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- W2039160052 abstract "Mutations in sarcoglycans (α-, β-, γ-, and δ-) have been linked with limb girdle muscular dystrophy (LGMD) types 2C–F in humans. We have cloned the zebrafish orthologue encoding δ-sarcoglycan and mapped the gene to linkage group 21. The predicted zebrafish δ-sarcoglycan protein is highly homologous with its human orthologue including conservation of two of the three predicted glycosylation sites. Like other members of the dystrophin-associated protein complex (DAPC), δ-sarcoglycan localizes to the sarcolemmal membrane of the myofiber in adult zebrafish, but is more apparent at the myosepta in developing embryos. Zebrafish embryos injected with morpholinos against δ-sarcoglycan were relatively inactive at 5 dpf, their myofibers were disorganized, and swim bladders uninflated. Immunohistochemical and immunoblotting experiments show that δ-, β-, and γ-sarcoglycans were all downregulated in the morphants, whereas dystrophin expression was unaffected. Whereas humans lacking δ-sarcoglycan primarily show adult phenotypes, our results suggest that δ-sarcoglycan plays a role in early zebrafish muscle development." @default.
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- W2039160052 date "2005-03-10" @default.
- W2039160052 modified "2023-10-16" @default.
- W2039160052 title "δ-Sarcoglycan is required for early zebrafish muscle organization" @default.
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- W2039160052 doi "https://doi.org/10.1016/j.yexcr.2004.10.032" @default.
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