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Pantothenate and L-Carnitine Supplementation Improves Pathological Alterations in Cellular Models of KAT6A Syndrome

Mutations in several genes involved in the epigenetic regulation of gene expression have been considered risk alterations to different intellectual disability (ID) syndromes associated with features of autism spectrum disorder (ASD). Among them are the pathogenic variants of the lysine-acetyltransfe...

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Autores principales: Munuera-Cabeza, Manuel, Álvarez-Córdoba, Mónica, Suárez-Rivero, Juan M., Povea-Cabello, Suleva, Villalón-García, Irene, Talaverón-Rey, Marta, Suárez-Carrillo, Alejandra, Reche-López, Diana, Cilleros-Holgado, Paula, Piñero-Pérez, Rocío, Sánchez-Alcázar, José A.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9778406/
https://www.ncbi.nlm.nih.gov/pubmed/36553567
http://dx.doi.org/10.3390/genes13122300
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author Munuera-Cabeza, Manuel
Álvarez-Córdoba, Mónica
Suárez-Rivero, Juan M.
Povea-Cabello, Suleva
Villalón-García, Irene
Talaverón-Rey, Marta
Suárez-Carrillo, Alejandra
Reche-López, Diana
Cilleros-Holgado, Paula
Piñero-Pérez, Rocío
Sánchez-Alcázar, José A.
author_facet Munuera-Cabeza, Manuel
Álvarez-Córdoba, Mónica
Suárez-Rivero, Juan M.
Povea-Cabello, Suleva
Villalón-García, Irene
Talaverón-Rey, Marta
Suárez-Carrillo, Alejandra
Reche-López, Diana
Cilleros-Holgado, Paula
Piñero-Pérez, Rocío
Sánchez-Alcázar, José A.
author_sort Munuera-Cabeza, Manuel
collection PubMed
description Mutations in several genes involved in the epigenetic regulation of gene expression have been considered risk alterations to different intellectual disability (ID) syndromes associated with features of autism spectrum disorder (ASD). Among them are the pathogenic variants of the lysine-acetyltransferase 6A (KAT6A) gene, which causes KAT6A syndrome. The KAT6A enzyme participates in a wide range of critical cellular functions, such as chromatin remodeling, gene expression, protein synthesis, cell metabolism, and replication. In this manuscript, we examined the pathophysiological alterations in fibroblasts derived from three patients harboring KAT6A mutations. We addressed survival in a stress medium, histone acetylation, protein expression patterns, and transcriptome analysis, as well as cell bioenergetics. In addition, we evaluated the therapeutic effectiveness of epigenetic modulators and mitochondrial boosting agents, such as pantothenate and L-carnitine, in correcting the mutant phenotype. Pantothenate and L-carnitine treatment increased histone acetylation and partially corrected protein and transcriptomic expression patterns in mutant KAT6A cells. Furthermore, the cell bioenergetics of mutant cells was significantly improved. Our results suggest that pantothenate and L-carnitine can significantly improve the mutant phenotype in cellular models of KAT6A syndrome.
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spelling pubmed-97784062022-12-23 Pantothenate and L-Carnitine Supplementation Improves Pathological Alterations in Cellular Models of KAT6A Syndrome Munuera-Cabeza, Manuel Álvarez-Córdoba, Mónica Suárez-Rivero, Juan M. Povea-Cabello, Suleva Villalón-García, Irene Talaverón-Rey, Marta Suárez-Carrillo, Alejandra Reche-López, Diana Cilleros-Holgado, Paula Piñero-Pérez, Rocío Sánchez-Alcázar, José A. Genes (Basel) Article Mutations in several genes involved in the epigenetic regulation of gene expression have been considered risk alterations to different intellectual disability (ID) syndromes associated with features of autism spectrum disorder (ASD). Among them are the pathogenic variants of the lysine-acetyltransferase 6A (KAT6A) gene, which causes KAT6A syndrome. The KAT6A enzyme participates in a wide range of critical cellular functions, such as chromatin remodeling, gene expression, protein synthesis, cell metabolism, and replication. In this manuscript, we examined the pathophysiological alterations in fibroblasts derived from three patients harboring KAT6A mutations. We addressed survival in a stress medium, histone acetylation, protein expression patterns, and transcriptome analysis, as well as cell bioenergetics. In addition, we evaluated the therapeutic effectiveness of epigenetic modulators and mitochondrial boosting agents, such as pantothenate and L-carnitine, in correcting the mutant phenotype. Pantothenate and L-carnitine treatment increased histone acetylation and partially corrected protein and transcriptomic expression patterns in mutant KAT6A cells. Furthermore, the cell bioenergetics of mutant cells was significantly improved. Our results suggest that pantothenate and L-carnitine can significantly improve the mutant phenotype in cellular models of KAT6A syndrome. MDPI 2022-12-06 /pmc/articles/PMC9778406/ /pubmed/36553567 http://dx.doi.org/10.3390/genes13122300 Text en © 2022 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).
spellingShingle Article
Munuera-Cabeza, Manuel
Álvarez-Córdoba, Mónica
Suárez-Rivero, Juan M.
Povea-Cabello, Suleva
Villalón-García, Irene
Talaverón-Rey, Marta
Suárez-Carrillo, Alejandra
Reche-López, Diana
Cilleros-Holgado, Paula
Piñero-Pérez, Rocío
Sánchez-Alcázar, José A.
Pantothenate and L-Carnitine Supplementation Improves Pathological Alterations in Cellular Models of KAT6A Syndrome
title Pantothenate and L-Carnitine Supplementation Improves Pathological Alterations in Cellular Models of KAT6A Syndrome
title_full Pantothenate and L-Carnitine Supplementation Improves Pathological Alterations in Cellular Models of KAT6A Syndrome
title_fullStr Pantothenate and L-Carnitine Supplementation Improves Pathological Alterations in Cellular Models of KAT6A Syndrome
title_full_unstemmed Pantothenate and L-Carnitine Supplementation Improves Pathological Alterations in Cellular Models of KAT6A Syndrome
title_short Pantothenate and L-Carnitine Supplementation Improves Pathological Alterations in Cellular Models of KAT6A Syndrome
title_sort pantothenate and l-carnitine supplementation improves pathological alterations in cellular models of kat6a syndrome
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9778406/
https://www.ncbi.nlm.nih.gov/pubmed/36553567
http://dx.doi.org/10.3390/genes13122300
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