Drosophila auditory organ genes and genetic hearing defects

Cell. 2012 Aug 31;150(5):1042-54. doi: 10.1016/j.cell.2012.06.043.


The Drosophila auditory organ shares equivalent transduction mechanisms with vertebrate hair cells, and both are specified by atonal family genes. Using a whole-organ knockout strategy based on atonal, we have identified 274 Drosophila auditory organ genes. Only four of these genes had previously been associated with fly hearing, yet one in five of the genes that we identified has a human cognate that is implicated in hearing disorders. Mutant analysis of 42 genes shows that more than half of them contribute to auditory organ function, with phenotypes including hearing loss, auditory hypersusceptibility, and ringing ears. We not only discover ion channels and motors important for hearing, but also show that auditory stimulus processing involves chemoreceptor proteins as well as phototransducer components. Our findings demonstrate mechanosensory roles for ionotropic receptors and visual rhodopsins and indicate that different sensory modalities utilize common signaling cascades.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Axonemal Dyneins / metabolism
  • Basic Helix-Loop-Helix Transcription Factors / metabolism
  • Drosophila / anatomy & histology
  • Drosophila / genetics
  • Drosophila / physiology*
  • Drosophila Proteins / genetics
  • Drosophila Proteins / metabolism
  • Hair Cells, Auditory / metabolism
  • Hearing / physiology
  • Ion Channels / genetics
  • Ion Channels / metabolism
  • Nerve Tissue Proteins / metabolism
  • Oligonucleotide Array Sequence Analysis
  • Rhodopsin / genetics
  • Rhodopsin / metabolism
  • Signal Transduction*
  • Transient Receptor Potential Channels / genetics
  • Transient Receptor Potential Channels / metabolism


  • Basic Helix-Loop-Helix Transcription Factors
  • Drosophila Proteins
  • Ion Channels
  • Nerve Tissue Proteins
  • Transient Receptor Potential Channels
  • ato protein, Drosophila
  • Rhodopsin
  • Axonemal Dyneins