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BREATHE DMD:在杜氏肌营养不良症治疗性低氧发作后增强呼吸效果。

BREATHE DMD: boosting respiratory efficacy after therapeutic hypoxic episodes in Duchenne muscular dystrophy.

机构信息

Department of Physiology, University College Cork, Cork, Ireland.

出版信息

J Physiol. 2024 Jul;602(14):3255-3272. doi: 10.1113/JP280280. Epub 2024 Jun 4.

DOI:10.1113/JP280280
PMID:38837229
Abstract

Duchenne muscular dystrophy (DMD) is a fatal genetic neuromuscular disorder, characterised by progressive decline in skeletal muscle function due to the secondary consequences of dystrophin deficiency. Weakness extends to the respiratory musculature, and cardiorespiratory failure is the leading cause of death in men with DMD. Intermittent hypoxia has emerged as a potential therapy to counteract ventilatory insufficiency by eliciting long-term facilitation of breathing. Mechanisms of sensory and motor facilitation of breathing have been well delineated in animal models. Various paradigms of intermittent hypoxia have been designed and implemented in human trials culminating in clinical trials in people with spinal cord injury and amyotrophic lateral sclerosis. Application of therapeutic intermittent hypoxia to DMD is considered together with discussion of the potential barriers to progression owing to the complexity of this devastating disease. Notwithstanding the considerable challenges and potential pitfalls of intermittent hypoxia-based therapies for DMD, we suggest it is incumbent on the research community to explore the potential benefits in pre-clinical models. Intermittent hypoxia paradigms should be implemented to explore the proclivity to express respiratory plasticity with the longer-term aim of preserving and potentiating ventilation in pre-clinical models and people with DMD.

摘要

杜氏肌营养不良症(DMD)是一种致命的遗传性神经肌肉疾病,其特征是由于肌营养不良蛋白缺乏的继发后果,骨骼肌功能逐渐下降。肌肉无力会扩展到呼吸肌,心肺衰竭是 DMD 男性患者死亡的主要原因。间歇性低氧已成为一种通过诱发呼吸的长期易化来对抗通气不足的潜在治疗方法。在动物模型中,已经很好地阐明了呼吸感觉和运动易化的机制。已经设计并实施了各种间歇性低氧方案,并在脊髓损伤和肌萎缩侧索硬化症患者的临床试验中达到了顶峰。考虑到这种毁灭性疾病的复杂性,将治疗性间歇性低氧应用于 DMD 与讨论进展的潜在障碍一起进行。尽管间歇性低氧疗法治疗 DMD 面临着相当大的挑战和潜在的陷阱,但我们认为研究界有责任在临床前模型中探索潜在的益处。应实施间歇性低氧方案,以探索表达呼吸可塑性的倾向,其长期目标是在临床前模型和 DMD 患者中保存和增强通气。

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J Physiol. 2024 Jul;602(14):3255-3272. doi: 10.1113/JP280280. Epub 2024 Jun 4.
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