@misc{oai:niigata-u.repo.nii.ac.jp:00006049, author = {Kitamura, Atsushi}, month = {Mar}, note = {The primary cilia play a critical role in many biological processes including bone and cartilage formation. 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, it remains unclear whether the primary cilia are involved in mandibular development. To address this question, we generated mice with a mesenchymal deletion of ciliary protein, Ift88 (Ift88^; Wnt1Cre) and Ofd1 (Ofd1^; Wnt1Cre). Both Ofd1and Ift88 mutant mice showed ectopic mandibular bone, which was found to be associated with the downregulation of Shh signaling. Unlike mandibular bone, Meckel’s cartilage showed different phenotypes between Ofd1^; Wnt1Cre and Ift88^; Wnt1Cre mice. Bifurcated and enlarged Meckel’s cartilage were observed in the posterior mandible of Ofd1^; Wnt1Cre mice, whereas Ift88^; Wnt1Cre mice exhibited enlarged Meckel’s cartilage in the entire mandible. Chondrogenesis and osteogenesis during mandibular development is thus regulated by the primary cilia, and each ciliary protein is likely to have a distinct role, depending on the regions., 学位の種類: 博士(歯学). 報告番号: 甲第4432号. 学位記番号: 新大院博(歯)甲第395号. 学位授与年月日: 平成30年3月23日, 新大院博(歯)甲第395号}, title = {Primary cilia regulate mandibular development}, year = {2018} }