Dalgaard Kevin, Landgraf Kathrin, Heyne Steffen, Lempradl Adelheid, Longinotto John, Gossens Klaus, Ruf Marius, Orthofer Michael, Strogantsev Ruslan, Selvaraj Madhan, Lu Tess Tsai-Hsiu, Casas Eduard, Teperino Raffaele, Surani M Azim, Zvetkova Ilona, Rimmington Debra, Tung Y C Loraine, Lam Brian, Larder Rachel, Yeo Giles S H, O'Rahilly Stephen, Vavouri Tanya, Whitelaw Emma, Penninger Josef M, Jenuwein Thomas, Cheung Ching-Lung, Ferguson-Smith Anne C, Coll Anthony P, Körner Antje, Pospisilik J Andrew
Max Planck Institute of Immunobiology and Epigenetics, Stübeweg 51, 79108 Freiburg, Germany.
Department of Women's and Child Health, Center for Pediatric Research Leipzig, University Hospital for Children & Adolescents, University of Leipzig, 04103 Leipzig, Germany; Integrated Research and Treatment Center (IFB) Adiposity Diseases, University of Leipzig, 04103 Leipzig, Germany.
Cell. 2016 Jan 28;164(3):353-64. doi: 10.1016/j.cell.2015.12.025.
More than one-half billion people are obese, and despite progress in genetic research, much of the heritability of obesity remains enigmatic. Here, we identify a Trim28-dependent network capable of triggering obesity in a non-Mendelian, "on/off" manner. Trim28(+/D9) mutant mice exhibit a bi-modal body-weight distribution, with isogenic animals randomly emerging as either normal or obese and few intermediates. We find that the obese-"on" state is characterized by reduced expression of an imprinted gene network including Nnat, Peg3, Cdkn1c, and Plagl1 and that independent targeting of these alleles recapitulates the stochastic bi-stable disease phenotype. Adipose tissue transcriptome analyses in children indicate that humans too cluster into distinct sub-populations, stratifying according to Trim28 expression, transcriptome organization, and obesity-associated imprinted gene dysregulation. These data provide evidence of discrete polyphenism in mouse and man and thus carry important implications for complex trait genetics, evolution, and medicine.
超过5亿人肥胖,尽管基因研究取得了进展,但肥胖的许多遗传力仍然难以捉摸。在这里,我们确定了一个依赖Trim28的网络,它能够以非孟德尔式的“开/关”方式引发肥胖。Trim28(+/D9)突变小鼠表现出双峰体重分布,同基因动物随机出现正常或肥胖,很少有中间状态。我们发现肥胖的“开”状态的特征是包括Nnat、Peg3、Cdkn1c和Plagl1在内的印记基因网络表达降低,并且对这些等位基因的独立靶向重现了随机双稳态疾病表型。对儿童脂肪组织转录组的分析表明,人类也根据Trim28表达、转录组组织和肥胖相关印记基因失调分为不同的亚群。这些数据提供了小鼠和人类中离散多型现象的证据,因此对复杂性状遗传学、进化和医学具有重要意义。