1] Division of Human Genetics, Institute of Infectious Disease and Molecular Medicine, University of Cape Town, Cape Town, South Africa [2] Centre for Molecular Medicine and Therapeutics, Child and Family Research Institute, University of British Columbia, Vancouver, British Columbia, Canada.
Eur J Hum Genet. 2013 Oct;21(10):1120-7. doi: 10.1038/ejhg.2013.2. Epub 2013 Mar 6.
Huntington disease (HD) is a neurodegenerative disorder resulting from the expansion of a CAG trinucleotide repeat in the huntingtin (HTT) gene. Worldwide prevalence varies geographically with the highest figures reported in populations of European ancestry. HD in South Africa has been reported in Caucasian, black and mixed subpopulations, with similar estimated prevalence in the Caucasian and mixed groups and a lower estimate in the black subpopulation. Recent studies have associated specific HTT haplotypes with HD in distinct populations. Expanded HD alleles in Europe occur predominantly on haplogroup A (specifically high-risk variants A1/A2), whereas in East Asian populations, HD alleles are associated with haplogroup C. Whether specific HTT haplotypes associate with HD in black Africans and how these compare with haplotypes found in European and East Asian populations remains unknown. The current study genotyped the HTT region in unaffected individuals and HD patients from each of the South African subpopulations, and haplotypes were constructed. CAG repeat sizes were determined and phased to haplotype. Results indicate that HD alleles from Caucasian and mixed patients are predominantly associated with haplogroup A, signifying a similar European origin for HD. However, in black patients, HD occurs predominantly on haplogroup B, suggesting several distinct origins of the mutation in South Africa. The absence of high-risk variants (A1/A2) in the black subpopulation may also explain the reported low prevalence of HD. Identification of haplotypes associated with HD-expanded alleles is particularly relevant to the development of population-specific therapeutic targets for selective suppression of the expanded HTT transcript.
亨廷顿病 (HD) 是一种神经退行性疾病,由亨廷顿 (HTT) 基因中的 CAG 三核苷酸重复扩展引起。全球患病率因地理位置而异,欧洲血统人群的报告发病率最高。南非的 HD 已在白种人、黑人和混合人群中报告,白种人和混合人群的估计患病率相似,黑人群体的估计患病率较低。最近的研究将特定的 HTT 单倍型与不同人群中的 HD 相关联。欧洲扩张的 HD 等位基因主要发生在单倍群 A 上(特别是高危变体 A1/A2),而在东亚人群中,HD 等位基因与单倍群 C 相关联。特定的 HTT 单倍型是否与南非黑人中的 HD 相关联,以及这些单倍型与欧洲和东亚人群中的单倍型相比如何,目前尚不清楚。本研究对每个南非人群中的未受影响个体和 HD 患者进行了 HTT 区域的基因分型,并构建了单倍型。确定了 CAG 重复大小并进行了单倍型相位测定。结果表明,来自白人和混合患者的 HD 等位基因主要与单倍群 A 相关,表明 HD 具有类似的欧洲起源。然而,在黑人患者中,HD 主要发生在单倍群 B 上,这表明该突变在南非有几个不同的起源。黑人亚群中缺乏高危变体 (A1/A2) 也可能解释了报告的 HD 低患病率。确定与 HD 扩展等位基因相关的单倍型对于开发针对选择性抑制扩展 HTT 转录本的特定人群治疗靶点尤其重要。