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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...
Autores principales: | , , , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
MDPI
2022
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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. |
format | Online Article Text |
id | pubmed-9601687 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | MDPI |
record_format | MEDLINE/PubMed |
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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