Dana Farber/Boston Children's Cancer and Blood Disorders Center, Harvard Medical School, Boston, MA.
Blood. 2019 Mar 7;133(10):1071-1085. doi: 10.1182/blood-2018-10-844662. Epub 2019 Jan 22.
Myelodysplastic syndrome (MDS) typically presents in older adults with the acquisition of age-related somatic mutations, whereas MDS presenting in children and younger adults is more frequently associated with germline genetic predisposition. Germline predisposition is increasingly recognized in MDS presenting at older ages as well. Although each individual genetic disorder is rare, as a group, the genetic MDS disorders account for a significant subset of MDS in children and young adults. Because many patients lack overt syndromic features, genetic testing plays an important role in the diagnostic evaluation. This review provides an overview of syndromes associated with genetic predisposition to MDS, discusses implications for clinical evaluation and management, and explores scientific insights gleaned from the study of MDS predisposition syndromes. The effects of germline genetic context on the selective pressures driving somatic clonal evolution are explored. Elucidation of the molecular and genetic pathways driving clonal evolution may inform surveillance and risk stratification, and may lead to the development of novel therapeutic strategies.
骨髓增生异常综合征(MDS)通常发生在老年患者中,与获得与年龄相关的体细胞突变有关,而在儿童和年轻患者中发生的 MDS 更常与种系遗传易感性相关。随着年龄的增长,在 MDS 中也越来越多地认识到种系易感性。虽然每个个体遗传疾病都很少见,但作为一个群体,遗传 MDS 疾病占儿童和年轻患者 MDS 的重要部分。由于许多患者缺乏明显的综合征特征,因此基因检测在诊断评估中起着重要作用。本综述概述了与 MDS 遗传易感性相关的综合征,讨论了其对临床评估和管理的影响,并探讨了从 MDS 易感性综合征研究中获得的科学见解。探讨了种系遗传背景对驱动体细胞克隆进化的选择压力的影响。阐明驱动克隆进化的分子和遗传途径可能有助于监测和风险分层,并可能导致新的治疗策略的发展。