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Management and treatment for the behavioral symptom of Prader-Willi syndrome

Research Project

Project/Area Number 15K19630
Research Category

Grant-in-Aid for Young Scientists (B)

Allocation TypeMulti-year Fund
Research Field Pediatrics
Research InstitutionNational Center for Child Health and Development

Principal Investigator

Ayabe Tadayuki  国立研究開発法人国立成育医療研究センター, 生体防御系内科部, 研究員 (80566555)

Co-Investigator(Renkei-kenkyūsha) NAGAI Toshiro  東埼玉中川の郷療育センター, 小児科, 医師
IHARA Hiroshi  獨協医科大学, 埼玉医療センター・精神科, 教授
KANBAYASHI Takashi  秋田大学, 医学(系)研究科(研究院), 准教授 (50323150)
OMOKAWA Mayu  秋田大学, 医学部, 医員
Project Period (FY) 2015-04-01 – 2018-03-31
Project Status Completed (Fiscal Year 2017)
Budget Amount *help
¥4,160,000 (Direct Cost: ¥3,200,000、Indirect Cost: ¥960,000)
Fiscal Year 2017: ¥520,000 (Direct Cost: ¥400,000、Indirect Cost: ¥120,000)
Fiscal Year 2016: ¥650,000 (Direct Cost: ¥500,000、Indirect Cost: ¥150,000)
Fiscal Year 2015: ¥2,990,000 (Direct Cost: ¥2,300,000、Indirect Cost: ¥690,000)
Keywordsプラダー・ウィリ症候群 / 日中過眠 / オレキシン / 糖尿病 / SGLT2阻害薬 / 食行動異常
Outline of Final Research Achievements

People with Prader-Willi syndrome have clinical problems which are greatly different with age.
In this study, we first focused on excessive daytime sleepness (EDS) deprived after adolescence, and clarified that the clinical condition is difficult to maintain arousal due to reduction in cerebrospinal fluid orexin level.
Next, we examined the effect of SGLT inhibitors on those with diabetes mellitus. Although improvement of glycemic control can be obtained in the short term, it is difficult to maintain good glycemic control in the long term.

Report

(4 results)
  • 2017 Annual Research Report   Final Research Report ( PDF )
  • 2016 Research-status Report
  • 2015 Research-status Report
  • Research Products

    (14 results)

All 2018 2017 2016 2015

All Journal Article (6 results) (of which Int'l Joint Research: 4 results,  Peer Reviewed: 4 results,  Open Access: 1 results) Presentation (8 results) (of which Int'l Joint Research: 1 results,  Invited: 3 results)

  • [Journal Article] Aberrant autistic, and food-related behaviors in adults with Prader-Willi syndrome: The comparison between young adults and adults.2018

    • Author(s)
      Hiroyuki Ogata, Hiroshi Ihara, Masao Gito, Masayuki Sayama, Nobuyuki Murakami, Tadayuki Ayabe, Yuji Oto, Toshiro Nagai, Kazutaka Shimoda.
    • Journal Title

      Research in Development Disabilities

      Volume: 73 Pages: 126-134

    • DOI

      10.1016/j.ridd.2017.12.020

    • Related Report
      2017 Annual Research Report
    • Peer Reviewed / Int'l Joint Research
  • [Journal Article] Delayed peak response of cortisol to insulin tolerance test in patients with Prader-Willi syndrome2018

    • Author(s)
      Yuji Oto, Keiko Matsubara, Tadayuki Ayabe, Masahisa Shiraishi, Nobuyuki Murakami, Hiroshi Ihara, Tomoyo Matsubara, Toshiro Nagai.
    • Journal Title

      American Journal of Medical Genetics part A

      Volume: 印刷中 Issue: 6 Pages: 1369-1374

    • DOI

      10.1002/ajmg.a.38713

    • Related Report
      2017 Annual Research Report
    • Peer Reviewed / Int'l Joint Research
  • [Journal Article] Autistic, Aberrant, and Food-Related Behaviors in Adolescents and Young Adults with Prader-Willi Syndrome: The Effects of Age and Genotype2017

    • Author(s)
      Atsuhi Ishii, Hiroshi Ihara, Hiroyuki Ogata, Masayuki Sayama, Masao Gito, Nobuyuki Murakami, Tadayuki Ayabe, Yuji Oto, Asami Takahashi, Toshiro Nagai.
    • Journal Title

      Behavioural Neurology

      Volume: - Pages: 1-10

    • DOI

      10.1155/2017/4615451

    • Related Report
      2017 Annual Research Report
    • Peer Reviewed / Int'l Joint Research
  • [Journal Article] プラダー・ウィリ症候群をもつ子ども達への関わり方2016

    • Author(s)
      綾部匡之, 永井敏郎
    • Journal Title

      小児保健研究

      Volume: 75 Pages: 312-317

    • Related Report
      2016 Research-status Report
  • [Journal Article] Decline of CSF Orexin (Hypocretin) Levels in Prader-Willi Syndrome.2016

    • Author(s)
      Mayu Omokawa, Tadayuki Ayabe, Toshiro Nagai, Aya Imanishi, Ayumi Omokawa, Seiji Nishino, Yohei Sagawa, Tetsuo Shimizu, Takashi Kanbayashi.
    • Journal Title

      American Journal of Medical Genetics part A

      Volume: 170 Issue: 5 Pages: 1181-1186

    • DOI

      10.1002/ajmg.a.37542

    • Related Report
      2015 Research-status Report
    • Peer Reviewed / Open Access / Int'l Joint Research
  • [Journal Article] プラダー・ウィリ症候群をもつ子ども達への関わり方2016

    • Author(s)
      綾部匡之, 永井敏郎.
    • Journal Title

      小児保健研究

      Volume: 74

    • Related Report
      2015 Research-status Report
  • [Presentation] 糖尿病を合併したプラダー・ウィリ症候群患者に対するトホグリフロジンの長期使用経験2017

    • Author(s)
      綾部匡之, 大戸佑二, 村上信行, 永井敏郎
    • Organizer
      第60回日本糖尿病学会年次学術集会
    • Related Report
      2017 Annual Research Report
  • [Presentation] 糖尿病管理のコツ2016

    • Author(s)
      綾部匡之
    • Organizer
      Meet The Specialists Prader-Willi syndrome
    • Place of Presentation
      東京
    • Year and Date
      2016-09-25
    • Related Report
      2016 Research-status Report
    • Invited
  • [Presentation] Reduced cerebrospinal fluid orexin in Prader-Willi syndrome patients2016

    • Author(s)
      Tadayuki Ayabe Mayu Omokawa, Yuji Oto, Maki Fukami, Nobuyuki Murakami, Takashi Kanbayashi, Toshiro Nagai.
    • Organizer
      2016 International Prader-Willi Syndrome Organisation Conference
    • Place of Presentation
      Toronto, Canada
    • Year and Date
      2016-07-21
    • Related Report
      2015 Research-status Report
    • Int'l Joint Research
  • [Presentation] 小児・思春期糖尿病治療のさらなる課題:プラダー・ウィリ症候群に伴う糖尿病の管理のコツ2016

    • Author(s)
      綾部匡之
    • Organizer
      第22回日本小児・思春期糖尿病研究会年次学術集会
    • Place of Presentation
      大阪
    • Year and Date
      2016-07-10
    • Related Report
      2016 Research-status Report
  • [Presentation] プラダー・ウィリ症候群患者の日中過眠は視床下部のオレキシン機能低下が原因である2016

    • Author(s)
      綾部匡之, 大戸佑二, 村上信行, 松原知代, 永井敏郎
    • Organizer
      第119回日本小児科学会学術集会
    • Place of Presentation
      札幌
    • Year and Date
      2016-05-13
    • Related Report
      2015 Research-status Report
  • [Presentation] プラダー・ウィリ症候群とお昼寝の話2015

    • Author(s)
      綾部匡之
    • Organizer
      第10回PWSもこもこの会(患者会)
    • Place of Presentation
      大阪
    • Year and Date
      2015-12-19
    • Related Report
      2015 Research-status Report
    • Invited
  • [Presentation] 髄液オレキシン低値は、プラダー・ウィリ症候群患者の日中過眠の要因か?2015

    • Author(s)
      綾部匡之, 面川真由, 大戸佑二, 村上信行, 神林崇, 永井敏郎
    • Organizer
      第49回日本小児内分泌学会学術集会
    • Place of Presentation
      東京
    • Year and Date
      2015-10-08
    • Related Report
      2015 Research-status Report
  • [Presentation] 日中過眠とオレキシン2015

    • Author(s)
      綾部匡之
    • Organizer
      Meet The Specialists Prader-Willi syndrome
    • Place of Presentation
      東京
    • Year and Date
      2015-09-13
    • Related Report
      2015 Research-status Report
    • Invited

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Published: 2015-04-16   Modified: 2019-03-29  

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