Cilia-mediated Hedgehog signaling controls form and function in the mammalian larynx.
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.
Department of Molecular Biosciences, University of Texas at Austin, Austin, United States.
Jacqueline M
JM
Tabler
Department of Molecular Biosciences, University of Texas at Austin, Austin, United States.
Maggie M
MM
Rigney
Department of Biology, Emory University, Atlanta, United States.
Gordon J
GJ
Berman
Stem Cells and Development, CNRS UMR3738, Department of Developmental and Stem Cell Biology, Institut Pasteur, Paris, France.
Swetha
S
Gopalakrishnan
Stem Cells and Development, CNRS UMR3738, Department of Developmental and Stem Cell Biology, Institut Pasteur, Paris, France.
Eglantine
E
Heude
Department of Craniofacial Development and Stem Cell Biology, King's College London, London, United Kingdom.
Hadeel Adel
HA
Al-Lami
Department of Craniofacial Development and Stem Cell Biology, King's College London, London, United Kingdom.
Basil Z
BZ
Yannakoudakis
Department of Molecular Biosciences, University of Texas at Austin, Austin, United States.
Rebecca D
RD
Fitch
Department of Molecular Biosciences, University of Texas at Austin, Austin, United States.
Christopher
C
Carter
Department of Molecular Biosciences, University of Texas at Austin, Austin, United States.
Steven
S
Vokes
Department of Craniofacial Development and Stem Cell Biology, King's College London, London, United Kingdom.
Karen J
KJ
Liu
Stem Cells and Development, CNRS UMR3738, Department of Developmental and Stem Cell Biology, Institut Pasteur, Paris, France.
Shahragim
S
Tajbakhsh
Janelia Research Campus, Howard Hughes Medical Institute, Ashburn, United states.
Se Roian
SR
Egnor
Department of Molecular Biosciences, University of Texas at Austin, Austin, United States.
John B
0000-0002-6280-8625
JB
Wallingford
D000818:Animals; D002923:Cilia / Q000502:physiology*; D053823:Hedgehog Proteins / Q000378:metabolism*; D007830:Larynx / Q000002:abnormalities / Q000196:embryology*; D051379:Mice; D015398:Signal Transduction
D053823:Hedgehog Proteins
CPLANE; Fuz; cilia; ciliopathy; developmental biology; larynx; mouse; neural crest; stem cells