雷诺氏综合征的遗传和功能分析涉及血管和免疫系统中的基因位点。

IF 11.1 Q1 CELL BIOLOGY Cell genomics Pub Date : 2024-08-09 DOI:10.1016/j.xgen.2024.100630
Anniina Tervi, Markus Ramste, Erik Abner, Paul Cheng, Jacqueline M Lane, Matthew Maher, Jesse Valliere, Vilma Lammi, Satu Strausz, Juha Riikonen, Trieu Nguyen, Gabriella E Martyn, Maya U Sheth, Fan Xia, Mauro Lago Docampo, Wenduo Gu, Tõnu Esko, Richa Saxena, Matti Pirinen, Aarno Palotie, Samuli Ripatti, Nasa Sinnott-Armstrong, Mark Daly, Jesse M Engreitz, Marlene Rabinovitch, Caroline A Heckman, Thomas Quertermous, Samuel E Jones, Hanna M Ollila
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引用次数: 0

摘要

雷诺氏综合征是一种自律神经失调症,暴露于寒冷环境会导致血管收缩和缺氧,尤其是在四肢。我们对四个队列进行了荟萃分析,发现了八个位点(ADRA2A、IRX1、NOS3、ACVR2A、TMEM51、PCDH10-DT、HLA 和 RAB6C),其中 ADRA2A、ACVR2A、NOS3、TMEM51 和 IRX1 与表达定量性状位点(eQTLs)共定位,尤其是在远端动脉。CRISPR 基因编辑进一步表明,ADRA2A 和 NOS3 基因座改变了基因表达,原位 RNAscope 则明确了 ADRA2A 在小血管中的特异性和 IRX1 在皮肤小毛细血管周围的特异性。寒冷环境下的功能性收缩试验显示,ADRA2A 缺失的平滑肌细胞收缩力较低,而 ADRA2A 表达的平滑肌细胞收缩力较高。总之,我们的研究凸显了全基因组关联测试与功能跟踪作为一种了解复杂疾病的方法的强大威力。研究结果表明,在雷诺氏综合征中,温度依赖性肾上腺素能信号通过 ADRA2A、IRX1 对微血管的影响、NOS3 对内皮细胞信号的影响以及 HLA 位点对免疫机制的影响。
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Genetic and functional analysis of Raynaud's syndrome implicates loci in vasculature and immunity.

Raynaud's syndrome is a dysautonomia where exposure to cold causes vasoconstriction and hypoxia, particularly in the extremities. We performed meta-analysis in four cohorts and discovered eight loci (ADRA2A, IRX1, NOS3, ACVR2A, TMEM51, PCDH10-DT, HLA, and RAB6C) where ADRA2A, ACVR2A, NOS3, TMEM51, and IRX1 co-localized with expression quantitative trait loci (eQTLs), particularly in distal arteries. CRISPR gene editing further showed that ADRA2A and NOS3 loci modified gene expression and in situ RNAscope clarified the specificity of ADRA2A in small vessels and IRX1 around small capillaries in the skin. A functional contraction assay in the cold showed lower contraction in ADRA2A-deficient and higher contraction in ADRA2A-overexpressing smooth muscle cells. Overall, our study highlights the power of genome-wide association testing with functional follow-up as a method to understand complex diseases. The results indicate temperature-dependent adrenergic signaling through ADRA2A, effects at the microvasculature by IRX1, endothelial signaling by NOS3, and immune mechanisms by the HLA locus in Raynaud's syndrome.

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