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

Elife. 2017 Feb 13;6:e19153. doi: 10.7554/eLife.19153.


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.

Keywords: CPLANE; Fuz; cilia; ciliopathy; developmental biology; larynx; mouse; neural crest; stem cells.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Cilia / physiology*
  • Hedgehog Proteins / metabolism*
  • Larynx / abnormalities
  • Larynx / embryology*
  • Mice
  • Signal Transduction*


  • Hedgehog Proteins