Lee Hsiang-Ying, Gao Xiaofei, Barrasa M Inmaculada, Li Hu, Elmes Russell R, Peters Luanne L, Lodish Harvey F
Whitehead Institute for Biomedical Research, Nine Cambridge Center, Cambridge, Massachusetts 02142, USA.
Center for Individualized Medicine, Department of Molecular Pharmacology and Experimental Therapeutics, Mayo Clinic, Rochester, Minnesota 55905, USA.
Nature. 2015 Jun 25;522(7557):474-7. doi: 10.1038/nature14326. Epub 2015 May 11.
Many acute and chronic anaemias, including haemolysis, sepsis and genetic bone marrow failure diseases such as Diamond-Blackfan anaemia, are not treatable with erythropoietin (Epo), because the colony-forming unit erythroid progenitors (CFU-Es) that respond to Epo are either too few in number or are not sensitive enough to Epo to maintain sufficient red blood cell production. Treatment of these anaemias requires a drug that acts at an earlier stage of red cell formation and enhances the formation of Epo-sensitive CFU-E progenitors. Recently, we showed that glucocorticoids specifically stimulate self-renewal of an early erythroid progenitor, burst-forming unit erythroid (BFU-E), and increase the production of terminally differentiated erythroid cells. Here we show that activation of the peroxisome proliferator-activated receptor α (PPAR-α) by the PPAR-α agonists GW7647 and fenofibrate synergizes with the glucocorticoid receptor (GR) to promote BFU-E self-renewal. Over time these agonists greatly increase production of mature red blood cells in cultures of both mouse fetal liver BFU-Es and mobilized human adult CD34(+) peripheral blood progenitors, with a new and effective culture system being used for the human cells that generates normal enucleated reticulocytes. Although Ppara(-/-) mice show no haematological difference from wild-type mice in both normal and phenylhydrazine (PHZ)-induced stress erythropoiesis, PPAR-α agonists facilitate recovery of wild-type but not Ppara(-/-) mice from PHZ-induced acute haemolytic anaemia. We also show that PPAR-α alleviates anaemia in a mouse model of chronic anaemia. Finally, both in control and corticosteroid-treated BFU-E cells, PPAR-α co-occupies many chromatin sites with GR; when activated by PPAR-α agonists, additional PPAR-α is recruited to GR-adjacent sites and presumably facilitates GR-dependent BFU-E self-renewal. Our discovery of the role of PPAR-α agonists in stimulating self-renewal of early erythroid progenitor cells suggests that the clinically tested PPAR-α agonists we used may improve the efficacy of corticosteroids in treating Epo-resistant anaemias.
许多急性和慢性贫血,包括溶血、败血症以及诸如先天性纯红细胞再生障碍性贫血等遗传性骨髓衰竭疾病,都无法用促红细胞生成素(Epo)进行治疗,因为对Epo有反应的红系集落形成单位祖细胞(CFU-E)数量过少,或者对Epo不够敏感,无法维持足够的红细胞生成。治疗这些贫血需要一种作用于红细胞形成早期阶段并增强对Epo敏感的CFU-E祖细胞形成的药物。最近,我们发现糖皮质激素能特异性刺激早期红系祖细胞即爆式红系集落形成单位(BFU-E)的自我更新,并增加终末分化红系细胞的生成。在此我们表明,PPAR-α激动剂GW7647和非诺贝特激活过氧化物酶体增殖物激活受体α(PPAR-α)后,与糖皮质激素受体(GR)协同作用,促进BFU-E的自我更新。随着时间推移,这些激动剂能极大地增加小鼠胎肝BFU-E和动员的成人CD34(+)外周血祖细胞培养物中成熟红细胞的生成,并且一种新的有效培养系统被用于人类细胞,该系统能产生正常的去核网织红细胞。尽管Ppara(-/-)小鼠在正常和苯肼(PHZ)诱导的应激性红细胞生成过程中与野生型小鼠在血液学上没有差异,但PPAR-α激动剂能促进野生型小鼠而不是Ppara(-/-)小鼠从PHZ诱导的急性溶血性贫血中恢复。我们还表明,PPAR-α能缓解慢性贫血小鼠模型中的贫血症状。最后,在对照和皮质类固醇处理的BFU-E细胞中,PPAR-α都与GR共同占据许多染色质位点;当被PPAR-α激动剂激活时,额外的PPAR-α会被招募到GR相邻位点,推测这有助于GR依赖的BFU-E自我更新。我们对PPAR-α激动剂在刺激早期红系祖细胞自我更新中作用的发现表明,我们所使用的经过临床测试的PPAR-α激动剂可能会提高皮质类固醇治疗Epo抵抗性贫血的疗效。