| Project/Area Number |
16K09987
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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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| Research Field |
Pediatrics
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| Research Institution | Nagoya University |
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Principal Investigator |
Natsume Jun 名古屋大学, 医学系研究科, 寄附講座教授 (60422771)
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| Co-Investigator(Kenkyū-buntansha) |
城所 博之 名古屋大学, 医学部附属病院, 助教 (20647466)
前澤 聡 名古屋大学, 脳とこころの研究センター, 特任准教授 (90566960)
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| Project Period (FY) |
2016-04-01 – 2019-03-31
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| Project Status |
Completed (Fiscal Year 2018)
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| Budget Amount *help |
¥4,680,000 (Direct Cost: ¥3,600,000、Indirect Cost: ¥1,080,000)
Fiscal Year 2018: ¥1,300,000 (Direct Cost: ¥1,000,000、Indirect Cost: ¥300,000)
Fiscal Year 2017: ¥1,040,000 (Direct Cost: ¥800,000、Indirect Cost: ¥240,000)
Fiscal Year 2016: ¥2,340,000 (Direct Cost: ¥1,800,000、Indirect Cost: ¥540,000)
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| Keywords | てんかん / ウェスト症候群 / 脳波 / 機能的MRI |
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| Outline of Final Research Achievements |
We examined 7 patients with West syndrome (6 patients with cryptogenic West syndrome and 1 with tuberous sclerosis complex) by EEG-fMRI at the onset of West syndrome before ACTH therapy. The temporal information of periodic hypsarrhythmia was used to perform event-related analysis for detecting hypsarrhythmia-related blood oxygenation level-dependent (BOLD) changes on fMRI. As a result, positive BOLD changes were seen in brainstem and multiple cerebral cortices in 6 of 7 patients. After ACTH therapy epileptic spasms disappeared in all patients. However spasms relapsed in 2 patients and focal seizures were seen in 2. At 12 months of age, 3 patients had focal epileptiform discharges. EEG-fMRI at 12 months of age in these 3 patients revealed bilateral frontal positive BOLD in 2 patients and left hippocampal positive BOLD in 1. Positive BIOLD signal in the brainstem disappeared at 12 months of age in all 3 patients.
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| Academic Significance and Societal Importance of the Research Achievements |
今回の研究結果から、ヒプスアリズミアと呼ばれるWest症候群に特異的なてんかん性活動には、脳幹と大脳皮質の異常なネットワーク形成が関わっていることが示唆された。また、治療により脳波でヒプスアリズミアが焦点性てんかん放電に変化したのに伴いEEG-fMRIでは脳幹の異常活動が消失したことから、EEG-fMRIは治療効果の客観的評価にも有用と考えられた。1歳のEEG-fMRIで左海馬に陽性BOLDが存続する1例では同部位に局在性皮質形成異常が潜在する可能性があり、てんかん外科治療が有効な可能性があると考えられた。
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