肝糖原储存病:破解基因型-表型难题。

Arghya Samanta, Gautam Ray
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引用次数: 0

摘要

糖原储存病(GSDs)是由参与糖原产生或分解的各种酶的遗传缺陷引起的一组遗传性疾病。肝脏gsd通常具有重叠的临床特征,使得分型或预后困难。随着新一代测序技术的发展,大多数患者可获得明确的分子诊断,新的变异也越来越多地被发现。分子诊断有助于系统随访,预测并发症和预后。然而,在已发表的文献中报道的突变在种族和地理群体中表现出广泛的差异。因此,需要对各种肝脏gsd患者的自然病史、长期预后和基因型-表型相关性进行更深入的了解。考虑到肝脏gsd患者遗传谱的新证据,包括Vanduangden等人最近的研究,这篇文章旨在回顾各种肝脏gsd的各种临床亚型、基因突变谱和基因型-表型相关性。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Hepatic glycogen storage disease: Deciphering the genotype-phenotype conundrum.

Glycogen storage diseases (GSDs) are a group of inherited disorders caused by genetic defects in various enzymes involved in glycogen production or breakdown. Hepatic GSDs often have overlapping clinical features, making subtyping or prognostication difficult. With the availability and advancement of next-generation sequencing, definitive molecular diagnosis is now available for most patients, with newer variants being increasingly identified. Molecular diagnosis could help in systematic follow-up, anticipating complications and prognostications. However, the mutations reported in the published literature display wide variations across racial and geographical groups. Hence, natural history, long-term outcome, and genotype-phenotypic correlation studies in patients with various hepatic GSDs are needed for a deeper understanding. Considering the emerging evidence of genetic profiling of patients with hepatic GSDs, including the recent study by Vanduangden et al, this editorial aims to review the various clinical subtypes, the spectrum of genetic mutations, and genotype-phenotype correlations for various hepatic GSDs.

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