09:10: A Unique Transgenic Model for Hearing Loss

Francis, Howard W.; Cosetti, Maura K.; Culang, David; Kotla, Sumankrishna; O’Brien, Peter C.; Eberl, Daniel F.; Hannan, Frances

doi:10.1016/j.otohns.2007.06.280

Otolaryngol.--head neck surg.

2007

DOI: 10.1016/j.otohns.2007.06.280

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09:10: A Unique Transgenic Model for Hearing Loss

Howard W. Francis¹,

Maura K. Cosetti²,

David Culang³

et al.

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Unique Transgenic Animal Model for Hereditary Hearing Loss

Cosetti¹,

Culang²,

Kotla³

et al. 2008

Ann Otol Rhinol Laryngol

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Objectives This study capitalizes on the unique molecular and developmental similarities between the auditory organs of Drosophila and mammals, to investigate genes implicated in human syndromic and nonsyndromic hearing loss in a genetically tractable experimental animal model, the fruit fly Drosophila. Methods The Drosophila counterparts of 3 human deafness genes (DIAPH1/DFNA1, ESPN/DFNB36, and TMHS/DFNB67) were identified by sequence similarity. An electrophysiological assay was used to record sound-evoked potentials in response to an acoustic stimulus, the Drosophila courtship song. Results Flies with mutations affecting the diaphanous, forked, and CG12026/TMHS genes displayed significant reductions in the amplitude of sound-evoked potentials compared to wild-type flies (p < 0.05 to p < 0.005). The mean responses were reduced from approximately 500 to 600 μV in wild-type flies to approximately 100 to 300 μV in most mutant flies. Conclusions The identification of significant auditory dysfunction in Drosophila orthologs of human deafness genes will facilitate exploration of the molecular biochemistry of auditory mechanosensation. This may eventually allow for novel diagnostic and therapeutic approaches to human hereditary hearing loss.

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Unique Transgenic Animal Model for Hereditary Hearing Loss

Cosetti¹,

Culang²,

Kotla³

et al. 2008

Ann Otol Rhinol Laryngol

Self Cite

View full text Add to dashboard Cite

show abstract

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09:10: A Unique Transgenic Model for Hearing Loss

Cited by 1 publication

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Unique Transgenic Animal Model for Hereditary Hearing Loss

Unique Transgenic Animal Model for Hereditary Hearing Loss

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