Decoding Missense Variants by Incorporating Phase Separation via Machine Learning.


Journal

Nature communications
ISSN: 2041-1723
Titre abrégé: Nat Commun
Pays: England
ID NLM: 101528555

Informations de publication

Date de publication:
27 Sep 2024
Historique:
received: 28 12 2023
accepted: 12 09 2024
medline: 28 9 2024
pubmed: 28 9 2024
entrez: 27 9 2024
Statut: epublish

Résumé

Computational models have made significant progress in predicting the effect of protein variants. However, deciphering numerous variants of uncertain significance (VUS) located within intrinsically disordered regions (IDRs) remains challenging. To address this issue, we introduce phase separation, which is tightly linked to IDRs, into the investigation of missense variants. Phase separation is vital for multiple physiological processes. By leveraging missense variants that alter phase separation propensity, we develop a machine learning approach named PSMutPred to predict the impact of missense mutations on phase separation. PSMutPred demonstrates robust performance in predicting missense variants that affect natural phase separation. In vitro experiments further underscore its validity. By applying PSMutPred on over 522,000 ClinVar missense variants, it significantly contributes to decoding the pathogenesis of disease variants, especially those in IDRs. Our work provides insights into the understanding of a vast number of VUSs in IDRs, expediting clinical interpretation and diagnosis.

Identifiants

pubmed: 39333476
doi: 10.1038/s41467-024-52580-3
pii: 10.1038/s41467-024-52580-3
doi:

Substances chimiques

Intrinsically Disordered Proteins 0

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

8279

Informations de copyright

© 2024. The Author(s).

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Auteurs

Mofan Feng (M)

Bio-X Institutes, Key Laboratory for the Genetics of Developmental and Neuropsychiatric Disorders, Shanghai Jiao Tong University, Shanghai, China.
The Collaborative Innovation Center for Brain Science, and Brain Science and Technology Research Center, Shanghai Jiao Tong University, Shanghai, China.

Xiaoxi Wei (X)

Bio-X Institutes, Key Laboratory for the Genetics of Developmental and Neuropsychiatric Disorders, Shanghai Jiao Tong University, Shanghai, China.

Xi Zheng (X)

Bio-X Institutes, Key Laboratory for the Genetics of Developmental and Neuropsychiatric Disorders, Shanghai Jiao Tong University, Shanghai, China.
The Collaborative Innovation Center for Brain Science, and Brain Science and Technology Research Center, Shanghai Jiao Tong University, Shanghai, China.

Liangjie Liu (L)

Bio-X Institutes, Key Laboratory for the Genetics of Developmental and Neuropsychiatric Disorders, Shanghai Jiao Tong University, Shanghai, China.
The Collaborative Innovation Center for Brain Science, and Brain Science and Technology Research Center, Shanghai Jiao Tong University, Shanghai, China.

Lin Lin (L)

Bio-X Institutes, Key Laboratory for the Genetics of Developmental and Neuropsychiatric Disorders, Shanghai Jiao Tong University, Shanghai, China.

Manying Xia (M)

Bio-X Institutes, Key Laboratory for the Genetics of Developmental and Neuropsychiatric Disorders, Shanghai Jiao Tong University, Shanghai, China.

Guang He (G)

Bio-X Institutes, Key Laboratory for the Genetics of Developmental and Neuropsychiatric Disorders, Shanghai Jiao Tong University, Shanghai, China. heguang@sjtu.edu.cn.
The Collaborative Innovation Center for Brain Science, and Brain Science and Technology Research Center, Shanghai Jiao Tong University, Shanghai, China. heguang@sjtu.edu.cn.

Yi Shi (Y)

Bio-X Institutes, Key Laboratory for the Genetics of Developmental and Neuropsychiatric Disorders, Shanghai Jiao Tong University, Shanghai, China. yishi@sjtu.edu.cn.
The Collaborative Innovation Center for Brain Science, and Brain Science and Technology Research Center, Shanghai Jiao Tong University, Shanghai, China. yishi@sjtu.edu.cn.

Qing Lu (Q)

Bio-X Institutes, Key Laboratory for the Genetics of Developmental and Neuropsychiatric Disorders, Shanghai Jiao Tong University, Shanghai, China. luqing67@sjtu.edu.cn.
Department of Otorhinolaryngology-Head and Neck Surgery, Chongqing General Hospital, Chongqing, China. luqing67@sjtu.edu.cn.
Ear Institute, Shanghai Jiao Tong University School of Medicine, Shanghai, China. luqing67@sjtu.edu.cn.
Shanghai Key Laboratory of Translational Medicine on Ear and Nose Diseases, Shanghai, China. luqing67@sjtu.edu.cn.

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