Synchronous or metachronous occurrence of gastrointestinal stromal tumors (GISTs) and other primary gastrointestinal neoplasms has previously been reported. However, to the best of our knowledge, there are few studies regarding metachronous multiple GISTs and adenocarcinoma of the colon. The current case of an 80-year-old male patient who underwent a laparoscopic right hemicolectomy for colonic adenocarcinoma, located in the ascending colon, is presented. Twenty-one months after receiving the laparoscopic right hemicolectomy, two new disc-like bulge lesions in the descending colon and rectosigmoid were identified during an endoscopic follow-up examination, and a segmental bowel resection was performed. The final diagnosis of multiple colonic GISTs was established as a result of histopathological examination and immunohistochemistry. Gastrointestinal stromal tumors (GISTs) are a common mesenchymal tumor of the gastrointestinal tract. GISTs were mistaken for leiomyosarcoma and other mesenchymal sarcomas prior to 1983 and have an incidence of ~1–2/100,000 worldwide . The majPreviously, the synchronous occurrence of GISTs and other primary gastrointestinal malignancies has been reported in the literature –7. HowevIn June 2008, an 80-year-old male patient was admitted to the Department of Colorectal Surgery at The Liaoning Provincial Tumor Hospital  complaining of anemia and increasing paroxysmal abdominal pain during the previous month prior to admission. The patient’s medical history was unremarkable. On physical examination, the patient’s abdomen was flat and soft without tenderness and no mass was palpated. A blood count test revealed anemia, while the levels of carcinoembryonic antigen (CEA) and carbohydrate antigen (CA)19-9 were 14.14 ng/ml and 28.9 U/l, respectively. The chest X-ray was unremarkable and an abdominal computed tomography (CT)-scan, conducted to establish the tumor staging, demonstrated that there were no sites of distant metastasis. A fiberoptic colonoscopy showed an irregular bulging mass in the ascending colon, which occupied half of the intestinal lumen, however, no other abnormalities were detected in the remaining regions of the colon and rectum. Histological examination of the biopsy specimen revealed a well-differentiated adenocarcinoma.®; F. Hoffmann-La Roche, Basel, Switzerland) as adjuvant chemotherapy at a dose of 1,250 mg/m2 twice daily, administered on days 1–14 every 21 days over 24 weeks.A laparoscopy assisted right hemicolectomy was subsequently performed. During surgery, a tumor mass, measuring 3×2 cm, which was invading the serous membrane was detected in the ascending colon; there was no evidence of lymph node metastasis . HistoloA regular follow-up regime included a complete physical examination, basic serum chemistry, a chest X-ray, an abdominal ultrasound or CT-scan, and assessments of CEA and CA19-9 levels; this regime was performed every three months for two years. A fiberoptic colonoscopy was performed annually following treatment. In April 2010, two disc-like bulging lesions were identified in the descending colon and rectosigmoid during the follow-up colonoscopy. This included one lesion, which was a distance of 40 cm from the anus at the descending colon and measured 1.5×1.5 cm. The other lesion was located at the rectosigmoid and measured 1.2×1.2 cm . An explPrevious studies have demonstrated that ~20% of patients with GISTs develop other types of cancer, with the predominant GIST-associated cancers being gastrointestinal 47%), lymphoma/leukemia (7%), prostate (9%) and breast (7%) –7. Howev%, lymphoDespite recent progress regarding the diagnosis and treatment of GISTs, little is currently known concerning the rare case of synchronous or metachronous GISTs along with tumors of different histogenesis. The majority of previous case reports describe small GISTs that were discovered during surgical procedures for another primary malignancy . In the As a result of postoperative pathological examination and immunohistochemical analysis of the relevant factors, including CD117, the diagnosis was finally determined to be colonic multiple GISTs. Based on the location, size and number of mitotic figures, and according to the improved National Institute of Health risk classification , the patAs the natural history of GISTs is unknown, the method for the effective management of GISTs that are <2 cm in size remains elusive. However, the present case indicates that a resection may be essential for a colonic cancer patient, when the endoscopic diagnosis of new lesions in the colon, during an endoscopic follow-up, is complicated.In conclusion, metachronous GISTs and adenocarcinoma in the colon are rare. However, it is important to carefully investigate and differentiate between potential lesions during a routine postoperative colonoscopy following colorectal cancer surgery, as the patient may present with rare GISTs, which may be confused with the recurrence of colorectal cancer. In addition, a surgical resection may be necessary, when the diagnosis of new neoplastic lesions during the follow-up colonoscopy of a colonic cancer patient is in doubt.


Ovarian tumors in reptiles are uncommonly reported in the literature and for green iguanas previously reported cases include teratomas, one adenocarcinoma and one papillary cystadenocarcinoma. The present report is the first of a malignant ovarian teratoma in a green iguana. Complete and detailed pathological features, differential diagnosis and surgical management of malignant ovarian teratoma are discussed in this paper.Iguana iguana) with a clinical history of persistent anorexia and progressive abdominal distension was referred to the surgery department. On physical examination, a presumptive diagnosis of follicular stasis was established. Radiographic evaluation showed a large radioopaque mass within the abdomen, which was visible both in latero-lateral and ventro-dorsal exposures. Abdominal ultrasonography showed a large intra-abdominal mass, with numerous cyst-like structures filled with liquid and a heterogeneous aspect with hypoechoic areas. Exploratory laparatomy was thus suggested and the mass was removed surgically. The histologic findings of the neoplasm were consistent with those of ovarian malignant teratoma. Surgical excision of the mass in our case was considered curative and after a follow-up period of 6 months the animal has recovered completely.A 9-year-old intact female green iguana (A malignant ovarian teratoma has not been previously reported in green iguana and should be included in the list of differential diagnosis of ovarian tumors in this species. This report will contribute to a better understanding of the pathology of this rare tumor in green iguanas. In domestic mammals, primary ovarian tumors are classified into 3 different categories based on the embryological cell of origin of the predominating neoplastic cell: epithelial tumors (adenocarcinoma and adenoma), germ cell tumors (dysgerminomas and teratomas), and sex cord tumors  .Ovarian neoplasms have been reported in different species including reptiles –4, but aTeratoma is a gonadal germ cell tumour that predominantly occurs in the gonads: the testis and ovaries . The tumThe current report is the first of a malignant ovarian teratoma in a green iguana. Pathological features, differential diagnosis and surgical management of malignant ovarian teratoma are also discussed in this paper.Iguana iguana) was presented for consult with a history of 3 weeks anorexia and a distended abdomen  at 0.2 mg/kg and butorphanol  at 1 mg/kg were administered in the musculature of the right thoracic limb. After the intravenous induction of anesthesia with alphaxalone  at 15 mg/kg, the animal was intubated using a 3.5 endotracheal tube and was kept on IPPV ventilation, using Isoflurane 1–1.5 %  and 0.6 l/min air.The laparatomy was performed using a paramedian craniocaudal incision. Egg yolk content was present in the coelomic cavity Fig. , probablPostoperative, the iguana received 20 mg/kg cephtazidime  every 72 h, 10 administrations, 0.2 mg/kg meloxicam every 48 h, 4 administration and oral fluid therapy. Several days after surgery the animal was offered food, but refused to eat, and Emeraid Herbivore® Critical Care was administered. One month after surgery a biochemical recheck was done, using Avian/Reptile profile  proving no significant alterations and the patient was discharged after the removal of the skin suture. 6 months after surgery, the animal has recovered completely. An abdominal ultrasound was performed, with no evidence of regrowth.Grossly, rising from the left ovary, a well-demarcated mass expanding and compressing the surrounding vitellogenic follicles was present. The mass was 9 × 8.5cm in size, with a gray to reddish color and a weight of 340 g compared with the right ovary (203 g). The mass was surrounded by a variably thick, smooth and well vascularized capsule originating from outer layer of the ovary. On section, the neoplastic structure showed multiple necrotic and haemorrhagic areas and variably in size cystic filled cavities containing reddish to brownish fluid Fig. .Fig. 2PaFor histological examination, samples from the neoplastic mass were fixed in 10 % phosphate buffered formalin for 24 h, embedded in paraffin wax, cut into 3–5 μm sections, and stained with hematoxylin and eosin.Histologically, the neoplasm was composed by elements of all germ cell layers , haphazardly arranged within the mass. I) Endoderm: Two different epithelial populations were present in the sample. The first one was composed by pleomorphic epithelial cells arranged in cords and islets, multifocally circumscribing variable in size cysts filled with a pale eosinophilic and globous material  Fig. . The celThe second epithelial population was organized in acini and tubules lined by 1 to 7 layers of cells Fig. . The celAccording to the largest retrospective publication to date regarding the prevalence of neoplasia in reptiles , the tumAlthough most gonadal  and extrYolk coeliomitis may be the result of yolks being released from the reproductive tract into coelomic cavity or rupture of follicles while still on the ovary . In our Iguana ovarian tissue is diffuse and intimately associated with the vena cava and adrenal gland and this makes oophorectomy technically challenging. If the procedure is incomplete, even small remnants will regrow and folliculogenesis will develop . The useIn this reported case the bilateral oophorectomy was performed using microsurgical instruments and monofilament absorbable suture material.Six months later, the patient has recovered completely with no signs of ovarian regrowth.Iguana iguana). In our opinion the condition should be included in the list of differential diagnosis of ovarian and other intra-abdominal tumors in this species.To our knowledge, this is the first reported case of malignant ovarian teratoma described antemortem in green iguana (GCT, granulosa cell tumor


Multicentric gliomas are well-separated tumors in different locations of the brain, without anatomical continuity between lesions. We report a rare case of multicentric gliomas that occurred in both supra- and infratentorial regions with different histopathology.A 27-year-old man was admitted to our hospital with mild motor weakness of the right leg. Magnetic resonance imaging (MRI) showed a large tumor occupying the left insula, extending to the left basal ganglia, so tumor resection was performed. Histological diagnosis was diffuse astrocytoma. Tumor cells showed sporadic immunoreactivity for p53 and negative immunostaining for epidermal growth factor receptor (EGFR). Postoperative course was uneventful, and adjuvant therapy was not performed. At 7 months after surgery, MRI disclosed a left cerebellar tumor displaying an irregular ring formation on enhancement with gadolinium (Gd) and marked peritumoral edema. MRI studies including T2-weighted imaging demonstrated that this paravermian tumor had no contact with the initial left insular tumor. In addition, MRI studies of the whole neuraxis, cytological examination of the cerebrospinal fluid, and neurological findings demonstrated that no dissemination had occurred through the subarachnoid space or as intracerebral metastases. Therefore, the second surgery was performed. Histological diagnosis was glioblastoma. Immunohistochemistry revealed that most tumor cells were positively stained for both p53 and EGFR but negatively stained for isocitrate dehydrogenase 1 (IDH1).We reported a case of multicentric gliomas occurring in both supra- and infratentorial regions with different histopathology. Immunohistochemical examinations suggest that different genetic pathways may participate in the occurrence of these tumors. Multicentric gliomas are well-recognized entities, but their occurrence is still rare. Among these, gliomas arising from both supra- and infratentorial regions with different histology are very rare –4. We reA 27-year-old man began to experience disorientation and visited his family doctor. Although magnetic resonance imaging (MRI) showed an abnormal mass in the left insula, extending to the left basal ganglia, he was followed without therapy. The tumor gradually increased in size, and he was referred to our department. Neurological examination on admission revealed clear consciousness and no abnormality in cranial nerve functions other than mild weakness of the right leg . MRI on initial admission showed a tumor mass  extending from the left insula to the basal ganglia. The lesion was hypointense on T1-weighted imaging and hyperintense on T2-weighted imaging. The lesion was not enhanced on administration of gadolinium (Gd) Fig. . No otheMRI performed 3 months after surgery did not show any growth of the left insular tumor or any other lesion Fig. . The resThe clinical study of the abovementioned case report was approved by the Ethics Committee for Clinical Research of Ehime University Hospital, and informed consent was obtained from the patient prior to initiating the study.As for the definition of multicentric gliomas, Batzdorf and Malamud described the criteria that multifocal tumors should be distinguished from multicentric tumors . The mulMulticentric gliomas more often affect middle-aged men and show rapid tumor progression, and the prognosis is extremely poor. Many reported cases of multicentric gliomas occur in the supratentorial compartment, and occurrence in both supra- and infratentorial regions is very rare –7. To thThe pathogenesis of multicentric gliomas remains unclear. Many hypotheses have been proposed to explain the multicentricity. Willis et al. suggested that multicentric lesions could result from a two-step process . At firsReis et al. investigated genetic changes to compare primary and secondary glioblastomas and reported that these tumors were independent of each other, with showing a different pattern of genetic mutations . FurtherWe reported a case of multicentric gliomas occurring in the supra- and infratentorial regions with different histopathology. One tumor was diffuse astrocytoma of the left insula, while the other was glioblastoma of the cerebellum. Immunohistochemical analysis suggested that these tumors might have occurred by different pathways of neoplastic transformation in glial cells.EGFR, epidermal growth factor receptor; Gd, gadolinium; IDH1, isocitrate dehydrogenase 1; MRI, magnetic resonance imaging; PTEN, phosphatase and tensin homologue deleted from chromosome 10.


Cases of neonatal gastrointestinal system (GIS) obstruction are quite complex for pediatric surgery clinics. A rare cause of intestinal obstruction is the duplication cyst (DC). A three-day-old male patient presented at our clinic with a history of abdominal distension and bilious vomiting on the second day following birth. Although pathology had not yet been determined from observation and examination, surgery was performed when the patient could not tolerate oral feeding. An ileal DC forming an incomplete obstruction was observed. Ileoileal anastomosis was performed on the patient. Because DCs can present with different clinical symptoms, it is quite difficult to diagnose them in neonate patients. Lacking an imaging method that can provide an exact diagnosis, the diagnostic laparotomy is a suitable approach for both diagnosis and treatment to avoid delays in treatment. Duplication cysts (DCs), which may have either a spherical cystic or a tubular structure, are among the more rarely observed of congenital malformations of the gastrointestinal system (GIS). The first DC was described in 1733 by Calderin, followed by another in 1884 by Fitz. In 1937, Ladd defined DCs as having the following three properties: (a) the cyst is surrounded by smooth muscle, (b) the cyst must contain the GIS mucosa from which it takes its own origin, and (c) the cyst must have a wall in common with the anatomic region in which it is found [Though they are more often observed in the small intestine, DCs can also more rarely be rectal, duodenal, gastric, and thoracoabdominal. They have an incidence of 1/100,000 . They beThe literature demonstrates that delays may occur in treatment as DCs are rarely diagnosed in the neonatal period. We aim to present this rare case of ileal DC together with a literature review.A three-day-old newborn male was admitted to the emergency department with a history of abdominal distension and bilious vomiting on the second day following birth. No pathology was visualized on either direct abdominal radiography or abdominal ultrasound. Routine tests and thyroid hormone levels were normal. Contrast enema radiography was also performed to examine the patient for neonatal congenital bowel pathology. No pathology was observed. After two days of normal oral feeding vomiting began again. Upon obtaining drainage containing fecalith from nasogastric intubation, together with an increase in distension, an emergency operation was planned. The exploration, performed on the fifth day following birth, revealed a cystic DC approximately 2 × 1 cm in size that was creating pressure in the lumen of the terminal ileum and causing distension in the proximal ileum . Ileal rDCs of the GIS are the result of one or more congenital anomalies of uncertain etiology. Theories regarding etiology include the persistence of fetal bowel diverticula, vacuolization, caudal duplication, and split notochord, but the theory that has gained the most acceptance has been split notochord syndrome . DCs canUltrasound and barium studies are among the most widely used imaging methods in the diagnosis of duplication cysts. CT and MRI scans are considered less necessary. Duplication cysts are defined on ultrasound by the presence of a hypoechoic outer muscular layer with an echogenic internal mucosal layer. Surgical excision is the recommended therapy for DCs .DCs with different localizations and clinical symptoms may be encountered. Because there is no specific marker or imaging tool for them, we must include them among our differential diagnoses when patients become symptomatic. The diagnosis of DCs in the neonatal and infant periods can be quite difficult because patients present with nonspecific symptoms. Due to the difficulty of diagnosis in the period of infancy, fatal cases have been reported in the literature, diagnosed too late after clinical GIS obstruction . We too DCs may be of either a spherical cystic or a tubular type. In either case, the approach to treatment is the same: removal of the DC and anastomosis. We were unable to establish a preoperative diagnosis in our patient using ultrasound. We were able to diagnose the patient using explorative laparotomy, given the patient's prominent neonatal intestinal obstruction symptoms. The cystic-type DC we found in the ileum fit the classical definition and was exerting pressure on the lumen. We performed a resection and anastomosis as recommended by the literature, and we observed a complete recovery in the patient.In conclusion, DC may be the etiology in neonatal congenital intestinal obstruction, albeit rarely. Delays may occur in diagnosis due to the incomplete obstruction which occurs in most cases. We are of the opinion that DC is an important provisional diagnosis for patients with recurrent complaints of intestinal obstruction.


Serous psammocarcinoma is a rare variant of serous carcinoma arising from either ovary or peritoneum, characterized by massive psammoma body formation, low grade cytologic features, and invasiveness. Its clinical behavior is similar to serous borderline tumors with relatively favorable prognosis. We report herein a case of a 60-year-old postmenopausal woman who presented with abdominal distension. Contrast enhanced computed tomography (CECT) revealed calcified pelvic masses with ascites. Elevated serum CA-125 (970 U/mL) suggested malignant ovarian neoplasm. Patient underwent exploratory laparotomy with primary debulking surgery. Histopathology showed bilateral serous psammocarcinoma of ovary with invasive implants on omentum. Adjuvant chemotherapy was advised in view of advanced stage disease, although its benefits are poorly defined due to rarity of the tumor. However, patient opted out of it and is now on follow-up. Serous psammocarcinoma is a rare variant of serous carcinoma arising either from ovary or peritoneum . It is c3 on left side and 12 × 8 × 8 cm3 on the right  revealed ascites and calcified pelvic masses measuring 12 × 9 × 8 cmhe right . Serum COn exploratory laparotomy, 5 litres of ascites fluid was drained. Bilateral ovaries were enlarged with tumor nodules on surface . Periton3 and right ovary measuring 14 × 10 × 8 cm3 (Grossly, bilateral ovarian surface was bosselated with breach in capsule. Both ovaries were enlarged with left ovary measuring 14 × 11 × 10 cm × 8 cm3 . Cut sec × 8 cm3 . There w × 8 cm3 . In viewSerous psammocarcinoma was first described by Grimaldi et al. in 1916 . Gilks ePsammoma bodies are multiple, discrete, and laminated calcified bodies formed by accumulation of calcium on single necrotic or degenerated tumor cells. Psammoma bodies have rarely been demonstrated in cervicovaginal Pap smears and in cytology of peritoneal washing in cases of ovarian psammocarcinoma , 6. HistPsammocarcinoma of ovary is characterized by low proliferative activity with Ki67 and diploid DNA . ActivatClinically, patients usually present with abdominal discomfort and increase in abdominal girth. These tumors are usually unilateral, but rarely may these be bilateral as in our case . CA-125 Psammocarcinoma has been suggested to have more favourable prognosis compared to invasive serous adenocarcinoma with low recurrence following tumor resection . AlthougBecause of the rarity of this tumor, there are no standard treatment guidelines . Most auWe report this case of bilateral serous psammocarcinoma of ovary as it is a rare variant of serous carcinoma of ovary, usually associated with better prognosis in advanced stages also. Our case was also in advanced stage but there is no recurrence of disease either clinically or radiologically without chemotherapy. Her serum CA-125 level also decreased and is presently within normal range. Although the standard treatment of ovarian psammocarcinoma is surgery followed by few cycles of chemotherapy in the aggressive forms, we currently lack evidence of increasing the benefits that can bring chemotherapy in the management of advanced ovarian psammocarcinomas. Future trials in wide yard are required to establish the role of postoperative chemotherapy in such cases.


Angioleiomyoma is a benign smooth muscle tumor. It originates in the tunica media of veins. In general, it arises from the dermis, subcutaneous fat and fascia of lower extremities in middle-aged women. The case of intra-articular occurrence is very rare.This case report describes a 30-year-old Asian man with a 6-month history of recurrent pain localized in the lateral side of his left knee. Magnetic resonance imaging revealed an isolated intra-articular lesion. We performed a surgical excision of this lesion. On histological examination, the diagnosis of angioleiomyoma was confirmed. After the surgery, he recovered completely. Furthermore, no sign of recurrence was observed 1 year after surgery.In this study, we report a rare case of intra-articular angioleiomyoma of the knee. The mass showed atypical findings on magnetic resonance imaging including gadolinium-enhanced imaging in comparison to previously reported intra-articular lesions. This intra-articular tumor is difficult to diagnose prior to surgery due to paucity of information. Angioleiomyoma is a rare form of leiomyoma, originating from the tunica media of the vessel walls; it is a benign tumor with a predilection for the lower extremities in middle-aged women. In general, angioleiomyoma is painful and located in the subcutaneous fat and fascia ,2, but iA 30-year-old Asian man presented with a 6-month history of recurrent pain localized to the lateral side of his left knee. A physical examination revealed slight tenderness and swelling in the lateral side of his knee; however, no other abnormality was noted. Routine radiography showed no remarkable findings, and laboratory data were normal. MRI revealed an intra-articular space-occupying lesion (SOL) in his knee joint, measuring 1.5cm in diameter, which was invading his tibia Figure . No otheAngioleiomyoma generally presents as a solitary and painful subcutaneous lesion occurring in the extremities, most frequently in the lower limb. The first comprehensive review of this rare tumor was published by Stout in 1937 . To the Gd-enhanced MRI did not appear to be useful in this specific tumor, but it can still be useful in distinguishing between benign and malignant tumors, evaluating tumor extent, regional metastasis, and identifying satellite lesions. Intra-articular malignant conditions are rare, but cases have been reported such as synovial sarcoma and chondrosarcoma ,11. MRI To the best of our knowledge, no other cases with SOL in the knee joint, which is compatible with our MRI findings has been reported. Although the precise diagnosis can only be confirmed by histological examination, MRI with Gd enhancement before surgery could be helpful. Excision of the tumor generally results in complete resolution of symptoms, and a recurrence case has not been reported in the literature. The patient in this case recovered completely after the surgery and showed no signs of recurrence at the 1-year follow-up in an MRI examination.We describe a rare case of angioleiomyoma that arose in the knee joint. In this case, the symptoms of which the patient complained were different from those of typical subcutaneous angioleiomyoma. Atypical MRI findings in comparison to previously reported cases were observed especially in terms of Gd enhancement. Our patient became completely free of his preoperative symptoms, and no recurrence had occurred at 1 year follow-up. We should be aware of atypical locations of angioleiomyoma as in our case.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.TF and YA performed the surgical procedure and obtained the patient data. TF and TS were major contributors in writing the manuscript. All authors read and approved the final manuscript.


Pleuropulmonary blastoma is a very rare, aggressive, embryonal pulmonary neoplasm which mostly affects children under the age of 5. According to the histopathological features, three subtypes of pleuropulmonary blastoma have been recognized: type I (purely cystic), type II (grossly visible cystic and solid elements) and type III (purely solid). Characteristics of type I and type II blastoma allow an earlier diagnosis compared with type III. Here we present a case report of an unusual presentation of type III pleuropulmonary blastoma.We describe the case of a 7-month-old female baby of Italian mother and Kurdish father who was diagnosed with type III pleuropulmonary blastoma, which entirely occupied her right hemithorax.The reported case is an unusual presentation because type III pleuropulmonary blastoma typically occurs in older children. The complete re-expansion of her residual, previously totally compressed, right lung observed immediately after the resection of the lesion suggests an atypical rapid growth of this embryonal tumor in the late phase of gestation or after delivery. This case report suggests that, in addition to other childhood tumors, type III pleuropulmonary blastoma should be included in the differential diagnosis of solid nonhomogeneous thoracic large masses, compressing the mediastinal and chest wall structures in infants. This is an original case report of interest for several specialities such us pediatrics, radiology, surgery and oncology. Pleuropulmonary blastoma (PPB) is a rare malignant embryonal tumor of the lungs that appears to arise during organ development and is usually diagnosed in the pediatric population under the age of 5 ,2. The fSince type I PPB may recur after treatment as a type II or type III PPB and has a low recovery rate, it is vital to diagnose and treat the affected children at an early stage (type I PPB) and age, when the lesion is limited and most curable ,4. Unfor2) supplementation (an inspired fraction of O2=5L/minute) to achieve an O2 saturation of 95%. Her blood pressure was 50/90mmHg, heart rate 135 beats per minute and body temperature 36.5°C. An absence of breath sounds in her right hemithorax was perceived during her chest examination. Her white blood cells were 19.23×103/mm3 (neutrophils 62.6% and lymphocytes 27%), hematocrit 34.5%, mean corpuscular hemoglobin concentration 30.7g/dL, platelet counts 695×103/mm3, reticulocytes 182×109/L. Values lower than normal levels were detected for total proteins (5.3g/dL), albumin (2.8g/dL), iron (17μg/dL), immunoglobulin (Ig) A (30mg/dL) and IgG (625mg/dL), whereas her erythrocyte sedimentation rate (44mm/hour) and C-reactive protein  were elevated.Conventional chest radiography demonstrated a complete opacification of her right hemithorax with contralateral midline shift. Computed tomography (CT) scanning was performed and showed the presence of an enormous mass resulting in the contralateral displacement of her trachea, with total collapse of right lung parenchyma (Figure A 7-month-old female baby of Italian mother and Kurdish father was referred to our Pediatric Department for rapidly worsening dyspnea leading to severe shortness of breath. Respiratory symptoms, occurring intermittently in her first 2 to 3 months of life during upper respiratory tract infection, progressively worsened in length and severity and were subsequently associated with anorexia and failure to thrive. On admission, she appeared dyspneic and hypoxic and needed a high-level of oxygen (OThe occurrence of a type III PPB occupying the whole right hemithorax in a 7-month-old female baby is an unusual finding, suggesting an atypical rapid progression of this embryonal tumor in the late phase of gestation or after delivery. In our patient, nonspecific symptoms such as cough and mild dyspnea were noticed during infectious processes of the respiratory tract occurring 4 to 5 months before the final diagnosis. These symptoms rapidly worsened leading to respiratory failure. Presurgical imaging techniques clarified the features and the extension of the mass yet the specific diagnosis was based on histological examination. The radical resection of the mass and the absence of metastasis are favorable prognostic factors. Indeed, in addition to the histological type, the survival rate depends on the local recurrence and the risk of secondary localization in the lungs, thoracic structures, brain or spinal cord . From a Written informed consent was obtained from the parents of the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.2: Oxygen; PPB: Pleuropulmonary blastoma; IgA: Immunoglobulin A; IgG: Immunoglobulin G; CD56: Cluster of differentiation 56; IVADo: Ifosfamide, vincristine, actinomycin D and doxorubicin; 3D: Three dimensional.CT: Computed tomography; OAll authors declare that they have no competing interests.ADC, EP, GM contributed to acquisition of data. AC, CF, SL, GAR and MLR drafted the report and all authors contributed to revision of the report. All authors read and approved the final manuscript.


Ewing sarcomas are highly aggressive malignant tumours occurring predominantly in the long bones of the extremities in children and young adults. About 20 % of patients will present with metastases at diagnosis with the commonest sites being the lungs, bone and bone marrow. Cases of primary small bowel Ewing sarcomas have been described but are nonetheless exceedingly rare, even more so cases of metastasis to the small bowel.We describe a case of vertebral Ewing sarcoma in a 44 year-old female which metastasized to the jejunum causing intussusception.Ewing’s sarcoma is highly aggressive and presence of metastases, overt or subclinical, is thought to be present in almost all patients at diagnosis. As evidenced by our patient, metastatic disease can progress rapidly to cause further complications and confer a poorer survival. The possibility of metastasis, no matter how rare or unlikely the site is, should be considered and actively investigated to expedite treatment of the primary disease. Ewing sarcomas are rare small round cell tumours that arise predominantly in children and young adults with a slight male predilection , 4, 7. EA previously well 44-year-old female presented with acute lower limb numbness and weakness of 1-day duration. Prior to this, she suffered from back pain for 2 weeks that gradually progressed to pain in both hips as well. There were no complaints of urinary or bowel incontinence. She did not experience any abdominal pain, distension, vomiting or constipation.On examination, the patient had decreased sensation and power in the bilateral lower limbs and up-going plantar reflexes. Abdominal examination revealed no mass or distension. Bowel sounds were active. Anal tone was intact on digital rectal examination.Magnetic resonance imaging of her thoracic and lumbar spine with 10 ml intravenous Magnevist showed abnormal marrow replacement affecting the 11th thoracic vertebral body associated with enhancement causing compression of the spinal cord Fig.  was deteThe patient was planned for chemotherapy but, 3 weeks after her spine surgery, she developed symptoms of intestinal obstruction with persistent vomiting, intermittent upper abdominal pain and distension. A repeat CT scan showed multiple new lytic lesions in the vertebrae, right iliac bone and right femur, increase in size and number of liver nodules Fig.  and paraThe patient made an uneventful recovery from surgery and was started on palliative chemotherapy. She eventually succumbed to progressive metastatic disease 16 months after her initial diagnosis.EWS gene (22q12) and the 3ʹ half of members of the ETS family of transcription factors, leading to the understanding that ESFT represents a single neoplastic entity [The Ewing sarcoma family of tumors (ESFT) consists of Ewing sarcoma, peripheral primitive neuroectodermal tumor (PNET), extraosseous Ewing sarcoma (EES), and Askin’s tumor . ESFT tumours are of neural crest derivation that differentiate along a neuroendocrine lineage and are described as small round cell tumours. All ESFT tumours are characterized by a balanced chromosomal translocation between the 5ʹ half of the Ewing sarcoma predominantly affects children and young adults with a peak incidence between 10 and 20 years of age. About 30 % occur in adults over the age of 20 and fewer than 5 % occur in adults over the age of 40 . Ewing sThe presence of metastasis is the single most important factor in determining survival in ESFT patients. Patients with metastatic disease at diagnosis have a dismal 5-year survival rate varying from 0–25 %, compared with 40–79 % for those with localized disease . It is pIntussusception is the invagination of a bowel loop with its mesenteric fold (intussusceptum) into the lumen of a contiguous portion of the bowel (intussuscipiens) as a result of peristalsis. Intussusception is rare in adults, accounting for 1–5 % of all cases of intestinal obstruction and 5 % of all intussusceptions . Most caEwing’s sarcoma is a highly aggressive small round cell tumour that rarely arises in adults. The presence of metastases, overt or subclinical, is thought to be present in almost all patients at diagnosis and, as evidenced by our patient, can progress rapidly to cause further complications and confer a poorer survival. The possibility of metastasis, no matter how rare or unlikely the site is, should be considered and actively investigated to expedite treatment of the primary disease.Written informed consent was obtained from the patient for the publication of this report and any accompanying images. A copy of the written consent is available for re-view by the Editor-in-Chief of this journal.


Angiosarcoma of the urinary bladder is an extremely rare and poorly characterized tumor. We are presenting the 13th reported patient who was an 89-year-old man initially presented with massive hematuria. His past medical history included external-beam radiation for prostate cancer 12 years ago. His PSA was 0.26 ng/dL. His CT-Urography demonstrated a highly vascular mass originating from the bladder base. The mass was partially resected, transurethrally. The pathology was consistent with primary angiosarcoma of the urinary bladder. Bone scan and CT-U showed metastasis to spine. The patient was treated with palliative radiotherapy for back pain due to metastasis, and he refused chemotherapy. The patient died 3 months after his initial diagnosis. Nonurothelial tumors of the urinary bladder represent less than 5% of all bladder tumors, with sarcoma being the most common of which . AngiosaAn 89-year-old male presented to ER with massive hematuria. Two large caliber intravenous cannulas were placed and blood tests for CBC, biochemistry, and blood type were taken. A 3-way 22Fr catheter was urethrally inserted and bladder irrigation with normal saline started. His past medical history included T1 prostatic adenocarcinoma for which he received external beam radiotherapy 12 years ago. His physical examination and digital rectal examination were unremarkable. His creatinine was 0.9 mg/dL, PSA was 0.26 ng/mL, and cytology was negative for malignant cells. CT-Urography demonstrated a large highly vascular mass with irregular margins originating from the left bladder wall . Compute3 of grey tissue and blood clots. Microscopically, the tissue diagnosis was consistent with primary angiosarcoma of urinary bladder invading muscularis layer, with anastomosing vascular channels (Macroscopically, the specimen was 6 × 5 × 4 cmchannels . The tumchannels , three mchannels . No prosThe patient was referred to palliative radiotherapy because of symptomatic spinal metastasis. The patient was consulted about the option of chemotherapy but he denied. The patient died 3 months after the initial diagnosis.Nonurothelial neoplasms of the urinary bladder are extremely rare. Of these neoplasms, sarcoma represents the most common nonurothelial neoplasm of the urinary bladder . The mosFour cases of secondary angiosarcoma of the bladder were previously reported, originating from the penis , pelvis Angiosarcoma is a very aggressive tumor carrying poor prognosis . Most ofPrior radiation is a well-documented risk factor  and shouBecause of the small number of patients reported, there is no accepted gold-standard treatment. Suggested treatment options include chemotherapy, radiotherapy, and radical surgery, with some sort of combinations , 8.


A 71-year-old woman was referred to our institution for further investigation of epigastric pain. The patient had been detected to have a multilocular cyst in the medial segment of the liver measuring 69 mm in diameter at another hospital 2 years ago, and the diameter of the cyst had increased to 90 mm. Although the cyst had gradually increased in size, there was no evidence of mural nodules. As we were concerned about the malignant potential of the lesion, a left hepatic segmentectomy was performed. Pathologically, the cyst was lined by columnar and cuboidal epithelium with low-grade atypia. The epithelium covered an ovarian-like stroma, and the diagnosis was mucinous cystic neoplasm of the liver (MCN-L) with low-grade intraepithelial neoplasia. MCN-L is a rare disease and its characteristics are still poorly understood. MCN-L occurs at a lower frequency as compared to the counterpart of MCN of the pancreas, further investigations are necessary to clarify the biological malignancy of MCN-L. Previously, mucinous cystic neoplasm of the liver (MCN-L) had been classified as biliary cystadenoma or biliary cystadenocarcinoma. However, the World Health Organization (WHO) classification of 2010 defined MCN-L as a counterpart of MCN of the pancreas (MCN-P) [A 71-year-old woman visited a local hospital with the chief complaint of epigastric pain. Abdominal computed tomography (CT) revealed a large cystic mass in the liver measuring 69 mm in size, and a wait-and-watch approach was adopted. After 2 years of follow-up, the CA19-9 level, which was within normal range at first, increased to 188.5 U/ml, and CT revealed an increase in the diameter of the cystic lesion to 91 mm, and the patient was referred to our institution. At the time of her first visit to our institution, most of the laboratory data were within normal limits, and the serum CA19-9 level had increased further to 351.6 U/ml. Abdominal ultrasonography revealed a multilocular cystic lesion with a septum in the liver, measuring 110 mm in the largest diameter. CT revealed a multilocular cyst with a cyst-in-cyst appearance in the medial segment of the liver (Figure Based on the findings that the appearance of cyst is multiloculated, there is no communication with the large bile ducts, no bile duct dilatation, and no papillary lesion in the bile ducts, the patient was diagnosed clinically as having MCN-L. There was no evidence of malignancy; however, the cystic mass had gradually increased in size and become symptomatic. Malignant transformation could not be ruled out; therefore, we performed left segmentectomy of the liver. The cystic mass was in contact with the hilar plate, but could be separated easily.The cyst contents showed enhancement following postoperative injection of contrast medium into the bile duct of segment 4 Figure a. The cyPathologically, the cyst was lined by columnar and cuboidal epithelium with low-grade atypia Figure a. The epThe postoperative course was uneventful, and the patient was discharged from our institution on postoperative day 10. At present, 6 months since the surgery, the patient remains alive with no evidence of recurrence.MCN-L, defined as a cyst-forming epithelial neoplasm of the liver, is a rare entity and is reported to account for <5% of all liver cysts ,2. TypicMCN-L is defined as the pancreatic counterpart MCN-P. Shiono et al.  reportedWe have experienced a total of four resected cases of MCN-L, including the present case, from our institution. So far, 76 cases of MCN-L, defined as a cyst-forming epithelial neoplasm with ovarian-like stroma, have been reported in all ,6,7. TheComparison of the clinicopathological features between 156 cases of MCN-P, reported by the Japan Pancreas Society , and 80 The definition of MCN-L as a pancreatic counterpart MCN-P appears to be appropriate, because of the similarity of the clinicopathological features between MCN-L and MCN-P. Both diseases have a good prognosis. However, MCN-L is a rare disease and occurs at a lower frequency as compared to MCN-P; further investigations are necessary to clarify the biological malignancy of these tumors.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Isolated cerebral varix is a rare cerebrovascular anomaly, which is easily misdiagnosed as other brain tumors.A 59-year-old female patient with noncontributory medical history presented with headache and insomnia for the last 2 months. Upon admission, her neurological examination was unremarkable. Magnetic resonance imaging revealed a well-demarcated extra medullary mass, 11 × 11 mm in size, within the subdural space at the right frontal lobe. The lesion was initially interpreted as a convexity meningioma. After conducting a craniotomy on the patient, an extra-axial varix was exposed and resected subsequently. The patient's headache was resolved soon after surgery and charged without neurologic sequelae.Extra-axial isolated cerebral varix is mimicking convexity meningioma on MR images and should be considered as a differential diagnosis. The focal erosion in the inner table of the skull could be an important character of extra-axial isolated cerebral varix. An extremely round shape and smooth contour of the lesion was another important character. Isolated cerebral varix is rare vascular lesion that is treated surgically in the case of rupture or compression of adjacent structures. The information obtained with noninvasive imaging techniques should include CTA to make a clinical decision. Some cases, the isolated cerebral varix, could be described as incidental finding without any neurological symptoms. Due to its minor clinical manifestations, it is easily misinterpreted as cystic or tumoral lesion, which makes its diagnosis challenging.Cerebral varices are mostly associated with Galen aneurysms. In other cases, they are observed in arteriovenous malformation 2Ethical approval was given by the medical ethics committee of The Second Xiangya Hospital of Central South University. The patient signed the necessary documents to consent to the use of his data for teaching and publication.3A 59-year-old female without relevant medical history complained of 2 months of right, unilateral, intermittent headache, associated with insomnia that improved with nothing. A detailed neurological examination was performed and revealed no deficit. A plain and enhanced MR scan was performed and revealed a well-defined extra-axial round lesion with clear border located adjacent to the right frontal convexity Fig. A. The abA craniotomy was operated on the patient. At surgery, a 11-mm spindle-shaped, pliable structure was found. The structure, which pressed on the surrounding cortex, dura and the inner table of skull, was observed with filling Fig. A and loo44.1 Most of them are clinic silent and commonly found incidentally. Among the reported symptomatic cases, hemorrhage, thrombosis, seizure, and mass effect are the most common manifestations. Histological investigation showed that no signs of thickened or fibrotic intima were observed in the varix. Roda et al reported a case of a cerebrovascular varix located in the right lateral ventricle. The patient suffered intraventricular and subarachnoid hemorrhages caused by the isolated cerebral varix.Isolated cerebral varices are described as a focal dilatation of a single vein, involving no neural tissue and other vessels anomaly. Furthermore, as for other brain tumors, a large varix may produce symptoms from compression of adjacent structures. Inoue et al first reported a case of trigeminal neuralgia caused by compression from an isolated varix. Patient's plainness was removed immediately after the surgeon transposed the varix with its parent vein from the nerve. Isolated varix in brain lack clinical symptomatic specificities. Physicians need supplementary examinations to provide diagnosis clues and make the clinic decisions.Blood in isolate cerebral varices are prone to form turbulence and thrombus. Kondo et al introduced an epileptic patient caused by thrombosis of varix. The patient was well diagnosed as isolated cerebral varix and recommended regular follow-up. By 2 months later, she developed seizure of her left arm. The following CT scan and angiography demonstrated an acute thrombosis of the varix.4.2 Shibata et al reported a varix with a thin circular enhancement postcontrast in CT images. Tanohata et al, Dietrich and Forsting , and Hoell et al reported skull erosion adjacent to varices in their cases. In our case, we also find compression erosion in the inner table of skull. MR scan described isolated cerebral varices as extra-axial, cystic, and well-circumscribed lesions that are isointense with gray matter on noncontrast MR images and exhibit homogeneous-marked enhancement on contrast MR images. Sometimes 2 ends of isolate cerebral varix shows significant enhancement on contrast MR images, mimicking the “dura tail sign”. The lesion in CT or MR images is always well demarcated and spindle-shaped with clear border and smooth contours. A flow void phenomenon in MRI is expected, but not necessary. MR venography (MRV) and digital subtraction angiography (DSA) are recommended for they can reveal detailed venous anatomy of the varix, which are helpful to make conclusive diagnosis. However, it may be flow void phenomenon and angiographically negative for thrombosed varices.,15 At this time, the hyperintensity signal on both T1-WI and T2-WI images would be observed.Symptomatic patients may first receive a CT scan. CT images reveal isolated cerebral varix as isodense lesion precontrast and homogeneous-marked enhancement lesion postcontrast.4.3 Two features may be helpful in differential diagnosis. One of the features is the detection of focal erosion of the skull on CT images, which seems to be a helpful pattern to distinguish the varix from a meningioma that usually goes with skull hypertrophy. On the other hand, when lesion with extremely round shape and smooth contour was presented on CT or MR images, a vascular origin lesion should be considered. Patients may be free from unnecessary craniotomy for correct diagnosis. For patients whose preliminary diagnosis is convexity meningioma, if their CT/MRI images possessed either feature above, brain vessel MRV was recommended to exclude isolated cerebral varix.Isolated cerebral varices can be classified into 2 categories according to their locations: extra-axial and intra-axial. Isolated cerebral varix is easily misdiagnosed as other brain tumors, especially for patients without the presence of a subarachnoid hemorrhage or intracerebral hematoma. Extra-axial isolated cerebral varix mimics convexity meningioma on MR images. They are similar in several aspects, including the MR signal intensity, enhancement characteristics, and even the “dura tail sign,” which easily lead to imaging misdiagnosis. described a cystic mass within the deep left temporal lobe. Hyperintense was showed on both T1-WI and T2-WI images companied with hypointense on hemosiderin rim. This observation was initially suspected to be a neoplasm, such as cystic astrocytoma, abscess, or thrombosed aneurysm and was finally confirmed to be an intra-axial isolated cerebral varix by operation and pathology. Mouthuy et al provided a case of an isolated cerebral varix stimulating a cavernous angioma. In their case, the contrast CT scan revealed a lesion located in the left occipital lobe with circular enhancement. The MR appearance was extremely similar to Kelly's case. DSA is recommended to distinguish isolated cerebral varix from other common vascular anomalies.Intra-axial isolated cerebral varices are completely surrounded by parenchyma. The CT and MR appearance may stimulate other vascular anomalies. Kelly et al4.4For symptomatic patients whose complaints are associated with isolated cerebral varices, resection is recommended. To evaluate the feasibility of the resection, a DSA evaluation of the draining area of the feeding vein preoperative and a temporary blocking of the feeding vessel for 15 minutes intraoperative should be done before excision.,16 Reasons could be the balance between slight clinical manifestation and the high risk of craniotomy under general anesthesia. However, during outpatient observation period, hemorrhage, thrombosis, and seizures may develop, which may be potentially life threatening. In Kondo et al's case, the patient developed acute thrombosis of the varix during outpatient observation. In our opinion, neurosurgeons should be more aggressive about these varices. Different strategies between the nonsymptomatic extra-axial and intra-axial isolated cerebral varices should be taken. For nonsymptomatic extra-axial isolated cerebral varices, their feeding veins are superficial and the surgery is relative simple. Considered potentially life-threatening complications, we suggest early surgical interventions. For intra-axial isolated cerebral varices, the location and size of the lesion, patient's age, and other factors should be taken into account to decide whether surgical intervention or not.However, in most cases, the cerebral varices remain clinically silent and commonly found incidentally. For these varices, medical approaches are quite conflicting. Some clinicians recommended close outpatient observation and regular follow-up.5Isolated cerebral varix is extremely rare and can be classified into extra-axial isolated cerebral varix and intra-axial isolated cerebral varix. Extra-axial isolated cerebral varix has similar location and MR appearance with convexity meningioma, which is easily misdiagnosed. MR angiography and DSA are helpful in differential diagnosis. There is no unified view on treatment of these venous anomalies. Hemorrhage, thrombosis, and seizures may develop during outpatient observation period, which may be potentially life threatening. Surgical approaches are feasible after preoperative and intraoperative evaluation. Early resection of symptomatic extra-axial isolated cerebral varix is beneficial.


Dear SirDuodenal atresia usually presents with bilious vomiting. Presentation of duodenal atresia with non-bilious emesis can simulate gastric outlet obstruction [1-5]. Herein, we highlight technical aspects of identification of windsock anomaly of the duodenum that often deceives the operating team. A 5-day-old female neonate presented with persistent non-bilious projectile vomiting since birth. Baby girl was born at term by normal vaginal delivery. Antenatal scans at 12 and 20 weeks of pregnancy were reported as normal. Mother had polyhydramnios in late pregnancy. Baby was discharged home the same day of birth. The baby however developed non-bilious projectile emesis on the same day after feeding attempts. The baby was initially treated as a case of mucous gastritis and gastro-oesophageal reflux without any improvement. Laboratory tests showed metabolic alkalosis and acute renal failure. Capillary blood gas showed Ph 7.56, base excess -22 and HCO3 was 44 mmol/L, potassium 3 mmol/L and chloride 85 mmol/L. Plain x-ray abdomen showed gas in the stomach only . She was resuscitated, started on antibiotics and transferred to us. On arrival, baby was dehydrated but well perfused. There was visible left to right peristalsis in the epigastric region and the rest of the abdomen was scaphoid and soft. Pyloric tumour was not palpable during test feed. Complete blood count was normal. Biochemical profile showed sodium 133 mmol/L, potassium 3.6 mmol/L, urea 11.9 mmol/L, creatinine 193 mmol/L, total bilirubin 287 mmol/L with conjugated fraction being 12. Her renal functions gradually improved with adequate hydration.Ultrasound scan of the cranium was normal. Abdominal ultrasound scan showed fluid filled distended first part of duodenum and collapsed distal bowel loops with an appearance of luminal structure like a diaphragm in the lumen of the duodenum. She underwent upper gastrointestinal contrast study and the pilot film showed double bubble appearance . There was severe gastro-oesophageal reflux up to thoracic inlet level . The stomach was hypertrophied and dilated up to first part of duodenum which was dilated . Lateral view demonstrated clear windsock deformity but it was difficult to see the rim of the attachment of the diaphragm on the duodenal wall .At exploration, there was no discontinuity of the duodenum externally and there was no visible or palpable rim of the attachment of the diaphragm externally. The first and second parts of duodenum were uniformly dilated and it was difficult to determine the exact site of the obstruction. A wide bore orogastric tube was passed into the stomach and pushed tenting at the windsock deformity. This led to demonstration of a faint indentation at the junction of the first and second part of duodenum.A longitudinal incision centered on the indentation was made on the anterior duodenal wall and a complete mucosal duodenal diaphragm without any hole was identified. A distal windsock deformity was clearly evident .Initially a small central hole was made; to see both sides of the diaphragm and the site of attachment to the duodenal wall especially to safeguard it from ampullary area in the diaphragm; followed by complete resection leaving a small thin rim close to the duodenal wall more on the medial side than the lateral aspect . The duodenal incision was closed transversely after advancing a jejunal feeding tube distally . Her post operative period was uneventful. Nasojejunal feeds were started on second postoperative day which were tolerated well. The gastro-oesophageal reflux was severe and she failed to gain weight initially and needed anti-reflux medications. She was discharged home on anti-reflux medications at 2 weeks of age. At 8-month follow-up, she was free of any reflux symptoms. The anti-reflux medications were stopped. She is now 15 months of age, asymptomatic and thriving well.Type I duodenal atresia is an early embryonic event and should have been detected during 12 or 20th week antenatal scans. Polyhydramnios is associated with underlying congenital anomalies in over 50% of cases. Late pregnancy scan for polyhydramnios should have actively looked for evidence of bowel obstruction to rule out an organic cause. Although there is time pressure on discharge of mother and baby from the delivery suite after normal delivery, such cases should be observed till they tolerate feeds and pass meconium. Duodenal development and solid cord status is an early event in the embryogenesis and hence passage of meconium at birth is exceptional and invariably a very good clinical indicator of the underlying obstruction [2]. Congenital duodenal obstructions present with bilious vomiting and are relatively uncommon to be proximal to the ampulla of Vater, the most common site being just at the ampulla. In case of preampullary diaphragm, the presentation must be with non-bilious emesis as found in our case [3]. Plain radiographs demonstrate the double-bubble appearance with no distal gas. Upper gastrointestinal contrast studies may delineate the windsock deformity, but may not be able to show the site of attachment of the diaphragm. Sometimes there is an indentation externally to mark the site of attachment of the diaphragm or palpable scar tissue of the rim or annular pancreas around the site of stenosis. Passing a Fogarty or Foley catheter beyond the diaphragm and inflating balloon and withdrawing back may be helpful in the windsock deformity with a hole in the membrane but that option is not available in case of complete diaphragm. Endoscopic correction of incomplete duodenal diaphragm with or without windsock deformity is possible but in a complete diaphragm, it may be counterproductive and lead to serious complications [4]. Laparoscopy may have the same difficulties as it is difficult to palpate the rim and the soft indentation may require guiding the firm tube to enter in to the duodenum and proceed forward rather than curl back [5]. Duodenoplasty and diaphragm excision allowed minimal invasion and the trans-duodenoplasty feeding tube permitted early tube feeds to be started reducing the need for total parenteral nutrition.In conclusion, we believe that complete pre-ampullary duodenal membrane with windsock deformity could be a diagnostic and therapeutic challenge. Even in presence of polyhydramnios and modern prenatal screening programmes in the best centers, the prenatal diagnosis can be missed. Source of Support: NilConflict of Interest: None


The large skin defect was closed by two gluteus flaps. The rectum wall was reinserted in the remnant of the musculus sphincter ani externus. Postoperatively, parts of the flaps developed necrosis. Therefore, a vacuum sealing therapy was initiated. Subsequently, the remaining skin defects were closed by autologous skin transplantation. Six months later the colostomy could be reversed. To date, one year after first surgery, the patient has still a normal sphincter function and no recurrence of the condylomata acuminata. This case report demonstrates how giant condylomata acuminata can be successfully treated by extended surgical procedures including colostomy and plastic reconstruction of resulting defects upon resection. A condylomata acuminata infection is caused by human papillomaviridae (HPV). This sexually transmitted condition most often affects the perineal region. Importantly, infections with types 16 and 18 are associated with an increased risk for anal and cervix cancer. In most cases topical therapy is sufficient for successfully treating condylomata acuminata. Here, we report the case of a 51-year old patient who suffered from a giant perianal located condylomata acuminata which had developed over a period of more than 10 years. Imaging by MRI revealed a possible infiltration of the musculus sphincter ani externus. Because a topical treatment or a radiotherapy was considered unfeasible, a surgical treatment was the only therapeutic option in this unusual case. First, a colostomy was performed and subsequently a resection of the tumor  Condylomata acuminata is a skin manifestation caused by an infection with human papilloma viruses (HPV), in most cases by the subtypes 6, 11, 16, and 18 . Before We report the case of a 51-year-old patient who had been suffering from a perianal condylomata acuminata for more than 10 years. Anamnestically, the lesions had massively increased during the last decade. in toto including the resection of the external parts of the musculus sphincter ani externus (Figure 2 At time of presentation in our clinic the size of the condylomata acuminata amounted 16 x 12 cm (Figure 1A Figure 2 . The mucFigure 2 . BecauseFigure 2 . PostopeFigure 2 .Perianal giant condylomata acuminata is a very rare clinical condition . Due to The authors declare that they have no competing interests.


We report a case of 24-year-old male presenting with painless penoscrotal swelling for 3 years. On examination, a large soft bag of worm-like, superficial, nonpulsatile swelling was present in scrotum and penis. Color Doppler showed dilated tortuous vessels and on angiography no connections to corpora or vessels were seen. So a diagnosis of hemangioma was made and a surgical excision was carried out by circumcoronal and scrotal incisions. Postop course was uneventful. At 6 months of follow-up, no recurrence was seen and wound had healed with excellent cosmetic appearance. Cavernous hemangiomas involving genitalia are a rare clinical entity . GenitalA 24-year-old male presented with history of progressively increasing penoscrotal swelling for 3 years. It was associated with dull aching pain and discomfort. There was no history of trauma or previous surgery. No significant family history was present. On examination, a bosselated, nontender, nonpulsatile, noncompressible, soft bag of worm-like mass was felt on glans and the penile shaft circumferentially, also extending into the scrotum . No othePatient was given spinal anesthesia and catheterized preoperatively. A circumcoronal circumferential incision which is 1 cm from coronal sulcus was made and dissection was carried deep up to bucks fascia and the hemangiomatous tissue was carefully dissected from buck's fascia and skin of penile shaft . Then thAt 1-month follow-up no recurrence was seen and wound had healed well Figures . At 6-moOur case of a large cavernous hemangioma of penis and scrotum represents a very rare clinical entity. Many investigators consider them to be congenital vascular anomaly and a benign tumor . Some coSince the first report by Boullay in 1851, very few cases have been reported so far . The hemImaging studies are useful to identify and delineate the extent of the hemangioma, as well as detection of any associated anomalies. Color Doppler demonstrates blood flow within these lesions but the absence of flow does not rule out the presence of these lesions. Other imaging modalities like computed tomography (CT scan) and magnetic resonance imaging (MRI) are very useful for diagnosis and delineating their relationship with adjacent structures .2 laser, Nd: YAG laser, and yellow-light laser) and intralesional sclerotherapy  have been used primarily for smaller glanular lesion with satisfactory outcomes [Treatment decisions have to take into consideration the location of the lesion, size of lesion, cosmetic outcome, and cost of treatment. Among the various therapeutic options, nonsurgical treatments like laser (COoutcomes , 14. Dueoutcomes  also repHemal et al.  demonstrEarlier surgical excision was recommended for all lesions due to risk of traumatic rupture and bleeding but the risk is very low , 19. CosIn our case, we decided to operate on the patient due to the large size and extent of the hemangioma and an excellent surgical outcome was achieved due to meticulous dissection and hemostasis. Thus surgical excision remains a good option for large and multiple lesions and results are quite satisfactory with good surgical planning and technique.Cavernous hemangioma involving penis and scrotum is very rare and it poses both diagnostic and therapeutic challenges for treating physician. Surgical excision is preferable for large and multiple lesions with satisfactory outcomes.


Synovial chondromatosis, also termed synovial osteochondromatosis, is a rare benign disorder characterized by the presence of cartilaginous nodules in the synovium of the joints, tendon sheaths, and bursae. It most commonly involves large joints, such as the knee, hip, and shoulder, but its presence in smaller joints has also been reported. Nevertheless, ankle involvement is unusual. The diagnosis is commonly made following a thorough history, clinical, physical, and radiographic examination. We report a case of a young patient with primary synovial chondromatosis of the ankle joint and present the clinical, radiographic, and intraoperative findings. Synovial chondromatosis is a rare benign condition characterized by the formation of intra-articular cartilaginous nodules in the synovium of joints . These nSynovial chondromatosis originating from the ankle is an exceedingly rare condition , 7. HereA 21-year-old male patient presented to our clinic with 18-month history of pain in his left ankle which had deteriorated over the last year. He complained of stiffness, crepitation, and a catching sensation in the ankle; he felt as if he was “walking on pebbles.” Physical examination revealed a mildly tender swelling on the anterolateral aspect of the ankle joint. The patient was able to dorsiflex the ankle up to 10°, while plantar flexion was possible up to 20°. Anterior positive impingement was noted, while no instability was detected. There were no vascular or neurological abnormalities in the ankle and laboratory studies were within the normal range.Plain radiographs of the left ankle revealed a large speculated calcified mass of loose bodies emerging from the anterior aspect of the ankle . As therSynovial chondromatosis is a rare benign synovial lesion characterized by multiple pearl-like intra- and extra-articular osteochondral loose bodies , 8. InvoIt is broadly concurred that the exact etiology of synovial chondromatosis is unknown. Milgram et al. classified the disease process into three distinct stages , 13. DurClinically, patients with synovial chondromatosis usually present with pain, swelling, stiffness, and/or a detectable mass, and most of them have a long clinical history before an accurate diagnosis is made . There hThe goal for the treatment of synovial chondromatosis is to remove the loose bodies, improve pain symptoms, regain movement in the joint, and limit the development of early osteoarthritis . The treWe report a case of primary diffuse synovial chondromatosis of the ankle. This unusual, distinctive benign synovial neoplasm presents readily recognizable clinical, radiographic, and pathological features. Primary synovial chondromatosis is adequately treated by loose body resection either arthroscopically or through open surgery. Prognosis is excellent.


Primary synovial sarcoma of the prostate is an uncommon malignant tumor. There are few cases reported in the English medical literature to date. Here, we present a case of 22-year-old man with primary synovial sarcoma of the prostate metastatic to the liver and lung. To our knowledge, only six reports of synovial sarcoma involving the prostate have been previously published. We also reviewed the previous treatments and prognoses in previous case reports and evaluate the proper treatment for this disease. Primary synovial sarcomas have been described at unusual sites, including the heart, oesophagus, larynx, pleura, lung, kidney, prostate, liver, abdominal wall, retroperitoneum and gastrointestinal tract; involvement of the genitourinary tract is exceedingly rare -3. SynovA 22-year-old man presented elsewhere with lower urinary tract symptoms and was referred to us for further evaluation because of urinary retention. No particular family history or occupational hazard was elicited. Our patient complained of dysuria, urinary frequency and nocturia five to ten times per night before urinary retention. On digital rectal examination, a large prostatic mass was palpable, and the surface was smooth. His serum prostate-specific antigen was 1.2 ng/ml normal 4 or less). Pelvic computerized tomography (CT) imaging Figure . The pat or less.Synovial sarcoma is a clinically and histomorphologically well-defined soft tissue tumor that is extremely uncommon, with about 90% synovial sarcomas occurring on the extremities. In most patients with this disease, distant pulmonary metastases develop after primary tumor surgery. In the urinary system, most synovial sarcomas have been reported in the kidney . PrimaryPrimary prostatic sarcomas are rare tumors and most patients present with urinary obstruction, as did our patient. The pathologic findings were consistent with the diagnosis of synovial sarcoma arising from the prostate. In common with other synovial sarcomas, the immunohistochemical staining demonstrated some typical findings of synovial sarcoma: the tumor cells were positive for vimentin and CD99, but negative for CD34, Bcl-2, alpha smooth muscle actin, desmin, and S-100 protein. Finally, the presence of SYT-SSX fusion transcript confirmed the diagnosis ,8.Several clinicopathologic features were particular for primary synovial sarcoma of the prostate. As compared with the most common malignant tumor of the prostate adenocarcinoma, prostatic sarcoma occurs in relatively younger patients . AccordiThe survival of persons with primary prostatic sarcomas is poor, with most patients dying within a few months after diagnosis and surgery. Distant pulmonary metastases are found in most patients with this disease, whether the patient has undergone surgery or not. In our case, the tumor had metastasis to the liver and lung. Treatment for this sarcoma is uncertain due to extremely limited experience but it appears that aggressive resection has to be considered for the treatment of locally confined synovial sarcoma . The rolIn conclusion, we demonstrate a case of primary prostatic synovial sarcoma. Due to the paucity of cases of synovial sarcoma involving the prostate, protocols for definitive treatment are not well defined. Treatment for this tumor is uncertain due to extremely limited experience, but it appears that aggressive resection should be considered as the standard therapy. The role of adjuvant chemotherapy and radiation also remains unknown. Reporting of future cases is necessary to establish appropriate therapeutic guidelines for this disease.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.CT: computerized tomography; MRI: magnetic resonance imaging; RT-PCR: reverse transcription-polymerase chain reaction; DNA: Desoxvribose Nucleic Acid.The authors declare that they have no competing interests.QZ, LR, HW, XQ, FL and DZ participated in the admission and the care of this patient. All the authors participated in the conception, the design, data collection and interpretation, manuscript preparation and literature search. All authors have read and approved the final manuscript.


A 29-year-old woman with bipolar disorder presented to the plastic surgery clinic with a gradually growing mass of the lower lip. The patient reported that the lesion was irritating and she frequently found herself biting it. It had waxed and waned in size over several years.What is the differential diagnosis for a growing lip mass?What is the pathogenesis and classification of oral mucoceles?What are the management options?What is the risk of recurrence after removal?There is a broad differential diagnosis for a growing mass of the lower lip. The 2 most common lesions are traumatic fibromas, or focal fibrous hyperplasia, and mucoceles.5An oral mucocele is classified as either extravasation or retention type, depending on the pathogenesis. Extravasation type is due to mechanical trauma to the excretory duct of the salivary glands, leading to transection or rupture of the duct. Mucus extravasates into the connective tissue stroma, triggering an inflammatory response that leads to the formation of a fibrous pseudocapsule. This type is typically seen in the lower lip, buccal mucosa, and retromolar area. The retention type is due to obstruction of the duct and subsequent buildup of fluid. This type is less common overall and occurs mostly in the elderly.2 laser with good outcomes.The typical management of oral mucoceles is surgical. Small lesions can be completely excised, along with associated salivary gland tissue, and closed primarily. Larger mucoceles can be treated with marsupialization, since large excisions can carry a risk for dissection of vital structures . Micro-marsupialization is a variation of this technique that is particularly useful for pediatric patients because it is the least traumatic option. It involves incising a cyst and suturing the edges of the subsequent slit along its longest diameter.,6,The likelihood of recurrence after excision is partially dependent on technique and has been reported to occur in 5% to 18% of cases.3In our patient, we were able to make an incision directly over the lesion and bluntly dissect the mass out with the psuedocapsule intact along with the associated salivary gland see . After 2


Objective. Endolymphatic sac tumor (ELST) is a rare neoplasm arising from the intrapetrous portion of the endolymphatic sac, either isolated or in association with the von Hippel-Lindau disease. We report a sporadic case of ELST with an overview of the literature and a discussion of clinic-radiological, histopathologic, and surgical findings. Case Report. A young woman presented with a progressive hearing loss in the left ear. Otoscopy showed a reddish, bleeding hypotympanic mass. CT demonstrated an expansile lytic mastoid lesion extending to the middle ear, with bone erosion. MRI confirmed a lesion of increased signal on T1-weighted sequences. The patient underwent a canal wall-down tympanoplasty with complete removal of the tumor. Histopathology was consistent with a papillary ELST. Immunohistochemistry was positive for cytokeratin and chromogranin A. Conclusion. This paper highlights the rarity of ELST, the need for an accurate neuroradiological and immunohistochemical study at the early stages, and the timeliness of surgical treatment. Endolymphatic sac tumors (ELSTs) are very rare nonmetastasizing neuroectodermal tumors arising from the proximal, intrapetrous portion of the endolymphatic sac. The first report concerning the ELST was in 1984 .Since this time, a number of articles have reported on these tumors, along with their association with the von Hippel-Lindau (VHL) disease –7. The rWe report a sporadic case of a young woman with an ELST not vHL related, underlining the difficult differential diagnosis and emphasizing the role of immunohistochemical study and the surgery as the treatment of choice.A 37-year-old woman presented with a 3-month history of fullness and progressive hearing loss in the left ear. There was no tinnitus, otalgia, otorrhea, vertigo, or facial nerve paralysis. Otomicroscopy of the left ear showed the presence of a reddish, easily bleeding hypotympanic mass, extending to the medial third of the external auditory canal. Examination of the facial nerves, nasopharynx, oral cavity, larynx, and neck was normal. Neither the symptoms nor a family history of VHL disease was found in the patient. The pure-tone audiometry revealed a moderate, pantonal, conductive hearing loss in the left ear . ComputeELST is a primary neoplasm of the temporal bone; till now, less than 100 cases have been reported in literature. Most of them are sporadic, but 30% are associated with the VHL disease . Mean agThe endolymphatic sac is situated between the petrous periosteum and the dura mater of the posterior cranial fossa, posterior to the internal auditory canal. Tumors growing there slowly infiltrate and erode the petrous bone. Bambakidis et al. propose a grading system for ELSTs based on radiographic location and extent . In pracCT of these tumors is characterized by bony erosion centered on the posteromedial aspect of the petrous bone, although small tumors may show bony abnormalities other than widening of the vestibular aqueduct. Most cases demonstrate a thin calcified peripheral rim representing expanded cortex of the petrous bone. CT can be used to demonstrate normal architecture of the jugular foramen, thus, differentiating the mass from a jugular glomus or other jugulotympanic mass. In addition, CT will show typical sparing of the cochlea and middle ear , 23.On MR imaging, the ELSTs usually have a heterogeneous appearance. Multiple high-signal intensity foci on both T1- and T2-weighted images indicate the presence of blood-, methemoglobin-, or proteinaceous-filled cysts or cholesterol clefts. Such blood products are rare in other temporal bone lesions. Scattered low-signal foci represent hemosiderin deposition due to chronic parenchymal bleeding. Calcifications and vessels can demonstrate signal voids. The addition of contrast results in a nonhomogenous enhancement of the tumor's solid portions .ELSTs are papillary adenocarcinomas of low histologic grade. In the past, they have been confused with middle ear carcinomas and ceruminous gland tumors. Initial difficulties in the differentiation of ELSTs from choroid plexus papilloma were aided by the recognition of transthyretin as a site-specific marker for choroid plexus epithelium , 24. ManIn this case report the positivity for chromogranin A is a rare but interesting remark. The reactivity of the epithelial cells with antibodies against neuron specific enolase, chromogranin, and somatostatin, respectively, demonstrates a paracrine activity of the endolymphatic sac . In 1995The differential diagnosis of ELSTs includes other deep temporal bone lesions. Jugular glomus and jugulotympanic paragangliomas involve the jugular foramen and then the middle ear rather than retrolabyrinthine temporal bone. Irregular bone destruction by paragangliomas is similar to ELSTs, as are flow voids, marked enhancement, and a predominant external carotid artery supply. Bone spicules in the tumor matrix and high-signal hemorrhagic foci in paragangliomas are unusual. Hypervascular metastases such as renal cell or papillary thyroid carcinoma may also produce a destructive deep temporal bone lesion. They lack internal hemorrhagic foci on MRI examination. Schwannomas of the jugular foramen are well defined and centered in the jugular foramen, not in a retrolabyrinthine location. In the case of petrous apex granuloma, high signal is present on unenhanced T1-weighted MRI examination, but petrous apex granuloma is distinguished from ELSTs by its characteristic location .Concerning treatment, radical surgical local excision remains the mainstay of current therapy. In our case the timeliness of surgical treatment with a canal-down tympanoplasty allowed a complete removal of the lesion with a good long-term follow-up. The surgery may sometimes necessitate sacrifice of cranial nerves, and sometimes total resection of the advanced tumors may be impossible due to the anatomic complexity , 29, so The ELST shows quite a large variation in tumor extension (grade), hearing level, and comorbidity. Consequently, in our opinion, treatment must be individualized. Although surgery might be attractive as the first treatment option, radiotherapy and wait-and-scan are an alternative especially when hearing is still intact.


We report a case of a 32- year -old woman with multiple miliary peritoneal deposits mimicking advanced ovarian carcinoma.Peritoneal tuberculosis accounts 1-2% of all forms of tuberculosis. Peritoneal tuberculosis is an important differential diagnosis for ovarian cancer in women with ascites, adnexal mass and elevated cancer antigen 125 (CAA 32-year-old drug addicted woman presented with menometrorrhagia, fever and shivering, ascites and pelvis mass. Ultrasonography revealed a 53×65 mm cyst in left ovary and ascites. Multiple miliary peritoneal deposits were observed during laparatomy without any mass, histologic examination confirmed tuberculosis of uterus, tubes, omentum, liver and external surfaces of small intestine. Finally, the patient recovered with anti-tuberculosis treatment.125 even with negative cytology and bacteriology test results. These findings highlight considering tuberculosis in the differential diagnosis of any patients with adnexal mass, ascitis and elevated serum CA Pain was localized in the hypogastrium area for 3 months. The patient had menometrorrhagia, fever and shivering, and development ascites and diagnosis of pelvic mass required hospitalization.A 32-year-old-woman G She has been addicted to opium for 5 years and also used methadone. Her vital signs were stable and her lung sounds were clear but sometimes she had a fever of about 38. There was a 53×65 mm cyst in left ovary region in ultrasonography but the ovaries, uterus, liver, kidney and spleen were normal and ascites fluid was reported.isoniazid, pyrazinamide, ethambutol hcl and rifampin.An ovarian malignant mass was reported in spiral CT scan  with contrast. The patient underwent laparotomic surgery in which her uterine tubes were swollen with adhesion in both ovaries with multiple military peritoneal deposits. No mass has been observed in the abdomen and pelvis. The results of histologic examination demonstrated abdominal tuberculosis on her uterus, tubes, omentum, liver and intestinal surface. The results of laboratory test are shown in 125 can be considered as a marker for the evaluation of treatment response. This patient shows rapid decline in CA125 level paralleling clinical response to antituberculosis (Abdominal tuberculosis with series of clinical signs may mimic ovarian cancer . This ofrculosis . For prerculosis . High le125 may suggest the possibility of tuberculosis. Abnormal menstruation, pain, mass and distension of abdomen were the usual signs of 26 patients with ovarian malignancy diagnosed in a study in India that confirmed abdominal tuberculosis after laparoscopy and histopathology test . In addi125 even with negative cytology and bacteriological test results. The use of imaging techniques and laparoscopy or finally laparotomy is recommended. In endemic areas, physicians should consider tuberculosis in the differential diagnosis of any case with unusual manifestations.In conclusion, abdominal tuberculosis should be considered in the differential diagnosis of women with adnexal mass, ascites and elevated CA


Solitary fibrous tumor of the pelvic is an uncommon neoplasm with nonspecific symptoms. Reports of malignant transformation are especially rare. We report a case of solitary fibrous tumor in pelvic. A unique feature of our case compared with previously reported is that this patient relapsed with malignant transformation and had significant response to radiotherapy. The patient was initially treated with surgery, followed by postoperative dimensional conformal intensity modulated radiation therapy  to provide a radical cure for residual tumor.In this case, there were no signs of recurrence after six and a half years of further follow-up, indicating that postoperation radiotherapy may be an effective treatment for SFT with malignant transformation in pelvic. The main treatment for SFT is surgery. Herein, we report an SFT patient with postoperative recurrent malignancy transformation in pelvic. He was treated by radical surgical resection and subsequently underwent intensity-modulated radiation therapy (IMRT). It may provide a useful reference for the treatment of similar cases.A 58-year-old man was admitted to our hospital due to a pelvic mass, which was found by ultrasound in a private clinic in March 2007. Ultrasound demonstrated a large irregular marginated solid mass in the pelvic cavity. Computed tomography (CT) scan revealed a mass (5.5 cm in the longest diameter) in the cavity of the pelvic, the lesions border was clear, and no obvious swelling lymph nodes in double side basin wall and inguinal region Figure A. SubseqHe accidentally hit a mass in the right lower quadrant in February 2009. On the physical examination, the mass was hard, inactive, and painful if touched it. Laboratory tests, including tumor markers , were within the normal range. Pelvic CT revealed a tumor of ∼7.5 cm in diameter, mixed density showing multiple soft tissue mass Figure A. Immunomax 65.8 Gy; Dmean 34.8 Gy), femoral head-R , rectum . After radiation therapy, no other obvious radiation reaction was observed except the slight cystitis. The patient had refused adjuvant chemotherapy and has been followed up for six and a half years. There was no clinical evidence of disease recurrence.Due to the patient with recurrent malignant transformation, then we conducted post-operative IMRT to eliminate residual tumors, and avoid recurrence and/or metastases. Three weeks after the patient underwent operation, we developed a radiation treatment plan, and the patient underwent 3-dimensional conformal IMRT. The treatment machine was VRIAN 23 EX. LUNA TM 260 was used as the Eclipse planning system. The clinical tumor volume (CTV) was visualized on CT, and it was calculated including tumor bed. However, 0.3 cm outside of the CTV was defined as the planning target volume (PTV). We carried out the total PTV irradiation with a dose 60 Gy/30F, 2 Gy/F, 1 session/day, and 5 times/week. The DVH for each organ were as follows: bladder  high cellularity, (2) high mitotic activity (>4 mitoses per 10 high-power fields), (3) pleomorphism, (4) necrosis, and (5) hemorrhagic changes. In the present study, the diagnosis of SFT was made according to the above characteristic immunohistochemical findings and histological features.According to the previous reports, CD34 is strongly positive for diagnosis of SFT,18Currently, no standard therapy has been established for SFT with malignant transformation because of the rarity. The main method of treatment for SFT remains wide local excision of the tumor. Soft-tissue sarcomas have traditionally been managed by wide excisional surgery and radiation therapy, but in a study it was concluded that overall morbidity associated with adjuvant radiation was not significantly higher than that with surgery alone.19 However, it is controversial if RT is therapeutic only for high-grade tumors or also for low-grade tumors.21 Kawamura et al22 reported a case of huge pelvic malignant SFT with lung metastasis, chemotherapy did not work whereas the tumor shrank 12 months after 50 Gy irradiation of the pelvic region. Moreover, in a series of 11 SFT treated with definitive RT without surgery, no patient had a local recurrence, and 9 were disease-free at 3 to 20 years from diagnosis.23 Furthermore, Saynak et al13 also reported RT alone achieved significant effect, which was consistent with Saynak M’s report.13 On the contrary, Dietrich et al24 indicated an accelerated progression of the solitary fibrous chest wall tumor in the course of irradiation. Whether the development of sarcomatic growth occurred as a result of RT remains speculative. Nevertheless, RT in SFT could be conceived when surgery is not suitable, as in meningeal, retroperitoneal, and pleural presentations, and pathologic signs of “malignancy” are present at the onset.The treatment for unresectable malignant SFT has not been established yet. Radiation therapy (RT) alone can be considered for patients refusing or unsuitable for surgery, and it can attain 30% to 60% rate of control in sarcoma.27 Postoperative RT was given due to the high-grade malignancy, narrow excision margins, large size and rapid growing, but there are no published series to substantiate its routine usefulness for this disease.28 Xue et al29 reported four and a half years after postoperative RT, no local recurrences were observed. Furthermore, Wushou et al30 analyzed 227 SFT cases in the Surveillance, Epidemiology, and End Results database during the years of 2000 to 2009 and confirmed a survival benefit for patients treated with surgery in combination with adjuvant RT, whereas the effect was not appreciated with surgery alone. In another series, investigators found that the treatment protocol of combined adjuvant RT with surgery seemed to hinder tumor progression, but had no effect on overall survival.31Postoperative adjuvant therapy with chemotherapy and/or RT has been used sporadically, but the benefit of these adjuvant therapies remains unproven. The literature is inconclusive in regard of adjuvant RT in SFT.22 reported that SFT had no response to paclitaxel , and the tumor had continued to increase 22.2% in size in the pelvic CT scan. Gold et al2 found that chemotherapy regimen of doxorubicin and cisplatin was also ineffective for the SFT patient. However, Grobmyer et al32 revealed that combination of doxorubicin, ifosfamide, and mesna was associated with a significant improvement in disease-specific survival in patients with high-grade extremity soft tissue sarcomas. Stacchiotti et al33 found that anthracyclines have a degree of antitumor activity in the range of soft tissue sarcoma chemotherapy, but ifosfamide monotherapy seemed to have lower activity. Furthermore, Park et al34 found that combination therapy with temozolomide and bevacizumab is a generally well tolerated and clinically beneficial regimen for SFT patients. On the contrary, Stacchiotti et al35 found that the temozolomede-bevacizumab combination did not induce a better therapeutic activity than temozolomide alone, and dacarbazine as a single agent has antitumor activity in SFT.There are no standard chemotherapeutic indications or regimens for SFT. In some previous studies, Kawamura et al37 Stacchiotti et al38 found that sunitinib is also active in SFT, and response can be long-lasting. Additionally, malignant SFT can express platelet-derived growth factor receptor beta strongly and has missense mutation of 18 exons, providing promising treatment target for unresectable malignant SFT.39Several tyrosine kinase inhibitors have been reported, which have shown promise in treating SFT. For example, some previous studies reported that sorafenib has produced tumor responses in patients with SFTs.In the present study, we conducted postoperative IMRT to eliminate residual tumors, which can better control the primary tumor and avoid recurrence and/or metastases, but the patient refused adjuvant chemotherapy again. No recurrence was observed in the subsequent follow-up, indicating that the patient could be benefited from postoperative RT and clinicians can be referenced from this strategy.Our patient was initially diagnosed as benign SFT. After radical surgery, he refused to receive the subsequent adjuvant therapy with chemotherapy and/or RT, and did not go to the hospital regularly for re-examination. Currently, he had a relapse. The diagnosis was malignant SFT. Therefore, the first treatment of SFT was clearly insufficient. Following surgery, postoperative RT and chemotherapy should be performed in order to better control the primary tumor and avoid recurrence or metastases.In this case report, we described an SFT growing in the pelvic, an infrequent localization, which presented a unique associated with recurrent malignant transformation. The absence of chemotherapy and/or RT could be the reason of relapse. This patient underwent postoperation RT after recurrence, no recurrences or metastases occurred during long-term follow-up. Thus, postoperation RT may be an effective treatment for SFT with recurrent malignant transformation in pelvic. Additionally, chemotherapy also should be performed for the greatest degree to prevent relapse. Owing to the possibility of repeated recurrences or malignant transformation, a prolonged follow-up may be advisable in this rare disease.


Traumatic neuroma is a well-known disorder involving peripheral nerves, which occurs following trauma or surgery. The lesion develops most commonly in the soft tissues of the mental foramen area, lower lip and tongue. Intra-osseous lesions arising in jawbones are very uncommon. In this paper, we report a new case of an intra-osseous traumatic neuroma, discovered incidentally on a panoramic radiograph obtained for orthodontic documentation. In addition, the case herein described developed spontaneous remission, a situation not previously reported in the literature. Finally, we discuss relevant demographic, clinical, microscopic, immunohistochemical and treatment aspects of traumatic neuromas. Key words:Amputation neuroma, traumatic neuroma, mandible, spontaneous remission. Traumatic neuroma is a well-known disorder involving peripheral nerves, which occurs following trauma or surgery . The lesTraumatic neuromas may develop in any region of the body, including the head and neck . In thisThe aim of this paper is to report a new case of an intra-osseous traumatic neuroma, discovered incidentally on a panoramic radiograph obtained for orthodontic documentation. In addition, the case herein described developed spontaneous remission, a situation not previously reported in the literature.A 22-year-old white female was referred to the Oral Medicine Service of the Araujo Jorge Hospital for evaluation of an osteolytic, mandibular lesion detected on a panoramic radiograph obtained for orthodontic documentation purposes. During anamnesis the patient reported a previous history of complicated exodontias of an unerupted left mandibular third molar two years previously. Medical and familial histories were unremarkable. Extraoral and intraoral examinations were within normal limits. Radiographic examination revealed an ill-defined, multilocular lesion on the left side of the mandible, extending from the second premolar to the mandibular ramus Fig. . No expaet al. [2009] found only 15 cases among 21,476 specimens [0.07%] diagnosed in an oral pathology service (Traumatic neuromas are uncommon lesions. In 2005, Jones and Franklin reviewed 44,007 oral and maxillofacial specimens and found 149 cases [0.34%] (Traumatic neuromas can occur at any age, but are most often diagnosed in young and middle-aged adults ,7. WomenHistopathologically, traumatic neuromas typically present as non-encapsulated lesions, containing a large amount of haphazardly arranged nerve fascicles, within a densely collagenous and fibroblastic stroma. Occasionally, scar tissue may also be seen . ImmunohThe treatment of choice is nerve sparing surgical excision. An optimal technique, with minimal manipulation and severance of nerve fiber, is essential for adequate outcome. Other second-line therapeutic options found in the literature are stereotactic radiosurgery, local infiltration of steroids, sympathetic ganglion block, percussion, and ultrasonic therapy ,3. In th