Duchenne muscular dystrophy: disease mechanism and therapeutic strategies

被引:65
作者
Angulski, Addeli Bez Batti [1 ]
Hosny, Nora [1 ]
Cohen, Houda [1 ]
Martin, Ashley A. [1 ]
Hahn, Dongwoo [1 ]
Bauer, Jack [1 ]
Metzger, Joseph M. [1 ]
机构
[1] Univ Minnesota, Dept Integrat Biol & Physiol, Med Sch, Minneapolis, MN 55455 USA
基金
美国国家卫生研究院;
关键词
muscle disease; therapeutic strategies; skeletal muscle; dystrophin; Duchenne muscular dystrophy; pathophysiology; MDX MOUSE MODEL; SKELETAL-MUSCLE MASS; SICKLE-CELL-DISEASE; IN-VIVO; GENE-THERAPY; REACTIVE OXYGEN; MESSENGER-RNA; SYSTEMIC DELIVERY; FULL-LENGTH; STEM-CELLS;
D O I
10.3389/fphys.2023.1183101
中图分类号
Q4 [生理学];
学科分类号
071003 ;
摘要
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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收藏
页数:31
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