Dystrophin and dystroglycan, the main components of the dystrophin-glycoprotein complex, are essential for normal vital activity of the D. melanogaster during ontogenesis. Mutations in the genes, which encode these proteins, cause different phenotypes, among manifestations of which we noticed characters similar to those observed at human muscular dystrophy. Furthermore, several other phenotypes have beendescribed; their study will shed light on our understanding the development of different pathologies. In addition, it gives an opportunity to reveal new components of the dystrophin-glycoprotein complex as well as new regulatory proteins, which influence the functions of this complex. Thus, based on the D. melanogaster, an appropriate model has been developed to study human myopathies.

Keywords: Drosophila melanogaster, Dystrophin-Glycoprotein complex, muscular dystrophies, dystrophin (dystroglycan) expression