Although it is known that the complete knockout of Myo1e or its selective knockout in podocytes are sufficient to induce FSGS-like disease in mice (Chase et al., 2012; Krendel et al., 2009), the effects of substituting wild-type Myo1e with the mutant versions associated with human FSGS have not been tested in a model organism. Here, MYO1E is linked to focal segmental glomerulosclerosis.