将 Lipin1 作为杜兴氏肌肉萎缩症呼吸功能不全的治疗靶点。

IF 3.2 3区 医学 Q2 PHYSIOLOGY
Frontiers in Physiology Pub Date : 2024-11-12 eCollection Date: 2024-01-01 DOI:10.3389/fphys.2024.1477976
Alexandra Brown, Brooklyn Morris, John Karanja Kamau, Ryan J Rakoczy, Brian N Finck, Christopher N Wyatt, Hongmei Ren
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引用次数: 0

摘要

杜兴氏肌肉萎缩症(DMD)患者的横膈膜肌肉功能障碍会导致呼吸功能不全,这是导致患者死亡的主要原因。淀粉样蛋白基因突变导致肌细胞膜不稳定,造成膈肌组织结构退化。先前的研究表明脂蛋白1对维持骨骼肌膜完整性非常重要,因此我们探讨了脂蛋白1在肌营养不良性膈膜中的作用。我们发现,在肌营养不良膈肌中,脂蛋白1的蛋白表达水平降低了60%。进一步敲除营养不良膈肌中的lipin1会导致坏死增加,而恢复营养不良膈肌中的lipin1则会减少炎症和纤维化、减少肌纤维死亡并改善呼吸功能。我们的研究结果表明,恢复脂蛋白1可通过增强膜的完整性改善呼吸功能,并表明脂蛋白1可能是预防DMD呼吸功能不全和呼吸衰竭的潜在治疗靶点。要更好地了解这些发现背后的机制,并确定脂蛋白1在维持肌膜稳定性方面的作用,还需要继续进行研究。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Lipin1 as a therapeutic target for respiratory insufficiency of duchenne muscular dystrophy.

In Duchenne muscular dystrophy (DMD), diaphragm muscle dysfunction results in respiratory insufficiency which is a leading cause of death in patients. Mutations to the dystrophin gene result in myocyte membrane instability, contributing to the structural deterioration of the diaphragm muscle tissues. With previous works suggesting the importance of lipin1 for maintaining skeletal muscle membrane integrity, we explored the roles of lipin1 in the dystrophic diaphragm. We found that the protein expression levels of lipin1 were reduced by 60% in the dystrophic diaphragm. While further knockdown of lipin1 in the dystrophic diaphragm leads to increased necroptosis, restoration of lipin1 in the dystrophic diaphragm results in reduced inflammation and fibrosis, decreased myofiber death, and improved respiratory function. Our results demonstrated that lipin1 restoration improved respiratory function by enhancing membrane integrity and suggested that lipin1 could be a potential therapeutic target for preventing respiratory insufficiency and respiratory failure in DMD. Continued investigation is required to better understand the mechanisms behind these findings, and to determine the role of lipin1 in maintaining muscle membrane stability.

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来源期刊
CiteScore
6.50
自引率
5.00%
发文量
2608
审稿时长
14 weeks
期刊介绍: Frontiers in Physiology is a leading journal in its field, publishing rigorously peer-reviewed research on the physiology of living systems, from the subcellular and molecular domains to the intact organism, and its interaction with the environment. Field Chief Editor George E. Billman at the Ohio State University Columbus is supported by an outstanding Editorial Board of international researchers. This multidisciplinary open-access journal is at the forefront of disseminating and communicating scientific knowledge and impactful discoveries to researchers, academics, clinicians and the public worldwide.
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