Biomedical Sciences Training Program, University of Florida College of Medicine, Gainesville, FL.
University of Florida Genetics Institute, Gainesville, FL.
Diabetes. 2022 Feb 1;71(2):350-358. doi: 10.2337/db21-0194.
Signal regulatory protein SIRPγ (CD172G) is expressed on the surface of lymphocytes, where it acts by engaging its ligand, CD47. SIRPG, which encodes SIRPγ, contains a nonsynonymous coding variant, rs6043409, which is significantly associated with risk for type 1 diabetes. SIRPG produces multiple transcript isoforms via alternative splicing, all encoding potentially functional proteins. We show that rs6043409 alters a predicted exonic splicing enhancer, resulting in significant shifts in the distribution of SIRPG transcript isoforms. All of these transcript isoforms produced protein upon transient expression in vitro. However, CRISPR/Cas9 targeting of one of the alternatively spliced exons in SIRPG eliminated all SIRPγ expression in Jurkat T cells. These targeted cells formed fewer cell-cell conjugates with each other than with wild-type Jurkat cells, expressed reduced levels of genes associated with CD47 signaling, and had significantly increased levels of cell-surface CD47. In primary CD4+ and CD8+ T cells, cell-surface SIRPγ levels in response to anti-CD3 stimulation varied quantitatively by rs6043409 genotype. Our results suggest that SIRPG is the most likely causative gene for type 1 diabetes risk in the 20p13 region and highlight the role of alternative splicing in lymphocytes in mediating the genetic risk for autoimmunity.
信号调节蛋白 SIRPγ(CD172G)表达在淋巴细胞表面,通过与其配体 CD47 结合发挥作用。SIRPG 编码 SIRPγ,包含一个非同义编码变异体 rs6043409,与 1 型糖尿病的风险显著相关。SIRPG 通过选择性剪接产生多种转录本异构体,所有这些异构体都编码潜在的功能性蛋白质。我们表明 rs6043409 改变了一个预测的外显子剪接增强子,导致 SIRPG 转录本异构体的分布发生显著变化。所有这些转录本异构体在体外瞬时表达时都产生了蛋白质。然而,CRISPR/Cas9 靶向 SIRPG 中的一个选择性剪接外显子,消除了 Jurkat T 细胞中的所有 SIRPγ 表达。这些靶向细胞彼此形成的细胞-细胞连接比野生型 Jurkat 细胞少,表达与 CD47 信号相关的基因水平降低,细胞表面 CD47 水平显著增加。在原代 CD4+和 CD8+T 细胞中,抗 CD3 刺激后细胞表面 SIRPγ 水平的变化与 rs6043409 基因型有关。我们的结果表明,SIRPG 是 20p13 区域 1 型糖尿病风险的最可能致病基因,并强调了选择性剪接在淋巴细胞中介导自身免疫遗传风险的作用。