Abnormal cerebrospinal fluid dynamics due to ciliary movement disorder and molecular biological analysis of dynein
| Project/Area Number |
17K17792
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| Research Category |
Grant-in-Aid for Young Scientists (B)
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| Allocation Type | Multi-year Fund |
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| Research Field |
Neurosurgery
Biophysics
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| Research Institution | Nagoya University |
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Principal Investigator |
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| Project Period (FY) |
2017-04-01 – 2020-03-31
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| Project Status |
Completed (Fiscal Year 2019)
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| Budget Amount *help |
¥4,290,000 (Direct Cost: ¥3,300,000、Indirect Cost: ¥990,000)
Fiscal Year 2018: ¥1,950,000 (Direct Cost: ¥1,500,000、Indirect Cost: ¥450,000)
Fiscal Year 2017: ¥2,340,000 (Direct Cost: ¥1,800,000、Indirect Cost: ¥540,000)
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| Keywords | 水頭症 / 繊毛 / 脳室上衣細胞 / 電子顕微鏡 / ダイニン / Dpcd / サブユニット / 髄液ダイナミクス / 線毛 / Lrrc6 / 脳神経疾患 / 髄液 |
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| Outline of Final Research Achievements |
Using a hydrocephalic model mouse (Dpcd knockout mouse: Dpcd KO mouse) showing abnormal ciliary movement, the expression pattern and mechanism of hydrocephalus were investigated. This hydrocephalus showed lateral ventricle and third ventricle enlargement, but no fourth ventricle enlargement, suggesting that there is a phenotypic difference between the ventricles. Among the eight molecular species that compose the mouse inner arm dynein, it was revealed that the mRNA expression of Dnah1 was decreased and that of Dnah6 was increased. In addition, partial loss of the inner arm dynein was confirmed by structural analysis using a transmission electron microscope. It was clarified that the knockout of Dpcd gene resulted in gene polymorphism and structural abnormality of multiple dynein molecular species.
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| Academic Significance and Societal Importance of the Research Achievements |
繊毛運動障害に伴う各臓器の機能異常については報告が増えているが、脳室上衣繊毛の運動と水頭症形成の関連性については報告が少なく、特に脳室上衣繊毛ダイニンの構成タンパクの解析については未だ報告がなく、本研究は極めて新規性の高いものであった。
繊毛運動障害では非閉塞性水頭症を呈することが明らかとなった。非閉塞性水頭症は日常診療で遭遇する疾患であるが、その発症の原因、増悪の機序については未だ明らかにされていない。非閉塞性水頭症の一因として繊毛運動障害の存在が示唆され、また増悪の機序として繊毛運動ベクトルの経時的障害が存在することが示唆された。今後非閉塞性水頭症に対する治療への展開が期待される。
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Report
(4 results)
Research Products
(21 results)
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[Journal Article] H3F3A mutant allele specific imbalance in an aggressive subtype of diffuse midline glioma, H3 K27M-mutant.2020
Author(s)
Maeda S, Ohka F, Okuno Y, Aoki K, Motomura K, Takeuchi K, Kusakari H, Yanagisawa N, Sato S, Yamaguchi J, Tanahashi K, Hirano M, Kato A, Shimizu H, Kitano Y, Yamazaki S, Yamashita S, Takeshima H, Shinjo K, Kondo Y, Wakabayashi T, Natsume A.
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Journal Title
Acta Neuropathologica Communications
Volume: 8
Issue: 1
Pages: 8-19
DOI
Related Report
Peer Reviewed / Open Access
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