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Identification and characterization of zebrafish mutants defective in locomotion/behavior and application to associ ated human diseases

Research Project

Project/Area Number 20680023
Research Category

Grant-in-Aid for Young Scientists (A)

Allocation TypeSingle-year Grants
Research Field Laboratory animal science
Research InstitutionNagoya University

Principal Investigator

HIROMI Hirata  Nagoya University, 大学院・理学研究科, 助教 (60402450)

Project Period (FY) 2008 – 2009
Project Status Completed (Fiscal Year 2009)
Budget Amount *help
¥26,000,000 (Direct Cost: ¥20,000,000、Indirect Cost: ¥6,000,000)
Fiscal Year 2009: ¥12,350,000 (Direct Cost: ¥9,500,000、Indirect Cost: ¥2,850,000)
Fiscal Year 2008: ¥13,650,000 (Direct Cost: ¥10,500,000、Indirect Cost: ¥3,150,000)
Keywordsゼブラフィッシュ / 変異体
Research Abstract

I identified and analyzed eight zebrafish mutants, which have defects in locomotion and behavior. For instance, mutant mi310 harbored a point mutation in PIG-U gene, which is necessary for biogenesis of GPI-anchored proteins. In mutants, GPI-anchored proteins were not generated and surface expression of voltage-gated sodium channels was perturbed, leading to loss of action potentials. As a consequence, mutant zebrafish did not show escape behavior due to hypofunction of primary sensory neurons. The corresponding gene in human is a good candidate for a responsible gene for a human motor disorder.

Report

(3 results)
  • 2009 Annual Research Report   Final Research Report ( PDF )
  • 2008 Annual Research Report
  • Research Products

    (33 results)

All 2010 2009 2008

All Journal Article (13 results) (of which Peer Reviewed: 5 results) Presentation (16 results) Book (4 results)

  • [Journal Article] Biogenesis of GPI-anchored proteins is essential for surface expression of sodium channels in zebrafish Rohon-Beard neurons to respond to mechanosensory stimulation.2010

    • Author(s)
      Yuri Nakano, Morihisa Fujita, Kazutoyo Ogino, Taroh Kinoshita, Yoichi Oda, Hiromi Hirata*.
    • Journal Title

      Development 137

      Pages: 1689-1698

    • Related Report
      2009 Final Research Report
  • [Journal Article] Defective glycinergic transmission in zebrafish motility mutants.2010

    • Author(s)
      Hiromi Hirata, Eloisa Carta, Iori Yamanaka, Robert J. Harvey, John Y. Kuwada.
    • Journal Title

      Front. Mol. Neurosci. 2,26

      Pages: 1-17

    • Related Report
      2009 Final Research Report
  • [Journal Article] NaV1. 6a is required for normal activation of motor circuits normally excited by tactile stimulation.2010

    • Author(s)
      Sean E. Low, Weibin Zhou, Xinling Choong, Louis Saint-Amant, Shawn M. Sprague, Hiromi Hirata, Wilson W. Cui, Richard I. Hume, John Y. Kuwada.
    • Journal Title

      Dev Neurobiol. (In press)

    • Related Report
      2009 Final Research Report
  • [Journal Article] Biogenesis of GPI-anchored proteins is essential for surface expression of sodium channels in zebrafish Rohon -Beard neurons to respond to mechanosensory stimulation.2010

    • Author(s)
      中野由梨, 他
    • Journal Title

      Development 137

      Pages: 1689-1698

    • Related Report
      2009 Annual Research Report
    • Peer Reviewed
  • [Journal Article] NaVl.6a is required for normal activation of motor circuits nor mally excited by tactile stimulation.2010

    • Author(s)
      Low, S.E., 他
    • Journal Title

      Dev Neurobiol (印刷中)(未定)

    • Related Report
      2009 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Defective glycinergic transmission in zebrafish motility mutants.2010

    • Author(s)
      平田普三, 他
    • Journal Title

      Front. Mol.Neurosci. 2

      Pages: 1-17

    • Related Report
      2009 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Zebrafish muscular disease models towards drug discovery.2009

    • Author(s)
      Hiromi Hirata.
    • Journal Title

      Expert Opin. Drug Discov. 4

      Pages: 507-513

    • Related Report
      2009 Final Research Report
  • [Journal Article] Zebrafish muscular disease models towards drug discovery.2009

    • Author(s)
      平田普三, 他
    • Journal Title

      Expert Opin Drug Discov 4

      Pages: 507-513

    • Related Report
      2008 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Identification and expression of voltage-gated calcium channel b subunits in zebrafish.2008

    • Author(s)
      Weibin Zhou, Eric James Horstick, Hiromi Hirata, John Y. Kuwada.
    • Journal Title

      Dev. Dyn. 237

      Pages: 3842-3852

    • Related Report
      2009 Final Research Report
  • [Journal Article] The zebrafish ennui behavioral mutation disrupts acetylcholine receptor localization and motor axon stability.2008

    • Author(s)
      Saint-Amant, Shawn M. Sprague, Hiromi Hirata, Qin Li, Wilson W. Cui, Weibin Zhou, Olivier Poudou, Richard I. Hume, John Y. Kuwada.
    • Journal Title

      Dev. Neurobiol. 68

      Pages: 45-61

    • Related Report
      2009 Final Research Report
  • [Journal Article] Identification and expression of voltage-gated calcium channel beta subunits in zebrafish.2008

    • Author(s)
      Weibin Zhou, et al.
    • Journal Title

      Dev Dyn 237

      Pages: 3842-3852

    • Related Report
      2008 Annual Research Report
    • Peer Reviewed
  • [Journal Article] ゼブラフィッシュ運動と行動2008

    • Author(s)
      平田普三
    • Journal Title

      細胞工学 27

      Pages: 1125-1130

    • Related Report
      2008 Annual Research Report
  • [Journal Article] 硬骨魚類の逃避運動制御のメカニズム2008

    • Author(s)
      平田普三, 他
    • Journal Title

      実験医学 26

      Pages: 1869-1874

    • Related Report
      2008 Annual Research Report
  • [Presentation] GPI transamidase is essential for zebrafish Rohon-Beard neurons to respond to mechanosensory stimulation.2009

    • Author(s)
      Hiromi Hirata.
    • Organizer
      第32回日本分子生物学会年会
    • Place of Presentation
      パシフィコ横浜(横浜)
    • Related Report
      2009 Annual Research Report 2009 Final Research Report
  • [Presentation] 発生期のグリシン作動性シナプスの形成2009

    • Author(s)
      平田普三
    • Organizer
      日本発生生物学会秋期シンポジウム
    • Place of Presentation
      東レ総合研修センター(三島)
    • Related Report
      2009 Final Research Report
  • [Presentation] 感覚ニューロンの障害と細胞死2009

    • Author(s)
      平田普三、中野由梨、小田洋一
    • Organizer
      第32回日本神経科学大会
    • Place of Presentation
      名古屋国際会議場(名古屋)
    • Related Report
      2009 Final Research Report
  • [Presentation] Characterization of zebrafish motility mutant defective in voltage-gated sodium channel.2009

    • Author(s)
      Hiromi Hirata, Yuri Nakano, Yoichi Oda.
    • Organizer
      The 36th International Congress of Physiological Sciences.
    • Place of Presentation
      Kyoto, Japan
    • Related Report
      2009 Final Research Report
  • [Presentation] Rohon-Beardニューロンの異常による逃避運動の欠如2009

    • Author(s)
      平田普三
    • Organizer
      2009 NIG Zebrafish Meeting.
    • Place of Presentation
      国立遺伝学研究所(三島)
    • Related Report
      2009 Final Research Report
  • [Presentation] 発生期のグリシン作動性シナプスの形成2009

    • Author(s)
      平田普三
    • Organizer
      日本発生生物学会秋期シンポジゥム
    • Place of Presentation
      東レ総合研修センター
    • Related Report
      2009 Annual Research Report
  • [Presentation] 感覚ニューロンの障害と細胞死2009

    • Author(s)
      平田普三, 他
    • Organizer
      第32回日本神経科学大会
    • Place of Presentation
      名古屋国際会議場
    • Related Report
      2009 Annual Research Report
  • [Presentation] Characterization of zebrafish motility mutant defective in volt age-gated sodium channel.2009

    • Author(s)
      平田普三, 他
    • Organizer
      The 36th International Congress of Physiological Sciences
    • Place of Presentation
      京都国際会議場
    • Related Report
      2009 Annual Research Report
  • [Presentation] Rohon-Beardニューロンの異常による逃避運動の欠如2009

    • Author(s)
      平田普三
    • Organizer
      2009 NIG Zebrafish Meeting
    • Place of Presentation
      国立遺伝学研究所
    • Related Report
      2008 Annual Research Report
  • [Presentation] Zebrafish locomotion and swimming: contribution of fast and slow muscle.2008

    • Author(s)
      Hiromi Hirata.
    • Organizer
      MCDB Neuro Group Meeting.
    • Place of Presentation
      University of Michigan, USA
    • Year and Date
      2008-06-30
    • Related Report
      2009 Final Research Report
  • [Presentation] Zebrafish ryanodine receptor mutants show slow swimming and provide a model of Multi-minicore disease.2008

    • Author(s)
      Hiromi Hirata, Takaki Watanabe, Jun Hatakeyama, Shawn M. Sprague, Louis Saint-Amant, Ayako Nagashima, Wilson W. Cui, Weibin Zhou, John Y. Kuwada.
    • Organizer
      The 8th International Conference on Zebrafish Development and Genetics.
    • Place of Presentation
      Madison, Wisconsin, USA.
    • Related Report
      2009 Final Research Report
  • [Presentation] サカナの異常から見えてくる人の病気2008

    • Author(s)
      平田普三
    • Organizer
      第41回日本発生生物学会(ISDB共催)市民公開講座「発生生物学から見た生物の世界」
    • Place of Presentation
      徳島県郷土文化会館(徳島)
    • Related Report
      2009 Final Research Report
  • [Presentation] ゼブラフィッシュ触刺激応答異常変異体の解析2008

    • Author(s)
      中野由梨, 他
    • Organizer
      第14回小型魚類研究会
    • Place of Presentation
      岡崎カンファレンスセンター
    • Related Report
      2008 Annual Research Report
  • [Presentation] ゼブラフィッシュの運動異常変異体の解析2008

    • Author(s)
      中野由梨, 他
    • Organizer
      第31回日本神経科学大会
    • Place of Presentation
      東京国際フォーラム
    • Related Report
      2008 Annual Research Report
  • [Presentation] Zebrafish ryanodine receptor mutants show slow swimming and provide amodel of Multi-minicore disease.2008

    • Author(s)
      平田普三, 他
    • Organizer
      The 8th International Conference on Zebrafish Development and Genetics
    • Place of Presentation
      Madison, Wisconsin, USA
    • Related Report
      2008 Annual Research Report
  • [Presentation] サカナの異常から見えてくる人の病気2008

    • Author(s)
      平田普三
    • Organizer
      第41回日本発生生物学会市民公開講座「発生生物学から見た生物の世界」
    • Place of Presentation
      徳島県郷土文化会館
    • Related Report
      2008 Annual Research Report
  • [Book] 細胞工学(ゼブラフィッシュの運動と行動, vol.27, 11月号)2008

    • Author(s)
      平田普三
    • Related Report
      2009 Final Research Report
  • [Book] 実験医学(硬骨魚類の逃避運動制御メカニズム, vol.26, No.12)2008

    • Author(s)
      平田普三、小橋常彦、小田洋一
    • Related Report
      2009 Final Research Report
  • [Book] ブレインサイエンス・レビュー(ゼブラフィッシュを用いた運動制御の遺伝学的解析)2008

    • Author(s)
      平田普三
    • Related Report
      2009 Final Research Report
  • [Book] ブレインサイエンスレビュー20082008

    • Author(s)
      平田普三
    • Publisher
      ゼブラフィッシュを用いた運動制御の遺伝学的解析
    • Related Report
      2008 Annual Research Report

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Published: 2008-04-01   Modified: 2016-04-21  

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