Department of Pediatric Oncology and the Linde Program in Cancer Chemical Biology, Dana-Farber Cancer Institute, Boston, MA;
Blood. 2014 Feb 6;123(6):884-93. doi: 10.1182/blood-2013-04-499079. Epub 2013 Dec 18.
Mantle cell lymphoma (MCL) is a highly aggressive B-cell lymphoma resistant to conventional chemotherapy. Although defined by the characteristic t(11;14) translocation, MCL has not been recapitulated in transgenic mouse models of cyclin D1 overexpression alone. Indeed, several genetic aberrations have been identified in MCL that may contribute to its pathogenesis and chemoresistance. Of particular interest is the frequent biallelic deletion of the proapoptotic BCL-2 family protein BIM. BIM exerts its pro-death function via its α-helical BH3 death domain that has the dual capacity to inhibit antiapoptotic proteins such as BCL-2 and MCL-1 and directly trigger proapoptotic proteins such as the mitochondrial executioner protein BAX. To evaluate a functional role for Bim deletion in the pathogenesis of MCL, we generated cyclin D1-transgenic mice harboring Bim-deficient B cells. In response to immunization, Eμ(CycD1)CD19(CRE)Bim(fl/fl) mice manifested selective expansion of their splenic mantle zone compartment. Three distinct immune stimulation regimens induced lymphomas with histopathologic and molecular features of human MCL in a subset of mice. Thus, deletion of Bim in B cells, in the context of cyclin D1 overexpression, disrupts a critical control point in lymphoid maturation and predisposes to the development of MCL. This genetic proof of concept for MCL pathogenesis suggests an opportunity to reactivate the death pathway by pharmacologic mimicry of proapoptotic BIM.
套细胞淋巴瘤(Mantle cell lymphoma,MCL)是一种高度侵袭性的 B 细胞淋巴瘤,对常规化疗具有耐药性。尽管 MCL 以特征性的 t(11;14)易位为定义,但在单独过表达 cyclin D1 的转基因小鼠模型中并未重现。事实上,在 MCL 中已经鉴定出几种遗传异常,这些异常可能有助于其发病机制和化疗耐药性。特别引人关注的是促凋亡 BCL-2 家族蛋白 BIM 的频繁双等位基因缺失。BIM 通过其 α-螺旋 BH3 死亡结构域发挥其促凋亡功能,该结构域具有双重能力,既能抑制抗凋亡蛋白(如 BCL-2 和 MCL-1),又能直接触发促凋亡蛋白(如线粒体执行者蛋白 BAX)。为了评估 Bim 缺失在 MCL 发病机制中的功能作用,我们生成了携带 Bim 缺失 B 细胞的 cyclin D1 转基因小鼠。在免疫后,Eμ(CycD1)CD19(CRE)Bim(fl/fl) 小鼠表现出其脾脏套区隔间的选择性扩张。三种不同的免疫刺激方案在一部分小鼠中诱导了具有人类 MCL 的组织病理学和分子特征的淋巴瘤。因此,在 cyclin D1 过表达的情况下,B 细胞中 Bim 的缺失破坏了淋巴细胞成熟的一个关键控制点,并使 MCL 的发展易于发生。这种 MCL 发病机制的遗传概念验证表明,通过模拟促凋亡 BIM,可以有机会重新激活凋亡途径。