Dynamicasome—a molecular dynamics-guided and AI-driven pathogenicity prediction catalogue for all genetic mutations

Abstract Advances in genomic medicine accelerate the identification of mutations in disease-associated genes, but the pathogenicity of many mutations remains unknown, hindering their use in diagnostics and clinical decision-making. Predictive AI models are generated to combat this issue, but current...

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Main Authors: Naeyma N. Islam, Mathew A. Coban, Jessica M. Fuller, Caleb Weber, Rohit Chitale, Benjamin Jussila, Trisha J. Brock, Cui Tao, Thomas R. Caulfield
Format: Article
Language:English
Published: Nature Portfolio 2025-07-01
Series:Communications Biology
Online Access:https://doi.org/10.1038/s42003-025-08334-y
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author Naeyma N. Islam
Mathew A. Coban
Jessica M. Fuller
Caleb Weber
Rohit Chitale
Benjamin Jussila
Trisha J. Brock
Cui Tao
Thomas R. Caulfield
author_facet Naeyma N. Islam
Mathew A. Coban
Jessica M. Fuller
Caleb Weber
Rohit Chitale
Benjamin Jussila
Trisha J. Brock
Cui Tao
Thomas R. Caulfield
author_sort Naeyma N. Islam
collection DOAJ
description Abstract Advances in genomic medicine accelerate the identification of mutations in disease-associated genes, but the pathogenicity of many mutations remains unknown, hindering their use in diagnostics and clinical decision-making. Predictive AI models are generated to combat this issue, but current tools display low accuracy when tested against functionally validated datasets. We show that integrating detailed conformational data extracted from molecular dynamics simulations (MDS) into advanced AI-based models increases their predictive power. We carry out an exhaustive mutational analysis of the disease gene PMM2 and subject structural models of each variant to MDS. AI models trained on this dataset outperform existing tools when predicting the known pathogenicity of mutations. Our best performing model, a neuronal networks model, also predicts the pathogenicity of several PMM2 mutations currently considered of unknown significance. We believe this model helps alleviate the burden of unknown variants in genomic medicine.
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issn 2399-3642
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spelling doaj-art-328700c89ddc4e979e735b90860136212025-08-20T03:06:01ZengNature PortfolioCommunications Biology2399-36422025-07-018111410.1038/s42003-025-08334-yDynamicasome—a molecular dynamics-guided and AI-driven pathogenicity prediction catalogue for all genetic mutationsNaeyma N. Islam0Mathew A. Coban1Jessica M. Fuller2Caleb Weber3Rohit Chitale4Benjamin Jussila5Trisha J. Brock6Cui Tao7Thomas R. Caulfield8Department of Neuroscience, Mayo ClinicDepartment of Neuroscience, Mayo ClinicDepartment of Neuroscience, Mayo ClinicDepartment of Neuroscience, Mayo ClinicDepartment of Infectious Disease, Mayo ClinicInVivo Biosystems, Inc.InVivo Biosystems, Inc.Department of Artificial Intelligence and Informatics, Mayo ClinicDepartment of Neuroscience, Mayo ClinicAbstract Advances in genomic medicine accelerate the identification of mutations in disease-associated genes, but the pathogenicity of many mutations remains unknown, hindering their use in diagnostics and clinical decision-making. Predictive AI models are generated to combat this issue, but current tools display low accuracy when tested against functionally validated datasets. We show that integrating detailed conformational data extracted from molecular dynamics simulations (MDS) into advanced AI-based models increases their predictive power. We carry out an exhaustive mutational analysis of the disease gene PMM2 and subject structural models of each variant to MDS. AI models trained on this dataset outperform existing tools when predicting the known pathogenicity of mutations. Our best performing model, a neuronal networks model, also predicts the pathogenicity of several PMM2 mutations currently considered of unknown significance. We believe this model helps alleviate the burden of unknown variants in genomic medicine.https://doi.org/10.1038/s42003-025-08334-y
spellingShingle Naeyma N. Islam
Mathew A. Coban
Jessica M. Fuller
Caleb Weber
Rohit Chitale
Benjamin Jussila
Trisha J. Brock
Cui Tao
Thomas R. Caulfield
Dynamicasome—a molecular dynamics-guided and AI-driven pathogenicity prediction catalogue for all genetic mutations
Communications Biology
title Dynamicasome—a molecular dynamics-guided and AI-driven pathogenicity prediction catalogue for all genetic mutations
title_full Dynamicasome—a molecular dynamics-guided and AI-driven pathogenicity prediction catalogue for all genetic mutations
title_fullStr Dynamicasome—a molecular dynamics-guided and AI-driven pathogenicity prediction catalogue for all genetic mutations
title_full_unstemmed Dynamicasome—a molecular dynamics-guided and AI-driven pathogenicity prediction catalogue for all genetic mutations
title_short Dynamicasome—a molecular dynamics-guided and AI-driven pathogenicity prediction catalogue for all genetic mutations
title_sort dynamicasome a molecular dynamics guided and ai driven pathogenicity prediction catalogue for all genetic mutations
url https://doi.org/10.1038/s42003-025-08334-y
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