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In Silico Analyses of All STAT3 Missense Variants Leading to Explore Divergent AD-HIES Clinical Phenotypes
Autosomal dominant hyper-IgE syndrome (AD-HIES) is linked to dominant negative mutations of the STAT3 protein whose molecular basis for dysfunction is unclear and presenting with a variety of clinical manifestations with only supportive treatment. To establish the relationship between the impact of...
Autores principales: | , , , , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
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SAGE Publications
2023
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10134169/ https://www.ncbi.nlm.nih.gov/pubmed/37123531 http://dx.doi.org/10.1177/11769343231169374 |
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author | Mansouri, Mariam El Haddoumi, Ghyzlane Bendani, Houda Boumajdi, Nasma Hakmi, Mohammed Abbou, Hanane Bouricha, El Mehdi Elgharbaoui, Boutaina Kartti, Souad El Jaoudi, Rachid Belyamani, Lahcen Kandoussi, Ilham Ibrahimi, Azeddine El Hafidi, Naima |
author_facet | Mansouri, Mariam El Haddoumi, Ghyzlane Bendani, Houda Boumajdi, Nasma Hakmi, Mohammed Abbou, Hanane Bouricha, El Mehdi Elgharbaoui, Boutaina Kartti, Souad El Jaoudi, Rachid Belyamani, Lahcen Kandoussi, Ilham Ibrahimi, Azeddine El Hafidi, Naima |
author_sort | Mansouri, Mariam |
collection | PubMed |
description | Autosomal dominant hyper-IgE syndrome (AD-HIES) is linked to dominant negative mutations of the STAT3 protein whose molecular basis for dysfunction is unclear and presenting with a variety of clinical manifestations with only supportive treatment. To establish the relationship between the impact of STAT3 mutations in different domains and the severity of the clinical manifestations, 105 STAT3 mutations were analyzed for their impact on protein stability, flexibility, function, and binding affinity using in Silico approaches. Our results showed that 73% of the studied mutations have an impact on the physicochemical properties of the protein, altering the stability, flexibility and function to varying degrees. In particular, mutations affecting the DNA binding domain (DBD) and the Src Homology 2 (SH2) have a significant impact on the protein structure and disrupt its interaction either with DNA or other STAT3 to form a heterodomain complex, leading to severe clinical phenotypes. Collectively, this study suggests that there is a close relationship between the domain involving the mutation, the degree of variation in the properties of the protein and the degree of loss of function ranging from partial loss to complete loss, explaining the variability of clinical manifestations between mild and severe. |
format | Online Article Text |
id | pubmed-10134169 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2023 |
publisher | SAGE Publications |
record_format | MEDLINE/PubMed |
spelling | pubmed-101341692023-04-28 In Silico Analyses of All STAT3 Missense Variants Leading to Explore Divergent AD-HIES Clinical Phenotypes Mansouri, Mariam El Haddoumi, Ghyzlane Bendani, Houda Boumajdi, Nasma Hakmi, Mohammed Abbou, Hanane Bouricha, El Mehdi Elgharbaoui, Boutaina Kartti, Souad El Jaoudi, Rachid Belyamani, Lahcen Kandoussi, Ilham Ibrahimi, Azeddine El Hafidi, Naima Evol Bioinform Online Original Research Autosomal dominant hyper-IgE syndrome (AD-HIES) is linked to dominant negative mutations of the STAT3 protein whose molecular basis for dysfunction is unclear and presenting with a variety of clinical manifestations with only supportive treatment. To establish the relationship between the impact of STAT3 mutations in different domains and the severity of the clinical manifestations, 105 STAT3 mutations were analyzed for their impact on protein stability, flexibility, function, and binding affinity using in Silico approaches. Our results showed that 73% of the studied mutations have an impact on the physicochemical properties of the protein, altering the stability, flexibility and function to varying degrees. In particular, mutations affecting the DNA binding domain (DBD) and the Src Homology 2 (SH2) have a significant impact on the protein structure and disrupt its interaction either with DNA or other STAT3 to form a heterodomain complex, leading to severe clinical phenotypes. Collectively, this study suggests that there is a close relationship between the domain involving the mutation, the degree of variation in the properties of the protein and the degree of loss of function ranging from partial loss to complete loss, explaining the variability of clinical manifestations between mild and severe. SAGE Publications 2023-04-24 /pmc/articles/PMC10134169/ /pubmed/37123531 http://dx.doi.org/10.1177/11769343231169374 Text en © The Author(s) 2023 https://creativecommons.org/licenses/by-nc/4.0/This article is distributed under the terms of the Creative Commons Attribution-NonCommercial 4.0 License (https://creativecommons.org/licenses/by-nc/4.0/) which permits non-commercial use, reproduction and distribution of the work without further permission provided the original work is attributed as specified on the SAGE and Open Access page(https://us.sagepub.com/en-us/nam/open-access-at-sage). |
spellingShingle | Original Research Mansouri, Mariam El Haddoumi, Ghyzlane Bendani, Houda Boumajdi, Nasma Hakmi, Mohammed Abbou, Hanane Bouricha, El Mehdi Elgharbaoui, Boutaina Kartti, Souad El Jaoudi, Rachid Belyamani, Lahcen Kandoussi, Ilham Ibrahimi, Azeddine El Hafidi, Naima In Silico Analyses of All STAT3 Missense Variants Leading to Explore Divergent AD-HIES Clinical Phenotypes |
title | In Silico Analyses of All STAT3 Missense Variants Leading to Explore
Divergent AD-HIES Clinical Phenotypes |
title_full | In Silico Analyses of All STAT3 Missense Variants Leading to Explore
Divergent AD-HIES Clinical Phenotypes |
title_fullStr | In Silico Analyses of All STAT3 Missense Variants Leading to Explore
Divergent AD-HIES Clinical Phenotypes |
title_full_unstemmed | In Silico Analyses of All STAT3 Missense Variants Leading to Explore
Divergent AD-HIES Clinical Phenotypes |
title_short | In Silico Analyses of All STAT3 Missense Variants Leading to Explore
Divergent AD-HIES Clinical Phenotypes |
title_sort | in silico analyses of all stat3 missense variants leading to explore
divergent ad-hies clinical phenotypes |
topic | Original Research |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10134169/ https://www.ncbi.nlm.nih.gov/pubmed/37123531 http://dx.doi.org/10.1177/11769343231169374 |
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