Lerer E, Levi S, Salomon S, Darvasi A, Yirmiya N, Ebstein R P
Department of Human Genetics, Hebrew University, Jerusalem, Israel.
Mol Psychiatry. 2008 Oct;13(10):980-8. doi: 10.1038/sj.mp.4002087. Epub 2007 Sep 25.
Evidence both from animal and human studies suggests that common polymorphisms in the oxytocin receptor (OXTR) gene are likely candidates to confer risk for autism spectrum disorders (ASD). In lower mammals, oxytocin is important in a wide range of social behaviors, and recent human studies have shown that administration of oxytocin modulates behavior in both clinical and non-clinical groups. Additionally, two linkage studies and two recent association investigations also underscore a possible role for the OXTR gene in predisposing to ASD. We undertook a comprehensive study of all 18 tagged SNPs across the entire OXTR gene region identified using HapMap data and the Haploview algorithm. Altogether 152 subjects diagnosed with ASDs (that is, DSM IV autistic disorder or pervasive developmental disorder--NOS) from 133 families were genotyped (parents and affected siblings). Both individual SNPs and haplotypes were tested for association using family-based association tests as provided in the UNPHASED set of programs. Significant association with single SNPs and haplotypes (global P-values <0.05, following permutation test adjustment) were observed with ASD. Association was also observed with IQ and the Vineland Adaptive Behavior Scales (VABS). In particular, a five-locus haplotype block (rs237897-rs13316193-rs237889-rs2254298-rs2268494) was significantly associated with ASD (nominal global P=0.000019; adjusted global P=0.009) and a single haplotype (carried by 7% of the population) within that block showed highly significant association (P=0.00005). This is the third association study, in a third ethnic group, showing that SNPs and haplotypes in the OXTR gene confer risk for ASD. The current investigation also shows association with IQ and total VABS scores (as well as the communication, daily living skills and socialization subdomains), suggesting that this gene shapes both cognition and daily living skills that may cross diagnostic boundaries.
来自动物和人类研究的证据表明,催产素受体(OXTR)基因中的常见多态性可能是导致自闭症谱系障碍(ASD)风险的候选因素。在低等哺乳动物中,催产素在广泛的社会行为中很重要,最近的人类研究表明,给予催产素可调节临床和非临床群体的行为。此外,两项连锁研究和两项近期的关联调查也强调了OXTR基因在ASD易感性中的可能作用。我们对使用HapMap数据和Haploview算法确定的整个OXTR基因区域内的所有18个标签单核苷酸多态性(SNP)进行了全面研究。对来自133个家庭的总共152名被诊断为ASD(即DSM-IV自闭症障碍或广泛性发育障碍——非特定型)的受试者(父母和受影响的兄弟姐妹)进行了基因分型。使用UNPHASED程序集中提供的基于家庭的关联测试对单个SNP和单倍型进行关联测试。观察到与ASD存在单个SNP和单倍型的显著关联(置换检验调整后全局P值<0.05)。还观察到与智商和文兰适应行为量表(VABS)存在关联。特别是,一个五位点单倍型块(rs237897-rs13316193-rs237889-rs2254298-rs2268494)与ASD显著相关(名义全局P=0.000019;调整后全局P=0.009),该块内的一个单倍型(占人群的7%)显示出高度显著的关联(P=0.00005)。这是在第三个种族群体中进行的第三项关联研究,表明OXTR基因中的SNP和单倍型会导致ASD风险。当前的调查还显示与智商和VABS总分(以及沟通、日常生活技能和社交子领域)存在关联,表明该基因塑造了可能跨越诊断界限的认知和日常生活技能。