TY - JOUR
T1 - Duchenne muscular dystrophy
T2 - disease mechanism and therapeutic strategies
AU - Bez Batti Angulski, Addeli
AU - Hosny, Nora
AU - Cohen, Houda
AU - Martin, Ashley A.
AU - Hahn, Dongwoo
AU - Bauer, Jack
AU - Metzger, Joseph M.
N1 - Publisher Copyright:
Copyright © 2023 Bez Batti Angulski, Hosny, Cohen, Martin, Hahn, Bauer and Metzger.
PY - 2023
Y1 - 2023
N2 - 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.
AB - 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.
KW - Duchenne muscular dystrophy
KW - dystrophin
KW - muscle disease
KW - pathophysiology
KW - skeletal muscle
KW - therapeutic strategies
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U2 - 10.3389/fphys.2023.1183101
DO - 10.3389/fphys.2023.1183101
M3 - Review article
C2 - 37435300
AN - SCOPUS:85164605592
SN - 1664-042X
VL - 14
JO - Frontiers in Physiology
JF - Frontiers in Physiology
M1 - 1183101
ER -