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Axonal degeneration of rat culured nerve introduced mutant aprata in cDNA

Research Project

Project/Area Number 15591119
Research Category

Grant-in-Aid for Scientific Research (C)

Allocation TypeSingle-year Grants
Section一般
Research Field Pediatrics
Research InstitutionSapporo Medical University

Principal Investigator

TACHI Nobutada  Sapporo Medical University, School of Health Sciences, Assistant Professor, 保健医療学部, 助教授 (80136944)

Co-Investigator(Kenkyū-buntansha) YAMASITA Toshiharu  Sappro Medical University, School of Medicine, Assitant Professor, 医学部, 助教授 (50167706)
KOZUKA Naoki  Sappro Medical University, Schoold of Health Sciences, Assitant Professor, 保健医療学部, 助教授 (90225459)
Project Period (FY) 2003 – 2004
Project Status Completed (Fiscal Year 2004)
Budget Amount *help
¥3,800,000 (Direct Cost: ¥3,800,000)
Fiscal Year 2004: ¥1,100,000 (Direct Cost: ¥1,100,000)
Fiscal Year 2003: ¥2,700,000 (Direct Cost: ¥2,700,000)
Keywordsaprataxin / 689 ins T c DNA / cultured rat peripherl nerve / axonal degeneration / 変異aprataxin cDFNA導入 / 培養ラツト末鞘神経 / 軸策変性 / aprataxin cDNA / 689insT
Research Abstract

Koenig and we identified a responsible gene "aprataxin" for early-onset cerebellar ataxia with hypoalbuminemia(EOCA-HA) in Japan and cerebellar ataxia with ocular apraxia(AOA) in Portugal. The mutation of aprataxin gene in most EOCA-HA patients showed 689 insertion T(689 ins T) homozygously. First, we made a 689 ins T cDNA by site-directed mutagenesis bases on PCR. Second, we made a constructed adeno virus vector contained both wild and 689 ins T aprataxins. Third, we made a cultured rat peripheral nerve using dorsal root of fetal rat. Those cultured nerves were infected by constructed adeno virus vectors, Myelination and axonal sprouting of cultured nerves infected constructed adeno virus vector were observed by immunohistochenistory using anti-Po antibody and electron microscopy. No differences of myelination and axonal changes were observed in cultured nerves infected constructed adeno virus vector containing both a wild and 689 ins T aprataxin gene.
Further more, transgenic mouse containing 689 ins T should be obtained. Matured nerve of this mouse should be analyzed.

Report

(3 results)
  • 2004 Annual Research Report   Final Research Report Summary
  • 2003 Annual Research Report
  • Research Products

    (19 results)

All 2005 2003 Other

All Journal Article (15 results) Publications (4 results)

  • [Journal Article] 筋緊張性ジストロフィー3塩基反復配列(CTG repeat)と数と筋力との関係2005

    • Author(s)
      菊池 真, 舘 延忠, 小塚直樹 他
    • Journal Title

      総合リハ 33

      Pages: 167-173

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] A new mutation of IGHMBP2 gene in spinal muscular atrophy with2005

    • Author(s)
      Tachi N, Kikuchi S, Kozuka N
    • Journal Title

      Pediatr Neurol 32

      Pages: 288-290

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Study of correlation between CTG repeat lengths and muscle power in myotonic dystrophy patients2005

    • Author(s)
      Kikuchi S, Tachi N, Kozuka N et al.
    • Journal Title

      Sougou Rhiha 33

      Pages: 167-173

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] A new mutation of IGHMBP2 In spinal muscular atrophy with respiratory disgtress2005

    • Author(s)
      Tachi N, Kikuchi S, Kozuka N, Nogami A.
    • Journal Title

      Pediatr Neural 32

      Pages: 288-290

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] 筋緊張性ジストロフィー症における3塩基反復配列(CTGリピート)数と2005

    • Author(s)
      菊池 真, 舘 延忠, 小塚直樹 他
    • Journal Title

      総合リハ 33

      Pages: 167-173

    • Related Report
      2004 Annual Research Report
  • [Journal Article] A new mutation of IGHMBP2 gene in spinal muscular atrophy with2005

    • Author(s)
      Tachi N, Kikuchi S, Kozuka N, et al.
    • Journal Title

      Pediatr Neurol 32

      Pages: 288-290

    • Related Report
      2004 Annual Research Report
  • [Journal Article] 父親由来の筋緊張性ジストロフィーにおける研究2003

    • Author(s)
      菊池 真, 舘 延忠, 小塚直樹 他
    • Journal Title

      札幌医科大学保健医療学部紀要 6

      Pages: 43-48

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] 筋緊張性ジストロフィーCTG repeatのNon-RIサザン法による解析2003

    • Author(s)
      小塚直樹, 舘 延忠, 菊池 真, 他
    • Journal Title

      脳と発達 35

      Pages: 380-387

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] 「特異な脳症後てんかんの一群」と考えられた1女児例2003

    • Author(s)
      鈴木将史, 舘 延忠, 桜井のどか 他
    • Journal Title

      臨小医 51

      Pages: 115-120

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Study of myotonic dystrophy inherited from fathers2003

    • Author(s)
      Kikuchi S, Tachi N, Kozuka N et al.
    • Journal Title

      School of Healt Sciences, Sapporo University Kiyou 6

      Pages: 43-48

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Analysis of CTG repeat in Myotonic dystrophy using Non-RI southern2003

    • Author(s)
      Kozuka N, Tachi N, Kikuchi S et al.
    • Journal Title

      Nou to Hatatsu 35

      Pages: 380-387

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] A female case of specific epilepsy after encephalitis2003

    • Author(s)
      Suzuki S, Tachi N, Sakurai N et al.
    • Journal Title

      Rinsyou Syouni Igaku 51

      Pages: 115-120

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] 父親由来将に筋緊張性ジストロフィー症における研究2003

    • Author(s)
      菊池 真, 舘 延忠, 小塚直樹 他
    • Journal Title

      札幌医科大学保健医療学部紀要 6

      Pages: 43-48

    • Related Report
      2004 Annual Research Report
  • [Journal Article] 筋緊張性ジストロフィー症CTGリピートのNon-RIサザン法による解析2003

    • Author(s)
      小塚直樹, 舘 延忠, 菊池 真 他
    • Journal Title

      脳と発達 35

      Pages: 380-387

    • Related Report
      2004 Annual Research Report
  • [Journal Article] 「特異な脳炎脳症後てんかん一群」と考えられた1女児例2003

    • Author(s)
      鈴木将史, 舘 延忠, 桜井のどか 他
    • Journal Title

      臨小医 51

      Pages: 115-120

    • Related Report
      2004 Annual Research Report
  • [Publications] 菊池真, 舘 延忠ら: "父親由来小児筋緊張性ジストロフィーに関する研究"札幌医科大学保健医療学部紀要. 6. 43-48 (2003)

    • Related Report
      2003 Annual Research Report
  • [Publications] 小塚直樹, 舘 延忠ら: "黒龍江省小児脳性麻痺防防治療中心における肢体不自由療育の現状"札幌医科大学保健医療学部紀要. 6. 87-91 (2003)

    • Related Report
      2003 Annual Research Report
  • [Publications] 小塚直樹, 舘 延忠ら: "筋強直性ジストロフィー症CTGリピートのNon-RI PCRサザン法に"脳と発達. 35. 380-387 (2003)

    • Related Report
      2003 Annual Research Report
  • [Publications] Uchida E, Tachi N et al.: "Molecular genetic analysis of X-linked recessive myotubular myopathy"Bulletin of School of Health Sciences, Sappro Medical University. 6. 49-58 (2003)

    • Related Report
      2003 Annual Research Report

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Published: 2003-04-01   Modified: 2016-04-21  

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