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A more recent version of this article appeared on December 1, 2007
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Submitted on January 30, 2007
Accepted on September 11, 2007
Department of Biology, University of Toronto, Mississauga, ON, Canada L5L 1C6
Monitoring Editor: Jeffrey Brodsky
In vertebrates, mutations in Protein O-mannosyltransferase1 (POMT1) or POMT2 are associated with muscular dystrophy due to a requirement for O-linked mannose glycans on the Dystroglycan (Dg) protein. In this study we examine larval body wall muscles of Drosophila mutant for Dg, or RNAi knockdown for Dg, and find defects in muscle attachment, altered muscle contraction and a change in muscle membrane resistance. To determine if POMTs are required for Dg function in Drosophila we examine larvae mutant for genes encoding POMT1 or POMT2. Larvae mutant for either POMT, or doubly mutant for both, show muscle attachment and muscle contraction phenotypes identical to those associated with reduced Dg function, consistent with a requirement for O-linked mannose on Drosophila Dg. Together these data establish a central role for Dg in maintaining integrity in Drosophila larval muscles and demonstrate the importance of glycosylation to Dg function in Drosophila. This study opens the possibility of using Drosophila to investigate muscular dystrophy.
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