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ZFYVE19基因突变:进行性家族性肝内胆汁淤积症的一种新型变体。

ZFYVE19 gene mutation: A novel variant of progressive familial intrahepatic cholestasis.

作者信息

Ben Sabbahia Dalal, Atrasssi Meriem, Bennani Nissrine, Benmoussa Abdelhakim, Abkari Abdelhak

机构信息

The Department of Pediatrics III, Unit of Gastroenterology and Hepatology Pediatric, Abderrahim Harrouchi Children Hospital Ibn Rochd University Hospital Casablanca Morocco.

Faculty of Medicine and Pharmacy Hassan II University of Casablanca Casablanca Morocco.

出版信息

JPGN Rep. 2024 Aug 6;5(4):552-556. doi: 10.1002/jpr3.12111. eCollection 2024 Nov.

Abstract

A recent nonsyndromic phenotype, newly linked to mutations in the ZFYVE19 gene, is characterized by the appearance of cholestasis accompanied by an increase in serum gamma-glutamyltranspeptidase (GGT) from infancy or early childhood. Affected individuals generally present with hepatosplenomegaly and may develop portal hypertension. The disease is thought to be the result of cholangiocyte-specific ciliary dysfunction, indicating a ciliopathy that appears to be limited to the liver. Here, we describe the case of an infant born to first-degree consanguineous parents, in whom neonatal cholestasis accompanied by elevated GGT led to the discovery of a ZFYVE19 deficiency. The diagnosis was established following an in-depth analysis of the complete exome sequencing.

摘要

最近发现一种与ZFYVE19基因突变相关的非综合征性表型,其特征是自婴儿期或幼儿期起出现胆汁淤积,并伴有血清γ-谷氨酰转肽酶(GGT)升高。受影响个体通常表现为肝脾肿大,并可能发展为门静脉高压症。该疾病被认为是胆管细胞特异性纤毛功能障碍的结果,表明这是一种似乎仅限于肝脏的纤毛病。在此,我们描述了一例由一级近亲父母所生婴儿的病例,该婴儿的新生儿胆汁淤积伴GGT升高,从而发现了ZFYVE19缺乏症。通过对全外显子组测序的深入分析后确诊。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c3e8/11600356/9352808175a8/JPR3-5-552-g003.jpg

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