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人类疾病中的显性Gα突变:统一机制与治疗策略

Dominant Gα mutations in human disease: unifying mechanisms and treatment strategies.

作者信息

Katanaev Vladimir L, Solis Gonzalo P

机构信息

Translational Research Center in Oncohaematology, Department of Cell Physiology and Metabolism, Faculty of Medicine, University of Geneva, Geneva, CH-1211, Switzerland.

Translational Oncology Research Center, Qatar Biomedical Research Institute (QBRI), College of Health and Life Sciences, Hamad Bin Khalifa University (HBKU), Qatar Foundation, Doha, PO Box 34110, Qatar.

出版信息

EMBO Mol Med. 2025 Jul 31. doi: 10.1038/s44321-025-00274-8.

DOI:10.1038/s44321-025-00274-8
PMID:40745211
Abstract

Sixteen Gα-subunits transduce hundreds of G protein-coupled receptors and control countless cellular activities. Mutations in respective GNA genes underlie developmental, oncological, metabolic, neurological, and other pathologies. In addition to classical loss-of-function (LOF) and gain-of-function (GOF) mutations (the former represented by gene deletions/truncations, the latter by specific GTP hydrolysis-deficient mutations), multiple pathogenic dominant missense variants have been discovered in GNA genes, and their numbers constantly increase through advanced genetic diagnostics. While these mutations often have confusing features of hypomorphic, dominant-negative, and GOF mutations, many of the pathogenic Gαo (and by inference, other Gα-subunit) variants have recently emerged as neomorphic, i.e., leading to the creation of novel dominant pathogenic functions. Cross-family analysis of these missense variants scattered across GNA genes permits establishing mutational signatures underlying a wide range of Gα-pathies. These mutation patterns have a strong predictive power in the following aspects. First, new dominant mutations in further GNA genes will be discovered in rare diseases. Second, unifying mechanisms of pathogenic dominance emerge in different Gα-subunits. And third, drug(s) acting against some Gα-pathies may prove effective against others.

摘要

16种Gα亚基转导数百种G蛋白偶联受体并控制无数细胞活动。各个GNA基因的突变是发育、肿瘤、代谢、神经及其他疾病的基础。除了经典的功能丧失(LOF)和功能获得(GOF)突变(前者以基因缺失/截短为代表,后者以特定的GTP水解缺陷突变 为代表)外,在GNA基因中还发现了多种致病性显性错义变体,并且通过先进的基因诊断,它们的数量不断增加。虽然这些突变通常具有低表达、显性负性和GOF突变的混淆特征,但许多致病性Gαo(据推断,其他Gα亚基)变体最近已成为新形态的,即导致产生新的显性致病功能。对分散在GNA基因中的这些错义变体进行跨家族分析,有助于确定广泛的Gα疾病潜在的突变特征。这些突变模式在以下方面具有强大的预测能力。第一,在罕见病中将会发现更多GNA基因中的新显性突变。第二,不同Gα亚基出现致病性显性的统一机制。第三,针对某些Gα疾病的药物可能对其他疾病也有效。

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Clinical-molecular profiling of atypical patients: Novel pathogenic variants, unusual manifestations, and severe molecular dysfunction.非典型患者的临床分子特征分析:新型致病变异、不寻常表现及严重分子功能障碍
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GNAO1患者的表型多样性:变异与表型的全面概述
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Germline mutations in a G protein identify signaling cross-talk in T cells.胚系突变的 G 蛋白鉴定了 T 细胞中的信号串扰。
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Loss-of-function Thr347Ala Variant in the G Protein Subunit-Α11 Causes Familial Hypocalciuric Hypercalcemia 2.G蛋白亚基-α11中功能丧失性的苏氨酸347丙氨酸变体导致家族性低钙血症性高钙血症2型。
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