Williams C J, Grandal I, Vesprini D J, Wojtyra U, Danska J S, Guidos C J
Hospital for Sick Children Research Institute and Department of Immunology, University of Toronto, Toronto, Ontario, Canada.
Mol Cell Biol. 2001 Jan;21(2):400-13. doi: 10.1128/MCB.21.2.400-413.2001.
Defects in the nonhomologous end-joining (NHEJ) pathway of double-stranded DNA break repair severely impair V(D)J joining and selectively predispose mice to the development of lymphoid neoplasia. This connection was first noted in mice with the severe combined immune deficient (SCID) mutation in the DNA-dependent protein kinase (DNA-PK). SCID mice spontaneously develop thymic lymphoma with low incidence and long latency. However, we and others showed that low-dose irradiation of SCID mice dramatically increases the frequency and decreases the latency of thymic lymphomagenesis, but irradiation does not promote the development of other tumors. We have used this model to explore the mechanistic basis by which defects in NHEJ confer selective and profound susceptibility to lymphoid oncogenesis. Here, we show that radiation quantitatively and qualitatively improves V(D)J joining in SCID cells, in the absence of T-cell receptor-mediated cellular selection. Furthermore, we show that the lymphocyte-specific endonuclease encoded by the recombinase-activating genes (RAG-1 and RAG-2) is required for radiation-induced thymic lymphomagenesis in SCID mice. Collectively, these data suggest that irradiation induces a DNA-PK-independent NHEJ pathway that facilitates V(D)J joining, but also promotes oncogenic misjoining of RAG-1/2-induced breaks in SCID T-cell precursors.
双链DNA断裂修复的非同源末端连接(NHEJ)途径中的缺陷严重损害V(D)J连接,并选择性地使小鼠易患淋巴样肿瘤。这种联系最初在DNA依赖性蛋白激酶(DNA-PK)中具有严重联合免疫缺陷(SCID)突变的小鼠中被发现。SCID小鼠自发发生胸腺淋巴瘤的发生率低且潜伏期长。然而,我们和其他人表明,低剂量照射SCID小鼠会显著增加胸腺淋巴瘤发生的频率并缩短潜伏期,但照射不会促进其他肿瘤的发生。我们利用这个模型来探索NHEJ缺陷导致对淋巴样肿瘤发生具有选择性和深刻易感性的机制基础。在这里,我们表明,在没有T细胞受体介导的细胞选择的情况下,辐射在数量和质量上改善了SCID细胞中的V(D)J连接。此外,我们表明,重组激活基因(RAG-1和RAG-2)编码的淋巴细胞特异性核酸内切酶是SCID小鼠辐射诱导的胸腺淋巴瘤发生所必需的。总体而言,这些数据表明,辐射诱导了一种不依赖DNA-PK的NHEJ途径,该途径促进V(D)J连接,但也促进了SCID T细胞前体中RAG-1/2诱导的断裂的致癌性错误连接。