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A study on pahtophysiology and non-invasive treatment of dystonia

Research Project

Project/Area Number 15390274
Research Category

Grant-in-Aid for Scientific Research (B)

Allocation TypeSingle-year Grants
Section一般
Research Field Neurology
Research InstitutionThe University of Tokushima

Principal Investigator

KAJI Ryuji  The University of Tokushima, Graduate School, Institute of Health Bioscience, Professor, 大学院・ヘルスバイオサイエンス研究部, 教授 (00214304)

Co-Investigator(Kenkyū-buntansha) IZUMI Yuishin  The University of Tokushima, Graduate School, Institute of Health Bioscience, 大学院・ヘルスバイオサイエンス研究部, 講師 (10335812)
SAKAMOTO Takashi  The University of Tokushima, Graduate School, Institute of Health Bioscience, 大学院・ヘルスバイオサイエンス研究部, 助手 (20343303)
HARADA Masafumi  The University of Tokushima, Graduate School, Institute of Health Bioscience, 大学院・ヘルスバイオサイエンス研究部, 教授 (20228654)
NODERA Hiroyuki  The University of Tokushima, Graduate School, Institute of Health Bioscience, 大学院・ヘルスバイオサイエンス研究部, 講師 (40363147)
Project Period (FY) 2003 – 2004
Project Status Completed (Fiscal Year 2004)
Budget Amount *help
¥15,000,000 (Direct Cost: ¥15,000,000)
Fiscal Year 2004: ¥2,800,000 (Direct Cost: ¥2,800,000)
Fiscal Year 2003: ¥12,200,000 (Direct Cost: ¥12,200,000)
Keywordsdystonia / magnetic stimulation / X-linked dystonia-parkinsonism / persistent Na channel / PSTH / 前運動野 / 補足運動野 / silent period / 皮質-皮質間制御 / SPECT / プライミング
Research Abstract

Dystonia is a syndrome of sustained muscle contractions frequently causing repetitive or twisting movement or abnormal posture. It is a common disorder with its incidence being approximately one fifth of that of Parkinson's disease. Writer's cramp is a focal dystonia, affecting only writing in its onset. This task-specificity provides a unique opportunity of investigating the mechanism of normal motor control for performing a specific task. It is also known notorious because of the resistance to treatment. We tried to treat these patients with writer's cramp by decreasing the excitability of the premotor cortex, which is known to be hyperactive in previous PET studies. For this, we used subthreshold low-frequency repetitive transcranial magnetic stimulation (rTMS), which inhibits the cortex. Using a computerized system for evaluating handwriting, we demonstrated significant improvement in handwriting and normalization of the abnormal cortical silent periods. Therefore the premotor cortex, one of the major output receptive areas from the basal ganglia, was shown hyperactive in dystonia, and its correction by rTMS may provide a useful method for treating these patients.
We on the other hand had a chance to examine the immunohistochemichal findings in 7 autopsied patients with X-linked recessive dystonia-parkinsonism. We found the early loss of striosomal neurons in the neostriatum is the pathological hallmark of the dystonic phase of this disease. Striosomal cells are inhibiting the nigrastriatal dopaminergic projections from substantia nigra pars compacta, and their loss should lead to uncontrolled release of dopamine. Dystonia may therefore be caused by excess dopamine, which disinhibits premotor and other cortical areas.

Report

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

    (26 results)

All 2005 2004 2003 Other

All Journal Article (19 results) Book (2 results) Publications (5 results)

  • [Journal Article] Subthreshold low-frequency repetitive transcranial magnetic stimulation over the premotor cortex modulates writer's cramp.2005

    • Author(s)
      Murase, N., J.C.Rothwell, R., Kaji.R.et al.
    • Journal Title

      Brain 128(Pt 1):

      Pages: 104-115

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Puffer fish poisoning, Guillain-Barre syndrome and persistent sodium channels2005

    • Author(s)
      Kaji, R., H.Nodera
    • Journal Title

      Ann Neurol 52

      Pages: 309-309

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Functional anatomy of the basal ganglia in X-linked recessive dystonia-park insonism2005

    • Author(s)
      Goto, S.,...R.Kaji
    • Journal Title

      Ann Neurol (in press)

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Puffer fish poisoning, Guillain-Barre syndrome and persistent sodium channels.2005

    • Author(s)
      Kaji, R., H.Nodera
    • Journal Title

      Ann Neurol 57(3)

      Pages: 309-309

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Subthreshold low-frequency repetitive transcranial magnetic stimulation over the premotor cortex modulates writer's cramp.2005

    • Author(s)
      Murase, N., J.C.Rothwell, R.Kaji, R.Urushihara, K.Nakamura, N.Murayama, T.Igasaki, M.Sakata-Igasaki, T.Mima, A.Ikeda, H.Shibasaki
    • Journal Title

      Brain 128(Pt1)

      Pages: 104-115

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Subthreshold low-frequency repetitive transcranial magnetic stimulation over the nremotor cortex modulates writer's cramn2005

    • Author(s)
      Murase, N., J.C.Rothwell, R., Kaji, R.et al.
    • Journal Title

      Brain 128(Pt 1):

      Pages: 104-15

    • Related Report
      2004 Annual Research Report
  • [Journal Article] Puffer fish poisoning, Guillain-Barre syndrome and persistent sodium channels2005

    • Author(s)
      Kaji.R., H.Nodera
    • Journal Title

      Ann Neurol 57

      Pages: 309-309

    • Related Report
      2004 Annual Research Report
  • [Journal Article] Functional anatomy of the basal ganglia in X-linked recessive dystonia-parkinsonism2005

    • Author(s)
      Goto, S.,...R.Kaji
    • Journal Title

      Ann Neurol (In press)

    • Related Report
      2004 Annual Research Report
  • [Journal Article] Sensory deficits in dystonia and their significance.2004

    • Author(s)
      Kaji R, Murase N, Urushihara R, Asanuma K
    • Journal Title

      Ady Neurol 94

      Pages: 11-17

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Sensory deficits in dystonia and their significance.2004

    • Author(s)
      Kaji, R., N.Murase, R.Urushihara, K.Asanuma
    • Journal Title

      Adv Neurol 94

      Pages: 11-17

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Epidemiology of primary dystonias in Japan : comparison with Western countries.2004

    • Author(s)
      Matsumoto, S., M.Nishimura, H.Shibasaki, R.Kaji
    • Journal Title

      Mov Disord 18(1)

      Pages: 94-100

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Nerve excitability properties in Charcot-Marie-Tooth disease type 1A2004

    • Author(s)
      Nodera, H., H.Bostock, S...Kaji, R
    • Journal Title

      Brain 127(Pt 1)

      Pages: 203-211

    • Related Report
      2004 Annual Research Report
  • [Journal Article] Nucling recruits Apaf-1/pro-caspase-9 complex for the induction of stress-induced apoptosis2004

    • Author(s)
      Sakai, T., L.Liu, X...Kaji, R..
    • Journal Title

      J Biol Chem 279(39)

      Pages: 41131-41140

    • Related Report
      2004 Annual Research Report
  • [Journal Article] Modulation of the onset age in primary dystonia by APOE genotype.2003

    • Author(s)
      Matsumoto S, Nishimura M, Sakamoto T, Asanuma K, Izumi Y, Shibasaki H, Kamatani N, Nakamura T, Kaji R
    • Journal Title

      Neurology 60

      Pages: 2003-2005

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] The epidemiology of the primary dystonia in Japan : comparison to the western countries.2003

    • Author(s)
      Matsumoto S, Nishimura M, Shibasaki H, Kaji R
    • Journal Title

      Movement Disorders 18

      Pages: 1196-1198

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Premovement gating of somatosensory evoked potentials after tibial nerve stimulation2003

    • Author(s)
      Asanuma, K., R.Urushihara, R.Kaji
    • Journal Title

      Neuroreport 14(3)

      Pages: 375-379

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Modulation of the onset age in primary dystonia by APOE genotype.2003

    • Author(s)
      Matsumoto, S., M.Nishimura, T.Sakamoto, K.Asanuma, Y.Izumi, H.Shibasaki, N.Kamatani, T.Nakamura, R.Kaji
    • Journal Title

      Neurology 60(12)

      Pages: 2003-2005

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Movement-related cortical potentials before jaw excursions in oromandibular dystonia.2003

    • Author(s)
      Yoshida, K., R.Kaji, N.Kohara, N.Murase, A.Ikeda, H.Shibasaki, T.Iizuka
    • Journal Title

      Mov Disord 18(10)

      Pages: 1196-1198

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Movement-related cortical potentials before jaw excursions in oromandibular dystonia2003

    • Author(s)
      Yoshida, K., R.Kaji, et al.
    • Journal Title

      Mov Disord 18(1)

      Pages: 94-100

    • Related Report
      2004 Annual Research Report
  • [Book] Magnetic Stimulation in Clinical Neurophysiology(Editors : Hallett, Chokroverty, Chap 10, pp.155-164)2005

    • Author(s)
      Kaji, R., N.Kohara
    • Total Pages
      457
    • Publisher
      Elsevier
    • Related Report
      2004 Annual Research Report
  • [Book] Chapter 28 Dystonia (in Movement Disorders)2003

    • Author(s)
      Kaji R
    • Publisher
      Elsevier
    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Publications] Asanuma K, Urushihara R, Nakamura K, Kitaoka K, Sei H, Morita Y, Shibasaki H, Kaji R: "Premovement gating of somatosensory evoked potentials after tibial nerve stimulation"Neuroreport. 14. 375-379 (2003)

    • Related Report
      2003 Annual Research Report
  • [Publications] Matsumoto S, Nishimura M, Sakamoto T, Asanuma K, Izumi Y, Shibasaki H, Kamatani N, Nakamura T, Kaji R: "Modulation of the onset age in primary dystonia by APOE genotype"Neurology. 60. 2003-2005 (2003)

    • Related Report
      2003 Annual Research Report
  • [Publications] Matsumoto S, Nishimura M, Shibasaki H, Kaji R: "The epidemiology of the primary dystonia in Japan : comparison to the western countries"Movement Disorders. 18. 1196-1198 (2003)

    • Related Report
      2003 Annual Research Report
  • [Publications] Kaji R, Murase N, Urushihara R, Asanuma K: "Sensory deficits in dystonia and their significance"Adv Neurol. 94. 11-17 (2004)

    • Related Report
      2003 Annual Research Report
  • [Publications] Kaji R: "Chapter 28. Dystonia(in Movement Disorders)"Elsevier. 451-461 (2003)

    • Related Report
      2003 Annual Research Report

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

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