The role of retinoid signaling in pathogenesis of choanal atresia and stenosis
| Project/Area Number |
15H05687
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| Research Category |
Grant-in-Aid for Young Scientists (A)
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| Allocation Type | Single-year Grants |
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| Research Field |
Orthodontics/Pediatric dentistry
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| Research Institution | Osaka University |
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Principal Investigator |
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| Co-Investigator(Renkei-kenkyūsha) |
Yamashiro Takashi 大阪大学, 大学院・歯学研究科, 教授 (70294428)
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| Research Collaborator |
Nakaya Akihiro
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| Project Period (FY) |
2015-04-01 – 2018-03-31
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| Project Status |
Completed (Fiscal Year 2017)
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| Budget Amount *help |
¥23,790,000 (Direct Cost: ¥18,300,000、Indirect Cost: ¥5,490,000)
Fiscal Year 2017: ¥7,150,000 (Direct Cost: ¥5,500,000、Indirect Cost: ¥1,650,000)
Fiscal Year 2016: ¥7,800,000 (Direct Cost: ¥6,000,000、Indirect Cost: ¥1,800,000)
Fiscal Year 2015: ¥8,840,000 (Direct Cost: ¥6,800,000、Indirect Cost: ¥2,040,000)
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| Keywords | 顎顔面形成不全 / 後鼻孔閉鎖 / 口唇口蓋裂 / レチノイン酸シグナル / 発生・分化 / レチノイン酸 / 後鼻口閉鎖 / 後鼻孔閉鎖症 |
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| Outline of Final Research Achievements |
Craniofacial development is a complex process that involves sequential growth and fusion of the facial prominences. When these processes fail, congenital craniofacial anomalies can occur. For example, choanal atresia (CA) is a congenital craniofacial anomaly in which the connection between the nasal airway and nasopharynx is completely blocked. In this study, we discovered that mice with mutations in retinol dehydrogenase 10 (Rdh10), which perturbs Vitamin A metabolism and retinoid signaling, exhibit fully penetrant CA. Interestingly, we demonstrate Rdh10 is specifically required in non-neural crest cells prior to E10.5 for proper choanae formation, and that in the absence of Rdh10, Fgf8 is ectopically expressed in the nasal fin. Taken together, our findings demonstrate that RDH10 is essential during the early stages of facial morphogenesis for the formation of a functional nasal airway, and furthermore establish Rdh10 mutant mice as an important model system to study CA.
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Report
(4 results)
Research Products
(37 results)
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[Presentation] Novel mutation of DLC1 in familial cleft palate case2018
Author(s)
Yanran Wu, Qi Wang, Chisato Morita, Akihiro Nakaya, Atsuko Okazaki, Kaori Kobayashi, Masataka Kikuchi, Tomoji Mashimo, Yoshihiro Uno, Shinya Oki, Takashi Yamashiro
Organizer
Gordon Research Conference Craniofacial Morphogenesis and Tissue Regeneration
Related Report
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[Presentation] The role of Rdh10 in palatal development2015
Author(s)
Hiroshi Kurosaka, Qi Wang, Lisa Sandell and Paul Trainor.
Organizer
International Symposium 2015 Oral and Craniofacial Development and Diseases
Place of Presentation
吹田市、大阪府、大阪大学歯学部
Year and Date
2015-12-10
Related Report
Int'l Joint Research
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