Heterotopic Cutaneous Adnexa Arising from the Ascending Colon Presenting as a Chronic Abdominal Wall Sinus: A Rare Congenital Anomaly and Diagnostic Challenge.

IF 3.8 3区 医学 Q1 MEDICINE, GENERAL & INTERNAL
Gang Xiao, Dan-Dan Li, Qiu-Shi Huang, Shan He, Yuan Liang, Jian Shen
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Abstract

A chronic abdominal wall sinus tract caused by heterotopic cutaneous adnexa arising from the colonic serosa is extremely rare and readily misdiagnosed. We report a 39-year-old woman with congenital scoliosis and a 30-year history of intermittent, odorless, non-feculent discharge from a cutaneous opening in the right lower abdominal wall; she had no prior abdominal surgery. Contrast-enhanced computed tomography (CT) revealed a non-enhancing sinus tract extending from the ascending colon toward the abdominal wall, whereas colonoscopy showed normal colonic mucosa with no fistulous communication. Laparoscopic exploration revealed a tubular structure arising from the serosal surface of the middle ascending colon that bifurcated into an abdominal wall branch and a pelvic branch, the latter not identified on preoperative imaging. Because the lesion was considered benign but preoperative imaging could not determine the depth of involvement, and an unexpected pelvic branch was found intraoperatively, a laparoscopic limited right hemicolectomy was performed with en bloc excision of both branches. Histopathological examination confirmed the absence of intestinal epithelial components, and immunohistochemistry was positive for cytokeratin 7 (CK7), GATA-binding protein 3 (GATA-3), and p63, confirming a cutaneous adnexal origin. The patient recovered uneventfully, with no recurrence at the 6-month follow-up. This case may represent a rare congenital developmental anomaly related to abnormal persistence of ectodermal components during embryologic development; however, definitive embryologic proof is lacking. It emphasizes the importance of a detailed history, the inclusion of congenital heterotopic cutaneous adnexal lesions in the differential diagnosis of refractory abdominal wall sinus tracts, and complete excision of all sinus tract branches to prevent recurrence and repeated ineffective interventions.

源自升结肠的异位皮肤附件表现为慢性腹壁窦:一种罕见的先天性异常和诊断挑战。
摘要慢性腹壁窦道是由源自结肠浆膜的异位皮肤附件所引起,极为罕见且容易误诊。我们报告一位患有先天性脊柱侧凸的39岁女性,她有30年的间歇性、无臭、无排泄物的病史,从右下腹壁的皮肤开口流出;她之前没有做过腹部手术。增强计算机断层扫描(CT)显示从升结肠向腹壁延伸的非强化窦道,而结肠镜检查显示正常结肠粘膜,无瘘连。腹腔镜检查发现一管状结构起源于中升结肠浆膜表面,分为腹壁分支和骨盆分支,后者未在术前影像学中发现。由于病变被认为是良性的,但术前影像学无法确定受累的深度,且术中发现意外的盆腔分支,因此行腹腔镜有限右半结肠切除术,并将两个分支全部切除。组织病理学检查证实肠上皮成分缺失,免疫组化检测细胞角蛋白7 (CK7)、gata结合蛋白3 (GATA-3)和p63阳性,证实其起源于皮肤附件。患者恢复平稳,随访6个月无复发。本病例可能是一种罕见的先天性发育异常,与胚胎发育过程中外胚层成分的异常持久性有关;然而,尚无明确的胚胎学证据。它强调了详细病史的重要性,包括先天性异位皮肤附件病变在难治性腹壁窦束鉴别诊断中的重要性,以及完全切除所有窦束分支以防止复发和重复无效干预。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
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来源期刊
Diagnostics
Diagnostics Biochemistry, Genetics and Molecular Biology-Clinical Biochemistry
CiteScore
4.70
自引率
8.30%
发文量
2699
审稿时长
19.64 days
期刊介绍: Diagnostics (ISSN 2075-4418) is an international scholarly open access journal on medical diagnostics. It publishes original research articles, reviews, communications and short notes on the research and development of medical diagnostics. There is no restriction on the length of the papers. Our aim is to encourage scientists to publish their experimental and theoretical research in as much detail as possible. Full experimental and/or methodological details must be provided for research articles.
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