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Cloning and Characterization of the Hemifacial Microsomia Related Gene.

Research Project

Project/Area Number 10470456
Research Category

Grant-in-Aid for Scientific Research (B)

Allocation TypeSingle-year Grants
Section一般
Research Field 矯正・小児・社会系歯学
Research InstitutionShimane Prefectural Shimane Women's College

Principal Investigator

NAORA Hiroyuki  Shimane Women's College, Food Science, Instructor, 家政科, 助手 (70222156)

Co-Investigator(Kenkyū-buntansha) OTANI Hiroki  Shimane Medical University, Medicine, Professor, 医学部, 教授 (20160533)
Project Period (FY) 1998 – 1999
Project Status Completed (Fiscal Year 1999)
Budget Amount *help
¥11,100,000 (Direct Cost: ¥11,100,000)
Fiscal Year 1999: ¥5,100,000 (Direct Cost: ¥5,100,000)
Fiscal Year 1998: ¥6,000,000 (Direct Cost: ¥6,000,000)
KeywordsHemifacial Microsomia / Craniofacial Anomaly / Insertional Mutation / Chromosome 10 / Animal Model / Transgenic Mouse / 遺伝子導入マウス / 片側顔面萎縮症 / 片側顔面委縮 / 遺伝性疾患 / Hemi facial microsomia
Research Abstract

To get an insight into the genetical predisposition and pathogenesis of the hemifacial microsomia (HFM), we analyzed the mutant locus of the HFM model mouse. By the P1 phage library screening, we isolated the chromosomal DNA from the mutant region. By the DNA sequencing, we screened the coding regions that corresponded to the mutant gene(s). At present, we sequenced about 10 kb, then we identified some regions that had highly homologous sequences with those of some known genes. These genes locate the different loci with HFM locus, then it is suggested that the gene(s) that responds to HFM pathogenesis is a nobel gene, and shares some domain structures to the known genes.
We investigated the phenotype of homo-zygote of HFM mouse. By the natural mating between the hemi zygotes, we obtained 12 embryos at E9.5. Three embryos died as the rosy mass. PCR analysis showed that one of three embryos was a homo-zygote (another two embryos could not determine their genotypes due to DNA degradation). According to the size of the placenta, homo-zygotes embryo developed normally to E7.5. These results showed that the gene that disrupted in HFM mouse had an essential role for the normal development.
The pathological screening was carried out at the late prenatal days (E13.5-17.5). In addition to the branchial arch anomalies that described previously, other anomalies and asymmetries at the maxilla, mandible, zygoma and facial soft tissue ware identified. These observation further supported that HFM mouse is a suitable animal model for the human HFM at the point of the pathological characteristics.

Report

(3 results)
  • 1999 Annual Research Report   Final Research Report Summary
  • 1998 Annual Research Report
  • Research Products

    (21 results)

All Other

All Publications (21 results)

  • [Publications] 大谷浩: "初期発生:卵割から胚葉形成まで"生体の科学. 49. 54-60 (1998)

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      1999 Final Research Report Summary
  • [Publications] Kazuhiro Sugihara: "Racl is required for the formation of three germ layers during gastrulation"Oncogene. 17. 3427-3433 (1998)

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      1999 Final Research Report Summary
  • [Publications] Isao Oishi: "Spatio-temporally regulated expression of receptor tyrosine kinases, mRor1, mRor2, during mouse development : implications in development and function of the nervous system"Genes to Cells. 4. 41-56 (1999)

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      1999 Final Research Report Summary
  • [Publications] H. Naora: "Deuterated condition temporally modifies cell migration pattern of cultured fibroblasts"Jpn. J. Deuterium Sci.. 8. 3-10 (1999)

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      1999 Final Research Report Summary
  • [Publications] L. Zhang: "Myogenic Determination and differentiation of the mouse palatal muscle in relation to the developing mandibular nerve"J. Dent. Res.. 78. 1417-1425 (1999)

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      1999 Final Research Report Summary
  • [Publications] Ryuju Hashimoto: "Mouse embryo culture systems for postimplantation stage and expression of lim class homeodomain protein, Lim-1 in early mouse embryo geneses"Shimane J. Mes. Sci.. 17. 43-49 (1999)

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      1999 Final Research Report Summary
  • [Publications] N. Tamura: "Cardiac fibrosis in mice lacking brain natriuretic peptide"Proc. Natl. Acad. Sci. USA. (in press).

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      1999 Final Research Report Summary
  • [Publications] J. Udagawa: "Expression of the long form of leptin receptor (Ob-Rb)mRNA in the brain of mouse mbryos and newborn mice."Brain Res.. (in press).

    • Description
      「研究成果報告書概要(和文)」より
    • Related Report
      1999 Final Research Report Summary
  • [Publications] Kazuhiro Sugihara, Norio Nakatsuji, Kenji Nakamura, Kazuki Nakao, Ryuju Hashimoto, Hiroki Otani, Hiroyuki Sakagami, Hisatake Kondo, Shiro Nozawa, Atsu Aiba and Motoya Katsuki: "Rac1 is required for the formation of three germ layers during gastrulation."Oncogene. 17. 3427-3427 (1998)

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      1999 Final Research Report Summary
  • [Publications] Isao Oishi, Shigeto Takeuchi, Ryuju Hashimoto, Akira Nagabukuro, Takahiro Ueda, Zhao-Jun Liu, Toshihisa Hatta, Shizuo Akira, Yoichi Matsuda, Hirohei Yamamura, Hiroki Otani and Yasuhiro Minami: "Spatio-temporally regulated expression of receptor tyrosine kinases, mRor1, mRor2, during mouse development : implications in development and function of the nervous system."Genes to Cells. 4. 41-56 (1999)

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      1999 Final Research Report Summary
  • [Publications] H. Naora, K.Matoba, H.Omori and H.Otani: "Deuterated condition temporally modifies cell migration pattern of cultured fibroblasts."Jpn. J. Deuterium Sci.. 8. 3-10 (1999)

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      1999 Final Research Report Summary
  • [Publications] L.Zhang, Y.Yoshimura, T.Hatta and H.Otani: "Myogenic Determination and differentiation of the mouse palatal muscle in relation to the developing mandibular nerve."J. Dent. Res.. 78. 1417-1425 (1999)

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      1999 Final Research Report Summary
  • [Publications] Ryuju Hashimoto, Masayuki Ajiki and Hiroki Otani: "Mouse embryo culture systems for postimplantation stage and expression of lim class homeodomain protein, Lim-1 in early mouse embryogeneses."J. Dent. Res.. 78. 43-19 (1999)

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      1999 Final Research Report Summary
  • [Publications] N.Tamura, Y.Ogawa, H.Chusho, K.Nakamura, K.Nakao, M.Suda, M.Kasahara, R.Hashimoto, G.Katsuura, M.Mukoyama, H.Itoh, Y.Saito, I.Tanaka, H.Otani, M.Katsuki and K.Nakao: "Cardiac fibrosis in mice lacking brain natriuretic peptide."Proc. Natl. Acad. Sci. USA. (in press).

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      1999 Final Research Report Summary
  • [Publications] J.Udagawa, T.Hatta, H.Naora and H.Otani: "Expression of the long form of leptin receptor (Ob-Rb) mRNA in the brain of mouse mbryos and newborn mice."Brain Res.. (in press).

    • Description
      「研究成果報告書概要(欧文)」より
    • Related Report
      1999 Final Research Report Summary
  • [Publications] Isao Oishi: "Spatio-tempora11y regulated expression of receptor tyrosine kinases, mRor1, mRor2, during mouse development: implications in development and function of the nervous system"Genes to Cells.

    • Related Report
      1999 Annual Research Report
  • [Publications] H.Naora: "Deuterated condition tempora11y modifies cell migration pattern of cultured fibroblasts"Jpn.J.Deuterium Sci..

    • Related Report
      1999 Annual Research Report
  • [Publications] L.Zhang: "Myogenic Determination and differentiation of the mouse palatal muscle in relation to the developing mandibular nerve"J.Dent.Res..

    • Related Report
      1999 Annual Research Report
  • [Publications] Ryuju Hashimoto: "Mouse embryo culture systems for postimplantation stage and expression of lim class homeodomain protein, Lim-1 in early mouse embryogeneses"Shimane J.Mes.Sci..

    • Related Report
      1999 Annual Research Report
  • [Publications] N.Tamura: "Cardiac fibrosis in mice lacking brain natriuretic peptide"Proc.Natl.Acad.Sci.USA.

    • Related Report
      1999 Annual Research Report
  • [Publications] J.Udagawa: "Expression of the long form of leptin receptor (Ob-Rb) mRNA in the brain of mouse mbryos and newborn mice"Brain Res..

    • Related Report
      1999 Annual Research Report

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Published: 1998-04-01   Modified: 2016-04-21  

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