title: Genetic defects of pronephric cilia in zebrafish.
abstract: Cilia play key roles in many aspects of embryogenesis and adult physiology in vertebrates. Past genetic screens in zebrafish identified numerous defects of ciliogenesis, including several mutations in the components of the intraflagellar transport machinery. In contrast to previous studies, here we describe a collection of mutants that affect subpopulations of cilia. Mutant embryos are characterized by a shortening and an abnormal movement of kidney cilia, and in one case also a reduction of cilia length in the Kupffer's vesicle. In contrast to that, the cilia of sensory neurons, including photoreceptor cells, hair cells, and olfactory sensory cells, appear grossly intact. Motility defects of pronephric cilia vary in mutant strains from complete paralysis to an increased frequency of movement, and are associated with left-right asymmetry defects. While ciliary ultrastructure is normal in most mutants, one of the mutant loci is essential for the formation of proper microtubule architecture in the axoneme of pronephric cilia. Mutants characterized in this study reveal intriguing genetic differences between subpopulations of embryonic cilia, and provide an opportunity to study several aspects of cilia structure and function.
authors.affiliation: Department of Ophthalmology, Harvard Medical School, 243 Charles Street, Boston, MA 02114, USA.
authors.forename: Chengtian
authors.initials: C
authors.lastname: Zhao
authors.forename: Jarema
authors.initials: J
authors.lastname: Malicki
mesh_terms: D000818:Animals; D019521:Body Patterning; D002923:Cilia / Q000378:metabolism; D009154:Mutation; D009399:Nephrons / Q000002:abnormalities / Q000196:embryology*; D015027:Zebrafish / Q000002:abnormalities / Q000196:embryology* / Q000378:metabolism; D029961:Zebrafish Proteins / Q000235:genetics / Q000378:metabolism*
chemical_list: D029961:Zebrafish Proteins