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Abstract
Dystrophin/dystrobrevin superfamily proteins play structural and signalling roles
at the plasma membrane of many cell types. Defects in them or the associated multiprotein
complex cause a range of neuromuscular disorders. Members of the dystrophin branch
of the family form heterodimers with members of the dystrobrevin branch, mediated
by their coiled-coil domains. To determine which combinations of these proteins might
interact during embryonic development, we set out to characterise the gene expression
pattern of dystrophin and dystrobrevin family members in zebrafish. gamma-dystrobrevin
(dtng), a novel dystrobrevin recently identified in fish, is the predominant form
of dystrobrevin in embryonic development. Dtng and dmd (dystrophin) have similar spatial
and temporal expression patterns in muscle, where transcripts are localized to the
ends of differentiated fibres at the somite borders. Dtng is expressed in the notochord
while dmd is expressed in the chordo-neural hinge and then in floor plate and hypochord.
In addition, dtng is dynamically expressed in rhombomeres 2 and 4-6 of the hindbrain
and in the ventral midbrain. alpha-dystrobrevin (dtna) is expressed widely in the
brain with particularly strong expression in the hypothalamus and the telencephalon;
drp2 is also expressed widely in the brain. Utrophin expression is found in early
pronephros and lateral line development and utrophin and dystrophin are both expressed
later in the gut. beta-dystrobrevin (dtnb) is expressed in the pronephric duct and
widely at low levels. In summary, we find clear instances of co-expression of dystrophin
and dystrobrevin family members in muscle, brain and pronephric duct development and
many examples of strong and specific expression of members of one family but not the
other, an intriguing finding given the presumed heterodimeric state of these molecules.