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罕见变异在常见疾病中有多重要?

How important are rare variants in common disease?

作者信息

Saint Pierre Aude, Génin Emmanuelle

出版信息

Brief Funct Genomics. 2014 Sep;13(5):353-61. doi: 10.1093/bfgp/elu025. Epub 2014 Jul 8.

DOI:10.1093/bfgp/elu025
PMID:25005607
Abstract

Genome-wide association studies have uncovered hundreds of common genetic variants involved in complex diseases. However, for most complex diseases, these common genetic variants only marginally contribute to disease susceptibility. It is now argued that rare variants located in different genes could in fact play a more important role in disease susceptibility than common variants. These rare genetic variants were not captured by genome-wide association studies using single nucleotide polymorphism-chips but with the advent of next-generation sequencing technologies, they have become detectable. It is now possible to study their contribution to common disease by resequencing samples of cases and controls or by using new genotyping exome arrays that cover rare alleles. In this review, we address the question of the contribution of rare variants in common disease by taking the examples of different diseases for which some resequencing studies have already been performed, and by summarizing the results of simulation studies conducted so far to investigate the genetic architecture of complex traits in human. So far, empirical data have not allowed the exclusion of many models except the most extreme ones involving only a small number of rare variants with large effects contributing to complex disease. To unravel the genetic architecture of complex disease, case-control data will not be sufficient, and alternative study designs need to be proposed together with methodological developments.

摘要

全基因组关联研究已经发现了数百种与复杂疾病相关的常见基因变异。然而,对于大多数复杂疾病来说,这些常见基因变异对疾病易感性的贡献微乎其微。现在有人认为,位于不同基因中的罕见变异实际上可能比常见变异在疾病易感性中发挥更重要的作用。这些罕见的基因变异未被使用单核苷酸多态性芯片的全基因组关联研究捕获,但随着下一代测序技术的出现,它们已变得可被检测到。现在可以通过对病例和对照样本进行重测序或使用覆盖罕见等位基因的新型基因分型外显子阵列来研究它们对常见疾病的贡献。在这篇综述中,我们通过列举一些已经进行了重测序研究的不同疾病的例子,并总结迄今为止为研究人类复杂性状的遗传结构而进行的模拟研究结果,来探讨罕见变异在常见疾病中的贡献问题。到目前为止,除了极少数涉及仅有少量具有大效应的罕见变异导致复杂疾病的极端模型外,实证数据还无法排除许多模型。为了阐明复杂疾病的遗传结构,病例对照数据是不够的,需要提出替代研究设计并推动方法学的发展。

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How important are rare variants in common disease?罕见变异在常见疾病中有多重要?
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