Document Detail

The transmembrane inner ear (Tmie) protein is essential for normal hearing and balance in the zebrafish.
MedLine Citation:
PMID:  19934034     Owner:  NLM     Status:  MEDLINE    
Little is known about the proteins that mediate mechanoelectrical transduction, the process by which acoustic and accelerational stimuli are transformed by hair cells of the inner ear into electrical signals. In our search for molecules involved in mechanotransduction, we discovered a line of deaf and uncoordinated zebrafish with defective hair-cell function. The hair cells of mutant larvae fail to incorporate fluorophores that normally traverse the transduction channels and their ears lack microphonic potentials in response to vibratory stimuli. Hair cells in the posterior lateral lines of mutants contain numerous lysosomes and have short, disordered hair bundles. Their stereocilia lack two components of the transduction apparatus, tip links and insertional plaques. Positional cloning revealed an early frameshift mutation in tmie, the zebrafish ortholog of the mammalian gene transmembrane inner ear. The mutant line therefore affords us an opportunity to investigate the role of the corresponding protein in mechanoelectrical transduction.
Michelle R Gleason; Aaron Nagiel; Sophie Jamet; Maria Vologodskaia; Hernán López-Schier; A J Hudspeth
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Publication Detail:
Type:  Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't     Date:  2009-11-23
Journal Detail:
Title:  Proceedings of the National Academy of Sciences of the United States of America     Volume:  106     ISSN:  1091-6490     ISO Abbreviation:  Proc. Natl. Acad. Sci. U.S.A.     Publication Date:  2009 Dec 
Date Detail:
Created Date:  2009-12-18     Completed Date:  2010-02-12     Revised Date:  2010-09-28    
Medline Journal Info:
Nlm Unique ID:  7505876     Medline TA:  Proc Natl Acad Sci U S A     Country:  United States    
Other Details:
Languages:  eng     Pagination:  21347-52     Citation Subset:  IM    
Howard Hughes Medical Institute and Laboratory of Sensory Neuroscience, The Rockefeller University, New York, NY 10065, USA.
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MeSH Terms
Ear, Inner / pathology
Frameshift Mutation
Hair Cells, Auditory / pathology
Hearing / physiology*
Mechanotransduction, Cellular / genetics
Membrane Proteins / genetics,  physiology*
Postural Balance / physiology*
Zebrafish Proteins / genetics,  physiology*
Grant Support
DC00241/DC/NIDCD NIH HHS; GM07739/GM/NIGMS NIH HHS; //Howard Hughes Medical Institute
Reg. No./Substance:
0/Membrane Proteins; 0/Tmie protein, zebrafish; 0/Zebrafish Proteins

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