Geller Frank, Reichwald Kathrin, Dempfle Astrid, Illig Thomas, Vollmert Caren, Herpertz Stephan, Siffert Winfried, Platzer Matthias, Hess Claudia, Gudermann Thomas, Biebermann Heike, Wichmann H-Erich, Schäfer Helmut, Hinney Anke, Hebebrand Johannes
Institute of Medical Biometry and Epidemiology, Philipps-University, Marburg, Germany.
Am J Hum Genet. 2004 Mar;74(3):572-81. doi: 10.1086/382490. Epub 2004 Feb 17.
Several rare mutations in the melanocortin-4 receptor gene (MC4R) predispose to obesity. For the most common missense variant V103I (rs2229616), however, the previously reported similar carrier frequencies in obese and nonobese individuals are in line with in vitro studies, which have not shown a functional implication of this variant. In the present study, we initially performed a transmission/disequilibrium test on 520 trios with obesity, and we observed a lower transmission rate of the I103 allele (P=.017), which was an unexpected finding. Therefore, we initiated two large case-control studies (N=2,334 and N=661) and combined the data with those from 12 published studies, for a total of 7,713 individuals. The resulting meta-analysis provides evidence for a negative association of the I103 allele with obesity (odds ratio 0.69; 95% confidence interval 0.50-0.96; P=.03), mainly comprising samples of European origin. Additional screening of four other ethnic groups showed comparable I103 carrier frequencies well below 10%. Genomic sequencing of the MC4R gene revealed three polymorphisms in the noncoding region that displayed strong linkage disequilibrium with V103I. In our functional in vitro assays, the variant was indistinguishable from the wild-type allele, as was the result in previous studies. This report on an SNP/haplotype that is negatively associated with obesity expands the successful application of meta-analysis of modest effects in common diseases to a variant with a carrier frequency well below 10%. The respective protective effect against obesity implies that variation in the MC4R gene entails both loss and gain of function.
黑皮质素-4受体基因(MC4R)中的几种罕见突变易导致肥胖。然而,对于最常见的错义变体V103I(rs2229616),先前报道的肥胖个体和非肥胖个体中相似的携带者频率与体外研究结果一致,体外研究未显示该变体具有功能意义。在本研究中,我们首先对520个肥胖三联体进行了传递/不平衡测试,观察到I103等位基因的传递率较低(P = 0.017),这是一个意外发现。因此,我们启动了两项大型病例对照研究(N = 2334和N = 661),并将数据与12项已发表研究的数据合并,共计7713名个体。结果的荟萃分析为I103等位基因与肥胖的负相关提供了证据(优势比0.69;95%置信区间0.50 - 0.96;P = 0.03),主要包括欧洲血统的样本。对其他四个种族群体的额外筛查显示,I103携带者频率相当,均远低于10%。MC4R基因的基因组测序揭示了非编码区的三个多态性,它们与V103I表现出强烈的连锁不平衡。在我们的体外功能测定中,该变体与野生型等位基因没有区别,这与之前的研究结果一致。这份关于与肥胖负相关的单核苷酸多态性/单倍型的报告,将对常见疾病中适度效应的荟萃分析的成功应用扩展到了携带者频率远低于10%的变体。对肥胖的相应保护作用意味着MC4R基因的变异既有功能丧失也有功能获得。