Kay C, Skotte N H, Southwell A L, Hayden M R
Centre for Molecular Medicine and Therapeutics, Child and Family Research Institute, University of British Columbia, Vancouver, Canada.
Clin Genet. 2014 Jul;86(1):29-36. doi: 10.1111/cge.12385. Epub 2014 Apr 11.
Gene silencing offers a novel therapeutic strategy for dominant genetic disorders. In specific diseases, selective silencing of only one copy of a gene may be advantageous over non-selective silencing of both copies. Huntington disease (HD) is an autosomal dominant disorder caused by an expanded CAG trinucleotide repeat in the Huntingtin gene (HTT). Silencing both expanded and normal copies of HTT may be therapeutically beneficial, but preservation of normal HTT expression is preferred. Allele-specific methods can selectively silence the mutant HTT transcript by targeting either the expanded CAG repeat or single nucleotide polymorphisms (SNPs) in linkage disequilibrium with the expansion. Both approaches require personalized treatment strategies based on patient genotypes. We compare the prospect of safe treatment of HD by CAG- and SNP-specific silencing approaches and review HD population genetics used to guide target identification in the patient population. Clinical implementation of allele-specific HTT silencing faces challenges common to personalized genetic medicine, requiring novel solutions from clinical scientists and regulatory authorities.
基因沉默为显性遗传病提供了一种新的治疗策略。在特定疾病中,仅对一个基因拷贝进行选择性沉默可能比同时对两个拷贝进行非选择性沉默更具优势。亨廷顿舞蹈症(HD)是一种常染色体显性疾病,由亨廷顿基因(HTT)中CAG三核苷酸重复序列扩增引起。沉默HTT的扩增拷贝和正常拷贝在治疗上可能有益,但保留正常HTT表达更为可取。等位基因特异性方法可以通过靶向扩增的CAG重复序列或与该扩增处于连锁不平衡状态的单核苷酸多态性(SNP)来选择性沉默突变的HTT转录本。这两种方法都需要基于患者基因型的个性化治疗策略。我们比较了通过CAG特异性和SNP特异性沉默方法安全治疗HD的前景,并回顾了用于指导患者群体中靶点识别的HD群体遗传学。等位基因特异性HTT沉默的临床应用面临着个性化基因医学常见的挑战,需要临床科学家和监管机构提出新的解决方案。