To address the physiological importance of BIN1 and generate a BIN1-CNM model, we created and analyzed Bin1ex20−/− mice, in which the last exon, 20, is deleted (Fig. 1C), thus causing disruption of the SH3 domain similar to some CNM patients (Fig. S1B,C) (Cowling et al., 2017). Here, BIN1 is linked to centronuclear myopathy.