Research Project
Grant-in-Aid for Young Scientists (B)
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.
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。
All 2020 2019 2017
All Journal Article (8 results) (of which Peer Reviewed: 2 results, Open Access: 1 results) Presentation (13 results) (of which Int'l Joint Research: 3 results, Invited: 6 results)
Clinical Neurology and Neurosurgery
Volume: 193 Pages: 105764-105764
10.1016/j.clineuro.2020.105764
Acta Neuropathologica Communications
Volume: 8 Issue: 1 Pages: 8-19
10.1186/s40478-020-0882-4
Pituitary
Volume: 5 Issue: 5 Pages: 507-513
10.1007/s11102-019-00980-w
120006771422
World Neurosurgery
Volume: 132 Pages: e665-e669
10.1016/j.wneu.2019.08.051
Volume: 126 Pages: 53-58
10.1016/j.wneu.2019.02.134
120006718411
Volume: 122 Pages: e1202-e1210
10.1016/j.wneu.2018.11.017
Journal of Neurosurgery
Volume: 131 Issue: 5 Pages: 1625-1631
10.3171/2018.4.jns18366
Volume: 131 Issue: 1 Pages: 104-108
10.3171/2018.1.jns172393
