| Project/Area Number |
18K09854
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| Research Category |
Grant-in-Aid for Scientific Research (C)
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| Allocation Type | Multi-year Fund |
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| Section | 一般 |
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| Review Section |
Basic Section 57070:Developmental dentistry-related
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| Research Institution | Osaka University |
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Principal Investigator |
Okawa Rena 大阪大学, 歯学部附属病院, 講師 (80437384)
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| Co-Investigator(Kenkyū-buntansha) |
古々本 一馬 大阪大学, 歯学部附属病院, 特任助教(常勤) (00803107)
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| Project Period (FY) |
2018-04-01 – 2021-03-31
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| Project Status |
Completed (Fiscal Year 2020)
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| Budget Amount *help |
¥4,420,000 (Direct Cost: ¥3,400,000、Indirect Cost: ¥1,020,000)
Fiscal Year 2020: ¥1,430,000 (Direct Cost: ¥1,100,000、Indirect Cost: ¥330,000)
Fiscal Year 2019: ¥1,560,000 (Direct Cost: ¥1,200,000、Indirect Cost: ¥360,000)
Fiscal Year 2018: ¥1,430,000 (Direct Cost: ¥1,100,000、Indirect Cost: ¥330,000)
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| Keywords | 低ホスファターゼ症 / 骨系統疾患 / セメント質形成不全 / 乳歯早期脱落 / アルカリホスファターゼ / 乳歯の早期脱落 / 遺伝子治療 / 歯周組織由来細胞 |
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| Outline of Final Research Achievements |
A nationwide survey of HPP in Japan was conducted, with the results analyzed specifically for understanding prominent genetic and dental manifestations of odonto and other so-called “non-odonto” types. An autosomal dominant inheritance pattern was significantly more prevalent in odonto-type patients, while an autosomal recessive pattern was predominant in the non-odonto-type. Thus, such differences should be considered during clinical treatment of HPP patients. In addition, based on those results, we established a severe mouse model possessing the most frequent gene mutation noted in patients in Japan and a mild model mouse with the second most frequent gene mutation, as well as model mice with a gene mutation resulting in symptoms that only affect teeth. Furthermore, a method for extracting and culturing periodontal cells of primary teeth was developed, which resulted in establishment of three cell lines, including odonto-type, childhood type, and perinatal severe type.
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| Academic Significance and Societal Importance of the Research Achievements |
低ホスファターゼ症は稀少疾患であり、歯科症状の詳細は解明されておらず、根本的歯科治療法は確立されていない。また、近年、全身への治療法が開発され、重症型の生命予後が改善されたが、その歯科症状は不明であった。本研究は、重症型と軽症型では遺伝形式や歯科症状が異なることを明らかにし、病型に応じた歯科的対応法が必要であることが示された。また、重症型と軽症型の原因遺伝子変異部位の特徴の解明は、モデルマウス作製などの歯科治療法確立のための基礎研究につながってきている。さらに、患者由来の乳歯歯周細胞を樹立したことによって、細胞の性質や薬剤への反応の検討が可能となっている。
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