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Hydrogen sulfide supplementation as a potential treatment for primary mitochondrial diseases.
Slade, Luke; Deane, Colleen S; Szewczyk, Nathaniel J; Etheridge, Timothy; Whiteman, Matthew.
Afiliación
  • Slade L; University of Exeter Medical School, University of Exeter, St. Luke's Campus, Exeter EX1 2LU, UK; Leibniz-Institut für Analytische Wissenschaften-ISAS-e.V, Dortmund, Germany.
  • Deane CS; Human Development & Health, Faculty of Medicine, University of Southampton, Southampton General Hospital, Southampton, UK.
  • Szewczyk NJ; Medical Research Council Versus Arthritis Centre for Musculoskeletal Ageing Research, Royal Derby Hospital, University of Nottingham, Derby DE22 3DT, United Kingdom; Ohio Musculoskeletal and Neurologic Institute, Heritage College of Osteopathic Medicine, Ohio University, Athens, Ohio 45701, Greece.
  • Etheridge T; Public Health and Sport Sciences, Faculty of Health and Life Sciences, University of Exeter, Exeter EX1 2LU, United Kingdom. Electronic address: T.etheridge@exeter.ac.uk.
  • Whiteman M; University of Exeter Medical School, University of Exeter, St. Luke's Campus, Exeter EX1 2LU, UK. Electronic address: M.whiteman@exeter.ac.uk.
Pharmacol Res ; 203: 107180, 2024 May.
Article en En | MEDLINE | ID: mdl-38599468
ABSTRACT
Primary mitochondrial diseases (PMD) are amongst the most common inborn errors of metabolism causing fatal outcomes within the first decade of life. With marked heterogeneity in both inheritance patterns and physiological manifestations, these conditions present distinct challenges for targeted drug therapy, where effective therapeutic countermeasures remain elusive within the clinic. Hydrogen sulfide (H2S)-based therapeutics may offer a new option for patient treatment, having been proposed as a conserved mitochondrial substrate and post-translational regulator across species, displaying therapeutic effects in age-related mitochondrial dysfunction and neurodegenerative models of mitochondrial disease. H2S can stimulate mitochondrial respiration at sites downstream of common PMD-defective subunits, augmenting energy production, mitochondrial function and reducing cell death. Here, we highlight the primary signalling mechanisms of H2S in mitochondria relevant for PMD and outline key cytoprotective proteins/pathways amenable to post-translational restoration via H2S-mediated persulfidation. The mechanisms proposed here, combined with the advent of potent mitochondria-targeted sulfide delivery molecules, could provide a framework for H2S as a countermeasure for PMD disease progression.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Enfermedades Mitocondriales / Sulfuro de Hidrógeno / Mitocondrias Límite: Animals / Humans Idioma: En Revista: Pharmacol Res Asunto de la revista: FARMACOLOGIA Año: 2024 Tipo del documento: Article Pais de publicación: Países Bajos

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Enfermedades Mitocondriales / Sulfuro de Hidrógeno / Mitocondrias Límite: Animals / Humans Idioma: En Revista: Pharmacol Res Asunto de la revista: FARMACOLOGIA Año: 2024 Tipo del documento: Article Pais de publicación: Países Bajos