ZFYVE19基因突变:进行性家族性肝内胆汁淤积症的新变体。

JPGN reports Pub Date : 2024-08-06 eCollection Date: 2024-11-01 DOI:10.1002/jpr3.12111
Dalal Ben Sabbahia, Meriem Atrasssi, Nissrine Bennani, Abdelhakim Benmoussa, Abdelhak Abkari
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摘要

最近发现的一种与ZFYVE19基因突变有关的非综合征表型,其特征是出现胆汁淤积,并伴随婴儿期或幼儿期血清γ -谷氨酰转肽酶(GGT)升高。受影响的个体通常表现为肝脾肿大,并可能发展为门脉高压。该病被认为是胆管细胞特异性纤毛功能障碍的结果,表明纤毛病似乎仅限于肝脏。在这里,我们描述了一个一级近亲父母所生婴儿的病例,其中新生儿胆汁淤积伴随着GGT升高导致ZFYVE19缺乏症的发现。诊断是在对完整的外显子组测序进行深入分析后确定的。
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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ZFYVE19 gene mutation: A novel variant of progressive familial intrahepatic cholestasis.

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.

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