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Papers In Press, published online ahead of print July 22, 2004
Department of Bioinformatics, Laboratory of Cell Biology, Soka University, Faculty of Engineering, Tokyo, Tokyo 192-8577
Corresponding Author: shoko{at}t.soka.ac.jp
Waker-Warburg syndrome, caused by mutations in protein O-mannosyltransferase 1 (POMT1), is an autosomal recessive disorder characterized by severe brain malformation, muscular dystrophy, and structural eye abnormalities. As humans have a second POMT, hPOMT2, we cloned each Drosophila ortholog of the human POMTs and carried out RNAi knock-down to investigate the function of these proteins in vivo. Drosophila POMT2 (dPOMT2) RNAi mutant flies showed a "twisted-abdomen phenotype", in which the abdomen is twisted through 30 to 60, similar to the dPOMT1 mutant. Moreover, dPOMT2 interacted genetically with dPOMT1, suggesting that the dPOMTs function in collaboration with each other in vivo. We expressed dPOMTs in Sf21 cells and measured protein O-mannosyltransferase activity. dPOMT2 transferred a mannose to dystroglycan protein only when it was coexpressed with dPOMT1. Likewise, dPOMT1 showed protein O-mannosyltransferase activity only if coexpressed with dPOMT2, and neither dPOMT showed any activity by itself. Each dPOMT RNAi fly totally reduced protein O-mannosyltransferase activity, in spite of the specific reduction in the level of each dPOMT mRNA. The expression pattern of dPOMT2 mRNA was found to be similar to that of dPMOT1 mRNA using whole mount in situ hybridization. These results demonstrated that the two dPOMTs function as a protein O-mannosyltransferase in association with each other, in vitro and in vivo, to generate and maintain normal muscle development.
J. Biol. Chem, 10.1074/jbc.M404900200
Submitted on May 3, 2004
Revised on July 12, 2004
Accepted on July 22, 2004
The twisted-abdomen phenotype of drosophila POMT1 and POMT2 mutants coincides with their heterophilic protein O-mannosyltransferase activity
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