Synovial sarcoma (SS) occurs in various parts of the body, predominantly in the extremities. It also occurs in organs without synovial structures. The intracranial disease has been reported as metastasis, but primary intracranial SS has been reported rarely. We report a patient with hemiplegia and a mass on the brain CT. Pathology showed SS with no extracranial pathology. Synovial sarcoma (SS) is one of the most common sarcomas of soft tissue, and it mostly occurs in the lower extremities. It can cause metastasic disease, especially in the lungs, bones, and lymph nodes. Metastasis in the brain is very rare. Primary intracranial SS has also been reported in extremely rare cases. In this article, we study the case of a 28‐year‐old man with primary intracranial SS. With more prevalence in men than women. Most of the SS cases are located in extremities, predominantly lower extremities2The case study is devoted to investigating headache and left hemiplegia in a 28‐year‐old man 10 days before coming to the emergency room. Neurological examination revealed left‐sided hemiplegia, without gait instability and ataxia. Brain computed tomography (CT) scan revealed an intra axial mass lesion with a central necrotic component and surrounding vasogenic edema in the right frontal white matter causing a midline shift to the left side  scan revealed an intra axial mass lesion with a central necrotic component and surrounding vasogenic edema in right frontal white matter causing midline shift to the left side. The patient underwent MRI with gadolinium contrast for more evaluation, and the findings were as mentioned below: An Intra axial well‐circumscribed heterogeneous mass with central necrosis and irregular peripheral enhancement and significant surrounding vasogenic edema in right frontal white matter. Restriction of the peripheral solid component of mas was also seen. The patient was taken to the operation room and underwent total excision of the mass. Many hours later, the left hemiplegia was gradually improved. The histopathological assessment of the mass revealed neoplastic tissue composed of biphasic patterns: large irregular sheets of monotonous cells some with central necrosis set in the fibroblastic stroma. Tumoral cells are plump and have a very high N/C ratio. Frequent mitotic figures are present.IHC staining show positive reactivity for TLE1, FEI1, and INI1 and negative reactivity for Synaptophysin,Olig 2, SMA, CK CD 99. GFAP staining is nonspecific. The aforementioned findings are consistent with SS. The PET scan did not show any other metastatic disease. The abdominopelvic CT scan was also normal.4SS is one of the most common sarcomas of soft tissue, and it mostly occurs in the lower extremities. It can cause metastasic disease, especially in the lungs, bones, and lymph nodes. Metastasis in the brain is very rare. Primary intracranial SS has also been reported extremely rare. In this article, we present a 28‐year‐old man with primary intracranial SS. It can be misdiagnosed with other intracranial masses. So, it is important to confirm the diagnosis with pathology and resect the tumor and start radiotherapy treatment if needed.MAK and IM chose the case, reported the CT scan and MRI and supervised manuscript preparation. ZHT reviewed the published literature and wrote the case report manuscript. AS reviewed the published literature, interviewed the patient, and collected the patient past medical history. MAK, IM, and AS edited the written manuscript. All the authors read and approved the final manuscript.The authors declare that there are no competing interests.All the ethics standards and consent were obtained for this manuscript.Written informed consent for publication was obtained from the patient.


Desmoid tumors (DTs) are rare locally aggressive benign soft tissue tumors with an estimated annual incidence of two to four new cases per million people. The giant intra-abdominal mass presents a diagnostic challenge that includes a broad differential diagnosis of gastrointestinal stromal tumor (GIST), fibrosarcoma, retroperitoneal fibrosis, and other malignancies from adjacent organs. We report a case of a 38-year-old male patient with a giant intra‑abdominal mass. Magnetic resonance imaging (MRI) of the abdomen and pelvis indicated mucinous cystic neoplasm or gastrointestinal stromal tumor (GIST), but histopathology confirmed it to be a desmoid tumor. The patient was discharged, and on follow-up five months until now, there is no recurrence. This case highlighted the importance of including DT in the differential diagnosis of very large intra‑abdominal masses. Desmoid tumors (DTs) are rare locally aggressive benign soft tissue tumors with an estimated annual incidence of two to four new cases per million people in the general population . Giant mA 38-year-old male patient presented with progressive abdominal pain, swelling, and dyspnea on exertion for two to three months. Physical examination showed a massively distended, firm, but non-tender abdomen. He also had moderate pitting edema of bilateral lower extremities but was otherwise unremarkable. He denied fevers, chills, weight loss, night sweats, changes in bowel habits, or hematochezia. Serum CEA, CA-19-9, and AFP were all negative. He had no family history of colon cancer, familial adenomatous polyposis (FAP), or personal history of abdominal trauma. Computed tomography (CT) scan showed a massive peritoneal mixed density (solid and cystic components) mass encasing the ascending colon. Magnetic resonance imaging (MRI) was suggested to further elucidate the origin of this mass . It was predominantly infra-mesocolon and demonstrated areas of small solid enhancement. Based on imaging, the tumor was most likely arising from the right lower quadrant with encasement of the cecum and vermiform appendix. Considering the predominant T2 signal on MRI, the differential diagnosis included mucinous cystic neoplasm versus gastrointestinal stromal tumor (GIST). The patient underwent an elective exploratory laparotomy to remove a giant intra-abdominal mass occupying the whole abdomen and was densely adherent to the ileocecum. Right partial colectomy with ileocolonic anastomosis and ileocolostomy were performed after the mass was removed.The macroscopic pathological examination showed a very large, well-circumscribed, firm mass weighing 12,804 g and measuring 40 × 34 × 15 cm. The mass revealed a smooth external surface and was attached to the ileocecum and appendix . No tumor necrosis and hemorrhage were identified. Stroma was collagenous with occasional myxoid changes Figure . These fImmunohistochemical stains showed diffuse staining of beta-catenin, calretinin, and partial cyclin D1 Figure  and negaBacteroides fragilis, sensitive to piperacillin and tazobactam) requiring central line placement, intravenous infusion of piperacillin and tazobactam, and total parenteral nutrition (TPN). He was discharged after successful treatment of the abovementioned complications. Postoperative follow-up has been five months until the paper submission with no recurrence.The patient's postoperative course was complicated by the development of multiple intra-abdominal abscesses that required drain placement by interventional radiology. The patient also was found to have Gram-negative bacteremia , liposarcoma , low-graSurgical excision is the primary option for the treatment of intra-abdominal DT. Postoperative radiotherapy, chemotherapy, or a combination of the two has been used in cases of positive margins with variable successes . The recWe present a very rare, giant intra-abdominal DT in a male patient, which is the largest one in the reported literature to date. Differential diagnoses for giant intra-abdominal masses are very broad, including DT and inflammatory conditions, and benign and malignant neoplasms. Clinical history, histology features, immunohistochemical tests, and, in some cases, molecular studies are important to solve this challenging diagnosis.


Osteoid osteoma (OO) is a benign osteoblastic tumor characterized by nocturnal pain that responds well to non-steroidal anti-inflammatory drugs. This condition commonly affects adolescents and young adults, and patients between 5 and 24 years of age account for 85% of all OO cases; it occurs very rarely in patients under 5 years old. Tumors often occur in the cortical bone in the diaphysis and metaphysis of the appendicular skeleton and are more common in the lower extremities than upper extremities. Here, we present an extremely rare case of intramedullary OO that arose in the proximal metaphysis of the humerus in a 2-year-old boy, which mimicked subacute osteomyelitis on imaging studies. We also conducted a retrospective literature review and found that the intramedullary location was fairly common in very young patients (<6 years old) with OO. Osteoid osteoma (OO) is a benign osteoblastic tumor characterized by nocturnal pain that responds well to non-steroidal anti-inflammatory drugs (NSAIDs). This condition commonly affects adolescents and young adults, and patients between 5 and 24 years of age account for 85% of all OO cases 3/μL, C-reactive protein 0.01 mg/dL) showed no abnormal findings. Given the age, location of the lesion (metaphysis of the humerus), radiological findings, and recent history of BCG vaccination, subacute osteomyelitis, including BCG osteomyelitis, was primarily suspected, while OO was also considered as a differential diagnosis based on the characteristic pain at the time of waking up and the imaging findings. Curettage of the lesion was performed for both diagnostic and therapeutic purposes. Intraoperative findings showed that the lesion comprised red coarse tissue surrounded by highly firm bone without presence of abscess. Considering the high probability of OO, we further cauterized the surrounding hardened bone with an electric scalpel after simple curettage of the lesion  vaccination on his left arm at 7 months. On examination, his upper right limb was thinner than the left, and marked tenderness was observed in the right arm. He used only his left hand when he played with toys did not like to be touched on the right arm. There was no limit to passive range of motion of the shoulder and elbow joints. Plain radiographs of the right humerus showed an 8 × 3 mm oval lucent lesion in the proximal metaphysis surrounded by osteosclerotic bone, and the adjacent cortex was unilaterally thickened A. On come lesion . Patholoe lesion . No acide lesion A-E, and OO is a benign osteoblastic tumor, first described by Jaffe in 1935, which comprises approximately 10% of all benign osseous neoplasms and approximately 5% of all primary bone tumors OO characteristically appears as a lucent area called a nidus, with a surrounding reactive sclerotic bone and unilateral thickening of the cortical bone on radiographs and CT Kayser et al. classified OO into 4 subtypes according to the location of nidus on axial images of CT or MRI, namely subperiosteal type, cortical type, endosteal type, and intramedullary type Given the above-mentioned characteristic features, image findings of OO are similar to osteomyelitis. Above all, brodie abscesses occur predominantly at the metaphysis of the long bone canals in children, but often have a poorly elevated inflammatory response. Radiography shows lucent lesions with surrounding sclerotic margins in the marrow, very similar to an intramedullary OO. Typical MRI findings of Brodie abscess are indicated by the presence of high-intensity granulation tissue between central rotting/abscess and surrounding osteosclerosis, which shows low intensity on T1-weighted images. This layered appearance is called the penumbra sign. The specificity is 99% and the diagnostic value is high, but the sensitivity is 70%, and it is considered a good possibility that the initial image of Brodie abscess will show the MRI signal pattern, as in our case, without showing the penumbra sign Given that BCG osteomyelitis is a rare late-onset complication of BCG vaccination that often occurs about 1 year after vaccination, it was one of the predominant differential diagnoses in our case. According to an international survey on the complications of BCG vaccination, the incidence of osteomyelitis was 0.89-2.41 and 0.02-0.06 per 1 million among vaccine recipients at ages <1 year and ≥1 year, respectively Although it is known that pain arising from OO resolves spontaneously over a long period of time, the analgesic effect of NSAIDs is generally not sufficiently long-lasting, and surgery is performed for analgesic purposes. In children, such as in the present case, surgery is often chosen early because NSAIDs are inadvisable owing to the high risk of side effects, and overgrowth of the affected limb may lead to leg-length differences and bone and joint deformities ,16. The We conducted a retrospective literature review and summarized 15 cases of OO in children younger than 5 years  Because the lesion was in an intramedullary location with surrounding bone marrow edema, it was very difficult to differentiate it from osteomyelitis preoperatively, and it was not possible to diagnose OO only by imaging. We classified the localization of nidus based on Kayser's classification in past case reports of OO occurring in children younger than 5 years . AlthougIn conclusion, we report herein a case of intramedullary OO of the humerus in a 2-year-old boy. Because OO in children younger than 5 years is very rare and the symptoms are diverse, which makes it difficult to diagnose, careful imaging is important on considering the possibility of OO, especially when nocturnal limb pain that worsens in the early morning is reported. Given the many cases of intramedullary type OO in young children that have now been reported to date, when an intramedullary lesion is discovered in such patients, OO is one of the conditions that need to be ruled out together with osteomyelitis.When the patient's parents were informed that data concerning the case would be submitted for publication, they provided their consent.


Giant cell tumors (GCT) are benign but locally aggressive neoplasms composed of osteoclast-like giant cells and fusiform to ovoid mononuclear stromal cells. GCT generally comprise 5-10% of all benign bone tumors; they appear most frequently between the second and third decades of life. These tumors are also distributed throughout the vertebral column. Approximately half of all spinal GTCs develop in the sacrum. Many cases remain clinically silent and are discovered incidentally during the study of minor trauma. Symptomatic tumors often mimic other common spinal pathologies. Imaging studies ideal for diagnosis are CT and MRI. The techniques used in the treatment of giant cell tumors are curettage or intralesional surgery, block resection, radiotherapy, and chemotherapy. Herein, we report on a 23-year-old female patient diagnosed with a tumoral mass in the anterior part of the sacrum, suggestive of GCT. The lesion was completely excised in two consecutive surgeries, and lumbopelvic fixation was performed with favorable immediate postoperative results. Careful surgical planning with a multi-disciplinary approach leading to block resection still remains the most viable option for the treatment of vertebral GCT. Giant cell tumors (GCT) are rare and benign tumors of the bone. They are composed of osteoclast-like giant cells and fusiform to ovoid mononuclear stromal cells . GCT genImaging studies ideal for diagnosis are CT and MRI. CT shows an attenuation of the soft parts with defined margins and occasional sclerosis and osteolytic findings . VariablA female patient of 23 years of age with no hereditary family history of importance and an unremarkable personal medical history up-to-date was referred to the department of neurosurgery. She had presented with pelvic pain emanating from the lumbo-sacral region, specifically at night. The pain radiated to the lower left limb, causing an alteration of the gait and a loss of the tip-toe gait. She had a left-sided foot drop and an unintentional weight loss of 14 kg in the last year.A pelvic ultrasound was performed, which revealed an intrapelvic mass. Imaging studies were completed with lumbo-sacral and pelvic CTs and MRIs. The CT revealed a tumoral mass in the sacrum predominantly on the left side, extending superiorly to the left L5 transverse process, laterally to the lateral sacral crest, and obliterating the S1 and S2 neural foraminae  were performed. Approximately 80% of the tumor mass was resected. The excision of the tumor was very laborious and with a significant blood loss (mostly from the tumor bed and the bone margins), requiring a transfusion of two units of erythrocyte suspension. The surgical margins of the anterior pathway were contaminated with tumor rupture and dispersion of the material.After a week, the second surgical step was employed via a posterior lumbo-sacral approach. Through this approach, the remaining tumor was resected. A tumor mass was observed that invaded the left paraspinal muscles, which was resected with a wide surgical margin. A subperiosteal dissection of L3 to S3 was performed to clearly identify spinous processes. For partial sacrectomy, an S1 to S3 laminectomy was performed, and the S1-S2 roots were carefully identified and preserved. There was partial tearing of the dura mater, which was sutured. An L5-S1 discectomy was performed. The void space left, especially in the left sacroiliac joint, was filled with polymethylmethacrylate (PMMA). Bilateral transpedicular polyaxial screws were placed at the L4 and L5 levels. Two rods were placed, which were connected with a bar-to-bar connector to the screws placed in the iliac wing. There were no surgically sacrificed nerve roots.In the immediate postoperative period of the second surgical period, the patient presented paresis in the extension of the knee and dorsiflexion of the left ankle, accompanied by hypoesthesia of the L5-S1 dermatome. Postoperative CT and X-ray analysis Figure  showed tThe patient has consented to her radiological images and results being published for medical use as long as they are anonymized.The surgical treatment of sacral tumors is one of the great challenges for every spinal surgeon. The sacral canal and the pelvis are able to adapt to growing lesions, so sacrococcygeal tumors can be diagnosed after extensive growth . Tumors The need for careful preoperative planning and patient selection cannot be emphasized enough. Conventional radiological studies are usually insufficient in providing the detailed anatomical properties of the tumor and its surroundings. The surgeon must keep in mind that the oversized tumor may have displaced the normal positioning of anatomical structures and destroyed bony landmarks. Thus, all possible modern imaging technologies should be employed to prepare the surgeon for the upcoming challenge. The preoperative planning performed in a multi-disciplinary environment allows for the discussion of different approach strategies and the proper combination of techniques. Each surgical specialty has a different appreciation of the different anatomical structures located in the sacro-coccygeal region that prove beneficial in the treatment of the patient.Total and partial sacrectomy present a high risk of morbidity leading to motor, urinary, bowel, and sexual dysfunctions. The extent of resection of the sacrum can also cause lumbo-pelvic instability. Thus, the amount of resection, determining the surgical borders, and the possibility of root sacrifice are vital in predicting the functional outcome of the patient . The patGiant cell tumors are benign but locally aggressive and highly recurrent lesions, with poor responses to radiation and chemotherapy. The oncological goal of surgery is total excision.The broad resection of the tumor and the decompression of the neural elements associated with instrumented fusion allow for a stable spine, preserve or restore neural function, and prevent tumor recurrence . This caThe clinical presentation, the age of the patients, and the long-term surgical results make the reporting of complex cases treated by multidisciplinary teams extremely valuable. The broad resection of the tumor and the decompression of the neural elements associated with instrumented fusion allow for a stable spine, preserve or restore neural function, and prevent tumor recurrence. Tumor excision followed by fixation can reduce pain, decrease neurological deficits, and maintain or regain normal gait .The primary treatment of most sacral tumors remains surgical as chordoma, chondrosarcoma, and some giant cell tumors are resistant to radiation therapy and chemotherapy. Long-term control or even the cure of the disease can be achieved by a block resection of the tumor with sufficient margins. But due to the complexity of the sacro-coccygeal region and the need for lumbo-pelvic stability, a multi-disciplinary approach with meticulous preoperative planning is a must for sacral tumors. Even the most complex giant cell tumors of the sacrum can be successfully treated, but the patient should always be made aware of the possible complications and their implications for the remaining period of their life while including their views in the decision-making team.


Thyroid goiter is a benign chronic enlargement of the thyroid gland, which presents as a painless anterior neck mass with occasional extension to the mediastinum. Retropharyngeal goiter is a rare presentation and hardly reported in the literature.A 70-year-old male presented with a multinodular goiter with a large retropharyngeal component.Physical examination of the head and neck revealed a massive anterior neck mass. Laryngeal endoscopy with a 70-degree rigid endoscope demonstrated a large retropharyngeal mass completely obstructing the view of the larynx. Computed tomography (CT) scan revealed a large multinodular goiter with suprahyoid and retrosternal extension, resulting in displacement of the trachea.The mass was excised completely under general anesthesia and intubation was done under fiberoptic bronchoscopy guidance. The mass was sent for histological analysis, which confirmed the diagnosis of Hashimoto thyroiditis.Eventually, upon follow-up at three months post excision, no evidence of recurrence was detected. •A 70-year-old male presented with a multinodular goiter with a large retropharyngeal component.•The goiter was excised completely under general anesthesia and intubation was done under fiberoptic bronchoscopy guidance•The goiter was sent for histological analysis, which confirmed the diagnosis of Hashimoto thyroiditis.•Postoperative fiberoptic laryngoscopy revealed a significantly improved airway with bilateral symmetrical moving vocal cords. The incidence of thyroid goiter in developed countries is approximately 4%, but this is around 10% in countries with iodine deficiency 2This work has been reported in line with the SCARE 2020 criteria A 70-year-old male presented to the otorhinolaryngology (ORL) outpatient clinic at King Fahad Medical Military Complex in Al Dhahran, Saudi Arabia, for the evaluation of an anterior neck mass. He first noticed the mass one year prior to presentation. At that time, the mass was painless but was gradually increasing in size. He denied dysphagia, changes in voice quality, dyspnea, or signs of hypo- or hyperthyroidism. He also denied weight loss, night sweats, and had no history of recent foreign travel or radiation exposure. He was otherwise healthy and reported a long history of smoking but no alcohol consumption. No family history of malignancies was reported. On physical examination, there was a large firm painless anterior neck mass which did not move with deglutition. Laryngeal endoscopy with a 70-degree rigid endoscope demonstrated a huge retropharyngeal mass indenting the posterior pharyngeal wall at the level of the oropharynx which completely obstructed the view of the larynx. Thyroid function tests revealed that the patient was euthyroid. On CT scan, there was a large multinodular goiter with suprahyoid and retrosternal extension, resulting in mass effect upon the surrounding structures including displacement of the trachea . Total t3The thyroid gland lies in the anterior triangle of the neck, situated within the pretracheal layer and attached to the trachea via the ligament of Berry. The pretacheal layer of fascia extends between the hyoid bone superiorly and the mediastinum inferiorly. It has two parts: the muscular part which encloses the infrahyoid muscles, and the visceral part which encloses the thyroid gland, trachea, and esophagus. The posterior aspect of the visceral fascia is formed by contributions from the buccopharyngeal fascia (fascia covering the pharynx). The retropharyngeal space is located between the buccopharyngeal fascia and the prevertebral fascia; it extends from level of the skull base to the posterior mediastinum.The superior extension of the thyroid gland is limited by the superior attachment of the pretracheal fascia and the attachment of the infrahyoid muscles. Due to anatomic compartments created by fascial planes within the neck, goiters tend to grow into the mediastinum where it follows the path of least resistance, resulting in substernal or retrosternal goiter. This causes symptoms related to compression of the airway and/or esophagus. In a minority of patients, mediastinal extension is followed by growth superiorly behind the oro-hypopharynx, which may produce dysphagia and dysphonia In conducting a literature review, seven cases of retropharyngeal goiter have been reported In the present patient, due to the impending compressive symptoms our patient was managed surgically via a classical total thyroidectomy, and intubation in the OR was done under fiberoptic bronchoscopy guidance as there was a concern for upper airway obstruction and laryngeal compression.4Our case demonstrates the importance of understanding neck spaces in relation to visceral structures. In patients with thyroid goiter, it is important to recognize the possibility of a retropharyngeal or mediastinal extension of the thyroid gland and consider these as differential diagnoses of a neck mass. CT imaging of the neck is recommended in such presentations and surgical intervention is the treatment of choice to prevent airway obstruction. In addition, a multidisciplinary team approach with careful preoperative evaluation using fiberoptic laryngoscopy and an extensive discussion of the potential plans to secure the airway with the anesthesia team will prevent poor outcomes.This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.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.The case report was approved by the institutional review board at King Fahad Medical Military Complex, Dhahran, Saudi Arabia.Not commissioned, externally peer-reviewed.Alhanouf Alhedaithy: Wrote the manuscript. Part of the surgical team.Abdulaziz AlGhamdi: Part of the surgical team.Tariq Abualhamayel: Operated the patient, Reviewed the manuscript.Nada A Aldabal: Operated the patient, Reviewed the manuscript.Not applicable.Alhanouf Alhedaithy.None.


Paraganglioma-pheochromocytoma (PPGLs) are relatively rare catecholamine-secreting tumors of chromaffin origin. Due to the sympathetic effects of catecholamine excess, their presentation may range from non-specific symptoms to dangerous hypertensive crises. We present the case of a 36-year-old lady with recurrent paraganglioma (PGL) who presented in emergency with hypertensive crisis. She had a history of surgery for left-sided PGL 18 years earlier. Imaging showed local recurrence with pulmonary metastases and blood biochemistry showed raised urinary metanephrines. In view of her poor general condition, we undertook a staged surgical approach for management. She first underwent en-bloc excision of recurrent PGL with left nephrectomy. Nine weeks later, she underwent a pulmonary metastasectomy. This staged surgical approach resulted in the stabilization of blood pressure and normalization of urinary catecholamine. Although most of these tumors are indolent by nature, this case highlights the metastatic potential of apparently benign PGL. This case explores the possibility of a staged surgical approach in a high-risk patient and emphasizes the need for long-term follow-up in these cases. Paraganglioma (PGL) and pheochromocytoma (PCC) are neuroendocrine tumors of chromaffin origin. PGL arises from extra-adrenal paraganglia while PCC originates from the adrenal medulla . They arPresentation of PPGL often gets delayed due to the indolent nature of the disease and poor awareness among the patients. Even apparently benign PPGL may recur decades after initial diagnosis. Patients with recurrent disease manifest late in a debilitating condition. Such recurrences are difficult to manage. We are describing a case of PGL with recurrence 18 years after initial treatment. We have highlighted the staged surgical management of local and distant recurrence in the present report.A 36-year-old female presented in the emergency department for the evaluation of worsening headache and altered sensorium. On examination, her systolic blood pressure (BP) was 200 mmHg, Glasgow coma scale (GCS) was 14/15 with right-sided hemiparesis. There was no history of seizures, fever, ear discharge, head trauma, and bladder or bowel involvement. Investigations revealed deranged creatinine of 2.5 mg/dL . Magnetic resonance imaging (MRI) brain was suggestive of multiple infracts.2 encapsulated mass abutting the inferior pole of the left kidney was excised completely. Histopathology (HPE) of the operated specimen was compatible with PGL.Significant past clinical details included a history of surgery for left-sided PGL 18 years back. At that time, she had decreased vision (grade IV hypertensive retinopathy), resistant hypertension, and elevated 24-hours urinary norepinephrine. A 5×7 cm2 mass arising anterior to the left kidney with a few enlarged lymph nodes completely encasing the left renal artery and vein . Contrast-enhanced CT scan of the abdomen revealed a 2.6×2.7 cmn Figure  with lefA 68Ga-DOTANAC whole-body PET/CT scan revealed radiotracer uptake in the left paraaortic region of the abdomen, and right pulmonary lower lobe Figure  suggesti2 nodule in the right lower lobe, 0.6×0.5 cm2 nodule in the left lower lobe, and multiple centrilobular as well acinar nodules in the left lung suggestive of metastasis  nine weeks after laparotomy Figure . HistopaThe patient made an uneventful recovery after two surgeries. A repeat 68Ga-DOTANOC whole-body PET/CT at three months after pulmonary nodule resection showed no uptake in the left paraaortic and pulmonary areas  and PPGL associated with inherited syndromes are more likely to recur . PPGL wiSurgical resection is the treatment of choice for localized PPGL . DebulkiThe major debulking surgery in our patient may have resulted in the spontaneous resolution of smaller lung metastases. Few reports highlight the spontaneous regression of PPGL. Hammer et al. reported the spontaneous disappearance of the left carotid body tumor eight years after the excision of contralateral PGL . Chang ePPGLs are rare tumors with a high probability of recurrence even decades after the initial diagnosis. The Endocrine Society Clinical Practice Guideline (ESCPG) advises lifelong follow-up while the European Society of Endocrinology (ESE) suggests a 10-year follow-up for all operated cases of PPGL ,18. ESE The present case emphasizes the need for long-term regular follow-up of all cases of PPGL. Patients should be educated regarding the possible delayed recurrence of an apparently benign PPGL. In patients with poor performance status and extensive disease, staged surgery is an excellent option. Debulking surgery may lead to spontaneous resolution of small metastatic nodules and improve long-term survival.


The patient underwent a pretemporal transcavernous transtentorial approach for tumor removal. The pathological report showed an anaplastic astrocytoma. In this approach, a wider surgical corridor was obtained by opening the Meckel’s cave and cutting the tentorium, via which a safe entry point into the pons could be determined with neuromonitoring. In the authors’ opinion, this approach is safe and effective in selected ventrolateral pontine gliomas.A 39-year-old man, who had a history of spinal myxopapillary ependymoma with cerebrospinal seeding status post twice operations and radiation therapy, presented with aggravating headaches, diplopia, dysphagia, and unsteady gait for 2 weeks. The brain MRI revealed a parenchymal lesion at the left aspect of the pons, about 2.8 × 2.3 × 3.2 cmhttps://youtu.be/sUt-9QFGgCI.The video can be found here:  Pretemporal transcavernous transtentorial approach for left pontine glioma. A 39-year-old man had worsening headaches, diplopia, dysphagia, and unsteady gait for more than 2 weeks. The MRI showed an intraparenchymal lesion at the left aspect of the pons. The corticospinal tracts were pushed backwards. The tentative diagnosis was a left pontine glioma. We chose a pretemporal transcavernous transtentorial approach to target the lesion. Surgical approaches to the lesions of the ventrolateral brainstem include Dolenc and Kawase approaches. With Dolenc approach, we can access the floor of the third ventricle, the cerebral peduncle, and the upper pons; with Kawase approach, we can target the more lateral aspect of the middle and lower pons. With the combination of both, we have a panoramic view from the midbrain to the pons, which offers a wider area for the selection of suitable entry point to the brainstem. Under general anesthesia, the patient was in supine position with the head fixed by the Mayfield head holder. Intraoperative neuromonitoring was applied. A standard pterional craniotomy was performed with zygoma fracture. Flatten the sphenoid wing. Cut the meningoorbital fold. Expose the periorbita. Peel the lateral wall of the cavernous sinus. Inject tissue glues between V1 and V2. Coagulate and cut the MMA. Expose the GSPN and petrous apex. Expose and remove the ACP extradurally. Expose the third nerve to the oculomotor triangle extradurally. Remove the petrous apex. In this case, the tumor had a diffuse growth from the left cerebral peduncle to the whole left aspect of the pons. In our opinion, if we only used Kawase approach subtemporally, the pontine exposure would be limited, and the traction injury to the dominant temporal lobe was of concern. Hence, the combination of Dolenc and Kawase approaches was adopted. Cut the dura and tentorial incisura along the dashed red line. If the ACP was not removed, mobilization of the third nerve at the oculomotor triangle and the fourth nerve at its entry to the cavernous sinus would be difficult, and the following trans-Meckel’s cave/transtentorial procedures would be impeded as well. The goal of the transcavernous approach is to achieve neurovascular mobilization to gain a wider surgical corridor and to avoid traction injury to the cranial nerves. Cut the tentorium. Open the Meckel’s cave. Ligate the superior petrosal sinus. For a wider surgical exposure, we removed the ACP to access the cerebral peduncle and the upper pons; we cut the tentorial incisura and open the Meckel’s cave to target the middle pons; we removed the petrous apex to go down to the lower pons. This combination offers an immediate proximity to the whole ventrolateral pontine region. Use the neuromonitoring to determine the safe entry point. We tried to preserve the vein running on the surface of the pons. The tumor was removed carefully with the tumor forceps, suction tubes, and bipolar forceps. Debulk the tumor with CUSA. During tumor removal, the MEP and SSEP were constantly monitored. Because the size of the pontine incision was small, we had to remove the tumor from different angles of surgical trajectories. And this trans-Meckel’s cave transtentorial exposure in combination with Dolenc and Kawase approaches sufficed for our need. With the assistance of subcortical mapping, we confined our resection in the tumor, not violating the corticospinal tracts. There was no distinct boundary between the diffuse pontine tumor and the brainstem, so constant subcortical mapping was mandatory. The pathological diagnosis was an anaplastic astrocytoma. This approach offered a wide exposure and a panoramic view to determine the safe entry point to the pons. And it is effective for ventrolateral pontine lesion.0:32 Clinical vignette0:40 Brain MR0:53 DTI of corticospinal tracts0:59 Diagnosis and approach1:13 3D simulation of the surgical corridor1:49 Patient positioning2:01 Operation9:31 Immediate postop MRI9:51 Pathology report9:56 Postop neurological status10:00 Conclusion10:09 References


A 5 month old girl presented to the Paediatric Emergency Department with a rapidly growing neck mass over 24 hours. She was systemically well and otherwise asymptomatic. On examination she had a mobile, soft and non-tender 5 cm x 5 cm neck mass. Blood tests were unremarkable with normal inflammatory markers. Point of Care Ultrasound (POCUS) was done which showed a solid left sided neck mass with increased vascularity but no evidence of collection or abscess. Given the atypical presentation and rapid growth the patient was commenced on empirical antibiotics and was discussed with both tertiary ENT and Oncology teams. An MRI was performed which was indeterminate. Biopsy of the neck mass was positive for Ewing Sarcoma. This is a rare case of Ewing Sarcoma in an infant. POCUS can be used to rule out common pathology and abnormal lymph nodes, aiding in ongoing investigation and management in neck lumps. A 5 month old girl presented to the Paediatric Emergency Department (PED) after her parents noticed a large lump to the left side of her neck, seemingly appearing over the course of one day. The child was systemically well and had attended nursery the previous afternoon where it was not noticed by anyone. She had no fever, breathing difficulties or dysphagia and no restriction of neck movements or torticollis. A recent review of systemic symptoms was unremarkable apart from, on closer questioning, a cough which had been present intermittently over the past 6 weeks. The patient was born at term following an unremarkable pregnancy with normal antenatal scans. On examination a large mass was visible to the left side of her neck measuring approximately 5 cm x 5 cm . On palpation it was firm, not fluctuant and non-tender. There was no surrounding erythema or warmth. Ear, nose and throat examination was normal and she was apyrexial. Baseline blood tests were as follows; WBC 15.8 x 10'9 /L, Platelets 519 x 10'9 /L, Haemoglobin 125 g/L, Adjusted Calcium 2.70 mmol/L, Albumin 46 g/L, CRP 1.8 mg/L, and a normal coagulation screen. Point of Care Ultrasound (POCUS) was performed using the linear, high-frequency 12 Mhz probe by one of the authors as shown in Figures 2 and 3.  In the left neck level VB there was a large solid mass 3.4 cm x 4.1 cm. Rounded in shape with regular borders. Vascularity was increased. There was a maintained central echogenic hilum but also other abnormal increased echogenicity centrally. There was no fluid collection or evidence of abscess formation.Due to the appearances a formal radiology performed ultrasound (RADUS) was sought. This is shown in Figure 4. Differential diagnoses suggested by the radiology team included congenital cavernous haemangioma, vascular malformation, lymphangioma and dermoid cyst. A chest x-ray was performed to look for evidence of tuberculosis of which the child had no known contact. This was reported as normal with no lung lesions. The patient was commenced on IV Co-Amoxiclav and was reviewed by the local ENT team. Given the rapid evolution of the mass within 24 hours in an otherwise well child there was concern that the US report was not consistent with the clinical picture and therefore an MRI neck under sedation was planned. The MRI report stated: a large well-defined soft tissue lesion is seen in left side of neck in levels III and IV/V 5 x 4.1 x 3.6 cm. Showing inhomogeneous low T1 and indeterminate T2 signal with diffuse post contrast enhancement. No significant inflammatory stranding. Nature of lesion - indeterminate - although sinister possibilities (e.g. sarcoma) should be considered. .There was discussion regarding the nature of the mass between the local team and this was further discussed with the tertiary ENT and Oncology teams where it was agreed to transfer the patient for ongoing investigation and management. The mass was biopsied and histology showed Ewing Sarcoma. The patient underwent staging investigations and was planned to start chemotherapy. She she has since undergone Proton Beam therapy and is the youngest person in the United Kingdom to do so. This case demonstrates a very rare cause for a neck mass in an infant. Given the rapid evolution of the mass and age of the patient, use of POCUS was key in helping to identify concerning features associated with this mass and subsequently streamline further investigations and ongoing care. POCUS should be employed in order to answer binary questions which can help with hypothesis testing. In this case it was undertaken in order to differentiate less concerning swellings, such as a single or cluster of enlarged lymph nodes i.e. in lymphadenitis, from something more sinister. It was clear that this enlargement was not in keeping with a lymph node and although the appearances were not familiar to the operator, they were of sufficient concern that timely further investigation was sought. It was concluded that a radiology performed scan would be required urgently in order to stratify the risk associated with this potentially sinister mass. Clinical correlation prompted further action including specialty advice and further imaging.In our PED POCUS is used by those with appropriate training to help differentiate abnormal lymph node enlargement, such as lymphadenitis and abscess formation, from that of simple reactive lymphadenopathy. Normal lymph nodes should be discrete, well-defined, hypoechoic bean or oval shaped structures. They should have distinct borders and a short axis-to-long axis ratio of less than 0.5. The central echogenic hilum should be readily appreciable A lymph node with an abscess, however, loses its architectural integrity often appearing more circular, occasionally with variable degrees of internal echoes and lacking internal vascular flow. An enlarged lymph node with a more rounded appearance, i.e. an increase in diameter in the short axis, should be carefully interrogated. Thickened or irregular walls, loss of the normal echogenic hilum or internal features such as increased echogenicity  should prompt the user to consider a potentially sinister cause such as the algorithm in Figure 7 demonstrates  Simple cysts are mobile, often small and circular with thin walls with an anechoic centre. As they are entirely fluid-filled they demonstrate significant posterior acoustic enhancement. A sternocleidomastoid muscle tumour is another important differential for an infant with a neck lump which can lead to considerable distress for a parent. Fibromatosis coli can present within the early neonatal period as a firm, often large swelling within the territory of the sternocleidomastoid muscle and is entirely benign. It is most often related to birth trauma or instrumental delivery leading to fibrosis within the muscle. On POCUS this appears as fusiform enlargement of the sternocleidomastoid muscle belly where the muscle fibres can be seen running contiguous with the surrounding unaffected muscle fibres. There is no discrete mass observed Hemangiomas are congenital lesions made up of vascular tissue lined by endothelial cells. While often not present at birth they can grow rapidly within the first 3 months of life and become more prominent in size and number. Congenital cavernous haemangiomas are deeper and POCUS may reveal a cutaneous or subcutaneous soft tissue mass with prominent internal vascularity confirmed by enhanced colour flow Lymphangiomas are congenital abnormalities of the lymphatic system that most often affect the head and neck. Their classic appearances on ultrasound are that of multilocular cystic masses containing internal septa of varying thickness with occasional vascularity, though there is considerable heterogeneity Salivary gland pathology, though relatively uncommon in children, is another group of diagnoses to consider when faced with an acute undifferentiated neck swelling. In acute sialadenitis, the inflamed affected gland appears enlarged, hypoechoic and hyperaemic on POCUS. Sialolithiasis, by contrast, demonstrates hyperechoic internal areas with distal acoustic shadowing Ectopic thymus can arise within the midline or lateral aspect of the neck and present as a mass in infancy or during childhood. Its characteristic thymic pattern provides the classical “speckled” appearances owing to the multiple, linear hyperechoic septa and homogenously distributed hyperechoic foci Despite being the second most common primary sarcoma in children, Ewing Sarcoma is rare with only around 30 children in the UK diagnosed each year. There is a higher prevalence in males and adolescents POCUS is being increasingly utilised as part of the evaluation for the infant, child or young person presenting to the PED with an undifferentiated neck mass, as both a tool for aiding decision making and for streamlining patient flow. Studies have demonstrated the benefits it provides in terms of both diagnostic accuracy and an overall shorter length of stay in the PED None.


The ischial hygroma, also known as a perineal nodular induration, is a relatively rare and mostly cycling-specific injury that is often incorrectly diagnosed and managed. Here two cases with divergent managements are described to highlight the spectrum of treatment available to manage this condition. The presentation, assessment and management of two cases of perineal nodular induration are discussed.The management options, namely surgical excision vs conservative management, with saddle pressure mapping highlight that there is no single optimal method and that a multidisciplinary approach should be applied to treat these injuries successfully. Perineal nodular induration should be investigated appropriately to exclude less benign causes of perineal masses. Conservative management and surgical excision can both be successful. Clinicians should be familiar with the assessment and management of this relatively rare but debilitating condition in competitive cyclists. These lesions are known by several other terms, including a third or supernumerary testicle, accessory testicle, perineal hygroma or induration. ,3,4 Perineal nodular induration was first described by Vuong et al. in 1988 who described two cyclists with this condition. The biker's nodule or ischial hygroma is a relatively unique pathology that most commonly affects competitive male cyclists and to a lesser extent, females. There are also records of this condition occurring in female equestrians  In one of this study’s cases, the patient also had symptoms due to the close proximity to the sciatic nerve. Typically, a nodule is palpable on one or both sides of the midline, posterior to the scrotal sac, usually 2–3 cm in size. , 4, 5 This nodule is typically fibroelastic and may adhere to the adjacent connective tissue.  In females, the nodules are usually unilateral and may occur in the typical location as described above or may occur in either or both of the labium majora.As previously mentioned, a typical biker's nodule occurs mainly in male competitive cyclists. Symptoms include pain on pressure, such as when sitting in the saddle. A dull constant ache even when not riding has also been described. ,7,8 Thus mountain bikers are regarded as a particularly high-risk group for developing these lesions. The lesion is believed to be caused by increased friction and vibration between the ischial tuberosities and the hard saddle as a result of the constant rubbing of the superficial perineal fascia against bony structures..The variant which occurs in the labia majorus may show an admixture of adipose tissue, variably cellular hyalinised tissue containing bland, spindle-shaped fibroblasts, blood vessels, and nerve fibres. In some areas, thick cords of fibrous tissue imparting a keloid-like appearance can occur. Other histologic features include plump mesenchymal cells with round or ovoid nuclei and abundant eosinophilic cytoplasm resulting in an epithelioid, plasmacytoid, or ganglion-like appearance, lymphocytic infiltrate around blood vessels, foci of fat necrosis, and collections of elastic fibres Two typical cases of perineal nodular infiltration are described which are managed with differing modalities.A 45-year-old competitive female cyclist presented to her sports physician with a year-long history of a dull ache in the left inner buttock which radiated to the left hamstring. The pain was aggravated by cycling and sitting on her chair at work. She had initially been treated unsuccessfully by a physiotherapist for hamstring tendinosis.No visible abnormality was detected. On palpation, a nodule of approximately 2 × 2 cm was noted adjacent to the left ischial tuberosity. It was mildly tender on palpation and not fixed to the underlying muscle or bone.The patient was initially sent for diagnostic high-resolution ultrasound. This was followed by a MRI study of the pelvis.High-resolution greyscale ultrasound demonstrated a well-circumscribed predominantly hypo-echoic 2 × 1 cm nodule in the subcutaneous tissue just medial to the ischial tuberosity . MagnetiBased on these and clinical findings the diagnosis of an ischial hygroma was made.The sports physician injected a combination of local anaesthetic and corticosteroid into the nodule. There was no subsequent improvement. She was then referred for surgical excision of the lesion.Dissection through the skin and subcutaneous tissue revealed a fibrotic mass approximately 3 cm in size situated medial to the sciatic nerve and superficial to the hamstring origin at the ischial tuberosity. A white focus was noted centrally within the lesion compatible with the injected corticosteroid.Histology revealed a combination of dense hyalinised fibrous tissue, surrounding oedematous fibrous tissue and pseudocystic area compatible with a cyclist nodule . The patA 61-year-old competitive male cyclist presented to these authors’ clinic with a history of insidious onset pain in the left perineal area which occurred during cycling. Shortly prior to seeking medical attention, he noted a tender mass in his perineum.No visible abnormality was detected. On palpation, a nodule of approximately 1 cm in transverse and 4 cm in longitude was noted adjacent to the left ischial tuberosity. It was firm and rubbery in consistency, mildly tender on palpation, and not fixed to the underlying muscle or bone.The patient was referred for diagnostic high-resolution ultrasound.High-resolution greyscale ultrasound demonstrated a well-defined, heterogeneous, predominately cystic mass inferior to the left scrotum. It measured 5.0 × 2.4 × 4.0 cm and contained hypoechogenic material within it, with no increased vascularity.Based on the imaging and clinical findings, it was diagnosed as an ischial hygroma.The patient was reluctant to undergo surgery and specifically requested conservative management.A comprehensive assessment of his cycling biomechanics and saddle contact point was performed.TM (http://www.sciencetosport.com/ergofit) software and kinematic tools. Saddle pressure mapping was performed using Gebiomized® dynamic saddle pressure mapping systems (http://www.gebiomized.de).Pre-fitting and static kinematics were performed using ErgoFiT° using the Holmes10 method) and excessive saddle setback (10.5 cm). This was confirmed on initial saddle pressure mapping by a front: rear pressure balance ratio of 62:38.Salient findings were that underwear was being worn under the cycling-specific garment and that the cycling-specific garments were of relatively low quality, with chronic wear demonstrated on the padding material. The kinematic assessment demonstrated excessive saddle height (static knee flexion angle (KFA) of 22° and saddle setback of 6.8 cm) a series of minor adjustments were made and numerous different saddle types were tested to achieve the most favourable saddle pressure mapping possible  and MRI (magnetic resonance imaging) show lack of contrast uptake, which explains the hypovascular nature as seen on histology examinations.,7 On an MRI, the lesion would be hypointense on all sequences. The primary role of imaging is to determine the location and exact extent of the lesion and to exclude rarer and other important conditions, such as aggressive angiomyxoma. In most clinical scenarios, ultrasound would suffice. Imaging characteristics are rarely reported; however, an example can be found in a 2009 article by Van de Perre et al., 4, 8 Non-mass enhancement on MRI is a reassuring feature and readily excludes malignancy. Vuong et al. states that this pseudoneoplastic lesion has a relatively constant appearance, but if not realised to be a benign nonneoplastic lesion, there is a potential for misdiagnosis.  This again stresses the importance of suspecting this lesion in the correct clinical context and using imaging as an important diagnostic tool.If the diagnosis is unclear, imaging plays an important role in the differentiation from other causes of perineal swelling. These would include an abscess, epidermal cyst, cutaneous adnexal mass, lipoma or tumour (sarcoma or metastases).Secondly, the management of this entity can be successful by using both conservative and more invasive methods. Correct bike setup, saddle type and saddle position will go a long way in relieving symptoms and preventing recurrence of the lesions. Special attention should also be given to cycling shorts and the construction of the padding. Local infiltration with corticosteroids or hyaluronidase are described as first-line invasive treatment. If none of these therapies are efficacious, surgery should be considered. –5, 8In these authors’ experience, most clinicians limit management to avoidance of the causative factor, be it cycling or horse riding. This is sometimes not a practical option and, in these authors’ limited experience, it is generally not sufficient as therapy. Conservative management includes a period of rest from cycling to allow the area to settle which should reduce the size of the lesion but not eliminate it. Once the lesion has settled a comprehensive assessment of kinematics and saddle contact point can reduce loading of the area and prevent recurrent episodes of pain and enlargement. However, this requires dedicated expertise in the field of cycling biomechanics and specialised equipment to assess the saddle/cyclist interface. Clinicians should enquire about such expertise in their area and refer to the appropriate experts for management.In these authors’ experience, an injection of the lesion with corticosteroids has not resulted in lasting relief but can provide a temporary treatment option or adjunct to more specific management of the kinematics and contact area analysis. Clinicians should be aware of the risk of fat necrosis at the injection site which may cause further discomfort on the commencement of cycling.,4 However, the lesion is not well vascularised.  The margins of the lesion are not well circumscribed and there is no synovial lining. ,6,11 Focally, spindle cells showed mild degenerative atypia reminiscent of that seen in ischaemic fasciitis.Failure to resolve symptoms using conservative measures or a lack of expertise in this field in the clinician’s geographical area should warrant a referral for surgical excision. Surgical excision of large hygromas can be technically challenging due to the poorly defined boundary of the lesion and adherence to surrounding structures. As such, the patient may often experience a residual nodule, albeit smaller in size, and experience some persistent discomfort in the area post-surgery. The histopathological features show a myxoid degeneration of the fatty tissue, often with a pseudocyst formation. This is surrounded by dense hyalinised fibrous tissue which merges towards the periphery with oedematous fibrous tissue containing fibroblast-like spindle cells, thick collagen bundles, degenerate elastic fibres, and clusters of capillaries which may show diapedesis of erythrocytes or even haemorrhagic foci. In summary, clinicians should have a high index of suspicion for perineal nodular induration in cyclists presenting with perineal pain or pain in adjacent areas. They should be aware of the potential for more serious pathology and use imaging to differentiate these less benign lesions. Lastly, management principles can be dictated by the availability of expertise in cycling kinematics in their area but multiple management approaches can result in successful outcomes for the patient.


Mesenteric cysts are rare, usually benign, tumors that typically present asymptomatically and are found incidentally during evaluation for nonspecific abdominal symptoms. We present the case of a 41-year-old African American female who was found to have a mesenteric cyst in her jejunum during the evaluation of abdominal pain, nausea, and constipation that she had been experiencing for six weeks. Pre-operatively, an abdominal CT scan showed a 6x4x6 cm mesenteric cystic lesion in the right mid-abdomen, which was then successfully resected off the mesentery of the jejunum laparoscopically. Her postoperative course was uneventful and she was discharged home without complication a few hours after her procedure with appropriate follow-up.  Mesenteric cysts are rare, benign, intra-abdominal tumors with an incidence of one case per 250,000 hospital admissions. They are typically asymptomatic but can have variable presentations depending on location and morphology. Therefore, they are typically found incidentally during abdominal imaging or laparotomy when investigating non-specific abdominal symptoms . Cysts cA 41-year-old African American female presented to the emergency room with abdominal pain, nausea, and constipation for six weeks. Her abdominal pain was constant without precipitating or alleviating factors. The patient reported that her abdominal pain was moderate and originated at the right lower quadrant, and it started progressing and shooting up to her right shoulder in the past few days. The patient also reported that she was having constipation in the past few weeks, and she has tried ingesting charcoal to alleviate her symptoms. According to the patient, charcoal was helpful with her constipation and nausea, but the abdominal pain was unbearable. The patient denied any blood in her bowel movement, fevers, and sick contacts. The patient had a cesarean section in 2007, a laparoscopic gastric band procedure in 2010, an abdominoplasty procedure in 2018 for cosmetic purposes, and a preauricular cyst removal procedure in 2021. Due to leakage from the gastric band, she was scheduled at our outpatient surgery clinic for a laparoscopic gastric band revision procedure in three weeks. Beyond her past surgical history, the patient’s medical history was unremarkable. She was not taking any type of home medications. Based on the patient's clinical presentation and surgical history, an imaging study was suggested at the emergency department and the computerized tomography (CT) scan indicated a 6x4x6 cm mesenteric cystic lesion in the right mid abdomen  was then made with a scalpel. Subcutaneous tissue was divided with electrocautery, and the mass was discovered, excised, and sent to pathology. There was no gross involvement of the fascia. The wound was washed out. Hemostasis was achieved. Wounds were closed in layers anatomically. The patient tolerated the procedure well and was awoken and taken to the recovery room in stable condition. The patient was discharged from our hospital a few hours after the procedure without any postoperative complications. A mesenteric cyst is one of the rarest abdominal tumors. The incidence of this tumor is around one in 100,000-250,000 . MesenteThe origin of mesenteric cysts is still uncertain . Most ofThe diagnosis of a mesenteric cyst is usually done by imaging techniques such as CT scans, ultrasound, and MRI. For example, the CT scan will show a cystic lesion, and depending on the content of the cyst it can show anywhere from hypodense to dense lesions on the scan. Due to the rarity of this lesion, a mesenteric cyst can be misdiagnosed on imaging depending on its location. Common misdiagnoses include pancreatic pseudocysts and ovarian cysts. A case report by Theodoridis et al. describes a case where the initial diagnosis was presumed to be a para-ovarian cyst on ultrasound . It was Treatment for the cyst differs based on the symptoms. For an asymptomatic cyst, no treatment needs to be done. For a cyst that’s causing symptoms, the treatments include anywhere from drainage, aspiration, or excision. The definitive treatment is excision of the cyst via laparoscopic surgery as other methods have shown higher rates of recurrence . LaparosIn our case, the patient presented with abdominal pain in right lower quadrant, constipation, and nausea. In abdominal CT, a cystic lesion with fluid was identified in the patient. During the laparoscopic procedure, the mesenteric cyst in our patient was identified as wrapped around the jejunum. It was a multiloculated cyst and covered approximately 8 cm of the jejunum and around 70% of the circumference. We initially thought about resecting that part of the jejunum. However, the cyst came off fairly easily and was resected and sent to pathology Figure . The patBefore deciding on the optimal treatment plan for a patient presenting with a mesenteric cyst, a thorough evaluation should be conducted based on the patient's clinical presentation, imaging/laboratory studies, past medical history, and past surgical history to reach a provisional diagnosis. In our case, the patient’s mesenteric cyst was discovered in the jejunum, and the multilocular cyst contained chylous fluid, which was confirmed with postoperative pathology. For patients with clinical symptoms such as abdominal pain, nausea, and constipation, the definitive treatment is complete surgical removal to avoid recurrence and possible malignant transformation. Also, total cystectomy could be considered as a potential treatment in certain cases, such as our patient, to prevent potential secondary complications such as volvulus, spillage of infective fluid, herniation of bowel into an abdominal defect, or obstruction.


Surgery is an effective way for treating SFT, but it is often difficult to resect completely due to a large size, with a high recurrence rate and mortality rate after operation. Additionally, SFT is relatively resistant to chemotherapy, and there is a lack of effective systemic drug treatment. These lead to certain difficulties in the treatment of SFT. We report a case of a rare MSFT in the pelvic cavity. With a history of recurrence after two surgeries, this patient underwent surgical removal combined with  This approach can increase the radiation dose to the tumor target, and dramatically decrease the dose to surrounding normal tissues, showing great advantages of safety, minimal invasion, definite benefit, few complications, low level of radioactive contamination, and convenience for protection. In recent years, it has been used for treating solid tumors at various sites and has achieved good performance  is a rare spindle cell tumor originating from CD34-positive dendritic mesenchymal cells . It has formance –13. We r125I seed implantation was performed, in which 30 seeds (0.3 mCi) were implanted into the tumor bed near the right iliac vessels, with D90 of 62.0 Gy, and 32 seeds (0.5 mCi) into the tumor bed of the left pelvic cavity, with D90 of 58.0 Gy; the operation was smooth, and postoperative pathology came back as a recurrent low-grade MSFT. In August 2015, a residual tumor was observed on CT in the posterior wall of the bladder. On August 27, 2015, under the guidance of CT, 50 125I seeds (0.3 mCi) were placed percutaneously into the tumor in the left posterior wall of the bladder, with D90 of 64.8 Gy measured post implant. On September 21, 2015, under CT guidance, 28 125I seeds (0.3 mCi) were implanted into the tumor in the right posterior wall of the bladder, with D90 of 49.7 Gy measured post implant. Dose-volume histogram parameters were applied for the evaluation of target volume and organs at risk (OARs).The patient’s regular examinations after implants revealed a complete remission . On April 8, 2014, she underwent pelvic mass resection; postoperative pathology showed a borderline or low-grade malignant spindle cell tumor, consistent with SFT, and the immunohistochemistry results were as follows: Ki-67 (5% positive), Bcl-2 (-), Desmin (-), SMA (-), DOG-1 (-), CD68 (-), CD117 (-), S-100 (-), CD34 (blood vessels +), CD99 (+), and Calretinin (-). Two months later, her CT displayed a walnut-size mass each under the abdominal wall and in the vaginal stump, with no specific treatment. On June 29, 2015, the patient visited our hospital and had abdominal and pelvic CT examination, which indicated multiple space-occupying lesions in the abdominal wall and pelvic cavity (125I seeds (0.4 mCi) into the wall tumor, with D90 of 77.8 Gy. Her regular examinations thereafter demonstrated a complete remission all the way until October 2018, when CT displayed recurrent tumors in the abdominal wall and pelvic cavity. On October 16, 2018, with the help of CT guidance, 54 125I seeds (0.5 mCi) were embedded into the SFT (5.0×3.0×5.0 cm) under the right side of the bladder, with D90 of 97.6 Gy; on October 23, 2018, 30 125I seeds (0.5 mCi) were embedded into the SFT (3.6×1.5×3.0 cm) in the abdominal wall, with D90 of 101.3 Gy. The patient achieved a complete remission, no complications were found, and the brachytherapy was well tolerated by the patient, The patient had no recurrence of abdominal and pelvic tumors during the 4-month follow-up (In January 2016, CT showed that a tumor recurred in the abdominal wall, with a size of about 4.0×2.5×4.5 cm. On January 6, 2016, she underwent implantation of 29 The diagnosis of SFT mainly depends on the pathologic features and immunophenotypes. Histologically the tumor was composed of abundant and dense cells, which were separated by rope like collagenous fibrous stroma. The tumor cells were spindle to short-spindle shaped with marked nuclear atypia and increased mitotic activity. The tumor cells arranged in fascicular or swirl pattern. In focal areas, hemangiopericytoma-like structure, coagulative necrosis, focal hemorrhage and invasive margin were presented. MSFT is usually grossly indistinguishable from conventional forms, but it may also show a more irregular cut surface, with evidences of necrotic areas or infiltration of the nearest tissues . Immunoh125I seed implantation in MSFT, providing a novel solution to the treatment of the disease.Generally, the most effective treatment for SFT is surgical resection. However, in most cases, when patients feel discomfort and visit doctors, tumors have grown to large masses — beginning to press on adjacent organs to produce the warning symptoms — and there are usually abundant blood vessels and collateral circulation around the tumors, often making surgery difficult. Relevant literature has shown that patients with benign SFT undergoing surgical removal have a median 10-year overall survival rate of 54%-89% –23, and 125I seed implantation for the tumors in the left pelvic cavity and near the right iliac vessels showed a surprising benefit: the patient attained a complete remission, and no relapse was observed as of the end of follow-up, with a PFS of 43 months (125I seed implantation alone for the tumor behind the bladder continued to work: a complete remission was achieved, with a PFS of 35 months (et al. described that the median PFS with doxorubicin alone and combined with ifosfamide was 4.0 months and 6.7 months, respectively (et al. evaluated the efficacy of temozolomide combined with bevacizumab in 14 patients with advanced SFT, and the median PFS was 10.8 months (125I seeds than to chemotherapy, which often causes severe side effects. Moreover, seed implantation costs less than chemotherapy and targeted drug therapy. However, seed implantation is a local therapy. If there is widespread metastasis, a combination of seed implantation and chemotherapy or targeted drug therapy may be a possible solution.This patient was initially diagnosed with SFT in 2014 and then underwent tumor removal, but relapsed just two months later. In July 2015, surgical resection alone was performed on the tumor in the abdominal wall, and the tumor recurred in the abdominal wall six months later. While surgery alone failed to improve the outcome, surgical removal combined with ectively . Park et8 months . Compare125I seed implantation, which supports seed implantation as a potential solution to repeated local recurrence. As MSFT is very rare, there has been no more research than case reports, not to mention clinical randomized controlled trials. Our report could provide a reference for the treatment of MSFT.In this report, the patient experienced repeated recurrence after surgery, but benefited from The original contributions presented in the study are included in the article/supplementary material. Further inquiries can be directed to the corresponding author.ZG performed the bibliographic search and wrote the manuscript; ZG, HY, XD and JW revised the manuscript; YL, JZ and ZL took part to the equipment preparation and follow-up; HZ made the decision to submit the article for publication. All authors read and approved the final manuscript.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


In this paper, we present our own clinical-laboratory experience concerning three less obvious presentations of pemphigus vulgaris (PV) and discuss the pertinent literature. The involvement of the sacral dimple reported here for the first time, as well as the nipple and the eyes, could initially be misleading clinically. These less stereotypical localizations may occur due to the transition of different epithelia, each with varying levels of cadherin  and thus altered sensitivity to mechanical stress. The role of dermatologists who have experience in treating autoimmune blistering dermatoses is fundamental for identifying promptly the initial and exacerbating PV lesions in such unusual locations. Pemphigus is a group of rare, chronic, potentially life-threatening autoimmune bullous diseases of the skin and/or mucous membranes . PemphigThe skin lesions in PV typically can be found on the scalp, face, trunk, axilla, groin, and pressure points . Oral anThe initial manifestation of PV at a clinically non-typical body site is a diagnostic challenge for a dermatologist. In some cases, awareness that PV can infrequently affect locations around natural body orifices should help to establish a PV diagnosis. For patients already diagnosed with PV, skin lesions in an “unusual” body site always require a detailed differential diagnosis.In this paper, we present three PV patients showing sacral dimple, ocular, and nipple involvement and discuss the pertinent literature concerning these natural body orifices in pemphigus.A 48-year-old woman  with a 2Sacral dimples  are the most common cutaneous anomaly found during neonatal spinal examinations . They arAn expression of altered antigens linked with embryonic structures such as lining of the spinal cord  that would stimulate IgG autoimmunity against DSG isoforms is a plausible explanation for the presence of pemphigus lesions located in the area of the sacral dimple. Moderate expression of radially arranged DSG was present around the central canal of the spinal cord throughout all studied developmental stages . MechaniSkin lesions around the sacral dimple are very rare but always require detailed diagnostics to rule out pilonidal cysts, psoriasis, eczema, and hidden malignancy. Such lesions should be diagnosed subject to careful clinical examination and if necessary histopathological evaluation. In some cases, a direct immunofluorescence (DIF) examination is a crucial diagnostic procedure. For pemphigus, a diagnosis may be facilitated by presence of Nikolsky’s sign and a dermoscopy examination.A 73-year-old man with a three-month history of an apparent PV exacerbation involving mainly conjunctiva was seen in our outpatient clinic. An ophthalmological examination revealed ectropion of the lower eyelid and inflammatory changes in the eyelid conjunctiva. Minor erosions of the cornea of the right and left eyes were found. During this bilateral ocular exacerbation , taking Expression of the majority of desmosomal molecules in human conjunctiva is similar to the expression of DSGs in the middle of the epidermis . DSG 3 iIncubation of human specimens with PV-IgG for 12 h caused blistering in the suprabasal layers of the conjunctiva and a reduction of DSG 1 and DSG 3 protein levels . Only a The eyes are less frequently involved than oral mucosa , and ocuRecalcitrant conjunctivitis with conjunctival blisters should warrant a workup for PV . There iA 63-year-old woman was seen in our outpatient clinic because of a two-month history of widespread painful mucocutaneous lesions involving the right breast around the nipple with impetiginization, the lower abdomen, the area around navel, eyelids, and buccal mucosae. A DIF of perilesional skin showed pemphigus IgG (+), IgG1 (+), IgG4 (++), and C3 (+) deposits. A multi-analyte ELISA revealed elevated levels of IgG serum anti-DSG 1 (7.91) and anti-DSG 3 (1.73) antibodies (cut-off for both: 1.0). Both these findings confirmed a PV diagnosis . A H + EThe nipple is covered by pigmented squamous epithelium. Moreover, keratin-producing squamous cells of the epidermis extend into major ducts for 1–2 mm, and there is an abrupt transition from squamous cells to normal luminal/myoepithelial lining of ducts. The epithelium of exosecreting ducts extends to directly reach the skin of the nipple . DSG 3 sIn the literature, we found a description of PV and breast cancer coexisting in the area of the nipple. A 54-year-old woman had a 3-month history of a rash localized on her right breast and nipple. Six months before this presentation, she was diagnosed with PV. The skin overlying her right breast was covered with multiple confluent erosions, hyperkeratotic scales, and crusts. A DIF of the perilesional skin showed IgG and C3 pemphigus deposits, whereas an ELISA revealed high levels of IgG antibodies to DSG 1 and 3. A computed tomography scan and a subsequent breast biopsy confirmed the presence of an invasive triple negative ductal carcinoma . Garcia-For a differential diagnosis of PV located on the nipple, it is necessary to take into account other diseases. Nipple hyperkeratosis was associated with malignancies such as cutaneous lymphoma and malignant acanthosis nigricans . Nipple In this paper, we present three less obvious presentations of PV. The triggering factors of PV still remain unclear. These multifactorial triggers involve interactions between environmental influences and genetic predispositions . The strWe speculate that the transition between various kinds of epithelia lining natural body orifices may be accompanied by peculiarities of expression patterns of isoforms of both DSG and desmocollin, and therefore, it may facilitate the location of PV lesions in such sites of privilege. Conceivably, the skin immune system may act more vigorously at external–internal interfaces combating noxious environmental factors compared to other cutaneous areas, which may initiate the development of an autoimmune response characterizing PV in genetically prone individuals.Four DSG isoforms (DSG 1–4) have been identified in humans. These isoforms are differentially expressed in various epithelial tissues. DSG 2 and DSG 3 are mainly distributed throughout the lower layers of the epidermis, whereas DSG 1 is expressed at higher levels in the upper layers, with low to undetectable levels in the basal layers. DSG 3 has a strong basal distribution associated with proliferating cells . DSG 4 iMechanical stress could have triggered blisters in our patients at clinically non-typical body sites. Applying tangential pressure by a thumb to normal skin in a patient with pemphigus results in a force that dislodges the upper layers of the epidermis from the lower layers (Nikolsky’s sign). An additional explanation for such localization of skin lesions in pemphigus, apart from variable DSG expression, is mechanical trauma. This mechanism is feasible for our PV patients exhibiting lesions localized in the area of the nipple, the sacral dimple, and the conjunctivae. Most commonly ocular presentations of PV are unilateral. This may be due to pathergy in which in which the patient rubs one eye and induces lesions formations . Trauma-Finally, we suspect that the stubborn inflamed PV lesions around natural body orifices described here are the result of focal autoimmune response maintained by diffuse ectopic lymphoid-like structures (ELSs) , which mBlisters and their evolutionary lesions can be present in unobvious anatomic areas that are not always inspected during routine clinical examinations. Therefore, the role of the dermatologist is fundamental in taking into account PV, and in the evaluation of the real disease extension. Stubborn lesions around natural body orifices might be a valuable clinical hint for carrying out immunopathological testing to unequivocally diagnose incipient or exacerbated PV.


Echocardiography plays an important role in the diagnosis of heart disease. Sometimes, however, it may also provide misdiagnosis information.We report a rare case of a misdiagnosis of primary mitral valvular tumor with severe regurgitation by preoperative echocardiography. During the surgery, the true lesion was found to be mitral valve leaflet prolapse due to degenerative mitral valve disease.For individual patient, the best clinical decision not only needs the extensive application of echocardiography, but also needs the combination of clinical symptoms and more auxiliary examination.The online version contains supplementary material available at 10.1186/s12872-023-03131-z. Primary valvular tumor and valve degeneration are generally easy to distinguish. But in some special cases, they have a certain similarity in echocardiography. Especially for patients with atypical symptoms, it is often misdiagnosed one from another. Here, we report a case of mitral valve degenerative disease suspected to be diagnosed as primary valvular tumor.A 68-year-old female patient presented to the emergency department of our hospital with recurrent chest tightness and shortness of breath. On admission physical examination, systolic murmurs could be heard in the apex of the heart. The patient had no other comorbidities except hypertension. Transthoracic echocardiography revealed severe mitral regurgitation and mitral leaflet cystic nodules, suspected to be benign tumor or infective endocarditis with vegetations. Transesophageal echocardiography revealed multiple cystic structures with significant separation in the A1, C1, and P1 leaflets by the mitral Carpentier nomenclature [Echocardiography has developed rapidly since its appearance in the 1950s and is now the preferred choice for imaging occupying cardiac lesions. It provides high-quality, real-time images, which are invaluable in the evaluation of cardiac masses. Although transthoracic echocardiography is an excellent initial diagnostic technique for the evaluation and diagnosis of cardiac masses, transesophageal echocardiography provides superior image resolution and better visualization of cardiac masses in patients with poorly studied transthoracic echocardiography . Some diPrimary cardiac tumors are fairly uncommon, with an average incidence rate about 0.02%, while primary valvular tumors account for only 10% among them. The types that need differential diagnosis include myxoma, papillary elastic fibroma, lipoma, blood cysts, etc. –6. The fGenerally, only patients with severe mitral regurgitation combined with symptoms of cardiac insufficiency require surgery. This patient had severe mitral regurgitation before operation and the wrong judgment before operation did not cause any adverse effect. However, for suspected valve masses without valve regurgitation, more accurate evaluation and identification of primary valvular tumors and valve degenerative lesions is clearly necessary. For individual patient, it may require a combination clinical symptom, widen use of echocardiography and more auxiliary examinations such as three-dimensional echocardiography or a contrast study to make the optimal clinical decision.Additional file 1. Transesophageal echocardiography video-1.Additional file 2. Transesophageal echocardiography video-2.Additional file 3. Transesophageal echocardiography video-3.


Paragangliomas (PGLs) are frequently reported around the abdominal aorta; however, are extremely rare near the urachus.A 78‐year‐old woman was referred to the urology department of our hospital for further examination and treatment of a 1.2‐cm tumor in the lower abdominal wall, a tumor excision was then performed. On immunohistochemical staining, the tumor and supporting cells were positive for chromogranin A and the S 100 protein, respectively, and were diagnosed as PGL. The PGL was thought to be derived from chromaffin cells that migrated to the wall of the urachus during embryonic life and remained even after the wall regressed.We report a case of PGL near the urachus that can be explained by the distribution of the sympathetic network around the midline of the lower abdominal wall during embryonic development. Therefore, PGL should be considered in the differential diagnosis of periurachal tumors. CTComputed tomogrphyPGLParagangliomasSDHBSuccinate Dehydrogenase BWe report a case of PGL that arose around the urachus and was thought to be derived from the chromaffin cells that migrated to the wall of the urachus during embryonic development and remained there even after the involution of the urachus.Paragangliomas (PGLs) are rare neuroendocrine tumors that produce catecholamines. They are frequently reported around the abdominal aorta.A 78‐year‐old woman was referred to our hospital for treatment of duodenal papillary cancer. Fluorodeoxyglucose positron emission tomography‐computed tomography (FDG‐PET/CT) showed a 1.2‐cm tumor in the midline of the lower abdominal wall  scintigraphy, and no abnormal findings were observed.Postoperatively, she underwent 123 I MIBG scintigraphy and examinations to check for catecholamines in the blood and urine. The scintigraphy report revealed no significant integration, and the catecholamine test result was within the normal range. She was offered a genetic test but did not take it. Because the GAPP score was low, the follow‐up was requested to the endocrine medicine and the catecholamine inspection is periodically enforced. The imaging examination was periodically carried out in the recurrence follow‐up of the duodenal papilla cancer. There was no apparent recurrence 2 years after the operation.Chromaffin cells migrate from the neural crest at 5 weeks of gestation and contribute to the formation of the adrenal medulla.In fetal life, the muscular layer continues from the bladder wall to the allantoic duct; however, the muscular layer of the allantoic duct disappears by 15–20 weeks of gestation.Another possible hypothesis is that aberrant chromaffin cells accidentally originated from the peripheral nerve cells in the abdominal wall. However, chromaffin cells are said to arise from Schwann cell precursors that change to immature Schwann cells during embryonic development.The present case provides an implication of chromaffin cell distribution during development. Clinically, this case suggests that PGL should be considered as a differential diagnosis for tumors around the urachus.Masafumi Tsuruta: Conceptualization; data curation; writing – original draft. Takayuki Goto: Conceptualization; writing – review and editing. Jin Kono: Writing – review and editing. Yuki Kita: Writing – review and editing. Kimihiko Masui: Writing – review and editing. Takeshi Sano: Writing – review and editing. Masakazu Fujimoto: Data curation; writing – review and editing. Atsuro Sawada: Writing – review and editing. Shusuke Akamatsu: Writing – review and editing. Takashi Kobayashi: Conceptualization; supervision; writing – review and editing.The authors declare no conflict of interest.All procedures performed in this study involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.The protocol for this research project has been approved by our institutional reviewer board Approval No. R1531.Not applicable.Not applicable.


Rectal neuroendocrine neoplasms can induce liver metastasis. However, few reports exist on the associated long-term recurrence rates. We report a case of liver metastasis identified 15 years after rectal neuroendocrine neoplasm resection.A 50-year-old woman was on semi-annual follow-up after undergoing mastectomy for breast cancer (pT1N0M0) and low anterior resection for grade 1 rectal neuroendocrine neoplasm . Fifteen years postoperatively, a 7-mm hyperechoic mass was identified at liver segment 6. Magnetic resonance imaging revealed a slight growth of the mass. Positron emission tomography/computed tomography revealed radiotracer accumulation in the lesion. Laparoscopic hepatectomy was performed. The histopathological diagnosis was grade 2 neuroendocrine neoplasm. The pathological findings and clinical course indicated the tumor originated in the rectum.Our findings highlight the need to reassess the optimal postoperative follow-up period for patients with rectal neuroendocrine neoplasm. Liver metastasis from rectal neuroendocrine neoplasms (NENs) can occur even after curative resection. The recommended postoperative follow-up duration depends on the presence of risk factors for recurrence . Herein,A 50-year-old woman underwent left total mastectomy for breast cancer (pT1N0M0 by AJCC-8th) and low anterior resection for a localized NEN. A 10-mm mass was observed at the lower rectum. Submucosal and lymphovascular invasion  with negative surgical margins were noted. The mitotic count was < 1 per 10 high-power fields. Immunohistochemical staining revealed the cells were positive for synaptophysin, chromogranin A, and CD56 Fig. . ContrasTherefore, surgical resection was planned. Ascites, dissemination, and distant metastasis were not observed. Intraoperative ultrasonography revealed a single mass located at segment 6. No other hepatic lesion was identified. Laparoscopic partial hepatectomy was performed. The operation took 98 min. She was discharged on postoperative day 5 after uneventful postoperative course.Gross examination revealed an 11.0 × 10.0 × 10.0-mm mass. Histological examination revealed atypical cells with round nuclei arranged in a rosette-like pattern Fig. . The mitNENs originate from the diffuse system of neuroendocrine cells. The most common primary site of NENs is the gastrointestinal tract (67.5%) . The smaThis case can inspire discussion on the origin of hepatic NENs: primary or metastatic. Primary hepatic NENs are extremely rare 0.4%) , and the% , and tWe searched the English literature for reports of liver metastasis identified after a recurrence-free period of ≥ 5 years after rectal NEN resection  Table 11. The EuOur case has two remarkable features: (1) the extremely long observation period after surgical resection and (2) the implication that postoperative follow-up should be performed for a considerably long period in patients with risk factors for recurrence.


Isolated iliac artery aneurysms are rare and difficult to diagnose. It is more common in males. It can be asymptomatic at diagnosis or can present with frank rupture, or symptoms caused by compression on nearby organs.A 44 years old male was diagnosed with rectosigmoid adenocarcinoma and underwent low anterior resection. One year after the surgery, he presented with fistula between the rectal stump and left iliac artery that was managed by stenting.A fistula between the rectal stump and the left iliac artery is very rare. There are several treatment options for ilio-rectal fistula but no conclusive specific treatment. One of the major causes of morbidity and mortality in the elderly is lower gastrointestinal bleeding (LGIB) with the diverticular disease being the most common cause in this population . The morHerein we report a case of a patient with a fistula between the rectal stump and left iliac artery.A 44 years old male with no significant past medical or surgical history. Presented with a history of left lower abdominal pain, vomiting, flatulence and constipation for 2 months.On examination, his abdomen was soft with left lower abdominal tenderness.CBC and X-ray were unremarkable. An occult blood test was done and came out to be positive. Colonoscopy was performed and showed a lesion originating at 30 cm from the anal verge reaching the rectosigmoid junction. Sigmoid biopsy was obtained and was consistent with moderately differentiated invasive colonic adenocarcinoma.A computerized tomography (CT) scan abdomen obtained with IV, oral and rectal contrasts revealed a diffuse and annular wall thickening of the sigmoid/descending colon in the left iliac fossa, this affected area was measuring up to about 68 mm in length and up to 10 mm in wall thickness, associated with diffuse adjacent fat stranding and tiny lymph nodes, narrowing its lumen without complete obstruction with adjacent 16 mm soft tissue density. Metastatic workup was negative.The patient was referred to surgery and low anterior resection was done.The pathology report was consistent with moderately differentiated invasive colonic adenocarcinoma of the rectosigmoid. The tumor size was 3.5 cm in greatest dimension and was invading through the muscularis propria into pericolic fat. All margins were free of cancer. There were intramural and extramural lymphovascular as well as perineural invasion. 1 out of 16 lymph nodes was positive. The pathologic stage was pT3N1M0.The patient was started on adjuvant Capecitabine plus Oxaliplatin.Upon further investigations for follow-up, positron emission tomography (PET) and CT scans showed metastasis to the lung and abdominal wall. He was referred for chemotherapy followed by radiotherapy to the lung lesions. The patient was then completed stereotactic radiosurgery (SRS) to the lung lesions. Post SRS, he was started on Avastin® (bevacizumab) every 3 weeks.One year after the surgery, the patient presented to the emergency department (ER) unconscious. Upon thorough history, he was complaining of vomiting, diarrhea, lower left quadrant abdominal pain, and low-grade fever, 10 days before presenting to the ER. He was intubated and admitted to the intensive care unit (ICU) as a case of septic shock.Head CT scan was unremarkable. CT showed diffusely thickened colon with pericolonic stranding, associated with the possibility of localized collection and abnormal air foci suggestive of localized infection adjacent to the anastomotic site with possible involvement of the small bowel.After stabilization, the patient was taken for surgery. During laparotomy, there were extensive adhesions, a small bowel loop adhered to the site of the previous anastomosis. A moderate amount of free fluid was found. Features were in keeping with perforation with a contained leak. The small bowel loop was released from the old anastomosis, resection and anastomosis were done along with a diseased colon that was resected with adequate margin. End colostomy was created. He was then discharged in good condition.The pathology report showed mucosal ulceration with thrombosed blood vessels and inflammatory cells infiltration.2 weeks after the surgery, the patient presented to ER with massive bleeding per rectum, a portable ultrasound was done in the ER revealed that the rectosigmoid is distended with echogenic material that suggested blood. There were multiple pockets of free fluid in the abdomen, the largest is seen on the left measuring about 53 × 26 mm. Sigmoidoscopy was done, examining up to 16 cm from the anus, there were huge clots of blood with no visible source of bleeding. He was resuscitated with blood transfusion. An interventional radiologist was consulted. Selective crossover of left external iliac artery (EIA) small pseudoaneurysm and intimal irregularity with narrowing 2 cm segment in the proximal portion. That was covered with a stent. Post ballooning angiogram showed clear patency of the left EIA with a smooth outline of the wall, distal left lower limb angiogram showed mild spasm in the left superficial femoral artery (SFA). 4 days later he was discharged in good condition.Unfortunately, the patient passed away after developing cerebellum brain metastasis. Which he underwent craniotomy for cerebellum mass resection that was positive for metastatic deposits of adenocarcinoma.Aneurysms of the iliac artery can be classified into those related to abdominal aortic aneurysms, observed in 10–20% of cases and isolated aneurysms of the iliac artery . IsolateCauses recognized to be associated with iliac artery aneurysms include atherosclerosis, mycotic infections, intraoperative or trauma injuries, collagen vascular diseases, and connective tissue diseases such as Marfan syndrome and Ehlers-Danlos syndrome. Pseudoaneurysms are caused by erosions resulting from malignancies , 8.Formation of fistula can be noticed as a complication of low anterior resection (LAR), as blood vessels thinning and staple line tension with erosion that resulted from surgical manipulation . In the The natural history of these aneurysms is unclear however, the increase in the size of an aneurysm is associated with an increased risk of rupture. At diagnosis, the average size of an isolated internal iliac artery aneurysm (IIIAA) was 7.7 cm with a 33% incidence of rupture, although the overall mean size of ruptured aneurysms was 5.6 cm, as reported by Richardson and Greenfield. In another study by Brin and Busuttil, a rupture was seen in 38% with a 58% mortality rate although they could not find a direct relationship between the size of the aneurysm and the risk of rupture , 10.IIIAA can be asymptomatic at diagnosis and can present with frank rupture, or symptoms elicited by compression on adjacent organs, the ureter anteriorly, the internal iliac vein and the lumbosacral trunk posteriorly, the external iliac vein and obturator nerve laterally, and the colon and small bowel medially . A previAn aneurysmal rupture is greatly indicated in case of iliac artery aneurysm with unstable vitals. Iliac artery aneurysms can be palpated as a pulsatile mass during digital rectal or vaginal examinations and they are more beneficial than abdominal examinations . When ruThe treatment of iliac aneurysms has considerably changed over recent years. The management of choice now in patients with an anatomically suitable and uncomplicated IIAA is endovascular treatment. Proximal ligation with obliterative endoaneurysmorrhaphy is one of the surgical managements in case of rupture or fistula as ligation alone is associated with an increased risk of recurrence . PreviouIn conclusion, a fistula between the rectal stump and the left iliac artery is very rare. Diagnosis is difficult and no specific investigation is available. Patients with massive LGIB with failure of an endoscope may need to consider imaging to rule out a fistula. There are several treatment options for ilio-rectal fistula but no conclusive specific treatment, it depends on the patient’s situation and physician’s experience. Endovascular or embolization is a minimally invasive surgery and is suitable for patients with high operative risk. It is a simple emergency and effective procedure that saves a patient’s life and can be performed by a general surgeon Figs.  and 2.Fi