维普中文期刊产品整合服务

Acquired segmental colonic hypoganglionosis in an adult Caucasian male:A case report

查看全文 作　　者：Allan MF [1]Kwok;Andrew B [1]Still;Kimberly [2]Hart 高影响力作者 机构地区：[1]Department of Surgery, Wollongong Hospital;[2]Department of Anatomical Pathology, Wollongong Hospital高影响力机构 出　　处：《World Journal of Gastrointestinal Surgery》索引2019年第11卷第2期,共11页高影响力期刊 摘　　要：BACKGROUND Hypoganglionosis is a rare condition that most often presents with abnormal gastrointestinal transit and usually arises in early childhood or adolescence. Two types have been described(Type I and Type II). The adult-onset form(acquired hypoganglionosis) is extremely uncommon and is thought to arise due to cellular remodelling as a result of chronic inflammation. It differs from Hirschprung's disease in that there is a reduction in ganglion cells in the colonic neural plexuses as opposed to being completely absent.CASE SUMMARY A 31 year-old male presented to hospital with recurrent abdominal pain and vomiting over thirteen months. Abdominal computed tomography scans demonstrated thickening and stranding affecting the transverse, descending and sigmoid colon. Endoscopic appearances were non-specific but confirmed a mixed picture of mucosal inflammation and necrosis in various stages of healing.Numerous investigations were performed to elucidate an underlying aetiology but neither an infective nor ischaemic cause could be proven. Biopsy features were not typical of inflammatory bowel disease. Due to persistence of his symptoms and failure of medical management, a segmental colectomy was performed. Histological examination of the specimen revealed an unexpected finding of segmental hypoganglionosis. Complete surgical excision of the diseased segment of colon was curative and since his operation the patient has had no recurrence of symptoms requiring hospitalisation.CONCLUSION Our case serves to raise awareness of acquired hypoganglionosis as a rare condition that can result from chronic colitis. 关 键 词：ACQUIRED hypoganglionosis Hirschprung’s disease Adult GANGLION cells ACETYLCHOLINESTERASE CALRETININ Case report
相关文献

参考文献(19)

引证文献(5)

网站首页 | 关于我们 | 联系我们 | 产品服务 | 客服中心 | 广告服务 | 版权声明 | 网站联盟 | 友情链接 | 售卡网点

版权所有© 渝B2-20050021-1　渝公网安备 50019002500403号　违法和不良信息举报中心

互联网出版许可证 新出网证(渝)字10号 全国400电话 - 免长途话费