Deletion mapping of the head tilt (het) gene in mice: A vestibular mutation causing specific absence of otoliths

Rebecca A. Bergstrom, Yun You, Lawrence C. Erway, Mary F. Lyon, John C. Schimenti

Research output: Contribution to journalArticlepeer-review

44 Scopus citations

Abstract

Head tilt (het) is a recessive mutation in mice causing vestibular dysfunction. Homozygotes display abnormal responses to position change and linear acceleration and cannot swim. However, they are not deaf. bet was mapped to the proximal region of mouse chromosome 17, near the T locus. Here we report anatomical characterization of het mutants and high resolution mapping using a set of chromosome deletions. The defect in het mutants is limited to the utricle and saccule of the inner ear, which completely lack otoliths. The unique specificity of the het mutation provides an opportunity to better understand the development of the vestibular system. Complementation analyses with a collection of embryonic stem (ES)- and germ cell-induced deletions localized het to an interval near the centromere of chromosome 17 that was indivisible by recombination mapping. This approach demonstrates the utility of chromosome deletions as reagents for mapping and characterizing mutations, particularly in situations where recombinational mapping is inadequate.

Original languageEnglish (US)
Pages (from-to)815-822
Number of pages8
JournalGenetics
Volume150
Issue number2
StatePublished - 1998

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