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RFX transcription factors are essential for hearing in mice

Elkon, Ran and Milon, Beatrice and Morrison, Laura and Shah, Manan and Vijayakumar, Sarath and Racherla, Manoj and Leitch, Carmen C. and Silipino, Lorna and Hadi, Shadan and Weiss-Gayet, Michèle and Barras, Emmanuèle and Schmid, Christoph D. and Ait-Lounis, Aouatef and Barnes, Ashley and Song, Yang and Eisenman, David J. and Eliyahu, Efrat and Frolenkov, Gregory I. and Strome, Scott E. and Durand, Bénédicte and Zaghloul, Norann A. and Jones, Sherri M. and Reith, Walter and Hertzano, Ronna. (2015) RFX transcription factors are essential for hearing in mice. Nature communications, 6. p. 8549.

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Official URL: http://edoc.unibas.ch/39828/

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Abstract

Sensorineural hearing loss is a common and currently irreversible disorder, because mammalian hair cells (HCs) do not regenerate and current stem cell and gene delivery protocols result only in immature HC-like cells. Importantly, although the transcriptional regulators of embryonic HC development have been described, little is known about the postnatal regulators of maturating HCs. Here we apply a cell type-specific functional genomic analysis to the transcriptomes of auditory and vestibular sensory epithelia from early postnatal mice. We identify RFX transcription factors as essential and evolutionarily conserved regulators of the HC-specific transcriptomes, and detect Rfx1,2,3,5 and 7 in the developing HCs. To understand the role of RFX in hearing, we generate Rfx1/3 conditional knockout mice. We show that these mice are deaf secondary to rapid loss of initially well-formed outer HCs. These data identify an essential role for RFX in hearing and survival of the terminally differentiating outer HCs.
Faculties and Departments:09 Associated Institutions > Swiss Tropical and Public Health Institute (Swiss TPH)
09 Associated Institutions > Swiss Tropical and Public Health Institute (Swiss TPH) > Department of Medical Parasitology and Infection Biology > Tuberculosis Research (Gagneux)
UniBasel Contributors:Schmid, Christoph
Item Type:Article, refereed
Article Subtype:Research Article
Publisher:Nature Publishing Group
ISSN:2041-1723
Note:Publication type according to Uni Basel Research Database: Journal article
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Last Modified:30 Jun 2016 11:00
Deposited On:27 Jan 2016 08:59

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