Department of Twin Research and Genetic Epidemiology, King's College London, London, United Kingdom.
Section of Vascular Biology & Inflammation, BHF Centre for Cardiovascular Excellence, School of Cardiovascular Medicine and Sciences, King's College London, London, United Kingdom.
PLoS One. 2018 Apr 26;13(4):e0196279. doi: 10.1371/journal.pone.0196279. eCollection 2018.
Raynaud's phenomenon (RP) describes the phenomenon of recurrent vasospasm of digital arteries, associated with skin colour changes: pallor, cyanosis and erythema. Twin studies have indicated a genetic predisposition for RP; however, the precise aetiology of RP remains unknown. It is thought that genetic variation in temperature-responsive or vasospastic genes might underlie RP so performed a candidate gene study in a large, population based sample. We assessed the association between RP and single nucleotide polymorphisms (SNPs) in the TRPA1, TRPM8, CALCA, CALCB and NOS1 genes.
Analysis included a total of 4276 individuals from the TwinsUK database. RP status had been determined using validated, self-administered questionnaires and was diagnosed in 640 individuals (17.6%). 66 tag SNPs across the candidate genes were tested for association with RP status using a linear regression model, accounting for covariates. Adjustment was made for multiple testing. RegulomeDB and GTEx databases were used to assess possible functional effects of the polymorphisms.
Nominally significant associations between RP and four SNPs in NOS1 and one in CALCB were identified. After permutation testing, rs527590 SNP in NOS1 passed the significance threshold. RegulomeDB scores indicated an unlikely functional effect of this variant, while the survey of the GTEx database found the SNP and several variants in linkage disequilibrium to be cis-eQTLs in skin.
Results indicate that RP is associated with variation in gene NOS1. This finding may be related to the observation that the significant SNP in NOS1 is known to exhibit functional influence on the gene expression.
雷诺现象(RP)描述了手指动脉反复发作的血管痉挛现象,伴有皮肤颜色变化:苍白、发绀和红斑。双胞胎研究表明 RP 存在遗传倾向;然而,RP 的确切病因仍不清楚。人们认为温度反应或血管痉挛基因的遗传变异可能是 RP 的基础,因此在一个大型的基于人群的样本中进行了候选基因研究。我们评估了 RP 与 TRPA1、TRPM8、CALCA、CALCB 和 NOS1 基因中的单核苷酸多态性(SNP)之间的关联。
分析包括来自 TwinsUK 数据库的总共 4276 个人。使用经过验证的自我管理问卷确定 RP 状态,在 640 个人(17.6%)中诊断出 RP。使用线性回归模型,在考虑协变量的情况下,对候选基因中的 66 个标签 SNP 与 RP 状态进行关联测试。对多测试进行了调整。RegulomeDB 和 GTEx 数据库用于评估多态性的可能功能影响。
在 NOS1 中的四个 SNP 和 CALCB 中的一个 SNP 与 RP 之间发现了名义上显著的关联。在经过置换测试后,NOS1 中的 rs527590 SNP 通过了显著性阈值。RegulomeDB 评分表明该变体不太可能具有功能影响,而 GTEx 数据库的调查发现 SNP 及其几个连锁不平衡的变体是皮肤中的 cis-eQTL。
结果表明,RP 与基因 NOS1 的变异有关。这一发现可能与观察到的显著 SNP 在 NOS1 中对基因表达具有功能影响有关。