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杜氏肌营养不良症近期治疗进展概述

An Overview of Recent Therapeutics Advances for Duchenne Muscular Dystrophy.

作者信息

Mah Jean K

机构信息

Department of Paediatrics and Clinical Neurosciences, Cumming School of Medicine, University of Calgary, 2888 Shaganappi Trail NW, Calgary, Alberta, Canada, T3B 6A8.

出版信息

Methods Mol Biol. 2018;1687:3-17. doi: 10.1007/978-1-4939-7374-3_1.

Abstract

Duchenne muscular dystrophy (DMD) is the most common form of muscular dystrophy in childhood. Mutations of the DMD gene destabilize the dystrophin associated glycoprotein complex in the sarcolemma. Ongoing mechanical stress leads to unregulated influx of calcium ions into the sarcoplasm, with activation of proteases, release of proinflammatory cytokines, and mitochondrial dysfunction. Cumulative damage and reparative failure leads to progressive muscle necrosis, fibrosis, and fatty replacement. Although there is presently no cure for DMD, scientific advances have led to many potential disease-modifying treatments, including dystrophin replacement therapies, upregulation of compensatory proteins, anti-inflammatory agents, and other cellular targets. Recently approved therapies include ataluren for stop codon read-through and eteplirsen for exon 51 skipping of eligible individuals. The purpose of this chapter is to summarize the clinical features of DMD, to describe current outcome measures used in clinical studies, and to highlight new emerging therapies for affected individuals.

摘要

杜氏肌营养不良症(DMD)是儿童期最常见的肌营养不良症形式。DMD基因的突变会破坏肌膜中与肌营养不良蛋白相关的糖蛋白复合物的稳定性。持续的机械应力会导致钙离子不受控制地流入肌浆,激活蛋白酶,释放促炎细胞因子,并导致线粒体功能障碍。累积的损伤和修复失败会导致进行性肌肉坏死、纤维化和脂肪替代。虽然目前尚无治愈DMD的方法,但科学进步已带来许多潜在的疾病改善治疗方法,包括肌营养不良蛋白替代疗法、补偿蛋白上调、抗炎药物和其他细胞靶点。最近获批的疗法包括用于通读终止密码子的阿他芦醇和用于符合条件个体外显子51跳跃的依特普伦。本章的目的是总结DMD的临床特征,描述临床研究中使用的当前结局指标,并强调针对受影响个体的新兴疗法。

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