Mutations affecting cilia in zebrafish produce phenotypes that resemble human congenital disorders

Using a unique temperature sensitive mutation, we find that the Kurly protein is involved in both cilia motility and in cilia orientation in the zebrafish. Loss of Kurly leads to phenotypes associated with cilia including in left-right patterning and to cyst formation in the kidney. Kurly mutants raised at permissive temperatures are viable, but often develop severe skeletal defects, suggesting a role for this gene in skeletal formation or maintenance. Data will be presented on the cloning and characterization of this novel gene and insights into how it may function will be discussed