Darley R L, Hoy T G, Baines P, Padua R A, Burnett A K
Department of Haematology, University of Wales College of Medicine, Cardiff, United Kingdom.
J Exp Med. 1997 Apr 7;185(7):1337-47. doi: 10.1084/jem.185.7.1337.
RAS mutations arise at high frequency (20-40%) in both acute myeloid leukemia and myelodysplastic syndrome (which is considered to be a manifestation of preleukemic disease). In each case, mutations arise predominantly at the N-RAS locus. These observations suggest a fundamental role for this oncogene in leukemogenesis. However, despite its obvious significance, little is known of how this key oncogene may subvert the process of hematopoiesis in human cells. Using CD34+ progenitor cells, we have modeled the preleukemic state by infecting these cells with amphotropic retrovirus expressing mutant N-RAS together with the selectable marker gene lacZ. Expression of the lacZ gene product, beta-galactosidase, allows direct identification and study of N-RAS-expressing cells by incubating infected cultures with a fluorogenic substrate for beta-galactosidase, which gives rise to a fluorescent signal within the infected cells. By using multiparameter flow cytometry, we have studied the ability of CD34+ cells expressing mutant N-RAS to undergo erythroid differentiation induced by erythropoietin. By this means, we have found that erythroid progenitor cells expressing mutant N-RAS exhibit a proliferative defect resulting in an increased cell doubling time and a decrease in the proportion of cells in S + G2M phase of the cell cycle. This is linked to a slowing in the rate of differentiation as determined by comparative cell-surface marker analysis and ultimate failure of the differentiation program at the late-erythroblast stage of development. The dyserythropoiesis was also linked to an increased tendency of the RAS-expressing cells to undergo programmed cell death during their differentiation program. This erythroid lineage dysplasia recapitulates one of the most common features of myelodysplastic syndrome, and for the first time provides a causative link between mutational activation of N-RAS and the pathogenesis of preleukemia.
RAS突变在急性髓系白血病和骨髓增生异常综合征(被认为是白血病前期疾病的一种表现)中高频出现(20%-40%)。在每种情况下,突变主要发生在N-RAS基因座。这些观察结果表明该癌基因在白血病发生过程中起重要作用。然而,尽管其意义明显,但对于这个关键癌基因如何颠覆人类细胞中的造血过程却知之甚少。利用CD34+祖细胞,我们通过用表达突变型N-RAS以及选择标记基因lacZ的双嗜性逆转录病毒感染这些细胞,模拟了白血病前期状态。lacZ基因产物β-半乳糖苷酶的表达,通过用β-半乳糖苷酶的荧光底物孵育感染培养物,使得能够直接鉴定和研究表达N-RAS的细胞,这会在感染细胞内产生荧光信号。通过使用多参数流式细胞术,我们研究了表达突变型N-RAS的CD34+细胞在促红细胞生成素诱导下进行红系分化的能力。通过这种方法,我们发现表达突变型N-RAS的红系祖细胞表现出增殖缺陷,导致细胞倍增时间增加,细胞周期S + G2M期的细胞比例降低。这与分化速率减慢有关,通过比较细胞表面标志物分析确定,并且在发育的晚幼红细胞阶段分化程序最终失败。红细胞生成异常也与表达RAS的细胞在其分化程序中发生程序性细胞死亡的倾向增加有关。这种红系谱系发育异常概括了骨髓增生异常综合征最常见的特征之一,并首次提供了N-RAS突变激活与白血病前期发病机制之间的因果联系。