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Fanconi Anemia Patients from an Indigenous Community in Mexico Carry a New Founder Pathogenic Variant in FANCG

Fanconi anemia (FA) is a rare genetic disorder caused by pathogenic variants (PV) in at least 22 genes, which cooperate in the Fanconi anemia/Breast Cancer (FA/BRCA) pathway to maintain genome stability. PV in FANCA, FANCC, and FANCG account for most cases (~90%). This study evaluated the chromosoma...

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Autores principales: Reyes, Pedro, García-de Teresa, Benilde, Juárez, Ulises, Pérez-Villatoro, Fernando, Fiesco-Roa, Moisés O., Rodríguez, Alfredo, Molina, Bertha, Villarreal-Molina, María Teresa, Meléndez-Zajgla, Jorge, Carnevale, Alessandra, Torres, Leda, Frias, Sara
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8877758/
https://www.ncbi.nlm.nih.gov/pubmed/35216452
http://dx.doi.org/10.3390/ijms23042334
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author Reyes, Pedro
García-de Teresa, Benilde
Juárez, Ulises
Pérez-Villatoro, Fernando
Fiesco-Roa, Moisés O.
Rodríguez, Alfredo
Molina, Bertha
Villarreal-Molina, María Teresa
Meléndez-Zajgla, Jorge
Carnevale, Alessandra
Torres, Leda
Frias, Sara
author_facet Reyes, Pedro
García-de Teresa, Benilde
Juárez, Ulises
Pérez-Villatoro, Fernando
Fiesco-Roa, Moisés O.
Rodríguez, Alfredo
Molina, Bertha
Villarreal-Molina, María Teresa
Meléndez-Zajgla, Jorge
Carnevale, Alessandra
Torres, Leda
Frias, Sara
author_sort Reyes, Pedro
collection PubMed
description Fanconi anemia (FA) is a rare genetic disorder caused by pathogenic variants (PV) in at least 22 genes, which cooperate in the Fanconi anemia/Breast Cancer (FA/BRCA) pathway to maintain genome stability. PV in FANCA, FANCC, and FANCG account for most cases (~90%). This study evaluated the chromosomal, molecular, and physical phenotypic findings of a novel founder FANCG PV, identified in three patients with FA from the Mixe community of Oaxaca, Mexico. All patients presented chromosomal instability and a homozygous PV, FANCG: c.511-3_511-2delCA, identified by next-generation sequencing analysis. Bioinformatic predictions suggest that this deletion disrupts a splice acceptor site promoting the exon 5 skipping. Analysis of Cytoscan 750 K arrays for haplotyping and global ancestry supported the Mexican origin and founder effect of the variant, reaffirming the high frequency of founder PV in FANCG. The degree of bone marrow failure and physical findings (described through the acronyms VACTERL-H and PHENOS) were used to depict the phenotype of the patients. Despite having a similar frequency of chromosomal aberrations and genetic constitution, the phenotype showed a wide spectrum of severity. The identification of a founder PV could help for a systematic and accurate genetic screening of patients with FA suspicion in this population.
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spelling pubmed-88777582022-02-26 Fanconi Anemia Patients from an Indigenous Community in Mexico Carry a New Founder Pathogenic Variant in FANCG Reyes, Pedro García-de Teresa, Benilde Juárez, Ulises Pérez-Villatoro, Fernando Fiesco-Roa, Moisés O. Rodríguez, Alfredo Molina, Bertha Villarreal-Molina, María Teresa Meléndez-Zajgla, Jorge Carnevale, Alessandra Torres, Leda Frias, Sara Int J Mol Sci Article Fanconi anemia (FA) is a rare genetic disorder caused by pathogenic variants (PV) in at least 22 genes, which cooperate in the Fanconi anemia/Breast Cancer (FA/BRCA) pathway to maintain genome stability. PV in FANCA, FANCC, and FANCG account for most cases (~90%). This study evaluated the chromosomal, molecular, and physical phenotypic findings of a novel founder FANCG PV, identified in three patients with FA from the Mixe community of Oaxaca, Mexico. All patients presented chromosomal instability and a homozygous PV, FANCG: c.511-3_511-2delCA, identified by next-generation sequencing analysis. Bioinformatic predictions suggest that this deletion disrupts a splice acceptor site promoting the exon 5 skipping. Analysis of Cytoscan 750 K arrays for haplotyping and global ancestry supported the Mexican origin and founder effect of the variant, reaffirming the high frequency of founder PV in FANCG. The degree of bone marrow failure and physical findings (described through the acronyms VACTERL-H and PHENOS) were used to depict the phenotype of the patients. Despite having a similar frequency of chromosomal aberrations and genetic constitution, the phenotype showed a wide spectrum of severity. The identification of a founder PV could help for a systematic and accurate genetic screening of patients with FA suspicion in this population. MDPI 2022-02-20 /pmc/articles/PMC8877758/ /pubmed/35216452 http://dx.doi.org/10.3390/ijms23042334 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
Reyes, Pedro
García-de Teresa, Benilde
Juárez, Ulises
Pérez-Villatoro, Fernando
Fiesco-Roa, Moisés O.
Rodríguez, Alfredo
Molina, Bertha
Villarreal-Molina, María Teresa
Meléndez-Zajgla, Jorge
Carnevale, Alessandra
Torres, Leda
Frias, Sara
Fanconi Anemia Patients from an Indigenous Community in Mexico Carry a New Founder Pathogenic Variant in FANCG
title Fanconi Anemia Patients from an Indigenous Community in Mexico Carry a New Founder Pathogenic Variant in FANCG
title_full Fanconi Anemia Patients from an Indigenous Community in Mexico Carry a New Founder Pathogenic Variant in FANCG
title_fullStr Fanconi Anemia Patients from an Indigenous Community in Mexico Carry a New Founder Pathogenic Variant in FANCG
title_full_unstemmed Fanconi Anemia Patients from an Indigenous Community in Mexico Carry a New Founder Pathogenic Variant in FANCG
title_short Fanconi Anemia Patients from an Indigenous Community in Mexico Carry a New Founder Pathogenic Variant in FANCG
title_sort fanconi anemia patients from an indigenous community in mexico carry a new founder pathogenic variant in fancg
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8877758/
https://www.ncbi.nlm.nih.gov/pubmed/35216452
http://dx.doi.org/10.3390/ijms23042334
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