Department of Translational Hematology and Oncology Research, Taussig Cancer Institute, Cleveland Clinic Foundation, Cleveland, OH, USA.
Department of Hematology, National Hospital Organization Kyoto Medical Center, Kyoto, Japan.
Leukemia. 2023 Oct;37(10):2082-2093. doi: 10.1038/s41375-023-02003-x. Epub 2023 Aug 26.
Complete or partial deletions of chromosome 7 (-7/del7q) belong to the most frequent chromosomal abnormalities in myeloid neoplasm (MN) and are associated with a poor prognosis. The disease biology of -7/del7q and the genes responsible for the leukemogenic properties have not been completely elucidated. Chromosomal deletions may create clonal vulnerabilities due to haploinsufficient (HI) genes contained in the deleted regions. Therefore, HI genes are potential targets of synthetic lethal strategies. Through the most comprehensive multimodal analysis of more than 600 -7/del7q MN samples, we elucidated the disease biology and qualified a list of most consistently deleted and HI genes. Among them, 27 potentially synthetic lethal target genes were identified with the following properties: (i) unaffected genes by hemizygous/homozygous LOF mutations; (ii) prenatal lethality in knockout mice; and (iii) vulnerability of leukemia cells by CRISPR and shRNA knockout screens. In -7/del7q cells, we also identified 26 up or down-regulated genes mapping on other chromosomes as downstream pathways or compensation mechanisms. Our findings shed light on the pathogenesis of -7/del7q MNs, while 27 potential synthetic lethal target genes and 26 differential expressed genes allow for a therapeutic window of -7/del7q.
7 号染色体完全或部分缺失(-7/del7q)属于骨髓性肿瘤(MN)中最常见的染色体异常之一,并与预后不良相关。-7/del7q 的疾病生物学和导致白血病的基因尚未完全阐明。由于缺失区域中包含的杂合不足(HI)基因,染色体缺失可能会产生克隆脆弱性。因此,HI 基因是合成致死策略的潜在靶点。通过对 600 多个 -7/del7q MN 样本进行最全面的多模态分析,我们阐明了疾病生物学,并确定了一组最一致缺失和 HI 基因的清单。其中,有 27 个潜在的合成致死靶基因具有以下特性:(i)不受杂合/纯合 LOF 突变影响的基因;(ii)在敲除小鼠中具有产前致死性;(iii)CRISPR 和 shRNA 敲除筛选使白血病细胞脆弱。在 -7/del7q 细胞中,我们还鉴定了 26 个映射到其他染色体上的上调或下调基因,作为下游途径或补偿机制。我们的发现揭示了 -7/del7q MN 的发病机制,而 27 个潜在的合成致死靶基因和 26 个差异表达基因为 -7/del7q 提供了治疗窗口。