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心脏钠离子通道突变:为何有如此多的表型?

Cardiac Sodium Channel Mutations: Why so Many Phenotypes?

机构信息

The Warren Alpert Medical School of Brown University, Providence, RI, United States.

出版信息

Curr Top Membr. 2016;78:513-59. doi: 10.1016/bs.ctm.2015.12.004. Epub 2016 Mar 14.

Abstract

The cardiac Na(+) channel (Nav1.5) conducts a depolarizing inward Na(+) current that is responsible for the generation of the upstroke Phase 0 of the action potential. In heart tissue, changes in Na(+) currents can affect conduction velocity and impulse propagation. The cardiac Nav1.5 is also involved in determination of the action potential duration, since some channels may reopen during the plateau phase, generating a persistent or late inward current. Mutations of cardiac Nav1.5 can induce gain or loss of channel function because of an increased late current or a decrease of peak current, respectively. Gain-of-function mutations cause Long QT syndrome type 3 and possibly atrial fibrillation, while loss-of-function channel mutations are associated with a wider variety of phenotypes, such as Brugada syndrome, cardiac conduction disease, dilated cardiomyopathy, and sick sinus node syndrome. The penetrance and phenotypes resulting from Nav1.5 mutations also vary with age, gender, body temperature, circadian rhythm, and between regions of the heart. This phenotypic variability makes it difficult to correlate genotype-phenotype. We propose that mutations are only one contributor to the phenotype and additional modifications on Nav1.5 lead to the phenotypic variability. Possible modifiers include other genetic variations and alterations in the life cycle of Nav1.5 such as gene transcription, RNA processing, translation, posttranslational modifications, trafficking, complex assembly, and degradation. In this chapter, we summarize potential modifiers of cardiac Nav1.5 that could help explain the clinically observed phenotypic variability. Consideration of these modifiers could help improve genotype-phenotype correlations and lead to new therapeutic strategies.

摘要

心脏钠离子通道(Nav1.5)传导去极化内向钠离子电流,该电流负责产生动作电位的上升相 0 期。在心脏组织中,钠离子电流的变化会影响传导速度和冲动传播。心脏 Nav1.5 还参与动作电位时程的确定,因为在平台期某些通道可能会重新开放,产生持续或晚期内向电流。心脏 Nav1.5 的突变可分别通过增加晚期电流或减少峰值电流而导致通道功能的获得或丧失。功能获得性突变可引起长 QT 综合征 3 型,可能还会引起心房颤动,而功能丧失性通道突变与更广泛的表型相关,如 Brugada 综合征、心脏传导疾病、扩张型心肌病和病态窦房结综合征。Nav1.5 突变引起的外显率和表型也随年龄、性别、体温、昼夜节律和心脏不同区域而变化。这种表型变异性使得基因型-表型相关性变得困难。我们提出,突变只是表型的一个贡献因素,Nav1.5 上的其他修饰导致了表型的变异性。可能的修饰因子包括其他遗传变异和 Nav1.5 生命周期的改变,如基因转录、RNA 加工、翻译、翻译后修饰、运输、复合物组装和降解。在本章中,我们总结了心脏 Nav1.5 的潜在修饰因子,这些因子可能有助于解释临床观察到的表型变异性。考虑这些修饰因子可以帮助改善基因型-表型相关性,并导致新的治疗策略。

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