Iranian Research Center on Aging, University of Social Welfare and Rehabilitation Sciences, Tehran, Iran.
Chemical Injuries Research Center, Systems Biology and Poisonings Institute, Baqiyatallah University of Medical Sciences, Tehran, Iran.
Genetica. 2022 Feb;150(1):27-40. doi: 10.1007/s10709-021-00143-5. Epub 2022 Jan 5.
Across human protein-coding genes, the human neuron-specific genes, RIT2 and GPM6B, contain the two longest GA short tandem repeats (STRs) of 11 and 9-repeats, respectively, the length ranges of which are functional, and result in gene expression alteration. Here we sequenced the RIT2 and GPM6B STRs in 600 human subjects, consisting of late-onset neurocognitive disorder (n = 200), multiple sclerosis (n = 200), and controls (n = 200). Furthermore, we selected two large human databases, including the general-population-based gnomAD ( https://gnomad.broadinstitute.org ) and a mainly disease-phenotype-archiving database, TOPMed ( https://www.nhlbiwgs.org ), to compare allele frequencies in the general populations vs. the disease compartment. The RIT2 and GPM6B GA-repeats were monomorphic in the human subjects studied, at lengths of 11 and 9-repeats, respectively, and were predominantly human-specific in formula. Exception included a 9/11 genotype of the RIT2 GA-STR in an isolate case of female multiple sclerosis. Exceedingly rare alleles of the two GA repeats were significantly enriched in TOPMed vs. the gnomAD. We report prime instances of predominant monomorphism for specific lengths of STRs in human, and possible enrichment of rare divergent alleles in the disease phenotype compartment. While STRs are most attended because of their high polymorphic nature, STR monomorphism is an underappreciated feature, which may have a link with natural selection and disease.
在人类蛋白编码基因中,神经元特异性基因 RIT2 和 GPM6B 分别包含两个最长的 GA 短串联重复序列(STR),长度分别为 11 个和 9 个重复,其长度范围具有功能,并导致基因表达改变。在这里,我们对 600 个人类受试者的 RIT2 和 GPM6B STR 进行了测序,包括迟发性神经认知障碍(n=200)、多发性硬化症(n=200)和对照组(n=200)。此外,我们选择了两个大型人类数据库,包括基于一般人群的 gnomAD(https://gnomad.broadinstitute.org)和主要疾病表型存档数据库 TOPMed(https://www.nhlbiwgs.org),以比较一般人群与疾病组之间的等位基因频率。在研究的人类受试者中,RIT2 和 GPM6B GA 重复序列均为单态性,长度分别为 11 个和 9 个重复,在公式中主要是人类特异性的。例外的是一名多发性硬化症女性孤立病例的 RIT2 GA-STR 为 9/11 基因型。这两个 GA 重复的极其罕见等位基因在 TOPMed 中明显比 gnomAD 更丰富。我们报告了人类特定 STR 长度的主要单态性实例,以及疾病表型组中罕见的发散等位基因可能富集的情况。虽然 STR 因其高度多态性而备受关注,但 STR 单态性是一个未被充分认识的特征,它可能与自然选择和疾病有关。