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α-突触核蛋白可变剪接变体的淀粉样蛋白形成

Amyloid formation of alternatively spliced variants of α-synuclein.

作者信息

SanGiovanni Daniel Q, McGlinchey Ryan P, Lee Jennifer C

机构信息

Laboratory of Protein Conformation and Dynamics, Biochemistry and Biophysics Center, National Heart, Lung, and Blood Institute, National Institutes of Health, Bethesda, Maryland, USA.

出版信息

Protein Sci. 2025 Jul;34(7):e70195. doi: 10.1002/pro.70195.

Abstract

Parkinson's disease, dementia with Lewy bodies, and multiple system atrophy are disorders characterized by the presence of cytosolic α-synuclein (SNCA) amyloids. The gene SNCA is alternatively spliced, generating three variants of SNCA, missing exon 3 (SNCAΔ3) or 5 (SNCAΔ5), or both exons (SNCAΔ3Δ5). Despite purported upregulation in disease states, their pathological relevance is ill-defined. Here, we investigated the amyloid formation of alternatively spliced variants under physiological conditions. Aggregation kinetics, secondary structure, and fibril morphology of N-terminally acetylated SNCAΔ3, SNCAΔ5, and SNCAΔ3Δ5 were assessed by thioflavin-T fluorescence, circular dichroism spectroscopy, and transmission electron microscopy, respectively. Compared to SNCA, both SNCAΔ5 and SNCAΔ3Δ5 aggregate faster and adopt a more twisted fibril morphology, whereas SNCAΔ3 is more sensitive to solution conditions, exhibiting similar or modestly faster aggregation kinetics compared to SNCA. Cross-seeding experiments using spliced-variant fibrils and soluble SNCA showed that despite fibril morphological differences, SNCAΔ5 were competent seeds for SNCA, which is explained by their similar protease-K resistant regions. Contrastingly, neither SNCAΔ3 nor SNCAΔ3Δ5 fibrils cross-seed SNCA, indicating exon 3 (residues 41-54) is essential in modulating fibril structure. Notably, SNCA aggregation is stimulated by sub-stoichiometric amounts of soluble SNCAΔ5 and SNCAΔ3Δ5, but not SNCAΔ3, suggesting that exon 5 (residues 103-130) is more important in modulating aggregation kinetics. Taken together, we propose that alternatively spliced variants are pathogenic by exacerbating aggregation of the main SNCA isoform.

摘要

帕金森病、路易体痴呆和多系统萎缩是由胞质α-突触核蛋白(SNCA)淀粉样蛋白的存在所表征的疾病。基因SNCA可选择性剪接,产生三种SNCA变体,缺失外显子3(SNCAΔ3)或5(SNCAΔ5),或同时缺失两个外显子(SNCAΔ3Δ5)。尽管据称在疾病状态下会上调,但其病理相关性仍不明确。在这里,我们研究了在生理条件下选择性剪接变体的淀粉样蛋白形成。通过硫黄素-T荧光、圆二色光谱和透射电子显微镜分别评估了N端乙酰化的SNCAΔ3、SNCAΔ5和SNCAΔ3Δ5的聚集动力学、二级结构和纤维形态。与SNCA相比,SNCAΔ5和SNCAΔ3Δ5都聚集得更快,并呈现出更扭曲的纤维形态,而SNCAΔ3对溶液条件更敏感,与SNCA相比表现出相似或适度更快的聚集动力学。使用剪接变体纤维和可溶性SNCA进行的交叉接种实验表明,尽管纤维形态存在差异,但SNCAΔ5是SNCA的有效种子,这可以通过它们相似的蛋白酶K抗性区域来解释。相反,SNCAΔ3和SNCAΔ3Δ5纤维都不能交叉接种SNCA,表明外显子3(第41-54位氨基酸)在调节纤维结构中至关重要。值得注意的是,亚化学计量的可溶性SNCAΔ5和SNCAΔ3Δ5可刺激SNCA聚集,但SNCAΔ3则不能,这表明外显子5(第103-130位氨基酸)在调节聚集动力学方面更重要。综上所述,我们认为选择性剪接变体通过加剧主要SNCA异构体的聚集而具有致病性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c175/12168488/a5da80fc2afa/PRO-34-e70195-g001.jpg

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