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    Characterization of Vestibular Dysfunction in the Mouse Model for Usher Syndrome 1F

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    Author
    Alagramam, Kumar N.; Stahl, John S.; Jones, Sherri M.; Pawlowski, Karen S.; Wright, Charles G.
    Abstract
    The deaf-circling Ames waltzer (av) mouse harbors a mutation in the protocadherin 15 (Pcdh15) gene and is a model for inner ear defects associated with Usher syndrome type 1F. Earlier studies showed altered cochlear hair cell morphology in young av mice. In contrast, no structural abnormality consistent with significant vestibular dysfunction in young av mice was observed. Light and scanning electron microscopic studies showed that vestibular hair cells from presumptive null alleles Pcdh15av-Tg and Pcdh15av-3J are morphologically similar to vestibular sensory cells from control littermates, suggesting that the observed phenotype in these alleles might be a result of a central, rather than peripheral, defect. In the present study, a combination of physiologic and anatomic methods was used to more thoroughly investigate the source of vestibular dysfunction in Ames waltzer mice. Analysis of vestibular evoked potentials and angular vestibulo-ocular reflexes revealed a lack of physiologic response to linear and angular acceleratory stimuli in Pcdh15 mutant mice. Optokinetic reflex function was diminished but still present in the mutant animals, suggesting that the defect is primarily peripheral in nature. These findings indicate that the mutation in Pcdh15 results in either a functional abnormality in the vestibular receptor organs or that the defects are limited to the vestibular nerve. AM1-43 dye uptake has been shown to correlate with normal transduction function in hair cells. Dye uptake was found to be dramatically reduced in Pcdh15 mutants compared to control littermates, suggesting that the mutation affects hair cell function, although structural abnormalities consistent with significant vestibular dysfunction are not apparent by light and scanning electron microscopy in the vestibular neuroepithelia of young animals. Originally published J Assoc Res Otolaryngol, Vol. 6, No. 2, June 2005
    URI
    http://hdl.handle.net/10342/3199
    Subject
     Deafness; Vestibular defects; Ames waltzer; Protocadherin 15 (pcdh15); VsEP; AVOR 
    Date
    2005-06
    Citation:
    APA:
    Alagramam, Kumar N., & Stahl, John S., & Jones, Sherri M., & Pawlowski, Karen S., & Wright, Charles G.. (June 2005). Characterization of Vestibular Dysfunction in the Mouse Model for Usher Syndrome 1F. Journal of the Association for Research in Otolaryngology, 6(2), 106- 118. Retrieved from http://hdl.handle.net/10342/3199

    Display/Hide MLA, Chicago and APA citation formats.

    MLA:
    Alagramam, Kumar N., and Stahl, John S., and Jones, Sherri M., and Pawlowski, Karen S., and Wright, Charles G.. "Characterization of Vestibular Dysfunction in the Mouse Model for Usher Syndrome 1F". Journal of the Association for Research in Otolaryngology. 6:2. (106-118), June 2005. February 23, 2019. http://hdl.handle.net/10342/3199.
    Chicago:
    Alagramam, Kumar N. and Stahl, John S. and Jones, Sherri M. and Pawlowski, Karen S. and Wright, Charles G., "Characterization of Vestibular Dysfunction in the Mouse Model for Usher Syndrome 1F," Journal of the Association for Research in Otolaryngology 6, no. 2 (June 2005), http://hdl.handle.net/10342/3199 (accessed February 23, 2019).
    AMA:
    Alagramam, Kumar N., Stahl, John S., Jones, Sherri M., Pawlowski, Karen S., Wright, Charles G.. Characterization of Vestibular Dysfunction in the Mouse Model for Usher Syndrome 1F. Journal of the Association for Research in Otolaryngology. June 2005; 6(2): 106-118. http://hdl.handle.net/10342/3199. Accessed February 23, 2019.
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