Using computer-generated protein models to analyze mutations linked to Amelogenesis Imperfecta.

Amelogenesis Imperfecta (AI) is a disorder of tooth development caused by mutations in genes involved in several stages of tooth enamel formation. Few proteins involved in tooth development or developmental anomalies are explored in detail. Knowledge of 3D protein structure is essential to studying...

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Main Authors: Nazlee Sharmin, Jerald Yuan, Ava K Chow
Format: Article
Language:English
Published: Public Library of Science (PLoS) 2025-01-01
Series:PLoS ONE
Online Access:https://doi.org/10.1371/journal.pone.0326679
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author Nazlee Sharmin
Jerald Yuan
Ava K Chow
author_facet Nazlee Sharmin
Jerald Yuan
Ava K Chow
author_sort Nazlee Sharmin
collection DOAJ
description Amelogenesis Imperfecta (AI) is a disorder of tooth development caused by mutations in genes involved in several stages of tooth enamel formation. Few proteins involved in tooth development or developmental anomalies are explored in detail. Knowledge of 3D protein structure is essential to studying protein function. However, crystallized complete protein structures related to teeth and oral development are rare in the Protein Data Bank. Computational approaches for automated protein structure prediction have become a popular alternative for generating protein 3D structures. In this study, we aimed to explore the potential of using computer-generated protein models to analyze mutations linked to AI. We took a systematic approach to identify, screen, and analyze AI-linked protein variants. Proteins with AI-linked mutations were identified from the NCBI and OMIM databases, followed by screening of sequences for intrinsically disordered regions (IDRs). The iterative threading assembly refinement (I-TASSER) server was used to generate homology models for the wildtype and mutant proteins. PyMOL was used to analyze and compare the 3D structures of the proteins. Nineteen human genes with AI-associated mutations were identified from NCBI and OMIM. We identified multiple AI-associated protein variants with structural differences compared to their wildtype form. The current evidence aligns with several of the structural alterations identified in our study. Our findings suggest the potential of utilizing computer-generated protein models to investigate disease-associated mutations. However, careful consideration of models, templates, and alignments over the regions of interest is necessary to predict any potential structural impact of a disease-causing protein variant.
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spelling doaj-art-7c07c0b146ef4cfe8092600148104d7d2025-08-20T03:29:53ZengPublic Library of Science (PLoS)PLoS ONE1932-62032025-01-01206e032667910.1371/journal.pone.0326679Using computer-generated protein models to analyze mutations linked to Amelogenesis Imperfecta.Nazlee SharminJerald YuanAva K ChowAmelogenesis Imperfecta (AI) is a disorder of tooth development caused by mutations in genes involved in several stages of tooth enamel formation. Few proteins involved in tooth development or developmental anomalies are explored in detail. Knowledge of 3D protein structure is essential to studying protein function. However, crystallized complete protein structures related to teeth and oral development are rare in the Protein Data Bank. Computational approaches for automated protein structure prediction have become a popular alternative for generating protein 3D structures. In this study, we aimed to explore the potential of using computer-generated protein models to analyze mutations linked to AI. We took a systematic approach to identify, screen, and analyze AI-linked protein variants. Proteins with AI-linked mutations were identified from the NCBI and OMIM databases, followed by screening of sequences for intrinsically disordered regions (IDRs). The iterative threading assembly refinement (I-TASSER) server was used to generate homology models for the wildtype and mutant proteins. PyMOL was used to analyze and compare the 3D structures of the proteins. Nineteen human genes with AI-associated mutations were identified from NCBI and OMIM. We identified multiple AI-associated protein variants with structural differences compared to their wildtype form. The current evidence aligns with several of the structural alterations identified in our study. Our findings suggest the potential of utilizing computer-generated protein models to investigate disease-associated mutations. However, careful consideration of models, templates, and alignments over the regions of interest is necessary to predict any potential structural impact of a disease-causing protein variant.https://doi.org/10.1371/journal.pone.0326679
spellingShingle Nazlee Sharmin
Jerald Yuan
Ava K Chow
Using computer-generated protein models to analyze mutations linked to Amelogenesis Imperfecta.
PLoS ONE
title Using computer-generated protein models to analyze mutations linked to Amelogenesis Imperfecta.
title_full Using computer-generated protein models to analyze mutations linked to Amelogenesis Imperfecta.
title_fullStr Using computer-generated protein models to analyze mutations linked to Amelogenesis Imperfecta.
title_full_unstemmed Using computer-generated protein models to analyze mutations linked to Amelogenesis Imperfecta.
title_short Using computer-generated protein models to analyze mutations linked to Amelogenesis Imperfecta.
title_sort using computer generated protein models to analyze mutations linked to amelogenesis imperfecta
url https://doi.org/10.1371/journal.pone.0326679
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