Eps8 regulates hair bundle length and functional maturation of mammalian auditory hair cells
Hair cells of the mammalian cochlea are specialized for the dynamic coding of sound stimuli. The transduction of sound waves into electrical signals depends upon mechanosensitive hair bundles that project from the cell's apical surface. Each stereocilium within a hair bundle is composed of unif...
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creator | Zampini, Valeria Rüttiger, Lukas Johnson, Stuart L Franz, Christoph Furness, David N Waldhaus, Jörg Xiong, Hao Hackney, Carole M Holley, Matthew C Offenhauser, Nina Di Fiore, Pier Paolo Knipper, Marlies Masetto, Sergio Marcotti, Walter |
description | Hair cells of the mammalian cochlea are specialized for the dynamic coding of sound stimuli. The transduction of sound waves into electrical signals depends upon mechanosensitive hair bundles that project from the cell's apical surface. Each stereocilium within a hair bundle is composed of uniformly polarized and tightly packed actin filaments. Several stereociliary proteins have been shown to be associated with hair bundle development and function and are known to cause deafness in mice and humans when mutated. The growth of the stereociliar actin core is dynamically regulated at the actin filament barbed ends in the stereociliary tip. We show that Eps8, a protein with actin binding, bundling, and barbed-end capping activities in other systems, is a novel component of the hair bundle. Eps8 is localized predominantly at the tip of the stereocilia and is essential for their normal elongation and function. Moreover, we have found that Eps8 knockout mice are profoundly deaf and that IHCs, but not OHCs, fail to mature into fully functional sensory receptors. We propose that Eps8 directly regulates stereocilia growth in hair cells and also plays a crucial role in the physiological maturation of mammalian cochlear IHCs. Together, our results indicate that Eps8 is critical in coordinating the development and functionality of mammalian auditory hair cells. |
doi_str_mv | 10.1371/journal.pbio.1001048 |
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The transduction of sound waves into electrical signals depends upon mechanosensitive hair bundles that project from the cell's apical surface. Each stereocilium within a hair bundle is composed of uniformly polarized and tightly packed actin filaments. Several stereociliary proteins have been shown to be associated with hair bundle development and function and are known to cause deafness in mice and humans when mutated. The growth of the stereociliar actin core is dynamically regulated at the actin filament barbed ends in the stereociliary tip. We show that Eps8, a protein with actin binding, bundling, and barbed-end capping activities in other systems, is a novel component of the hair bundle. Eps8 is localized predominantly at the tip of the stereocilia and is essential for their normal elongation and function. Moreover, we have found that Eps8 knockout mice are profoundly deaf and that IHCs, but not OHCs, fail to mature into fully functional sensory receptors. We propose that Eps8 directly regulates stereocilia growth in hair cells and also plays a crucial role in the physiological maturation of mammalian cochlear IHCs. Together, our results indicate that Eps8 is critical in coordinating the development and functionality of mammalian auditory hair cells.</description><identifier>ISSN: 1545-7885</identifier><identifier>ISSN: 1544-9173</identifier><identifier>EISSN: 1545-7885</identifier><identifier>DOI: 10.1371/journal.pbio.1001048</identifier><identifier>PMID: 21526224</identifier><language>eng</language><publisher>United States: Public Library of Science</publisher><subject>Acoustic Stimulation ; Actin ; Action Potentials ; Adaptor Proteins, Signal Transducing - genetics ; Adaptor Proteins, Signal Transducing - metabolism ; Animals ; Biology ; Calcium - metabolism ; Calcium Channels - metabolism ; Cell Surface Extensions - metabolism ; Cell Surface Extensions - ultrastructure ; Cochlea - cytology ; Cochlea - growth & development ; Cochlea - physiology ; Cytoskeletal Proteins - genetics ; Cytoskeletal Proteins - metabolism ; Deafness ; Deafness - genetics ; Ears & hearing ; Evoked Potentials, Auditory, Brain Stem ; Exocytosis ; Gene Deletion ; Hair cells (Mechanoreceptors) ; Hair Cells, Auditory - metabolism ; Hair Cells, Auditory - ultrastructure ; Mechanotransduction, Cellular ; Mice ; Mice, Inbred C57BL ; Mice, Knockout ; Physiological aspects ; Potassium Channels - metabolism ; Proteins ; Rodents</subject><ispartof>PLoS biology, 2011-04, Vol.9 (4), p.e1001048</ispartof><rights>COPYRIGHT 2011 Public Library of Science</rights><rights>2011 Zampini et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited: Zampini V, Rüttiger L, Johnson SL, Franz C, Furness DN, et al. (2011) Eps8 Regulates Hair Bundle Length and Functional Maturation of Mammalian Auditory Hair Cells. 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regulates hair bundle length and functional maturation of mammalian auditory hair cells</title><author>Zampini, Valeria ; Rüttiger, Lukas ; Johnson, Stuart L ; Franz, Christoph ; Furness, David N ; Waldhaus, Jörg ; Xiong, Hao ; Hackney, Carole M ; Holley, Matthew C ; Offenhauser, Nina ; Di Fiore, Pier Paolo ; Knipper, Marlies ; Masetto, Sergio ; Marcotti, Walter</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c760t-d4daef37e5bda308bc74e180690e8638388c6e6503256ba3a2d4e1fe04d6f9793</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2011</creationdate><topic>Acoustic Stimulation</topic><topic>Actin</topic><topic>Action Potentials</topic><topic>Adaptor Proteins, Signal Transducing - genetics</topic><topic>Adaptor Proteins, Signal Transducing - metabolism</topic><topic>Animals</topic><topic>Biology</topic><topic>Calcium - metabolism</topic><topic>Calcium Channels - metabolism</topic><topic>Cell Surface 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the dynamic coding of sound stimuli. The transduction of sound waves into electrical signals depends upon mechanosensitive hair bundles that project from the cell's apical surface. Each stereocilium within a hair bundle is composed of uniformly polarized and tightly packed actin filaments. Several stereociliary proteins have been shown to be associated with hair bundle development and function and are known to cause deafness in mice and humans when mutated. The growth of the stereociliar actin core is dynamically regulated at the actin filament barbed ends in the stereociliary tip. We show that Eps8, a protein with actin binding, bundling, and barbed-end capping activities in other systems, is a novel component of the hair bundle. Eps8 is localized predominantly at the tip of the stereocilia and is essential for their normal elongation and function. Moreover, we have found that Eps8 knockout mice are profoundly deaf and that IHCs, but not OHCs, fail to mature into fully functional sensory receptors. We propose that Eps8 directly regulates stereocilia growth in hair cells and also plays a crucial role in the physiological maturation of mammalian cochlear IHCs. Together, our results indicate that Eps8 is critical in coordinating the development and functionality of mammalian auditory hair cells.</abstract><cop>United States</cop><pub>Public Library of Science</pub><pmid>21526224</pmid><doi>10.1371/journal.pbio.1001048</doi><oa>free_for_read</oa></addata></record> |
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subjects | Acoustic Stimulation Actin Action Potentials Adaptor Proteins, Signal Transducing - genetics Adaptor Proteins, Signal Transducing - metabolism Animals Biology Calcium - metabolism Calcium Channels - metabolism Cell Surface Extensions - metabolism Cell Surface Extensions - ultrastructure Cochlea - cytology Cochlea - growth & development Cochlea - physiology Cytoskeletal Proteins - genetics Cytoskeletal Proteins - metabolism Deafness Deafness - genetics Ears & hearing Evoked Potentials, Auditory, Brain Stem Exocytosis Gene Deletion Hair cells (Mechanoreceptors) Hair Cells, Auditory - metabolism Hair Cells, Auditory - ultrastructure Mechanotransduction, Cellular Mice Mice, Inbred C57BL Mice, Knockout Physiological aspects Potassium Channels - metabolism Proteins Rodents |
title | Eps8 regulates hair bundle length and functional maturation of mammalian auditory hair cells |
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