Granulomas are rarely seen in gastric biopsies mostly as an involvement of granulomatous diseases like sarcoidosis, Crohn's disease, infections, neoplasms, and vasculitis. Here, we claim cyanoacrylate as a foreign body type granuloma-causing agent in the stomach after vascular embolisation. We present cyanoacrylate associated gastric changes of three cases: two endoscopic biopsies and one gastric resection. In two cases, cyanoacrylate associated ulcers and granulomatous inflammation were observed in gastric mucosal biopsies following endoscopic examination after 7 months and 6 years of the glue injections, respectively. In the third case, the cyanoacrylate injection was performed 2 months prior to the surgery. Then the patient underwent distal pancreatectomy for pancreatic adenocarcinoma and during the operation a gastric mass was resected with a suspicion of tumoral infiltration. These three cases demonstrated that glue exposure causes active chronic inflammation with foreign body type granulomas, mucosal ulceration, and bleeding in the gastric mucosa. Even further, it can induce mass formation in the injection sites. Granulomas in the stomach can be due to several diseases, such as Crohn's disease, sarcoidosis, infections, lymphoma, adenocarcinoma, Whipple's disease, Langerhans cell histiocytosis, gastric perforation, and vasculitis . ForeignCyanoacrylate derivatives have been used as hemostatic or anastomotic agents . In the We report foreign body type giant cell reaction, inflammation, ulceration, and mass formation in the stomach caused by cyanoacrylate injections, in the context of endoscopic biopsies and gastric resections. Pathologists and clinicians should be aware of such long-term complications of “glue” injection into varices or bleeding sites and realise that this material can also form a discreet mass in the stomach wall.A 61-year-old male underwent distal pancreatectomy and splenectomy in September 2013, after 11 rounds of chemotherapy for metastatic pancreatic adenocarcinoma. A mass was detected in the stomach wall intraoperatively. This mass was considered to be a metastasis of pancreatic adenocarcinoma and was resected. The lesion had caused irregularity at the serosal surface of the stomach and was hard in consistency. The cut section of this 5 × 3 × 1.5 cm-sized gastric resection specimen revealed a poorly circumscribed white lesion, 1.5 cm in diameter, that spanned the whole gastric wall except for the mucosa, with grey-white necrotic material in its center Figures .A representative section revealed a bubbly, cystic, coarsely nodular lesion with occasional necrotic foci. Numerous foreign body type multinucleated giant cells surrounded empty-looking spaces. The outer margins of the lesion harbored a mild to moderate lymphoplasmacytic infiltrate. There were scattered hemosiderin-laden macrophages that suggested prior erythrocyte extravasation. The mucosa was intact; the lesion spanned the submucosa and destroyed the muscularis propria, reaching the serosa . Closer A 52-year-old female with a history of infertility was admitted with abdominal pain, nausea, and vomiting in December 2007. A hepatobiliary ultrasonography revealed portal vein thrombosis. Abdominal CT scan showed thrombosis in superior mesenteric and renal veins. Anticoagulant therapy was started. In August 2008, gastroesophageal varices were detected in upper gastrointestinal endoscopy. In July 2009, she presented with upper gastrointestinal bleeding. Endoscopy revealed varices in the distal esophagus and fundus of the stomach, with an overlying clot. 0,5 mL of glue was injected to esophageal varices. Afterwards the patient underwent gastrostomy and ligation. In August 2010 gastric variceal bleeding recurred and was embolised with 3% beta-blocker, alcohol, and 30% glue injection. In 2011, 2012, and 2013, follow-up endoscopies persistently displayed gastric varices and histoacryl glue injections were administered to ulcerated gastroesophageal varices.In 2015, a biopsy was taken from an ulcerated lesion in fundic and antral mucosa. On histopathologic examination, focal ulceration and exudate were seen in the mucosa. Beneath the intact part of the epithelium, there were extravasated erythrocytes . In the A 56-year-old female with chronic viral B hepatitis and cirrhosis was admitted in December 2011. Glue injection was performed to treat gastric variceal bleeding. In July 2012, the patient had an upper gastrointestinal endoscopic examination. An ulcerated lesion, 2 cm in diameter, was seen in the fundic mucosa and the edge of the ulcer was biopsied.The foveolar epithelium was intact, but beneath the epithelium there were extravasated erythrocytes and edema. A trace of foreign body giant cell reaction was detected in the lamina propria. Translucent foreign material was present in the cytoplasm of giant cells. An inflammatory reaction composed of eosinophils and lymphoid aggregates was detected Figures . The forGranulomatous gastritis can appear in a number of clinical settings: Crohn's disease, sarcoidosis, infections, lymphoma, adenocarcinoma, Whipple's disease, Langerhans cell histiocytosis, gastric perforation, and vasculitis . The conCyanoacrylate is a well-known tissue adhesive used in the treatment of varices related to portal hypertension, vascular malformations, and bleeding episodes of these lesions . It typiIn these three cases described above, none of them were diagnosed with a granuloma-causing disease during follow-up. Special stains for microorganisms were negative in all cases. There were no evidence for gastric pneumatosis in abdominal CTs. All cases had a history of glue injection to treat the bleeding. The gastric biopsies were sampled 2 months (Case 1), 7 months (Case 3), or 6 years (Case 2) after the initial vascular embolisation. All cases displayed a similar “bubbly” appearance with “slightly refractile” translucent material and surrounding chronic inflammation with the tissue findings of old and new hemorrhage.Cyanoacrylate-related findings in gastric biopsies have not been reported previously. Cyanoacrylate has the ability to harden and induce intense inflammatory reaction and fibrosis in and around the injection sites. These reactive tissue changes can be mistaken for a neoplastic mass, depending on the clinical context. The main morphological finding is to see “slightly refractile” wisps of foreign material in the empty-looking spaces with accompanying foreign body type giant cells. Ulceration and bleeding are also seen around the foreign material. The history of previous cyanoacrylate embolisation for variceal or gastric bleeding is critical for diagnosis. In association with the histopathological changes as described above, positive history for glue injection and absence of specific etiology to explain granulomatous gastritis are diagnostic for cyanoacrylate associated granulomatous gastritis.


Staphylococcus and Salmonella spp. infections. We herein report a unilateral acute scrotal abscess in a preterm infant born at 26 weeks in whom Propionibacterium acnes was isolated from the collected scrotal fluid culture. To our knowledge, this is the first case report implicating P. acnes as a causative agent of neonatal scrotal abscess. Based on such findings, P. acnes infection should be considered in differential diagnosis of acute scrotal abscess in neonates, particularly in preterm infants. Acute scrotal abscess is an extremely rare condition in neonates and may mimic testicular torsion. Most of these abscesses have reportedly been due to Staphylococcus and Salmonella spp.  and ampicillin (MIC < 2 μg/mL). Therefore, for deescalation of the antimicrobial agent, his meropenem therapy was changed to ampicillin. Further antibiotic therapy normalized CRP and WBC levels. The postoperative course was uneventful. At the time of drafting this case report, he is currently being followed up by the Divisions of NICU and Pediatric Surgery.Observed during surgery, the right tunicae vaginalis was markedly thickened and contained pus . The tes P. acnes during hospitalization and under steroid administration for LCC. As aforementioned, acute scrotal abscess is an extremely rare condition in neonates. Raveenthiran and Cenita reported nine cases of scrotal abscess, in which Staphylococcus spp. was most commonly isolated [ Salmonella enteritidis, and in their review, they reported five other cases in infants younger than 3 months due to the same pathogen [ Streptococcus, Bacteroides, E. coli, and Klebsiella spp. were also reported to cause scrotal abscess in neonates [ P. acnes. P. acnes is part of the skin and mucous membrane commensal microbial flora [To our knowledge, this is the first report of a neonatal scrotal abscess caused byisolated . Huang apathogen . As statneonates . Howeveral flora  and is mal flora , 10. P. acnes is rare. The only evidence present in the literature is a report by Yamamoto et al. that described the onset of an intrascrotal and vesicular granuloma likely sustained by P. acnes in an elderly patient [ P. acnes infection. Generally, this bacterium is sensitive to a large variety of antibiotics , highly resistant to metronidazole, and weakly sensitive to treatment with aminoglycosides [A review of the available literature indicated that intrastromal colonization of patient . In our ycosides .Conversely, managing acute scrotal swelling in neonates can be challenging, with scrotal infections often resembling testicular torsions. Neonatal scrotal abscesses often have none of the recognized signs of inflammation, and this limits the value of clinical assessment at this age . Moreove P. acnes and has gleaned insight into this rare neonatal condition. In case of swelling of newborn scrotum, it is imperative to distinguish between emergency acute scrotal abscess and testicular torsion. Therefore, neonatologists should consider such symptoms in male infants presenting with intractable scrotal abscess, albeit rare. In addition, in the case of newborn infants who are immunosuppressed by steroid administration, P. acnes should be considered as a causative agent in scrotal abscess, particularly in preterm infants.In summary, our case highlights the first premature infant to develop scrotal abscesses caused by


Hepatic artery pseudoaneurysm (HAP) incidence is rising due to more common use of endoscopic and percutaneous hepatic interventions. HAP is potentially fatal, as it could lead to sudden life-threatening hemorrhage. HAP can be intrahepatic or extrahepatic. On computed tomography angiogram (CTA) and magnetic resonance angiogram (MRA), HAP follows blood pool on multiphasic examination, with brisk arterial enhancement that washes out, similar to the abdominal aorta on later phases. We present a case of idiopathic giant HAP in an 82-year-old male. Currently, angioembolization is replacing surgery as the initial modality of choice for management of this condition. Pseudoaneurysms occur when there is a breach in the vessel wall with blood leaking through the wall but contained by the adventitia or surrounding soft tissue . VisceraThe patient is an 82-year-old male with history of atrial fibrillation, coronary artery disease, and hypertension. He was on anticoagulation. He presented to the emergency department with shortness of breath for which he underwent a CT of the chest without contrast. A large 14 cm hypodense liver mass was incidentally detected. Further evaluation with dedicated liver protocol magnetic resonance imaging (MRI) revealed a large mass in the right hepatic lobe showing intense and homogeneous arterial phase enhancement that persisted on delayed images. The mass followed the signal intensity of aorta throughout different phases of contrast enhancement. On abdominal duplex US, brisk internal color flow was seen as well as bidirectional blood flow “Ying-Yang sign” . ConfirmHAP is potentially fatal, as it could lead to sudden life-threatening hemorrhage. HAP can be intrahepatic or extrahepatic. Ultrasound demonstrates a cystic structure along the course of hepatic artery with brisk internal color flow, to-and-fro pattern and Ying-Yang sign . CTA of HAP incidence is rising. On CTA and MRA, HAP follows blood pool on multiphasic examination, with brisk arterial enhancement that washes out, similar to the abdominal aorta on later phases. Currently, angioembolization is replacing surgery as the initial modality of choice.


We present a case of two-year old boy with solitary atypical adenomatous hyperplasia (AAH) in extralobar sequestration (ELS), which was misdiagnosed as diaphragmatic hernia before surgery. Review of AAH and pulmonary sequestration (PS) revealed that the present case is the youngest of solitary AAH and also the first report of solitary AAH arising in ELS without a primary lung cancer. In a sense, the present case firstly supports the hypothesis that ELS may be an underlying cancer predisposition syndrome, so aggressive surgical therapy should be recommended for ELS. PS is often associated with other congenital anomalies, such as congenital diaphragmatic hernia (CDH) and congenital heart diseases. Atypical adenomatous hyperplasia (AAH) usually is associated with primary pulmonary adenocarcinoma. The review of the literatures reveals a few of cases of adenocarcinoma arising in PS, in which only one arising in extralobar sequestration (ELS), and solitary AAH had never been repoted in PS.Pulmonary sequestration (PS) consists 0.15%-6.4% of all congenital malformation of lungth partial costectomy. During the operation, congenital diaphragmatic eventration (CDE) was confirmed instead of CDH and diaphragm replication was executed. In addition, ELS was detected unexpectedly and removed, considering the history of pulmonary infection. In operation, we found the blood supply of ELS arise from the abdominal aorta. To our surprise, postoperative pathology showed AAH of bronchiolo-alveolar epithelium in the resected lung parenchyma  which shares common pleura with the normal lung tissue. The incidence of AAH has been reported to be about 9%-21% in primary lung cancer patients, while approximately 4%-10% in patients without lung cancer, and it is usually 0.5 cm or less and located in the peripheral area of the lung. Previously reported cases of AAH in children occurred in combination with a congenital cystic adenomatoid malformation, or metastatic osteosarcoma of the lung. To the best of our knowledge, this is the first report of solitary AAH arising in ELS without a primary lung cancer occurred in very early age. The youngest case of solitary AAH was a 12-year-old male in Korea, reported in 2016. In addition our review of the literatures reveals less than ten cases of carcinoma arising in PS, including four cases of pulmonary adenocarcinoma, but only one arising in ELS. Belchis et al hypothesized that the development of carcinoma in the sequestrations may be multifactorial and may include chronic inflammation and irritation. However, this hypothesis does not explain AAH in a patient under the age of three ages, who lacks a long-standing history of recurrent infection. Another hypothesis postulated that PS manifested as an underlying cancer predisposition syndrome and an innate propensity to undergo malignant degeneration. However, authors thought lack sufficient evidence, for all of the sequestration malignancies occurred in older patients. In a sense, the present case, a two years old AAH patient, supports the hypothesis that ELS may be an underlying cancer predisposition syndrome.AAH is involved in the early stage of a complex multistep carcinogenesis of pulmonary adenocarcinoma and thought to be a probable forerunner. As previously mentioned, AAH is a forerunner of pulmonary adenocarcinoma. Without any treatment, AAH arises from ELS may worsen a patient's condition and even develop into adenocarcinoma. Although the incidence of AAH arising from ELS is low, its existence provided an argument for surgery. At least, patients should receive regular follow-up after treatment.Surgical resection is the conventional standard treatment for PS to prevent possible infection, congestive heart failure, and hemoptysis. However, if the symptoms of patients with PS are not serious, they are often left un-intervened. The management of ELS is more controversial, it is known that these lesions can remain asymptomatic throughout the patient's life but the complications may developWe firstly reported the solitary AAH arising from ELS with CDE, and this AAH patient was the youngest in the world now. It is possible that AAH is one of the congenital anomalies associated with ELS, just like CDH and other congenital heart diseases, but the molecular mechanism is unclear. In our opinion, surgical treatment should be recommended in a patient with ELS regardless of their clinical symptoms since it might be an underlying cancer predisposition.


Internal herniation through lesser omentum hiatus and gastrocolic ligament with malrotation is extremely rare. This type of internal hernia has rarely been described before. Preoperative diagnosis is difficult and prone to misdiagnosis.A 38-year-old Chinese woman was an emergency admission to our hospital with a sudden onset of acute epigastralgia for the past 14 hours. We made a presumptive diagnosis of gastrointestinal perforation and septic shock. Due to the acute onset and rapid progress, she received timely surgical treatment. During operation, we observed that her small intestine herniated into the hepatogastric ligament and ligamentum gastrocolicum hiatus accompanied with intestinal malrotation that resulted in internal hernia. We found a diverticulum of approximately 3.0 × 6.0 cm sited at a distance of 80 cm from the ileocecal intestine. We resected the strangulated intestinal loop and the diverticulum, performed an appendicectomy, and closed the ligamentous fissure. Postoperation, she recovered smoothly, without any complications, and was discharged on day 6.A case of internal hernia formation is quite rare; accurate preoperative diagnosis and timely surgery are essential because it can cause strangulation of the ileus. However, the incidence of this internal herniation is low and preoperative diagnosis is difficult. An accurate preoperative diagnosis of internal hernia is still a challenge. An internal hernia is formed in a variety of ways, such as various intestinal mesentery defects of congenital or acquired peritoneal folds. Duodenal hernia (53%) and pericecal hernia 13%) are the most common internal hernias, followed by the foramen of Winslow (8%), transmesenteric hernia (2%), and transomental hernia (1%) [3% are th9/L, with 92% neutrophils), hemoglobin (Hb) 118 g/L, and normal liver and pancreatic enzymes. An emergency abdominal plain film  of the patients, and convergence of the mesenteric vessels at the superior lesser curvature of the stomach in 43% (3/7) [Preoperative radiography could provide evidence for the diagnosis of a lesser omental hernia. Abdominal orthostatic X-ray is a valuable method, which shows a round or arc gas and air-fluid level in lesser omental bursa, anterolateral displacement of the stomach, and presence of intestine loops beneath the liver in the right upper abdomen. Unusually, however, this case showed an arc loop of small intestine in subphrenic, not in lesser omental bursa or beneath the liver; the imaging led to the misdiagnosis of a hollow viscera perforation at first sight! Moreover, the examination of multiple abdominal computed tomography (CT) scans plays an important role in the diagnosis of lesser omental hernia. Konishi 3% (3/7) . HoweverWe speculate that the hernia occurred in this patient because an intestinal malrotation caused her internal anatomy to be abnormal, so that her intestine herniated into the vulnerable site of hepatogastric and gastrocolic ligament, then generated the internal hernia entity. Special attention should be paid to the diagnosis made before the operation. First, the abdominal plain film showed free gas below our patient’s diaphragm; from the first impression it was very easy to consider perforation of her abdominal cavity organs. Careful observation of the abdominal plain film reveals that under her diaphragm was gas loop of small intestine! The transverse arc subphrenic free gas is usually crescent shaped, while both ends of bowel loop shadow are often more rounded off Fig. . Second,The incidence of intra-abdominal hernia is rare, especially for patients who have not had abdominal surgery, so clinicians should be vigilant. Unfortunately, the preoperative diagnosis rate is low, patients often have obvious symptoms and late stage manifestation before emergency surgery. Because it is prone to incarceration and necrosis, delayed treatment could lead to dire consequences. Therefore, diagnosis and confirmation of preoperative diagnosis is still a challenge.


She was misdiagnosed with gastrointestinal disorder and ultimately had undergone imaging. Ultrasonography and computed tomography (CT) scan disclosed a huge solid-cystic mass originating from the ovary. On clinical examination the patient had no pain or tenderness and no gynecologic complaints. Laboratory tests showed normal tumor markers and hemoglobin at 8 g/dl. Laparotomy was carried out as diagnosis of ovarian serous cyst adenoma, but a huge tumor with attachment to uterus and ovaries and extension to pelvic floor, peripheral tissues of ureter, and upper abdomen was found. Hysterectomy with bilateral salpingooophorectomy was done. Pathology report demonstrated uterine leiomyosarcoma measuring 40 centimeters and weighing 10 kilograms. In conclusion, as pelvic masses even in a large size may present unspecific symptoms misdiagnosis may occur which lead to overgrowth, local invasion, or other complications. So, it is rather to suggest ultrasonography in patients with persistent abdominal or pelvic symptoms and if needed, more exact diagnostic modalities like magnetic resonance imaging (MRI) could be offered to avoid misdiagnosis and mismanagement.A 41-year-old woman (G Pelvic masses are common findings in general gynecology . Benign 3P2L2Ab1) was referred to gynecology clinic with chief complaints of abdominal distension and localized abdominal pruritus for three months. To rule out gastrointestinal disorders, she had visited a general physician given her symptoms. But she found no response to the drugs in spite of a long-term usage and was referred to our clinic after taking ultrasonography. Her past history showed menorrhagia but not menstrual irregularity and dysmenorrhea for several months. Laboratory tests including hemoglobin at 8 g/dl, hematocrit at 28,7%, ferritin at 6 ng/ml, and iron at 29 mcg/dl approved this claim but the patient had not expressed it initially as a complaint. Vital signs were in normal limits. In general appearance, the patient was not cachectic with full activity without vertigo, impairing appetite, constipation, nausea, vomiting, and urinary symptoms, but a distended abdomen that lacks rebound and tenderness. The origin of the mass was not detectable by vaginal examination. Ultrasonography had revealed a huge multicystic septated mass, 40 cm in diameter in abdominal cavity, probably a serous cystadenoma originating from ovary that was extended to epigastric area. Uterine and bladder were reported normal; however, these were impressed by extrinsic pressure of the mass. Uterine observation was not possible with vaginal sonography due to huge ovarian tumor. Tumor markers including cancer antigen-125, carcinoembryonic antigen, αFP, and CA19-9 were all within normal limits. Further assessment by computed tomography (CT) scan was done to show the nature of tumor which approved the diagnosis of ovarian serous cyst adenoma  are the most common sarcomatous malignancies of uterus that represent 1-2% of all uterine malignancies , 9. MostThe absolute diagnosis of uterine leiomyosarcoma is made by histologic confirmation. In most cases, the diagnosis of these tumors is made by specimen examination in hysterectomized patients who was managed as leiomyoma , 12.The main treatment of LMS is surgical excision which consists of total abdominal hysterectomy and debulking of any tumor invading outside the uterus. It is considered appropriate to preserve ovaries in young women and routine dissection of pelvic and para-aortic lymph nodes, since lymph node involvement is seen in less than 3% of patients . Its fivIn conclusion, pelvic masses even in a large size may be without any specific symptoms. Delay in diagnosis may lead to overgrowth, torsion, or hemorrhagic tear of tumors and invasion to surrounding visceral organs. So, it is considered more appropriate to suggest ultrasonography in patients with persistent abdominal pain or distension who do not respond to recommended drugs and in case of presence of a mass, MRI as a preferable imaging could be offered to avoid misdiagnosis.


A 65-year-old male was brought to our hospital with right upper abdominal fullness sensation and recent body weight loss of about 3 kg. The patient had developed episodes of melena following progressive abdominal muscular guarding and drop of haemoglobin level to 6.3 g/dL. An abdominal computed tomography scan disclosed a ruptured hepatocellular cell carcinoma. A segmental arterial mediolysis was found on the superior mesenteric artery in the process of repairing the ruptured right hepatic artery with the assistance of angiography. Transarterial embolization was carried out and permanent haemostasis was achieved. Segmental arterial mediolysis (SAM) is an uncommon non-inflammatory and non-atherosclerotic vascular disease first described by Slavin in 1976, which is characterized by dissecting aneurysms, haematoma, occlusion and haemorrhage following vacuolization, and lysis of the outer media of the arterial wall . It occasionally occurs in the middle-aged and elderly, but rarely in young patients , 3. SAM A 65-year-old male, who had previously been in good health, presented to the emergency department with a 2-month history of weight loss of about 3 kg. He reported poor appetite and a distended sensation over the right upper quadrant region of the abdomen. General vital signs revealed a body temperature of 37°C, a rapid heart rate of 109 beats per minute, and a blood pressure of 129/90 mmHg. Physical examination revealed a distended right upper epigastric region with a palpable mass lesion. No muscular guarding or tenderness was observed initially. Laboratory examination results are shown in The most common clinical presentations of SAM include abdominal pain and bleeding. The haemoperitoneum in our case is related to hepatocellular cell carcinoma with rupture; however, the tarry stool episode with unremarkable findings from panendoscopy of the upper gastrointestinal tract could suggest the underlying SAM . RadiogrThe differentiated diagnosis may include infection (e.g. mycotic aneurysm and endocarditis) and connective tissue diseases  and Behcet syndrome) , 15–18. polyarteritis nodosa, which can have an angiographical appearance identical to SAM. The treatment of SAM is composed of vascular embolization or surgical resection [Although angiographical patterns are suggestive of SAM, histopathology remains the ‘gold standard’ for definitive diagnosis. This is especially important in the case of esection , 19. Theesection , 21. To Segmental arterial mediolysis is an acute, limited disease. In patients with malignancy-related intra-abdominal bleeding, it is difficult to locate the origin of bleeding—as in our case with a ruptured hepatocellular carcinoma. Conventional angiography is more sensitive than CT or magnetic resonance angiography, and should be used if the more commonly-used methods fail to reveal a diagnosis. If a diagnosis of SAM is suspected a multi-disciplinary approach—involving consultation with interventional radiology and vascular or general surgery—should be promptly pursued.Conflict of interest: none declared.


The aim of this study is to present a rare case of Müllerian adenosarcoma of the cervix with ovarian metastasis and sarcomatous overgrowth. A gravida 2, para 2 woman aged 32 years with vaginal bleeding was admitted to the gynecology department. A 3-4 cm polypoid mass protruding from the cervix was detected in a pelvic examination. Total abdominal hysterectomy and bilateral salpingoopherectomy was performed because of metastatic implants on the right ovary. The pathologic evaluation revealed Müllerian adenosarcoma of the cervix with sarcomatous overgrowth and ovarian metastasis. After surgery, the patient was planned to undergo chemo- and radiotherapy. This is the first cervical Müllerian adenosarcoma case mentioned in the literature with metastasis to the ovary in a young woman. There is no optimal management option for cervical adenosarcomas due to the rarity of this phenomenon. Nevertheless, even if the patient is young and imaging techniques do not elucidate metastatic disease, surgeons should evaluate the ovaries for the spread of tumor, especially if histology reveals sarcomatous overgrowth. MA mostly occurs in the uterus but also in extra-uterine sites, particularly in the ovary.Müllerian adenosarcoma (MA) is relatively a rare type of mixed epithelial and mesenchymal tumor of the uterus. It is usually composed of a benign epithelial component and a low-grade stromal-sarcomatous component. Occasionally, the epithelial component may be atypical and sarcomatous overgrowth may be seen in the stromal component,4. Cervical adenosarcoma, especially with sarcomatous overgrowth, is extremely rare. Distant metastasis is also extremely rare because the epithelial component is usually benign and the stromal component is generally low-grade. In this report, we present a MA originating from the endocervical canal with sarcomatous overgrowth including heterologous elements and ovarian metastasis in a young woman.The tumors generally originate from the uterine corpus/endometrium, and less frequently from the uterine cervix. Adenosarcoma usually presents with abnormal vaginal bleeding. If the tumor originates from the cervix, the tumor is commonly seen as a polypoid tissue protruding from the external cervical ostium like a cervical polyp on gross evaluationA gravida 2, para 2 woman aged 32 years with vaginal bleeding was admitted to the gynecology department. She reported having abnormal vaginal bleeding for a couple of years and a benign endometrial biopsy in another gynecology center 6 months ago. The pelvic examination revealed a 3-4 cm polypoid mass protruding from the cervix to the vagina. The uterus and the ovaries were normal in size. Transvaginal sonography revealed an irregular-shaped polypoid structure lying from the internal cervical ostium level to the vagina whose thickness was 16 mm. Magnetic resonance imaging (MRI) revealed a lesion starting from the endocervical canal lying to the vagina and there were no pathologic lymph nodes. Her serum CA125 and CA19-9 levels were within normal limits. A biopsy from the polypoid lesion revealed MA with sarcomatous overgrowth.During surgery, metastatic implants were observed over the right ovary and the left ovary was solid in appearance. The outer surface of the uterus was normal. Total abdominal hysterectomy and bilateral salpingoopherectomy was performed. Three polyps were seen in the endocervical canal, which measured between 0.6x0.5x0.3 and 2x0.8x0.5 cm. The cut surfaces of the polyps were solid and grey-white in appearance. Hemorrhage was seen because of previous biopsy. The tumor had invaded the outer half of the myometrium macroscopically , 1b.The final report confirmed that the tumor originated from the endocervical canal. It invaded the lower uterine segment and the myometrium, less than the outer half, and consisted of elongated spindle cells with a myxoid background and few benign glandular formations. The sarcomatous stroma made up 80% of the tumor. In these areas, paucicellular and hypercellular morphology with myxoid stroma were seen. Low-grade and high-grade stromal components were mixed. Mitotic activity was 4 per 10 high-power fields (HPFs) and 2 per 10 HPFs in paucicellular and hypercellular areas, respectively. Immunohistochemistry showed positive immunoreactivity with vimentin and desmin, and negative immunoreactivity to actin, s-100, and CD99. The glandular epithelium was primarily of endocervical type without atypia and mitotic activity. Large foci of benign cartilage were seen in the sarcomatous areas , 1d, 1e.,6,7,8. The age at clinical presentation in cervical adenosarcoma is lower than that with uterine corpus, which is about 27 years,4,5,6. This is concordant with our patient whose age was 32 years. Chin et al. also showed that 89% of patients were premenopausal. The most frequent symptom is abnormal vaginal bleeding, as in our case. Generally, the most common finding in physical examination is cervical polypoid lesion protruding from external cervical ostium to the vagina. The diagnosis can be made through biopsy from the lesion.MA is an infrequent type of sarcoma of the female genital system. It mostly comprises a low-grade malignant stromal component and benign epithelial component. It commonly arises from the uterine corpus/endometrium. Cervical adenosarcoma is seen in 2-9% of all MAs. This subtype has a more malignant behavior than the classic adenosarcoma. The rate of myometrial invasion and extension to the serosal surface, recurrence rate, and death because of tumor progression are all higher in adenosarcomas with sarcomatous overgrowth,11.Sarcomatous overgrowth is defined as overgrowth of the neoplasm by a pure sarcomatous component occupying at least 25% of the lesion,4. Mostly, these elements are cartilage or striated muscle. Charfi et al. also reported a patient with adenosarcoma of the cervix with sarcomatous overgrowth and heterologous elements that contained cartilage and presented as a recurrent cervical polyp.Heterologous elements are rarely reported, accounting for 8-42% of cervical adenosarcoma reported that ovarian metastases were not identified in any patients undergoing bilateral salpingoopherectomy in 16 patients with MA. The present case is one of the first with cervical adenosarcoma metastases to the ovary.Cervical adenosarcomas with sarcomatous overgrowth in young patients are very rare. In particular, ovarian metastasis at the time of diagnosis is almost not mentioned in the literature. A recent study by Tanner et al., reported 9 cases of cervical adenosarcoma. They performed a cervical wedge resection for a patient who desired to preserve fertility. Hysterectomy, bilateral salpingoopherectomy, and lymphadenectomy were performed for other patients, none of whom had sarcomatous overgrowth. Park et al. performed hysterectomy, bilateral salpingoopherectomy, and lymphadenectomy for a 37-year-old woman with adenosarcoma of the cervix with sarcomatous overgrowth. However, there was no tumor in the ovaries and no lymph node metastasis. Manoharan et al. reported three patients with cervical adenosarcoma. The first one was a 28-year-old woman with sarcomatous overgrowth. She underwent radical hysterectomy with preservation of the ovaries. The histology revealed lymphovascular space and right parametrial invasion, so she had chemotherapy followed by both pelvic radiotherapy and brachytherapy. The second case was a 26-year-old woman who had cervical MA with spindle cell stroma and benign glands. Vaginal hysterectomy with preservation of the ovaries and laparoscopic lymph node dissection was performed. The lymph nodes were free of tumor. She was given adjuvant pelvic radiotherapy because of deep stromal infiltration and high-grade tumor. The third case was a 41-year-old woman who had cervical adenosarcoma with mild-to-moderate stromal cytological atypia and underwent abdominal hysterectomy with preservation of ovaries. She received no adjuvant therapy because the tumor was localized to the polyp. Our patient was also a young woman and we were planning to preserve the ovaries before surgery. However, we came across the metastatic implants on the right ovary intraoperatively. MRI and ultrasonography did not facilitate the management preoperatively.The optimal therapy and the prognostic factors for these are still unclear because the reports of these cases are insufficient and long-term follow-up data are absent. The extensiveness of surgery may differ from patient to patient. Chin et al.As previously mentioned, this is a very rare case and there is no consensus on either surgical or adjuvant treatment strategy. The first point that physicians should keep in mind is the number of mitotic figures in the mesenchymal part because the epithelial part is benign. Secondly, sarcomatous overgrowth may determine the behavior of the disease, as it was in our patient. If there is sarcomatous overgrowth, surgeons should carefully evaluate the patient for distant or locoregional spread.As a result, there is no optimal management option for cervical adenosarcomas due to the rarity of this phenomenon. Nevertheless, even if the patient is young and imaging techniques fail to reveal metastatic disease, surgeons should evaluate the ovaries for the spread of tumor, especially if the histology reveals sarcomatous overgrowth.


Scrotoschisis is a rare congenital anomaly of the scrotal wall with idiopathic etiology and unknown prevalence. This pathology is extremely rare. We report a new case and review the literature for relevant data.A 3-day-old full-term baby boy of African ethnicity, who had a homebirth, with birth weight of 2.7 kg presented to our emergency department with exteriorization of left testis; after clinical examination and proper investigations the diagnosis was scrotoschisis. Surgical treatment was performed by primary closure with excellent follow-up. We reviewed the literature to elaborate on the etiology of this pathology and its management.Scrotoschisis is a rare congenital anomaly affecting healthy babies. Early management is substantial. Further studies are recommended to learn more about the etiology and long-term results, including the effect on the fertility. Scrotoschisis is a rare congenital anomaly of the scrotal wall with idiopathic etiology and unknown prevalence. This pathology is extremely rare. In this anomaly, the testis eviscerates through an opening high on the anterior wall of the scrotum . The exaA 3-day full-term baby boy of African ethnicity presented to our emergency department with exteriorization of left testis from his left hemi-scrotum. He was born at home at 41 weeks of gestational age by normal vaginal delivery: birth weight. 2.7 kg, birth length of 44 cm, and head circumference of 46 cm. His 27-year-old mother, primigravida, is a farmer without a particular past or medical history: there is no family history of the same pathology and no history of consanguinity. An examination revealed no relevant findings for his other organs. There was neither antenatal nor ultrasonography follow-up. A physical examination was remarkable for left scrotal anterior wall defect of 1.5 × 2 cm with normal aspect of inguino-genital region. His left testis was exteriorized within its coverage of tunica vaginalis, mildly edematous with a thin fine exudative inflammatory membrane but with normal vascular appearance. All blood analyses and urine examinations were within normal range. Broad-spectrum antibiotics (ceftriaxone) were started on his arrival at our emergency unit. Under general anesthesia, surgical debridement and scrotal defect exploration showed complete left scrotoschisis with free and mobile extracorporeal testis, no calcified masses, and no meconium residua (Fig. Scrotoschisis is a rare congenital scrotal wall pathology. Its prevalence is unknown. Only 16 cases were reported in the literature , 3. It iScrotoschisis is a rare congenital anomaly affecting otherwise healthy babies. Early management is substantial. Further studies are recommended to learn more about the etiology and long-term results, including fertility.


A rare case of birooted primary maxillary central incisor is reported. A 5-year-old male patient reported with carious maxillary primary anterior teeth. Radiographic examination incidentally revealed root bifurcation in primary maxillary central incisors bilaterally. This represents a rare finding. There are few cases that have reported birooted permanent incisors, but there is no case reported till date with birooted primary incisors. This report discusses the findings in the context of the available literature. The possible etiology and the need for careful assessment prior to root canal therapy are highlighted. Jajoo SS. Primary Maxillary Bilateral Central Incisors with Two Roots. Int J Clin Pediatr Dent 2017;10(3):309-312. The present report describes a case in which the maxillary primary central incisors are birooted in mesial and distal directions.Primary teeth have fewer abnormalities with respect to size and morphology as compared with permanent teeth. Rarely primary teeth have additional roots and those that do are usually primary molars and the primary canines.3 According to Ingle,4 one of the most important causes of endodontic treatment failure is the incomplete obturation of the root canal system. Similarly, Vertucci,5 DeGrood, and Cunningham6 reported that a considerable number of failures could be assigned to anatomical variations, such as the presence of unusual root canals.10 The purpose of the present study is to present and describe a clinical case of endodontic treatment of primary maxillary central incisors with two roots demonstrated by radiographic examination.One of the main objectives of nonsurgical endodontic treatment is the elimination of infection from root canal system and prevention of its reinfection. Thus, a prerequisite for the success of conventional endodontic procedure is the clear understanding of root canal morphology and that the entire root canal system must be shaped, disinfected, and filled.A 5-year-old male patient came to the Department of Pedodontics and Preventive Dentistry, M.A. Rangoon-wala College of Dental Sciences and Research Centre, Pune, Maharashtra, India, with a chief complaint of decayed upper front teeth. The patient’s medical history was noncontributory. Patient’s mother gave a history of breastfeeding for 1 year after which the child was bottle fed for 2 years. Intraoral examination revealed primary dentition with 51, 52, 61, 62, 72, 73, 74, 75, 82, 83, 84, 85 carious teeth .The intraoral periapical (IOPA) radiograph revealed pulpal involvement with 51, 52, 61, 62, 74, 75, 84, 85 . In addiDiet analysis, counseling, and oral prophylaxis were undertaken. The treatment planned for the pulpally involved teeth was pulpectomy, followed by composite buildup with anteriors and stainless steel crown with the posteriors. Extraction was planned for 74, 84 as the teeth were grossly decayed followed by functional space maintainer.The treatment plan was divided into two phases for 51, 52, 61, and 62. Phase 1: Endodontic phase Phase 2: Restorative buildupAt the first appointment, an infraorbital block was administered. A relative isolation of the upper anterior primary teeth was obtained as the child was not sufficiently cooperative.Gross carious lesions were removed with a no. 330 round carbide steel bur .The pulp chamber was opened and working length determination IOPA was taken with 52, 62 using 8 no. K-file. During canal negotiation with 51, one K-file was placed in distal canal, while the other K-file was placed in the mesial direction and working length was determined . SimilarGlass-ionomer cement (type II) was placed in close proximity to canal orifice to avoid contact between zinc oxide eugenol and composite . After c11 demonstrated that bifurcated roots may be related to an in-growth of Hertwig’s epithelial root sheath. Other researchers have suggested that fusion or germination may be related to the clinical presentation of supernumerary roots.12 The development of the roots begins after enamel and dentin formation has reached the future cementoenamel junction. The enamel organ plays an important part in root development by forming Hertwig’s epithelial root sheath, which molds the shape of roots and initiates dentin formation. There is a pronounced difference in the development of Hertwig’s epithelial root sheath in teeth with one root and in those with two or more roots.13 These findings suggest that in this case a defect in the dental lamina for the early stage of root formation could be an etiological factor in birooted incisors. It is also possible that an abnormality in the morphodifferentiation of the incisors may have occurred. Such abnormalities may be genetically determined or be associated with environmentally induced cellular changes.1The etiology of teeth with supernumerary roots is poorly understood. Several authors have postulated theories for the occurrence of this phenomenon. Kelly14 used the term supernumerary roots in describing the development of increased number of roots in a tooth compared with a classical description in dental anatomy. When a maxillary incisor presents two roots or two root canals, conditions, such as fusion, gemination, dens in dente, and some variation in the normal development of Hertwig’s epithelial root sheath must be considered.1516 In the present case, the pretreatment radiograph showed no evidence of enamel or dentinal invagination, thus making dens in dente or dens invagination unlikely causative factors.Neville et al16In this case, clinical examination and pretreatment radiograph revealed a crown of normal size and shape side. Therefore, there are no chances for fusion or gemination, which results in either a single larger crown or a fused or joined crown.13 normal root development occurs when Hertwig’s root sheath is horizontally bent at the cementoenamel junction to narrow the cervical opening of the tooth germ. In this case report, the clinical crown has normal shape , and it seems that during the epithelial diaphragm formation some incident caused the development of a horizontal flap of the Hertwig’s epithelial root sheath, and then the horizontal flap fused and resulted in the formation of a second root.According to BhaskarEndodontic success in teeth with a number of canals above that normally found requires a correct diagnosis and careful clinical radiographic inspection. Morphological variations in pulpal anatomy must be considered before treatment onset. The case presented a maxillary central incisor with two root canals, determining the developmental origin of this anatomical anomaly that appeared to have clinical significance.Knowledge of dental anatomy is fundamental for proper endodontic practice. When root canal treatment is performed, the clinician should be aware that both external and internal anatomy may be abnormal. Radiograph and computer tomography can help identify abnormal tooth anatomy.


Pulmonary artery sarcoma is a rare tumor with varying presentations including pericardial effusion and pulmonary metastasis. Single‐nucleotide polymorphism array is a novel method that can be used to define tumor genome and be used as a guidance to choose the proper treatment regimen. Pulmonary artery sarcomas (PAS) are rare tumors with varying presentations including pulmonary artery obstruction, pulmonary hypertension, and intrathoracic or extrathoracic metastasis A 69‐year‐old woman was hospitalized because of palpitation, nausea, and vomiting. She reported a history of dyspnea and pleuritic chest pain one month prior to hospitalization that was gradually worsened. Her past medical history included hypertension and chronic kidney disease. The physical examination showed several remarkable elements, including hypoxemia at room air, decreased blood pressure, and muffling of heart sounds. Because cardiac tamponade was our first concern in the diagnosis, bedside transthoracic echocardiography (TTE) was performed, which revealed moderate pericardial effusion with tamponade physiology. Patient underwent emergent pericardiocentesis and 800 cc of bloody fluid was drained. TTE was also revealing a large lesion in the main pulmonary artery, with extensions into the right and left pulmonary arteries Fig. . In ordeAnalysis of pericardial effusion showed lymphocyte‐predominant fluid with negative cytology. Subsequently, 18‐fluorodeoxyglucose (18‐FDG) positron emission tomography–CT (PET‐CT) from the vertex to the thighs was performed and revealed an increased 18‐FDG‐standardized uptake value in the right upper lobe nodule and in a soft tissue density mass in the perivascular space in the anterior mediastinum Fig. .2 (days 1 and 8) and docetaxel 100 mg/m2 (day 8) with 21 days’ interval were prescribed. After 3 months, TTE and CT scans were repeated, but unfortunately, we found no improvement. At this time, a single‐nucleotide polymorphism (SNP) analysis of tumor's genome was planned. Venous blood sample of 5 mL has been collected to EDTA‐covered tube and the DNA was extracted. The analysis of rs2032582 (SNP) in tandem repeats of ABCB1 gene was carried out using allele‐specific polymerase chain reaction. This test revealed tumor susceptibility to doxorubicin. Based on these findings, a new chemotherapy regimen was started, containing ifosfamide 5000 mg/m2 (day 1) and Adriamycin 75 mg/m2 (day 1) with 21 days’ interval. Patient received four course of chemotherapy and subsequent CT scans following the end of the forth course of chemotherapy revealed partial response to chemotherapy and partial resolution of the pulmonary nodule and pulmonary artery mass (Fig. The patient received antibiotics for diagnosis of pneumonia and a CT‐guided biopsy of right parenchymal nodule was performed. A pathologic examination showed neoplastic tissue composed of pleomorphic cells, while immunohistochemistry of the tumor cells was positive for vimentin and negative for Pan CK, EMA, S‐100, HMB‐45, SM‐actin, desmin, CD 31, CD 34, and CD 68, while 30% of tumor cells showed reactivity to Ki67. All findings were suggestive of high‐grade pleomorphic sarcoma (Fig. Pulmonary artery sarcomas are rare tumors that the literature suggests are often mistaken for pulmonary artery thrombi Although surgical resection is the standard treatment for pulmonary artery sarcoma, these tumors usually present as late, bilateral, locally advanced, and/or metastatic, which decrease the possibility of successful operation SH and SK: wrote the draft of the manuscript and obtained the consent. NA: performed data interpreting regarding to SNP analysis and participated in the writing. AS, AI, and AA: performed literature review and language editing and gave final approval of the manuscript. All authors have read and approved the final manuscript.The authors declare that they have no competing interests.


The coexistence of Rosai-Dorfman disease (RDD) with Langerhans cell histiocytosis (LCH) is very rare, as to date only 17 cases have been reported in the english literature. The pathophysiology of this uncommon co-occurrence still remains enigmatic and a subject of various speculations.We report a case of a 30-year-old female patient who presented with a pathologic fracture of the left proximal femur. Her medical history was unremarkable, there were no fever, skin lesions, lymphadenopathy or other organomegaly at physical examination. X-ray radiograph of the fractured femur showed an isolated and ill-defined osteolytic lesion. The histopathological analysis of biopsies from this lesion were consistent with a combined RDD-LCH of the bone.Combined RDD-LCH is a very rare phenomenon, whose pathophysiology still remains unclear and a subject of various speculations. Histiocytic disorders are a are and heterogenous entity comprising a large variety of diseases with a wide spectrum of clinical, histological, molecular and prognostic features , 2. LangRecent studies suggested that LCH is a clonal proliferation of abnormal dendricytic cells (Langerhans cells) with a particular phenotype, CD1a+, S-100+ and CD68+ . The disIn contrast, RDD is a non-neoplastic disease characterized by a polyclonal proliferation and a tissue accumulation of CD1a- histiocytes , 3, 5. LHowever, the co-occurrence of RDD and LCH is a very rare phenomenon. Since the first case of combined RDD-LCH reported in 2002 by Wang KH et al. , to the A 30-year-old woman was admitted at emergency department for a pathologic fracture of the left femur bone, after a mild trauma. The patient’s medical history, as well as her family history were unremarkable. The physical examination showed a painful and swollen left hip with inability to move the lower limb because of the pain. Apart from these trauma-related signs, there were no fever, skin lesions, lymphadenopathy or other organomegaly. An X-ray radiograph has been performed and revealed at the traumatic site, a relatively well-limited trochanteric osteolityc lesions at the fracture’s site Fig. . No otheAfter treatment with 10% nitric acid , the histological examination of the submitted bone biopsies showed a dense inflammatory infiltrate destroying the bone tissue Fig. . It consThe association between RDD and LCH is extremely rare, since the first case reported by Wang KH et al., 16 otheThe morphologic diagnosis often shows typical features of both types of histiocytosis, RDD and LCH , 5. The BRAF mutation [Anyway, transitional cells present or not, the main enigmatic and challenging issue is the exact pathological and molecular mechanism underlining the association between RDD and LCH. In fact, previous studies have supported a similar cytokine-mediated mechanisms in both RDD and LCH, and it has been speculated that the RDD presents as a reaction to LCH, or that both LCH and RDD could result from divergent differenciation from a common precursor, or that the histiocytes have undergone a phenotypic switch , 4, 5. Lmutation , 3. In cmutation , 5. Howemutation , suggestmutation , 15. JXGmutation , 15, wasAs suggested by previous authors, the co-existence of RDD and LCH is more than a simple coincidence, and more studies in the future are required to highlight the complex relationship between the two entities.Association between RDD and LCH is a very rare occurrence; a few cases have been reported in the current literature. The mechanism by which these entities occur together still remains a subject of various speculations, and future studies are required to unveil this enigmatic and challenging fact.


Insular gliomas were previously considered inoperable lesions and were typically treated via biopsy, chemotherapy and/or radiation, if not observation alone. Stereotactic biopsies of low grade insular gliomas can underestimate tumor grade or fail to establish malignancy. Moreover, the survival advantages of maximal safe resection for insular lesions are increasingly being recognized. As such, early surgical resection is increasingly being performed. As with most lesions, a differential diagnosis exists for apparent insular gliomas, with definitive diagnosis generally obtained upon resection. We report an illuminating case that presented similarly to an insular glioma undergoing malignant transformation, but resolved spontaneously following a nondiagnostic biopsy. A 53-year-old female patient presented with aphasia and dizziness, followed by syncope and a 30-minute loss-of-consciousness. Imaging findings included a 12 mm region of contrast enhancement and central necrosis within a larger 3.5 cm expansile, T2-hyperintense lesion involving most of the left insula and extending into the anterior left temporal lobe. Imaging was felt most consistent with high-grade glioma. Stereotactic biopsy demonstrated nonspecific gliosis without definitive neoplastic tissue. A systemic workup was unrevealing, and thus an open procedure was subsequently planned in the intraoperative magnetic resonance imaging (MRI) suite. Preoperative MRI demonstrated interval resolution of the original lesion, despite profound T2 hyperintensity along the needle tract; thus, the planned resection was aborted. Subsequent imaging and systemic studies failed to establish a definitive infectious, neoplastic, autoimmune, or other diagnosis. However, poor dentition, history of a recent dental procedure, and the tiny central focus of diffusion restriction on the index MRI rendered abscess the most parsimonious explanation. On follow-up imaging, the lesion was noted to have further resolved without intervention. Our case illustrates the complexity of managing insular lesions and highlights the potential for alternate pathologies that can mimic insular glioma. Additionally, it provides a humbling reminder that, even in the presence of seemingly pathognomonic imaging findings, a differential diagnosis of insular lesions must be thoughtfully considered in patient counseling and presurgical planning. Insular gliomas represent a uniquely challenging subset of primary brain tumors, which typically arise from white matter tracts adjacent to the mesocortex or allocortex . This peIn most supratentorial locations, MRI adequately distinguishes tumor from abscess; however, in the compact and anatomically complex insula, edema surrounding an abscess or metastatic lesion may masquerade as the typical T2 hyperintensity of a glial tumor, creating a diagnostic dilemma . Given tA 53-year-old woman with no prior neurological history presented to the emergency department after an isolated episode of word finding difficulty and dizziness, followed by syncope and a reported 30-minute loss-of-consciousness. No tongue biting, bowel/bladder incontinence, or other sign of ictal activity was observed, and she recovered to her neurologic baseline. Her medical history was significant for occasional nighttime headaches—severe enough to wake her from sleep—and chronic right-sided hearing impairment associated with prior chronic otitis media status post mastoidectomy. Her physical exam was unremarkable, with no other focal neurologic deficits; her family history was significant for malignancy in two second- and third-degree relatives.A head computed tomography (CT) demonstrated low attenuation changes suggestive of vasogenic edema in the left anterior perisylvian region, with loss of insular grey-white differentiation and ambiguous mass effect Figure .A follow-up MRI demonstrated a 3.5 cm expansile, T2-hyperintense lesion involving most of the left insula and extending into the anterior left temporal lobe, within which was noted a 12 mm region of contrast enhancement and central necrosis. The imaging findings were felt most consistent with high-grade glioma , erythrocyte sedimentation rate (ESR), echocardiogram, and cerebrospinal (CSF) analysis. The microbiology testing of the biopsy specimen was also negative, including Gram, mycobacteria, Nocardia, fungal, and acid-fast bacillus stains. Given the nondiagnostic biopsy and confounding clinical course, the case was referred to our surgical neuro-oncology team and presented at a multidisciplinary conference, from which we recommended awake craniotomy in the intraoperative MRI suite to obtain tissue diagnosis and potentially proceed with maximal safe resection, as indicated. The patient consented to awake craniotomy in the intraoperative MRI suite. However, the preoperative MRI failed to demonstrate the substantial previously observed insular T2 hyperintensity despite new significant fluid-attenuated inversion recovery (FLAIR) signal surrounding the needle tract. The prior area of contrast enhancement was similarly greatly reduced Figure .Given these imaging findings, the possibility of insular glioma was now felt to be decidedly unlikely. Although the still-awake patient was prepped in pins for the procedure, a mutual decision was reached to abort the case. All further studies failed to establish a definitive diagnosis; however, in light of what was subsequently determined to be extremely poor dentition and a history of recent self-extraction of a tooth, as well as a small central focus of diffusion-restriction on the original MRI, an abscess was thought to be the most likely diagnosis. The patient elected to observe the lesion, and follow-up MRI at three months demonstrated further resolution of the lesion with overall restoration of normal regional anatomy Figure .As of last follow-up at six months, the patient has remained completely asymptomatic with no evidence of ictal activity, paroxysmal spells, or other neurologic abnormalities, and she has displayed continued radiographic stability and no clinical evidence indicating an underlying diagnosis.The insula is a challenging neurosurgical structure from both diagnostic and surgical perspectives due to its complex anatomy, proximity to critical vasculature, and white matter tracts. Although glioma surgeons at high volume referral centers may see an abundance of what are ultimately pathologically proven to be insular gliomas, the present case illustrates the potential for other lesions in this region to mimic the classic expansile T2 hyperintensity of an insular glioma. Although arguments are sometimes made to observe small T2-hyperintense insular lesions to document growth prior to accepting the potential risks of surgery, evidence of contrast enhancement typically implies a more aggressive histology, discouraging conservative observation. We here illustrate a case wherein the opportunity for spontaneous resolution—perhaps facilitated by perioperative antibiotics at the time of biopsy—enabled a craniotomy to be avoided.Advances in techniques and technologies including intraoperative MRI and awake mapping techniques have empowered safe resection of insular lesions with an acceptable risk-benefit profile for the vast majority of patients . The impIn retrospect, though our case with glioma-like imaging findings and seizure-like presentation was within typical limits for a neoplastic lesion, certain radiographic features did put abscess on the differential diagnosis. Specifically, a dark rim of T2 hypointensity surrounding the lesion, together with central restricted diffusion and radial stranding enhancement are findings consistent with abscess . ConversIn terms of the generalizability of this case to the neurosurgical readership, our experience with this patient has not prompted us to change overall practice patterns in our institution; we continue to advocate for early surgical intervention for most patients with a significant clinical suspicion for glioma. Nevertheless, in the setting of increasingly favored surgery for insular gliomas at institutions worldwide, we felt this case to be an excellent and timely reminder of the imperative to critically scrutinize imaging findings and thoroughly explore all aspects of the clinical history. Moreover, in all such neuro-oncologic cases, a forthright discussion with the patient regarding diagnostic uncertainty must complement the surgical informed consent.The case we have presented herein illustrates the complexity of managing insular lesions and highlights the potential for alternate pathologies that can mimic insular glioma. In addition, it provides a humbling reminder that, even in the presence of seemingly pathognomonic imaging findings, a differential diagnosis of insular lesions must be thoughtfully considered in patient counseling and presurgical planning.


The cementoma is a common disease of the dental root apex, which generally occurs in the maxilla and the mandible, but the cementoma occurring in the long bone is rare. Moreover, the incidence of cementoma in the calcaneus is extremely infrequent.The present study reports an unusual case of a 19-year-old girl, who complained of pain in the left heel. Subsequent radiographs and computed tomography (CT) were used in the diagnosis. The imaging features of the lesion included a radiopaque matrix and radiolucent tissue, particularly an arc-shaped fat band. An excisional biopsy was performed. Histopathological examination confirmed the diagnosis of cementoma in the calcaneus. After the operation, the patient was followed up without recurrence.Imaging examination plays an important role in the differential diagnosis of cementoma of the calcaneus. Extragnathic cementoma of long bone as a benign bone neoplasm is very rare, which was reported to mainly locate in the lower limb in the literature –4. In 19We report an extragnathic cementoma of the calcaneus in a 19-year-old woman, who presented with intermittent pain at the left heel half a year ago. The imaging features including radiography and computerized tomography (CT) are described in our study. To the best of our knowledge, no report has previously introduced this tumor occurring in the calcaneus. Written informed consent was obtained from the patient.A 19-year-old woman presented with intermittent pain over a period of 6 months in the left heel, which was severe after activity and relieved after taking rest. There was no history of trauma to the left foot. She visited our hospital for treatment and diagnosis because of worsening pain in last 10 days. The pain was limited to the heel. Tenderness was noted in the posterior part of the calcaneus. No subcutaneous mass was palpated. Normal range of motion was demonstrated in the ankle. Radiograph of the left foot revealed a well-defined osteolytic lesion with marginal sclerosis, involving the calcaneal tuberosity. The size of the lesion was 3.5 × 2.5 × 2.0 cm Fig. . Taken tDuring operation, the cortex of the affected bone was found to be thin. Granulation with fibrous tissue was observed in the lesion. Calcified sample particles mixed together were also seen. A large stiff sclerotic mass was took out of the calcaneus and appeared pinkish-grey or grey. An excisional biopsy was performed. Histopathological examination revealed there was abundant fibrous tissue containing calcified spherules and few cells in the excised specimens. Cementum-like material was found within the calcified spherules, which irregularly formed acellular trabeculae. Some cementum corpuscles were mixed together Fig. . Thus a The patient was treated by curettage, bone grafting and bone packing with the iliac crest in the same side. There was no evidence of recurrence at the 2-year follow-up Fig. .Fig. 4PoAccording to the World Health Organization (WHO), the classification of odontogenic tumor keeps changing , 7. CemeIn the English literature, the radiographic appearances in the cementomas of the long bones were quite similar . The lesThis new observation of this rare entity found in our patient differs from others by its localization . The radiographic and microscopic appearance of the cementoma found in the present patient was consistent with the findings reported by previous authors. On radiograph, the lesion presented a cavity surrounded by an incomplete sclerotic rim and a radiopaque mass was present within it. On CT, it was cortical expansive, and had a well defined sclerotic border, peripheral radiolucency, and an amorphous eccentric focus of matrix calcification. These radiographic features resembled those of their maxillary counterparts and other previously reported lesions of the long bones containing cementum or cementum-like material , 9, 10. Radiologically, cementoma of extragnathic bone has been classified into three stages –14: osteThe radiographic differential diagnosis of cementoma of the calcaneus includes enchondroma , osteoblSince this study was a retrospective study, no MR examination was done and intraoperative photos were not acquired.We first present a rare case of cementoma of the calcaneus. When compared with dental cementoma, cementoma of the extragnathic bone is rare, mainly occurring in the long bone. The radiologic imaging features of this lesion, including the radiopague deposit and radiolucent tissue with an arc-shaped fat band should be concerned by the radiologists. When cementum corpuscle was detected, the diagnosis of cementoma should be considered. We report this case and hope to provide informative data that confirm that the cementoma of the extragnathic bone exists as a distinct entity.


Ewing’s sarcoma (ES) is an uncommon aggressive bone malignancy that mainly affects children and adolescents. Mandible involvement is quite rare and usually represents metastasis from another skeletal site. Combined therapy including wide surgical resection and preoperative and postoperative chemotherapy has been demonstrated as the mainstay of therapeutic approach. As improved therapeutic modalities have significantly increased survival over the last decades, functional and esthetic reconstruction of the postmandibulectomy defect is usually necessary for patient’s total rehabilitation. We report a case of ES of the mandible with special consideration to the postresection spontaneous structural and functional regeneration of the mandible.Chatzistefanou I, Kabesi S, Paraskevopoulos K, Koliouskas D, Antoniades K. Ewing’s Sarcoma of Mandible: An Impressive Case of Spontaneous Mandible Regeneration. Int J Clin Pediatr Dent 2016;9(3):273-277. More than 50% of ES cases originate in the pelvis and long bones,256 with distant dissemination at diagnosis being the rule rather than the exception, reflecting its aggressive biological behavior.7Ewing’s sarcoma (ES) is a poorly differentiated primary bone malignancy that mainly affects children and adolescents.11 The mandibular ramus is the predominant site of occurrence, with only few cases reported in the anterior mandible or maxilla.17 As improved therapeutic modalities have significantly increased survival, the current maxillofacial surgeon may need to face the challenge of an extensive surgical resection followed by a demanding functional and esthetic reconstruction of the ablative defect. We report a case of an ES developed in mandibular ramus of a 2-year-old girl with special consideration to the postmandibulectomy spontaneous structural and functional bone regeneration that made any plans for secondary mandible reconstruction unnecessary.Facial bones are rarely involved (1-2%) and when do so their involvement usually represents metastatic disease from a primary skeletal lesion.A 2-year-old girl with a 3-month history of a painless, progressively deteriorating swelling in the right mandible was referred to the Oral and Maxillofacial Department for evaluation. A fixed, hard in consistency, irregular, nontender, expansive mass of the right mandible was observed on clinical examination. No trismus or other signs of odontogenic infection were mentioned. The panoramic radiograph revealed a mixed radiolucent and radiopaque lesion with ill-defined borders extending from tooth 84 to the mandible angle. A computed tomography scan revealed a suspicious expansive lytic mass of approximately 4.0 × 4.5 cm with cortical erosion and periosteal reaction, suggesting a potential neoplastic process . MagnetiAn open incisional biopsy of the lesion was performed, showing a high-density infiltration of small, round, hyperchromatic cells. Immunohistochemical staining with positive and negative controls was also performed, revealing positivity for CD99, CD117, vimentin, BCL2, and epithelial membrane antigen. Antibodies to cytokeratin AE1/3, synaptophysin, desmin, alpha smooth muscle, osteonectin, neuron-specific enolase, CD45, S100, Myo-D1, and CD34 were not reactive with the specimen. Histopathological and immunohistochemical findings were compatible with ES/primitive neuroectodermal tumor (PNET).After diagnosis, the patient was referred to the Pediatric Oncology department for further care. Total body bone-scan revealed increased uptake in the right pelvis, which was considered as the primary ES lesion based on tumor’s demographics. The patient was assigned to vincristine, ifosfamide, doxorubicin, and etoposide (VIDE) (EURO-EWING 99) Oncology Protocol, receiving six courses of induction chemotherapy with an almost complete clinical response. A segmental mandibulectomy, extending from the neck of the right mandibular condyle to the distal edge of the right lower primitive 1st molar, was subsequently performed through a submandibular approach . The uniClinical and radiological evaluation was performed 2 years after surgery, showing no evidences of locore-gional recurrence. An imprecise reparative reaction of the healthy osseous tissue, leading to an almost complete structural and functional regeneration of the resected mandible, was reported , making 18 Pain, sensory dysfunctions, loosening teeth, or remitted fever may also be observed in some cases, increasing the suspicion of a malignant process.921 Radiographically, ES could mimic a variety of pathological entities involving the jaws, and thus, it is aptly characterized as the “great imitator of bone pathology” by many authors.22 An ill-defined, moth-eaten lytic lesion with or without cortical erosion and bone expansion is the most characteristic radiographic feature,31325 while the commonly seen long bone periosteal “onion skin” reaction is rarely encountered in jaw lesions.172627 Computed tomography scans and MRI have been well demonstrated as the most accurate approach for evaluating the extent of the bone destruction and the soft tissue involvement respectively.3724 Microscopically, ES is characterized by poorly differentiated, small, round, blue cells.232829A rapidly extensive mass of the mandible in a young-age patient is the most dominant and invariable manifestation of an ES involving the facial bones.81032 immunohistochemical analysis is crucial for the final diagnosis. Recent genetic studies demonstrated that ES shares a common chromosomal translocation   with PNETs in more than 90% of cases.14182735 The identification of this genetic abnormality, using antibodies to CD99 (Mic-2), is highly sensitive and specific for EW/PNET tumors.40 Positive staining to vimentin, CD117, BCL2, EMA, FLI1, S-100, glycogen, or pancytokeratin could further support the diagnosis.71214242941As ES has similar clinical, radiological, and histo-pathological features to various other malignancies of childhood,2742 recent multidisciplinary therapeutic protocols and improved chemotherapeutic regimens have significantly improved the 5-year survival from less than 15% to more than 75%.2134344 Combined therapy including wide surgical resection and preoperative and postoperative chemotherapy has been demonstrated as the mainstay of therapeutic approach.284546 Radiotherapy is generally avoided as it may induce secondary cancer formation or interfere with facial growth.49 Vincristine, doxorubicin, cyclophosphamide, ifosfamide, and actinomycin-D are the most widely used anticancer agents for definitive chemotherapy.5051 Demanding function and esthetic reconstruction efforts are usually required for maintaining an acceptable quality of life.Although the overall prognosis is generally poor due to the tumor’s aggressiveness and the high incidence of early hematogenous dissemination,57 In contrast with other tissues, bone heals by regeneration rather than scar formation. Patient’s age545658 and preservation of peri-osteum53545659 have been described as the main factors that favorably influence this process.However, this is not the first time that an extensive spontaneous renaissance of a postresection mandible defect has been reported.5557 Ihan Hren and Miljavec,60 studying the spontaneous healing in 33 patients with large mandible defects, demonstrated that bone-healing capacity was significantly higher (p = 0.006) in patients younger than 20 years. The fact that the cellular activity associated with sequential bone absorption and regeneration is higher in young patients, in conjunction with the presence of abundant mesenchymal cells that can differentiate into osteo-genic cells, offers a satisfactory theoretical background for this statement.545658 Periosteum is a well-recognized source of osteoprogenitor cells, and thus, its preservation is crucial in the attempt to maximize the spontaneous regeneration potential.535456 Other factors including anatomical location of the defect,60 genetic behavior,52 and infection55 have also been suggested as potential influential factors on mechanism of bone regeneration, but without adequate scientific documentation.Most cases of spontaneous mandible regeneration, reported in the literature, are in children and young individuals.63 It is widely accepted that there are three main growth sites of the mandible. The mandibular condyle is responsible for its vertical growth through endochondral bone formation. Length is gained through constant remodeling of the ramus in response to muscle forces. New bone is deposited at the alveolar process to support teeth development and eruption.6164 Preserving the condylar growth center, as in our case, could, however, minimize the potential of asymmetry or deformity.The mandible is the last facial bone that reaches skeletal maturity , and thus, surgical management of jaw pathologic lesions affecting childhood or early adolescence may restrict the mandibular growth, leading to malocclusion and facial asymmetry.The ES/PNET is a highly aggressive malignancy of childhood that rarely affects the bones of the maxillofa-cial region. As it is characterized by undefined clinical, radiological, and histopathological presentation, the immunohistochemical identification of specific chromosomal translocations is the basis of final diagnosis. Current multidisciplinary oncology therapeutic protocols have significantly improved patients’ survival rates, while maintaining high standards of quality of life.


Peri-ictal magnetic resonance imaging (MRI) findings following seizure activity are a recognized phenomenon that is not well understood . Transient changes are not usually expected to be present in postictal MRI studies because of their rarity. Here, we present a unique case of peri-ictal MRI findings located in the occipital lobe, present in a 34-year-old female with recurrent occipital seizures occurring twice in four years. MRI changes completely resolved after both episodes with no residual focal damage. The peri-ictal occipital changes on MRI in this patient are unique because they have been captured on more than one occasion. Peri-ictal MRI findings are a known phenomenon with unknown pathophysiology, although attempts have been made to understand these findings. Though the MRI findings and presentation appear to be stroke-like or PRES-like, seizures should be kept in the differential for better treatment outcomes. Peri-ictal magnetic resonance imaging (MRI) findings following seizure activity are a recognized phenomenon that is not well understood . AlthougA 34-year-old female with a history of recurrent occipital seizures presented with a four-hour history of right visual field changes associated with left hemicranial headache. She described seeing a bright spinning beach ball in her right visual field intermittently, and physical exam showed constant right homonymous hemianopia. These symptoms were similar to those associated with a previous episode of clustered seizures that happened four years ago, at which point she was prescribed an antiepileptic agent, Levetiracetam. Continuous video EEG during the patient's most recent episodes showed left occipital sharp waves evolving into brief, frequent electrographic and clinical seizures, lasting 60–70 seconds .MRI with DWI, ADC, and T2/FLAIR showed diffusion restriction and hyperintense signal throughout the left occipital cortex Figures . Given tMRI scans that were obtained four years ago showed increase in diffuse cortical and FLAIR signal abnormality involving the left occipital lobe with more focal diffusion restriction and hyperintense T2 signal medially Figures . EEG shoFollowing seizure activity, MRI is routinely ordered to look for brain structural abnormalities. Nonstructural MRI changes are rarely seen in patients with epilepsy, especially in patients with focal seizures. Partial complex seizures associated with hippocampal involvement are reported in some case series , as wellThe pathophysiology to explain abnormal peri-ictal MRI findings is unclear at this point. It is believed that peri-ictal T2/FLAIR and DWI abnormalities that can sometimes be seen in patients with seizures reflect cytotoxic and vasogenic edema, induced by seizure activity in the brain . VariousAt first glance, the brain MRI imaging suggests stroke, which may lead to stroke management erroneously. To confirm reversibility of the brain findings, follow-up MR imaging should be performed. Resolution of initial abnormal changes favors seizures, while, in stroke, residual encephalomalacia will be present. Additionally, it is important to consider the patient's clinical presentation and medical history, as well as the imaging results, before deciding on a treatment plan.Another condition with similar findings on imaging is Posterior Reversible Encephalopathy Syndrome (PRES). On MRI, lesions appear as high signal intensity on T2-weighted images and FLAIR. The lesions are typically symmetric between the right and left hemispheres, although there is asymmetry in about 28% of cases . On DWI,Evidence of recurrent peri-ictal MRI changes has rarely been reported . There hTransient changes are not usually expected to be present in postictal MRI studies because of their rarity. Therefore, we recommend follow-up MRI to confirm transient nature and to confirm that no permanent changes have occurred. No guidelines have been set regarding when to repeat imaging, but we would recommend approximately 60 days between initial and follow-up imaging. Larger studies are needed to determine the minimum time to expect reversibility of abnormalities on MRI, duration of antiseizure treatment in such rare cases, and a more detailed understanding of pathophysiology in these changes.


Paraduodenal hernias are rare congenital internal hernias accounting for <2% of intestinal obstruction. Left paraduodenal hernias (LPDHs) into the fossa of Landzert are the more common type and result from abnormal rotation of the midgut and failure of peritoneal fusion. Sequelae of these hernias usually occur spontaneously in the 4th or 5th decade of life and are more common in males and have a significant risk of incarceration and subsequent strangulation. We describe a case of a 15-year-old female who develops a LPDH following laparoscopic appendectomy, resulting in jejunal incarceration and subsequent small intestinal obstruction. The patient discussed is from an atypical demographic, being young and female. In addition, the precipitating event prompting incarceration of the hernia appears to be the application of pneumoperitoneum, placement in the Trendelenburg position, and manipulation of small intestine for the purpose of facilitating laparoscopic appendectomy. To our knowledge, this is the first reported case of LPDH exacerbated by laparoscopic procedure. Paraduodenal hernias are rare congenital internal hernias accounting for <2% of intestinal obstruction, most commonly involving jejunum . Left paA 15-year-old female presented with a 12-hour history of gradual onset right iliac fossa abdominal pain associated with nausea, vomiting, anorexia, and fevers. She denied urinary symptoms. Her last menstrual period was 3 months prior and was an irregular occurrence. She had no similar previous episodes. She had no significant past medical or surgical history and took no regular medications.On examination, she was alert, orientated, and appeared well. Vital signs were within normal limits, and she was afebrile. Abdominal examination revealed tenderness in the lower abdomen, worse in the right iliac fossa. Rovsing's sign was present.Investigations revealed a normal urine analysis, and subsequent culture was negative. White blood cell count was 19.1 × 109/L with neutrophilia. C-reactive protein was 3.0 mg/L. Abdominal ultrasound showed a noncompressible, avascular, hypoechoic tubular structure with well-defined margins measuring 9.2 mm, extending from the mid-pelvis to the left side. This was suspicious for appendicitis.The patient underwent a diagnostic laparoscopy and appendectomy. This was performed using a 10 mm infraumbilical hasson port and two further 5 mm working ports. The patient was positioned supine with bed tilted head-down (Trendelenburg position) and rolled leftward. High-flow carbon dioxide insufflation of 12 mmHg was used, and small intestine was manoeuvred out of the pelvis into the upper abdomen. Early acute appendicitis with no perforation was found, and laparoscopic appendectomy was performed. Histopathology confirmed acute appendicitis.The following day, the patient complained of severe and worsening abdominal pain. She was unable to tolerate oral intake. On examination, she appeared to be in pain. Tachycardia of 120 beats per minute was noted; otherwise, vital signs were within normal limits. Her abdomen was distended and tender with signs of generalised peritonism. Blood tests were unremarkable, with reduced white cell count compared with initial presentation. Due to this clinical picture, no further imaging studies were arranged, and she was taken back to the operating theatre for exploratory laparoscopy.Upon laparoscopy, dilated small intestinal loops and copious free fluid were found. Conversion to laparotomy was performed due to impeded laparoscopic view. This revealed a small bowel obstruction with transition point at the paraduodenal region. An LPDH diagnosed with a long segment of jejunum was seen herniating through a defect to the left of the fourth part of the duodenum and accumulating in the fossa of Landzert Figures  and 2. RInternal hernias result from protrusion of the viscera through an opening in the peritoneum or mesentery . They acPatients most commonly present in the 4th to 5th decades or life , and theThe most effective preoperative diagnostic tool is the computed tomography (CT) scan . This maTreatment methods reported in the literature include laparoscopy or laparotomy and repair. However, exploratory laparotomy is more often reported, especially in the setting of an acute complication such as strangulation, perforation, or large distension from obstruction . In addiA literature search of PubMed back to 1980 discovered only approximately 50 reported cases of intestinal obstruction secondary to a LPDH. However, to our knowledge, this is the first reported case of LPDH exacerbated by laparoscopic pneumoperitoneum and intraoperative patient positioning. This case is particularly interesting for a few reasons. Firstly, the patient demographic is not typical of this disease. Our patient was a young female, while reported cases more commonly describe males in the 4th to 5th decades of life. Secondly, the patient described no symptoms at any time prior to the day of her appendectomy. Although the patient clearly possessed the congenital anomaly of a left paraduodenal fossa, the application of pneumoperitoneum, placement in the Trendelenburg position, and manipulation of small intestine for the purpose of facilitating laparoscopic appendectomy appear to have precipitated the LPDH and subsequent incarceration. In this instance, the diagnosis was not made prior to laparotomy as in most cases presenting with an acute abdomen. Although diagnostic laparoscopy was performed upon return to theatre, gross small bowel distension from obstruction prevented further progression via the laparoscopic approach.


Bilateral papilledema secondary to obstructive or communicating hydrocephalus in patients with vestibular schwannomas is a known presentation; however, papilledema in the absence of hydrocephalus is rarely reported and its mechanism is poorly understood. We report a case of a 20-year-old woman presenting with visual deterioration and bilateral papilledema on fundoscopy. Magnetic resonance imaging (MRI) revealed a giant vestibular schwannoma with no sign of hydrocephalus. The only imaging evidence of increased pressure on preoperative imaging studies was seen on a T2-weighted MRI, where there was subtle dilatation of the arachnoid space of the optic sleeve. We presume that this patient developed papilledema by some mechanism not connected to hydrocephalus. In a young patient, papilledema may be a sign preceding hydrocephalus, or she may have had pseudotumor cerebri concomitant with her vestibular schwannoma. In either case, removal of the vestibular schwannoma solved the problem. She had complete visual recovery, irrespective of the mechanism. Vestibular schwannoma accounts for approximately 8% of all intracranial tumors and represents the most common neoplasm of the cerebellopontine angle (CPA) -3. TypicA 20-year-old female, otherwise healthy, presented for ophthalmological evaluation due to a one-month history of decreasing visual acuity and transient episodes of darkened vision bilaterally. The examination revealed bilateral papilledema. Magnetic resonance imaging (MRI) demonstrated a giant (40 mm diameter) enhancing cerebellopontine angle (CPA) tumor extending into the right auditory canal, characteristically compressing the brainstem, Hannover class T4a Figure  6]. The. The6]. The patient was referred for neurosurgical evaluation. She reported decreased hearing, dizziness, imbalance, unstable gait, and headache of several weeks duration but denied any incontinence or cognitive symptoms. On physical examination, there was right facial hypoesthesia, right peripheral facial palsy (House-Brackmann Grade 2 (HB2)), right beating nystagmus, diadochokinesis (DDK), positive Romberg sign, and dysmetria in the right extremities . A lumbaVisual acuity was 6/12 bilaterally. Fundoscopy confirmed bilateral papilledema with normal visual fields Figure . On optiSurgical procedureThe tumor was resected via a right retrosigmoid approach. The facial nerve was very adherent in the premeatal area; thus, a 10 × 10 mm “fingernail” residual was left to ensure facial nerve preservation in this young woman. The surgery was uneventful.Postoperative courseFacial nerve function was unchanged (HB2) following surgery. Postoperative head CT showed favorable resection with no sign of significant bleeding or hydrocephalus. The early and late postoperative course was uneventful except for a significant pseudomeningocele in the surgical wound. The patient was discharged home with ambulatory physiotherapy because of mild ataxia. The pseudomeningocele had contracted significantly and her ataxia improved gradually.Follow-upAt the three-month follow-up examinations, her visual acuity was normal (6/6), the papilledema had subsided on fundoscopy, and the thickness of the retinal nerve fiber layer had normalized bilaterally on OCT Figure .MRI performed three months after surgery showed a small residual plaque of tumor. There were no signs of hydrocephalus. On T2-weighted MRI, dilatation of the arachnoid space of the optic sleeve was markedly reduced and the globes had regained their normal curvature Figure . No chanPapilledema is a known sequela of elevated intracranial pressure (ICP), which generally develops due to obstructive hydrocephalus, venous obstruction, supratentorial mass effect, or pseudotumor cerebri. In spite of the large tumor diameter with significant brainstem compression, the aqueduct of Sylvius appeared patent and the ventricles were of normal size. The only imaging evidence of increased pressure was seen on a T2-weighted MRI, where there was dilatation of the arachnoid space of the optic sleeve and flattening of the posterior aspect of the globes Figure .Bilateral papilledema in this young patient may have been a relatively early sign preceding ventricular dilatation. Pressure on the brainstem would have produced a tendency for the aqueduct to occlude, with resulting resistance to normal CSF flow. ICP elevation could have served as a mechanism to maintain aqueduct patency in this case, as evident in the coronal view Figure .Harada, et al. described a 26-year-old patient with neurofibromatosis Type 2 (NF2) who presented with bilateral vestibular schwannomas associated with bilateral optic disc edema . There wMore recently, Grainger and Dias  and MatoThere was a documented resolution in the papilledema after tumor resection. Regardless of the mechanism, since the tumor was removed soon after the patient’s vision was compromised and before there was permanent damage to the optic nerve, we were not only successful in treating the papilledema, but also in facilitating recovery to normal visual acuity. We can speculate that there are two possible mechanisms here: papilledema may precede ventricular dilatation, regardless of whether it is of obstructive or absorptive origin, or the young woman could have two concomitant diseases, a vestibular schwannoma that became enlarged on the background of a formerly silent pseudotumor cerebri syndrome, causing its decompensation. It may well be that elevated intracranial pressure (ICP) contributed to the development of the postoperative pseudomeningocele, which could have played a positive role in replacing and obviating the need for a CSF diversion procedure.A 20-year-old woman presented with visual deterioration and bilateral papilledema on fundoscopy. Her MRI was characteristic for a giant vestibular schwannoma without signs of hydrocephalus. The papilledema was permanently resolved after surgery. This illustrates the possibility that visual decline and severe papilledema may be manifestations of a giant vestibular schwannoma without the signs of hydrocephalus. Another possible explanation is a mechanism similar to pseudotumor cerebri or a preexisting silent pseudotumor cerebri syndrome.


Corrosive ingestion can cause serious damage to the upper gastrointestinal tract. The acute consequences could range from mild erythema to gangrene and perforation. Delayed consequences could be strictures of the oesophagus presenting as dysphagia or gastric, pyloric and antral stenosis presenting as gastric outlet obstruction. Gastro-colic fistula as a complication of acid ingestion is a rare clinical entity. We report this case of a patient who presented with complaints of dysphagia and recurrent vomiting of foul smelling brownish vomitus two months following suicidal acid (sulphuric acid) ingestion. The presence of a gastro-colic fistula was confirmed with a barium meal examination. Corrosive ingestion is a common cause of upper gastrointestinal tract injury in India. Esophageal burns, gastric perforation and peritonitis present with acute life threatening emergencies. However, it is the delayed complications of esophageal strictures and gastric outlet obstruction that lead to considerable morbidity. The present case here is highlighting an extremely uncommon delayed manifestation of acid ingestion in the form of gastro-colic fistula which to date has been scantly reported in literature.A 45 years old unemployed man following a bout of alcohol binge presented to the hospital emergency with history of suicidal ingestion of approximately 100 ml of sulphuric acid. Examination of the oral cavity revealed superficial ulcers on the inner cheeks, the posterior pharyngeal wall and erythema of the epiglottis. There was no respiratory discomfort or hemodynamic instability. A chest X-ray and abdominal X-ray done to look for perforation was normal. After the initial resuscitation, he was taken up for upper gastrointestinal endoscopy which revealed deep ulcers of esophagus at approximately 20 cm from the incisors and scattered superficial ulcers in the rest of the esophagus along with diffuse erythema. There were deep ulcers in the antrum of the stomach extending to the pylorus along with ulcers in the mid body on the posterior surface. Duodenum was normal.The patient was managed conservatively and discharged after 10 days of hospitalization. Two months after discharge he presented with complaints of dysphagia and recurrent vomiting of brownish foul smelling liquid. An upper gastrointestinal examination revealed a tight stricture at 20 cm, which did not allow the endoscope (Pentax videoscope EG 3840) to go beyond. A barium swallow and meal follow through examination showed the presence of a 3 cm long stricture in the upper esophagus. The rest of the esophagus was normal. The stomach showed slight lack of distensibility with slight thickening of the folds in the fundus. There was very little flow of barium into the duodenum, but instead the transverse colon showed opacification along with descending colon, suggesting a possibility of gastro-colic fistula in the proximal body of the stomach from the greater curvature .Subsequent upper gastrointestinal endoscopy of the stomach done after esophageal dilatation with Savary Gilliard dilators revealed severe antral and pyloric stenosis with scarring. The endoscope could not be negotiated into the duodenum. No definite gastro-colic opening could be appreciated. A colonoscopic examination done to look for any abnormal opening in the colon also revealed no abnormal opening.A feeding jejunostomy was done and patient was planned for a second stage definitive surgery. Laparatomy was done three months later. The procedure performed was distal gastrectomy along with Billroth 1. Operative findings were pyloric stenosis, scarring and adhesions between the stomach and colon. The scarred site was probably the site of fistula, which had healed. Subsequently the feeding jejunostomy was removed. The patient is doing well and is gaining weight.Caustic ingestion can produce serious injury to the upper gastrointestinal tract . The injFistula between various abdominal organs following corrosive ingestion has been extremely rarely reported. A pancreaticocolic fistula has been seen at surgery, which was done in a patient having extensive burns of the upper gastrointestinal tract following acid ingestion . Gastro-