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尺侧腕伸肌在杜氏肌营养不良症犬中出现出乎意料的慢肌向快肌纤维类型转变。

Extensor carpi ulnaris muscle shows unexpected slow-to-fast fiber-type switch in Duchenne muscular dystrophy dogs.

机构信息

Department of Molecular Microbiology and Immunology, School of Medicine, The University of Missouri, Columbia, MO 65212, USA.

National Center for Advancing Translational Sciences, National Institutes of Health, Bethesda, MD 20892, USA.

出版信息

Dis Model Mech. 2021 Dec 1;14(12). doi: 10.1242/dmm.049006. Epub 2021 Dec 16.

Abstract

Aged dystrophin-null canines are excellent models for studying experimental therapies for Duchenne muscular dystrophy, a lethal muscle disease caused by dystrophin deficiency. To establish the baseline, we studied the extensor carpi ulnaris (ECU) muscle in 15 terminal age (3-year-old) male affected dogs and 15 age/sex-matched normal dogs. Affected dogs showed histological and anatomical hallmarks of dystrophy, including muscle inflammation and fibrosis, myofiber size variation and centralized myonuclei, as well as a significant reduction of muscle weight, muscle-to-body weight ratio and muscle cross-sectional area. To rigorously characterize the contractile properties of the ECU muscle, we developed a novel in situ assay. Twitch and tetanic force, contraction and relaxation rate, and resistance to eccentric contraction-induced force loss were significantly decreased in affected dogs. Intriguingly, the time-to-peak tension and half-relaxation time were significantly shortened in affected dogs. Contractile kinetics predicted an unforeseen slow-to-fast myofiber-type switch, which we confirmed at the protein and transcript level. Our study establishes a foundation for studying long-term and late-stage therapeutic interventions in dystrophic canines. The unexpected myofiber-type switch highlights the complexity of muscle remodeling in dystrophic large mammals. This article has an associated First Person interview with the first author of the paper.

摘要

老年肌营养不良症(DMD)模型犬是研究杜氏肌营养不良症(DMD)实验疗法的极佳模型,DMD 是一种由肌营养不良蛋白缺乏引起的致命肌肉疾病。为了建立基线,我们研究了 15 只终末期(3 岁)雄性患病犬和 15 只年龄/性别匹配的正常犬的尺侧腕伸肌(ECU)。患病犬表现出肌营养不良的组织学和解剖学特征,包括肌肉炎症和纤维化、肌纤维大小变化和中央化核、以及肌肉重量、肌肉与体重比和肌肉横截面积的显著减少。为了严格表征 ECU 肌肉的收缩特性,我们开发了一种新的原位测定法。患病犬的抽搐和强直力、收缩和松弛速度以及抵抗离心收缩引起的力损失显著降低。有趣的是,患病犬的最大张力时间和半松弛时间显著缩短。收缩动力学预测了一种意想不到的慢肌纤维向快肌纤维的转换,我们在蛋白质和转录水平上进行了证实。我们的研究为研究 DMD 犬的长期和晚期治疗干预奠定了基础。出乎意料的肌纤维类型转换突出了大型肌肉营养不良动物中肌肉重塑的复杂性。本文附有该论文第一作者的第一人称采访。

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