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赫尔曼斯基-普德拉克综合征所致肉芽肿性结肠炎

Granulomatous Colitis Due to Hermansky-Pudlak Syndrome.

作者信息

Koulali Hajar, Azzmouri Samira, Tajir Mariam, Zerrouki Khawla, Haloui Anass, Elmqaddem Ouiam, Zazour Abdelkrim, Ismaili Zahi, Kharrasse Ghizlane

机构信息

Department of Hepato-Gastroenterology, Mohammed VI University Hospital, Oujda, Morocco.

Digestive Diseases Research Laboratory (DSRL), Faculty of Medicine and Pharmacy, Mohammed First University, Oujda, Morocco.

出版信息

ACG Case Rep J. 2024 Oct 31;11(11):e01477. doi: 10.14309/crj.0000000000001477. eCollection 2024 Nov.

Abstract

Hermansky-Pudlak syndrome (HPS) is a rare genetic disorder characterized by oculocutaneous albinism, bleeding diathesis, and multiorgan involvement. Granulomatous enterocolitis may occur in a subset of patients. Distinguishing HPS from other diseases such as Crohn's disease can be challenging, and managing HPS-associated colitis is complex. Recent reports suggest potential efficacy of infliximab in treating HPS-related granulomatous colitis. Here, we document the case of a 27-year-old patient with genetically confirmed HPS type 1, presenting with granulomatous colitis and successfully treated with corticosteroids and infliximab.

摘要

Hermansky-Pudlak综合征(HPS)是一种罕见的遗传性疾病,其特征为眼皮肤白化病、出血素质和多器官受累。部分患者可能会出现肉芽肿性小肠结肠炎。将HPS与其他疾病(如克罗恩病)区分开来可能具有挑战性,并且管理与HPS相关的结肠炎很复杂。最近的报告表明英夫利昔单抗在治疗HPS相关的肉芽肿性结肠炎方面可能有效。在此,我们记录了一例27岁经基因确诊为1型HPS的患者,该患者患有肉芽肿性结肠炎,并成功接受了皮质类固醇和英夫利昔单抗治疗。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d20a/11527421/fd7493950c98/ac9-11-e01477-g001.jpg

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