Jeong Jun-Hyeon, Jo Areum, Park Pilgu, Lee Hyunsook, Lee Hae-Ock
Department of Biological Sciences and Institute of Molecular Biology and Genetics, Seoul National University, Seoul 151-742, Korea.
Samsung Genome Institute, Samsung Medical Center, Seoul 135-710, Korea.
Mol Cells. 2015 Mar;38(3):251-8. doi: 10.14348/molcells.2015.2302. Epub 2015 Feb 4.
Germline mutations in the breast cancer type 2 susceptibility gene (BRCA2) are linked to familial breast cancer and the progressive bone marrow failure syndrome Fanconi anaemia. Established Brca2 mouse knockout models show embryonic lethality, but those with a truncating mutation at the C-terminus survive to birth and develop thymic lymphoma at an early age. To overcome early lethality and investigate the function of BRCA2, we used T cell-specific conditional Brca2 knockout mice, which were previously shown to develop thymic lymphoma at a low penetrance. In the current study we showed that the number of peripheral T cells, particularly naïve pools, drastically declined with age. This decline was primarily ascribed to improper peripheral maintenance. Furthermore, heterozygous mice with one wild-type Brca2 allele manifested reduced T cell numbers, suggesting that Brca2 haploinsufficiency might also result in T cell loss. Our study reveals molecular events occurring in Brca2-deficient T cells and suggests that both heterozygous and homozygous Brca2 mutation may lead to dysfunction in T cell populations.
乳腺癌2型易感基因(BRCA2)的种系突变与家族性乳腺癌以及进行性骨髓衰竭综合征范科尼贫血有关。已建立的Brca2基因敲除小鼠模型表现出胚胎致死性,但那些在C端有截短突变的小鼠能存活至出生,并在幼年时发展为胸腺淋巴瘤。为了克服早期致死性并研究BRCA2的功能,我们使用了T细胞特异性条件性Brca2基因敲除小鼠,此前已证明这些小鼠会以低外显率发展为胸腺淋巴瘤。在当前研究中,我们发现外周T细胞的数量,尤其是初始T细胞库,会随着年龄的增长而急剧下降。这种下降主要归因于外周维持不当。此外,携带一个野生型Brca2等位基因的杂合小鼠表现出T细胞数量减少,这表明Brca2单倍剂量不足也可能导致T细胞丢失。我们的研究揭示了Brca2缺陷型T细胞中发生的分子事件,并表明杂合和纯合Brca2突变都可能导致T细胞群体功能障碍。