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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...

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Autores principales: 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
Formato: Online Artículo Texto
Lenguaje:English
Publicado: SAGE Publications 2023
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.
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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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