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Roles of the Primary Cilium Component Polaris in Synchondrosis Development

¹ Department of Orthopaedic Surgery, Thomas Jefferson University College of Medicine, 1015 Walnut Street, Curtis Building, Room 501, Philadelphia, PA 19107, USA;
² Department of Oral Pathology, Asahi University School of Dentistry, Mizuho, Gifu, 501-0296, Japan 1581;
³ Department of Cell Biology, University of Alabama at Birmingham, Birmingham, AL 35294, USA; and
⁴ Department of Oral Pathology, Matsumoto Dental University School of Dentistry, Shiojiri, Nagano, 399-0781, Japan

Correspondence: ^* eiki.koyama{at}jefferson.edu

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 intramembranous 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.

Key Words: Polaris • primary cilium • cranial base synchondrosis • hedgehog signaling • heparan sulfate proteoglycans

Journal of Dental Research, Vol. 88, No. 6, 545-550 (2009)
DOI: 10.1177/0022034509337775


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