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Study for the development of screening method for the teratogenic medicine using spina bifida model mice

Research Project

Project/Area Number 16591026
Research Category

Grant-in-Aid for Scientific Research (C)

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

Principal Investigator

SAKAI Norio  Osaka University, Graduate School of Medicine, Assistant Professor, 医学系研究科, 講師 (30314313)

Co-Investigator(Kenkyū-buntansha) OKINAGA Takeshi  Osaka University, Gaduate School of Medicine, Assistant, 医学系研究科, 助手 (30362734)
TANIIKE Masako  Osaka University, Gaduate School of Medicine, Associate Professor, 医学系研究科, 助教授 (30263289)
OZONO Keiichi  Osaka University, Graduate School of Medicine, Professor, 医学系研究科, 教授 (20270770)
Project Period (FY) 2004 – 2005
Project Status Completed (Fiscal Year 2005)
Budget Amount *help
¥3,400,000 (Direct Cost: ¥3,400,000)
Fiscal Year 2005: ¥1,300,000 (Direct Cost: ¥1,300,000)
Fiscal Year 2004: ¥2,100,000 (Direct Cost: ¥2,100,000)
Keywordsspina bifida / Lrp6 / Wnt signaling / pQCT / 葉酸 / 催奇性薬剤 / スクリーニング
Research Abstract

1)Selection of highly susceptible model mice for spina bifida : We prepare KO mice of Wnt receptor Lrp6 and KO mice of Pax1 after cleaning for SPF room. We observed the ratio of embryo having spina bifida produced with mating of Lrp6 heterozygous pair. We found this ratio was as high as 40% of all embryo produced with this mating.
2)Medicine treatment of pregnant mice ; We prepare mating of heterozygous mouse for each mutant and wild mouse. We treated the carbamazepine or valproic acid with several dose and different schedule. We produced the embryo at E12 and observed the ratio of embryos showing spina bifida.
3)Phenotypic analysis of embryo : We also produced homozygous embryo of rs (hypomorphic mutation of Lrp6) with heterozygous mating. The embryos were stained with Alcianblue and Alizarin for analyzing the abnormality of cartilage or bone formation. We found the abnormality of bone formation in rs/rs embryo. We analysed the bone measurement using pQCT method for bone density. In this observation we found loss of bone density at cancellous tissue in the tibia of rs/rs mouse for young and old mouse. We analysed the expression of Rankl gene in the osteoblast cells produced from rs/rs was increased.
4)Analysis of protective effect of folic acid aginst spina bifida ; We treated pregnant mice produced from rs heterozygous mating with folic acid for three days. We checked the ratio of spina bifida in the embryo of wild type and heterozygous type. We could not detect clear difference in this observation.

Report

(3 results)
  • 2005 Annual Research Report   Final Research Report Summary
  • 2004 Annual Research Report
  • Research Products

    (22 results)

All 2006 2005 2004 Other

All Journal Article (22 results)

  • [Journal Article] Novel mutation of gene coding for glial fibrillary acidic protein in a Japanese patient with Alexander disease2006

    • Author(s)
      Kawai M, Sakai N, Miyake S, Tsukamoto H, Akagi M, Inui K, Mushiake S, Taniike M, Ozono K.
    • Journal Title

      Brain Dev 28.6

      Pages: 60-62

    • NAID

      10017319316

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2005 Final Research Report Summary
  • [Journal Article] Characterization of the sensitivity of lymphoblastoid cell lines to various stress agents in Roberts syndrome2006

    • Author(s)
      Gordillo M, Vega H, Sakai N, Tsukamoto H, Ozono K, Inui K
    • Journal Title

      Med J Osaka Univ 49(1-4)

      Pages: 29-41

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2005 Final Research Report Summary
  • [Journal Article] Novel mutation of gene coding for glial fibrillary acidic protein in a Jananese patient with Alexander disease2006

    • Author(s)
      Kawai M, Sakai N, Miyake S, Tsukamoto H, Akagi M, Inui K, Mushiake S, Taniike M, Ozono K.
    • Journal Title

      Brain Dev 28(6)

      Pages: 60-62

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2005 Final Research Report Summary
  • [Journal Article] Novel mutation of gene coding for glial fibrillary acidic protein in a Japanese patient with Alexander disease2006

    • Author(s)
      Kawai M, Sakai N, Miyake S, Tsukamoto H, Akagi M, Inui K, Mushiake S, Taniike M, Ozono K.
    • Journal Title

      Brain Dev 28(6)

      Pages: 60-62

    • NAID

      10017319316

    • Related Report
      2005 Annual Research Report
  • [Journal Article] Related Articles, Links, Renal cell carcinoma in a pediatric patient with an inherited mitochondrial mutation2005

    • Author(s)
      Sangkhathat S, Kusafuka T, Yoneda A, Kuroda S, Tanaka M, Sakai N, Fukuzawa M.
    • Journal Title

      Pediatr Surg Int 21.9

      Pages: 745-748

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2005 Final Research Report Summary
  • [Journal Article] Enzyme replacement therapy in Japanese Fabry disease patients : the results of a phase 2 bridging study2005

    • Author(s)
      Eto Y, Ohashi T, Utsunomiya Y, Fujiwara M, Mizuno A, Inui K, Sakai N, Kitagawa T, Suzuki Y, Mochizuki S, Kawakami M, Hosoya T, Owada M, Sakuraba H, Saito H.
    • Journal Title

      J Inherit Metab Dis 28.4

      Pages: 575-583

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2005 Final Research Report Summary
  • [Journal Article] Roberts syndrome is caused by mutations in ESCO2, a human homolog of yeast ECO1 that is essential for the establishment of sister chromatid cohesion2005

    • Author(s)
      Vega H, Waisfisz Q, Gordillo M, Sakai N, Yanagihara I, Yamada M, van Gosliga D, Kayserili H, Xu C, Ozono K, Jabs EW, Inui K, Joenje H.
    • Journal Title

      Nat Genet 37.5

      Pages: 468-470

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2005 Final Research Report Summary
  • [Journal Article] Renal cell carcinoma in a pediatric patient with an inherited mitochondrial mutation2005

    • Author(s)
      Sangkhathat S, Kusafuka T, Yoneda A, Kuroda S, Tanaka M, Sakai N, Fukuzawa M.
    • Journal Title

      Pediatr Surg Int 21(9)

      Pages: 745-748

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2005 Final Research Report Summary
  • [Journal Article] Enzyme replacement therapy in Japanese Fabry diseas patients : the results of a phase 2 bridging study2005

    • Author(s)
      Eto Y, Ohashi T, Utsunomiya Y, Fujiwara M, Mizuno A, Inui K, Sakai N, Kitagawa T, Suzuki Y, Mochizuki S, Kawakami M, Hosoya T, Owada M, Sakuraba H, Saito H.
    • Journal Title

      J Inherit Metab Dis 28(4)

      Pages: 575-583

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2005 Final Research Report Summary
  • [Journal Article] Roberts syndrome is caused by mutations in ESCO2, a human homolog of yeast ECO1 that is essential for the establishment of sister chromatid cohesion2005

    • Author(s)
      Vega H, Waisfisz Q, Gordillo M, Sakai N, Yanagihara I, Yamada M, van Gosliga D, Kayserili H, Xu C, Ozono K, Jabs EW, Inui K, Joenje H.
    • Journal Title

      Nat Genet 37(5)

      Pages: 468-470

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2005 Final Research Report Summary
  • [Journal Article] Related Articles, Links, Renal cell carcinoma in a pediatric patient with an inherited mitochondrial mutation2005

    • Author(s)
      Sangkhathat S, Kusafuka T, Yoneda, A, Kuroda S, Tanaka M, Sakai N, Fukuzawa M.
    • Journal Title

      Pediatr Sure Int 21(9)

      Pages: 745-748

    • Related Report
      2005 Annual Research Report
  • [Journal Article] Enzyme replacement therapy in Japanese Fabry disease patients : the results of phase 2 bridging study2005

    • Author(s)
      Eto Y, Ohashi T, Utsunomiya Y, Fujiwara M, Mizuno A, Inui K, Sakai N, Kitagawa T, Suzuki Y, Mochizuki S, Kawakami M, Hosoya T, Owada M, Sakuraba H, Saito H.
    • Journal Title

      J Inherit Metab Dis 28(4)

      Pages: 575-583

    • Related Report
      2005 Annual Research Report
  • [Journal Article] Roberts syndrome is caused by mutations in ESCO2, a human homolog of yeast ECO1 that is essential for the establishment of sister chromatid cohesion2005

    • Author(s)
      Vega H, Waisfisz Q, Gordillo M, Sakai N, Yanagihara I, Yamada M, van Gosliga D, Kayserili H, Xu C, Ozono K, Jabs EW, Inui K, Joenie H.
    • Journal Title

      Nat Genet 37(5)

      Pages: 468-470

    • Related Report
      2005 Annual Research Report
  • [Journal Article] Skeletal defects in ringelschwanz mutant mice reveal that Lrp6 is required for proper somitogenesis and osteogenesis2004

    • Author(s)
      Chikara Kokubu Ulrich Heinzmann2, Tomoko Kokubu, Norio Sakai, et al.
    • Journal Title

      Development 131(21)

      Pages: 5469-5480

    • Related Report
      2004 Annual Research Report
  • [Journal Article] In Vivo Studies of Phenylalanine Hydroxylase by Phenylalanine Breath Test : Diagnosis of Tetrahydrobiopterin-Responsive Phenylalanine Hydroxylase Deficiency.2004

    • Author(s)
      Okano Y, Hase Y, Kawajiri M, Nishi Y, Inui K, Sakai N, et al.
    • Journal Title

      Pediatr Res. 56

      Pages: 714-719

    • Related Report
      2004 Annual Research Report
  • [Journal Article] Involvement of phosphoinositide 3-kinase signaling pathway in chondrocytic differentiation of ATDC5 cells : Application of a gene-trap mutagenesis.2004

    • Author(s)
      Ihara-Watanabe M, Uchihashi T, Miyauchi Y, Sakai N, et al.
    • Journal Title

      J Cell Biochem 93(2)

      Pages: 418-426

    • Related Report
      2004 Annual Research Report
  • [Journal Article] Analysis of recombinant human saposin A expressed by Pichia pastoris.2004

    • Author(s)
      Yamada M, Inui K, Hamada D, Nakahira K, Yanagihara K, Sakai N et al.
    • Journal Title

      Biochem Biophys Res Commun 318(2)

      Pages: 588-593

    • Related Report
      2004 Annual Research Report
  • [Journal Article] Six novel mutations detected in GALC gene in 17 Japanese patients with Krabbe disease and new genotype-phenotype correlation

    • Author(s)
      Xu C, Sakai N, Taniike M, Inui K, Ozono K
    • Journal Title

      J Hum Genet in press

    • NAID

      10017608409

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2005 Final Research Report Summary
  • [Journal Article] Characterization of the sensitivity of lymphoblastoid cell lines to various stress agents in Roberts syndrome

    • Author(s)
      Gordillo M, Vega H, Sakai N, Tsukamoto H, Ozono K, Inui K
    • Journal Title

      Med J Osaka Univ in press

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      2005 Final Research Report Summary
  • [Journal Article] Six novel mutations detected in GALC gene in 17 Japanese patients with Krabbe disease and new genotype-phenotype correlation

    • Author(s)
      Xu C, Sakai N, Taniike M, Inui, Ozono K
    • Journal Title

      J Hum Genet (in press)

    • NAID

      10017608409

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      2005 Final Research Report Summary
  • [Journal Article] Six novel mutations detected in GALC gene in 17 Japanese patients with Krabbe disease and new genotype-phenotype correlation

    • Author(s)
      Xu C, Sakai N, Taniike M, Inui, Ozono K
    • Journal Title

      J Hum Genet in press

    • NAID

      10017608409

    • Related Report
      2005 Annual Research Report
  • [Journal Article] Characterization of the sensitivity of lymphoblastoid cell lines to various stress agents in Roberts syndrome

    • Author(s)
      Gordillo M, Vega H, Sakai N.Tsukamoto H, Ozono K, Inui K
    • Journal Title

      Med J Osaka Univ in press

    • Related Report
      2005 Annual Research Report

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Published: 2004-04-01   Modified: 2016-04-21  

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