Although chronic diarrhea is frequent, some of its causes are rare, namely, neuroendocrine tumors (NETs). Due to their rarity and non-specific symptoms, such as diarrhea, they are often underdiagnosed.An 80-year-old woman presented to the emergency department due to emesis and watery diarrhea. Blood tests showed acute kidney injury, hypokalemia, and metabolic acidosis. An abdominal computed tomography revealed a 51 mm pancreatic lesion. An endoscopic ultrasound-guided biopsy raised the hypothesis of a NET. The patient refused surgery and was lost to follow-up. At the eighth hospitalization, 11 months later, the suspicion of a vasoactive intestinal peptide tumor (VIPoma) was raised and confirmed by assessing the vasoactive intestinal peptide (VIP) levels (>100 pmol/L). Octreotide was started with the resolution of the symptoms. 68Ga-DOTANOC positron emission tomography/computed tomography excluded metastatic disease. After six months of octreotide therapy, the tumor shrunk 13 mm in maximum diameter. At the last follow-up, one year later, she remained asymptomatic.The delayed diagnosis of VIPoma led to multiple life-threatening episodes. This case highlights the importance of considering all potential differential diagnoses of common symptoms such as diarrhea. Although VIPomas are rare, clinicians should be aware of this entity and suspect this diagnosis in patients with chronic diarrhea with a poor response to standard antidiarrheal agents. Somatostatin analogs should be promptly prescribed for symptom control and tumor progression prevention in patients who refuse surgery or have unresectable tumors. Tumor shrinkage might also be observed in these cases. There are multiple possible causes of chronic diarrhea, some of them exceptionally rare, namely, neuroendocrine tumors (NETs) . VasoactAn 80-year-old woman went to the emergency department with a history of asthenia, emesis, and multiple daily episodes of watery non-bloody diarrhea initiated with a one-week duration. Her medical history included dyslipidemia and atrial fibrillation, being chronically medicated with simvastatin 20 mg daily and rivaroxaban 20 mg daily. She denied recent travel, dietary changes, sick contacts, the addition of new medications, ingestion of alcohol, smoking, or use of illicit drugs.+ 131 mmol/L), mild hypokalemia (K+ 3.1 mmol/L), and severe metabolic acidosis . C-reactive protein levels and leukocyte counts were within the normal range. During the hospital stay, an abdominal computed tomography (CT) revealed a 51 mm heterogeneous mass in the pancreatic body.Blood tests revealed an acute kidney injury (urea nitrogen 267 mg/dL and creatinine 2.91 mg/dL), mild hyponatremia . Given her clinical background, the elevated VIP levels, and the pancreatic lesion, the diagnosis of VIPoma was ultimately established. She was then referred to our department. Treatment with a long-acting somatostatin analog was initiated  with the resolution of diarrhea and metabolic disturbances. A 68Ga-DOTANOC PET/CT was performed showing no evidence of metastatic disease. Six months after starting octreotide, the maximum diameter of the tumor had reduced by approximately 25% (from 51 mm to 38 mm) . Although this neuropeptide also inhibits gastric acid secretion, achlorhydria is not an obligatory finding, and hypochlorhydria, or even normal acid secretion, can occur ,5. This Giardia lamblia and consumption of unsupervised water possibly acted as confounding factors; however, these two situations were uncovered and resolved on the second hospitalization. We believe the most preponderant factor for the delay was the fact that, in general, physicians do not have NETs as a differential diagnosis right away due to their rarity and non-specific symptoms, such as diarrhea. Indeed, an international survey found a mean 52-month delay in the diagnosis of NETs and patients must see an average of six different clinicians before they get the correct diagnosis [Nevertheless, for clinicians to prescribe these potentially life-saving medications and prevent numerous complications, they need to promptly diagnose this disease, which was not the case here. Notwithstanding the cytological report raising the possibility of a NET, the patient was discharged after refusing surgery, without being offered an effective treatment alternative or referral to other specialties. After being lost to follow-up, the patient had multiple visits to the emergency department. It could be argued the diagnosis should have been made sooner. History of iagnosis . Our patiagnosis . The usuiagnosis ,10,11. Biagnosis ,10,11,20iagnosis ,5.Although VIPomas are rare, clinicians should be aware of this potentially life-threatening entity and suspect this diagnosis in patients with chronic watery diarrhea that persists with fasting and has a poor response to standard antidiarrheal agents. Somatostatin analogs should be promptly prescribed for symptom control and tumor progression prevention in patients who refuse surgery or have unresectable tumors. Tumor or metastasis shrinkage might also be observed in these cases.


Leiomyosarcoma (LMS) is a mesenchymal neoplasm with smooth muscle differentiation, being considered one of the most common soft tissue sarcomas. However, it rarely affects the eye, and when it does, it is usually located in the orbit, being extremely rare in the conjunctiva.We report a case of a 45 years old male patient, with a recurrent rapid growing conjunctival mass on the temporal limbus of his left eye, which was excised, and the anatomopathological report was suggestive of a grade 1 leiomyosarcoma. Since the lesion was recurrent, we decided to perform an extended enucleation for treating this condition. Nevertheless, the patient is being followed up to 30 months, with systemic metastasis screening, showing no other lesions or recurrences.Conjunctival leiomyosarcoma is an extremely rare ocular tumor, which can be clinically indistinguishable from other conditions such as squamous cell carcinoma, so, biopsy is essential. Albeit there is no standard treatment, complete surgical removal with safety margins is mandatory. It accounts for up to 25% of all soft tissue sarcomas,2A 45 years old male patient was referred to the Ocular Oncology ambulatory of the Department of Ophthalmology and Visual Sciences at Escola Paulista de Medicina, due to a rapidly growing mass in the temporal conjunctiva of his left eye, associated with redness and irritation. He had a history of excision of a similar lesion in the same topography, performed in another hospital one year before, with an anatomopathological report suggestive of leiomyosarcoma.On ophthalmological examination, he presented a vascularized whitish lesion in the temporal limbus, with an infiltrative aspect. The lesion extended to the cornea , temporal conjunctiva, and causing retraction of the tarsal conjunctiva . Slides Fédération National des Centres de Lutte Contre Le Cancer (FNCLCC).We decided to perform an extended enucleation, mostly because the infiltrative aspect of the lesion and also due to the fact that the lesions was previously biopsed, and the material was sent to further analysis. Biopsy showed fascicles of spindle cells with eosinophilic cytoplasm and areas of atypia. Necrosis was absent; however, areas of haemorrhage were present. Up to 2 mitosis per high-power field (HFP) was noticed, some of them being atypical. Scleral and corneal deep stroma invasion was present. The chamber angle was not invaded. Immunohistochemistry was positive for Vimentin, Calponin, Desmin, Caldesmon, CD68, AE1/AE3 , and SMA , Fig. 4 The patient is being followed by a clinical oncologist, with imaging exams of the central nervous system (CNS), chest, and abdomen for 30 months without recurrence.3Leiomyosarcomas are malignant tumours showing smooth muscle differentiation, with multiple genomic aberrations. It is characterised by non-recurrent structural and copy number alterations.Several orbital leiomyosarcomas have been reported in the literature, as well as metastatic LMS to the orbit and choroid. However, to the best of our knowledge, only 12 cases of primary conjunctival LMS have been previously reported.The heterogeneous clinical appearance of LMS makes it difficult to distinguish among several other conjunctival conditions such as squamous carcinoma, lymphomas, and amelanotic melanoma. Other possible differential diagnosis would be myofibroma, leiomyoma and nodular fascitis. For that reason, a biopsy is mandatory for the correct diagnosis. The typical histological pattern of LMS is characterised by intersecting sharply marginated fascicles of spindle cells with eosinophilic cytoplasm and elongated hyperchromatic nuclei.Immunohistochemistry analysis of the lesions usually shows Vimentin, Desmin, Calponin, and Smooth Muscle Actine (SMA) reactivity. AE1/AE3 anti-cytokeratin antibodies can also be positive, due to the anomalous presence of keratin in these tumours.The overall incidence of LMS increases with age, as in soft tissue sarcomas in general, peaking in the seventh decade of life.Regarding the location of the tumor, De GrootSome risk factors were previously proposed for LMS. The most important is Epstein-Barr virus (EBV) infection, mainly in the setting of immunosuppression such as acquired immunodeficiency syndrome (AIDS), and immunosuppressants or steroids use.LMS is considered an aggressive type of tumor, capable of hematogenous dissemination , local recurrence, and metastasis .Surgical resection with negative margins is the preferred treatment for LMS.4Conjunctival LMS is a rare tumor, which should be suspected in patients with conjunctival tumours that look sub-epithelial. The diagnosis can only be made by histopathological and immunohistochemistry analysis. Moreover, due to the rarity of this condition, there is no standard treatment. However, the consensus is that a complete surgical removal with safety margins is mandatory. Globe-sparing procedures can be an option if the tumor is restricted to bulbar conjunctiva. In other cases, a more aggressive surgery, such as enucleation or evisceration is necessary, mostly because of the high recurrences rates of LMS.Patient has provided written informed consent.No funding or grant support.There are no conflicts of interest or financial disclosures for any of the authors listed.All authors attest that they meet the current ICMJE criteria for Authorship.We wish to confirm that there are no known conflicts of interest associated with this publication and there has been no significant financial support for this work that could have influenced its outcome.No funding was received for this work.We confirm that we have given due consideration to the protection of intellectual property associated with this work and that there are no impediments to publication, including the timing of publication, with respect to intellectual property. In so doing we confirm that we have followed the regulations of our institutions concerning intellectual property.We further confirm that any aspect of the work covered in this manuscript that has involved human patients has been conducted with the ethical approval of all relevant bodies and that such approvals are acknowledged within the manuscript.All listed authors meet the ICMJE criteria. We attest that all authors contributed significantly to the creation of this manuscript, each having fulfilled criteria as established by the ICMJE.We confirm that the manuscript has been read and approved by all named authors.We confirm that the order of authors listed in the manuscript has been approved by all named authors.The Corresponding Author declared on the title page of the manuscript is:Conjunctival Leiomyosarcoma.This author submitted this manuscript using his/her account in EVISE.We understand that this Corresponding Author is the sole contact for the Editorial process (including EVISE and direct communications with the office). He/she is responsible for communicating with the other authors about progress, submissions of revisions and final approval of proofs.aluisio.gameiro@unifesp.br.We confirm that the email address shown below is accessible by the Corresponding Author, is the address to which Corresponding Author's EVISE account is linked, and has been configured to accept email from the editorial office of American Journal of Ophthalmology Case Reports:


Retroperitoneal cavernous hemangioma (RCH) is a rare benign vascular malformation. Only a few cases of RCH were reported. Here we present a case of RCH in a 66-year-old female complaining of long-standing progressive dull abdominal pain. A 66-year-old female was visiting our teaching hospital complaining of long-standing dull abdominal pain radiating to the back, which become progressive and more evident in the last few months.She had a chronic history of hypertension and was on regular treatment. She underwent left hip replacement surgery for years, otherwise is no recent history of surgical intervention or recent trauma.trans-abdominal ultrasound examination, which confirms the presence of a left para-aortic large retroperitoneal mass lesion; then we requested a contrast-enhanced CT scan of the abdomen and pelvis.A palpable mass was felt by clinical examination, and then the patient was sent for a The CT scan was confirming the presence of a relatively well-defined retroperitoneal lobulated mass lesion seen extending along the left lateral wall of the distal abdominal aorta and left common iliac artery and anteromedial to the left psoas muscle. It showed heterogeneous intense peripheral enhancement during the porto-venous phase, the fat planes were preserved between the lesion and adjacent structures with no gross evidence of local invasion –3. The fSurgical resection of the tumor was performed using a lower midline incision and a Kocher maneuver was used for the dissection of the retroperitoneum. The mass was found, lying posterior to the left ureter and gonadal vessels (both of them isolated by a rubber tube) and compressing medially on the aorta above the bifurcation. The feeding arteries of the tumor were found to originate from retroperitoneal tissue instead of from the abdominal aorta, and each vessel was ligated before the tumor was removed. There was no evidence intraoperatively of the invasion of the surrounding structures. The mass was well capsulated showing a lobulated surface and dark fleshy color  and 5. TThe histopathology shows a well-capsulated mass composed of multiple variable sizes dilated vascular spaces consistent with a benign vascular tumor (cavernous hemangioma) with no evidence of atypia or malignancy .Hemangioma is a benign vascular tumor that may involve the skin, mucosa, or viscera such as the liver and spleen. It rarely involves the retroperitoneal organs such as kidneys, pancreas, and adrenal glands .Retroperitoneal hemangiomas are of the cavernous type and are commonly seen in pediatrics .Adult hemangiomas are distinct from pediatric hemangiomas, which proliferate during infancy and then involute slowly for several years, followed by eventual regression .Primary retroperitoneal cavernous hemangioma (RCH) is an extremely rare type of hemangioma , also itRCH are usually asymptomatic . The maiRCH can cause local invasion of adjacent structures and lead to aggressive symptoms and complications . IncreasOur patient was an old-aged female. In general, no sex predilection had been found for RCH, except for adrenal glands, which are more common in women with a female:male ratio of ~2:1 .Physical abdominal examination may reveal a palpable mass when it becomes large enough as in our case. Laboratory studies usually provide no additional indications for RCH .It is difficult to make a diagnosis of RCH even by various imaging modalities and the diagnosis of all reported cases is made by histopathological examination .However, the imaging findings of RCH may differ depending on the organ of origin, but in general, it shows well-defined hypoechoic and homogeneous lobulated lesions with positive internal vascularity by ultrasound examination . On CT aIn our case, the US and CT imaging characteristics were benign and near classical to RCH, so the differential diagnosis of benign retroperitoneal mass lesion was made including paraganglioma, neurofibroma, lipoma, teratoma and neurilemoma . ParaganOperative and nonoperative approaches are possible for RCH patients. Because of the complexity of retroperitoneal anatomy, RCHs often involve vital structures, making resection extremely technically challenging, and high-volume centers are required for optimal opportunities for surveillance and multidisciplinary care , 9.In our case, the tumor was localized and there was no evidence of local invasion or metastasis, we performed curative resection of the tumor to relieve the patient’s symptoms and to confirm the diagnosis of the benign lesion by histopathology.RCH is a rare adult mass lesion. The clinical diagnosis is difficult and imaging findings may also not be conclusive.The optimal management of RCH has not yet been standardized and based on the lesion location, size, growth rate, local effect and patient’s symptoms. However, surgical resection is a curative choice for RCH. It reduces the risk of hemorrhage and relieves the pressure effect and patient’s symptoms.None declared.None.


Liomyomas of the nasal cavity and paranasal sinuses are rare. They make up less than 1% of all leiomyomas in the human body. This is due to the paucity of smooth muscle in the nose. They are classified in three groups: leiomyoma, angiomyoma and epithelioid leiomyoma. Only 15 cases of vascular leiomyomas have been found in the literature. The treatment of choice is surgical excision. Hereby we present a new case and review the literature. The lesion grew slowly with small volume epistaxis episodes. After 3 years she developed nasal obstruction in he left side and local pruritus. In the physical exam and under nasal endoscopy we noticed a red lesion inserted on the left turbinate head, well outlined, measuring about 1 × 2cm, partially obstructing the nasal cavity. CT scan showed a round nodular lesion, which reacted intensely to contrast injection, having 1.2cm in diameter . We biopShe did not have any complications in her postoperative and has been disease-free for 10 months now.,Nasal cavity and paranasal sinuses vascular leiomyomas are extremely rare, making up less than 1% of all human leiomyomas. This rarity is partially due to the scarcity of smooth muscle in the nasal cavity, except for the vessels' walls.,Histological classification of tumors by the World Health OrganizationThere are reports of malignant tumor variants of this line. Lack of mitosis seems to indicate the tumor's benign characteristics.,13These tumors grow slowly and may persist for a long time.4Comparing literature data, we observed that the most frequent location of the vascular leiomyoma in the nasal cavity has been in the inferior turbinate. Of the 15 cases described , 5 origiAccording to Barr et al.,,Of the 15 cases aforementioned, 13 were women; making up a female predominance of 87.5% if we include the present case.,,,,At the time of diagnosis, the patients' ages ranged from 25 to 76 years, with an average of 55.6 years.Radiologic studies, such as CT scan or MRI do not help much in diagnosis, however it is important to establish the extension of the lesion and treatment planning.,,,,,Treatment for these tumors is based on local resection, and there are no reports of recurrence after total excision.Nasal cavity vascular leiomyomas are extremely rare. The exact origin of this neoplasia is still uncertain, however it is believed that it comes from the muscle cells present in blood vessels' walls. Resection is the procedure of choice and it has high cure rates. The endoscopic procedure is a good option for small to moderate extension tumors.


To explore the incidence, imaging and treatment of solid pseudopapillary tumor of pancreas in children, and Summarize the experience of treatment.The clinical data of 18 children with a solid pseudopapillary tumor of the pancreas treated in our hospital from January 2012 to June 2021 were analyzed retrospectively. The age range was 8–16 years old, the average age was 11.67 years old, and the median age was 11.5 years old, namely, three boys with an average age of 10 years old and 15 girls with an average age of 12 years old. In total, two cases were admitted to the hospital because of trauma, seven cases were found in physical examination, and nine cases were admitted with the abdominal pain as the main complaint. Enhanced CT examination was performed before operation, nuclear magnetic resonance examination and abdominal ultrasound examination were performed in some children, and plain CT scan was performed after operation.Solid pseudopapillary tumor of the pancreas can occur in all parts of the pancreas, especially in the body and tail of the pancreas. Among the 18 cases, SPN occurred in the head of pancreas in 5 cases (27.78%), the neck of pancreas in 2 cases (11.11%), and the body and tail of pancreas in 11 cases (61.11%). All the 18 children were treated by operation. among them, 4 cases underwent choledochal pancreatico duodenectomy (1 case), 4 cases underwent pancreaticoduodenectomy combined with splenectomy (3 cases), 6 cases underwent spleen-preserving pancreatectomy / tail pancreatectomy (1 case), 3 cases underwent enucleation of pancreatic tumor due to exogenous growth, 1 case underwent laparoscopic partial pancreatectomy and pancreaticoenterostomy. Laparotomy was performed in 12 cases and endoscopic surgery in 6 cases. Postoperative pathology confirmed solid pseudopapillary tumor of the pancreas. None of the patients received radiotherapy and chemotherapy after operation and were followed up for 6 months to 2 years. There was no recurrence, metastasis or pancreatic dysfunction.Solid pseudopapillary tumor of pancreas in children is a rare, low-grade malignant solid tumor with no specific clinical manifestations and laboratory examinations. Preoperative diagnosis mainly depends on enhanced CT. Surgical resection of tumor is a reliable treatment, and the specific operation is mainly based on the experience of the chief surgeon, the location of the tumor and the invasion of surrounding tissue. At present, there is no evidence of the effectiveness of other treatment options, and surgical resection of the tumor has a good prognosis. Solid pseudopapillary neoplasm (SPN) of the pancreas is a rare pancreatic tumor with malignant potential, accounting for ~1%−3% of the pancreatic tumors . The disThis was a single-center retrospective case series. All the surgeries were carried out by members of the same surgical team. Medical records were analyzed by two surgeons (AY and WH) for data, namely, radiological findings, operation records, and medical files. The clinical data of 18 patients with SPN in the First Affiliated Hospital of Xinjiang Medical University from January 2012 to June 2021 were analyzed retrospectively. The clinical manifestations, imaging features, tumor location, pathology, surgical treatment, and prognosis were analyzed to summarize the treatment experience. The main postoperative follow-up was an outpatient follow-up from 6 months to 2 years. Abdominal routine ultrasound or plain CT examination was performed at 6, 12, and 24 months after the operation. During the follow-up period, there were no long-term complications, such as tumor recurrence, metastasis, diabetes with dyspepsia, or pancreatic dysfunction. There was no significant difference in growth and development between children and children of the same age. Patients who were lost to follow-up or not willing to participate in the present study were also excluded. All the enrolled patients were informed and consented to about this study after permission from the Ethics Committee of our institute.All the patients with SPN were examined by the pancreatic computed tomography (CT), magnetic resonance imaging (MRI), or ultrasound after admission. According to the imaging findings, the tumors were divided into three types: cystic, solid, and cystic-solid. At the same time, the relationship between the lesion and the pancreas  and whether adjacent organs were involved were recorded. The tumor diameter was defined as the maximum cross-sectional diameter of the tumor on CT/MRI and measured.Statistics on the methods of laparotomy and laparoscopic surgery were made by consulting surgical records. All the 18 children were treated by operation, which depends on the location, size, and close relationship with the surrounding organs and tissues of the tumor. These include local resection of the focus, pancreaticoduodenectomy, resection of the head of the pancreas with preservation of the duodenum, resection of the body and tail of the pancreas, resection of the body and tail of the pancreas + splenectomy, surgical biopsy, lymph node dissection and pancreatic focus combined with other operations.The judgment of postoperative complications was based on the postoperative course records and the results of postoperative blood routine, biochemical routine, drainage fluid biochemistry, and bacterial culture. According to the Clavien-Dindo classification of the postoperative complications , postopeStatistical analysis was performed using SPSS 25.0 statistical software . A descriptive study was used to study the clinical characteristics of the patients. The measurement data are expressed as the mean ± SD, and the counting data are expressed as a percentage (%).Of the 18 patients, three (16.7%) were males, and 15 (83.33%) were females, with an average age of 11.6 years (8–16 years). Among the 18 children, two (11.11%) were found after admission because of the trauma, and seven (38.89%) were found during physical examination. A total of nine (50.00%) cases were admitted to the hospital with abdominal pain as the main complaint. The nature of abdominal pain was intermittent epigastric pain and no radiation pain elsewhere, of which three cases were accompanied by vomiting. In the physical examination of admission, the abdominal mass was palpable in four (22.22%) cases, which was hard, had a poor range of motion, and had no tenderness .All 18 children underwent enhanced CT examination before the operation, showing solid, cystic solid, or cystic round-like masses, namely, seven cases (38.89%) with solid or solid components, six cases (33.33%) with cystic solid components, and five cases (27.78%) with cystic or cystic components. The tumor was located in the head of the pancreas in five cases (27.78%), the neck of the pancreas in two cases (11.11%), and the body and tail of the pancreas in 11 cases (61.11%). A total of 11 cases of SPN were considered to be diagnosed after perfecting CT, and the sensitivity was 61.11%. The typical CT features were cystic-solid mass, low density, or isodensity on the solid part . On contOf the five children with tumors located in the head of the pancreas, four underwent choledochal pancreaticoduodenectomy (one case was performed under laparoscopy). Tumor enucleation was performed in one case because the tumor was exophytic with an intact capsule . Among tThe postoperative hospital stay was 5–15 days, and complications occurred in two cases (11.11%). Among them, one case had pancreatic fistula, which was improved after fasting water, enzyme inhibition, and drainage. In total, one case had incomplete intestinal obstruction, which was improved after fasting water and acid inhibition treatment. No complications were found in other cases.All the 18 children achieved complete resection of the tumor. The average maximum diameter of the tumor was 6.7 ± 2.43 cm. Ten cases (55.56%) had intact capsules, and four cases (22.22%) had peripheral tissue invasion and lymph node dissection, but no metastasis was detected. It is generally a round or quasi-round mass, the surface is smooth, the capsule is intact, and the section can be solid, cystic, or cystic , 11. TheSolid pseudopapillary neoplasm can occur in all parts of the pancreas, mostly, in the body and tail of the pancreas . Of the Solid pseudopapillary neoplasm has no specific tumor markers, and other routine laboratory tests, such as blood glucose and amylase, are not special, which have no diagnostic significance for the disease. At present, the preoperative diagnosis of the disease mainly depends on CT and MRI. Its special pathological manifestations are solid, cystic-solid, and cystic structures, and also its lack of blood supply . It is sPathologically, SPNs are similar to pancreatic neuroendocrine tumors, but it is sometimes difficult to distinguish them by routine pathology, and most of them can be distinguished by immunohistochemistry. The tumor AAT β-the expression rates of catenin, vimentin, CD56, and CD10 were high . With thAt present, surgical resection of tumors is the only effective way to treat the disease. At present adjuvant therapy is reported in individual cases, and the drugs used are not the same. All kinds of guidelines actively recommend surgical treatment, but the scope and method of surgical resection are not standardized at present. The postoperative life expectancy for children is longer than that of adults, so more destructive surgical procedures, such as standard pancreaticoduodenectomy and combined splenectomy, should be used with caution. The biological characteristics of SPNs are potentially malignant or low-grade malignancy, poor invasiveness, and low-metastasis and low-recurrence rates . Even ifFor SPNs located in the head of the pancreas, in addition to traditional pancreaticoduodenectomy, pancreaticoduodenectomy with preservation of the common bile duct, pancreaticoduodenectomy with preservation of the pylorus, and resection of the head of the pancreas with preservation of the duodenum can be selected . If the The spleen is an important immune organ that may cause explosive infection and septicemia after splenectomy, so preserving the spleen is more important for children than adults. If the adhesion between the tumor and the splenic vein is serious, a combined splenectomy can be selected when it is difficult to separate during the operation. However, during the operation, we can also try to remove the splenic vein and preserve the splenic artery and spleen. The difficulty of this operation is to deal with the splenic vessels well. If the splenic vessels cannot be treated properly, it may lead to splenic infarction and splenic abscess . BecauseWith the increasing maturity and popularity of laparoscopic technology, an increasing number of operations are being performed through laparoscopy . CompareSolid pseudopapillary neoplasm is a rare and potentially malignant tumor in children. Its clinical manifestations are not specific, and there is no specific laboratory examination. Preoperative diagnosis depends mainly on imaging examination, and the gold standard of diagnosis is histopathological examination. The degree of malignancy of SPNs is low, and surgical resection can achieve a clinical cure. Therefore, to completely remove the tumor, retain glandular function as much as possible, and reduce postoperative complications, we should strictly grasp the surgical indications and formulate a reasonable surgical plan so that children can benefit more from the process of diagnosis and treatment.The original contributions presented in the study are included in the article/supplementary material, further inquiries can be directed to the corresponding author.The studies involving human participants were reviewed and approved by Medical Ethics Committee of the First Affiliated Hospital of Xinjiang Medical University (K2021110-05). Written informed consent to participate in this study was provided by the participants' legal guardian/next of kin. Written informed consent was obtained from the minor(s)' legal guardian/next of kin for the publication of any potentially identifiable images or data included in this article.AM and HW: conducted the study, collected, analyzed and interpreted the data, and wrote the manuscript. YW: statistically analyzed and interpreted the data. WL: planned the project and reviewed the manuscript. All authors contributed to the article and approved the submitted version.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.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


A serval  presented for progressive enlargement of the right adrenal gland, which had been found incidentally on abdominal ultrasound 2 years previously and upon subsequent ultrasound examinations enlarged progressively from 1.26 to 1.43 cm.Clinical signs had not been recorded by the zookeeper, however, progressive weight gain and lethargy were reported. Computed tomography (CT) confirmed the presence of a right caudal pole adrenal mass measuring 1.8 cm.via a ventral midline laparotomy that included the resection of the right phrenicoabdominal vein in association with the mass. Histopathological examination identified the mass as an epithelial cyst. Chromogranin A staining for a possible pheochromocytoma was performed but was negative. The serval recovered at the zoo without complication and has been on display for 18 months.The right adrenal with associated mass was surgically resected Epithelial adrenal cysts have not been previously reported in felids. An adrenal cyst should be included on the differential list for any animal with an enlarged, slowly growing adrenal gland mass with non-specific clinical signs. Adrenal cysts are rare and have not been reported in a feline to the authors' knowledge. As adrenal cysts are uncommonly reported in humans, there is no current consensus regarding the best management and treatment . MultiplAn 11-year-old female serval  from Wellington Zoo was presented for a right adrenal mass, which had been progressively enlarging over the previous several months. The mass was initially found incidentally on abdominal ultrasound approximately 2 years previously while undergoing evaluation following a foreign body small intestinal obstruction (wood chip mulch). At initial diagnosis, the right adrenal gland was measured as 1.26 cm in comparison to the left adrenal gland, which measured 0.5 cm .via small intestinal enterotomy at exploratory laparotomy surgery 2 years previously was reported by the zookeeper as uneventful, and no further gastrointestinal signs were noted.The zookeeper did not report any clinical signs other than lethargy and progressive weight gain over the last 2 years. Recovery following wood chip foreign body removal Successive abdominal ultrasounds indicated that the adrenal mass was expanding without evidence of invasion into the surrounding caudal vena cava or renal vasculature. The mass was encroaching on the right phrenicoabdominal vein but invasion into the vein could not be determined using ultrasound, and computed tomographic evaluation was recommended .Physical examination on the presentation for computed tomographic evaluation was limited due to the felid's fractious demeanor. While under general anesthesia, vital parameters, such as pre-operative blood pressure , heart rate (80 bpm), respiratory rate (20 bpm), and thoracic auscultation, were within normal limits for felid species under general anesthesia, however, her body condition score was 9/9. A blood sample was collected and found incompatible on major and minor cross-match with two donor domestic cats.A triple phase-contrast abdominal computed tomography (CT) was performed to fully assess the adrenal mass and thoracic CT for metastasis evaluation. The cranial pole of the right adrenal was markedly enlarged at 1.8 cm diameter × 3.4 cm craniocaudal length with a hypoattenuating nodule in the caudal pole measuring 1.8 cm in diameter. The nodule had marked rim enhancement and mild homogenous central enhancement in the arterial and venous phases. The right phrenicoabdominal vein was in close apposition to the caudal aspect of the adrenal nodule and the right adrenal gland was in close apposition to the caudal vena cava without visible flattening or filling defects . The lefThe CT findings were consistent with a right adrenal mass without invasion into the caudal vena cava or renal vasculature. Differential diagnoses included possible neoplastic processes, such as adrenocortical adenoma and carcinoma or pheochromocytoma, although nodular hyperplasia could not be ruled out. Surgical exploration and right adrenal resection were recommended for excisional biopsy to determine the diagnosis.via ventral midline celiotomy under general anesthetic, the previous enterotomy site was visualized in the proximal to mid jejunum with omental adhesions. The right kidney and adrenal gland were palpable, however, they could not be visualized due to abundant retroperitoneal fat. Malleable retractors were utilized to improve visualization and the overlying fat was carefully dissected following incision into the peritoneum overlying the right kidney until the adrenal gland and associated mass were visible . Recovery from anesthesia was uneventful. The sample was submitted for special immunohistochemical staining with Chromogranin A, as the intraoperative hypertension raised the possibility of an unusual pheochromocytoma.Upon exploratory laparotomy  visible . The cauHistopathology showed an expansile, encapsulated cyst lined by a single layer of plump to attenuated to cuboidal to columnar epithelial cells . The cysSince the surgery, the serval has been doing well, showing no clinical signs with a healthy appetite and normal activity.Adrenal cysts are rare and have not been reported in a feline in the literature to the authors' knowledge. Other than compression atrophy of the adrenal parenchyma and potential for the rupture with associated inflammation, they are considered benign incidental findings.An attempt has been made to categorize adrenal cysts in humans and the currently adopted method categorizes them into four main subtypes; epithelial, endothelial, pseudocysts, and parasitic hydatid cysts , 6, 7. AAdrenal cysts are also uncommonly reported in humans and therefore a consensus has not been reached in terms of management and treatment . DefinitIn humans, some authors have recommended surgical excision of all symptomatic functional cysts, cysts >5 cm in size due to risk of hemorrhage, and cysts with a hemorrhagic or heterogeneous nature on imaging suggestive of malignancy –4. PercuThe blood pressure of this patient was increased during the surgical manipulation of the adrenal epithelial cyst. Intra-operative hypertension is often seen during surgical removal of pheochromocytomas due to surges in catecholamine concentration in the blood . This inLimitations of the case reported in this article include limited conscious examination of the animal due to its fractious nature and reporting on the surgical intervention and outcome of a single patient. However, having repeating ultrasound examinations followed by a triple phase-contrast CT scan of the abdomen allowed a thorough depiction of the adrenal epithelial cyst and histopathology of the excised mass with the inclusion of special stains allowed for a definitive novel diagnosis.In conclusion, benign adrenal cysts should be included on the differential list for an enlarged adrenal gland or an adrenal mass found in imaging studies. Current treatment and monitoring recommendations must be extrapolated from the human literature as there have not been adrenal cysts previously described in the veterinary literature for felines. As adrenal cysts are rare in humans, controversy still exists as to the best treatment option. Due to the non-specific clinical signs and imaging characteristics of cysts, surgical excision should be considered for all masses as this will allow a definitive pathological diagnosis and prevent interpretation of a malignant tumor on imaging as a benign incidentaloma .The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.Ethical review and approval was not required for the animal study because this study was a retrospective analysis of the medical records from the School of Veterinary Science and therefore did not require approval by an Animal Ethics Committee. Written informed consent was obtained from the owners for the participation of their animal in this study.SD drafting of manuscript and literature review. WB editing of manuscript. SP histopathological analysis and drafting summary of findings for this article. MO imaging interpretation and drafting summary of findings for this article. All authors contributed to the article and approved the submitted version.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.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


Thecoma is a commonly benign ovarian tumor of the group “Sex cord‐stromal neoplasms.” This group represents <5 percent of ovarian tumors. Thecoma is extremely rare in pregnancy. Here, we describe a 17‐week pregnant woman presenting with acute ovarian pedicle torsion as a result of an ovarian thecoma. Thecoma is a type of sex cord‐stromal neoplasm, which is an infrequent ovarian tumor and extremely rare in pregnancy. It is important to consider such neoplasms in pregnant women suspected of adnexal mass, as the probability of ovarian tumor rupture or pedicle torsion increases during the course of pregnancy. They are often benign tumors that arise from perifollicular stromal cells and mostly presented in postmenopausal women.2A 35‐year‐old gravida 3 para 0 (abortion 2) woman presented to the department of obstetrics emergencies with chief complaint of abdominal pain. The pain was predominantly in the hypogastric and right lower quadrant regions, had initiated a week earlier, and exacerbated since the night before. The patient expressed the presence of nausea and occasional vomiting since the beginning of pregnancy. She had no specific past medical history other than the two abortions  and a surgical correction of ocular strabismus at the age of 22. She had regular menstruation of 28‐day cycles.On physical examination, the patient was relatively ill but not toxic, vital signs were stable, and abdominal examination revealed hypogastric tenderness. The patient was admitted and monitored in the emergency ward, and color Doppler ultrasound was requested with the initial diagnosis of ovarian torsion. Ultrasound study reported a normal 20 × 36 mm left ovary, and a 34 × 87 millimeter right adnexal mass, with whirlpool sign on the infero‐medial region Figure . Due to The patient was transferred to the operating room, and a mini‐laparotomy incision was performed. A cyst with a white appearance and firm consistency was seen in the right ovary with three rotations. Tumor detorsion was first performed, and then the tumoral mass was resected and sent for pathologic examination  arising from the ovarian stroma. Clinical manifestations are not very much specific from other ovarian tumors and preoperative diagnosis is challenging, as it may cause abdominal pain, abdominal distension, and pelvic mass. These tumors rise in postmenopausal women and are rare before the age of 40. Most of thecoma lesions are unilateral, various in size, shape, and even pathologic features as they may coexist with other sex cord‐stromal tumors like fibroma and granulosa cell tumors. Nearly, one‐third of these tumors can manifest hormonal production such as estrogen with clinical presentations such as abnormal menstrual bleeding, virilization, menstrual irregularities, and even infertility. Thecoma and fibromatous tumors can also be associated with ascites and sometimes pleural effusion.In the present case, the main complaint was abdominal pain, and pelvic fluid was noticed in ultrasound examination, in addition to the ovarian torsion. However, the patient's emergency situation made it impossible to assess other clinical and paraclinical manifestations.The general sonographic features of ovarian thecoma‐fibroma group are adnexal masses, which are hypoechoic with well‐demarcated borders and acoustic attenuation. There are minimal Doppler flow signals unless a complication like malignancy or torsion is present. Most of the time, the tumor is unilateral with a diameter smaller than 5 cm. The tumor diameter is reported to be significantly correlated with CA125 level and the volume of ascites fluid.The present case was not diagnosed with an ovarian tumor until the increase in tumor size along with the enlargement of the uterus led to torsion and acute pain. Ultrasound demonstrated the whirlpool sign of ovarian torsion, which is characterized by the appearances of a twisted ovarian pedicle.In terms of pathologic examination, microscopic examination of thecoma usually demonstrates a primarily diffuse growth with varying degrees by hyaline plaques, nodular pattern, and calcification. Forty percent of the tumors are reported to have a fibroma component. The differential diagnosis is predominantly with other sex cord‐stromal neoplasms, particularly other stromal tumors, steroid cell tumor, and adult granulosa cell tumor.Fertility‐sparing surgery is the general approach for sex cord‐stromal tumors, as they tend to present in young patients with early stage disease. Complete surgery  is considered in women older than child‐bearing age, and chemotherapy is reserved for advanced disease or recurrence.Ovarian sex cord‐stromal tumors, especially thecomas, are rare complications in pregnancy. The behavior of the disease, the clinical course, and fetomaternal outcomes are not fully understood in such cases. Blake et al. performed a systematic review on fetomaternal outcomes of pregnancies complicated by ovarian sex cord‐stromal tumors. The most common histologies were granulosa cell tumor (22.0%), thecoma (18.6%), and Sertoli–Leydig cell tumor (8.5%), respectively. The majority of cases resulted in successful live birth, with most cases being benign localized tumors which underwent unilateral adnexectomy. Despite the favorable results mentioned above, these conditions are considered high‐risk pregnancy. Risk factors such as younger age (<30 versus ≥30), large tumor (size ≥15 cm versus <15 cm), and stage (higher than I) increase the chance of adverse events.Considering the specific diagnosis of thecoma in pregnancy, reported experiences in literature are merely case reports. Okada et al. reported thecoma in a pregnant woman in first trimester which presented with abdominal distention due to ascites, and elevated serum CA125 levels. The patient underwent ascites aspiration and unilateral adnexectomy.4Thecoma is a predominantly benign tumor of ovary, which belongs to sex cord‐stromal tumors' category. Ovarian torsion in pregnancy can occur due to an ovarian thecoma. Although most tumors of this group are safely treated with fertility‐sparing surgery and have favorable fetomaternal outcomes similar to the present case, it is important to differentiate the lesions from malignant tumors through careful pathological examination and consider evaluation of risk factors, which are reported to be associated with worse outcomes.A.M. and M.K. contributed to conception, design, and drafting of the manuscript. Authors A.M. and A.z.M. contributed to data collection.The authors have no conflict of interest to declare.Written informed consent was obtained from the patient to report their case, and the manuscript was approved at the Ethics Committee of Mashhad University of Medical Sciences.Published with written consent of the patient.


Cavernous haemangiomas of the rectosigmoid colon are rare benign vascular neoplasms of the GI tract. Patients usually present at a younger age with various degree of rectal bleeding ranging from mild painless episodic bleeding to life-threatening hemorrhage. High index of suspicion and early diagnosis is crucial to avoid unnecessary biopsy and inappropriate management. We report a case of a 26-year-old male patient with a long history of recurrent rectal bleeding. Contrast-enhanced CT scan of the abdomen and pelvis and MRI confirmed the diagnosis of cavernous hemangioma. Further surgical treatment with rectosigmoid resection and colo-anal anastomosis represents the optimum path of management for our patient. Bleeding is not associated with defecation and the amount of blood is variable. At times, it is associated with abdominal pain and loose stools. The patient gave a surgical history of undergoing surgical treatment of a rectal vascular malformation at the age of 8 years. No other significant medical history was given.On physical examination, the patient vitals were within the normal limits. Chest was clear and abdomen was soft to palpation with evidence of a transverse scar as well as a colostomy scar suggestive of previous surgery. Per rectal examination denoted multiple nodularities in the rectum and fresh blood was observed. Laboratory blood work-up revealed a picture of microcystic anaemia and occult blood stool was positive.Colonoscopy showed multiple congested large swellings extending 7 to 15 cm from the anal verge with normal appearing mucosa above that level suspicious for a vascular aetiology thus no biopsy was taken . Upper GContrast-enhanced computed tomography of the abdomen and pelvis showed nodular chunky calcifications with central lucencies likely representing phleboliths with diffuse circumferential thickening of the recto-anal wall and a right-sided perirectal serpiginous structure likely of vascular origin. Nonetheless no contrast enhancement was appreciated. The rest of the bowel appeared grossly normal. No evidence of bowel obstruction was seen. Solid organs appeared normal. Focal nodal calcification was seen in the small bowel mesentery likely representing an old healed inflammatory or infectious process. Gadolinium-enhanced magnetic resonance imaging of the abdomen and pelvis revealed circumferential submucosal thickening of approximately 7 cm in length up to 2.5 cm from the anal verge appearing low on T1 sequences and high on T2 sequences with heterogenous enhancement and multiple signal voids suggestive of calcifications. No evidence of restricted diffusion. Multiple prominent enhancing vessels in the post-contrast images were noted. The prostrate and seminal vesicles were slightly displaced due to the rectal wall thickening. Urinary bladder appears normal with no evidence of free fluid or pelvic collection. A digital subtraction angiogram of the internal iliac arteries identified prominent middle rectal artery branches supplying the rectal wall swellings . Nonethe1,2 Intestinal haemangiomas are rare accounting for only 0.05% of all the GI tumors and usually present in children and young adults with history of painless, recurrent rectal bleeding and anaemia.2,4 The rectosigmoid colon is the commonest site of involvement accounting for 50–70% of the cases.4,5Intestinal haemangiomas are usually solitary and occasionally associated with certain syndromes such as Osler-Weber-Rendu syndrome, Maffucci syndrome and Klippel Trenaunay-Weber syndrome.6 There are three types of haemangiomas: cavernous, capillary and mixed haemangiomas with cavernous hemangiomas being the commonest type.1Haemangiomas are benign vascular lesions that can involve any part of the body including the gastrointestinal tract.1,2 The gold standard diagnostic tool of choice is video-colonoscopy as it is a non-invasive technique that requires no radiation and which accurately localises and depicts the morphology and size of the lesion as well as excludes other synchronous lesions.1 On colonoscopy, cavernous haemangiomas appear as solitary or cluster of bluish or red submucosal soft lesions that are easily compressible on insufflation with adjacent engorged veins in the rectal wall.2,3 Due to the vascular nature of the lesion, biopsy is not recommended.5Due to rarity of intestinal haemangiomas these lesions are usually misdiagnosed in 80% of cases for other commoner etiologies of lower GIT bleeding including internal hemorrhoids, inflammatory bowel disease and colorectal cancer.3 CT scan of the pelvis can confirm the diagnosis as it shows concentric thickening of the rectosigmoid colon with multiple intralesional phleboliths and heterogeneously enhancing perirectal soft tissues.4,6 Due to risk of radiation from multiple follow-up scans on these young patients, MRI is a suitable alternative imaging modality. Rectosigmoid haemangiomas appear as wall thickening with high T2 signal intensity due to slow flow voids within the vascular malformations and progressive nodular enhancement on post-contrast images.6 Moreover, 2T-weighted images aid in differentiating colorectal carcinoma from haemangiomas which usually show intermediate signal on T2 images in comparison with the adjacent high signal intensity rectal fat and low signal intensity colonic muscular layer.4Abdominal radiographs usually show multiple scattered pelvic phleboliths which represent thrombosed and calcified intralesional vascular channels.7 Abdominoperineal resection (APR) was considered the standard treatment previously, however due to the resultant permanent stoma and common complications such as sexual and urinary dysfunction, it is currently a less desirable approach.8 Advocated colo-anal sleeve anastomosis is instead the new popular approach that allows resection of the lesions without a permanent stoma.8 DCHR may not be completely resolved by non-operative therapies such as sclerosing injections, microwave treatment, and trans-fixation or ligation of the internal iliac artery and recurrent rectal bleeding is often common after these treatments.8Surgical resection is reserved for larger and diffuse rectosigmoid haemangiomas, while smaller submucosal haemangiomas can be resected endoscopically.We reported a case of a 26-year-old male patient with rectosigmoid hemangioma. While this entity is a rare cause of rectal bleeding, it should be kept in the differential diagnosis especially in young patients presenting with a long history of episodic hematochezia. Video colonoscopy is the gold standard diagnostic tool and CT and MR imaging play a crucial role in confirming the diagnosis and evaluating the extent of the disease prior to surgical management.Proper evaluation of diffuse cavernous haemangiomas with diagnostic imaging modalities is necessary while investigating patients with rectal bleeding in order to reach a definitive diagnosis.Colonoscopy and MRI are cornerstones for the correct diagnosis of rectal cavernous haemangiomas.Bleeding is the most feared complication because of the high vascularity of the rectal cavernous haemangiomas.Careful assessment of the whole of the gastrointestinal tract is needed to rule out diffuse involvement.Surgical resection is the definitive management.


Solitary rectal ulcer syndrome (SRUS) is a benign and chronic disorder well known in young adults that is characterized by a series of symptoms such as rectal bleeding, copious mucus discharge, prolonged excessive straining, perineal and abdominal pain, a feeling of incomplete defecation, constipation and, rarely, rectal prolapse. The etiology of this syndrome remains obscure, and the diagnosis is easily confused with that of other diseases, contributing to difficulties in treatment. We present a case of a 37-year-old male with a nonulcerated rectal lesion grossly resembling a superficial depressed rectal cancer misdiagnosed in another hospital and describe its appearance on endoscopy and in the analysis of its pathological manifestations. The aim of this case report is to report an easily misdiagnosed case of SRUS, which needs to be distinguished from superficial rectal cancer, which should be educational for endoscopists. Solitary rectal ulcer syndrome (SRUS) is a rare benign rectal disease that is characterized by a combination of symptoms, clinical findings and histological abnormalities . HoweverA 37-year-old male had recurrent abdominal pain, diarrhea and hematochezia for 1 year. The patient had several bad habits such as tobacco use, alcohol consumption and betel quid chewing, and had no previous family history of cancer or special sexual behavior. This patient was recommended to receive endoscopy. Regrettably, he was diagnosed with superficial depressed rectal cancer in other two hospitals. He was then transferred to our hospital in preparation for surgery. The preoperative endoscopy discovered a reddish and irregular but well-defined 0-IIc lesion in the anterior wall of the rectum 4–6 cm from the anal margin a. MagnifSRUS is an unusual benign rectal disorder ,5. SeverIn this case, the lesion appeared as a nonulcerated rectal lesion, with a superficial depressed area. The patient was misdiagnosed with superficial depressed rectal cancer in other two hospitals. Related studies reported that superficial depressed cancers arose through the de novo pathogenic sequence and had a higher tendency for early invasions ,10. As aSRUS is an already well-known but easily misdiagnosed condition; the proper diagnosis and treatment of SRUS remain important challenges. It is worth noting that its rare occurrence usually leads to the fact that it is not properly diagnosed due to the lack of knowledge or lack of experience of doctors. The diagnosis of SRUS can usually be performed by a combination of symptomatology, endoscopy and histology. However, patients sometimes have typical symptoms without typical endoscopic findings. As mentioned above, this lesion did not present with typical ulcerative changes, but presented with superficial depressed changes. We used magnifying NBI and chromoendoscopy to observe this lesion and biopsied again, thus ruling out the possibility of rectal cancer, and finally reached the correct diagnosis. Although there is a little regret due to the fact that the patient strongly demanded ESD for a definitive diagnosis, we believe this is the most fortunate outcome for the patient, as he avoided surgery or even the risk of a permanent fistula.We consider this case to be a good learning opportunity for gastroenterologists, as when they encounter similar cases, SRUS should be one of the options in the differential diagnosis list.Not all SRUS cases present ulcers. Patients with typical symptoms and nonulcerated rectal lesions should be differentiated from those with superficial rectal cancer. Magnifying NBI and chromoendoscopy are useful, and histopathological examination should be performed to confirm the diagnosis.


Low-grade appendiceal mucinous neoplasm is a tumor of the appendix whose major clinical relevance derives from its inherent potential for peritoneal dissemination as pseudomyxoma peritonei. It sometimes poses challenges in clinical, radiological, and pathological diagnosis, and it may be confused with gynecological conditions in females. We report a case of low-grade appendiceal mucinous neoplasm presenting as firm sclerotic cystic mass and was initially suspected to be an ovarian mass in a postmenopausal woman. We review the literature for the pathogenesis and clinical implication of sclerohyalinization in mucinous appendiceal tumors. Neoplasms of the vermiform appendix are rare, estimated to occur in 2% percent of all appendectomy tissues . PrimaryA 67-year-old African woman, 20 years postmenopausal, and on treatment for diabetes and hypertension, presented with a right lower abdominal swelling of five-month duration. The swelling was of gradual growth with intermittent dull ache that preceded the swelling by a few months. On physical examination, her vital signs were normal. Abdominal palpation revealed a mobile, nontender, soft right lower abdominal mass. A provisional diagnosis of right iliac fossa mass probably of ovarian origin was made prompting a review by a gynecologist. Transabdominal ultrasound scan showed a large, irregular shaped, complex heteroechoic mass, that abuts the right aspect of the urinary bladder and the uterus. The mass had a well-defined, relatively hyperechogenic thick margin and measured 15 × 7.17 × 10.5 cm. Computed tomography (CT) scan was requested for, but the patient could not afford it. She was transferred to the general surgery unit where she underwent exploratory laparotomy.The finding at laparotomy was a large caecal mass which was adherent to the uterus and bladder. A right hemicolectomy and ileo-transverse 2-layer anastomosis was done with good outcome. She has remained in good health 2 years post-surgery.Pathology: Gross examination showed a right hemicolectomy tissue. The appendix was transformed into a firm whitish cystic mass measuring 8 cm × 7 cm × 7 cm. The cyst contained gelatinous mucin, and its wall measured 1 cm in thickness . No perfHistologic sections of the appendix showed mainly intense sclerosis and hyalinizing fibrosis with the mucosal or epithelial tissue denuded in most sections. The lumen showed adherent mucin. Extensive sampling of the mass revealed foci of flat single layer of neoplastic epithelium resting on a fibrotic wall. The epithelial cells were dysplastic with some showing cytoplasmic mucin. The layer of neoplastic epithelium showed outpouchings into the fibrous wall, but no areas of destructive invasion were seen Review of existing literature on appendiceal neoplasm portrayed some controversy which revolved around what should be the right taxonomy for mucinous neoplasms, a group of neoplasm that often showed a wide range of clinical behavior varying from indolent to aggressive , 4. DiffThe present case was cystic with thick hyalinized wall and was adherent to adjacent organs. Long-standing cases of LAMN can grow to a massive size of up to 29 cm with dense fibrous adhesion to surrounding structures . The preLow-grade mucinous neoplasms, having little capacity for deep or widespread visceral invasion, produce dire clinical consequences through copious mucin secretion. MUC2 and MUC5AC genes responsible for secretion and deposition of gel-forming mucin are strongly expressed in all mucinous tumors of the appendix and most PMP irrespective of nuclear grade . Mucin tImaging plays a role in the pre-surgical diagnosis and evaluation of LAMN, and aids in follow up of patients for possible complications, such as PMP. Modalities used include ultrasonography, computed tomography (CT), and magnetic resonance imaging (MRI). The characteristic well-demarcated appearance seen at CT imaging of LAMN has been ascribed to high incidence of fibrosis and mural hyalinization .Low-grade appendiceal mucinous neoplasm has prominent fibrogenic potencies. It sometimes poses challenges in clinical, radiological, and pathological diagnosis; it may be confused with gynecological conditions in females, while some cystic fibrous tumors require extensive sampling before the characteristic neoplastic epithelium can be identified.


Complex regional pain syndrome (CRPS) is a potentially devastating condition that can result in severe psychological and social morbidity. It is a diagnosis of exclusion, and other pathologic entities must be ruled out first. Glomus tumors are exquisitely painful benign vascular tumors that are most common in the hand and are rarely found in the lower extremity. Here, we present a case of a patient who developed a focus of severe anterior knee pain and tenderness a few months after a car accident that had been misdiagnosed as CRPS for 15 years. She coincidentally developed a sarcoma of her ipsilateral leg distal to this site. Magnetic resonance imaging of the sarcoma included the area of knee pain where, interestingly, it identified a separate small soft-tissue mass. A glomus tumor was diagnosed histologically in a needle biopsy specimen from this mass, which was resected along with the sarcoma. For the first time in 15 years, despite the additional sarcoma surgery, she reported relief of her pain and complete resolution of her “CRPS.” The Budapest diagnostic criteria comprise the most widely used system for diagnosing CRPS3456 and emphasize, “There is no other diagnosis that better explains the symptoms.” Patients often present with skin-related changes, as well as severe and often debilitating pain. The diagnosis and treatment of CRPS may be challenging. Other maladies often considered in the differential diagnosis include infection, deep vein thrombosis, Raynaud disease, thoracic outlet syndrome, vascular insufficiency, and psychiatric diseases.8 However, the diagnostic pathways described in the literature often neglect to mention soft-tissue tumors.Complex regional pain syndrome (CRPS) is characterized by continuous localized pain that is disproportionate to any causative trauma or lesion. The pain is considered regional because it is not in an identifiable nerve distribution or dermatome, and it may consist of a constellation of abnormalities in sensation or motor function along with vasomotor, sudomotor, or trophic findings.9 rare cases have been reported in the lower extremity.1011121314 Glomus tumor characteristics include pain and tenderness at the location of the nodule and temperature sensitivity, especially to cold.15 They are commonly misdiagnosed and can cause persistent pain and discomfort if they are not treated effectively. In one series of glomus tumors from 1995 to 2009, the average time to diagnosis was 3.3 years.9 Surgical excision is the standard of care.10Glomus tumors are rare, benign, vascular neoplasms that arise most commonly subungually in fingers. Although they occur almost exclusively in the upper extremity, with ∼75% in the hand,This study describes a patient misdiagnosed with CRPS affecting the anterior knee region for 15 years. Magnetic resonance imaging (MRI) of the affected area had not been done. She coincidentally presented with a soft-tissue sarcoma distal to her area of knee pain; this prompted an MRI, which revealed, in addition to the soft-tissue sarcoma, a small mass in the area corresponding to her “CRPS.”A 68-year-old woman was referred by a trauma surgeon for an orthopaedic oncology consultation about an expanding anterior left leg soft-tissue mass. She had returned to the trauma surgeon for a routine follow-up 1 year after cephalomedullary nailing of an ipsilateral intertrochanteric fracture. Her medical history included a distal pancreatectomy and splenectomy for pancreatic cancer 6 years previously with no known metastases. She was also diagnosed with diabetes, hypertension, hyperlipidemia, osteoporosis, and a transient ischemic attack. Her surgical history included resection of the carotid body and adrenal tumors 12 years before presentation, and workup for pheochromocytoma was negative. She had no endocrine or oncological concerns since then.A diagnosis of CRPS of the left lower extremity was made after a motor vehicle accident 15 years before her recent presentation with the leg mass. After the motor vehicle accident, she had mild knee pain, but within 1 year, the pain had worsened, was severe, was burning in nature, and was accompanied by occasional bouts of severe allodynia, which prevented her from actively extending her knee. She had been seen by five neurologists, who had all diagnosed CRPS, and an outside orthopaedic surgeon who recommended physical therapy and pain control. Her pain was uncontrolled on oral pain medications, and she was unable to complete physical therapy. She was referred to pain management where she had four rounds of intravenous infusion therapy with lidocaine, ketamine, and midazolam. She was offered a left L3 sympathetic nerve block but declined it.The orthopaedic oncologist observed a 5-cm ovoid, firm mass in her proximal anterolateral leg that was mildly tender to palpation. She had no associated skin changes and was neurovascularly intact distally. She resisted examination of the area of focal “CRPS” located proximal to the mass because it was extremely sensitive to touch but allowed palpation of the tissues around it, including the soft-tissue mass.Radiographs of the left knee showed no acute fracture, dislocation, or destructive bony changes Figure . An MRI The larger mass was rapidly growing and could be a concern for malignancy, and an ultrasound-guided core needle biopsy of that mass was expedited and conducted at bedside without general anesthesia. However, the patient's hypersensitivity in the region of her previously presumed “CRPS” prevented biopsy under local anesthesia. Therefore, an ultrasound-guided core needle biopsy was conducted of the T2-hyperintense region under general anesthesia.Histologic examination of the biopsy specimen of the rapidly growing leg mass showed a spindle cell tumor with myxoid stroma, focal sclerosis, moderate nuclear pleomorphism, multinucleated giant cells, mast cells, and up to seven mitoses/10 high-power fields, with no tumor necrosis. Immunohistochemistry showed that the tumor cells had focal membranous expression of CD99, nuclear/cytoplasmic expression of B-cell lymphoma 2, and cytoplasmic/membranous expression of smooth muscle actin and CD34, consistent with an intermediate-grade spindle cell sarcoma . The patient did well postoperatively and remained hospitalized until reconstructive surgery using a medial gastrocnemius rotational flap was conducted for the sarcoma excision 1 week later, to allow for a complete histologic analysis of surgical margins.After the biopsy results were analyzed, the patient was admitted for wide resection of the sarcoma and marginal resection of the glomus tumor simultaneously. A longitudinal incision was made over the site of the previous glomus tumor biopsy; a small round mass was identified in the subcutaneous fat and this mass was resected 16 A diagnosis of high-grade myxofibrosarcoma with focally epithelioid features was rendered.The sarcoma specimen comprised a 10.5 × 7.0 cm skin ellipse with attached subcutaneous tissue and muscle, which contained a white-tan 4.0 × 3.5 × 3.3 cm centrally located nodule bulging under the skin surface. Serial sectioning revealed that the lesion was well circumscribed and multinodular, with pink-tan and yellow-tan cut surfaces. Histologic analysis showed tumor cells with spindle, round, and epithelioid features; focally marked nuclear pleomorphism; and multinucleated tumor giant cells in a myxoid background, with dense fibrous bands separating myxoid and solid regions, up to 21 mitoses/10 high-power fields, but no necrosis Figures  and 7. IThe patient was given postoperative radiation therapy for 6 weeks, and a repeat MRI scan showed no residual sarcoma. She is doing well at more than 2 years postoperatively with no evidence of disease on surveillance MRI scans of the knee and CT scans of the chest. She remains free of leg pain.17 Although typical glomus tumors share the triad of pain, point tenderness, and cold hypersensitivity,18 extradigital glomus tumors are rarer and often do not share this constellation of symptoms.20 Extradigital glomus tumors can appear in the upper and lower extremities and multiple other sites in the body.21 Owing to their unusual nature and nonuniform symptoms, they tend to present with a longer duration of time to diagnosis. Lower extremity extradigital tumor case reports have involved patients with burning pain, difficulty with ambulation, and even unilateral limb atrophy.22 Often, extradigital glomus tumors are not diagnosed until an MRI is done. In several reported cases of lower extremity glomus tumor, MRI of the painful regions was often done several months to years after the patient began experiencing pain.19-21 Unfortunately, the patient in this case experienced a delay in diagnosis as well.Glomus tumors are uncommon and, in one study, comprised only 1.6% of 500 consecutive soft-tissue tumors.23 MRI has been shown to have a high positive predictive value for glomus tumors as small as 2 mm in diameter.24MRI is a useful tool in the diagnosis of glomus tumor. Findings of glomus tumors on MRI are often consistent with homogenous well-circumscribed lesions with hypointensity on T1-weighted images and hyperintensity on T2-weighted images along with contrast enhancement.26 As in this case, these patients noticed a significant decrease in symptoms following excision of the tumors. Glomus tumors are rare in the lower extremity, which might account for the repeated misdiagnosis of CRPS by an interdisciplinary team of physicians in this case. In addition, a glomus tumor near a soft-tissue sarcoma in the lower extremity has not been reported previously.Although glomus tumors are rare, they can cause symptoms, which were misdiagnosed as CRPS in the upper extremity in two previously reported cases.27 and, in this case, was used to provide a tissue diagnosis before initiating treatment. General anesthesia was necessitated because the patient had exquisite local hypersensitivity to the extent that even contact with clothing could cause pain in the region of her anterolateral leg.Although in this case, the orthopaedic oncologist opted to first have the painful soft-tissue mass biopsied before excision, a primary excisional biopsy could have been justified. The history was suggestive of glomus tumor, the mass was small and superficial enough to address safely through an excisional biopsy, and the core needle biopsy itself was not trivial because it required general anesthesia because of the patient's hypersensitivity. However, the senior author (F.A.K.) elected for a core needle biopsy because of the highly unusual nature of this case. Several years of misdiagnosis by multiple physicians and the coincidental fact that the patient developed a large soft-tissue sarcoma near the undiagnosed soft-tissue mass raised questions about the mass representing something other than a glomus tumor and the possibility of a relationship to the nearby malignant mass. Core needle biopsy has been shown to provide high diagnostic accuracy for soft-tissue masses28 Three-phase bone scanning has been reported to have a high negative predictive value to rule out CRPS type I,30 which presents without an identifiable nerve lesion and accounts for approximately 80% to 90% of CRPS cases.4 In a three-phase bone scan, abnormal flow patterns and vasomotor instability are seen in the first and second phases while increased periarticular uptake is seen in the third phase.30 CRPS type II, which presents with an identifiable nerve lesion, may show a decrease in nerve conduction velocity and increased sensorimotor latency on electromyography.31 Other diagnostic tools for CRPS include bone densitometry, infrared thermography, sweat testing, sympathetic blocks, bedside clinical examination methods, and targeted questionnaires.32It is important to appreciate that CRPS is a diagnosis of exclusion. On plain radiography, CRPS has been associated with periarticular osteopenia and patchy osteoporosis.33 Owing to nonuniform MRI findings, such as an absence of bone edema up to 50% of the time, MRI has been viewed as only an adjunct to help rule out alternative diagnoses.34 Overall, most of the research on MRI and CRPS is regarding brain MRIs and analyzing the neurologic involvement of this poorly understood condition.36 Nonetheless, MRI is useful in diagnosing pathologic entities, such as tumor or infection. Although a tumor may be unlikely to cause symptoms mimicking CRPS, it did in this case. In addition, advanced imaging was included in the workup only after the development of a visible palpable mass close by the painful region. Knowledge of CRPS-like symptoms as a possible presentation of glomus tumor and a thorough clinical workup, including advanced imaging, could result in a prompter diagnosis, reduce the burden of pain, and facilitate appropriate treatment.MRI has been largely excluded in the CRPS literature because it has been shown to have lower sensitivity and specificity than a three-phase bone scan for CRPS.


Gastrointestinal attacks are frequent symptoms in patients diagnosed with hereditary angioedema (HAE). Cases of self-limited bowel intussusception and unneeded exploratory laparotomy due to lack of knowledge about HAE have been reported. Furthermore, after the introduction of C1-esterase inhibitor (C1-INH) concentrate, the recommended medication for HAE attacks, treatment has become typically medical in nature. We share a rare case where operative exploration was indicated to resolve a mechanical small bowel obstruction secondary to an HAE attack.An 80-year-old woman with HAE presented with lower left abdominal pain, vomiting, and nausea. Computed tomography (CT) showed edema of the small bowel and stomach as well as possible signs of mechanical small bowel obstruction. The patient was treated with C1-INH concentrate but showed only mild signs of relief, warranting diagnostic laparoscopy. Intraoperative findings showed internal herniation and strangulation of the small bowel caused by adhesions forming a band. After surgical intervention, no bowel resection was needed.Although C1-INH concentrate remains the principal treatment for HAE, gastrointestinal attacks may potentially cause surgical emergencies. Hereditary angioedema (HAE) is a rare genetic disorder characterized by recurrent episodes of angioedema, which often affects mucosal tissue of the skin and respiratory and gastrointestinal tracts . The mosAn 80-year-old woman with a history of HAE, angina pectoris, and cerebral infarction was referred to the emergency department due to severe pain in her lower left abdomen. She reported a one-day history of abdominal pain, nausea, and vomiting. She showed no other signs of edema of her skin or airway. Her previous attacks included abdominal pain and were occasionally accompanied by skin symptoms. Although she had experienced multiple HAE gastrointestinal attacks throughout her lifetime, they were all self-limited without administration of C1-INH concentrate. Her vital signs were a blood pressure of 163/65 mmHg, a heart rate of 68 beats per minute, and a temperature of 37.1 °C. On examination, the abdomen was soft and nondistended with tenderness and rebound in the left lower quadrant. She claimed that the pain was similar to her previous episodes of gastrointestinal attacks; however, her history of angina pectoris and cerebral infarction suggested increased vascular risks, making it necessary to exclude other diagnoses, such as mesenteric ischemia, mesenteric embolism, and ischemic enteritis.5/μL and a lactate concentration of 2.80 mmol/L. Other test results were unremarkable, including C-reactive protein, liver function, and electrolytes. Subsequent laboratory tests revealed a serum C4 level of 6 mg/dL (reference range: 20–50 mg/dL) and C1-INH activity as low as 15% (reference range: 70–130%). Computed tomography (CT) showed edema of the stomach, which was most pronounced at the gastric angle  postoperatively due to possible laryngeal edema after extubation. However, she was successfully extubated; no further doses of C1-INH concentrate were necessary, and she was moved to the general ward. She was discharged with no apparent complications on hospital day 7. Subsequent laboratory tests showed elevation of C1-INH activity (45%) and serum C4 levels (16 mg/dL) compared with those observed on admission .HAE is a rare autosomal dominant disorder, affecting 1 in 50–100,000 people of any ethnicity. The mean age at onset of symptoms is approximately 8–12 years, and swellings occur as frequently as once per week to less than once per year , 5. HAE Gastrointestinal tract involvement is one of the most common features of HAE. Symptoms are present in 33–50% of patients, such as nausea, vomiting, abdominal pain, and diarrhea, which are the result of intestinal edema . SymptomAbdominal adhesions are fibrous bands that span intra-abdominal organs. Although the mechanism of how adhesions are formed is unclear, 90% of adhesions form post-surgically. Other etiologies include radiation, formation secondary to inflammation such as endometriosis, pelvic inflammatory disease, Crohn’s disease and idiopathic formation , 12. ThiHAE attacks often mimic other abdominal conditions, and unneeded procedures can be avoided by administering appropriate medication. Nevertheless, our report suggests that recurrent episodes of HAE may lead to a patient with adhesions becoming symptomatic due to internal hernia with an edematous bowel wall. Physicians need to be aware that abdominal pain from HAE attacks that does not resolve with C1-INH concentrate may have an underlying condition, including surgical emergencies.


A 48-year-old man presented with chronic headaches since 2014 in addition to dizziness, somnolence, and forgetfulness. He also reported episodes of transient focal neurological deficits, especially motor impairment, which resolved spontaneously.An initial brain computed tomography (CT) scan revealed multiple parenchymal calcified nodules, a focal lesion with peripheral enhancement, and perilesional edema on the left frontal lobe. These findings, as well as the accompanying symptoms, were consistent with neurocysticercosis in the calcified nodular and colloidal vesicular stage.2O) and presence of white blood cells . An angiographic CT scan revealed hydrocephalus and segmental narrowing of the left middle cerebral artery


Aneurysmal bone cysts (ABCs) rarely involve the cranium. We report a case arising in the ethmoid sinus with orbital and intracranial invasion. Imaging suggested an associated fibro-osseous lesion. The lesion was completely resected. Histology confirmed the imaging diagnosis of ABC on top of an ossifying fibroma. A multidisciplinary approach is essential for optimal surgical outcomes.We report a case of an 8-year-old boy with a 5 week history of painless, increasing prominence of the left eye. Clinical examination revealed non-pulsatile left proptosis. Visual acuity and ocular movements were normal. CT and MRI scans of the maxillofacial regions showed a large space-occupying lesion involving the left ethmoid air cells with left orbital and left inferior frontal intracranial extension. Multiple fluid levels with blood products were seen. Areas of the bony component of the lesion showed ground-glass density on CT. Imaging was consistent with an ABC with an underlying fibro-osseous lesion; probably fibrous dysplasia which was confirmed after surgical removal of the lesion. A multidisciplinary team of maxillofacial, neurosusugery and ENT surgeons performed the surgery.ABC arising from an osseous fibroma of the skull is rare. Total resection can be achieved with a multidisciplinary surgical approach. Post-operative histology confirmed by the imaging findings. Involvement of the skull, however, is rare.2Aneurysmal bone cysts (ABCs) were first described in 1942.We report a rare case of a giant ABC associated with a fibro-osseous lesion involving the ethmoid sinus, orbit and anterior cranial fossa. Gross total surgical resection was achieved. Imaging and histopathological features are described.An 8-year-old boy presented with a 5 week history of painless, increasing prominence of the left eye. Parents noticed the swelling after the child had sustained a minor injury while playing. No visual disturbance was reported. Clinical examination revealed non-pulsatile left proptosis measuring 22 mm by exophthalmometer compared to the normal right eye (18 mm). Visual acuity and ocular movements were normal.CT and MRI  demonstrA multidisciplinary team of Maxillofacial, ENT and Neurosurgery performed a total surgical resection of the lesion. Pre-operative nasal endoscopy confirmed origin of the lesion from the left ethmoid sinus and medial wall of the left orbit. A bicoronal flap was raised along with a vascularised pericranial flap and a bifrontal craniotomy was performed to access the lesion. An irregular thick walled fluid filled cystic lesion was seen coexisting with stippled hypervascular irregular bone that was indenting the medial and superior walls of left orbit. The lesion extended into the ethmoids and posteriorly along the orbital roof, encased in thin bone. Using the microscope, plane of cleavage was developed between the lesion wall and the overlying dura mater. To permit further mobilisation, the mass was opened and gelatinous liquid was released decompressing the cyst. Following resection of the lesion, the large anterior fossa skull base defect was repaired with a pedicled nasal septal flap from below and a vascularised pericranial flap from above in conjunction with a durasealant Xact . The patient made an excellent recovery and was discharged 4 days post-surgery.Specimens of bone tissue around the tumour, left orbital bone and left frontal bone showed features of ABC with spaces filled with RBCs. The wall of the cyst showed spindle cells with benign cytomorphology, a large number of multinucleated giant cells and cement like material in an area of fibrous stroma. No evidence of malignancy .3 However, it can affect any bone in the body. Only 3–6% of ABCs affect the skull.2 ABCs in the head and neck area are present in only 2% of cases.4 The mandible and maxilla are the most frequently involved sites with a mandible predilection of 2:1 over the maxilla4 ; with over 90% of cases reported above 30 years of age.3 The involvement of the ethmoid bone as in our case is rare.5ABCs are uncommon lesions, accounting for 2% of all primary bone tumours predominantly involving the metaphysis of long bones.4 Neurologic deficits are more likely to happen when the skull base is involved. No neurologic abnormality was found in our patient where painless proptosis was the presenting symptom.Clinically, presentation depends on location, size and compressive effects of the mass on surrounding structures.2 The most common prior lesion is giant cell tumour. Other lesions include fibrous dysplasia, nonossifying fibroma, fibromyxoma, chondroblastoma, osteoblastoma and osteosarcoma.2ABCs are classified as primary or secondary, with primary lesions appearing in isolation. Secondary lesions can develop in a number of benign and malignant bone lesions or trauma.6The pathogenesis of ABCs remains controversial. It may be related to genetic predisposition, post-traumatic or reactive vascular malformation.6 This helps distinguish ABCs from hemangiomas. Reports also documented a pathological appearance of woven bone with proliferating fibrous tissues and blood-filled chambers with bony islands.6Histopathology provides supportive data to confirm the diagnosis of an ABC and distinguishing it from other haemorrhagic bone tumours. Pathology reveals large areas of haemorrhage with fibroblastic septations lacking an endothelium.ABCs are benign locally destructive rapidly growing tumours. In our patient, the ABC is probably secondary to an ossifying fibroma. ABCs secondary to ossifying fibroma remain a relatively uncommon finding in the facial bones.3 Benign fibro-osseous lesions include fibrous dysplasia, ossifying fibroma and cement-osseous dysplasia.Few cases in the literature describe a close association of ossifying fibroma with juvenile ABCs as in our case.7 Reports state that 87% appear as radiolucent and only 2% are radiopaque with the rest having mixed opacity.7,8 Other findings also include ground-glass density, osseous expansion, narrowing of the foramina and contrast enhancement.8The imaging diagnostic workup for ABCs usually consists of a CT scan, followed by an MRI. CT usually shows a heterogeneous mass with solid and cystic regions.9 The fluid level is suggestive of a mixture of proteinaceous and blood products.7 The mass also presents with associated bony remodelling, as well as areas of non-aggressive bony resorption.8Fluid levels are present on CT in 35% of cases, with increased attenuation of the dependent level.9 However, fluid levels may also be seen in simple bone cysts, soft tissue cavernous hemangiomas and cystic hygromas.10 Other MRI findings include multiple internal septations and lobulations with varying blood degradation intensities.9MRI shows fluid levels better than CT.8However, imaging findings alone are not enough to distinguish ABCs from other pathologies. Differential diagnoses include fibrous dysplasia, haemorrhagic cyst, giant cell reparative granuloma, metastasis and plasmacytoma.8 Telangiectatic osteosarcoma is much more aggressive in nature and appears in older age groups.8 Plasmacytoma shows a diffuse homogeneous enhancement after i.v. contrast. Metastatic lesions characteristically show ring enhancement with gadolinium.8Fibrous dysplasia like ABC is also an expansile mass frequently involving the ethmoid sinuses, but it shows ground-glass appearance on CT.10 which is generally curative. This may be more difficult with lesions involving the skull base.Treatment of choice is gross total resectionABCs of the skull are rare. Involvement of the ethmoid bone in the setting of fibro-osseous lesion as in our patient has been reported in only a handful of previous case reports. Lesions can often be identified based on characteristic imaging findings of multi loculated septated haemorrhagic mass with fluid levels. Areas of ground-glass density on CT suggest the underlying fibro-osseous lesion. CT and MRI help pre-operative anatomical location and extent of intracranial and orbital involvement. Histological examination is essential to make an accurate diagnosis.Patients presenting with proptosis should be investigated by cross-sectional imaging.Imaging findings of multiloculated septated haemorrhagic mass with fluid levels are highly suggestive of ABC, but other pathologies cannot be ruled out.Areas of ground-glass density on CT suggest the underlying fibro-osseous lesion.CT and MRI provide pre-operative anatomic details of the ABC as well as invasion of surrounding structures.Histological examination is essential to make an accurate diagnosis.