391': 2, '19652916': 1, '29192135': 1, '19574261': 1, '4902772': 1, '9974077': 1, '12140223': 1, '24604937': 2}

{'5134170-1': 1}

3932577-1

noncomm/PMC003xxxxxx/PMC3932577.xml

Cone-beam computed tomography in the management of dentigerous cyst of the jaws: A report of two cases

A 17-year-old male patient was referred by an orthodontist for evaluation of a cystic radiolucency surrounding the crown of impacted 47 observed on a routine OPG []. The OPG showed an unerupted 47 with a well-demarcated radiolucency around the coronal portion of the tooth, suggestive of a dentigerous cyst. The tip of the roots of forty seven appeared to be impinging on the inferior alveolar canal. Also, the margins of the lesion were in close proximity to the distal root of 46 and the mesial root of forty eight. The patient was asymptomatic and examination did not reveal any abnormalities. He was advised surgical removal of the cyst alongwith impacted tooth and was prescribed a CBCT as part of the presurgical workup to aid in preserving the adjacent teeth and preventing nerve damage.\nBased on the findings of CBCT study, the nerve was traced carefully and identified lingual to the roots and just touching the apex of the roots of forty seven. Also, the tip of the distal root of 46 was seen close to the lesion and hence an intentional RCT was performed. Tooth 48 was preserved and kept under observation. The CBCT images revealed that the roots of the impacted tooth were intimately associated with the inferior alveolar canal. This information, which was not revealed on the OPG [Figure –], enabled the surgeon to employ the necessary high level of intrasurgical care and dissect the tooth with minimal trauma to the nerve.\nThe cyst was enucleated and fixed in 10% formalin. Histological examination identified it as a dentigerous cyst. There were no postoperative complications and on a recent follow-up visit, the patient did not exhibit any symptoms of neural damage such as anesthesia/paresthesia or tingling or burning sensation.

[[17.0, 'year']]

M

{'21731270': 1, '10379084': 1, '18404914': 1, '19461061': 1, '18848113': 1, '19639086': 1, '20587656': 1, '14663801': 1, '18238255': 1, '21507612': 1, '16934625': 1, '18082349': 1, '17315829': 1, '16528556': 1, '24604939': 2}

{'3932577-2': 2}

3932577-2

noncomm/PMC003xxxxxx/PMC3932577.xml

Cone-beam computed tomography in the management of dentigerous cyst of the jaws: A report of two cases

A 23-year-old male patient reported with a complaint of mild continuous pain and swelling of the left cheek. The mild swelling had been first noticed by the patient 2 years back but it had been asymptomatic. Extraorally, a nontender, firm, diffuse swelling of the left malar region, causing mild facial asymmetry, was observed. Intraorally, no swelling was detected. The maxillary left third molar (28) was unerupted, the gingival, alveolar, and palatal mucosa appeared normal.\nThe OPG revealed a radiolucent lesion, with the impacted tooth displaced to the region of the inferior orbital rim []. The patient was advised a CBCT scan [Figure –]. The CBCT image revealed the presence of a unilocular radiolucent lesion, measuring 24.3 mm × 26.6 mm × 32.5 mm in its largest dimension, occupying most of the left maxillary sinus. The lesion had well-defined sclerotic borders, except at the inferolateral wall of the sinus and the crestal region of twenty eight. The lesion caused slight expansion of the antrum and left lateral wall of the maxillary sinus. A well-defined radiodensity, morphologically resembling a tooth with open apices, was observed embedded in the lateral wall of the middle meatus.\nDuring surgery, a vestibular flap was raised from twenty three to twenty seven and a 1-cm window was made in the anterolateral wall of the sinus. The cyst lining was lifted and the cyst was enucleated, and the impacted tooth was also removed. The cyst was diagnosed as a dentigerous cyst on histopathological examination. There were no postoperative complications. The patient had recovered well on a recent follow-up.

[[23.0, 'year']]

M

{'21731270': 1, '10379084': 1, '18404914': 1, '19461061': 1, '18848113': 1, '19639086': 1, '20587656': 1, '14663801': 1, '18238255': 1, '21507612': 1, '16934625': 1, '18082349': 1, '17315829': 1, '16528556': 1, '24604939': 2}

{'3932577-1': 2}

3932579-1

noncomm/PMC003xxxxxx/PMC3932579.xml

Percutaneous transhepatic portal vein stenting in a patient with benign non-transplant postoperative portal vein stenosis: A case report

A 45-year-old female patient presented to the gastrointestinal surgery out-patient department with history of pain in right hypochondrium and jaundice of 3 months duration. Ultrasonography (USG) revealed dilated intrahepatic biliary radicles (IHBR) in both lobes of liver with an ill-defined mass lesion in the region of porta hepatis. Multiphase contrast-enhanced computed tomography (CT) scan of the abdomen showed an ill-defined mass lesion involving the left hepatic duct and the primary biliary confluence with resultant dilatation of the IHBR of both lobes []. The mass showed delayed persistent contrast enhancement. Left branch of portal vein was completely encased by the mass with atrophy of the left lobe of liver. The main and right portal vein and the right, left, and proper hepatic arteries were normal. No abdominal lymphadenopathy was seen. The imaging diagnosis was infiltrating cholangiocarcinoma involving left hepatic duct and primary biliary confluence with invasion of left portal vein. The patient was surgically treated with left hepatectomy, hepaticojejunostomy, and portal vein repair. Histopathology confirmed the diagnosis. Subsequently, the patient was treated with chemotherapy and radiotherapy.\nDuring follow-up, the patient presented with short history of abdominal pain and distension after about 8 months of surgery. USG revealed gross ascites and Doppler study showed severely narrowed portal vein. This was followed by contrast-enhanced CT scan of the abdomen which revealed narrowing of extrahepatic portal vein at the site of repair with multiple periportal portosystemic collaterals and gross ascites. No mass lesion was seen in liver or at porta hepatis []. A diagnosis of postoperative benign portal vein stenosis was made and she was treated with percutaneous transhepatic balloon angioplasty of the portal vein, which showed disappearance of most of the collaterals []. Although the symptoms relieved initially, they recurred after 3 weeks and USG showed persistent stenosis of the extrahepatic portal vein. Since the angioplasty showed a positive outcome, percutaneous transhepatic portal vein stenting was planned. After initial balloon dilatation of the stenotic segment, a self-expandable metallic stent (Bloomington Indiana, USA), measuring 4 cm in length and 10 mm in diameter, was placed across the stenosis []. Portal venography after the stenting showed disappearance of the collaterals []. Patient's symptoms improved with minimal ascites on follow-up USG after 1 month. Four months after stenting, the patient again developed moderate ascites with deranged liver functions and vomiting. USG showed ascites with coarsened liver echotexture and patent portal vein stent with normal flow. Two weeks later, the patient died of hepatic failure, which was presumed to be due to postoperative chemo-radiotherapy.

[[45.0, 'year']]

F

{'15929192': 1, '20353467': 1, '19339200': 1, '19370787': 1, '21427310': 1, '18956224': 1, '16257135': 1, '18086428': 1, '11425988': 1, '12749245': 1, '1543202': 1, '24604941': 2}

{}

3932584-1

noncomm/PMC003xxxxxx/PMC3932584.xml

Unconventional abdominal uses of FIESTA (CISS) sequence

A 38-year-old male presented with vague epigastric pain. MRI (1.5 T) axial FIESTA images [] showed superior mesenteric vein (SMV) swirling around superior mesenteric artery (SMA), which is suggestive of midgut volvulus. This was confirmed by plain CT [Figure –]. Coronal FIESTA images [Figure and ] showed prominent duodenal loop that returned to right side of the abdomen (suggestive of volvulus); coronal T2 fat-saturated images [Figure and ] showed jejunal loops on right and ileal loops on the left of abdomen (suggestive of malrotation).

[[38.0, 'year']]

M

{'17729346': 1, '18086992': 1, '16799782': 1, '591236': 1, '16314698': 1, '18196324': 1, '18635634': 1, '17373755': 1, '9210631': 1, '19959507': 1, '19458102': 1, '20518091': 1, '18806147': 1, '10966702': 1, '6604422': 1, '19829910': 2, '15908538': 1, '16546032': 1, '21431484': 1, '17208678': 1, '3510519': 1, '11932222': 1, '19669973': 1, '12072607': 1, '16861559': 1, '11598254': 1, '17981166': 1, '24604946': 2}

{'3932584-2': 2, '3932584-3': 2, '3932584-4': 2, '2740257-1': 1}

3932584-2

noncomm/PMC003xxxxxx/PMC3932584.xml

Unconventional abdominal uses of FIESTA (CISS) sequence

A 70-year-old man presented with abdominal discomfort and history of blood in stools. Coronal MR (1.5 T) FIESTA image [] showed an abnormally dilated bowel loop in left lower abdomen, suggestive of sigmoid volvulus. This was confirmed by coronal T2 fat-saturated images [Figure –], which showed air-filled loop [] and fluid in the limbs of loop [Figure and ], CT scanogram [], and coronal CT section [].

[[70.0, 'year']]

M

{'17729346': 1, '18086992': 1, '16799782': 1, '591236': 1, '16314698': 1, '18196324': 1, '18635634': 1, '17373755': 1, '9210631': 1, '19959507': 1, '19458102': 1, '20518091': 1, '18806147': 1, '10966702': 1, '6604422': 1, '19829910': 2, '15908538': 1, '16546032': 1, '21431484': 1, '17208678': 1, '3510519': 1, '11932222': 1, '19669973': 1, '12072607': 1, '16861559': 1, '11598254': 1, '17981166': 1, '24604946': 2}

{'3932584-1': 2, '3932584-3': 2, '3932584-4': 2, '2740257-1': 1}

3932584-3

noncomm/PMC003xxxxxx/PMC3932584.xml

Unconventional abdominal uses of FIESTA (CISS) sequence

A 40-year-old female presented with acute exacerbation of right upper quadrant pain. MR (3 T) axial T2 [] and T2 fat-saturated [] images showed a hydatid cyst in the right lobe of the liver, reaching up to liver hilum. MR axial FIESTA [Figure and ] images differentiated biliary tract from portal vein branches and clearly demonstrated biliary communication []. Hydatid material seen within common bile duct (CBD) on magnetic resonance choangiopancreatography (MRCP) images [Figure and ] confirmed biliary rupture.

[[40.0, 'year']]

F

{'17729346': 1, '18086992': 1, '16799782': 1, '591236': 1, '16314698': 1, '18196324': 1, '18635634': 1, '17373755': 1, '9210631': 1, '19959507': 1, '19458102': 1, '20518091': 1, '18806147': 1, '10966702': 1, '6604422': 1, '19829910': 2, '15908538': 1, '16546032': 1, '21431484': 1, '17208678': 1, '3510519': 1, '11932222': 1, '19669973': 1, '12072607': 1, '16861559': 1, '11598254': 1, '17981166': 1, '24604946': 2}

{'3932584-1': 2, '3932584-2': 2, '3932584-4': 2, '2740257-1': 1}

3932584-4

noncomm/PMC003xxxxxx/PMC3932584.xml

Unconventional abdominal uses of FIESTA (CISS) sequence

A 63-year-old male came for follow-up of resolving liver lesion. MR (1.5 T) axial [Figure and ] and coronal FIESTA [Figure and ] images showed well-defined lesions with black outline within small bowel. These lesions were hyperintense on T1 [] and suppressed on fat-saturated images [] suggestive of fat content. CT sections [Figure and ] confirmed the fat density of small bowel lipomas. Wall thickening noted around a lipoma on MR [] and CT [] images suggested intussusception, a known complication of lipoma.

[[63.0, 'year']]

M

{'17729346': 1, '18086992': 1, '16799782': 1, '591236': 1, '16314698': 1, '18196324': 1, '18635634': 1, '17373755': 1, '9210631': 1, '19959507': 1, '19458102': 1, '20518091': 1, '18806147': 1, '10966702': 1, '6604422': 1, '19829910': 2, '15908538': 1, '16546032': 1, '21431484': 1, '17208678': 1, '3510519': 1, '11932222': 1, '19669973': 1, '12072607': 1, '16861559': 1, '11598254': 1, '17981166': 1, '24604946': 2}

{'3932584-1': 2, '3932584-2': 2, '3932584-3': 2, '2740257-1': 1}

3932600-1

noncomm/PMC003xxxxxx/PMC3932600.xml

Fanconi anemia presenting as an “evolving” acute leukemia-diagnostic challenges

A 14-year-old male patient presented with the complaints of generalized weakness and off and on high grade fever for 6 months. On examination, he was well-looking, afebrile and had pallor without any evidence of mucocutaneous bleeding. There was no lymphadenopathy or organomegaly. His complete blood counts (CBC) showed a hemoglobin (Hb) of 8.7 g/dl, total leucocyte count (TLC) of 1.5 × 109/L and a platelet count of 38 × 109/L. Red cell indices showed hematocrit of 25.2 %, mean corpuscular volume of 102.5 fl, mean corpuscular Hb of 35.3 pg and mean corpuscular Hb concentration of 34.4 g/dl. A clinical diagnosis of aplastic anemia (AA) was made and bone marrow examination was carried out. The marrow aspirate smears were hemodilute and pauci-particulate with very few marrow cell trails showing presence of blast cells, mature monocytes and cells with promonocytoid features (~10%, ~13% and ~4% of the evaluable marrow cells, respectively) []. Erythroid cells showed megaloblastic erythropoiesis with dyserythropoiesis. There was a prominence of macrophages with hemophagocytosis. The blast cells were negative for myeloperoxidase (MPO) cytochemical staining. Bone marrow trephine biopsy was suboptimal and insufficient for opinion. In view of the hemodilution and lack of appreciable dyshemopoietic features, a diagnosis of an “evolving” MPO negative acute leukemia was given and flowcytometric immunophenotyping (FCIM) was suggested for further evaluation. Meanwhile, the clinical course of patient was apparently uneventful except for an acute onset submandibular swelling that subsided with antibiotic therapy. His subsequent CBC at next presentation after 2 weeks showed persistent pancytopenia with Hb of 10.2 g/dl, TLC of 2.3 × 109/L and 21 × 109/L platelets. At this time, his marrow aspirate smears showed hypocellular marrow cell trails with myelogram comprising 18% blasts, 10% promonocytes, 04% monocytes, 10% polymorphs, 01% metamyelocytes, 03% eosinophils, 42% lymphocytes, 04% plasma cells and 08% erythroid precursor cells []. A diagnosis of hypocellular marrow with AML was made. The conventional cytogenetics revealed a normal karyotype []. At subsequent presentation, the patient was apparently maintained except for worsening anemia and thrombocytopenia. There was no bleeding and his clinical examination did not reveal any new findings. His peripheral blood was sent for the stress cytogenetics and human leukocyte antigen (HLA) typing. However, after 3 months from initial presentation, he came with high grade fever and bone pains and finally consented for admission. His CBC showed 6.6 g/dl Hb, 0.8 × 109/L leucocytes and 3 × 109/L platelets. Bone marrow aspirate revealed hypocellular particles with ~68% blasts and promonocytes. ~3% blast cells showed MPO positivity. The 6-color FCIM (FACS Canto II, BD Biosciences) on bone marrow revealed significant replacement by a prominent cell population (red) with variable CD45 expression. This abnormal cluster spanned from the blast region into the monocyte position with bulk of the neoplastic cells falling into the medium cell size range []. A small number of lymphocytes (blue), few residual myeloid precursors (purple) as well as maturing monocytic elements (green) were also present. The leukemic cells comprised ~45% of the cells in the sample and showed homogeneously bright expression of CD33, variable expression of CD13, CD34 and displayed a complex pattern of CD14/CD64 co-expression, indicating monocytic maturation [Figure and ]. These blast cells also showed heterogeneous expression of CD11b, CD15, CD4 and bright HLA DR []. The maturing monocytic elements (comprising ~3% of cells in the sample) showed normal expression of brighter CD33, CD16, CD13, HLA DR and CD11b. Based on the shape of the abnormal cell cluster on Side Scatter/CD45 dot-plot and immunophenotypic findings including pattern of CD14/CD64 co-expression, diagnosis of AML with monocytic differentiation was made. Stress cytogenetics [] showed 88% chromosome breaks and rearrangements in patient's peripheral lymphocytes with mitomycin C (MMC) []. The diagnosis of FA was thus established. Patient consequently received packed red cell and platelet support and was started on a low dose cytosine arabinoside 100 mg/M2/daily (adverse drug event induction therapy). Subsequently, he developed typhlitis and was conservatively managed until he left the hospital care against medical advice.

[[14.0, 'year']]

M

{'12584146': 1, '12393424': 1, '11389461': 1, '8500573': 1, '8068955': 1, '2917181': 1, '10704682': 1, '12393516': 1, '939547': 1, '24604962': 2}

{}

3932601-1

noncomm/PMC003xxxxxx/PMC3932601.xml

Primary mediastinal seminoma; resistance and relapse: An aggressive entity

A 22-year-old male presented with a history of progressive dyspnea, cough, hoarseness of voice, and unquantified weight loss for the last 6 months in Feb 2010. On clinical examination, there were signs of superior vena cava syndrome, with 5 cm × 6 cm left supraclavicular mass. The whole body computed tomography (CT) scan showed heterogeneously enhancing 12 cm × 8.2 cm × 4.2 cm firms soft-tissue mass in the mediastinum encasing ascending aorta, arch of aorta, right main pulmonary artery, and proximal bronchi, infiltrating superior vena cava []. The ultrasound of scrotum was unremarkable. The biopsy of left supraclavicular mass was consistent for extragonadal seminoma with imunohistochemistry positive for C-kit and negative for leucocyte common antigen, epithelial membrane antigen and PLAP. The serum lactate dehydrogenase (LDH) was raised to 1963 U/L (normal range 100-190 U/L) while β- human chorionic gonadotropin (β- HCG < 1.20 mlU/ml) and alpha fetoprotein AFP (2.73 ng/ml) were normal. The baseline complete blood counts, renal and liver function tests were found to be normal.\nThe patient received chemotherapy comprising of bleomycin, etoposide, and cisplatin (BEP; Bleomycin 18 U/m2 IV, Etoposide 100 mg/m2, Cisplatin 20 mg/m2) 3 weekly for four cycles until July 2010. The patient tolerated chemotherapy well with no dose reductions. There was a serial reduction in serum LDH values. The response assessment CT showed residual disease of 5.8 cm × 4 × 4 cm and elevated serum LDH of 221 U/L. To consolidate the partial response and vascular involvement precluding surgery; external beam radiation to a dose of 30 Gy in 17 fractions over 3 weeks was given to the mediastinum and supraclavicular region. The 6 MV photons with the tomotherapy (high precision arc intensity modulated radiation with image guidance) were used to irradiate the planning target volume, which included clinical target volume (CTV) with a margin of 5 mm for set-up error. The CTV included gross residual tumor as seen on planning CT with 5 mm margin. The post-radiation positron emission tomography-CT (PET-CT) showed complete metabolic regression of the disease and the normal serum LDH. Patient was on regular follow-up with tumor markers and CT thorax abdomen pelvis every 3 monthly.\nAt 1 year patient presented with rapidly progressing right side cheek swelling (5 cm × 6 cm) and progressive lower backache. On examination, there was local tenderness in cheek swelling and in the region of 5th lumbar vertebrae. The CT showed a soft-tissue mass involving subcutaneous plane of right cheek of 6 cm × 6 cm × 4 cm []; increased size of mediastinal mass (10 cm × 8 cm × 6 cm) and multiple lytic lesions in ribs. The fine needle aspiration cytology from check swelling showed cells consistent with the metastatic seminoma collaborated with raised serum LDH values (353U/L). The serum β-HCG and AFP were normal. The technetium-99 m- methylene diphosphonate MDP bone scintigraphy revealed multiple metastases in ribs and 5th lumbar vertebra.\nAfter discussion in institutional joint tumor board meeting keeping the view of disease status, poor performance status and patient wishes; he was offered short course palliative radiotherapy to right face and lumbar spine with the supportive care. Review after 4 weeks showed poor pain relief and progressive disease. Taking an overall view of patient performance status advanced disease, distant geographical location, challenged socio-economic status and discussions with the patient and family members; further anti-neoplastic therapy was precluded and best supportive care was offered. Patient succumbed to progression of the disease shortly.

[[22.0, 'year']]

M

{'21651648': 1, '28467339': 2, '4044269': 1, '21161157': 1, '11919246': 1, '19937408': 1, '25395217': 1, '27441078': 2, '19395465': 1, '20530278': 1, '21460378': 1, '20933444': 1, '21724441': 1, '18751412': 1, '32857912': 1, '19289622': 1, '22243760': 1, '18040999': 1, '30430771': 1, '24604963': 2}

{'6166189-1': 1, '4935827-1': 1}

3932602-1

noncomm/PMC003xxxxxx/PMC3932602.xml

Successful outcome of mucormycosis in two children on induction therapy for acute lymphoblastic leukemia

A 10-year-old male was on induction chemotherapy (BFMBerlin-Frankfurt-Münster-95 protocol) for moderate risk T cell ALL. On day 11 of induction, he was observed to have a scabbed wound (2 cm × 2 cm) on the lateral aspect of his left forearm. There was a history of trauma 4 days ago. As the wound was in close proximity to the central line, parenteral antibiotics were started immediately. Antibiotics were switched from cefoperazone-sulbactam to meropenem and amikacin after 48 hour of non-response. Although absolute neutrophil count (ANC) = 60/mm3, child remained afebrile, non-toxic, capillary refill was brisk and radial pulsations were well-felt. Repeated blood and wound swab cultures were sterile. The lesion progressed and conventional amphotericin-B (CAB) was started on D18 of induction, after taking a skin biopsy for culture and histology. Skin scrapping showed growth of non-septate hypha, branching at right angles, diagnostic of mucormycosis. CAB was continued for 12 weeks, along with radical serial debridement of the necrotic wound. Computed tomography (CT) chest was normal. Induction chemotherapy was continued with prednisolone, vincristine, and L-asparaginase, however, only first dose of daunorubicin could be administered. Skin grafting by rotation flap was carried out to cover the exposed ulna proximal to the styloid process []. He achieved complete remission (CR) and subsequent cycles of chemotherapy were administered as per the protocol. He completed his treatment and is off therapy for the past 7 months.

[[10.0, 'year']]

M

{'15003897': 1, '11714058': 1, '30039238': 1, '18780010': 1, '18063603': 1, '14715813': 1, '32684769': 2, '15776383': 1, '34367400': 1, '18627579': 1, '9475685': 1, '26500748': 2, '19155544': 1, '16315848': 1, '25608979': 1, '11405452': 1, '24604964': 2}

{'3932602-2': 2, '4594447-1': 1, '7344120-1': 1}

3932602-2

noncomm/PMC003xxxxxx/PMC3932602.xml

Successful outcome of mucormycosis in two children on induction therapy for acute lymphoblastic leukemia

A 4-year-old male diagnosed with standard risk pre-cursor B ALL was hospitalized on day 4 of induction chemotherapy for fever and cellulitis on the dorsum of the right hand, at venous cannula site. As he was neutropenic (ANC = 100/mm3) broad spectrum antibiotics were administered to cover both gram positive and gram negative bacteria. The wound progressed to an abscess and pus culture grew antibiotic sensitive Pseudomonas aeruginosa. Despite the specific therapy (cefoperazone-sulbactam, amikacin), the patient developed a necrotic area at the tip of the little finger of the same hand, suggestive of ecthyma gangrenosum []. He subsequently developed pain and swelling over the right shoulder and as he had a central line on the same arm (8 cm from the wound), the line was removed despite normal coagulation profile and Doppler study. Shoulder pain resolved within 24 h of removing the line. As child remained afebrile, non-toxic and hemodynamically stable, induction chemotherapy was continued with some modification (no daunorubicin after D8). On D19, he developed another black skin lesion on the lateral aspect of left leg [] and skin scrapping from the lesion grew Streptococci on initial culture. However, due to progression, biopsy from the leg lesion was done and liposomal amphotericin B (LAB) was started. Histopathological examination showed broad ribbon-like non-septate hypha branching at the right angle, suggestive of mucormycosis. The fungal culture showed fluffy, grey, cotton candy-like growth that was identified as Rhizomucor pussilus []. CT scan of the chest revealed multiple, bilateral pulmonary lesions with right pleural effusion (at this time patient also had a cough and tachypnea). Successful control of mucormycosis was achieved by serial surgical debridement of both cutaneous lesions, wedge resection of two large lung lesions along with LAB and posaconazole (12 weeks). After completion of induction patient achieved CR and subsequent chemotherapy was administered as per protocol. He has completed his treatment and is off therapy for 4 months. The clinical characteristics of both cases are summarized in .

[[4.0, 'year']]

M

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{'3932602-1': 2, '4594447-1': 1, '7344120-1': 1}

3932603-1

noncomm/PMC003xxxxxx/PMC3932603.xml

Follicular dendritic cell sarcoma with paraneoplatic pemphigus: Rare case and a brief review of literature

A 20-year-old boy presented with 7 months history of multiple polymorphous vesiculobullous mucocutaneous eruptions around the lips and subsequently in buccal mucosa causing dysphagia. He later developed conjuntivitis associated with blurred vision and atypical targetoid lesions with crusts surrounded by erythema scattered on the trunk and extremities including prepuce and perianal region []. He lost 5 kg during this period. Direct immunoflorescence of biopsy specimen from Skin lesion showed deposition of Immunoglobulin G (+) and C3 in the intercellular region of epidermal cells mainly in the suprabasal region. Linear deposition of IgG is also seen in dermo-epidermal junction suggestive of PNP. Computed tomography (CT) scan revealed a well defined intensely enhancing mass lesion with central necrosis in the right hemipelvis, 6 cm in size, abutting the right obturator internus muscle and displacing the urinary bladder anteriorly. Engorged vessels were seen around the mass []. Exploratory laprotomy was done but the mass was found to be unresectable. Histopathlogy of the mass showed oval to spindle cells in a background of vascular stroma infiltrated by lymphocytes. The cells were immunopsitive for CD21, CD35, CD23, S-100, Claustrin, CD34, CD31, S-100; Leucocyte common antigen, and H uman leukocyte antigen-DR were also positive in some cells. Hyaline-vascular follicles were identified at the peripheral portion or occasionally scattered in the center of the tumor. Rich vascularity was highlighted by the CD34 immunostaining [Figures and ]. Overall features favoured FDCS. The presence of rich vascularity around the tumor and few hyalanized vascular follicles found in histopathological examination (HPE) gave the clue that the tumor might have developed from CD. He tested negative for human immunodeficiency virus. He was started on systemic steroids. His truncal lesions marginally improved after steroids but there was no improvement in arm lesions. He was then given Rituximab, cyclophosphamide, Adriamycin, vincristin, prednisolone. He expired post 1st course of chemotherapy due to pseudomonas infection.

[[20.0, 'year']]

M

{'34430462': 1, '10025851': 1, '33371089': 2, '9010103': 1, '17596748': 1, '27190659': 2, '18477166': 1, '16029358': 1, '21777224': 1, '8712294': 1, '32775392': 2, '12269857': 1, '31312243': 1, '25825580': 1, '15063382': 1, '12417613': 1, '17373675': 1, '2420185': 1, '24604965': 2}

{'4842386-1': 1, '7385594-1': 1, '7748373-1': 1}

3932604-1

noncomm/PMC003xxxxxx/PMC3932604.xml

Inflammatory myofibroblastic tumor of kidney with heterotopic bone formation: An unusual case mimicking a renal malignancy

A 32-year-old male patient came with complaints of pain in the lower abdomen and urinary tract symptoms since 1 year. The patient had a history of renal lithiasis 8 years back. Laboratory investigations including complete blood count, serum biochemistry and urine analysis were within the normal limits. Computed tomography (CT) scan revealed a heterogeneously enhancing mass lesion measuring 4.5 cm × 4.4 cm arising from the lower pole of the right kidney with an exophytic component. Calcific areas were also noted in the mass. It was confined to the cortex with no evidence of extension into renal calyces. Right renal vein was patent and there was no evidence of renal vein thrombosis. The contralateral kidney was normal. Radiological and clinical features strongly suggested a renal cell carcinoma (RCC) for which the patient underwent a right radical nephrectomy. Macroscopically, a well-circumscribed hard grey white tumor was noted in the lower pole of nephrectomy specimen with exophytic component also. The tumor was hard and had a gritty sensation on cutting through it. Histological examination revealed a tumor composed of cellular and hypocellular hyalinized areas. The cellular foci were composed of spindle cells in short fascicles and prominent storiform pattern. Spindle cells showed mild atypia and mitotic activity ranged from 4 to 7/10 high power field (HPF). There were lymphoid aggregates with germinal center formation and plasma cells interspersed amongst these spindle cells. Extravasated red blood cells were also seen prominently at many foci. Within the hypocellular hyalinized zones, the spindle cells were arranged in an unusual perivascular whorled pattern resembling meningeal whorls. In addition, heterotopic bone formation was seen bordered by osteoblastic cells []. Mitotic rate up to 3-6/10 HPF was appreciated focally in the cellular areas. However, no atypical mitosis was appreciated. No areas of conventional RCC were demonstrable even after studying additional sections from the tumor. On immunohistochemistry, the spindle cells were strongly positive for smooth muscle antibody [] and were negative for CD 34, ALK-1, CK7, CD 23, CD21 and h-caldesmon. p53 was positive in few of the spindle cells. The remaining renal parenchyma was unremakable. Surgical margins of excision were clear with no evidence of extension into the renal vein or renal pelvis. The patient is asymptomatic 18 months after his surgery.

[[32.0, 'year']]

M

{'3791160': 1, '7611533': 1, '17892577': 2, '16230166': 1, '32577144': 1, '21864369': 2, '31911920': 2, '26893784': 1, '17938159': 1, '7557942': 1, '8434707': 1, '11342774': 1, '12717250': 1, '24604966': 2}

{'2034571-1': 1, '3177925-1': 1, '6940336-1': 1}

3932605-1

noncomm/PMC003xxxxxx/PMC3932605.xml

Human immunodeficiency virus Infection in a patient of chronic myelogenous leukemia

A 50-year-old male patient was a known case of diabetes mellitus and was being treated for that since 1989. He was; however, quite irregular with this treatment and his diabetes was often out of control.\nIn November 2005, he presented with complaints of fever, cough with expectoration and weakness. The fever was low grade and there was no history of hemoptysis, dysuria and hematuria. Routine laboratory investigations showed high White Blood Cell count which was 59,000/mm3, and there were premature leucocytes on peripheral smear. He also had elevated platelet count which was 726,000/mm3. His diabetes was out of control with FBS level 264 mg/dl and PPBS value 372 mg/dl.\nA provisional diagnosis of CML with uncontrolled diabetes mellitus was made and he was further investigated.\nHis routine urinalysis showed glycosuria. Renal Function Tests and Liver Function Tests were within normal limits. Test for HIV infection was non-reactive. Chest X-ray did not reveal any abnormality. Bone marrow aspiration study was consistent with the diagnosis of CML. Test for Philadelphia chromosome was not carried out.\nHe was given hydroxyurea 500 mg 3 times a day and subsequently the doses were titrated with reference to WBC count.\nDuring the follow-up that spanned over next several months, he experienced occasional seasonal attacks of respiratory infection. For this, he was treated with antibiotics and insulin which controlled the infection promptly.\nIn October 2010, he complained of occasional fever, cough with slight expectoration and loss of appetite, weakness and weight loss. He was diagnosed to have pulmonary tuberculosis on the basis of his clinical picture and positive TB ELISA test (IgM) for which he received anti tubercular treatment for next 10 months.\nIn February 2011, he complained of retrosternal burning sensation while walking which was relieved by rest. He also experienced tightness around the chest. The pain did not radiate but was associated with the mild breathlessness. ECG did not reveal any abnormality. The Treadmill Test was strongly positive for exercise induced coronary insufficiency.\nAs his symptoms and stress test suggested coronary heart disease, he was referred to a cardiac center for further evaluation. 2D-echo was carried out which showed echo-concentric LVH, normal resting systolic function, presence of diastolic dysfunction, no regional wall motion abnormalities, and normal PA pressure. Coronary angiography was done on 1st March 2011 which showed LVEF 50% and triple vessel disease. Revascularization was contemplated; however, his routine investigation showed a positive HIV test; hence no further interventional treatment was carried out.\nThis new development necessitated further investigations. Philadelphia chromosome was tested which showed a positive result. CD3, CD4 and CD8 count results were 2940 cells/mm3, 393 cells/mm3 and 2477 cells/mm3 respectively. Viral load was tested on 5th November 2011and it showed 43356 HIV-1 RNA copies/ml.\nHe was advised to start anti-retroviral therapy along with Imatinib, however, patient declined to take this treatment due to lack of resources. He was therefore continued on hydroxyurea.\nIn May 2012, the retroviral disease tests were repeated. The results of both the tests are given in .\nPatient continues to take hydroxyurea regularly and in spite of advanced HIV he has not developed any major complications of HIV or CML during his follow-up period.

[[50.0, 'year']]

M

{'18184078': 1, '22527850': 1, '19492322': 1, '9250808': 1, '17203680': 1, '19025802': 1, '34186065': 1, '18452074': 1, '11361394': 1, '15597521': 1, '24604967': 2}

{}

3932606-1

noncomm/PMC003xxxxxx/PMC3932606.xml

Acute promyelocytic leukemia relapsing into acute myeloid leukemia-M2 with normal cytogenetics

A 32-year-old housewife presented with 1 month history of fever, bleeding per vaginum, and pancytopenia. Peripheral smear and bone marrow were suggestive of infiltration of 90-95% abnormal promyelocytes and blasts, which were strongly myeloperoxidase positive. Coagulation screen was normal. Flourescence in situ hybridization FISH for detection of translocation which fuses the promyelocytic leukemia gene (PML) on chromosome 15q22-q24 to the retinoic acid receptor alpha (RARA) gene on 17q21 PML/RARA translocation carried out by vysis probe/dual color dual fusion method identified PML/RARA translocation. After the first induction course, with ATRA (45 mg/m2/d per oral in 2 divided doses) with daunorubicin (45 mg/m2/d IV over days 1-3) the patient was in morphological complete remission (CR). This was followed by consolidation and maintenance which was completed by April 2009. The patient remained in CR until December 2010, when she presented with fever, weakness, and pancytopenia. Bone marrow examination was compatible with acute myeloid leukemia (AML) French-American-British M2; the karyotype was normal, PML/RARA was negative by both FISH and PCR methods which were repeated twice. The patient attained complete hematological remission after 7 + 3 induction cytarabine (100 mg/m2/d over days 1-7) and idarubicin (12 mg/m2/d over days 1-3) followed by first consolidation with high dose cytarabine. She did not have a full sibling Human leukocyte antigen HLA match. She was taken for autologous hematopoietic stem cell transplant (HSCT) using busulphan and cyclophosphamide conditioning. After a treatment free interval of 9 months, the patient again presented with fever, weakness and pancytopenia and diagnosed with second relapse (AML-M2). Patient was explained about the nature of the disease and the available treatment options and their resultant toxicity. She opted for supportive care and finally succumbed to her illness.

[[32.0, 'year']]

F

{'12886249': 1, '7832190': 1, '22480783': 1, '1551085': 1, '19806661': 1, '11122141': 1, '11522536': 1, '188440': 1, '32023731': 1, '11806982': 1, '8574167': 1, '11120332': 1, '8518183': 1, '29892552': 1, '11999558': 1, '12393590': 1, '24604968': 2}

{}

3932607-1

noncomm/PMC003xxxxxx/PMC3932607.xml

Cisplatin induced paroxysmal supraventricular tachycardia

Our patient was a 55-year-old male who had been recently diagnosed as having small cell carcinoma of the right lung. Patient was normotensive and had no underlying cardiac illness. Baseline electrocardiogram (ECG) and Echocardiography were normal as were other parameters []. He was planned for cisplatin based chemotherapy Cisplatin (62 mg D1, D2 + Etoposide 165 mg D1, D2, D3). Patient received first cycle of chemotherapy uneventfully. During the second cycle while on cisplatin infusion, patient complained of palpitations. An ECG was performed that revealed PSVT []. Cisplatin infusion was stopped and the patient was given diltiazem 15 mg intravenous. Meanwhile, blood samples were collected for serum Na+, K+, Ca2+ and Mg2+, which were reported as normal. Echocardiography and electrophysiological studies were carried out later, which were normal. Besides, subsequent ECGs were also normal []. Patient received tab bisoprolol 5 mg prior to each cycle and didn’t develop any arrhythmia in subsequent cycles.

[[55.0, 'year']]

M

{'28766171': 1, '29310415': 1, '29534446': 1, '20025547': 1, '29521316': 1, '33408748': 2, '21034734': 1, '27113369': 1, '7201366': 1, '29644475': 1, '29434790': 1, '21595514': 1, '33437376': 1, '27717837': 1, '28764226': 1, '375794': 1, '21878427': 1, '24604969': 2}

{'7781763-1': 1, '7781763-2': 1}

3932609-1

noncomm/PMC003xxxxxx/PMC3932609.xml

Flagellate erythema induced by bleomycin toxicity

A 15-year-old boy diagnosed as intracranial germ cell tumor underwent surgery, followed by cranial radiotherapy (45 Gy) and later treated with Bleomycin, Etoposide and Cisplatin(BEP) regimen. After third cycle of chemotherapy, he developed asymptomatic erythematous rashes over the back, chest and thighs, which later subsided with persistence of hyperpigmentation [].\nBleomycin is a chemotherapeutic antibiotic associated with various skin related toxicities such as Raynaud's phenomenon, flagellate erythema, and sclerodermoid reaction. In the acute stage they are erythematous rashes later may persist as hyperpigmentation marks.[] Other conditions which may be associated with flagellate erythema are docetaxel, adult Still's disease, dermatomyositis and ingestion of shiitake mushrooms.[] During the acute phase, topical with or without oral steroids may reduce symptoms.

[[15.0, 'year']]

M

{'27688147': 1, '21127762': 1, '7528043': 1, '24604971': 2}

{}

3932756-1

noncomm/PMC003xxxxxx/PMC3932756.xml

Acupuncture treatment for hypertension: a case study

A 56-year-old man (height 165 cm, weight 75 kg) was admitted to The First Affiliated Hospital of Tianjin University of Traditional Chinese Medicine on 24 September 2012 with hypertension and side effects of antihypertensive drugs. He was diagnosed with hypertension in 2007 and his usual BP was 160/100 mm Hg. He started taking levamlodipine 5 mg once daily in March 2011. However, his BP remained the same and he reported side effects such as flushing and palpitations. He was switched to irbesartan 150 mg once daily in July 2012. His BP remained at 140/85 mm Hg but he reported side effects of diarrhoea, fatigue and decreased sexual function, among others.\nHe denied a family history of alcoholism and cardiovascular disease and did not have a preference for a salty diet. He exercised about 2 h a day.\nInvestigations revealed mild hypocalcaemia (2.11 mmol/L) and reduced high density cholesterol (0.88 mmol/L). Transcranial cerebral Doppler showed increased blood flow in the right intracranial internal carotid artery while the blood flow frequency spectrum in the other cerebral vessels was normal. Carotid artery ultrasound revealed a plaque with a thickness of 2.0 mm in the left common carotid artery wall, which accounted for 25.6% of the blood vessel. Cardiac colour ultrasound revealed no findings in left ventricular hypertrophy: interventricular septum thickness at end-diastole (IVSTd)=10.9 mm, left ventricular posterior wall depth (LVPWd)=9.7 mm, E/A=0.87, left ventricular mass index (LVMI)=112 g/m2, relative wall thickness (RWT)=0.39.\nIn view of the side effects of his medication, the patient asked for a course of acupuncture. He received a total of 60 sessions of acupuncture treatment over the course of 12 weeks (5–6 treatments per week) from 24 September to 24 December 2012. Each acupuncture treatment session lasted 30 min. The acupuncture was stopped because he no longer had side effects from the antihytertensive drugs and he decided to return to work.\nDuring the treatments, acupuncture points LI4, LI11, ST36 and ST9 were used bilaterally for antihypertensive purposes. Needles were inserted 0.8–1.0 cun perpendicularly. Twirling with tonifying or reducing manipulation was applied (tonifying or reducing manipulation is to inspire jing qi through proper acupuncture technique). LI11, ST36 and ST9 were needled with tonifying manipulation for 1 min at a frequency of 120–160 twirls/min; LI4 was needled with reducing manipulation for 1 min at a frequency of 40–60 twirls/min. CV4, CV6 and SP6 were also needled to treat adverse drug reactions (eg, diarrhoea, fatigue).\nThe acupuncturists who administered the treatment had at least 10 years of experience. Stainless steel needles (Suzhou, China), 0.25×40 mm, were employed at corresponding points. Appropriate stimulation was applied in order to attain the de qi sensation, which was associated with physiological changes that played an important role in the effects of acupuncture.\nThe patient received individualised acupuncture treatments for hypertension and for adverse drug reactions such as diarrhoea and fatigue. Frequently used antihypertensive acupuncture points as described in systematic reviews are LR3, LI11, GB20, ST36 and ST40.\nIn this case, the antihypertensive point selection was based on Traditional Chinese Medicine meridian theory. In traditional concepts, points located on the Yangming meridian, such as LI4, LI11 and ST36, can ‘reconcile qi and blood’. We also selected ST9 Renying, a meeting point of the Stomach and Gallbladder channels that regulates qi and blood. Located next to the carotid sinus, ST9 is the pressure sensor of the human body that regulates BP. Based on TCM theory, CV4, CV6 and SP6 were chosen to treat diarrhoea and fatigue.\nOffice BP (OBP) was taken at each visit using an automated sphygmomanometer while seated and repeated after a 5 min break. If the systolic BP (SBP) or diastolic BP (DBP) changed more than 5 mm Hg within the 5 min period, a third measurement was taken. The average value was recorded.\nIn addition to OBP taken with an automatic sphygmomanometer, 24 h ambulatory BP monitoring (ABPM) was also applied to measure the daytime and night time BP. As an adjunct to OBP, ambulatory measurements of BP can assist in identifying ‘white coat hypertension’ and predicting the risk of organ damage and cardiovascular disease.

[[56.0, 'year']]

M

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{}

3932847-1

noncomm/PMC003xxxxxx/PMC3932847.xml

Mutation in SNAP25 as a novel genetic cause of epilepsy and intellectual disability

The proband is a 15 y old female, delivered at 41 weeks of gestation. Early prenatal ultrasounds showed bilateral choroid plexus cysts in the brain which were no longer observed by 21 weeks of gestation. APGAR scores were 8 and 9 at 1 and 5 min, respectively. Birth weight was appropriate at 3360 g. The newborn physical exam identified hip dysplasia requiring a Pavlik harness for 4 mo.\nDevelopmentally, she was always delayed in reaching milestones. At 6 weeks of age, the proband’s mother was concerned that she was not making eye contact. At 2.5 mo she was unable to lift her head when prone. Generalized hypotonia was noted at 4 mo. She first sat unsupported at 3 y of age. She can now ambulate with assistance, but does so infrequently. She is non-verbal. She gained weight well from 0–5 mo, maintaining her weight between the 50th–75th centiles but dropped to the 5th centile by 8 mo. Head circumference was also maintained at the 50th centile from 0–5 mo but dropped to the 5th centile by 9 mo. There was no history of developmental regression, but rather a severe delay in development from early life. To optimize nutrition and increase her caloric intake, nasogastric feeds were started at 1 y of age followed shortly thereafter with gastrostomy tube placement.\nSeizures began at 5 mo of age with episodes of repetitive blinking, upward eye deviation and impaired awareness, and were later associated with bilateral kicking and arm abduction accompanied by eye deviation to either side. Video EEG monitoring revealed mild generalized slowing, 2–2.5 Hz generalized spike-and-slow wave complexes. Subsequent video EEG studies revealed (1) mild generalized slowing, (2) interictal partial and symptomatic generalized discharges, and (3) ictal partial and generalized seizures with left (95%) or right-sided (~5%) hemiclonic seizures and lateralized frontocentral discharges (~90% left-sided, ~10% right sided) as well as seizures with bilateral motor features at onset, including tonic-clonic seizures, with generalized discharges. She currently averages 3–10 focal clonic seizures per hour, with an average duration of 15 s. Tonic-clonic seizures occur once every two weeks. Her most recent video EEG showed frequent epileptiform discharges but did not comprise more than 30% of her waking or sleep background. Seizures remain refractory to more than eight antiepileptic drugs in up to three drug combinations, as well as a ketogenic diet and intravenous methylprednisolone trials.\nAt 5 y of age, she required bilateral varus rotation osteotomies of the proximal femurs. She required spinal fusion at 11 y of age for scoliosis.\nHer current neurological examination reveals that she is nonverbal, with global developmental delays. She can intermittently follow simple commands and make intermittent, but fleeting eye contact. Eye tracking is impaired. She is microcephalic with diminished oralmotor tone, excessive drooling and spastic quadriparesis.\nThe proband is an only child. There is no family history of birth defects, seizures, intellectual disability or consanguinity.\nBrain MRI at 8 mo showed delayed myelination, but no structural abnormalities. MR spectroscopy showed increased lactate, raising concern for a mitochondrial disorder but given normal cerebrospinal fluid lactate levels it was thought that the MR spectroscopy lactate peaks were secondary to frequent seizure activity.\nMetabolic screening including quantitative plasma amino acids, acylcarnitine profile, carbohydrate deficient transferrin electrophoresis, urine organic acid analysis, and cerebrospinal fluid neurotransmitters, amino acids, lactate and glucose were all normal. Sulfite oxidase testing for molybdenum cofactor deficiency was normal. Muscle biopsy pathology and electron microscopy, and mitochondrial oxidative phosphorylation enzyme activity in muscle were normal. Electromyogram was normal. Karyotype, subtelomeric FISH, and chromosome microarray were normal. Molecular genetic testing for MECP2, CDKL5, Angelman syndrome methylation, ACAT1, FOXG1, Mitochondrial DNA 3243 allele was negative, MERRF testing on muscle, 101 nuclear encoded mitochondrial genes, 53 epilepsy genes, and 64 genes associated with X linked intellectual disabilities were all normal.

[[15.0, 'year']]

F

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{}

3933023-1

noncomm/PMC003xxxxxx/PMC3933023.xml

Posterior reversible encephalopathy syndrome following an inadvertent dural puncture during an emergency laparotomy for ischemic colitis – a case report

A 62-year-old woman presented acutely with severe sudden onset left-sided abdominal pain and several episodes of diarrhea with blood mixed in with stool. Her past medical history included coeliac disease and hypertension that was well controlled with ramipril 10 mg daily. On admission, observations were: temperature 36.4°C, blood pressure 115/50 mmHg, heart rate 75 bpm, respiratory rate 16, and oxygen saturations of 99% breathing on air. Examination revealed localized peritonitis of the left abdomen, and rectal examination showed altered blood with no stool. A computed tomography (CT) scan of her abdomen and pelvis showed an appearance consistent with colitis involving the transverse and sigmoid colon, the distribution suggesting ischemic colitis. She was initially managed conservatively with analgesia and intravenous antibiotics. Flexible sigmoidoscopy the following day showed an ischemic splenic flexure, and the decision was made for the patient to undergo a laparotomy. Preoperatively, the patient remained normotensive.\nOn the second day of her admission, the patient was taken to theater for a laparotomy. A low thoracic epidural was attempted for postoperative pain relief; however, the procedure was abandoned after one attempt due to a dural tap. Consequently, the patient had general anesthesia with systemic opioid analgesia and a rectus sheath block. Intraoperative findings revealed an ischemic left colon and proximal sigmoid colon; therefore, she underwent a Hartmann’s procedure. Intraoperatively, the patient remained hemodynamically stable, with an average blood pressure of 120/60 mmHg. Postoperatively, the patient was commenced on a morphine patient-controlled analgesia for pain relief and was anti-coagulated for the ischemic colitis. Investigations towards underlying conditions likely to have caused the ischemic colitis were also carried out. Screening for presence of the anti-phospholipid antibody, JAK2 mutation, and thrombophilia proved negative.\nPostoperatively, the patient developed an ileus. This was managed conservatively with nasogastric drainage and intravenous fluids. Her blood pressure rose from an average of 150/80 mmHg on postoperative day 1 to 190/80 mmHg on postoperative day 3, despite remaining on her usual dose of ramipril. On postoperative day 3, the patient complained of a frontal headache and “cloudy vision.” The headache and rising blood pressure was initially thought to be due to postoperative pain and dural tap; on postoperative day 3, she was commenced on a second antihypertensive, amlodipine. On postoperative day 4, headache, severe hypertension, and worsening vision continued. Neurological examination revealed no focal abnormality; however, fundoscopy showed blurred discs but no retinal hemorrhages or exudates. The patient was commenced on bisoprolol and underwent an urgent CT head scan ().\nThe CT head scan revealed bilateral areas of low attenuation in the left occipital lobe and to a lesser extent in the right occipital lobe. There were also some patchy areas of low attenuation in the right and left cerebellar hemispheres. There was neither mass effect nor enhancement.\nA subsequent magnetic resonance imaging scan of the head and magnetic resonance venography () showed patchy white matter edema most markedly in the occipital lobes but extending anteriorly to reach the frontal lobes, consistent with PRES.\nDespite these findings, blood pressure continued to be controlled with oral medication. However, on postoperative day 6, the patient developed status epilepticus. Repeat head CT excluded an intracerebral bleed. The patient was loaded with intravenous phenytoin, intubated, and blood pressure was tightly controlled with a mean arterial pressure of 85 mmHg. The patient was extubated the following day. Blood pressure continued to be tightly controlled with regular antihypertensives, and she remained on phenytoin to prevent further seizures.\nThe patient continued to improve and was discharged 21 days postoperatively with blood pressure controlled (systolic readings of 120–130 mmHg) on her usual dose of ramipril plus 10 mg amlodipine daily. The only residual neurological defects that she experienced on discharge were minor visual disturbances and memory problems.

[[62.0, 'year']]

F

{'10080724': 1, '7282395': 1, '26106495': 1, '15997391': 1, '21716568': 2, '8623651': 1, '3178528': 1, '18084001': 1, '8559202': 1, '2925919': 1, '32655488': 2, '18403560': 1, '28367283': 1, '17717238': 1, '13318967': 1, '19151296': 1, '17110690': 1, '21276384': 1, '453603': 1, '28695050': 2, '1865208': 1, '24600245': 2}

{'5473084-1': 1, '5473084-2': 1, '3110298-1': 1, '7324723-1': 1}

3933713-1

noncomm/PMC003xxxxxx/PMC3933713.xml

Olanzapine use in a manic patient during second and third trimester pregnancy

A 31-year-old woman with a history of four psychiatric hospitalizations (for a depressive episode in 2002 and three manic episodes thereafter) visited our clinic in 2010 and began a course of valproic acid 500 mg/day and olanzapine 5 mg/day. She demonstrated stable progress and was in a euthymic state while discussing pregnancy. The clinical staff planned to taper out the mood stabilizer slowly, use only quetiapine and clonazepam just prior to pregnancy, and cease all medication by June 1, 2012. After one month, her pregnancy was confirmed.\nIn September 2012, the patient experienced nausea and vomiting and was admitted to the obstetric department for conservative care. She then complained of depressed mood, avolition, and loss of appetite in weeks 9–10 of gestation. After some time, the patient developed a manic episode with elated mood, decreased need for sleep, talkativeness, hyperactivity, and distractibility, at which time she was admitted to the inpatient psychiatric department (January 2013).\nAfter hospitalization, the clinical team decided to control the patient’s symptoms by close monitoring without pharmacotherapy. However, due to her persistent irritability and violent behavior, it was decided that overall harm to the fetus from the disorder exceeded that from pharmacotherapy. Electroconvulsive therapy, which the clinical team considered as the first-line treatment, was not carried out due to her family’s refusal. Therefore, a course of olanzapine, which was shown to be effective in previous years, was reinstituted at week 25 of gestation with a dose of up to 7.5 mg/day. However, she was discharged early, even with symptom persistence, due to the family’s demands. Despite using a 7.5 mg/day dose of olanzapine, she was readmitted in March 2013 (week 33 of gestation) for persecutory delusions and delusions of infidelity in addition to manic symptoms.\nDespite amelioration of the delusions after taking olanzapine up to 20 mg/day, the patient still continued to show irritability and behavioral disturbances. Therefore, during week 35 of gestation, her medication was switched from olanzapine to quetiapine. Her sleep duration lengthened and she showed some improvements in irritability and hyperactivity afterwards with 800 mg/day of quetiapine, but still appeared to be talkative and sometimes temperamental. She was found to have oligohydramnios on follow-up sonogram, and emergency cesarean section was carried out at week 37 of gestation. The patient experienced no postsurgical complications, and the newborn’s Apgar score was 9. The baby was deemed to be healthy by the pediatrician and there were no congenital malformations.\nBeginning the day after surgery, the patient was started on valproic acid 1,000 mg/day and quetiapine 800 mg/day and showed improvement in her irritability, loss of impulse control, and distractibility. After agreeing not to breastfeed, the valproic acid was increased to 1,500 mg/day based on blood levels of 44.53 μg/mL while taking 1,000 mg/day. She tolerated the medication even after quetiapine was increased to_1,200 mg/day; she showed stable progress and was discharged on the 59th inpatient day.

[[31.0, 'year']]

F

{'22938889': 1, '16683078': 1, '12085677': 1, '11099738': 1, '18056236': 1, '10917399': 1, '11769820': 1, '14571052': 1, '1346886': 1, '15056503': 1, '23137820': 1, '15816786': 1, '17151155': 1, '12861154': 1, '15928351': 1, '17764378': 1, '18378767': 1, '17671284': 1, '12004800': 1, '11925316': 1, '24196828': 1, '24570586': 2}

{}

3934607-1

noncomm/PMC003xxxxxx/PMC3934607.xml

Gastrointestinal Endometriosis Causing Subacute Intestinal Obstruction with Gradual Development of Weight Loss and Misdiagnosed as Irritable Bowel Syndrome

A 44-year-old woman noted the onset of upper and lower abdominal pain associated with constipation approximately 4 years ago. Her symptoms were worse following meals, especially if they included fried foods, fatty foods, coffee or carbonated beverages. Episodes occurred every 2–3 days with occasional emesis but no history of hematemesis, melena or hematochezia. Her appetite remained good, but she noted a weight loss of 25 lb over the past year. There was no history of alcohol, coffee, aspirin or non-steroidal inflammatory medication abuse. She denied dyspareunia or painful menstrual periods. Her only operations were a cesarean section in 2003 and cholecystectomy in 2004.\nOn physical examination she appeared to be well-developed, well-nourished and in no acute distress. Her abdomen was non-tender and without palpable masses, organomegaly or distention. Right upper quadrant laparoscopy and suprapubic surgical scars were noted. Rectal examination demonstrated no masses or tenderness, with brown guaiac negative stool present in the rectal ampulla. Papanicolaou smear and gynecological examination performed by her gynecologist 1 month previously were normal. Her diagnostic work-up included normal complete blood count, liver and kidney function tests, thyroid function and C-reactive protein. Upper endoscopy and colonoscopy examinations were normal except for internal hemorrhoids. Abdominal and pelvic sonography were normal except for evidence of a prior cholecystectomy, mild fatty liver and a simple right ovarian cyst. A lactose tolerance test was positive.\nOn the basis of her symptoms and negative diagnostic work-up, the diagnoses of IBS, gastroesophageal reflux and lactose intolerance were made. Treatment with antacids and an anti-cholinergic did not provide relief. Due to her recurrent symptoms, a Computed tomography enterography examination was performed to evaluate the small intestine. This revealed a partial small bowel obstruction located approximately 5 cm proximal to the ileocecal valve (fig. ). Subsequent surgical resection and histology identified the cause of the partial obstruction to be endometriosis with focal submucosal hemorrhage and serosal scarring (fig. , fig. ). The patient became totally asymptomatic postoperatively.

[[44.0, 'year']]

F

{'22720303': 1, '18528943': 1, '29033768': 2, '12454865': 1, '22997597': 2, '18549937': 1, '15618831': 1, '30705873': 2, '23066308': 1, '15341718': 1, '21297534': 1, '20846366': 2, '15967329': 1, '22891009': 1, '12425553': 1, '16803612': 1, '24574950': 2}

{'6328977-1': 1, '3446655-1': 1, '2949747-1': 1, '5624264-1': 1}

3934608-1

noncomm/PMC003xxxxxx/PMC3934608.xml

A Rare Case of Retinal Artery Occlusion in the Context of Mediterranean Spotted Fever

A 55-year-old healthy Caucasian female with no history of ocular disease was observed in the emergency department with fever, myalgia, nasal congestion with 3 days of evolution, and a generalized rash for 1 day. She had contact with dogs. Physical examination revealed a cooperating and well-oriented patient with stable vital parameters and a tympanic temperature of 38.2°C. There was a maculopapular rash on the upper limbs, abdomen, and the dorsal region. A tache noir, a small crust surrounded by a violet halo, was visible in the region of the left thigh. There were no meningeal signs and no cardiorespiratory or abdominal changes. Serology for R. conorii was positive with an IgM titer of 1/320. Autoimmune and thrombophilia studies were negative. Hypercholesterolemia and systemic hypertension were excluded. The patient was admitted to our department and therapy with oral doxycycline 100 mg every 12 h was initiated. Oral doxycycline was given for 10 days.\nOn the first day of hospitalization, the patient reported decreased visual acuity (VA) of the right eye (RE). A CT scan was performed which was normal. No ophthalmologic examination was requested.\nThere was a systemic improvement and the patient was discharged after 6 days. Five days thereafter, the patient returned to the emergency room with complaints of worsening of RE VA. Observation revealed an RE VA of 20/100 and there was a superior temporal artery branch occlusion with perivascular sheathing in the ocular fundus as well as cotton wool spots and edema (fig. ). The retinal edema affected the macular region (fig. ). Campimetric examination revealed an inferior nasal scotoma, consistent with fundus lesions (fig. ).\nIntravitreal injection of triamcinolone acetonide 3.2 mg/0.08 ml was given only once and showed significant improvement of the retinal edema and a recovery of RE VA to 20/25 after 1 month. The vascular sheathing and the occlusion persisted (fig. ), and so did the corresponding scotoma in the visual field.\nThe total follow-up period is 1 year. No ocular side effects related to intravitreal triamcinolone were detected.

[[55.0, 'year']]

F

{'11037929': 1, '10892433': 1, '15019331': 1, '1554861': 1, '32952967': 1, '19319229': 1, '9059259': 1, '22034560': 1, '18084718': 1, '24596555': 2}

{}

3934610-1

noncomm/PMC003xxxxxx/PMC3934610.xml

Solitary Idiopathic Choroiditis

An asymptomatic 26-year-old previously healthy white male was referred to us following a routine optometrist visit. Visual acuity was 6/6 in both eyes, with no sign of anterior segment inflammation, but with the presence of a single, inactive, yellow choroidal lesion, 0.75 mm in diameter, located inferior to the left optic disc (fig. ).\nTime domain OCT of the lesion showed thinning and compression of the overlying choroidal vasculature with an apparently normal retina above it. Spectral domain OCT with enhanced depth imaging (fig. ) helped locate the lesion on the choroid and sclera, under the attenuated choroidal vasculature, without evidence of disruption of the outer or inner retinal layers and the retinal pigment epithelium (RPE). FAF (fig. ) revealed a hyperautofluorescent area corresponding with the mass.\nUltrasound B-scans failed to show any elevated lesion or thickening of the ‘coats’ since conventional B-scans cannot separate choroidal from scleral layers. Also, there was no evidence of the sub-Tenon fluid and the T-sign was negative.\nThe common systemic causes of a choroidal granuloma were tested for sarcoidosis (serum angiotensin-converting enzyme and calcium) and tuberculosis (QuantiFERON Gold), and both were negative. Other less common causes of SIC such as Toxocara, Brucella and Wegener's disease were excluded either by anamnesis and/or additional tests. Also, inflammatory markers (erythrocyte sedimentation rate and C-reactive protein) were within normal limits and the chest X-ray showed no abnormalities. Taking the funduscopic appearance and the image characteristics on the OCT and FAF into account, the diagnosis of SIC was made.

[[26.0, 'year']]

M

{'4034179': 1, '33750335': 2, '32971610': 2, '9258222': 1, '1555132': 1, '12578786': 1, '7335317': 1, '7162778': 1, '3051466': 1, '9261313': 1, '6720813': 1, '23246121': 1, '6719335': 1, '2401424': 1, '6671099': 1, '33796517': 1, '9747695': 1, '7422265': 1, '3897938': 1, '11879134': 1, '24575031': 2}

{'7727985-1': 1, '7942170-1': 1}

3934611-1

noncomm/PMC003xxxxxx/PMC3934611.xml

Malignant Melanoma with Probable Smooth Muscle Differentiation

A 63-year-old woman presented with an ulcerated reddish nodule on the left toe (fig. ). No pigmentation was observed in the lesion. The patient had no other pigmented lesion, which was suspected as malignant melanoma. Leiomyosarcoma was highly suspected based on an incisional biopsy performed at another hospital; thus, the patient was referred to our institution. Subsequently, the left toe was amputated. Histopathologically, the neoplasm occupied the entire dermis (fig. ) and was composed of pleomorphic spindle cells with eosinophilic cytoplasm, blunt-ended nuclei, and perinuclear vacuoles (fig. ). Mitosis was frequently observed (47 mitosis/10 high-power fields). Alfa smooth muscle actin (α-SMA; fig. ) and desmin (fig. ) were diffusely positive. S-100 was focally positive (fig. ). At that time, these findings suggested a diagnosis of cutaneous leiomyosarcoma. Seven months after the amputation, the patient presented with left groin lymph node swelling. Analysis of the excised lymph node showed replacement of lymph node tissue with a cytologically uniform eosinophilic spindle cell neoplasm (fig. ). Immunohistochemically, the spindle cells were diffusely positive for S-100 (fig. ) and α-SMA (fig. ). Staining for microphthalmia transcription factor (MiTF; fig. ) and HMB45 (fig. ) was focally positive. Melan-A-positive cells were scarcely observed (fig. ). Since whole tissues were embedded in paraffin block, electron microscopic analysis could not be performed. Finally, we made the diagnosis of spindle cell melanoma with probable smooth muscle differentiation. Two years after lymph node dissection followed by 5 courses of chemotherapy (dacarbazine, nimustine hydrochloride, vincristine sulfate, and interferon beta), no local recurrence or distant metastasis was observed.

[[63.0, 'year']]

F

{'8915854': 1, '30374898': 1, '27047932': 1, '31684113': 1, '8944620': 1, '17172493': 1, '10792480': 1, '17825057': 1, '24575006': 2}

{}

3934612-1

noncomm/PMC003xxxxxx/PMC3934612.xml

Recurrent Upside-Down Stomach after Endoscopic Repositioning and Gastropexy Treated by Laparoscopic Surgery

An 88-year-old woman with periodic vomiting and inability to eat was admitted to our hospital on three separate occasions where she received conservative treatment for a few days. Upside-down stomach was diagnosed. Because of her advanced age, complications (bedridden state, left hemiplegia after cerebral infarction, heart failure) and poor general condition, the minimally invasive method of endoscopic repositioning and gastropexy was performed. The stomach was reduced to the normal anatomic position, and the anterior stomach wall was fixed to the abdominal wall in three places as widely as possible (fig. ). She became symptom-free and was discharged on postoperative day 12.\nThree months later, she was hospitalized again because of vomiting. Chest and abdominal computed tomography showed the gastric fornix in the abdominal cavity, but the gastric corpus was above the diaphragm with organoaxial volvulus (fig. ). She was then diagnosed with a recurrent upside-down stomach after endoscopic repositioning and gastropexy. She was initially managed conservatively and had a nasogastric tube inserted, but her condition did not improve.\nAlthough her anterior stomach wall had been fixed to the abdominal wall in three places as widely as possible in the last hospitalization, she again presented with upside-down stomach. We concluded that it was impossible to prevent the recurrence of an upside-down stomach by endoscopic therapy. Therefore, we treated the condition with a laparoscopic repair of hernias and gastropexy.\nUsing a laparoscope, we observed that the anterior stomach wall was adhered to the abdominal wall in three places from the antrum to the lower corpus of the stomach (fig. ). The upper corpus of the stomach was rotated and herniated into the esophageal hiatus (fig. ). The herniated part of the stomach was repositioned easily. However, it quickly returned into the esophageal hiatus with rotation because of the adhesion between the omentum and the esophageal hiatus (fig. ). This adhesion was peeled off and the crura were closed by a primary suture (fig. ). The cardial part of the stomach wall was fixed to the crura, and the anterior stomach wall was fixed in three places to the abdominal wall widely from the upper corpus to the antrum (fig. ).\nOral ingestion was started the day after surgery. After rehabilitation, the patient was discharged 18 days after the operation. The suture used for fixing the stomach wall was removed 44 days after the operation. Three months after removal of the suture, the patient remained asymptomatic.

[[88.0, 'year']]

F

{'8379715': 1, '5647546': 1, '23238375': 1, '23711265': 1, '16151684': 1, '33376618': 1, '23364700': 1, '8338098': 1, '4043688': 1, '14585430': 1, '19067074': 1, '33364036': 2, '10801022': 1, '23579532': 1, '28275016': 1, '22874707': 1, '25320536': 1, '24574947': 2}

{'7750025-1': 1}

3934613-1

noncomm/PMC003xxxxxx/PMC3934613.xml

Successful Treatment of Congenital Lymphangioma Circumscriptum of the Vulva with CO2 and Long-Pulsed Nd:YAG Lasers

A 16-year-old Japanese girl presented with a 9-year history of vesicles on both sides of the vulva (fig. ). These lesions had developed at around 7 years of age and no treatment had been administered. The patient sought treatment at our hospital because of increased lymphatic leakage. Clinical examination revealed grouped vesicular lesions on the vulva, giving it a typical ‘frog spawn’ appearance. The medical history of the patient showed no other dermatologic or systemic abnormalities, and no family history of similar lesions was evident. A biopsy was taken from the middle of the affected area. Histopathologic examination revealed multiple thin-walled, dilated lymphatic spaces that contained lymphocytes and erythrocytes in the papillary dermis (fig. ). Clinical and pathologic findings were consistent with a diagnosis of LC.\nWe decided to use different kinds of lasers for the right and left lesions. Once a month, we treated the right-side vulval lesion with a CO2 laser (SHARPLAN 30C, Lumenis, Yokneam, Israel) and the left-side lesion with a long-pulsed Nd:YAG laser from the XeoTM multi-platform device (Cutera, Brisbane, Calif., USA) under local anesthesia with 1% lidocaine plus 1:100,000 epinephrine. The CO2 laser was set to a wavelength of 10,600 nm at 4 W with a 1-mm spot size. Visually confirming cauterization of lymphatic channels on the surface, we treated the lesions little by little every month, as treatment of a wide area in a single session may lead to ulceration. The long-pulsed Nd:YAG laser was set to a wavelength of 1064 nm with a fluence of 80–100 J/cm2 at 20 or 30 ms and a 7-mm spot size. Energy and pulse length were adjusted to achieve only partial coagulation.\nAfter the fourth session, the region on the right vulva treated with the CO2 laser was greatly improved, with no lesions visible to the naked eye. The region on the left vulva was greatly improved after only 3 treatments using the Nd:YAG laser. The patient preferred the results from the Nd:YAG laser as lymphatic fluid discharge was temporarily increased (for 1 week) after each CO2 laser irradiation, but stopped after Nd:YAG laser irradiation. However, the second session of irradiation with the Nd:YAG laser resulted in the development of a 4-mm linear scar after 1 week, which appeared as a slight textural change that disappeared within 3 months.\nAfter completion of the laser treatment (4 sessions), the patient no longer needed to use incontinence diapers and her quality of life improved considerably. No recurrence has been detected yet, 2 years after the last treatment, although follow-up is continuing (fig. ).

[[16.0, 'year']]

F

{'27331134': 1, '34712579': 1, '831620': 1, '26156111': 1, '15928635': 1, '16808671': 1, '27329721': 2, '16792643': 1, '26167062': 2, '16681790': 1, '24575003': 2}

{'4481748-1': 1, '5217005-1': 1, '5217005-2': 1}

3934614-1

noncomm/PMC003xxxxxx/PMC3934614.xml

nab-Paclitaxel in Combination with Carboplatin for a Previously Treated Thymic Carcinoma

A 40-year-old man was admitted to our hospital in May 2013 because of progressive right-sided back pain. The patient had been surgically diagnosed with squamous cell carcinoma of the thymus in July 2009 (designated as stage II according to the Masaoka classification []) and had received postoperative thoracic irradiation. However, computed tomography (CT) scans of the chest in May 2010 showed pleural dissemination in the right thorax. The patient worked as an engineer and requested to avoid paclitaxel-induced neuropathy at that time. Therefore, he received combined chemotherapy consisting of cisplatin and docetaxel, and achieved a minor response. After the patient became refractory to this regimen, various antitumor agents, including gemcitabine, S-1, and amrubicin, were administered. However, pleural dissemination recurred and the patient's back pain gradually worsened, even while using opioids.\nUpon admission to our hospital, CT scans of the chest showed multiple pleural disseminated nodules with a pleural effusion in the right thorax (fig. ). The serum cytokeratin 19 fragment (CYFRA) level was increased (13.9 ng/ml; normal level <3.5 ng/ml). The patient received nab-paclitaxel in combination with carboplatin as described in a previous phase III trial []. He experienced grade 4 neutropenia, according to the NCI-CTCAE (National Cancer Institute – Common Terminology Criteria for Adverse Events) version 4.0, 2 weeks after beginning chemotherapy. Therefore, the nab-paclitaxel administration was omitted on day 15, and the patient received granulocyte colony-stimulating factor (G-CSF) for several days. The severity of thrombocytopenia and nonhematological toxicities such as reversible neuropathy did not exceed grade 1 during the treatment. Objective tumor shrinkage was obtained after 4 cycles of chemotherapy, and the pleural effusion disappeared (fig. ). The serum CYFRA level decreased to 1.7 ng/ml, and the patient's back pain was relieved without any opioids.

[[40.0, 'year']]

M

{'21742653': 1, '19409644': 1, '28868014': 2, '34581013': 1, '16489089': 1, '12963221': 1, '33564291': 2, '20629617': 1, '34771601': 1, '27123268': 1, '7296496': 1, '22547591': 1, '24575009': 2}

{'5567109-1': 1, '7841741-1': 1, '7841741-2': 1}

3934615-1

noncomm/PMC003xxxxxx/PMC3934615.xml

Two Case Reports of Resensitization to Previous Chemotherapy with the Novel Hypoxia-Activated Hypomethylating Anticancer Agent RRx-001 in Metastatic Colorectal Cancer Patients

A 62-year-old man with a chief complaint of hematochezia was diagnosed with KRAS wild-type cancer of the sigmoid colon in 2007. The patient was initially treated with FOLFIRI+bevacizumab, and when the disease progressed, he received several therapies both alone and in combination: cetuximab, bevacizumab, capecitabine, gemcitabine, oxaliplatin, axitinib and irinotecan. On enrollment in the phase-1 study of the experimental drug RRx-001, a computed tomography (CT) scan revealed multiple metastases in the liver, bone and lungs. The patient received RRx-001 at a dose of 10 mg/m2 weekly over a period of approximately 9 months, receiving a total of 39 doses. RRx-001 was well tolerated, with the only treatment-related toxicity being pain on infusion that was readily managed. During this time, the patient had stabilization of his disease and was essentially asymptomatic. In addition, his ECOG performance status improved from 1 to 0, and he not only returned to full-time employment but also began to exercise vigorously.\nAfter nearly 10 months of treatment with RRx-001, he had progressive disease with elevation of the carcinoembryonic antigen (CEA) and increased tumor size, and was therefore discontinued from the trial. During the 2-week period between discontinuation of RRx-001 and resumption of a new therapy, there was a rapid increase in the slope of his CEA levels, suggesting that RRx-001 had suppressed tumor growth even after he met the criteria for progression.\nThe principal investigator (T.R.) restarted FOLFIRI, and the patient was treated for approximately 5 months, resulting in a sharp decline in the CEA levels from 511 to 162 (fig. ). After 5 months of therapy, a rise in CEA (table ) and a CT scan confirmed progression, and the treatment regimen was discontinued.

[[62.0, 'year']]

M

{'26125013': 1, '26678620': 1, '26933421': 2, '28978082': 1, '25389457': 1, '29254266': 1, '26280276': 1, '27229330': 1, '24893730': 1, '26296953': 1, '21358960': 1, '27330954': 1, '27377482': 1, '27084426': 1, '22699395': 1, '25674537': 1, '26841903': 1, '26657731': 1, '22589277': 1, '34880756': 1, '21037809': 1, '26280277': 1, '24575021': 2}

{'3934615-2': 2, '4772628-1': 1}

3934615-2

noncomm/PMC003xxxxxx/PMC3934615.xml

Two Case Reports of Resensitization to Previous Chemotherapy with the Novel Hypoxia-Activated Hypomethylating Anticancer Agent RRx-001 in Metastatic Colorectal Cancer Patients

A 66-year-old male with unresectable mCRC was treated with the FOLFOX therapy as first-line chemotherapy and with FOLFIRI combined with cetuximab upon progression after first-line therapy. Although the patient achieved a response to this regimen, the course was complicated by a persistent grade 2/3 skin rash. After 6 months on the second-line regimen, his disease again progressed, and the patient entered into the RRx-001 phase-1 clinical trial, receiving RRx-001 at 83 mg/m2 once weekly, which he tolerated well. His disease stabilized for 6 months before progressing again (fig. , fig. ). In the interval between stopping RRx-001 and restarting treatment (approx. 40 days), his disease progressed significantly, suggesting that RRx-001 had suppressed tumor growth even after he met the criteria for progression. Given the lack of other effective options, he was rechallenged with FOLFIRI, resulting in moderate tumor shrinkage after only 4 weeks and improvement of symptoms. The treatment of this patient is currently ongoing.

[[66.0, 'year']]

M

{'26125013': 1, '26678620': 1, '26933421': 2, '28978082': 1, '25389457': 1, '29254266': 1, '26280276': 1, '27229330': 1, '24893730': 1, '26296953': 1, '21358960': 1, '27330954': 1, '27377482': 1, '27084426': 1, '22699395': 1, '25674537': 1, '26841903': 1, '26657731': 1, '22589277': 1, '34880756': 1, '21037809': 1, '26280277': 1, '24575021': 2}

{'3934615-1': 2, '4772628-1': 1}

3934617-1

noncomm/PMC003xxxxxx/PMC3934617.xml

Primary Synovial Sarcoma of the Thyroid Gland: Case Report and Review of the Literature

The patient was a 55-year-old Caucasian male with a personal medical history of high blood pressure and smoking. In December 2012, he underwent a cervicothyroid ultrasonography because of a rapidly growing palpable neck mass for 1 month, associated with dysphagia. Physical examination revealed a 7-cm, firm, macronodular, fixed, dipping thyroid mass. No cervical adenopathy was palpable. The WHO performance status was equal to 0. Ultrasonography revealed a 7-cm mass of the left thyroid lobe which was heterogeneous, hypervascularized, and mixed (solid and liquid). Biological thyroid tests, including serum tumor markers, were normal.\nBecause of suspicion of anaplastic thyroid carcinoma, a total thyroidectomy was planned in February 2013, without any preoperative cytology. Intraoperatively, the right thyroid lobe was hypertrophic, firm, but not nodular. The left lobe was under tension, ovalized, and enlarged (more than 12 cm) and extended from the top of the neck to the superior mediastinum. Its mobilization was difficult and led to capsular rupture and effusion of necrotic, hemorrhagic, and gelatinous liquid. Perioperative pathological analysis of tumor fragments showed a spindle cell proliferation suggesting sarcoma. Thyroidectomy was performed without lymph node dissection. Because of perioperative rupture and tumor fragmentation, the resection was classified as incomplete (R2). The postoperative course was uneventful.\nPathological examination (fig. ) of multiple white-tan tumor fragments (between 2 and 3.5 cm each) revealed a dense cellular proliferation composed of ovoid-shaped to frankly spindle-shaped cells, without overt nuclear atypia, and disposed in a fascicular pattern. The mitotic index was moderate, inferior to 10 mitoses per 10 high-power fields. A rich vasculature was composed of branching vessels with a staghorn or hemangiopericytoma-like pattern. There were large hemorrhagic areas, without actual necrosis. On immunohistochemistry (IHC), tumor cells stained heterogeneously but strongly for epithelial membrane antigen (EMA). They were negative for pan-cytokeratin (AE1-AE3), CD34 (only staining the rich vasculature), desmin, H-caldesmon, PS100, HMB-45, and MDM2 antibodies. The diagnosis of monophasic SVS was suspected, and slides were sent to our expert institution for confirmation. Molecular analysis, performed on formalin-fixed paraffin-embedded tissue by fluorescent in situ hybridization (FISH), showed a SYT gene rearrangement, highly consistent with the diagnosis of a monophasic SVS, fibrous type. The histologic grade was 2 in the FNCLCC system (differentiation 3, mitosis count 1, and necrosis 0).\nThe patient was thus referred to our institution. Only 15 days after surgery, clinical examination detected a local disease evolution with palpation of a 4-cm neck mass, associated with dysphagia and pain. Whole-body computed tomography (CT) scan revealed a 5-cm mass, in contact with the frontal parts of the left primitive carotid artery and internal jugular vein and the thyroid cartilage. No lymphadenopathy or distant metastasis was present. Cervical magnetic resonance imaging (MRI) confirmed a 4.2-cm mass in contact with large vessels and likely invasion of the paralaryngeal fat tissue above the thyroid cartilage (fig. ). Because of immediate postoperative relapse, local extension, and known relative chemosensitivity of SVS, we delivered primary chemotherapy combining doxorubicin and ifosfamide, before possible surgery. After 3 cycles, MRI and CT scan showed tumor size stabilization, with a small volume reduction (4 × 3 × 5.5 vs. 4.5 × 3 × 6 cm), associated with extension of necrosis within the mass (60 vs. 20% before chemotherapy). MRI also showed a decrease in fat invasion and contact with the large vessels, whereas perichondrium invasion of the thyroid cartilage remained stable. Three additional chemotherapy cycles were thus delivered. After the last cycle, MRI and CT scan showed small tumor reduction (4 × 2.5 × 4.5 vs. 4 × 3 × 5.5 cm) with further extension of tumor necrosis and improvement of tumor contact with the internal carotid artery and the internal jugular vein, without a resectability margin relative to the thyroid cartilage (fig. ). Chemotherapy was stopped and the patient was operated on. After discussion with the patient, it was decided to perform conservative surgery, without laryngectomy. Tumor resection was done including resection of the previous cutaneous scar, prelaryngeal muscles, thyroid cartilage perichondrium, superior horn of the thyroid cartilage, and left longer horn of the hyoid bone. An additional resection was performed in the thyroidectomy area with removal of fat and lymph nodes in the upper mediastinum. Ipsilateral cervical lymph node dissection was also performed. During the procedure, the left inferior laryngeal nerve was resected, but all other vascular and nervous elements could be preserved (fig. ). No postoperative complications occurred and the patient was discharged after 5 days. Pathological examination of the operative specimen found a 7-cm monophasic SVS. Unfortunately, the resection margins (left cricothyroid muscle) were microscopically involved by tumor cells. No further mutilating surgery was chosen and treatment was completed with ongoing adjuvant radiotherapy.

[[55.0, 'year']]

M

{'31560833': 1, '9865642': 1, '27026769': 1, '30758754': 1, '12789939': 1, '29259650': 2, '23456589': 1, '17923744': 2, '22232717': 2, '24575008': 2}

{'3250586-1': 1, '2694401-1': 1, '5721497-1': 1}

3934618-1

noncomm/PMC003xxxxxx/PMC3934618.xml

A Case of a Giant Growing Serous Cystic Neoplasm of the Pancreas

In 2003, during a routine health checkup, a pancreatic cyst measuring 8 cm in diameter was detected in a 51-year-old woman. The cyst was subsequently managed with follow-up care by the previous physician on an outpatient basis. In 2010, the cyst had increased to 12 cm in size and then to 19 cm within approximately the next 6 months. At this time, the patient was referred to our hospital for treatment. At the initial visit, she reported abdominal bloating. However, no medical problems were evident from the patient's health history, and she had no history of pancreatitis, abdominal trauma or alcohol intake. A physical examination showed no sign of anemia in the palpebral conjunctiva or jaundice of the bulbar conjunctiva; the abdomen was soft, flat, and nontender, and no obvious tumor mass was detected by palpation. Blood tests revealed no elevation of pancreatic enzyme levels or tumor markers. Abdominal ultrasonography and contrast-enhanced abdominal computed tomography (CT) scans showed the presence of a partially septated giant cystic lesion measuring 20 × 11 × 13 cm in the abdominal cavity (fig. ). The lining membrane of the cyst was thin, and no solid components or calcifications were noted in the cyst. A beak sign was observed in the pancreas, and the main pancreatic duct in the tail of the pancreas was slightly dilated. Therefore, the mass was considered a cystic tumor of pancreatic origin. On abdominal magnetic resonance imaging, the pancreatic body lesion detected by the CT scan was depicted as high-intensity area on T2-weighted images. These findings were consistent with the characteristics of cystic lesions (fig. ). A septum-like structure was present in the cyst, and dilation was noted in the pancreatic duct in the tail of the pancreas. Endoscopic ultrasonography showed extramural compression of the middle part of the gastric body. Although imaging studies of the same site could not reveal the entire extent of the cyst because of its enormous size, the cyst demonstrated low-level internal echoes and did not show any obvious solid component. A septum-like structure was observed, but there was no microcystic lesion with a characteristic honeycomb pattern. Endoscopic retrograde cholangiopancreatography displayed only the main pancreatic duct in the head of the pancreas; the cyst compressed the tail side. Based on these findings, the differential diagnosis included serous cystic neoplasm (SCN), mucinous cystic neoplasm, and branch duct-type intraductal papillary mucinous neoplasm in the neoplastic category as well as pancreatic pseudocyst in the nonneoplastic category. Because this mass was increasing in size and abdominal bloating was present, surgical resection was deemed appropriate. Therefore, a distal pancreatectomy with splenectomy was performed. The cystic content was serous fluid of a dark, muddy color, suggesting previous bleeding (fig. ). On histopathological examination, the lining membrane had a papillary structure composed of cuboidal cells with round nuclei. The cells stained PAS-positive and diastase PAS-negative, thus indicating the presence of glycogen (fig. ). These findings led to the definitive diagnosis of SCN.

[[51.0, 'year']]

F

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{}

3934671-1

noncomm/PMC003xxxxxx/PMC3934671.xml

Stereotactic Body Radiation Therapy as a Bridge to Transplantation and for Recurrent Disease in the Transplanted Liver of a Patient with Hepatocellular Carcinoma

A 52-year-old male, undergoing surveillance because of long-standing hepatitis C, was found to have a rising α fetoprotein (AFP) level and a new 1.7-cm liver lesion consistent with HCC on a CT scan in October 2007. He was placed on the transplant list and underwent transarterial chemoembolization as bridging therapy in February 2008. The AFP level declined to normal, but within several months began to rise again. In August 2008, the patient was referred for SBRT.\nThe Novalis ExacTrac patient positioning platform (BrainLab AG, Heimstetten, Germany) was used for immobilization. Treatment planning was performed using the BrainScan system (BrainLab AG) and BrainLab treatment software. Treatment was prescribed to the 100% isodose line, with the 80% isodose line covering the planning target volume. Conformal arcs were used to deliver 50 Gy in ten 5 Gy daily fractions using 6-MV photons.\nOn May 10, 2009, the patient underwent an OLT. Pathologic examination of the native liver showed a 2.2-cm necrotic mass with no viable tumor identified. He did well following transplant until March 2011, when a rise in AFP up to 328 IU/ml was found.\nA CT scan revealed an enlarged pericaval lymph node, which was biopsied and showed metastatic carcinoma. The lymph node was treated in May 2011 with SBRT to a dose of 50 Gy in 10 fractions. There was a good response to treatment based on repeat imaging studies as well as a significant decline in the AFP level.\nHowever, in August 2012 there was a rapid rise in the patient's AFP level, reaching 1,590 IU/ml by October. In an October 2012 CT scan, a new 2.2-cm mass was seen in the left lobe of the liver and was treated with SBRT, 62.5 Gy in 5 fractions (fig. ). At an initial follow-up after treatment, the lesion was stable on CT, and the AFP level had declined to 76 IU/ml. The most recent CT scan, almost 1 year after treatment, shows complete resolution of the treated lesion with no new suspicious disease. Follow-up examination also did not reveal any signs of liver disease or radiation-related toxicity (fig. ).

[[52.0, 'year']]

M

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{}

3934674-1

noncomm/PMC003xxxxxx/PMC3934674.xml

Spontaneous Healing of Corneal Perforation after Temporary Discontinuation of Erlotinib Treatment

A 65-year-old Japanese man was diagnosed in April 2010 with stage IV lung adenocarcinoma, manifesting multiple metastases in the lung, adrenal gland and brain. He received 31 Gy of basicranial radiation for the brain metastases. Given that he was found to harbor an epidermal growth factor receptor (EGFR) mutation, he was also treated with the EGFR-specific tyrosine kinase inhibitor (TKI) gefitinib (250 mg/day). As a result of the development of adverse effects including fatigue, dermatitis, and liver dysfunction, however, the dose of gefitinib was initially reduced by half and treatment with the drug was subsequently discontinued after a total of 2 months. Computed tomography and magnetic resonance imaging revealed that the size of the original lesion and the number of brain metastases had increased. Treatment with the combination of cisplatin (125 mg) and pemetrexed (0.75 mg) was initiated. After 8 weeks, this regimen was switched to vinorelbine (32 mg) and docetaxel (80 mg) because of the development of adverse effects. This latter treatment was also not tolerated well, and so administration of the EGFR-TKI erlotinib (150 mg/day) was initiated. The patient again experienced general adverse effects including dermal exanthema. At this time, he developed a foreign body sensation in both eyes and visited his local ophthalmologist, who referred him to the corneal service at Yamaguchi University Hospital for the treatment of corneal epithelial disorders with corneal thinning.\nSlitlamp microscopy revealed bilateral superficial punctate keratopathy and conjunctival epithelial disorders. Corneal ulceration on his right eye was also observed, but no signs of infection or inflammation were apparent. His visual acuity was 20/25 OD and 20/25 OS. Schirmer's test detected bilateral mild hypolacrimation (5 mm), and his corneal and conjunctival epithelial disorders were thus attributed to aqueous-deficient dry eye. We treated him by insertion of punctal plugs in both lower lacrimal puncta. Three weeks after plug insertion, his corneal and conjunctival epithelial disorders had apparently improved. However, 2 months after his first visit to our clinic, he was referred to us again for the treatment of bilateral corneal ulcers, which were confirmed by slitlamp microscopy (fig. ). Slight stromal edema, ulceration with an opaque epithelium, and a shallow anterior chamber were also observed in the right eye. The next day, the anterior chamber of the right eye was flat, and so we treated this eye with a bandage soft contact lens. The right anterior chamber remained flat, however, and the patient was diagnosed with noninfectious corneal perforation of unknown cause and was prepared for tectonic keratoplasty. The oral administration of erlotinib was interrupted in preparation for general anesthesia, and the patient was treated with gatifloxacin eyedrops only. Two days later, the anterior chamber of his right eye had spontaneously reformed and the epithelial defects of both eyes had healed (fig. ). The keratoplasty was therefore canceled and we monitored the patient carefully, but his corneal condition remained stable. Treatment with erlotinib at half the original dose was reinitiated and both eyes were maintained by the administration of artificial tears, with the punctal plugs remaining in place. Corneal ulceration or other corneal disorders did not recur.

[[65.0, 'year']]

M

{'7698260': 1, '33788182': 1, '3305411': 1, '22584020': 1, '28562552': 2, '34572058': 1, '29503911': 1, '19512896': 1, '23642754': 1, '2544455': 1, '32724688': 2, '29286080': 1, '33811466': 1, '16086962': 1, '21400063': 1, '11328735': 1, '11311043': 1, '415981': 1, '24575032': 2}

{'7366197-1': 1, '5459717-1': 1}

3934679-1

noncomm/PMC003xxxxxx/PMC3934679.xml

Sustained Complete Response after Maintenance Therapy with Topotecan and Erlotinib for Recurrent Cervical Cancer with Distant Metastases

A 53-year-old woman was diagnosed with stage IB2 poorly differentiated squamous cell carcinoma of the cervix with lymphovascular invasion. She received definitive pelvic radiation therapy with concurrent cisplatin chemotherapy followed by brachytherapy. Six months after completion of treatment, positron emission tomography-computed tomography (PET-CT) showed recurrent disease to the common iliac and para-aortic lymph nodes above the previous radiation field. The patient was treated with additional radiation therapy to the common iliac and para-aortic lymph nodes. Two years later, PET-CT showed a hypermetabolic left supraclavicular lymph node, and fine-needle aspiration showed poorly differentiated carcinoma consistent with cervical cancer recurrence. The patient received additional chemoradiation with cisplatin encompassing the left supraclavicular area, cervical regions, and upper mediastinum. She subsequently received 3 cycles of adjuvant cisplatin and topotecan. PET-CT showed a complete response. Given the patient's high likelihood of recurrence, she was offered maintenance therapy with erlotinib based on previous study findings in NSCLC. She received therapy with low-dose topotecan (2 mg/m2 intravenous every 14 days) combined with erlotinib (100 mg orally daily). PET-CT scans were performed every 3–4 months during maintenance therapy and were negative for disease. Maintenance therapy was well tolerated and produced minimal side effects including mild fatigue and nausea. After 20 months of maintenance therapy, the treatment was discontinued given the long interval without any evidence of disease. Since then, the patient has undergone PET-CT yearly. The patient is currently without evidence of disease 5 years after completing the topotecan-erlotinib maintenance therapy. Of note, EGFR mutational testing was not performed in this case.\nA copy of the written consent form is available for review by the Editor-in-Chief of this journal on request.

[[53.0, 'year']]

F

{'16014882': 1, '15337563': 1, '18657909': 1, '21296855': 1, '19574787': 1, '20493771': 1, '25842084': 1, '9770378': 1, '22285168': 1, '10100709': 1, '17452677': 1, '22949150': 1, '24575024': 2}

{}

3934680-1

noncomm/PMC003xxxxxx/PMC3934680.xml

Successful Concurrent Chemoradiotherapy with Cisplatin plus Vinorelbine for Locally Advanced Thymic Carcinoma

A previously healthy 53-year-old man was admitted to a local hospital because of an abnormality on chest radiography detected during health screening in October 2009. Chest computed tomography (CT) revealed an abnormal mass in the anterior and middle mediastinum (fig. ). 18F-fluorodeoxyglucose positron emission tomography (FDG-PET) showed positive uptake in the mediastinal mass and right supraclavicular lymph node (fig. ). The patient suddenly developed hoarseness, and left recurrent nerve paralysis was detected. He was referred to our hospital for further examination. Physical examination and laboratory studies revealed no specific findings. Endobronchial ultrasound-guided transbronchial needle aspiration was performed for the mediastinal mass. The histological findings revealed undifferentiated type of carcinoma and the tumor cells were positive for CD 5. These findings were consistent with thymic carcinoma. Brain magnetic resonance imaging (MRI) revealed no brain metastasis. According to the classification of Masaoka et al. [], the patient had advanced disease with supraclavicular lymph node metastasis (IVb).\nConcurrent chemoradiotherapy was selected as treatment. The chemotherapy regimen consisted of cisplatin (80 mg/m2, day 1) and vinorelbine (25 mg/m2, days 1 and 8) every 4 weeks and concurrent thoracic radiation therapy (2 Gy × 30 fractions, total 60 Gy). Partial response was achieved when 4 cycles of chemotherapy had been completed (fig. ). He has remained well for approximately 4 years without any evidence of relapse.

[[53.0, 'year']]

M

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{}

3934681-1

noncomm/PMC003xxxxxx/PMC3934681.xml

Concurrent Reactivation of Herpes Simplex and Varicella Zoster Viruses Confirmed by the Loop-Mediated Isothermal Amplification Assay

A 69-year-old man, with a background of recurrent outbreaks of herpes labialis approximately once every 2 years and childhood varicella, noticed vesicles on the lower lips. Five days later, a skin eruption developed on the right side of his face. Although he had no history of atopic dermatitis, Kaposi varicelliform eruption with secondary bacterial infection was suspected at another hospital, so he was treated with oral acyclovir and levofloxacin for 2 days. The patient subsequently presented to our outpatient clinic for consultation after the lack of symptomatic improvement.\nOn examination, erythema and small vesicles were present on the right side of the face in the dermatome innervated by the maxillary branch of the trigeminal nerve; the dorsum nasi, philtrum and upper lip were also affected. Crusting of these lesions was observed, and the right eyelids were red and swollen. Small vesicles were also noted at the bottom of the lower lip (fig. ). Vesicles with red halos were scattered on the trunk and extremities. Laboratory investigations revealed an elevated level of C-reactive protein of 2.4 mg/dl, suggesting a mild inflammatory reaction; however, other blood tests, including immunoglobulin levels and HIV antibody results, were either normal or negative. No evidence of immunological dysfunction was seen. A small number of coagulase-negative staphylococci were identified on bacterial culture of a swab taken from a skin lesion.\nThe differential diagnosis included disseminated herpes zoster, varicella, herpes simplex and impetigo. We performed the LAMP assay (Eiken Chemical, Tokyo, Japan) using swab samples taken from the following areas: an erosion on the right cheek, vesicles on the upper and lower lips (fig. ; arrows 1, 2 and 3) and a vesicle with a red halo on the right thigh. HSV-1 and VZV were detected by LAMP in samples taken from all 3 sites on the face, and VZV was detected in the sample taken from the vesicle on the right thigh. LAMP results were confirmed by real-time PCR (table ). Serologically, HSV-1- and VZV-specific IgG were positive. Based on these findings, the patient was diagnosed with facial herpes simplex and disseminated herpes zoster.\nThe patient was treated with acyclovir at 750 mg/day for 7 days and cefcapene pivoxil at 300 mg/day due to the possibility of secondary bacterial infection. After 7 days of treatment, the facial swelling improved and all facial vesicles had crusted. The eruption on the trunk and extremities also became crusted. Postherpetic neuralgia or ocular lesions did not complicate disease resolution.

[[69.0, 'year']]

M

{'11835389': 1, '15695716': 1, '15714482': 1, '6259942': 1, '15484264': 1, '18312326': 1, '29165905': 1, '12037117': 1, '15308854': 2, '9154763': 1, '11534912': 1, '10640851': 1, '16191900': 1, '32038653': 1, '7601930': 1, '10871386': 1, '24303946': 1, '24575004': 2}

{'2816897-1': 1}

3934682-1

noncomm/PMC003xxxxxx/PMC3934682.xml

A Case of Choroidal Neovascularization Secondary to Unilateral Retinal Pigment Epithelium Dysgenesis

An 8-year-old boy was referred to us because of a unilateral unique funduscopic appearance in the left eye (OS). Funduscopic findings revealed a geometric lesion at the RPE level of the interpapillomacular area, contiguous with the slightly hyperemic optic nerve (fig. ). The lesion was composed of marginal RPE hyperplasia and atrophic RPE in its center. Findings from the right fundus examination were normal. Based on these characteristic findings, the patient was diagnosed with URPED. Best corrected Landolt ring chart visual acuity (BCVA) was 1.0 in both eyes. Twenty-three months after the first visit, he presented with visual disturbance OS. Funduscopic examination showed enlargement of the lesion and development of subfoveal CNV (fig. ). FA findings revealed hypofluorescence from RPE hyperplasia, hyperfluorescence from RPE atrophy and dye leakage from the optic nerve and CNV (fig. ). Optical coherence tomography (OCT) revealed type 2 CNV, but the exudative change was not obvious (fig. ). The patient's BCVA was 0.4 OS. Two-time STTA (40 mg/1 ml) was performed at the onset of CNV and 6 months later. Additional IVB (1.25 mg/0.05 ml) was performed 10 months later for the treatment of CNV. Both STTA and IVB were performed with the written informed consent from both the patient and his father as well as with an approval of the institutional review board of Kobe University of Medicine and the Tenets of the Declaration of Helsinki. Despite the treatment, the geometric lesion and CNV were resistant to the treatment and expanded gradually. Seven years after the first visit, the geometric lesion and the CNV kept expanding steadily, and new CNV developed at the inferior retina (fig. ).

[[8.0, 'year']]

M

{'19733831': 1, '32002409': 1, '11934331': 1, '32631022': 2, '34041416': 1, '18971849': 1, '32819322': 2, '24575036': 2}

{'7441658-1': 1, '7338740-1': 1}

3934684-1

noncomm/PMC003xxxxxx/PMC3934684.xml

Optimizing Evaluation of Split Renal Function in a Living Kidney Donor Using Scintigraphy and Calculation of the Geometric Mean: A Case Report

We report the case of a 28-year-old healthy male applying to donate a kidney to his 61-year-old father. Preliminary assessment showed no contraindications. The computed tomography (CT) angiogram performed showed 2 short renal veins and 2 arteries of the right kidney (fig. ). Due to the complex anatomy and consequently increased risks for the donor and recipient, a living kidney transplantation of the right kidney was not considered by mutual agreement. However, the left kidney had a relative function of 60% in conventional posterior Tc99m-mercaptoacetyltriglycin (MAG3) scintigraphy (fig. ), and MAG3 total clearance was in the lower normal range (242 ml/min/1.73 m2). Considering that the position of the kidney (e.g. malrotation) determines the activity measured, a second scintigraphy using anterior and posterior Tc99m-dimercaptosuccinic acid (DMSA) scans was performed. As expected, the posterior DMSA scan confirmed the results of the posterior MAG3 scan: left kidney 60%. In the additional anterior scan, however, the left kidney now had a relative function of only 46% (fig. ). Calculating the geometric mean (✓ a × p), the adapted relative function of the left kidney was 53%, now meeting the inclusion criteria for living kidney donation. Confirmatory, CT-based renal cortex volume calculation was almost the same on both sides (97.23 and 97.31 ml on the left and right, respectively). After extensive education of the donor and recipient, live donor transplantation was carried out without any intra- or postoperative complications. Postoperative renal function (eGFR) of the donor was 62.59 ml/min/1.73 m2 one week after transplantation (vs. 124.13 ml/min/1.73 m2 preoperatively). Serum creatinine levels of the donor and recipient were normal at the time of discharge. Six months after transplantation, the eGFR of the donor and recipient were 90.75 and 73.64 ml/min/1.73 m2, respectively.

[[28.0, 'year']]

M

{'2395028': 1, '16894324': 1, '31144061': 1, '3893837': 1, '26286903': 1, '15785361': 1, '28634839': 1, '10321826': 1, '23770137': 1, '15446304': 1, '15657508': 1, '2822198': 1, '24575115': 2}

{}

3934695-1

noncomm/PMC003xxxxxx/PMC3934695.xml

Acute Macular Neuroretinopathy in a 15-Year-Old Boy: Optical Coherence Tomography and Visual Acuity Findings

A 15-year-old Japanese boy presented with a 6-day history of acute painless blurred vision and a small central scotoma in the left eye. Best corrected visual acuity (BCVA) was 0.8 and 0.3 in the right and left eyes, respectively. Intraocular pressure was 16 mm Hg in both eyes. The patient had no specific past medical history. No inflammatory cells were observed in the anterior segment or vitreous of either eye. Fundus examination revealed no specific abnormalities in the right eye (fig. ). In the left eye, there was a small dark area in the fovea (fig. , fig. ). OCT (RS-3000; Nidek, Japan) revealed no specific abnormalities in the right eye; however, in the left eye, disruption of the photoreceptor inner segment (IS)/outer segment (OS) junction line and the OS/retinal pigment epithelium (RPE) line in the foveal area (fig. ) was found. The patient was monitored without treatment. After 1 week, BCVA had improved to 0.6 in the left eye and OCT revealed partial restoration of the IS/OS line with focal thinning of the OS/RPE line in the foveal area (fig. ). After 1 month, the patient's complaints had resolved and BCVA had improved to 0.8 in the left eye. OCT showed complete restoration of the IS/OS line and the OS/RPE line (fig. ). Fluorescein angiography, indocyanine green angiography, and multifocal electroretinogram were not available in this case.

[[15.0, 'year']]

M

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{}

3934696-1

noncomm/PMC003xxxxxx/PMC3934696.xml

Olanzapine and Betamethasone Are Effective for the Treatment of Nausea and Vomiting due to Metastatic Brain Tumors of Rectal Cancer

A 78-year-old male presented with nausea, vomiting, anorexia and slight right hemiplegia. He was diagnosed with rectal cancer in August 2007, and laparoscopic low anterior resection was performed. Pathological examination confirmed the diagnosis of rectal adenocarcinoma pSE,N2,M0, stage IIIb. He received adjuvant chemotherapy consisting of oral uracil and tegafur plus leucovorin for 1 year. In November 2008, lung metastases were detected, and the chemotherapy was switched to fluorouracil, levoleucovorin and irinotecan-based chemotherapy (FOLFIRI). In May 2009, lung metastases developed, and the chemotherapy was combined with bevacizumab. However, bevacizumab was discontinued due to angina in February 2011. The lung metastases worsened in January 2012, and the chemotherapy was changed from FOLFIRI to fluorouracil, levoleucovorin and oxaliplatin-based chemotherapy (FOLFOX4). In September 2012, brain imaging using magnetic resonance imaging (MRI) showed multiple brain metastases (maximum size 10 mm) in the left lobe with surrounding cerebral edema (fig. ).\nOn October 11, 2012 the patient was admitted for nausea, vomiting, anorexia and vertigo during body movement. Right hemiplegia appeared on hospital day 6, and he underwent whole brain radiation therapy with 30 Gy in 10 fractions starting on hospital day 7 in addition to therapy with glycerol 400 ml daily and the antiemetics metoclopramide and granisetron hydrochloride. The treatment failed to relieve his nausea and vomiting, and he was referred to the department of palliative care on hospital day 34. Treatment with olanzapine was initiated at a dose of 1.25 mg every night. The constant nausea disappeared the next day. The vomiting during body movement decreased from 2–3 times a day to 0–1 times a day, and the patient's appetite increased. However, 6 days after the administration of olanzapine, he developed a sudden intense headache in addition to nausea and vomiting, and right hemiplegia and anarthria rapidly progressed (on hospital day 39). On MRI, the size of the main brain metastatic lesion had increased to 20 mm and the surrounding cerebral edema had worsened (fig. ). The patient received betamethasone 4 mg per day in addition to olanzapine 1.25 mg. For approximately 3 weeks until death (on hospital day 64), the nausea and vomiting were relieved, without anorexia. However, the sudden periodic intense headaches and paralysis did not improve.

[[78.0, 'year']]

M

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{}

3934697-1

noncomm/PMC003xxxxxx/PMC3934697.xml

Intestinal Neuronal Dysplasia Type B in Adults: A Controversial Entity

A 71-year-old male patient presented to the surgical emergency department of our hospital complaining of abdominal pain and constipation. Basic laboratory tests on admission were within normal range. Clinical examination revealed abdominal distention, dehydration and no evidence of bowel sounds. Systemic and neurologic diseases as well as infections and malabsorption syndromes were excluded by medical history, physical and blood examination. From the patient's history, chronic constipation had achieved 20 points in the Wexner Constipation Scale (WCS) [] since his infancy. He had occasionally received treatment with laxatives and rectal enemas. His last surgery had been 10 years before and included sigmoidectomy. Histopathology analysis of the resected sigmoid colon showed a dilated bowel with no evidence of malignancy, and no other immunohistological analysis was done. The patient stated that the symptoms of constipation had never resolved, while during the last 6 months he had been admitted to hospital twice due to bowel obstruction and treated without surgery.\nDuring the last admission to our hospital, an abdominal X-ray showed a massively distended large bowel (fig. ), and an abdominal computed tomography scan did not reveal any mechanical obstruction (fig. ). Due to excess abdominal tenderness and radiological findings, it was decided that an exploratory laparotomy would be appropriate. During laparotomy (fig. ) no obvious mechanical cause was found and a subtotal colectomy and Hartmann's procedure was performed. The patient had an uneventful postoperative course and was discharged on the 12th postoperative day with a temporary ileostomy.\nIn the pathology department, gross examination showed that the colon and especially the cecum was distended and its mucosal surface appeared flattened, with a polypoid lesion measuring 2.5 cm found in the cecum. Microscopic examination and immunohistochemical study of the polypoid lesion proved that it was a villoglandular adenoma with low-grade dysplasia, and full-thickness analysis of the resected colon revealed enlarged myenteric and submucosal neurons as well as an increased number of giant cells. The nerve bundles appeared hypertrophic, containing increased numbers of thickened disorganized axons. A small number of isolated ganglion cells and giant submucosal ganglia (containing 7–15 ganglion cells) (fig. ) were also detected in the submucosa. Immunohistochemistry was applied by using antibodies against protein S-100, neuron-specific enolase, glial fibrillary acidic protein and synaptophysin. Protein S-100 and synaptophysin revealed an increase in ganglion cells, and glial fibrillary acidic protein staining showed increased nerve plexus around blood vessels. The diagnosis of IND was established.\nThree months later, during his second admission for restoration of bowel continuity, preoperative rectoscopy and rectal manometry showed no significant findings. During surgery for bowel continuity restoration, the upper third of the rectum was removed and an ileorectal (side to end) low anterior anastomosis to the posterior wall of the rectum 6 cm above the dentate line was performed. The patient was discharged on postoperative day 10 and during a 2-year follow-up has remained asymptomatic with normal bowel motility. The WCS score is 8 points, showing a significant difference compared to 20 points preoperatively.

[[71.0, 'year']]

M

{'15564160': 1, '15630639': 1, '16772835': 1, '10448584': 1, '15359389': 1, '16772824': 1, '4130757': 1, '34908804': 1, '28700491': 1, '23480925': 1, '7857887': 1, '21327554': 1, '5429378': 1, '8646957': 1, '14761826': 1, '15660323': 1, '10770242': 1, '12483654': 1, '7738747': 1, '9504852': 1, '28756361': 1, '27602240': 1, '24574943': 2}

{}

3934770-1

noncomm/PMC003xxxxxx/PMC3934770.xml

Trigeminal Neuralgia as the First Clinical Manifestation of Anti-Hu Paraneoplastic Syndrome Induced by a Borderline Ovarian Mucinous Tumor

On June 5, 2012, a 76-year-old female patient was transported to the Intensive Care Unit of Mehr General Hospital (Tehran, Iran) via ambulance. She had a 3-week history of progressive somnolence, confusion, agitation, symptoms of depersonalization, and visual hallucinations. Her disorder began 12 weeks prior to her current admission, with daily and repeated short, sharp, lancinating, electric shock-like pain involving the left lower division of the fifth cranial nerve. She was taken to a local hospital in another city, where she underwent neurological evaluation by magnetic resonance imaging (MRI) and electroencephalography (EEG), with normal results. The patient was instructed to take 200 mg carbamazepine 3 times a day, which partially relieved her pain episodes. Eight weeks later, and following attacks of nocturnal hallucinations, a neurologist prescribed 5 mg olanzapine to be taken each night. However, her mental and psychiatric state deteriorated, and she was transferred to Mehr General Hospital.\nOn physical examination, the patient was afebrile. Scars from a coronary artery bypass surgery performed in 2008 were present on her anterior chest wall. Her lungs were clear, and no lymphadenopathy or organomegaly was detected. Neurological evaluation revealed an awake, agitated, confused woman with garbled and incoherent speech. She was easily distracted by irrelevant stimuli and could not sustain attention. No carotid bruit or stiff neck was detected. Cranial nerves were grossly normal, and no sensory or motor abnormalities were detected in the fifth cranial nerve. Corneal, orbicularis oculi, and sneeze reflexes were intact. Her motor power was 5/5 in all 4 extremities, her deep tendon reflexes were 2/4 in the arms and 1/4 in the legs, she had normal flexor plantar responses, and no abnormalities were seen in gait or coordination.\nBrain MRI, MR angiography, and MR venography were normal. EEG showed moderately diffuse slowing (fig. ). A lumbar puncture was performed, and the results of the cerebrospinal fluid analysis are shown in table . To avoid any possible treatable diagnosis, intravenous phenytoin and acyclovir were started, but acyclovir was stopped 5 days later when the final diagnosis was achieved. Quetiapine was administered to keep the patient calm.\nBlood tests showed an elevated erythrocyte sedimentation rate (40 mm after 1 h; normal: <20 mm) with normal complete blood count, and low hemoglobin (10.6 mg/dl; normal: 12–16 mg/dl). Liver and kidney function tests and blood sugar levels were normal. Immunologic tests showed negative anti-Ro, anti-nuclear, and anti-neutrophil cytoplasmic antibodies. Serum protein electrophoresis and angiotensin-converting enzyme levels were normal. Broad serological screening assays for infectious conditions, including HIV, herpes simplex virus 1 and 2, hepatitis, Lyme disease, syphilis, and tuberculosis using the intermediate-strength purified protein derivative skin test, were negative. Tumor markers, including carbohydrate antigen (CA)-125, CA-19.9, CA-15.3, CA-242, carcinoembryonic antigen, serum alpha-fetoprotein, and anti-Ri and anti-Yo antibodies were not detected, but anti-Hu antibodies were present in the serum, as visualized by dot blot and indirect immunofluorescence techniques.\nTo localize the suspected malignancy, the patient underwent computed tomography scans of the chest and abdomen, which disclosed a large mass in the left ovary (fig. ). She underwent surgery, and a 20-cm ovarian intestinal-type mucinous tumor (fig. ) was removed. After surgery, the patient recovered quickly. After 2 weeks, she was alert and oriented with normal speech. Her second EEG at this stage was normal (fig. ). Two months after the operation, the patient was pain-free, and her carbamazepine and phenytoin were gradually discontinued. At her last follow-up visit in March 2013, the patient was well and free of symptoms.

[[76.0, 'year']]

F

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{'5030447-1': 1}

3934772-1

noncomm/PMC003xxxxxx/PMC3934772.xml

Subglottic Chondrosarcoma Presenting Only Mild Acute-Onset Dyspnea: A Case Report and Review of the Literature

A 68-year-old Asian male presented with a 1-year history of hoarseness, mild dysphagia, and a 1-month history of dyspnea. On examination, the patient had no stridor. Transnasal flexible endoscopy (fig. ) revealed a smooth mass below the glottis distorting the larynx. Half of the subglottic area was obstructed, and he also had left vocal cord paresis. A computed tomography (CT) scan of the neck revealed a solid mass in the supraglottic region, extending from the cricoid cartilage to the left thyroid cartilage area, with no neck lymphadenopathy (fig. ). The CT scan showed gross expansion of the cricoid cartilage with marked reduction of the airway, but the overlying mucosa was smooth. Some intermittent calcification spots within the extending tumor mass were also noted. On admission, the patient received laryngomicrosurgery for biopsy and tracheostomy to secure the airway. Multiple biopsies were taken which revealed features consistent with low-grade chondrosarcoma (fig. ). As the patient is running a company, he needs to talk frequently. In agreement with the patient and his family, and considering the low-grade nature of the tumor, endoscopic debulking was decided. As a result, we performed a laryngomicrosurgery operation. The subglottic lesion was debulked with electrocauterization to widen the compromised airway. Histologic diagnosis revealed a low-grade chondrosarcoma of the cricoid. The patient was well after removal of the tracheostomy tube 2 months later. During the 12-month follow-up, the lesion did not increase in size and no metastasis was detected on the CT image. The patient receives ambulatory treatment and is doing well, and transnasal flexible endoscopy revealed that his airway is relatively patent (fig. ).

[[68.0, 'year']]

M

{'12131151': 1, '8215099': 1, '2243522': 1, '18533555': 1, '6810266': 1, '890662': 1, '9591557': 1, '23404226': 1, '6497238': 1, '6503573': 1, '17908358': 1, '10740194': 1, '2451440': 1, '19566974': 1, '32495672': 2, '24575022': 2}

{'7273573-1': 1}

3934774-1

noncomm/PMC003xxxxxx/PMC3934774.xml

A Case of Pulmonary Infiltrates in a Patient with Colon Carcinoma

A 31-year-old white male with a known history of colon carcinoma was referred to the Interventional Pulmonary service for right lower lobe infiltrates and mucous plugging on computed tomography (CT) with concern for pneumonia (fig. ). Bronchoscopy was performed revealing a broad based mass completely obstructing the bronchus intermedius (fig. ). It was possible to pass a probe into the right lower lobe, and subsequent photoablation and mechanical debulking revealed that the mass was arising near the origin of the superior basal segment of the right lower lobe (RB6) and could be resected (fig. ). Pathology confirmed that this was consistent with the patient's known primary colon carcinoma.

[[31.0, 'year']]

M

{'25337391': 1, '25337406': 1, '8361198': 1, '6499611': 1, '25337403': 1, '11243940': 1, '25774306': 1, '10836010': 1, '8691837': 1, '25861608': 1, '25774308': 1, '25774309': 1, '25337405': 1, '25774305': 1, '25861609': 1, '19057274': 1, '25774304': 1, '25774310': 1, '25815301': 1, '18812690': 1, '25337402': 1, '25815302': 1, '8989073': 1, '11243955': 1, '25861610': 1, '25815303': 1, '25774311': 1, '25815304': 1, '7359928': 1, '24575014': 2}

{}

3934776-1

noncomm/PMC003xxxxxx/PMC3934776.xml

Unmasking Cryptococcal Meningitis Immune Reconstitution Inflammatory Syndrome due to Granulocyte Colony-Stimulating Factor Use in a Patient with a Poorly Differentiated Germ Cell Neoplasm

A 56-year-old male visited his primary care physician to be evaluated for abdominal pain, and the subsequent workup revealed a large retroperitoneal mass measuring 21.8 × 17.5 × 11 cm. A diagnostic workup revealed a poorly differentiated germ cell neoplasm, most likely an embryonal carcinoma. Two months after his initial presentation he began chemotherapy (bleomycin, etoposide and cisplatin). By day 10 of cycle 3 (approximately 4 months after initial presentation), imaging showed a drastic decrease in tumor size.\nSix days after his most recent dose of chemotherapy, he presented to the emergency department noting intermittent fatigue, nausea, vomiting and diarrhea since his last chemotherapy treatment. He also noted headache and fever over the past day. In the emergency department (hospital day 0), he was found to be neutropenic with a total white blood cell (WBC) count of 0.6 × 109 cells/l and an absolute neutrophil count (ANC) of 0.4 × 109 cells/l. He appeared to be in no acute distress but febrile with a temperature of 38.7°C. No obvious signs of an infectious source were noted, including examination of his port-a-cath. The patient was started on cefepime and admitted to the inpatient oncology service for treatment of neutropenic fever.\nThe admitting physician noted tachycardia, but the patient's blood pressure was within normal limits. The only possible localizing sign of infection was a questionable cellulitis of the right lower leg. As the patient had previously been on prophylactic levofloxacin and had frequently been to infusion clinics, vancomycin was added for methicillin-resistant Staphylococcus aureus coverage. The patient was also started on G-CSF (480 μg/day) for neutropenia. Over the subsequent 2 days, his fever and tachycardia continued, and on hospital day 3 the patient became lethargic and confused. A chest radiograph (which showed no sign of pneumonia), blood and urine cultures and a CT scan of the head without contrast showed no acute changes. Throughout this period his ANC remained <1.0 × 109 cells/l.\nBy hospital day 4, the patient was unable to respond to commands and was transferred to our institution for further care; his WBC count had risen to 1.8 × 109 cells/l (ANC not measured) and he remained febrile. His vancomycin was continued while cefepime was changed to ceftazidime out of concern for cefepime-induced encephalopathy []; G-CSF was continued. Shortly after arrival, the patient had 2 tonic clonic seizures, and lorazepam and fosphenytoin were given. He became more lethargic, with periods of apnea, and was transferred to the intensive care unit for endotracheal intubation. An MRI of the brain and MRA of the brain and neck with and without contrast showed an acute punctate infarct in the right hippocampus and no major vascular findings. Continuous EEG monitoring was started. His WBC count shortly after intubation was 11.4 × 109 cells/l with an ANC of 7.0 × 109 cells/l. Fig. shows ANC, WBC count, G-CSF dosing and the patient's symptoms over time.\nA lumbar puncture was performed on hospital day 5 and revealed 64 WBC/μl, 30% neutrophils, 39% lymphocytes, glucose 37 mg/dl and protein 99 mg/dl. No organisms were seen on Gram staining. No cerebrospinal fluid opening pressure was measured. A thorough cerebrospinal fluid workup for infection was sent for evaluation, and empiric acyclovir was started. Cerebrospinal fluid India ink stain was completed in the early evening of hospital day 5 and showed rare encapsulated yeast, and the cerebrospinal fluid cryptococcal antigen (Latex Agglutination, IMMY, Inc., Norman, Okla., USA) was positive at a dilution of 1:32.\nThe infectious disease service was informed of the result, amphotericin B lipid complex was started at a dose of 5 mg/kg, and flucytosine 100 mg/kg/day was also ordered but was not but available until hospital day 7. On hospital day 6, the WBC count rose to 28.0 × 109 cells/l and G-CSF, acyclovir, ceftazidime and vancomycin were stopped.\nThroughout hospital days 6 and 7, the patient was minimally responsive despite no sedation since intubation; however, he did withdraw from painful stimuli. No additional seizures occurred. On hospital day 8, the patient moved his lower extremities on command, and his neurological function rapidly improved. Repeat lumbar puncture was performed on hospital day 9 with an opening pressure of 16 cm H20. By hospital day 10, the patient's neurologic status had improved, and he was extubated. His culture showed Cryptococcus neoformans resistant to flucytosine, and so flucytosine was replaced with fluconazole 800 mg daily.\nAfter 14 days total of amphotericin-based combination therapy, the patient continued on fluconazole 800 mg daily consolidation monotherapy. Repeat lumbar puncture at the time of stopping amphotericin showed a decreased cryptococcal antigen titer of 1:4 and an opening pressure of 22 cm H20. On hospital day 21, the patient was discharged to a transitional care unit. Since hospital discharge, the patient has continued to do well in terms of his CM, although he has required hospitalization for pericarditis. He is being maintained on fluconazole 200 mg daily for secondary prophylaxis, and further chemotherapy has been postponed pending improvement of his other health issues.

[[56.0, 'year']]

M

{'24173584': 1, '21253011': 1, '28111998': 1, '17516390': 1, '23509356': 1, '15909263': 1, '26814182': 1, '21029993': 1, '20230635': 1, '29670625': 1, '16344542': 1, '21094071': 1, '11860010': 1, '31138748': 1, '24575007': 2}

{}

3934778-1

noncomm/PMC003xxxxxx/PMC3934778.xml

Caudal Cingulate Infarction Manifesting Astasia

A 58-year-old male with a history of hypertension and diabetes mellitus presented to our emergency room. He complained of dysarthria and inability to sit, stand and walk since his awakening. On admission, he was alert and oriented. He was slightly dysarthric. His Mini-Mental State Examination score was 30. He showed neither hemispatial neglect nor disconnection syndrome. Muscle strength of all extremities and the trunk was not decreased. On sensory examination, vibratory sensation was slightly decreased on both feet, which was attributed to diabetes mellitus. Thermal, pinprick and positional sensation was perceived normally in all extremities and trunk. The finger-to-nose test revealed terminal dysmetria in the right arm. However, the heel-to-shin test showed no asymmetry. Rapid alternating hand movement was not disturbed on both sides. Deep tendon reflexes were normoactive without pathological reflexes. He did not show any resting or intentional tremor nor asterixis. When he attempted to sit, he grasped the bed rails only with both hands to pull himself up without using his axial and leg muscles, but retropulsion occurred soon after detaching his hands from the bed rails. When tested lying on the bed, he could maintain his legs elevated for more than 10 s. However, he could not stand by himself unless his caregiver supported him.\nCT of the brain did not show any intracranial hemorrhage. Then, an MRI of the brain was performed and diffusion-weighted imaging showed a high-intensity lesion at the medial frontal lobe on the left side (fig. ). A sagittal section of the double inversion recovery sequence revealed a high-intensity lesion at the cingulate gyrus between the VCA and VCP line and a corpus callosum body (fig. ).\nAfter 3 weeks of rehabilitation, he could keep standing and walk freely without assistance. His gait was not broad-based and the steps were regular in amplitude and direction. However, when standing on one foot and walking in tandem gait, lateropulsion to the right side persisted. With his eyes closed, lateropulsion to the right remained. After discharge, he could return to work, although the slight lateropulsion on tandem gait and standing on one foot persisted even at 1 year after onset.

[[58.0, 'year']]

M

{'22205761': 1, '16322493': 1, '8437689': 1, '29264041': 1, '8042930': 1, '18506392': 1, '22767673': 1, '4961812': 1, '10388793': 1, '8670662': 1, '17179714': 1, '25126430': 2, '8027764': 1, '2841901': 1, '8179307': 1, '15804250': 1, '24575027': 2}

{'4122058-1': 1}

3934780-1

noncomm/PMC003xxxxxx/PMC3934780.xml

Acquired Ichthyosis Triggered by an Osseous Hemangiopericytoma: A Case Report and Review of the Literature

A 34-year-old woman presented to our Department with a 2-month history of a widespread, scaling, skin eruption. She also complained of a 7-kg weight loss over the last 2 months as well as fatigue and low back pain. On physical examination, she had generalized xerosis with large rhomboidal scales covering most of her body and scalp, painful stomatitis and palmar and plantar hyperkeratosis (fig. ). Bilateral ectropion due to eyelid involvement was present. There was no personal or family history of ichthyosis. A skin biopsy was performed, which confirmed the clinical diagnosis of AI by revealing hyperorthokeratosis and an absence of granular layers (fig. ). However, her past medical history was significant for osseous HP, for which she had successfully undergone tumor embolization, followed by a total tumor resection 2 years ago. Based on her past medical history and the current signs, a possible recurrence of HP was suspected, and therefore, an abdominal computed tomography (CT) scan was recommended. The CT scan revealed a multilobular tumor mass in the minor pelvis, infiltrating the sacrum and perirectal space and spreading to the lower lumbar muscles (HP recurrence) (fig. ). The presence of multiple liver metastases was also reported. Intensive topical emollients were prescribed for the AI, and the patient was referred to the Oncology Department. Treatment with paclitaxel at a dose of 80 mg/m2 at days 1, 8 and 15, followed by 1 week of rest and repeated every 4 weeks. Unfortunately, despite the chemotherapy, no improvement of either the tumor mass and metastases or the AI was observed over the following 3 months. On the contrary, at that point, the patient's general condition started to deteriorate rapidly. The patient passed away 6 months after her initial presentation.

[[34.0, 'year']]

F

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{}

3934781-1

noncomm/PMC003xxxxxx/PMC3934781.xml

Combined Intracorneal and Subconjunctival Bevacizumab Injections for Recurrent Visual Loss and Intraocular Hemorrhage from Vascularized Fibrous Downgrowth

This 44-year-old housewife sustained ocular injuries 23 years ago after a motor vehicle accident and was operated bilaterally. Visual acuity was since lost in the right eye and the left eye was maintained on ocular hypotensive drops. Two weeks before presentation, she had fallen from a ladder and had sustained extensive corneoscleral laceration of the left eye with uveal prolapse and total hyphema. The laceration was sutured in an outside hospital.\nVisual acuity on presentation was light perception with through-and-through vertical corneal wound, dense hyphema and large inferior conjunctival bleb. Intraocular pressure was 5 mm Hg. B-scan ultrasonography revealed total retinal detachment. The corneoscleral wound was resutured because it was gaping. A scleral buckle was placed at 360°. There was profuse continuous bleeding from the iris and ciliary body area throughout surgery despite pars plana cleanout of the anterior segment (lens remnant, vitreous and blood clot). During posterior vitrectomy, a one-disc diameter macular hole was suspected. Silicone oil fill of the vitreous cavity was followed by endolaser of an extensive inferior giant retinal tear. Postoperatively, the patient had multiple sessions of laser retinopexy to seal a localized inferior retinal detachment. A large retrocorneal membrane was evident 6 weeks postoperatively over the temporal cornea (fig. ). Postoperative best spectacle-corrected visual acuity was 6/60. A rise in intraocular pressure was again controlled by topical β-blocker. She had 5 recurrent episodes of visual loss to the level of hand motion from intraocular bleeding (interface hemorrhage, hyphema and anterior vitreous hemorrhage) (fig. ). Visual loss used to resolve 1 week after subconjunctival (0.1 ml; 2.5 mg) and intracorneal (0.05 ml; 1.25 mg) bevacizumab (after formal consent). The rationale was the vascular nature of the fibrovascular membrane with feeder vessels consisting of several branches of limbal and corneal stromal neovascularization (fig. ). The injections were done in a sterile fashion using a speculum and a 30-gauge needle. The intrastromal (corneal) and subconjunctival injections were aimed at the site of the feeder vessels (fig. ). We avoided intracameral injections because of the concomitant presence of glaucoma and intravitreal silicone oil. The patient was instructed to have monthly injections. When she was late for follow-up, intraocular bleeding would occur. The patient was followed for 40 months after presentation with a final vision of 6/120. She received a total of 21 injections over a period of 2.5 years with a total of 5 episodes of vitreous hemorrhage.\nWritten informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

[[44.0, 'year']]

F

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{}

3934782-1

noncomm/PMC003xxxxxx/PMC3934782.xml

Revision of Perineal Urethrostomy Using a Meshed Split-Thickness Skin Graft

A 45-year-old male patient underwent partial penectomy and distal urethrostomy for erythroplasia of Queyrat (i.e. carcinoma in situ of squamous cell carcinoma) in 2008. Since he was extremely dissatisfied with the remaining penile stump, he consulted our center about the option of phallic reconstruction. On June 14, 2010, phallic reconstruction was performed. The native urethra was lengthened by means of a radial forearm free flap. The phallus was reconstructed with an anterolateral thigh flap. The urethra was positioned inside the phallus according to the ‘tube-in-a-tube’ principle. In October 2012, the patient presented with obstructive voiding symptoms. A diagnostic urethroscopy showed a suspicious lesion in the remaining native, bulbar urethra, and biopsy confirmed the recurrence of a poorly differentiated squamous cell carcinoma of the urethra. On December 13, 2012, he underwent excision of the remaining native, bulbar urethra with the creation of a perineal urethrostomy at the membranous urethra. The perineal urethrostomy was performed using the technique as described by Blandy et al. []. Unfortunately, the postoperative period was complicated by bleeding, wound dehiscence and infection (fig. ). Finally, the perineal urethrostomy became completely obliterated and a suprapubic catheter was placed (fig. ). As the patient accepted this only as a temporary solution, we proposed the revision of the perineal urethrostomy using a meshed split-thickness skin graft. This procedure was performed on April 23, 2013. The patient was placed in the lithotomy position and a midline perineal incision was made. Dissection was continued up to the level of the urogenital diaphragm. A flexible cystoscope was inserted along the suprapubic tract and brought through the bladder neck into the posterior urethra. This marked the place of obliteration as the light of the cystoscope could be seen through the perineal incision. The urethra was opened on the tip of the cystoscope. All fibrotic tissue was further excised until only healthy urethral mucosa remained. The urethral stump was spatulated over 1.5 cm. Vicryl 4.0 stay sutures were placed at the mucosa to facilitate identification and to prepare the anastomosis for the graft (fig. ). A split-thickness graft (10 × 15 cm) was harvested from the skin of the left thigh and meshed 1:1. In the middle of the graft, an opening was made to anastomose with the urethral opening. The edges of the graft were further tailored and anastomosed with the perineal skin edges. Several additional quilting sutures were placed to fix the graft against the perineal graft bed (fig. ). A compressive dressing and a 20-Fr urethral catheter were left in place for 5 days. After 5 days, the dressing was removed with satisfactory graft take. Subsequently, the catheter was also removed. At present (6 months of follow-up), the patient is voiding well. Maximum and average urinary flow are 22 and 13 ml/s, respectively. His International Prostate Symptom score is 5/35. The perineal wound has healed well, with minimal contraction of the grafted area (fig. ). Through the neomeatus, a 20-Fr catheter can be passed without any difficulties, indicating a sufficient diameter.\nThis study was approved of by the local ethics committee.

[[45.0, 'year']]

M

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{'6458805-1': 1}

3934784-1

noncomm/PMC003xxxxxx/PMC3934784.xml

The Use of Lanreotide Autogel® in the Treatment of Intestinal Obstruction in a Patient with Adenocarcinoma

In January 2012, a 60-year-old woman presented with a 3-month history of increasing abdominal pain and a change in the stool pattern with a single episode of anal red blood loss in the past week. She had a medical history of hypercholesterolemia and had previously undergone appendectomy. An ultrasound of the abdomen showed several masses dispersed over the peritoneum, compatible with significant peritoneal metastasis. Diffuse ascites fluid was also noted and could not be tapped. Clinical examination and auscultation showed a slightly enlarged abdomen with good peristalsis of the intestine. No clangor was noted. Colonoscopy revealed a large ulcerating lesion at the level of the sigmoid colon, which was diagnosed as a moderately differentiated adenocarcinoma after biopsy.\nNo surgery was performed due to the presence of peritoneal carcinomatosis. Chemotherapy with 18 biweekly cycles (1 cycle every 2 weeks) of FOLFIRI and bevacizumab (Avastin®, Roche) was started. FOLFIRI was administered according to the following schedule: day 1, irinotecan 180 mg/m2; days 1 and 2, leucovorin 100 mg/m2, 5-fluorouracil bolus 400 mg/m2 and 5-fluorouracil infusion 600 mg/m2. Avastin was given at a dose of 5 mg/kg every 2 weeks. If the patient had responded to this treatment, the possibility of performing debulking surgery and hyperthermic intraperitoneal chemotherapy would have been considered. The abdominal pain disappeared after the first month of chemotherapy, while the ascites disappeared after 6 weeks. There was also a slight regression of the peritoneal masses.\nDuring the 5th, 6th and 7th cycle of chemotherapy, the patient was hospitalized because of clinical signs of intestinal subocclusion (vomiting, absence of stools and flatus) that evolved into complete obstruction. A CT scan of the abdomen did not reveal the cause of the obstruction or deterioration of the oncological lesions. Based on this observation, a FOLFIRI-induced mucositis, motility disturbances due to peritoneal metastasis or a combination of these two factors were suspected. During hospitalization, fluids, alizapride (Litican®, 300 mg/day; Sanofi-Aventis) and methylprednisolone sodium succinate (Solu-Medrol®, 40 mg/day; Pfizer Inc.) were administered intravenously, paracetamol (1 g/dose) was administered intravenously 4 times a day and octreotide (Sandostatin®, 0.5 mg/dose; Novartis) was administered subcutaneously 3 times a day until the obstruction resolved. Oral consumption of food and fluids was withheld, and a nasogastric tube was placed. At each of the 3 hospitalization episodes, an enema was performed. After the first episode of obstruction, the dose of FOLFIRI was decreased to 75%, while the dose of Avastin was not decreased. All episodes of obstruction resolved after 3 days, and the chemotherapy was reintroduced after 1–2 weeks following resolution of the obstruction. After the third episode of obstruction, we initiated treatment with lanreotide Autogel 120 mg, injected deep subcutaneously once every 4 weeks. The 120-mg dose was chosen based on our experience that the 90-mg dose has a lower efficacy. The treatment with lanreotide Autogel continued during the remaining 11 cycles of chemotherapy, and no further episodes of intestinal subocclusion or obstruction occurred.\nBecause tumor response following the 18 cycles of chemotherapy was not sufficient, debulking surgery or hyperthermic intraperitoneal chemotherapy treatment were not recommended. After a 3-month break in the chemotherapy treatment requested by the patient, a second-line chemotherapy course was initiated. Lanreotide Autogel administration was continued during this treatment break. The patient is currently undergoing second-line chemotherapy with FOLFOX4 at 75% of the usual dose, administered once every 2 weeks in 12 cycles according to the following schedule: day 1, oxaliplatin 85 mg/m2 at 75%; days 1 and 2, 75% of leucovorin 100 mg/m2, 5-fluorouracil bolus 400 mg/m2 and 5-fluorouracil infusion 600 mg/m2. The 4-weekly lanreotide Autogel injections have been continued without interruption to support the quality of life of the patient. No new episodes of obstruction occurred until November 2013, and the patient reported no side effects due to lanreotide Autogel.

[[60.0, 'year']]

F

{'9204656': 1, '23109694': 1, '16376743': 1, '4682131': 1, '11430417': 1, '15357519': 1, '11980627': 1, '10600282': 1, '22277783': 1, '22265994': 1, '26933420': 2, '24575015': 2}

{'4772641-1': 1}

3934786-1

noncomm/PMC003xxxxxx/PMC3934786.xml

Rapidly Progressive Lupus Nephritis with Extremely High Levels of Antineutrophil Cytoplasmic Antibodies

A 43-year-old Caucasian woman was admitted to the hospital due to acute renal failure and proteinuria in the nephrotic range. She presented with a 3-month history of fatigue, anaemia and lymphadenopathy. Before admission, a CT scan was performed, which revealed lymphadenopathy of the neck as well as generalized abdominal and mediastinal lymphadenopathy. Lymph node excision showed reactive lymphadenopathy. No history of skin rashes, photosensitivity, joint swelling, hair loss or oral ulcers was identified. On physical examination, the patient's height was 170 cm, her weight 56 kg (BMI 19.4), she was afebrile and her blood pressure was 117/77 mm Hg.\nLaboratory examinations revealed (table ) a white blood cell count of 3.2 × 109/l (reference range, 4–10 × 109/l), lymphocytes 13.5% (reference range, 10–50%), haemoglobin 102 g/l (reference range, 115–165 g/l), platelets 147 × 109/l (reference range, 150–350 × 109/l), total protein 71 g/l (reference range, 64–83 g/l), albumin 27 g/l (reference range, 35–50 g/l), lactate dehydrogenase 161 U/l (reference range, <250 U/l), haptoglobulin 1.85 g/l (reference range, 0.3–2.0 g/l), urea 12.6 mmol/l (reference range, 2.7–6.8 mmol/l), creatinine 174 μmol/l (reference range, 45–84 μmol/l), C-reactive protein <5 mg/l (reference range, <5 mg/l) and an increased erythrocyte sedimentation rate of 81 mm/h. Urine analysis showed proteinuria of 6.4 g/day and an active sediment containing dysmorphed red blood cells. Serum complement levels were low with a C3c of 0.2 g/l (reference range, 0.8–1.8 g/l), C4 0.02 g/l (reference range, 0.1–0.4 g/l), and CH50 14 U Eq/ml (reference range, 70–180 U Eq/ml). Antinuclear antibodies were elevated to 1:80 (reference range, <40-fold), anti-double-stranded DNA antibodies were not detectable and anti-C1q was 20 IU/ml (reference range, <15 U/ml). ANCA titres on immunofluorescence were increased up to 1:640 (reference range <1:20). Using enzyme-linked immunosorbent assay (ELISA), MPO-ANCAs were positive with 3,622 U/ml (reference range, <5 U/ml) and PR3-ANCAs were not detectable. Anti-SS-A/Ro52 were 22 U/ml (reference range, <10 U/ml) and anti-SS-A/Ro60 were >240 U/ml (reference range, <10 U/ml). Cryoglobulines were not detectable. Serologic tests for HIV, hepatitis B/C and cytomegalovirus were all negative. Epstein-Barr-virus IgG was positive, IgM was negative and EBNA1 IgG was positive.\nRenal biopsy was performed on day 2, which revealed a diffuse, segmental, proliferative, immune-complex GN with crescents in 4 of 19 glomeruli corresponding to an LN class IV-S (A/C; fig. ). Immunofluorescence staining showed deposition of IgA, IgM, C1q, C3 and C5–9 in the mesangial area and peripheral capillaries. Electron microscopy revealed mesangial and peripheral electron-dense deposits without obvious structuring. Most of the peripheral deposits were located subendothelially, there were also some subepithelial deposits. Tubulo-reticular structures within one endothelial cell were found.\nAfter renal biopsy, methylprednisolone pulses were initiated (500 mg/day for 3 days) followed by oral prednisone 50 mg/day and cyclophosphamide 1 g intravenously. However, the serum concentration of creatinine gradually increased, so that again methylprednisolone pulses (500 mg/day for 2 days) were repeated on day 13, and high volume plasma exchange was started for 5 consecutive days to eliminate MPO-ANCAs (fig. ). After the plasma exchanges, the MPO-ANCA levels were decreased to 104 U/l and renal function improved so that the patient was discharged on day 39. Cyclophosphamide therapy was continued with 500 mg intravenously every second week for 3 months, so that the patient received a cumulative dose of 3.5 g. The maintenance therapy with azathioprine had to be switched to mycophenolate mofetil due to gastrointestinal side effects. Eighteen months after starting treatment, the patient is still in remission. MPO-ANCAs are not detectable and renal function has recovered completely with a creatinine concentration of 88 μmol/l (reference range, 45–84 μmol/l) and a minimal proteinuria of 0.2 g/day – using a treatment regime consisting of mycophenolate mofetil 2 g, prednisone 7.5 mg and hydroxychloroquine 200 mg per day.

[[43.0, 'year']]

F

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{}

3934787-1

noncomm/PMC003xxxxxx/PMC3934787.xml

Carcinomatous Myelitis and Meningitis after a Squamous Cell Carcinoma of the Lip

The patient was a 33-year-old Caucasian man without any specific personal or familial history. He was an active smoker without alcoholic intoxication. In 2009, he presented a lesion of the lower lip. He underwent resection of this lesion, and the first pathological analysis revealed a salivary ductal ecstasy.\nOne year later, he developed a peripheral facial paralysis associated with left trigeminal neuralgia. MRI showed perineural invasion of the trigeminal nerve within the temporal fossa (fig. ). Surgical biopsies performed in July 2010 identified secondary lesions from a well-differentiated SCC localized around the V and VII nerve sheaths. According to this diagnosis, a new pathological analysis of the previous resection of the lip was performed in a cancer reference center and diagnosis of SCC of the lip was made. The patient started radio-chemotherapy in September 2010. He received 70 Gy in 35 fractions, and five courses of an association of 5-fluorouracil and cisplatinum. First radiological assessments, 4 months after the end of radio-chemotherapy showed no residual disease.\nHowever, in November 2011, the patient presented with new neurological symptoms: paraparesis and back pain. MRI showed medullary myelitis without meningeal enhancement. Lumbar puncture revealed inflammatory cells without atypical cells within the cerebrospinal fluid (CSF). Cerebral MRI and positron emission tomography scan were normal with no evidence of visceral metastasis. Multiple sclerosis was suspected and the patient received several corticosteroid flashes who led to partial improvement of symptoms. Nevertheless, other explorations were negative, and it was not possible to certify the diagnosis of multiple sclerosis. In early 2012, the patient relapsed and developed paraparesis once again. Sequential lumbar punctures found no abnormal cells. He was given a symptomatic treatment combining corticosteroid and plasma depletion. In March 2012, CSF analysis showed intrathecal immunoglobulin syntheses with oligoclonal immunoglobulin bands. Medullary MRI revealed intramedullary hyperintensity without enhancement after gadolinium injection. The diagnosis of dysimmune myelitis was made and the patient received an immunosuppressive drug (mycophenolate mofetil). It is of note that his neurological symptoms partially improved under this treatment.\nIn August 2012, he subsequently developed progressive back pain, sphincter disorders, and lower limb weakness. A new MRI performed in November showed enhancement of the meningeal nodules and abnormal leptomeningeal contrast enhancement (fig. ). There was no evidence of brain lesions. New lumbar punctures revealed several atypical cells consistent with metastatic squamous cells (fig. ) associated with low glycorrhachia and high proteinorrhachia levels (1.25 g/l). These cells were well-differentiated with nuclear abnormalities consistent with meningeal infiltration by a well-differentiated SCC. Intrathecal immunoglobulin synthesis was no longer seen. Moreover, chest and abdominal CT-scans did not identify other secondary lesions, especially bone metastasis. Nevertheless, a PET scan showed an intramedullary spinal cord metabolism enhancement. All these data were consistent with the diagnosis of CM associated with a carcinomatous myelitis.\nThe patient underwent intrathecal weekly infusions of methotrexate (15 mg) and methylprednisolone combined with systemic treatment based on carboplatin every 3 weeks and weekly cetuximab (250 mg/m2). After two cycles, the disease was clinically stable with no extension of symptoms, the proteinorrhachia level was still high (1.5 g/l), and metastatic cells were still identified in the CSF. However, MRI showed a slight amelioration and cytological analysis revealed only isolated metastatic cells. A new chest and abdominal CT-scan showed no other secondary lesions. As the patient developed a febrile neutropenia, we decided to stop systemic chemotherapy. He received cetuximab 500 mg/m2 and intrathecal methotrexate 15 mg every 2 weeks. After three courses of treatment, clinical and MRI evaluation showed no neurological worsening and a stabilization of the medullar lesions. These observations were confirmed 6 months after treatment start.

[[33.0, 'year']]

M

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{}

3934789-1

noncomm/PMC003xxxxxx/PMC3934789.xml

Brain Dopamine Transporter Binding and Glucose Metabolism in Progressive Supranuclear Palsy-Like Creutzfeldt-Jakob Disease

A 58-year-old male was referred to a neurologist specialised in movement disorders in June 2009. He had a 6-month history of increasing clumsiness, forgetfulness, and apathy. The patient had a history of atrial fibrillation and concomitant mild dilative cardiomyopathy. He was taking warfarin, bisoprolol, digitalis, and candesartan. A 1.5-T brain MRI did not show clinically significant findings. A neurological examination revealed mildly diminished arm swing bilaterally, slight hypomimia, hypokinesia, cervical axial rigidity, and decreased frequency of blinking. The combination of hypokinesia, axial rigidity, and symmetrical extrapyramidal symptoms, in the absence of tremor, gave rise to suspicion of PSP. Levodopa was initiated with increasing doses up to 400 mg/day. One month later, increased latency in saccadic eye movements in downward gaze was noted, and postural retropulsion was evident. No favorable response to levodopa was noted, and the patient reported visual hallucinations. Consequently, the medication was withdrawn.\nThe patient was referred to the Neurology Outpatient Clinic of the University Hospital for further diagnostics and clinical follow-up. Dopamine transporter imaging with [123I]-FP-CIT-SPECT was performed 9 months after the onset of symptoms. The scan showed clear reductions in tracer binding in the left caudate nucleus compared to the right side and bilaterally in the posterior putamen (striatal to occipital uptake ratio: 2.42 on the right and 2.02 on the left) (fig. ). Neuropsychological testing revealed marked cognitive impairment manifesting as slowed executive functions and impaired working and short-term memory as well as visual recognition and processing. Psychomotor slowing and mild apraxia were also noted. FDG-PET showed widespread reductions in FDG binding bilaterally in cortical and deep grey matter, with a small area of preserved cortical binding in the frontoparietal region (fig. ). The FDG-PET findings were clinically interpreted to be suggestive of Lewy body dementia.\nDuring follow-up, rigidity further increased axially and also became present in the lower extremities. The patient's gaze was staring, and saccadic eye movements were slow. No tremor was present, but bradykinesia had increased substantially. At this time, in January 2010, he was still ambulatory, although he needed bilateral walking aids intermittently. In April 2010, the patient was admitted to an emergency unit due to deteriorating condition. He was now bedbound and aphasic, his upper limbs were in constant flexion, and intermittent myoclonic jerks, especially in the left upper limb, were present. CT of the head only revealed increased central atrophy. An EEG showed posterior theta-delta background activity with 3 slowly generalising spike-slow-wave discharges frontally without clinical motor symptoms. After intravenous lorazepam, the abnormal discharge subsided without improvement in the clinical status. The EEG was interpreted to be consistent with nonconvulsive status epilepticus, and the patient was treated with an intravenous loading dose of fosphenytoin and further with sodium valproate. However, his clinical condition remained essentially the same, and the suspicion of prion disease was raised. A control EEG, performed the next day, showed no epileptiform discharges, whereas semi-rhythmic, slow, mixed activity periods alternating with rhythmic, triphasic PED activity were evident. Per oral clonazepam (via nasogastric tube) alleviated, but did not stop, the myoclonic jerks. A brain MRI showed no structural abnormalities, whereas pathological changes in diffusion tensor imaging concurrent with CJD were seen (fig. ); however, other infectious or inflammatory etiology could not be excluded.\nIn cerebrospinal fluid (CSF) analysis, the protein content was elevated to 1,216 mg/l (normal range 150–450 mg/l), but no discoloration or pleocytosis was present, and comprehensive examinations revealed no signs of active autoimmune or infectious diseases. CSF Western blot analysis showed small amounts of 14-3-3 protein. The patient was unable to communicate or react meaningfully to physical stimuli, and at this point, 1.5 years since the first symptoms, he was transferred to a nursing home for palliative care. The patient succumbed 22 months after the onset of initial symptoms.\nIn order to confirm the diagnosis, a neuropathological examination was performed. Under macroscopic inspection, the cerebral cortex appeared atrophic and slightly spongiform. Atrophy of the caudate nucleus was apparent, and moderate dilatation of brain ventricles was noted. However, the striatum did not show any marked macroscopic changes. There were no major changes in the substantia nigra, pons or medulla, whereas marked atrophy of the cerebellum was observed. The histological samples showed marked spongiform changes in the cerebral cortex (fig. ). Abundant spongiform changes and gliosis as well as nerve cell loss were found in the caudate nucleus, putamen, and thalamus (fig. ). The substantia nigra did not show any obvious loss of neurons, but slight spongiosis was noted. In the cerebellum, a marked loss of Purkinje cells as well as granular cells was observed with numerous torpedoes in the granular cell layer. Immunohistochemically, a positive finding for prion protein was discovered using the monoclonal antibody 12F10, confirming the diagnosis of CJD.\nThis case study was conducted according to the World Association's Declaration of Helsinki. Since it was an observational retrospective study of a clinical patient case following the local guidelines of investigation and treatment procedures, no separate permission from the local ethics committee was needed. Written informed consent was obtained from the patient's caregiver.

[[58.0, 'year']]

M

{'352478': 1, '6381559': 1, '7931389': 1, '6351815': 1, '12636242': 1, '16364674': 1, '18084908': 1, '12082046': 1, '17353478': 1, '9083566': 1, '17234215': 1, '15142229': 1, '18638039': 1, '22691528': 1, '12111302': 1, '24575030': 2}

{}

3934803-1

noncomm/PMC003xxxxxx/PMC3934803.xml

Diagnostic Evaluation of Ocular Toxocariasis Using High-Penetration Optical Coherence Tomography

The patient was a 50-year-old man who reported blurred vision and floaters in the right eye that persisted for about 6 months. He then consulted a local ophthalmologist. He was diagnosed with panuveitis and referred to our department. There was no previous ophthalmic history and his general health was good. He had a 10-year history of contact with dogs, but no history of ocular trauma. On presentation, the best-corrected visual acuity (BCVA) in the affected eye was 20/25, and the ocular pressure was normal. Results of slit lamp examination showed inflammatory cells (2+) and infiltration in the anterior chamber of the right eye. The fundus examination showed (1+) vitreous opacities, (2+) vitreous cells, phlebitis, moderate disc swelling, reddening, yellowish-white retinal lesions in the inferotemporal region of the optic disc, and a white exudative lesion near the inferior arcade (fig. ). The left eye was normal. Fluorescein angiography showed a hyperfluorescent lesion and dye leakage from the middle to late phases with a hyperfluorescent optic disc. Peripheral blood tests, including blood cell count, erythrocyte sedimentation test, C-reactive protein, and serum angiotensin converting enzyme, were within the normal ranges. Serum toxoplasma antibody and viral antibodies were negative. Chest X-rays were normal. No signs suggested endogenous uveitis, and no clinical findings suggested uveitis due to tuberculosis or syphilitic, fungal, or viral infection. Examination of the patient's serum for antibody to T. canis larvae by ELISA showed an elevated antibody level (fig. ).\nBased on characteristic funduscopic findings, a diagnosis of ocular toxocariasis was made. Systemic albendazole (ALBENZA®, GlaxoSmithKline, Brentford, UK) (800 mg twice daily) was prescribed with oral steroids (initial dose 30 mg/day), which resulted in the resolution of the vitreous opacity. However, the lesion near the arcade seemed to be white and enlarged. It was alleviated because the exudative lesion was dried out with treatments after 1 month (fig. ). However, 3 months after treatment initiation, the toxocariasis recurred during a treatment-free period (fig. ). In addition to the primary lesion, 2 other yellowish-white retinal lesions with vitreous flares appeared. Anthelmintic albendazole (ALBENZA 800 mg twice daily) and oral steroids (initial dose 20 mg/day) were resumed, and the infectious lesions were controlled gradually after another 2 months (fig. ). The BCVA in the right eye improved to 20/12.5. Two months after the retreatment was terminated, an intraretinal lesion was observed protruding from the retinal surface into the vitreous cavity (fig. ). Examination of this site by HP-OCT confirmed an elevated lesion protruding from the inner retina to the vitreous cavity (fig. ). The lesion was in the inner retinal layer, and the retinal pigment epithelium (RPE) and choroid were intact. The anthelminthic medication (800 mg twice daily) was resumed, and the elevated lesion resolved on OCT within the following 8 months (fig. ). One year after the therapy, signs of inflammation and the elevated retinal lesion had disappeared along with the reduction of blood anti- Toxocara canis antibody titer (fig. ). Unexpectedly, after 4 months, the elevated lesion reappeared (fig. ) and anthelmintic monotherapy (800 mg twice daily) was resumed, resulting in lesion control after 2 months (fig. ). The patient was subsequently treated with anthelminthic drug and steroid tapering. At the last examination after 21 months of follow-up, the BCVA was 20/12.5 with no evidence of active inflammation. The patient continues to be followed untreated without complications.

[[50.0, 'year']]

M

{'19892874': 1, '15838735': 1, '14798648': 1, '6463400': 1, '22548084': 2, '31762765': 1, '29895324': 1, '11520433': 1, '5726673': 1, '12834693': 1, '23095726': 1, '13794768': 1, '5553025': 1, '24575034': 2}

{'3324167-1': 1}

3934804-1

noncomm/PMC003xxxxxx/PMC3934804.xml

Chemotherapy and Surgical Approach with Repeated Endovascular Embolizations: Safe Interdisciplinary Treatment for Kasabach-Merritt Syndrome in a Small Baby

A 40-day-old female baby was referred to our division for an abdominal mass that had been diagnosed in another institution. Upon physical examination, she presented a pelvic mass extending to the gluteus and left thigh. Left lower-extremity paralysis was noted. Blood tests showed severe anemia and thrombocytopenia. In the following hours, her hematologic status worsened due to consumptive coagulopathy and the increasing volume of the mass. The CT scan done on admission showed an infiltrating pelvic mass (12 × 8 × 6 cm) extending through the sciatic foramen to the left gluteus and thigh (fig. ). The clinical aspect was pathognomonic of KMS.\nTransfusion, steroids and chemotherapy (vincristine, prednisone, rapamycin and cyclophosphamide) were started immediately. Coagulation parameters were normalized and the patient stabilized. Three months later, regression of the tumor volume (8 × 4 × 3 cm) was observed and interventional treatment became possible. Surgical dissection and ligature of the left internal iliac artery and sacral artery were performed. The main arterial supply of the pelvic mass was then embolized using a gelatin sponge (Spongostan Haemostatic®) under the fluoroscopy guidance of a portable C-arm tube in the operating room. At the control, complete regression of the pelvic mass and its vascularization were seen on the CT scan (fig. ). A clear sensitive-motor improvement was observed.\nThree months later, a second, similar, combined approach for the tumor occupying the left thigh was adopted. Surgical dissection of the left femoral artery and then partial embolization of the remaining tumor using a gelatin sponge (Spongostan Haemostatic) were successfully performed (fig. ). A third selective embolization of the arterial supply of the remaining tumor localized at the left gluteus region was planned for 2 months afterwards.\nA hybrid approach to the contralateral femoral artery was achieved in the angiographic room using Philips Allura flat-panel angiography. The right femoral artery was dissected and a 4-french, valved introducer was placed in the right common femoral artery. The contralateral, left lower-limb angiography done with a 4-french, shaped Cobra catheter (Glidecath, Terumo, Tokyo, Japan) showed the main supply of the hypervascular tumor in the gluteus (fig. ). Using a 2.7-french Echelon (EV3) microcatheter, selective catheterization of the profunda femoris artery feeders supplying the tumor was performed. Angiographic devascularization was achieved under fluoroscopy after injection of 0.8 ml of a nonadhesive, liquid glue, Onyx-18 (ethylene vinyl alcohol copolymer, Micro Therapeutics, Irvine, Calif., USA) (fig. ). Four months after treatment, the patient was stable.

[[40.0, 'day']]

F

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{'6039087-1': 1, '5308515-1': 1}

3934805-1

noncomm/PMC003xxxxxx/PMC3934805.xml

Primitive Neuroectodermal Tumor in an Ovarian Cystic Teratoma: Natural Killer and Neuroblastoma Cell Analysis

A 31-year-old, gravida 3, para 3, Caucasian woman, was referred with abdominal distension and general fatigue for 2 months. Pelvic ultrasound and computed tomography of the pelvis and abdomen showed a large left ovarian mass measuring 17 × 11 cm in diameter. Her preoperative serum cancer markers showed an elevated cancer antigen 125 (Ca125) level of 102 U/ml (normal range <35 U/ml) and a lactate dehydrogenase level of 511 U/l (normal range 125–220 U/l). Serum levels of cancer antigen 19.9, CEA and calcium were within normal ranges. She underwent exploratory laparotomy, which revealed a large, solid left ovarian mass with diffuse and nodular disseminations on the peritoneum, omentum, with retroperitoneal pelvic and periaortic lymph nodes enlargement. The left ovarian mass was removed and sent for pathologic evaluation. Immunohistochemistry showed positive cells for synaptophysin, neuron-specific enolase, chromogranin, CD56 and neurofilaments and confirmed the diagnosis of neuroblastoma arising in ovarian teratoma.\nWritten informed consent for publication of this case report was obtained from the patient. Approval was obtained from the Review Board of the Spedali Civili of Brescia. Informed consent was provided according to the Declaration of Helsinki.

[[31.0, 'year']]

F

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{}

3934807-1

noncomm/PMC003xxxxxx/PMC3934807.xml

Gastrointestinal Obstruction due to Solitary Lymph Node Recurrence of Alpha-Fetoprotein-Producing Gastric Carcinoma with Enteroblastic Differentiation

A 75-year-old woman with a history of diabetes mellitus, hyperlipidemia and osteoporosis consulted her local physician for occasional upper abdominal discomfort. She was referred and admitted to our hospital for evaluation and treatment. No abdominal tumor mass or superficial lymph nodes were palpable. Routine laboratory findings were unremarkable, with the exception of slightly elevated serum AFP (10.5 ng/ml). Gastroscopy revealed a 20-mm type 2 tumor, primarily in the pyloric region. Histological examination of gastroscopic biopsies confirmed gastric adenocarcinoma (moderately differentiated tubular adenocarcinoma), but no detailed information was obtained. Barium enema confirmed a type 2 tumor in the pyloric region. No obvious lymph node metastasis or remote metastasis was observed on contrast-enhanced computed tomography (CT).\nThus, with a provisional diagnosis of gastric adenocarcinoma, we performed laparoscopic distal gastrectomy with lymph node dissection. Macroscopically, the type 2 tumor was located near the pylorus and measured 20 × 15 mm (fig. ). It invaded vertically into the muscularis propria, and two regional lymph node metastases were observed microscopically. Histological examination demonstrated the presence of adenocarcinoma with a tubulopapillary growth pattern (fig. ). Tumor cells were cuboidal in shape with characteristically clear cytoplasm. Periodic acid-Schiff (PAS) stain showed that the clear cytoplasm was almost filled with fine granular glycogen (fig. ). The tumor cells were negative in mucin in either alcian blue-PAS stain, but also in the immunohistochemistry for MUC2 (goblet cell mucin) or MUC5AC (foveolar mucin).\nImmunohistochemically, tumor cells were positive for AFP (fig. ), carcinoembryonic antigen (CEA) (fig. ), caudal-type homeobox transcription factor 2 (CDX2) (fig. ) and common acute lymphoblastic leukemia antigen (CD10) (fig. ). AFP was mainly expressed in the cytoplasm whereas CEA was positive in the apical surfaces of tumor cells. CDX2 was expressed strongly in the tumor cell nuclei. CD10 was positive in the luminal surfaces in some tumor cells. The tumor was immunohistochemically negative in synaptophysin, chromogranin A and CD56 (NCAM), though a few chromogranin A and few synaptophysin-positive neuroendocrine cells were found scattering among clear carcinoma cells. Human chorionic gonadotropin was not found in tumor cells. The final diagnosis was AFP-producing gastric carcinoma with enteroblastic differentiation. Lymph node metastases showed similar histological and immunohistochemical results as the primary site.\nThe patient had an uneventful postoperative course, and AFP levels normalized to 3.7 ng/ml. She was followed without adjuvant chemotherapy because of her advanced age. However, 15 months after the initial surgery, serum levels of AFP increased to 279.4 ng/ml, and fluorodeoxyglucose positron emission tomography (FDG-PET) revealed No. 8p lymph node recurrence (fig. ). Therefore, chemotherapy combining cisplatin (CDDP) and etoposide (VP-16) was conducted for six courses. The size of lymph node recurrence initially decreased, but she consulted our hospital for upper abdominal pain and vomiting 26 months after the initial surgery. A CT scan revealed gastrointestinal obstruction near the anastomotic site of gastroduodenostomy due to No. 8p lymph node recurrence (fig. ). As it was difficult to continue chemotherapy, she underwent palliative gastrojejunostomy. The surgery temporarily restored adequate oral intake, but she received repeated blood transfusions for uncontrollable gastrointestinal bleeding, and serum levels of AFP increased to 8,634 ng/ml. 29 months after initial surgery, the patient died.

[[75.0, 'year']]

F

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{'6136504-1': 1}

3934809-1

noncomm/PMC003xxxxxx/PMC3934809.xml

Pancreatic Arteriovenous Malformation

A 59-year-old man was admitted to our hospital with intermittent upper abdominal pain. He had a history of cervical disc herniation, lumbar compression fracture and abdominal aortic aneurysm. Physical examination revealed slight tenderness in the left upper quadrant of the abdomen, and no masses were palpable. Laboratory data on admission showed: erythrocyte count 411 × 104/mm3 (normal 410–530), hemoglobin 13.7 g/dl (normal 14–18), leukocyte count 9,800/mm3 (normal 4,000–8,000), platelet count 27.1 × 104/mm3 (normal 15–40), serum total protein 6.8 g/dl (normal 6.5–8.0), total bilirubin 0.67 mg/dl (normal 0.2–1.0), aspartate aminotransferase 16 IU/l (normal 10–40), alanine aminotransferase 12 IU/l (normal 5–40), alkaline phosphatase 339 IU/l (normal 115–360), lactate dehydrogenase 256 IU/l (normal 119–229), γ-glutamyltranspeptidase 56 IU/l (normal 10–47), serum amylase 100 U/l (normal 33–116), blood urea nitrogen 14.0 mg/dl (normal 8–22), creatinine 0.8 mg/dl (normal 0.6–1.1), and C-reactive protein 1.71 mg/dl (normal 0–0.3). The serum level of the tumor marker carcinoembryonic antigen (CEA) was slightly elevated to 6.5 ng/ml (normal 0–5), and the carbohydrate antigen (CA19-9) level was elevated to 159.3 U/ml (normal 0–37).\nContrast-enhanced computed tomography (CT) showed numerous strongly enhanced abnormal vessels and a hypovascular lesion in the area of the pancreatic tail (fig. ). Angiographic study of the celiac artery confirmed racemose vascular networks in the tail of the pancreas and early venous return to the portal system (fig. ). Endoscopic retrograde pancreatography (ERP) revealed narrowing and displacement of the main pancreatic duct in the tail of the pancreas (fig. ). Screening esophagoscopy showed a 0-IIa+IIc type tumor in the lower thoracic esophagus and multiple Lugol-voiding lesions. Histological examination of esophagoscopic biopsies showed squamous cell carcinoma.\nBased on these findings, P-AVM and chronic pancreatitis or pancreatic cancer with esophageal cancer were diagnosed. Video-assisted thoracoscopic esophagectomy and distal pancreatectomy with partial transverse colectomy were performed. The operative time was 402 min, and the amount of blood loss was 956 ml. There was a honeycomb-like structure with collected vessels in the pancreas. The splenic vein was occupied by thrombi (fig. ).\nHistological examination of the resected pancreas revealed rupture of the dilated pancreatic duct with bleeding (fig. ), pancreatic stones (fig. ) and severe atrophy with fibrosis of the pancreatic parenchyma (fig. ). These findings were consistent with chronic pancreatitis. In addition, many abnormal vessels, some containing thrombi, were observed in the pancreas (fig. ). The final diagnosis was P-AVM consequent to severe chronic pancreatitis and esophageal carcinoma. The patient had an uneventful postoperative course, and CA19-9 levels normalized to 27.6 U/ml.

[[59.0, 'year']]

M

{'5656734': 1, '11984154': 1, '15290955': 1, '6969839': 1, '22477452': 1, '11180867': 1, '26943391': 2, '11291928': 1, '18953516': 1, '16670626': 1, '27081231': 2, '22129532': 1, '24574946': 2}

{'4813082-1': 1, '4747923-1': 1}

3934810-1

noncomm/PMC003xxxxxx/PMC3934810.xml

Aggressive Gastric Carcinoma Producing Alpha-Fetoprotein: A Case Report and Review of the Literature

In September 2011, a 65-year-old Caucasian man was admitted to our hospital after a 1-month history of epigastric pain, dyspepsia, and anorexia. He had been treated with a proton-pump inhibitor for 2 months. He had a history of type 2 diabetes mellitus, arterial hypertension, and dyslipidemia. At physical examination, intense abdominal pain in the upper abdominal quadrants was noted. Laboratory tests showed elevated aspartate transaminase 107 U/l (reference range 10–40 U/l), alanine transaminase 89 U/l (reference range 10–40 U/l), gamma-glutamyl transpeptidase 627 U/l (reference range 10–40 U/l) and alkaline phosphatase 309 U/l (reference range 40–130 U/l); total bilirubin was 1.07 mg/dl (reference range 0.3–1.0 mg/dl). Ultrasonography showed a remarkable and irregular enlargement of the liver with multiple and diffuse nodules in the right hepatic lobe (the largest measured 5 cm in diameter) and a unique large mass (15 × 9 cm) in the left lobe. Abdomen computer tomography scans showed multiple nodules in the liver, suspected to be metastatic lesions, portal vein thrombosis, multiple adenopathies along the lesser curvature of the stomach, the mesenteric root and the lombo-aortic region as well as a thickening of the gastric lesser curvature wall (fig. a, b). Tumor markers were: alpha-fetoprotein (AFP) 209093 U/ml (reference range 0–10 U/ml), Ca 19.9 170,5 U/ml (reference range 0–39 U/ml), and Ca 125 52,2 U/ml (reference range 035 U/ml); other markers were found to be in a normal range, in particular CEA and Ca 72.4. Markers for hepatitis B and C were negative (HBsAg, HBV-DNA and HCV IgG). An esophagogastroduodenoscopy showed a 4 cm ulcer on the lesser curvature of the stomach (fig. ). At the pathologic examination, the biopsy revealed a gastric adenocarcinoma. The biopsy of one of the hepatic nodules showed a poorly differentiated adenocarcinoma, similar to that of a gastric biopsy. Immunohistochemical findings showed a positivity for CAM 5.2, CDX 2 CK 20 and a negativity for CD10, TTF1, sinaptophysin and chromogranin A, therefore supporting a diagnosis of metastasis of gastric adenocarcinoma (fig. ). One week later, the patient's condition quickly worsened (Performance Status 3 in accordance with the European Cooperative Oncology Group scale). He presented jaundice, abdominal pain and, at palpation, the appearance of a solid mass in the left upper quadrant of the abdomen and liver enlargement. Levels of transaminases, gamma-glutamyl transpeptidase and alkaline-phosphatase increased; total bilirubin was 19.6 mg/dl and the conjugated one was 12.8 mg/dl. Abdominal ultrasonography showed, with respect to the previous one, an increased number and size of the hepatic nodules as well as a biliary tract dilatation. Tumor markers were found to be increased further: AFP 470396 U/ml, Ca 19.9 354.0 U/ml, and Ca 125 113.7 U/ml. A chemotherapeutic approach was excluded due to the patient's poor condition, his performance status and the altered liver function tests. The patient was discharged and received the best possible supportive care. He died 15 days later.

[[65.0, 'year']]

M

{'12435906': 1, '12931013': 1, '5426134': 1, '4111729': 1, '20632272': 1, '24083471': 1, '20740583': 1, '7541181': 1, '28693208': 1, '2446735': 1, '11940206': 1, '24575023': 2}

{}

3934811-1

noncomm/PMC003xxxxxx/PMC3934811.xml

Unilateral Optic Disc Papilloedema following Administration of Carboplatin Chemotherapy for Ovarian Carcinoma

A 48-year-old woman with a positive BRCA1 gene mutation was referred with a pelvic mass. Ca-125 was >4,000 and a CT scan confirmed the presence of large bilateral ovarian tumours. She underwent debulking surgery, and histology revealed completely resected stage 3b high-grade endometrioid ovarian carcinoma. She was therefore referred to Oncology for consideration of adjuvant chemotherapy. She was well, with a performance status of 0 and no medical history of note. EDTA glomerular filtration rate was measured to be 98 ml/min.\nShe was prescribed the regime of weekly paclitaxel 80 mg/m2 (total dose 150 mg) with concurrent 3-weekly carboplatin AUC 6 (total dose 740 mg) []. During the first cycle of paclitaxel, she suffered an immediate allergic reaction; therefore, subsequently her regime was switched to single-agent carboplatin at the previously calculated dose. She received 2 cycles without incident, but her third cycle was delayed by 1 week owing to thrombocytopaenia (platelet count 77 × 109/l), and her fourth was delayed by a further week due to neutropaenia (neutrophil count 0.9 × 109/l). At review prior to her fourth cycle, she mentioned an incidental finding of a blot haemorrhage at a routine optometrist visit, which was asymptomatic. Diabetic screening was performed at this stage in view of a strong family history, but was negative.\nFive days after her fifth cycle of carboplatin, she awoke with new-onset blurred vision in her left eye. An ophthalmology review showed left-sided disc oedema (fig. ) with normal optic nerve function tests and 6/24 visual acuity. No abnormality was seen in the right eye with 6/6 visual acuity. A CT scan of the head and orbits was performed which showed no evidence of metastases or raised intracranial pressure. An autoimmune screen was performed which did not reveal any explanation for her visual symptoms. Fundus fluorescein angiography showed bilateral intense late disc leakage with no evidence of vasculitis (fig. ). In view of her low platelet count and no clinically evident disc oedema in the right eye, a lumbar puncture was not performed.\nThe diagnosis was felt to be that of carboplatin-induced unilateral disc oedema. At this point, her chemotherapy was stopped in view of a radiological and biochemical remission and her visual symptoms were monitored. She was also started on prednisolone 40 mg daily. Her symptoms did not improve with steroids, however, and the dose was gradually tapered down. Five months after the initial review, she has developed left optic disc atrophy with 6/18 visual acuity, while the right eye remains asymptomatic.

[[48.0, 'year']]

F

{'32160941': 1, '14750566': 1, '1346911': 1, '20740148': 1, '8518227': 1, '16414359': 1, '19767092': 1, '24575012': 2}

{}

3934813-1

noncomm/PMC003xxxxxx/PMC3934813.xml

Suspected de novo Hepatitis B in a Patient Receiving Anti-Tumor Necrosis Factor Alpha Therapy for the Treatment of Crohn's Disease

A 41-year-old woman had abdominal pain, diarrhea and fever in July 2006. Because of persistent symptoms, she was hospitalized in August 2006. A cobblestone-like appearance was seen in the sigmoid colon upon colonoscopy. On the basis of the results of detailed examination, she was diagnosed as having colonic CD. Since she had severe CD with a Crohn's disease activity index (CDAI) of 373 and needed prompt and effective treatment, treatment with IFX at 5 mg/kg and 6-mercaptopurine (6-MP) at 30 mg/day was initiated in September 2006. Laboratory findings at the initiation of treatment included an aspartate transaminase (AST) level of 22 IU/l, an alanine transaminase (ALT) level of 22 IU/l and negative HBsAg. She was not tested for anti-HBsAb and anti-HBcAb. At week 10 of IFX therapy, symptoms were alleviated (CDAI: 56), and remission was successfully induced. IFX was administered at weeks 0, 2 and 6 and every 8 weeks thereafter, and remission was successfully maintained. Colonoscopy about 1 year after the initiation of treatment revealed colonic mucosal healing with scarring. However, about 1 year after the initiation of IFX therapy, she began to experience delayed-onset hypersensitivity reactions to IFX such as fever and joint pain. We thus considered switching biologic therapy from IFX to ADA. On October 8, 2010, 4 years and 1 month after the first administration of IFX, the 30th and final administration of IFX was given, and her hepatic function was normal (AST 16 IU/l, ALT 21 IU/l). On December 12, 2010, hepatic function test immediately before the first administration of ADA revealed mild hepatic dysfunction (AST 61 IU/l, ALT 54 IU/l). Since no abnormal hepatic function had been found before and the cause was unknown, she received 160 mg of ADA on that day and was followed up thereafter. On December 17, 2010, although hepatic enzyme levels were still high, she received 80 mg of ADA.\nOn January 4, 2011, since she felt generalized malaise with an AST of 322 IU/l and an ALT of 527 IU/l, she was hospitalized for detailed examination and treatment (fig. ). Examinations revealed HBsAg of 250 IU/ml and HBV DNA of >9.0 log copies/ml. HBV genotype was type C, and anti-hepatitis C virus antibody (anti-HCVAb) was negative (table ). Virological testing of a sample obtained from her husband revealed negative results for HBsAg and anti-HBcAb and no detection of HBV DNA. Sexual transmission of HBV from her husband and others was ruled out. Abdominal echo and computed tomography examination showed no evidence of chronic hepatic disorder. Liver biopsy revealed mild fibrosis (A2/F1). These findings supported the presence of hepatic disorder due to HBV. Although no data on HBV DNA and anti-HBcAb before onset were available, her clinical course suggested de novo hepatitis B due to reactivation of HBV related to the treatment with IFX and 6-MP.\nOn January 5, 2011, antiviral treatment with ETV at 0.5 mg/day was initiated. Treatment with ADA was suspended after the first two administrations, but treatment with 6-MP was continued. Ten sessions of granulocytapheresis were conducted to maintain remission of CD. AST and ALT levels peaked at 440 IU/l and 624 IU/l, respectively, on January 14, 2011, and then decreased gradually over time. On March 29, 2011, hepatic function returned to normal (AST 30 IU/l, ALT 24 IU/l). Since negative HBV DNA results were obtained in June 2011, treatment with ADA was reintroduced (fig. ). Since then she has continued treatment with ETV and has maintained normal levels of AST, ALT and HBV DNA without recurrence of hepatitis B. 10 months after treatment with ETV, her anti-HBsAb turned positive. She has also continued ADA and has maintained mucosal integrity and clinical remission without gastrointestinal symptoms.

[[41.0, 'year']]

F

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{}

3935073-1

noncomm/PMC003xxxxxx/PMC3935073.xml

Translocation Renal Cell Carcinoma t(6;11)(p21;q12) and Sickle Cell Anemia: First Report and Review of the Literature

A 27-year-old woman from the French West Indies who was followed at Pointe-a-Pitre University Hospital for a sickle cell anemia was referred for a left renal tumor. She had a personal history of cholecystectomy and acute thoracic syndrome.\nA left renal mass issuing from the renal sinus was discovered incidentally on an abdominal ultrasound performed for the patient's annual sickle cell anemia checkup. The clinical examination revealed a weight loss of 3 kilograms. There was no hematuria, no flank pain, no asthenia, and no lumbar contact (ECOG=0).\nA computed tomography (CT) scan confirmed a left renal tumor of 34 mm×86 mm, which seemed to develop from the renal medulla, that was heterogeneously enhanced with contrast agent () and several perihilar adenopathy (the largest being 17 mm). Thoracic CT scan revealed no metastasis.\nA left laparoscopic radical nephrectomy was performed after specific preoperative care for sickle cell disease: exchange transfusion for hemoglobin S <40%, hyperhydration, chest physiotherapy, and prevention against acidosis and hypothermia. There were no postoperative complications.\nFinal pathological examination showed a 60-mm well-defined medio-renal tumor with a brownish-red appearance. The 11 removed lymph nodes were negative. Microscopically, the tumor showed biphasic growth patterns consisting of nests of eosinophilic and clear cells (). Immunohistochemical analysis was performed, using a panel of antibodies including CD10, RCC marker, vimentin, α-methylacyl-coenzyme A racemase (p504s), human melanoma black 45 (HMB45), microphthalmia transcription factor (MiTF), TFE3, and TFEB. The tumor cells revealed diffuse nuclear TFEB staining, demonstrating a translocation RCC t(6;11)(p21;q12). They were also positive for vimentin, p504s, and HMB45 but in less than 10% of tumor cells. This tumor was classified as stage pT1bN0M0 Fuhrman grade 2.\nTwelve months after the surgery, the patient was in excellent condition and no recurrence was noticed on the CT scan.

[[27.0, 'year']]

F

{'18344867': 1, '12475675': 1, '18278810': 1, '19494850': 1, '31600176': 1, '19393983': 1, '15848741': 1, '15644781': 1, '17633988': 1, '7528470': 1, '24578813': 2}

{}

3935074-1

noncomm/PMC003xxxxxx/PMC3935074.xml

Primary Mature Cystıc Teratoma Mimickıng an Adrenal Mass in an Adult Male Patient

A 54-year-old previously healthy male patient developed acute, left upper quadrant abdominal and left flank pain. This pain lasted several hours and then resolved spontaneously. When he was admitted to the hospital, on ultrasound and an abdominal and pelvic CT scan, he was found to have an 8-cm×7-cm×6-cm solid and cystic mass in the left (surrenal area) retroperitoneum containing bone and multiple soft tissue densities (). The tumor had calcification and was not well enhanced. There was no evidence of distant metastasis. The tumor was also examined by magnetic resonance imaging (MRI). We also diagnosed that the tumor originated in the left adrenal gland, because the normal adrenal gland could not be recognized by CT or MRI. The patient had hypertension for which he was taking amlodipine 5 mg daily. However, plasma levels of catecholamines, rennin, aldosterone, adrenocorticotropic hormone, and cortisol were within the normal range. Tumor markers, such as serum alpha-fetoprotein, lactate dehydrogenase, carcinoma antigen 125, neuron-specific enolase, and carbohydrate antigen 19-9, were examined because it was possible that the tumor did not originate from the adrenal gland.\nThe patient underwent resection of the lesion through a flank incision by 11th rib resection. The retroperitoneal dissection was tedious and difficult, but the mass was excised in its entirety (). The left adrenal gland was normal. The resected retroperitoneal cystic mass measured 8 cm×7 cm×6 cm and weighed 153 g (). The final pathological evaluation of the tumor was a mature teratoma without malignant components. Macroscopically, the specimen was a 6 cm ×5 cm ×4 cm fluctuating mass. On incision, cystic material came off and a thinly walled cyst was left. The cyst wall thickness was approximately 1 mm with only some minor excrescents. Histologically, the cystic cavity was lined by ciliated columnar cells () and the excrescents were hyalin cartilage islands under this epithelium with some lobules of seromucinous glands (). No immature elements were seen and the diagnosis was a mature cystic teratoma pathologically. Because these tumors usually represent metastasis from other primary sites, additional imaging with CT of the chest and scrotal ultrasonography was performed. No other primary tumor was identified. Therefore, we diagnosed the mass as a primary retroperitoneal teratoma. After 12 months of follow-up, the patient was free of recurrence.

[[54.0, 'year']]

M

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{'5954807-1': 1}

3935113-1

noncomm/PMC003xxxxxx/PMC3935113.xml

Identification of a novel mutation in the CHD7 gene in a patient with CHARGE syndrome

A 21 day-old girl was transferred to Samsung Medical Center due to extubation failure. The baby was born at the 35+1 weeks of gestation by emergency cesarean section due to spontaneous rupture of the membranes and premature labor. Apgar scores were 2 at 1 minute and 8 at 5 minutes. When the baby was born, she did not cry and became cyanotic and lethargic. Positive pressure ventilation was not effective, so endotracheal intubation was performed in the delivery room. At birth, weight, length, and head circumference were 2,030 g (25th percentile), 49 cm (75th-90th percentile), and 35 cm (>90th percentile), and she had low set ears. A large patent ductus arteriosus (PDA) was found and ligated 12 days after birth.\nWe conducted osteomeatal unit computed tomography (CT) to find the cause of extubation failure. CT scan showed bilateral choanal atresia, atresia of the left external auditory canal, and stenosis of the right auditory canal (). Thirty-five days after birth, she underwent a repair operation of the choanae, and we tried to extubate again. However, she was reintubated with an endotracheal tube because of sustained desaturation and tachypnea. We considered that the cause of extubation failure was an upper airway problem. She was identified to have postglottic stenosis through laryngomicrosurgery (). Forty-two days after birth, a tracheostomy was performed, and respiration stabilized without respiratory support.\nThe patient had a left aortic arch with aberrant origin of the right subclavian artery on chest CT and minimal left pulmonary artery hypoplasia without focal stenosis on echocardiography. Based on these results, we conducted the ophthalmic examination to identify other features of CHARGE syndrome, and found coloboma in her both eyes. Because she met two out of the three major criteria and two of the five minor diagnostic criteria, we concluded that she had typical CHARGE syndrome.\nWritten informed consent was obtained for a molecular genetic analysis of the CHD7 gene mutation from the parents. Direct sequencing analysis of the CHD7 gene was performed with genomic DNA isolated from peripheral blood leukocytes. As a result, a c.3896T>C (p. leu1299Pro) missense mutation in exon 16 of the CHD7 gene was detected, which has not been reported previously (). A familial study revealed that neither of the parents had the mutation. The polymorphism phenotyping program and the sorting intolerant from tolerant program predicted "possibly damaging" and "not tolerated." The amino acid residue L1299 is highly conserved across different species, and the L1299P variation does not occur in 200 control chromosomes from 100 healthy individuals.\nAfter discharge at 2 months of age, she has been followed up at the outpatient clinic regularly. At present, she is 11 months old and is using a hearing aid because of sensorineural hearing loss. Since she has had recurrent aspiration; thus G-tube feeding is required. Her height was 10th-25th percentile, body weight was 5th-10th percentile, and head circumference was 10th-25th percentile. The patient can sit alone but cannot stand with support. Hypoplasia of both kidneys was identified by abdominal ultrasonography, which was performed at 5 months of age. She has met two of the three major criteria and four of the five minor diagnostic criteria for CHARGE syndrome.

[[21.0, 'day']]

F

{'16118347': 1, '15637722': 1, '16169932': 1, '9932584': 1, '2317068': 1, '26538304': 1, '16400610': 1, '15890414': 1, '15666308': 1, '15672384': 1, '19159393': 1, '20624498': 1, '1999835': 1, '22461308': 1, '21041284': 1, '16615981': 1, '25553296': 2, '24578717': 2}

{'4272946-1': 1}

3935114-1

noncomm/PMC003xxxxxx/PMC3935114.xml

A novel PRF1 gene mutation in a fatal neonate case with type 2 familial hemophagocytic lymphohistiocytosis

The proband was a 29-day-old female infant who was admitted to our institution because of four days of mild fever and rapid breathing, moaning, and mottling. She was the second child of a nonconsanguineous couple, born at 38+4 weeks of gestation with a birth weight of 3,500 g. Family history revealed that her sister died at 28 days of age from sepsis with rapidly progressive clinical manifestations of acute hepatitis, gastrointestinal bleeding, and pulmonary hemorrhage. The initial neonatal course of the proband was not remarkable until she developed respiratory distress and uncontrolled fever on postnatal day 29. She subsequently experienced significant bleeding and bruising from coagulopathy and had hepatosplenomegaly. Her condition deteriorated, and she developed lactic acidosis, 22.38 mmol/L (normal range, 0.5-2.2 mmol/L), which required mechanical ventilation and continuous veno-venous hemodiafiltration ().\nLeukopenia and anemia slowly developed along with thrombocytopenia. The 7th day of the admission, laboratory finding revealed leukocytes level at 4,010×103/µL, thrombocytes 28×103/µL, hemoglobin 9.7 g/dL, absolute neutropenic counts 960×103/µL, triglyceride 774 mg/dL, ferritin 165,000 ng/mL, fibrinogen 93 mg/dL.\nLaboratory workup for infection revealed no diagnostic findings; however, on day 31, broad-spectrum antibiotics and intravenous immunoglobulin and corticosteroid were administered empirically with a presumptive diagnosis of neonatal sepsis. Since the patient's manifestations met the criteria for HLH 2004, a bone marrow study was performed on day 35, which revealed frequent active hemophagocytic histiocytes. As the first step of a diagnostic workup for FHL, we performed flow cytometric analysis for the intracytoplasmic perforin expression of cytotoxic cells using the proband's peripheral blood sample, as previously described with some modifications. We observed a complete absence of perforin expression in the proband's natural killer (NK) cells and CD8+ T cells. The experiments using her parents' blood samples demonstrated partial deficiency of cytoplasmic perforin in NK cells and complete deficiency in CD8+ T cells (). Based on the results of flow cytometric analysis, we obtained written informed consent from the parents and proceeded with a molecular genetic test for FHL2 by direct sequencing analysis of the PRF1 gene, as previously described). As a result, the proband was shown to have two deleterious point mutations of PRF1, c.65delC [p.Pro22Argfs*2] and c.1090_1091delCT [p.Leu364Glufs*93] (. And no other mutation has been found in direct sequencing analysis of the PRF1 gene. Her parents were heterozygous carriers of the respective mutant alleles (compound heterozygous for the c.65delC mutation (of maternal origin; novel) and the c.1090_1091delCT mutation (of paternal origin; known), confirming the compound heterozygous status of the two mutations in the proband. Based on the diagnosis of FHL2, the patient received HLH-2004 chemotherapy with cyclosporine, etoposide, and dexamethasone. She experienced hypotensive episodes that were initially responsive to fluid resuscitation and medications but that eventually progressed to refractory status, leading to renal failure. She died from multiorgan failure on day 43.

[[29.0, 'day']]

F

{'20383897': 1, '32909006': 1, '28029005': 1, '19131769': 1, '20015888': 1, '16188649': 1, '32194620': 2, '29152263': 2, '31396234': 1, '34671419': 1, '11756153': 1, '16860143': 1, '19595804': 1, '12716377': 1, '17151879': 1, '34381049': 1, '21653941': 1, '24578718': 2}

{'5676276-1': 1, '7064636-1': 1, '7064636-2': 1, '7064636-3': 1, '7064636-4': 1}