Department of Immunology, Graduate School of Pharmaceutical Sciences, Osaka University, Suita, Osaka, Japan.
Am J Pathol. 2010 May;176(5):2414-24. doi: 10.2353/ajpath.2010.090887. Epub 2010 Mar 19.
Duchenne muscular dystrophy (DMD) is the most common lethal genetic disorder of children. The mdx (C57BL/10 background, C57BL/10-mdx) mouse is a widely used model of DMD, but the histopathological hallmarks of DMD, such as the smaller number of myofibers, accumulation of fat and fibrosis, and insufficient regeneration of myofibers, are not observed in adult C57BL/10-mdx except for in the diaphragm. In this study, we showed that DBA/2 mice exhibited decreased muscle weight, as well as lower myofiber numbers after repeated degeneration-regeneration cycles. Furthermore, the self-renewal efficiency of satellite cells of DBA/2 is lower than that of C57BL/6. Therefore, we produced a DBA/2-mdx strain by crossing DBA/2 and C57BL/10-mdx. The hind limb muscles of DBA/2-mdx mice exhibited lower muscle weight, fewer myofibers, and increased fat and fibrosis, in comparison with C57BL/10-mdx. Moreover, remarkable muscle weakness was observed in DBA/2-mdx. These results indicate that the DBA/2-mdx mouse is a more suitable model for DMD studies, and the efficient satellite cell self-renewal ability of C57BL/10-mdx might explain the difference in pathologies between humans and mice.
杜氏肌营养不良症(DMD)是儿童中最常见的致命性遗传疾病。mdx(C57BL/10 背景,C57BL/10-mdx)小鼠是 DMD 的广泛使用模型,但 DMD 的组织病理学特征,如肌纤维数量减少、脂肪和纤维化积累以及肌纤维再生不足,除了在膈肌外,在成年 C57BL/10-mdx 中并不明显。在这项研究中,我们表明 DBA/2 小鼠在反复退化-再生循环后表现出肌肉重量减轻和肌纤维数量减少。此外,DBA/2 卫星细胞的自我更新效率低于 C57BL/6。因此,我们通过将 DBA/2 与 C57BL/10-mdx 杂交产生了 DBA/2-mdx 品系。与 C57BL/10-mdx 相比,DBA/2-mdx 小鼠的后肢肌肉重量降低,肌纤维数量减少,脂肪和纤维化增加。此外,DBA/2-mdx 还观察到明显的肌肉无力。这些结果表明,DBA/2-mdx 小鼠是 DMD 研究的更合适模型,而 C57BL/10-mdx 中卫星细胞高效的自我更新能力可能解释了人类和小鼠之间病理差异的原因。