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亨廷顿舞蹈病修饰基因的探索——单基因疾病的多因素特征

The search for modifier genes in Huntington disease - Multifactorial aspects of a monogenic disorder.

作者信息

Arning Larissa

机构信息

Department of Human Genetics, Ruhr-University Bochum, Germany.

出版信息

Mol Cell Probes. 2016 Dec;30(6):404-409. doi: 10.1016/j.mcp.2016.06.006. Epub 2016 Jul 12.

DOI:10.1016/j.mcp.2016.06.006
PMID:27417534
Abstract

It is becoming increasingly evident that the underlying mutation of a single locus is often insufficient for the prediction of the comprehensive phenotype in human Mendelian disorders, implicating that there is no clear distinction between monogenic and complex traits. By definition, monogenic traits show a classic pattern of inheritance and are strongly influenced by variation within a single gene. However, many Mendelian traits that result in genetic disorders can have phenotypes that differ in subtle or profound ways such as severity, onset age and other associated phenotypic characteristics. Among the factors that may explain these differences in disease expression are modifier genes. This review focuses on the role of modifier genes using the example of Huntington Disease (HD), an autosomal dominantly transmitted, progressive neurodegenerative disorder. The advantages and limitations of candidate gene approaches versus genome-wide association studies (GWAS) as well as its implications for diagnostic, prognostic, and therapeutic interventions are discussed.

摘要

越来越明显的是,在人类孟德尔疾病中,单个基因座的潜在突变往往不足以预测综合表型,这意味着单基因性状和复杂性状之间没有明确的区别。根据定义,单基因性状表现出经典的遗传模式,并受到单个基因内变异的强烈影响。然而,许多导致遗传疾病的孟德尔性状可能具有在严重程度、发病年龄和其他相关表型特征等方面存在细微或显著差异的表型。修饰基因可能是解释这些疾病表达差异的因素之一。本综述以亨廷顿舞蹈病(HD)为例,重点探讨修饰基因的作用,HD是一种常染色体显性遗传的进行性神经退行性疾病。文中讨论了候选基因方法与全基因组关联研究(GWAS)的优缺点及其对诊断、预后和治疗干预的影响。

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