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NEUROMUSCULAR DISEASES CAUSED BY ABANORMAL EXCITATION-CONTRACTION COUPLING

Research Project

Project/Area Number 08670742
Research Category

Grant-in-Aid for Scientific Research (C)

Allocation TypeSingle-year Grants
Section一般
Research Field Neurology
Research InstitutionOKINAKA MEMORIAL INSTITUTE FOR MEDICAL RESEARCH(OMIMR)

Principal Investigator

TAKAGI Akio  OMIMR,CHIEF RESEARCHER,OKINAKA MEMORIAL INSSTITUTE FOR MEDICAL RESEARCH, 研究員 (20010142)

Co-Investigator(Kenkyū-buntansha) 上坂 義和  (財)冲中記念成人病研究所, 研究員
IDA Masayoshi  OMIMR,RESEARCHER,OKINAKA MEMORIAL INSTITUTE FOR MEDICAL RESEARCH, 研究員
WATANABE Tomoji  OMIMR,RESEARCHER,OKINAKA MEMORIAL INSTITUTE FOR MEDICAL RESEARCH, 研究員
KOJIMA Susumu  OMIMR,RESEARCHER,OKINAKA MEMORILA INSTITUTE FOR MEDICAL RESEARCH, 研究員
NAKASE Hirohumi  OMIMR,RESEARCHER,OKINAKA MEMORIAL INSTITUTE FOR MEDICAL RESEARCH, 研究員 (80155738)
UESAKA Kazuyoshi  OMIMR,RESEARCHER,OKINAKA MEMORIAL INSTITUTE FOR MEDICAL RESEARCH
Project Period (FY) 1996 – 1997
Project Status Completed (Fiscal Year 1997)
Budget Amount *help
¥2,300,000 (Direct Cost: ¥2,300,000)
Fiscal Year 1997: ¥500,000 (Direct Cost: ¥500,000)
Fiscal Year 1996: ¥1,800,000 (Direct Cost: ¥1,800,000)
KeywordsMALIGNANT HYPERTHERMIA / CENTRAL CORE DISEASE / RYANODINE RECEPTOR / CALCIUM ION / CAFFEINE / MUTATION / 点変異 / 筋小胞体
Research Abstract

Malignanthyperthermia(MH)is a serious complication of general anesthesia, presenting high fever, tachyarrhithmia, acidosis and myoglobinuria. MH is a heterogeneous disorder, but half of MH cases are associated with various mutation of ryanodine receptor gene of the skeletal muscle(ryr1). Central core disease(CCD), a form of congenital myopathy, frequently developed MH.In some of CCD cases, mutation of ryr1 is also reported. In the first part, we studied pharmacological aspects of caffeine to supplement the caffeine contracture test.
Using theskinned muscle fiber preparation, influence of caffeine on the muscle fiber was compared between mdx and control mouse. Tension generataion was depressed by 30mM caffeine in high calcium ion(Ca)such as-pCa4.5. In type 2 muscle fibers of mdx, increase of tension by caffeine was always more marked than in controls. On the other hand, influence of caffeine ontype 1 fiber was the same between mdx and control. Reasons for this difference between type 1 and 2 is not clear yet.
In familial patients with CCD,who had recovered from episode of MH,contracture by caffeine was markedly exaggerated. In a case of the postoperative MH,caffeine response was abnormal only in the type 2 fiber. In twocaseswith suspected MH,results of caffeine contracture were equivocal. On thefamily with CCD,complicated with MH,all the known mutation of ryr1 was not detected.

Report

(3 results)
  • 1997 Annual Research Report   Final Research Report Summary
  • 1996 Annual Research Report
  • Research Products

    (19 results)

All Other

All Publications (19 results)

  • [Publications] 高木 昭夫: "悪性高熱・セントラルコア病の分子遺伝学" 日本臨床. 55(12). 3307-3314 (1997)

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      1997 Final Research Report Summary
  • [Publications] 高木 昭夫: "筋ジストロフィー研究最前線" 診断と治療. 85(8). 1204-1209 (1997)

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      1997 Final Research Report Summary
  • [Publications] 井田 雅祥: "筋緊張性ジストロフィーと痴呆" Dementia Japan. 11・3. 313-316 (1997)

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      1997 Final Research Report Summary
  • [Publications] 上坂 義和: "Rimmed vauolesや細胞質封入体を伴う常染色体優性遠位型ミオパチー" 臨床神経学. 37・1. 1-6 (1997)

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      1997 Final Research Report Summary
  • [Publications] 高木 昭夫: "悪性高熱-病態と遺伝子異常" 医学のあゆみ. 179・5. 354-358 (1996)

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      1997 Final Research Report Summary
  • [Publications] 高木 昭夫: "悪性高熱、最新内科学大系71" 中山書店, 8 (1996)

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      1997 Final Research Report Summary
  • [Publications] Takagi A: "malignant Hyperthermia" Springer-Verlag(Tokyo), 67-73 (1996)

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      1997 Final Research Report Summary
  • [Publications] Takagi A et al.: "Caffeine contracture of the skinned fiber in malignant hyperthermia." in "Malignant Hyperthermia"(edit by Morio et al.). Springer-Verlag(Tokyo). 67-73 (1996)

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      1997 Final Research Report Summary
  • [Publications] 高木 昭夫: "悪性高熱/セントラルコア病の分子遺伝学" 日本臨床. 55(12). 3307-3314 (1997)

    • Related Report
      1997 Annual Research Report
  • [Publications] 高木 昭夫: "筋ジストロフィー研究最前線" 診断と治療. 85・8. 1204-1209 (1997)

    • Related Report
      1997 Annual Research Report
  • [Publications] 井田 雅祥, 高木 昭夫: "筋緊張性ジストロフィーと痴呆" Dementia Japan. 11・3. 313-316 (1997)

    • Related Report
      1997 Annual Research Report
  • [Publications] 上坂 義和ほか: "Rimmed vacuolesや細胞質封入体を伴う常染色体優性遠位型シオパチ" 臨床神経学. 37.1. 1-6 (1997)

    • Related Report
      1997 Annual Research Report
  • [Publications] 高木 昭夫: "悪性高熱-病態と遺伝異常" 医学のあゆみ. 179・5. 354-358 (1996)

    • Related Report
      1997 Annual Research Report
  • [Publications] Takagi A et al: "Caffeine contracture of the skinnedfiber in malignant hyper thermia" ″Malignant Hyperthermia, Springer-Verlag(Tokyo). 67-73 (1996)

    • Related Report
      1997 Annual Research Report
  • [Publications] 高木 昭夫: "悪性高熱、最新内科学大系71" 中山書店, 8 (1996)

    • Related Report
      1997 Annual Research Report
  • [Publications] 高木昭夫: "悪性高熱-病態と遺伝子異常" 医学のあゆみ. 179(5). 354- 368 (1996)

    • Related Report
      1996 Annual Research Report
  • [Publications] 高木昭夫: "リアノジン受容体" 内科. 77(6). 1309-1310 (1996)

    • Related Report
      1996 Annual Research Report
  • [Publications] Takagi A: "Caffeine contracture of the okinned muscle fiber" Malignant Hyper the rmia(Springer Verlag). 67-73 (1996)

    • Related Report
      1996 Annual Research Report
  • [Publications] 高木昭夫: "最新内科学大系-71(ミオパチー,分担)" 中山書店, 289-296 (1996)

    • Related Report
      1996 Annual Research Report

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Published: 1996-04-01   Modified: 2016-04-21  

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