Hedgehog activity controls opening of the primary mouth

Research output: Contribution to journalArticlepeer-review

20 Citations (Scopus)


To feed or breathe, the oral opening must connect with the gut. The foregut and oral tissues converge at the primary mouth, forming the buccopharyngeal membrane (BPM), a bilayer epithelium. Failure to form the opening between gut and mouth has significant ramifications, and many craniofacial disorders have been associated with defects in this process. Oral perforation is characterized by dissolution of the BPM, but little is known about this process. In humans, failure to form a continuous mouth opening is associated with mutations in Hedgehog (Hh) pathway members; however, the role of Hh in primary mouth development is untested. Here, we show, using Xenopus, that Hh signaling is necessary and sufficient to initiate mouth formation, and that Hh activation is required in a dose-dependent fashion to determine the size of the mouth. This activity lies upstream of the previously demonstrated role for Wnt signal inhibition in oral perforation. We then turn to mouse mutants to establish that SHH and Gli3 are indeed necessary for mammalian mouth development. Our data suggest that Hh-mediated BPM persistence may underlie oral defects in human craniofacial syndromes.
Original languageEnglish
Pages (from-to)1-7
Number of pages7
JournalDevelopmental Biology
Issue number1
Publication statusPublished - 1 Dec 2014


  • buccopharyngeal membrane
  • fibronectin
  • hedgehog
  • mouse
  • xenopus
  • primary mouth
  • stomodeum
  • WNT


Dive into the research topics of 'Hedgehog activity controls opening of the primary mouth'. Together they form a unique fingerprint.

Cite this