Liu Lifeng, Wang Pengfei, Meng Jingbo, Chen Lili, Zhu Wensheng, Ma Weijun
School of Mathematical Sciences, Heilongjiang University, Harbin150080, China.
Key Laboratory for Applied Statistics of MOE, School of Mathematics and Statistics, Northeast Normal University, Changchun130024, China.
Genet Res (Camb). 2019 Dec 13;101:e13. doi: 10.1017/S0016672319000120.
In recent years, there has been an increasing interest in detecting disease-related rare variants in sequencing studies. Numerous studies have shown that common variants can only explain a small proportion of the phenotypic variance for complex diseases. More and more evidence suggests that some of this missing heritability can be explained by rare variants. Considering the importance of rare variants, researchers have proposed a considerable number of methods for identifying the rare variants associated with complex diseases. Extensive research has been carried out on testing the association between rare variants and dichotomous, continuous or ordinal traits. So far, however, there has been little discussion about the case in which both genotypes and phenotypes are ordinal variables. This paper introduces a method based on the γ-statistic, called OV-RV, for examining disease-related rare variants when both genotypes and phenotypes are ordinal. At present, little is known about the asymptotic distribution of the γ-statistic when conducting association analyses for rare variants. One advantage of OV-RV is that it provides a robust estimation of the distribution of the γ-statistic by employing the permutation approach proposed by Fisher. We also perform extensive simulations to investigate the numerical performance of OV-RV under various model settings. The simulation results reveal that OV-RV is valid and efficient; namely, it controls the type I error approximately at the pre-specified significance level and achieves greater power at the same significance level. We also apply OV-RV for rare variant association studies of diastolic blood pressure.
近年来,在测序研究中检测与疾病相关的罕见变异受到了越来越多的关注。大量研究表明,常见变异只能解释复杂疾病表型变异的一小部分。越来越多的证据表明,部分缺失的遗传力可以由罕见变异来解释。考虑到罕见变异的重要性,研究人员提出了大量用于识别与复杂疾病相关的罕见变异的方法。针对罕见变异与二分、连续或有序性状之间的关联性检测,已经开展了广泛的研究。然而,到目前为止,对于基因型和表型均为有序变量的情况,几乎没有相关讨论。本文介绍了一种基于γ统计量的方法,称为OV-RV,用于在基因型和表型均为有序变量时检测与疾病相关的罕见变异。目前,在对罕见变异进行关联分析时,对于γ统计量的渐近分布了解甚少。OV-RV的一个优点是,它通过采用Fisher提出的置换方法,对γ统计量的分布提供了稳健的估计。我们还进行了广泛的模拟,以研究OV-RV在各种模型设置下的数值性能。模拟结果表明,OV-RV是有效且高效的;也就是说,它将I型错误大致控制在预先指定的显著性水平,并在相同显著性水平下具有更高的检验效能。我们还将OV-RV应用于舒张压的罕见变异关联研究。