A 40-year-old male presented with facial puffiness and edema of lower extremities of 7 months duration. He had no history of bony pain, oral ulcers, arthralgia, skin rashes or any features suggestive of connective tissue disorder. On examination patient had pitting edema of lower limbs with no pallor, icterus or skin rashes. Blood pressure was 110/70 mm Hg. Systemic examination was normal. Urine microscopic examination revealed 4 + proteinuria with no active sediments. Twenty-four hours urine protein was 6.9 g. Renal function tests revealed blood urea nitrogen of 25 and serum creatinine of 2.06 mg/dl respectively. Patient had hemoglobin of 138 g/L, total leucocyte count of 11 × 109/L with normal differential counts and platelet count of 3.4 lakhs/micro litre. Serum total protein and albumin were 5.5 and 2.5 g/dl respectively. Liver function tests were normal. Patient's serum tested negative for hepatitis B surface antigen, antibodies for hepatitis C virus and human immunodeficiency virus-I/II. Antinuclear factor, rheumatoid factor, cryoglobulin and antineutrophil cytoplasmic antibodies were negative and serum complement levels were normal (C3 = 110 mg/dl and C4 = 20 mg/dl). Serum total cholesterol and triglyceride were 352 and 328 mg/dl. Ultrasonography revealed normal sized kidneys. With clinical diagnosis of nephrotic syndrome, ultrasound guided percutaneous kidney biopsy was performed.
Light microscopic examination showed eight glomeruli, all showed diffuse thickening of glomerular capillary walls with lifting up of podocytes. Jones silver stain revealed spikes []. No endocapillary or mesangial proliferative, tubulointerstitial and vascular alterations were noted. Immunofluorescence showed granular polyclonal IgG, kappa light chain and deposition of monoclonal IgG3 and C3 along glomerular capillary loops with intensity being 3 + on a scale of 0-3+ []. IgA, IgM, lambda light chain and complement (C1q) were negative. No deposition along tubular basement membranes was observed. Therefore, a diagnosis of MGN with monoclonal IgG3-kappa deposition was made on renal biopsy.
Urine and serum immunoelectrophoresis showed no monoclonal proteins. Serum kappa and lambda light chain concentrations were 1088 and 208 mg/dl respectively with κ/λ ratio of 5.2 (normal - 0.26-1.52). Skeletal survey did not reveal any abnormalities. Bone marrow examination revealed 3% plasma cells. With a diagnosis of monoclonal gammopathy with MGN, patient was started on treatment with 6 monthly cycles of velcade-thalidomide-dexamethasone (VTD) regimen (bortezomib - 1.3 mg/m2 on 1st, 4th, 7th and 11th days, thalidomide - 200 mg/day and dexamethasone - 30 mg/kg on 1st-4th and 7th-11th days of month). After 6 monthly cycles of VTD therapy, patient had reduction in proteinuria to 3.1 g/day with serum creatinine and albumin of 0.9 mg/dl and 3.1 g/dl respectively. Repeat serum free light assay revealed kappa and lambda light chain of 29 and 14 mg/dl respectively (κ/λ ratio: 2.01).


A 51-year-old woman presented to our breast clinic with a 2-year history of a self-palpated left breast mass. The patient underwent breast augmentation 12 years earlier with the insertion of bilateral subpectoral saline breast implants which required cosmetic revisions due to malposition and were subsequently positioned subglandularly.
Breast imaging by mammography and ultrasonography revealed a 0.8-cm well-defined mass which was radiographically reported to be consistent with fibroadenoma at an outside institution. She refrained from 6-month surveillance scans until she noticed an increase in size of the mass. Despite its reported benign appearing nature on imaging, the patient desired removal of the lesion.
Prior to excision, a repeat US and US-guided FNAB were performed by a radiologist, and immediate assessment for specimen adequacy was performed by the cytopathologist. Direct smears were made from the aspirated material and fixed in Carnoy's solution for Papanicolaou staining and also air-dried for Diff-Quik staining. The syringe used for the aspiration was rinsed in Roswell Park Memorial Institute solution which was centrifuged. The cell pellet that was generated was sufficient to prepare a single cytospin which was stained by Papanicolaou method.
The FNAB smears showed high cellularity with many dispersed single intact atypical epithelial cells and very few loose clusters. The epithelial cells exhibited a striking plasmacytoid appearance with a moderate amount of granular eosinophilic cytoplasm. The nuclear chromatin of the cells was fine without conspicuous nucleolus. Rare mitotic figures were identified. There was no evidence of mucin vacuoles in the atypical cells. Naked bipolar nuclei were absent in the background of the smears. The overall cytomorphological findings were consistent with a well- to moderately-differentiated mammary carcinoma with plasmacytoid features, raising concern for a neuroendocrine carcinoma []. Due to the lack of cell block material from the FNAB, a CNB was subsequently performed under US guidance [].
Ancillary testing on the CNB showed that the tumor was positive for estrogen receptor (30% of tumor cells; moderate intensity) and progesterone receptor (90% of tumor cells; strong intensity) and negative for human epidermal growth factor receptor 2 (HER2/neu) protein overexpression (1 + staining in 10% of tumor cells). The tumor cells expressed synaptophysin (90% of cells) and chromogranin (50% of cells) while negative for CDX-2. The tumor was classified as invasive ductal carcinoma with features consistent with primary neuroendocrine carcinoma of the breast based on the FNAB and CNB findings.
The patient underwent bilateral total mastectomies with sentinel lymph node dissection. Histological examination confirmed the diagnosis of invasive neuroendocrine carcinoma, intermediate nuclear grade, and Nottingham histologic grade 2. The invasive tumor demonstrated small areas of extracellular mucin. Focal lymphovascular invasion was identified, and isolated tumor cells were found to be present in one sentinel lymph node upon keratin immunohistochemical analysis.


A 13-year-old boy presented with sudden onset of painless blurring of vision in his both eyes. He had a history of exanthematous fever 20 days back and was treated symptomatically. There were healing skin lesions on face, abdomen and upper and lower limbs []. On examination, the un-corrected visual acuity in oculi uterque (OU) was 3/60, and best corrected visual acuity was 6/6; N-8 with − 3.00 Diopter spherical (DS) correction. Intraocular pressure (IOP) was 14 and 15 mm Hg, respectively in the right and left eye. Anterior chambers (AC) were quiet and shallow, and on gonioscopy angles were open to anterior trabecular meshwork. By immersion A-scan ultrasonography AC depth (ACD) were 3.47 and 3.41 mm, and lens thickness were 3.64 mm and 3.61 mm, respectively in the right and left eye. Pupils were symmetrical, round, regular and reactive to light. Fundus examination was significant for the presence of bilateral retinal folds in the macular region bilaterally []. B scan ultrasonography showed features of annular cilliochoroidal effusion []. A dermatology opinion was taken, which pointed toward varicella, and serum was positive for the specific immunoglobulin M (1.14) antibodies. Patient was started on oral prednisone (50 mg/day) and topical homatropine 2% thrice daily.
At 1 week follow up unaided distance vision OU had improved to 6/6, and near vision was N-24. The oral prednisolone and homatropine was stopped over 2 weeks. At 4 weeks, unaided vision OU was 20/20; N-6. The AC angle was open to sclera spur on gonioscopy and fundus examination was un-remarkable.


A 31-year-old Caucasian, military dependent, female presented to the Naval Medical Center Emergency Department with a chief complaint of “chest discomfort”, further described as a “burning sensation that started in (her) throat and migrated down to (her) chest”. This symptom occurred once before, 3 days prior to this presentation. She denied radiation of symptoms, dyspnea, or any other associated symptoms. She had not experienced these symptoms in the past. Her past medical history was significant for being 4 months status post, uncomplicated, right tympanoplasty as treatment for a middle ear cholesteatoma, irritable bowel syndrome, and adenoid cyst carcinoma 5 years past. Review of her current, active medications includes a chloramphenicol, sulfanilamide, fungizone, and hydrocortisone otic insufflation combination (CSF + HC) for an otitis of her right ear. The medication was started approximately 2 weeks prior to her presentation. She noted burn-like throat pain that migrated down to her chest occurring twice after starting this treatment. Review of her social and family history revealed no risk factors for cardiopulmonary disease and was otherwise noncontributory. She was a nonsmoker and marginal alcohol consumer.
On physical exam, the patient was comfortable, pleasant, in no acute distress and breathing with normal oxygen saturation at a regular rate. Her vitals were within normal limits and stable. Heart, lungs, and vascular exam were normal. On head, ears, eyes, nose, and throat (HEENT) examination; the right tympanic membrane appeared to have postsurgical changes with a noted defect; otherwise the ear canal was clean without discharge or erythema. Oropharyngeal exam was unremarkable. The remainder of her exam was unremarkable. An electrocardiogram (ECG) was obtained which demonstrated a normal sinus rhythm. Her symptoms resolved completely after consuming a gastrointestinal cocktail. She was monitored for 2 h with no change in status and continued resolution of symptoms. She was discharged with an H2 blocker and referred to her otolaryngologist for evaluation.


The case we present here is about a 49-year-old male patient who had a history of HBV related chronic liver failure and HCC for 2 years developed multiple osteoblastic bone metastasis [] and bone pain was refractory to analgesic treatments (tramadol, fentanyl). SM-153 treatment was planned for palliation of bone pain. He was mildly decompensated in terms of liver disease, and there was no significant finding except minimal pleural effusion and a chronic infiltrative area at chest X-ray graphic examination taken before the procedure []. Laboratory findings of patient at the day of treatment were Hb: 9.3 g/dl, Plt: 66.000 and white blood cell (WBC): 3400; creatinine 0.6 mg/dl, glomerular filtration rate (GFR): Normal and total bilirubin: 5.09 mg/dl. Sm-153 intravenous (IV) bolus (1 mCi/kg) and 100 cc %0.9 NaCl 1 h IV infusion was applied for pain palliation. Sm-153 accumulation at bone metastatic sites was shown by total body scintigraphic imaging at 24thh. Patient had dyspnea and the cardiac shadow was more prominent at chest X-ray taken 2 days after the SM-153 treatment []. Massive pericardial effusion was determined at echocardiography. Bilateral pleural effusion, massive pericardial effusion and subtotal collapse of right lung were noticed at thorax CT []. Pericardiotomy was performed, and the effusion was hemorrhagic. No malignant cell was determined at cytologic examination, and there was no proliferation in culture of pericardial fluid. Approximately, 500 cc/day pericardial hemorrhagic fluid was drained. The bilirubin level increased up to 30 mg/dl and the patient died 20 days after the application of Sm-153.


A 56-year-old woman was admitted to our clinic with a history of sore throat for three days. Her pain was also referring to her left ear. In her head and neck examination, otoscopy and anterior rhinoscopy findings were insignificant. In her oropharyngeal examination, pharyngeal arches and posterior pharyngeal wall were hyperemic and edematous with an accompanying left upper jugular lymphadenopathy. In addition, we examined a uvula-like elongation from the middle of the soft palate, apart from the original uvula (). In her medical history, we learned that the patient was aware of her soft palate anomaly; however, she had used to live with this without any complaint. We administered an oral empiric antibiotic therapy and anti-inflammatory drug for acute lymphadenitis. After two weeks and with a ten-day antibiotic therapy, patient was free of lymphadenopathy or any complaints.


A 15-year-old male presented to the emergency department following a direct blow from his bicycle handlebars to his groin. His chief complaint was pain and swelling in the right inguinal region. His abdominal examination was unremarkable, except for a small abrasion and visible mass in his right groin. Peripheral pulses were palpable bilaterally. After exclusion of other abdominal or chest injuries, he was discharged on the same day with a diagnosis of right groin haematoma and follow-up in 6–8 weeks.
He presented to his general practitioner 2 weeks later with claudication. His symptoms included pain and paraesthesia in his right buttocks on mobilization >100 m. He was found to have absent peripheral pulses in his right leg, although it appeared well perfused with a normal capillary refill. An arterial Doppler ultrasound showed a right external iliac artery thrombus occluding the proximal two-thirds of the vessel. A computed tomography scan of the abdomen and pelvis with intravenous contrast identified complete occlusion of the right external iliac artery ∼1 cm beyond its origin. However, the common femoral artery and profunda femoris remained patent via collaterals (Fig. ).
He underwent a right external iliac thromboendarterectomy with patch repair using a saphenous vein graft. A suprainguinal incision was initially made in attempt to expose the proximal external iliac artery. Due to its extent, retrieval of the entire thrombus was incomplete and a second groin incision near the junction of the common femoral with external iliac (at the inguinal ligament level) was required. The patient subsequently underwent a proximal and distal thrombectomy, and the arteriotomy was extended between the two incisions identifying an intimal stricture with significant fibrosis (Fig. ). An intimal flap was identified in the distal region and tacked down with a 7-0 prolene suture. The long saphenous vein was harvested and the defect closed with patch repair extending from proximal iliac to proximal femoral vessel. He had an uneventful recovery. At the initial 2-week follow-up, the vessels remained patent and patient was progressing well. Subsequently, regular biannual follow-up was planned to monitor for any longer-term complications.


A 27-year-old woman presented with sensory disturbance along the radial three and half digits of her left hand. She had noticed a soft tissue swelling on volar surface of her wrist for six years and sometimes had experienced paresthesia and numbness in her radial three and half digits. Her symptoms worsened during the preceding year. On examination, results of Phalen’s test and Tinel’s sign were positive. Nerve conduction study revealed severe latency of the median nerve conduction velocity at carpal tunnel, consistent with severe CTS. Sonography of the swelling demonstrated enlarged cross-section of the median nerve (4.3 cm2; normal cross-section, 5.78 ± 0.9 mm2) under the flexor retinaculum with hypoechoic cable-like neural fascicle separated by hyperechoic fat (). On ultrasonographic evaluation, the median nerve involvement was extended to the proximal forearm at the level of pronator teres muscle (). Magnetic resonance imaging (MRI) of distal forearm demonstrated fusiform enlargement of the median nerve with low signal intensity of neural bundles in T1 and T2 sequences, surrounded by high signal intensity substratum representing fat. On axial image, the lesion demonstrated a characteristic “coaxial cable-like” appearance (). She underwent carpal tunnel release because of severe secondary CTS. At surgery, enlargement of the median nerve under the flexor retinaculum in the distal forearm and carpal tunnel was observed (). A small biopsy sample was obtained from the perineurium. No further treatment was performed. Histopathologic study demonstrated infiltration of perineurium with mature fibrous and adipose tissue (). The patient had unremarkable postoperative course with complete resolution of her symptoms and remained asymptomatic at the four-month follow-up course.


A 42-year-old woman presented with exacerbations of palpitation and dyspnea that she had been suffering from since her childhood. She had no history of trauma. Upon admission, the patient’s blood pressure was normal and an apical systolic murmur was heard during cardiac auscultation. Also detected was a thrill at the left side of the neck and an audible bruit over the left mandibular angle. Electrocardiography showed evidence of left ventricular hypertrophy and the chest X-ray revealed mild cardiac enlargement. Transesophageal echocardiography (TEE) demonstrated severe left ventricular enlargement, mild right ventricular enlargement, severe mitral regurgitation, moderate tricuspid regurgitation and moderate pulmonary hypertension. The cardiac ejection fraction was approximately 55%. With respect to chronic symptoms, attributable to valvular problems, mitral valve replacement was planned. Furthermore, abnormally dilated vascular structures with turbulent flow on the left side of the neck were also visualized on TEE. Presence of bruit in the physical exam as well as echocardiographic findings raised strong suspicion of AVF; therefore, the patient underwent CT-angiography of the cervical vasculature. Consequently, an AVF was identified between the distal part of the left external carotid artery and the left external jugular vein. Significant dilatation of the left common carotid artery, 20 mm in diameter, and the left external carotid artery were seen, accompanied by dilatation and tortuosity of the left external jugular vein; however, the major branches of the left external carotid artery had normal size and appearance (). An incidental finding was made of hypoplasia of the mid and distal portions of the ipsilateral internal jugular vein. Thereafter, the patient was scheduled for ligation of the external carotid-external jugular AVF before mitral valve replacement.


A 63-year-old female was admitted to Kyung Hee University Medical Center after an incidental finding of a right colonic mass during her regular health check-up. She underwent surgical excision of a submandibular gland due to a Wharton's duct stone. She was asymptomatic. On physical examination, there were no signs of fever or acute abdomen and no palpable abdominal mass. Colonoscopy revealed a mucosal tumor in the cecum without mucosal abnormalities. On abdominal computed tomography (CT), there was a 3.8-cm exophytic mass with internal low attenuation between the cecum and the terminal ileum (). The radiographic impression was that this mass was a gastrointestinal stromal tumor (GIST) and likely was not an adenocarcinoma. On further exploration, a round, soft, exophytic mass was identified at the cecal wall, 2.0 cm from the appendix orifice. After surgical investigation of the mass, an ileocecectomy was performed.
Macroscopically, the surface of the mass revealed an irregular cystic lesion filled with a yellowish-gray, cheesy material (). No connection between the cyst and the cecal lumen was observed. The mucosal and serosal surfaces of the cecum were unremarkable. Histologically, the wall of the cyst was surrounded in its entirety by a cecal muscularis propria. Cells lining the cyst formed a mature, keratinized, and stratified squamous epithelium with a granular layer (). The focal area of cystic wall showed nonkeratinizing squamous epithelium without a granular layer (). The lumen of the cyst was filled with mature, dense keratin. Skin adnexa were completely absent. No other ectodermal, endodermal or mesenchymal elements were noted. The pathological diagnosis was an epidermoid cyst.


A 60-year-old Japanese woman presented with watery diarrhea. She denied nausea, vomiting, or abdominal pain. She noted a 4-week history of extremity weakness. Her past medical history involved schizophrenia, treated with paliperidone 6 mg and flunitrazepam 2 mg per day. She had no history of diabetes, malignancy, or steroid use. On admission, her physical findings included a masked face and slight rigidity. No electrolyte abnormalities were detected and magnetic resonance imaging showed no abnormal findings. Her cerebrospinal fluid revealed no inflammatory changes and albuminocytologic dissociation was not detected. Her family doctor had increased her dose of paliperidone 1 month prior. We suspected her weakness might be caused by drug-induced parkinsonism. On admission, her white blood cell count was 22,500/mm3 and C-reactive protein was 1.91mg/dL. Aspartate transaminase was slightly elevated (66 U/L; normal reference 9–32 U/L), compared with normal alanine transaminase (28 U/L; normal reference 3–38 U/L). Urine white blood cells and nitrites were positive. We suspected concomitant urinary tract infection and administered cefotaxime. Nine days after admission, her weakness improved gradually, but she developed abdominal pain and bloody diarrhea. She had no fever, nausea, or vomiting. On abdominal computed tomography, bowel wall thickening, and nonspecific inflammatory changes of surrounding fat tissue were seen from the sigmoid colon to rectum. Firstly, we suspected ischemic colitis and treated her with bowel rest. Four days after bowel rest, her symptoms did not improve. We performed colonoscopy, which revealed mucosal erosion and ulceration from the sigmoid colon to rectum (). Clostridium difficile was detected from culture of bowel lavage. We suspected pseudomembrane colitis and treated her with a 10-day course of metronidazole 250 mg four times daily. After treatment, her symptoms persisted and we performed follow-up colonoscopy, which showed only minimally improved ulceration (). Hematoxylin-eosin stained biopsy specimens of ulceration showed no inclusion bodies (), but immunostained specimens revealed CMV-infected cells (). Laboratory findings showed elevated CMV-immunoglobulin M (0.87; normal reference 0.00–0.79). We made a diagnosis of CMV colitis. HIV serology was negative and upper gastrointestinal endoscopic examination revealed that she had no malignancy, in addition to the result of the colonoscopy described above. She was considered as immunocompetent. By the time of diagnosis, her symptoms had substantially improved. Two months later, we performed follow-up colonoscopy, which showed complete resolution of ulceration (); biopsy demonstrated no CMV-infected cells.


A 2-year-old girl was brought to our outpatient clinic with a complaint of irritable crying after being taken to a stream in Hualien 1 day previous, where she played in the water. The parents noticed that she rubbed her right eye a lot. There was no history of trauma to the eye or any other chronic eye disorders.
Upon examination, the girl had good fix and follow in either eye. Slit-lamp examination showed conjunctival injection with a moving dark black–brown foreign body partly attached in the lower conjunctiva near the limbus, resembling uveal prolapse. The dark black–brown foreign body was partially movable, with tail hanging. It was then identified as a live leech grasping the bulbar conjunctiva (). After applying topical anesthetics, the leech was extracted under microscope with forceps, and it measured 1 cm in length (). The patient began using topical antibiotic and corticosteroid agents afterward.
By 1 week after extraction, the patient had no obvious symptoms or signs, except for a mild congestion and patchy subconjunctival hemorrhage. There was no evidence of scleral perforation, corneal injury, uveal prolapse, or uveitis.


A 42-year-old female from Dibrugarh, Assam visited us with history of minor injury to the right eye due to fall into a sewer when in a drunken state. She presented 6 days after the injury with chief complaints of swelling, itching, and blood-stained, foul-smelling discharge from the wound. Her husband also noticed small whitish-yellow worm-like structures crawling out from the wound (). She had no significant past history, and systemic examination was normal.
On local examination, best corrected visual acuity of the right eye was 6/18. There was generalized right periorbital edema, with matting of the eyelashes and severe conjunctival vascular engorgement. A lesion of approximately 3×2 cm was noted at the medial canthus. The ulcer had indurated margins with scanty blood-stained, foul-smelling discharge. Small freely moving maggots were visible in the wound. The cornea was hazy, and further detailed examination was not possible. Computed tomography (CT) scan of the orbit showed no signs of bone erosion or paranasal sinus involvement, except for the soft tissue swelling (). The other eye was also examined for maggots and was found to be normal.
Turpentine oil packing was done to immobilize the maggots. Mechanical debridement of the wound was done, and freely moving maggots were removed with the help of forceps, under local anesthesia using 4% xylocaine (). Regular dressing of the wound was done, with removal of maggots, for next 4 days. The patient was treated with intravenous amoxicillin sodium/potassium clavulanate 1.2 gm given 8 hourly and ibuprofen 400 mg twice daily for 3 days, followed by oral amoxicillin/clavulanic acid 625 mg twice daily for 5 days. A single dose of albendazole 400 mg tablet was also given. Local application of moxifloxacin 0.5% ointment to the right eye for 1 week was advised. The wound started to heal, with granulation tissue formation, and healing was completed by the end of 2 weeks ().
A total of 26 maggots were removed. The maggots were preserved in 10% formalin and sent for entomological evaluation to the Department of Microbiology. The larvae were creamy white in color, with cuticular spines, and varied in size due to different stages of presentation, from 5 to 15 mm. They had strong robust mouth hooks, with four to six papillae on the anterior spiracles, incomplete posterior spiracular peritreme, and pigmented dorsal tracheal trunks in the terminal twelfth larval segment. Based upon these findings they were confirmed to be larvae of C. bezziana (–).


A 67-year-old male patient presented with painless jaundice, dark-brown urine and acholic stools. The general practitioner had diagnosed onset of a diabetic condition with a HbA1c of 8.5% and a weight loss of 4 kg, corresponding to a weight loss of 5%. His antecedent history showed prostate cancer treated by prostatectomy 6 years before and a malignant melanoma on his back 5 years before. Both malignant conditions were followed periodically and did not show any signs of recurrence. Furthermore the patient had had borreliosis 30 years earlier and polymyalgia rheumatica for 2 years. The latter had been treated with low-dose steroids for one and a half years. During the first 6 months, 5 mg of prednisone was given daily, then prednisone was tapered to 2.5 mg twice a week.
A CT scan showed a double duct sign without clear delineated tumor, no calcification of the pancreas nor infiltration of the vessels (fig. ). A sonography was performed, which showed a mass of 15 × 23 mm in the pancreatic head. Blood tests showed normal values of the tumor markers CEA (2.4 μg/l) and CA19-9 (<3 kU/l) as well as total bilirubin of 50 µmol/l. Surgery was indicated since we suspected pancreatic cancer due to the clinical and radiological findings.
Infiltration of the mesocolic artery found during surgery made the planned Whipple procedure impossible. Biopsies were taken from the pancreatic head. A cholecystectomy and a biliodigestive anastomosis were performed. The histological results showed chronic inflammation with fibrosis and fat necrosis, but no signs of malignancy. After discussion of the case on a multidisciplinary tumor board, a secondary percutaneous biopsy was taken. This one showed chronic periductal inflammation with infiltration of plasma cells, sclerosis, venulitis, parenchymal atrophy (fig. , fig. ) and strong positive immunohistochemical staining for IgG4 (fig. ). This led to the diagnosis of type 1 autoimmune pancreatitis in the absence of granulocytic epithelial lesions.
We determined IgG4 in the serum, which was elevated (3.4 g/l, normal range 0.03–2.01 g/l), although the other autoimmune rates (rheumatoid factors, antinuclear antibodies, c-ANCA, p-ANCA) were normal. A follow-up CT scan 6 weeks later showed a 15% reduction in the craniocaudal dimension, resulting in a less prominent pancreas with inhomogeneous alteration of the structure. Nevertheless, neither calcification nor tumor mass was found (fig. ). As the patient was meanwhile asymptomatic, further steroid therapy was omitted.


A 78-year-old woman complained of erythroderma and was hospitalized. She had been suffering from psoriasis vulgaris since 47 years of age and used over-the-counter drugs for treatment. She had hypertension and chronic heart failure which were managed with acetazolamide, candesartan cilexetil and amlodipine besylate. Her brother had psoriasis vulgaris and her niece had psoriatic arthritis. Her daughter had arthritis with a high anti-nuclear antibody (ANA) titer (×1,240), but her diagnosis was not established (fig. ). A toxic eruption was suspected for the patient and 30 mg daily prednisolone was administered. However, it was ineffective, and the dose of prednisolone was decreased and instead cyclosporine 150 mg daily was given for 12 days. Cyclosporine was effective and the dose was decreased to 100 mg daily, but 2 weeks later, a sudden fever, generalized edema, erythroderma with multiple pustules and lakes of pus occurred (fig. ). A skin biopsy showed infiltrates of neutrophils into the upper spinous and subcorneal layers of the epidermis which formed a Kogoj spongiform pustule (fig. ). The diagnosis of GPP was made and etretinate 20 mg daily was administered. The pustules subsided but the fever and erythroderma with edema continued, so she was referred to us.
On examination, edematous generalized erythroderma with exudations and scales was seen, but pustules were absent (fig. , left). A blood test showed a white blood cell count of 7,380/μl (segment 78.5%, lymphocytes 15.3%, monocytes 6.0%, eosinophils 0.1%, basophils 0.1%), and a high C-reactive protein level of 13.5 mg/dl. The severity score for GPP [] was 12 (skin score 6 plus score for general symptoms and blood tests 6). She was treated with bed rest and a combination of cyclosporine and etretinate for 1 week, but the GPP score remained at 10. For supportive therapy, GMA was introduced once weekly. After the initial session of GMA, the white blood cell and C-reactive protein levels began to decrease and edema, skin redness and scales became less severe. Before GMA, the neutrophil cell count was 4,500 cells/µl, but after three sessions of GMA, neutrophils had decreased to 1,608 cells/µl. At 6 days after the last GMA, the severity score for GPP had improved to 5 (fig. , center). Further sessions of GMA were held, and neutrophils recovered to normal levels by 19 days, at which time the GPP severity score was 4 (fig. , right).
During hospitalization, the patient complained of a dry mouth feeling and had a positive ANA titer (×320) and a high titer of anti-SSA and anti-SSB auto-antibodies. Sjögren syndrome was diagnosed and the symptoms were controlled by cevimeline hydrochloride. After treatment with GMA, the dose of cyclosporine was decreased and the drug was eventually discontinued, with no exacerbation of GPP. She was followed up with etretinate 10 mg daily and topical vitamin D3 and steroids for the remaining lesions of psoriasis vulgaris. Relapse of GPP has been absent for 2 years to date.
The IL36RN gene was examined under approval of the Institutional Review Boards and informed consent from the patient and her daughter. PCR products from their genomic DNAs were subjected to Sanger sequencing. Genetic analysis revealed homozygous mutations c.[28C>T];[28C>T] which led to p.[Arg10Ter];[Arg10Ter] in the patient (fig. ). Her daughter had a heterozygous mutation c.[28C>T];[=] in IL36RN (fig. ). The HLA types of the patient were HLA-A*0201, HLA-B*0201, HLA-DRB1*0803 and HLA-DRB1*1405.
The serum cytokine levels of IL-1β, IL-6, IL-8 and TNF-α were examined at the patient's first visit, 1 day before the first session of GMA, 2 days after the second session of GMA and 19 days after the last session of GMA (fig. ). Those cytokine levels had already decreased before the first session of GMA. The serum levels of IL-6 and IL-8 further decreased along with the improvement of the GPP score by GMA, but the levels of IL-1β and TNF-α did not.


In February 2013, a 39-year-old woman presented with limited mouth opening suspected by her dentist to be caused by TMD. She could not open her mouth widely since her elementary school days, and restricted mouth opening gradually developed. She always ate sushi after tearing it into small pieces. Her facial configuration was characterized by a square mandible (). Oral examination revealed no pain on motion, tenderness, clicking or injury of the temporomandibular joint. The lower jaw moved smoothly, but unaided maximal mouth opening was only 27 mm (). There was thus no evidence of TMDs. Magnetic resonance imaging (MRI) showed bilateral enlargement of the masseter muscles. In addition, a ‘thick’ aponeurosis of the anterior aspect of the masseter muscle was noted bilaterally (Fig. ). On maximal mouth opening, intraoral palpation along the anterior border of the masseter muscle confirmed a hard cord-like structure, consistent with the findings on MRI. MMTAH, recently established disease category [–], was thus diagnosed.
After obtaining her consent, surgical treatment was performed (Figs and ; ). Resection of the ‘thick’ masseter muscle aponeurosis and coronoidectomy, performed under general anesthesia, increased maximal mouth opening from 29 to 53 mm. Although the coronoid process did not interfere with the zygomatic bone on mouth opening, the coronoidectomy was needed to completely resect the temporal muscle tendon from the posterior of the coronoid process. Five days after surgery, postoperative mouth-opening training began. The current mouth opening is 49 mm ().


A 61-year-old Indian woman was referred to our cancer centre with adenocarcinoma of right lung with stage IV due to bone metastasis. Patient initially received four cycles of palliative carboplatin and Pemetrexed combination chemotherapy. After four cycles, patient discontinued platinum-based doublet chemotherapy for generic Erlotinib due to poor chemotherapy tolerance, financial constraints, and EGFR mutation positivity for deletion E746-A750 of codon 19. Approximately after 18 weeks of erlotinib therapy, she developed significant lengthening, rigid and curly overgrowth of her eyelashes which is called trichomegaly of eyelashes. (Figure ) Patient continued on Erlotinib therapy for 13 months in view of good clinical response of tumour despite trichomegaly of eyelashes requiring frequent trimming.


A 63-year-old male patient was submitted to CBCT (i-CAT, Imaging Sciences International, Hatfield, PA, USA) of the mandible for the planning of rehabilitating treatment with dental implants. Intraoral and extraoral examinations showed no significant findings, and the patient was asymptomatic.
Sequential axial sections in the craniocaudal direction (), sequential parasagittal sections obtained every 1 mm in the distomesial direction (), and coronal sections in the posteroanterior direction () revealed the presence of two mental foramina on the right side of the mandible, while only one foramen could be identified on the left side. At the level of the second premolar, before its opening in the vestibular portion of the mandible, the mandibular canal on the right side showed a ramification with a distinct and independent intraosseous course in the posterior direction and an additional opening in the vestibular cortical bone of the mandible. The mental foramen and accessory mental foramen, measuring 2.83 and 1.75 mm in diameter, respectively, were present at the same height, with the accessory mental foramen in a slightly posterior position. The mental foramen on the left side was a single structure located at the same height as the others, but with a diameter of 4.84 mm.
Three-dimensional reconstruction demonstrated the presence of two mental foramina on the right side and only one, of wider diameter, on the left side (). In the panoramic radiograph, we could not observe the presence of the accessory mental foramen ().


There were 2 cases involved in the same MVC (driver and passenger).
A 41- year old female, front-seat passenger restrain by seatbelt (no air bag), presented to our trauma center with grade IV shock, heart rate of 120 bpm, blood pressure of 80/45 mmHg, respiratory rate of 28 per min, oxygen saturation of 89%, and GCS of 14. Though, airway was clear, there was decreased air entry on left lateral side. Seat belt sign was noted over left chest and lower abdomen with degloving injury of lower abdomen and left lateral lumbar area. She also had a 15 cm laceration on left forehead with active bleeding, grade IV open fracture of the right distal radius with obvious transected right radial artery.
The patient was resuscitated with oxygen mask, crystalloid infusion and two units of ‘O’ negative (uncrossed match) PRBC in the trauma room and massive blood transfusion was activated. Focus Assessment with sonography of trauma (FAST) was positive, chest X-ray demonstrated herniation of abdominal content into the left pleural cavity [] and pelvic X-ray was normal.
Patient was immediately transferred for exploratory laparotomy and found to have grade V left diaphragmatic rupture with significant left hemothorax, transected 10 cm of jejunum with its mesentery [], and multiple bleeding mesenteric lacerations in mid small bowel and distal ileum. There was also a small paracecal hematoma, lacerated serosa of sigmoid colon with laceration of its mesentery, and small non-expanding retroperitoneal hematoma in left zone-2. Patient underwent diaphragmatic repair, irrigation of left hemothorax, left thoracostomy tube insertion and primary anastomosis of jejunum. A large soft tissue hematoma of (degloving/shearing injury) lower abdomen and left lateral lumbar areas were evacuated and drained with three large J-vac closed suction []. Right radial artery was ligated after positive triphasic Doppler signal of right ulnar artery, followed by external fixation of right radius, wound debridement and closure.
Patient was hemodynamically stable and was underwent head, chest and abdominal CT scan post-operatively. The CT findings revealed significant left lung contusion, fracture of left 10-11 ribs, fracture of 7th rib, non-perfused left kidney surrounded by small hematoma with thrombosed distal left renal artery, and unstable chance fracture of L2 with no cord compromise.
The patient maintained her vital signs after 12 hours with normalization of the acid-base balance. On tertiary trauma survey, patient was unable to move both lower limbs. Emergent MRI revealed congenital tethered spinal cord to the level of lower L2 with cord contusion extended from upper L1 to lower L2. X-ray of the extremities showed fractures of right lateral malleolus, 2nd and 3rd right metatarsal and query right talus, and fractured right foot 1st and 2nd phalanx, and fracture of right 5th MCB. She was extubated after 11 days; pedicle screw fixation was performed after 17 days with back brace applied for continuous physiotherapy, and she was able to walk normally after 2 months.
The second patient involved in the same MVC with the first patient was a male of 40 years old. This patient was using seatbelt and the airbag was working during the accident. He arrived with a stable hemodynamic status, GCS of 15, and decreased air entry to left lateral field of lung. He had seatbelt marks over his left chest and lower abdomen with moderate degloving injury of left lateral abdominal wall. Secondary survey revealed blood at the external urethral meatus, and had incidental right inguinal hernia. FAST was negative, fracture left mid femur, right radius, fibula and talus. Chest X-ray showed herniation of bowel to left hemi-thorax. CT scan demonstrated grade V left diaphragmatic injury with herniation of abdominal content into left hemothorax [], bilateral lung contusions, suspicious of extravasated contrast from prostatic urethra. Patient was shifted to the operating room for repair of diaphragm, insertion of left thoracostomy tube and repair of mesenteric tear of ileocecal junction, rectosigmoid junction, and serosal tear of rectum []. He was extubated after 10 days; open reduction internal fixation for left femur was done after 12 days. The patient was doing well and transferred to trauma floor after 14 days.


A 60-year-old female patient with a history of nephroureterectomy and cuff excision due to renal pelvic TCC was admitted to our outpatient clinic with complaints of red skin lesion in the near vicinity of the operational incision scar for 3 months. Her medical history revealed nothing but nephroureterectomy operation on her upper urinary system; moreover, it was learned that she had been ignoring what was recommended to her for routine controls. A thorough search through the database regarding patient's past medical reports revealed that the pathology report about the past operation had been compatible with high-grade TCC and that the tumor had been multifocally located in the renal pelvis with the largest one to be 2.5 cm in diameter. The past pathology report had further indicated presence of lymphovascular invasion, compatible with pT2 stage (Muscle invasion is present in the absence of parenchymal and peripelvic fat tissue invasion) and absence of tumoral tissue within the surgical border. In the physical examination, a hard, painless and immobile mass of 2*2 cm dimension was palpated 3 cm below the incision line. Thoraco-abdominal computed tomographic (CT) examination performed on the basis of the aforementioned findings depicted a mass lesion of 78*65*50 mm dimension with heterogeneous contrast uptake, which originated in the abdomen at the level of left renal vein drainage into the inferior vena cava, completely surrounding the inferior vena cava and extending inferiorly to the level of aortic bifurcation. In addition, a mass lesion of 24*20 mm dimension with high-contrast uptake was detected within the subcutaneous fat tissue in the right abdominal wall. The other abdominal and thoracic structures were evaluated to be normal [].
The skin lesion depicted in CT was surgically excised. The pathological examination of the excised material was reported to be compatible with TCC metastasis localized within the reticular dermis and hypodermis without any invasion into the epidermis and papillary dermis, and with surgical border negativity.
The patient was referred due to abdominal lesion to medical oncology after the operation. Followed up under chemotherapy protocol, the patient died 3 months after the metastasectomy operation.


The patient, a 41-year-old woman with a non-relevant past medical history, concluded her fourth pregnancy by cesarean delivery owing to macrosomic product (July 2013). Tubal ligation was performed on postoperative day one by the Kroener fimbriectomy method, which coursed uneventfully and without any remarkable findings. The specimens obtained were sent for routine histopathological assessment, and the patient was discharged after full recovery. To date she is healthy and without any complaints.
Both distal tubes and fimbriae were identified; the large tube was bulky with a nodular appearance (3.5×2×1.3 cm) (). Transverse sections taken from this dilated duct revealed an overfilled lumen with a caramel-like substance surrounding an ill-defined peripheral spot, which, when seen under a magnifying glass, showed a fractal pattern similar to a snowflake (). Histologically, this hazy frame was sketched by a slender stalk with progressively branching papillae lined by columnar non-ciliated cells with plump eosinophilic cytoplasm showing focal pseudostratification and budding (, ). The nuclei of these cells were centrally located, enlarged, and rounded or oval. The nuclei displayed a variable appearance, either dense and hyperchromatic, or vesicular with a prominent nucleolus, and sometimes having coarse chromatin, membrane foldings, pseudoinclusions or grooves. Mitotic activity was not identified.
Despite the presence of luminal extracellular mucin, several subnuclear mucin-filled vacuoles were also identified, occasionally expanding into cyst-like structures (). Periodic acid– Schiff and Alcian blue stains highlighted the extracellular and intracellular mucin, while the intracytoplasmic vacuoles were stained with Mayer’s mucicarmine (–).
Immunoperoxidase stains demonstrated an intensely positive reaction for cytokeratin cocktail (CK AE1/AE3) in the membrane and cytoplasm () of cells, as well as for epithelial membrane antigen (EMA) with weak cytoplasmic expression and luminal border reinforcement (). Detection of progesterone receptors (PR) was markedly positive in 95% of cells, while estrogen receptors (ER) and androgen receptors were moderately and weakly positive in 70% and 10% of cells, respectively (–). The proliferation index labeled with Ki-67 was rated in approximately 3% of cells (). Surprisingly, cyclin D1 staining was observed in nearly 90% of cells (). No reaction was detected toward a variety of factors, including carcinoembryonic antigen (CEA), human chorionic gonadotropin, human epidermal growth factor receptor 2 (HER2/Neu), or B-cell lymphoma 2 protein (Bcl-2).
Accordingly, the proliferation was diagnosed as a metaplastic papillary tumor of the Fallopian tube, with a note to the clinician about the benign behavior of the alteration.


A 62-year-old Italian man developed slowness and loss of strength of his right arm and hand. In July 2010, after a first neurological examination, he was prescribed l-DOPA, 250 mg/day, resulting in significant improvement of his parkinsonism. PD was therefore indicated using an ex juvantibus reasoning. Subsequent magnetic resonance imaging carried out in October 2010 excluded other conditions. At this point, the diagnosis of PD was confirmed and the neurologist who followed the patient at that time made the decision to change treatment to 1.05 mg/day of pramipexole and the complete cessation of l-DOPA. He was checked twice over a period of 12 months and his condition was considered stable each time.
The patient had a history of cervical spondylosis, a very common condition in the aging population caused by the progressive degeneration of the neck bones and neck tissues. He also had a straightforward inguinal hernia for which he undertook a corrective operation on 21 November 2011. The operation started at 9:00 am and ended at ∼9:30 am. He did not take pramipexole on the day of the operation. Given the option of general anesthesia or epidural he chose the former, carried out as follows:
– Stage 1: Analgesic administration: [ultiva (remifentanil); infusion pumps were used] – Stage 2: Anesthesia induction: (propofol and curare immediately after) – Stage 3: Intubation: [halogenated anesthetic (sevorane)]; – Stage 4: Before wake up: prostigmina was used to eliminate curare and sevorane was stopped.
There were no postoperative complications and the patient was discharged on the same day. He restarted taking pramipexole (1.05 mg) on the morning after the operation. On the same day, he developed neck rigidity and his head dropped forward. The local general practitioner treated him with painkillers, but these proved ineffective.
From the immediate onset of antecollis the patient's life was severely impaired: he needed assistance at work, could not drive his car and experienced difficulties when eating and speaking. On 3 April 2012, the patient decided to consult a professional physiotherapist who objectively confirmed severe antecollis: his neck was bent forward at an angle of 80–90°. He could actively extend his head up only of about 50–40° with obvious compensation of the posterior and inferior (lumbar) muscles of the vertebral trunk. Therefore, this was not a real extension of the head and could not be sustained beyond ∼30 s. The neck rotation on both left and right sides of the head was severely restricted. He undertook 10 physiotherapy sessions with the aim of reducing the strong contraction of his neck muscles. However, no improvement was observed on treatment completion.
In June 2012, 6 months from the onset of antecollis, following similar cases reported in the literature [, ], it was decided to withdraw pramipexole and reintroduce l-DOPA. The improvement was almost immediate: on the first day, the patient felt less pressure on the neck and gained more flexibility. After a few days, he could easily turn his neck and he started to drive his car again.
On 13 August 2012, about 2 months after the cessation of pramipexole, the patient decided to undertake a second cycle of physiotherapy. His condition objectively appeared significantly improved, despite the extended period of time in which his neck movements were severely impaired. The only change in his treatment regime from June to August 2012 was the cessation of pramipexole.
Since this second cycle of physiotherapy, neck angle remained about 30° at rest. The patient can now perform extension and rotation movement easily, as shown in Fig. .
Unlike the cited cases in which antecollis appeared within a few weeks after taking pramipexole, this patient developed antecollis several months after its introduction for the treatment of PD. This relatively extended period might indicate that some factor(s) other than pramipexole may have induced antecollis, but the improvement upon cessation suggests otherwise. Intriguingly, antecollis emerged immediately after the inguinal hernia operation, raising the possibility that onset was triggered by the surgical procedure—however, this interpretation is highly speculative.


A 40-year-old male was evaluated in the Allergy clinic as a self-referral for systemic mastocytosis. The patient reported a 13-year history of generalized hyperpigmented cutaneous lesions with a wheal-and-flare reaction after scratching, and with exposures to warm and hot baths, and to exercise. His face was spared from cutaneous lesions. He had associated lightheadedness after exposure to hot baths and exercise without need for emergency room visits. He denied other symptoms such as angioedema, hives, shortness of breath/wheezing or anaphylaxis. He also denied previous adverse reactions to hymenoptera stings. Previous workup included an elevated serum tryptase level of 49 (range 2–10 ng/ml) and a bone marrow biopsy with 30% of aggregates of spindle CD117-positive/CD25-positive MCs, consistent with a diagnosis of systemic mastocytosis. Skin biopsy report showed an increase number of MCs around dilated blood vessels within the superficial dermis. Leder stain confirmed an increased quantity of MCs (Fig. ). The patient had been started on loratadine 10 mg every other day and diphenhydramine 25 mg as needed for acute exacerbations with no further workup follow-up.
During his initial evaluation at our clinic, he was found to have generalized hyperpigmented lesions (Fig. a) that flared upon scratching (Fig. c) with a tongue depressor (Fig. b) and hepatomegaly. We requested bone marrow biopsy slides for further review by our Pathology Department, and our pathology review was that immunohistochemical stains demonstrated lymphoid aggregates to contain numerous CD3-positive cells admixed with slightly fewer CD20-positive cells (Fig. a). The atypical spindled MCs were positive for CD117 (Fig. b). A bone scan showed no lytic osseous lesions. A c-kit gene analysis reported no known pathogenic mutations in exons 8, 9, 11, 13 or 17 of the kit gene. CT of the chest and abdomen showed no liver, spleen or lymph node enlargement. The patient received a diagnosis of indolent systemic mastocytosis. He was instructed to decrease sport activities, such as participating in marathons, since this type of exercise could exacerbate his condition. Medical therapy was recommended and this included self-injectable epinephrine prescription, fexofenadine 180 mg twice a day and ranitidine 150 mg twice a day. After 4 months, when the patient was seen again in follow-up, the patient was asymptomatic on medical therapy.


A 24-year-old female presented with history of backache, nausea, vomiting and abdominal pain of 2 weeks duration. Clinical examination was noteworthy for pallor and tachycardia. Laboratory evaluation was remarkable for anaemia (7.2 g/dl), leucocytosis (97 000/μl) with 70% plasma cells (Fig. ), elevated serum creatinine (9.9 mg/dl), total protein, and proteinuria (2.72 g/day). IgG kappa monoclonal paraprotein was identified and kappa light chain was elevated (Table ). Bone marrow showed diffuse infiltration with plasma cells (CD 138+, kappa restriction). Cytogenetic analysis showed a complex karyotype. Skeletal survey revealed two tiny lytic lesions in the skull. Based on the above presentation, a diagnosis of pPCL was made.
The patient received therapy with bortezomib, thalidomide and dexamethasone (VTD). She achieved complete remission (CR) after six cycles. She then underwent autologous haematopoietic stem cell transplant (HSCT) with high-dose melphalan (200 mg/m2) as the conditioning regimen. Six months after the autologous transplant (Table ) atypical plasma cells were noted in the peripheral blood (CD138+, CD56+, CD38+, kappa restriction).
Based on one case report [] the patient was treated with hyper-CVAD. After two cycles of chemotherapy, she achieved VGPR. As there was no matched sibling/unrelated donor, she underwent an allogeneic HSCT from a haplo-identical donor []. She received myeloablative conditioning with Fludarabine-Busulfan and post-transplant Cyclophosphamide along with Tacrolimus and Mycophenolate as GVHD prophylaxis. She engrafted on D +14 and was discharged on D+21.
Five months after allogeneic HSCT, she developed a nodular subcutaneous lesion in the left lumbar region. Fine needle aspiration from this lesion showed many atypical plasmacytoid cells (strongly positive for CD 138). Further testing revealed systemic relapse (Table ). She was treated with palliative radiotherapy to the affected area along with donor lymphocyte infusion. She could not receive any further therapy as she developed severe graft vs. host disease (grade III skin + gut). Despite the above measures, she died of progressive disease 24 months after diagnosis.


A previously healthy 5-year-old boy was admitted to our hospital with a short history of a rash and abdominal pain. His medical history was unremarkable, and no drug intake was reported. His parents reported a palpable non-blanching purpuric rash over his lower extremities that had been present for 2 days. The rash extended to the upper extremities, and the patient complained of abdominal pain on the day before admission. Laboratory data obtained on admission showed a hemoglobin level of 12.5 g/dL, a white blood cell count of 11,020/mm3 with a normal differential count, and slightly elevated IgA (211 mg/dL). Other laboratory data were within normal limits. A urinalysis showed no proteinuria or hematuria. The patient was treated with intravenous fluids as conservative treatment under the impression of HSP. After admission, he complained of pain in the right knee and elbow joint with swelling and was started on 1 mg/kg/day of oral prednisolone.
The joint and abdominal pain improved by hospital day 17. However, both sides of the scrotum became edematous and tender with a purpuric rash. The penis also showed swelling and a purpuric rash with a focal reddish mass-like lesion at the penile tip. The reddish mass-like lesion caused skin twisting at the base with mild deviation of the penile axis (). The patient did not complain of pain in the penile shaft or in the reddish mass-like lesion at the penile tip. No history of trauma was reported, and no evidence of a urinary tract infection or hematuria was detected. The phallus was uncircumcised, and the patient voided without difficulty or pain and had a normal urinary stream. The patient was referred for emergency scrotal ultrasound examination. On ultrasonography, bilateral scrotal soft tissue thickening was noted. It was more severe on the right side (), and the right epididymis was swollen with increased vascularity, mimicking bacterial epididymitis (-). The size, echogenicity, and vascularity of both testicles were within normal limits. The penile shaft and foreskin were also swollen (), and there was a well-defined hypoechoic mass-like lesion (dimensions, ~1.4 cm×1.3 cm) without vascularity on the penile tip (). This lesion was not connected with the corpus cavernosum, corpus spongiosum, or glans, and seemed to originate from the tip of the foreskin. Although the lesion did not show vascularity in a Doppler study, the patient did not complain of any pain in the penile area or voiding difficulty. Follow-up ultrasonography was recommended under the impression of HSP-related large ecchymosis or purpura with a blister on the penile tip. The edema, purpuric rash, and pain in the scrotum completely subsided as did the reddish mass-like lesion on the penile tip after 2 days of continuous treatment with oral prednisolone. The penile axis also became normal. Followup ultrasonography showed no scrotal or penile edema and no epididymal swelling (), and the hypoechoic mass-like lesion on the penile tip had disappeared ().


A 40-year-old female patient presented with multiple raised growths over both feet since four years. The growths started over soles and progressed to involve the dorsum of both feet. It was not associated with pain or itching. There was no history of trauma or application of any irritant prior to onset. There was no history of diabetes, smoking or alcohol addiction. Contractures, bony deformities and keloids were absent. On examination, symmetrical, well-defined, firm nodules coalescing to form lobulated plaques were noted over the dorsum of both feet extending from base of the 2nd, 3rd, 4th, and 5th toes to the mid-foot [Figures and ]. The skin over swelling was not pinchable. Nails were normal. Clinically, differential diagnoses of callosity, fibromatosis, and keloids were considered. Routine blood and biochemical investigations were within normal limits. HIV and HBsAg were negative. Radiographs of both feet showed no bony deformity and ultrasound of both feet showed no significant soft tissue thickening. An elliptical biopsy of lesion taken for histopathology showed epidermis with hyperkeratosis, acanthosis, mild parakeratosis and prominent granular layer. Superficial dermis was unremarkable and deep dermis showed fibroblastic proliferation with variable vascularity and lack of inflammatory infiltrate, and thick bundles of irregular collagen characteristic of fibromatosis [Figures and ]. With these clinical and histopathological features, a diagnosis of fibromatosis was made. The patient was advised topical keratolytics along with intralesional steroids for 2 months with minimal response. The patient was counseled about the benign nature of the disease and measures for lifestyle modification and proper footwear.


A 32-year-old man presented to our department with a 6-year history of multiple nodules on the scalp. The patient had experienced intermittent pain and an itching sensation on the lesions. On physical examination, 6 protruding, subcutaneous, firm nodules of varying size were observed on the scalp (). The nodules were several centimeters in size. An earlier ultrasonographic examination at a different hospital had indicated that the lesions could be suspected lipomas. For a further diagnosis, an excisional biopsy was performed for a single lesion. Histopathological examination revealed a poorly circumscribed, paucicellular lesion composed of thick, haphazardly arranged collagen fibers with sparsely scattered fibroblast-like cells in the subcutis (). Adipose tissue islands and peripheral nerve fibers were entrapped by the collagen fibers (). These findings were consistent with those of NTF. The other lesions were also excised, and they displayed the same histopathological features.


This is a case report of a 37-year-old female who was referred to our renal outpatient clinic with the incidental finding of proteinuria determined by dipstick. She had been diagnosed with MS 9 years previously. She had been on long-term IFN-β 44 µg three times weekly for most of this time. She had a normal pregnancy 10 years previously (a year prior to her MS diagnosis). She was not on any other medication. She denied haematuria, rash, arthralgia, weight loss, nose bleeds and haemoptysis. At the time of presentation in clinic, she had a urine albumin creatinine ratio (UACR) of 268 mmol/L, equivalent to a protein leak of about 2.5 g/24 h. Her serum creatinine (Cr) was 60 µmol/L, albumin 28 g/L, haemoglobin 11.5 g/dL, adjusted calcium 2.3 mmol/L, C-reactive protein (CRP) of 2 mg/L and normal complement levels (C3 0.98 g/L and C4 0.19 g/L) (). On examination, her blood pressure was 127/80 mmHg. She had normal heart sounds. Her lungs were clear and her abdomen was soft and non-tender. She had no pedal oedema. In view of the proteinuria, an angiotensin-converting enzyme (ACE) inhibitor was introduced at this point (). Subsequently, urine culture, anti-glomerular basement membrane antibodies, myeloma screen, hepatitis B and C serology and human immunodeficiency virus were all negative. The ultrasonography showed unequal size kidneys, the right kidney measuring 10 cm and the left 12.5 cm in length.
The renal biopsy showed 11 glomeruli with a general increase in the cellularity predominantly in the mesangium (A). There was no tuft necrosis or endocapillary proliferation. The glomerular capillary basement membrane showed foci of basement membrane irregularity and few foci of ‘double contours’ (B). The tubules and interstitium appeared essentially normal. The electron micrographs confirmed focal mesangial cell interposition (D), with new basement membrane material deposited on the internal surface. This was associated with a few small and rather ‘woolly’ accumulations of slightly electron-dense material, but these did not have the typical morphology of immune complex-type electron deposit (C). Endothelial cells appeared somewhat swollen, with infrequent areas of fenestration. The epithelial cell foot processes appeared remarkably normal. There were mild mesangial cell interposition and basement membrane irregularities, with no more than a hint of glomerular hyper cellularity. The immunofluorescence showed a partly particulate deposition of predominantly C3 within the mesangium with a lesser deposition of IgG, M and A. The renal biopsy report concluded that in the absence of immune-complex deposition (or linear dense deposits), these findings justified a search for other possible causes of low-grade endothelial damage, such as the use of interferon therapy.
Unfortunately, following the renal biopsy, she bled and required embolization.
In view of the biopsy report, IFN-β was switched to Glatiramer acetate (Copaxone®) 20 mg daily with a resultant progressive improvement of the proteinuria (). The proteinuria remains negative at 5 months even after stopping the ACE inhibitor therapy.


A 65-year-old male patient diagnosed to have right lower lobe lung adenocarcinoma was referred for FDG PET/CT for staging evaluation. FDG PET-CT showed intense tracer uptake in the right lower lobe lung lesion along with multiple mediastinal and right supraclavicular lymph nodes []. Furthermore, increased tracer uptake noted in the entire left hemipelvis with a maximum standardized uptake value (SUVmax) of 5.3. CT showed cortical thickening, intra trabeculation with course thick trabeculae, bone expansion, and multiple areas of sclerosis, consistent with Paget's disease []. The level of alkaline phosphatase (ALP) was 456 IU/l. He underwent follow-up FDG PET-CT after three cycles of chemotherapy showed intense FDG uptake with decrease in size and metabolism of the lung primary and mediastinal nodes with complete response in supraclavicular and some mediastinal nodes. But left hemipelvis uptake remains same, and he was asymptomatic for Paget's disease [].


A 55-year-old, 167 cm, 70 kg male suffering from alcoholic end-stage liver cirrhosis with a 'model for end stage liver disease' score of 15 points, and Child-Pugh class C, was scheduled for deceased-donor liver transplantation. He had a history of hepatic encephalopathy, refractory ascites, and undergoing a repetitive paracentesis over the previous 6 months. When he first arrived at our hospital 3 months earlier, he had suffered from hepatic hydrothorax, which waxed and waned. Pleural fluid analysis revealed transudative fluid. The preoperative transthoracic echocardiography revealed elevated right ventricular systolic pressure (46 mmHg) without coexisting cardiopulmonary disease. The preoperative chest computed tomography showed massive pleural effusion and related passive atelectasis in the right middle and lower lobe (). On the day of surgery, he was in mild respiratory distress and a plain chest radiograph revealed massive pleural effusion of the right lung and associated lung collapse (). An 8.5-French percutaneous pigtail catheter was placed in the right pleural space 3 h before surgery and 600 ml of transudative pleural fluid were drained naturally.
Anesthesia was induced with fentanyl (100 µg), midazolam (3 mg), thiopental sodium (250 mg), and rocuronium (40 mg). Anesthesia was maintained with a 50% oxygen/air mixture and sevoflurane. Neuromuscular block was maintained with cisatracurium. Hemodynamic monitoring included the right radial and femoral arterial, central venous, pulmonary arterial, and pulmonary capillary wedge pressures, and cardiac output measurements (Vigilance monitoring, Edwards Lifesciences, Irvine, CA, USA) using pulmonary artery catheter (CCOmbo V; 7.5 French, Edwards Lifesciences).
The prehepatic and anhepatic phase was uneventful and during that period, 500 ml of transudate were drained through the pigtail catheter. After reperfusion of a graft, he required a bolus of epinephrine (10 µg), dobutamine infusion (5-10 µg/kg/min) and norepinephrine infusion (0.05-0.1 µg/kg/min) to maintain mean arterial pressure > 65 mmHg. At this time, his ventilation was well controlled and the results of arterial blood gas analysis were unremarkable, as shown in . However, 30 min after reperfusion, we found a straw-colored fluid emanating from the ET tube. The trachea was intermittently suctioned and total volume suctioned via ET tube was over 500 ml in 30 min. The characteristics of the regurgitated fluid were similar to those of fluid drained through the pigtail catheter, which decreased visibly at that time. Hypoxemia did not occur with a FIO2 of 0.5, as indicated by the stable SpO2 during the entire intraoperative period. Thus, we suspected the presence of a connection between the airway and pleural space on the affected side and that the pleural fluid on the right side was regurgitated towards the ET tube. We changed a standard ET tube to a 37-French double-lumen tube (DLT; Covidien, Mansfield, MA, USA) to provide differential lung ventilation. A regurgitated fluid was obtained continuously from the right bronchus, but not from the left bronchus. After a discussion with the operating surgeons and a thoracic surgeon, we decided to open the diaphragm to check for the presence of APF. Air leakage via a 1.0 cm defect on the right lung surface during positive-pressure ventilation was detected through a 1.5 cm incision of the right side of the diaphragm ( and ). The APF and incised diaphragm was repaired with a suture with a minimal residual APF. The remaining surgical procedure was otherwise uneventful. At the end of surgery, the chest tube was inserted in the right pleural cavity. During the 550-min anesthesia, 7,400 ml of crystalloid, 200 ml of 20% albumin, 12 units of leukocyte depleted red blood cell, and 10 units of fresh frozen plasma were infused. Urine output was 880 ml and the estimated blood loss was about 10,000 ml.
Following the surgery, the patient was transferred to the intensive care unit with the placement of a DLT. An immediate postoperative chest radiograph suggested a decreased pleural effusion and mild bilateral pulmonary edema (). As the amount of regurgitated fluid decreased, the DLT was replaced with a standard ET tube on the day after the operation. On the second postoperative day, pneumothorax developed on the right side (), which resolved 2 days later. The ET tube was removed on the fourth postoperative day. In total, 500-1,000 ml of drainage were obtained daily via the chest tube during 2 weeks with ongoing ascites. Drainage decreased slowly thereafter, and the chest tube was removed on postoperative day 21 ().


The patient was a 34-year-old gentleman, a known case of chronic hepatitis B with multifocal HCC who underwent right lobe hepatectomy with cholecystectomy 4 years ago. He had recurrent HCC at segment III and caudate lobe, thus he underwent three TACE procedures followed by wide caudate lobe resection couple years before. In this episode he presented with jaundice, high-grade fever and severe abdominal pain for 3 days. His blood works showed leukocytosis with neutrophilia. Liver chemistry showed hyperbilirubinemia which total/direct bilirubin 12.9/11.2 mg/dl (reference 0.2–1.0 mg/dl), aspartate transaminase and alanine transaminase were 75 and 53 U/l (reference 0–38 U/l), and alkaline phosphatase was 209 U/l (reference 39–117 U/l). Computed tomography showed a 6-mm common bile duct with mild left intrahepatic duct dilatation. Proximal left intrahepatic duct stricture was suspected; therefore, he was scheduled for endoscopic retrograde cholangiopancreatography and biliary drainage.
The patient was positioned in the left lateral decubitus position and sedated using intravenous propofol under full anesthetic monitoring. After duodenal intubation with a duodenoscope, a large duodenal ulcer was seen in the duodenal bulb, so the scope was changed to be standard gastroscope. The endoscopic view showed a 1.5–2.0-cm duodenal ulcer covered by yellowish debris tissue. After carefully checking the ulcer, two openings were seen at the ulcer base (fig. ) with whitish epithelial lining of the larger one, therefore a 0.035-inch guidewire was passed through these two nonsurgical openings (fig. ) followed by contrast injection. The authors found that one of the openings was a choledochoduodenal fistula and the other one a hepaticoduodenal fistula (fig. ). After revision of his surgical procedures, no surgical bypass was performed at all in this particular patient. The authors hypothesized that these fistulas were spontaneously created and might be the consequence of TACE procedures which had caused ischemia and necrosis of the liver. The obstructive jaundice was due to the obstruction of the openings by food particles and a strictured bile duct at segments II and III, so two plastic stents (10 Fr, 7 cm) were inserted with good bile flow (fig. ). The patient was followed up a month later. He was doing well and his liver chemistries were normal.


The patient is a 58-year-old male with a history of hypertension and type II diabetes mellitus who presented to an outside hospital after developing chin and right arm tremors, followed by a seizure. The seizure was complicated by a fall and urinary incontinence; the seizure resolved on its own. A CT completed at an outside hospital revealed a large, complex cystic mass in the frontal lobe and left hemispheric subarachnoid hemorrhage. The patient was administered levetiracetam and transferred to a specialty hospital for further evaluation. MRI analysis revealed a 5.6 × 8 × 5 cm complex cystic lesion in the left frontoparietal lobe, with a mass effect on the left lateral ventricle (fig. ). This lesion included several nodular T2 bright and T1 isointense solid components. There was some enhancement in the cystic components that was also visualized on T1 imaging with gadolinium. Surgery was performed, and a left frontoparietal brain lesion, which was initially suspected to be a low-grade glioma, was removed. However, the tumor was surprising in its grayness and more firm than it would be expected for a low-grade glioma. The surgery was repeated 3 days later for microdissection and tissue harvesting. During his hospital stay, the patient developed waxing and waning expressive aphasia, with the development of global aphasia, a right facial droop, and right-sided weakness. A CT demonstrated hemorrhage in the resection cavity, effacement of the left superior frontoparietal sulci suggestive of cerebral edema, and a slight increase in left to right midline shift from 1.3 to 1.5 cm. This required surgery on postoperative day 6 for removal. However, it was unclear if the aphasia stemmed from the clot or cerebral edema following the surgery. On the day following evacuation of the hematoma, the patient's symptoms improved. The patient's symptoms included residual slurred speech, which improved over the subsequent month. At his last visit, 18 months following surgery, he continued to do well with no evidence of clinical or radiographic recurrence.
Pathological analysis revealed brain tissue infiltrated by well-differentiated piloid cells with low-to-moderate cellularity (fig. ). There was no microvascular proliferation or necrosis identified. Analysis of immunohistochemical staining revealed glial fibrillary acidic protein-positivity solely in reactive astrocytes. The proliferative index of the tumor was low, with only 2% of cells positive for Ki67 staining. Immunohistochemistry was also negative for mutant isocitrate dehydrogenase 1 (IDH1R132H). Immunohistochemistry against epithelial membrane antigen revealed a ‘dot-like’ positivity. A histological analysis revealed a tanycytic ependymoma (WHO Grade II).


A 52-year-old man presented with a 12-month history of a swelling of the distal phalanx of the right middle finger without prior trauma or infection (). The tumor was not palpable because the fingertip was plump and sometimes painful. There was no redness, discoloration, or inflammation of the skin. Sensation, motion, and perfusion were not impaired. X-ray findings showed no bony involvement. Intraoperatively, we observed a cystic brown tumor with a soft consistency (). Histologic examination revealed a cystic tubulopapillary epithelial tumor. A computed tomographic scan of the chest, ultrasound of the abdomen, and bone scintigraphy showed no evidence of metastasis. We chose an aggressive surgical approach consisting of amputation of the middle phalanx and regional lymph node dissection. The surgical margins of the finger and the lymph nodes were free of tumor. Ten-year follow-up did not show any recurrence.


A 54-year-old man presented with a 1×1 cm solid painless tumor on the dorsal side of the middle phalanx of the right middle finger. The tumor showed slow growth over 14 months. The tumor had been previously resected at another institution under local anesthesia. The histologic report disclosed an aggressive digital papillary adenocarcinoma with margins not free of tumor (–). Physical examination at our institution showed a bland scar on the middle dorsal phalanx with full function of the hand. X-ray imaging of the hand, magnetic resonance imaging, and a computed tomographic scan of the chest and abdomen were not suspicious for tumor spread. The patient refused finger amputation. An excision of the scar with adequate safety margins was done once again. There were no tumor cells detected in the histological specimen. The defect was covered with a full-thickness skin graft (). The sentinel lymph node was free of tumor. Postoperative wound healing was very good, and function of the right hand was not impaired. Staging procedures, including colonoscopy, bronchoscopy, gastroscopy, and ultrasound of the thyroid gland revealed no further tumor localization. No recurrence was detected at follow-up after 18 months.


A 30-year-old man diagnosed with DSM-IV schizophrenia, paranoid type, for 3 years. The patient had taken olanzapine 15 mg daily initially for nearly 3 years, and his weight sharply jumped from 55 to 100 kg in the first 6 months. He was unwilling to take olanzapine for the first 3 months. However, when the medication was stopped, persistent psychotic symptoms recurred. So, he had taken olanzapine 15 mg/d again for the following two years, and his weight had jumped up to 112 kg when he came to our clinic. The patient finally refused to take this medicine any more. Compared with some other atypical antipsychotics, aripiprazole is associated with fewer metabolic disturbances. So we tapered his dose of olanzapine down to 5 mg/d quickly and administered aripiprazole 5 mg/d initially. Then, olanzapine was stopped and aripiprazole was titrated up to 10 mg/d on day 5. However, he still experienced auditory hallucinations and reference delusions. So aripiprazole was increased progressively by 5 mg every 4 days until his psychotic symptoms were controlled. His psychotic symptoms improved significantly when aripiprazole was titrated up to 25 mg/d. Follow-up treatment was conducted once a month at the outpatient department. His family told us afterwards that there was occasional eye blinking and neck jerking from when the aripiprazole dose was titrated to 10 mg/d. He could generally suppress these symptoms for a while, but they eventually reemerged and had been more frequent gradually when aripiprazole was titrated up to 25 mg/d at day 19. When the patient came to our clinic again 1 month after he had taken aripiprazole initially, we used the Yale Global Tic Severity Scale to measure the severity of tic symptoms, and the score was 39. He had had childhood-onset simple motor tics since he was 10 years old, with varying characteristics, including blinking and shoulder shrugging. No abnormality was detected on a brain magnetic resonance imaging scan and electroencephalogram. Laboratory examination was normal: white blood cells, 7.12×109/L; red blood cells, 4.50×109/L; hemoglobin, 135.00 g/L; platelets, 150×109/L; aspartate transaminase, 32 μg/L; alanine transaminase, 31 μg/L; anti-streptolysin O, 32 IU/mL; and rheumatoid factor, negative. No family history of tic disorder was found.
Therefore, the dose of aripiprazole was quickly decreased to 5 mg/d in 1 week. These tic symptoms gradually improved and greatly resolved after 1–4 weeks. However, with a dose of aripiprazole 5 mg/d, the psychotic symptoms appeared again. Aripiprazole was titrated to 25 mg/d again. Two weeks later, the psychotic symptoms resolved, but the tic symptoms occurred again. For that reason, medication was changed to risperidone 5 mg daily to control his psychotic symptoms. At his follow-up visit 7 months later, he appeared to have no symptoms of schizophrenia and no tics in response to the treatment (risperidone, 5 mg/d).


A 7-year-old female, delivered at intrauterine period 38 weeks, 2.9 kg, by normal spontaneous vaginal delivery in another hospital, was diagnosed with DiGeorge's syndrome at 2 weeks of birth by chromosomal assay () following a hypocalcemic seizure. At that time, there was missing thymic shadow on her chest x-ray. There was no cardiac anomaly with presence of minimal patent ductus arteriosus. She initially came to our hospital for rehabilitation outpatient treatment at the age of 10 months. She visited pediatric department frequently because of recurrent pneumonia and upper respiratory tract infection.
At 7 years of age, she was admitted for pneumonia. Comparing with her initial chest x-ray, a bulging contour of mediastinal shadow was noted on the follow, initially thought to be an enlarged thymus (). As her previous chest ultrasonography reviewed no thymus, we first considered chest computed tomography (CT) for further evaluation. There was no abnormal blood test except mild leukocytosis, and elevated erythrocyte sedimentation rate and C-reactive protein which thought to be the result of infection. On her chest CT, ovoid enhancing lesion (1.5 cm×2.2 cm×2.3 cm) in right paratracheal area was seen (), and chest sonography was recommended.
Chest ultrasonography was done. The ovoid enhancing lesion that was seen on the chest CT was a slightly hyperechoic lesion on ultrasonography. Parenchymal echogenic pattern of the thymus was not seen (). As it was a nonspecific finding, excluding thymus was impossible. Follow-up evaluations showed up, mass size showed slight decrease as her pneumonia improved.
Regular checkup was done through 6 months. Mass shadow still existed, and magnetic resonance imaging (MRI) was recommended. Size of right upper paratracheal mass and lymph nodes were increased. According to the MRI image, there were pneumonic consolidation on both lung fields, and increased size of right upper paratracheal mass and lymphnodes. Lymphoma or lymphoproliferative disorder was suspected. Biopsy was recommended. Video assisted thoracoscopic surgery was done. The thoracic scope showed a 2-cm-sized solid mass in anterior mediastinal mass between superior vena cava and aortic arch was observed (). There was no adhesion or effusion. The mass was removed without any complication, and pathologic review was done. No immunological stain was done. Numerous enlarged follicles were irregularly distributed in various sizes and shapes. Reactive follicles showed well demarcated mantle zone and germinal center (). As it showed normal architecture of lymph node with reactive hyperplasia and there was no evidence of specific infection, special stain was not done. Final diagnosis was lymphoproliferative lesion.


A 25-year-old female presented with severe headache and was diagnosed with migraine. She was started on TPM 25 mg/day; however, she stopped the treatment after 3 days without consultation as her headache was not relieved. After 5 days of stopping the treatment with TPM, she presented at the emergency clinic of a hospital with complaints of blurred vision and severe pain in both the eyes, which were of a few hours in duration. She also complained of colored halos and headache associated with nausea with no family history of eye-related disorders. On ophthalmic examination, the visual acuity was found to be 3/60 in both the eyes, and did not show improvement in visual acuity in the pinhole test. There was bilateral lid edema, ciliary congestion, and chemosis. Both anterior chambers were found to be shallow, appeared occluded in the periphery, and pupils were reactive. Applanation tonometry revealed high intraocular pressure (IOP) of 34 and 32 mmHg, in the right and left eyes, respectively (Fig. ).
A diagnosis of AACG, precipitated following oral administration of TPM, was made. While, laser peripheral iridotomy would have been an ideal procedure for AACG, due to presence of choroidal effusion along with anterior migration of anterior structures this treatment option was not considered in this case. She was hence started on acetazolamide tablets 250 mg four times a day (QID), pilocarpine 2% eye drops QID, travoprost 0.004% OD, and dorzolamide 2% eye drops three times a day (TID). Since she had already stopped TPM, she was advised not to take it again and was reviewed the next day. The repeat ophthalmic examination on the second day showed improved vision (6/60) in both the eyes, reduction in conjuctival chemosis, and improved depth of anterior chamber, while it continued to be shallow peripherally. IOP measurements were repeated using applanation tonometry and were found to be 20 and 18 mmHg, in the right and left eyes, respectively. On the third day, her vision improved to 6/12 in the right eye and 6/6 partial (p) in the left with IOP 10 and 14 mmHg, respectively. The ophthalmoscopic examination of disc and macula was normal in both the eyes. Subsequent examination on the fifth day showed improved visual acuity 6/6 p in both the eyes and IOP was 14 and 12 mmHg and the anterior chamber appeared well formed.
She was advised to taper the anti-glaucoma medication and was examined a month later when her visual acuity was 6/6, with IOP 14 mmHg in both the eyes and was then advised via evaluation by a glaucoma specialist.
All procedures followed were in accordance with the ethical standards of the responsible committee on human experimentation (institutional and national) and with the Helsinki Declaration of 1975, as revised in 2008. Written informed consent was obtained from the patient for which identifying information is included in this case report.


First patient was a 50-year-old woman without any additional disease or previous abdominal operation. The preoperative body mass index (BMI) was 50.4, and the American Society of Anesthesiology (ASA) score was II []. Preoperative endoscopic examination of the stomach and duodenum were normal. LRYGB was performed with hand-sewn procedure. The operative time was 90 minutes with negligibly blood loss. A nasogastric tube was placed to check the anastomosis with methylene blue and a 26 French silicone tube was placed behind the gastrojejunostomy (GJ) anastomosis to provide abdominal drainage. The patient was monitored in the intensive care unit. A leakage was diagnosed on postoperative day 3 in the presence of saliva drainage with no tachycardia, fever or hemodynamic failure. Initially daily drainage volume was 300 ml. The upper gastrointestinal (UGI) series with water-soluble contrast showed a filiform leak and the drain catheter was evacuating contrast totally [].


Second patient was a 31-year-old woman with a preoperative BMI of 61, and the American Society of Anesthesiology (ASA) score was II. On gastroduodenoscopic examination there was no pathology. The patient underwent LRYGB. Two operations were performed on the same day, and the same operative procedure was applied. Approximately 300 ml daily flow rate of a leakage was observed on postoperative day 4. Patient's oral intake was stopped, and total parenteral nutrition (2500 kcal/24h) was added to treatment. The amount of drainage on the 10th day fell to 150 ml, and gastroscopic examination and covered metallic stent placement or fibrin sealent therapy were planned. The gastroscopy demonstrated the presence of the drain catheter inside the gastric pouch, and drainage catheter was withdrawn 5 cm [Figures and ]. Region of leakage was regular edged and small. Therefore, leakage was treated with endoscopic fibrin sealant injection (4 ml of TISSEEL; Baxter Healthcare, Norfolk, UK) [Figures and ]. The fistulas healed progressively and oral nutrition was authorized on fifth day after endoscopic intervention. All of the drains were removed on sixth day. The patients were discharged on seventh and ninth day.


A 48-year-old woman presented with a few months history of chronic dyspepsia and epigastric abdominal pain and discomfort. Her past medical history was unremarkable. Her physical examination on initial presentation revealed minimal tenderness in the epigastric region with no peritoneal signs. Vital signs were stable. Laboratory examination showed no biochemical abnormalities. A gastroscopy was performed and demonstrated a subepithelial lesion that was about 3 cm in size at the prepyloric area on the greater curvature of the stomach. Computed tomography scan of the abdomen and pelvis with oral and intravenous contrast showed an ill-defined, 3 cm, deeply seated mass at the gastric antrum. There was neither infiltration of the surrounding adipose tissue, nor perilesional lymph node enlargement.
An endoscopic ultrasound (EUS) examination demonstrated a 3 cm localized, intramural mass in the prepyloric region. It showed a heterogeneous appearance with an indistinct margin and mainly a hypoechoic parenchyma. Fine-needle aspiration cytology material was obtained.
Air-dried slides were first examined in the endoscopy suite using the Diff-Quik method to assess for adequacy. This was followed by processing other slides for fixation with 95% ethanol and staining by the Papanicolaou method. The smears revealed plentiful clusters of benign-appearing, uniform, cohesive, medium-sized epithelial cells with moderate amounts of granular cytoplasm and small to medium slightly hyperchromatic nuclei with delicate and thin membranes, and inconspicuous nucleoli []. Some clusters showed peripherally located nuclei mimicking acinar architecture []. Neither frank nuclear pleomorphism, nor mitotic activity was identified. The chronicity of her symptoms and persistence of gastric wall thickening prompted a decision to proceed with surgery. The patient underwent surgery. Intraoperatively, the patient was diagnosed to be a solid deeply seated intramural lesion (3 cm in diameter) located in the distal part of the stomach, without infiltration of other organs. Regional lymph nodes revealed no abnormalities.
The resected specimen measured 14 × 7 × 4 cm with attached perigastric fat measuring 5 cm in maximum thickness. Cut sections revealed a subserosal, well-circumscribed tan/yellow 3 cm solid mass with central cystic change [].
Histopathologic examination of the hematoxylin and eosin permanent slides of the mass showed pancreatic heterotopia with ducts, acini, islets of Langerhans, and intervening connective tissue []. Areas of focal chronic inflammatory reaction and fibrosis were seen. The postoperative course proved uneventful.


A 22-year-old female patient was admitted for surgery to revise her Waterston central aortopulmonary shunt. She had the history of TF, pulmonary valve stenosis, ventricular septal defect and major aortopulmonary collateral artery. Her shunt was placed at the age of 5 years then complicated with stenosis for which a stent was placed and re-dilated 1 year ago. After echocardiography reported minimal aorto-pulmonary shunt flow, a decision was made to operate the patient for revision of the central shunt. Midsternal incision was done and a polytetrafluoroethylene (PTFE) tube was inserted between the ascending aorta and right pulmonary branch, where the previous Gortex was located. Patient recovery and rehabilitation after surgery was optimal until the patient experienced abrupt left-sided pleuritic chest pain and dyspnea. Arterial O2 saturation diminished to 75%. The patient was referred to the department of nuclear medicine for perfusion and ventilation lung scanning. Perfusion lung scan was done with 4 mCi 99m Technetium labeled macroaggregated albumin (total particle count 120,000) in planar anterior, posterior, lateral and multiple oblique projections (). The left lung was generally hypoperfused more in the upper zone. No clear evidence of PTE was detected in the planar views. Additionally, the brain, renal parenchyma and the thyroid demonstrated uptake due to the right to left shunt (). Ventilation scan was not performed due to logistic problems; the available ventilation tracer in Iran is 81Kr, which is distributed only on Wednesdays. Afterward, a 2-hour delayed SPECT imaging was done (). There was a large wedge-shaped peripheral defect in the apicoposterior segment of the left upper lobe. The finding was interpreted as either acute/chronic PTE or secondary to developmental/iatrogenic vascular abnormality. Therapeutic dose of intravenous IV heparin was administrated (500 U/hour). Meanwhile, the patient underwent CT angiography reporting postoperative atelectatic changes in the bilateral lower lobes without evidence in support of PTE. The IV heparin was changed to warfarin after 5 days when the symptoms improved gradually and the arterial O2 saturation increased to 92% in room atmosphere. Pulmonary angiography was not performed as the patient was improving. She was discharged with good general condition and no specific pulmonary symptoms.


A 10-year-old boy presented with inability to walk since 2 days with difficulty in swallowing, dysphonia and drooling of saliva. There was no history of recent infection, fever, diarrhea, convulsions or trauma or recent immunization. His antenatal, postnatal details were not significant.
On clinical evaluation, he was conscious and irritable. Neurological examination revealed the generalized hypotonia; weakness started with lower limbs then progressed to involve both upper limbs. However, respiration was normal. The anterior abdominal muscles were weak, and the patient could support himself with difficulty in sitting position. He had lower motor neuron type of facial paralysis.
The patient had irritability and fundoscopy was suggestive of bilateral papilledema. Hence, cerebrospinal fluid analysis was deferred in our patient. He was treated for raised intracranial pressure with mannitol and 3% NaCl. Emergency computed tomography (CT) scan head was done which revealed vasogenic edema in bilateral fronto-parietal and left temporal regions and infarcts. Further evaluation with MRI study was suggested.
Magnetic resonance imaging brain study was done based on CT scan findings MRI revealed white matter lesions in the brain, pons and thoracic levels of the spinal cord. It revealed peripherally enhancing lesions in right fronto-parietal and left fronto-temporo-parieto-occipital region involving grey and white matter, subcortical region and corpus callosum; suggestive of ADEM. Multiple altered signal intensity areas, largest size 5.3 cm × 3.6 cm, were seen involving grey matter and bilateral periventricular deep white matter [].
Electro-diagnosis showed an important reduction of conduction velocity that is, mild generalized motor sensory axonal polyneuropathy (acute motor and sensory axonal neuropathy-GBS).
Fundus evaluation revealed bilateral optic neuritis after 3 weeks of illness [Figure and ]. However, there was no clinical deterioration of vision or retro-orbital pain.
Based on clinical findings and electromyography-nerve conduction velocity, a diagnosis of GBS was made and based on MRI findings and fundoscopy, concurrent diagnosis of ADEM and GBS was considered.
Hence, the treatment followed with intravenous immunoglobins (2 g/kg) for 2 days and intravenous methylprednisolone (30 mg/kg/day) for 3 days.
There was gradual improvement in motor power to 3/5 within about 2 weeks. The patient showed significant clinical improvement after 4 weeks and could stand with support and walk with assistance.
The patient was advised physiotherapy and discharged on oral steroids prednisolone at (1 mg/kg/day) in tapering doses over 4 weeks period. However, this could be considered as the first episode of MS with optic neuritis, therefore, our patient will be evaluated on follow-up for further episodes and treatment with immunomodulators.


NK is a Caucasian female, 15 years old, in ninth grade at presentation, with mild intellectual disability (ID), (intellectual quotient [IQ] =55–69), atrioventricular canal repair at age 6 months, and no previous psychiatric history. She presented with a 6-month history of gradual development of slow motor activity, reduced speech with increased latency, unusual movements such as repetitive movements and posturing, blunted and inappropriate affect, mood lability, sleep disturbance, slow eating, and a general decline in previously attained skills, all of which supported the catatonia diagnosis. Family history was significant for maternal anxiety. Previous medical work-up included brain magnetic resonance imaging (MRI), electroencephalograms (EEGs), computed tomography scan of sinuses, thyroid function tests, and polysomnogram to rule out sleep apnea, all with normal findings. The only exceptions were evidence of previous Mycoplasma and Epstein–Barr virus infections. Recent stressors included transition to high school and the death of a grandmother a year earlier.
At presentation, she had received low-dose clonazepam for 1 month with some improvement in symptoms. Previous medication trials included ethosuximide for 2 weeks (no effect); a trial of lisdexamfetamine (resulted in agitation, mood and sleep dysregulation, and possible hallucinations); and citalopram 20 mg, which may have led to further decline in ADL. Based on this presentation, NK was diagnosed with unspecified catatonia. Treatment with lorazepam 1.5 mg three times daily was initiated, and gradually titrated to a total dosage of 12 mg daily with some improvement in functioning. However, psychomotor slowing and mood lability persisted and was impairing. She was therefore hospitalized and started on ECT; lorazepam was continued and citalopram discontinued. She responded well to ECT and continued a tapering course of ECT, ending having had a total of 26 treatments. During this time, she was also started on lithium carbonate and amiloride 5 mg for renal protection. When her symptoms recurred 3 months later, she received a second course of 21 bilateral ECT over 7 months with 100% recovery.
She continues to receive maintenance ECT at 2-week intervals, primarily to control motor symptoms, and has received a total of 73 treatments over the past 31 months. Lithium was discontinued due to lack of response and side effects (polyuria and weight gain).