Chan Esther Ern Hwei, Merchant Khurshid, Othman Mohd Yusran, Laksmi Narasimhan Kannan
Department of Paediatric Surgery, KK Women's and Children's Hospital, Singapore
Department of Pathology and Laboratory Medicine, KK Women's and Children's Hospital, Singapore.
BMJ Case Rep. 2023 Apr 25;16(4):e252484. doi: 10.1136/bcr-2022-252484.
Hirschsprung's disease (HD) is one of the most well-known gastrointestinal motility disorders. Diagnosis and management of other lesser-known motility disorders are often challenging and tedious. We describe a teenager who was severely constipated from birth and needed intensive care admissions for life-threatening enterocolitis. She also had concomitant anal stenosis. Several rectal biopsies were unable to yield a conclusive diagnosis. Surgical level of resection had to be identified based on the motility of the bowel as determined by transit studies using oral ingestion of a milk feed labelled with Technetium-99m colloid. After completion of all operative stages, histopathological examination of the excised specimens concluded that she had short-segment HD associated with reduced interstitial cells of Cajal in the large bowel. She is currently continent, evacuating voluntarily approximately four times a day and is relieved of all her symptoms.
先天性巨结肠(HD)是最广为人知的胃肠动力障碍之一。诊断和处理其他不太知名的动力障碍往往具有挑战性且繁琐。我们描述了一名自出生就严重便秘且因危及生命的小肠结肠炎而需要重症监护的青少年。她还伴有肛门狭窄。多次直肠活检未能得出确定性诊断。必须根据口服标记有锝-99m胶体的奶液进行转运研究确定的肠道动力来确定手术切除水平。在完成所有手术阶段后,对切除标本的组织病理学检查得出结论,她患有短段先天性巨结肠,伴有大肠中Cajal间质细胞减少。她目前排便正常,每天自主排便约四次,所有症状均已缓解。