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1993 Fiscal Year Final Research Report Summary

Establishment of prenatal diagnosis of epidermolysis bullosa in Japan

Research Project

Project/Area Number 04557045
Research Category

Grant-in-Aid for Developmental Scientific Research (B)

Allocation TypeSingle-year Grants
Research Field Dermatology
Research InstitutionKeio Gijuku University

Principal Investigator

NISHIKAWA Takeji  Keio Univ.Sch.Med., Professor, 医学部・皮膚科, 教授 (50051579)

Co-Investigator(Kenkyū-buntansha) SUZUMORI Kaoru  Medical School, Nagoya City University, Assist.Prof., 医学部・産科婦人科, 助教授 (80117829)
SHIMIZU Hiroshi  Keio Univ.Sch.Med., Instructor, 医学部・皮膚科, 助手 (00146672)
HASHIMOTO Takashi  Keio Univ.Sch.Med., Assist.Prof., 医学部・皮膚科, 講師 (20129597)
Project Period (FY) 1992 – 1993
Keywordsprenatal diagnosis / epidermolysis bullosa / fetal skin biopsy / GB3 / LH7.2 / 19-DEJ-1 / electron microscopy
Research Abstract

With the Grant-in-Aid for Scientific Research (B), we have made a prenatal diagnosis of epidermolysis bullosa for the first time in Japan. Prenatal diagnosis of epidermolysis bullosa using fetal skin biopsy specimens has been successfully performed in Europe and America, but this technique has not previously been attempted in Asia. We, for the first time in Asia, attempted tomake a prenatal diagnosis of epidermolysis bullosa in high-risk fetus by fetal skin biopsy. A skin biopsy was obtained from the fetus as risk of gravis type ofjunctional epidermolysis bullosa of Herlitz. The biopsy specime was studied by electron microscopy and by immunohistochemistry using various monoclonal antibodies against epidermal basement membrane zone. There were were no ultrastructural abnormalities in the basement membrane zone, including the hemidesmosomes. Indirect immunofluorescence study showed normal expression of GB3 antigen. The pregnancy was continued, and a normal, healthy infant was born. The prenatal diagnosis of epidermolysis bullosa is now available in Tokyo. This clinical diagnostic service is available to families from various parts of Asia.

  • Research Products

    (23 results)

All Other

All Publications (23 results)

  • [Publications] 清水宏: "遺伝性皮膚疾患の遺伝外来" 臨床皮膚科. 47. 162-167 (1993)

    • Description
      「研究成果報告書概要(和文)」より
  • [Publications] Akiyama,M.: "Ultrastructural localization of Brazilian pemphigus foliaceus(fogo selvagem)antigens in cultured human squamous cell carcinoma cells." Br J Dermatol. 128. 378-383 (1993)

    • Description
      「研究成果報告書概要(和文)」より
  • [Publications] Ishiko,A.: "Human autoantibodies against the 230-kD bullous pemphigoid antigen(BPAG1)bind only to the intracellular domain of the hemidesmosome whereas those against the plasma membrane of the hemidesmosome in normal human and swine skin." J Clin Invest. 91. 1608-1615 (1993)

    • Description
      「研究成果報告書概要(和文)」より
  • [Publications] Hashimoto,T.: "Desmoyolcin,a 680kDa keratinocyte plasma membrane-associated protein,is holnologous to the protein encoded by human gene AHNAK." J Cell-Science.105. 275-286 (1993)

    • Description
      「研究成果報告書概要(和文)」より
  • [Publications] Shimizu,H.: "Recent advances in electron microscopic immunocytochemistry in dermatology." Eur J Dermatol. 3. 635-642 (1993)

    • Description
      「研究成果報告書概要(和文)」より
  • [Publications] Shimizu,H.: "Prenatal diagnosis of epidermolysis bullosa:First successful trial in Asia." Dermatology. 188. 46-49 (1994)

    • Description
      「研究成果報告書概要(和文)」より
  • [Publications] Shimizu,H.: "Proceeding of the 8th Japan-Korea joint meeting of Dermatology 1993" The 8th Japan-Korea Joint Meeting of Dermatology, 5 (1993)

    • Description
      「研究成果報告書概要(和文)」より
  • [Publications] 西川武二: "標準皮膚科学 第4版" 医学書院, 6 (1994)

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      「研究成果報告書概要(和文)」より
  • [Publications] Shimizu, H., Ishida-Yamamoto, A., Eady, R.A.J.: "The use of silver-enhanced 1-nm gold probes for light and electron microscopic localization of intra- and extracellular antigens in skin." J Histochem Cytochem. 40(6). 883-888 (1992)

    • Description
      「研究成果報告書概要(欧文)」より
  • [Publications] Dmochowski, M., Hashimoto, T., Nishikawa, T.: "The analysis of IgG subclasses of anti-intercellular antibodies in pemphigus by an immunoblot technique." Arch Dermatol Res. 284(5). 309-311 (1992)

    • Description
      「研究成果報告書概要(欧文)」より
  • [Publications] Shimizu, H., Ishiko, A., Kikuchi, A., Akiyama, M., Suzumori, K., Nishikawa, T.: "Prenatal diagnosis of tyrosinase-negative oculocutaneous albinism." Lancet. 340. 739-740 (1992)

    • Description
      「研究成果報告書概要(欧文)」より
  • [Publications] Onodera, Y., Shimizu, H., Yamashita, S., Nishikawa, T.: "Cryofixed, freeze-dried and paraffin-embedded skin enables successful immunohistochemical staining of skin basement membrane antigens." Histochemistry. 98(2). 87-91 (1992)

    • Description
      「研究成果報告書概要(欧文)」より
  • [Publications] Akiyama, M., Hashimoto, T., Sugiura, M., Nishikawa, T.: "Ultrastructural localization of autoantigens of intercellular IgA vesiculopustular dermatosis in cultured human squamous cell carcinoma cells." Arch Dermatol Res. 284(6). 371-373 (1992)

    • Description
      「研究成果報告書概要(欧文)」より
  • [Publications] Iwatsuki, K., Hashimoto, T., Ebihara, T., Teraki, Y., Nishikawa, T., Kaneko, F.: "Intercellular IgA vesiculo-pustular dermatosis and related disorders : diversity of IgA anti-intercellular autoantibodies." EJD. 3(1). 7-11 (1993)

    • Description
      「研究成果報告書概要(欧文)」より
  • [Publications] Shimizu, H., Nishikawa, T.: "Application of an image analyzer to gold labeling in immunoelectron microscopy to achieve better demonstration and quantitative analysis." J Histochem Cytochem. 41(1). 123-128 (1993)

    • Description
      「研究成果報告書概要(欧文)」より
  • [Publications] Ebihara, T., Hashimoto, T., Kudoh, J., Gamou, S., Shimizu, N., Nishikawa, T.: "Detection of the 170-kDa bullous pemphigoid antigen by immunoprecipitation." J Invest Dermatol. 100(2). 176-179 (1993)

    • Description
      「研究成果報告書概要(欧文)」より
  • [Publications] Hashimoto, T., Amagai, M., Ebihara, T., Gamou, S., Shimizu, N., Tsubata, T., Hasegawa, A., Miki, K., Nishikawam, T.: "Further analyzes of epitopes for human monoclonal anti-basement membrane zone antibodies produced by stable human hybridoma cell lines constructed with epstein-barr virus transformants." J Invest Dermatol. 100(3). 310-315 (1993)

    • Description
      「研究成果報告書概要(欧文)」より
  • [Publications] Dmochowski, M., Hashimoto, T., Bhogal, B.S., Black, M.M., Miyakawa, K., Miyamoto, R., Nishikawa, T.: "Concomitant occurrence of circulating IgA anti-intercellular and anti-basement membrane zone antibodies in autoimmune blistering diseases : immunofluorescence and immunoblot atudies." J Dermatol. 20(3). 131-137 (1993)

    • Description
      「研究成果報告書概要(欧文)」より
  • [Publications] Hashimoto, T., Ebihara, T., Ishiko, A., Shimizu, H., Bhogal, B.S., Black, M.M., Stanley, J.R., Nishikawa, T.: "Comparative study of bullous pemphigoid antigents among Japanese, British, and U.S.patients indicates similar antigen present both in the basement membrane and on the keratinocyte cell membrane." J Invest Dermatol. 100(4). 385-389 (1993)

    • Description
      「研究成果報告書概要(欧文)」より
  • [Publications] Ishiko, A., Shimizu, H., Kikuchi, A., Ebihara, T., Hashimoto, T., Nishikawa, T.: "Human autoantibodies against the 230-kD bullous pemphigoid antigen(BPAG1) bind only to the intracellular domain of the hemidesmosome whereas those against the plasma membrane of the hemidesmosome in normal human and swine skin." J Clin Invest. 91(4). 1608-1615 (1993)

    • Description
      「研究成果報告書概要(欧文)」より
  • [Publications] Hashimoto, T., Amagai, M., Parry, D.A.D., Dixon, T.W., Tsukita, S., Tsukita, S., Miki, K., Sakai, K., Inokuchi, Y., Kudoh, J., Shimizu, N., Nishikawa, T.: "Desmoyokin, a 680 kDa keratinocyte plasma membrane-associated protein, is homologous to the protein encoded by human gene AHNAK." J Cell Science. 105(2). 275-286 (1993)

    • Description
      「研究成果報告書概要(欧文)」より
  • [Publications] Shimizu, H., Nishikawa, T.: "Recent advances in electron microscopic immunocytochemistry in dermatology." Eur J Dermatol. 3(8). 635-642 (1993)

    • Description
      「研究成果報告書概要(欧文)」より
  • [Publications] Shimizu, H., Onodera, Y., Ikeda, S., Ogawa, H., Suzumori, K., Nishikawa, T.: "Prenatal diagnosis of epidermolysis bullosa : First successful trial in Asia." Dermatology. 188(1). 46-49 (1994)

    • Description
      「研究成果報告書概要(欧文)」より

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Published: 1995-03-27  

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