Human Medical Genetics and Genomics Program, University of Colorado, Anschutz Medical Campus, Aurora, CO 80045, USA.
Department of Mathematical and Statistical Sciences, University of Colorado Denver, Denver, CO 80217, USA.
Am J Hum Genet. 2019 Aug 1;105(2):364-372. doi: 10.1016/j.ajhg.2019.06.013. Epub 2019 Jul 18.
Vitiligo is an autoimmune disease that results in patches of depigmented skin and hair. Previous genome-wide association studies (GWASs) of vitiligo have identified 50 susceptibility loci. Variants at the associated loci are generally common and have individually small effects on risk. Most vitiligo cases are "simplex," where there is no family history of vitiligo, though occasional family clustering of vitiligo occurs, and some "multiplex" families report numerous close affected relatives. Here, we investigate whether simplex and multiplex vitiligo comprise different disease subtypes with different underlying genetic etiologies. We developed and compared the performance of several different vitiligo polygenic risk scores derived from GWAS data. By using the best-performing risk score, we find increased polygenic burden of risk alleles identified by GWAS in multiplex vitiligo cases relative to simplex cases. We additionally find evidence of polygenic transmission of common, low-effect-size risk alleles within multiplex-vitiligo-affected families. Our findings strongly suggest that family clustering of vitiligo involves a high burden of the same common, low-effect-size variants that are relevant in simplex cases. We furthermore find that a variant within the major histocompatibility complex (MHC) class II region contributes disproportionately more to risk in multiplex vitiligo cases than in simplex cases, supporting a special role for adaptive immune triggering in the etiology of multiplex cases. We suggest that genetic risk scores can be a useful tool in analyzing the genetic architecture of clinical disease subtypes and identifying subjects with unusual etiologies for further investigation.
白癜风是一种自身免疫性疾病,导致皮肤和毛发出现色素脱失斑块。先前对白癜风的全基因组关联研究 (GWAS) 已经确定了 50 个易感性位点。关联位点的变异通常是常见的,对风险的影响个体较小。大多数白癜风病例是“单纯性”的,即没有白癜风家族史,但偶尔会出现白癜风家族聚集,有些“多发性”家族报告有许多近亲受影响。在这里,我们研究单纯性和多发性白癜风是否包含不同的疾病亚型,具有不同的潜在遗传病因。我们开发并比较了几种不同的基于 GWAS 数据的白癜风多基因风险评分的性能。通过使用表现最佳的风险评分,我们发现多发性白癜风病例相对于单纯性病例,GWAS 确定的风险等位基因的多基因负担增加。我们还发现,在多发性白癜风受影响的家族中,常见的低效应大小风险等位基因存在多基因传递的证据。我们的研究结果强烈表明,白癜风的家族聚集涉及到相同的常见、低效应大小变异的高负担,这些变异与单纯性病例有关。我们还发现,主要组织相容性复合体 (MHC) 区域内的一个变体在多发性白癜风病例中的风险贡献不成比例地高于单纯性病例,支持适应性免疫触发在多发性病例发病机制中的特殊作用。我们建议遗传风险评分可以成为分析临床疾病亚型遗传结构和识别具有异常病因的患者的有用工具,以便进一步研究。