[常染色体显性阿尔波特综合征发病机制的新遗传变异:家族病例报告]。

Q4 Medicine
Sandra La Rosa, Stefania Pesce, Simona Lombardo, Marilena C Fiorino, Antonio Granata
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引用次数: 0

摘要

Alport综合征是一种以血尿、蛋白尿和进行性肾衰竭为特征的遗传性疾病,常伴有肾外表现。COL4A5基因的致病变异与x连锁Alport综合征有关,而COL4A3和COL4A4基因的致病变异与常染色体隐性(AR)或显性(AD)形式有关。这种疾病的特点是与不同的基因和不同的突变相关的相当大的表型变异性,因此症状根据病例以不同的频率表现出来。近年来,由于下一代基因测序(NGS)技术的发展,发现了一种常染色体显性的阿尔波特综合征,这使得发现未知的阿尔波特综合征遗传变异成为可能。我们研究的家族存在COL4A3基因的杂合性改变(c.1029+5G>A与MAF 0和c.3211-7A>G与MAF 1:10万),MTHFR基因的杂合性改变(C677T和A1298C)和PAI-1基因的纯合性改变。遗传数据库(ClinVar)显示,c.3211-7A>G变异似乎是良性的,而内含子变异c.1029+5G>A(由外显子跳变引起)由于其特征以及它与家族内表型共分离的事实,可以归类为致病性的。其中一名姐妹的组织学资料强调了离散性全局肾小球硬化的存在,超微结构检查显示肾小球基底膜变薄。COL4A3基因的新突变变体可能在慢性肾脏疾病的发展中发挥风险变异的作用。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
[New Genetic Variants Involved in the Pathogenesis of Autosomal Dominant Alport Syndrome: A Familial Case Report].

Alport syndrome is a hereditary disorder characterized by hematuria, proteinuria and progressive renal failure, frequently associated with extrarenal manifestations. The pathogenic variants of the COL4A5 gene are associated with X-linked Alport syndrome while those of the COL4A3 and COL4A4 genes are associated with the autosomal recessive (AR) or dominant (AD) form. The disease is characterized by considerable phenotypic variability linked to the different genes involved and the different mutations present, so the symptoms manifest themselves in different frequencies depending on the case. The existence of an autosomal dominant form of Alport syndrome has been identified in recent years thanks to next generation gene sequencing (NGS) techniques which have made it possible to highlight unknown genetic variants of Alport syndrome. The family studied by us presents concomitant heterozygous alterations of the COL4A3 genes (c.1029+5G>A with MAF 0 and c.3211-7A>G with MAF 1:100000), heterozygous alterations of the MTHFR gene (both C677T and A1298C) and homozygous alteration of the PAI-1 gene. While the variant c.3211-7A>G, as shown by genetic databases (ClinVar), appears to be benign, the intronic variant c.1029+5G>A (caused by exon skipping) can be classified as pathogenic due to its characteristics and the fact that it co-segregates with the phenotype within the family. The histological data, in one of the sisters, highlighted the presence of a discrete global glomerular sclerosis and the ultrastructural investigation a thinning of the glomerular basement membrane. New mutational variants of the COL4A3 gene may play a role as risk variants for the development of chronic kidney disease.

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来源期刊
CiteScore
0.70
自引率
0.00%
发文量
62
期刊介绍: Il Giornale Italiano di Nefrologia (GIN) è la rivista di educazione continua della Società Italiana di Nefrologia SIN ed è pubblicato bimestralmente. E" il più autorevole organo di informazione nefrologia disponibile a livello nazionale. Il giornale Italiano di Nefrologia offre la più aggiornata informazione medico-scientifica rivolta al nefrologo sotto forma di rassegne, casi clinici e articoli finalizzati all’Educazione Continua in Medicina, oltre ai notiziari ed agli atti dei congressi di questa prestigiosa Società Scientifica
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