Department of Biostatistics, CIMH Mannheim, Mannheim Medical School of the University of Heidelberg, Mannheim, Germany.
Department of Medical Biostatistics, Epidemiology & Informatics, University Medical Center of the Johannes Gutenberg University Mainz, Mainz, Germany.
PLoS One. 2019 Feb 21;14(2):e0212344. doi: 10.1371/journal.pone.0212344. eCollection 2019.
The problem of checking the genotype distribution obtained for some diallelic marker for compatibility with the Hardy-Weinberg equilibrium (HWE) condition arises also for loci on the X chromosome. The possible genotypes depend on the sex of the individual in this case: for females, the genotype distribution is trinomial, as in the case of an autosomal locus, whereas a binomial proportion is observed for males. Like in genetic association studies with autosomal SNPs, interest is typically in establishing approximate compatibility of the observed genotype frequencies with HWE. This requires to replace traditional methods tailored for detecting lack of fit to the model with an equivalence testing procedure to be derived by treating approximate compatibility with the model as the alternative hypothesis. The test constructed here is based on an upper confidence bound and a simple to interpret combined measure of distance between true and HWE conforming genotype distributions in female and male subjects. A particular focus of the paper is on the derivation of the asymptotic distribution of the test statistic under null alternatives which is not of the usual Gaussian form. A closed sample size formula is also provided and shown to behave satisfactorily in terms of the approximation error.
对于 X 染色体上的某些二等位标记,检查所获得的基因型分布是否与 Hardy-Weinberg 平衡(HWE)条件相匹配的问题也会出现。在这种情况下,可能的基因型取决于个体的性别:对于女性,基因型分布是三项式的,就像常染色体上的基因座一样,而男性则观察到二项式比例。与常染色体 SNP 的遗传关联研究一样,人们通常感兴趣的是确定观察到的基因型频率与 HWE 的近似兼容性。这需要用等效检验程序代替专门用于检测对模型拟合不足的传统方法,该程序通过将对模型的近似兼容性作为替代假设来推导。这里构建的检验基于上限置信区间和一个简单的解释,用于衡量女性和男性受试者中真实和符合 HWE 的基因型分布之间的距离的综合度量。本文的一个特别关注点是在零假设替代下推导检验统计量的渐近分布,该分布不是通常的高斯形式。还提供了一个封闭的样本量公式,并显示在近似误差方面表现良好。