Roles of the primary cilium component polaris in synchondrosis development

T. Ochiai, M. Nagayama, T. Nakamura, T. Morrison, D. Pilchak, N. Kondo, H. Hasegawa, B. Song, R. Serra, M. Pacifici, E. Koyama

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12 Citations (Scopus)

Abstract

Primary cilia regulate several developmental processes and mediate hedgehog signaling. To study their roles in cranial base development, we created conditional mouse mutants deficient in Polaris, a critical primary cilium component, in cartilage. Mutant post-natal cranial bases were deformed, and their synchondrosis growth plates were disorganized. Expression of Indian hedgehog, Patched-1, collagen X, and MMP-13 was reduced and accompanied by decreases in endochondral bone. Interestingly, there was excessive intramem- branous ossification along the perichondrium, accompanied by excessive Patched-1 expression, suggesting that Ihh distribution was wider and responsible for such excessive response. Indeed, expression of heparan sulfate proteoglycans (HS-PGs), normally involved in restricting hedgehog distribution, was barely detectable in mutant synchondroses. Analyses of the data provides further evidence for the essential roles of primary cilia and hedgehog signaling in cranial base development and chondrocyte maturation, and point to a close interdependence between cilia and HS-PGs to delimit targets of hedgehog action in synchondroses.

Original languageEnglish
Pages (from-to)545-550
Number of pages6
JournalJournal of dental research
Volume88
Issue number6
DOIs
Publication statusPublished - 2009 Jun 1
Externally publishedYes

Keywords

  • Cranial base synchondrosis
  • Hedgehog signaling
  • Heparan sulfate proteoglycans
  • Polaris
  • Primary cilium

ASJC Scopus subject areas

  • Dentistry(all)

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    Ochiai, T., Nagayama, M., Nakamura, T., Morrison, T., Pilchak, D., Kondo, N., Hasegawa, H., Song, B., Serra, R., Pacifici, M., & Koyama, E. (2009). Roles of the primary cilium component polaris in synchondrosis development. Journal of dental research, 88(6), 545-550. https://doi.org/10.1177/0022034509337775