Makishima Hideki
Pathology and Tumor Biology, Graduate School of Medicine, Kyoto University.
Rinsho Ketsueki. 2021;62(4):278-288. doi: 10.11406/rinketsu.62.278.
Recent advances in sequencing technologies have increased the detection rate for identifying germline mutations that predispose an individual to various myeloid neoplasms and somatic mutations acquired during progression from myelodysplastic syndromes (MDS) to acute myeloid leukemia (AML). In addition to pediatric subjects, adult patients were analyzed in order to obtain a complete spectrum of driver mutations in germline cells and/or somatic tumor samples. As shown in several recent studies, such driver mutations are acquired in a gene-specific fashion. DDX41 mutations are observed in germline cells long before MDS presentation. SAMD9/SAMD9L germline mutations associated with defective hematopoiesis account for recurrent and familial -7/del (7q) lesions, which result in the removal of the disadvantageous allele. Additionally, MDS cases in younger population display compound heterozygous germline mutations in the Shwachman-Diamond syndrome-associated SBDS gene. In peripheral blood samples from healthy elderly individuals, DNMT3A, TET2, and ASXL1 somatic mutations are usually detected due to age-related clonal hematopoiesis and are considered to be a risk factor for hematological neoplasms. In MDS, mutations of genes, such as NRAS and FLT3, designated as type-1 genes, are significantly associated with leukemic evolution. On the other hand, mutations in type-2 genes, including RUNX1 and GATA2, are related to progression from low risk MDS to high risk MDS.
测序技术的最新进展提高了对种系突变的检测率,这些突变使个体易患各种髓系肿瘤,以及在从骨髓增生异常综合征(MDS)进展为急性髓系白血病(AML)过程中获得的体细胞突变。除了儿科受试者外,还对成年患者进行了分析,以获得种系细胞和/或体细胞肿瘤样本中驱动突变的完整谱。正如最近的几项研究所表明的,这种驱动突变是以基因特异性方式获得的。在MDS出现之前很久就在种系细胞中观察到DDX41突变。与造血功能缺陷相关的SAMD9/SAMD9L种系突变导致复发性和家族性-7/del(7q)病变,从而导致不利等位基因的去除。此外,年轻人群中的MDS病例在与施瓦茨曼-戴蒙德综合征相关的SBDS基因中表现出复合杂合种系突变。在健康老年人的外周血样本中,由于年龄相关的克隆性造血,通常会检测到DNMT3A、TET2和ASXL1体细胞突变,这些突变被认为是血液肿瘤的危险因素。在MDS中,被指定为1型基因的NRAS和FLT3等基因突变与白血病进展显著相关。另一方面,包括RUNX1和GATA2在内的2型基因突变与从低风险MDS进展为高风险MDS有关。