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    The lhfpl5 Ohnologs lhfpl5a and lhfpl5b Are Required for Mechanotransduction in Distinct Populations of Sensory Hair Cells in Zebrafish

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    Author
    Erickson, Timothy; Pacentine, Itallia V; Venuto, Alexandra; Clemens, Rachel; Nicolson, Teresa
    Abstract
    Hair cells sense and transmit auditory, vestibular, and hydrodynamic information by converting mechanical stimuli into electrical signals. This process of mechano-electrical transduction (MET) requires a mechanically gated channel localized in the apical stereocilia of hair cells. In mice, lipoma HMGIC fusion partner-like 5 (LHFPL5) acts as an auxiliary subunit of the MET channel whose primary role is to correctly localize PCDH15 and TMC1 to the mechanotransduction complex. Zebrafish have two lhfpl5 genes (lhfpl5a and lhfpl5b), but their individual contributions to MET channel assembly and function have not been analyzed. Here we show that the zebrafish lhfpl5 genes are expressed in discrete populations of hair cells: lhfpl5a expression is restricted to auditory and vestibular hair cells in the inner ear, while lhfpl5b expression is specific to hair cells of the lateral line organ. Consequently, lhfpl5a mutants exhibit defects in auditory and vestibular function, while disruption of lhfpl5b affects hair cells only in the lateral line neuromasts. In contrast to previous reports in mice, localization of Tmc1 does not depend upon Lhfpl5 function in either the inner ear or lateral line organ. In both lhfpl5a and lhfpl5b mutants, GFP-tagged Tmc1 and Tmc2b proteins still localize to the stereocilia of hair cells. Using a stably integrated GFP-Lhfpl5a transgene, we show that the tip link cadherins Pcdh15a and Cdh23, along with the Myo7aa motor protein, are required for correct Lhfpl5a localization at the tips of stereocilia. Our work corroborates the evolutionarily conserved co-dependence between Lhfpl5 and Pcdh15, but also reveals novel requirements for Cdh23 and Myo7aa to correctly localize Lhfpl5a. In addition, our data suggest that targeting of Tmc1 and Tmc2b proteins to stereocilia in zebrafish hair cells occurs independently of Lhfpl5 proteins.
    URI
    http://hdl.handle.net/10342/7836
    Subject
    hair cell, mechanotransduction, deafness, lateral line, zebrafish, LHFPL5, TMC1, PCDH15
    Date
    2020-01-15
    Citation:
    APA:
    Erickson, Timothy, & Pacentine, Itallia V, & Venuto, Alexandra, & Clemens, Rachel, & Nicolson, Teresa. (January 2020). The lhfpl5 Ohnologs lhfpl5a and lhfpl5b Are Required for Mechanotransduction in Distinct Populations of Sensory Hair Cells in Zebrafish. Frontiers in Molecular Neuroscience, (. Retrieved from http://hdl.handle.net/10342/7836

    Display/Hide MLA, Chicago and APA citation formats.

    MLA:
    Erickson, Timothy, and Pacentine, Itallia V, and Venuto, Alexandra, and Clemens, Rachel, and Nicolson, Teresa. "The lhfpl5 Ohnologs lhfpl5a and lhfpl5b Are Required for Mechanotransduction in Distinct Populations of Sensory Hair Cells in Zebrafish". Frontiers in Molecular Neuroscience. . (.), January 2020. March 08, 2021. http://hdl.handle.net/10342/7836.
    Chicago:
    Erickson, Timothy and Pacentine, Itallia V and Venuto, Alexandra and Clemens, Rachel and Nicolson, Teresa, "The lhfpl5 Ohnologs lhfpl5a and lhfpl5b Are Required for Mechanotransduction in Distinct Populations of Sensory Hair Cells in Zebrafish," Frontiers in Molecular Neuroscience 12, no. (January 2020), http://hdl.handle.net/10342/7836 (accessed March 08, 2021).
    AMA:
    Erickson, Timothy, Pacentine, Itallia V, Venuto, Alexandra, Clemens, Rachel, Nicolson, Teresa. The lhfpl5 Ohnologs lhfpl5a and lhfpl5b Are Required for Mechanotransduction in Distinct Populations of Sensory Hair Cells in Zebrafish. Frontiers in Molecular Neuroscience. January 2020; 12() . http://hdl.handle.net/10342/7836. Accessed March 08, 2021.
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