The role of primary cilia in amelogenesis

• Project/Area Number: 16K20641
• Research Category: Grant-in-Aid for Young Scientists (B)
• Allocation Type: Multi-year Fund
• Research Field: Orthodontics/Pediatric dentistry
• Research Institution: Niigata University
• Principal Investigator: kawasaki katsushige 新潟大学, 医歯学系, 助教 (40529640)
• Project Period (FY): 2016-04-01 – 2018-03-31
• Project Status: Completed (Fiscal Year 2017)
• Budget Amount: ¥3,900,000 (Direct Cost: ¥3,000,000、Indirect Cost: ¥900,000); Fiscal Year 2017: ¥1,560,000 (Direct Cost: ¥1,200,000、Indirect Cost: ¥360,000); Fiscal Year 2016: ¥2,340,000 (Direct Cost: ¥1,800,000、Indirect Cost: ¥540,000)
• Keywords: 一次繊毛 / エナメル質 / エナメル質形成不全 / primary cilia

Outline of Final Research Achievements

Key features of the oral-facial-digital syndrome type 1 (OFD1) include amelogenesis imperfecta (AI). We generated epithelium-specific mutations of Ofd1 using the Cre/loxP -recombination system, and analyzed the role of in amelogenesis.
The cKO teeth by SEM analysis showed dental attrition of the molar cusps in three-days-old mice as compared to the control. Real-time PCR analysis showed that Gli1 mRNA levels decreased significantly in ameloblasts of cKO mice.
These results demonstrate that Ofd1 has important roles in Ameligenesis in murine teeth. We suggested that the amelogenesis imperfecta on the Ofd1 cKO mice associated with Shh pathway.