In May 2009, a 60 year-old female on regular prednisolone and methotrexate for focal myositis and vasculitis presented to her rheumatologist with a 1-year history of right shoulder pain and swelling over the right acromioclavicular joint (ACJ). She underwent a subacromial aspiration in clinic, which helped relieve her symptoms. The aspiration showed no growth. Four weeks later, the swelling increased in size and she was again aspirated in clinic. Following this, it began discharging and swabs were taken. The swab results showed no growth. Following two courses of flucloxacillin, her symptoms resolved. In June 2010, she was admitted after developing a discharging abscess anterior to the right ACJ. Investigations revealed that both her C-reactive protein (CRP) and estimated sedimentation rate (ESR) were raised at 82.7 mg/L and 89 mm/h, respectively. A radiograph was taken []. Wound swabs and shoulder joint aspirations were taken. A microbiology opinion was sought and she was commenced on oral rifampicin 300 (mg) twice daily (bd) and intravenous flucloxacillin 1 g four times a day (qds). She failed to improve and was commenced on oral sodium fusidate 500 mg three times a day (tds) and oral ciprofloxacin 500 mg bd. Following a rheumatologic consultation, methotrexate was stopped and prednisolone was continued. A magnetic resonance imaging (MRI) scan of the shoulder was performed []. Following referral to a shoulder surgeon, she underwent an arthroscopic washout of the shoulder joint, open lateral clavicle excision, and closure of wound. In view of no frank pus being observed, no drains were used. Extended cultures were requested. Multiple tissue biopsies were taken, and joint fluid was sent to the laboratory. She was commenced on oral ciprofloxacin 500 mg bd and rifampicin 600 mg bd. The latter had to be stopped due to gastrointestinal disturbance. Initial microbiology and histopathology results were negative for gram stain, ZN stain, Wade-fite stain, and periodic acid-Schiff (PAS) stain. There were foci of granulomatous inflammation. Following surgery, there was a clinical and biochemical improvement. In view of the negative results from microbiology and histology, antibiotics were stopped. Eight weeks later, M. malmoense was isolated which was sensitive to ethambutol, rifampicin, and clarithromycin. There was resistance to isoniazid and ciprofloxacin. Ethambutol 1 g once a day (od) and oral clarithromycin 500 mg bd were commenced. Twelve months following surgical treatment, the patient had clinically, functionally, and biochemically improved. In July 2011, the patient developed bilateral central scotoma secondary to ethambutol toxicity. Ethambutol was stopped with continuation of clarithromycin. She continues to be followed up with normal shoulder function and sinus resolution. However, her ocular symptoms still persist.


A 60-year-old female was referred to our radiotherapy department with vaginal stump recurrence of endometrial cancer.
The patient was diagnosed at the age of 55 years with stage IC uterine body cancer of histological grade 1 endometrioid adenocarcinoma. She underwent total abdominal hysterectomy and bilateral salpingo-oophorectomy, and received no adjuvant therapy. She visited hospital monthly for 1.5 years, at which time she suddenly stopped attending.
The patient re-presented at hospital at the age of 59 years, following a relapse of genital bleeding. Recurrence was histologically confirmed; her serum CA19-9 level was 1216.4 U/ml. Chemotherapy was started and a total of 17 cycles administered intravenously was carried out for 12 months consisting of four cycles with paclitaxel 90 mg/m2 and carboplatin (area under the plasma concentration time curve [AUC] = 2), one with cisplatin 50 mg/m2 and docetaxel 55 mg/m2, one with cisplatin 70 mg/m2 and docetaxel 70 mg/m2, followed by 11 cycles with docetaxel 55 mg/m2 and carboplatin [AUC] = 5). During the chemotherapy, tumor markers decreased for about 1 year (Fig. ), while tumor size was stable; the patient then began to complain of abdominal pain and constipation, at which time the tumor marker level showed a steep re-increase up to approximately 8 cm in maximal diameter and 149.8 cc in gross tumor volume, magnetic resonance imaging revealed tumor encasement of the bowel, and positron emission tomography (PET) showed strong fluorodeoxyglucose (FDG) accumulation at the tumor site (Fig. ).
We planned two-step radiotherapy: (i) 50 Gy of conformal EBRT in 2 Gy per fraction for 5 weeks aiming to reduce the initial tumor volume (150 ml) and to release the bowel from encasement; and (ii) a boost brachytherapy to the supposedly residual target, with pararectal spacing with gel injection to preserve the bowel. Both treatments were scheduled on an outpatient basis. Informed consent was obtained from the patient prior to treatment, which was performed with standard institutional approval. The interventional and radiotherapeutic procedures were performed by qualified specialists.


A 41-year-old man presented with left hydronephrosis, detected incidentally during a regular checkup in July 2008. A computed tomography (CT) scan of his abdomen showed an infiltrative homogeneous mass involving the left renal sinus, with low attenuation on CT (). Results of a ureteroscopic biopsy revealed nonspecific inflammation. However, result of urine analysis and microscopic examination showed no renal dysfunction or abnormality. Until July 2010, follow-up CT scans showed no significant change in perirenal infiltration ().
In March 2010, the patient was admitted to the hospital complaining of bone pain that had lasted for 10 months, with no significant history of trauma. He reported mild pain in both knees, which gradually extended to both ankles and both upper limbs. However, the pain he described had not affected his daily life. He showed no sign of fever or weight loss. His erythrocyte sedimentation rate was 23 mm/hr and his C-reactive protein was 3.09 mg/dL. Results of urine analysis, complete blood count, renal and liver function tests, and lipid profiles were normal. Plain radiography of the left knee () showed heterogeneous patchy medullary sclerosis and coarse trabeculae and osteopenia of the covered femur and tibia. Symmetric and diffuse sclerotic changes were observed on gadolinium-enhanced magnetic resonance imaging (MRI) of his knees and ankles. Involvement of mainly the meta- and diaphyses, with relative epiphyseal sparing, was observed (). Diffuse and symmetric uptake in the bilateral tibiae, fibulae, distal femora, radii, ulnae, humeri, and clavicles was observed by whole-body bone scintigraphy (). MRI of the brain and CT scan of the chest showed no abnormalities. Biopsy of the proximal tibia revealed sclerotic bony trabeculae, with lipid-laden histiocytic and lymphocytic infiltration (). As a result of immunohistochemical staining, lipid-laden histiocytes were found to be positive for CD68 () and negative for S-100 and CD1a (). Results of a bone marrow examination also revealed two focal lymphohistiocytic infiltrations, which were found to be positive for CD68 and negative for CD1a. Bone marrow examination showed no atypical cells. Bone pain described by the patient did not require intervention; therefore, a wait-and-see policy was adopted in the outpatient clinic.


A 79-year-old woman received definitive RT for International Federation of Gynecology and Obstetrics (FIGO) stage IIIB cervical cancer. EBRT was performed at a total dose of 50.4 Gy in 1.8 Gy fractions for five days each week using a 15-MV photon. Intracavitary HDR brachytherapy (microSelectron-HDR) of 24 Gy in six fractions was conducted twice each week using a tandem and colpostat. Following the completion of RT, the cervical cancer completely disappeared. After one year, the patient returned to the hospital complaining of intermittent rectal bleeding. A barium study revealed a 3-cm-long severe narrowing of the sigmoid wall, which was suspicious for sigmoid colon cancer (). The patient was referred to our hospital. Sigmoidoscopy revealed luminal narrowing with a friable hyperemic nodular mucosal change 15 cm from the anal verge (). Biopsies were performed, and the results showed focal chronic active inflammation. The patient was diagnosed as having radiation sigmoiditis and was managed conservatively.


A 58-year-old woman received definitive RT with concurrent chemotherapy for FIGO stage IIIA cervical cancer. EBRT was performed at a total dose of 50.4 Gy in 1.8 Gy fractions for five days each week using a 15-MV photon. Intracavitary HDR brachytherapy (microSelectron-HDR) of 24 Gy in six fractions was conducted twice each week using a tandem and colpostat. Six cycles of weekly cisplatin (30 mg/m2) were given during RT. The patient has been followed with no evidence of recurrence. After five years, the patient visited our emergency department with complaints of poor oral intake, lower abdominal pain and no stool passage over ten days. Sigmoidoscopy revealed an approximately 5-cm-long section of diffuse luminal narrowing in the sigmoid colon and hyperemic mucosal nodularities with mucosal edema (). The endoscopist suspected primary sigmoid colon cancer or local invasion of recurrent cervical cancer. Pathologic examination reported diffuse active inflammation with inflamed granulation tissue. A computed tomography (CT) scan also revealed thickening of the wall of the rectosigmoid colon probably caused by radiation or inflammation; however no evidence of any cancer recurrence was found (). She has been managed conservatively with laxatives and has been clinically followed with improved symptoms.


A 58-year-old woman was admitted to the hospital on 15 August, 2003 with severe left loin pain, and hematuria. Abdominal ultrasonography revealed left loin mass. Subsequent computed tomography (CT) scan confirmed a left renal mass of 6×7 cm confined to the left kidney with no evidence of spread or vascular invasion. The patient underwent left radical nepherectomy on 25 August, 2003. Pathological examination demonstrated renal cell carcinoma, clear cell type grade II. The tumor was limited to the kidney with no invasion of perinephric tissue or vascular invasion. Surgical margins were free and the patient was classified as tumor stage II (T2aN0M0). The patient was followed up every 3 months for 2 years then annually for 3 years guided by clinical examination, annual laboratory investigations and radiological imaging. Her last CT scan evaluation was September 2008, and was unremarkable with no detected disease compared with previous scans. Her annual visit was 4 months away when she discovered her breast lesion. She was disease free clinically and radiologically until March 2009, when the patient presented to the breast clinic with a painless, palpable, and rapidly growing left breast mass discovered during breast self examination 2 months previously. Clinical examination confirmed a non-tender, palpable, mobile, and firm mass in the upper outer quadrant of left breast at the 3 o'clock position with no skin infiltration. There was no clinically palpable axillary lymphadenopathy.
Bilateral mammography showed a dense well circumscribed solid mass, speculated intramammary lesion measuring 4×3 cm located in the lower outer quadrant of the left breast with adjacent parenchymal distortion and no signs of micro or macrocalcifications, and no skin or nipple retraction (Breast Imaging Reporting and Data System [BIRADS] score was IV of left breast) (). No evidence of enlarged lymph nodes. For the right breast, the BIRAD score was I, and no intramammary lesion. Breast ultrasonography showed a large well defined hypoechoic heterogeneous solid mass intramammary lesion with internal solid components that showed some vascularity. The adjacent parenchyma was just displaced without obvious distortion with no skin thickening or nipple retraction or duct ectasia. No evidence of enlarged lymph nodes. The right breast was unremarkable.
Fine needle aspiration biopsy (FNAB) was carried out and the morphological diagnosis was malignant epithelial neoplasm, consistent with renal clear cell carcinoma, metastatic deposits (). Tumor cells were strongly positive for vimentin.
The patient denied any chest, abdominal, urinary or neurological symptoms and her clinical abdominal examination was unremarkable. A whole body CT scan was carried out and CT of the chest showed a left breast mass with central nonenhanced hypodensity and surrounded by fat stranding and skin density (). Multiple pulmonary deposits of variable sizes were identified with most of the nodules subpleural in location (, ). Abdominal CT scans revealed a left renal mass in the left renal bed which showed necrotic nonenhanced area with involvement of the left psoas muscle (). There was thrombus noted in inferior vena cava. A whole body bone scan with 99mTc was negative for bone metastatic spread. The patient's hemoglobin was 9.1 g/dL, her calcium level was 9.2 mg/dL, and LDH was 720 IU/L (patient's predictors of short survival score was 3). Her case was discussed in a multidisciplinary meeting and, the breast lump was excised on 26 April 2009. Pathological examination showed metastatic clear cell renal carcinoma, with free surgical margins. The patient was referred to the renal oncologist for further management. Her performance status was II ECOG, and she was asymptomatic regarding local recurrence and pulmonary metastatic deposits. She received systemic therapy oral tyrosine kinase inhibitor sunitinib with good tolerance and a stationary course.
Six months after breast lump excision, the patient presented to the emergency room (ER) with repeated vomiting, and headache. CT of the brain with contrast revealed two metastatic deposits, the largest measuring 4×3×1.5 cm in the cerebellar region (). Craniotomy and excision biopsy was carried out and the morphological pattern demonstrated, metastatic deposit of renal clear cell carcinoma ().
The patient received whole brain irradiation 30 Gy/10 fractions/2 weeks, and she tolerated her treatment very well. Two months later she developed progressive intolerable left renal pain with radiological disease progression. The pulmonary deposits and left renal mass had both increased in size. Sunitinib was stopped and she was not a candidate for second line active therapy because of her performance status (PS III ECOG). The patient referred to the palliative care unit for palliative therapy. She survived for 81 months after diagnosis of her primary disease and 14 months following the development of breast secondaries.


A female child aged 10 years came with complaint of irritable behavior, and communication problem. The patient was absolutely normal till age of 5 years. The child is a product of consanguineous marriage wherein her father is her mother's maternal uncle. The child is born at full term normal vaginal delivery, no intranatal maternal infections, no complications after birth, the child attained age appropriate motor and language milestones till 5 years of age. The child was toilet trained and was able to control her bowels and bladder. The child also attended school wherein she learnt to recite poems and stories. She also used to take bath all by herself with soap and water under supervision.
At the age of 4 years the patient developed a severe attack of upper respiratory tract infection for which she suffered with fever and cough for 6 months and had pleural effusion for which the fluid is drained. The parents were instructed to use medications for 1 month with regular follow ups but they could not do so due to financial restraint. The patient from then on started to get severe attacks of fever and was only treated by an unqualified health worker. She stopped going to school and started to lose all the communicative milestones like talking sentences, calling her family members by name. She used to sit alone all day self-absorbed in play and showed increased anger and irritability, wherein she would hit or bite anyone who disturbed her. She stopped playing with her friends which she previously used to enjoy doing. She also stopped asking for food and would only cry if she is hungry. She also stopped taking personal hygiene and used to pick up bits of sticks and stones from floor and put in her mouth. She also did not sleep all night and used to cry for no apparent reason. She even lost the toilet training she acquired previously and started to pass stools and urine in clothes. With the above complaints the patient was brought to Mamata General Hospital to the Psychiatric OP. The patient was admitted in psychiatry ward all the investigations done like complete blood picture to rule out and blood dyscrasias, liver function tests for any metabolic abnormalities, blood urea and creatinine for renal abnormalities, urine test for sugar and proteinuria. All the investigations were normal. Computerized tomography of brain showed marked reduction of brain volume with less sulci and gyri and enlarged ventricles. IQ test revealed that the patient has an IQ of 37.5. The patient was started on resperidon 1 mg once a day and over the next 3 week she improved, the symptoms of anger and irritability have reduced and she also started to sleep normally. Her motor coordination improved, and she was able to feed herself which she previously was not able to. On subsequent follow-ups for the next 6 months child has shown improvement in communication in the form of naming objects and also her social interaction also improved wherein she started to play with other children at her home. She has been referred to a higher center wherein she is being given special training to improve her cognitive abilities. The child is scheduled for clinical interview for every 2 months with the above medication and training to assess the improvement. Till now 3 follow-up visits have take place and substantial improvement was found in above-mentioned areas.


A 57-year-old male patient presented with persistent massive bleeding per rectum for one month that developed immediately after administration of herbal enema by a religious quack for the treatment of chronic constipation. He was admitted at some other hospital for the same complaint and was managed conservatively with oral glucocorticoid, 5-aminosalicylate preparation, metronidazole, entofoam (hydrocortisone acetate) and sucralfate enema. He had history of 48 units of blood transfusions during the course of conservative management to raise the haemoglobin above 8 mg/dl, but the condition did not improve and the patient was referred to our centre for further management.
On clinical examination, the patient was pale, had tachycardia (pulse, 136/min.) and hypotension (systolic blood pressure, 70 mmHg). Abdominal examination revealed normal findings. Rectal examination showed altered blood. His haemoglobin was 5.9 g/dl with haematocrit 20%. All other blood investigations were normal. Colonoscopy revealed extensive ulcerations and friability of entire colon, more on left colon and ileal intubation also showed multiple ulcerations []. CECT of abdomen showed distended small and large bowel with no air-fluid level.
We gave the patient a trial of conservative management because the condition was not permissible to withstand a major abdominal surgery. Conservative management did not work and once again patient developed massive lower gastrointestinal bleed bringing haemoglobin down to 4.9 g%. Patient was planned for emergency laparoscopic total colectomy. On laparoscopy, there was evidence of mild ascites, thick oedematous inflamed and friable large bowel and distal 30 cm of ileum. Large bowel was very friable and developed 3 small inadvertent perforations by bowel holding grasper during dissection and manipulation of colon []. The perforations were managed by intracorporeal sutures to avoid peritoneal contamination. Total colon with distal inflamed ileum was resected laparoscopically. Hartmann procedure was done with end ileostomy at right spino-umbilical line. Specimen was retrieved by a small Pfannenstiel incision. The patient developed hyponatremia and paralytic ileus in early postoperative period that were managed conservatively. Oral liquids were started on postoperative day (POD) 3 and after that stoma started functioning. There was no wound infection. Drains were removed on POD5 and patient was discharged on POD 7 with advice of restoration of bowel continuity after 8-10 weeks.


A 3 years old girl born of non-consanguineous marriage presented with multiple yellowish papular lesions and plaques over Achilles tendons, wrists, knees, and gluteal folds [] and also involving gluteal cleft []. Lesions were asymptomatic and started when she was 6 months old. They have progressively increased in size and extent thereafter. Her father also had multiple nodular swellings over elbows [] and legs. The lipid profile of the child showed serum cholesterol level 760.3 mg/dl (normal < 200), HDL cholesterol 35.3 mg/dl (normal 45-65), LDL cholesterol 598.6 mg/dl (normal <100), VLDL cholesterol 126.4 mg/dl (normal <40) and serum triglyceride 117.7 mg/dl (normal <140). Serum cholesterol level and triglyceride level in her father were also deranged, i.e., 240mg/dl and 237 mg/dl respectively. A skin biopsy of both showed eruptive xanthomas. Her elder male sibling and mother had a normal lipid profile. On eliciting further history from other members of the family, the maternal sister of her father had similar complaints and died of acute myocardial infarction at the age of 40.
This girl was developmentally appropriate for her age with no significant history of loss of weight or appetite. There was no lymphadenopathy or organomegaly. CVS and CNS examination was normal. X-ray chest, ultrasound abdomen, ECG, echocardiogram, blood sugar, and LFTs were all normal. A diagnosis of homozygous FH was made on the basis of family history, characteristic cutaneous xanthomas since early childhood, and elevated levels of cholesterol and LDL cholesterol. After consultation with a cardiologist, the child was started on atorvastatin 10 mg daily besides dietary control of fats. The parents were counseled about the facts related to the disease and advised for regular follow up.


In May 2010, a 65-year-old male patient presented at our hospital with complaints of numerous papules and nodules in the pubic and suprapubic region and the scrotum. According to him the lesions had been present for the last 4 months. In October 2009, he had undergone partial amputation of the penis at a tertiary care hospital for an ulceroproliferative growth involving the glans penis, which was histologically proven to be a moderately differentiated SCC.
A fine needle aspiration cytology (FNAC) of the left inguinal nodes at the same teritiary care hospital where patient underwent surgery, revealed well-differentiated metastatic SCC on the left side and reactive lymphoid hyperplasia on the right side. Bilateral block resection of inguinal lymph nodes, wth skin grafting from a donor site from the thigh, was performed. However, 4 months later he developed edema of both lower limbs, scrotum, and the suprapubic region. He also developed multiple painless papules and nodules in the above areas, with some of the lesions discharging a clear fluid. A diagnosis of post-lymphadenectomy lymphangiectasia, was entertained at that time at the tertiary care hospital and the patient was treated conservatively and reassured.
He presented at our hospital in May, 2010, with multiple papules and nodules (some of them ulcerated) on the scrotum and the pubic and suprapubic regions. Local examination revealed edema of the scrotal skin and suprapubic region, a short stump of the amputated penis, postsurgical scars in both inguinal regions, and postinflammatory pigmentary changes – both hypo- and hyperpigmentation – at the donor site on the thigh []. There were multiple firm papules and nodules of about 2–3 mm in size, some of them ulcerated and discharging serous fluid, in the pubis, suprapubic region, and scrotum. A biopsy from one of the papules revealed a metastatic deposit. [] Roentgenogram of the chest and ultrasonogram of the abdomen did not reveal any metastatic deposits. Other than anemia (with hemoglobin of 8.1 gm%), the routine hematologic and biochemical investigations did not reveal any abnormality.
A diagnosis of SCC with in- transit cutaneous metastasis was made and the patient was referred back to the tertiary hospital for further management. However, he was later lost to follow-up.


A 75-year-old woman underwent uneventful phacoemulsification with intraocular lens (IOL) implantation for the cataract occurring in the right eye at a local clinic. From day 1 on, the patient complained of ocular pain. On day 3, the patient's symptoms were aggravated, and her visual acuity had decreased; the patient was transferred to our clinic. At the time of admission, the patient had light perception. Upon slit lamp examination, the patient was observed to have severe conjunctival congestion, edema and infiltration over the cornea, hypopyon of 5 mm in height, and a thickened inflammatory fibrous membrane within the anterior chamber (). We also observed wound dehiscence and severe corneal stromal melting at the site of the incision. At the time of admission, the patient underwent pars plana vitrectomy, IOL explantation and silicone oil tamponade. Aqueous humor and vitreous samples were aspirated to identify the causative organism prior to the main surgery. Additionally, the patient received intravitreal injections of vancomycin (1 mg/0.1 mL; Hanomycin, Samjim pharmaceutical, Seoul, Korea) and ceftazidime (2 mg/0.1 mL; Fortum, GlaxoSmithKline, Uxbridge, UK) at the end of the procedure before the site was sutured. At the time of surgery, the pus that had spread over the retina's surface was removed with a flute needle, but some pus remained because it was strongly adhered. Due to severe infiltrations of the cornea and the presence of fine inflammatory substances on the surface of the IOL, the fundus could not be well observed. It was possible that the IOL was contaminated, so the IOL and the lens capsule were both removed. Postoperatively, the patient received systemic antibiotics, levofloxacin 500 mg intravenous injection qd (Cravit; Daiichi Pharmaceutical, Tokyo, Japan), and levofloxacin eye drops (Cravit; Santen Pharmaceutical, Osaka, Japan) at 2-hour intervals. On postoperative day 2, silicone oil leaked from the site of corneal wound, which had been sutured using 10-0 nylon. To resolve the leakage, this site was sutured again. Due to further progression of corneal melting on postoperative day 3, there was a multiple pin-point leakage of silicone oil in the overall area of the cornea. On organism identification test of the anterior chamber and vitreous, we identified that ESBL-producing E. coli had an extensive range of resistance to penicillins, cephalosporins, aminoglycosides and levofloxacin. Therefore, levofloxacin therapy was discontinued. A subtenon injection of meropenem (Meropen; Yuhan Co., Seoul, Korea), a carbapenem antibiotic, was administered at a dosage of 50 mg/cc concomitantly with eye drops at the same concentration each hour. A 120-degree periotomy was done to allow good penetration of the antibiotics. Intravenous injection of meropenem 500 mg bid was also administered. From postoperative day 6 the silicone leakage decreased significantly. After postoperative day 10, epithelialization was initiated from the corneal limbus. Hyaluronic acid eyedrops were inoculated at two-hour intervals. On postoperative day 13 (), stromal lysis of the cornea decreased. New blood vessels began to grow from the peripheral cornea. Systemic antibiotics were used for 3 weeks. Meropenem eye drops were continued for 2 months. By postoperative month 2, inflammation had decreased. However, there were still findings suggestive of conjunctivalization of the corneal surface and the phthisis bulbi (). The patient had no light perception visual acuity.


A 52-year-old male with a 2-day history of left lower abdominal pain was admitted to a local hospital. He had been treated as left renal colic and given analgesic. Because of no recovery, he was transferred to our hospital. Physical examination revealed left lower quadrant tenderness and rebound tenderness without guarding. Laboratory test showed an elevated WBC count (20.1 × 109/l) with neutrophils of 85%. Computed tomography (CT) showed incomplete intestinal malrotation findings, including a right-sided duodenojejunal junction, left-sided hepatic flexure, SMV inversion sign (is the name given to an SMV vertically above or left lateral to the superior mesenteric artery), situation of transverse colon on the left side of the spine, a midline-positioned dilated appendix with wall enhancement and small abscess formation []. These findings were diagnostic of nonrotation of the proximal loop associated with partial rotation of the distal loop form of malrotation with ruptured appendicitis. At laparotomy, the cecum was found in the midline small bowel was located in the right abdomen []. Appendectomy was performed. Pathology revealed ruptured appendicitis. He had an uneventful recovery.


A 7-year-old boy presented with left flank pain and intermittent fever for 10 days. He was antenatally diagnosed as bilateral PUJO. Postnatal differential renal function on Diethylenetriamine pentaacetic acid (DTPA) scan was 17%. He underwent an open dismembered pyeloplasty on the left side followed by the right side at the age of 4 months and 1.5 years respectively. He was on regular follow-up for 5 years and doing well. However, he had not visited our pediatric urology clinic in the last 1 year. The current presentation was attributed to left sided pyonephrosis and empirical intravenous antibiotics were started. The patient was hemodynamically stable and the systemic examination was unremarkable except for tenderness in the left flank. The leucocyte count, kidney function tests and blood sugar levels were within normal range. The urine culture was negative. ELISA for HIV was also negative. An ultrasonography showed grossly dilated pelvicalyceal system with thinning of renal parenchyma and echogenic debris. A contrast enhanced CT scan [] corroborated the sonographic findings.
A needle biopsy was done to rule out suspicion of a neoplastic lesion and the histopathology revealed presence of broad fungal hyphae interspersed with inflammatory cells. The branching pattern of the hyphae was suggestive of mucormycosis. The urine and blood samples for fungal cultures were negative. Intravenous amphotericin B (0.6–1 mg/kg/day) was initiated and his fever subsided. After 4 weeks of therapy, a repeat CT scan showed only partial resolution of the mass []; hence nephrectomy was done []. Relative function of the left kidney was 20% on pre-nephrectomy DTPA scan. Amphotericin B was continued for 1 more week in the post operative period. The histopathology of the nephrectomy specimen revealed fungal hyphae [Figure and ]. Propensity of the organism to invade the vessels was also demonstrated []. His recovery was uneventful and is asymptomatic at 2 months follow up.


This 5-year-old girl presented with history of tonic clonic seizures for 8 months. Magnetic resonance imaging (MRI) of the brain revealed right temporo-parietal mixed intensity lesion, which was predominantly cystic. Patchy ill-defined enhancement was noted with midline shift and subfalcine herniation []. Magnetic resonance spectroscopy showed significant increase in choline and lactate peaks. She underwent right temporo-parietal craniotomy and gross total excision of the tumor. Histopathological examination (HPE) was suggestive of glioblastoma multiforme. Tumor cells were glial fibrillary acidic protein (GFAP) positive and were occasional synaptophysin positive, suggesting neuroblastic differentiation. Post-operative computerized tomography (CT) showed radical excision of lesion []. She received post-operative radiotherapy of 54 Gy in 30 fractions and 2 cycles of chemotherapy (etoposide and cisplatin). 8 months later, she presented with features of increased intracranial pressure (ICP) with history of headache, multiple episodes of seizures and an altered sensorium. Fundus examination revealed papilloedema. Repeat imaging revealed large irregularly, enhancing recurrent lesion with central cystic and hemorrhagic components in right temporo-parietal region with perilesional edema, causing mass effect and midline shift []. Imaging also showed multiple small enhancing lesions in the periventricular region around the frontal and occipital horn of the other side. She underwent right temporo-parietal re-craniotomy and gross total excision of the lesion. Post-operatively, she improved in her neurological status. Histopathological examination confirmed a glioblastoma multiforme with high mitotic index and radiation induced changes. Histopathology also revealed that GBM had primitive neuro-ectodermal tumor (PNET) like differentiation, similar to the previous HPE. In the post-operative period, the patient was neurologically stable with no new neurological deficits. Post-operative CT, done on 6th post-operative day, showed radical excision with a small amount of residual tumor in the right parietal region []. To our surprise, the periventricular lesion around the left occipital region had grown very rapidly []. Even the frontal lesion had increased in size, and new lesion was seen around the 4th ventricle []. Poor prognosis was explained to the patient's attendants, and the patient was discharged. She continued to have seizures 2 weeks after the surgery. 6 weeks after the surgery, the child developed multiple seizures, followed by decreased conscious level and expired.


A 35.5-year-old non-smoking Caucasian G2 P1011 attended for reproductive endocrinology consultation with a history of bilateral tubal disease and unilateral oophorectomy. Two years previously, the patient underwent IVF with a two-ET (at day 3) which resulted in a healthy singleton term livebirth; that treatment cycle also produced two embryos which were cryopreserved at day 3 but did not survive thaw. She returned for a second fresh IVF cycle, but follicular recruitment was poor and only one blastocyst was available for transfer. Unfortunately, her transfer was cancelled secondary to an intrauterine fluid collection identified on transvaginal ultrasound. The single blastocyst was frozen at day 6 with spontaneous resolution of the endometrial fluid the following week.
The patient elected to begin a third fresh IVF cycle 2 months later, using a combined 600 IU/day FSH (Puregon®, Organon (Ireland) Ltd; Swords, Co. Dublin) + 75 IU/day LH (Luveris®, MSD Serono Ltd; Middlesex, UK) ovulation induction protocol beginning on cycle day 2 without pituitary downregulation. Based on the endocrine status and follicular growth monitoring, we initiated 0.25 mg/day GnRH-antagonist (Orgalutran®, Organon (Ireland) Ltd; Swords, Co. Dublin) to suppress native LH activity on day 9. Five oocytes were retrieved after a 12-day follicular recruitment phase, but only one advanced to the 2pn stage following intracytoplasmic sperm injection. This single eight-cell embryo was transferred fresh under transabdominal ultrasound guidance on day 3; the remaining cryopreserved blastocyst was thawed next and transferred on day 6. A positive pregnancy test was noted 12 days after the second-stage transfer and serial hCG determinations were consistent with a normal pregnancy. Early obstetrical ultrasound was performed on day 55 and two distinct intrauterine implantation sites were noted. These structures were asymmetric and the smaller sac appeared empty. Follow-up sonograms confirmed the regression of the smaller sac (see Fig. ), and the larger gestational sac included a foetal pole with a heart rate of 110 per min. Combined estradiol/progesterone support was continued until the 12th week. An uncomplicated obstetrical course followed, and a healthy male infant (birth weight = 3,913 g) was delivered at 40 2/7 weeks’ gestation by Cesarian. Mother and baby were discharged home in good condition after 5 days; both continue to do well.


A nine-year-old boy presented to our clinic with a one-week history of a productive cough, blood-streaked sputum and increased wheezing and a five-day history of fever. He complained of a one-year history of asthma, which had previously been treated with inhaled corticosteroids with the addition of montelukast. A BCG scar confirmed previous immunization against tuberculosis. He had no history of travel to helminth endemic areas and showed no weight loss. The boy was admitted because of the exacerbation of his symptoms and a chest radiograph revealing infiltration in both lungs in April 2011 (). The possibility of pneumonia was considered. After taking a five-day course of antibiotics, which included ceftriaxone for five days and azithromycin for three days, his respiratory symptoms and physical condition improved. However, several days later, his condition worsened, with cough, severe dyspnea, perioral cyanosis, urticaria-like lesions and vesicular rashes on the skin of both lower extremities, and purpura on the hands. Additionally, he began to experience lower back pain, whole body fatigue, pyknocardia, hepatomegaly, mild splenomegaly, hyperpyrexia, bilateral swelling of the parotid gland, submaxillary gland enlargement, discrete cervical lymphadenectasis, acute abdominal pain, chest pain, polyarthralgia, myalgia, and muscle weakness. His chest X-ray demonstrated worsening of the pulmonary infiltration ().
The initial laboratory examination at the time of admission showed a peripheral white blood cell (WBC) count of 14.9×109/L (normal: 4-10×109/L) with 30.7% eosinophils (normal: 0%–5%), 41.4% neutrophils, 21.6% lymphocytes, and a hemoglobin level of 138 g/L. Notably, 11 days later, his eosinophil count increased to 72.2% with an absolute eosinophil count of 27.9×109/ L and a WBC count of 38.6×109/L. The patient's erythrocyte sedimentation rate was 77 mm/hour, and immunoglobulin E (IgE) was elevated to 1258 KU/L (reference range: 0–100 KU/L). Tests for cytoplasmic (classical) antineutrophil cytoplasmic antibodies (c-ANCA) and peri-nuclear (protoplasmic-staining) antineutrophil cytoplasmic antibodies (p-ANCA) were negative. The levels of proteinase 3 (PR3) and myeloperoxidase (MPO) were normal. Computed tomography of the chest demonstrated interstitial infiltration. Cultures and/or stains of blood and sputum for viruses, fungi, acid-fast bacilli and bacteria were all negative. His parents declined bone marrow aspiration. A peripheral blood smear analysis produced normal results. His lactate dehydrogenase level was 235 U/L (normal, 120-300 U/L). Peripheral blood lymphocyte immunophenotyping indicated that his T-cell subsets were normal. All tumor markers and antinuclear antibodies were negative. Stool cultures for parasites and the results of serum analyses for anti-Schistosoma and anti-Sparganum species antibodies were all negative, and serum samples were negative for HIV and hepatitis B and C. No other significant laboratory abnormalities were found, including blood gas levels, hepatic, renal and myocardial function, coagulation function, antistreptolysin o, complement levels, 1-3-β-D glucosan (G test) and galactomannan (GM test). Cardiac evaluations, including electrocardiography and echocardiography, revealed no abnormalities.
A skin biopsy specimen obtained from a rash on the patient's left lower extremity revealed a dermal infiltration with a considerable quantity of eosinophils (). Flexible bronchoscopy revealed moderate secretions. Bronchoalveolar lavage cytology revealed a predominance of in flammatory cells with 93% eosinophils, 2% macrophages (), and no microorganisms. A diagnosis of Churg-Strauss syndrome (CSS) was suggested. Corticosteroid therapy (intravenous methylprednisolone) was initiated at a dose of 80 mg daily (2.7 mg/kg/day), which improved the symptoms. Five days later corticosteroid (methylprednisolone tablets) was tapered at 48 mg daily (1.7 mg/kg/day). After two weeks of corticosteroids treatment, his condition stabilized and a peripheral eosinophil count decreased to 1.3%, WBC count decreased to 8.07×109/L, and the sedimentation rate decreased from 77 to 16 mm/hour. Chest computed tomography () and x-ray () revealed an improvement in pulmonary infiltration. Ten months later, the last flexible fiberoptic examination showed a clear airway, and the bronchoalveolar lavage cytology assay was normal. The corticosteroids were tapered without any systemic vasculitis over the following 11 months. At the latest follow up in late February 2012, the child was started on methylprednisolone tablets at a dose of 8 mg daily (0.3 mg/Kg/day) for two months.


A previously healthy 5-year-old girl presented with a 24-hour history of sore throat, chest pain, and shortness of breath at the Pediatric Intensive Care Unit, University Hospital, Heraklion, Greece. On physical examination there were decreased breath sounds and wheezing on the right side. There was no history of viral illness, asthma, or FBA. A posteroanterior chest radiograph revealed right lung collapse and emphysema of the left lung, with tracheal deviation and mediastinal shift []. Thoracic computed tomography scanning showed extensive multiple obstructions of the distal airways of the right lung which were initially suggestive of disseminated FBA []. Emergency bronchoscopy was performed and multiple small chewing gummi bear (HARIBO) particles impacted in the orifices of the right main bronchus and right lobar and segmentalinic bronchi were successfully removed and aspirated. Next day chest radiograph result was normal and the patient was discharged uneventfully without any complication.


A 21-year-old woman in apparently good general health was referred to us with the chief complaint of swelling on the lower left side of the face since 5 years []. It was not associated with pain, paresthesia, or discharge.
Intraoral examination revealed a fixed swelling present with respect to the symphysis and left body of the mandible. Radiographic examination showed presence of ground glass radiolucency with distinct borders and extended from the right mandibular lateral incisor to the left ramus. An impacted molar tooth was present in relation to the left ramus. There was no evidence of root resorption [Figures –]. Laboratory investigations revealed that the serum calcium, serum phosphorus, and alkaline phosphatase levels were within normal limits. The clinical impression was that this lesion was either a vascular malformation or an odontogenic cyst or tumor.
In attempt to take incisional biopsy, the lesion was explored surgically from an intraoral approach. Excessive bleeding was encountered. Vascular malformation was suspected and patient was asked to get an angiogram done. Patient did not give consent for an angiogram to be done. Surgeons on the basis of provisional diagnosis decided to resect the mandible. Patient's consent was obtained. Reconstruction was done following resection of the tumor mass.
Part of the resected specimen was submitted to the Department of Oral Pathology. The accompanying provisional diagnoses were “vascular malformation,” “hemangioma,” and “central giant cell granuloma.” After recording the gross macroscopic feature, a part of bone was removed and kept for decalcification. The bone bit was typically floating in the acid solution.
On histological examination, the lesion consisted of well-delineated lobules of mature fat separated by anatomizing septa composed of loose fibrous connective tissue. Interspersed throughout these lobules of fat were numerous endothelium-lined spaces of varying sizes [Figures and ]. These endothelium-lined spaces were congested with erythrocytes, and thrombus formation was evident at their periphery. Except for numerous mast cells present throughout the fibrous connective tissue septa, there was an absence of inflammatory cells in the lesion. There was no evidence of a hematopoietic marrow component.
A microscopic diagnosis of angiolipoma was made.


A 32 year old male patient reported to the department of Conservative Dentistry and Endodontics at Rajasthan Dental College and Hospital, with the chief complaint of pain in the posterior right mandibular tooth for the past 1 week. Patient's medical history was non contributory. Clinical examination revealed a carious and occlusally attrited right mandibular first premolar. The tooth was tender on percussion. Radiographic examination of the tooth indicated an unusual anatomy of two roots, and also there was widening of the apical periodontium, indicating periapical pathology and the necessity for root canal treatment [].
The clinical examination, radiographic examination and vitality tests led to a diagnosis of acute apical periodontitis of the right mandibular first premolar requiring endodontic therapy. The tooth was anaesthetized by way of right inferior alveolar nerve block using a 2% solution of lignocaine hydrochloride containing 1:80000 adrenaline (Lignox 2% A, Warren, Indoco). Subsequently, the tooth was isolated with a rubber dam. Endodontic access was prepared with a round diamond bur in a high speed airotor handpiece. The pulp chamber was inspected with the aid of a magnifying loupe (Seiler loupes) and a sharp DG 16 explorer was used to locate the canal orifice. After obtaining the canal patency, a #10 K file (Dentsply, Maillefer) was precurved and inserted in a distolingual direction to traverse the canal bifurcation into the second root []. A working length radiograph confirmed the presence of a single coronal canal bifurcating in the middle one third, and coinciding with the separation of the two roots. The two canals exited in separate apical foramina located in the respective roots []. Cleaning and shaping of the canals was performed using crown-down technique under copious irrigation with 5.25% sodium hypochlorite solution. The canals were dried with paper points, and the tooth was temporized. After three days, the canals were obturated with cold, lateral compaction of gutta percha cones (Dentsply) and zinc oxide eugenol sealer. A post obturation radiograph was taken to evaluate the quality of obturation [].


A 35 year old woman, Hosp. No. – A 8332 came to our OPD on 05.08.05 with chief complaints of growth in the vulva - 2 month, bleeding from the growth - 1 month, irregular period - 6 month. She is P4+1. She chews betel nut and leaf. There is no family history of malignancy. No history of HTN, DM, TB. Tubectomy was done a year ago.
On examination, GC is fair, pallor +, no palpable LN. P/A- no organomegaly, others NAD, groin- NAD. Local examination of the vulva revealed a 8 × 5 cms pedunculated growth [] having its thick pedicle of about 1.5 cm thickness hanging from the clitoreal region slightly on the left side []. P/S/V- cervix- NAD, vaginal walls- NAD, Uterus – N/S, mobile, adnexae free. P/R- NAD.
On investigation, Hb %- 9.8 gm%, ABO gr.- O +ve, RBS- 101 mg/dl, RFT-WNL, LFT-WNL. PAP Smear-NAD, CXR-NAD, USG (W/A) – NAD. Pre operative biopsy revealed Aggressive Angiomyxoma.
She was planned for wide excision of the vulval growth; accordingly it was done under SA on 18.08.05 []. On sectioning, the mass had focally infiltrating margins and a rubbery glistening grey/white surface []. Post operative period was uneventful, and patient was discharged on 27.08.05.
Post op HPE revealed Aggressive Angiomyxoma [Figures and ], cut margins negative. IHC studies revealed Desmin and Vimentin +ve, ER, PR +ve and S100 protein negative. She is under regular follow-up, and doing well for the last 5 years.


On November 17, 2011, A 44-yr-old male underwent CPB to treat a 2 vessel disease involving occlusions in the left anterior descending and right coronary arteries. He was previously diagnosed with hypertension and type 2 diabetes mellitus. There was no prior history of polydipsia, polyuria, or head injury. In addition, the patient did not have a familial history of polyuria. On the day of admission, his blood pressure was 127/66 mmHg and heart rate was 79 beats per minute. The CABG operation lasted 6 hr with a CPB time of 115 min and aortic cross clamp time of 69 min. The myocardium was protected by cold cardioplegia. There were no intra-operative hypotensive episodes or other major complications. Intra-operative mean blood pressure was maintained between 75 mmHg to 90 mmHg.
One hour after the operation, the patient's urinary output increased to about 1,000 mL/hr. His mental status was normal and there were no clinical signs of neurologic deficit. Serum creatinine levels increased slightly from 1.15 mg/dL to 1.3 mg/dL. Serum glucose and sodium concentrations were 137 mg/dL and 140 mEq/L (). Specific gravity of the urine was 1.020. The urine was also negative for protein, glucose, and occult blood. Other laboratory findings did not reveal any meaningful changes compared to the patient's pre-operative data. Over-hydration with hypertonic solutions nor large amounts of diuretics were not administered during the operation.
On the first post-operative day, the patient's urinary output was continued up to 1,050 mL/hr and he was referred to the nephrology clinic. His clinical and laboratory findings revealed a serum osmolality of 333 mOsm/kg, urine osmolality of 80 mOsm/kg, and serum sodium of 155 mEq/L (). Considering the patient's serum sodium level and osmolality, 10 mcg of a desmopressin nasal spray was administered for a desmopressin stimulation test () without water deprivation. His urine output declined dramatically to 125 mL/hr 30 min after desmopressin use. At 60 min after nasal desmopressin administration, urine osmolality increased to 518 mOsm/kg and urine output declined from 1,050 mL/hr to 80 mL/hr. Based on the laboratory data and an increase in urine osmolality of more than 100% measured by the desmopression stimulation test, the patient was diagnosed with complete central DI.
On the third post-operative day, magnetic resonance imaging (MRI) was performed to exclude brain injury that may have occurred during operation. However, there were no abnormal findings (). Urine output was stabilized by intermittent nasal desmopressin administration until the fourth post-operative day at amount of 150-200 mL/hr. Thereafter, no further therapy was necessary and the patient has remained symptom-free with no complaints of altered urinary output or excessive thirst.


A 33-year-old male was referred to our hospital with infection of the distal end of the catheter 20 days after the ventriculoperitoneal shunt. He has a past history of head injury in a road traffic accident six months ago. He presented with loss of consciousness and right temporal lobe uncal herniation. CT scan revealed right temporal lobe contusion with hematoma. Emergency craniotomy was performed for drainage of hematoma with decompressive craniectomy. Postoperative period was marked by lost consciousness. A left ventriculoperitoneal shunt was performed for traumatic hydrocephalus. Cranioplasty was accomplished after three months of decompressive craniectomy. On postoperative day 15, the patient experienced fever with temperature of 39℃ with increased intracranial tension. CT scan revealed recurrence of hydrocephalus. Blockage of peritoneal end of catheter was confirmed followed by replacement. Catheter tip was positive for staphylococcus epidermis infection. Peritoneal fluid did not show any infection. He was started on antibiotics. On postoperative day 20, oozing of pus from the abdominal incision site was noted, with wound gapping. Immediate debridement of the wound was done followed by delayed suture. The patient was then referred to our hospital. On admission he was unconscious with purulent fluid oozing out from the abdominal incision. Abdominal X-ray showed peritoneal end of the shunt was located accurately ().
Further replacement of peritoneal shunt might increase the chances of failure owing to the recurrent abdominal infection and inflammation. VA shunt was then considered. We decided to conserve the ventricular shunt and the pressure adjusted pump, with removal of the peritoneal shunt from the pump. A new distal catheter was connected to the pump. Under general anesthesia, the incision was made across the anterior border of the left sternomastoid muscle and the left common facial vein was identified. A small opening was made into the common facial vein and the shunt was then expected to pass down to the left jugular vein, subclavian vein and then to the right atrium. The passage was smooth till 6 cm of shunt remained out, then we encountered a resistance, which was subdued due to continued movement. After the complete insertion of the shunt catheter we severed the left common facial vein, ligated the distal end of the vein and the proximal end was ligated to the catheter. The chest X-ray done in the recovery room did not reveal the shunt at the T6 level (). An X-ray of neck demonstrated that the distal catheter tangled back in the jugular vein before the entry into subclavian vein (). Endovascular intervention via femoral vein intervention was then planned to draw the distal shunt into right atrium. Under local anesthesia, a 6F sheath was inserted into the right femoral vein by Seldinger method. A 5F guiding catheter was then advanced into the left subclavian vein, followed by imaging to determine the location of the shunt. A 4 mm Gooseneck loop snare was promoted through the left jugular vein to capture and ligate the tip of the shunt catheter. Finally, the shunt end was dragged into the right atrium smoothly. Post procedure chest X-ray confirmed the accurate position of the distal catheter of the shunt in right atrium (). Following endovascular re-positioning, the shunt function was optimal.


An 80-year-old male was admitted owing to recurrent chest pain for elective PCI of a chronic total occlusion (CTO) in a left circumflex (LCX) lesion. Six months previously, the patient had undergone emergent PCI in the left anterior descending artery (LAD) owing to unstable angina. Follow-up coronary angiography revealed no significant in-stent restenosis in the LAD and CTO in the LCX (). We decided to revascularize the LCX lesion. A 7-Fr JL4 guiding catheter was engaged and predilation with a 2.0×20-mm balloon was done. After ballooning, we attempted to insert the 2.5×30-mm Resolute-Integrity (Medtronic, Minneapolis, MN, USA) stent into the LCX, but the stent failed to cross the CTO lesion in the LCX. We used the deep intubation technique, but the stent could not cross the lesion entirely because the lesion was severely calcified and angulated. While attempting to push the stent, we noticed that the stent was stuck within the lesion. After a struggle, the guiding catheter was taken out but we observed that the stent was half-degloved off the balloon (). Because of the risk of stent loss and embolization, we decided to pull out the catheter and stent strut by use of an Amplatz GooseNeck snare (EV3, Plymouth, MN, USA). We pushed down the guiding catheter and stent strut together to the iliac bifurcation level. The left femoral artery was punctured and we used a 6Fr Balkin sheath to grab the stent with the snare and both the stent strut and the balloon catheter were removed successfully by the snare (). We completed the procedure and decided on medical therapy instead of PCI for the LCX lesion. Two days later, the patient was discharged and there have been no adverse events during 3 months of clinical follow-up.


A 46-year-old man with a history of radical extirpated melanoma was transferred to our hospital under the suspicion of cerebral vasculitis. One week before, he presented with spontaneous acute severe headache, followed a few days later by progressive confusion and a mild left-sided hemiparesis. Magnetic imaging resonance (MRI) of the brain showed ischemic lesions bilateral occipital and right parietal. MR angiography (MRA) showed no abnormality; however, on second review the examination was without gadolinium and of poor quality and therefore not reliable. Patient did not use any medication, smoked 20 cigarettes and 3–4 times cannabis a day. The admission blood pressure was 184/89 and the following day 154/90. On neurological examination, he appeared confused and scored 15 of 30 items on the mini-mental state examination. There was a hemiparesis MRC grade 4 on the left side. Ancillary laboratory investigations in serum (including blood count, serum electrolytes, liver, renal and thyroid function tests, erythrocyte sedimentation rate, C-reactive protein level, antinuclear and antineutrophil cytoplasmic antibody tests, complement C1q, C3 and C4, lupus anticoagulant, rheumatoid factor) and cerebrospinal fluid (CSF) showed no abnormalities. Selective catheter cerebral angiography showed multiple caliber changes in the intracranial blood vessels (Fig. ). Transcranial Doppler ultrasonography (TCD) examination showed increased intracranial blood flow velocities among others of medial cerebral artery (154 cm/s), and anterior cerebral artery (168 cm/s). Because of the acute presentation with TCH, normal CSF examination and the findings on TCD the diagnosis RCVS was established. Calcium-channel antagonist nimodipine 60 mg six times daily was started. Few days after treatment was started, the clinical condition improved. A follow-up TCD showed a normalization of blood flow velocities (medial cerebral artery: 94 cm/s and anterior cerebral artery: 90 cm/s). The follow-up MRI showed reduction in size of the hyperintense lesions. The patient was transferred to a rehabilitation center and discharged home after 3 weeks under good clinical condition.


A 48-year-old woman presented at the emergency department with spontaneous acute severe headache. She had a history of migraine, but did not recognize this type of headache as her migraine headache. She frequently used sumatriptan and 1 week before admission, because of severe attacks, she had used sumatriptan daily with temporally good response. On examination, the blood pressure was 140/88 and neurological examination was normal. SAH was ruled out by CT scan and CSF examination was completely normal. Following day, she became confused with behavioral changes. Brain MRI/MRA revealed no abnormalities. TCD showed increased blood flow velocities over the medial cerebral artery (109 cm/s) and anterior cerebral artery (108 cm/s). Under the suspicion of RCVS, calcium antagonist verapamil 120 mg twice daily was started. Within a day, the patient improved clinically and TCD after 4 days showed normal blood flow velocities of medial cerebral artery (80 cm/s) and anterior cerebral artery (66 cm/s). She was discharged at home without any complaints and was strongly advised not resume any triptans.


A case of 14-year-old girl presented to the department of Periodontics, in government dental college and hospital Aurangabad (India), with deep periodontal pockets with mandibular first molars bilaterally on distal aspect.
Radiographs were taken at 90° and other with a 30° mesial shift on both mandibular molars which depicted presence of three roots in mandibular molars Figures and . The extra distolingual root contributed for chronic localized periodontitis with mandibular first molars. Patient was referred to endodontic department and teeth were found to be vital on pulp vitality test.
On surgical interventions under local anesthesia, conventional flaps were reflected and thorough debridement of root surfaces was done. Three walled defects were explored bilaterally Figures and with grade II furcation involvement between distobuccal and distolingual roots Figures and . Regenerative therapy with hydroxyapatite grafting was carried out Figures and and flaps were approximated with direct sutures. On radiographic evaluation after six months, some bone gain was seen on right side, while on left side no change in level of bone Figures and .


A 46-year-old man presented to the urology outpatient department with history of high-grade fever and dull aching left flank pain for the past 6 months. Abdominal examination revealed a large lump in the left flank. His total leukocyte count was elevated with an increase in the absolute eosinophil count. An ultrasonography of abdomen showed a large multilocular cyst replacing the left kidney, with floating membranes inside []. A contrast-enhanced computed tomography (CECT) scan revealed the same findings without increase in enhancement or calcification of the cyst wall [Figure and ]. Serum antibody test for hydatidosis was positive. The patient was given oral albendazole 400 mg twice daily for 4 weeks preoperatively. Then, a laparoscopic transperitoneal surgery was done using the hydatid trocar cannula system.
The hydatid trocar cannula system consists of a 12-mm assembly of laparoscopic trocar and cannula along with 5-mm and 3-mm reducers.[] The trocar is 29 cm long. It is fenestrated at the tip and hollow throughout its length to accommodate a suction cannula []. Its tip is pyramidal shaped, with each facet of the pyramid bearing a fenestration to enable any fluid leaking on its insertion to be sucked into its hollow body by the suction cannula placed within. Its long shaft also bears two fenestrations opposite to each other, at a distance of 17 cm from the tip. The cannula is 26 cm long with an inner diameter of 12 mm. It has two side channels, one for irrigation and another for suction [Figure and ]. The suction channel has an inner diameter of 10 mm.
For a left-sided renal surgery, left lateral position is made with a 60° tilt from the horizontal. Initially, three ports are placed; the first one is at the umbilicus (10 mm for the camera), the second is midway between the left anterior superior iliac spine and the umbilicus on the spino-umbilical line (10 mm, working port), and the third port is 2 cm below the rib margin in the left hypochondrium (5 mm, working port). The colon is mobilized and the renal area is dissected as in standard transperitoneal laparoscopic exposure for the kidney. The cyst is exposed all around by a combination of sharp and blunt dissection, especially near the tail of the pancreas, adrenal gland, and vessels. The cyst is isolated on all sides by packing it off using gauze pieces soaked with 10% povidone iodine, especially near the proposed site of introduction of the hydatid trocar and cannula, to minimize the spillage of the hydatid contents [].
The Palanivelu hydatid system (PHS) trocar with cannula is introduced through the peritoneal cavity directly over the hydatid cyst via a 15-mm incision in the left flank. Once inside the peritoneal cavity, the trocar is removed and the cannula alone is advanced till its tip reaches over the surface of the hydatid cyst []. At this stage, suction is applied through the side channel to maintain the contact between the cyst and the cannula opening. Thereafter, the fenestrated trocar with a 5-mm suction nozzle inside connected to another suction machine is introduced into the cannula and by a steady pressure it is introduced into the cyst. Care is taken to ensure that the suction is already switched on before the trocar is introduced in the cyst. At this level, utmost care is taken to avoid any spillage. Any fluid spillage on puncture of the cyst wall is immediately suctioned either into the body of the hollow trocar through its fenestrated tip and then into the suction cannula or into the outer cannula and then into the suction side channel. Once the PHS enters into the hydatid cyst, the trocar is removed and the cavity is irrigated with a scolicidal solution through the main channel while continuous suction is simultaneously maintained all the time. In this way, fragments of laminated membrane, daughter cysts, and debris are easily removed [Figure and ]. Once the retaining fluid is clear, CO2 is insufflated at low pressure (3–4 mm Hg) and the telescope is introduced into the cavity through the cannula to visualize the interior. After confirming the complete aspiration of cyst, hypertonic saline is instilled as the scolicidal agent for 10 min. After 10 min, the scolicidal agent is suctioned and the cyst is marsupialized or excised depending upon its location. Specimen is then retrieved through a specimen-retrieval bag and a thorough lavage is given in the peritoneal cavity with normal saline.


Our patient, a 50-year-old man, a known case of hypertension, on two antihypertensive drugs, was admitted with history of progressively increasing weakness with stiffness and numbness in both lower limbs since 9 months. The patient also had history of urinary and fecal incontinence. Neurological evaluation revealed spastic paraparesis with power 3/5 (MRC grading system) and decreased sensation below L1 dermatome bilaterally. Perianal sensation and anal tone were decreased. Magnetic resonance imaging of the lumbosacral spinal segment, before and after gadolinium administration, demonstrated a lobulated lesion extending from lower border of D12 to L2 vertebra level, intimately related to conus medullaris. It was isointense on T1W images [], heterogeneous on T2W images [Figure and ] and showed intense postcontrast enhancement []. Significant scalloping of posterior margin of vertebral bodies and their posterior elements were seen []. There was extension of the lesion into bilateral L1-2 neural foramina. The patient was planned for surgery keeping the possibility of ependymoma. D12 to L2 laminectomy with subtotal excision of the tumor was done. The tumor was hyper-vascular. The rostral part of the tumor was adherent to conus and was inseparable, so left behind. During surgery, blood pressure increased considerably on few occasions, but was controlled. On postoperative day 1 the patient had flushing all over the body, specially over the face and chest region, palpitation, dysphagia, and uncontrolled blood pressure. The patient was shifted to the intensive care unit where his blood pressure was not controlled in spite of giving three antihypertensive drugs. His blood and urine were sent for all necessary investigation including the serum catecholamine level. On the same day the patient collapsed and expired after an episode of hematemesis. The histopathological examination revealed a diagnosis of paraganglioma. Typical “Zellballen nests” of neoplastic cells were present surrounded by a delicate fibro-vascular stroma. [Figure and ] The cells possessed granular eosinophilic cytoplasm and demonstrated nuclear pleomorphism. Immunohistochemical testing showed a positive reaction for chromogranin and S100 protein. Serum noradrenaline level was 2951 pg/ml (normal 100-450 pg/ml) with normal adrenaline level.


A 19-year-old male patient of Malay ethnicity was referred to our department for the treatment of a decayed upper right second premolar (). Initial periapical radiograph confirmed endodontic involvement of the offending tooth. Additionally, it revealed the presence of two radiopacities in the maxillary canine-premolar region (). The first radiopacity was cast due to the presence of a paramolar, whereas the second radiopacity gave an image of a transversely positioned "Y" shaped structure, consisting of supernumerary crowns and a short root and root canal, suggestive of a geminated supernumerary tooth. A pericoronal radiolucency of approximately 2 mm surrounding the geminated crown was found.
Panoramic radiograph revealed the presence of supernumerary teeth in both the maxilla and mandible (). To gain further insight into the buccolingual positioning of the supernumeraries, maxillary and cross sectional mandibular occlusal radiographs were recommended ().
After obtaining informed consent from the patient, computed tomographic (CT) examination was performed with a 64-slice multidetector computed tomography (MDCT) scanner (Brilliance CT 64-channel, Philips Healthcare, Andover, MA, USA) using the dental software program DentaScan (GE Healthcare, Milwaukee, WI, USA). CT slices of 0.7 mm thickness were obtained () along with the 3D reconstructed images () which revealed the precise internal and external morphology of the supernumeraries. The 'Y' shaped structure consisted of a common root and root canal for three crowns: a medially directed molariform crown with a slight widening of the pericoronal space and two partially separated "premolar-like" crowns directed inferiorly, thereby confirming the diagnosis of geminated supernumerary tooth with trifid crown. Loss of the cortical plate of the palatal vault was also evident in the axial slices.
Owing to the possibility of causing interference in the restorative treatment plan, the erupted maxillary paramolar was extracted, whereas the other impacted supernumerary teeth, except for their slight pericoronal radiolucency, displayed no associated pathology or danger to the adjacent vital structures. Hence, a decision was made to keep the patient under periodic follow-up evaluation.


A 19-year-old female sought treatment at our department complaining of a missing right maxillary incisor. Clinical and radiological (panoramic and periapical) examinations revealed that the alveolar ridge height was normal. Ridge mapping showed only 2 mm of bucco-lingual bone (Seibert class I defect) [Figures and ]. Adequate zone of keratinized gingiva was present. Bucco-lingual atrophy of the edentulous alveolar ridge made it impossible to place an implant in this area. It was decided to augment the alveolar crest horizontally. The mandibular symphysis area was selected as the donor site for augmentation.
Under local anesthesia, access to surgical site was obtained by crestal incision and by mesial and distal vertical releasing incision on facial side []. A full thickness flap was raised to expose the defect []. Subsequently, a monocortical autogenous bone graft was obtained from the symphysis area of the mandible by using piezosurgery unit [Figures and ]. Before placing the autogenous graft onto the missing tooth area, the recipient site was perforated to induce vascularity. Then, block bone graft was placed in the recipient site and screwed with a 9-mm titanium miniplate screw [], and small gaps in the edge of the autogenous bone graft was also filled []. A tension-free suturing was done for primary closure of the elevated flap. Donor and recipient site were sutured using horizontal mattress suture with simple loop for proper closure of flap [Figures and ]. Medications prescribed for postoperative use by the patients included amoxicillin 500 mg and paracetamol 650 mg thrice a day for 5 days. Chlorhexidine mouthwash of 0.2% was prescribed for use after surgery.
The patient was reviewed after a week. There was no postoperative swelling, less patient discomfort, and uneventful healing [Figures and ]. Six months postoperatively, bone mapping revealed that the patient gained bucco-lingual width of about 5 mm which was sufficient for implant placement.


A 20-year-old man presented with dizziness and swaying sensation for 3 days after a boat trip. He had experienced seasickness or dizziness either during or after the trip, which lasted 2-3 hours. However, he began to feel dizzy and a swaying sensation when he came home after an additional bus trip of about 4 hours. He complained of fatigue and drowsiness during the day. Ear symptoms were denied. He had no recent history of medication and infection. He also denied previous history of ear disease, migraine, seizure, or syncope. His family history
was unremarkable.
An examination produced no abnormal neurological or neurotological findings. Vestibular function tests including vestibular evoked myogenic potential, subjective visual vertical, caloric test, and pure-tone audiometry were normal. Brain MRI and cerebrospinal fluid findings were also normal. Eight days later the patient began to improve without medication, and his symptoms disappeared completely a further 2 days later.
The patient underwent EEG while he suffered from dizziness and disequilibrium (3 days after symptom onset), and had a follow-up EEG 7 days later when the symptoms had disappeared completely. Compared with the follow-up EEG, the EEG while having MdD symptoms indicated a significantly decreased alpha-activity current source density (8-12 Hz) at the precentral gyrus (Brodmann area 6) of the left frontal lobe () and increased beta-2 activity (19-21 Hz) at the parahippocampal gyrus (Brodmann areas 27, 30, 35, and 36) of the right mesial temporal region (). In contrast, the delta, theta, and lower beta bands did not differ significantly between the EEGs.


A 36-year old otherwise healthy male patient was referred to oral surgeon with an asymptomatic, swelling in the right buccal mucosa that he had been aware of for 2 years.
In clinical examination an obvious submucosal swelling with intact overlying mucosa was observed. The clinical appearance was similar to mesenchymal tumors, salivary gland tumors, or even bone tumor with soft tissue invasion. An omnipaque contrast CT scan was taken, in which a well-circumscribed lesion with a density similar to adipose tissue was shown [].
With a provisional diagnosis of oral lipoma under local anesthesia, surgical excision of the lesion was performed. When a deep seated, yellow, well-circumscribed soft tissue mass with a lobulated surface 4 × 3.5 × 1.2 (length × width × thickness) centimeters in size was totally excised and submitted for microscopic examination in 10% buffered formalin [Figures and ].
In pathology laboratory, the submitted specimen was processed and embedded in paraffin using standard procedures. From the paraffin block a 3 μ section was prepared and stained with Hematoxylin and Eosin stain (H and E) to allow histopathologic examination.
In microscopic examination of H and E stained slides, using light microscope (Olympus, Japan), a well-circumscribed nodular mass was seen. Which showed a distinct lobular pattern composed of mature adipocytes, cells with large clear cytoplasm and a flat dark nucleus on periphery. Thin septa of fibrous tissue was seen between the closely packed normal looking fat cells. At the borders, lesion was surrounded by a thin fibrous capsule. According to histopathologic features a definitive diagnosis of lipoma was made []. In an 18-month postsurgery follow-up no recurrence or complication has occurred.


A nine year old female patient weighing 19 kg was admitted to the pediatric ward of our hospital, with complaints of severe colicky abdominal pain, vomiting, throat pain and tingling with numbness over both upper and lower limbs since 3 days. The patient had complained of weakness since last 1 month. Patient was conscious at the time of admission with normal pulse and respiratory rate. Blood pressure was 98/70 mmHg. On admission, there was the tonic posture of both the upper limbs. Extremities were cold. Patient was on antiretroviral therapy (ART) since 3 years and 8 months. She was taking zidovudine + lamovudine + nevirapine (ZLN) combination for the initial 3 years. Due to the development of anemia, zidovudine was replaced with stavudine 8 months prior and stavudine + lamovudine + nevirapine (SLN) regimen was started. Cluster of differentiation 4 (CD4) cells count was 247/7.5% (World Health Organization - WHO clinical stage 2) before 8 months. Patient was taking cotrimoxazole since last 8 months. A few days before the admission, patient's CD4 count was 179/10% (WHO clinical stage 3). There was no past history of similar complaints and no history of trauma, viral infections (mumps, viral hepatitis), ascariasis, choledochal cysts, hyperlipidaemia and hereditary pancreatitis. Provisional diagnosis of AP associated with tetany was suspected due to ART, and ART was discontinued.
Laboratory parameters like hemoglobin, differential leukocyte count, peripheral smear examination, platelet count, erythrocyte sedimentation rate (ESR), red blood cells (RBC) indices, Random blood sugar (RBS), serum billirubin, blood urea, serum potassium, serum protein, serum glutamic pyruvic transaminase (SGPT), serum glutamic oxaloacetic transaminase (SGOT) and serum widal were normal except high total white blood cells (WBC) count (19000/cumm: reference value: 4400 to 11000/cumm). The other tests conducted and their values were as follows: Serum amylase (1186 U/L; reference value: 28 to 100 U/L), serum lipase (5410 U/L; reference value: 25 to 160 U/L), serum ionized calcium (0.56 mmol/L; reference value: 1.16 to 1.32 mmol/L), serum sodium (129 mEq/L; reference value: 135 to 145 mEq/L) and serum alkaline phosphotase (378 IU/L; reference value: 98 to 279 IU/L). Ultrasound (USG) of abdomen showed moderate amount of free fluid in the peritoneal cavity. Diagnosis was confirmed with computed tomography (CT) scan abdomen showing signs of acute necrotizing pancreatitis with peripancreatic inflammatory changes.
The patient was treated with calcium gluconate (2 ml/kg, iv, 6 hourly for 8 days), Isolyte-P (340 ml iv, 6 hourly for 5 days), cefotaxime (50 mg/kg, iv, 12 hourly for 5 days), gentamycin (4 mg/kg, iv, 24 hourly for 2 days), and dopamine (5 mcg/kg/ min, iv, 24 hourly for 9 days; infusion rate adjusted according to blood pressure). Ciprofloxacin (10 mg/kg, iv, 12 hourly for 3 days) was started on the 3rd day. Amikacin (7.5 mg/kg, iv, 12 hourly for 6 days) and metronidazole (5 mg/kg, iv, 12 hourly for 6 days) were started on the 4th day. Levofloxacin (10 mg/kg, iv, 24 hourly for 4 days) and ceftriaxone (50 mg/kg, iv, 12 hourly for 4 days) were started on the 6th day.
After dechallenge and treatment, serum amylase (186 U/L) and serum ionized calcium (1.18 mmol/L) were normal; however, the total WBC count remained to be high (20200/cumm). Symptoms of tetany disappeared on the 3rd day of admission; however, the patient's general condition was not improving. Patient was intubated due to respiratory distress, and she died on the 9th day of admission. Causality assessment with Naranjo's scale and WHO-UMC scale showed that the relationship between stavudine and acute necrotizing pancreatitis was probable. According to Modified Schumock and Thornton's criteria, this reaction was not preventable, and the Modified Hartwig and Siegel's scale showed that the reaction was severe (level 7).


A 81 year old woman was admitted to emergency department complaining of a purple discoloration and tenderness on her skin of chest wall and back, which developed rapidly over a few hours. The patient denied trauma or any other medication except clopidogrel that may cause skin hemorrhage. The patient suffered from hypertension since 10 years and had undergone coronary artery bypass grafting ten years ago. She was discharged on aspirin, ramipril, carvedilol and pantoprazole. However ouicy to be gastric irritation, clopidogrel 75 mg/day had been added instead of aspirin. The physical examination revealed a normal heart rate and regular rhythm, normal oxygenation and normal blood pressure (110/70 mmHg). The lungs were clear to auscultation bilaterally. The abdomen was soft and nontender. Examination revealed a large hematoma originating from the skin of her left chest wall and back [Figures , ]. Laboratory tests showed mildly decreased hemoglobin and hematocrit levels (10,3 g/dl and 31,6%, respectively). Platelet count (349.000 K/uL), creatinine level (1.3 mg/dl) and albumin (5.3 mg/dl) were normal. Prothrombin time, activated partial thromboblastin time, bleeding time and clotting time were within normal range. Enhanced computed tomography (CT) of thorax showed subpectoral hematoma on the left side []. Clopidogrel therapy was withheld. The patient was treated with intravenous fluids. Repeated hemogram tests showed no significant decrease in the values and the patient did not require hemo-transfusion. The patient was discharged 12 hours after admission. The hematoma completely disappeared within two weeks.


A 54-year-old woman with gravidity 3 and parity 1, who had been postmenopausal for two years, presented with a 6-month history of vaginal bleeding and general weakness. The patient looked generally anemic and fatigued, and had no past history of taking estrogen replacement therapy. In addition, the patient had a body mass index of 24.5 kg/m2 and a blood cancer antigen 125 level of 24.2 U/mL (normal range, 0 to 35 U/mL). The patient had a normal Papanicolaou smear but no notable gynecological history. Pelvic examination revealed an enlarged uterus without palpable ovaries. Transvaginal ultrasound showed a 7-cm protruding mass in the endometrial cavity with an endometrial thickness of 22.3 mm. In addition, the mass was surrounded by echogenic blood flow and it showed an ill-defined margin with the myometrium.
The patient underwent total hysterectomy with a bilateral salpingo-oophorectomy under the laparoscopic guidance under a provisional diagnosis of submucosal leiomyoma. The tumor did not involve the serosal surface of the uterus or the abdominal cavity. The cervix, adnexa and vagina appeared normal. Grossly, there was an ill-defined, relatively solid endometrial mass with a spotty necrosis, measuring 7×6×5 cm. In addition, it involved the myometrium at the fundus and body but not its serosal tissue. Following a careful examination, the endometrial mass was divided into 12 sections in total. There were no notable findings in the uterine cervix and both adnexae. The entire uterine cervix was divided into 12 sections in total from the 12 o'clock position.
Microscopically, the tumor showed a clear evidence of squamous differentiation, such as presence of intercellular bridges and keratinization (), thus confirming a well-differentiated, invasive SCC of the endometrium. The tumor involved more than 80% of the myometrial thickness and it showed a vascular invasion to the myometrium. In the adjacent non-neoplastic endometrium, there were glands with both a pseudostratification and a ciliary change and their stroma was infiltrated by inflammatory cells, plasma cells and neutrophils in particular (). There were no evidences demonstrating the presence of squamous metaplasia of the endometrium, endometrial adenocarcinoma, cervical SCC and a connection between the endometrial tumor and the squamous epithelium of the cervix. Both fallopian tubes and ovaries were normal. Moreover, there was no evidence of SCC at other sites. These findings confirmed the diagnosis of PESCC rather than submucosal leiomyoma. On immunohistochemistry, there was a positive immunoreactivity for cytokeratin 7, p63 (), and p16INK4a (), but not for cytokeratin 20 and estrogen receptor (ER) and progesterone receptor (PR). Ki-67 was stained in 80% of the tumor cells. But HPV was not detected on either in situ hybridization (ISH) or HPV DNA chip analysis. The patient underwent radiotherapy for two months. The patient showed a disease-free course during a 13-month follow-up period.


A 50-year-old Indian woman presented with dark pigmentation symmetrically distributed over cheeks, forehead, chin, and neck. She had developed asymptomatic brown-colored pigmentation over the right cheek 25 years ago, which gradually involved the left cheek. The pigment gradually increased in extent while on topical treatment over last 7-8 years, to involve both cheeks, forehead, and chin, and the color became dark brown to black. She gave a history of prolonged topical use of 2-5% hydroquinone. There was no history of pigmentation at other sites and complaints pertaining to systemic involvement. On examination, there were multiple dark brown to black discrete pin-head-sized macules coalescing to form larger macules present in a reticulate and cribriform pattern over both malar and mandibular area, chin, and forehead [Figure , ]. Atrophy and telangiectasias were present over some lesions. Rest of the skin examination was unremarkable. Histologic examination revealed ochre-colored fibers in the dermis, and homogenization of the collagen fibers in the dermis []. A diagnosis of exogenous ochronosis due to prolonged use of topical hydroquinone was made.


An edentulous 22-year-old female was referred from the Oral and Maxillofacial Surgery Department to the Prosthodontics Department at Tehran University of Medical Sciences. The chief complaint was extreme dissatisfaction with appearance, mastication and speech. A detailed medical, dental and social history was obtained.
The medical history and general physical condition were unremarkable. Dental history showed that the patient had hypodontia with some impacted teeth because of Amelogenesis Imperfecta (AI).
All the impacted teeth apart from the mandibular first right premolars due to proximity to the inferior border of the mandible and theconsequent fracture risk were surgically removed in the oral and maxillofacial surgery department.
Although clinical and radiographic examination of the patient revealed pseudo long face, malar deficiency, maxillary deficiency, mandibular prognathism, class III deformityand a wide gonial angle (). Preliminary impressions were made using irreversible hydrocolloid impression material (Alginoplast, HeraeusKulzer, South Bend, IN) and final impressions were made with zinc oxide eugenol paste (Luralite, Kerr). Maxillary cast was mounted using an arbitrary facebow (Dentatus, Dentatus Ltd., NY) on a semi-adjustable articulator (Dentatus ARH, Dentatus Ltd., NY).
The mandibular cast was connected to the maxilla using a centric relation record.Anterior tooth set-up (Vivotac/Orthotak, IvoclarVivadent, Schaan, Liechtenstein) was accomplished on the mounted casts in class III occlusion ().
The anterior acrylic teeth were coated with barium sulfate. This made the teeth opaque so that the occlusal plane could be detected in the cephalogram for Epker prediction procedure [] (). After presurgical analysis, the patient was diagnosed as having skeletal mandibular prognatism, maxillary retrognatism and an excessive vertical growth pattern. Therefore, to achieve improvement in both form and function, sagittal split ramus osteotomy (SSRO) and Lefort I osteotomy were the selected surgical procedures. Epker prediction procedure was carried out for predicting hard and soft tissue movement and the patient’s final profile.
Based on the results of this prediction and some clinical considerations (e.g., zygomatic regions, paranasal area, nasolabial and gonial angles, and SNA, SNB and ANB angles), the surgeon decided to reposition the maxilla about 2.0 mm superiorly and an advancement of about 5.0 mm and to setback the mandible 7.0 mm.
These repositionings were applied on the casts to model surgery.
Then, the posterior teeth were arranged and the dentures were fabricated in new articulation and were remounted for occlusal adjustment. This denture plays the role of a definitive splint for the edentulous patient.
Mandibular cast with dentures were set approximately 7.0 mm forward compared to its original position. So an intermediate splint (IMS) was made with self cure acrylic resin (Meliodent Cold, HerausKulzer, Ltd, Newbury, UK) at this position ().
Subsequently, the complete denture was finished and polished as the final splint.
Arch bars were bonded to the appliance with multiple holes drilled into the denture bases to accommodate circummandibular wires or palatal screws that would be used to fix these appliances to their respective jaws.
In the surgical procedure, after dissection and maxillary Lefort I osteotomy, the final splint was used and fixed to the jaws (). The IMS was put between the final splint and was fixed with wire between arch bars.
Then the maxilla was fixed with screws and miniplates (Synthes, SynthesGmblt, Switzerland).
The surgeon opened the arch bar and performed mandibular sagittal split osteotomy. The final splint was fixed by arch bar () then mandibular fixation was performed in that position by screws. The deficient alveolar ridge in the premaxilla and mandible was augmented by autogenous iliac bone graft harvested during the orthognathic surgery.


A 3-year-old symptomatic girl was diagnosed to have a large PDA, left to right (L-R) shunt, left atrial and left ventricular volume overload (LALVVO), and moderate mitral regurgitation (MR). Moderate MR was considered secondary to annular dilation from the marked LALVVO. On angiogram the PDA measured 6 mm at the pulmonary arterial end and 13 mm at the aortic ampulla. She underwent device closure of PDA using a 12 × 10 mm Cocoon duct occluder (Vascular Innovations Co., Thailand) using a standard technique.[] The pulmonary arterial pressure was documented to be less than 50% of the systemic pressure prior to device deployment. The final angiogram revealed a well-deployed device with a moderate (L-R) residual shunt []. The shunt was expected to resolve with progressive thrombus formation and the child was discharged on diuretics. At 6-month follow-up, echocardiogram revealed a significant (L-R) shunt persisting through the duct occluder, with LALVVO and moderate MR. Although there was no hemolysis, the child was symptomatic with exertional dyspnea and poor weight gain.
Deployment of a coil inside the Cocoon duct occluder followed by deployment of an Amplatzer duct occluder (ADO II) adjacent to the Cocoon device successfully reduced the large shunt to a trivial one. Hemodynamic data revealed half-systemic pulmonary artery pressure. An aortogram done in lateral, right anterior oblique and anteroposterior view revealed good positioning of the previously implanted PDA device with no deformation or change in orientation. However, there was a large left to right shunt, with contrast flowing through the device as well as exiting superolaterally from around the body of the device. Endothelialization around the central part of the device was evident angiographically with at least 1 mm separation between the device and vascular lumen similar to the observation by Javois,[] suggesting that the device was secure. The tip of a 5F Judkin's right coronary artery (RCA) catheter was forced from the retrograde route into the device. The RCA catheter was exchanged for an M-Vert 5F catheter (Merit Medical Systems, South Jordan, USA) through which a 38-8-6 free embolization coil (Cook, Bloomington, USA) was deployed inside the previously deployed device []. As the shunt remained undiminished, it was decided to deliver two additional smaller Cook embolization coils (38-3-3) inside the device. Both these smaller coils, on delivery, migrated across the device into distal left pulmonary artery branches and being inaccessible were not retrieved.
A lateral projection during delivery of both smaller coils confirmed that the catheter tip had not crossed the pulmonary end of the large PDA device. However, we presume that the delivery catheter tip may have been abutting the wall of the device with inadvertent delivery of the two small coils across the device into the space around the pulmonary end of the device. This was the evidence of the presence of a large space between the device and the wall of the PDA and the prime reason for persistence of the large residual shunt despite one large coil within the device [].
On observation over the next 48 hours, the shunt did not decrease and hence the child was catheterized again. The PDA occluder was again entered from the retrograde route with a 5Fr RCA catheter over a straight tip 035 Terumo wire. Persistent attempts to cross the device with the wire resulted in the wire exiting the body of the device into the ductal lumen superolaterally and from there into the main pulmonary artery (MPA) []. The wire entry point at the aortic disc of the device was balloon dilated with a 2 mm coronary balloon catheter over a 0.014” BMW PTCA guide wire (Abbott, Santaclara, USA). A 5Fr right coronary guiding catheter was then manipulated over the wire into the MPA. A 6 × 4 mm ADO II (AGA Medical Corporation, North Plymouth, USA) was then deployed through the guiding catheter with the distal disc at the pulmonary end, the middle cylindrical portion adjacent to the body of the Cocoon occluder and the proximal disc in the descending aorta, flush with the aortic disc of the Cocoon occluder []. Immediate post-procedure aortogram showed a significantly reduced shunt [], which further decreased to show only a restrictive trivial shunt by echocardiography 24 hours later []. Five months post-procedure, the child is reportedly asymptomatic with good weight gain and with no further episodes of respiratory tract infections. There was also no evidence of hemolysis after the second procedure.


The patient was a 68-year-old woman who was diagnosed with bilateral mixed severe hearing loss due to chronic otitis media (right ear, postoperative cholesteatoma plus tympanosclerosis; left ear, tympanosclerosis). She had undergone a tympanoplasty for the right cholesteatoma at the age of 55 years. She had worn a hearing aid on her left ear since she was 57. Revision surgery for cholesteatoma recurrence was conducted at Shinshu University Hospital when she was 66 years old. No ear ossicles were found except for the footplate, which was fixed, and tympanosclerotic lesions were found in the tympanic cavity. Reconstruction surgery was then conducted at the age of 67 years. The ossicular chain was reconstructed using a cartilage and the mastoid cavity was obliterated, with no resulting hearing improvement. She suffered with bilateral hearing loss and was not satisfied with hearing aid usage.
Otoscopic examination revealed an expansion of the right external auditory canal and a sclerotic lesion on her left tympanic membrane (). A pure-tone audiometric examination showed bilateral mixed hearing loss (). Average air conduction hearing loss was 73.8 dBHL in the right ear and 68.8 dBHL in the left ear. The average air–bone gap was 41.3 dB in the right ear and 36.3 dB in the left ear. Her maximum speech discrimination score with monosyllables was 55% (100 dB) in the right ear and 85% (100 dB) in the left ear. We evaluated the effect of her conventional hearing aids as follows. (1) Speech discrimination scores with and without the hearing aids. (2) Questionnaire regarding background noise with the hearing aids. (3) Warble tone threshold level with and without the hearing aids. (4) Speech discrimination scores with the hearing aids in quiet and in noise. (5) Questionnaire with and without the hearing aids. She did not pass items number 2–5, according to the guideline for adaptation tests of conventional hearing aids (2010) in Japan. Computed tomography (CT) of the temporal bone showed poor development of both mastoid cavities, sclerotic lesions in both tympanic cavities, and sclerosis in both RWs ().
We judged that it was difficult to improve the hearing loss in the patient's left ear by tympanoplasty, because her left conductive hearing impairment was also caused by tympanosclerosis, as in the right ear. The hearing aid evaluation tests revealed that the hearing aid's effect had been inadequate ( and ). The maximum speech discrimination score in the right ear was much worse than that in the left ear. We performed a RW-Vibroplasty with VSB in her left ear.
The surgical procedure consisted of a VSB implantation using a retroauricular approach. We found sclerotic lesions in the middle ear cavity and the RW and fixation of the stapes. The bony lip of the RW was drilled down until the RW membrane could be seen. However, drilling down the bony lip until the FMT could be placed completely on the RW membrane meant that a perilymphatic fistula might possibly be induced because of injury to the RW membrane. Therefore, to prevent the occurrence of a fistula, we decided to use the RW-Coupler, which is designed to fit smaller RW diameters, and were able to confirm good coupling (). Perichondrium was placed in contact with the RW membrane, with the FMT interposed and wrapped with the perichondrium.
Fitting of the VSB's audio processor (Amadė®) was performed at 2 months following implantation. Warble tone threshold level at the first fitting according to the postoperative audiogram () showed that functional gain at low frequencies (0.25–1 kHz) was low (). Amplification was elevated at the low frequencies and the final threshold level was calculated in . The audiologic outcomes 3 month after VSB fitting are shown in . The evaluation tests consisted of the following. (1) Speech discrimination test for monosyllables at 60, 70, and 80 dBSPL. (2) Questionnaire for environmental noise (sentences 65 dBSPL and noise 60 dBSPL in the following conditions: a train station platform, a major intersection, a rub sound with a plastic bag, the sound of dishes being washed). (3) Speech discrimination tests for monosyllables in noise (noise at 60 dBSPL and monosyllables at S/N + 0, 5, 10 dB). Speech and noise signals were delivered by two speakers frontally placed at a distance of 1 m from the center of the subject's head. Speech discrimination scores in quiet and noise and functional gain with the VSB were better than those with the conventional hearing aid. Good placement of the FMT with RW-Coupler at the RW was confirmed 3 months after implantation ().
This study was approved by the Ethics Committee of Shinshu University School of Medicine and written consent was obtained from the patient.