Genetic aspects of the oxidative phosphorylation dysfunction in dilated cardiomyopathy.

Dilated cardiomyopathy Mitochondrial DNA Mitochondrial biogenesis Oxidative phosphorylation Pathogenicity criteria Pathologic mutation

Journal

Mutation research. Reviews in mutation research
ISSN: 1388-2139
Titre abrégé: Mutat Res Rev Mutat Res
Pays: Netherlands
ID NLM: 101632211

Informations de publication

Date de publication:
Historique:
received: 22 03 2020
revised: 19 08 2020
accepted: 20 08 2020
entrez: 19 12 2020
pubmed: 20 12 2020
medline: 4 2 2021
Statut: ppublish

Résumé

Dilated cardiomyopathy is a frequent and extremely heterogeneous medical condition. Deficits in the oxidative phosphorylation system have been described in patients suffering from dilated cardiomyopathy. Hence, mutations in proteins related to this biochemical pathway could be etiological factors for some of these patients. Here, we review the clinical phenotypes of patients harboring pathological mutations in genes related to the oxidative phosphorylation system, either encoded in the mitochondrial or in the nuclear genome, presenting with dilated cardiomyopathy. In addition to the clinical heterogeneity of these patients, the large genetic heterogeneity has contributed to an improper allocation of pathogenicity for many candidate mutations. We suggest criteria to avoid incorrect assignment of pathogenicity to newly found mutations and discuss possible therapies targeting the oxidative phosphorylation function.

Identifiants

pubmed: 33339579
pii: S1383-5742(20)30054-5
doi: 10.1016/j.mrrev.2020.108334
pii:
doi:

Substances chimiques

DNA, Mitochondrial 0

Types de publication

Journal Article Research Support, Non-U.S. Gov't Review

Langues

eng

Sous-ensembles de citation

IM

Pagination

108334

Informations de copyright

Copyright © 2020 Elsevier B.V. All rights reserved.

Déclaration de conflit d'intérêts

Declaration of Competing Interest The authors report no declarations of interest.

Auteurs

M Pilar Bayona-Bafaluy (MP)

Departamento de Bioquímica, Biología Molecular y Celular, Universidad de Zaragoza. C/ Miguel Servet, 177. 50013, Zaragoza, Spain; Instituto de Investigación Sanitaria (IIS) de Aragón, Av. San Juan Bosco, 13., 50009, Zaragoza, Spain; Centro de Investigaciones Biomédicas en Red de Enfermedades Raras (CIBERER), Av. Monforte de Lemos, 3-5, 28029, Madrid, Spain. Electronic address: pbayona@unizar.es.

Eldris Iglesias (E)

Departamento de Bioquímica, Biología Molecular y Celular, Universidad de Zaragoza. C/ Miguel Servet, 177. 50013, Zaragoza, Spain; Instituto de Investigación Sanitaria (IIS) de Aragón, Av. San Juan Bosco, 13., 50009, Zaragoza, Spain. Electronic address: eiglesia@unizar.es.

Ester López-Gallardo (E)

Departamento de Bioquímica, Biología Molecular y Celular, Universidad de Zaragoza. C/ Miguel Servet, 177. 50013, Zaragoza, Spain; Instituto de Investigación Sanitaria (IIS) de Aragón, Av. San Juan Bosco, 13., 50009, Zaragoza, Spain; Centro de Investigaciones Biomédicas en Red de Enfermedades Raras (CIBERER), Av. Monforte de Lemos, 3-5, 28029, Madrid, Spain. Electronic address: esterlop@unizar.es.

Sonia Emperador (S)

Departamento de Bioquímica, Biología Molecular y Celular, Universidad de Zaragoza. C/ Miguel Servet, 177. 50013, Zaragoza, Spain; Instituto de Investigación Sanitaria (IIS) de Aragón, Av. San Juan Bosco, 13., 50009, Zaragoza, Spain; Centro de Investigaciones Biomédicas en Red de Enfermedades Raras (CIBERER), Av. Monforte de Lemos, 3-5, 28029, Madrid, Spain. Electronic address: seortiz@unizar.es.

David Pacheu-Grau (D)

Department of Cellular Biochemistry, University Medical Center, Georg-August University,Humboldtalle, 23., 37073, Göttingen, Germany. Electronic address: david.Pacheu-grau@med.uni-goettingen.de.

Lorenzo Labarta (L)

Unidad de Cuidados Intensivos, Hospital San Jorge, Av. Martínez de Velasco, 36., 22004, Huesca, Spain. Electronic address: llabarta@salud.aragon.es.

Julio Montoya (J)

Departamento de Bioquímica, Biología Molecular y Celular, Universidad de Zaragoza. C/ Miguel Servet, 177. 50013, Zaragoza, Spain; Instituto de Investigación Sanitaria (IIS) de Aragón, Av. San Juan Bosco, 13., 50009, Zaragoza, Spain; Centro de Investigaciones Biomédicas en Red de Enfermedades Raras (CIBERER), Av. Monforte de Lemos, 3-5, 28029, Madrid, Spain. Electronic address: jmontoya@unizar.es.

Eduardo Ruiz-Pesini (E)

Departamento de Bioquímica, Biología Molecular y Celular, Universidad de Zaragoza. C/ Miguel Servet, 177. 50013, Zaragoza, Spain; Instituto de Investigación Sanitaria (IIS) de Aragón, Av. San Juan Bosco, 13., 50009, Zaragoza, Spain; Centro de Investigaciones Biomédicas en Red de Enfermedades Raras (CIBERER), Av. Monforte de Lemos, 3-5, 28029, Madrid, Spain; Fundación ARAID, Av. de Ranillas, 1-D., 50018, Zaragoza, Spain. Electronic address: eduruiz@unizar.es.

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