TY - JOUR
T1 - Genetic analysis of vertebrate sensory hair cell mechanosensation
T2 - The zebrafish circler mutants
AU - Nicolson, Teresa
AU - Rüsch, Alfons
AU - Friedrich, Rainer W.
AU - Granato, Michael
AU - Ruppersberg, Johann Peter
AU - Nüsslein-Volhard, Christiane
N1 - Funding Information:
We thank Dr. Hans-Georg Frohnhöfer for providing us with the tk256e allele of mercury and Brian Crawford for help with the fish stocks. We are grateful to Angie Ribera, Suresh Jesuthasan, and Darren Gilmour for helpful discussions and comments on the manuscript. We also thank Prof. Zenner, ENT-Hospital, Tübingen, for technical support. We are also grateful to Dr. F. Bonhoeffer for continuous support. R.W.F. was supported by a fellowship from the Boehringer Ingelheim Fonds.
PY - 1998/2
Y1 - 1998/2
N2 - The molecular basis of sensory hair cell mechanotransduction is largely unknown. In order to identify genes that are essential for mechanosensory hair cell function, we characterized a group of recently isolated zebrafish motility mutants. These mutants are defective in balance and swim in circles but have no obvious morphological defects. We examined the mutants using calcium imaging of acoustic-vibrational and tactile escape responses, high resolution microscopy of sensory neuroepithelia in live larvae, and recordings of extracellular hair cell potentials (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 normal microphonics and thus appear to affect events downstream of mechanotransduction. Mutant orbiter, mercury, and gemini larvae have normal hair cell morphology and yet do not respond to acoustic- vibrational stimuli the microphonics of lateral line hair cells or orbiter, mercury, and gemini larvae are absent or strongly reduced. Therefore, these genes may encode components of the transduction apparatus.
AB - The molecular basis of sensory hair cell mechanotransduction is largely unknown. In order to identify genes that are essential for mechanosensory hair cell function, we characterized a group of recently isolated zebrafish motility mutants. These mutants are defective in balance and swim in circles but have no obvious morphological defects. We examined the mutants using calcium imaging of acoustic-vibrational and tactile escape responses, high resolution microscopy of sensory neuroepithelia in live larvae, and recordings of extracellular hair cell potentials (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 normal microphonics and thus appear to affect events downstream of mechanotransduction. Mutant orbiter, mercury, and gemini larvae have normal hair cell morphology and yet do not respond to acoustic- vibrational stimuli the microphonics of lateral line hair cells or orbiter, mercury, and gemini larvae are absent or strongly reduced. Therefore, these genes may encode components of the transduction apparatus.
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U2 - 10.1016/S0896-6273(00)80455-9
DO - 10.1016/S0896-6273(00)80455-9
M3 - Article
C2 - 9491988
AN - SCOPUS:0032006996
SN - 0896-6273
VL - 20
SP - 271
EP - 283
JO - Neuron
JF - Neuron
IS - 2
ER -