Histiocytic sarcoma (HS) is a rare malignant neoplasm showing morphologic and immunohistochemical features of histiocytes. It is characterized typically by extranodal presentation and a poor clinical course, particularly in cases with disseminated disease.This report documents a case of bifocal and aggressive HS in small bowel and lung revealed by acute peritonitis in a 63-year-old man.Despite its rarity, we believe that the correct diagnosis of HS is crucial for clinical treatment and prognostic prediction.The collection of additional cases of HS are important to obtain further progress in prognosis and guide treatment decisions. •Histiocytic sarcoma is a rare malignant neoplasm showing morphologic and immunohistochemical features of histiocytes.•The diagnosis of histiocytic sarcoma can be extremely challenging because histologic overlap with diverse mimics.•Judicious application of immunohistochemical markers is crucial to confirm the diagnosis.•The collection and evaluation of additional cases are pivotal to obtain further progress in prognosis and guide treatment decisions. Abdominal examination revealed a rebound tenderness suggesting for acute peritonitis. Abdominal Computed Tomography (CT) scan with contrast injection, showed a pneumoperitoneum with a perihepatic intraperitoneal effusion. Urgent surgical exploration was made by a university assistant in general surgery and found a purulent peritonitis with a mass of 7 × 4 cm of the small intestine complicated by perforation and adhering to the omentum . A 20 cm3Histiocytic sarcoma is an extremely rare malignant neoplasm included within the tumors of the macrophage-dendritic cell lineage . Its patClinical presentations of HS are variable, ranging from localized disease to widely disseminated disease with multifocal masses ,4. AccorOn histology, the tumor consists of a diffuse non-cohesive proliferation of oval to round large cells >20 p.m.). Tumor cells have usually an abundant eosinophilic cytoplasm and pleomorphic nuclei. The extent of mitotic activity parallels closely the degree of nuclear pleomorphism. Large multinucleated cells are commonly seen. Features of haemophagocytosis can be observed in the neoplastic cells. An intense and marked inflammatory infiltrate of small lymphocytes, neutrophils or eosinophils is often seen ,2,4,6.0 p.m.. TImmunohistochemistry is pivotal in the diagnosis of HS, to confirm histiocytic differentiation with positive markers and to exclude morphologic mimics with negative markers. Most HSs express CD68, CD163, PU.1and CD45 . It is rThe differential diagnosis of HS is notably broad and includes other malignant neoplasms such as large-cell non-Hodgkin's lymphomas, metastatic carcinoma, malignant melanoma and soft-tissue sarcoma. Benign entities with histiocytic proliferation, like storage disorders and hemophagocytic syndrome can be excluded because of malignant cytologic features ,4,18. HSThe gold standard of treatment of HS is surgical resection with wide margins. Other treatment options include chemotherapy and radiotherapy in case of incomplete tumor resection . The sit4HS is a very rare neoplasm with malignant proliferation of cells showing morphological and immunohistochemical features of mature tissue histiocytes. The diagnosis of histiocytic sarcoma can be extremely challenging because its rarity and histologic overlap with diverse mimics. Thus, the recognition of morphologic features, as well as judicious application of immunohistochemical markers is crucial to confirm the diagnosis. The collection and evaluation of additional cases are pivotal to obtain further progress in prognosis and guide treatment decisions of this aggressive disease.Not commissioned, externally peer reviewed.This is not applicable for our manuscript.Exemption from ethnical approval.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.Ahlem Bellalah and Seifeddine Ben Hammouda: data analysis and writing the paper. Manel Njima and Nouha Ben Abdeljelil: bibliography, coordination and helped to draft the manuscript. Ibtissem Korbi, Asma Achour and Amira Daldoul: specimen contribution and data collection. Abdelfatteh Zakhama, Rim Hadhri and Leila Njim: revision.This is not applicable to our case report.Dr. Seifeddine Ben Hammouda.None.


Adenomyomatous hyperplasia is an extremely rare lesion encountered in the ampulla of Vater. Less than 50 cases have been described, most of them with clinical consequences of biliary obstruction, misdiagnosing it as a malignancy. The authors present a concomitant case with a renal tumor, its diagnosis, management, and clinical relevance, as well as a brief revision of the literature. Ampullar and renal tumors were found in a 74-year-old female, in the imagiologic study of a low back pain, in the emergency department. Both were considered malign after further study, and pancreatoduodenectomy with partial nephrectomy was proposed. There is no accurate diagnostic tool to differentiate the benign nature of adenomyomatous hyperplasia and extensive operations are often performed. As an incidental finding in the study of another tumor, this case raises the concern about which and how to treat both tumors, taking into account the morbidity of the respective interventions. Benign tumors constitute 6% of all extrahepatic biliary tree neoplasms and are responsible for 0.1% of all biliary tract operations -4.The histopathogenesis and natural history of these lesions in the common bile duct (CBD), including the ampulla of Vater, is not clear and malignant transformation in other organs as well as recurrence after local resection has been described ,5. DiagnUnlike the other counterparts in the gastrointestinal tract, the ampulla of Vater´s tumors is responsible for the biliary obstruction, which leads to significant clinical consequences. Jaundice and abdominal pain are the main complaints of these patients although an incidental finding in the study of another neoplasm has been described as well . The latWe report a case of a patient AH of the ampulla of Vater and a concomitant renal tumor, its diagnosis, management, and clinical relevance, as well as a brief revision of literature.A 74-year-old female, with a past history of renal lithiasis and lumbar hernia, recurred to the emergency department (ED) complaining of acute low back and abdominal pain. An abdominopelvic CT scan in the ED revealed not only the main cause of the pain, calculous hydronephrosis but also two incidentally distinct lesions: a left renal tumor with malignant features and a 15-mm nodular lesion conditioning CBD and wirsung ectasia. No hepatic changes or cholestasis were observed in the laboratory study. Medical treatment for ureterolithiasis was done with complete symptomatic resolution.Further study was done for both tumors. Laboratory tests showed normal hepatic and renal function, with no cholestasis. Serum carcinoembryonic carcinogen and carbohydrate antigen levels were not increased.A magnetic resonance cholangiopancreatography (MRCP) confirmed bicanalar dilation (CBD and wirsung) with abrupt stenosis at the ending of both channels, although no evident nodular lesion, as well the renal tumor Figure . FluorodEndoscopic ultrasound was attempted and no ampullar changes were demonstrated, but a cystic lesion was apparent at the terminus of the CBD (choledococele), so biopsies were not taken.Three months later, an MRCP was repeated which revealed at this time an ampullar mass causing bicanalar obstruction, and so, a CBD malignancy could not be ruled out Figure .A multidisciplinary team discussed the case and the patient was proposed to pylorus-preserving pancreatoduodenectomy in combination with partial nephrectomy, in the same surgical time, to which the patient consented.Macroscopic examination of the surgical specimen revealed a polypoid lesion in the ampullar region with 1.2 cm.Pathologic examination demonstrated chronic polypoid fibrous ampullitis with glandular AH. No malignant cells were identified. The partial nephrectomy specimen revealed a clear cell renal carcinoma (pT1aNxVx).The patient's condition was complicated with postoperative intra-abdominal abscess followed by surgical drainage. Full recovery and discharge were possible one month after surgery.Eight months later, the patient is doing well, with no complaints or recurrence of both lesions.AH is an extremely rare lesion encountered in the ampulla of Vater and can be misdiagnosed as an ampullar adenoma or carcinoma. To our knowledge, less than 50 cases have been described in the indexed English literature . This isIt is difficult to determine the real incidence, but a frequency of 0.13% has been reported in a series of consecutive 3,131 endoscopic retrograde cholangiograms (four cases) . Higher Histologically, AH is defined as a nodular lesion or a mucosal thickening with a proliferation of smooth muscle and epithelial components, as well as glandular hyperplasia without cellular atypia. Its histogenesis is not clear and many theories have been proposed. Incomplete heterotopic pancreas (type III according to Heinrich classification) seems to be the most widely accepted theory, although some authors defend that this lesion may have an inflammatory origin ,10. BaggThe preoperative diagnose is challenging and imaging is not accurate enough to distinguish an AH from adenoma or carcinoma. A definitive diagnose is only possible with histological examination of the completed resected mass ,12,13. PDespite AH is considered a slow-growing benign lesion, in this case, rapid growth was observed during the investigation, with the development of an ampullar mass noted in the second MRCP, three months after the first with no visible lesion. A similar case was described by Ulich et al, who suggest that some of these lesions might be truly neoplastic in nature, in accordance with some authors that consider the AH of the ampulla of Vater a premalignant lesion ,15.There is no widely accepted approach to managing these patients. Ampullectomy, local resections with endoscopic snare excisions, or simply biliary tree drainage with sphincterotomy have been offered when a preoperative diagnosis is possible, avoiding postoperative morbidity and mortality, which is not neglectable in a pancreaticoduodenectomy. Sphincterotomy was unsatisfactory in previous cases in severely symptomatic patients or showed recurrence . ExtensiThis particular case raises a dilemma: both tumors are suspicious of malignancy, being an ampullar tumor more aggressive; in spite of the good physical condition of the patient for the age, the physiological status is limited, and the decision to operate involves substantial risk. Nevertheless, radical surgery is still the chosen approach when an ampullar carcinoma cannot be ruled out, as in this case. Further studies have to be done to improve the diagnostic accuracy and provide less aggressive interventions.Adenomyomatous hyperplasia of the ampulla of Vater is an extremely rare tumor of the biliary tree. Unlike in other organs, this location has important clinical consequences, being misdiagnosed as an adenoma or carcinoma in most of the cases. There is no accurate method to distinguish these lesions from an adenoma or carcinoma, and so, extensive surgery is frequently adopted.Efforts must be done to improve the diagnostic tools. Cases with concomitant neoplasms are not negligible in the literature, with decision-making implications and increased surgical risk.


Preduodenal portal vein (PDPV) is a rare congenital anomaly commonly associated with other gastrointestinal abnormalities. We report the case of a female neonate with a PDPV, intestinal malrotation and jejunal atresia. This is the second account of this association reported in the literature. The previously reported case underwent a gastroduodenostomy to correct the presumed duodenal obstruction caused by the PDPV. In our case, the PDPV was not corrected and the child remains well and asymptomatic. We propose that in this rare association, the PDPV is not a cause of obstruction and does not need correction. Preduodenal portal vein (PDPV) is a rare congenital anomaly, first described by Knight in 1921 ,4 and isHere we report the second case of a neonate with a PDPV, intestinal malrotation and jejunal atresia where the PDPV was not corrected.On day 1 of life, shortly after an uneventful vaginal delivery at 36+6 weeks of gestation, a female neonate was referred to the surgical team. The parents had received antenatal counselling by the paediatric surgical team at 31 weeks of gestation for distended proximal bowel loops and a likely diagnosis of intestinal atresia  jejunal atresia of approximately 5mm in length Figure  and 3 weThe patient was extubated on postoperative day 2, and feeding was commenced postoperative day 6. By postoperative day 22, the infant was breastfeeding on demand and able to be discharged home. At 6-month follow-up baby is thriving and had started taking solid foods, with no abdominal concerns.After the first description in 1921 by Knight, we are aware of 323 cases of PDPV previously described in the literature . It is rA review of the literature demonstrated only one previously reported case of PDPV associated with malrotation and jejunal atresia . In the As there was no clear proximal duodenal obstruction, a corrective procedure for the PDPV was not performed in our case. Gastroduodenostomy and duodenoduodenostomy should not be performed without significant consideration in the neonate as there are potential long-term complications associated such as gastroesophageal reflux, anastomotic stricture and bile reflux gastritis ,9. Our dPDPV rarely causes duodenal obstruction and is usually diagnosed incidentally at the time of surgery for one of its common associations such as malrotation, situs inversus, duodenal stenosis, atresia or web, pancreatic or biliary abnormalities. In the absence of calibre change or evidence of obstruction at the proximal duodenum, PDPV correction is unlikely to be of benefit. We propose that in the rare case of PDPV associated with malrotation and jejunal atresia, surgical correction of the PDPV might not be necessary.


Insulinoma is a functioning pancreatic neuroendocrine tumor primarily leading due to hypoglycemia due to hypersecretion of insulin. This case illustrates the real challenges faced in the detection of an occult insulinoma, which resulted in a protracted course of the disease.A 33-year-old female presented with recurrent hypoglycemia. Endogenous hyperinsulinemia was confirmed by a prolonged fast, however serial imaging was negative. Incidental finding of an ovarian mass gave rise to the suspicion of an insulin-producing ovarian tumor. Subsequent multimodality pancreatic imaging remained negative, requiring more invasive investigations. The tumor was localized by specialized arteriography using calcium stimulation to support the diagnosis of an insulinoma. However, repeated negative imaging led to further delays in definitive management, with worsening hypoglycemia. The surgery was finally performed three years after the initial presentation with successful removal of the tumor using intra-operative ultrasound.It is important to emphasize that preoperative radiological imaging is useful to localize pancreatic lesions. However, most insulinomas could only be detected intraoperatively. The absence of suggestive radiological evidence should not deter surgeons from proceeding with definitive surgical intervention.The case highlights the importance of a multidisciplinary approach in the management of a complicated case. •Insulinoma is a difficult disease to diagnose due to its ambiguous location and insidious course.•Insulinoma and type 2 diabetes mellitus coexistence is rare.•Radiological imaging has improved in aiding the detection of pancreatic lesion but falsely negative results remain high.•Surgical intervention is imperative for explorative diagnosis as well as treatment.•A multidisciplinary approach could have been more successful in early detection and thus providing definitive treatment. Episodes of hypoglycemia started 3 months later, prompting the cessation of both these medications. However, her symptoms persisted and she experienced episodes of palpitation, sweating and dizziness at her workplace. Upon arrival at the Emergency Room, her blood glucose was confirmed to be below 3 mmol/L, and her symptoms improved following dextrose infusion. Physical examination revealed a well-built woman with a body mass index of 28.4kg/m2 and other examinations were unremarkable. She gave further history of intermittent dizziness, palpitation and sweating during exercise and while performing house chores within the last few months in the absence of any anti-diabetic medications. These symptoms occurred at any time of the day, with no clear predilection for the fasting periods. She had also been experiencing gradual weight gain over the last few months, but denied changes in behavior or morning headaches, and has not been taking traditional medications or supplements. She had no previous medical, surgical nor psychological history, and she had not been taking any prescribed medication. Interestingly, her late father who had hypertension and end-stage renal disease on dialysis was diagnosed with insulinoma 2 years previously, but unfortunately succumbed to septicemic shock secondary to pneumonia. Her 8 siblings had been well with no known medical illness. She had no allergies and never smoked. She lived with her sister and was financially independent as a shop assistant.She was admitted and further investigated for unprovoked hypoglycemia. Her baseline blood results, including her hemoglobin, white cell count, platelets, renal and liver profiles were all within normal ranges. She was commenced on a prolonged fast, and developed hypoglycemia within 9 hours of fasting, with symptoms of sweating, palpitations and drowsiness, confirmed by a capillary blood glucose of 2.8mmol/l.The results were suggestive of insulinoma, with venous blood glucose of 2.3 mmol/l, Insulin 24 uIU/L (6-27) and C-Peptide 1685 pmol/L (165-662). However, subsequent computed tomography (CT) scan with pancreatic protocol did not reveal any significant pancreatic lesions. In view of the family history of insulinoma, the possibility of Multiple Endocrine Neoplasia (MEN) 1 was entertained. However, further investigations revealed negative results . She wasShe presented six months later to a gynecologist with lower abdominal pain and irregular menses. She had learnt to overcome the intermittent symptoms of hypoglycemia in the interim by eating regular small meals. A computed tomography scan of the abdomen showed no pancreatic lesions, but revealed a left ovarian mass measuring 3.6 × 3.7 × 4cm, with features suggestive of a teratoma. Refer She underwent another battery of investigations for localization of the insulin-producing lesion, including a repeated CT pancreatic protocol, endoscopic ultrasound and magnetic resonance imaging (MRI) of the pancreas, all of which failed to reveal any significant abnormalities. Refer The results of the SACST  was high3Insulinomas are rare with an estimated incidence of 1–3 per million population per year .Unprovoked hypoglycemia is the pathognomonic feature of insulinoma, with an episodic nature, which reflects the intermittent secretion of insulin by the tumor. The main symptoms are either autonomic symptoms including palpitation, irritability and sweating, or due to the effects of hypoglycemia on the central nervous system, such as confusion, headache and behavioral changes ,3. AlthoIn endogenous hyperinsulinism, the cause for hypoglycemia is the low levels of glucose production rather than increased utilization of glucose. Hence the main point is the failure of suppression of insulin secretion during this low glucose level . This leInsulinomas are almost universally located within the pancreas, generally described as 1/3 in the head, body and tail of the pancreas ,7. Of spLocalization of the insulin-producing tumor was indeed the most challenging part in this patient's journey towards a definitive cure. She underwent an extensive series of imaging modalities with no amenable positive result. Conventional studies namely transabdominal ultrasonography, computed tomography and magnetic resonance imaging have varying detection rates, where some series are positive in only 10-40% of cases, versus a sensitivity approaching 70-85% for CT and MRI in other reports ,4. This The subsequent protracted course of insulinoma in this case further highlights the challenges in localization of cases with negative imaging. After the negative imaging results, she underwent another invasive modality, namely selective arterial calcium stimulation test (SACST) for localization of this elusive tumor. Similar to other pancreatic neuroendocrine tumors (p-NET), insulinoma is a vascular tumor. Hence, selective angiography has been shown to be positive in 60% of cases and when combined with hepatic venous sampling after intra-arterial calcium administration it regionalizes insulinoma with high sensitivity, with reported positive detection rates of 88–100% ,4. The SInterestingly, although some authors insist on preoperative localization with SACST, others have suggested that it may not be essential as most insulinomas can be detected intraoperatively ,11. In a4Although rare, insulinomas remain as one of the commonest neuroendocrine tumors of the pancreas, with episodes of debilitating hypoglycemia and reduced quality of life, requiring early detection and intervention. The sensitivity of a variety of imaging examinations are variable, with SACST having the highest success rate. Surgery is a definitive treatment but frequently delayed because of limited radiographic evidence. This case, therefore, clearly illustrates some of the challenges posed in the detection and management of occult insulinoma. It certainly underscores the importance of a multidisciplinary approach and cooperation from all members to be well informed in the most recent scientific knowledge in managing an extremely complicated and intriguing case.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.None declared.10.13039/501100004625Universiti Teknologi MARA.Partial reimbursement of publication fee from All authors provided clinical input to the case report and comments towards the manuscript.None.Prof Dr Rohana Abdul Ghani, Consultant Endocrinologist, UiTM.Not commissioned, externally peer-reviewed.None declared.


Pancreas is an uncommon site of hydatid cysts (HCs) even in endemic countries. Primary pancreatic hydatid cysts (PHCs) mainly occur through hematogenous dissemination. Their rarity and the absence of clinical manifestations in most cases explain their challenging preoperative diagnosis. In symptomatic cases, clinical findings may be similar to those of other diseases. We report a case of a 54-year-old female presented with a six-month history of abdominal pain, although her abdominal examination was normal. Radiological imaging revealed a serous cyst in the body and tail of the pancreas. Biliopancreatic endoscopic ultrasound (EUS) suggested a peritoneal hydatid cyst. Intraoperatively, it was diagnosed as a PHC. The patient underwent resection of the PHC and was then placed on albendazole. She did not have any symptoms for the last seven months. Through this case report, we can conclude that peritoneal hydatid cyst of the pancreas should be considered in the differential diagnosis of the cystic lesions of the pancreas. Moreover, surgery achieves a definitive treatment of the disease. Echinococcus granulosus. It is an endemic zoonosis in the Mediterranean area [Hydatid cyst (HC) is a disease caused by a parasite called ean area . It ofteean area . They caean area . In thisWe present a case of a 54-year-old female living in Morocco, who has a past medical history of hypothyroidism on levothyroxine sodium. She presented with a six-month history of abdominal pain in the epigastrium and left hypochondrium region, with no other digestive or extradigestive symptoms. She had no history of pancreatitis, anorexia, or weight loss. Abdominal examination and the biology report were normal. Abdominal ultrasonography (USG) was suspicious for a cystic mass of the pancreas. Abdominal magnetic resonance imaging (MRI) showed a serous cyst located in the isthmus of the pancreas measuring 48 × 36 mm Figure .The biliopancreatic EUS showed a heterogeneous extrapancreatic mass, it was oval, hyperechoic, and measured 49.5 × 38 mm. It had an echogenic double wall and contained serpiginous elements evoking hydatid membranes, suggesting a peritoneal HC Figure . HydatidPHCs are a rare cause of cystic lesions of the pancreas . They arThe most accepted mechanism of the pancreatic infestation of hydatid disease is hematogenous dissemination. Other possible mechanisms are the passage of cystic elements to the pancreas through biliary or lymphatic system .PHCs are asymptomatic in most cases; therefore, they are incidentally discovered. Their symptomatology depends on their size, location, and potential complications . They caThe complications of PHC depend on their location in the pancreas. When located in the head of the pancreas, it can cause obstructive jaundice, cholangitis, acute and chronic pancreatitis, duodenal stenosis or fistula, and pancreatic abscess. When located in the pancreatic tail, it can cause splenomegaly, portal hypertension, abscess formation, or it can rupture into the peritoneal cavity and gastrointestinal tract .Echinococcus antigens in enzyme-linked immunosorbent assay can distinguish PHC from other types of cystic lesions [Abdominal USG is the most sensitive tool for the detection of floating membranes, hydatid sand, and floating daughter cysts. The highly suggestive computed tomography (CT) scanning signs of HC are the water lily sign, water attenuation, and calcifications. EUS can provide a characterization of the cystic lesions of the pancreas, but is limited for making the definitive diagnosis . However lesions . However lesions .The differential diagnoses of PHC are cystic diseases like pseudocyst, serous cystadenoma, and intraductal papillary mucinous neoplasm . In thisThe treatment modalities depend on the location of the PHC. When it is located in the tail of the pancreas, distal pancreatectomy can be a successful treatment option. The treatment option for PHC located in the body and head of the pancreas is proper evacuation, pericystectomy, and omentoplasty. In the case of obstructive jaundice, ultrasound-guided drainage of the HC can be performed. In the case of cholangitis due to PHCs, endoscopic retrograde cholangiopancreatography and mini-sphincterotomy can be an effective treatment strategy . PostopePHCs are rare even in endemic areas. They are diagnosed incidentally in most cases. When symptomatic, the clinical manifestation depends on their location in the pancrea, they are an unusual cause of abdominal pain. Despite radiological imaging, including abdominal USG, CT scanning, EUS, and hydatid serology, the preoperative diagnosis of PHC remains difficult. This diagnosis should be considered in the differential diagnosis of cystic lesions of the pancreas, especially in endemic geographical regions. Surgery offers a complete cure of the disease, and the risk of recurrence decreases with the administration of albendazole.


A foreign body granulomatous reaction is a rare complication of the subcutaneous injection of fillers. We describe a 57-year-old female that presented with a six-month history of a non-painful, right lower eyelid mass. She had a dermal filler in the zygomatic region several months before the onset of her symptoms. Anterior orbitotomy and pathology revealed multinuclear giant cells consistent with a hyaluronic acid-based filler. This case describes the clinical presentation, histopathologic features, and treatment of an orbital granuloma secondary to dermal filler injection. Our case was uncommon because the zygomatic filler migrated across the orbital septum. Additionally, transconjunctival orbitotomy was used instead of a hyaluronidase injection due to the inferior location of the granuloma. Subcutaneous injection of fillers has been used as a nonsurgical treatment for facial rejuvenation. Fillers can be injected with a quick recovery and minimal discomfort making them a popular procedure for correcting soft tissue loss, rhytids, and folds. The aesthetic plastic surgery national databank estimates that over 1.3 million dermal fillers were injected in 2020 . One sucA 57-year-old female presented to the office for evaluation of a painless, right lower eyelid mass. The mass had been present for six months. She had a history of multiple dermal filler injections to her face over the last four years. She had a previous adverse reaction after a dermal filler full-face rejuvenation three years prior characterized by multiple periocular and perioral hard nodules that were successfully treated using oral steroids and injected hyaluronidase. Examination revealed a right lower eyelid, palpable, painless, well-circumscribed, mobile mass that appeared posterior to the orbital septum. Her visual acuity was 20/20 in the right eye with normal pupillary function, full extraocular motility, and unremarkable Hertel exophthalmometry and optic nerve function. Upon further questioning, the patient revealed that she had been injected with a Juvederm Voluma injection in the lower eyelid and lateral cheek junction in the zygomatic region to augment her cheeks approximately eight months prior to presentation. She denied any history of filler injections to her eyelid or tear troughs. She underwent a successful anterior orbitotomy through a transconjunctival incision that revealed a cystic encapsulated lesion Figure .The lesion was sent for histologic analysis, which showed acellular grey-blue material surrounded by multinucleate foreign body giant cells consistent with hyaluronic acid-based filler Figure . The patForeign body reactions occur when a material or object, such as a dermal filler originating outside the body, resists degradation by individual macrophages, which then aggregate and coalesce, leading to multinucleate giant cell formation . This prHyaluronic acid (HA) is a glycosaminoglycan polysaccharide constituted from alternating residues of the monosaccharide D-glucuronic acid and N-acetyl-D-glucosamine . HA-baseFiller migration is described as filler at a location other than the primary injection site ,6,12. FiWe conclude that the evaluation and management of foreign body granulomas secondary to dermal fillers include a proper history, recognition of the substance, and imaging and/or tissue diagnosis if necessary to rule out any other sinister underlying condition in patients presenting with new orbital masses. Our case was uncommon, as it was inside the orbit, demonstrating that migrated fillers can cross the orbital septum. Most orbital granulomas secondary to fillers can be treated with injected hyaluronidase. However, our patient underwent transconjunctival orbitotomy for tissue diagnosis due to the location in the inferior orbit. Patients should be given information on the type of filler injected and counseled to seek evaluation to perform an appropriate treatment if a complication arises.


Rectus sheath hematoma (RSH) is an infrequent condition that occurs when epigastric arteries bleed into the rectus sheath and sometimes acts like an acute abdomen. In view of the fact that it is a rare case without specific clinic signs, misdiagnosis and use of invasive manipulations for patients are possible. A 27-year-old woman applied to our clinic with abdominal pain, weakness, and nausea. Acute abdominal pain was in the periumbilical region and physical examination revealed tenderness, rebound phenomenon, and local mass in the right periumbilical region. Rectus sheath hematoma was identified by magnetic resonance imaging. The cause in this case was abdominal trauma due to domestic violence. After the legal procedures, symptomatic treatment was prescribed to the patient. Interestingly in this case, we did not get to the diagnosis through history, we reached the history after diagnosis. Besides the treatment of the patient, maybe our team prevented further violence against a woman. Rectus sheath hematoma (RSH) is an infrequent condition that occurs when epigastric arteries bleed into the rectus sheath and sometimes acts like an acute abdomen. The first case in the literature was reported by McKim . Rectus A 27-year-old woman applied to our clinic with abdominal pain, weakness, and nausea. Acute abdominal pain was in the periumbilical region and physical examination revealed tenderness, rebound phenomenon, and local mass in the right periumbilical region with a 130mm x 44mm size. No sign of contusion, scar, or other skin lesions was seen in the abdominal wall. Her vital signs were heart rate 98 per minute, blood pressure 95/70 mmHg, oxygen saturation 97%, respiratory rate 12 per minute, and temperature 36.9 Celsius. Hemoglobin levels and leukocytes were 9.2 gr/dl and 13.400 IU, respectively. Initially, the patient underwent abdominal ultrasonography, which confirmed a large complex, avascular, heteroechoic mass along the lower rectus sheath. Given the described ultrasound features and in the context of this clinical presentation together with pain and palpable mass, it was suspected as a hematoma. However, because of its size, it could not be concluded with certainty whether the patient had an additional intra-abdominal pathology as a free fluid. Thus, on suspicion, an abdominopelvic computer tomography without intravenous or oral contrast was ordered. Computer tomography showed the right rectus abdominis muscle was markedly enlarged compared to the normal left due to a large heterodense (27 to 61 Hounsfield unit) mass that extended to the subperitoneal and inferiorly prevesical spaces, causing bladder compression , peritoneal irritation, a positive Carnett sign, vomiting, nausea, constipation, scrotal swelling, and tachycardia ,8. OccasDiagnosis of rectus sheath hematoma is done by imaging methods. Ultrasonography is a simple and cost-effective method to diagnose rectus sheath hematoma; the main demonstrator sign is heterogeneity. However, in some cases like obese or pregnant patients, ultrasonography is suboptimal for diagnosing and other imaging methods such as computer tomography and magnetic resonance imaging can be used to evaluate the patient. In computer tomography imaging with intravenous contrast, hematoma is evaluated by its confinement to the abdominal wall, lack of enhancement, and high attenuation. Berna et al.'s research illustrates that the role of computer tomography imaging in rectus sheath hematoma diagnosis is crucial, as sensitivity and specificity are 100% [The causes of rectus sheath hematoma are variable, starting from anticoagulant usage to trauma. Sheth et al. reported in their study that chronic kidney disease, cancer, liver diseases, and abdominal injections are among the causes .Abdominal traumas are also one of the main causes of rectus sheath hematoma. In our patient, the reason was abdominal trauma related to domestic violence. Since about 36% of the women who face domestic violence tend to not disclose because of ignominy, discomfiture, and disgrace, doctors ought to be more cautious when they confront a woman with abdominal trauma . VariousRectus sheath hematoma is a rare outcome of abdominal traumas. Thus, it is possible to misdiagnose the disease and patients can face redundant procedures. The underlying reason should be investigated thoroughly. The underlying cause in our case was domestic violence. In the publications there is an absence of studies on the percentage of abdominal traumas caused by violence against women.Diagnostic evaluation and treatment approach in abdominal trauma-caused rectus sheath hematoma are crucial during the course of the disease.


Although laparoscopic incisional hernia repair, especially laparoscopic intraperitoneal onlay mesh, is a widely used technique, it can cause serious complications, including mesh erosion, adhesive bowel obstruction, and chronic pain. The enhanced-view totally extraperitoneal (eTEP) technique has been reported to prevent such complications by placing the mesh in the retrorectus space. Here, we report the case of a patient with post-robot-assisted laparoscopic radical prostatectomy (RARP) incisional hernia repaired using the eTEP technique.A 67-year-old man, who underwent RARP for prostate cancer 4 years ago developed an incisional hernia. Abdominal computed tomography showed the presence of an epigastric incisional hernia measuring 4 cm long and 3.7 cm wide. We performed an eTEP repair. We closed the hernia defect using a 0 barbed suture and placed a self-gripping mesh measuring 20 cm long and 15 cm wide in the developed retrorectus space with no fixation. There were no postoperative complications, and the patient was discharged on postoperative day 2.eTEP repair is considered an extremely effective surgical treatment option for incisional hernias because of its few resulting postoperative mesh-and-tacker-related complications. Recently, robot-assisted procedures have become widely used in various surgeries; in particular, robot-assisted laparoscopic radical prostatectomy (RARP) for prostate cancer has been one of the most accepted surgeries. RARP has clinical, functional, and oncological benefits in patients with prostate cancer , 2. HoweIncisional hernia is one of the most common postoperative complications experienced by patients, which not only causes pain and cosmetic problems, but also causes incarceration of the small bowel . EmergenThe enhanced-view totally extraperitoneal (eTEP) technique, an endoscopically performed Rives–Stoppa technique, was first described by Miserez for ventral hernia repair . The eTETo the best of our knowledge, no other study has reported a case of eTEP repair in a patient with incisional hernia after RARP. Here, we report a case of a patient with post-RARP incisional hernia repaired using the eTEP technique.A 67-year-old man presented to our department with a swelling in the upper abdomen for half a year. His surgical history included RARP for prostate cancer in 2017. He had been medicated with a few anti-anxiety drugs for depressive anxiety disorder. Physical examination revealed a tennis ball-sized irreducible subcutaneous mass on the previous RARP incision scar, where the specimen was removed from the upper abdominal midline. Blood tests, including blood cell counts, liver enzymes, renal function, and HbA1c, were within the respective reference ranges. Abdominal computed tomography showed the presence of an epigastric incisional hernia 4 cm long and 3.7 cm wide (Fig. The patient was placed in the supine position with his arms tucked at the sides under general anesthesia. Figure Above the level of the hernia defect, the medial portion of the left posterior rectus sheath was incised from the cephalad in the caudal direction and crossing to the preperitoneal space under the white line to the right posterior rectus sheath. The medial portion of the right posterior rectus sheath was incised and a 12-mm trocar was inserted at the port 4 position after sufficient dissection in the right retrorectus space. Retrorectus dissection was performed in the caudal direction, incising bilateral posterior rectus sheaths to the hernia defect Fig. .Fig. 3InWhen the hernia sac was sharply dissected, we found very mild adhesions between the hernia sac and omental tissue Fig. . CarefulThere were no postoperative complications and the patient was discharged on postoperative day 2. Postoperative pain was well controlled with prophylactic oral analgesics for 5 days after surgery. No hernia recurrences occurred after 9 months of follow-up Fig. .Fig. 6PoRecent large sample size cohort studies reported that the incisional hernia incidence rate was higher with minimally invasive radical prostatectomy, including RARP, than with traditional open radical prostatectomy , 4. The The laparoscopic approach to incisional hernia repair with the IPOM technique was first described by LeBlanc in 1993 . It has The eTEP technique, which is an endoscopically performed Rives–Stoppa technique, was first described by Miserez for ventral hernia repair . The eTEWhen dissecting the hernia sac for eTEP repair, careful procedures are required to avoid injury to intra-abdominal organs, such as the small bowel. We consider that dissecting the hernia sac with a laparoscopic scissors in small steps is useful to avoid damaging the intra-abdominal organs. It is controversial whether a prosthesis can use for the incisional hernia repair if a bowel resection is performed as a result of a bowel injury , 21. AlteTEP repair is a very useful minimally invasive procedure for patients with incisional hernias after RARP. Although long-term follow-up is necessary to establish the safety and efficacy of eTEP repair, we consider this procedure to be the best option for patients with incisional hernia because of its few postoperative complications.


Benign fibro-osseous lesions of the craniofacial region are a diverse group of entities with overlapping histologic characteristics. One of these fibro-osseous lesions is ossifying fibroma and it is seen rarely in the head and neck region. Only a few cases of temporal bone involvement were reported in the literature. Patients with ossifying fibroma located in the temporal bone may have the following complaints: conductive hearing loss, swelling, localized pain, headache, and ear discharge. The lesion should be removed surgically and obtaining negative surgical margins is crucial to prevent any recurrence. A 29-year-old female patient who applied to our clinic with the complaint of a mass behind the left ear was treated, and the pathological diagnosis was an ossifying fibroma. In this study, a case of ossifying fibroma is presented. Ossifying fibroma (OF) was first described by Mezel in 1872 . The folA 29-year-old female patient was admitted to our clinic with a mass posterior to the left ear that had been enlarging slowly for about three years. The patient stated that her main complaint was cosmetic deformity. The patient had no history of head trauma or surgery. On physical examination, a rigid mass, approximately 30 × 25 mm in size, was palpated on the left mastoid process, with regular borders, fixed, without hyperemia and fever. Other than that, the patient's physical examination, audiological tests, complete blood count, and routine biochemical parameters were normal. The patient was evaluated with temporal bone computed tomography (CT). CT showed a well-circumscribed nodular lesion posterior of the left auricle, within subcutaneous fatty tissue, with a size of approximately 23 × 12 mm in axial sections, which did not destroy the adjacent bone structures, and contained extensive millimetric-sized amorphous calcifications  were excised. The remaining part of the pedicle was removed with the help of a drill, and the suture lines were visible Figure .The patient was discharged on the first postoperative day, and there were no complications. Histopathological examination of the lesion reported OF. Microscopic examination revealed thick collagen bundles in the mass and disorganized bone trabeculae that did not tend to merge in the hypocellular stroma. No osteoclasts were seen in the bone tissue, and there was no significant osteoblastic activity Figure . No recuOF was first described by Mezel in 1872 and is a rare fibro-osseous lesion of the head and neck region with a benign character and locally aggressive course ,5. AlthoOFs in the head and neck region are most commonly seen in the mandible, followed by the maxilla ,2. It maFor the clinical differential diagnosis of OFs located in the temporal bone, fibrous dysplasia, osteoma, exostosis, osteoblastoma, giant cell tumor, myelomas, eosinophilic granuloma, and metastatic lesions should be considered . Among tIn addition to conservative approaches such as enucleation and curettage, radical approaches can also be chosen for treatment. In a retrospective study in which eight case series were analyzed, the recurrence rate after surgical treatment was found to be 15.3%. It was stated that the recurrence rate in radical approaches is lower than in conservative approaches. In addition, it was revealed that 75% of recurrences are seen within the first year after surgery . In our OF is a very rare mass in the head and neck region. Temporal bone involvement especially is an exceptional case of OF. It should not be forgotten in the differential diagnosis of temporal bone masses and treatment should be surgical. The most confusing disease histopathologically in the differential diagnosis is fibrous dysplasia. OFs should be excised with broad surgical margins to prevent a recurrence.


Hepatic angiosarcoma (HAS) is a malignant mesenchymal neoplasm composed of highly atypical, pleomorphic endothelial cells, which diffusely grow along liver sinusoids and other preformed vascular channels, especially at the periphery of the tumor, replacing normal endothelial cells. More solid areas, resembling sarcomas, can be found . Far lesBecause HAS is an extremely rare tumor, both radiological and histological diagnosis could be really challenging and their integration with clinical data and history has a key role in reaching the correct diagnosis.Herein the case of a primary multifocal HAS is described, with initially misleading unspecific radiological findings. Suggesting the biopsy a malignant vascular lesion of the liver, an autopsy was performed, which confirmed the diagnosis of HAS.A 73-year-old man with a history of hypertension and congestive cardiac failure was admitted at our hospital for progressive jaundice of unknown etiology. During prior hospitalizations multiple hepatic nodular lesions of uncertain nature had been reported at various imaging exams; furthermore, two ultrasound-guided liver biopsies had already been formerly performed, leading to a diagnosis of nodular regenerative hyperplasia of uncertain interpretation.After admission to our hospital, laboratory tests revealed increased direct bilirubin levels (9.59 mg/dl), while liver enzymes were within the recognized normal ranges. Moreover, search for autoantibodies and serologic tests for hepatotropic viruses were negative and no anomaly was noted for blood iron parameters.Abdominal contrast-enhanced US revealed multiple hyperechoic, anaechoic, and isoechoic hepatic nodules Fig. . AbdominAfter multidisciplinary team discussion, a laparoscopic liver biopsy was performed. The histological findings were suspicious for a vascular lesion but, before a definitive diagnosis could be made, 3 days after the procedure the patient’s conditions rapidly worsened, evolving in acute respiratory distress syndrome (ARDS) and severe liver failure. Ultimately, the patient developed hepato-renal syndrome and died.In order to find the cause of the sudden deterioration of the liver function, an autopsy was requested. Grossly, the liver had an irregular surface and revealed blood filled cavities and gray-brownish spongy areas surrounded by recognizable residual normal hepatic parenchyma at cut examination Fig. . No otheTwenty-four tissue samples were taken from the liver and then fixed in 10% formalin solution and embedded in paraffin blocks. Four-micrometer-thick sections were obtained and stained with hematoxylin and eosin for histopathological examination. Immunohistochemical staining for CD 31 , CD34 , and ERG  was performed.Microscopically, multiple cavernous spaces covered by highly atypical endothelial cells were observed Fig. , along wA chronic occupational vinyl chloride exposure was retrospectively discovered. Based on these clinical data and the morphologic features, the diagnosis of multifocal HAS was established.Hepatic angiosarcoma (HAS) is a very aggressive malignant mesenchymal tumor arising from liver endothelial cells composed of atypical, plump, pleomorphic cells with hyperchromatic nuclei and high mitotic rate. Although solid areas of sarcomatous-looking neoplastic cells, admixed with giant multinucleated cells, can be found, HAS’s neoplastic cells tend to grow mainly within preexisting vascular channels, especially along hepatic sinusoids, without gathering in a distinct and compact mass . Tumor cells show an endothelial phenotype, staining commonly positive for CD31, CD34, ERG, and FVIII; a small percentage of cases is reported to express pan-cytokeratins. HAS is frequently a multifocal disease, with the liver’s parenchyma not uncommonly almost completely wiped out by the tumor.HAS is a rare entity, accounting for less than the 2% of all the primary malignant hepatic neoplasms, with about 200 cases worldwide diagnosed every year. Although a broad part of all HASs do not recognize any specific etiology, some chemical carcinogens have been claimed to be responsible for occupational cases, above all vinyl chloride monomer (VCM), Thorotrast (thorium dioxide), and arsenic . VCM is HAS’s diagnosis may sometimes be extremely challenging, potentially representing both a radiological and histological pitfall. Namely, radiologists can face difficulties in identifying HAS for several reasons. Firstly, due to its rarity, physicians are not so likely to have to deal with these tumors in their daily routine. Secondly, HASs’ imaging findings are generally not specific, with extremely variable radiological patterns appreciated, mainly regarding the number of neoplastic nodules and the prevalence of necrotic or hemorrhagic areas within them , 6, 8–11Therefore, finally, for all the reported reasons a thorough evaluation of the patient’s clinical data and history and its integration with imaging and pathological findings it is mandatory for correctly assessing the diagnosis of HAS.Hepatic angiosarcoma is the most common primary malignant mesenchymal neoplasm of the liver, which has been linked to chronic occupational exposure (10–40 years) to different chemical carcinogens, such as vinyl chloride monomer, Thorotrast, and arsenic. Due to its extremely aggressive clinical behavior, which generally causes patients to die within a very short time, it is therefore mandatory to promptly reach the proper diagnosis. However, for several reasons HAS represents a potential radiological and histological pitfall. Firstly, physicians are not so likely to deal with these tumors due to their rarity, both for lowered permissible exposure limit and for more accurate worker surveillance programs. Secondly, unspecific imaging findings have been repeatedly reported, suggesting various different hepatic diseases, either of a benign, malignant or, as in our case, unknown nature. Finally, as neoplastic cells commonly use to grow singularly along sinusoidal spaces, without forming a distinct mass, histological evaluation of bioptic material could be incredibly tough work. Therefore, integration of pathological and radiological findings with patients’ clinical history is the key for avoiding wrong interpretations and reaching the correct diagnosis.The case reported perfectly depicts all the obstacles that physicians might encounter in diagnosing such a rare and fatal condition. In fact, the initial radiological picture was that of an unspecific disease, probably vascular in origin: a proper definitive diagnosis of hepatic angiosarcoma could be only assessed following autopsy evaluation and its integration with the retrospective discovery of a chronic occupational vinyl chloride exposure.


Sclerosing rhabdomyosarcoma presentations are widely variable and non-specific initial features. We report a rare case of non-orbital sclerosing rhabdomyosarcoma presented with optic neuropathy. A 15-year-old female patient initially presented with upper gum swelling and pain for 3 months. It was associated with loosening of teeth. Subsequently, the patient developed recurrent epistaxis follow by left facial swelling and blurring of vision. Examination showed marked left facial swelling with mild proptosis. Visual acuity in the left eye was no perception of light with the presence of relative afferent pupillary defect. Fundoscopy showed left optic atrophy. Neuroimaging showed large aggressive soft tissue mass on the left infratemporal, masticator, and parapharyngeal space with a local extension to the sphenoid sinus. There was also an intracranial extension to the left temporal lobe with the base of skull bone destruction. Transnasal endoscopic biopsy revealed sclerosing rhabdomyosarcoma. Management was with chemotherapy. Sclerosing rhabdomyosarcoma may present with optic nerve involvement that may carry a guarded prognosis to the eyes. Rhabdomyosarcoma (RMS) is an aggressive soft tissue sarcoma, a malignant neoplasm that is composed of cells with histopathologic features of striated muscle in various stages of embryogenesis. Sclerosing rhabdomyosarcoma (SRMS) is a rare subtype of RMS that was first described by Mentzel and Katenkamp in 2000 that is characterized by hyaline sclerosis and pseudovascular pattern . CurrentA 15-year-old female patient presented with left-sided upper gum pain for three months. She was seen by a dentist and treated with antibiotics as an infected molar. She developed progressive loosening of teeth that worsened despite multiple dental treatments. In the following two months, she developed painful left facial swelling, nasal block, anosmia, and recurrent epistaxis. Her condition worsened and she started to have blurring of vision in the left eye. She went to the emergency department for treatment and was referred to the otorhinolaryngology team and ophthalmology team.Examination showed facial asymmetry with prominent left facial swelling extended from the periorbital region to the mandibular region Figure . The sweContrast-Enhanced Computed Tomography (CECT) brain and orbit revealed an ill-defined heterogeneous enhancing mass on the left side of the face. It occupied the left infratemporal, masticator and parapharyngeal space measuring 7.2 x 5.8 x 7.2 cm. There was a local and intracranial extension to the left temporal lobe. Bony destruction was observed at the base of the skull, lateral wall of the sphenoid and left maxillary sinus Figure . There wThe patient was treated with four cycles of IVADo chemotherapy , and six cycles of IVA . Magnetic Resonance Imaging (MRI) of the brain and orbit was done after six months of chemotherapy showed that the tumour size had reduced, indicating a good response to treatment. There was no enhancement of the optic nerve or any extension to posterior orbit Figure . HoweverSclerosing RMS (SRMS) presents at a wide age range (1-79 years) and has a bimodal age distribution, with a high number of patients identified in the first and second decades or around the fifth decade . Males aThe most common symptoms of head and neck RMS tumors are facial swellings, dysphagia, nasal obstruction, epistaxis, and difficulty opening the mouth and speaking . PersistOptic nerve infiltration may occur as a result of hematogenous spread, perineural invasion, or leptomeningeal metastatic dissemination as has been reported in earlier cases of leukemia, lymphoma, sarcoma, breast carcinoma, lung carcinoma, prostate cancer, pinealoblastoma, and rectal carcinoma -10.Contrast-Enhanced Computed Tomography (CECT) and Magnetic Resonance Imaging (MRI) can provide crucial information in determining the size and location of the primary RMS tumor and its extension into adjacent tissues . RMS appThe International Rhabdomyosarcoma Study Group (IRSG) has been leading protocols to help define the shifting therapy paradigms for RMS since 1972. RMS is usually treated with multimodality therapy combines chemotherapy with surgery and/or radiotherapy . In our The overall survival of 16 pediatric cases of SRMS with a short follow-up of <2 years was found to be 87.5% and disease-free survival was 62.5%. But the incidence of pediatric SRMS treatment failure was significantly high at 43.75% [Early optic neuropathy may be a sign of head and neck SRMS, and non-specific symptoms may cause a delay in diagnosis. Fortunately, treatment outcome with chemotherapy is promising whereas radiotherapy is more challenging due to its size. Hence the need for a multidisciplinary approach in managing SRMS is of paramount importance.


Although Vernix caseosa peritonitis (VCP) is a rare complication of cesarean section (CS). It possesses a high rate of morbidity and its exact underlying pathology is not fully understood yet. However, it is assumed that the leakage of amniotic fluid into the maternal abdominal cavity triggers the inflammatory process. We discussed herein a 25-year-old patient who developed acute peritonitis three days after cesarean section. CT abdomen showed multiple fluid collections with no obvious other pathologies. At laparotomy, we found cheesy exudates covering the peritoneal surface of the abdominal viscera and multiple turbid fluid collections, but no bowel, uterine, or any another organ injury could be identified. Abdominal washing was done and, as the appendix was queried, an appendectomy was added. Histopathology study of the omental biopsy revealed mixed inflammatory infiltrate, fibrin, fetal hair, and squames. These findings suggest the diagnosis of VCP, but the removed appendix was normal. The postoperative course was long and complicated. A few cases of VCP were reported in the world; the majority of these cases are from the USA and a few are from the UK. According to the information we have, our case is the first reported case of VCP in Saudi Arabia. Typically, VCP is manifested as acute abdomen hours to weeks after CS or vaginal delivery. As VCP is usually mistaken for the other causes of acute peritonitis, and the diagnosis is only established by the histopathology examination of peritoneal or omental biopsies, the case is commonly managed by urgent laparotomy or laparoscopy with the removal of suspected organs, which are later confirmed to be normal on histopathology study. The typical intraoperative findings are adhesive exudate, white or cheese-like membranes covering the intraabdominal viscera, and fluid collections, but no visceral injuries can be identified. Therefore, it is crucial to include the VCP in the differential diagnosis of the acute abdomen after CS or vaginal delivery to avoid unnecessary laparotomy and removal of normal organs. Vernix caseosa (VC) is a material that covers the surface body of a human neonate. The VC is specific to human babies; it consists of a cheesy sebaceous substance, soft hair, and epithelial squames of the baby skin . VC has VCP is a rare but critical complication after CS or vaginal delivery. VCP is hypothesized to be due to the dribble of the fetal amniotic fluid or meconium into the maternal abdominal cavity . VCP is To our best of knowledge, this is the first reported and published case of VCP in Saudi Arabia. This study set out to increase awareness among obstetrics, pathologists, and surgeons about this rare complication after CS and to raise the point that not all cases of acute peritonitis after CS need laparotomy.A 25-year-old Saudi woman, gravida 1, para 0, has no past medical or surgical history of note. She has been on regular prenatal follow up since the 27 th week of gestation. In the 37th gestational week, the patient was admitted through the obstetric clinic for labor induction due to fetal growth restriction. A straightforward emergency CS was done because of fetal distress and failure of labor progression on medical induction. The first two post-CS days were unremarkable, and she was planned discharge possibly on the next day. On postop day three, the patient developed abdominal and chest pain with tachycardia 110/minute and shortness of breath. The clinical suspicion of pulmonary embolism was excluded by chest CT that only showed bilateral pleural and pericardial effusion  for close monitoring and respiratory support. The postoperative course was lengthy and complicated. Although the patient was on broad antibiotics , high-flow oxygen, and chest physiotherapy, she persistently had tachycardia, tachypnea, dyspnea, fever, leukocytosis, abdominal pain, distention, tenderness, and increase oxygen demand. The workup of the third postoperative day revealed negative bacterial culture . CT abdomen showed a new fluid collection anterior to the right lobe of the liver and CT chest revealed bilateral lung infiltrates and consolidation, which suggest an acute respiratory distress syndrome (ARDS). There was no DVT on venous doppler of both lower extremities, and the histopathology report diagnosed the case as VCP. Intravenous steroids (methylprednisolone 30 mg every 12 hours for seven days) were added to the current medications. After commencing the steroids, our patient had gradual and significant improvement over the subsequent days, and she was discharged home on the 10th postoperative day. Surprisingly, after two days, she presented to the emergency room complaining of blood-stained secretions from the laparotomy wound. On local wound exploration, we observed full wound dehiscence. Hence, the patient was transferred to the operating room; formal abdominal closure under general anesthesia was done. The patient was discharged home after five days of an eventless hospital course. We have been following up with our patient regularly, for six months and, fortunately, she is free of significant complications, apart from intermittent crampy abdominal pain.The pathologic analysis of the removed appendix showed an appendicular wall lined with non-atypic, cylindrical, and not ulcerated epithelium; these findings mean normal epithelium. The mucosa and underlying muscularis propria are within the normal histopathological features. However, in the serosa, there is a mild peri-appendicular inflammatory infiltrate centered around a focal area of fetal squamous and keratin deposits. The histopathologic diagnosis is peri-appendicular peritonitis with a focal area of fetal squamous deposits. It is highly suggestive of vernix caseosa peritonitis Figure . The hisVCP was first described in 1976. It is a rare peripartum condition and is thought to be secondary to the dribbling of fetal amniotic fluid into the maternal abdominal cavity. Typically, the amniotic fluid reaches the mother’s abdomen during CS or from uterine perforation -4; alterClinically, VCP manifests hours to weeks after CS or vaginal delivery. Frequently, patients have signs and symptoms simulating acute abdomen: diffuse abdominal pain, distention; tenderness and rebound tenderness; fever 38.8°C); tachycardia (100-110/min) ,8-9. Sho.8°C; tacRadiologically, X-ray, ultrasound, CT, and MRI of the abdomen are normal or inconclusive . The comNumerous intraoperative findings were reported: a variant amount of bloody ascites, turbid fluids, yellowish plaques on the liver surface, extensive adhesions, fibrinous omental plaques, granulomatous inflammation, mass lesions resembling bowel perforation, and widespread white adhesive exudate covering the surface of the abdominal organs and parietal peritoneum. In most cases, no uterus or bowel perforation nor obvious intra-abdominal emergencies can be detected -4,6,9-10In the literature, the treatment of VCP falls into three categories: first, ultrasound-guided aspiration of the intraabdominal fluids and conservative treatment. Second, laparoscopy, peritoneal or omental biopsy, abdominal lavage, and intravenous antibiotics with steroids . Third, The histopathology examination of biopsies from the omentum, ascitic fluids, and peritoneum have numerous findings. These findings are basically influenced by the time between the amniotic fluid spillage into the peritoneal cavity and the microscopic examination ,3. In thBoth our study and literature share a number of key points: acute peritonitis following CS, indeterminate findings of diagnostic imaging, no clinical improvement on antibiotic therapy, and lack of awareness regarding the diagnosis among health providers. All these points made us suspect an intra-abdominal emergency and subsequent laparotomy with removal of the questioned appendicitis. On the other hand, our case is unique because, to our best of knowledge, this is the first reported and published case of VCP in Saudi Arabia, as the majority of reported cases are from the USA and a few are from the UK . This caTherefore, the clinical diagnosis of VCP should be kept in mind in patients who present with an acute abdomen following CS, particularly when the findings on CT abdomen are inconclusive and there is no improvement on antibiotics alone. Nevertheless, CT abdomen is the most helpful imaging modality. Peritoneal, omental, and ascitic fluid biopsies are diagnostic -3. Once These data may help us create the following cautious strategy when dealing with post-CS acute abdomen: First, it is critical to maintain the diagnosis of VCP. Second, if the laboratory and imaging results are inconclusive, an image-guided aspiration of the intraabdominal fluid for histopathological evaluation could be undertaken. If the diagnosis of VCP is confirmed by a histopathological study, conservative treatment (intravenous antibiotics and steroids) will suffice. Third, if the intraabdominal fluid is not accessible, diagnostic laparoscopy is recommended. Intraoperatively, the presence of characteristic findings without any injury suggests the diagnosis of VCP; thus, the therapeutic options include fluid, omental, or peritoneal biopsy with peritoneal lavage. This approach might be useful in reducing the frequency of unnecessary laparotomies and normal organ removal.Despite its rarity, VCP is a serious complication of CS. Frequently, it is misdiagnosed and undervalued since it mimics the other types of acute peritonitis. Therefore, it is crucial to increase awareness of this rare diagnosis among healthcare providers and include it in the differential diagnosis of post-CS acute peritonitis especially when the imaging studies are inconclusive. Having this awareness may help doctors avoid unnecessary laparotomies and the removal of normal abdominal organs. Further research is needed to determine the exact underlying relationship between peritonitis and the spillage of amniotic fluid into the mother's abdomen during CS and why it does not always occur.


An adrenocortical tumor is a rare tumor in pediatrics, which can be functional or nonfunctional. Functional tumors present with virilization, feminization, or hypercortisolism. Feminizing adrenal tumors, though rare in pediatrics, need to be excluded in any child presenting with features of feminization. We report a case of a 4-year-old Sudanese girl who presented with gradually progressive bilateral breast enlargement and accelerated growth since the age of 6 months. The family had sought medical advice several times in numerous health facilities without much gain. Investigations showed pubertal luteinizing hormone levels, high estradiol E2, and dehydroepiandrosterone sulfate, with normal early morning cortisol level. Abdominal ultrasound revealed a right-sided hypoechoic suprarenal mass. Abdominal computed tomography scan showed a right adrenal mass. The diagnosis of feminizing adrenal neoplasm was confirmed and right adrenalectomy was done. Histopathological examination of the resected adrenal gland showed adrenocortical adenoma. The patient was started on gonadotrophin-releasing hormone agonist for secondary central precocious puberty.Adrenocortical tumors, though rare in pediatrics, are a documented cause of precocious puberty; biochemical and imaging screening protocol should be adopted for patients with precocious puberty, even in a resource-limited setting, for early detection and treatment. An adrenocortical tumor (ACT) is a rare disease in pediatrics. The annual worldwide incidence is 0.3–0.38 per million children, with a peak incidence in those younger than 5 years .ACT can be functional or nonfunctional. Functional tumors can present with peripheral precocious puberty with predominant virilization or feminization, hypercortisolism alone or in combination with peripheral precocious puberty, or rarely with hypoaldosteronism .Owing to the rarity of these tumors and their variable presentation, they can be missed by general physicians and pediatricians in primary care facilities, and this is what had happened to our patient. She had suffered from progressive peripheral isosexual precocious puberty caused by long-lasting excess estrogen production from a right adrenal tumor, which had been complicated by central precocious puberty as a result of the delayed diagnosis and management.We report this case to show the importance of adopting biochemical and imaging screening for early detection of ACT, and the need to train doctors in primary health care facilities in our limited-resource setting about the causes of precocious puberty and the importance of early diagnosis and management.A 53-month-old Sudanese female presented with progressive bilateral breast enlargement and accelerated growth since the age of 9 months. Her family had sought medical advice several times in different primary health care facilities and were reassured. She had no vaginal bleeding and no pubic or axillary hair.1, P1, and B3. She had reddish mucoid vagina. She had no clitoromegaly, acne, hirsutism, or palpable abdominal mass  and height 108 cm (90th centile) using the Centers for Disease Control and Prevention growth chart. Mid-parental height was 175 cm and predicted adult height was 167 cm using the JM Tanner formula. No previous documented follow-up growth data were available. Her Tanner staging was ALeft wrist X-ray revealed a bone age of 8 years.The hormonal evaluation using fluorometric enzyme immunoassay showed basal luteinizing hormone of 3.1 mIU/L, which increased to 8.8 mIU/L 45 minutes post-gonadotrophin-releasing hormone stimulation. Elevated levels of estradiol E2 29,000 pg/ml (5–15 pg/ml), and dehydroepiandrosterone sulfate 90 ng/mL (2.3 ng/mL), with normal early morning cortisol level 16 ng/mL (7–28 ng/mL). Due to financial difficulties, we did not measured the follicular-stimulating hormone level.3, uterine volume of 3 cm3, and endometrial thickness of 1.2 cm. The abdominal CT scan showed a 25 × 22 mm well-defined rounded focal lesion with a smooth outline, at the level of the right adrenal gland with homogeneous attenuation, HU-7 on a noncontrast scan, and no evidence of local tissue invasion . Findings suggestive of benign adrenal cortical adenoma.The postoperative evaluation revealed normal cortisol and dehydroepiandrosterone sulfate. Estradiol E2 40 pg/mL was consistent with secondary central precocious puberty, so the patient was started on monthly gonadotrophin-releasing hormone agonist, with a regular follow-up plan for the possibility of recurrence of adrenal adenoma.Six months after the operation, the patient was well and compliant to monthly gonadotrophin agonist injections. She showed partial regression of her secondary sexual characters and a growth velocity of 5 cm/year. The abdominal CT scan was normal with no evidence of recurrence.ACT is a rare disease in pediatrics. The annual worldwide incidence is 0.3–0.38 per million children, with a peak incidence in those younger than 5 years .ACT can be functional or nonfunctional. Functional tumors can present with peripheral precocious puberty with predominant virilization or feminization, hypercortisolism alone or in combination with peripheral precocious puberty, or rarely it can present with hyperaldosteronism .Feminizing adrenocortical tumors (FAT) are rare in pediatrics. They present mainly with peripheral precocious puberty; isosexual in girls and heterosexual in boys. Excessive estrogen secretion, with or without other adrenal hormones, is the main biochemical finding. Imaging and biochemical assessment of other adrenal hormones can discriminate adrenal from ovarian secretion of estrogen . Due to P53 gene are the most common inherited genetic abnormalities associated with increased ACTs frequency in familial cancer syndromes. Overexpression of steroidogenic factor-1 and insulin-like growth factor-2 plays a pivotal role in ACTs pathogenesis [The etiology of ACTs is currently not very clear. Mutations in the Li Fraumeni, Beckwith–Wiedeman syndromes, isolated hemihypertrophy syndromes, congenital anomalies of the kidney, and congenital adrenal hyperplasia are commonly associated with ACTs . Our patThe laboratory characteristics of ACTs include disturbance of the normal circadian rhythm of cortisol secretion, and discordantly elevated serum levels of sexual corticosteroids including dehydroepiandrosterone sulfate, estrogen, and testosterone. Elevated testosterone is the predominant hormonal abnormality in virilizing adrenal tumors, while raised estrogen is the main biochemical abnormality in feminizing tumors . Our patImaging using ultrasonography is cheap and easy, but it has low sensitivity and specificity in the characterization of adrenocortical neoplasms. It can play a pivotal role in neonates and young children as the adrenal gland has a bigger size in this age group . ComputeTo define the benign or malignant nature of the adrenal tumor is difficult, even in pathology. Three histopathological scoring systems can be used to predict the malignant nature of the adrenal tumors. Weiss score and modified Weiss score, which are used to differentiate adrenocortical adenoma (ACA) from adrenocortical carcinoma (ACC) in adults have low accuracy in the pediatric age group, so are replaced by the Wieneke index .Therefore, to define malignant from the benign nature of adrenal tumor clinical behavior, laboratory, imaging, and pathological findings need to be considered.ACAs can be successfully treated by total excision. Either laparoscopically if the tumor size is less than 10 cm, or by open surgery for larger tumors. Early detection and total excision by open surgical approach are the main steps in managing ACC as partial excision and advanced stage are associated with poor outcome. Mitotane can be used as adjuvant therapy in ACC . AromataThe prognosis for benign adrenocortical tumors is good, except for the risk of recurrence, while malignant adrenocortical tumors have a poor prognosis with a 5-year survival rate of 49–55% .Estradiol E2 assessment using fluorometric immunoassay, which is available in our setting, can detect high estrogen secretion, the main biochemical finding in FAT. The peak incidence of adrenocortical tumors is in those younger than 5 years, so ultrasonography can be used as a screening method in our limited-resource setting for these young patients with peripheral precocious puberty. They have relatively larger adrenal glands that can be visualized using ultrasonography. So recommending estradiol level assessment and ultrasonography as part of standard assessment tools of patients with precocious puberty can aid early diagnosis and management of FATs.Adrenocortical tumors, though rare in pediatrics, are documented cause of precocious puberty, so biochemical and imaging screening protocol should be adopted even in a resource-limited setting for early detection and treatment. Assessment of estradiol level combined with abdominal ultrasound can aid in the early detection of feminizing adrenocortical tumors.Finally, the family had found an answer to why their kid was developing breasts since the age of 9 months, why she was growing faster compared with her peers, and they were happy with their management, which helped to stop early pubertal progression.


Brunner's gland hamartoma (BGH) is a rare benign small bowel tumor, mostly encountered in the duodenum. Massive upper gastrointestinal (UGI) hemorrhage is an unusual presentation rarely reported in English literature. Symptomatic patients mostly present with features of gastric outlet obstruction, occult bleeding, or intussusception. Herein, we report a case of BGH presenting with overt UGI bleed and features of gastric outlet obstruction. Esophagogastroduodenoscopy revealed a smooth polypoidal swelling in the posterior wall of the duodenal bulb. An endoscopic ultrasound (EUS) guided fine-needle-aspiration was performed, which was inconclusive. Contrast-enhanced computed tomography showed the absence of any extraluminal component of the lesion. Endoscopic polypectomy was attempted but failed due to the broad base of the lesion, and hence the patient was managed by open surgical excision. Histological examination of the resected specimen confirmed the diagnosis to be BGH. The patient had an uneventful recovery and was doing well at the 15-month follow-up. BGH should be considered as a differential diagnosis of a polypoidal lesion of the duodenum. Any lesion larger than 2 cm or symptomatic should be removed either by endoscopic or surgical intervention. Brunner’s gland hamartoma is a rare tumor with an incidence of <0.01% accounting for approximately 5-10% of benign duodenal tumors [ A 44-year-old female presented with a history of hematemesis and melena for seven days. She also complained of multiple episodes of non bilious vomiting for the last eight months associated with mild abdominal discomfort and loss of appetite. Pallor was present on physical examination. Her hemoglobin was 7 gm/dL and was treated with two units of blood transfusion. Post-transfusion hemoglobin was 11 gm/dL. Esophagogastroduodenoscopy revealed a smooth sessile polypoidal swelling with a large base in the first part of the duodenum measuring >2 cm in the posterior wall of the duodenal bulb with no ulcer or bleeding Figure . EndoscoBrunner’s glands are located in the submucosa of the proximal duodenum, which secretes alkaline mucin and protects duodenal mucosa from gastric chyme ,3. BrunnThese are generally solitary, sessile, or pedunculated polypoidal lesions with equal sex distribution, diagnosed in the fifth decade of life, found in the posterior wall of the duodenal bulb ,5. MostlDiagnosis is made by radiological and endoscopic findings. Endoscopic biopsy findings are often non-contributory as they cannot reach deep submucosal tumor tissue ,7. ContrTreatment regarding asymptomatic, incidentally found BGH is controversial. Larger lesions (>2 cm) are shown to complicate with acute bleeding leading to shock ,8. HenceBrunner’s gland hamartoma is an important differential to be considered in polypoidal lesions of the duodenum. Removal of lesions, either by endoscopic or surgical methods, is required in all symptomatic cases and asymptomatic cases where the size of the lesion is >2 cm. The endoscopic snare cautery technique, although more cost-effective and less invasive, is often not possible if the polyp is large enough or has a large base. In these cases, surgical excision is the treatment of choice.


Accessory liver lobe (ALL) is a rare liver malformation. An ALL develops due to malformation of the endodermal caudal foregut and segmentation of the hepatic bud in the third week of gestation. Most ALLs are asymptomatic and are detected incidentally during abdominal surgery. The incidence of ALL is < 1% in patients who undergo abdominal surgery. However, some ALLs twist and cause acute abdomen. We experienced a pediatric case of ALL torsion in a patient who underwent elective laparoscopic surgery.The 5-year-old girl had a 3-month history of epigastralgia and vomiting, which occurred every 2 weeks. Abdominal ultrasonography with color Doppler imaging revealed an 11.8 × 13.6 mm nonvascular lesion with mixed echogenicity near the round ligament of the liver. Enhanced computed tomography confirmed a 14 × 16 × 20 mm low-attenuation mass surrounded by a hyperdense line and disproportionate fat stranding on the right side of the round ligament of the liver. There was no ascites or hemorrhage. These findings suggested an abscess of the round ligament of the liver. Her symptoms improved with the administration of oral antibiotics; thus, we planned to perform elective exploratory laparoscopy and subsequent resection. Two trocars (5 mm) were inserted through a multichannel port device at the umbilicus and one trocar (3 mm) was inserted at the right lateral abdomen. Upon observation of the abdominal cavity, the omentum was observed adhering to the round ligament of the liver. Macroscopic observation revealed no apparent mass lesions. We performed adhesiolysis of the omentum from the round ligament of the liver using a vessel sealing system. We performed resection at the site at which adhesion had formed between the round ligament of the liver with the surrounding tissue using a vessel sealing system and the resected specimen was extracted through the umbilical wound. The postoperative course was uneventful. A pathological examination revealed necrotic liver tissue. The resected tissue was founded to be an ALL with ischemic change.The recurrent abdominal pain was induced by torsion of the ALL. Pediatric surgeons should consider ALL torsion as a differential diagnosis for epigastralgia of unknown etiology.The online version contains supplementary material available at 10.1186/s40792-021-01231-6. Accessory liver lobe (ALL) is a rare liver malformation. An ALL develops as a result of the malformation of the endodermal caudal foregut and segmentation of the hepatic bud in the third week of gestation . Most ALA 5-year-old girl with history of mackerel allergy, atopic dermatitis and chronic constipation, had a 3-month history of epigastralgia and vomiting, which occurred repeatedly, every 2 weeks. A pediatrician performed physical and laboratory examinations when she had no symptoms; however, no abnormal findings were recognized. Four months later, she was transferred to our institution for a detailed workup because of recurrent epigastralgia.On abdominal examination, a small palpable mass was identified in the upper abdomen. A blood analysis revealed the following: white blood cell count, 10,100 /μl; hemoglobin, 13.2 g/dl; and C-reactive protein, 0.66 mg/dl. The patient’s liver enzyme levels were within the normal ranges. On abdominal ultrasonography with color Doppler imaging, an 11.8 × 13.6 mm nonvascular lesion with mixed echogenicity was detected near the round ligament of the liver . In addition, a 3-mm trocar (operator’s left hand) was inserted at the right lateral abdomen under a laparoscopic view . Observation of the abdominal cavity revealed adhesion between the omentum and the round ligament of the liver  was applied. Two 5-mm trocars  were inserted through multichannel port device  to attach to the wound retractor and pneumoperitoneum was established  large ALL > 30 g) attached to the liver by a stalk; (2) small ALL (≤ 30 g) attached to the liver; (3) ectopic accessory liver without any connection to the liver; and (4) microscopic ectopic liver 0 g attac. Types 1An ALL may prevent fusion of the anterior abdominal wall and is frequently associated with anterior abdominal wall defects . CorbittThe exact cause of ALL torsion is often unknown, but some studies reported that it might be caused by strenuous exercise or trauma . After tUltrasonography, non-contrasted or enhanced CT and magnetic resonance imaging (MRI) are used for the diagnosis of ALL. However, most cases were not diagnosed preoperatively , 12. In The surgical indications for asymptomatic ALL are controversial. Generally, cases of asymptomatic ALL are observed. However, torsion of a large ALL with strangulation of the vascular supply to the liver may potentially lead to severe hepatic ischemia. Ladurner et al. reported a case of torsion of a large ALL in which liver transplantation was required . A largeLaparoscopic resection of ALL torsion has been reported in pediatric cases , 11. TheWe successfully treated a pediatric patient with ALL torsion. A definitive diagnosis of small ALL is difficult to make before surgery. However, laparoscopic elective resection is acceptable for a pediatric patient with a small ALL with no symptoms. Pediatric surgeons include ALL torsion in the differential diagnosis epigastralgia of unknown etiology.Additional file 1. Video.


An epigastric hernia is a rare type of abdominal hernia, described in the literature mostly as small, containing only preperitoneal fat. A large true epigastric hernia with herniation of the abdominal viscera is even rarer. Only a few case reports have given an account of strangulation in such an epigastric hernia. This case report describes a middle-aged, morbidly obese man with a big epigastric hernia presenting with incarceration and acute abdominal pain. Emergency surgical exploration revealed a 7 cm midline defect in the rectus sheath and a 30 cm segment of the jejunum and a 6 cm segment of the transverse colon were gangrenous. The gangrenous bowel segments were resected, and an end-to-end jejuno-jejunal and colo-colic anastomosis were done. The patient had an uneventful postoperative recovery. A hernia is the protrusion of a viscus or part of a viscus through a defect in its containing cavity wall. The common types of external abdominal hernias are inguinal (75%), umbilical 15%), femoral (8.5%), and incisional (3.5-11%). Epigastric hernias account for around 1.6-3.6% of all abdominal hernias and 0.5-5% of all operated abdominal hernias [5%, femor2) presented with epigastric swelling for one year which was initially reducible but became irreducible for one day. It was associated with sudden onset of upper abdomen pain with abdominal distension and non-bilious vomiting for one day.A 42-year-old gentleman with severe obesity  but was maintaining blood pressure. On examination, a 10x15 cm tender irreducible swelling was present in the epigastrium. The skin over the swelling was normal. The rest of the abdomen was soft, but bowel sounds were absent. Ultrasonography (USG) of the abdomen revealed a supra-umbilical midline defect in the anterior abdominal wall. There was herniation of bowel loops and omentum, showing decreased vascularity and absent peristalsis. He was taken up for emergency laparotomy. Intra-operatively, a 7 cm midline defect was present in the rectus sheath. A hernial sac containing a 30 cm gangrenous segment of the jejunum  and a gangrenous segment of the transverse colon for a length of 6 cm was noted (Figure The gangrenous bowel segment was resected, and an end-to-end jejuno-jejunal and colo-colic anastomosis were done. He had an uneventful postoperative recovery and was discharged on postoperative day 10.Any protrusion of tissue through a defect in the linea alba between the xiphoid process and the umbilicus is designated an epigastric hernia . Most ofThe oldest theory for epigastric hernia is the perforating vessel theory by Moschowitz. The preperitoneal fat herniates through the defect for the perforator vessels in the rectus, forming an epigastric hernia . This miEpigastric hernias are generally small, containing only preperitoneal fat. Therefore, patients present with periodical sharp localized pain in the epigastric region, often associated with dyspepsia and nausea without any relation to food intake . ClinicaEpigastric hernias, similar to other hernias, need surgery because of their symptomatic nature. Early surgical intervention of an obstructed epigastric hernia is crucial as delay can result in strangulation and the need for bowel resection with prolonged recovery and increased complication rate. If preperitoneal fat or intraperitoneal hernial contents are healthy, they can be reduced and defects in the rectus sheath can be repaired. The repair has to be augmented with mesh placement . HoweverA true epigastric hernia with a peritoneal sac and bowel contents is a rare occurrence. Therefore, a high index of suspicion for strangulation should be kept in cases of a big epigastric hernia with acute abdominal pain and recent-onset irreducibility. Early diagnosis and surgical management are the keys to avoiding bowel resection with prolonged recovery and increased complication rate.


Osteoid osteoma is the most common benign bone forming tumor characterized by a nidus surrounded by reactive sclerotic bone typically associated with nocturnal pain and most common in males less than 30 years. Diagnosis may be difficult in cases of atypical presentation, intraarticular localizations or very small size. Computed tomography guided radiofrequency ablation is one of the promising treatment methods being used with an advantage of minimal invasion, faster recovery and shorter hospitalization. We present a case of an 8-year-old boy with osteoid osteoma of neck of femur managed successfully with radiofrequency ablation. To our knowledge, this is the first case reported on radiofrequency ablation in Nepal. Late presentations can lead to muscle wasting because of disuse atrophy of affected limb in children. CT scan is one of the useful investigations used to diagnose this condition. Antiinflammatory medications have been traditionally used for pain management. Although surgery is the definitive treatment, difficulty in lesion localization and the need for extensive dissection pose a problem. Radiofrequency ablation (RFA) has been found to be a safe, fast, and reliable method of treating osteoid osteomas.7Osteoid osteoma is the most common benign, slow growing bone forming tumor.An eight-year-old male child presented with a history of pain in the left hip for past 8 months. The pain was insidious in onset, progressive and aggravated at night affecting sleep and caused difficulty in walking, squatting and playing. Within two months of onset, he developed limping and wasting of left lower limb which led to antalgic gait. There was no history of trauma, fever, pain or swelling of any other joints, chest pain, weight loss or loss of appetite, and recent urinary/ gastrointestinal or respiratory tract infections. There was no family history suggestive of hemoglobinopathies. He was under muscle strengthening physiotherapy for 6 months but his symptoms were not improving.He came to us for further consultation with persistent pain and antalgic gait. On clinical examination, the child was cooperative and active, with normal height and weight for age. There was mild tenderness over the anterior aspect of left hip joint with normal range of motion. Numeric Rating Scale (NRS) assessment of pain was 5 at rest and 7 on walking. Neurological and musculoskeletal examination revealed intact cranial nerves with intact bilateral lower limb sensation. On motor examination, muscle wasting of left lower limb was seen with mid-thigh girth difference of 2 cm and decreased power (4/5) with normal tone and reflexes. On examining the upper limb no abnormalities were found.Investigations showed slightly raised ESR of 12 mm/1st hour. However, the complete blood count, renal function and liver function tests, serum calcium, phosphate, vitamin D3, vitamin B12 levels were within normal range. Review of X-ray of pelvis and bilateral thighs were unremarkable .As suspicion was made on left hip synovitis, MRI scan of hip and thigh region was done which revealed well defined 9x8x5 mm eccentric subcortical intramedullary lesion with hypointense signal (T2w) with a hyperintense (T2w) peripheral rim in anterior half of neck of femur with perilesional marrow edema .Hence, a provisional diagnosis of osteoid osteoma was made and CT scan of the pelvis was further done which showed a 7x6 mm well margined lytic lesion in the anteromedial aspect of femoral neck suggestive of osteoid osteoma .7 The parents were given detailed explanations about the procedure and the surgical and medical alternatives available. Informed written consent was obtained.This led to the diagnosis of osteoid osteoma left neck of femur. Among available treatment modalities, we chose CT-guided radiofrequency ablation (RFA) of the tumor which has shown promising results circumventing the need of surgery and its resulting complications, especially, as the tumor was near the growing end of femur.The procedure was performed with joint effort from the Departments of Orthopedics, Anesthesiology and Radiology .The CT scan suite was equipped with anesthesia machine and all essential drugs and resuscitation equipment required for the administration of general anesthesia. After a thorough pre-anesthetic evaluation and adequate fasting period, general anesthesia was induced on the patient with oxygen and Propofol and Laryngeal Mask Airway was inserted for airway management. Anesthesia was maintained with oxygen and inhalation anesthesia with adequate dose of Fentanyl as analgesic. The patient was positioned supine and skin preparation and draping were done as per standard protocol. Lidocaine 0.5% was infiltrated under the skin site marked for the tumor and 14-gauge bone biopsy needle was introduced into the lesion under CT guidance. The lesion was localized with the help of 16-slice multidetector-row CT scanner  with multiplanar evaluation to confirm accurate needle position within the nidus of osteoid osteoma. After confirmation of needle in the nidus, an 11-gauge, 12-cm long conventional Radiofrequency (RF) probe was introduced and the electrode was connected to the RF generator (HALYARD). The tip temperature of probe was set to 95°C for 4 minutes duration. Due to high impedance, the machine could not operate initially. Local anesthetic 0.5% Lidocaine 2 ml was injected through the biopsy needle cannula and after restarting the machine, two cycles of ablation were successfully performed. After the procedure, the cannula was removed and a small pressure dressing was applied at the percutaneous puncture site. Total duration of procedure was about 1 hour. Recovery of the child from anesthesia was uneventful. The next day his pain score (NRS) was assessed to be ‘zero’ both at rest and while walking. Physiotherapy was started the same day when the pain score was 0 to regain his muscle mass and for improvement of gait.He was discharged from ward on the next day of the procedure with advice for follow up and prescribed simple home exercises for strengthening his thigh muscles. On follow up examination after two months, there was no complaint of pain and he gained his left thigh muscle mass with mid-thigh girth difference of 1 cm and gait was completely normal. In 12 months follow up, he was playful, without any complaint of pain and his left thigh girth was normal. His CT findings were unremarkable and no obvious residual lesion seen 12 months post RFA .1 Despite its relatively small size, pain is excruciating and persistent which is usually worst at night. Typically, the pain is relieved by salicylates. It generally has a self-limiting course with pain subsiding within 5 to 6 years.8 If the tumor is near the growing end of a bone in a child, increased blood flow may cause deformities in the form of either overgrowth or undergrowth of the limb.9Osteoid osteoma is the most common benign osteoblastic tumor of bone.10 such as in the case described, because of its small size. It may require a thin cut CT scan which is the investigation of choice.11 MRI is also useful in its diagnosis which shows half-moon sign due to related bone marrow edema.12 Biopsy prior to treatment is not mandatory due to a remarkable amount of false negative findings in clinically and morphologically unambiguous cases of osteoid osteoma.13 With delayed diagnosis, features other than pain such as limping or muscle wasting and weakness can appear, which was prominent in this particular case.Diagnosis of osteoid osteoma can be done with the help of X-ray which shows radiolucent nidus surrounded by a zone of sclerosis. If there is extensive sclerosis or nidus <4 mm, nidus may not be seen in plain radiographs9 Difficulty in lesion localization during surgery is a major problem.15 Moreover, successful surgery necessitates complete removal of the tumor and thus extensive resection which causes tissue damage and may lead to structural weakening.15 Surgery also carries a high risk of complications and requires a long recovery period.14 Surgery for tumors in weightbearing areas also entails a long period of limited weight bearing.9In the past, treatment modalities included conservative treatment with NSAIDs, waiting for spontaneous resolution of the tumor or surgical methods including en-bloc resection, curettage of tumor or arthroscopic excision for intra-articular lesions. Surgery was the only treatment available for osteoid osteomas until recently.16 and CT-guided Radiofrequency ablation of tumor (RFA). RF ablation for treating osteoid osteoma was first described in 1989, with initial results published in 1992.17 RFA is the gold standard treatment for osteoid osteoma in all locations as it is an effective, safe method with lowest recovery time and rate of complications.16 Percutaneous RFA is a widely used interventional technique. It allows the precise delivery of heat under image guidance to the targeted tissue. It can be done in a day care basis and the symptoms are relieved immediately after the procedure. The resolution of pain is the primary parameter used to define a successful treatment. Radiographically, partial or complete infilling of the nidus with sclerotic bone is expected over 2-27 months, although little or no change in lesion appearance is also possible.2 So, in a nutshell, osteoid osteoma can sometimes be difficult to diagnose initially when it presents with atypical clinical features, complications or proximity of lesion to a joint. But, once diagnosed, it can be easily treated with CT-guided radiofrequency ablation (RFA) with very good clinical outcomes and an added advantage of early recovery and lesser hospital stay.Other methods which are less invasive include percutaneous bone resection and drilling (PBRD)3 RFA is preferred over other treatment methods because it is not associated with significant complications, does not necessitate hospitalization, and requires only a short convalescence period.18 Difficulty in localizing the lesion, conventional method of wide dissection leading to prolonged recovery, incomplete removal and recurrence of tumor have been major issues in the conventional treatment of osteoid osteoma, which makes surgery a less preferred option at present.RFA is usually performed under general or spinal anesthesia. We performed RFA using general anesthesia. The procedure described above is similar to the procedure described by other authors.RFA has proved to be a safe, quick, and minimally invasive method of osteoid osteoma treatment in many countries. We have been able to perform this procedure for the first-time in Nepal with collaborative effort from multidisciplinary faculties with a successful outcome for the patient. RFA should be considered the current method of choice for the treatment of osteoid osteomas.