Clinical Neuroscience Group, Eskitis Institute for Cell and Molecular Therapies, Griffith University, Brisbane, Don Young Road, Nathan, Queensland, Australia.
Parkinsonism Relat Disord. 2012 May;18(4):351-7. doi: 10.1016/j.parkreldis.2011.11.024. Epub 2011 Dec 14.
Genes involved in familial dystonia syndromes (DYT genes) are ideal candidates for investigating whether common genetic variants influence the susceptibility to sporadic primary dystonia. To date, there have been few candidate gene studies for primary dystonia and only two DYT genes, TOR1A and THAP1, have been assessed. We therefore employed a haplotype-tagging strategy to comprehensively assess if common polymorphisms in eight DYT genes (TOR1A, TAF1, GCH1, THAP1, MR-1 (PNKD), SGCE, ATP1A3 and PRKRA) confer risk for sporadic primary dystonia. The 230 primary dystonia cases were matched for age and gender to 228 controls, recruited from movement disorder clinics in Brisbane, Australia and the Australian electoral roll. All subjects were genotyped for 56 tagging SNPs and genotype associations were investigated. Modest genotypic associations (P<0.05) were observed for three GCH1 SNPs (rs12147422, rs3759664 and rs10483639) when comparing all cases against controls. Associations were also seen when the cases were stratified based on presentation. Overall, our findings do not support the hypothesis that common TOR1A variants affect susceptibility for sporadic primary dystonia, and that it is unlikely that common variants around the DYT genes confer substantial risk for sporadic primary dystonia. Further work is warranted to follow up the GCH1 SNPs and the subgroup analyses.
参与家族性肌张力障碍综合征(DYT 基因)的基因是研究常见遗传变异是否影响散发性原发性肌张力障碍易感性的理想候选基因。迄今为止,原发性肌张力障碍的候选基因研究较少,只有两个 DYT 基因(TOR1A 和 THAP1)得到了评估。因此,我们采用了单倍型标签策略,全面评估了八个 DYT 基因(TOR1A、TAF1、GCH1、THAP1、MR-1(PNKD)、SGCE、ATP1A3 和 PRKRA)中的常见多态性是否与散发性原发性肌张力障碍相关。230 例原发性肌张力障碍病例与澳大利亚布里斯班运动障碍诊所和澳大利亚选举名单中的 228 名对照者进行了年龄和性别匹配。所有受试者均进行了 56 个标记 SNP 的基因分型,并对基因型关联进行了研究。在比较所有病例与对照组时,三个 GCH1 SNP(rs12147422、rs3759664 和 rs10483639)观察到适度的基因型关联(P<0.05)。当根据表现对病例进行分层时,也观察到了关联。总体而言,我们的研究结果不支持常见 TOR1A 变异影响散发性原发性肌张力障碍易感性的假设,并且 DYT 基因周围的常见变异不太可能导致散发性原发性肌张力障碍的风险显著增加。需要进一步的工作来跟进 GCH1 SNP 和亚组分析。