Department of Molecular Biology and Biochemistry, Simon Fraser University, Burnaby, BC, Canada.
Office of Cancer Genomics, National Cancer Institute, National Institutes of Health, Bethesda, MD.
Blood. 2023 Feb 23;141(8):904-916. doi: 10.1182/blood.2022016534.
Burkitt lymphoma (BL) accounts for most pediatric non-Hodgkin lymphomas, being less common but significantly more lethal when diagnosed in adults. Much of the knowledge of the genetics of BL thus far has originated from the study of pediatric BL (pBL), leaving its relationship to adult BL (aBL) and other adult lymphomas not fully explored. We sought to more thoroughly identify the somatic changes that underlie lymphomagenesis in aBL and any molecular features that associate with clinical disparities within and between pBL and aBL. Through comprehensive whole-genome sequencing of 230 BL and 295 diffuse large B-cell lymphoma (DLBCL) tumors, we identified additional significantly mutated genes, including more genetic features that associate with tumor Epstein-Barr virus status, and unraveled new distinct subgroupings within BL and DLBCL with 3 predominantly comprising BLs: DGG-BL (DDX3X, GNA13, and GNAI2), IC-BL (ID3 and CCND3), and Q53-BL (quiet TP53). Each BL subgroup is characterized by combinations of common driver and noncoding mutations caused by aberrant somatic hypermutation. The largest subgroups of BL cases, IC-BL and DGG-BL, are further characterized by distinct biological and gene expression differences. IC-BL and DGG-BL and their prototypical genetic features (ID3 and TP53) had significant associations with patient outcomes that were different among aBL and pBL cohorts. These findings highlight shared pathogenesis between aBL and pBL, and establish genetic subtypes within BL that serve to delineate tumors with distinct molecular features, providing a new framework for epidemiologic, diagnostic, and therapeutic strategies.
伯基特淋巴瘤(BL)占大多数儿科非霍奇金淋巴瘤,在成人中诊断时较为罕见,但致死率显著更高。迄今为止,BL 的遗传学知识主要源于儿科 BL(pBL)的研究,因此其与成人 BL(aBL)和其他成人淋巴瘤的关系尚未完全探索。我们试图更彻底地确定 aBL 中淋巴发生的体细胞变化以及与 pBL 和 aBL 内和之间临床差异相关的任何分子特征。通过对 230 例 BL 和 295 例弥漫性大 B 细胞淋巴瘤(DLBCL)肿瘤进行全面全基因组测序,我们确定了其他显著突变基因,包括更多与肿瘤 EBV 状态相关的遗传特征,并在 BL 和 DLBCL 中揭示了新的不同亚组,其中 3 个主要由 BL 组成:DGG-BL(DDX3X、GNA13 和 GNAI2)、IC-BL(ID3 和 CCND3)和 Q53-BL(quiet TP53)。每个 BL 亚组的特征是由异常体细胞超突变引起的常见驱动基因和非编码突变的组合。BL 病例中最大的两个亚组,IC-BL 和 DGG-BL,进一步以不同的生物学和基因表达差异为特征。IC-BL 和 DGG-BL 及其原型遗传特征(ID3 和 TP53)与 aBL 和 pBL 队列中不同的患者结局有显著关联。这些发现突出了 aBL 和 pBL 之间的共同发病机制,并在 BL 内建立了遗传亚型,用于描绘具有不同分子特征的肿瘤,为流行病学、诊断和治疗策略提供了新的框架。