title: Analysis and functional evaluation of the hair-cell transcriptome.
abstract: An understanding of the molecular bases of the morphogenesis, organization, and functioning of hair cells requires that the genes expressed in these cells be identified and their functions ascertained. After purifying zebrafish hair cells and detecting mRNAs with oligonucleotide microarrays, we developed a subtractive strategy that identified 1,037 hair cell-expressed genes whose cognate proteins subserve functions including membrane transport, synaptic transmission, transcriptional control, cellular adhesion and signal transduction, and cytoskeletal organization. To assess the validity of the subtracted hair-cell data set, we verified the presence of 11 transcripts in inner-ear tissue. Functional evaluation of two genes from the subtracted data set revealed their importance in hair bundles: zebrafish larvae bearing the seahorse and ift 172 mutations display specific kinociliary defects. Moreover, a search for candidate genes that underlie heritable deafness identified a human ortholog of a zebrafish hair-cell gene whose map location is bracketed by the markers of a deafness locus.
authors.affiliation: Howard Hughes Medical Institute and Laboratory of Sensory Neuroscience, The Rockefeller University, 1230 York Avenue, New York, NY 10021-6399, USA.
authors.forename: Brian M
authors.initials: BM
authors.lastname: McDermott
authors.forename: Jessica M
authors.initials: JM
authors.lastname: Baucom
authors.forename: A J
authors.initials: AJ
authors.lastname: Hudspeth
mesh_terms: D000818:Animals; D003638:Deafness / Q000235:genetics / Q000378:metabolism; D018507:Gene Expression Regulation, Developmental / Q000235:genetics*; D006198:Hair Cells, Auditory / Q000196:embryology / Q000378:metabolism* / Q000502:physiology; D022781:Hepatocytes / Q000378:metabolism; D006801:Humans; D051379:Mice; D009154:Mutation; D020411:Oligonucleotide Array Sequence Analysis; D014158:Transcription, Genetic; D015027:Zebrafish / Q000196:embryology / Q000235:genetics* / Q000378:metabolism