| Project/Area Number |
15K19642
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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 |
Pediatrics
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| Research Institution | Kurume University |
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Principal Investigator |
KIDA HIROSHI 久留米大学, 医学部, 助教 (80529454)
|
|---|
| Research Collaborator |
TAKAHASHI tomoyuki
MATSUISHI toyojiro
TANIWAKI takayuki
TANAKA eiichiro
MURAI yoshinaka
|
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| Project Period (FY) |
2015-04-01 – 2019-03-31
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| Project Status |
Completed (Fiscal Year 2018)
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| Budget Amount *help |
¥3,900,000 (Direct Cost: ¥3,000,000、Indirect Cost: ¥900,000)
Fiscal Year 2017: ¥1,690,000 (Direct Cost: ¥1,300,000、Indirect Cost: ¥390,000)
Fiscal Year 2016: ¥1,170,000 (Direct Cost: ¥900,000、Indirect Cost: ¥270,000)
Fiscal Year 2015: ¥1,040,000 (Direct Cost: ¥800,000、Indirect Cost: ¥240,000)
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| Keywords | Rett症候群 / モデルマウス / ES細胞 / イオンチャネル / 神経伝達物質 / 誤嚥性配線 / レット症候群 / 誤嚥性肺炎 / MeCP2 |
|---|
| Outline of Final Research Achievements |
Rett syndrome (RTT) is a neurodevelopmental disorder predominantly affecting females. Most cases of RTT are caused by de novo mutations in methyl-CpG binding protein 2 (MeCP2) gene on X chromosome. In this study, we analyzed the effect of MeCP2 mutation on the expression of central nervous ion channels and neurotransmitters. Glutamatergic neurons induced from RTT model ES cells were tended to immature. In RTT model mice, it was revealed that abnormal neurotransmitter expression in the brainstem caused aspiration pneumonia from dysphagia and affected its prognosis.
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| Academic Significance and Societal Importance of the Research Achievements |
本研究によって、RTTマウスでの脳幹機能不全からの嚥下反射障害、それに惹起される誤嚥性肺炎がその自然歴に与える影響が明らかになった。RTT症例においても、同様の機序からの誤嚥性肺炎はその生命予後に大きな影響を与えていると考えられる。本研究の成果により、今後、脳幹嚥下中枢の機能是正をターゲットとした、RTTに対する予後改善治療の開発に繋がることが期待される。
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