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Duchenne muscular dystrophy: disease mechanism and therapeutic strategies

Duchenne muscular dystrophy (DMD) is a severe, progressive, and ultimately fatal disease of skeletal muscle wasting, respiratory insufficiency, and cardiomyopathy. The identification of the dystrophin gene as central to DMD pathogenesis has led to the understanding of the muscle membrane and the pro...

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Autores principales: Bez Batti Angulski, Addeli, Hosny, Nora, Cohen, Houda, Martin, Ashley A., Hahn, Dongwoo, Bauer, Jack, Metzger, Joseph M.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10330733/
https://www.ncbi.nlm.nih.gov/pubmed/37435300
http://dx.doi.org/10.3389/fphys.2023.1183101
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author Bez Batti Angulski, Addeli
Hosny, Nora
Cohen, Houda
Martin, Ashley A.
Hahn, Dongwoo
Bauer, Jack
Metzger, Joseph M.
author_facet Bez Batti Angulski, Addeli
Hosny, Nora
Cohen, Houda
Martin, Ashley A.
Hahn, Dongwoo
Bauer, Jack
Metzger, Joseph M.
author_sort Bez Batti Angulski, Addeli
collection PubMed
description Duchenne muscular dystrophy (DMD) is a severe, progressive, and ultimately fatal disease of skeletal muscle wasting, respiratory insufficiency, and cardiomyopathy. The identification of the dystrophin gene as central to DMD pathogenesis has led to the understanding of the muscle membrane and the proteins involved in membrane stability as the focal point of the disease. The lessons learned from decades of research in human genetics, biochemistry, and physiology have culminated in establishing the myriad functionalities of dystrophin in striated muscle biology. Here, we review the pathophysiological basis of DMD and discuss recent progress toward the development of therapeutic strategies for DMD that are currently close to or are in human clinical trials. The first section of the review focuses on DMD and the mechanisms contributing to membrane instability, inflammation, and fibrosis. The second section discusses therapeutic strategies currently used to treat DMD. This includes a focus on outlining the strengths and limitations of approaches directed at correcting the genetic defect through dystrophin gene replacement, modification, repair, and/or a range of dystrophin-independent approaches. The final section highlights the different therapeutic strategies for DMD currently in clinical trials.
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spelling pubmed-103307332023-07-11 Duchenne muscular dystrophy: disease mechanism and therapeutic strategies Bez Batti Angulski, Addeli Hosny, Nora Cohen, Houda Martin, Ashley A. Hahn, Dongwoo Bauer, Jack Metzger, Joseph M. Front Physiol Physiology Duchenne muscular dystrophy (DMD) is a severe, progressive, and ultimately fatal disease of skeletal muscle wasting, respiratory insufficiency, and cardiomyopathy. The identification of the dystrophin gene as central to DMD pathogenesis has led to the understanding of the muscle membrane and the proteins involved in membrane stability as the focal point of the disease. The lessons learned from decades of research in human genetics, biochemistry, and physiology have culminated in establishing the myriad functionalities of dystrophin in striated muscle biology. Here, we review the pathophysiological basis of DMD and discuss recent progress toward the development of therapeutic strategies for DMD that are currently close to or are in human clinical trials. The first section of the review focuses on DMD and the mechanisms contributing to membrane instability, inflammation, and fibrosis. The second section discusses therapeutic strategies currently used to treat DMD. This includes a focus on outlining the strengths and limitations of approaches directed at correcting the genetic defect through dystrophin gene replacement, modification, repair, and/or a range of dystrophin-independent approaches. The final section highlights the different therapeutic strategies for DMD currently in clinical trials. Frontiers Media S.A. 2023-06-26 /pmc/articles/PMC10330733/ /pubmed/37435300 http://dx.doi.org/10.3389/fphys.2023.1183101 Text en Copyright © 2023 Bez Batti Angulski, Hosny, Cohen, Martin, Hahn, Bauer and Metzger. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Physiology
Bez Batti Angulski, Addeli
Hosny, Nora
Cohen, Houda
Martin, Ashley A.
Hahn, Dongwoo
Bauer, Jack
Metzger, Joseph M.
Duchenne muscular dystrophy: disease mechanism and therapeutic strategies
title Duchenne muscular dystrophy: disease mechanism and therapeutic strategies
title_full Duchenne muscular dystrophy: disease mechanism and therapeutic strategies
title_fullStr Duchenne muscular dystrophy: disease mechanism and therapeutic strategies
title_full_unstemmed Duchenne muscular dystrophy: disease mechanism and therapeutic strategies
title_short Duchenne muscular dystrophy: disease mechanism and therapeutic strategies
title_sort duchenne muscular dystrophy: disease mechanism and therapeutic strategies
topic Physiology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10330733/
https://www.ncbi.nlm.nih.gov/pubmed/37435300
http://dx.doi.org/10.3389/fphys.2023.1183101
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