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Global analysis of congenital bone and joint diseases using a chondrogenic differentiation system from iPS cells

Research Project

Project/Area Number 24591507
Research Category

Grant-in-Aid for Scientific Research (C)

Allocation TypeMulti-year Fund
Section一般
Research Field Pediatrics
Research InstitutionKyoto University

Principal Investigator

UMEDA KATSUTUSGU  京都大学, 医学(系)研究科(研究院), 助教 (80397538)

Project Period (FY) 2012-04-01 – 2015-03-31
Project Status Completed (Fiscal Year 2014)
Budget Amount *help
¥5,200,000 (Direct Cost: ¥4,000,000、Indirect Cost: ¥1,200,000)
Fiscal Year 2014: ¥1,560,000 (Direct Cost: ¥1,200,000、Indirect Cost: ¥360,000)
Fiscal Year 2013: ¥1,560,000 (Direct Cost: ¥1,200,000、Indirect Cost: ¥360,000)
Fiscal Year 2012: ¥2,080,000 (Direct Cost: ¥1,600,000、Indirect Cost: ¥480,000)
KeywordsiPS細胞 / 骨・関節疾患 / 軟骨分化 / 軟骨 / 先天異常
Outline of Final Research Achievements

We addressed whether the pathogenesis of CINCA syndrome, a congenital bone and joint disease characterized by epiphyseal overgrowth, could be recapitulated by using neural crest-derived chondroprogenitor differentiation from using patient-derived iPS cells. 3D chondrogenic assays showed mutant iPS cells produced significantly huge chondrogenous pellets than wild type iPS cells. Furthermore, in vivo chongrogenic assays by subcutaneous xenotranplation into immunodeficient mice showed that huge chondrogenenous tissues, consisting of immature chondroprogenitors, were obtained, which recapitulates the pathological condition of the syndrome.

Report

(4 results)
  • 2014 Annual Research Report   Final Research Report ( PDF )
  • 2013 Research-status Report
  • 2012 Research-status Report
  • Research Products

    (5 results)

All 2015 2013 2012

All Journal Article (3 results) (of which Peer Reviewed: 3 results,  Open Access: 1 results,  Acknowledgement Compliant: 1 results) Presentation (2 results)

  • [Journal Article] Enhanced chondrogenesis of induced pluripotent stem cells from patients with neonatal-onset multisystem inflammatory disease occurs via the caspase 1-independent cAMP/protein kinase A/CREB pathway2015

    • Author(s)
      Yokoyama, K. Ikeya, M. Umeda, K. Oda, H. Nodomi, S. Nasu, A. Matsumoto, Y. Izawa, K. Horigome, K. Kusaka, T. Tanaka, T. Saito, M. K. Yasumi, T. Nishikomori, R. Ohara, O. Nakayama, N. Nakahata, T. Heike, T. Toguchida, J.
    • Journal Title

      Arthritis Rheumatol

      Volume: 67 Issue: 1 Pages: 302-314

    • DOI

      10.1002/art.38912

    • Related Report
      2014 Annual Research Report
    • Peer Reviewed / Open Access / Acknowledgement Compliant
  • [Journal Article] Selective development of myogenic mesenchymal cells from human embryonic and induced pluripotent stem cells2012

    • Author(s)
      Awaya T., Kato T., Mizuno Y., Chang H., Niwa A., Umeda K., Nakahata T., Heike T.
    • Journal Title

      PLoS ONE

      Volume: 7(12) Issue: 12 Pages: e51638-e51638

    • DOI

      10.1371/journal.pone.0051638

    • NAID

      120005122354

    • Related Report
      2012 Research-status Report
    • Peer Reviewed
  • [Journal Article] Human chondrogenic paraxial mesoderm, directed specification and prospective isolation from pluripotent stem cells.2012

    • Author(s)
      Umeda K, Zhao J, Simmons P, Stanley E, Elefanty A, Nakayama N
    • Journal Title

      Sci Rep

      Volume: 2 Issue: 1 Pages: 455-455

    • DOI

      10.1038/srep00455

    • Related Report
      2012 Research-status Report
    • Peer Reviewed
  • [Presentation] ヒト多能性幹細胞を用いた神経堤由来軟骨前駆細胞の大量増幅システムの開発2013

    • Author(s)
      梅田雄嗣、納富誠司郎、平家俊男、中山直樹
    • Organizer
      日本再生医療学会
    • Place of Presentation
      横浜市
    • Related Report
      2012 Research-status Report
  • [Presentation] ヒト多能性幹細胞を用いた神経堤由来軟骨前駆細胞の大量増幅システムの開発2013

    • Author(s)
      梅田雄嗣、納富誠司郎、平家俊男、中山直樹
    • Organizer
      日本小児科学会総会
    • Place of Presentation
      広島市
    • Related Report
      2012 Research-status Report

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

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