Department of Hematology Oncology, Fondazione Istituto di Ricovero e Cura a Carattere Scientifico Policlinico San Matteo and University of Pavia, Pavia, Italy.
Blood. 2011 Dec 8;118(24):6239-46. doi: 10.1182/blood-2011-09-377275. Epub 2011 Oct 12.
In a previous study, we identified somatic mutations of SF3B1, a gene encoding a core component of RNA splicing machinery, in patients with myelodysplastic syndrome (MDS). Here, we define the clinical significance of these mutations in MDS and myelodysplastic/myeloproliferative neoplasms (MDS/MPN). The coding exons of SF3B1 were screened using massively parallel pyrosequencing in patients with MDS, MDS/MPN, or acute myeloid leukemia (AML) evolving from MDS. Somatic mutations of SF3B1 were found in 150 of 533 (28.1%) patients with MDS, 16 of 83 (19.3%) with MDS/MPN, and 2 of 38 (5.3%) with AML. There was a significant association of SF3B1 mutations with the presence of ring sideroblasts (P < .001) and of mutant allele burden with their proportion (P = .002). The mutant gene had a positive predictive value for ring sideroblasts of 97.7% (95% confidence interval, 93.5%-99.5%). In multivariate analysis including established risk factors, SF3B1 mutations were found to be independently associated with better overall survival (hazard ratio = 0.15, P = .025) and lower risk of evolution into AML (hazard ratio = 0.33, P = .049). The close association between SF3B1 mutations and disease phenotype with ring sideroblasts across MDS and MDS/MPN is consistent with a causal relationship. Furthermore, SF3B1 mutations are independent predictors of favorable clinical outcome, and their incorporation into stratification systems might improve risk assessment in MDS.
在之前的一项研究中,我们在骨髓增生异常综合征(MDS)患者中发现了 SF3B1 的体细胞突变,SF3B1 是 RNA 剪接机制的核心组成部分之一。在这里,我们定义了这些突变在 MDS 和骨髓增生异常/骨髓增殖性肿瘤(MDS/MPN)中的临床意义。使用大规模平行焦磷酸测序筛选了 MDS、MDS/MPN 或由 MDS 发展而来的急性髓系白血病(AML)患者的 SF3B1 编码外显子。在 533 例 MDS 患者、83 例 MDS/MPN 患者和 38 例 AML 患者中发现了 SF3B1 体细胞突变,分别占 28.1%、19.3%和 5.3%。SF3B1 突变与环形铁幼粒细胞的存在显著相关(P<.001),且突变等位基因负荷与其比例相关(P=.002)。突变基因对环形铁幼粒细胞的阳性预测值为 97.7%(95%置信区间,93.5%-99.5%)。在包括已建立的危险因素的多变量分析中,SF3B1 突变被发现与更好的总生存(风险比=0.15,P=0.025)和更低的向 AML 发展的风险(风险比=0.33,P=0.049)独立相关。SF3B1 突变与环形铁幼粒细胞在 MDS 和 MDS/MPN 中密切相关,表明其与疾病表型具有因果关系。此外,SF3B1 突变是有利临床结果的独立预测因素,将其纳入分层系统可能会改善 MDS 的风险评估。