Hinds David A, Barnholt Kimberly E, Mesa Ruben A, Kiefer Amy K, Do Chuong B, Eriksson Nicholas, Mountain Joanna L, Francke Uta, Tung Joyce Y, Nguyen Huong Marie, Zhang Haiyu, Gojenola Linda, Zehnder James L, Gotlib Jason
23andMe, Inc, Mountain View, CA;
Division of Hematology & Medical Oncology, Mayo Clinic, Scottsdale, AZ;
Blood. 2016 Aug 25;128(8):1121-8. doi: 10.1182/blood-2015-06-652941. Epub 2016 Jun 30.
We conducted a genome-wide association study (GWAS) to identify novel predisposition alleles associated with Philadelphia chromosome-negative myeloproliferative neoplasms (MPNs) and JAK2 V617F clonal hematopoiesis in the general population. We recruited a web-based cohort of 726 individuals with polycythemia vera, essential thrombocythemia, and myelofibrosis and 252 637 population controls unselected for hematologic phenotypes. Using a single-nucleotide polymorphism (SNP) array platform with custom probes for the JAK2 V617F mutation (V617F), we identified 497 individuals (0.2%) among the population controls who were V617F carriers. We performed a combined GWAS of the MPN cases plus V617F carriers in the control population (n = 1223) vs the remaining controls who were noncarriers for V617F (n = 252 140). For these MPN cases plus V617F carriers, we replicated the germ line JAK2 46/1 haplotype (rs59384377: odds ratio [OR] = 2.4, P = 6.6 × 10(-89)), previously associated with V617F-positive MPN. We also identified genome-wide significant associations in the TERT gene (rs7705526: OR = 1.8, P = 1.1 × 10(-32)), in SH2B3 (rs7310615: OR = 1.4, P = 3.1 × 10(-14)), and upstream of TET2 (rs1548483: OR = 2.0, P = 2.0 × 10(-9)). These associations were confirmed in a separate replication cohort of 446 V617F carriers vs 169 021 noncarriers. In a joint analysis of the combined GWAS and replication results, we identified additional genome-wide significant predisposition alleles associated with CHEK2, ATM, PINT, and GFI1B All SNP ORs were similar for MPN patients and controls who were V617F carriers. These data indicate that the same germ line variants endow individuals with a predisposition not only to MPN, but also to JAK2 V617F clonal hematopoiesis, a more common phenomenon that may foreshadow the development of an overt neoplasm.
我们开展了一项全基因组关联研究(GWAS),以确定普通人群中与费城染色体阴性骨髓增殖性肿瘤(MPN)及JAK2 V617F克隆性造血相关的新的易感等位基因。我们招募了一个基于网络的队列,其中包括726例真性红细胞增多症、原发性血小板增多症和骨髓纤维化患者,以及252637名未因血液学表型而被选择的人群对照。使用带有针对JAK2 V617F突变(V617F)的定制探针的单核苷酸多态性(SNP)阵列平台,我们在人群对照中鉴定出497名(0.2%)V617F携带者。我们对MPN病例加对照人群中的V617F携带者(n = 1223)与其余V617F非携带者对照(n = 252140)进行了联合GWAS。对于这些MPN病例加V617F携带者,我们复制了种系JAK2 46/1单倍型(rs59384377:优势比[OR]=2.4,P = 6.6×10^(-89)),该单倍型先前与V617F阳性MPN相关。我们还在TERT基因中鉴定出全基因组显著关联(rs7705526:OR = 1.8,P = 1.1×10^(-32))、在SH2B3中(rs7310615:OR = 1.4,P = 3.1×10^(-14))以及在TET2上游(rs1548483:OR = 2.0,P = 2.0×10^(-9))。这些关联在一个单独的复制队列中得到证实,该队列包括446名V617F携带者和169021名非携带者。在对联合GWAS和复制结果的联合分析中,我们鉴定出与CHEK2、ATM、PINT和GFI1B相关的其他全基因组显著易感等位基因。对于MPN患者和V617F携带者对照,所有SNP的OR值相似。这些数据表明,相同的种系变异不仅使个体易患MPN,还易患JAK2 V617F克隆性造血,这是一种更常见的现象,可能预示着显性肿瘤的发生。