The neurological foundation of behavior in Prader-Willi syndrome: a 7T MRI study

• Project/Area Number: 17K10049
• Research Category: Grant-in-Aid for Scientific Research (C)
• Allocation Type: Multi-year Fund
• Section: 一般
• Research Field: Pediatrics
• Research Institution: Niigata University
• Principal Investigator: Yamada Kenichi 新潟大学, 脳研究所, 准教授 (70436773)
• Co-Investigator(Kenkyū-buntansha): 鈴木 雄治 新潟大学, 脳研究所, 准教授 (90529851) ; 鈴木 清隆 新潟大学, 脳研究所, 准教授 (40303169) ; 渡辺 将樹 新潟大学, 脳研究所, 助教 (40345517)
• Project Period (FY): 2017-04-01 – 2020-03-31
• Project Status: Completed (Fiscal Year 2019)
• Budget Amount: ¥4,680,000 (Direct Cost: ¥3,600,000、Indirect Cost: ¥1,080,000); Fiscal Year 2019: ¥1,690,000 (Direct Cost: ¥1,300,000、Indirect Cost: ¥390,000); Fiscal Year 2018: ¥1,170,000 (Direct Cost: ¥900,000、Indirect Cost: ¥270,000); Fiscal Year 2017: ¥1,820,000 (Direct Cost: ¥1,400,000、Indirect Cost: ¥420,000)
• Keywords: プラダー・ウィリ症候群 / 7テスラ / NODDI / 拡散強調画像 / 依存症 / 肥満 / 衝動制御障害 / 自閉症 / ７テスラ / ＮＯＤＤＩ / 発達 / 発達障害 / 磁気共鳴画像 / 拡散画像 / 脳 / 脳発達 / 拡散テンソル画像

Outline of Final Research Achievements

This work investigated the brain developmental characteristics of microstructure in individuals with Prader-Willi syndrome (PWS) using an ultra-high field (7-Tesla) MRI. Representative indices derived from diffusion imaging analysis were assessed in PWS participants and healthy controls with typical development. The altered indices were observed in the scattered brain regions, which were correlated with the behavioral scores. The results add the cortical evidence of altered connectivity which would contribute to behavioral characteristic in PWS.

Academic Significance and Societal Importance of the Research Achievements

長らく推定されてきたが可視化できなかった脳神経系の発達異常を、当事者自身で実証したことで、知的発達症や衝動制御障害の生物学的証拠として神経病理標本では得られない機能情報も提供する。PWS のライフサイクルに伴う行動発達変化を支える脳発達軌跡を追跡する際の基礎データとなる。当事者自身の脳発達特徴は、個別性も反映した結果となる一方で、症候の共通点を持つ他の神経疾患（ナルコレプシー, 認知症等）への応用もできる。最終的に、悩めるPWS 支援者ネットワークへのサポートにつながる。

Research Products

• [Journal Article] Developmental abnormalities of the brain exposed to childhood maltreatment detected by diffusion tensor imaging. (2019)
  • Author(s): Yamada K, Suzuki Y, Okuyama M, Watanabe M, Nakada T
  • Journal Title: Neurological Research Volume: 41 Issue: 1 Pages: 19-25
  • DOI: 10.1080/01616412.2018.1522413
  • Peer Reviewed
• [Journal Article] Novel Modified 3D Shepp-Logan Phantom Model for Simulation of MRI Radiofrequency Coil Design (2018)
  • Author(s): Watanabe M, Matsuzawa H, Yamada K, Igarashi H, Nakada T
  • Journal Title: Advanced Biomedical Engineering Volume: 7 Issue: 0 Pages: 156-159
  • DOI: 10.14326/abe.7.156
  • NAID: 130007529668
  • ISSN: 2187-5219
  • Peer Reviewed
• [Journal Article] Maturational decrease of glutamate in the human cerebral cortex from childhood to young adulthood: a 1H-MR spectroscopy study. (2017)
  • Author(s): Shimizu M, Suzuki Y, Yamada K, Ueki S, Watanabe M, Igarashi H, Nakada T.
  • Journal Title: Pediatr Res Volume: 82 Issue: 5 Pages: 749-752
  • DOI: 10.1038/pr.2017.101
  • Peer Reviewed
• [Journal Article] MRI characteristics of the glia limitans externa: A 7T study. (2017)
  • Author(s): Suzuki K, Yamada K, Nakada K, Suzuki Y, Watanabe M, Kwee IL, Nakada T
  • Journal Title: Magn Reson Imaging Volume: 44 Pages: 140-145
  • DOI: 10.1016/j.mri.2017.08.012
  • Peer Reviewed
• [Presentation] プラダー・ウィリ症候群における行動発達特性の脳神経基盤：高磁場ＭＲＩ研究 (2020)
  • Author(s): 山田謙一
  • Organizer: 日本行動科学学会 ウィンターカンファレンス
  • Invited
• [Presentation] Gray matter microstructural alteration of the brain in individuals with Prader-Willi syndrome: a 7T MRI study (2019)
  • Author(s): Yamada K, Suzuki K, Watanabe M, Suzuki Y
  • Organizer: International Prader-Willi Syndrome Organization Conference 2019
  • Int'l Joint Research
• [Presentation] プラダー・ウィリ症候群のニューロイメージング (2019)
  • Author(s): 山田謙一
  • Organizer: PWSMTS2019シンポジウム・ワークショップ
  • Invited
• [Presentation] 無鎮静小児MRI脳撮像におけるゼロテスラ・プレパレーションシステムの有用性 (2019)
  • Author(s): 山田謙一、鈴木雄治、植木智志、渡辺将樹、鈴木清隆、五十嵐博中、中田力
  • Organizer: 第30回臨床MR脳機能研究会