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以部分性肠梗阻为表现的炎性肌纤维母细胞瘤:一例报告

Inflammatory Myofibroblastic Tumor Presenting as a Partial Bowel Obstruction: A Case Report.

作者信息

Kaspar Kolton, Jackson Anthony, Hunt Christina M, Williams Seth, Trudeau Donovan, Mohammed Ricardo

机构信息

Department of Research, Alabama College of Osteopathic Medicine, Dothan, USA.

Department of General Surgery, Santa Rosa Medical Center, Milton, USA.

出版信息

Cureus. 2023 Mar 28;15(3):e36798. doi: 10.7759/cureus.36798. eCollection 2023 Mar.

Abstract

Intussusception, or telescoping of the bowel, is a rare condition in the adult population that can lead to serious complications, such as obstruction or ischemia. Most cases of intussusception are idiopathic and present with a pathognomonic "target sign" on imaging. Rarely, in adults, intussusceptions can be found with lead points, some of which may be neoplastic. Treatments for intussusception include air enemas or surgical intervention if enemas are unsuccessful in resolving the telescoped bowel. This case report discusses an atypical presentation of intussusception in an adult female with a "whirlpool sign" on imaging rather than the typical "target sign." She was found to have incorporation of mesenteric fat into telescoping bowel causing edema and partial bowel obstruction. The affected bowel was removed laparoscopically, and an end-to-end anastomosis was formed. Pathology of the resected bowel revealed a non-immunoreactive inflammatory myofibroblastic neoplasm as the lead point. Most inflammatory myofibroblastic tumors stain positive for desmin, smooth muscle actin, and anaplastic lymphoma kinase (ALK), whereas this patient was non-immunoreactive. The patient tolerated surgery well and is now pain-free with normal gastrointestinal function. This case report hopes to heighten awareness of atypical presentations of intussusceptions, the use of imaging to help aid in uncertain diagnoses, and the appropriate surgical treatment for symptomatic patients.

摘要

肠套叠,即肠管套叠,在成人中是一种罕见疾病,可导致严重并发症,如肠梗阻或肠缺血。大多数肠套叠病例为特发性,影像学上表现为具有诊断意义的“靶征”。在成人中,肠套叠很少伴有引导点,其中一些可能是肿瘤性的。肠套叠的治疗方法包括空气灌肠,如果灌肠未能成功解除套叠肠管,则进行手术干预。本病例报告讨论了一名成年女性肠套叠的非典型表现,其影像学表现为“漩涡征”而非典型的“靶征”。发现她的套叠肠管内有肠系膜脂肪嵌入,导致水肿和部分肠梗阻。通过腹腔镜切除受累肠管,并进行端到端吻合。切除肠管的病理显示,作为引导点的是一种非免疫反应性炎性肌纤维母细胞瘤。大多数炎性肌纤维母细胞瘤对结蛋白、平滑肌肌动蛋白和间变性淋巴瘤激酶(ALK)染色呈阳性,而该患者无免疫反应。患者手术耐受性良好,目前无痛,胃肠功能正常。本病例报告希望提高对肠套叠非典型表现、利用影像学辅助不确定诊断以及对有症状患者进行适当手术治疗的认识。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e300/10134782/1418ece8806b/cureus-0015-00000036798-i01.jpg

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