{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,12]],"date-time":"2026-06-12T01:53:19Z","timestamp":1781229199571,"version":"3.54.1"},"reference-count":35,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2019,10,28]],"date-time":"2019-10-28T00:00:00Z","timestamp":1572220800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["18K09762"],"award-info":[{"award-number":["18K09762"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Journal of Anatomy"],"published-print":{"date-parts":[[2020,2]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>The mandible is a crucial organ in both clinical and biological fields due to the high frequency of congenital anomalies and the significant morphological changes during evolution. Primary cilia play a critical role in many biological processes, including the determination of left\/right axis patterning, the regulation of signaling pathways, and the formation of bone and cartilage. Perturbations in the function of primary cilia are known to cause a wide spectrum of human diseases: the ciliopathies. Craniofacial dysmorphologies, including mandibular deformity, are often seen in patients with ciliopathies. Mandibular development is characterized by chondrogenesis and osteogenesis; however, the role of primary cilia in mandibular development is not fully understood. To address this question, we generated mice with mesenchymal deletions of the ciliary protein, <jats:italic>Ift88<\/jats:italic> (<jats:italic>Ift88<\/jats:italic><jats:sup><jats:italic>fl\/fl<\/jats:italic><\/jats:sup><jats:italic>;Wnt1Cre<\/jats:italic>). <jats:italic>Ift88<\/jats:italic><jats:sup><jats:italic>fl\/fl<\/jats:italic><\/jats:sup><jats:italic>;Wnt1Cre<\/jats:italic> mice showed ectopic mandibular bone formation, whereas <jats:italic>Ift88<\/jats:italic> mutant mandible was slightly shortened. Meckel's cartilage was modestly expanded in <jats:italic>Ift88<\/jats:italic><jats:sup><jats:italic>fl\/fl<\/jats:italic><\/jats:sup><jats:italic>;Wnt1Cre<\/jats:italic> mice. The downregulation of Hh signaling was found in most of the mesenchyme of <jats:italic>Ift88<\/jats:italic> mutant mandible. However, mice with a mesenchymal deletion of an essential molecule for Hh signaling activity, <jats:italic>Smo<\/jats:italic> (<jats:italic>Smo<\/jats:italic><jats:sup><jats:italic>fl\/fl<\/jats:italic><\/jats:sup><jats:italic>;Wnt1Cre<\/jats:italic>), showed only ectopic mandibular formation, whereas <jats:italic>Smo<\/jats:italic> mutant mandible was significantly shortened. <jats:italic>Ift88<\/jats:italic> is thus involved in chondrogenesis and osteogenesis during mandibular development, partially through regulating Sonic hedgehog (Shh) signaling.<\/jats:p>","DOI":"10.1111\/joa.13096","type":"journal-article","created":{"date-parts":[[2019,10,28]],"date-time":"2019-10-28T10:56:51Z","timestamp":1572260211000},"page":"317-324","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":19,"title":["<i>Ift88<\/i> is involved in mandibular 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