Howard Hughes Medical Institute, Harvard Medical School, Boston, Massachusetts 02115, USA.
Nature. 2009 Dec 10;462(7274):803-7. doi: 10.1038/nature08633.
B-cell malignancies, such as human Burkitt's lymphoma, often contain translocations that link c-myc or other proto-oncogenes to the immunoglobulin heavy chain locus (IgH, encoded by Igh). The nature of elements that activate oncogenes within such translocations has been a long-standing question. Translocations within Igh involve DNA double-strand breaks initiated either by the RAG1/2 endonuclease during variable, diversity and joining gene segment (V(D)J) recombination, or by activation-induced cytidine deaminase (AID, also known as AICDA) during class switch recombination (CSR). V(D)J recombination in progenitor B (pro-B) cells assembles Igh variable region exons upstream of mu constant region (Cmu) exons, which are the first of several sets of C(H) exons ('C(H) genes') within a C(H) locus that span several hundred kilobases (kb). In mature B cells, CSR deletes Cmu and replaces it with a downstream C(H) gene. An intronic enhancer (iEmu) between the variable region exons and Cmu promotes V(D)J recombination in developing B cells. Furthermore, the Igh 3' regulatory region (Igh3'RR) lies downstream of the C(H) locus and modulates CSR by long-range transcriptional enhancement of C(H) genes. Transgenic mice bearing iEmu or Igh3'RR sequences fused to c-myc are predisposed to B lymphomas, demonstrating that such elements can confer oncogenic c-myc expression. However, in many B-cell lymphomas, Igh-c-myc translocations delete iEmu and place c-myc up to 200 kb upstream of the Igh3'RR. Here we address the oncogenic role of the Igh3'RR by inactivating it in two distinct mouse models for B-cell lymphoma with Igh-c-myc translocations. We show that the Igh3'RR is dispensable for pro-B-cell lymphomas with V(D)J recombination-initiated translocations, but is required for peripheral B-cell lymphomas with CSR-associated translocations. As the Igh3'RR is not required for CSR-associated Igh breaks or Igh-c-myc translocations in peripheral B-cell lymphoma progenitors, we conclude that this regulatory region confers oncogenic activity by long-range and developmental stage-specific activation of translocated c-myc genes.
B 细胞恶性肿瘤,如人类伯基特淋巴瘤,通常含有将 c-myc 或其他原癌基因与免疫球蛋白重链基因座(IgH,由 Igh 编码)连接起来的易位。在这种易位中激活癌基因的元件的性质一直是一个长期存在的问题。Igh 内的易位涉及由 RAG1/2 内切酶在可变、多样性和连接基因片段(V(D)J)重组过程中引发的 DNA 双链断裂,或由激活诱导的胞嘧啶脱氨酶(AID,也称为 AICDA)在类别转换重组(CSR)过程中引发。祖细胞 B(pro-B)细胞中的 V(D)J 重组将 IgH 可变区外显子组装到 mu 恒定区(Cmu)外显子的上游,这是几个 C(H) 外显子(“C(H) 基因”)中的第一个,跨越数百个千碱基(kb)。在成熟 B 细胞中,CSR 删除 Cmu 并将其替换为下游的 C(H) 基因。可变区外显子和 Cmu 之间的内含子增强子(iEmu)促进发育中的 B 细胞中的 V(D)J 重组。此外,Igh 3' 调控区(Igh3'RR)位于 C(H) 基因座的下游,通过远距离转录增强 C(H) 基因来调节 CSR。携带 iEmu 或 Igh3'RR 序列融合到 c-myc 的转基因小鼠易患 B 淋巴瘤,表明此类元件可赋予致癌 c-myc 表达。然而,在许多 B 细胞淋巴瘤中,Igh-c-myc 易位删除 iEmu,并将 c-myc 放置在 Igh3'RR 上游多达 200 kb 处。在这里,我们通过在具有 Igh-c-myc 易位的两种不同的 B 细胞淋巴瘤小鼠模型中使 Igh3'RR 失活来解决其致癌作用。我们表明,Igh3'RR 对于由 V(D)J 重组引发的前 B 细胞淋巴瘤是可有可无的,但对于 CSR 相关易位的外周 B 细胞淋巴瘤是必需的。由于 Igh3'RR 在外周 B 细胞淋巴瘤祖细胞中的 CSR 相关 Igh 断裂或 Igh-c-myc 易位中不是必需的,因此我们得出结论,该调节区通过远距离和发育阶段特异性激活易位的 c-myc 基因赋予致癌活性。