title: Mutations affecting development of the zebrafish ear.
abstract: In a large scale screen for genetic defects in zebrafish embryogenesis we identified mutations affecting several aspects of ear development, including: specification of the otic placode, growth of the otic vesicle (otocyst), otolith formation, morphogenesis of the semicircular canals and differentiation of the otic capsule. Here we report initial phenotypic and genetic characterization of 20 of these mutations defining 13 independent loci. Embryos mutant at the quadro locus display abnormal specification of the otic placode. As revealed by dlx-3 expression, the otic field in the mutant embryos is smaller or split into two fields. At later stages of development the ear of quadro mutants is frequently divided into two smaller, incomplete units. Four loci affect ear shape shortly after formation of the otic vesicle. All of them also display abnormal brain morphology. Mutations in five loci result in the absence of otolith formation; two of these also produce changes of ear morphology. Two loci, little richard and golas, affect morphology of the otic vesicle shortly before formation of the semicircular canals. In both cases the morphogenesis of the semicircular canals is disrupted. Finally, the antytalent locus is involved in late expansion of the ear structure. Analysis of mutations presented here will strengthen our understanding of vertebrate ear morphogenesis and provide novel entry points to its genetic analysis.
authors.affiliation: Cardiovascular Research Center, Massachusetts General Hospital, Charlestown 02129, USA.
authors.forename: J
authors.initials: J
authors.lastname: Malicki
authors.forename: A F
authors.initials: AF
authors.lastname: Schier
authors.forename: L
authors.initials: L
authors.lastname: Solnica-Krezel
authors.forename: D L
authors.initials: DL
authors.lastname: Stemple
authors.forename: S C
authors.initials: SC
authors.lastname: Neuhauss
authors.forename: D Y
authors.initials: DY
authors.lastname: Stainier
authors.forename: S
authors.initials: S
authors.lastname: Abdelilah
authors.forename: Z
authors.initials: Z
authors.lastname: Rangini
authors.forename: F
authors.initials: F
authors.lastname: Zwartkruis
authors.forename: W
authors.initials: W
authors.lastname: Driever
mesh_terms: D000818:Animals; D004423:Ear / Q000196:embryology*; D047108:Embryonic Development; D018507:Gene Expression Regulation, Developmental; D008297:Male; D016296:Mutagenesis; D010037:Otolithic Membrane / Q000196:embryology; D010641:Phenotype; D015027:Zebrafish / Q000033:anatomy & histology / Q000196:embryology* / Q000235:genetics*