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显性遗传性肌肉疾病:理解其复杂性并探索治疗方法。

Dominantly inherited muscle disorders: understanding their complexity and exploring therapeutic approaches.

机构信息

Washington University Saint Louis, Neuromuscular Disease Center, 660 S. Euclid Ave., St Louis, MO 63110, USA.

出版信息

Dis Model Mech. 2024 Oct 1;17(10). doi: 10.1242/dmm.050720. Epub 2024 Nov 6.

Abstract

Treatments for disabling and life-threatening hereditary muscle disorders are finally close to becoming a reality. Research has thus far focused primarily on recessive forms of muscle disease. The gene replacement strategies that are commonly employed for recessive, loss-of-function disorders are not readily translatable to most dominant myopathies owing to the presence of a normal chromosome in each nucleus, hindering the development of novel treatments for these dominant disorders. This is largely due to their complex, heterogeneous disease mechanisms that require unique therapeutic approaches. However, as viral and RNA interference-based therapies enter clinical use, key tools are now in place to develop treatments for dominantly inherited disorders of muscle. This article will review what is known about dominantly inherited disorders of muscle, specifically their genetic basis, how mutations lead to disease, and the pathomechanistic implications for therapeutic approaches.

摘要

针对致残和危及生命的遗传性肌肉疾病的治疗方法终于即将成为现实。迄今为止,研究主要集中在肌肉疾病的隐性形式上。由于每个细胞核中都存在正常染色体,因此通常用于隐性、功能丧失性疾病的基因替换策略不易转化为大多数显性肌病,这阻碍了这些显性疾病的新疗法的开发。这主要是由于它们复杂的、异质的疾病机制需要独特的治疗方法。然而,随着基于病毒和 RNA 干扰的疗法进入临床应用,现在已经有了用于开发肌肉显性遗传性疾病治疗方法的关键工具。本文将回顾关于肌肉显性遗传性疾病的已知内容,特别是它们的遗传基础、突变如何导致疾病以及对治疗方法的病理机制影响。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3033/11574355/a2300c86dca0/dmm-17-050720-g1.jpg

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