Paratesticular liposarcoma is a sporadic urological tumor. We report the clinical presentation, treatment course, and prognosis of 2 cases of primary paratesticular liposarcoma with different pathological types, with the aim of further understanding the diagnosis and treatment of this rare disease. Case 1 was a 53-years-old male patient who presented with left scrotal enlargement with swelling 3 years ago and was considered to have a testicular malignancy on preoperative CT scan and enhanced MRI. The patient underwent resection of the left scrotal mass and left orchiectomy under general anaesthesia. Histopathological study confirmed the diagnosis of dedifferentiated liposarcoma. At the 4-months follow-up, no local recurrence or distant metastasis was observed. Case 2 is a 42-years-old male patient with a left scrotal mass which was discovered six months ago. Preoperatively, he underwent CT plain and enhanced MRI examinations suggesting an intra-scrotal occupancy. The patient underwent scrotoscopic excision of the left scrotal mass under general anesthesia. Histopathological studies confirmed the diagnosis of highly differentiated liposarcoma. At the 10-months follow-up, no local recurrence or distant metastasis was observed. Preoperative differential diagnosis of paratesticular liposarcoma should be noted with testicular tumor and extra-abdominal hernia. Extensive local excision and, if necessary, concomitant radical testicular resection is the treatment of choice. If the tumor in the scrotum spreads to the inguinal region, surgical removal with the aid of a scrotoscope may be attempted. This procedure avoids the formation of a large incision in the inguinal region compared to traditional open surgery. Patients commonly experience local recurrence and less distant metastases after surgery, so long-term follow-up is recommended. Paratesticular liposarcoma (PLS) accounts for approximately 7% of the scrotal tumor. And it is a sporadic urologic tumor. Only about 265 cases have been reported worldwide . The 202A 53-years-old male patient was admitted to the hospital with “an enlarged left scrotum with a feeling of swelling for more than 3 years”. The patient was previously healthy, had no smoking history, and did not consume alcohol. There was no history of disease or surgery. There was no specific family history, including cancer.The left side of the scrotum was enlarged, hard, without tenderness, with poorly defined borders, measuring about 15 cm×10 cm, and the mass extended to the left inguinal region. The left testis and epididymis could not be palpated. The right testis and epididymis were well developed. The right testicle and epididymis were developed. The transillumination test was negative.  (MDM2 gene amplification (MDM2/CSP12 ratio:8.58)   showed that concomitant organ resection was not associated with a survival benefit in RP WDLPS . IKOMA NWe performed the first scrotoscopic scrotal mass excision for PLS in case 2. We found preoperatively that the mass in case 2 had grown along the spermatic cord to the inguinal region. A conventional open procedure would have created an incision approximately 10 cm long in the inguinal region. Moreover, the patient was young and had a strong desire for minimally invasive surgery, so we chose to use the scrotoscopic approach for minimally invasive surgical treatment. After making a small incision in the scrotum, we created a pneumoperitoneum by piercing the trocar needle so that the scrotal tract fitted closely to the trocar needle, thus ensuring a gas-tight seal to keep the CO2 inside the scrotum, and we did not find any CO2 severe leakage during the operation. In case of air leakage, we recommend reinforcing the incision with sutures. The external ring of the inguinal canal was not opened during the operation, which did not lead to gas entering the abdominal cavity through the opening of the external ring of the inguinal canal and forming a peritoneal pneuperitoneum. In the past, scrotoscopy was performed by injecting saline into the scrotum to provide visualization, which was prone to postoperative scrotal edema, but our use of CO2 to dilate the scrotum compensated for this defect .There is still clinical controversy regarding the role of adjuvant radiotherapy or chemotherapy for PLS. In a study that included 265 cases of PLS, adjuvant radiation therapy was found to have no statistical impact on recurrence-free survival . AlthougIn conclusion, PLS is a rare tumor, which should be differentiated from testicular tumor and external abdominal hernia before surgery. Local wide excision and, if necessary, concurrent radical orchiectomy are the preferred treatments. If the tumor in the scrotum spreads to the groin area, surgical resection with the aid of a scrotoscope can be tried. This procedure avoids the formation of a large incision in the inguinal region compared to traditional open surgery. We will collect more cases later to explore this procedure’s indications and verify its superiority. Margin-positive cases should be re-excised to improve recurrence-free survival. Patients have a higher risk of local recurrence after surgery but less distant metastasis, so long-term follow-up is recommended. Chemotherapy and targeted drug therapy can be tried for patients with existing metastases or inoperable.The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.JL composed the manuscripts and literature review. JW and HH provided figures and pathology review. HY and LT had the acquisition, analysis or interpretation of clinical data for the work, revising it critically for important intellectual content and agreement to be accountable for all aspects of the work. All authors contributed to the article and approved the submitted version.The authors would like to express their gratitude to the pathologist Hongying Zhao of Beijing Chaoyang Hospital, Capital Medical University, who provided the pathological figures.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.


Gut malrotation may result in failure of descent of the cecum to the right iliac fossa, resulting in the anomaly where the cecum and appendix are situated in the subhepatic/gallbladder region. Although the true incidence of subhepatic cecum or appendix is not known, there is a handful of case reports in the literature describing the diagnosis and management of subhepatic appendicitis and associated challenges. Some case reports describe subhepatic appendicitis, where the cecum is in a normal position, but the subhepatic tip of the long appendix gets perforated or inflamed, resulting in the process being in the subhepatic region. We report a patient with subhepatic appendicitis, who had multiple episodes of abdominal pain for almost one year, treated with antibiotics, but was never diagnosed properly. The case was diagnosed by abdominal sonography and was managed successfully in our institution. Subhepatic appendicitis is a very uncommon entity, accounting for only 0.08 % of all cases of acute appendicitis according to a study of 7210 patients by Palanivelu et al. in 2007 [Acute appendicitis is one of the commonest causes of acute abdomen in children and young adults and there are countless studies in the literature on the subject. Although diagnosis and management of acute appendicitis are often straightforward, not infrequently surgeons face dilemmas in both diagnosis and management, due to variations in the anatomic position of the appendix . SubhepaDiagnosis of subhepatic appendicitis is usually made by contrast-enhanced computed tomography (CECT) of the abdomen, or upon diagnostic laparoscopy. Abdominal sonography has poor sensitivity for diagnosing unusually located appendicitis, especially in retrocecal cases . We presA 25-year-old male patient presented to the emergency department (ED) of our district general hospital with a one-day history of right-sided abdominal pain, fever, nausea, anorexia, and vomiting. The patient admitted to having repeated similar attacks for the last nine months, which were managed by antibiotics with an improvement of the condition, only to recur after some time. He did not have abdominal sonography or computed tomography done for recurrent abdominal pain prior to presentation to our hospital. The patient had no surgery in the past and was not suffering from any chronic illness. On examination, he was febrile with a core body temperature of 38,2 ° C, but his remaining vital signs were within normal range. Abdominal examination revealed right-sided abdominal tenderness, which was more pronounced over the right upper quadrant with muscle guarding and positive rebound tenderness. His laboratory blood work showed an elevated leukocyte count of 15.0x10^9/L, with significant left shift, neutrophilia being 89%. Renal and liver function tests were normal. Radiographs of the chest and abdomen were unremarkable, but abdominal sonography showed evidence of appendicitis, with the liver and the biliary tree being normal. The sonographer reported that the inflamed appendix appears to be just under the gallbladder. Since laparoscopic set-up was not available in the hospital, open surgery was the only option for the management of our patient. The patient was taken to the operating room (OR) after antibiotic and thromboembolism prophylaxis, the abdomen was entered through the right pararectal incision in order to have good access to the subhepatic region. Upon exploration, we found the inflamed appendix and cecum in the subhepatic position , followed by the pelvic (21%) . Some ra4%, folloAfter the first report of acute subhepatic appendicitis by King in 1955 , many siOnce the diagnosis of subhepatic appendicitis has been made, the optimal management approach must be contemplated, since there is no standard approach or internationally accepted consensus, considering the rare nature of the condition. It appears that the laparoscopic approach is preferred by most surgeons, which is subject to the availability of skilled surgeons and laparoscopic set-up, especially in the developing world ,13. AlthSubhepatic appendicitis is a rare condition, which poses a significant challenge to clinicians in terms of timely diagnosis and optimal management. The great majority of the cases of subhepatic appendicitis either get perforation with abscess, peritonitis, sepsis, and increased morbidity, or suffer from repeated attacks of abdominal pain before getting an accurate diagnosis. We recommend using abdominal sonography initially, which can be helpful in a good number of cases, but maintaining a lower threshold for CRCT for equivocal cases. Diagnostic laparoscopy, coupled with laparoscopic appendectomy, is a very useful tool in expert hands for both diagnosis and safe management of this rare and challenging condition.


Juxtaglomerular cell tumors (JGCTs) are rare, typically benign neoplasms; only rare cases are clinically or histologically malignant. We herein report the histologic, immunophenotypic, and molecular features of a clinically unsuspected, diagnostically challenging case of malignant JGCT in a 23-year-old man. The diagnosis is confirmed with electron microscopy. The case is notable for its marked mitotic activity, which has not been previously reported in JGCTs, and novel finding of GATA3 immunohistochemical positivity. We herein report the histologic, immunophenotypic, and molecular features of a clinically unsuspected, diagnostically challenging case of malignant JGCT.22 identified. No necrosis or vascular invasion was identified. A panel of immunohistochemical stains showed the neoplasm was diffusely positive for CD34 . Other than a mildly low potassium (3.4 mmol/L), a basic metabolic panel was unremarkable. Abdominal ultrasound and computed-tomography were performed and identified a 4.8 cm solid-appearing, well-marginated mass in the right kidney A. The pafor CD34 A and GATfor CD34 B, with wA next generation sequencing (NGS) sarcoma fusion panel  showed no gene fusions. In addition, targeted NGS analysis  showed no tier I/II ACMG/AMP DNA mutational variants, gene fusions, or copy number alterations. However, electron microscopy revealed rhomboid, electron-dense renin protogranules in neoplastic cells C and a d3JGCTs, or reninomas, arise from the modified smooth muscle cells in the juxtaglomerular apparatus. JGCTs present predominantly in young adults, with a peak incidence in the second and third decades of life, and show a slight female predominance.,The aforementioned gross, microscopic, and immunohistochemical features are fairly typical of previously reported JGCT. Though not present here, variable muscle marker expression is often noted.NOTCH fusions characteristically seen in glomus tumors. The absence of common fusions and mutations suggests a unique pathogenesis from glomus tumor (and other pericytomatous tumors).Interestingly, JGCTs show some morphologic and immunohistochemical overlap with glomus tumors, and given the presumed origin from the juxtaglomerular apparatus, it is appealing to postulate a relationship with tumors that show “true” pericytic differentiation. Strong, diffuse expression of CD34 may favor JGCTs over glomus tumor, however, the specificity is questionable due to the rarity of renal glomus tumors.At least three malignant JCGTs have been published with common reported clinical features including increased size (greater than 8 cm in largest dimension) and advanced age at time of diagnosis. Common reported histologic features included lymphovascular invasion, marked cytologic atypia, and a mildly increased mitotic rate. On clinical follow-up, two cases developed distant metastasis and one case showed local recurrence after one year.4In conclusion, we report a rare case of malignant JCGT with the novel finding of GATA3 positivity, which may help resolve differential diagnostic considerations. Despite application of available NGS panels, a driver mutation remains elusive.This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.DG, FA, IJ, GQG, RS, SB contributed with investigation, formal analysis, resources, review, and editing. DG and FA also contributed with original draft writing.The authors have no conflicts of interest to disclose.


The aims of this study were to analyze the clinical characteristics, auxiliary examinations, and treatment measures of small intestinal lymphangioma and to improve the clinical diagnostic ability of clinicians.This paper reports two cases of small intestinal lymphangioma in the Department of Gastroenterology, the First Affiliated Hospital of Soochow University, and makes a comprehensive analysis.A 31-year-old woman went to the hospital with complaints of dizziness, fatigue, and anemia. A 52-year-old woman complained of upper abdominal pain and went to the hospital with abdominal pain awaiting investigation. Both patients were subjected to three major routine examinations, tumor complete set, CT, capsule endoscopy, and deep enteroscopy, and both of them underwent complete resection of the affected intestinal segment. Pathology showed that both patients had small intestinal lymphangioma.The clinical manifestations of small intestinal lymphangioma lack specificity. Capsule endoscopy and deep enteroscopy are helpful for clinical diagnosis, and pathological examination is still the gold standard. Surgical treatment can achieve better results. Small intestinal lymphangioma is a rare disease of the digestive tract. It often presents as chronic gastrointestinal bleeding due to mucosal compression, bleeding, necrosis, or ulceration caused by enlarging the tumor. It accounts for 6% of small intestinal tumors in children and 1.4%–2.4% in adults  of 38 g/L, and esophagogastroduodenoscopy showed chronic superficial gastritis. Reexamination after blood replenishment treatment still showed severe anemia, and the symptoms of dizziness and fatigue did not improve significantly. Therefore, the patient was in the hospital due to “anemia of unknown origin”. During hospitalization, the patient did not have nausea, vomiting, abdominal pain, abdominal distension, black stool, and bloody stool. Physical examination showed anemic appearance, soft abdomen, no tenderness, rebound pain, no obvious mass, and bowel sounds six times per minute. Admission diagnosis was as follows: anemia of unknown origin. Blood routine showed HGB 45 g/L; urine routine revealed 94 RBC/μl; stool occult blood test was positive; tumor full set showed erythropoietin >762.00 mIU/ml. Contrast-enhanced CT of chest, abdomen, and pelvis revealed a few fibrosis foci in the left lower lung, small hepatic cysts, and slightly reduced density of heart cavity, consistent with anemia. Capsule endoscopy showed that the mucosa at the proximal end of the jejunum is polypoid, with congestion, edema, erosion, white moss attachment, and active bleeding, as shown in Patient B, a 52-year-old woman, went to the hospital on 3 November 2020 because of “recurrent upper abdominal pain for more than 20 days”. The patient had abdominal pain more than 20 days ago. The pain was mainly in the middle and upper abdomen, which was like a paroxysmal knife cutting and relieved after several seconds. There was no radiating pain in the waist and back, no abdominal distension, and no nausea and vomiting. Physical examination showed tenderness in the middle and right upper abdomen without rebound pain and muscle tension. The abdomen is flat without gastrointestinal type or peristalsis wave, there was no obvious mass, and the bowel sounds are normal. Admission diagnosis revealed abdominal pain of unknown origin. There were no abnormalities in blood routine, CRP, biochemistry, AFP, CA19-9, and CEA. Abdominal enhanced CT showed local intestinal wall edema, structural disorder, and mesenteric edema, as shown in Lymphangioma is a benign tumor caused by congenital malformation of the lymphatic system . It usuaThrough literature review, we found that there are no characteristic clinical manifestations in the early stage of small intestinal lymphangioma. With the enlargement of the tumor, it could cause various clinical symptoms. The common symptoms were abdominal pain caused by intestinal irritation, followed by gastrointestinal bleeding and anemia, intestinal obstruction, and hypoproteinemia. Some cases presented with non-specific gastrointestinal symptoms, such as nausea, vomiting, abdominal distension, diarrhea, and anorexia –9. In thSmall intestinal lymphangioma should be distinguished from intestinal endometriosis. Endometriosis refers to the growth of endometrial tissue outside the uterine cavity and myometrium. When ectopic endometrium invades the intestine, it is intestinal endometriosis. The disease has no specific clinical manifestations. When it invades the rectum and sigmoid colon, a series of intestinal symptoms such as abdominal pain, diarrhea, constipation, hematochezia, and even intestinal obstruction can occur. It is difficult to distinguish the two diseases based on clinical manifestations, and thus, it could easily result in misdiagnosis. The common diagnostic methods of this disease include ultrasound, CT, colonoscopy, and laparoscopy. Ultrasonography mainly showed thickening of the affected intestinal wall and hypoechoic changes , 16. ColIn this paper, two patients went to the hospital with anemia and abdominal pain as the main symptoms. CT examination of patient A showed no abnormality, while patient B showed intestinal wall edema and disorder; thus, a diagnosis could not be made. Capsule endoscopy and deep enteroscopy also failed to diagnose small intestinal lymphangioma. As patient B showed symptoms of IBD, other intestinal manifestations may cover up lymphangioma-related symptoms, leading to a missed diagnosis. The diagnosis is clear by pathological results, which further proved that pathological examination is still the gold standard for the diagnosis of small intestinal lymphangioma. According to the size and shape of the proliferative lymphatic vessels in the tumor, lymphangioma can be classified into three types: capillary lymphangioma, cavernous lymphangioma, and cystic lymphangioma . SometimIn conclusion, small intestinal lymphangioma is a rare tumor of the small intestine with a lack of clinical specificity. Imaging examination is of certain significance, and capsule endoscopy and deep enteroscopy are helpful for clinical diagnosis. The pathological results can make a definite diagnosis of small intestinal lymphangioma. Small intestinal lymphangioma is a benign tumor, but malignant transformation may exist. Surgical treatment is the first choice, with the advantages of quick recovery and good prognosis.The original contributions presented in the study are included in the article/Supplementary Material. Further inquiries can be directed to the corresponding author.The studies involving human participants were reviewed and approved by Ethics Committee of the First Affiliated Hospital of Soochow University. The patients/participants provided their written informed consent to participate in this study. Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.FL contributed to conception and design of the study. LW wrote the first draft of the manuscript. HF and BC wrote sections of the manuscript. All authors contributed to manuscript revision, read, and approved the submitted version.


Carotid body paragangliomas (CBPs) clinically present as highly vascularized cervical masses with a pathognomonic localization at the carotid artery bifurcation. Following ultrasonography and MRI/CT imaging, surgical resection with optional preoperative embolization is considered as the treatment of choice in most cases. We herein present the case of a 60-year-old female with characteristic clinical signs and imaging findings of a right-sided CBP who finally went to surgical treatment. Intraoperatively, the tumor showed an adherent growth to the hypoglossal nerve that had to be partially resected, resulting in a postoperative nerve palsy. Histological examination of the resected tumor revealed the unexpected diagnosis of a hypoglossal nerve schwannoma. To the best of our knowledge, we herein present the third case reported in the literature of a unilateral hypoglossal schwannoma located at the carotid bifurcation mimicking clinical symptoms, imaging and intraoperative findings of a CBP. SDH, VHL, and TMEM127 are the most frequently altered genes and can thus be characterized as tumor drivers in this entity. Depending on the tumor size, compression of adjacent structures, and potential catecholamine production, clinical symptoms can comprise a pulsatile cervical mass, palsy of cranial nerves IX/X/XII and difficult-to-control hypertension [Carotid body paragangliomas (CBPs), also known as chemodectomas or carotid glomus tumors, represent rare neuroendocrine tumors and account for about 0.5% of all head and neck neoplasms . CBPs orrtension . Imagingrtension . DependiTreatment options include surgical resection with optional preoperative embolization, conservative imaging follow-up, and radiotherapy ,8. The cA 60-year-old Caucasian female presented at a tertiary care medical center with a three-month history of intermittent visual disturbance, bilateral weakness of arm elevation, and chronic headache. Past medical history included hypertension treated with Ramipril, cervical spondyloarthrosis, and latex allergy. After initially consulting her family doctor, an MRI scan of the head and neck was performed, showing a 2.6 × 2.1 cm measuring round-shaped tumor at the level of the right carotid bifurcation with an irregular but strong contrast enhancement as an incidental finding see a. The tuClinical examination of the patient showed no signs of cranial nerve palsy, laboratory tests including catecholamine blood levels, urinary metanephrines, and vanillylmandelic acid showed no pathological findings. Ultrasonography of the neck revealed a 2.6 × 2.1 × 1.3 cm tumor at the bifurcation of the common carotid artery with splaying but not compressing the branching internal and external carotid artery see . The tumThe clinical diagnosis of a right-sided carotid paraganglioma was made, and treatment options were discussed with the patient including surgery, radiotherapy, and periodic imaging follow-up. Based on the comparably small size of the tumor that was classified as Shamblin Type I being associated with a relatively low risk for surgical complications tumor resection with preoperative embolization was planned. Twenty-four hours before surgery, the patient was transferred to the radiology department for digital subtraction angiography and embolization. Herein, only small-sized feeding vessels originating from the internal carotid artery were found that were not accessible for selective catheterization and subsequent embolization see b. NonethHistopathological examination of the resected tissue showed an in toto resected tumor surrounded by a fibrous capsule and being composed of vascular structures, regressive fibrous tissue, and predominantly spindle-shaped cells. Immunohistochemical staining showed positive reactions of these lesional cells for neuron-specific enolase (NSE), CD56, S100, vimentin, Sox10, and collagen type IV with negativity for P14, P63, chromogranin A, synaptophysin, glial fibrillary acidic protein (GFAP), desmin, melan A, HMB45, CK20, and AE1/3 see .NF2, SMARCB, and LZTR1 gene mutations with no suspicious findings.Based on histological architecture and immunohistochemical analysis, the diagnosis of a schwannoma was made, which was confirmed by an external pathology laboratory consulted for a second opinion. In a follow-up examination four weeks after surgery, the patient presented with an inconspicuous cervical scar and improved hypoglossal nerve function under ongoing physical and speech therapy. To exclude a schwannomatosis as a pre-disposition to develop further schwannomas, the patient was tested for To the best of our knowledge, we present herein only the third case reported in the literature of a hypoglossal schwannoma located at the carotid bifurcation mimicking clinical symptoms, imaging and intraoperative findings of a CBP.Schwannomas are benign peripheral nerve sheath tumors and predominately affect the vestibular nerve in about 90% of cases . Of the Surgical treatment of carotid body tumor bears a relevant risk of cranial nerve injury that occurs in about 25% of patients, with hypoglossal nerve being the most common nerve injured . The samTaken together, this singular and illustrative case shows that hypoglossal schwannoma should remain a consideration in the evaluation of tumors located at the carotid bifurcation. As this report demonstrates, clinical and radiological as well as intraoperative findings can closely mimic that of carotid body paragangliomas and should therefore be considered as a differential diagnosis.


Retroperitoneal vascular leiomyosarcoma (RVLMS) are rare soft-tissue sarcomas that most commonly arise from large blood vessels and have a poor prognosis. We present the case of a 61-year-old woman who presented with isolated left flank pain. Abdominal computed tomography and magnetic resonance imaging revealed a 5 cm retroperitoneal soft-tissue mass that was contiguous with the left ovarian vein and connected to the proximal ureter, causing hydronephrosis. As ureteroscopy suggested extrinsic compression of the ureter, a percutaneous biopsy of the mass was obtained, whereupon diagnosis of leiomyosarcoma was made. Radical en-bloc excision of the tumor, including the involved upper urinary tract and the gonadal vein, was performed. The tumor proved to be a leiomyosarcoma arising from the ovarian vein wall. No adjuvant therapy was planned, and no recurrence was noticed at her 24-month follow-up. Primary RVLMS of the ovarian vein is an uncommon condition. To date, only a few sporadic cases have been reported in the literature. What makes the present case further interesting is the unusual tumor’s relationship with the patient’s ureter, raising both diagnostic and management challenges. To our best knowledge, this is so far the fourth reported case of its kind to cause ureteral obstruction. Leiomyosarcoma (LMS) is a rare malignant mesenchymal tumor of smooth muscle origin. Among adults 55 and older, it is the second most common subtype of soft-tissue sarcoma to affect the retroperitoneum 28%) , 2. Most8% , 2. MA 61-year-old woman with an unremarkable past medical history presented with intermittent and isolated left flank pain of six months duration. Her physical examination was normal. Abdominal ultrasonography showed left hydronephrosis. Routine blood tests, urinalysis, culture, and cytology were normal.Contrast-enhanced computed tomography (CECT) of the abdomen revealed a 5 cm retroperitoneal mass, projecting at the left L3 level and causing obstruction of the proximal ureter. No tissue plan was noticed between the mass and the psoas muscle Figure . Tumor mOn magnetic resonance imaging (MRI), the mass was completely separated from the psoas muscle by a fat plane, and the left ovarian vein was seen to be running through the mass. However, no abnormal macroscopic fat was detected in the lesion Figure . ConsequExploratory laparotomy revealed a retroperitoneal firm mass located about 1.5 cm beneath the ureteropelvic junction, completely wrapping approximately 4 cm of the proximal ureter and tightly adherent to the ovarian vein. Based on these intraoperative findings, radical en bloc excision of the tumor, including the involved upper urinary tract and the gonadal vein, was performed ; it typically appears as a large macroscopic-fat free soft-tissue mass that is separate from retroperitoneal organs and is contiguous with the vascular structure, often with areas of necrosis, hemorrhage, or cystic changes , 2. In tOwing to its various pulse sequences and multiplanar capacity , MRI wasThe diagnosis of LMS in our patient, despite a high index of radiologic suspicion, had required a pretreatment biopsy, which had proved to be safe, feasible, and effective using a retroperitoneal route and immunohistochemical staining. Indeed, patients with no history of malignancy and normal biomarkers should have their macroscopic fat-free retroperitoneal mass sampled on a utility-based approach whenever histological findings may influence management .Approximately 60% of RVLMS present an extravascular growth pattern , 2. The However, the origin of retroperitoneal LMSs may have no clinical relevance in such clinical scenarios as they all have similar treatments. Nonetheless, it is important to distinguish a non-vascular soft-tissue LMS  from an LMS of vascular origin, as the latter tends to metastasize through hematogenous spread, leading to a worse prognosis . None ofThere are currently no formal guidelines on the management of RVLMS. Nevertheless, complete excision of the tumor offers the best chance of cure; neither neoadjuvant nor adjuvant chemo- and /or radiotherapy have proved to be effective at improving local control and survival ,2, 5. HoTumor size, depth of invasion, differentiation, mitosis count, and microscopic necrosis have been described as prognostic factors of vascular LMS . BesidesIn the setting of an indeterminate retroperitoneal mass, RVLMS should be considered if the lesion is contiguous with a vessel. Multisequence and multiplanar MRI may be of great help in predicting the histologic subtype and the vascular origin of such neoplasms. However, the ovarian vein is a rare and unusual location of the LMS, and cases of gonadal vein LMS causing obstructive uropathy are even rarest. Hence, pretreatment ureteroscopy and biopsy should be obtained. Conservative surgery can be used in selected cases. However, radical en-bloc excision of the tumor and the ipsilateral upper urinary tract is the standard treatment option in our patient with ureteral invasion.


The diagnosis of pulmonary leiomyosarcoma using bronchoscopy is difficult, and surgical resection is often performed for definitive diagnosis and curative therapy. We report a case of pulmonary leiomyosarcoma, successfully diagnosed using repeated transbronchial cryobiopsy (TBCB). A 69‐year‐old‐woman was found to have an oval mass in the left hilar region extending into the left main bronchus on computed tomography (CT). All transbronchial biopsy specimens were necrotic, but repeated TBCB removed the necrotic tissue from the tumour and finally led to the diagnosis of pulmonary leiomyosarcoma. Proton therapy was administered, which caused shrinkage of the tumour. Thus, TBCB is useful for definitive diagnosis of leiomyosarcoma without surgical biopsy. Repeated TBCB can reduce tumour volume, eliminate atelectasis, and reduce the extent of radiotherapy exposure. Pulmonary leiomyosarcoma is a rare tumour. Diagnosis by bronchoscopy is difficult because of significant necrotic tissue in the tumour and laborious exfoliation of the cells. We report a case of pulmonary leiomyosarcoma, successfully diagnosed using repeated transbronchial cryobiopsy (TBCB). Pulmonary leiomyosarcoma is a rare tumour that accounts for 0.5% of all lung malignancies.A 69‐year old woman presented with a 2‐week history of cough and dysponea. Computed tomography (CT) revealed a 35 × 32 × 39 mm oval mass in the left hilar region, extending into the left main bronchus Figure . We perfWe report a case of pulmonary leiomyosarcoma that was successfully diagnosed using repeated TBCB. To the best of our knowledge, this is the first report on the usefulness of TBCB for the definitive diagnosis of pulmonary leiomyosarcoma. Previously, several cases have reported that a definitive diagnosis cannot be made using TBB because of necrotic tissue. Many specimens are required to obtain viable tissue, and a considerable mechanical force is required to collect specimens from a firm tumour characterized by smooth muscle tissue. Brushing and washing tumour cells are of little value since they are difficult to exfoliate.We believe that a non‐invasive definitive diagnosis can promote non‐surgical treatment options. Surgery is the first‐line treatment for pulmonary leiomyosarcomas. Previous studies have reported prolonged overall survival in surgical cases compared with nonsurgical cases.In addition, repeated TBCB can reduce the tumour volume, thus resolving atelectasis and reducing the area requiring irradiation. Previously, several cases of atelectasis due to the extension of pulmonary leiomyosarcomas into the bronchus were reported.We report the first case of pulmonary leiomyosarcoma that was successfully diagnosed using repeated TBCB. Repeated TBCB may also be useful in diagnosing pulmonary malignancy with significant necrosis regardless of pulmonary leiomyosarcoma. However, further accumulation of similar cases is expected to substantiate TBCB as a powerful diagnostic technique.Shinya Tsukamoto drafted the manuscript. Takamasa Kitajima, Satoshi Marumo and Motonari Fukui revised the manuscript. All authors approved the final manuscript.None declared.The authors declare that appropriate written informed consent was obtained for the publication of this manuscript and accompanying images.


Spindle cell lipoma (SCL) is an uncommon benign tumor. A 67-year-old male with multiple comorbidities, presented with a complain of swelling in the neck, which was excised, and the diagnosis of spindle shape lipoma was made based on pathological examination. •Spindle cell lipoma (SCL) is a rare tumor that arises from subcutaneous tissue.•The definitive diagnosis requires focused gross examination of it varies morphological features is essential, in addition to the Cytological examination. SCL frequently manifest in middle-aged male patients . The dia2A 67-year-old comorbid male, presented to the clinic complaining of swelling in the left side of the neck, the swelling was round, soft and mobile and growing slowly in the past two years. He denied any history of pain, discharge, fever, and constitutional symptoms. For past medical history he is known to have diabetes mellitus type 2, Hypertension and dyslipidemia on oral antihypertensives, statins, and oral hypoglycemic agents, Patient also has history of Ischemic heart disease, status post percutaneous coronary intervention, and he's on aspirin since then.He denies any family history of related malignancies. On physical examination: there was a palpable soft swelling at posterior aspect of the neck measuring 3 cm in diameter with intact neck and shoulders range of movement. Laboratory investigations including complete blood count, Renal and Hepatic profiles were done and were unremarkable. Furthermore, the lesion was investigated initially with an Ultrasound which demonstrated a 3.6 × 1 cm in size, subcutaneous soft tissue mass . DiffereThe option of surgical excision was offered, and an informed consent was obtained The dissection was made around the lesion till it was completely excised including its capsule. The specimen of homogenous yellow cut surface. The specimen was sent for pathological review. Histopathological findings were mature adipocytes collagen fibers and spindle cells, which was consistent of SCL and negative for malignancy .Fig. 2ShThe patient was followed up in the clinic 2 weeks post operatively with unremarkable physical examination with the wound being completely healed with no swelling nor recurrence.3Adipose tissue lesions can vary from a benign lipoma to a more serious malignant lesions including liposarcomas .Other subtypes of lipoma may consist of lipomatosis, lipoblastoma, angiolipoma, and myolipoma .SCL is a benign histopathological variant of lipoma . It is mMoreover, clinical and radiological workoup does not always aid in the diagnosis of SCL. A focused gross examination of it varies morphological features is essential, in addition to the Cytological exam which consists of mature adipocytes and spindle cells with myxoid matrix. In cytogenetic basis it's has been linked to CD34 positivity and often associated with lost 12q ± 16q in karyotyping . On the Management of SCL involved a simple excision of the lesion . A Chen,4Spindle cell lipoma is a benign tumor that arises from adipose tissues. It can be diagnosed with microscopic evaluation and managed with excision with good prognosis.Not commissioned, externally peer reviewed.Not applicable.No funding.Dr. Rema AlRashed: writing the paper, Dr. Abdulllah albudah: review, Dr. Feras sanna: Primary consultant & surgeon final modification.IT’S A CASE REPORT, NOT A RESEARCH.Name of the registry:Unique Identifying number or registration ID:Hyperlink to your specific registration (must be publicly accessible and will be checked).Not applicable.No conflict of interest.


The coexistence of calcium pyrophosphate dihydrate crystal deposition (CPP) and synovial chondromatosis (SC) in the temporomandibular joint (TMJ) is rarely reported. CPP disease (CPPD) is complex arthritis synonymous with excessive pyrophosphate production and variable aberrations in mineral and organic phase metabolism of the joint cartilage, leading to local inundated CPP and crystal deposition of partially deciphered predispositions. Meanwhile, SC is a rare benign synovial joint proliferative disease of unclear etiology and has a low risk of malignant transformation. However, SC manifests severe joint disability and dysfunction because of connective tissue metaplasia of the synovial membrane, which forms cartilaginous nodules with or without calcifications or ossifications. These nodules often detach and form intra-articular loose bodies and very rarely within extraarticular spaces.We report the case of a 61-year-old man to expand the body of literature on these unusual coexisting arthropathies of the TMJ. The patient presented to our hospital in 2020 with complaints of pain in the right TMJ and trismus for over 6 months. Radiographic assessments of the TMJ provided a preoperative provisional diagnosis of SC. However, the histopathology of the open biopsy revealed tumor-like lesions comprising several deposits of rhomboid and rod-shaped crystals that displayed positive birefringence in polarized light, confirming a coexistence of CPPD. A second-stage operation was performed for the complete removal of the loose bodies and chalk-like lesions including synovectomy. No evidence of recurrence was recorded after a follow-up of nearly 1.5 years.Isolated CPPD and SC of the TMJ are prevalent in the literature however, monoarticular coexistence of these diseases is rare, due to the lack of consistency in the diagnostic criteria in clinical practice. Moreover, optimal treatment depends on several considerations. This report delineated the molecular etiopathology and underscored the need for continued deciphering of the causal mechanisms of coexisting CPPD and SC of the TMJ. In addition, the importance of confirmatory testing for accurate diagnosis, and appropriate management of these diseases were discussed. ANKH) [ANKH activity. The increased activity leads to the accumulation of PPi in the cartilage, which in combination with calcium results in CPP crystal formation [ANKH mutations also contribute, at least in part, to the pathophysiology of the non-genetic CPPD type [β and IL-18, which eventually cause joint inflammation [First identified as pseudogout by McCarty et al. in 1962 , calciumANKH) , 16. Theormation . FurtherCPPD of the TMJ poses diagnostic challenges; a likely justification for its rarity in the literature –21. AlthFibronectin 1 (FN1)–Activin receptor 2A (ACVR2A) gene fusions, which were first identified in SC and reported by Totoki et al. [KMT2A)–BCL6 corepressor (BCOR) gene fusion in a rare case of tenosynovial chondromatosis [Synovial chondromatosis (SC) is a rare benign joint neoplasm that is associated with the metaplastic proliferation of cartilaginous nodules within the synovial membrane that commonly manifests as intraarticular loose bodies. Although very rare, with a prevalence of approximately 1 per 100,000, loose bodies can form within extraarticular spaces, resulting in a condition referred to as tenosynovial chondromatosis –27. SC ai et al.  and latei et al. . The automatosis .The clinical presentation of SC that involves three cardinal signs and symptoms was previously described . The sigSimultaneous occurrence of CPPD and SC is rarely reported in the TMJ , 41, 42.The TMJ disorders were assessed following validated diagnostic criteria for temporomandibular disorders (DC/TMD) protocol .A 61-year-old Japanese man was referred to our oral and maxillofacial surgery department in October 2020, with a chief complaint of persistent preauricular pain in the right cheek and restricted mouth opening for over 6 months. The patient was referred by a primary physician a month earlier for further investigations and surgical management when the frequency and intensity of pain increased and no longer responded to pharmacological treatment; mainly nonsteroidal anti-inflammatory drugs (NSAIDs). The patient had a history of clenching and grinding of teeth and medical history of controlled hypertension and type 2 diabetes mellitus (DM) by his family physician for many years. Hypertension was controlled at daily self-automated blood pressure (BP) measurements of systolic BP less than 130 mmHg and diastolic BP less than 80 mmHg, on oral amlodipine besylate 10 mg once daily. DM was controlled at hemoglobin A1c measurement values of 40–51 mmol/mol (5.8–6.8%), by oral administration of 5 mg linagliptin once a day after taking breakfast. The patient’s previous medical records also indicated that renal and hepatic function measurements were within normal limits, with nonremarkable history of any familial predisposition and surgical or traumatic injuries.On examination, the patient was well-built, stood 178 cm, weighed 78 kg, and was in good general condition with no: pallor, edema, cyanosis, jaundice, cervical, or generalized lymphadenopathy. Systemic examinations were within normal limits. However, local examination revealed facial asymmetrical with mild swelling at the right preauricular region and limited mouth opening  of 15 mm, accompanied by replicated arthralgia involving masticatory muscles  at the visual analog scale of 8–10 in the right TMJ region Fig. . There wRadiography involved panoramic examination, contrast-enhanced computed tomography (CT), and magnetic resonance imaging (MRI) of the bilateral TMJ using protocols and recommendations highlighted in the recent literature . Other iConsidering the patient’s cardinal clinical features and radiological findings, the differential diagnosis of a tumor or tumor-like diseases such as pigmented villonodular synovitis (PVNS), CS, and a possibility of malignant chondrosarcoma of the right TMJ were made. Under general anesthesia, an open biopsy was performed to rule out malignancy and obtain a definitive diagnosis. Suffice it to say that, preparations for excisional biopsy amid overt features of malignancy were made. Briefly, surgery was approached through a right-sided preauricular incision, the shallow temporal artery rly identified and dissected, and the superior articular space was opened, which revealed a mass covered with a fibrous capsule. The mass had numerous white, cartilage-like hard tissues and chalk-like soft tissues of mixed sizes Fig. A, B. TheConsidering all these findings, a pathological diagnosis of coexisting SC and CPPD in the right TMJ was made. In early January 2021, second-stage surgery was performed under general anesthesia to completely remove all the lesions, including synovectomy of the TMJ to prevent recurrence and improve mandibular function. Postoperatively, a mild motor disturbance was observed in the temporal branch of the right facial nerve, but the patient gradually recovered and was discharged. A 6 months post-surgery review indicated that although there was mild replicable pain in the right TMJ when opening the mouth, AROM had improved to 43 mm. Moreover, no obvious mandibular deviation, suggesting the possibility of a transient TMD-associated malocclusion, and no recurrence of symptoms was observed after nearly 1.5 years postoperatively Fig. A, B.Fig.Due to the likelihood of a persistent malocclusion albeit of reduced severity, and recurrence of the TMD manifestations, an extended follow-up including a validated DC/TMD assessment is planned. Notwithstanding, the patient was very satisfied with the treatment outcomes and was very grateful to the entire medical team Fig. .Fig. 9HiTMJ is indispensable to key facial activities such as jaw mobility, mastication, and verbal and emotional expression. CPPD and CS are among inflammatory arthropathies and neoplasm pathologies respectively, which commonly contribute to the annual incidence (4%) and prevalence (5–31%) of chronic TMJ pain, which is an economic burden to society , 47. CPPA definitive diagnosis of CPPD is based on the presence of criteria I–IIb and considered likely with IIa–IIIb, as shown in Table Therefore, taken together, the aforementioned findings suggested a coexistence of CPPD and SC in the right TMJ in the present case, the rare pathology. The mechanism by which SC and CPPD coexist is not well understood , 41, 42.A recent report by Stack and Geraldine  well sumThe coexistence of both SC and CPPD is managed through excision of the lesion, anti-inflammatory therapy with NSAIDs, cleansing therapy, and follow-up. Excision of the loose bodies and pathological synovium is necessary, and some reports suggest that mandibulectomy is required if the lesion is large and invasive. However, Blankestijn et al.  reportedIn summary, although isolated cases of CPPD and SC of the TMJ are more prevalent in the literature, a monoarticular coexistence of these diseases is rare. This is often attributed to factors such as difficulties in diagnosing TMJ diseases, despite the advances in imaging technology and the reported interest in research and application of several different diagnostic approaches. Moreover, optimal treatment depends on several considerations. The present case delineated the molecular etiopathology of CPPD and SC and unveiled the need for continued deciphering mechanisms leading to the diseases' coexistence in the TMJ. In addition, the importance of confirmatory testing for accurate diagnosis was recapitulated and, in our opinion, appropriate management of the case was considered.


Malakoplakia is a rare entity on inflammatory base that mostly occurs in immunocompromised individuals which is thought to be secondary to a bactericidal defect in macrophages. The genitourinary tract is typically affected. The appendix is a very rare localization. We report a case of malakoplakia in the appendix of a young healthy patient with a recent history of abdominal pain associated with diarrhea and nausea. The colonscopy and CT scan showed an extramucosal bumping mass pressing on the cecum and covered by normal mucosa. The patient underwent to laparoscopic appendectomy. The histology showed a malakoplakia of the appendix. Gastrointestinal localization of malakoplakia is often associated with preexisting diseases, which are probably responsible for an immune disorder underlying the etiopathogenesis of the disease. However, in our case, the patient had no comorbidities. Probably, a clinically unknown immune predisposition plays an important role. Further studies are needed to clarify this nexus. Malakoplakia is an inflammatory reaction to organisms, which include bacteria, mycobacteria, fungi and occasionally parasites. Usually makes its presence known as a papule, plaque or ulceration which generally occurs in the genitourinary tract; nevertheless, it has been described in almost all bodily organs.Malakoplakia arises on average around 50 years of age; pediatric cases are rare . Women aMalakoplakia, from the Greek ‘malakos’, meaning soft, and ‘plakion’, meaning plaque, is the term that was coined by von Hanseman to describe the first human case in 1903, a postmortem bladder finding of a 66-year-old man who died of pulmonary tuberculosis, characterized by the microscopic relief of soft and elevated, yellow-to-brownish, lesions. Subsequent early cases of malakoplakia were all showed in the genitourinary tract .Within the genitourinary tract, which remains the most affected site, the urinary bladder is the most frequent region of onset . HoweverWe describe a case of malakoplakia of the appendix, probably the seventh reported in literature.A 33-year-old male presented himself to our clinic with a 1-year history of increasing abdominal pain, diarrhea and nausea.The colonoscopy showed an extraluminal bumping mass pressing on the cecum of about 4 cm covered by regular mucosa compatible with gastrointestinal stromal tumor (GIST) or lipoma; it was not possible to identify the appendicular dimple.The CT scan of the abdomen found a pseudonodular relatively inhomogeneous submucosal mass located by the median wall of the cecum, of about two centimeters in size with relatively thickened margins and fluid dense content. Apparent continuity with the appendicular lumen which appears relatively thinned and occupied by material of moderate density. The finding could refer to a locoregional inflammatory collection in continuity with the cecum and the appendicular lumen.The patient was submitted to laparoscopic appendectomy, with and intraoperative finding of an intraluminal mass of the base of the appendix. A stapled section of the appendix with a partial inclusion of the wall of the cecum was performed. The patient was discharged 2 days after the surgery; no post-operative complications occurred.The resected specimen consisted of 8-cm-long appendix with enlarged lumen, thickened walls for 1.5 cm. The cecum resection margin was free of disease.On microscopy, the appendix showed a thickening of his walls due to an accumulation of rounded elements with rich granular cytoplasm containing amorphous corpuscles and surrounded by microcalcifications, macrophage elements with adjacent de-epithelialized, ulcerated and eroded mucous tracts. No positive S100/pancytokeratin elements were found in the numerous sections examined. The morphological findings therefore indicated a form of malakoplakia –3, confiAt 1, 3 and 6 months follow-up, the patient results in good condition.et al. [et al. in 2008 [In 1970, W. M. Blackshear  describeet al.  in 2000.et al.  or tuberet al. , and witet al. . Shaktaw in 2008  describeMalakoplakia is thought to be due to a defect in the response to bacterial infection. Macrophages and monocytes show deficient phagolysosomal activity; they phagocytose bacteria but are unable to digest them completely. Partially digested bacteria accumulate in the cell cytoplasm and produce a granulomatous immune cell reaction. Diagnosis is not easy, as it can also run asymptomatically and be an entirely incidental finding. In cases with gastrointestinal involvement, the most common symptoms are abdominal pain, diarrhea, bleeding and dyspepsia. Recognizing malakoplakia is difficult because it is not associated with either specific clinical symptoms or characteristic imaging pictures. Diagnosis is based on endoscopy findings, which reveal the presence of yellowish vascularized plaques or white polypoid nodules. Confirmation is made by biopsy of the affected tissues. Histologic examination demonstrates the presence of von Hansemann cells, or rather histiocytes with small nuclei and acidophilic granular cytoplasm containing Michaelis–Gutmann bodies . This lesion is pathognomonic for malakoplakia.Treatment is based on administration antibiotics capable of penetrating inside the cells, as fluoroquinolones and trimethoprim-sulfamethoxazole. The duration of therapy is not yet standardized. In pseudotumor forms, as in our case, surgical removal of lesions is indicated.Gastrointestinal malakoplakia is often associated with diseases underlying immunosuppressive states. However, this association has only been hypothesized in the case of appendicular localization. Indeed, in our case, the patient, did not suffer from any known comorbidities, posing a not indifferent question mark on the etiopathogenesis of the condition. Given the patient’s young age, it cannot be ruled out that an unknown immune predisposition may have led to the development of appendicular malakoplakia. However, further studies are needed to better elucidate the predisposing factors for this condition.


Carotid body tumors (CBTs) are rare neuroendocrine neoplasms arising near the carotid bifurcation with a reported incidence of 1 to 2 cases in 100,000 patients. Most CBTs are sporadic, benign, slow-growing, and non-secreting, but untreated CBTs can grow locally to compress the nearby blood vessels, esophagus, and airway. Regional metastases can occur in 5% to 10% of cases, but distant metastases are exceedingly rare, occurring in roughly 1–2% of cases. As such, the optimal treatment for metastatic CBTs is not well-defined. We report a rare case of a patient with CBT distant metastases causing spinal cord compression.A 40-year-old African American female presented with a right neck mass, headaches, vertigo, tinnitus, hoarseness, and dysphagia. Imaging demonstrated a Shamblin II right neck mass; subsequent transcervical resection and pathology showed a carotid body paraganglioma. The patient recurred locally near the carotid bifurcation, so she underwent Stereotactic Body Radiation Therapy to the recurrent right neck disease. She later re-presented with new onset bilateral lower extremity weakness, dysmetria, and numbness. She was found to have metastatic disease to the thoracic spine causing spinal cord compression. She underwent laminectomy, tumor resection, and posterior fixation followed by adjuvant radiation therapy. She was started on systemic therapy with sunitinib. She eventually progressed with metastatic disease to the right iliac bone, which was treated with palliative radiotherapy. Second line systemic therapy with capecitabine and temozolomide was started. At last follow up, the patient was asymptomatic with stable persistent disease.Paragangliomas often exhibit a prolonged interval to the development of progression; locoregional recurrences or rare distant metastases have been reported to occur as many as 20 years from diagnosis. The natural course of CBTs in other cases as well as the present case call into question the idea that CBTs are truly benign; instead CBTs may be indolent tumors with metastatic potential. Treatment choices for CBTs include surgical resection, radiation therapy, and systemic therapy, though the optimal treatment regimen for metastatic CBTs is not well-defined. A more advanced understanding of CBT pathophysiology, disease classification, risk stratification, and treatment options is needed to improve outcomes for patients. Carotid body tumors (CBTs), generally referred to as paragangliomas, chemodectomas, and glomus tumors, are rare, highly vascular neuroendocrine neoplasms. They arise near the carotid bifurcation within glomus bodies (paraganglia) and are derived from the embryonic neural crest .Most paragangliomas arise in the adrenal gland with only 3% of extra-renal tumors manifesting in the head and neck . Of thesSurgical resection is the treatment of choice for CBTs. The Shamblin classification system uses the tumor’s size and association with the carotid vessels to assess the possibility of surgical resection and associated mortality. Based on this relationship and risk, tumors are classified into 3 groups . Group 1Only about 5–10% of incident cases of CBT present with regional metastasis and distant disease is very rare . Over a A 40-year-old African American female presented in 2014 with complaints of a right neck mass that was first appreciated 9 months earlier. The appearance of the neck mass coincided with a constellation of symptoms: frequent severe headaches, periods of lightheadedness, vertigo and tinnitus, hoarseness, dysphagia, blurred vision and tearing of the right eye. On physical examination, a non-pulsatile, non-tender mass was palpated, deep to the right sternocleidomastoid muscle. The patient underwent CT neck imaging, which revealed a 6.1 × 4.0 × 4.1 cm right neck mass extending to the base of the skull and encompassing the internal and external carotid artery  . Radiatip= 0.047) [Radiation therapy has traditionally served as a second line intervention in the treatment of CBTs, but conventional radiotherapy with fractionated external beam radiotherapy (EBRT) has more recently been examined as a primary treatment in multiple studies. One study reported on 13 patients with CBTs, 10 of which underwent definitive EBRT. Local control was achieved in all patients . A meta-= 0.047) . The patCurrent studies support systemic therapy options for paragangliomas with distant metastases including octreotide or lanreotide; sunitinib; temozolomide; capecitabine; combination chemotherapy with cyclophosphamide, vincristine, and dacarbazine; or radionuclide therapy with 131Iodine or 177Lutetium-dotatate –32. The Risk stratification and investigation into the role of the various treatments for CBT is needed in order to create a protocol that will improve outcomes for patients. Distant metastases is a rare event in CBT but can occur many years after primary treatment, so while routine systemic imaging is not currently recommended for all patients, long term clinical follow up and attention to patient-reported symptoms may help to identify distant metastasis sooner.


Dermatofibrosarcoma protuberans (DFSP) is a rare, locally aggressive spindle cell mesenchymal tumor arising in the dermis, with low metastatic potential. The most commonly affected sites are the trunk and proximal extremities; rarely are acral sites involved. Atrophic DFSP is a rare form of DFSP, that is morphologically different but histologically similar to DFSP. It commonly affects young adults between the ages of 20 to 50 years. The current management strategy for atrophic DFSP is surgical excision with long-term follow-up to detect any recurrence. Only one known case of atrophic DFSP with eosinophilic infiltration is what makes our case an exceptionally rare presentation. Dermatofibrosarcoma protuberans (DFSP) is a rare cutaneous tumor representing 1.8% of all soft tissue sarcomas ,3. The WThe patient is a 27-year-old female who presented to the dermatology office with a skin lesion on the right anterior shoulder. The lesion had been present first as an area of discoloration for 2–3 years. Several weeks before presentation, the lesion changed texture and developed a focal papular area. The patient had no pertinent past medical or surgical history and no known drug allergies. A 2.5 cm erythematous to violaceous atrophic patch with an isolated, slightly firm papule, was noted in the right anterior shoulder distribution . At the Various subtypes of DFSP have been described, including atrophic, granular, myxoid, sclerosing, fibrosarcomatous, and pigmented (Bednar Tumor) ,11,12,13Tissue eosinophilia in this lesion is a highly unusual finding. Eosinophilia is a frequently encountered finding in malignant neoplasms that is considered a favorable host response to malignant tissue . EosinopAtrophic DFSP can be confused with morphea, atrophic dermatofibroma, morpheaform basal cell carcinoma, resolving panniculitis, and lipoatrophy ,16. The Surgical excision remains the mainstay of treatment . HistoloIn our case, these changes were thought to be consistent with a diagnosis of dermatofibrosarcoma protuberans with eosinophilic infiltration. However, the differential diagnosis included a fibrotic reaction to an insect bite, or an unusual inflammatory response to a possible cyst or traumatic event. Granulomatous diseases were not considered in our differential diagnoses, as no granulomatous or histiocytic component was noted in either of the biopsy specimens. Demonstration of diffuse expression of CD34, in the initial and subsequent biopsy and characteristic histopathology in the subsequent biopsy, confirmed the diagnosis of DFSP.We report a rare case of atrophic DFSP with eosinophilic infiltration. DFSP should be considered in the differential diagnosis, while evaluating an atrophic cutaneous plaque to avoid delays in diagnosis. An evidenced-based defensive immunological role of eosinophils has been postulated in certain malignancies . However


Ingested wooden toothpick (WT) represents a rare cause of acute abdomen. Preoperative diagnosis of ingested WT is a challenge because of its unspecific clinical presentation, the low sensitivity rate of radiological investigations and the patient's inability to often recall the event of swallowing a WT. Surgery represents the main treatment in case of ingested WT-induced complications.loop ileostomy were performed. The postoperative course was uneventful.A 72-year-old Caucasian male presented to the Emergency Department with a two-day history of left lower quadrant (LLQ) abdominal pain, nausea, vomiting and fever. Physical examination revealed LLQ abdominal pain and rebound tenderness with muscle guarding. Laboratory tests reported high levels of C-reactive protein and neutrophilic leukocytosis. Abdominal contrast-enhanced computed tomography (CECT) showed colonic diverticulosis, wall thickening of the sigmoid colon, pericolic abscess, regional fatty infiltration, a suspicion of sigmoid perforation secondary to a foreign body. The patient underwent diagnostic laparoscopy: a sigmoid diverticular perforation caused by an ingested WT was noticed and a laparoscopic sigmoidectomy with end-to-end Knight-Griffen colorectal anastomosis, partial omentectomy and protective The ingestion of a WT represents a rare but potentially fatal condition which may cause GI perforation with peritonitis, abscesses and other rare complications if it migrates out of the GI tract.Ingested WT may cause serious GI injuries with peritonitis, sepsis or death. Early diagnosis and treatment are crucial for reducing morbidity and mortality. Surgery is mandatory in case of ingested WT-induced GI perforation and peritonitis. •A wooden toothpick (WT)-induced perforation of a sigmoid diverticulum is a rare cause of acute abdomen.•Diagnosis of WT-induced gastrointestinal (GI) perforation is a challenge because of the absence of specific clinical presentation.•WT impaction can be difficult to diagnose through radiology.•Radiology plays an important role in the diagnosis of WT-induced GI perforation•Surgery represents the main treatment in case of WT-induced open GI perforation and peritonitis. The patient was initially managed with fluids, intravenous broad-spectrum antibiotics, antipyretic drugs and bowel rest. After an abdominal ultrasound exam, the patient was evaluated by abdominal contrast-enhanced computed tomography (CECT) which confirmed colonic diverticulosis and revealed wall thickening of the sigmoid colon, pericolic abscess, regional fatty infiltration, a suspicion of sigmoid perforation secondary to a FB  abdominal pain, nausea, vomiting and fever (38 °C); others vital signs were normal. His past medical history included arterial hypertension, colonic diverticulosis, and appendectomy. The patient, wearing dentures, was on hypertensive medications for ten years and referred habit on smoking but denied alcohol consumption; his familial medical history was normal. He was retired from the work, married and of medium socio-economic status. Physical examination revealed mild abdominal distention, LLQ abdominal pain on superficial and deep palpation with obvious muscle guarding and rebound tenderness, hypoactive bowel sound. Laboratory tests reported high levels of C-reactive protein (90.5 mg/L) and neutrophilic leukocytosis (WBC 16.700 10 to a FB A,B. The  to a FB  and coveformalin , consistformalin . Stapled3This rare clinical case describes a delayed presentation of WT-induced perforation of a sigmoid diverticulum leading to acute abdomen. FB ingestion represents a common problem in everyday emergency clinical practice. Foreign bodies include a wide range of materials, from biological ones such as bones 4Ingested WT is a rare event which may cause serious GI injuries with peritonitis, sepsis or death. When risk factors for FB ingestion are present, physicians should be aware of the possibility of FB ingestion like as rare cause of acute abdomen Imaging, endoscopy and/or laparoscopy are helpful in detecting WT ingestion and its complications. Surgery is mandatory in case of WT-induced GI perforation and peritonitis.Ethical approval has been exempted by our institution because this is a case report and no new studies or new techniques were carried out.Not applicable.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.Not commissioned, externally peer-reviewed.The guarantor for this case report is Giuseppe Evola.Not applicable.Giuseppe Evola: Drafting the manuscript, literature research.Giulia Impellizzeri: Operated on the patient, drafting the manuscript.Elia Pulvirenti: Operated on the patient, drafting the manuscript.Maria D’Angelo: Drafting the manuscript, literature research.Martina Reina: Drafting the manuscript, literature research.Giuseppe Angelo Reina: Operated on the patient, revising the manuscript.Not applicable.


Xanthogranulomatous cholecystitis, a rare variant of cholecystitis, may infrequently be complicated by spontaneous cholecystocutaneous fistula.We report the case of a 75-year-old Saudi Arabian man who presented with “a painful area of redness” (cellulitis) over his right upper abdomen. Abdominal computed tomography revealed multiple collections, which were drained surgically. A discharging sinus was identified, and a fistulogram revealed cholecystocutaneous fistula during his follow-up visit. The patient underwent laparoscopic management and recovered uneventfully. Final histopathological evaluation confirmed acute-on-chronic xanthogranulomatous cholecystitis .Although rare, surgeons should consider cholecystocutaneous fistula in the differential diagnosis of anterior abdominal wall abscesses, particularly in patients with concurrent or background symptoms of gallbladder disease. We report the first case of laparoscopic management for cholecystocutaneous fistula in Saudi Arabia. Biliary fistulization refers to the formation of an abnormal tract between the gallbladder and adjacent epithelial surfaces, which could be the gastrointestinal tract  or the abdominal wall . External fistulization maybe spontaneous, post-traumatic, or iatrogenic in origin .Spontaneous cholecystocutaneous fistula (CCF) is a rare complication of neglected gallbladder disease and is infrequent in modern surgical practice because symptoms associated with the gallbladder are unlikely to be ignored by patients or missed by physicians. This entity was first described by Thilesus in 1670, and to date, less than 100 cases have been reported in the literature . We repoA 75-year-old Saudi Arabian man with a known clinical history of diabetes and hypertension presented to the emergency department with a 4-day history of “a painful area of redness” over his right upper abdomen. He denied any other symptoms at the time of presentation. The patient’s past history was remarkable for multiple episodes of colicky, transient, but tolerable upper abdominal pain associated with heavy meals. His past surgical history included bilateral inguinal hernia and hydrocele repair. He denied any relevant drug or family history.Upon examination, the patient was ambulatory, conscious, and oriented. He looked well, not pale or jaundiced. He was afebrile with stable vital signs. Except for the area of redness over the right upper quadrant, his abdomen was soft, lax, nontender, and nondistended. The right upper abdomen showed a raised area of cellulitis . A review of other systems revealed no significant findings.9/L (4–10 × 109/L), and hypokalemia of 3.1 mmol/L (3.5–5 mmol/L). Liver function test results were within the reference ranges.Routine laboratory workup showed a slightly elevated white blood cell count of 10.4 × 10Abdominal computed tomography (CT) revealed multiple collections involving the anterior right upper abdominal wall with superficial and deep components between the abdominal wall muscles. The superficial component measured approximately 2.0 × 5.0 × 5.0 cm , and the deep component measured approximately 1.5 × 5.0 × 8.5 cm . The patient’s postoperative course was uneventful, and he was discharged on oral antibiotics. He was pain free and had recovered uneventfully at the first postoperative visit at 2 weeks. However, examination showed an opening at the edge of the wound site with mucoserous discharge. Therefore, the patient was admitted for further evaluation.We performed fluoroscopy guided fistulography Fig. , and obsIntraoperatively, we performed meticulous dissection to separate adhesions around the fistulous tract until the fistulous tract was clearly visible Fig. . The traThe patient showed an uneventful postoperative recovery. The drain was removed on the second postoperative day, and the patient was discharged. He was followed up 2 and 4 weeks postoperatively and recovered well with complete symptom resolution. Final histopathological evaluation confirmed diagnosis of acute-on-chronic xanthogranulomatous cholecystitis.We present a case of spontaneous CCF, a rare complication of gallbladder disease. Overall, fewer than 100 cases have been reported in the literature . To the Most cases of spontaneous CCF are associated with neglected calculous gallbladder disease, as observed in our patient. However, a small number of studies have reported that such complications were secondary to gallbladder adenocarcinoma , 5. SimiThe clinical manifestations vary and depend on the patient’s status and time of presentation; however, most patients present with systemic  and local  features . AlthougImaging is necessary for diagnostic confirmation. Ultrasonography may show abscesses, gallstones, and evidence of inflammation. However, it is unlikely to demonstrate a fistulous tract, and CT (or magnetic resonance imaging) is necessary. Additionally, a fistulogram (radiography or CT) may be a useful aid in confirming and accurately assessing the fistulous tract. Magnetic resonance cholangiopancreatography is an essential diagnostic modality if biliary obstruction is suspected. Biliary obstruction should be suspected on the basis of clinical findings (jaundice) or investigation results .Conclusive guidelines are unavailable for the diagnostic approach and optimal treatment of CCF. Therefore, an individualized management regimen should be implemented on the basis of the underlying etiology, time, and nature of presentation, and treatment largely depends on the surgeon’s expertise. Decision-making should necessarily be based on thorough discussion with the patient.et al. have described laparoscopic surgery in such cases [The management of the acute phase of this condition includes resuscitation, pain control, and antibiotic therapy. Watchful waiting to allow healing and spontaneous closure of the fistulous tract may be considered in patients deemed unfit for surgery. However, the outcomes of such approaches remain unclear. Several studies recommend an open approach, considering its feasibility and lower complication rates , 8. We ach cases . In addich cases , 10.Finally, following the final histopathological diagnosis is of the utmost importance, as gallbladder carcinoma is a possibility. In our case, histopathological evaluation confirmed diagnosis of acute-on-chronic xanthogranulomatous cholecystitis (XGC). XGC is a rare inflammatory disease of the gallbladder. It is characterized by destructive inflammation and fibrosis of the gallbladder wall and sometimes adjacent structures. This makes it difficult to differentiate from carcinoma radiologically, hence the importance of histopathological evaluation. It presents similarly to calculous gallbladder disease and has comparable complications, yet they are more common .This case report highlights that surgeons should consider CCF in the differential diagnosis of anterior abdominal wall abscesses, particularly in patients with concurrent or background symptoms of gallbladder disease. Being a rare differential, some features, such as history of longstanding and neglected gallbladder disease or the presence of bile drainage through the external opening, might further point to this diagnosis. Imaging is necessary for diagnostic confirmation, and fistulography is recommended. Decision-making regarding the optimal surgical approach for this rare complication warrants careful evaluation of the disease stage and the patient’s general health condition, and should be based on the surgeon’s expertise and the patient’s preference.


Gastric hyperplastic polyp (GHP) commonly arises in the abnormal background mucosa, which makes it easy to be misdiagnosed and missed, and has a potential risk of malignant transformation over time. Here, we present a case of neoplastic transformation of GHP in a context of autoimmune gastritis (AIG).In 2020, a 67-year-old woman was admitted for endoscopic review 6 years after gastric polyp resection, the histological diagnosis of gastric polyp was neoplastic transformation of GHP as before. The patient had undergone multiple polypectomies at the same part. Then histological examination revealed that partial epithelial hyperplasia and dysplasia, and the neoplastic areas were interlaced with normal mucosa.We further found that the background diagnosis was AIG. These results supported the diagnosis of neoplastic transformation of GHP in a context of AIG. With the doubt of missed diagnose, we retrospectively analyzed the medical history in 2014, 2015 and 2016, confirmed the presence of AIG. Unfortunately, serological tests and special treatment were not performed.The correct diagnosis was eventually confirmed in 2020, which enables patients to receive normal treatment and monitoring, and avoids further deterioration of the disease.The purpose of this case report is to increase clinical awareness of neoplastic transformation of GHP in a context of AIG, and hope promise for early diagnosis and treatment. GHP occurs in all ages, but the high incidence age is 65 to 70 years old. Of which, 58% to 70.5% of patients with GHP are female, strongly indicating a gender difference in the occurrence and development of GHP. Morphologically, GHP ranges in size from a few millimeters to a few centimeters, leading to be misdiagnosed as tumor or cancer under endoscopy. Some of patients with GHP may present with dyspepsia, heartburn, abdominal pain, or anemia, but most of them are usually asymptomatic, which makes it difficult to be diagnosed in advance. GHP was considered to be a non-tumor lesion previously. However, an increasing number of studies,7 have shown that GHP could be complicated with dysplasia and canceration, especially for polyps larger than 2 cm in diameter have a higher risk of neoplastic transformation.Gastric hyperplastic polyp (GHP) is one of the most common polyps encountered in the stomach, especially in the antrum, and detected in 1.9% of 110,000 patients subjected to gastroscope. In the context of AIG, GHP is often multiple, large, pedunculate or subpedunculate, and easy to produce neoplastic transformation. Zhang et al reported that the incidence of neoplastic transformation of GHP in AIG is approximately 2.8%. In this paper, we report a case of GHP in a context of AIG misdiagnosed as adenomatous polyp. Fortunately, the correct diagnosis was eventually confirmed by comprehensive analysis of gastroscopy, pathology and serology. Therefore, when patients recurrent GHP in the body of gastric with a history of anemia, we should be alert to the possibility of AIG, especially repeated recurrence in the same position after polyp resection, formulate a reasonable gastroscopy and follow-up program to achieve early diagnosis and treatment, to avoid complications and neoplastic transformation.In general, GHP occurs in the abnormal background mucosa, particularly in autoimmune gastritis (AIG), also known as type A gastritis.In August 2020, a 67-year-old Chinese woman was admitted to our hospital for review 6 years after gastric polyp resection. Her symptoms included fatigue, poor appetite and lost 4 kg in a year, and she was accompanied by a 6-year history of iron deficiency anemia, which has been taking orally with iron and folic acid. The results from blood tests performed showed an iron deficiency anemia and decreased hemoglobin. No abnormalities were found in the Female Tumor Markers or Gastrointestinal Tumor Markers Table . Upper eInterestingly, we discovered that the patient had undergone multiple polypectomies in the same part of the corpus, and was accompanied with anemia all the time. Therefore, we did histological examination and revealed that partial epithelial hyperplasia and dysplasia, and the neoplastic areas were interlaced with normal mucosa, as indicated by positive Ki-67 staining in the whole layer of tumor area and a decrease in mucus-secreting goblet cells in the surface epithelium. Fig. C–F. ThesThrough further clinical and pathological collaboration, we further found that the normal mucosa around polyps showed atrophy of inherent glands in gastric mucosa with intestinal metaplasia and pyloric gland metaplasia Fig. G and H, The patient had multiple surgeries with a long history of anemia, the onset of AIG, so what was the relationship between neoplastic polyps and AIG? Is there any possibility of misdiagnosis in this patient before? To address these questions, we reviewed the medical history.In November 2014, the first visit of this patient was due to a physical examination. The results from positron emission tomography-computed tomography revealed gastric space occupation, as demonstrated by the vegetable pattern mass of the gastric wall on the greater curvature of the gastric body protrudes into the gastric cavity, and metabolism increases. Therefore, neoplastic lesions were considered. Upper endoscopy showed that there were 1.5 × 2.0 cm polyps in lobulated shape on the greater curvature of the gastric body, with scattered erosion on the surface ,YX collected patient information, analyzed results and wrote the first draft of the manuscript. HH collected information and performed the research. YW had pathological staining. ZS, LW, DP, XG, NY, YY, WZ did the upper gastrointestinal examination. TM supervised and interpreted the experimental data, critically revised the manuscript. YL designed the research, analyzed the data, and revised the manuscript critically. All authors read and approved the final manuscript.Conceptualization: Yunqi Xing, Haixiao Han.Formal analysis: Yunqi Xing, Yuxuan Wang, Zhongmei Sun, Linheng Wang, Dan Peng, Xiaoyuan Guo, Na Yao, Yali Yuan, Wenji Zhang.Methodology: Haixiao Han.Supervision: Tangyou Mao, Yuyue Liu.Writing – original draft: Yunqi Xing.Writing – review & editing: Tangyou Mao, Yuyue Liu.


Symptomatic Meckel's diverticulum (MD) is easily neglected in the acute abdomen during pregnancy. MD is the most common congenitally anomalous development of the intestines, with an incidence of 2% in the general population, although it is not easily diagnosed because of variable clinical features. Especially when complicated with pregnancy, doctors can easily overlook this disease, which directly threatens maternal and foetal life.We report the case of a 25-year-old at 32 + 2 weeks of gestation complicated with MD volvulus who presented with progressive abdominal pain and finally peritonitis. She underwent exploratory laparotomy and small-bowel resection. The mother and the baby successfully recovered.MD-complicated pregnancy is not easily diagnosed. Once highly suspiciously diagnosed, especially with peritonitis, surgery should be arranged, which helps preserve maternal and foetal life. MD is the most common congenital anomaly of the gastrointestinal tract, caused by incomplete obliteration of the omphalomesenteric duct in the developing embryo, which has an incidence of 2% , 2. PatiHere, we report a young woman at 32 + 2 weeks of gestation complicated with MD volvulus who presented with progressive abdominal pain and finally peritonitis. Meckel's diverticulum is the third trimester of pregnancy is rare. We summarized five such cases that we found in the existing literature in A 25-year-old at 32 + 2 weeks gestation arrived at the hospital because of abdominal pain with a recent ingestion of spicy food. Approximately 4 h later, she felt abdominal colic pain, which improved spontaneously or by changing position, with vomiting 5 times. Without diarrhoea, she defecated normal soft stool twice. She had no fever. The pain was not relieved after she received the phloroglucinol at a dose of 40 mg iv in the outpatient department. Then, she was transferred to the inpatient department for treatment at 17:00.Physical examination showed a pregnant abdomen, which was consistent with the gestational age. The patient had total abdominal pain, mainly in the lower abdomen, with tenderness and rebound pain. She had no noticeable uterine contractions, pain, or feelings of tightness. The cervix was closed.Ultrasound testing of the foetus and the placenta was normal. Antepartum testing was also normal. Foetal surveillance was normal, and there were no obvious uterine contractions.The patient was not considered to be in preterm labour. The cause of her abdominal pain was investigated, and acute gastroenteritis was considered. When she arrived at our inpatient department, we did not use dexamethasone considering that there was no uterine contraction pain.Four hours after admission (at approximately 21:30), the patient felt increased pain in her left lower quadrant. She had total abdominal pain, mainly in the left lower abdomen, with tenderness and rebound pain. Strong, regular contractions were occurring every 3 min , and we The white blood cell count was 16,530, and the percentage of neutrophils was 86.20%. Procalcitonin, interleukin-6, C-reactive protein, pancreatic amylase, and pancreatic lipase levels were normal. As the patient's abdominal pain worsened, we examined the routine blood and infection indicators again. The white blood cell count was 20,670, and the percentage of neutrophils was 90.30%. Procalcitonin was 0.03 ng/ml, interleukin-6 was 44.90 pg/ml, and C-reactive protein was 7.90 mg/L. Computed tomography (CT) examinations are associated with ionizing radiation and are not advised in pregnancy, which could have a negative effect on the foetus, mainly depending on the the absorbed radiation dose and the gestational age at the time of exposure. However, CT examination may improve the diagnosis accuracy in the acute abdomen, especially intestinal obstruction and appendicitis. The patient was informed of the advantages and disadvantages of CT examinations. With the patient's permission, we immediately contacted the imaging department to perform B-ultrasound and CT examination. CT examination showed that the left lower abdominal part of the small intestine was in a vortex shape, and the local intestinal lumen was dilated. The intestinal inflammation was distinguished from the incomplete small intestinal torsion . No obviThe next morning, the patient continued to feel abdominal pain, and she presented obvious tenderness and rebound pain, which showed peritonitis. We consulted radiologist and surgeon and they considered that the patient could have volvulus. The surgeon suggested that exploratory laparotomy be performed. Due to the large size of the pregnant uterus, which would obstruct the surgical field of view, combined with the gestational stage, the surgeon recommended emergency caesarean section in obstetrics, and the surgeon would assist in the exploration. The complete 10 mg dexamethasone injection was empirically administered before the operation.We performed a caesarean section. The Apgar score at 1, 5, 10, 15 and 20 min was 10, 5 (each item was deducted one point), 9 (reflex irritability was deducted one point), 9 (reflex irritability was deducted one point) and 9 (reflex irritability was deducted one point), respectively. The foetus looked healthy and got full score at 1 min. However, the heart rate, respiratory effort, muscle tone, reflex irritability, and color from the foetus were not the best, so the foetus got 5 points when 5 min. Subsequently, the foetus was rescued by keeping warm and positive pressure ventilation by mask resuscitation bag. And then the foetus got 9 points because the reflex irritability was not the best after rescue. The foetus was finally transferred to the neonatology department. During the operation, the surgeon explored the small intestine MD, with a 360° twist and local necrosis without perforation . The MD Early diagnosis of acute abdomen in obstetrics is of great significance to improve the life and health of mothers and foetuses. Common obstetrical acute abdomen includes digestive inflammatory diseases, such as appendicitis, acute cholecystitis, acute pancreatitis, and intestinal obstruction. In addition, urinary calculi and torsion of the ovarian cyst pedicle are also seen in the hospital. However, MD is rare. In this study, we reported pregnancy-complicated MD volvulus, which attracted our attention to the diagnosis of obstetrical acute abdomen. When we managed the acute abdomen, we could also consider MD.MD is the most common anatomic abnormality of the gastrointestinal tract, resulting from the failure of the omphalomesenteric duct to involute during the 5th to 7th weeks of gestation  and it hOur patient presented primarily with abdominal pain, which became predominantly left lower abdominal pain, and eventually developed peritonitis. The first case presented with right lower abdominal pain reminiscent of acute appendicitis, with subsequent finding of MD at surgery. In fact, appendicitis could be the most common surgical pathology in the acute abdomen, but some rare diseases should be also considered. MD should not be neglected as one of the differential diagnoses of acute abdominal complaints in pregnancy.Doctors find it difficult to diagnose MD-complicated pregnancy because it can present unspecific symptoms, such as abdominal pain, nausea, vomiting, and obstipation, which overlap with other conditions and can also occur physiologically during pregnancy. Moreover, moderate leucocytosis can be normal for pregnant women but indicates inflammation in the population without pregnancy, making it more difficult to perform timely evaluations. During pregnancy, symptomatic MD should be distinguished from obstetric diseases such as preterm labour, placental abruption or chorioamnionitis .Ultrasonography is a common radiological investigation in the pregnant patient presenting with an acute abdomen . UltrasoCT is common, but doctors can easily overlook MD, which could be considered a small intestinal loop without the presence of complications . When MDIn our report, CT showed the “whirlpool sign”, indicating small bowel volvulus. Combining CT results and peritonitis, the surgical chief thought it was more likely to be small bowel volvulus and suggested exploratory laparoscopy, by which we found MD complicated by volvulus.The mainstay of management for symptomatic Meckel's patients is surgical intervention. In the asymptomatic patient, a conservative approach is well justified giving the rarity of complications . Every pMany diseases can lead to acute abdomen, while MD is a rare cause of acute abdomen. CT is valuable for the suspicious diagnosis of MD accompanied by symptoms. When managing the acute abdomen, we must not forget the possibility of MD. For the pregnant woman with symptomatic MD, surgery is the preferred mode of management.