Institute for Stem Cell and Regenerative Medicine, University of Washington, Seattle, Washington, USA.
Department of Rehabilitation Medicine, University of Washington, Seattle, Washington, USA.
WIREs Mech Dis. 2024 Nov-Dec;16(6):e1653. doi: 10.1002/wsbm.1653. Epub 2024 Oct 23.
Duchenne muscular dystrophy (DMD) is a severe degenerative muscle disease caused by mutations in the DMD gene, which encodes dystrophin. Despite its initial description in the late 19th century by French neurologist Guillaume Duchenne de Boulogne, and identification of causal DMD genetic mutations in the 1980s, therapeutics remain challenging. The current standard of care is corticosteroid treatment, which delays the progression of muscle dysfunction but is associated with significant adverse effects. Emerging therapeutic approaches, including AAV-mediated gene transfer, CRISPR gene editing, and small molecule interventions, are under development but face considerable obstacles. Although DMD is viewed as a progressive muscle disease, muscle damage and abnormal molecular signatures are already evident during fetal myogenesis. This early onset of pathology suggests that the limited success of current therapies may partly be due to their administration after aberrant embryonic myogenesis has occurred in the absence of dystrophin. Consequently, identifying optimal therapeutic strategies and intervention windows for DMD may depend on a better understanding of the earliest DMD disease mechanisms. As newer techniques are applied, the field is gaining increasingly detailed insights into the early muscle developmental abnormalities in DMD. A comprehensive understanding of the initial events in DMD pathogenesis and progression will facilitate the generation and testing of effective therapeutic interventions.
杜氏肌营养不良症(DMD)是一种严重的退行性肌肉疾病,由 DMD 基因突变引起,该基因突变编码肌营养不良蛋白。尽管 19 世纪末法国神经学家 Guillaume Duchenne de Boulogne 首次描述了这种疾病,并在 20 世纪 80 年代确定了导致 DMD 的遗传突变,但治疗方法仍然具有挑战性。目前的标准治疗方法是皮质类固醇治疗,虽然可以延缓肌肉功能障碍的进展,但会引起明显的不良反应。目前正在开发新兴的治疗方法,包括 AAV 介导的基因转移、CRISPR 基因编辑和小分子干预,但面临着相当大的障碍。尽管 DMD 被视为一种进行性肌肉疾病,但在胎儿肌发生过程中已经可以明显看到肌肉损伤和异常的分子特征。这种早期发病机制表明,目前治疗方法的有限成功可能部分是由于在缺乏肌营养不良蛋白的情况下,异常的胚胎肌发生发生后才进行治疗。因此,确定 DMD 的最佳治疗策略和干预窗口可能取决于对最早的 DMD 疾病机制的更好理解。随着新技术的应用,该领域对 DMD 早期肌肉发育异常的认识越来越深入。全面了解 DMD 发病机制和进展的初始事件将有助于生成和测试有效的治疗干预措施。