Centronuclear myopathies (CNM) are a group of congenital myopathies characterized by skeletal muscle weakness, fatigability and atrophy. In affected muscles, myonuclei are abnormally located at the center of mature muscle fibers since they never migrate to the cell periphery as in healthy muscles. Initially considered as a consequence of the pathology, myonuclear mispositioning has recently emerged as one of the possible causes of muscle functionality defects observed in CNM. So far, the entire molecular machinery responsible for myonuclear positioning during myofibers formation is not known but recent clues involve cytoskeleton proteins.

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