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

Author:

Tabler Jacqueline M1,Rigney Maggie M1,Berman Gordon J2,Gopalakrishnan Swetha3,Heude Eglantine3,Al-lami Hadeel Adel4,Yannakoudakis Basil Z4,Fitch Rebecca D1,Carter Christopher1,Vokes Steven1,Liu Karen J4,Tajbakhsh Shahragim3,Egnor SE Roian5,Wallingford John B1ORCID

Affiliation:

1. Department of Molecular Biosciences, University of Texas at Austin, Austin, United States

2. Department of Biology, Emory University, Atlanta, United States

3. Stem Cells and Development, CNRS UMR3738, Department of Developmental and Stem Cell Biology, Institut Pasteur, Paris, France

4. Department of Craniofacial Development and Stem Cell Biology, King’s College London, London, United Kingdom

5. Janelia Research Campus, Howard Hughes Medical Institute, Ashburn, United states

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.

Funder

National Institute of Dental and Craniofacial Research

National Institutes of Health

Howard Hughes Medical Institute

Publisher

eLife Sciences Publications, Ltd

Subject

General Immunology and Microbiology,General Biochemistry, Genetics and Molecular Biology,General Medicine,General Neuroscience

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