Ra. Bergstrom et al., DELETION MAPPING OF THE HEAD TILT (HET) GENE IN MICE - A VESTIBULAR MUTATION CAUSING SPECIFIC ABSENCE OF OTOLITHS, Genetics, 150(2), 1998, pp. 815-822
Head tilt (het) is a recessive mutation in mice causing vestibular dys
function. Homozygotes display abnormal responses to position change an
d linear acceleration and cannot swim. However, they are not deaf. hct
was mapped to the proximal region of mouse chromosome 17 near the T l
ocus. Here we report anatomical characterization of hpt mutants and hi
gh 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 mut
ation provides an opportunity to better understand the development of
the vestibular system. Complementation analyses with a collection of e
mbryonic stern (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 ch
romosome deletions as reagents for mapping and characterizing mutation
s, particularly in situations where recombinational mapping is inadequ
ate.