Pikachurin interaction with dystroglycan is diminished by defective O-mannosyl glycosylation in congenital muscular dystrophy models and rescued by LARGE overexpression

▶ O-mannosyl glycosylation of α-dystroglycan is required for its binding to pikachurin. ▶ LARGE overexpression restores pikachurin-dystroglycan interactions. ▶ Pikachurin expression at the ribbon synapses requires dystroglycan and its glycosylation.