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Experimental study of immune responses in cell transplantation therapy for genetic neurodegenerative disorders

Research Project

Project/Area Number 20591229
Research Category

Grant-in-Aid for Scientific Research (C)

Allocation TypeSingle-year Grants
Section一般
Research Field Pediatrics
Research InstitutionOsaka City University

Principal Investigator

TANAKA Akemi  Osaka City University, 大学院・医学研究科, 准教授 (30145776)

Co-Investigator(Kenkyū-buntansha) MAEDA Mitsuyo  大阪歯科大学, 大学院・医学研究科, 助教 (40122080)
SETO Toshiyuki  大阪市立大学, 大学院・医学研究科, 講師 (60423878)
Project Period (FY) 2008 – 2010
Project Status Completed (Fiscal Year 2010)
Budget Amount *help
¥4,550,000 (Direct Cost: ¥3,500,000、Indirect Cost: ¥1,050,000)
Fiscal Year 2010: ¥910,000 (Direct Cost: ¥700,000、Indirect Cost: ¥210,000)
Fiscal Year 2009: ¥1,040,000 (Direct Cost: ¥800,000、Indirect Cost: ¥240,000)
Fiscal Year 2008: ¥2,600,000 (Direct Cost: ¥2,000,000、Indirect Cost: ¥600,000)
Keywords神経遺伝性疾患 / 細胞移植 / 免疫応答 / 移植免疫 / 再生医療 / 遺伝性神経疾患
Research Abstract

We performed cell transplantation therapy to treat the brain involvement of lysosomal storage diseases. The grafted cells were gradually depleted because of immunological rejection by the host animals. To avoid deleterious immune attack and to achieve sufficient long-term efficacy in brain, development of methods to steer the immune response away from cytotoxic responses or to induce tolerance to the products of therapeutic genes is needed. To overcome this immunological rejection, we carried out brain injection and intravenous administration of mesenchymal stem cells simultaneously to a model mouse of β-galactosidase deficiency. However, we could not show a significant deference in the duration of engrafting between the brain injection only and the simultaneous injection of brain and vein.

Report

(4 results)
  • 2010 Annual Research Report   Final Research Report ( PDF )
  • 2009 Annual Research Report
  • 2008 Annual Research Report
  • Research Products

    (46 results)

All 2011 2010 2009 2008 Other

All Journal Article (22 results) (of which Peer Reviewed: 21 results) Presentation (20 results) Book (4 results)

  • [Journal Article] Enzyme replacement therapy in a patient with Fabry disease and the development of IgE antibodies against agalsidase beta but not agalsidase alpha.2010

    • Author(s)
      Tanaka A, Takeda T, Hoshina T, Fukai K, Yamano T.
    • Journal Title

      J Inherit Metab Dis (Epub ahead of print)

    • Related Report
      2010 Final Research Report
    • Peer Reviewed
  • [Journal Article] Japan Elaprase Treatment (JET) Study : Idursulfase Enzyme Replacement Therapy in Adult Patients with Attenuated Hunter Syndrome.2010

    • Author(s)
      Okuyama T, Tanaka A, Suzuki Y, Ida H, Tanaka T, Cox JF, Eto Y, Orii T.
    • Journal Title

      Mol Genet Metab 99

      Pages: 18-25

    • Related Report
      2010 Final Research Report
    • Peer Reviewed
  • [Journal Article] Nerve injury-activated microglia engulf myelinated axons in a P2Y12 signaling-dependent manner in the dorsal horn.2010

    • Author(s)
      Maeda M, Tsuda M, Tozaki-Saitoh H, Inoue K, Kiyama H.
    • Journal Title

      Glia (Epub ahead of print)

    • Related Report
      2010 Final Research Report
    • Peer Reviewed
  • [Journal Article] Damage-induced neuronal endopeptidase is critical for presynaptic formation of neuromuscular junctions.2010

    • Author(s)
      Nagata K, Kiryu-Seo S, Maeda M, Yoshida K, Morita T, Kiyama H.
    • Journal Title

      J Neurosci 30

      Pages: 6954-6962

    • Related Report
      2010 Final Research Report
    • Peer Reviewed
  • [Journal Article] Japan Elaprase Treatment (JET) Study : Idursulfase Enzyme Replacement Therapy in Adult Patients with Attenuated Hunter Syndrome2010

    • Author(s)
      Okuyama T, Tanaka A, Suzuki Y, Ida H, Tanaka T, Cox JF, Eto Y, Orii T
    • Journal Title

      Mol Genet Metab

      Volume: 99 Pages: 18-25

    • Related Report
      2010 Annual Research Report
    • Peer Reviewed
  • [Journal Article] ムコ多糖症親の会患者家族に対する酵素補充療法の意識調査(酵素補充療法承認前調査)2010

    • Author(s)
      田中あけみ、澤田智、山野恒一
    • Journal Title

      日本小児科学会雑誌

      Volume: 114 Pages: 465-467

    • Related Report
      2010 Annual Research Report
    • Peer Reviewed
  • [Journal Article] ファブリー病透析患者における酵素補充療法の治療効果とアガルシダーゼアルファの体内動態について2010

    • Author(s)
      岡田志緒子、稲荷場ひろみ、崔吉永、河野仁美、寺柿政和、岡村幹夫、吉本充、田中あけみ、根来伸夫、葭山稔
    • Journal Title

      日本透析学会雑誌

      Volume: 43 Pages: 945-951

    • NAID

      10027893550

    • Related Report
      2010 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Japan Elaprase Treatment (JET) Study : Idursulfase Enzyme Replacement Therapy in Adult Patients with Attenuated Hunter Syndrome.2010

    • Author(s)
      Okuyama T, Tanaka A, Suzuki Y, Ida H, Tanaka T, Cox JF, Eto Y, Orii T
    • Journal Title

      Mol Genet Metab 99

      Pages: 18-25

    • Related Report
      2009 Annual Research Report
    • Peer Reviewed
  • [Journal Article] ムコ多糖症親の会患者家族に対する酵素補充療法の意識調査(酵素補充療法承認前調査)2010

    • Author(s)
      田中あけみ、澤田智、山野恒一
    • Journal Title

      小児科学会雑誌 114

      Pages: 463-467

    • Related Report
      2009 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Intracerebral cell transplantation therapy for murine GM1 gangliosidosis.2009

    • Author(s)
      Sawada T, Tanaka A, Higaki K, Takamura A, Nanba E, Seto T, Maeda M, Yamaguchi E, Matsuda J, Yamano T.
    • Journal Title

      Brain & Development 31

      Pages: 717-724

    • NAID

      10026412886

    • Related Report
      2010 Final Research Report 2009 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Mucolipidosis II and III alpha/beta : mutation analysis of 40 Japanese patients showed genot ype-phenotype correlation.2009

    • Author(s)
      Otomo T, Muramatu T, Yorifuji T, Oku yama T, Nakabayashi H, Fukao T, Oh ura T, Yoshino M, Tanaka A, Okamoto N, Ozono K, Sakai N.
    • Journal Title

      J Hum Ge net 54

      Pages: 145-151

    • Related Report
      2010 Final Research Report
    • Peer Reviewed
  • [Journal Article] Mucolipidosis II and III alpha/beta : mutation analysis of 40 Japanese patients showed genotype-phenotype correlation.2009

    • Author(s)
      Otomo T, Muramatu T, Yorifuji T, Okuyama T, Nakabayashi H, Fukao T, Ohura T, Yoshino M, Tanaka A, Okamoto N, Ozono K, Sakai N
    • Journal Title

      J Hum Genet 54

      Pages: 145-151

    • NAID

      10030729556

    • Related Report
      2009 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Mucolipidosis II and III alpha/beta : mutation analysis of 40 Japanese patients showed genotype-phenotype correlation.2009

    • Author(s)
      Otomo T, Muramatu T, Yorifuji T, Okuyama T, Nakabayashi H, Fukao T, Ohura T, Yoshino M, Tanaka A, Okamoto N, Ozono K, Sakai N.
    • Journal Title

      J Hum Genet 54

      Pages: 145-151

    • NAID

      10030729556

    • Related Report
      2008 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Intracerebral cell transplantation therapy for murine GM1 gangliosidosis.2009

    • Author(s)
      Sawada T, Tanaka A, Higaki K, Takamura A, Nanba E, Seto T, Maeda M, Yamaguchi E, Matsuda J, Yamano T.
    • Journal Title

      Brain & Development 31(In press)

    • NAID

      10026412886

    • Related Report
      2008 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Fluorescence in situ hybridization analysis is useful for the diagnosis of the carrier state of X-linked ichthyosis.2008

    • Author(s)
      Hosomi N, Fukai K, Tanaka A, Fujita H, Ishii M.
    • Journal Title

      Internat J Dermatol 47

      Pages: 529-530

    • Related Report
      2010 Final Research Report 2008 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Non-invasive high-risk screening for Fabry disease hemizygotes and heterozygotes.2008

    • Author(s)
      Kitagawa T, Suzuki K, Ishige N, Ohashi T, Kobayashi M, Eto Y, Tanaka A, Odaka H, Owada M.
    • Journal Title

      Pediatr Nephrol 23

      Pages: 1461-1471

    • Related Report
      2010 Final Research Report
    • Peer Reviewed
  • [Journal Article] Vascular type of Ehlers-Danlos syndrome associated with haemophilia A.2008

    • Author(s)
      Umekoji A, Fukai K, Hosomi M, Ishii M, Tanaka A, Murakami K, Kamoi H, Mizoguchi M, Utani A.
    • Journal Title

      Clin Exper Dermatol 34

      Pages: 101-101

    • Related Report
      2010 Final Research Report
    • Peer Reviewed
  • [Journal Article] Expression of pancreatitis associated proteins in urothelium and urinary afferent neurons following cyclophosphamide induced cystitis.2008

    • Author(s)
      Takahara Y, Suzuki A, Maeda M, Kawashima H, Nakatani T, Kiyama H.
    • Journal Title

      J Urol 179

      Pages: 1603-1609

    • Related Report
      2010 Final Research Report
    • Peer Reviewed
  • [Journal Article] Non-invasive high-risk screening for Fabry disease hemizygotes and heterozygotes. 23 : 1461-1471, 2008.2008

    • Author(s)
      Kitagawa T, Suzuki K, Ishige N, Ohashi T, Kobayashi M, E to Y, Tanaka A, Odaka H, Owada M.
    • Journal Title

      Pediatr Nephrol 23

      Pages: 1461-1471

    • Related Report
      2008 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Vascular type of Ehlers-Danlos syndrome associated with haemophilia A. 34 : 101, 20082008

    • Author(s)
      Umekoji A, Fukai K, Hosomi M, Ishii M, Tanaka A, Murakami K, Kamoi H, Mizoguchi M, U tani A.
    • Journal Title

      Clin Exper Dermatol 34

      Pages: 101-101

    • Related Report
      2008 Annual Research Report
    • Peer Reviewed
  • [Journal Article] ムコ多糖症のマス・スクリーニング(総説)2008

    • Author(s)
      田中あけみ
    • Journal Title

      日本マス・スクリーニング学会誌 18

      Pages: 224-228

    • Related Report
      2008 Annual Research Report
  • [Journal Article] Enzyme replacement therapy in a patient with Fabry disease and the development of IgE antibodies against agalsidase beta but not agalsidase alpha

    • Author(s)
      Tanaka A, Takeda T, Hoshina T, Fukai K, Yamano T
    • Journal Title

      J Inherit Metab Dis

      Volume: (Epub)

    • Related Report
      2010 Annual Research Report
    • Peer Reviewed
  • [Presentation] Efficacy of hematopoietic cell transplantation on the patients with mucopolysaccharidosis type II : Multicenter survey in Japan.2010

    • Author(s)
      Tanaka A, Okuyama T, Suzuki Y, Sakai N, Sawada T, Tanaka T, Takakura H, Ohashi T, Wada M, Yabe H, Ohura T, Suzuki N, Kato K, Adachi S, Mugishima H, Kato S
    • Organizer
      11^<th> International Symposium on Mucopolysaccharide and Related Diseases
    • Place of Presentation
      Adelaide, Australia
    • Related Report
      2010 Final Research Report
  • [Presentation] Japanese perspectives of optimal treatment with ERT and HSCT for mucopolysaccharidoses Symposium2010

    • Author(s)
      田中あけみ
    • Organizer
      The 1^<st> Asian Congress for Inherited Metabolic Diseases
    • Place of Presentation
      Fukuoka
    • Related Report
      2010 Final Research Report
  • [Presentation] ムコ多糖症II 型における造血幹細胞移植の中枢神経系への効果について(多施設共同研究)2010

    • Author(s)
      田中あけみ、酒井規夫、奥山虎之、鈴木康之、澤田智、大橋十也、大浦敏博、麦島秀雄、田中藤樹、大友孝信
    • Organizer
      第52回日本小児神経学会
    • Place of Presentation
      福岡
    • Related Report
      2010 Final Research Report
  • [Presentation] 分野別シンポジウム5:先天性代謝異常症に対する造血幹細胞移植2010

    • Author(s)
      田中あけみ
    • Organizer
      第113回日本小児科学会
    • Place of Presentation
      盛岡市民文化ホール・いわて県民情報交流センター(招待講演)
    • Related Report
      2010 Annual Research Report
  • [Presentation] Efficacy of hematopoietic cell transplantation on the patients with mucopolysaccharidosis type II : Multicenter survey in Japan2010

    • Author(s)
      Tanaka A, Okuyama T, SuzukiY, Sakai N, Sawada T, Tanaka T, Takakura H, Ohashi T, Wada M, Yabe H, Ohura T, Suzuki N, Kato K, Adachi S, Mugishima H, Kato S
    • Organizer
      11^<th> International Symposium on Mucopolysaccharide and Related Diseases
    • Place of Presentation
      アデレード・オーストラリアアデレードコンベンションセンター
    • Related Report
      2010 Annual Research Report
  • [Presentation] シンポジウム:新しい治療法の適応とガイドライン:ムコ多糖症2010

    • Author(s)
      田中あけみ
    • Organizer
      第52回日本先天代謝異常学会
    • Place of Presentation
      大阪国際会議場(招待講演)
    • Related Report
      2010 Annual Research Report
  • [Presentation] ライソゾーム病マス・スクリーニングの試みと遺伝カウンセリング2010

    • Author(s)
      田中あけみ
    • Organizer
      第55回日本人類遺伝学会
    • Place of Presentation
      大宮ソニックシティ
    • Related Report
      2010 Annual Research Report
  • [Presentation] Enzyme replacement therapy and hematopoietic stem cell transplantation for MPS II patients in Japan.2009

    • Author(s)
      Tanaka A
    • Organizer
      The 5^<th> Meeting of the Mucopolysaccharidoses Association in Taiwan
    • Place of Presentation
      台北・中華人民共和国
    • Year and Date
      2009-06-14
    • Related Report
      2009 Annual Research Report
  • [Presentation] ムコ多糖症II 型に対する造血幹細胞移植の効果に関する多施設共同調査研究(第1報)2009

    • Author(s)
      澤田智、田中あけみ、奥山虎之、田中藤樹、鈴木康之、酒井規夫、大橋十也、高倉広充、矢部晋正、加藤俊一
    • Organizer
      第51回日本先天代謝異常学会
    • Place of Presentation
      東京
    • Related Report
      2010 Final Research Report
  • [Presentation] Enzyme replacement therapy for a boy with Fabry disease showing IgE positive against agalsidase beta, not against agalsidase alpha.2009

    • Author(s)
      Takeda T, Tanaka A, Hoshina T, Fukai K, Yamano T
    • Organizer
      11^<th> International Congress of Inborn Errors of Metabolism
    • Place of Presentation
      サンディエゴ・アメリカ合衆国
    • Related Report
      2009 Annual Research Report
  • [Presentation] Efficacy of enzyme replacement therapy on the patients with late stage of mucopolysaccharidoses Type I and II.2009

    • Author(s)
      Sawada T, Tanaka A, Aso K, Tsuruhara A, Morita K, Takeda T, Tada A, Hozumi T, Yokoi T, Hattori H, Yamano T
    • Organizer
      11^<th> International Congress of Inborn Errors of Metabolism
    • Place of Presentation
      サンディエゴ・アメリカ合衆国
    • Related Report
      2009 Annual Research Report
  • [Presentation] Pharmacokinetics of agalsidase alpha and beta in three patients with Fabry disease.2009

    • Author(s)
      S Okada, A Tanaka, T Takeda, I Kobayashi, E Ishimura, N Negoro, Y Nishizawa, T Yamano, M Yoshiyama
    • Organizer
      11^<th> International Congress of Inborn Errors of Metabolism
    • Place of Presentation
      サンディエゴ・アメリカ合衆国
    • Related Report
      2009 Annual Research Report
  • [Presentation] Pitfalls in Diagnosis of MPS and How to Differentiate Between MPS.2009

    • Author(s)
      Tanaka A
    • Organizer
      11^<th> Asian Symposium of Lysosomal Storage Disease
    • Place of Presentation
      台北・中華人民共和国
    • Related Report
      2009 Annual Research Report
  • [Presentation] 先天性代謝異常症の遺伝カウンセリングを始める前に2009

    • Author(s)
      田中あけみ
    • Organizer
      第51回日本先天代謝異常学会
    • Place of Presentation
      東京
    • Related Report
      2009 Annual Research Report
  • [Presentation] ハンター病兄弟例にみる臨床像に影響を及ぼす因子と酵素補充療法および造血幹細胞移植の効果について2008

    • Author(s)
      田中あけみ、澤田智、麻生和良、鶴原昭史、森田こころ、多田明央、山野恒一
    • Organizer
      第13回日本ライソゾーム病研究会
    • Place of Presentation
      東京
    • Related Report
      2010 Final Research Report
  • [Presentation] ムコ多糖症骨髄移植症例の骨髄抑制下における酵素製剤の薬物動態について2008

    • Author(s)
      岡崎伸、田中あけみ、原純一、西垣五月、東浦壮志、木村志保子、澤田智、山野恒一
    • Organizer
      第50回日本先天代謝異常学会
    • Place of Presentation
      米子
    • Related Report
      2010 Final Research Report
  • [Presentation] ムコ多糖症の酵素補充療法 : I型5例、II型4例の使用経験2008

    • Author(s)
      澤田智, 田中あけみ, 麻生和良, 鶴原昭史, 横井俊明, 服部英司, 多田明央, 山野恒一
    • Organizer
      第111回日本小児科学会
    • Place of Presentation
      東京
    • Related Report
      2008 Annual Research Report
  • [Presentation] Efficacy of enzyme replacement therapy in 12 Japanese patients with mucopolysaccharidosis type II (Hunter disease)2008

    • Author(s)
      Sawada T, Tanaka A, Aso K, Yamano T.
    • Organizer
      10^<th> International Symposium on Mucopolysacc haridoses and Related Disease
    • Place of Presentation
      Vancouver, Canada
    • Related Report
      2008 Annual Research Report
  • [Presentation] MPS II patient reports2008

    • Author(s)
      Tanaka A.
    • Organizer
      11^<th> Annual Asia LSD symposium
    • Place of Presentation
      Beijing, China
    • Related Report
      2008 Annual Research Report
  • [Presentation] ムコ多糖症11例の酵素補充療法の効果の検討2008

    • Author(s)
      澤田智, 田中あけみ, 森田こころ, 麻生和良, 鶴原昭史, 横井俊明, 服部英司, 多田明央, 山野恒一
    • Organizer
      第50回日本先天代謝異常学会
    • Place of Presentation
      米子
    • Related Report
      2008 Annual Research Report
  • [Book] Neurochemistry of Metabolic Disease, Chaptor 8: Enzyme Replacement Therapy for Lysosomal Storage Dseases. Editors: Sanker Surendran2011

    • Author(s)
      Tomatsu S, Montano AM, Molano ACS, Roman D, Hintze J, Carvalho CG, Federhen A, Vieira TA, Giugliani R, Wegrzyn G, Tanaka A, Suzuki Y, Orii T.
    • Publisher
      Nova Science Publishers, Inc. in press
    • Related Report
      2010 Final Research Report
  • [Book] Neurochemistry of Metabolic Disease2011

    • Author(s)
      Tomatsu S, Montano AM, Molano ACS, Roman D, Hintze J, Carvalho CG, Federhen A, Vieira TA, Giugliani R, Wegrzyn G, Tanaka A, Suzuki Y, Orii T
    • Publisher
      Chaptor 8 : Enzyme Replacement Therapy for Lysosomal Storage Dseases(In press)
    • Related Report
      2010 Annual Research Report
  • [Book] ポンペ病(糖原病II型)2009

    • Author(s)
      田中あけみ, 他
    • Total Pages
      171
    • Publisher
      診断と治療社
    • Related Report
      2009 Annual Research Report
  • [Book] 症例から学ぶ先天性代謝異常症2009

    • Author(s)
      田中あけみ, 他
    • Total Pages
      237
    • Publisher
      診断と治療社
    • Related Report
      2009 Annual Research Report

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Published: 2008-04-01   Modified: 2016-04-21  

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