Consistent with the results of the present study in Atoh1‐Pex5−/− mice, BK α‐subunit knockout mice were also reported to show identical hearing loss phenotype.[57] Further, mutations in the BK‐encoding sol gene are a susceptibility factor for hearing loss in human.[56] Of note, Pejvakin genes were found to be potentially associated with peroxisome proliferation, and abnormalities in BK channels were detected in Pjvk−/− mice.[19] At this point, it is not clear whether the peroxisome deficiency affects BK channels directly or indirectly through another factor(s) or pathway(s). Here, PJVK is linked to hearing loss disorder.