Two new mouse mutants with vestibular defects that map to the highly mutable locus on chromosome 4 Dos nuevos ratones mutantes con defectos vestibulares hallados en el altamente mutable locus del cromosoma 4
- 1 March 2005
- journal article
- Published by Taylor & Francis in International Journal of Audiology
- Vol. 44 (3) , 171-177
- https://doi.org/10.1080/14992020500057434
Abstract
The purpose of this study was to characterise two new mouse mutants, carousel, and whirligig. Both were derived from a large-scale mutagenesis programme which screened for dominantly inherited mutations that cause hearing impairments and balance defects. Genetic mapping placed both mutations on the proximal region of chromosome 4. Paint-filling and clearing techniques revealed abnormalities of the lateral semicircular canal. Scanning electron microscopy showed increased numbers of outer and inner hair cells in the apical region of the organ of Corti. The behavioural, genetic, and morphological characteristics lead us to the conclusion that both mutants are probably alleles of seven previously identified mutants which all map to proximal chromosome 4 and share similar defects of the lateral semicircular canal. We suggest that this region may be particularly susceptible to ENU mutagenesis independent of genetic background.Keywords
This publication has 13 references indexed in Scilit:
- Hereditary Familial Vestibular Degenerative DiseasesAnnals of the New York Academy of Sciences, 2006
- The homeobox gene Emx2 underlies middle ear and inner ear defects in the deaf mouse mutant pardonJournal of Neurocytology, 2003
- ENU mutagenesis reveals a highly mutable locus on mouse Chromosome 4 that affects ear morphogenesisMammalian Genome, 2002
- Epidemiology of the UK population of hearing-impaired children, including characteristics of those with and without cochlear implants—audiology, aetiology, comorbidity and affluence: Epidemiología de la población infantil de hipoacúsicos en el Reino Unido, incluyendo las características de aquellos con y sin implante coclear-audiología, etiología, co-morbilidad y nivel económicoInternational Journal of Audiology, 2002
- The Wheels Mutation in the Mouse Causes Vascular, Hindbrain, and Inner Ear DefectsDevelopmental Biology, 2001
- A genetic approach to understanding auditory functionNature Genetics, 2001
- Neurogenin 1 Null Mutant Ears Develop Fewer, Morphologically Normal Hair Cells in Smaller Sensory Epithelia Devoid of InnervationJournal of the Association for Research in Otolaryngology, 2000
- Descriptive and Experimental Analysis of the Epithelial Remodellings That Control Semicircular Canal Formation in the Developing Mouse Inner EarDevelopmental Biology, 1993
- Genetic epidemiological studies of early‐onset deafness in the U.S. school‐age populationAmerican Journal of Medical Genetics, 1993
- A Technique for Preparation of Cochlear Specimens for Assessment with the Scanning Electron MicroscopeActa Oto-Laryngologica, 1978