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Generation of dominant dystrophic epidermolysis bullosa model mice using humanized COL7 mice

Research Project

Project/Area Number 24659516
Research Category

Grant-in-Aid for Challenging Exploratory Research

Allocation TypeMulti-year Fund
Research Field Dermatology
Research InstitutionHokkaido University

Principal Investigator

SHIMIZU Hiroshi  北海道大学, 大学院・医学研究科, 教授 (00146672)

Co-Investigator(Kenkyū-buntansha) SHINKUMA Satoru  北海道大学, 大学院・医学研究科, 助教 (00613788)
Project Period (FY) 2012
Project Status Completed (Fiscal Year 2012)
Budget Amount *help
¥3,770,000 (Direct Cost: ¥2,900,000、Indirect Cost: ¥870,000)
Fiscal Year 2012: ¥3,770,000 (Direct Cost: ¥2,900,000、Indirect Cost: ¥870,000)
Keywords表皮水疱症 / モデルマウス / ドミナントネガティブ効果 / グリシン置換変異 / 7型コラーゲン
Research Abstract

Dominant dystrophic epidermolysis bullosa (DDEB) is a clinically heterogeneous disorder characterized by blistering and scarring of the skin and mucous membranes in response to mechanical force. The DDEB phenotype arises because of a dominant negative effect of mutations in type VII collagen gene, resulting in the formation of structurally abnormal anchoring fibrils. There have not been DDEB model mouse. In thisstudy, we attempt to generate the DDEB model mice.

Report

(2 results)
  • 2012 Annual Research Report   Final Research Report ( PDF )
  • Research Products

    (7 results)

All 2013 2012 Other

All Journal Article (4 results) (of which Peer Reviewed: 4 results) Presentation (3 results)

  • [Journal Article] Jacyk、Ken Natsuga、Hideyuki Ujiie、Hideki Nakamura、Masashi Akiyama、Hiroshi Shimizu2013

    • Author(s)
      Satoru Shinkuma、Wataru Nishie、 Witold K
    • Journal Title

      A Novel Keratin 5 Mutation in an African Family with Epidermolysis Bullosa Simplex Indicates the Importance of the Amino Acid Located at the Boundary Site Between the H1 and Coil 1A Domains

      Volume: 印刷中 Issue: 5 Pages: 585-587

    • DOI

      10.2340/00015555-1538

    • Related Report
      2012 Final Research Report
    • Peer Reviewed
  • [Journal Article] Long-term Follow-up of Cultured Epidermal Autograft in a Patient with Recessive Dystrophic Epidermolysis Bullosa2013

    • Author(s)
      Shinkuma S, Sawamura D, Fujita Y, Kawasaki H, Nakamura H, Inoie M, Nishie W, Shimizu H
    • Journal Title

      Acta Dermato-Venereologica

      Volume: In press

    • Related Report
      2012 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Type VII collagen deficiency causesdefective tooth enamel formation due to poor differentiation of ameloblasts2012

    • Author(s)
      Umemoto H, Akiyama M, Domon T, Nomura T, Shinkuma S, Ito K, Asaka T, Sawamura D, Uitto J, Uo M, Kitagawa Y, Shimizu H
    • Journal Title

      Am J Pathol

      Volume: 181 Issue: 5 Pages: 1659-71

    • DOI

      10.1016/j.ajpath.2012.07.018

    • Related Report
      2012 Final Research Report
    • Peer Reviewed
  • [Journal Article] Long-term follow-up of cultured epidermal autograft in a recessive dystrophic epidermolysis bullosa patient

    • Author(s)
      Satoru Shinkuma、Daisuke Sawamura、Yasuyuki Fujita、Hiroyuki Kawasaki、Hiroyuki Nakamura、Masukazu Inoie、ataru Nishie、Hiroshi Shimizu
    • Journal Title

      Acta Dermato-Venereologica

      Volume: (in press)

    • Related Report
      2012 Final Research Report
    • Peer Reviewed
  • [Presentation] ケラチン5のコイル1A領域N末 端部にミスセンス変異を生じた単純型表皮水疱症2012

    • Author(s)
      新熊悟
    • Organizer
      第76回日本皮膚科学会東部支部学術大会
    • Place of Presentation
      ロイトン札幌(北海道)
    • Year and Date
      2012-09-30
    • Related Report
      2012 Final Research Report
  • [Presentation] Long-term follow-up ofcultured epidermal autograft in a recessive dystrophic epidermolysis bullosa patient2012

    • Author(s)
      新熊悟
    • Place of Presentation
      Raleigh Convention Center(米国)
    • Year and Date
      2012-05-10
    • Related Report
      2012 Final Research Report
  • [Presentation] Long-term follow-up of cultured epidermal autograft in a recessive dystrophic epidermolysis bullosa patient2012

    • Author(s)
      Shinkuma S, Sawamura D, Kawasaki H, Nakamura H, Inoie M, Nishie W, Shimizu H
    • Organizer
      72nd Annual Meeting of the SOCIETY FOR INVESTIGATIVE DERMATOLOGY
    • Place of Presentation
      Raleigh Convention Center(米国)
    • Related Report
      2012 Annual Research Report

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Published: 2013-05-31   Modified: 2019-07-29  

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