International Journal of Surgery Case Reports最新文献

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An unusual lead point: jejunal leiomyosarcoma causing jejuno-jejunal intussusception - a case report. 一个不寻常的引子:空肠平滑肌肉瘤引起空肠-空肠肠套叠1例。
IF 0.6
International Journal of Surgery Case Reports Pub Date : 2026-09-14 eCollection Date: 2026-10-01 DOI: 10.1097/RC9.0000000000001003
Mayin Uddin Mahmud, Sanjoy Kumar Nath
{"title":"An unusual lead point: jejunal leiomyosarcoma causing jejuno-jejunal intussusception - a case report.","authors":"Mayin Uddin Mahmud, Sanjoy Kumar Nath","doi":"10.1097/RC9.0000000000001003","DOIUrl":"https://doi.org/10.1097/RC9.0000000000001003","url":null,"abstract":"<p><strong>Introduction: </strong>Adult intussusception is rare. Unlike in pediatric cases, adult intussusception typically arises from an underlying pathology. Primary jejunal leiomyosarcoma is an exceptionally rare malignant tumor originating from the smooth muscle cells of the small intestine.</p><p><strong>Case presentation: </strong>A 52-year-old man presented with signs and symptoms of acute intestinal obstruction, including abdominal pain, constipation, vomiting, and abdominal distension of 2 days duration. Contrast-enhanced computed tomography revealed a jejunojejunal intussusception with a lead-point intraluminal mass. Exploratory laparotomy showed a jejuno-jejunal intussusception 40 cm distal to the duodenojejunal flexure. Segmental resection with primary anastomosis was performed. The postoperative period was uneventful. Histopathology demonstrated a spindle-cell neoplasm consistent with leiomyosarcoma. Immunohistochemistry showed strong positivity for smooth muscle actin and desmin, and negativity for CD117, DOG-1, and S-100.</p><p><strong>Discussion: </strong>Primary jejunal leiomyosarcomas often present with nonspecific symptoms. Imaging, especially CT, is critical for diagnosis, but confirmation requires histopathology and immunohistochemistry. Complete surgical resection remains the treatment of choice. This case contributes a complete immunohistochemical panel that distinguishes leiomyosarcoma from gastrointestinal stromal tumors, the formal AJCC 8th edition staging, and a rationale for adjuvant chemotherapy based on soft tissue sarcoma protocols - thereby providing a more standardized diagnostic and management template than existing jejunal leiomyosarcoma case reports.</p><p><strong>Conclusion: </strong>Jejunal leiomyosarcoma presenting as jejuno-jejunal intussusception is exceptionally rare. Early recognition and prompt surgical intervention are essential for favorable outcomes.</p>","PeriodicalId":48113,"journal":{"name":"International Journal of Surgery Case Reports","volume":"138 10","pages":"4365-4369"},"PeriodicalIF":0.6,"publicationDate":"2026-09-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13652428/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149169652","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
The vertebral axis in retroperitoneal fetus in fetu: a case report on the definitive diagnosis. 腹膜后胎儿椎轴畸形一例确诊。
IF 0.6
International Journal of Surgery Case Reports Pub Date : 2026-09-14 eCollection Date: 2026-10-01 DOI: 10.1097/RC9.0000000000001000
Mostafa Abrishami, Hossein Savadi, Armin Vahabi Sani, Afsaneh Hokmabadi, Ali Samady Khanghah, Khashayar Atqiaee
{"title":"The vertebral axis in retroperitoneal fetus in fetu: a case report on the definitive diagnosis.","authors":"Mostafa Abrishami, Hossein Savadi, Armin Vahabi Sani, Afsaneh Hokmabadi, Ali Samady Khanghah, Khashayar Atqiaee","doi":"10.1097/RC9.0000000000001000","DOIUrl":"https://doi.org/10.1097/RC9.0000000000001000","url":null,"abstract":"<p><strong>Background: </strong>Fetus in fetu (FIF) is an exceptionally rare congenital anomaly (1 in 500 000 live births) involving a fetiform mass within a host twin. It is characterized by tissue organization around a vertebral axis rather than the mere presence of three germ layers. Distinguishing retroperitoneal FIF from a mature teratoma is critical, given the latter's malignant potential.</p><p><strong>Case presentation: </strong>A 4-year-old boy presented with progressive abdominal distension. Imaging revealed a 150 × 100 × 136 mm retroperitoneal mass containing calcifications and axial structures, suggesting FIF. The patient underwent a successful <i>en bloc</i> complete excision via a transverse infraumbilical laparotomy without capsular breach. Histopathology confirmed the diagnosis by identifying vertebral bodies, limb-bud-like structures, and tissues from all three germ layers, with a complete absence of immature or malignant elements. The boy's recovery was uneventful, and follow-up confirmed normal alpha-fetoprotein (AFP) levels with no recurrence.</p><p><strong>Conclusions: </strong>Distinguishing FIF from mature teratomas relies on identifying a vertebral axis and organized internal structures. Complete surgical excision is the definitive treatment and is associated with an excellent prognosis. While standard clinical follow-up is sufficient for completely excised, mature FIFs, long-term AFP surveillance may be considered, particularly in cases exhibiting immature elements with malignant potential.</p>","PeriodicalId":48113,"journal":{"name":"International Journal of Surgery Case Reports","volume":"138 10","pages":"4527-4531"},"PeriodicalIF":0.6,"publicationDate":"2026-09-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13652471/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149168349","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Limb salvage for an aorto-bifemoral vascular graft infection using multilayer negative pressure wound therapy: a case report. 应用多层负压创面治疗主动脉双侧血管移植感染保肢1例。
IF 0.6
International Journal of Surgery Case Reports Pub Date : 2026-09-14 eCollection Date: 2026-10-01 DOI: 10.1097/RC9.0000000000000998
Giorgio Caddia, Andrea Magi, Salvatora Dettori, Chiara Santoro, Giulio J Jaber, Corrado Rubino
{"title":"Limb salvage for an aorto-bifemoral vascular graft infection using multilayer negative pressure wound therapy: a case report.","authors":"Giorgio Caddia, Andrea Magi, Salvatora Dettori, Chiara Santoro, Giulio J Jaber, Corrado Rubino","doi":"10.1097/RC9.0000000000000998","DOIUrl":"https://doi.org/10.1097/RC9.0000000000000998","url":null,"abstract":"<p><strong>Introduction: </strong>Exposure and infection of aorto-bifemoral grafts are severe complications associated with a high risk of catastrophic hemorrhage and limb loss.</p><p><strong>Case presentation: </strong>A 71-year-old man presented with rupture of a femoral anastomotic pseudoaneurysm after previous aorto-bifemoral bypass. Following emergency vascular reconstruction, extensive debridement resulted in a large groin defect with exposure of the vascular graft and distal anastomotic sites, complicated by infection and multi-organ failure. In the absence of viable reconstructive options, low-pressure negative-pressure wound therapy (NPWT) was initiated using a customized three-layer protective interface incorporating a silver-impregnated non-adherent dressing placed directly over the graft. Progressive granulation tissue formation allowed staged wound closure and complete healing without flap reconstruction. At follow-up, no clinical evidence of recurrent infection was observed.</p><p><strong>Clinical discussion: </strong>NPWT over exposed vascular grafts remains controversial because of the risk of catastrophic hemorrhage. In this patient, extensive soft-tissue destruction and unfavorable vascular anatomy created a true \"no-option\" reconstructive scenario, making low-pressure NPWT followed by staged wound closure the only feasible limb- and graft-preserving strategy. The customized three-layer protective interface was designed to combine local antimicrobial activity with mechanical protection of the exposed graft.</p><p><strong>Conclusion: </strong>This case suggests that a tailored three-layer NPWT protective interface incorporating a silver-impregnated dressing may represent a useful adjunct to optimize local infection control in exposed vascular grafts. In carefully selected patients without reconstructive options, low-pressure NPWT followed by staged wound closure may represent a valuable limb- and graft-preserving strategy.</p>","PeriodicalId":48113,"journal":{"name":"International Journal of Surgery Case Reports","volume":"138 10","pages":"4413-4417"},"PeriodicalIF":0.6,"publicationDate":"2026-09-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13652429/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149170461","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
A case of giant cholangioblastic cholangiocarcinoma (12 kg) with left hepatectomy and hilar reconstruction: a surgical and pathological challenge. 巨大胆管母细胞胆管癌(12公斤)左肝切除及肝门重建1例:手术及病理挑战。
IF 0.6
International Journal of Surgery Case Reports Pub Date : 2026-09-11 eCollection Date: 2026-10-01 DOI: 10.1097/RC9.0000000000000956
Doaa Abdulaziz, Navneet Tiwari, Akshadar Koneti, Viktorija Nara, Jennifer Watkins, Joerg Matthias Pollok
{"title":"A case of giant cholangioblastic cholangiocarcinoma (12 kg) with left hepatectomy and hilar reconstruction: a surgical and pathological challenge.","authors":"Doaa Abdulaziz, Navneet Tiwari, Akshadar Koneti, Viktorija Nara, Jennifer Watkins, Joerg Matthias Pollok","doi":"10.1097/RC9.0000000000000956","DOIUrl":"https://doi.org/10.1097/RC9.0000000000000956","url":null,"abstract":"<p><strong>Introduction: </strong>Cholangioblastic cholangiocarcinoma is a rare variant of intrahepatic cholangiocarcinoma affecting young adults. Fewer than 30 cases have been reported. We describe a giant tumor in a 32-year-old man with morbid obesity (BMI of 54).</p><p><strong>Case presentation: </strong>The patient presented with a 34 cm mass, pain, persistent vomiting, inability to tolerate oral intake, and a 30 kg weight loss. Biopsy confirmed cholangioblastic intrahepatic cholangiocarcinoma. As mass-effect symptoms and malnutrition precluded initial systemic treatment, he underwent high-risk palliative cytoreduction. A fresh 12 kg tumor was removed by left hepatectomy. Inadvertent hilar stapling in severely distorted anatomy required immediate portal vein and hepatic artery reconstruction and Roux-en-Y hepaticojejunostomy. Hepatic inflow was compromised for approximately 75 minutes. Estimated blood loss was 20 L. He was discharged on postoperative day 14.</p><p><strong>Pathology: </strong>The specimen measured 53 × 33.5 × 22 cm and weighed 7.914 kg after fixation. Histology showed extensive venous and lymphovascular invasion. An involved transection margin and residual intrahepatic and nodal disease constituted an R2 resection; six of nine nodes were metastatic (pT2, pN1).</p><p><strong>Discussion: </strong>This case illustrates the diagnostic, surgical, and anesthetic hazards of palliative resection for a giant hilar tumor, including the need for anatomical confirmation before hilar division and for preparedness for vascular reconstruction and massive hemorrhage.</p><p><strong>Conclusion: </strong>Palliative cytoreduction relieved obstruction and enabled systemic therapy. The procedure was neither radical nor curative, and its potential symptomatic benefit must be balanced against substantial operative risk.</p>","PeriodicalId":48113,"journal":{"name":"International Journal of Surgery Case Reports","volume":"138 10","pages":"4309-4314"},"PeriodicalIF":0.6,"publicationDate":"2026-09-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13652379/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149167676","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Staged surgical management of advanced MRONJ in a nonagenarian with severe infection: extensive segmental mandibulectomy and reconstruction. 高龄严重感染的晚期MRONJ分阶段手术治疗:广泛的下颌节段切除术和重建。
IF 0.6
International Journal of Surgery Case Reports Pub Date : 2026-09-11 eCollection Date: 2026-10-01 DOI: 10.1097/RC9.0000000000000988
Miki Yamada, Yosuke Iijima, Miyabi Mori, Shunsuke Hino, Norio Horie, Takahiro Kaneko
{"title":"Staged surgical management of advanced MRONJ in a nonagenarian with severe infection: extensive segmental mandibulectomy and reconstruction.","authors":"Miki Yamada, Yosuke Iijima, Miyabi Mori, Shunsuke Hino, Norio Horie, Takahiro Kaneko","doi":"10.1097/RC9.0000000000000988","DOIUrl":"https://doi.org/10.1097/RC9.0000000000000988","url":null,"abstract":"<p><strong>Introduction: </strong>The management of advanced medication-related osteonecrosis of the jaw (MRONJ) in very elderly patients remains controversial because of concerns regarding perioperative risk. This report describes the clinical course and outcome of staged surgical management in a nonagenarian patient with advanced MRONJ complicated by severe infection.</p><p><strong>Methods: </strong>A 92-year-old woman with Stage 3 MRONJ, according to the 2022 American Association of Oral and Maxillofacial Surgeons Position Paper, presented with extensive mandibular destruction and severe infection accompanied by a marked systemic inflammatory response. Initial management included airway protection, incision and drainage, and systemic infection control measures. After resolution of systemic inflammation, definitive surgical treatment was performed based on disease severity, the patient's general condition, and treatment preference.</p><p><strong>Results: </strong>A segmental mandibulectomy and reconstruction with a titanium reconstruction plate were performed under general anesthesia. The postoperative course was uneventful, and oral intake was resumed successfully. Imaging confirmed restoration of mandibular continuity. Over a 4-year follow-up period, a fracture of the reconstruction plate occurred secondary to trauma, but no recurrence of MRONJ was observed.</p><p><strong>Conclusion: </strong>Staged management with initial infection control followed by definitive surgical resection was safely performed in a carefully selected nonagenarian patient with Stage 3 MRONJ. Reports of such extensive surgical management in nonagenarian patients remain extremely limited. Surgical decision-making should be based on disease severity and systemic condition rather than chronological age alone.</p>","PeriodicalId":48113,"journal":{"name":"International Journal of Surgery Case Reports","volume":"138 10","pages":"4462-4467"},"PeriodicalIF":0.6,"publicationDate":"2026-09-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13652469/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149170432","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Non-ampullary duodenal mixed neuroendocrine-non-neuroendocrine neoplasm initially diagnosed as adenocarcinoma: a case report. 最初诊断为腺癌的非壶腹十二指肠混合神经内分泌-非神经内分泌肿瘤1例。
IF 0.6
International Journal of Surgery Case Reports Pub Date : 2026-09-11 eCollection Date: 2026-10-01 DOI: 10.1097/RC9.0000000000000967
Maho Takuwa, Yukinori Yamagata, Toshihiro Haga, Hirokazu Shoji
{"title":"Non-ampullary duodenal mixed neuroendocrine-non-neuroendocrine neoplasm initially diagnosed as adenocarcinoma: a case report.","authors":"Maho Takuwa, Yukinori Yamagata, Toshihiro Haga, Hirokazu Shoji","doi":"10.1097/RC9.0000000000000967","DOIUrl":"https://doi.org/10.1097/RC9.0000000000000967","url":null,"abstract":"<p><strong>Introduction: </strong>Mixed neuroendocrine-non-neuroendocrine neoplasms (MiNENs) are rare tumors composed of neuroendocrine and non-neuroendocrine components. Their occurrence in the duodenum is extremely uncommon. Preoperative diagnosis is difficult because the neuroendocrine component may not be detected in biopsy specimens. We report a rare case of non-ampullary duodenal MiNEN initially diagnosed as adenocarcinoma.</p><p><strong>Case presentation: </strong>A 56-year-old man was found to have a tumor in the duodenal bulb during routine endoscopic examination. Biopsy revealed moderately to poorly differentiated adenocarcinoma, and computed tomography demonstrated bulky regional lymph node metastasis without distant disease. Neoadjuvant chemotherapy with S-1 plus oxaliplatin was administered based on treatment strategies used for advanced gastric cancer in Japan. Post-treatment evaluation showed stable disease, and pancreaticoduodenectomy with regional lymph node dissection achieved R0 resection. Histopathological examination revealed both adenocarcinoma and neuroendocrine carcinoma (NEC) components, leading to a diagnosis of MiNEN. Lymph node metastases demonstrated component-specific heterogeneity, with NEC involvement in station No. 8a and adenocarcinoma in the remaining metastatic nodes. Postoperative platinum-based chemotherapy was administered, and the patient remains recurrence-free at 16 months after surgery.</p><p><strong>Discussion: </strong>Duodenal MiNEN presents diagnostic and therapeutic challenges because biopsy specimens may fail to detect the neuroendocrine component preoperatively. In the present case, component-specific lymph node metastases suggested biological heterogeneity and may have influenced treatment selection.</p><p><strong>Conclusion: </strong>Non-ampullary duodenal MiNEN is a rare and diagnostically challenging disease. This case highlights the importance of accurate pathological evaluation and multidisciplinary management.</p>","PeriodicalId":48113,"journal":{"name":"International Journal of Surgery Case Reports","volume":"138 10","pages":"4320-4324"},"PeriodicalIF":0.6,"publicationDate":"2026-09-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13652443/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149167522","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Complicated hepatic cyst with an incidental pelvic hydatid cyst: a case report. 并发肝囊肿伴盆腔包虫病1例。
IF 0.6
International Journal of Surgery Case Reports Pub Date : 2026-09-11 eCollection Date: 2026-10-01 DOI: 10.1097/RC9.0000000000000973
Nawwar Shaheen, Samer Sara
{"title":"Complicated hepatic cyst with an incidental pelvic hydatid cyst: a case report.","authors":"Nawwar Shaheen, Samer Sara","doi":"10.1097/RC9.0000000000000973","DOIUrl":"https://doi.org/10.1097/RC9.0000000000000973","url":null,"abstract":"<p><strong>Introduction: </strong>Hydatid disease, a zoonosis caused by <i>Echinococcus granulosus</i>, most commonly affects the liver and lungs. Primary pelvic localization is exceptionally rare (< 2% of cases) and poses significant diagnostic challenges, particularly in patients without classic epidemiological risk factors. This case report highlights the complexities of managing such an atypical presentation.</p><p><strong>Case presentation: </strong>A 56-year-old Arab man from an urban area, with a history of mechanical aortic valve replacement and treatment with warfarin, presented with acute abdominal pain and fever. Imaging revealed a complicated, likely infected hepatic cyst in segment IV and an incidental presacral cystic lesion. Serology was positive for <i>E. granulosus</i>. Emergency laparotomy confirmed that both cysts were hydatid in origin. Management included warfarin reversal, surgical evacuation of both cysts with scolicidal irrigation, and post-operative albendazole therapy. The patient remained healthy, with a good recovery at the 6-month follow-up.</p><p><strong>Clinical discussion: </strong>The concurrent presence of a symptomatic hepatic cyst and an asymptomatic primary pelvic cyst is rare. This case underscores that hydatid disease can occur in urban patients without a typical exposure history, necessitating a high index of suspicion based on imaging. The acute presentation due to cyst superinfection required urgent surgical intervention. Successful management hinges on meticulous surgical technique to prevent spillage, combined with adjunctive anthelmintic therapy. Long-term surveillance is essential to monitor for recurrence.</p><p><strong>Conclusion: </strong>Hydatid disease should be considered in the differential diagnosis of cystic abdominal masses even in low-risk populations. A structured approach involving timely imaging, careful surgical management, peri-operative albendazole, and prolonged follow-up may be crucial for optimal outcomes in complex, multi-focal cases.</p>","PeriodicalId":48113,"journal":{"name":"International Journal of Surgery Case Reports","volume":"138 10","pages":"4436-4440"},"PeriodicalIF":0.6,"publicationDate":"2026-09-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13652442/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149170801","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Holt-Oram syndrome presenting with failure to thrive due to a hemodynamically significant patent ductus arteriosus: the importance of early cardiac evaluation in infants with radial ray abnormalities. Holt-Oram综合征表现为由于血流动力学上显著的动脉导管未闭而导致发育不全:早期心脏评估对桡骨射线异常婴儿的重要性。
IF 0.6
International Journal of Surgery Case Reports Pub Date : 2026-09-11 eCollection Date: 2026-10-01 DOI: 10.1097/RC9.0000000000000968
Rahaf Ibrahim, Alwaleed Al-Dairy, Raghad Ibrahim, Samir Srour
{"title":"Holt-Oram syndrome presenting with failure to thrive due to a hemodynamically significant patent ductus arteriosus: the importance of early cardiac evaluation in infants with radial ray abnormalities.","authors":"Rahaf Ibrahim, Alwaleed Al-Dairy, Raghad Ibrahim, Samir Srour","doi":"10.1097/RC9.0000000000000968","DOIUrl":"https://doi.org/10.1097/RC9.0000000000000968","url":null,"abstract":"<p><strong>Background: </strong>Holt-Oram syndrome (HOS) is a rare autosomal dominant disorder caused by pathogenic variants in the TBX5 gene and characterized by radial ray malformations associated with congenital heart defects and/or cardiac conduction abnormalities. Although atrial and ventricular septal defects are the most common cardiac manifestations, other congenital cardiac lesions may occur. Early recognition of cardiac involvement is essential to prevent complications and improve outcomes.</p><p><strong>Case presentation: </strong>We report the case of an 8-month-old female infant presenting with failure to thrive, feeding difficulties, and mild respiratory distress. She had unilateral radial ray abnormalities, including complete absence of the left thumb and index finger with associated radial, carpal, and humeral hypoplasia. Physical examination revealed bounding peripheral pulses and a continuous machinery murmur. Transthoracic echocardiography demonstrated a large hemodynamically significant patent ductus arteriosus (PDA) with pulmonary hypertension and left-sided volume overload. Computed tomography angiography confirmed the diagnosis and excluded associated great vessel anomalies. Based on the characteristic upper limb abnormalities, congenital heart disease, and sinus bradycardia, a clinical diagnosis of HOS was established. Surgical ligation of the PDA was successfully performed without complications. At 6-month follow-up, the patient showed complete resolution of heart failure symptoms, satisfactory catch-up growth, and normal postoperative cardiac findings.</p><p><strong>Discussion: </strong>This case expands the recognized cardiac phenotype of HOS by describing a hemodynamically significant PDA presenting with failure to thrive and pulmonary hypertension. It also illustrates that the severity of skeletal abnormalities does not predict the type or severity of associated cardiac disease, emphasizing the need for comprehensive cardiovascular assessment in all infants with radial ray abnormalities.</p><p><strong>Conclusion: </strong>Early recognition of HOS and prompt cardiovascular evaluation are critical for identifying treatable congenital heart defects. Timely intervention, combined with multidisciplinary long-term care, can significantly improve clinical, functional, and developmental outcomes in affected children.</p>","PeriodicalId":48113,"journal":{"name":"International Journal of Surgery Case Reports","volume":"138 10","pages":"4341-4345"},"PeriodicalIF":0.6,"publicationDate":"2026-09-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13652367/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149170838","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Adult-onset primary retroperitoneal cystic lymphangioma displacing the inferior vena cava: laparoscopic management of a triple-rarity case from Nepal and review of recent literature. 成人发病原发性腹膜后囊性淋巴管瘤移位下腔静脉:尼泊尔三罕见病例的腹腔镜治疗和近期文献回顾。
IF 0.6
International Journal of Surgery Case Reports Pub Date : 2026-09-11 eCollection Date: 2026-10-01 DOI: 10.1097/RC9.0000000000000966
Manaswi Acharya, Rajani Neupane, Suman Supratik, Ranjit Kumar Yadav, Akash Giri, Dinesh Nalbo
{"title":"Adult-onset primary retroperitoneal cystic lymphangioma displacing the inferior vena cava: laparoscopic management of a triple-rarity case from Nepal and review of recent literature.","authors":"Manaswi Acharya, Rajani Neupane, Suman Supratik, Ranjit Kumar Yadav, Akash Giri, Dinesh Nalbo","doi":"10.1097/RC9.0000000000000966","DOIUrl":"https://doi.org/10.1097/RC9.0000000000000966","url":null,"abstract":"<p><strong>Introduction and importance: </strong>Lymphangiomas are rare benign lymphatic malformations, predominantly affecting children in the head and neck region. Retroperitoneal cystic lymphangioma (RCL) accounts for fewer than 1% of all lymphangiomas. Adult-onset primary RCL is exceptionally rare, with fewer than 200 cases documented in the global literature. We report what appears to be the first such case from Nepal, based on a review of available published literature.</p><p><strong>Case presentation: </strong>A 40-year-old woman presented with right back pain, a lumbar mass, and early satiety. Tumor markers were unremarkable. Initial ultrasonography misidentified the lesion as a probable ovarian cyst. Contrast-enhanced computed tomography (CECT) revealed a septated retroperitoneal cyst of size 7.0 × 5.9 × 5.7 cm causing significant medial displacement of the inferior vena cava (IVC). Laparoscopic exploration confirmed the retroperitoneal origin. Following controlled aspiration of serous fluid, laparoscopic marsupialization was performed with meticulous IVC and ureteric preservation. Histopathology confirmed cystic lymphangioma. The patient was discharged on postoperative day 4 without complications.</p><p><strong>Clinical discussion: </strong>RCL frequently mimics ovarian or mesenteric pathology on ultrasonography, making CECT essential for operative planning. When complete excision poses prohibitive hemorrhagic risk due to IVC proximity, laparoscopic marsupialization represents a justified and safe alternative. A recurrence risk of 10-13.6% following conservative surgical management mandates structured long-term annual surveillance. Immunohistochemistry (D2-40, CD31, and CD34) is recommended for definitive diagnosis where laboratory resources permit.</p><p><strong>Conclusions: </strong>This case involving a rare tumor type, an exceptionally rare anatomical location, and an atypical adult age of onset demonstrates that laparoscopic management of RCL with major vascular displacement is feasible in resource-limited settings. At 6-month follow-up, no recurrence was detected and the IVC had returned to its anatomical position. This represents a provisional early outcome; long-term surveillance remains mandatory given the established late recurrence risk.</p>","PeriodicalId":48113,"journal":{"name":"International Journal of Surgery Case Reports","volume":"138 10","pages":"4325-4331"},"PeriodicalIF":0.6,"publicationDate":"2026-09-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13652394/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"149166242","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Locked patellar dislocation due to osteophytes: two case reports with a focused literature review. 骨赘引起的锁定髌骨脱位:两例报告并集中文献回顾。
IF 0.6
International Journal of Surgery Case Reports Pub Date : 2026-09-11 eCollection Date: 2026-10-01 DOI: 10.1097/RC9.0000000000000936
Ngoc Quyen Le, Trong Xuan Cuong Tran
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