Identification and characterization of zebrafish mutants defective in locomotion/behavior and application to associ ated human diseases

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I identified and analyzed eight zebrafish mutants, which have defects in locomotion and behavior. For instance, mutant mi310 harbored a point mutation in PIG-U gene, which is necessary for biogenesis of GPI-anchored proteins. In mutants, GPI-anchored proteins were not generated and surface expression of voltage-gated sodium channels was perturbed, leading to loss of action potentials. As a consequence, mutant zebrafish did not show escape behavior due to hypofunction of primary sensory neurons. The corresponding gene in human is a good candidate for a responsible gene for a human motor disorder.