Martin Lisa J, Ding Lili, Zhang Xue, Kissebah Ahmed H, Olivier Michael, Benson D Woodrow
1] Division of Human Genetics, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA [2] Division of Biostatistics and Epidemiology, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA [3] Department of Pediatrics, University of Cincinnati School of Medicine, Cincinnati, OH, USA.
1] Division of Biostatistics and Epidemiology, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA [2] Department of Pediatrics, University of Cincinnati School of Medicine, Cincinnati, OH, USA.
Eur J Hum Genet. 2014 Feb;22(2):243-7. doi: 10.1038/ejhg.2013.120. Epub 2013 Jun 5.
The Human Genome Project was expected to individualize medicine by rapidly advancing knowledge of common complex disease through discovery of disease-causing genetic variants. However, this has proved challenging. Although linkage analysis has identified replicated chromosomal regions, subsequent detection of causal variants for complex traits has been limited. One explanation for this difficulty is that utilization of association to follow up linkage is problematic given that linkage and association are not required to co-occur. Indeed, co-occurrence is likely to occur only in special circumstances, such as Mendelian inheritance, but cannot be universally expected. To overcome this problem, we propose a novel method, the Variant Impact On Linkage Effect Test (VIOLET), which differs from other quantitative methods in that it is designed to follow up linkage by identifying variants that influence the variance explained by a quantitative trait locus. VIOLET's performance was compared with measured genotype and combined linkage association in two data sets with quantitative traits. Using simulated data, VIOLET had high power to detect the causal variant and reduced false positives compared with standard methods. Using real data, VIOLET identified a single variant, which explained 24% of linkage; this variant exhibited only nominal association (P=0.04) using measured genotype and was not identified by combined linkage association. These results demonstrate that VIOLET is highly specific while retaining low false-negative results. In summary, VIOLET overcomes a barrier to gene discovery and thus may be broadly applicable to identify underlying genetic etiology for traits exhibiting linkage.
人类基因组计划期望通过发现致病基因变异来迅速推进对常见复杂疾病的认识,从而实现个性化医疗。然而,事实证明这具有挑战性。尽管连锁分析已经确定了重复的染色体区域,但随后对复杂性状因果变异的检测却很有限。造成这一困难的一个原因是,鉴于连锁和关联并非必然同时出现,利用关联来跟进连锁存在问题。实际上,只有在特殊情况下,如孟德尔遗传,才可能同时出现,而不能普遍预期。为克服这一问题,我们提出了一种新方法——变异对连锁效应测试(VIOLET),它与其他定量方法不同,其设计目的是通过识别影响由数量性状基因座解释的方差的变异来跟进连锁。在两个具有数量性状的数据集里,将VIOLET的性能与测量基因型和联合连锁关联进行了比较。使用模拟数据时,与标准方法相比,VIOLET检测因果变异的能力很强,且假阳性减少。使用真实数据时,VIOLET识别出一个单一变异,该变异解释了24%的连锁;使用测量基因型时,这个变异仅显示出名义上的关联(P = 0.04),联合连锁关联未识别出该变异。这些结果表明,VIOLET具有高度特异性,同时保持低假阴性结果。总之,VIOLET克服了基因发现的一个障碍,因此可能广泛适用于识别表现出连锁的性状的潜在遗传病因。