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2007 Fiscal Year Final Research Report Summary

The molecular genetic analyses of acute disseminated encephalomyelitus

Research Project

Project/Area Number 17591095
Research Category

Grant-in-Aid for Scientific Research (C)

Allocation TypeSingle-year Grants
Section一般
Research Field Pediatrics
Research InstitutionKyushu University

Principal Investigator

TORISU Hiroyuki  Kyushu University, Kyushu University Hospital, Assistant Professor (10398076)

Co-Investigator(Kenkyū-buntansha) KIRA Ryutaro  Kyushu University, Kyushu University Hospital, Lecturer (70304805)
Project Period (FY) 2005 – 2007
KeywordsAcute disseminated encephalomyelitis / Acute encephalitis / Multiple sclerosis / Host factor / Gene polymorphism
Research Abstract

1. Case accumulation
We asked 78 departments of neurology in university hospitals and 90 facilities where the councilors of the Japanese society of child neurology worked, to participate in the acute disseminated encephalomyelitis (ADEM) research. 15 facilities participated and this study was approved by the gene ethics committee in each facilities. Clinical material of the ADEM patient was collected after it approved, and DNA had been extracted from the collection blood.
2. Clinical features of ADEM
Thirty-one cases (male 15, female 16) were accumulated by the end of 2007. The median age of onset was seven years old (from 11 months to 51 years old).Twenty-three cases were infection related ADEM. Four cases were related to vaccination. Seventy four percentages of the patients had fever. First symptoms were gait disturbance (58%) and impaired consciousness (45%). WBCs and CRP were mildly elevated. Pleocytosis was found in cerebrospinal fluid (average 92/μl). The lesions were found by brain MR images in subcortical white matter (57%), periventricular white matter (63%), brainstem (43%), cerebellum (33%), and spinal cord (27%).
3. Gene analysis of ADEM
We analyzed ADEM patients' genotypes of the disease susceptibility genes of multiple sclerosis (MS). In the correlational analysis of the CTLA4 gene and the VDR gene, a significant difference was not found between the patient group and the control group. In addition, we investigated the MS susceptible single nucleotide polymorphisms (SNPs) in the HLA-DRA gene, the IL2RA gene, and the IL7R gene, shown by the genome wide research in a large-scale group of MS. However, a significant difference between the genotypic frequency and the allele frequency of the reported SNPs was not found though the correlational analysis.

  • Research Products

    (55 results)

All 2009 2008 2007 2006 2005 Other

All Journal Article (42 results) (of which Peer Reviewed: 19 results) Presentation (12 results) Book (1 results)

  • [Journal Article] Interleukin-lbeta enhances susceptibility to hyperthermia-induced seizures in developing rats.2009

    • Author(s)
      M. Fukuda
    • Journal Title

      Seizure (in press)

    • Description
      「研究成果報告書概要(欧文)」より
  • [Journal Article] Fulminant sepsis/meningitis due to Haemophilus influenzae in a protein C-deficient heterozygote treated with activated protein C therapy.2009

    • Author(s)
      M. Ishimura
    • Journal Title

      European Journal of Pediatrics (in press)

    • Description
      「研究成果報告書概要(欧文)」より
  • [Journal Article] Association of toll-like receptor 3 gene polymorphism with subacute sclerosing panencephalitis.2008

    • Author(s)
      Yoshito Ishizaki
    • Journal Title

      Journal of Neurovirology 18

      Pages: 1-6

    • Description
      「研究成果報告書概要(和文)」より
    • Peer Reviewed
  • [Journal Article] Epstein-Barr virus load in cerebrospinal fluid of patients with chronic active Epstein-Barr virus infection.2008

    • Author(s)
      Shoichi Ohga
    • Journal Title

      The Pediatric Infectious Disease Journal 27

      Pages: 1027-30

    • Description
      「研究成果報告書概要(和文)」より
    • Peer Reviewed
  • [Journal Article] Epstein-Barr virus-associated meningoencephalomyelitis : intrathecal reactivation of the virus in an immunocompetent child.2008

    • Author(s)
      Masafumi Sanefuji
    • Journal Title

      Journal of Child Neurology 23

      Pages: 1072-7

    • Description
      「研究成果報告書概要(和文)」より
    • Peer Reviewed
  • [Journal Article] Sjogren's syndrome-associated meningoencephalomyelitis : cerebrospinal fluid cytokine levels and therapeutic utility of tacrolimus.2008

    • Author(s)
      Takayuki Hoshina
    • Journal Title

      Journal of the Neurological Sciences 267

      Pages: 182-186

    • Description
      「研究成果報告書概要(和文)」より
    • Peer Reviewed
  • [Journal Article] Analysis of MxA, IL-4, and IRF-1 genes in Filipino patients with subacute sclerosing panencephalitis.2008

    • Author(s)
      Judy R Pipo-Deveza
    • Journal Title

      Neuropediatrics 37(4)

      Pages: 222-8

    • Description
      「研究成果報告書概要(和文)」より
    • Peer Reviewed
  • [Journal Article] 小児急性散在性脳脊髄炎(ADEM)小児例の臨床像と疫学.2008

    • Author(s)
      吉良龍太郎
    • Journal Title

      Neuroinfection 13

      Pages: 64-67

    • Description
      「研究成果報告書概要(和文)」より
  • [Journal Article] Association of toll-like receptor 3 gene polymorphism with subacute sclerosing panencephalitis.2008

    • Author(s)
      Y. Ishizaki
    • Journal Title

      Journal of Neurovirology 18

      Pages: 1-6

    • Description
      「研究成果報告書概要(欧文)」より
  • [Journal Article] Epstein-Barr virus load in cerebrospinal fluid of patients with chronic active Epstein-Barr virus infection.2008

    • Author(s)
      S. Ohga
    • Journal Title

      The Pediatric Infectious Disease Journal 27

      Pages: 1027-1030

    • Description
      「研究成果報告書概要(欧文)」より
  • [Journal Article] Epstein-Barr virus-associated meningoencephalomyelitis: intrathecal reactivation of the virus in an immunocompetent child.2008

    • Author(s)
      M. Sanefuji
    • Journal Title

      Journal of Child Neurology 23

      Pages: 1072-1077

    • Description
      「研究成果報告書概要(欧文)」より
  • [Journal Article] Sjogren's syndrome-associated meningoencephalomyelitis: cerebrospinal fluid cytokine levels and therapeutic utility of tacrolimus.2008

    • Author(s)
      T. Hoshina
    • Journal Title

      Journal of the Neurological Sciences 267

      Pages: 182-186

    • Description
      「研究成果報告書概要(欧文)」より
  • [Journal Article] Analysis of MxA, IL-4, and IRF-1 genes in Filipino patients with subacute sclerosing panencephalitis.2008

    • Author(s)
      JR. Pipo-Deveza
    • Journal Title

      Neuropediatrics 37(4)

      Pages: 222-228

    • Description
      「研究成果報告書概要(欧文)」より
  • [Journal Article] Interleukin-10 is associated with resistance to febrile seizures- Genetic association and experimental animal studies.2008

    • Author(s)
      Y. Ishizaki
    • Journal Title

      Epilepsia (in press)

    • Description
      「研究成果報告書概要(欧文)」より
  • [Journal Article] Moyamoya syndrome in a splenectomized patient with beta-thalassemia intermedia.2008

    • Author(s)
      M. Sanefuji
    • Journal Title

      Journal of Child Neurology 21

      Pages: 75-76

    • Description
      「研究成果報告書概要(欧文)」より
  • [Journal Article] Serum levels of matrix metalloproteinase-9 and tissue inhibitors of metalloproteinases 1 in subacute sclerosing panencephalitis.2007

    • Author(s)
      Takashi Ichiyama
    • Journal Title

      Journal of the Neurological Sciences 252(1)

      Pages: 45-8

    • Description
      「研究成果報告書概要(和文)」より
    • Peer Reviewed
  • [Journal Article] The relationship between retrieval success and task performance during the recognition of meaningless shapes : an event-related near-infrared spectroscopy study.2007

    • Author(s)
      Masafumi Sanefuji
    • Journal Title

      Neuroscience Research 59

      Pages: 191-198

    • Description
      「研究成果報告書概要(和文)」より
    • Peer Reviewed
  • [Journal Article] 亜急性硬化性全脳炎(SSPE)宿主側の遺伝要因2007

    • Author(s)
      楠原浩一
    • Journal Title

      日本臨床 65

      Pages: 1467-1474

    • Description
      「研究成果報告書概要(和文)」より
  • [Journal Article] 進行性多巣性白質脳症2007

    • Author(s)
      鳥巣浩幸
    • Journal Title

      小児内科 39

      Pages: 579-580

    • Description
      「研究成果報告書概要(和文)」より
  • [Journal Article] Serum levels of matrix metalloproteinase-9 and tissue inhibitors of metalloproteinases 1 in subacute sclerosing panencephalitis.2007

    • Author(s)
      T. Ichiyama
    • Journal Title

      Journal of the Neurological Sciences 252(1)

      Pages: 45-48

    • Description
      「研究成果報告書概要(欧文)」より
  • [Journal Article] The relationship between retrieval success and task performance during the recognition of meaningless shapes: an event-related near-infrared spectroscopy study.2007

    • Author(s)
      M. Sanefuji
    • Journal Title

      Neuroscience Research 59

      Pages: 191-198

    • Description
      「研究成果報告書概要(欧文)」より
  • [Journal Article] CSF cytokine and chemokine profiles in acute disseminated encephalomyelitis.2006

    • Author(s)
      Takaaki Ishizu
    • Journal Title

      Journal of Neuroimmunology 175(1-2)

      Pages: 52-8

    • Description
      「研究成果報告書概要(和文)」より
    • Peer Reviewed
  • [Journal Article] Gene expression profiles in peripheral blood mononuclear cells from patients with subacute sclerosing panencephalitis using oligonucleotide microarrays.2006

    • Author(s)
      Megumi Takemoto
    • Journal Title

      Journal of Neurovirology 11(3)

      Pages: 299-305

    • Description
      「研究成果報告書概要(和文)」より
    • Peer Reviewed
  • [Journal Article] A novel R275X mutation of the SLC25A15 gene in a Japanese patient with the HHH Syndrome2006

    • Author(s)
      Hiroyuki Torisu
    • Journal Title

      Brain & development 28

      Pages: 332-335

    • Description
      「研究成果報告書概要(和文)」より
    • Peer Reviewed
  • [Journal Article] Persistent diffusion abnormalities in the brain stem of three children with mitochondrial diseases.2006

    • Author(s)
      Yasunari Sakai
    • Journal Title

      American Journal of Neuroradiology 27

      Pages: 1924-1926

    • Description
      「研究成果報告書概要(和文)」より
    • Peer Reviewed
  • [Journal Article] Benign convulsion with mild gastroenteritis and benign familial infantile seizure.2006

    • Author(s)
      Yasunari Sakai
    • Journal Title

      Epilepsy Research 68

      Pages: 269-271

    • Description
      「研究成果報告書概要(和文)」より
    • Peer Reviewed
  • [Journal Article] Moyamoya syndrome in a splenectomized patient with beta-thalassemia in termedia.2006

    • Author(s)
      Masafumi Sanefuji
    • Journal Title

      Journal of Child Neurology 21

      Pages: 75-76

    • Description
      「研究成果報告書概要(和文)」より
    • Peer Reviewed
  • [Journal Article] Novel NPC1 mutations and characterization of the concomitant acid sphingomyelinase deficiency.2006

    • Author(s)
      Hiroaki Tamura
    • Journal Title

      Molecular Genetics and Metabolism 87

      Pages: 113-121

    • Description
      「研究成果報告書概要(和文)」より
    • Peer Reviewed
  • [Journal Article] Magnetic resonance studies of brain lesions in patients with Kawasaki disease.2006

    • Author(s)
      Jun Muneuchi
    • Journal Title

      Brain & development 28

      Pages: 30-33

    • Description
      「研究成果報告書概要(和文)」より
    • Peer Reviewed
  • [Journal Article] CSF cytokine and chemokine profiles in acute disseminated encephalomyelitis.2006

    • Author(s)
      T. Ishizu
    • Journal Title

      Journal of Neuroimmunology 175(1-2)

      Pages: 52-58

    • Description
      「研究成果報告書概要(欧文)」より
  • [Journal Article] Gene expression profiles in peripheral blood mononuclear cells from patients with subacute sclerosing Panencephalitis using oligonucleotide microarrays.2006

    • Author(s)
      M Takemoto
    • Journal Title

      Journal of Neurovirology 11(3)

      Pages: 299-305

    • Description
      「研究成果報告書概要(欧文)」より
  • [Journal Article] A novel R275X mutation of the SLC25A15 gene in a Japanese patient with the HHH svndrome2006

    • Author(s)
      H. Torisu
    • Journal Title

      Brain & development 28

      Pages: 332-335

    • Description
      「研究成果報告書概要(欧文)」より
  • [Journal Article] Persistent diffusion abnormalities in the brain stem of three children with mitochondrial diseases.2006

    • Author(s)
      Y. Sakai
    • Journal Title

      American Journal of Neuroradiology 27

      Pages: 1924-26

    • Description
      「研究成果報告書概要(欧文)」より
  • [Journal Article] Benign convulsion with mild gastroenteritis and benign familial infantile seizure.2006

    • Author(s)
      Y. Sakai
    • Journal Title

      Epilepsy Research 68

      Pages: 269-271

    • Description
      「研究成果報告書概要(欧文)」より
  • [Journal Article] Novel NPC1 mutations and characterization of the concomitant acid sphingomyelinase deficiency.2006

    • Author(s)
      H. Tamura
    • Journal Title

      Molecular Genetics and Metabolism 87

      Pages: 113-121

    • Description
      「研究成果報告書概要(欧文)」より
  • [Journal Article] Magnetic resonance studies of brain lesions in patients with Kawasaki disease.2006

    • Author(s)
      J. Muneuchi
    • Journal Title

      Brain & development 28

      Pages: 30-33

    • Description
      「研究成果報告書概要(欧文)」より
  • [Journal Article] Genetic susceptibility to simple febrile seizures : interleukin-lbeta promoter polymorphisms are associated with sporadic cases.2005

    • Author(s)
      Ryutaro Kira
    • Journal Title

      Neuroscience Letter 384(3)

      Pages: 239-244

    • Description
      「研究成果報告書概要(和文)」より
    • Peer Reviewed
  • [Journal Article] Genetic susceptibility to simple febrile seizures: interleukin-lbeta promoter polymorphisms are associated with snoradic cases.2005

    • Author(s)
      R. Kira
    • Journal Title

      Neuroscience Letter 384(3)

      Pages: 239-244

    • Description
      「研究成果報告書概要(欧文)」より
  • [Journal Article] Interleukin-10 is associated with resistance to febrile seizures : Genetic association and experimental animal studies.

    • Author(s)
      Yoshito Ishizaki
    • Journal Title

      Epilepsia (in press) (印刷中)

    • Description
      「研究成果報告書概要(和文)」より
    • Peer Reviewed
  • [Journal Article] Interleukin-lbeta enhances susceptibility to hyperthermia-induced seizures in developing rats.

    • Author(s)
      Mitsumasa Fukuda
    • Journal Title

      Seizure (in press) (印刷中)

    • Description
      「研究成果報告書概要(和文)」より
    • Peer Reviewed
  • [Journal Article] Fulminant sepsis/meningitis due to Haemophilus influenzae in a protein C-deficient heterozygote treated with activated protein C therapy.

    • Author(s)
      Masataka Ishimura
    • Journal Title

      European Journal of Pediatrics (in press) (印刷中)

    • Description
      「研究成果報告書概要(和文)」より
    • Peer Reviewed
  • [Journal Article] Hyperornithinemia-hyperammonemia-homocitrullinuria症候群(HHH症候群)

    • Author(s)
      鳥巣浩幸
    • Journal Title

      小児科臨床(in press) (印刷中)

    • Description
      「研究成果報告書概要(和文)」より
  • [Presentation] 急性散在性脳脊髄炎における宿主遺伝素因の検討-CTLA4遺伝子の解析-2007

    • Author(s)
      鳥巣浩幸
    • Organizer
      第49回日本小児神経学会
    • Place of Presentation
      大阪
    • Year and Date
      20070705-20070707
    • Description
      「研究成果報告書概要(和文)」より
  • [Presentation] ADEM小児例の臨床像と疫学2007

    • Author(s)
      吉良龍太郎
    • Organizer
      日本神経感染症学会
    • Place of Presentation
      福岡市
    • Year and Date
      2007-10-11
    • Description
      「研究成果報告書概要(和文)」より
  • [Presentation] Association of PD-1 gene to subacute sclerasing panencephalitis.2006

    • Author(s)
      Yoshito Ishizaki
    • Organizer
      2nd Congress of the Asian Society for Pediatric Research
    • Place of Presentation
      Yokohama, Japan
    • Year and Date
      20061208-20061210
    • Description
      「研究成果報告書概要(和文)」より
  • [Presentation] Association of PD-1 gene to subacute sclerosing panencephalitis.2006

    • Author(s)
      Y. Ishizaki
    • Organizer
      2nd Congress of the Asian Society for Pediatric Research
    • Place of Presentation
      Yokohama
    • Year and Date
      20061208-20061210
    • Description
      「研究成果報告書概要(欧文)」より
  • [Presentation] Acute disseminated encephalomyelitis in Japan : a population-based study of pediatric patients.2006

    • Author(s)
      Hiroyuki Torisu
    • Organizer
      The 8th International Congress of Neuroimmunology
    • Place of Presentation
      Nagoya, Japan
    • Year and Date
      20061015-20061019
    • Description
      「研究成果報告書概要(和文)」より
  • [Presentation] Acute disseminated encephalomyelitis in Japan: a population-based study of pediatric patients.2006

    • Author(s)
      H. Torisu
    • Organizer
      The 8th International Congress of Neuroimmunology
    • Place of Presentation
      Nagoya
    • Year and Date
      20061015-20061019
    • Description
      「研究成果報告書概要(欧文)」より
  • [Presentation] 新規NPC1遺伝子変異を認めた乳児期発症Niemann-Pick病C型の日本人同胞例2006

    • Author(s)
      鳥巣浩幸
    • Organizer
      第48回日本小児神経学会
    • Place of Presentation
      浦安市
    • Year and Date
      20060601-20060603
    • Description
      「研究成果報告書概要(和文)」より
  • [Presentation] Pelizaeus-Merzbacher病の遺伝子解析 : PLP欠損を招く新たな機構2005

    • Author(s)
      岩城明子
    • Organizer
      第28回日本分子生物学会
    • Place of Presentation
      福岡市
    • Year and Date
      20051207-20051210
    • Description
      「研究成果報告書概要(和文)」より
  • [Presentation] Molecular and genetic basis of subacute sclerosing panencephalitis : Microarray analysis of PBMCs.2005

    • Author(s)
      Ryutaro Kira
    • Organizer
      1^<st> Congress of the Asian Society for Pediatric Research
    • Place of Presentation
      Tokyo, Japan
    • Year and Date
      20051124-20051125
    • Description
      「研究成果報告書概要(和文)」より
  • [Presentation] Molecular and genetic basis of subacute sclerosing panencephalitis:Microarray analysis of PBMCs.2005

    • Author(s)
      R. Kira
    • Organizer
      1^<st> Congress of the Asian Society for Pediatric Research
    • Place of Presentation
      Tokyo
    • Year and Date
      20051124-20051125
    • Description
      「研究成果報告書概要(欧文)」より
  • [Presentation] PLP1遺伝子完全欠失によるPelizaeus-Merzbacher病(PLP null症候群)の男児例2005

    • Author(s)
      鳥巣浩幸
    • Organizer
      第47回日本小児神経学会
    • Place of Presentation
      熊本市
    • Year and Date
      20050519-20050521
    • Description
      「研究成果報告書概要(和文)」より
  • [Presentation] SSPEにおける末梢血単核球の網羅的遺伝子発現解析2005

    • Author(s)
      武本環美
    • Organizer
      第10回日本神経感染症学会
    • Place of Presentation
      東京
    • Year and Date
      2005-10-20
    • Description
      「研究成果報告書概要(和文)」より
  • [Book] 多発性硬化症の診断と治療 : 小児の急性散在性脳脊髄炎(分担執筆)2008

    • Author(s)
      吉良龍太郎
    • Total Pages
      6
    • Publisher
      新興医学出版社
    • Description
      「研究成果報告書概要(和文)」より

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Published: 2010-06-09  

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