T. Nicolson et al., GENETIC-ANALYSIS OF VERTEBRATE SENSORY HAIR CELL MECHANOSENSATION - THE ZEBRAFISH CIRCLER MUTANTS, Neuron, 20(2), 1998, pp. 271-283
The molecular basis of sensory hair cell mechanotransduction is largel
y unknown. In order to identify genes that are essential for mechanose
nsory hair cell function, we characterized a group of recently isolate
d zebrafish motility mutants. These mutants are defective in balance a
nd swim in circles but have no obvious morphological defects. We exami
ned the mutants using calcium imaging of acoustic-vibrational and tact
ile escape responses, high resolution microscopy of sensory neuroepith
elia in live larvae, and recordings of extracellular hair cell potenti
als (microphonics). Based on the analyses, we have identified several
classes of genes. Mutations in sputnik and mariner affect hair bundle
integrity. Mutant astronaut and cosmonaut hair cells have relatively n
ormal microphonics and thus appear to affect events downstream of mech
anotransduction. Mutant orbiter, mercury, and gemini larvae have norma
l hair cell morphology and yet do not respond to acoustic-vibrational
stimuli. The microphonics of lateral line hair cells of orbiter, mercu
ry, and gemini larvae are absent or strongly reduced. Therefore, these
genes may encode components of the transduction apparatus.