Dystrophin in adult zebrafish muscle

Mutations in the human dystrophin gene are implicated in the fatal muscle w asting disease Duchenne Muscular Dystrophy (DMD). This gene expresses a sar colemmal-associated protein that is evolutionarily conserved, underpinning its important role in the architecture of muscle. In terms of DMD modelling , the mouse has served as a suitable vertebrate species but the pathophysio logy of the disease in the mouse does not entirely mimic human DMD. We have examined the zebrafish in order to expand the repertoire of vertebrate spe cies for muscle disease modelling, and to dissect further the functional in teractions of dystrophin. We report here the identification of an apparent zebrafish orthologue of the human dystrophin gene that expresses a 400-kDa protein that is localised to the muscle membrane surface. These data sugges t that the zebrafish may prove to be a beneficial vertebrate model to exami ne the role and functional interactions of dystrophin in disease and develo pment. (C) 2001 Academic Press.