
pmid: 2250176
The mdx mouse has a myopathy caused by dystrophin deficiency, and is therefore biochemically and genetically homologous to human Duchenne muscular dystrophy. While mdx mouse muscle shows no dystrophin by immunoblotting, a very small percentage of myofibers appear clearly dystrophin-positive by immunofluorescence microscopy. We have characterized these rare positive-staining fibers, and conclude that they are indeed expressing dystrophin despite a nonsense mutation within the dystrophin gene. Thus, the dystrophin-positive fibers probably represent somatic reversion or suppression of the mdx mutation. Cardiac muscle and skeletal muscle from mdx mice showed dramatically different patterns of dystrophin-positive cells. However, this difference is expected given the apparent clonal nature of the reversion/suppression events, the inability of cardiac muscle to regenerate, and other differences in the developmental programs of myofibers and cardiocytes. The prevalence of dystrophin-positive cells in mdx cardiac muscle was determined to be approximately 2 x 10(-5). The observed prevalence of dystrophin-positive cardiocytes in the mdx mouse is a possible estimate of the somatic reversion rate of the mdx mutation in vivo.
Dystrophin, Mice, Inbred C57BL, Mice, Muscles, Myocardium, Animals, Muscular Dystrophy, Animal, Mice, Mutant Strains
Dystrophin, Mice, Inbred C57BL, Mice, Muscles, Myocardium, Animals, Muscular Dystrophy, Animal, Mice, Mutant Strains
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