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Abstract
Zebrafish serve as a key model for auditory and vestibular research. We developed a novel method to quantify vestibular defects in larvae using the vestibulo-ocular reflex, demonstrating functional eye movements by 72 hours post-fertilization dependent on anterior/utricular otolith organs. Investigating synaptic mechanisms, we identified a point mutation (nsfI209N) that impairs synaptic transmission without altering ribbon synapse structure, while nsf null mutations cause synaptic degeneration. Nsf activity is essential for ribbon synapse maintenance, and Vamp2/Vamp8 were detected in hair cells, suggesting non-conventional SNARE-mediated transmission. Additionally, a yeast two-hybrid screen identified Tmc2a as a candidate mechanotransduction partner for Pcdh15a, warranting further study.