Ingested toothpicks are a relatively rare event, but they may cause serious gut injuries and can be listed among rare causes of perforation, peritonitis, sepsis or death. Unless the foreign bodies were intentionally swallowed, many patients who ingested them fail to remember the event and they do not refer it during the medical history collection; this makes diagnosis problematic. In this work, a case of perforation of the sigmoid colon is described, caused by a toothpick ingestion. The patient had to be surgically treated because of a complication: the formation of an entero-iliac fistula with subsequent development of a pseudoaneurysm of the right external iliac artery. Vascular perforation due to toothpick ingestion has rarely been reported. In similar cases, it could be difficult to establishing the correct diagnosis because of the low sensitivity and accuracy rates of diagnostic investigations. The ingestion of foreign bodies should be kept in mind as an important differential diagnosis in patients with acute abdomen or chronic abdominal pain of unknown origin. •Ingested toothpicks are a relatively rare event.•These can be listed among rare causes of perforation, peritonitis, sepsis or death.•It could be difficult to establishing the correct diagnosis because of the low sensitivity of diagnostic investigations.•Vascular perforation due to toothpick ingestion has rarely been reported.•The ingestion of foreign bodies should be kept in mind as an important differential diagnosis. Most fo2A 77-year-old man presented to the emergency department with two-days history of lower abdominal pain followed by rectal bleeding. His personal history revealed a stage 1 chronic kidney disease (CKD) and the presence of a heart arrhythmias treated with an implantable device. No other relevant information was reported. Laboratory testing revealed white blood count of 9,81 cells/mm3, a haemoglobin count of 10,6 g/dL and C-reactive protein value of 1,45 mg/dl; creatinine level was 1,62 mg/dl. Others laboratory tests did not report specific findings. The patient's clinical symptoms were lower abdominal tenderness and digital rectal examination (DRE) positive for blood. He underwent abdominal x-ray and US, which just highlighted distention of small intestine and transverse colon. Then, the computed tomography scan (CT) confirmed intestinal distention and also reported the presence of sigmoid diverticular formations with wall thickening of distal sigmoid loops. An area of inhomogeneous thickening with fluid density was visible between the anti-mesenteric site of sigma, external iliac artery and iliopsoas muscle, suggesting a diagnosis of diverticulitis/peri-diverticulitis. With this suspected diagnosis, patient was recovered, and a fluid and antibiotic therapy was set up. On the 4th day after recovery the patient presented an important gastrointestinal bleeding . He was transfused and underwent an urgent CT Angio-scan, that confirmed the presence of fluid-like density alterations in correspondence to the right external iliac artery and in close proximity of anti-mesenteric site of sigma, as already reported in the previous CT exam. Moreover, it was reported the presence of roundish formation of about 1 cm with intense arterial enhancement referable in first hypothesis to a pseudoaneurysm of the right external iliac artery of unknown origin A. DecisiA recto-sigmoidoscopy was performed, and it was diagnostic for the presence of ingested toothpick which caused the entero-iliac fistula . The tooFive days after angioplasty, a control CT scan was performed, which documented a right sided hydroureteronephrosis A. After Despite of medical therapy and procedures, the patient still had fever (>38 °C) . An urgent exploratory laparotomy was performed (A.G. and L.P.), and it was found a right iliac artery inflammation due to infected vascular endoprosthesis, with millimetric serous defect on the adjacent part of sigma. A femoro-femoral bypass and a direct closure of the sigmoid wall lesion were performed. The patient conditions gradually improved, and he was discharged on postoperative day X, with indications for post-recovery vascular surgery consultation and urological controls. Oral antibiotic therapy was suggested for ten days more. The patient was weekly followed on an outpatient basis, and his clinical conditions remained good. His last instrumental (TC scan) follow-up at 3 months did not reveal any particular problems, allowing to program the removal of the ureteral stent.3Toothpick ingestion is a relatively rare event that can cause serious gastrointestinal injuries like perforations, bleedings, peritonitis, sepsis and even death. Unless the foreign bodies were intentionally swallowed, many patients who ingested them fail to remember the event and they do not refer it during the medical history collection; this makes diagnosis problematic .In the case reported, the perforation site was the sigmoid colon. It was difficult to establish the correct diagnosis of intestinal perforation caused by toothpick ingestion because of the low sensitivity and accuracy rate of diagnostic investigations. In particular, the accuracy of CT scan to detect an ingested toothpick it is only 42,6% ; in factThe patients did not have any symptoms suggesting a peritonitis, but he had to be surgically treated because of another complication: the formation of an entero-iliac fistula with subsequent development of a pseudoaneurysm of the right external iliac artery. Otherwise, for patients who do not have complications or symptoms of peritonitis, the endoscopic removal of toothpick has proven to be successful .4The ingestion of foreign bodies should be kept in mind as an important differential diagnosis in patients with acute abdomen or chronic abdominal pain of unknown origin. We want to share our experience about this because, although rare, in these cases a rapid and accurate diagnosis is necessary to avoid severe outcomes.Ethical approval was not required.No funding.Sayali Valiyeva, Lucia Romano: Writing.Francesco Maffione: Data collection.Marco Leopardi, Aldo Victor Giordano, Loreto Lombardi: Images and contribution to the text.Mario Schietroma, Francesco Carlei, Antonio Giuliani: Study design and review.1. Name of the registry:2. Unique Identifying number or registration ID:3. Hyperlink to your specific registration (must be publicly accessible and will be checked):Dott. Antonio Giuliani.Not commissioned, externally peer reviewed.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.No conflict of interest.


Primitive aortic sarcomas are rare tumors characterized by resistance to medical treatment and a poor prognosis with high metastatic rates and local recurrences. Surgery remains the mainstay treatment and is based on challenging and technically demanding resections with high rate of major intraoperative and postoperative complications. We report the case of a patient with primitive intimal sarcoma of the aorta, who underwent a descending aortic resection and reconstruction with a prosthetic tube. Intimal sarcomas are extremely rare tumors involving the great vessels, primarily the pulmonary artery and the aorta, with an unfavorable prognosis.We report the case of a surgically treated primary sarcoma of the aorta.A 48-year-old male with a history of chronic cardiac disease was referred to our division for 3 months of persistent back pain.A computer tomography (CT) scan and a magnetic resonance imaging MRI, revealeTo obtain a histological diagnosis of the tumor, a CT-guided biopsy was attempted without success. The diagnosis was later achieved with a left thoracoscopic surgical biopsy and the final histopathology revealed a primitive sarcoma of the aortic intima.The patient underwent neoadjuvant chemotherapy treatment with one cycle of epirubicin and taxolo. A new MRI  revealeConsidering the patient characteristics, the clinical investigations, and the poor response to chemotherapy, a multidisciplinary discussion suggested a salvage surgical resection.A left thoracoabdominal approach was chosen. Under full heparinization, the descending thoracic aorta was isolated, clamped, resected, and reconstructed with a prosthetic tube of Dacron 18 mm;, positiVideo 1Aorta reconstruction with a prosthetic tube of Dacron.Histopathological analysis confirmed the diagnosis of intimal sarcoma with greater than 50% necrosis, pleomorphism, and a mitotic index exceeding to 10 × 10 power fields. The immunohistochemical staining was positive for ETS-related gene protein and ubiquitin carboxyterminal hydrolase and focally positive for CD 31, factor VIII, and S100. The intimal layers of the resected aorta were involved with margins microscopically free.At the end of surgery, the patient showed flaccid paraplegia with loss of anal sphincter control and a tactile hypoesthesia. These signs were associated with a spinal cord injury at the L1/L2 level. The patient was discharged on the 10th postoperative day. However, he was readmitted due to dyspnea after 2 days. CT-scan revealed a right iliac venous thrombosis extending to the femoral vein, requiring an urgent inferior vena cava filter placement.After 2 months of follow-up another MRI showed a local recurrence and liver metastases, for which the patient started an immunotherapy treatment with pazopanib 800 mg/day. The local recurrence and the hepatic metastases were reduced after 2 months of treatment and metabolically silent on 18-F–FDG PET imaging.After 1 year, the patient is still alive with persistent paraplegia for which he has started an intensive neurological rehabilitation program.Primary sarcomas of the aorta are extremely rareThe prognosis of aortic sarcomas is very poor, related to the tumor histological grade, possibility of resection and anatomic location. The average survival rate is 15.6 months after diagnosis.Although several measures have been developed to reduce the incidence of spinal ischemia and neurological complications, this type of surgery is still associated with a high rate of postoperative complications, with up to 16% paraplegia.Adjuvant chemotherapy with doxorubicin is indicated in case of tumor emboli, metastases, or unresectable tumors, with response rates of 10 to 25%. Other proposed medical treatments include ifosfamide, gemcitabine, or docetaxel. The use of the immunotherapy shows promising success and worthy of further evaluation.In conclusion, the optimal treatment for aortic angiosarcoma is still under investigation and may be considered a multimodal treatment. Surgery mainly represents a salvage option and is particularly indicated in young patients to achieve a radical resection even if the postoperative complication rates remain high.


Angiolipomas are rare primary benign tumors that can arise in the epidural canal and cause stenosis. Of the few cases of spinal angiolipomas described, most lesions have been located in the thoracic spine, and presentation of angiolipoma in the lumbar spine is very rare. The surgical management of a 39-year-old morbidly obese woman with angiolipoma that caused stenosis with neurogenic claudication and urinary changes is described. The lesion spanned L1-L2 and surgical management consisted of T12-L2 laminectomy and en-bloc resection of the lesion. During the latest follow-up, four years after the surgery, the patient’s neurological symptoms showed improvement and there was no recurrence. Extradural spine tumors usually arise from metastatic spread or hematologic  malignancies . PrimaryThe patient was informed that data and images concerning the case would be submitted for publication and provided consent.A 39-year-old woman presented to our spine center with a two-year history of back pain and bilateral leg pain. Her pain began spontaneously. She reported pain in her upper back (left greater than right) and bilateral lower extremities (right greater than left). The pain radiated anteriorly on her left leg and posteriorly on her right. The pain improved with squatting and leaning forward, but worsened with prolonged standing or walking. She had urinary urgency and occasional urinary incontinence. She did not have bowel incontinence, no recent weight loss, and no pain that wakes her up from sleep at night. On physical examination, the patient was afebrile, alert, and oriented. The patient had a Body mass index (BMI) of 49.8. Examination of bilateral lower extremities demonstrated normal strength except 4/5 motor strength in the right tibialis anterior. Sensation was diminished on the right leg compared to the left in the L2-S1 distribution. Radiography of the thoracolumbar spine and magnetic resonance imaging (MRI) of the lumbar spine were performed. An extradural lesion, dorsal to the thecal sac, spanning L1 - L2 and causing stenosis was noted  were in the thoracic spine, followed by 12 (11%) in the lumbar spine, two (1.85%) in the cervical spine, and one in the sacrum (0.9%). Most of the studies do not mention the patients’ BMI, however, one case report described a patient with a BMI of 34.3 who developed thoracic angiolipoma . In a laTreatment options for angiolipoma include surgery with complete resection or subtotal removal. Both options are favorable for patients, but complete resection appears to be curative. Wider resection and radiotherapy have been described if complete removal is not easily achievable, however, radiation for this benign tumor is debatable.In conclusion, successful surgical resection of a very rare lumbar extradural epidural soft tissue tumor, angiolipoma, is described. As more cases are described and biopsy specimens collected a better understanding of the genetic etiologies contributing to this rare condition would be obtained. The presence of the lesion in the epidural canal and associated compression can result in neurological symptoms and urinary changes as described in this case.


Lipomas are rarely found in primary mesenchymal tumors of the hypopharynx and larynx. When they do appear, they often macroscopically resemble a retention cyst or laryngeal nodule. The laryngeal spindle cell lipoma, a variant, is extremely rare.A 65-year-old woman presented with a 3-month history of pharyngeal paraesthesia. Laryngoscopy revealed the presence of a well-encapsulated, smooth-surfaced, yellowish, pedicled mass on the left epiglottis. Magnetic resonance imaging confirmed the epiglottic mass.Following excision of the mass, the diagnosis of an spindle cell lipoma was established and confirmed by immunohistochemistry.Surgical excision of the lesion using a controlled-temperature plasma technique (coblation).At the last (4-year) follow-up evaluation, the patient was asymptomatic and without recurrence.SLC involvement of the epiglottis is rare. Coblation is an effective means to remove it. We report our experience with a 65-year-old woman who presented with pharyngeal paraesthesia.Lipomas are the most frequently observed soft tissue tumors in adults. One subtype is the spindle cell lipoma (SCL), which typically presents as a benign lipomatous neoplasm in the posterior neck and back of older men. It accounts for approximately 1.5% of all lipomas.2This study was approved by the Ethics Committee of the First Affiliated Hospital, College of Medicine, Zhe Jiang University, China . Written informed consent was obtained from the patient for publication of this case report and the accompanying images.A 65-year-old woman was admitted to our hospital with a 3-month history of pharyngeal paraesthesia. She did not complain of, or exhibit, hoarseness, choking spells, stridor, or dyspnea. She was a non-smoker. Laryngoscopy revealed the presence of a well-encapsulated, smooth-surfaced, yellowish, pedicled mass arising from the left epiglottis. Magnetic resonance imaging subsequently confirmed the presence of the non-homogeneous soft tissue mass Fig. . There wHistologic examination of the specimen showed proliferation of spindle cells with elongated cytoplasmic processes on a loose, edematous, myxoid background Fig. . ImmunohThe patient's course during the follow-up period was uneventful, with no evidence of disease recurrence at the 4-year postoperative follow-up evaluation.3 It has a 10:1 predilection for the male sex, with the mean age of occurrence at 56 years. In the present case, the patient was a 65-year-old woman. Clinically, the lingual SCL often presents as a painless, slowly enlarging mass in patients 31 to 78 years of age, with male predominance. The yellowish color and soft consistency of the lesion in our patient suggested a lipoma, although it required immunohistochemical staining for a definitive diagnosis.SCL is rare, accounting for approximately 1.5% of all lipomas. Immunohistochemistry had revealed spindle cell positivity for CD34, which has been used as a diagnostic marker for SCL. In the present case, the tumor cells did indeed exhibit immunoreactivity for CD34.Histologically, SCLs are classically composed of mature fat cells, bland spindle cells with low mitotic activity, ropey collagen, and a myxoid matrix. The differential diagnosis is important for tumors that share histological features with SCL, such as the well-differentiated sclerosing liposarcoma.,4 Histopathologically, SCLs are characterized by mature fat cells admixed with spindle cells arranged in prominent collagen bundles in a lobular pattern. The proportion of spindle cells can vary. For example, some tumors are predominantly composed of mature adipocytes and a few scattered spindle cells, whereas others contain mostly spindle cells and only a small number of mature adipocytes. In the current case, the spindle cell component represented a moderate proportion of the tumor. Overall, the clinical, histopathological, and immunohistochemical profile of the present case supports the SCL diagnosis.In contrast to the well-differentiated liposarcoma, which occurs in various locations, SCL mainly appears in subcutaneous regions of the upper back, neck, or shoulder, with uncommon occurrences at other sites.,9 Complete resection is possible with this approach because of the nature and location of these lesions, as shown in the current patient who underwent coblation via microlaryngoscopy. The patient was monitored during a 4-year follow-up period despite the low recurrence rates reported for SCLs, and we found no evidence of symptoms or recurrent disease. Thus, complete surgical resection with the temperature-controlled plasma technique is a useful procedure for addressing an SCL, a rare tumor involving the epiglottis.The preferred method of treatment for laryngeal and hypopharyngeal lipogenic tumors is radical excision of the lesion, ideally under endoscopic vision.www.liwenbianji.cn/ac), for editing the English text of a draft of this manuscript.We thank Nancy Schatken BS, MT(ASCP), from Liwen Bianji, Edanz Group China (Data curation: wang qinying.Investigation: wang qinying.Resources: wang qinying, liu yongcai.Software: chen haihong.Supervision: zhou shuihong.Visualization: chen haihong.Writing – original draft: liu yongcai, chen haihong.Writing – review & editing: zhou shuihong.


Isolated gallbladder tuberculosis is a rare entity, even in endemic zones. Preoperative diagnosis is usually not possible, with most of the cases being diagnosed either as cholecystitis or malignancy. Histopathological examination of the resected specimen clinches the diagnosis. We present a middle-aged man with obstructive jaundice who was diagnosed to have gallbladder malignancy clinically and radiologically and on microscopy, and was diagnosed as gallbladder tuberculosis with the involvement of the common bile duct. On microscopy, diagnosis of gallbladder tuberculosis (GBTB) involving the common bile duct (CBD) was given. Postoperatively, the patient underwent relevant investigations and repeat meticulous clinical examination, and tuberculosis (TB) of other sites was excluded. Isolated GBTB extending to the CBD and presenting as obstructive jaundice has not been reported so far.Gallbladder involvement with tubercular bacilli in isolation is extremely rare.A 55-year-old man presented with persistent pain in the right upper abdomen for 6 months, relieved with medication, which recurred. After 4 months, he noticed progressive jaundice, clay-colored stools, itching, significant weight loss, and anorexia. There was no history of awareness of lump, abdominal distension, or chronic cough. On general examination, he was icteric and had no lymphadenopathy. Abdominal examination revealed palpable gallbladder, which was smooth and firm.The patient's serum total bilirubin was 6.93 mg/dL with direct component of 4.26 mg/dL, serum aspartate aminotransferase was 175 U/L, serum alanine aminotransferase was 153 U/L, and serum alkaline phosphate was 1100 U/L. Abdominal ultrasound scan showed echogenic soft-tissue thickening along the gallbladder neck and proximal CBD with dilatation of intrahepatic biliary radicals. Contrast-enhanced computed tomography showed enhancing thickening at the gallbladder neck (up to 5.5 mm) with periductal spread along the cystic duct and proximal CBD causing its obstruction and proximal intrahepatic biliary radical dilatation . Abdominal drain was removed on the third POD, and he was discharged on the fifth POD. At the time of discharge, his serum total bilirubin was 1.56 mg/dL with direct of 0.72 mg/dL. Retrospectively, TB infection from the abdomen and chest were excluded. Immune-compromised status was ruled out. Antitubercular therapy with isoniazid (I), rifampicin (R), ethambutol (E), and pyrazinamide (P) for 6 months  was started. After the 9-month follow-up, he is doing well.5 GBTB is uncommon because high alkalinity of bile and bile acids prohibit the growth of tubercle bacillus.6 However, cystic duct obstruction causing low bile acid concentration and damage to the gallbladder mucosa predisposes to GBTB. TB can involve the gallbladder by hematogenous or lymphatic route from adjacent positive foci, by serosal spread from a peritoneal lesion or canalicular dissemination.6 In the present case, the blockage of the cystic duct most likely predisposed to tubercular infection. Furthermore, cholelithiasis, which acts as a nidus for the development of TB, has been reported in nearly two-thirds of patients with GBTB,6 but it was not noted in the present case.Hepatobiliary TB accounts for nearly 1% of abdominal TB.7 as seen in the present case. The symptomatology may mimic acute cholecystitis to frank malignancy.4 Hence, GBTB should be considered as a differential diagnosis for gallbladder mass, especially in individuals from an endemic area or in an immunocompromised patient.GBTB has myriad presentations as a component of miliary or disseminated abdominal TB in immunocompromised states or as isolated involvement,5 The existing literature for biliary TB on magnetic resonance imaging (MRI) is scarce and mostly described as biliary strictures.Preoperatively, GBTB poses a diagnostic dilemma despite advanced imaging modalities, leading to missed diagnosis. Contrast-enhanced computed tomography imaging of GBTB may show thickened gallbladder wall, nodular lesion with homogeneous enhancement, calcific flecks, and necrosis.8 can be extrapolated to biliary tubercular lesions as demonstrated in our case.On a retrospective review of MRI findings of this case, we feel that T2 hypointense nature, diffusion restriction, and peripheral enhancement of soft-tissue thickening could have been a clue for a differential of biliary TB. Because biliary malignancies with sclerosing component can show similar features, diagnosis of gallbladder malignancy with periductal spread was made. We suggest that all these findings well described for hepatic tuberculomas4 In the present case, the lymph nodes showed granulomas but not compressing the CBD. Thirdly, GBTB is preferentially noted in young women.5 In the present case, the patient is an older man. Finally, most of the literature mentions classical microscopic findings suggesting the diagnosis of TB. However, documentation of Mycobacterium tuberculosis on ZN stain has only been reported once,2 but image for the same was not shown. The current case demonstrates acid-fast bacilli on ZN stain.Various peculiarities in the present case distinguish it from previously reported cases. First and foremost, the rare presentation of GBTB extending from the cystic duct into CBD leading to obstructive jaundice has not been reported so far. Secondly, the usual causes of obstructive jaundice in context of abdominal TB or GBTB are due to CBD compression by pericholedochal lymph nodes.This case highlights that biliary TB should be included in the preoperative imaging differentials on MRI despite its rarity. This report reiterates the value of meticulous histopathological examination of all gallbladder specimens.Author contributions: B. Varshney, P. Elhence, S. Soni, T. Yadav, and S. Saha wrote the manuscript. B. Varshney, P. Elhence, and VK Varshney revised the manuscript for intellectual content and approved the final manuscript. P. Elhence is the article guarantor.Financial disclosure: None to report.Informed consent was obtained for this case report.


Lymphoepithelial carcinoma (LEC) of the salivary glands is a rare, but distinct, poorly-differentiated neoplasm that resembles undifferentiated nasopharyngeal carcinomas (NPC). Among primary salivary gland LECs, they most commonly arise in the parotid gland. These lesions have a noticeable racial predilection, mostly occurring in Asians and Arctic region native populations. They are strongly associated with Epstein-Barr virus (EBV), and are more common in EBV-endemic areas. The most common presenting symptoms of primary parotid LEC are parotid mass development and cervical lymphadenopathy. We report an unusual case of EBV-negative LEC in a Canadian Caucasian woman.A 40-year-old female from Atlantic Canada presented with a rapidly enlarging preauricular mass and cervical lymphadenopathy. Preoperative diagnostics via radiology and fine needle aspiration alluded to an ambiguous primary parotid malignancy. She underwent a total parotidectomy with facial nerve sacrifice and ipsilateral selective neck dissection. The facial nerve was reconstructed with a lateral antecubital nerve graft. Pathology came back positive for LEC, and she began adjuvant radiotherapy. At her 1-year follow up, she was disease-free and obtained a House-Brackmann facial nerve function grade of 3.The case report highlights the importance of having a high index of suspicion and the need for interdisciplinary collaboration in reaching the diagnosis of primary parotid LEC. LEC can afflict patients of any ethnicity in non-EBV endemic areas and should therefore be considered in all patients with a painless parotid mass regardless of ethnicity. Further studies are required to elucidate the oncogenic role of EBV in these cancers. They are histopathological analogues to undifferentiated nasopharyngeal carcinomas (NPC) and most commonly occur in the nasopharynx. However, LEC has been reported to arise primarily in various organs such as lungs, stomach, breast, ovaries, uterus, renal pelvis, bladder and skin ,20. In aIt is important to distinguish between primary LEC of the salivary glands from secondary metastatic NPC to the salivary glands as the treatment for each differs. Similar to NPC, primary LEC also has a very strong racial and geographical predilection, predominantly affecting the Asian, and Arctic circle native populations, with less than 15% of all primary parotid LECs involving patients of Caucasian descent . Like NPClinically, the most common presenting symptoms of LEC of the parotid gland is a non-tender parotid mass of variable duration, although pain or discomfort may be present. Approximately 20% of cases present with concurrent facial nerve palsy, and 40% with cervical lymphadenopathy, both ipsilateral to the primary lesion. , and som2A 40-year-old Caucasian female from Prince Edward Island, Canada, presented for evaluation of a left preauricular mass of two years’ duration. The patient indicated that the mass had recently seemed firmer and she had become more aware of its presence in the few months leading up to presentation. From a symptomatology perspective, the patient denied pain or tenderness, and hadn’t noticed any facial weakness on the involved side. Her past medical history included a remote sleeve gastrectomy due to obesity, an ankle injury repair, and a cholecystectomy. She was not on any medications. She did not smoke or consume alcohol and had no known allergies. Her family history was negative for head and neck malignancies. On physical examination, a rubbery, fixed, 5-centimeter (cm) nontender mass was easily palpable in the left parotid. She did not have any palpable cervical lymph nodes. Facial nerve function was unremarkable. Flexible nasopharyngoscopy was unremarkable. An initial fine needle aspiration (FNA) was done on the parotid mass at a satellite head and neck clinic on Prince Edward Island, revealing atypical cells. Imaging was also requested, and a computed tomography (CT) scan demonstrated a heterogeneously enhancing solid mass in the left parotid, measuring 3.5 cm × 3.2 cm × 4.6 cm with a component of deep lobe extension and ill-defined margins. Multiple pathological lymph nodes were also seen along the vicinity of the mass and in the neck . A chestAs such, she was scheduled for surgery. A total parotidectomy with facial nerve sacrifice and ipsilateral selective neck dissection were performed. Intraoperatively, the mass was found adherent to surrounding structures, encasing vessels, and also the main trunk of the facial nerve. This required resection of the main nerve trunk along with a component of the upper and lower divisions, while preserving the peripheral branches via retrograde dissection, and ligation of the external carotid. A lateral antecubital nerve graft was used to anastomose the resected temporal and buccal facial nerve segments to the main trunk. All frozen section margins of the proximal and distal facial nerve were negative. The mass was submitted enbloc and an ipsilateral neck dissection of levels II and III was performed without any issues. On microscopic evaluation, however, there was extraparotid extension with margin involvement. There was also extensive neurovascular involvement, including perineural and intraneural invasion, as well as angioinvasion. Multiple intraglandular lymph nodes came back positive, as well as neck nodes from levels II and III, with one node demonstrating extranodal extension. The mass was determined to be a poorly differentiated carcinoma, consistent with a primary lymphoepithelial carcinoma of the parotid on final histopathology. Microscopically, the tumor showed a syncytial growth pattern of undifferentiated, high-grade malignant cells with a patchy infiltrate of small lymphocytes in the background. No glandular or squamous differentiation was distinguished on hematoxylin and eosin sections. . HoweverShe was referred for radiotherapy for the primary site and the neck basin, and then followed up on an annual basis. One year following surgery, she had no evidence of recurrence and was able to close her eye completely. She obtained a House-Brackmann grade 3 out of 6 for her facial nerve function. At the five year follow up mark, she remained recurrence free at the primary site, but unfortunately developed multiple pulmonary metastases and received chemotherapy.3Lymphoepithelial carcinoma of the salivary glands is a distinct and rare tumor, closely associated to anaplastic carcinoma, poorly differentiated carcinoma, or undifferentiated carcinoma. LECs of the head and neck primarily involve either the parotid gland or submandibular gland at a ratio of 7:1 . MicroscThese tumors have a noticeable racial predilection, mostly occurring in Asians and Arctic region natives. They are uncommon among Americans and Europeans, with Caucasians comprising less than 15% of reported cases . FurtherClinically, the average age at presentation is typically during the fourth to fifth decade of life. This neoplasm affects more females than males with a ratio of approximately 3:2 . The mosFNA is a safe and relatively low-cost procedure used to detect malignancy of the salivary glands ,25. WhilGiven the limited diagnostic accuracy of FNA, radiological imaging is a valuable tool in the preoperative evaluation of parotid LEC. Ban et al. (2014) found that most parotid LEC present on CT and MRI as poorly defined masses with lobulated or plaque-like appearance. These lesions typically show homogeneous signal intensity on unenhanced CT and MRI, with no signs of cystic degeneration or calcification. Absence of tumor necrosis may also be diagnostically useful as most salivary gland tumors lack necrotic regions. These radiological features in conjunction with clinical presentation, FNA results, and an absence of nasopharyngeal lesions on nasopharyngoscopy/nasopharyngeal CT and/or biopsy suggests a diagnosis of parotid LEC.Treatment modalities for LEC of the salivary glands include surgical excision, radiation therapy, and chemotherapy . EstabliThe introduction of microsurgery and other techniques made the practice of facial nerve resection in the setting of parotid malignancy when the nerve is uninvolved completely unacceptable . The facBecause of the rarity of this neoplasm, it is difficult to formulate prognostic and survival data. Some reports suggest that LEC has a better prognosis than other undifferentiated carcinomas of the salivary glands, in part due to their radiosensitivity, and because the lymphoid stroma is thought to have a role in limiting the aggressiveness of the tumor . Local r4LEC of the parotid gland is a rare malignant neoplasm that requires complete surgical resection of the primary tumor, ipsilateral selective neck dissection, and postoperative adjuvant radiation therapy. The facial nerve, if involved, should be resected and primarily reconstructed to avoid morbidity. Patient evaluation should include a proper history and careful physical examination including fiberoptic nasopharyngoscopy or nasopharyngeal CT to rule out NPC clinically before the diagnosis of LEC is accepted. FNA of the lesion should be obtained as part of the evaluation and repeated when in doubt or if the results are inconclusive. Imaging in the form of CT and/or MRI are useful diagnostic aids and should be obtained as part of the preoperative diagnosis and planning. Rarely, as is highlighted in this case, it can affect Caucasians in non-EBV endemic areas, illustrating the need to consider primary parotid LEC even in ethnically and demographically low-risk patients. Therefore, a high index of suspicion and interdisciplinary collaboration are required to reach the diagnosis. Moreover, this case report highlights the need for further studies that are required to elucidate the oncogenic role of EBV in these cancers.The authors declare that they have no competing interests.No funding was required for this case report.This case report was performed under the Nova Scotia Health Authority Research Ethics Board guidelines for case reports. No formal research ethics board approval was necessary and therefore no reference number was generated.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.Ashley Whelan: investigation and writing .Ahmed A. Al-Sayed: investigation, validation and writing .Martin Bullock: formal analysis and writing (review and editing).S. Mark Taylor: conceptualization, supervision, and writing (review and editing).1.Name of the registry: N/A.2.Unique identifying number or registration ID: N/A.3.Hyperlink to your specific registration (must be publicly accessible and will be checked): N/A.Ashley Whelan.Data sharing is not applicable to this article as no datasets were generated or analysed during the current study. Data sharing is unavailable for this study as it would compromise patient privacy. However, further information regarding the case is available, within limits of patient privacy, upon request.Not commissioned, externally peer-reviewed.


Congenital dermal sinuses are a rare form of spinal dysraphism. The developmental defects are located along the midline neuroaxis, with sinuses in the cervical region being the least common. Congenital dermal sinuses can be associated with intraspinal infection as they act as a direct route from the skin and subcutaneous tissues into the spinal cord.The authors present two cases of cervical dermal sinuses complicated by intramedullary abscess. Both children presented with neurological decline and febrile illness. MRI showed intraspinal abscess. Both underwent prompt surgical excision of the sinus tract, exploration of the cord and intravenous antibiotics.Both patients demonstrated excellent neurological recovery.Complete surgical excision of the sinus and tract in addition to long-term antimicrobials can yield excellent neurological outcomes. At surgery, do not expect to find pus when exploring the intramedullary component. Long-term follow-up is advocated due to potential late recurrence. Congenital dermal sinuses are a rare form of spinal dysraphism occurring in approximately 1 in 2500 live births.Dermal sinus tracts may occur along the midline neuroaxis, but only 1% of all tracts are in the cervical region.A 3-year-old girl was admitted with gait disturbance. Physical examination showed an excoriated midline cervical dermal sinus over the C6 spinous process. The child had a wide-based, hypertonic gait with hyperreflexia in the lower limbs bilaterally. Magnetic resonance imaging (MRI) of the spine showed an enhancing intradural lesion at C5/C6 which communicated with the subcutaneous tissues posteriorly. This was suggestive of an intradural abscess resulting in a swollen cord with oedematous changes extending from the medulla down to T9.The child underwent surgical excision of the dermal sinus. The tract was dissected down to the dura where the wall of the sinus was in continuity with the spinal dura. No pus was encountered within the cord, only inflamed-looking dermoid-type tissue. Samples were sent for histopathological and microbiological examination. It revealed an epithelial-lined cyst, fibro-fatty connective tissue and skeletal muscle fibres with inflammatory cell infiltration, confirming the diagnosis of dermal sinus. There was no growth on bacterial culture. The child received 6 weeks of intravenous and 4 weeks of oral antimicrobials. Clinical examination at 6 months demonstrated a complete neurological recovery. There has been no recurrence to date after 9 years of follow-up Fig A 16-month-old girl presented with rapid onset neck swelling, torticollis and lethargy. On examination, temperature was 38.2 °C, and she was tachycardiac and tachypnoeic with an increased work of breathing for which she needed intubation and ventilation. On inspection, there was a midline birthmark with a pinhole opening in the mid cervical region. MRI of the spine showed an intraspinal abscess originating from a dermal sinus tract at C4 with oedema extending from the pons to T1.Propionibacterium species. Post-operatively her left arm was plegic. She received 6 weeks of intravenous antimicrobials and made a full neurological recovery. At 14 months follow-up, she remains well. Fig The child was taken to theatre for exploration of the spinal abscess, and the dermal sinus was excised, and inflamed-looking intramedullary dermoid tissue was biopsied. No intramedullary pus was found. Tissue pathology revealed an epithelial-lined cyst with a central punctum and keratinous debris with polymorph infiltrate in keeping with a dermal sinus and intramedullary dermoid tissue. Bacterial culture grew Congenital dermal sinuses are persistent epithelium-lined tracts that connect the skin surface to deeper structures extending down to variable depths.14Historically, the first presentation of a congenital dermal sinus was following an infection of the central nervous system, presenting through rapid neurological decline from inflammation or mass effect.Once diagnosed, prompt surgical excision of the sinus tract with exploration of the abscess should be performed. This should include excision of the tract down to the dura and liberal wash out of the abscess.7Staphylococcus aureus is the most commonly cultured organism from intramedullary spinal cord abscesses resulting from congenital dermal sinusesPropionibacterium species, an organism rarely found in association with an intramedullary abscesses, documented only once previously in the literature.3Alongside surgical management, long-term appropriate intravenous antimicrobials should be commenced.Both of our patients suffered neurological deficits as a result of cervical congenital dermal sinuses complicated by infection. The need for prompt surgical intervention seems reasonable in these cases; however, the management of congenital dermal sinuses identified through cutaneous findings in routine examinations is unclear. Many authors advocate prophylactic surgical resection with complete removal of the sinus to prevent delayed presentations which are associated with worse outcomes.1Both of our case studies presented with intradural abscesses secondary to cervical dermal sinus tracts. We advocate emergency excision of the sinus and tract down to dura and exploration of the cord abscess; however, pus may not be encountered, and extensive cord dissection should be avoided. The role of prophylactic removal of the sinus tract in the cervical region is currently undocumented in the literature.


Ectopic cervical thymus is a rare congenital anomaly, which results because of the failure of complete descent of the thymus. They are incidental findings in the young and may disappear during the early years of life; however, they have rarely been described in adults. Some of them may undergo hyperplasia or neoplastic transformation and become visible or cause symptoms. We report two rare cases of incidentally detected cervical thymic tissue in adults. In one case, the thymic tissue was seen adjacent to a parathyroid adenoma of the left inferior parathyroid gland. In the other, it was seen adjacent to the left inferior parathyroid gland in a case of papillary thyroid carcinoma. In both these cases, the ectopic thymic tissue was diagnosed as a result of pathological examination, not clinically by macroscopic appearance during operation or radiological evaluation. The finding needs to be noted as these ectopic foci can occasionally give rise to hyperplasia and neoplasms of the thymus. Ectopic cervical thymic tissue (ECT) is a rare anomaly; it may be associated with the parathyroid and thyroid glands because of their embryologic relationship . It is aWe report two rare cases of ECT in adults, one associated with left inferior parathyroid adenoma and the other with papillary thyroid carcinoma. In both the cases, the finding was incidental on histopathology examination, not clinically by macroscopic appearance during operation or radiological evaluation. The cases should remind clinicians of the infrequent occurrence of ectopic thymic tissue caused by maldescent during early embryologic development.μg/dL, and TSH-3.85 mIU/mL). On ultrasonography, both the thyroid lobes were normal in size and architecture. There was a focal oval hypoechoic area in the left lobe measuring 6 × 7.8 mm with no vascularity or calcification. Parathyroid glands were not visualized. Few enlarged discrete nodes were seen in the left supraclavicular region with the largest measuring 1.1 cm in diameter. The nodes revealed a normal hilum. Otolaryngology examination was normal.A 42-year-old lady presented with a history of bony pains, multiple fractures, and weight gain over the last 4 years and was bed bound for the last 12 months. Imaging studies revealed severe osteopenia with multiple lytic skeletal lesions. Serum calcium was 12.4 mg/dL, and phosphorus was 2.8 mg/dL. Serum parathyroid hormone was elevated to 1058 pg/mL, and vitamin D3 level was low. The patient was euthyroid . On ultrasound, a solitary nodule 4 × 3 × 1 cm was seen in the left lobe of the thyroid. There was no significant cervical lymphadenopathy. Otolaryngology examination confirmed the presence of palpable left thyroid nodule, else was normal.A 30-year-old lady presented with midline neck swelling which moved with deglutition and was present since last 6 years. The patient was clinically and biochemically euthyroid (T3-0.86 ng/mL, T4-8.3 On fine-needle aspiration cytology, the case was diagnosed as papillary thyroid carcinoma. Cytology showed papillae with and without vascular core, monolayered sheets, and cells with metaplastic and well-defined cytoplasm. Epithelial swirls, chewing-gum colloid, and many cells with intranuclear cytoplasmic inclusions were noted Figures . The patHistopathology confirmed the FNA diagnosis. Sections showed a tumor with arborizing papillae having delicate fibrovascular core. The tumor cells were polygonal with lightly eosinophilic cytoplasm. The nuclei were large, ovoid, and showed ground glass or hypochromatic appearance. Some showed nuclear grooves and pseudoinclusions . In one The patient is on regular follow-up for the last 2 years. There has been no recurrence of the tumor.There is an intimate anatomical and embryological relationship between the inferior parathyroid gland and the thymus. Both are derived from the third pharyngeal pouch and descend down the neck to reach their anatomical position. Any maldescent can lead to ectopic thymic tissue or its lesion being encountered anywhere from the angle of the mandible to the superior mediastinum . These aECT has been commonly reported in the pediatric literature, mostly at autopsy, and uncommonly presents as a neck mass , 5, 6. MECT has been described in the vicinity of a parathyroid adenoma , 10. EbrECT has been peroperatively suspected to be a lymph node metastasis adjacent to a papillary thyroid carcinoma. Histopathology, however, revealed the nodule to be composed of ECT and parathyroid and thyroid tissue . Case 2 Two rare cases of ectopic cervical thymic tissue in an adult adjacent to a parathyroid adenoma and a papillary thyroid carcinoma have been presented. Any ectopic tissue and their neoplasm such as thymic or parathyroid, even though very rare, should always be considered in the differential diagnosis when a neck mass is evaluated, especially in the pediatric age group. The cases should remind pathologists of the infrequent occurrence of ectopic thymic tissue caused by maldescent during early embryologic development.


Postlaryngectomy stomal recurrence can infiltrate the adjacent tissues of tracheal stoma. However, to the best of our knowledge, there is no report about postlaryngectomy stomal recurrence invading the thoracic duct with intra-mass accumulation of chylous fluid.Our patient was a 52-year-old Han man who presented with a cystic-solid mass on the left side of the tracheal stoma after total laryngectomy. A diagnosis of postlaryngectomy stomal recurrence was confirmed by fine-needle aspiration and surgical dissection of the parastomal mass.In the case of parastomal masses in a total laryngectomized patient, the rare differential diagnosis of postlaryngectomy stomal recurrence invading the thoracic duct with intra-mass accumulation of chylous fluid should be considered a possibility. Postlaryngectomy stomal recurrence (PSR) is the diffuse infiltration of a tumor at the junction of trachea and skin after laryngectomy. The occurrence rate of PSR reported in the literature ranges between 5 and 25% , 2. PSR 9/L, with 96% lymphocytes. The fluid was confirmed to be chyle on the basis of these results. The parastomal mass became smaller after the fine-needle aspiration, but returned to its original size on the following day. The same fluid was aspirated by fine-needle aspiration. Subsequently, he was admitted to our department. An ultrasonography of his neck described a 43 mm × 36 mm mixed echoic mass located between the left lobe of the thyroid gland and the great vessels. Other routine preoperative examinations were normal. According to his medical history, computed tomography, and the results of the fine-needle aspiration, a diagnosis of PSR invading the thoracic duct with intra-mass accumulation of chylous fluid was suspected.Our patient was a 52-year-old Han man who underwent partial laryngectomy in our department for squamous cell carcinoma of the glottis in September 2011. He came to our department again for recurrence of laryngeal carcinoma and underwent total laryngectomy and cervical lymph node dissection in November 2016. Then, he received radiotherapy at 60 Gy for 1.5 months. There was no obviously abnormal family history. He healed well until April 2018, when a slowly enlarging mass on the left side of the tracheal stoma was noticed. Since then, he had experienced progressively aggravating dyspnea. He came to our out-patient department in October 2018. A physical examination revealed an immobile, well-defined, partially compressible and non-pulsatile mass that measured approximately 4 cm × 4 cm on the left side of the tracheal stoma (Fig. On October 29, 2018, a parastomal mass excision was performed through both cervical and thoracic incision. An intraoperative frozen section showed moderate differentiated squamous cell carcinoma Fig. . The uppet al. classified PSR into four types according to the location of tumor, which is correlate with the selection of appropriate treatment [PSR is defined as the diffuse infiltration of recurrent cancer at the junction of the cervical skin and the amputated trachea as well as in the soft tissues adjacent to the tracheal stoma after total laryngectomy , 2. Mostreatment . Type I reatment , 7. The reatment –10. In oAfter originating from the cisterna chyli, penetrating through the thorax, the thoracic duct enters the lower left side of the neck posterior to the common carotid artery and the internal jugular vein, and arches 3 to 4 cm above the clavicle. It lies anterior to the phrenic nerve, sympathetic trunk and thyrocervical trunk, and finally terminates into the venous system. The thoracic duct is generally supposed to terminate into the angle between the internal jugular and subclavian veins , but it Chyle has a very characteristic milky-white appearance and a very unusual composition in accordance with both biochemical and cytological analysis. The most notable among all results of biochemical analysis is the high concentration of triglycerides and chylomicrons. The results of cytological analysis will show lymphocytes to be the most predominant cell type, which may be another sign of chyle . HoweverIt is known that the tracheal stoma is located just above the suprasternal fossa which is close to the thoracic duct. The wall of the cervical thoracic duct, which is composed of a layer of connective tissue and smooth muscle fibers without any elastic lamina, is extremely thin . AccordiDiagnostic considerations for a parastomal, partially compressible cervical mass which manifests as a cystic-solid and non-enhancing mass lesion in a total laryngectomized patient, in our opinion, include PSR or enlarged lymph node accompanying central necrosis. However, a diagnosis of PSR invading the thoracic duct with intra-mass accumulation of chylous fluid may not be considered. Therefore, in order to better deal with the tumor and thoracic duct during the operation and reduce postoperative complications, this rare differential diagnosis of parastomal masses should be considered before operation. Fine-needle aspiration is essential for a mass of this type in a total laryngectomized patient because of its important role in preoperative diagnosis.In the case of parastomal masses in a total laryngectomized patient, the rare differential diagnosis of PSR invading the thoracic duct with intra-mass accumulation of chylous fluid should be considered a possibility.


Case Presentation. A 3-year-old female with Currarino syndrome presented with abdominal distention, blood-flecked stools, and prolonged cecostomy flush time. For her anorectal malformation, a colostomy was initially placed. A new colostomy was created at posterior sagittal anorectoplasty (PSARP) to allow the distal rectum to reach the anus without tension. Differentials for her presenting symptoms included a mislocation of the anus, stenosis at the anoplasty site, stricture within the colon, or sacral mass from Currarino syndrome, causing obstructive symptoms. Workup at our hospital included an anorectal exam under anesthesia (EUA), which showed a well-located anus with without stenosis at the anoplasty site, and an antegrade contrast study revealed a featureless descending colon with a 3-4 mm stricture in the distal transverse colon at the site of the previous colostomy, without an obstructing presacral mass. To alleviate this obstruction, the child underwent removal of the chronically ischemic descending colon and a redo-PSARP, where the distal transverse colon was brought down to the anus. She is now able to successfully perform antegrade flushes.  Anorectal malformations (ARM) are complex disorders that often require staged reconstructions. We present a case and imaging findings of a child who developed issues following colostomy closure due to segmental colonic ischemia.  Patients who have had prior surgeries for ARM repair are at a higher risk of complications, including strictures or ischemic complications at areas of previous surgery or colostomy placement. A thorough preoperative workup, including contrast studies, can alert the surgeon to these potential pitfalls. Anorectal malformations (ARM) affect 1 in 5000 children ; problemA 3-year-old female was diagnosed one week after birth with a rectovestibular fistula and presacral mass. Her initial surgery consisted of an anorectal exam under anesthesia (EUA) and diverting divided sigmoid colostomy. Other congenital defects included an atrial septal defect, bilateral vesicoureteral reflux, left hemi-sacral agenesis, tethered cord, anterior myelomeningocele, and didelphys uterus. At 3 months of age, she underwent an intrapelvic myelomeningocele repair and tethered cord release. At 7 months of age, a posterior sagittal anorectoplasty (PSARP), takedown of the sigmoid colostomy, and creation of a transverse colostomy were performed as the rectum was unable to reach the anoplasty site with the current colostomy still in place. Following colostomy reversal, the patient developed episodes of bloody stools, severe constipation requiring senna and lactulose, as well as intermittent saline irrigations. At 2 years of age, given the difficulty with various laxative regimens, a cecostomy tube was placed.The patient presented to our institution at 3 years of age with abdominal bloating, a history of blood-flecked stools, and prolonged cecostomy flush evacuation. Her flush consisted of 400 mL of saline and 25 mL of glycerin. The differential diagnosis for her symptoms included a mislocated anus from her previous PSARP, a stricture at the anoplasty site, a stricture, or other anatomic abnormality of the colon (such as a twist of the pull-through), or a presacral mass obstructing the colon. Rectal EUA revealed a well-located anus and rectal vault without stricture at the anoplasty site. An abdominal radiograph  and retrAntegrade contrast study through the cecostomy  demonstrated a focal transition point measuring 3-4 mm in the distal transverse colon and a small, featureless descending colon .To further delineate the pathology and allow for ascending colon decompression, the patient underwent an exploratory laparoscopy. The anastomosis was patent; however, the descending colon appeared abnormally thick-walled, was narrow in caliber, and featureless, with a thin mesentery from the previous transverse colostomy site to the peritoneal reflection and significant scarring at the level of the previous sigmoid colostomy and rectum . Given tThe patient subsequently underwent excision of the descending colon to include the previous transverse and sigmoid colostomy sites, a redo PSARP, and neoappendicectomy placement. Removal of the colon from the transverse to the sigmoid colostomy site was performed due to the focal narrowing at the transverse colostomy site and the descending colon's ischemic nature, which we were concerned would act as an obstruction point from lack of motility if it remained in place. This was confirmed with the use of indocyanine green fluorescence angiography (SPY), which revealed poor perfusion to the descending colon. The area of the previous sigmoid colostomy was densely adhered to the pelvis with unclear mesentery. After complete mobilization of the colon through the laparotomy incision, SPY technology was utilized to confirm where the blood supply attenuated on the colon. Although the anus was free of stricture, the colon remaining in the pelvis was <5 cm; therefore, a colorectal anastomosis was unable to be performed and a redo PSARP was performed. The patient was placed in lithotomy, and a lone-star retractor was placed to facilitate the anorectoplasty. Multiple sutures were placed through the mucosa of the rectum; circumferential dissection of the distal rectum was performed. Derotation of the colon at the level of adequate blood supply as seen on SPY was performed; the colon was pulled through the anus, ensuring correct orientation. The mucosa was secured to the skin using interrupted sutures . The anaCurrently, the child is in a 1-year post ileostomy closure and redo-PSARP and remains clean for stool with morning flushes with the aid of 2 mg of Imodium in the morning and 4 mg in the evening. Genetic testing has since been performed; both the patient and parent were found to be positive for a pathogenic MNX-1 variant that is consistent with Currarino syndrome.We present a child who experienced abdominal bloating during antegrade colonic flushes due to a long ischemic descending colon stricture situated between two previous colostomy sites. From this case, there are three unique issues to highlight. First, the relatively larger size of the proximal colon alerts that an obstruction may be present. Second, while nonspecific, the lack of descending colonic haustra can be due to ischemia and may indicate that this part of the colon may not function properly, acting as a functional obstruction. This child had two colostomies with subsequent closure, which sandwiched an ischemic descending colon (likely from disruption of the intervening colonic arcade). Any child who presents with abdominal bloating issues and soiling may have an issue from a colostomy closure site. This goes along with the third issue that the focal stricture could be why this child was experiencing the bloating and prolonged flush times during cecostomy flushes.While the creation of a colostomy is necessary for initial fecal diversion in a majority of patients, they are not without risk of complications –4. In onIn patients with ARMs, the best chance of obtaining continence is with a successful repair at the first attempt ; howeverThe diagnosis of colonic ischemia or stricture at a previous colostomy site should be considered in children who have issues with fecal incontinence or abdominal bloating following colostomy closure. While a retrograde contrast enema often is reliable in obtaining the diagnosis, an antegrade enema may help to define any proximal colonic dilation and antegrade obstructions. A high index of suspicion should be maintained in patients with abdominal bloating and fecal incontinence in the setting of a previous colostomy.


Mycobacterium bovis (M. bovis) osteitis is highly suspected. She was treated with antimycobacterium drugs and showed good results. Osteomyelitis can manifest by mimicking bone tumors. Without a biopsy, the pathogen may go undetected. So, interventions such as biopsy are warranted and avoid mass resection without indication. High C-reactive protein (CRP), alkaline phosphatase (ALP), periosteal reaction of radiating spicules, and penumbra sign in magnetic resonance imaging (MRI) are helpful for discriminating osteomyelitis from bone tumor.Chest wall mass in infancy is rare. Malignant lesions are more common than infection or benign tumors. This is a case of a 12-month-old girl who presented with a 2 cm mass at the right costal margin and poor weight gain. Chest radiograph demonstrated a moth-eaten osteolytic lesion at the 8th rib. The resection was performed, and a mass with pus content was found. The positive acid fast stain (AFB) organism was noted. Pathology confirmed caseous granulomatous inflammation compatible with mycobacterial infection. However, QuantiFERON-TB Gold was negative, so  Chest wall mass in infancy is rare . As an iInfection in the form of chest wall osteomyelitis can be caused by bacteria, fungi, and mycobacteria, whether from direct extension of empyema and pneumonia or hematogenous spread . World iH. influenza, and intradermal tuberculosis vaccine at birth.A 12-month-old girl came from Nakornsawan province, who presented with painless progressive palpable round shape mass at the right costal margin. She had no fever. Her appetite was good, but still she had poor weight gain with height and weight both lower than fifth percentile. No history of contact with tuberculosis was found, but her mother had a positive history of TB lymphadenitis and completed treatment more than ten years ago. The natal history was unremarkable. She completed the Thai national immunization program and was given complementary vaccines including pneumococcus, th rib, shown in N: 20.4%, L: 73.1%), PLT: 339,000/mcL, alkaline phosphatase: 201 (150–420) U/L, and hs-CRP: 20.98 (0–5) mg/L. Chest magnetic resonance imaging (MRI) is shown in Physical examination revealed a 2 cm round fixed firm mass at the right anterior lower chest wall. Bacillus Calmette–Guérin (BCG) scar was found at right upper leg. Otherwise, it was unremarkable. Chest radiograph demonstrated a moth-eaten osteolytic lesion at the right anterior 8Mycobacterium tuberculosis complex (MBTC). Pathology, shown in Mycobacterium bovis (M. bovis) osteitis is highly suspected. She was treated with HRZE standard regimen  for 2 months and switched to HR (isoniazid and rifampicin) for a total of 12 months. Outpatient 3-year follow-up demonstrates excellent weight gain, good appetite, and normal wound healing without mass progression.Resection of the mass was arranged. Intraoperative examination revealed a firm round 5 cm mass with signs of osteomyelitis on the rib and pus. The pus was further processed and revealed positive acid fast bacilli stain (AFB) and was reactive of polymerase chain reaction (PCR) for M. tuberculosis. Therefore, other species should be suspected including M. bovis.The diagnostic guidelines recommend that the diagnosis of TB should follow the cautious history of TB contact, symptoms and signs, abnormal chest radiograph, sputum/gastric aspiration smear AFB/culture, and tuberculin skin test . NegativM. bovis infection is more likely to present with extrapulmonary infection [Bacillus Calmette–Guérin (BCG), Tokyo 172-strains, was employed for preventing tuberculosis and has shown excellent results in preventing miliary and meningeal tuberculosis . Althougnfection , 10. Ostnfection . Only 2%nfection .M. bovis to M. tuberculosis. Infected sites are often the rib (6 of 8 cases), and the others are sternum, chest wall, and pulmonary and hematologic dissemination. M. bovis infection was only found in the infant period. Therefore, if age is more than 12 months, M. tuberculosis should be considered first [Review from 8 cases of chest wall mycobacterial infection  shows thed first , 12–18.M. bovis is resistant to pyrazinamide. So, the combination of 2HRZE + 10HR is strongly recommended for osteomyelitis and a longer course is considered in severe disease [Naturally,  disease .Staphylococcus aureus and Klebsiella pneumoniae [Osteomyelitis can manifest by mimicking bone tumors. The most common pathogen in nontuberculous endemic areas are bacteria such as eumoniae . WithoutIt is not possible to distinguish between osteomyelitis and a bone tumor clinically. The majority of osteomyelitis cases did not show concurrent fever and mild or no local reactions . But mulM. bovis should be proven due to difference in drug regimens.This case report shows an atypical finding of a rib mass in infancy. As atypical infection, benign and malignant tumors must be considered. Although infection is in the differential, tissue biopsy or resection may be necessary. CRP, ALP, and penumbra sign on MRI may support the diagnosis of osteomyelitis and favor a biopsy rather than mass excision. For younger patients with mycobacterium osteomyelitis or abscess,


Primary malignant tumors of the aorta are extremely rare. They are frequently located in the abdominal aorta, followed by the thoracic aorta. Sarcomas are the most common histological type. These tumors originate from the middle or intimal layer, the latter being the most common. Symptoms and radiological findings are generally nonspecific. Since their growth is endovascular, embolic phenomena can occur leading to occlusive signs and symptoms.We describe the case of a 75-year-old Hispanic man, a former tobacco smoker, with a history of pain and epigastric tenderness, dysphagia, and weight loss of approximately 6 kg. A thorax computed tomography scan showed a mass within the posterior mediastinum with poorly defined borders and heterogeneous density, located between thoracic vertebra 5–8, with a size of 78 × 53 × 76 mm, with left main bronchus compression. Endobronchial ultrasound-guided transbronchial needle aspiration was performed; it found an extrinsic posterior compression of the left main bronchus with no endobronchial injury. An intimal angiosarcoma of the thoracic aorta was diagnosed.Tumors of the aorta are rare and difficult to diagnose; they are a challenge during the diagnosis, since they usually require open surgical procedures. Endobronchial ultrasound-guided transbronchial needle aspiration associated with rapid on-site examination offered, in this case, the possibility of a successful diagnosis, avoiding major procedures. This is the first case reported in the literature of an intimal angiosarcoma of the thoracic aorta diagnosed using endobronchial ultrasound-guided transbronchial needle aspiration. Primary malignant tumors of the aorta are extremely rare and have heterogeneous histology. They were first described by Brodowski in 1873 . UsuallyA 75-year-old Hispanic man, a former tobacco smoker with a history of chronic obstructive pulmonary disease for which he was on inhaled bronchodilators, consulted with a 4-month history of diarrhea, persistent epigastric abdominal pain, dysphagia, and weight loss of approximately 6 kg. During initial examination, his vital signs were normal and his oropharynx unremarkable; no abnormalities were detected in his head and neck or heart auscultation. Respiratory sounds were generally decreased without any other abnormalities. During an abdominal examination, epigastric tenderness was found. His extremities were symmetrical, without edema, and he had normal peripheral pulses. Abdomen ultrasound, colonoscopy, and upper gastrointestinal endoscopy from the out-patient consult were normal. An abdominal computed tomography (CT) scan showed an irregular mass located close to the posterior esophagus wall, causing extrinsic compression, with heterogeneous highlight and infiltration of the descending aorta, which had an intramural thrombus. A thoracic CT scan showed a mass in the posterior mediastinum, with poorly defined borders and heterogeneous density, located between thoracic vertebra 5–8 (T5–T8), with size of 78 × 53 × 76 mm, with left main bronchus compression, posterior displacement of the esophagus without cleavage plane, which surrounded and infiltrated the aorta with an intraluminal thrombus causing a 50% obstruction Fig. a.Fig. 1A positron emission tomography (PET)/CT scan showed a hypermetabolic mass with malignant features in the posterior mediastinum Fig. b. The maFive passes were needed for a successful sample. Liquid-base cytology was performed, building a block which underwent Papanicolaou staining Fig. b, c. SamAngiosarcomas are classified histopathologically as intimal or mural subtypes, which is clinically relevant since they have different behaviors and prognoses. Intimal sarcoma is polypoid, involves the intima of blood vessels, and grows in the direction of the aortic lumen; it frequently generates occlusion and embolic phenomena. Mural angiosarcoma originates in the media or adventitia and it predominantly grows toward adjacent structures. Immunohistochemistry is essential to confirm the diagnosis. Positivity of CD31 is of great value for the confirmation of an angiosarcoma. Due to the extension of the tumor, no surgical treatment was considered. The oncology team proposed to do two consecutive cycles of chemotherapy: doxorubicin 10 mg/intravenous for 3 days, Isofosfamide (ifosfamide) 1000 mg/intravenous for 4 days, and mesna 400 mg for 4 days was given. After the second course of chemotherapy there was no response to treatment and our patient presented multiple adverse effects, mainly intense abdominal pain and dysphagia. The palliative care team was consulted, who decided to begin management with analgesia; they supported our patient and his family in the process of bereavement. One week later, our patient died.This is the first case of an intimal aorta angiosarcoma diagnosed by EBUS-TBNA, which in this case was the only option, anatomically, to approach the lesion and to guarantee a timely diagnosis without any further interventions. Although the literature supports that core biopsies have a better sensitivity and a lesser risk of localized spread compared to needle aspiration, we believed the primary tumor was inside the bronchi and the rest of the alterations corresponded to metastasis, being irresectable, which placed EBUS-TBNA as the optimal technique considering it is minimally invasive. Furthermore, the mass strongly adhered to the aorta and it was in an anatomical location that was very difficult to reach by open surgery, as considered by the thoracic surgeon who was in agreement with the less invasive approach. EBUS-TBNA is the preferred method to diagnose and stage lung cancer. This technique allows mediastinum evaluation, including extrathoracic metastasis with real-time visualization and sampling . ComplicIn some hospitals, endobronchial ultrasound-guided transvascular needle aspiration (EBUS-TVNA) is performed without complications, when sites for biopsy are not accessible using traditional endobronchial ultrasound, such as the aortopulmonary window , 8. In t


Intramural duodenal hematoma is an uncommon entity, usually associated with trauma. Spontaneous intramural duodenal hematoma is an even more rare phenomenon reported in patients with anticoagulation therapy, gastrointestinal endoscopy procedure or coagulopathy. We report a case of spontaneous intramural duodenal hematoma in a 30-year-old male as a pancreatitis complication, very few cases have been known in the past and still a lot is to be discovered about this rare hematoma associated with pancreatitis. This condition can have catastrophic consequences and should be managed appropriately. In 1838, during an autopsy, McLauchlan first introduced intramural duodenal hematoma ,3. SpontA 30-year-old-male with history of alcohol abuse presented with a complaint of severe epigastric pain. The pain was diffuse across his abdomen, 10/10 in intensity and sharp in quality. The patient was diagnosed with acute pancreatitis and was discharged three days ago. On physical examination, everything was normal except diffuse abdominal tenderness. A nasogastric tube was inserted with improvement in his pain along with fluid management. Analgesics in the form of morphine was used to control the pain. Lab investigation as in Table Chest X-ray and ECG were ordered with assumption of transferring patient for surgery because of presentation with acute abdomen. The patient was evaluated for pancreatitis complications with ultrasonography which showed gallbladder with microlithiasis, sludge and diffuse echogenicity of liver. CT scan showed a 13-cm duodenal hematoma as shown in Figure The surgery team was consulted but they refused any intervention due to low hemoglobin and fear of further worsening of hematoma. The patient was managed conservatively with total parenteral nutrition, nasogastric suction, electrolyte correction and pain control. Repeat CT scan a few days later showed regression of hematoma. After 14 days of hospitalization, the patient became asymptomatic and was discharged.The first case of intramural duodenal hematoma of pancreatic origin was reported in 1938 by Oppenheimer; very few cases have been reported in the past . PancreaIntramural duodenal hematoma is a rare condition in itself which is traumatic in origin. Spontaneous intramural duodenal hematoma as a complication of pancreatitis is an even more rare occurrence, and only few cases were found to be reported in the database. Familiarity of this condition is important to establish timely diagnosis and ruling out other complications of pancreatitis.


We subsequently appraise the related literature and discuss the diagnostic challenges.Dermatofibrosarcoma protuberans (DFSP) is a rare cutaneous sarcoma with an indolent early course that may be misdiagnosed for benign skin pathology. In this case-report we highlight an instance of DFSP arising from a keloid scar  Dermatofibrosarcoma protuberans (DFSP) is a rare, locally invasive, low to intermediate-grade cutanaeous sarcoma arising from the dermis with an incidence of 0.8–4.5 cases/million/year.A 31-year-old female presented to the plastic surgery OPD with a painful, intermittently bleeding mass on her left anterior chest. Clinical history revealed a keloid scar present at the same site for over a decade resulting from a cat scratch during early adulthood. The patient clearly recalled that the wound on her chest had taken months to heal following the injury with the scar eventually becoming lumpy and keloid. The scar had previously been managed with steroid injections. The patient reported a recent change in the appearance of the keloid scar over the preceding year. She was otherwise fit and well. Clinical examination revealed a 3 × 2 cm fungating lesion at the superior pole of a partially involuted scar, with no associated palpable lymphadenopathy see  and 2.FiAn incisional biopsy demonstrated a spindle cell lesion infiltrating the dermis and subcutaneous fat with immunohistochemical features suggestive of dermatofibrosarcoma protuberans (DFSP). The patient was discussed at a specialist skin cancer MDT where recommendation for wide local excision was made. She underwent a staged surgical excision, with reconstruction carried out once histological margins were confirmed as clear and adequate. The defect was both wide and deep and the tumour was found to involve both breast tissue and pectoralis fascia. The defect was reconstructed using a pedicled latissimus dorsi myocutaneous flap see . The pat,DFSPs represent 0.1% of all cutaneous malignancies and account for 2–6% of soft-tissue sarcomas.9n=38) for conventional surgery against 0%  for MMS. When the study data was pooled with all other available published studies this finding was confirmed with recurrence rates of 20.7%, 95% CI: 18.6–22.9%, n=1394) and 1.3% , respectively. Such findings have been supported further by more recent systematic reviewsDFSPs are most frequently diagnosed during their indolent phase when superficial, less than 5 cm in size, non-fixed and confined to the dermis. In view of their malignant potential with high-risk for local recurrence, surgery represents the mainstay of treatment for DFSP on diagnosis.DFSPs have an excellent prognosis if completely and widely excised. Ten-year survival rates are high (>99%)16The treatment of metastatic DFSP can be challenging given its nature as a chemo-resistant tumour. Anecdotally, methotrexate has been employed with limited evidence of efficacy.,In our case, the DFSP arose from a pre-existing keloid scar. Reviewing the literature, there appears to be only one instance where DFSP transformation from a keloid scar has reported. This was in a 26-year-old black male with a history of multiple keloids who presented with a progressive right post-auricular neck lesion present since 10-years-of-age, which was then excised and histologically confirmed to be a keloid. The patient subsequently underwent re-excision of recurrence 8 years later where DFSP was demonstrated and unfortunately died 2 years later from widespread metastatic disease.24We chose to reconstruct the defect for our patient with a pedicled latissimus dorsi flap - a flap commonly used for breast reconstruction. We chose this regional option for its reliability, ease of harvest and transposition as well as the ability to provide a large skin paddle leaving a cosmetic donor site hidden within the patient's bra strap. With this flap we were able to provide supple uniform tissue for her chest wall and replace the volume of breast tissue excised with a single flap. Alternative options considered for this area include a superiorly-based pedicled VRAM , a pectoralis major turnover flap with skin grafting or free-flap reconstruction.de novo. It should be included in any differential diagnosis for apparently benign indolent fibrous skin lesions, particularly in younger patients. As a cutaneous sarcoma DFSPs should be managed aggressively in conjunction with a sarcoma or specialist skin multi-disciplinary team. Despite emerging oncological therapy, surgical excision represents the mainstay of treatment with good 5-year outcomes. Follow-up is advised given the predilection of delayed recurrence.In summary, this case demonstrates how DFSPs are often mistaken for keloids, and highlights that DFSPs may arise from keloids The authors declare no conflict of interests.


Cystic adrenal lesions are rare and uncommon manifestation with few cases reported so far. Different types of adrenal cysts have been described with heterogeneous etiology and overlapping clinical findings, ranging from benign to malignant cystic neoplasm. They are usually asymptomatic or may rarely present with abdominal pain or fullness. Optimum management of adrenal cysts still remain controversial, owing to its low incidence. In this study, we report our institutional experience on diagnosis and management of different histological types of cystic adrenal lesions.During 4 years period, 55 patients underwent adrenalectomy with five cases presenting as adrenal cysts. All the five patients were biochemically nonfunctional and underwent adrenalectomy . The primary indications for surgery were larger size and/or suspicion of malignancy. Histological evaluation revealed two epithelial cysts, one endothelial cyst, one pseudocyst, and a very rare case of adrenocortical carcinoma arising in a pseudocyst.Cystic adrenal lesions are rare with varied etiologic and clinical presentation that may sometimes lead to diagnostic and management dilemma. These cases must undergo biochemical and radiological evaluation to rule out underlying malignancy followed by referral for surgical intervention. He had no history suggestive of a functional adrenal mass. The patient had no comorbidities or significant past history. On clinical examination, abdomen was soft with no evidence of any organomegaly. His upper GI endoscopy revealed normal study. Serum morning cortisol  and 24 hours urinary metanephrine  and nor metanephrine  levels were also within normal limits.Computerized tomography (CT) scan of abdomen revealed well defined, round to oval, hypo dense lesion with hyper dense thin wall measuring 6.3 × 6.4 × 6.6 cm in the retroperitoneum closely abutting pancreas. There was subtle calcification in inferior aspect however no enhancing solid component was identified. Although the patient had no significant symptoms, since the size of the lesion was >6 cm, surgery was planned and he was managed by laparoscopic adrenalectomy via anterior approach. Preoperatively, there was a well‐circumscribed cystic mass lesion, filled with serous fluid, at left suprarenal region. Grossly, the outer surface was smooth, congested, and covered with fibro fatty tissue. Histopathological evaluation revealed a cystic cavity variable wall thickness (2‐3 mm) composed of fibrocollagenous wall which was lined by flattened epithelial cells along with remnants of adrenocortical tissue at the periphery Figure . Hence, Case 2A similar case of 38‐year‐old female patient presented with chief complain of pain in hypochondria, which radiated to back, for 2 years. Serum morning cortisol level was 5.84 μg/dl. Twenty‐four hours urinary metanephrine and normetanephrine levels were 25.90 and 45.93 μg, respectively. CT scan revealed a large well‐defined round to oval non enhancing thin‐walled cystic lesion in suprarenal region on right side leading to significant mass effect to adjacent viscera measuring 66 × 86 × 80 mm. This patient was also managed by laparoscopic adrenalectomy via anterior approach and grossly specimen was received as cystic structure with smooth outer surface weighing 19 g. Final diagnosis was epithelial adrenal cyst.Case 3A 35‐year‐old female who was under investigation for hepatic cystic lesion was referred to the Department of Endocrine Surgery for further evaluation. She had initially presented with complaints of pain in right upper abdomen for 5 months which was insidious in onset, dull aching, non‐radiating and was relieved only on medication. There was no abdominal distension, vomiting, altered bowel, or bladder habits. Serum cortisol (11 μg /dl) and 24 hours urinary metanephrine (50.67 μg) and nor metanephrine (250.56 μg) levels were within normal limits. Biochemical investigations also showed normal liver and kidney function tests as well as negative ELISA for Echinococcus. Ultrasonography of the abdomen, which was performed before the referral revealed an irregular cystic shadow, measuring 35 × 33 mm in the posterior segment of right lobe of liver with foci of calcification, suggestive of right hepatic hydatid cyst. CECT of the abdomen revealed a multiloculated cystic lesion measuring 31 × 32 × 36 mm with multiple wall and septal calcification in right adrenal region. Right adrenal gland was not visualized separately from the lesion. The image was suggestive of an adrenal cystic lesion.This is a retrospective descriptive study including all the patients who visited the Department of Endocrine Surgery (between June 2016 and December 2020) in King George's Medical University, Lucknow in Uttar Pradesh, India with an adrenal mass and were referred to Department of Pathology for histopathological evaluation. Hematoxylin and eosin‐stained sections were reviewed by two pathologists for verification of diagnosis. Macroscopic pathologic features were obtained through review of the pathology reports, macroscopic photographs, and surgical reports. Clinical data as well as pathological details were retrieved from the pathology and clinical files. The adrenal masses which were cystic in nature were identified and studied in detail as well as described in result section of this article. Fifty‐five cases of adrenal mass were evaluated histopathologically for various adrenal pathologies in the Department of Pathology, King George's Medical University between June 2016 and March 2020. Mean age of the cohort was 36 years (age range 1.5‐63 years) with female predominance (M:F = 1:1.7). There were five unusual cases of cystic adrenal lesions including two cases of epithelial adrenal cyst as well as one case each of endothelial cyst, pseudocyst, and a very rare case of adrenocortical carcinoma arising from a pseudocyst. These cases are described below.Case 4A 29‐year‐old normotensive female presented with dull aching pain in right hypochondrial region, radiating to back. She had similar episodes 7 and 3 years back for which she underwent image‐guided aspiration. There was no history of fever, weight loss, visual disturbance, or palpitation. Serum morning cortisol, 24 hours urinary metanephrine and 24 hours urinary normetanephrine levels were 5.80 μg/dl , 118.65 μg , and 228.72 μg . CECT abdomen showed well‐defined right adrenal cyst as rounded hypodense lesion measuring approx. 79 × 75 × 78 mm with internal hemorrhage as slight hyperdense echogenic area in it. No fat component or enhancing nodule was identified. Part of adrenal gland was normally visualized. Per abdomen findings were within normal limits.In view of patient's symptoms, surgical management was planned and retroperitoneoscopic adrenalectomy was performed. Intraoperatively, the lesion appeared multiloculated containing straw‐colored fluid. It measured 4 × 3 cm in size, with all borders free. Both intraoperative and postoperative periods were uneventful and the patient was discharged on postoperative day 2. The postoperative histopathology showed a cystic lesion with fibrocollagenous wall and no evident lining. Remnant of adrenal tissue is seen at the outer aspect. Histology was consistent with adrenal pseudocyst  where seven adrenal pseudocysts were associated with tumor including two pheochromocytomas, one neuroblastoma, one adrenal cortical carcinoma, one adrenal cortical adenoma, one myelolipoma, and one schwannoma. He concluded that because of their heterogeneous etiology and overlapping clinical findings, definite diagnosis relies on extensive sampling and thorough microscopic examination in order to exclude the possibility for coexisting tumor.Optimum management of adrenal cysts still remain controversy, owing to its low incidence. Surgical management, open or minimally invasive, depends on a surgeon's preference, tumor characteristics, and expertise. Surgery is usually indicated in functional cysts, malignant or potentially malignant cysts, symptomatic cysts of any size, asymptomatic cysts of size more than 5 cm, and those patients with uncertain follow upConservative management is apt in those with uncomplicated/asymptomatic cysts <5 cm.The limitation of our study may seem to be lesser number of cases, however looking at the short study duration (~4 years) as opposed to 20 to 25 years data published in larger studies, the spectrum of adrenal cyst documented in our study seems to be appropriate.4In conclusion, cystic adrenal lesions are not as uncommon as mentioned in past literature. These lesions present with varied manifestations and may also sometime present as diagnostic dilemma. Preoperative evaluation plays an important role to rule out other differential diagnosis. Proper investigation including CT or MRI is essential for defining adrenal cystic lesion and differentiating from cystic lesion of adjacent organs. Benign cyst may harbor underlying malignancy mandating a histopathological evaluation of all cystic adrenal lesions. Surgery is the treatment of choice in symptomatic cases as well asymptomatic cases with a large diameter or increasing dimensions during follow‐up or with any anomaly of adrenal hormones.The present study has been approved by the ethical committee of the institute and consent has been taken from the patients for publication of cases.There are no conflicts of interest.Divya Goel: Conceptualization; data curation; formal analysis; methodology. Loreno Enny: Conceptualization; data curation; formal analysis; methodology. chanchal rana: Conceptualization; data curation; formal analysis; investigation; methodology; project administration; supervision; validation; writing‐original draft; writing‐review and editing. Pooja Ramakant: Conceptualization; investigation; writing‐review and editing. Kulranjan Singh: Supervision; writing‐review and editing. Suresh Babu: Writing‐review and editing. Anand Mishra: Supervision; writing‐review and editing.


Ileocecal knitting should be considered as a differential diagnosis of small intestinal obstruction. Aggressive resuscitation and early surgery will be life‐saving. One should anticipate finding extensive bowel gangrene which requires aggressive resection. Ileocecal knitting should be considered as a differential diagnosis of small intestinal obstruction. Aggressive resuscitation and early surgery will be life‐saving. One should anticipate finding extensive bowel gangrene which requires aggressive resection. Most common causes of small bowel obstruction are adhesions, hernias, and small intestinal volvulus while large bowel obstruction is often caused by tumors and volvulus.Advanced bowel obstruction leads to bowel dilation and retention of fluid within the lumen proximal to the obstruction, while bowel decompression occurs distal to the obstruction as luminal contents pass. If bowel dilation is excessive, or strangulation occurs, perfusion to the intestine can be compromised, leading to necrosis or perforation, complications that increase the mortality associated with small bowel obstruction.Intestinal knot syndromes are rare but life‐threatening cause of closed double‐loop obstruction. Various types of intestinal knot syndromes have been described such as ileosigmoid, ileoileal, appendico‐ileal, ileocecal, and knotting by meckel's diverticum. Ileosigmoid knotting is the commonest variety reported in the literatures. In this situation, devitalization of both segments occurs, and the prognosis is grave unless urgent surgical intervention is done.Preoperative diagnosis of intestinal obstruction due to knotting is quite difficult if not impossible.Ileocecal knotting occurs when a loop of ileum wraps around a mobile cecum and ascending colon. It commonly presents with acute small bowel obstruction and needs urgent resuscitation and surgical intervention.In this paper, we present a 21‐year‐old male Ethiopian patient who presented in a critical condition with signs and symptoms of small bowel obstruction but found to have ileocecal knotting with mobile cecum and ascending colon.22 presented with abdominal cramps, bilious vomiting, and abdominal distention of 48 hours duration to Zewditu Memorial Hospital, Addis Ababa, Ethiopia. He had no history of similar illness before, and there was no history of prior surgery. The pain was severe, centrally located with no radiation. There was no history of fever and bloody stool.A 21‐year‐old young Ethiopian man with a BMI of 24.6 kg/mAt presentation, he was acutely sick looking with feeble pulses and a blood pressure of 60/40 mm of mercury. Only 60 mL of urine was collected at initial catheterization. Abdomen was moderately distended with direct and rebound tenderness all over and absent bowel sounds. Rectum was empty on per rectal examination.Two IV lines were secured with wide bore cannulas and aggressive resuscitation with 5 L of crystalloids started. His blood pressure improved to 100/60 mm of mercury and his pulse become well palpable and he produced adequate urine. Investigations revealed white cell count of 18 000 and hemoglobin of 15.49 g/dL. Erect abdominal X‐ray showed multiple air‐fluid level and distended bowel loops suggestive of small bowel obstruction , and the proximal ileum and jejunum were wrapped around the base cecum as well as ascending colon. Most of jejunum, whole of ileum, cecum, and most of ascending colon were gangrenous.The knot was unwrapped, and the gangrenous intestine packed with warm saline for 10 to 15 minutes Figure . IntestiHe was kept in an intensive care for 3 days but there was no need for mechanical ventilation. Later, he developed frequent diarrhea of ten to fifteen times a day starting the 4th postoperative day. This was managed with fluid replacement, antimotility drugs (loperamide), and proton pump inhibitors. Parenteral nutrition was initiated after the 10th postoperative day. The diarrhea decreased gradually, and he was discharged home after 20 days of hospital stay.He was followed for 6 months. During this period, he lost significant weight. He also complains of weakness, crampy abdominal pain, occasional vomiting, and loose stool. Although he was being given a commercially available nutritional formula as supplement, he showed evidences of nutritional deficiencies. The patient was not checked for vitamin B12, folate, vitamins A, D, E, and K deficiencies as these tests are not routinely available here.3Intestinal knot syndromes occur when part of an intestine wraps around the base of a loop of another bowel. The most common variety is ileosigmoid knotting which occurs when an ileum wraps around the base of the sigmoid and passes beneath itself forming a knot. The earliest reported case was that of Parker in 1845, entitled, "Case of Intestinal Obstruction: Sigmoid Flexure Strangulated by the Ileum”.Since then, there are a lot of reports of iliosigmoid knotting in world literature especially from Africa including Ethiopia.The other rarer varieties reported are ileocecal, ileoileal knots, and appendiculoileal knotting.Intestinal obstruction by knotting is a very dangerous acute abdominal condition resulting in the development of gangrene in both loops of bowel involved in a short time. Our patient presented with a 2 days history of symptoms in a state of shock and had majority of small bowel, cecum, and ascending colon gangrenous.In this patient in addition to ileum, most of jejunum was involved in wrapping around the mobile cecum and ascending colon and all these structures were found to be gangrenous. There was only 40 cm of viable jejunum remaining, and anastomosis between jejunum and transverse colon has to be done.2 which is ideal.The presence of a mobile cecum and ascending colon is known to predispose to midgut volvulus as well as knotting by ileum and appendix.The patient developed symptoms of short bowel syndrome which was aggressively treated with fluid and electrolyte replacement as well as parenteral hyperalimentation.Short bowel syndrome in adult is often the consequence of repeated resections for inflammatory bowel disease and occasionally for a gangrenous small bowel volvulus or mesenteric ischemia and has been reported rarely for intestinal knot syndromes.4Ileocecal knotting should be considered as a possible cause of intestinal obstruction and extensive bowel gangrene. Aggressive resuscitations, timely surgical intervention with resection of gangrenous bowel, and use of parenteral nutrition for short bowel syndrome have a good outcome.No competing interest to disclose.The author is the surgeon who operated up on him.Ethical approval was gained from the hospital for the report.Consent to publish was gained from the patient.