Abstract
Mechanosensory hair bundles are assembled from actin-based stereocilia that project from the apical surface of hair cells in the inner ear. Stereocilia architecture is critical for the transduction of sound and accelerations, and structural defects in these mechano-sensors are a clinical cause of hearing and balance disorders in humans. Unconventional myosin motors are central to the assembly and shaping of stereocilia architecture. A sub-group of myosin motors with MyTH4-FERM domains (MYO7A, MYO15A) are particularly important in these processes, and hypothesized to act as transporters delivering structural and actin-regulatory cargos, in addition to generating force and tension. In this review, we summarize existing evidence for how MYO7A and MYO15A operate and how their dysfunction leads to stereocilia pathology. We further highlight emerging properties of the MyTH4/FERM myosin family and speculate how these new functions might contribute towards the acquisition and maintenance of mechano-sensitivity.