Introduction: The presentation of eosinophilic myenteric ganglionitis (EMG) can be similar to that of Hirschsprung's disease (HD). In a limited number of cases of pediatric patients, the diagnosis of both EMG and HD are reported. A case of pseudo-obstruction in EMG occurring in a child with HD diagnosis is discussed with literature review.Case Presentation: A boy aged 2 years and 6 months presented with intractable constipation and abdominal distension. Histological HD diagnosis was carried out and transanal Soave pullthrough was performed. At the age of 3 years and 2 months, an infectious enterocolitis occurred. One month later, he presented with constipation, marked abdominal distension and melena. Full thickness colonic biopsies revealed eosinophilic myenteric ganglionitis. Specific IgE tests were positive for several foods. Dietary exclusion was adopted with resolution of clinical symptoms and histologic remission.Conclusion: EMD may occur in patients with HD. At the onset, EMD may be associated with functional intestinal obstruction. The use of an elimination diet proved effective for the relief of symptoms. Long term follow-up is mandatory to define the timing of the reintroduction of foods. Eosinophilic myenteric ganglionitis (EMG) is an inflammatory neuronal enteropathy, characterized by infiltration of the Auerbach myenteric plexus by eosinophils, which develops mainly in children . EMG canHirschprung's disease (HD) is the most common cause of congenital intestinal dysmotility due to aganglionosis in the colon, rectum, and possibly the small intestine, which leads to abnormal intestinal motility or obstruction. The presentation of EMG can be similar to that of HD .In a limited number of pediatric patients, the diagnosis of EMG and concomitant HD have been reported , 3.A case of pseudo-obstruction in EMG occurring in a child with HD diagnosis is discussed along with a pediatric literature review.A boy aged 2 years and 6 months presented with refractory constipation and abdominal distension and was admitted to the Pediatric Surgical Unit. He had passed meconium within 24 h of birth in the postnatal ward with no signs of abdominal distension; progressively he developed chronic constipation.At the clinical evaluation, his abdomen was found to be tender and distended, although quiet. Abdominal radiography showed a distal obstruction . SuctionAt the age of 3 years and 2 months, an infectious enterocolitis (C. Difficile) confirmed by the finding of the specific toxin by microbiological laboratory testing occurred and an antibiotic treatment was administered, with resolution of symptoms.One month later, he presented with marked abdominal distension and melena; occasional episodes of vomiting was also reported. Abdominal obstruction was detected at radiography . Upper eThe patient had peripheral eosinophilia (8%) and an elevated age-related IgE concentration (423kU/l). A panel of specific IgE tests (immuno-CAP system) was then performed, showing sIgE positive for casein, α-lactalbumin, β-lactoglobulin, egg white, wheat, and maize.Avoidance diet for cow's milk proteins, egg, wheat and maize was then prescribed. An elemental formula was introduced as a milk substitute; the symptoms resolution was observed.After 3 months of dietary modification, colonoscopic mucosal biopsies showed a mild chronic inflammatory cell infiltrate of lymphocytes in rectal tissue, with evidence of histologic remission of intestinal eosinophilic infiltrate  .At 15 months from the start of the diet, colon biopsies showed a complete resolution of esosinophilic infiltrate and the child's well-being persisted.Intestinal pseudo-obstruction is characterized by the impairment of gastrointestinal propulsion of the gut content in the absence of obstructive lesions . It may A limited number of cases has been described of different presentations of acquired and reversible functional intestinal obstructive syndrome in association with eosinophilic ganglionitis and eosinophilic colitis in pediatrics. Schappi et al.  describeThe presence of EMG with concomitant HD is rarely reported in pediatrics. Phillips et al. described a neonate with eosinophilic infiltrates extending into the aganglionic segments of the bowel . LowichiThe pathogenesis of EMD has yet to be fully elucidated. As reported paraneoplastic syndrome, infection, or primary neurologic disorders have been considered as factors involved in EMG ; in our EMD diagnosis is made based on symptoms and histological examination results. Two-third of patients had symptoms, mainly diarrhea and abdominal pain, poor feeding, vomiting and more rarely, as in this reported case, a pseudo-intestinal obstruction may occur; . PathognSurgical resection of the aganglionic intestinal segment and restoration of intestinal continuity is the gold standard for HD treatment. In some patients with HD, postoperative intestinal dysmotility persists, most often manifested as chronic constipation and recurrent episodes of enterocolitis . PostopeTo our knowledge, only one case of EMG which responded to dietary modification alone has been described . In contIn summary, we described the first case of postoperative infectious enterocolitis due to EMG occurring in HD. At the onset, EMG may be associated with postoperative functional intestinal obstruction, secondary to mechanical damage as a consequence of eosinophilic infiltration of the intestinal musculature . DietaryThe raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.Ethical review and approval was required for the study on human participants in accordance with the local legislation and institutional requirements. Written informed consent to participate in this study was provided by the participants' legal guardian/next of kin.ED'A performed allergological assessment, baseline evaluation and follow up. ED'A and VC wrote and supervised the manuscript with input from all of the authors. MM, LM, and GS performed the surgical interventions. VV and MN performed the histological evaluations and supervised the manuscript. GP supervised the manuscript. All authors discussed the results and approved the final version of the manuscript.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


Synovial sarcoma (SS) is one of the reported sarcomas in the pediatric and adult populations. Delay in diagnosis and treatment is common in SS cases. SS may be excised before the correct diagnosis is made.we present a case involving a 4-year-old boy who visited our service with complaints of left knee pain and limited knee flexion. Initially, the child was diagnosed with osteochondromatosis. Surgical excision was opted, and initial histopathological examination revealed a fibrous histiocytoma. The slide and blocks were then brought to the King Faisal Specialist Hospital Research Center (KFSH&RC) and histopathologic analysis has shown a well-circumscribed nodule in the synovium with a sub-synovial monomorphic spindle cell sarcoma, confirmed by fluorescence in situ hybridization (FISH).Therefore, we strongly recommend considering all differential diagnoses for soft-tissue masses when planning surgical management. Synovial sarcoma (SS) is one of the sarcomas which reported in the pediatric and adult populations . The peaAlthough it can occur anywhere in the body, SS commonly arises in soft tissues adjacent to large joints of the upper and lower extremities . The symRadiological examinations, such as plain X-ray, computed tomography (CT), and magnetic resonance imaging (MRI) are the “first-line” examinations used to evaluate SS. However, SS is definitively diagnosed by histological examination of a biopsy sample.Despite its name, SS does not develop from synovial tissue . SS was There is a strong relationship between the histologic subtype of the tumor and either of these two fusion proteins. The majority of the SYT-SSX2 tumors present a monophasic phenotype involving only a spindle cell component , while aThe treatment of SS depends on several factors. Surgical excision is the mainstay of treatment. We present a case of SS in a 4-year-old child who was initially misdiagnosed. The case adheres to CARE guidelines .The patient’s parents were informed that data concerning the case would be submitted for publication, and they provided consent for the same. A 4-year-old boy with global developmental delay and bronchial asthma presented to a private clinic with complaints of left knee pain and limited knee flexion. This pain was irregularly experienced on movement. He mainly showed localized tenderness over the anterolateral aspect of the left knee.Initial knee radiographs revealed no remarkable findings Fig. . ComputeAfter discussion with the family, we opted for surgical excision through a longitudinal incision. We observed a pedunculated 2 × 2 cm small lesion resembling a blood clot arising from the border of the lateral femoral condyle Fig. ; the lesThe pathologist at that clinic thought that the lesion was benign and labeled it as a fibrous histiocytoma. He had performed limited staining; therefore, we brought the slide and blocks to the King Faisal Specialist Hospital Research Center (KFSH&RC). While the lesion was reported to be 2 × 2 cm in size, the whole resected surgical specimen measured 2 × 1.5 × 0.7 cm. Histopathology revealed a well-circumscribed nodule in the synovium with sub-synovial monomorphic spindle cell sarcoma with brisk mitotic activity in the hematoxylin and eosin stained sections. The tumor cells showed patchy cytoplasmic positivity for cytokeratin 19 and diffuse nuclear positivity for transducin-like enhancer of split 1 , and non-contrast CT of the chest was performed. No evidence of metastatic disease was observed. Increased abnormal fluorodeoxyglucose (FDG) uptake was noticed at the site of the known primary tumor, indicating postoperative inflammatory change.The patient was evaluated at the sarcoma clinic at 2 and 6 weeks, and at 3 months thereafter. Clinical, radiological, and laboratory evaluations were performed at each follow-up. The patient was followed-up for a year with no evidence of local recurrence or metastatic disease on surveillance MRI and CT scans of the chest, abdomen, and pelvis. These were done 3 times, and the final PET-CT scan was with no significant FDG avid residual/recurrent or metastatic disease. So, our plan was not to do unless there are clinical symptoms.Delay in diagnosis and treatment is common in SS cases . SS may Since the synovial membrane covers a large part of the knee, the intra-articular lesions of the knee are common. Lesions in the knee joint are commonly benign and arise from inflammatory or degenerative articular disease . HoweverHere, we present an unfortunate scenario where a mesenchymal tumor that was potentially aggressive was incorrectly diagnosed as a benign tumor preoperatively. This pitfall is a great learning point for those who make daily decisions regarding surgical treatment. Such lesions should be diagnosed systematically. The location of the lesion must be ascertained, whether it is intra or extra-articular. The anatomical structure involved must then be defined.Synovial osteochondromatosis, pigmented villonodular synovitis (PVNS), or synovial hemangiomas could be differential diagnoses for tumor-like lesions in the knee joint. While MRI plays a major role in the evaluation and description of intra-articular lesions of the knee, distinguishing intra-articular synovial sarcomas from benign tumors is challenging since no specific radiological features of SS have been established . SS is hMoreover, the other main concern in this report was the incorrect diagnosis by the pathologist in the private center. However, the incorrect diagnosis had not affected our final management since complete resection with clear margins remains the treatment of choice for localized SS in children . AlthougAlthough the use of adjuvant radiotherapy and/or chemotherapy was suggested for children who undergo surgery , the oncTumor stage, grade, size and male sex have adverse prognostic significance in SS, and the impact must be confirmed in prospective studies . HoweverOur report emphasizes the importance of teamwork since the patient was misdiagnosed initially although appropriate diagnostic evaluations were performed. Therefore, we strongly recommend considering all differential diagnoses for soft-tissue masses when planning surgical management. Therefore, selection of proper diagnostic modalities, careful assessment of biopsy samples, and adequate surgical excision are essential for SS treatment.


Nonocclusive mesenteric ischemia (NOMI) has been reported to be a life-threating disease. Gastric conduit necrosis is known as a critical postoperative complication after esophagectomy for esophageal cancer. We encountered a rare case of NOMI of a wide area of the intestine accompanied by gastric conduit necrosis after esophagectomy, which was successfully treated with an emergency operation.A 67-year-old man presented with dysphagia. He was diagnosed with middle thoracic advanced esophageal cancer. After neoadjuvant chemotherapy, he underwent subtotal esophagectomy with lymphadenectomy and gastric conduit reconstruction. On postoperative day (POD) 2, he had diarrhea, high fever, and low blood pressure, which were treated with catecholamines. Laboratory data revealed acidosis and severe sepsis with multi-organ failure, including the kidneys. Although enhanced computed tomography did not exhibit definite findings of bowel ischemia, upper gastrointestinal endoscopy revealed necrotic mucosal changes in the whole gastric conduit. Therefore, we made a diagnosis of septic shock caused by gastric conduit necrosis and performed an emergency operation. When we explored the abdominal cavity, we found not only gastric conduit necrosis but also intermittent necrotic changes in the intestinal wall from the jejunum to the rectum. Therefore, NOMI was diagnosed. We performed an excision of the gastric conduit and 2 m of the small intestine, as well as total colectomy. After the second operation, prostaglandin E1 was administered intravenously as the treatment for NOMI, and sepsis was improved. On POD 122, he was self-discharged. He died of recurrence of lung metastasis from the esophageal cancer 9 months after the first operation.When a patient has a critical status, including severe sepsis or severe acidosis, after esophagectomy, we should consider the possibility of NOMI in addition to gastric conduit necrosis and aim to diagnose and treat it immediately with an urgent operation. Nonocclusive mesenteric ischemia (NOMI) causes ischemia of the mesentery due to absence of mechanical obstruction of the mesenteric vessels by thrombus or embolism –3. NOMI A 67-year-old Japanese man presented with dysphagia. He had a medical history of exploratory laparotomy for ileus but no history of anticoagulant medication. Upper gastrointestinal (GI) endoscopy revealed type III esophageal cancer in the middle thoracic esophagus Fig. a. An uppAs a result, he was diagnosed with middle thoracic esophageal cancer (squamous cell carcinoma) and T3N3M0 in clinical stage III according to the criteria of the Japan Esophageal Society. He was treated with combination chemotherapy of docetaxel, cisplatin, and fluorouracil as neoadjuvant therapy. Chemotherapy for 2 months resulted in shrinkage of the primary tumor of the esophagus and lymph nodes in the mediastinum Fig. e–g. FDG-After chemotherapy, he underwent subtotal esophagectomy with three-field lymphadenectomy and gastric conduit reconstruction through the retrosternal route by thoracoscopy and laparotomy. Postoperative care was performed under mechanical ventilation in the intensive care unit (ICU). On postoperative day (POD) 1, mechanical ventilation was terminated because of a good respiratory condition. However, he suffered from dyspnea and severe acidosis and chest X-ray examination revealed right pneumothorax, which was observed 5 h after extubation. Furthermore, air leakage was observed from the water seal chamber of the chest drain system. We thought that the respiratory disorder was caused by the right pneumothorax. Therefore, mechanical ventilation was started again. After that, it became clear that the cause of the pneumothorax was the air sucked from his drain insertion site. A few sutures were added at his drain insertion site. And we decided to continue his treatment under mechanical ventilation until the respiratory problems were improved.On POD 2, the pneumothorax was improved. However, he had diarrhea, fever, and low blood pressure, which were treated with catecholamines. At the time, his abdomen was flat and soft. Laboratory findings revealed worsened inflammation with a decline in the white blood cell count, deviated enzyme levels, renal failure, and acidosis Table . All thiThe pathological findings revealed shedding of the epithelium, hemorrhagic necrosis of the mucosa, capillary dilation of the submucosa, and transmural necrobiosis in the gastric conduit, small intestine, and large intestine, which were compatible with NOMI. Almost all of the lesions had no thrombosis in the resected specimens. After the second operation, sepsis was improved. Furthermore, 0.01 μg/kg/min of prostaglandin E1 had been administered intravenously for a week, and enhanced CT findings revealed no ischemic changes in the residual small intestine after the second operation. On POD 16, the patient was discharged from the ICU. On POD 122, he was self-discharged from our hospital after a long period of nutritional support, including total parenteral nutrition and enteral nutrition.The pathological diagnosis of esophageal cancer was squamous cell carcinoma and pT3N1M0 stage III. On POD 64, follow-up CT revealed the recurrence of esophageal cancer at the para-aortic lymph node. Although, we considered re-reconstruction of the esophagus, we abandoned this strategy, because his condition could not tolerate surgical stress, and recurrence of esophageal cancer was confirmed. He died of the recurrence of lung metastasis from esophageal cancer 9 months after the operation.We report a rare case of nonocclusive mesenteric ischemia of a wide area of the intestine accompanied by gastric conduit necrosis following esophagectomy, which was treated successfully with an emergency operation.Previously, to our knowledge, NOMI following esophagectomy was reported in only 12 cases (Table As presented in Table Regarding treatment of the previous 12 cases of NOMI following esophagectomy, an emergency laparotomy was performed in 10 cases, and nonsurgical treatments, including intra-arterial or intravenous infusion of papaverine, were performed in two. Previous reports revealed that management of NOMI was initially needed improvement of hemodynamics and exclusion of vasoconstrictors . AdditioAmong the reported cases, 5 of 12 patients died during the early postoperative periods. The widespread intestine was resected in many of the terminal cases. Resection of a large part of the small and large intestines was also required in our patient. Nevertheless, our patient was alive after the surgery for NOMI. The reasons might be that an adequate area was excised and that the necrotic lesion of the small intestine was narrower than that in the previous report.Furthermore, only in our patient, NOMI was accompanied by gastric conduit necrosis following esophagectomy. To our knowledge, this phenomenon was not reported in other cases. Gastric conduit necrosis is also a rare critical complication following esophagectomy , 5. In aWe encountered a case of NOMI accompanied by gastric conduit necrosis following esophagectomy, and the patient was treated with adequate resection of the necrotic digestive tract and provided with intensive care. When a patient is in critical status, including severe sepsis or severe acidosis, following esophagectomy, we should consider the possibility of NOMI in addition to gastric conduit necrosis and aim to diagnose and treat it immediately with an emergency operation.


Thus far, only a single case describing an atypical appearance of elastofibroma dorsi at a prior thoracotomy site has been published in the literature. We describe a series of three cases recently imaged at our institution with the same atypical appearance, in order to highlight and increase recognition of this more unusual morphology in post-operative patients. Elastofibroma dorsi are benign reactive masses with characteristic mixed fatty and soft tissue morphology and subscapular location that are typically readily recognizable on ultrasound, CT and MRI of the chest. Widened intercostal spaces at thoracotomy sites, however, may result in elastofibroma formation, enlargement and medial herniation into the thoracic cavity. The resulting atypical appearance may present a diagnostic challenge on imaging, and may be mistaken for aggressive processes, precipitating unnecessary work-up or even resection.A 79-year-old asymptomatic female with a history of right upper lobectomy 10 years earlier for a Stage 1A adenocarcinoma of the lung had a unilateral right chest wall mass noted on chest CTs performed at an outside institution . The finAn 80-year-old asymptomatic female patient with history of left upper lobe wedge resection for atypical adenomatous hyperplasia 13 years earlier had a unilateral left-sided chest wall soft tissue mass noted on post-operative chest CTs  performeAn 84-year-old asymptomatic female underwent a left lower lobe wedge resection for typical carcinoid tumor 9 years earlier. Post-operative chest CTs  were ini1 thought to result from repetitive mechanical stresses on the chest wall soft tissues due to friction between the scapula and ribs. Lesions are often asymptomatic and only detected as an incidental finding on imaging, however, larger lesions may manifest as a chest wall mass on physical exam, and approximately 50% of patients may complain of a clunking sensation or local swelling on movement.2 Although initially described as rare, with a reported prevalence of 2% on one CT study,3 autopsy series have shown an incidence of up to 24% of females and 11% of males older than 55 years of age,3 and pre-elastofibroma changes (defined as a few or many degenerated elastin fibers) identified in up to 81%.4 The true incidence of elastofibroma may be underestimated on imaging, as lesions may be overlooked when small or symmetric, given the often similar morphologic appearances to normal chest wall soft tissues. Imaging findings are in general pathognomonic, showing a subscapular mass with variable mixed fatty and soft tissue on ultrasound, CT or MRI,5 with no need for biopsy for typical appearances, and surgery only reserved for symptomatic cases. The incidence of bilaterality has a wide reported range of 10–60%6 ; although a contralateral lesion effectively excludes a malignant process, a unilateral lesion with typical appearances should therefore not raise concern. In Case 1, the avid enhancement on MRI was interpreted as a suspicious finding, however, radiologists should be aware that highly variable enhancement patterns are recognized, including marked enhancement,6 and should not raise concern in a lesion with otherwise typical morphology and location for an elastofibroma. The healthcare costs and morbidity associated with the repeated chest CT follow-up, chest MRI, biopsy, two separate surgeries, inpatient stay, and post-operative recuperation for this particular case were not insignificant, for an incidental benign lesion in a previously asymptomatic elderly patient.Elastofibroma dorsi are benign reactive masses composed of variable amounts of fibroblasts, collagen, elastin fibers and fat,In two of our cases, the elastofibroma dorsi were not present pre-operatively, but developed at the widened rib interspace after an interval of several years following surgery, and subsequently enlarged at follow-up. Widened rib interspaces at thoracotomy sites may therefore have an etiologic role in both elastofibroma formation and enlargement, by creating a deeper pocket medial to the scapula with altered dynamic mechanical stresses on the chest wall tissues.7 Iwata et al also published a case report of an enlarging, morphologically identical mass at a thoracotomy site in an elderly female patient, with pathology demonstrating muscle and connective tissue, and ascribed the finding to “an inverted intercostal hernia”8 ; however, as that entity has no other descriptions in the literature, and the history and imaging findings were identical to our three cases, it is the authors’ opinion that this was much more likely an unrecognized elastofibroma, lending further support that this atypical appearance provides a diagnostic challenge.A literature search reveals only one other published case of an atypical presentation of an elastofibroma dorsi indenting into the thoracic cavity through a widened rib space following thoracotomy in a middle-aged female patient, involving a lesion that was already present pre-operatively.In conclusion, in addition to the already well-described imaging radiologic appearances of elastofibroma dorsi, lesions may also develop, enlarge or herniate medially at widened intercostal spaces following thoracotomy. The finding of three cases with identical CT appearances all imaged within the last year at our institution would support that this may be a less rare constellation of findings than already reported in the literature. Recognition of this more unusual appearance of elastofibroma dorsi by radiologists, clinicians and surgeons should obviate the need for other costly imaging tests, follow-up, biopsy or surgical resection in patients with this incidental benign finding.Elastofibroma dorsi may develop, enlarge and herniate medially at widened rib spaces in post-thorcotomy patients, particularly in elderly females.Avid gadolinium enhancement is one of several recognized enhancement patterns in elastofibroma dorsi and should not be interpreted as suspicious in a lesion with typical morphology and location.Biopsy, follow-up and resection are typically not required for incidental, asymptomatic elastofibroma dorsi.


Giant intra-abdominal cystic lesions are seldom encountered and can post a diagnostic challenge pre-operatively. These often present as increasing abdominal size and from its mass effect.Here, we present a case of a 58 year-old gentleman with worsening bloating and abdominal distension. A contrasted CT scan of the abdomen revealed a giant intra-abdominal cyst with no definite organ of origin. He underwent a laparotomy and excision of the giant cyst which was not found to be attached to any organ or mesentery. This resulted in resolution of his symptoms and a drastic improvement in his appetite.It is often difficult to identify the origin of giant intra-abdominal cysts as pre-operative imaging may show it abutting multiple organs due to its size. Common intra-abdominal cysts include mesenteric, ovarian or peritoneal cysts. A precipitating history such as pancreatitis or surgical implants can suggest pseudocysts. Surgical excision alone is curative but can be difficult due to the size. Controlled intra-operative aspiration can aid in visualization and dissection.Giant intra-abdominal cystic lesions cause significant discomfort due to its mass effect. Pre-operative investigations may not identify its cause. Surgical excision is recommended to diagnostic and therapeutic purposes. This is due to the availability of radiological imaging that allow for an earlier diagnosis at a smaller size. Intra-abdominal cysts may remain asymptomatic until they achieve a certain size of which the patient may encounter non-specific complaints such as bloating and abdominal distension. The majority of abdominal pseudocysts occur as a result of a ventriculoperitoneal shunt or as sequelae of pancreatitis. Here, we present a case of a giant intra-abdominal pseudocyst with no risk factors or contributory cause and which represented a diagnostic dilemma. The case is reported in line with the SCARE criteria .2A 58 year-old gentleman presented to the hospital with worsening bloating and a gradual increase in his abdominal girth. He had also noted a loss of weight of more than 10 kg over the last 2 years. The patient otherwise denied any abdominal pain or change in his bowel habit. He was known to have a history of well-controlled diabetes mellitus, hypertension, hyperlipidaemia and atrial fibrillation. There was no previous history of pancreatitis or abdominal surgery. The patient had recently undergone a gastroscopy and colonoscopy the previous year for iron deficiency anaemia. This had shown gastritis as well as the presence of pandiverticular disease and a sub-centimeter colonic polyp. Histology showed the polyp to be a tubular adenoma with low-grade dysplasia , Fig. 6.Clinical examination showed an adequately nourished gentleman but with a large abdominal mass occupying most of his abdomen. It was possible to feel over the superior edge but the inferior edge extended into the pelvis. The mass was non-tender on palpation. Digital rectal examination was unremarkable.In view of the above findings, the patient underwent a computed tomography (CT) of the abdomen and pelvis. This showed a large 25 × 17 × 22 cm cystic lesion extending from the mid-abdomen to the pelvis. The lesion was thin walled and contained homogenous low density fluid (14 Hounsfield unit). There was no septations, irregularity or abnormal thickening of the cyst wall. The cyst was noted to have a mass effect but not invading the surrounding bowel loops and the urinary bladder. It was found to be separate from the liver and the kidneys. The pancreas was normal in appearance. The CT scan was otherwise unable to identify the origin of the giant cyst.As this was a thin walled cyst with no irregular or solid component, a fine needle aspiration (FNA) was not suitable as there was no specific area to target. Aspiration of the fluid was also unlikely to yield any meaningful finding for diagnosis. Further imaging such as a MRI would also not help in identifying the origin or the diagnosis of the cyst. The possibility of a mesenteric or omental cyst was therefore discussed and surgical excision was offered to the patient. Tumour markers were not performed as it would not have affected the management.An elective exploratory laparotomy was performed via a midline incision. A giant cyst was immediately identified but was found to have multiple adhesions to the peritoneum, omentum, mesentery, urinary bladder as well as the small and large intestines. However, the cyst was not found to be originating from any of the above organs or the vas deferens. The cyst was entirely within the abdominal cavity and did not originate from within the mesentery. A controlled decompression of the cyst was made through a purse-string encircled incision in the anterior wall. This was performed to aid with retraction and visualization of the posterior surface. Thick purulent fluid was aspirated until dry. The cyst was subsequently excised in whole.The patient underwent an uneventful recovery with a brief period of expected post-operative ileus. He was discharged on post-operative day 6. Follow-up visits showed complete resolution of his initial symptoms and a vast improvement in his appetite.Streptococcus agalactiae. Fungal culture and tuberculosis polymerase chain reaction (TB PCR) tests were negative. Cytology of the fluid showed mainly neutrophils.Culture of the fluid was positive for Histological examination of the cyst showed thick fibrous walls covered with coarse fibrillary strands admixed with fibrin. There were also large numbers of mature IgG plasma cells with aggregates of neutrophils and scattered lymphocytes. No viable epithelial lining was identified. The walls stained positive for AE1/3 suggesting myofibroblasts. They were negative for CD117 and DOG-1 and therefore not suggestive of a gastrointestinal stromal tumour.This was therefore treated as a benign cyst of an undetermined origin.The patient was last seen in clinic two months after his surgery with a significant improvement in his appetite and oral intake. He was thereafter discharged from follow-up as the likelihood of recurrence was low considering that the cyst had been excised entirely with no remnant wall left behind.3GIC are seldom encountered in the recent times as they are often picked up at a smaller size due to the ease of medical access and imaging. Patients tend to present with non-specific symptoms such as abdominal fullness, distension, change in bowel habit, pain or nausea and vomiting ,3. TheseDiagnosis of a cyst or pseudocyst is difficult to ascertain pre-operatively as tissue biopsy or cytology can be difficult to attain. Ultrasound or CT scans done pre-operatively are a useful guide if the cyst can be seen to be arising from or located within an organ . HoweverAbdominal cystic lesions can be broadly categorized as cysts or pseudocysts. Large cysts are uncommon and are often ovarian , mesenteric  or peritoneal  in origin and can be benign or malignant . MesenteSurgical excision of the cyst is sufficient for treatment. However, dissection of large cysts can prove difficult especially in the deep posterior surface where the view is limited and retraction limited due to the size and weight. In this case, the author encountered difficulty freeing adhesions between the cyst and the intestines safely. In order to aid visualization, the cyst was decompressed under control. This drastically improved the surgical view and allowed the cyst to be easily retracted for adhesiolysis.4All patients with GIC should be offered surgical excision in view of the mass effect. Surgical excision alone is sufficient and will allow for resolution of the symptoms. The cystic lesion should be sent for histology for definite diagnosis.The author declares there was no funding involved.No ethical approval was required as the author’s institution does not require approval for case reports of less than three patients. Consent was also obtained from the patient and the patient is not identifiable from the case report.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Nern Hoong Kao was involved in all stages of the article.N/A.Nern Hoong Kao.Editorially reviewed, not externally peer-reviewed.The author declares no conflict of interest.


Rasmussen's encephalitis (RE) is a rare progressive inflammatory disease of the central nervous system. It is characterized by unilateral hemispheric atrophy, pharmacoresistant focal seizures, and progressive neurological deficit. The exact etiopathogenesis still remains unknown. Brain imaging plays an important role in diagnosis and follow-up. Fluctuation of lesions in brain imaging was reported in few cases. Herein, we report an additional pediatric case of Rasmussen encephalitis with fluctuating changes in brain MRI. Rasmussen's encephalitis (RE) was first described in the late 1950s. It is a rare neurological disease of childhood characterized by unilateral hemispheric atrophy, pharmacoresistant focal seizures, and progressive neurological deficits. The exact etiopathogenesis still remains unknown. Brain imaging plays a pivotal role in diagnosis and control of disease progression. Few cases with atypical MRI features of RE represented by improvement and reoccurrence of signal abnormalities were reported.We report on the clinical, electrophysiological, and imaging data of an additional pediatric case of RE with atypical MRI features.An 11-year-old boy was born to nonconsanguineous parents. He had a family history of febrile seizures in his sister, epilepsy in 2 cousins, and ulcerative colitis in his mother. He had no significant antenatal and perinatal history. Psychomotor development was normal. He was treated for adrenal insufficiency and dysthyroïdism. On August 2014, he was referred to our department, at the age of 11, with focal clonic right-sided seizures, which were preceded by gastroenteritis 1 month ago. Neurological examination showed right hemidystonia, myoclonia, right pyramidal syndrome, and right hemihypoesthesia. Interictal electroencephalogram (EEG) showed left frontotemporal discharge persisting during sleep . Brain mWe report the case of an 11-year-old boy presenting with focal seizures. The diagnosis of Rasmussen's encephalitis (RE) was made due to the clinical and radiological findings. Our patient illustrates a rare case of RE with fluctuating signal abnormalities on brain MRI.RE is a progressive chronic inflammatory disease of the central nervous system. It was first reported by Theodore Rasmussen in 1958 , 3. It iBrain MRI is an important tool for diagnostic assessment and follow-up in RE , 5. The In early disease stages, electroencephalograms (EEG) may contribute to the diagnosis of RE. Various abnormalities are seen in patients with RE. Some unihemispheric findings such as impairment of background activity with persistent polymorphic delta waves and sleep spindles, focal slow activity, subclinical ictal discharges, and multifocal ictal discharges are strongly suggestive of RE , 3. The Brain biopsy can also help the diagnosis, but it is not required in all RE cases. The characteristic histopathological features are microglial and lymphocytic nodules, neuronal loss, neuronophagia, and perivascular cuffing, confined to one cerebral hemisphere with frontoinsular predilection , 5.RE can be treated by antiepileptic drugs, immunosuppressive and immunomodulator regimens, and surgery. The aim of these treatments is to reduce seizure severity and improve the motor and cognitive performance , 5. FreqIntravenous immunoglobulin (IVIG) was used in some patients having RE with good results. The recommended dose is 2 g/kg monthly. The association of steroids and IVIG may be indicated when the two treatments alone are ineffective .Plasmapheresis has good effects on seizures and neurological functions. It contributes to assessing the mental and residual motor function before surgery. The frequency was three to six single volume exchanges on consecutive or alternate days every 2 to 8 weeks .Other medical treatments, such as using tacrolimus, rituximab, cyclophosphamide, azathioprine, and interferon, have been reported . Our patSurgery  seems to be the only cure for the seizures and to improve cognitive outcome. However, inevitable sequelae  should be considered . RehabilRasmussen's encephalitis is a progressive inflammatory disease of one cerebral hemisphere characterized by frequent focal seizures, hemiparesis, and mental deterioration. Brain imaging findings, associated with electroencephalogram and clinical data, may indicate early diagnosis and could be an indicative of prognosis. Knowledge of atypical radiological aspects is necessary in order to establish the right diagnosis. Rapid diagnosis and management can modify the progression of disease.


However, it should be noted that this tumor can be detected in older age groups as well.•Although extremely rare, osteoid osteoma may occur in the hallux.•CT is the method of choice in the diagnosis of osteoid osteoma.•Osteoid osteoma should be included in the differential diagnosis list in all patients presenting with foot pain. Osteoid osteomas are benign osteoblastic bone tumors mostly seen in patients in the second or third decade of life, and they most frequently involve the femur and tibia. Hallux osteoid osteoma is an extremely rare occurrence with only 14 reported cases to date.A 46-year-old woman with a right hallux pain for the last 18 months was admitted. Her foot radiographs showed a small sclerotic focus on the distal phalanx and degenerative changes in the interphalangeal joint of the hallux. The complaints of the patient were attributed to osteoarthritis involving the interphalangeal joint and non-steroidal anti-inflammatory drugs were administered; however, the patient’s condition did not improve, and hallux osteoid osteoma could not be correctly diagnosed until a CT scan was performed.We present a case of hallux osteoid osteoma in which the diagnosis was delayed greatly due to the unusuality of the lesion location, the advanced age of the patient, and the uncertainty of the clinical and radiographic appearance.Although rare, osteoid osteoma may occur in the hallux. Even if the patient age, pain pattern, and radiographic findings do not exactly meet the classical definitions for osteoid osteoma, this tumor should always be included in the differential diagnosis list in patients presenting with foot pain. Most cases are reported to occur in the second or third decade of life . The mos2A 46-year-old woman was admitted to the Department of Orthopedic Surgery with the complaint of a right hallux pain for the last 18 months. She had been to a doctor several times, but she had not benefited from the prescribed non-steroidal anti-inflammatory drugs (NSAID). The pain was occurring after long periods of standing and fatigue. There was no history of trauma. Anteroposterior and lateral oblique radiographs of the right foot revealed a cancellous, oval-shaped opacity of 7 × 3 mm in the distal phalanx of the hallux that was interpreted as a bone island or a sclerotic focus of a healed benign bone lesion. A joint space narrowing accompanied by periarticular sclerosis and osteophyte formation in the interphalangeal joint of the hallux was evident in radiographs . The patTwo months later, the patient was admitted with a progressed and worsened right hallux pain that was unresponsive to the NSAID medication. This time, the finger was swollen with obvious erythema and tender to palpation . The patFollowing the diagnosis, salicylate treatment was started immediately and a significant decrease was recorded in the patient's complaints. A percutaneous radiofrequency ablation under CT guidance was planned for the treatment of the lesion; however, because the patient feared the planned intervention and was significantly less complaining under salicylate, she requested that the intervention be postponed.3We presented a case of hallux OO in which the diagnosis was delayed greatly due to the unusuality of the case in terms of the lesion location and the patient age as well as the uncertainty of its clinical and radiographic appearances.Foot and ankle OOs constitute 2–11% of all OOs with a predilection for the talus and calcaneus ,6. HalluOO is classically known as a tumor of childhood and adolescence . AccordiOOs typically present with pain that worsens at night and diminishes under salicylates. Swelling and erythema in the affected region may also be present in the cases with OO ,9; howevRadiography, CT, MRI and bone scintigraphy are the imaging modalities used for the diagnosis of OO ,6. The tAlthough surgical excision is the traditionally adopted treatment of OOs, imaging-guided laser and radiofrequency ablation have gained significant popularity in recent years ,12. CT-g4Although rare, OO may occur in the hallux. Even if the patient age, pain pattern, and radiographic findings do not exactly meet the classical definitions for OO, this tumor should always be included in the differential diagnosis list in patients presenting with foot pain.This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.This article does not include research involving patients, so it does not require ethical approval.Informed consent for publication was obtained from the patient.MÖ: Acquisition of data, analysis and interpretation of data, drafting the article, final approval of the version to be published.RPK: Acquisition of data.BDŞ: Final approval of the version to be published.ED: Analysis and interpretation of data.All authors read and approved the final manuscript.N/A.Meltem Özdemir, MD.Not commissioned, externally peer-reviewed.The authors declare that they have no conflict of interest.


Among various adrenal tumors, metastatic ones are the most common. PET/CT scanning facilitates early detection. Occurrence of isolated and synchronous metastasis is very rare and poses serious diagnostic and therapeutic challenges. Among various adrenal tumors, metastatic ones are the most common. PET/CT scanning facilitates early detection. Occurrence of isolated and synchronous metastasis is very rare and poses serious diagnostic and therapeutic challenges. Left adrenal resection confirmed the adrenal metastasis.A 76‐year‐old man was found with a 12.3 cm, hormonally inactive, left adrenal mass by abdominal CT. FDG PET/CT scan showed a malignant adrenal tumor (SUVMAX uptake: 18 and revealed an increased SUVMAX: 5,9 of a 1.1 cm nodule at the upper right lung field (Figure A 76‐year‐old man was found with a left adrenal mass after complaints of weight loss and anorexia during the past 2 months. His past medical history included heavy cigarette smoking and signs/symptoms of generalized vascular disease. Abdominal CT revealed a 12.3 cm inhomogeneous mass with irregular borders (Figure 1.1Left adrenal resection confirmed the presence of adrenal metastatic adenocarcinoma from the lung primary. The adrenal gland is a potential site of metastasis from various malignancies. Adrenal metastases are the second most common type of adrenal masses after benign adenomas, and most of them are seen in patients with disseminated cancer and rarely are isolated. Timewise, metastases can be synchronous or metachronous.None declared.Andreas Kiriakopoulos MD—authorThere are no identification details of patient, and an informed consent was taken.


Lipomatous tumors are the most common type of soft-tissue tumors. Benign lipomatous tumors are lipomas and lipoblastoma. We herein report a case of benign mesenteric lipomatous tumor and the largest collection of known benign mesenteric lipomatous tumors in children in the literature.A 3-year-old girl presented with repeated dull abdominal pain and left abdominal mass swelling. On a physical examination, the child had a soft, moderately distended left abdomen that was not tender when palpated. Computed tomography and magnetic resonance imaging demonstrated a large fatty mass within the mesentery, measuring approximately 8 × 6 cm. The mass extended from the right upper quadrant to the lower pole of the kidneys. Laparotomy with resection of the mesenteric tumor was performed under general anesthesia. A well-capsuled tumor was a soft, yellow mass and found loosely attached to the mesenterium of the ileum. A histopathological examination demonstrated the lobular proliferation of mature adipocytes. Atypical lipoblasts were not seen. These features are compatible with benign lipomatous tumor, such as lipoma or lipoblastoma with maturation.In conclusion, benign mesenteric lipomatous tumors tend to be large in size over 10 cm in longitudinal length. However, resection is well tolerated in the vast majority of cases with benign post-operative courses. Lipomatous tumors are the most common type of soft-tissue tumors. Benign lipomatous tumors are “lipomas” and “lipoblastoma”. Lipoma is a rare benign lesion of mature adipose tissue. It is a well-defined, noninvasive, and encapsulated mass that can be discovered in asymptomatic patients or may cause variable nonspecific symptoms, depending on its size and location , 2. LipoWe herein report a case of benign mesenteric lipomatous tumor and the largest collection of known benign mesenteric lipomatous tumors in children in the literature.A 3-year-old girl presented with repeated dull abdominal pain and left abdominal mass swelling. There were no evident congenital abnormalities at birth nor any familial history of disease. On a physical examination, the child had a soft, moderately distended left abdomen that was not tender when palpated. The hemoglobin, alphafetoprotein, and beta-hCG levels were normal.Abdominal ultrasound showed a heterogeneous soft-tissue mass measuring φ8 cm. Computed tomography demonstrated a large fatty mass within the mesentery, measuring approximately 8 × 6 cm. The mass extended from the right upper quadrant to the lower pole of the kidneys Fig. . MagnetiLaparotomy with resection of the mesenteric tumor was performed under general anesthesia. A well-capsuled tumor was a soft, yellow mass and found loosely attached to the mesenterium of the ileum. The tumor was located 6 cm from the ileocecal valve. The tumor was not separated from small intestine. Tumor was resected with small intestine which was 5 cm and end-to-end anastomosis of the small intestine was performed without injury to adjacent structures Fig. . The tumA histopathological examination demonstrated the lobular proliferation of mature adipocytes. Atypical lipoblasts were not seen Fig. a. ImmunoFollow-up at 2 years revealed no evidence of recurrence.Mesenteric lipomatous tumors are slow-growing, mobile, soft masses that do not infiltrate the surrounding organs. Ultrasound can detect abdominal lipomatous tumors as homogenous echogenic masses; however, they may present a confusing picture, from a homogenous to a heterogeneous echo pattern . AccordiAccording to the pathological findings, the maturation pattern of lipoblastoma tends to be characterized by a high proportion of mature adipocytes at the center of the lobule with lipoblasts on the periphery. Some reports suggest that lipoblastoma may spontaneously mature or regress . Coffin The recent use of cytogenetics has proven useful for making the diagnosis, as translations involving the long arm of chromosome 8, particularly 8q11-13, with or without pleomorphic adenoma gene 1 (PLAG1) oncogene rearrangements, have been found to be associated with lipoblastomas –9. This The immunohistochemical trio of CDK4, MDM2, and p16 is a useful ancillary diagnostic tool that provides strong support for distinguishing differentiated liposarcoma from other adipocytic neoplasms . A previA literature search was performed using the electronic database “PubMed” for all patient reports in the English literature with benign mesenteric lipomatous tumors using the search term “mesenteric lipoma”, “mesenteric lipoblastoma”, and “benign mesenteric lipomatous tumor”. Relevant data were extracted from all primary reported patients. Patients included in multiple reports were used only once for the analysis. All patients under 15 years old were combined to create this report. The clinical features of the current case were consistent with those previously reported, including her age, gender, onset, size and location of tumor, preoperative diagnosis, pathological diagnosis, operation, and complications.All patient data from 1956 to 2020 were combined to create this report , 12–45. In conclusion, benign mesenteric lipomatous tumors tend to be large in size over 10 cm in longitudinal length. However, resection is well tolerated in the vast majority of cases with benign post-operative courses.


Leiomyomas are rare benign tumors that grow in the tunica media of smooth muscle cells. Leiomyomas occur most frequently in the uterus or gastrointestinal tract and only very rarely in the area of the cheek. This study reports on a rare case of a leiomyoma in the cheek of a 43-year-old woman, who presented with a well-circumscribed, asymptomatic, mobile swelling in the right cheek. This swelling was slightly purplish in color and measured approximately 4 cm × 3 cm. Surgical excision was the treatment of choice, and the diagnosis was based on histopathological and immunohistochemical stains, which were positive for actin and desmin and negative for AE1/AE3, CD34, and S100. The patient's follow-up, two years later, showed no recurrence, and she has been asymptomatic since the surgery. Leiomyomas are benign mesenchymal tumors arising from nonepithelial tissues, such as smooth muscle , 2. ThesA 43-year-old woman, a nonsmoker, with no medical antecedents was referred to the Departments of Oral and Maxillofacial Surgery of a dental college in northern Brazil for evaluation of a well-circumscribed, solitary, asymptomatic, mobile mass in the right cheek, which was slightly purplish in color, causing facial asymmetry Figures .The patient reported having had a swelling in the right cheek over the previous two years. She also reported that she was not on any drug therapy and did not consume alcohol regularly. Physical and oral examination revealed a well-demarcated, palpable, hard elastic mass measuring approximately 4 cm × 3 cm, located in the submucosal layer of the right cheek. The overlying mucosa appeared clinically normal and was not ulcerated. A CT scan revealed a homogenous mass with well-defined margins and no evidence of maxillary sinus infiltration or bone resorption Figures .The patient underwent an excisional biopsy . MicroscBaden et al. described leiomyomas as a benign neoplasm that arises in the smooth muscle , 19. As Previous studies –24 have Leiomyomas recur only very rarely, although prognosis depends on the completeness of the surgical excision of the tumor. In one case, an angioleiomyoma was excised from the region of the hard palate, but 9 weeks after the first procedure, the authors noted the recurrence . In the A leiomyoma of the cheek is a rare benign neoplasm. We report a case of a leiomyoma located in the buccal mucosa of the oral cavity. Tumors of this type are asymptomatic and may develop for months or even years. The diagnosis of this lesion requires histopathological evaluation.


Ileosigmoid knotting (ISK) is one of the rare causes of acute intestinal obstruction, which has a rapid course for forming gangrene. ISK is considered a variant of sigmoid volvulus, which otherwise is called compound volvulus. The difficulty in ISK diagnoses is owing to its rarity, uncommon presentation, and non-specific radiological findings. The physiological status of the patient and intraoperative findings are the key factors in deciding the operative procedure of choice. Herein the author describes a rare case of ISK in a 70-year-old man with gangrenous ileum and sigmoid colon, which were treated successfully with resection and double stoma. Ileosigmoid knotting (ISK) is a rare condition to encounter at an emergency doorstep, where the ileal loop twists around the sigmoid colon and its mesentery or the opposite way. It is more common in males, with a mean age of 40 years. It culminates in double-loop obstruction, which can be gangrenous or non-gangrenous at presentation. Effective resuscitation, prompt diagnosis, and early surgical intervention are the key factors in deciding postoperative mortality and morbidity. Surgical management of ISK is controversial and is based on the patient’s hemodynamic status and intraoperative findings. The primary aim of surgery is to untwist the knot, resect any gangrenous segment, and restore bowel continuity. However, in patients with unstable vital signs, advanced age, comorbidities, or peritonitis, or in whom anastomosis is difficult and precarious, resection with stoma may be necessary .A 70-year-old male presented to the surgical emergency with complaints of abdominal pain for three days. The pain was insidious in onset, diffuse, non-radiating, and continuous. It was associated with a few episodes of bilious vomiting, obstipation, and abdominal distension. There was no history of fever, jaundice, or urinary complaints. The patient had no past surgical history but had a personal history of addiction to opium. On general examination, the patient was conscious, oriented, and afebrile. His pulse rate was 100 beats per minute, respiratory rate was 20 breaths per minute, and blood pressure was 100/50 mm of Hg. Examination of the abdomen revealed a distended abdomen with diffuse tenderness, guarding, rigidity, and evidence of free fluid. There was no obliteration of liver dullness, and bowel sounds were absent. The digital rectal examination revealed an empty rectum with ballooning. X-ray of the abdomen showed distended large bowel and multiple air-fluid levels in the small bowel with no free air under the right hemidiaphragm Figure . AdditioISK is one of the uncommon but potentially life-threatening causes of bowel obstruction, in which a portion of the ileum and sigmoid colon wrap around each other, forming a closed-loop obstruction. It is commonly seen in the Africa, Asia, and the Middle East populations, with a male preponderance and mean age of presentation around 40 years ,3. EndemISK is a rare and life-threatening condition of closed-loop obstruction. It is sporadic and with high morbidity and mortality if it occurs in old age. Diagnosis of this disease is often difficult due to the rarity of the disease and variation in clinical presentation. Initial resuscitation and prompt diagnosis followed by early surgical intervention are vital in determining the morbidity and mortality of this disease. The surgical procedure performed is based on the patient’s physiological status and intraoperative findings. Resection of gangrenous bowel and restoration of the continuity should be the primary aim, but stoma can be created in the presence of high-risk factors to prevent undue mortality.


An asymptomatic 63-year-old woman presented for annual ophthalmic examination at University of Texas Medical Branch eye clinic and was found to have a simple congenital hamartoma of the retinal pigment epithelium (RPE) in the left eye. The best corrected visual acuity was 20/20 and the visual field was within normal limits. On fundus examination, a circular hyperpigmented retinal lesion measuring less than the diameter of an optic disc was detected superonasal to the fovea. Old records indicated that the lesion had been present since at least 12 years before, showing no significant morphological changes on serial fundus photographs [Figures 1A and 1B].The lesion showed hypoautofluorescence in autofluorescence imaging [Figure 2A] and optical coherence tomography (OCT) revealed high surface reflectivity with deep optical shadowing and full-thickness retinal involvement that had been stable over time [Figures 2A and 2B]. B-scan ultrasonography revealed a flat lesion showing medium to high internal reflectivity [Figure 2C].,2 It is a rare tumor and presents as a pigmented mass protruding from the RPE into the retina and at times into the vitreous cavity, usually at the posterior pole.,2 Shields et al reported the presence of a minimally dilated feeding retinal arteriole and a draining venule in five cases. We did not observe this finding in the present case.Simple congenital hamartoma of the RPE is a benign tumor composed of a hyperplastic RPE with variable vascularity and is often discovered incidentally in asymptomatic children and young adults. which was not present in the current case. These lesions showed early hypofluorescence in fluorescein angiography with some variable hyperfluorescence seen in later frames due to either RPE atrophy or intrinsic tumor vasculature. OCT features of the lesion show a hyper-reflective surface and dense shadowing. Simple hamartomas of RPE are usually small and cannot be easily seen by ultrasonography. If visible, they have medium to high internal reflectivity on ultrasound.According to previous studies, the retinal tissue surrounding the hamartoma may show minimal signs of traction, The absence of a history of trauma, retinal traction, time-related growth features, and the typical funduscopic features of the lesion can help differentiate between these diagnoses.Differential diagnoses include combined hamartoma of the retina and RPE, adenoma or adenocarcinoma of the RPE, congenital hyperplasia of the RPE, choroidal nevus, choroidal melanoma, RPE hyperplasia, and intraocular foreign body.The present case is particularly interesting because the stability of lesion is documented by fundus photographs spanning a duration of 10 years and by OCT images that are 8 years apart.Nil.There are no conflicts of interest.


Synchronous multiple primary lung cancers (sMPLC) are rare forms of lung cancer, and their diagnosis remains as a significant challenge. Distinguishing sMPLC from advanced disease is important as their prognoses and therapeutic management vary dramatically.BRAF V600E oncogenic mutation was exclusive to L5. In addition, the KRAS G12C mutation was identified in three lesions (L1-L3) located in the right lung, which may have resulted from convergent evolution.The patient was a 56-year-old Chinese male who exhibited six synchronous invasive adenocarcinomas at diagnosis [T2(6)N0M0], and who achieved durable clinical benefit under adjuvant chemotherapy for 41 months following wedge resection and lobectomy. Whole-exome sequencing revealed that two lesions (L4 and L6) in the left upper lobe of the patient’s lung shared 28 nonsynonymous mutations; thus, suggesting that the lesions may have arisen from a common ancestor at the early stages of tumorigenesis, and spread into distinct histologic subtypes. Moreover, while L5 was in the same lobe as L4 and L6, it represented a distinct lineage as it did not share any mutations with other lesions. Notably, the We report a patient with six synchronous invasive adenocarcinomas who demonstrated durable clinical benefits under adjuvant chemotherapy following surgical treatment. While cancer staging is one of the many challenges associated with sMPLC, the data generated through next-generation sequencing can provide information on lesion origins, and thus, advance the era of precision medicine. An increasing number of lung cancers  exhibit two or more malignant pulmonary lesions ; howeverA 56-year-old Chinese male patient was admitted to the Hospital following the accidental discovery of GGOs in his lung during a routine physical examination. The patient was a heavy smoker for 30 years. Routine laboratory workups and the levels of serum tumor markers including carcinoembryonic antigen (CEA) were normal. Microbiological blood tests were negative, and no abnormal cells were detected during the sputum cytology test. Computed tomography (CT) scans revealed bilateral and ill-defined GGOs, including three lesions (L4-L6) in the left upper lobe (LUL), and three additional lesions, L1-L3, in the right upper lobe (RUL), right middle lobe (RML), and right lower lobe (RLL), respectively. A Corona radiata sign was observed in L1, which was in the apical segment of the RUL with the convergence of the supplying blood vessels. Cavitation was present in L3 and there was no associated lymphadenopathy in the mediastinum.max) ranging from 1.3 to 3.8. No distant metastases were detected  scan revealed increased 18F-fluorodeoxyglucose (FDG) avidity in these multifocal lesions. The size of the lesions ranged from 0.7 cm to 2.4 cm, with maximum standardized uptake values  data revealed that the lesions exhibited different histological subtypes, including 50% papillary + 50% acinar in L1, L3 and L5; 90% papillary in L2; 90% lepidic in L4; and 90% acinar in L6. The detailed clinicopathological characteristics of the lesions are summarized in Table According to the Martini-Melamed classification (1975), the patient was diagnosed with sMPLC . The patTo investigate the molecular profiles of the different lesions, genomic DNA was extracted from formalin-fixed, paraffin-embedded tumor specimens using QIAamp DNA FFPE Tissue Kit (Qiagen), and library preparations were performed with KAPA Hyper Prep Kit (KAPA Biosystems). Target enrichment was performed using the xGen Exome Research Panel and Hybridization and Wash Reagents Kit (Integrated DNA Technology) according to the manufacturer’s protocol. Sequencing was performed on Illumina HiSeq4000 platform using PE150 sequencing chemistry (Illumina) . To deteBRAF V600E oncogenic mutation was exclusive to L5  provided a new approach to understanding sMPLC in the era of precision medicine.Additional file 1: Figure S1. Heatmap of the alternate allelic frequencies of heterozygous SNPs on chromosome 1q across all lesions. The color indicates the alternate allelic frequency ranging from red to white (range: 0–100%). Lesions are clustered based on the similarities of the patterns of the alternate allelic frequency of heterozygous SNPs.


Cavernous hemangioma (CH) is not commonly found within the abdomen or the retroperitoneum. We report the first case of CH originating from the gonadal vein.A retroperitoneal tumor was found incidentally in a 57-year-old female patient. The differential diagnoses from the initial imaging studies included gastrointestinal stromal tumor, carcinoid tumor, neurogenic tumor, metastasis, lymphadenopathy, or another rare tumor.A surgical en-bloc excision was performed via a subcostal incision and intravenous CH arising from a gonadal vein was diagnosis by the urological pathologist.After the surgery, no complications were noted. A computed tomography scan was performed after 3 months follow-up and no tumor recurrence was found.This case reminds us that CH should be listed as one of the differential diagnoses for a retroperitoneal tumor. A definite diagnosis of CH relies on surgical resection. The prognosis is well if adequate resection is performed. CHs that originate in the retroperitoneum are even rarer.–6 Retroperitoneal CHs tend to be asymptomatic, especially in the early stages of their development. However, if they grow large enough to compress the adjacent anatomical structures, the patient starts to get symptoms. The radiographic features of CHs are diverse and may be observed using ultrasonography, computed tomography (CT), angiography, and magnetic resonance imaging (MRI). Thus, it is hard to make a definitive diagnosis for retroperitoneal CH through any non-surgical method. Here we share the case of a 57-year-old female patient who had CH arising from the gonadal vein.Cavernous hemangiomas (CHs) are benign endothelial cell neoplasms that are typically absent at birth. However, it may grow rapidly during infancy with spontaneous involution later in life.2This study presents the case of a 57-year-old female patient who had a history of hypertension, diabetes mellitus, and left breast cancer post a modified radical mastectomy in April 1989. The patient gave informed consent for the publication of her case. She was transferred to our urological clinic because of an incidentally identified heterogenous tumor in the right retroperitoneal cavity, which was 4.5 × 3.9 cm in size, and causing right hydronephrosis Fig. A and B rWe created a subcostal incision, and the retroperitoneal tumor was found medial to the right upper third ureter, lateral to the duodenum, and above the inferior vena cava. One draining vein into the right renal vein, and another 2 feeding arteries arising from the renal artery were noted. It was not possible to clearly identify the gonadal vessel within the operative field. In addition, a slight adhesion was noted between the tumor, hydroureter, and second portion of the duodenum area.Gross examination of the resection specimen Fig.  revealedNo complications were noted after the surgery and the patient was discharged after 5 days. A CT scan was performed after 3 months follow-up and no tumor recurrence was found.3,8 CH is usually observed in infants and children but seldom in adults. The probability of getting CH is similar for both sexes.,9–11 There are no specific symptoms for patients who have abdominal or retroperitoneal CH. Flank pain, symptomatic or asymptomatic hematuria, anemia, thrombocytopenia, renal vein thrombosis (rarely), and even life-threatening bleeding have been reported as symptoms of renal CH. In the current case, the patient did not have flank pain despite hydronephrosis. Maybe slow progression of retroperitoneal CH is the reason for this.Vascular tumors are non-epithelial and can be divided into hemangioma and vascular malformations. CH is a benign vascular malformation often seen in the orbital, skin, and mucosal areas. However, it is rarely found within the retroperitoneal cavity.,14 Within the kidney, the most common site is the renal papilla.The most common sites for retroperitoneal CH include the peripancreatic, perirenal, or periureteric locations, and the iliopsoas muscle area. The kidney is the most common site of genitourinary CH, followed by the urinary bladder.,10 It may also present as a homogeneous, hypodense inner component tumor without enhancement during the artery or portal phase. The reason for atypical findings on the CT scan might be neovascularity, hemorrhage, thrombosis, and arteriovenous shunting. An MRI did help with the diagnosis of retroperitoneal CH. On T1 imaging they usually show low signal intensity. Whereas on T2-weighted imaging, hemorrhage, and hyalinization of tissue can often be interpreted with high intensity within the tumor.–17 Although we did offer the option of a MRI examination before the operation, the patient declined and instead surgery was performed after a physical exam.Retroperitoneal CH is very difficult to diagnose before surgery. In this case, the differential diagnosis included GIST, carcinoid tumor, neurogenic tumor, metastasis, lymphadenopathy, or another rare tumor. GIST was the first diagnosis of the uro-radiologist because of the heterogeneous enhancement. According to the literature, CH may have features of homogeneous enhancement relative to the renal cortex.,18 Thus, there are no golden standard therapeutic option to follow. The generally recommended treatment for retroperitoneal hemangioma was surgical resection.,17 Even though retroperitoneal hemangioma is a benign tumor, patients still have the risk of tumor rupture and bleeding without monitoring nor treatment. As for the prognosis, no obvious complication nor recurrence are found in these cases in follow-up, although local recurrence had been reported sporadically if primary resection was inadequate.Retroperitoneal hemangioma is very rare and only confirmed in <5% of all retroperitoneal tumors in the adult. Less than 30 cases had been reported since 1950.In conclusion, this case reminds us that CH should be listed as one of the differential diagnoses for retroperitoneal tumors. The definite diagnosis of CH relies on surgical resection. The prognosis is well if adequate resection is performed.Conceptualization: Chun-Yo Laih, Guang-Heng Chen, Han Chang, Wei-Ching Lin.Writing – original draft: Chun-Yo Laih, Chun-Ming Lai.Writing – review & editing: Chun-Yo Laih, Po-Fan Hsieh, and Chao-Hsiang Chang.


Intramedullary cervical spinal cord teratomas (ICTs) are extremely rare, and diagnosis and treatment are challenging. We conducted a systematic review of the literature on the diagnosis and treatment of ICT.The presentation, imaging manifestations, diagnosis, management, surgery findings, prognosis and histology were reviewed following Preferred Reporting Items for Systematic Reviews and Meta Analyses guidelines. English-language studies and case reports published from inception to 2018 were retrieved. Data on presentation, imaging characteristics, diagnosis, management, surgery findings, outcomes, and histopathology were extracted.Ten articles involving 10 patients were selected. The lesions were located in the upper cervical vertebrae in 4 cases, whereas in the lower cervical vertebrae in the remaining 6 cases. In 5 cases, the lesions were located on the dorsal side of the spinal cord, and in the center of the spinal cord in the remaining 5 cases. Quadriparesis (60%), paraplegia (30%), monoplegia (10%), and neck pain (50%) were the main presentations. The lesion appeared as a intramedullary heterogeneous signal during an MRI scan, and the lesion signal would be partially enhanced after the contrast medium was applied. All patients underwent surgical intervention through a posterior approach. Neurological function improved postoperatively in all patients. Two patients with pathology confirmed to be immature teratomas experienced recurrence.ICTs are extremely rare entities that are mainly located in the center or dorsal part of the spinal cord which mainly manifest as quadriplegia and neck pain. MRI is a useful modality that provides diagnostic clues. Surgery from a posterior approach is the primary treatment, and the effect of adjuvant therapy remains uncertain. The prognosis is mainly related to the pathological nature of the tumor and not the method of resection. A majority of the studies describing ICTs are case reports, and there are no systematic reviews. Therefore, we conducted a systematic review of the ICTs to provide a summary on their incidence and prevalence rates, presentation, imaging characteristics, diagnosis, management, prognosis, and histological characteristics.Teratomas account for about 0.1% of all spinal tumors, and intramedullary spinal cord teratomas, which are less common, are found mainly in the lumbar and thoracic segments.22.1 Potentially relevant publication was retrieved from PubMed, Embase, and the Cochrane library from inception to 2018. The search strategy included combinations of the terms “intramedullary teratoma,” “spine teratoma,” cervical teratoma,” either as keywords or as MeSH terms. The reference lists of retrieved literatures were manually searched for relevant articles, and the abstracts were read for possible full text review and inclusion. Articles were screened and selected independently by 2 reviewers. Disagreements were resolved by discussion, and a third author conducted an independent review if agreement was not reached.The systematic review was conducted following the PRISMA .2.2The full text of articles written in English, published after 1980, and involving human subjects were included. Prospective clinical trials, retrospective studies, reports of case series, and case reports with data on intramedullary cervical teratoma were eligible for inclusion. Cadaver studies, laboratory or animal studies and reports of teratoid tumor were excluded. Meta-analyses and systematic reviews were excluded.2.3The names of the first and corresponding authors, type of study, publication date, number of patients, presentation, imaging manifestation, lesion level, surgical procedure, findings during surgery, outcome, histology and duration of follow-up were extracted.33.1,8,10–17Figure The initial literature search yielded 504 articles. After eliminating duplicates, 349 articles were retained for further screening. Of these, 313 were excluded following a review of the titles and abstracts, and 26 articles were excluded because they did not meet the inclusion criteria. Finally, 10 articles describing ten patients were included in this systematic review.3.2All the studies included in the systematic review were case reports, each describing one patient, were published between 1982 and 2015, and included 4 males and 6 females  and quadriplegia combined with neck pain , was the most common symptom , followed by paraplegia combined with neck pain 20%, n = 2), monoplegia , and monoplegia combined with chest pain . Neck pain was reported in 50% (n = 5) of the patients. Upon physical examination, 3 patients (30%) were found to have a skin dimple or nodular mass with surrounding hair at the midline of the dorsal cervical region , computed tomography (CT), X-ray, and myelography were the most frequently used imaging modalities. In this review, eight patients were evaluated with MRI, whereas the two patients reported in the 1980s underwent myelography and X-ray. CT and X-ray were able to reveal coexisting spinal deformities. Table 3.5None of the lesions were diagnosed as a teratoma before surgery, and all the lesions were confirmed as ICTs by postoperative pathological examination. All the patients underwent surgery after the discovery of the intramedullary lesion, and all the procedures are performed with a posterior approach for laminectomy. Among these 10 cases, subtotal resection, total resection, and partial resection were performed in 5, 4, and 1 case, respectively. Postoperatively, 1 patient suffered cerebrospinal fluid leak at the incision site, which was resolved by oversewing the wound and placing a lumbar drain. The symptoms in all the patients were resolved to different degrees in the early postoperative period. Two patients with immature teratomas had recurrence in the fourth and tenth months after the initial operation, respectively, whereas none of the 8 patients with mature teratomas experienced relapse during a follow-up period of 3 to 48 months  with intramedullary teratomas had deformities of the cervical spine, including spinal bifida, incomplete segmentation, congenital nonunion of vertebral body, hypoplasia of the spinous process, and skin dimple on the dorsum of the neck, which provide evidence for the dysembryogenic theory. No spinal deformities were found in the remaining 5 patients. All the tumors included in the current review were located in the central or dorsal part of the cervical spinal cord, supporting the misplaced germ cell theory. A new hypothesis proposes a cause-and-effect relationship between the spinal cord malformations and the migration of the pluripotential cells, which get entrapped in an abnormal environment that leads to teratoma formation.The pathogenesis of intradural spinal teratoma is controversial. The dysembryogenic theory and the misplaced germ cell theory are the two most widely discussed theories. In the current review, there was no recurrence in 8 patients with mature cervical intramedullary teratomas, although some of the tumors were removed with partial or subtotal excision. In contrast, both of the patients with immature teratomas who underwent total and near-total resection suffered relapse.,8Intramedullary teratomas can be categorized as mature and immature teratomas based on the degree of cell differentiation. A mature teratoma with well differentiated cells shows the characteristics of a benign tumor, while an immature teratoma harboring primitive, immature, and undifferentiated cell characteristics often has the features of malignant tumors.The clinical presentations of cervical intramedullary teratomas were variable and related to the location and size of the tumor. The main manifestations were symptoms and signs related to cervical spinal cord injury. Quadriparesis was the primary manifestation and included motor dysfunction and/or sensory changes. The patients presented with varying degrees of strength reduction and deep and shallow sensory disturbances in the extremities. Hyperactive deep tendon reflexes and ankle clonus were present in some patients. In addition, paraplegia and monoplegia were observed in some patients with relatively small tumor volumes. In the current review, the voiding difficulty was reported in one patient only. A more prominent feature of patients with intramedullary teratomas was the relatively high incidence of neck pain, which was reported in 50% of the patients included in the current review. A posterior skin dimple with a hair around in the neck region was an important clue for teratoma.,30,31 X-ray and CT can show changes in the bony structure of the spine due to the presence of tumors, such as widening of the spinal canal, congenital nonunion, incomplete segmentation, and spina bifida. MRI is the most useful imaging method for cervical intramedullary teratomas that can provide information on several characteristics of the tumor such as location and size. On MRI, the lesion is observed as a lobular mass with homogeneous intermediate signal intensity that arises from within the spinal cord and displays an indistinct enhancement with contrast medium. Some of the lesions have cystic cavities that are well displayed in MRI. Intramedullary teratomas of the spinal cord were predominantly located in the lower thoracic and thoracolumbar regions.,21 Therefore, it is necessary to perform an MRI scan of the thoracic and lumbar spine when a cervical intramedullary teratoma is found. All the ten literatures did not described the biochemical changes in cerebrospinal fluid and its role in the diagnosis of intramedullary cervical teratoma. We believe that it is necessary to pay attention to research in this area in the future. Teratomas also occur in the intracranial medulla.,41 However, we have not found any literature describing the association between intracranial intramedullary teratomas and spinal intramedullary teratomas.Evaluation using imaging studies is an important diagnostic tool, which can provide some diagnostic evidence but cannot confirm the diagnosis of teratoma.,37,38 However, some scholars have pointed out that intraoperative monitoring may limit the scope of tumor resection. The lesions may have solid and cystic components. Therefore, to avoid postoperative aseptic chemical meningitis, surgeons should aim at reducing the spillage of cystic components intraoperatively.,36 There may be a firm attachment between the lesion and the spinal structure at certain areas or a relative cleavage plane, although no real capsule was found. Laminectomy or laminoplasty from a posterior approach was applied in all patients reported to date. Following the tract of the skin dimple during surgery may aid in determining the tumor position. Total resection of the tumor without damaging the spinal cord is the surgical goal. However, due to the adhesion of the tumor with the spinal cord during surgery, this may not be fully realized in all patients. To avoid damage to normal spinal cord tissue, subtotal resection is sometimes required.,32 Some studies reported that total and subtotal resection had comparable recurrence rates. We believe that the recurrence rate is related primarily to the nature of the tumor and is less likely associated with the resection method. The recurrence incidence of mature teratomas is low, and the probability of recurrence in immature teratomas is high. In the current review, there were no recurrences among the eight mature teratoma cases during the follow-up period regardless of the resection method . However, the two patients with immature teratomas undergoing total resection and partial resection, respectively, relapsed within a short period of time after surgery.Surgical resection is the primary treatment for ICTs. For intramedullary tumor resection, intraoperative electrophysiological monitoring can reduce the risk of neurological damage.,34 Conversely, immature teratomas are composed of poorly differentiated cells and tissues, and some primitive structures may be seen in the lesion. Intramedullary teratomas should be differentiated from other intramedullary lesions such as astrocytomas, ependymomas, hemangioblastomas, schwannomas, neurofibromas, and enterogenous cysts, which have structures resembling a teratoma, which is a source of dilemma for the differentiation of teratomas. Teratomas are subdivided into 3: group I is characterized by the presence of only endodermally derived tissues, group II characterized by endodermally and mesodermally derived tissues, and group III characterized by endodermally, mesodermally, and ectodermally derived tissues.,12Histopathology of the resected tissue ultimately determines the definitive diagnosis of teratoma and identifies mature and immature lesions. A typical mature teratoma includes a mixture of well differentiated tissues from the epithelial and mesenchymal elements of the 3 germ layers: endoderm, mesoderm, and ectoderm. The tumor tissue, which is always disorganized, may be composed of less than three germ layers, as derivatives of 1 or 2 layers may overgrow the others.,32 Additionally, whether adjuvant radiotherapy after resection reduces recurrence of immature teratomas remains unclear. Some authors believed that postoperative radiotherapy and chemotherapy may have a role in reducing the recurrence and progression of malignant intramedullary teratoma. In Dr Han's report, they used a dose of 14Gy to 36Gy for local radiotherapy after tumor resection. In addition, they used etoposide phosphate  and cisplatin  for chemotherapy.There is no consensus on adjuvant therapy for teratomas. For mature teratomas, due to its indolent growth characteristics, the recurrence rate is very low even without postoperative adjuvant radiotherapy or chemotherapy.,13 In long-term follow-up, the main indicator of prognosis is recurrence, which is closely related to the nature of the tumor, that is, whether it is a mature or immature teratoma.One of the most important factors in determining prognosis is the pathological nature of the tumor. In most patients receiving successful resection, postoperative symptoms are relieved to varying degrees, regardless of whether the tumor was benign or malignant.Although informative, this review was limited by the selection of retrospective case reports. Also, reporting bias and selection bias must be considered because not all cases of ICTs reported in the literature were included, and articles in languages other than English were excluded.5Our systematic review reveal ICTs as an extremely rare entity that are mainly located in the center or dorsal part of the spinal cord which mainly manifest as quadriplegia and neck pain, accompanied with a skin dimple in the posterior neck that has a tract entering the dura in some cases. MRI is a useful modality that provides diagnostic clues. Surgery from a posterior approach is the primary treatment, and the effect of adjuvant therapy remains uncertain. The prognosis is mainly related to the pathological nature of the tumor and not the method of resection.Data curation: Tongxiang Li.Formal analysis: Lang Li.Investigation: Fei Xing.Methodology: Min Gong.Project administration: qing guan.Software: Rui Xiao.Supervision: qing guan.Writing – original draft: Lishuai Wang.


Inflammatory pseudotumour has been used to describe an inflammatory or fibrosing tumoral process of an undetermined cause that may involve a variety of organ systems, including the lungs, spleen, liver, lymph nodes, pancreas and extrahepatic bile duct with potential for recurrence and persistent local growth. In this article, we report a patient with a big mass of uncertain nature and behavior.A 60-year-old woman presented with a 1-week history of abdominal pain, fever and jaundice. Six months before she had had right upper quadrant pain that was interpreted as biliary colic. A contrast-enhanced CT scan showed a big mass of soft tissue with diffuse infiltration of the gallbladder, displacement of the transverse colon, hepatic flexure and duodenum. For diagnostic distinction between a chronic inflammatory disease or a neoplasm, exploratory laparotomy was required. Intraoperative exploration disclosed a big mass of hard texture involving the gallbladder, with multiple concrements, hepatoduodenal ligament, right and transverse mesocolon, stomach and duodenum.Cholecystectomy was performed, preserving adjacent organs with macroscopic desmoplastic reaction. Histopathologic examination of the gallbladder showed a spindle cell proliferation with diffuse chronic inflammatory infiltrate of lymphocytes, plasma cells and hyalinized fibrous stroma. No vascular invasion or cellular atypia were evident.Inflammatory pseudotumour is a rare condition and diagnostic distinction from a chronic inflammatory disease or other neoplasm is only possible by histopathologic examination. There is a limited number of case reports in the literature indicating tumor location in the gallbladder. Inflammatory pseudotumour is a rare lesion that has been described in various organs and tissues. Intra-abdominal variants of the disease are reported to occur most frequently in the liver, spleen, mesentery, and extrahepatic bile duct. The location of the gallbladder is even more uncommon.Malignant transformations and recurrences of inflammatory pseudotumour have been reported years after surgery, therefore long-term follow-up is necessary even for patients successfully treated by surgical resection.Elevated IgG4 serum levels have been reported in association with this illness as well as abundant IgG4 positivity in tumor infiltrating plasma cells, signs suggestive of an IgG4-related disease. A high serum IgG4 concentrations, thus provides a useful means of distinguishing this disorder from other differential diagnoses.Pharmacologic treatments have also been reported for IgG4-associated inflammatory pseudotumor and there are even cases of complete resolution of the disease with steroids treatments. However in the presented case, this condition did not occur, so treatment was exclusively surgical.A 60-year-old woman presented to the Emergency Room with abdominal pain, fever, pruritus and jaundice since 1 week. The patient had a history of smoking and a family history of pancreatic cancer.On physical examination, a hard and painful mass was identified on the right hypochondrium. Blood laboratory examination showed extrahepatic cholestasis enzymes  and Pan-Cytokeratin (PAN-CK). Masson’s trichrome stain showed intense positivity on collagen fibers. Less than 10% of the tumor cells sample were Ki67 positive, and 11 plasma cells were IgG4 positive per high power field. Taken together these findings confirmed the diagnosis of inflammatory pseudotumor of the gallbladder with sclerosing cholangitis associated with a normal level of serum of Immunoglobulin G4 of 24 mg/dl (10–67 mg/dL).No local recurrence was detected at the three-years follow-up on CT scan.The term inflammatory pseudotumour has been used to describe an inflammatory or fibrosing tumoural process of undetermined cause that may involve a variety of organ systems, including the lungs, spleen, liver, lymph nodes, pancreas and extrahepatic bile duct with potential for recurrence and persistent local growth , 2. TherInflammatory pseudotumour appear to be more common in non-European populations. They usually occur in infancy and young adults but can occur in the elderly . The IgGHigh serum IgG4 concentrations might provide a useful means of distinguishing this disorder from other lesions –9.The histopathological examinations showed sclerosing fibrous tissue with histiocytes, chronic lymphocytic inflammatory infiltrate and plasma cells with isolated eosinophils compatible with IgG4-related disease. However as IgG4 serum levels were within normal range and the IgG4 tissue expression was very weak, there is no strong evidence of a clear association with IgG4-related disease.Inflammatory pseudotumour can generally be considered to be a relatively rare disease of undefined origin, with a great variety of symptoms, causing diagnostic challenges in the distinction of chronic inflammatory disease and neoplasm.Inflammatory pseudotumor is defined as non-neoplastic but is currently considered as a tumour with low-grade malignant transformations. It has been reported in the liver, urinary bladder, kidney, breast, stomach, pancreas, spleen and retroperitoneum. There is also a limited number of case reports in the literature indicating the gallbladder location. These patients must be observed with close and regular long-term follow-up as recurrences have been reported to occur four to 7 years after surgery .


Colonic lipomas are rare benign, non-epithelial tumors of mesenchymal origin. They are often solitary lesions of submucosal origin found in the proximal colon and typically measure less than 2 cm in size. Giant colonic lipomas are greater than 4 cm and present with non-specific gastrointestinal symptoms such as abdominal pain, abdominal distention, constipation, or gastrointestinal bleeding. Traditionally, giant colonic lipomas have been surgically rather than endoscopically resected due to concerns for bowel wall perforation and life-threatening hemorrhage. However, in recent years, advances in endoscopic tools and hemostatic techniques have lessened these risks. The following case details the successful endoscopic resection of an intermittently obstructing giant colonic lipoma (6 cm) located in the descending colon utilizing the loop-assisted-snare resection technique.  Intestinal lipomas are rare benign, non-epithelial tumors of mesenchymal origin that can form throughout the gastrointestinal tract. They are more frequently encountered in the colon with an overall incidence of 0.3-4.4% . These lA 40-year-old African American female with a past medical history of hypertension and lupus profundus was referred to the gastroenterology clinic by her primary physician for endoscopic evaluation of a one-year history of intermittent left lower quadrant pain, constipation, and hematochezia. The patient described noticing a bulge located in her left lower quadrant when symptomatic that would subsequently disappear after having a bowel movement. She often needed to massage the area and change position to facilitate a bowel movement. Upon further questioning, the patient denied use of nonsteroidal anti-inflammatory drugs (NSAIDs), opiates or other medications known to cause constipation. She denied any personal or family history of colon cancer. Her physical exam, including a digital rectal exam, was unremarkable. Laboratory tests including hemoglobin and hematocrit were also within normal range. A colonoscopy was performed which revealed a large broad-based 6 cm pedunculated polypoid lesion at the descending colon that appeared to be causing a ball-valve type intermittent colonic obstruction Figure .Grossly, the mass had normal overlying mucosa with evidence of slight erosions at the tip. Using biopsy forceps, the polypoid lesion was investigated and demonstrated a positive pillow sign. An endoloop device was then deployed at the stalk and the entire mass was endoscopically resected with snare cautery en-bloc and removed from the colon using a net retrieval device Figure .Upon inspection of the snare cautery site, the endoloop was noted to have been inadvertently cut and a small, colonic bowel wall defect was noted. The defect was completely closed utilizing seven hemoclips Figure .Pathology of the large polypoid mass revealed colonic mucosa with underlying mature adipose tissue consistent with a submucosal colonic lipoma. The patient reported complete resolution of all her symptoms upon follow-up.Colonic lipomas are uncommon benign tumors composed of mature adipose tissue. They are often solitary lesions of submucosal origin, and very rarely of subserosal and muscularis origin . Most arGiven the nonspecific nature of the gastrointestinal symptoms related to colonic lipomas, its clinical diagnosis can be very challenging and requires additional diagnostic radiological or endoscopic modalities. An abdominal CT scan can demonstrate the lesion’s characteristic uniform low-density adipose tissue signal with any overlying hyper-intensity stranding near the mucosal surface indicating the presence of ulcerations . EndoscoTraditionally, endoscopic removal of lipomas had been reserved for smaller lesions (<3 cm) due to concerns of increased risk of bowel wall perforation and life-threatening hemorrhage. However, in recent years, advances in endoscopic procedures and hemostatic tools such as hemoclips, endoloops, and hemostatic powders have gained it favor over more invasive surgeries . DependiCurrently, there is limited literature comparing the efficacy and safety of these various endoscopic techniques for the resection of colonic lipomas, highlighting their relative symptomatic rarity. The most comprehensive systematic review available was done by Bronswijk et al. , encompaPreviously, surgical resection was recommended for the management of giant colonic lipomas due to a higher risk of bowel wall perforation and life-threatening hemorrhage with endoscopic resection. However, endoscopic techniques such as unroofing, endoscopic mucosal resection (EMR), “loop and let go”, loop-assisted-snare resection, and endoscopic submucosal dissection (ESD) have been effectively used in recent years to excise giant colonic lipomas. This case highlights the endoscopic resection of an obstructing giant descending colonic lipoma utilizing loop-assisted-snare resection technique while also addressing possible intraprocedural complications including endoloop failure and small, colonic bowel wall perforation.