Cilia-mediated Hedgehog signaling controls form and function in the mammalian larynx

• Type of Material: Article
• Authors: Jacqueline M. Tabler, University of Texas at Austin; Maggie Rigney, University of Texas at Austin; Gordon Berman, Emory University; Swetha Gopalakrishnan, Institut Pasteur; Eglantine Heude, Institut Pasteur; Hadeel Al-lami, King's College London; Basil Z Yannakoudiadkis, King's College London; Rebecca D Fitch, University of Texas at Austin; Christopher Carter, University of Texas at Austin; Steven Vokes, University of Texas at Austin; Karen J Liu, King's College London; Shahragim Tajbakhsh, Institut Pasteur; SE Roian Egnor, Howard Hughes Medical Institute; John B Wallingford, University of Texas at Austin
• Language: English
• Date: 2017-02-13
• Publisher: eLife Sciences Publications
• Publication Version: Final Published Version
• Copyright Statement: © 2017, Tabler et al
• License: Creative Commons Attribution 4.0 International
• Final Published Version (URL): http://dx.doi.org/10.7554/eLife.19153
• Title of Journal or Parent Work: eLife
• ISSN: 2050-084X
• Volume: 6
• Start Page: e19153
• End Page: e19153
• Grant/Funding Information: Howard Hughes Medical Institute to Roian Egnor, John B Wallingford.; National Institutes of Health R01HD073151 to Steven Vokes.; National Institutes of Health R01HD085901 to John B Wallingford.; This work was supported by an NRSA to JT from the NIDCR (F32DE023272); funding to ST from the Institut Pasteur, Association Française contre le Myopathies, and Agence Nationale de la Recherche (Laboratoire d’Excellence Revive, Investissement d’Avenir; ANR-10-LABX-73); and NIH R01HD073151 to SAV and R01HD085901 to JBW SERE is supported by the Howard Hughes Medical Institute; JBW was once an early career scientist of the HHMI.; National Institute of Dental and Craniofacial Research F32DE023272 to Jacqueline M Tabler.
• Research Categories: Biology, Genetics; Biology, Cell; Biology, Molecular

Abstract

Acoustic communication is fundamental to social interactions among animals, including humans. In fact, deficits in voice impair the quality of life for a large and diverse population of patients. Understanding the molecular genetic mechanisms of development and function in the vocal apparatus is thus an important challenge with relevance both to the basic biology of animal communication and to biomedicine. However, surprisingly little is known about the developmental biology of the mammalian larynx. Here, we used genetic fate mapping to chart the embryological origins of the tissues in the mouse larynx, and we describe the developmental etiology of laryngeal defects in mice with disruptions in cilia-mediated Hedgehog signaling. In addition, we show that mild laryngeal defects correlate with changes in the acoustic structure of vocalizations. Together, these data provide key new insights into the molecular genetics of form and function in the mammalian vocal apparatus.