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Glycosylation of dystroglycan

The dystroglycan protein is represented by a dumbbell-shaped structure to which sugars are sequentially added, starting with O-mannosylation of a threonine/serine site. The sites of action of 3 enzymes are shown. The sites of action of Fukutin, FKRP and LARGE are unknown, yet deficiency of these enzymes also causes dystroglycanopathy. (Adapted from Hewitt 2009.)