Inokuchi K, Yamaguchi H, Tarusawa M, Futaki M, Hanawa H, Tanosaki S, Dan K
Division of Hematology, Department of Internal Medicine, Nippon Medical School, Tokyo, Japan.
Leukemia. 2002 Feb;16(2):170-7. doi: 10.1038/sj.leu.2402341.
Chronic myelogenous leukemia (CML) is characterized by the Philadelphia (Ph) chromosome and bcr/abl gene rearrangement which occurs in pluripotent hematopoietic progenitor cells expressing the c-kit receptor tyrosine kinase (KIT). To elucidate the biological properties of KIT in CML leukemogenesis, we performed analysis of alterations of the c-kit gene and functional analysis of altered KIT proteins. Gene alterations in the c-kit juxtamembrane domain of 80 CML cases were analyzed by reverse transcriptase and polymerase chain reaction-single strand conformation polymorphism (RT-PCR-SSCP). One case had an abnormality at codon 564 (AAT --> AAG, Asn --> Lys), and six cases had the same base abnormality at codon 541 (ATG --> CTG, Met --> Leu) in the juxtamembrane domain. Because the change from Met to Leu at codon 541 was a conservative one which was also observed in the normal population and normal tissues of CML patients, it probably represents a polymorphic variation. Although samples of hair roots and leukemic cells from the chronic phase of one CML patient showed no abnormality, an abnormality at codon 541 (ATG --> CTG, Met --> Leu) was found only at blastic crisis (BC) of this case. In the case with the abnormality at codon 564, the mutation was detected only in a sample of leukemic cells collected at BC. To examine the biological consequence and biological significance of these abnormalities, murine KIT(L540) and KIT(K563) expression vectors were introduced into interleukin-3 (IL-3)-dependent murine Ba/F3 cells to study their state of tyrosine phosphorylation and their growth rate. Ba/F3 cells expressing KIT(WT), KIT(L540) and KIT(K563) showed dose-dependent tyrosine phosphorylation after treatment with increasing concentrations of recombinant mouse stem cell factor (rmSCF). The cells expressing KIT(L540) and KIT(K563) were found to have greater tyrosine phosphorylation than cells expressing KIT(WT) at 0.1 and 1.0 ng/ml of rmSCF. The Ba/F3 cells expressing KIT(K563) proliferated in response to 0.1 and 1.0 ng/ml of rmSCF as well as IL-3. The Ba/F3 cells expressing KIT(L540)showed a relatively higher proliferative response to 0.1 ng/ml of rmSCF than the response of cells expressing KIT(WT). These mutations and in vitro functional analyses raise the possibility that the KIT abnormalities influence the white blood cell counts (P < 0.05) and survival (P < 0.04) of CML patients.
慢性粒细胞白血病(CML)的特征是存在费城(Ph)染色体以及bcr/abl基因重排,这种重排发生在表达c-kit受体酪氨酸激酶(KIT)的多能造血祖细胞中。为了阐明KIT在CML白血病发生中的生物学特性,我们对c-kit基因的改变进行了分析,并对改变后的KIT蛋白进行了功能分析。通过逆转录酶和聚合酶链反应-单链构象多态性(RT-PCR-SSCP)分析了80例CML病例c-kit近膜结构域的基因改变。1例在密码子564处存在异常(AAT→AAG,天冬酰胺→赖氨酸),6例在近膜结构域的密码子541处存在相同的碱基异常(ATG→CTG,甲硫氨酸→亮氨酸)。由于密码子541处由甲硫氨酸变为亮氨酸是一种保守变化,在CML患者的正常人群和正常组织中也有观察到,所以它可能代表一种多态性变异。虽然1例CML患者慢性期的发根和白血病细胞样本未显示异常,但仅在该病例的急变期(BC)发现密码子541处存在异常(ATG→CTG,甲硫氨酸→亮氨酸)。在密码子564处存在异常的病例中,仅在BC期采集的白血病细胞样本中检测到该突变。为了研究这些异常的生物学后果和生物学意义,将小鼠KIT(L540)和KIT(K563)表达载体导入依赖白细胞介素-3(IL-3)的小鼠Ba/F3细胞,以研究它们的酪氨酸磷酸化状态和生长速率。用浓度递增的重组小鼠干细胞因子(rmSCF)处理后,表达KIT(WT)、KIT(L540)和KIT(K563)的Ba/F3细胞呈现剂量依赖性酪氨酸磷酸化。在0.1和1.0 ng/ml的rmSCF作用下,发现表达KIT(L540)和KIT(K563)的细胞比表达KIT(WT)的细胞具有更高的酪氨酸磷酸化水平。表达KIT(K563)的Ba/F3细胞对0.1和1.0 ng/ml的rmSCF以及IL-3有增殖反应。表达KIT(L540)的Ba/F3细胞对0.1 ng/ml的rmSCF的增殖反应相对高于表达KIT(WT)的细胞。这些突变和体外功能分析增加了KIT异常影响CML患者白细胞计数(P<0.05)和生存率(P<0.04)的可能性。