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Investigation of causative gene of familial occurrence of idiopathic occlusion of Willis ring

Research Project

Project/Area Number 14370441
Research Category

Grant-in-Aid for Scientific Research (B)

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

Principal Investigator

HOUKIN Kiyohiro  Sapporo Medical University, School of Medicine, Professor, 医学部, 教授 (90229146)

Co-Investigator(Kenkyū-buntansha) TOKINO Takashi  Sapporo Medical University, School of Medicine, Professor, 医学部, 教授 (40202197)
TADA Mitsuhiro  Hokkaido University, Institute for Genetic Medicine, Professor, 遺伝子病制御研究所, 助教授 (10241316)
Project Period (FY) 2002 – 2004
Project Status Completed (Fiscal Year 2004)
Budget Amount *help
¥13,600,000 (Direct Cost: ¥13,600,000)
Fiscal Year 2004: ¥4,400,000 (Direct Cost: ¥4,400,000)
Fiscal Year 2003: ¥4,400,000 (Direct Cost: ¥4,400,000)
Fiscal Year 2002: ¥4,800,000 (Direct Cost: ¥4,800,000)
KeywordsMoyamoya disease / microsatelite analysis / familial occurrence / angiogenesis
Research Abstract

Purpose and Background:
It is estimated that some genetic factors are closely related to the familial moyamoya disease. Previous microsatellite analysis has suggested that related genes may be located on chromosomes 3,8,12 and 17. However, the responsible gene has not been identified yet. This study aimed to identify the responsible genes that are located in the 17825 locus. In addition, clinical anticipation and the presence of triplet repeat was investigated and the basic FGF and HGF of the cerebrospinal fluid of moyamoya patients were measured.
Methods and Results:
Considering the function, we selected nine genes as candidates from a total of 65 genes identified in the 9-cM region of D17S785-D17S836 in chromosome 17825, and performed sequence analysis on the DNA samples obtained from a pedigree of familial moyamoya disease, which showed a complete linkage to the region by a haplotype analysis. Also, we attempted to identify candidate genes that have not been known but might be function … More ally relevant to the disease among a total of 2,100 expressed sequence tag (EST) sequences using bioinformatics techniques. As results, the sequence analysis could detect no mutation in the nine genes. Nor could we identify a novel candidate gene by the EST analysis.
Clinical anticipation study was done based on 141 cases with moyamoya disease. This analysis revealed that apparent clinical anticipation was observed in familial moyamoya disease. No triplet repeat was found in 17g25 locus.
Cerebrospinal fluid was obtained from the patients during surgery. Control value was measured using asymptomatic cerebral aneurysm patients and other ischemic cerebrovascular disease. As results, high value of basic FGF and HGF was seen in the cerebrospinal fluid of moyamoya patients compared to the control group.
Conclusions:
Clinical anticipation observed in familial moyamoya patients suggests that some triplet repeat may located in some gene in moyamoya disease. Further studies using alternative approaches are warranted to clarify the pathogenesis of moyamoya disease. However, our results of high level of some cytokines including basicFGF and HGF suggests that some gene abnormality related to these cytokines are closely involved the pathogenesis of moyamoya disease. Less

Report

(4 results)
  • 2004 Annual Research Report   Final Research Report Summary
  • 2003 Annual Research Report
  • 2002 Annual Research Report
  • Research Products

    (65 results)

All 2005 2004 2003 2002 Other

All Journal Article (42 results) Book (6 results) Publications (17 results)

  • [Journal Article] Sequence analysis and bioinformatics analysis of chromosome 17q25 in familial moyamoya disease2005

    • Author(s)
      Nanba R
    • Journal Title

      Childs Nervous System 21

      Pages: 62-68

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] 無症候性もやもや病の治療指針について-成人もやもや病の進行はまれか?-2005

    • Author(s)
      黒田 敏
    • Journal Title

      厚生労働科学研究費補助金難治性疾患克服研究事業モヤモヤ病(ウィリス動脈輪閉塞症)に関する調査研究 平成16年度総括・分担研究報告書

      Pages: 45-46

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] モヤモヤ病の外科治療の周術期リスク2005

    • Author(s)
      宝金清博
    • Journal Title

      厚生労働科学研究費補助金 治性疾患克服研究事業モヤモヤ病(ウィリス動脈輪閉塞症)に関する調査研究 平成16年度総括・分担研究報告書

      Pages: 47-50

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] PLGF遺伝子導入による血管新生療法の基礎的研究2005

    • Author(s)
      本望 修
    • Journal Title

      厚生労働科学研究費補助金難治性疾患克服研究事業モヤモヤ病(ウィリス動脈輪閉塞症)に関する調査研究 平成16年度総括・分担研究報告書

      Pages: 51-52

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] MRAによるモヤモヤ病の新病期分類2005

    • Author(s)
      宝金清博
    • Journal Title

      厚生労働科学研究費補助金難治性疾患克服研究事業モヤモヤ病(ウィリス動脈輪閉塞症)に関する調査研究平成14年度〜16年度総合研究報告書

      Pages: 35-40

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] 家族性もやもや病の原因遺伝子の探索2005

    • Author(s)
      黒田 敏
    • Journal Title

      厚生労働科学研究費補助金難治性疾患克服研究事業モヤモヤ病(ウィリス動脈輪閉塞症)に関する調査研究平成14年度〜16年度総合研究報告書

      Pages: 63-65

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Sequence analysis and bioinformatics analysis of chromosome 17q25 in familial moyamoya disease2005

    • Author(s)
      Nanba R, Tada M, Kuroda S, Houkin K, Iwasaki Y.
    • Journal Title

      Childs Nervous System 21

      Pages: 62-68

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Sequence analysis and bioinformatics analysis of chromosome 17q25 in familial moyamoya disease2005

    • Author(s)
      Nanba R.
    • Journal Title

      Childs Nerv Syst 21

      Pages: 62-68

    • Related Report
      2004 Annual Research Report
  • [Journal Article] A case of moyamoya disease presenting with chorea2005

    • Author(s)
      Zheng W.
    • Journal Title

      Childs Nerv Syst (in press)

    • Related Report
      2004 Annual Research Report
  • [Journal Article] Pitfalls in cerebral revascularization surgery2004

    • Author(s)
      Houkin K
    • Journal Title

      International Congress Series 1259

      Pages: 313-320

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] 家族性もやもや病の臨床像と最近の研究の動向2004

    • Author(s)
      難波理奈
    • Journal Title

      脳神経外科 32

      Pages: 7-16

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] 家族性もやもや病家系における17q25のtriplet repeatの伸長に関する研究2004

    • Author(s)
      難波理奈
    • Journal Title

      厚生労働科学研究費補助金難治性疾患克服研究事業モヤモヤ病(ウィリス動脈輪閉塞症)に関する調査研究 平成15年度総括・分担研究報告書

      Pages: 47-49

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] もやもや病患者の脳脊髄液サイトカイン値と新治療法の検討2004

    • Author(s)
      宝金清博
    • Journal Title

      厚生労働科学研究費補助金難治性疾患克服研究事業モヤモヤ病(ウィリス動脈輪閉塞症)に関する調査研究 平成15年度総括・分担研究報告書

      Pages: 55-58

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Determinants of intellectual outcome after surgical revascularization in pediatric moyamoya disease : a multivariate analysis2004

    • Author(s)
      Kuroda S
    • Journal Title

      Childs Nervous System 20

      Pages: 302-308

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] How does angiogenesis develop in pediatric moyamoya disease after surgery? A prospective study with MR angiography2004

    • Author(s)
      Houkin K
    • Journal Title

      Childs Nervous System 20

      Pages: 734-741

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Increased expression of hepatocyte growth factor in cerebrospinal fluid and intracranial artery in moyamoya disease2004

    • Author(s)
      Nanba R
    • Journal Title

      Stroke 35

      Pages: 2837-2842

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Pitfalls in cerebral revascularization surgery2004

    • Author(s)
      Houkin K.
    • Journal Title

      International Congress Series 1259

      Pages: 313-320

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Determinants of intellectual outcome after surgical revascularization in pediatric moyamoya disease : a multivariate analysis2004

    • Author(s)
      Kuroda S, Houkin K, Ishikawa T, Nakayama N, Ikeda J, Ishii N, Kamiyama H, Iwasaki Y.
    • Journal Title

      Childs Nervous System 20

      Pages: 302-308

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] How does angiogenesis develop in pediatric moyamoya disease after surgery? A prospective study with MR angiography2004

    • Author(s)
      Houkin K, Nakayama N, Kuroda S, Ishikawa T, Nonaka T.
    • Journal Title

      Childs Nervous System 20

      Pages: 734-741

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Increased expression of hepatocyte growth factor in cerebrospinal fluid and intracranial artery in moyamoya disease2004

    • Author(s)
      Nanba R, Kuroda S, Ishikawa T, Houkin K, Iwasaki Y.
    • Journal Title

      Stroke 35

      Pages: 2837-2842

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Determinants of intellectual outcome after surgical revascularization in pediatric moyamoya disease ; a multivariate analysis2004

    • Author(s)
      Kuroda S.
    • Journal Title

      Childs Nerv Syst 20

      Pages: 302-308

    • Related Report
      2004 Annual Research Report
  • [Journal Article] Increased expression of hepatocyte growth factor in cerebrospinal fluid and intracranial artery in moyamoya disease2004

    • Author(s)
      Nanba R.
    • Journal Title

      Stroke 35

      Pages: 2837-2842

    • Related Report
      2004 Annual Research Report
  • [Journal Article] How does angiogenesis develop in pediatric moyamoya disease after surgery? A prospective study with MR angiography2004

    • Author(s)
      Houkin K.
    • Journal Title

      Childs Nerv Syst 20

      Pages: 734-741

    • Related Report
      2004 Annual Research Report
  • [Journal Article] MRAによるもやもや病の病期分類2003

    • Author(s)
      中山若樹
    • Journal Title

      厚生科学研究研究費補助金 特定疾患対策研究事業モヤモヤ病(ウィリス動脈輪閉塞症)に関する調査研究 平成14年度総括・分担研究報告書

      Pages: 9-13

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] 家族性もやもや病の原因遺伝子の探索2003

    • Author(s)
      黒田 敏
    • Journal Title

      厚生科学研究研究費補助金 特定疾患対策研究事業モヤモヤ病(ウィリス動脈輪閉塞症)に関する調査研究 平成14年度総括・分担研究報告書

      Pages: 51-53

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] もやもや病患者髄液中サイトカインの測定2003

    • Author(s)
      宝金清博
    • Journal Title

      厚生科学研究研究費補助金 特定疾患対策研究事業モヤモヤ病(ウィリス動脈輪閉塞症)に関する調査研究 平成14年度総括・分担研究報告書

      Pages: 59-61

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] モヤモヤ病に対する血行再建術2003

    • Author(s)
      宝金清博
    • Journal Title

      脳神経外科速報 13

      Pages: 247-252

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] もやもや病に対する直接的血行再建術2003

    • Author(s)
      宝金清博
    • Journal Title

      脳神経外科ジャーナル 12

      Pages: 301-301

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] もやもや病の診断・治療に関する現状と今後の展望2003

    • Author(s)
      黒田 敏
    • Journal Title

      脳卒中 25

      Pages: 215-229

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] モヤモヤ病2003

    • Author(s)
      宝金清博
    • Journal Title

      臨床医-これだけは知っておきたい臨床医の画像診断- 29(増刊号)

      Pages: 710-711

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] 乳幼児もやもや病の臨床像2003

    • Author(s)
      黒田 敏
    • Journal Title

      脳神経外科 31

      Pages: 1073-1078

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Source localization of the re-build up phenomenon in pediatric moyamoya disease-a dipole distribution analysis using MEG and SPECT2003

    • Author(s)
      Qiao F
    • Journal Title

      Childs Nervous System 19

      Pages: 760-764

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] 成人無症候性もやもや病の臨床像と予後2003

    • Author(s)
      難波理奈
    • Journal Title

      脳神経外科 31

      Pages: 1291-1295

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Source localization of the re-build up phenomenon in pediatric moyamoya disease-a dipole distribution analysis using MEG and SPECT2003

    • Author(s)
      Qiao F, Kuroda S, Kamada K, Houkin K, Iwasaki Y.
    • Journal Title

      Childs Nervous System 19

      Pages: 760-764

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] Moyamoya Disease. Batjer and Loftus: Textbook of Neurological Surgery2003

    • Author(s)
      Houkin K, Kuroda S.
    • Journal Title

      Lippincott Williams and Wilkins 1

      Pages: 1149-1159

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] 小児および成人もやもや病に対する脳血行再建術の長期効果2002

    • Author(s)
      黒田 敏
    • Journal Title

      脳卒中の外科 30

      Pages: 369-374

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] 脳画像におけるアポトーシス検出の展望2002

    • Author(s)
      宝金清博
    • Journal Title

      分子脳血管病 1

      Pages: 277-283

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] もやもや病の遺伝子異常-もやもや病病因研究の展望-2002

    • Author(s)
      宝金清博
    • Journal Title

      脳と循環 7

      Pages: 301-305

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] (学会記事)北日本脳神経外科連合会第26回学術集会-家族性もやもや病の臨床的特徴-2002

    • Author(s)
      難波理奈
    • Journal Title

      新潟医学会雑誌 116

      Pages: 508-509

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] もやもや病における後大脳動脈狭窄性病変の臨床的意義2002

    • Author(s)
      黒田 敏
    • Journal Title

      脳神経外科 30

      Pages: 1295-1300

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Journal Article] A case of moyamoya disease presenting with chorea

    • Author(s)
      Zheng W
    • Journal Title

      Childs Nervous System 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] A case of moyamoya disease presenting with chorea

    • Author(s)
      Zheng W, Houkin K.
    • Journal Title

      Childs Nervous System (in press)

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Book] もやもや病2004

    • Author(s)
      宝金清博
    • Publisher
      株式会社中山書店
    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Book] もやもや病-1.疾患の概略,2.疫学および病因,3.臨床症状,4.画像診断,5.治療2004

    • Author(s)
      宝金清博
    • Publisher
      金芳堂
    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Book] 脳神経外科体系13小児脳神経外科(山浦 晶編集)2004

    • Author(s)
      宝金清博
    • Publisher
      株式会社中山書店
    • Related Report
      2004 Annual Research Report
  • [Book] 脳神経外科学I改訂9版(太田富雄, 松谷雅生編集)2004

    • Author(s)
      宝金清博
    • Publisher
      金芳堂
    • Related Report
      2004 Annual Research Report
  • [Book] Textbook of Neurological Surgery : Batjer and Loftus2003

    • Author(s)
      Houkin K
    • Total Pages
      1212
    • Publisher
      Lippincott Williams and Wilkins
    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Book] モヤモヤ病の画像診断2002

    • Author(s)
      宝金清博
    • Publisher
      先端医療技術研究所
    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2004 Final Research Report Summary
  • [Publications] 宝金清博, 本望 修, 備前明子: "モヤモヤ病患者の脳脊髄液サイトカイン値と新治療法の検討"厚生省もやもや病研究班平成15年度報告書. (印刷中). (2004)

    • Related Report
      2003 Annual Research Report
  • [Publications] 難波理奈, 黒田 敏, 石川達哉, 多田光宏, 宝金清博, 岩崎喜信: "家族性もやもや病における17q25のtriplet repeatの伸張に関する研究"厚生省もやもや病研究班平成15年度報告書. (印刷中). (2004)

    • Related Report
      2003 Annual Research Report
  • [Publications] 宝金清博: "モヤモヤ病"臨床医-これだけは知っておきたい臨床医の画像診断-. 29(増刊号). 710-711 (2003)

    • Related Report
      2003 Annual Research Report
  • [Publications] 宝金清博: "もやもや病に対する直接的血行再建術"脳外誌. 12. 301 (2003)

    • Related Report
      2003 Annual Research Report
  • [Publications] 宝金清博: "モヤモヤ病に対する血行再建術"脳神経外科速報. 13. 247-252 (2003)

    • Related Report
      2003 Annual Research Report
  • [Publications] 黒田 敏, 中山若樹, 難波理奈, 七戸秀夫, 石川達哉, 鈴木明文, 西野晶子, 宝金清博, 峰松一夫: "もやもや病の診断・治療に関する現状と今後の展望"脳卒中. 25. 215-229 (2003)

    • Related Report
      2003 Annual Research Report
  • [Publications] Qiao F, Kuroda S, Kamada K, Houkin K, Iwasaki Y: "Source localization of the re-build up phenomenon in pediatric moyamoya disease a dipole distribution analysis using MEG and SPECT"Childs Nerv Syst. 19. 760-764 (2003)

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  • [Publications] Houkin K: "Pitfalls in cerebral revascularization surgery"International Congress Series. 1259. 313-320 (2004)

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  • [Publications] 難波理奈, 黒田 敏, 石川達哉, 多田光宏, 宝金清博, 岩崎喜信: "家族性もやもや病の臨床像と最近の研究の動向"脳外. 32. 7-16 (2004)

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  • [Publications] Houkin K, Kuroda S: "Textbook of Neurological Surgery"Lippincott. 1212 (2003)

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  • [Publications] 宝金 清博, 本望 修, 備前明子: "もやもや病患者髄液中サイトカインの測定"厚生省もやもや病研究班平成14年度報告書. (印刷中). (2003)

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  • [Publications] 中山 若樹, 宝金 清博, 黒田 敏: "MRAにより病期分類"厚生省もやもや病研究班平成14年度報告書. (印刷中). (2003)

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  • [Publications] 宝金清博: "モヤモヤ病の画像診断"先端医療シリーズ17・脳神経外科 脳血管障害の最新医療. 292-297 (2002)

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  • [Publications] 宝金清博, 難波理奈, 黒田 敏, 多田光宏: "もやもや病の遺伝子異常-もやもや病病因研究の展望-"脳と循環. 7. 301-305 (2002)

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  • [Publications] 黒田 敏, 石川達哉, 宝金清博, 岩崎喜信: "もやもや病における後大脳動脈狭窄性病変の臨床的意義"脳外. 30. 1295-1300 (2002)

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  • [Publications] 難波理奈, 黒田 敏, 岩崎喜信, 多田光宏, 宝金清博: "(学会記事)北日本脳神経外科連合会第26回学術集会-家族性もやもや病の臨床的特徴-"新潟医学会雑誌. 116. 508 (2002)

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  • [Publications] Houkin K, Kuroda S: "Textbook of Neurological Surgery"Lippincott. 1212 (2003)

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Published: 2002-04-01   Modified: 2016-04-21  

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