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Title: Mutation in the δ-subunit of the nAChR suppresses the muscle defects caused by lack of dystrophin
Authors: Etard, C.
Behra, M.
Ertzer, R.
Fischer, N.
Jesuthasan, S. 
Blader, P.
Geisler, R.
Strähle, U.
Keywords: Acetylcholine receptor
Delta subunit
Issue Date: Dec-2005
Citation: Etard, C., Behra, M., Ertzer, R., Fischer, N., Jesuthasan, S., Blader, P., Geisler, R., Strähle, U. (2005-12). Mutation in the δ-subunit of the nAChR suppresses the muscle defects caused by lack of dystrophin. Developmental Dynamics 234 (4) : 1016-1025. ScholarBank@NUS Repository.
Abstract: Normal motility of the zebrafish embryo requires a large number of gene loci, many of which have human orthologues implicated in myasthenies and other myopathies. We have identified a mutation in the zebrafish that abolishes body motility. Embryos have narrower myofibrils and lack clusters of nicotinic acetylcholine receptors (nAChRs) on the surface of the somitic muscle. We mapped the mutation to the δ-subunit of the nAChR, showing this mutant to be a new allele of the previously named sofa potato (sop). The mutant allele carries a missense mutation in the extracellular domain altering the cysteine at position 150 to an arginine. The δ-subunit is expressed in all striated muscles in embryonic and early larval stages together with the α1, β1, ε, and γ-subunits of nAChR. In contrast to mammals that show switching from the γ embryonic to the adult ε-subunit, the two subunits are coespressed in zebrafish embryos. We, furthermore, demonstrated that the sop/δ-nAChR mutation is a suppressor of the myopathy caused by lack of Dystrophin. The myofiber detachment phenotype of Dystroglycan-deficient embryos was not suppressed, suggesting that Dystrophin and Dystroglycan play distinct roles in muscle formation and maintenance of muscle integrity. © 2005 Wiley-Liss, Inc.
Source Title: Developmental Dynamics
ISSN: 10588388
DOI: 10.1002/dvdy.20592
Appears in Collections:Staff Publications

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