Institut National de la Santé et de la Recherche Médicale (INSERM) U1065, Université Côte d'Azur, Centre Méditerranéen de Médecine Moléculaire (C3M), Atip-Avenir, Fédération Hospitalo-Universitaire (FHU) Oncoage, 06204 Nice, France.
Human Oncology and Pathogenesis Program and Leukemia Service, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA.
Cell Rep. 2020 Mar 10;30(10):3397-3410.e5. doi: 10.1016/j.celrep.2020.02.056.
Defective cholesterol efflux pathways in mice promote the expansion of hematopoietic stem and progenitor cells and a bias toward the myeloid lineage, as observed in chronic myelomonocytic leukemia (CMML). Here, we identify 5 somatic missense mutations in ABCA1 in 26 patients with CMML. These mutations confer a proliferative advantage to monocytic leukemia cell lines in vitro. In vivo inactivation of ABCA1 or expression of ABCA1 mutants in hematopoietic cells in the setting of Tet2 loss demonstrates a myelosuppressive function of ABCA1. Mechanistically, ABCA1 mutations impair the tumor-suppressor functions of WT ABCA1 in myeloproliferative neoplasms by increasing the IL-3Rβ signaling via MAPK and JAK2 and subsequent metabolic reprogramming. Overexpression of a human apolipoprotein A-1 transgene dampens myeloproliferation. These findings identify somatic mutations in ABCA1 that subvert its anti-proliferative and cholesterol efflux functions and permit the progression of myeloid neoplasms. Therapeutic increases in HDL bypass these defects and restore normal hematopoiesis.
在小鼠中,胆固醇外排途径的缺陷会促进造血干细胞和祖细胞的扩增,并偏向髓系,就像在慢性粒单核细胞白血病(CMML)中观察到的那样。在这里,我们在 26 名 CMML 患者中鉴定出 ABCA1 中的 5 个体细胞错义突变。这些突变赋予单核白血病细胞系体外增殖优势。在 Tet2 缺失的情况下,体内失活 ABCA1 或在造血细胞中表达 ABCA1 突变体,证明了 ABCA1 的骨髓抑制功能。在机制上,ABCA1 突变通过 MAPK 和 JAK2 增加 IL-3Rβ 信号,随后进行代谢重编程,从而损害 WT ABCA1 在骨髓增生性肿瘤中的肿瘤抑制功能。人载脂蛋白 A-1 转基因的过表达可抑制骨髓增生。这些发现确定了 ABCA1 的体细胞突变,这些突变破坏了其抗增殖和胆固醇外排功能,并允许髓系肿瘤的进展。HDL 的治疗性增加可绕过这些缺陷并恢复正常造血。