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Clinical, Biochemical, and Molecular Characterization of Two Families with Novel Mutations in the LDHA Gene (GSD XI)

Lactate dehydrogenase (LDH) catalyzes the reversible conversion of L-lactate to pyruvate. LDH-A deficiency is an autosomal recessive disorder (glycogenosis type XI, OMIM#612933) caused by mutations in the LDHA gene. We present two young adult female patients presenting with intolerance to anaerobic...

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Autores principales: Serrano-Lorenzo, Pablo, Rabasa, María, Esteban, Jesús, Hidalgo Mayoral, Irene, Domínguez-González, Cristina, Blanco-Echevarría, Agustín, Garrido-Moraga, Rocío, Lucia, Alejandro, Blázquez, Alberto, Rubio, Juan C., Palma-Milla, Carmen, Arenas, Joaquín, Martín, Miguel A.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9601687/
https://www.ncbi.nlm.nih.gov/pubmed/36292720
http://dx.doi.org/10.3390/genes13101835
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author Serrano-Lorenzo, Pablo
Rabasa, María
Esteban, Jesús
Hidalgo Mayoral, Irene
Domínguez-González, Cristina
Blanco-Echevarría, Agustín
Garrido-Moraga, Rocío
Lucia, Alejandro
Blázquez, Alberto
Rubio, Juan C.
Palma-Milla, Carmen
Arenas, Joaquín
Martín, Miguel A.
author_facet Serrano-Lorenzo, Pablo
Rabasa, María
Esteban, Jesús
Hidalgo Mayoral, Irene
Domínguez-González, Cristina
Blanco-Echevarría, Agustín
Garrido-Moraga, Rocío
Lucia, Alejandro
Blázquez, Alberto
Rubio, Juan C.
Palma-Milla, Carmen
Arenas, Joaquín
Martín, Miguel A.
author_sort Serrano-Lorenzo, Pablo
collection PubMed
description Lactate dehydrogenase (LDH) catalyzes the reversible conversion of L-lactate to pyruvate. LDH-A deficiency is an autosomal recessive disorder (glycogenosis type XI, OMIM#612933) caused by mutations in the LDHA gene. We present two young adult female patients presenting with intolerance to anaerobic exercise, episodes of rhabdomyolysis, and, in one of the patients, psoriasis-like dermatitis. We identified in the LDHA gene a homozygous c.410C>A substitution that predicts a p.Ser137Ter nonsense mutation in Patient One and a compound heterozygous c.410C>A (p.Ser137Ter) and c.750G>A (p.Trp250Ter) nonsense mutation in Patient Two. The pathogenicity of the variants was demonstrated by electrophoretic separation of LDH isoenzymes. Moreover, a flat lactate curve on the forearm exercise test, along with the clinical combination of myopathy and psoriatic-like dermatitis, can also lead to the diagnosis.
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spelling pubmed-96016872022-10-27 Clinical, Biochemical, and Molecular Characterization of Two Families with Novel Mutations in the LDHA Gene (GSD XI) Serrano-Lorenzo, Pablo Rabasa, María Esteban, Jesús Hidalgo Mayoral, Irene Domínguez-González, Cristina Blanco-Echevarría, Agustín Garrido-Moraga, Rocío Lucia, Alejandro Blázquez, Alberto Rubio, Juan C. Palma-Milla, Carmen Arenas, Joaquín Martín, Miguel A. Genes (Basel) Case Report Lactate dehydrogenase (LDH) catalyzes the reversible conversion of L-lactate to pyruvate. LDH-A deficiency is an autosomal recessive disorder (glycogenosis type XI, OMIM#612933) caused by mutations in the LDHA gene. We present two young adult female patients presenting with intolerance to anaerobic exercise, episodes of rhabdomyolysis, and, in one of the patients, psoriasis-like dermatitis. We identified in the LDHA gene a homozygous c.410C>A substitution that predicts a p.Ser137Ter nonsense mutation in Patient One and a compound heterozygous c.410C>A (p.Ser137Ter) and c.750G>A (p.Trp250Ter) nonsense mutation in Patient Two. The pathogenicity of the variants was demonstrated by electrophoretic separation of LDH isoenzymes. Moreover, a flat lactate curve on the forearm exercise test, along with the clinical combination of myopathy and psoriatic-like dermatitis, can also lead to the diagnosis. MDPI 2022-10-11 /pmc/articles/PMC9601687/ /pubmed/36292720 http://dx.doi.org/10.3390/genes13101835 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 Case Report
Serrano-Lorenzo, Pablo
Rabasa, María
Esteban, Jesús
Hidalgo Mayoral, Irene
Domínguez-González, Cristina
Blanco-Echevarría, Agustín
Garrido-Moraga, Rocío
Lucia, Alejandro
Blázquez, Alberto
Rubio, Juan C.
Palma-Milla, Carmen
Arenas, Joaquín
Martín, Miguel A.
Clinical, Biochemical, and Molecular Characterization of Two Families with Novel Mutations in the LDHA Gene (GSD XI)
title Clinical, Biochemical, and Molecular Characterization of Two Families with Novel Mutations in the LDHA Gene (GSD XI)
title_full Clinical, Biochemical, and Molecular Characterization of Two Families with Novel Mutations in the LDHA Gene (GSD XI)
title_fullStr Clinical, Biochemical, and Molecular Characterization of Two Families with Novel Mutations in the LDHA Gene (GSD XI)
title_full_unstemmed Clinical, Biochemical, and Molecular Characterization of Two Families with Novel Mutations in the LDHA Gene (GSD XI)
title_short Clinical, Biochemical, and Molecular Characterization of Two Families with Novel Mutations in the LDHA Gene (GSD XI)
title_sort clinical, biochemical, and molecular characterization of two families with novel mutations in the ldha gene (gsd xi)
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9601687/
https://www.ncbi.nlm.nih.gov/pubmed/36292720
http://dx.doi.org/10.3390/genes13101835
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