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心脏钠通道突变:为何会有如此多的表型?

Cardiac sodium channel mutations: why so many phenotypes?

作者信息

Liu Man, Yang Kai-Chien, Dudley Samuel C

机构信息

Warren Alpert Medical School, Brown University, 593 Eddy Street, APC730, Providence, RI 02903, USA.

出版信息

Nat Rev Cardiol. 2014 Oct;11(10):607-15. doi: 10.1038/nrcardio.2014.85. Epub 2014 Jun 24.

Abstract

Mutations of the cardiac sodium channel (Nav1.5) can induce gain or loss of channel function. Gain-of-function mutations can cause long QT syndrome type 3 and possibly atrial fibrillation, whereas loss-of-function mutations are associated with a variety of phenotypes, such as Brugada syndrome, cardiac conduction disease, sick sinus syndrome, and possibly dilated cardiomyopathy. The phenotypes produced by Nav1.5 mutations vary according to the direct effect of the mutation on channel biophysics, but also with age, sex, body temperature, and between regions of the heart. This phenotypic variability makes genotype-phenotype correlations difficult. In this Perspectives article, we propose that phenotypic variability not ascribed to mutation-dependent changes in channel function might be the result of additional modifiers of channel behaviour, such as other genetic variation and alterations in transcription, RNA processing, translation, post-translational modifications, and protein degradation. Consideration of these modifiers might help to improve genotype-phenotype correlations and lead to new therapeutic strategies.

摘要

心脏钠通道(Nav1.5)的突变可导致通道功能的获得或丧失。功能获得性突变可引起3型长QT综合征并可能导致心房颤动,而功能丧失性突变则与多种表型相关,如Brugada综合征、心脏传导疾病、病态窦房结综合征以及可能的扩张型心肌病。Nav1.5突变产生的表型不仅根据突变对通道生物物理学的直接影响而变化,还随年龄、性别、体温以及心脏不同区域而变化。这种表型变异性使得基因型与表型的相关性难以确定。在这篇观点文章中,我们提出,那些不能归因于通道功能突变依赖性变化的表型变异性,可能是通道行为的其他调节因素的结果,如其他基因变异以及转录、RNA加工、翻译、翻译后修饰和蛋白质降解的改变。考虑这些调节因素可能有助于改善基因型与表型的相关性,并带来新的治疗策略。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1512/4878851/6e5c6d95c0d9/nihms783702f1.jpg

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Cardiac sodium channel mutations: why so many phenotypes?心脏钠通道突变:为何会有如此多的表型?
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