This review covers, in general terms, the salient features and impact of molecular myology under the following headings: its role in providing clues for the understanding of molecular etiology and pathogenesis of genetic myopathies, its contribution to the modernization and rationalization of the classification of muscle diseases, providing means of precise diagnosis and prevention of myopathies, development of radically new cell and gene therapies, and determination of future research directions. Myology appears to be among the medical disciplines that have benefited a great deal from molecular science. This remarkable progress will hopefully translate into effective treatment capabilities in the near future.