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Establishment of iPS cells from McCune-Albright syndrome and it application

Research Project

Project/Area Number 16K15791
Research Category

Grant-in-Aid for Challenging Exploratory Research

Allocation TypeMulti-year Fund
Research Field Pathobiological dentistry/Dental radiology
Research InstitutionTokyo Dental College

Principal Investigator

Yamaguchi Akira  東京歯科大学, 歯学部, 客員教授 (00142430)

Co-Investigator(Kenkyū-buntansha) 小野寺 晶子  東京歯科大学, 歯学部, 講師 (90637662)
齋藤 暁子  東京歯科大学, 歯学部, 助教 (90722835)
Project Period (FY) 2016-04-01 – 2018-03-31
Project Status Completed (Fiscal Year 2017)
Budget Amount *help
¥3,380,000 (Direct Cost: ¥2,600,000、Indirect Cost: ¥780,000)
Fiscal Year 2017: ¥1,690,000 (Direct Cost: ¥1,300,000、Indirect Cost: ¥390,000)
Fiscal Year 2016: ¥1,690,000 (Direct Cost: ¥1,300,000、Indirect Cost: ¥390,000)
KeywordsMcCune-Albright 症候群 / iPS細胞 / GNAS / McCune-Albright症候群 / 線維性骨異形成症 / GNAS1 / 歯学 / 病理学
Outline of Final Research Achievements

Since distribution of the gene mutated cells in McCune-Albright syndrome (MAS) occurs depending somatic mosaicism, we applied gene editing technique using CRISPR/Cas9 system to generate human iPS cells exhibiting GNAS1 mutation observed in MAS. We obtained 2 clones exhibiting GAS1 mutation among 24 clones. These iPS cells showed undifferentiated markers and those identifying three germ layers. Thus we successfully established human iPS cells retaining GNAS1 mutaion observed in MAS.

Report

(3 results)
  • 2017 Annual Research Report   Final Research Report ( PDF )
  • 2016 Research-status Report
  • Research Products

    (19 results)

All 2018 2017 2016

All Journal Article (14 results) (of which Int'l Joint Research: 2 results,  Peer Reviewed: 14 results,  Open Access: 12 results,  Acknowledgement Compliant: 1 results) Presentation (4 results) (of which Invited: 4 results) Book (1 results)

  • [Journal Article] Leukemia inhibitory factor produced by fibroblasts within tumor stroma participates in invasion of oral squamous cell carcinoma.2018

    • Author(s)
      Ohata Y, Tsuchiya M, Hirai H, Yamaguchi S, Akashi T, Sakamoto K, Yamaguchi A, Ikeda T, Kayamori K
    • Journal Title

      PLos One

      Volume: 13 Issue: 2 Pages: e0191865-e0191865

    • DOI

      10.1371/journal.pone.0191865

    • Related Report
      2017 Annual Research Report
    • Peer Reviewed / Open Access
  • [Journal Article] Targeted reversion of induced pluripotent stem cells from patients with human cleidocranial dysplasia showed corrected osteoblastic differentiation both in vitro and in vivo.2018

    • Author(s)
      Ooki A, Nakamura T, Onodera S, Hayashi K, Hasegawa D, Kato H, Onda T, Watanabe A, Kosaki K, Nishimura K, Ohtaka M, Nakanishi M, Sakamoto T, Yamaguchi A, Sueishi K, Azuma T
    • Journal Title

      Stem Cell Res Ther

      Volume: 9 Issue: 1 Pages: 12-12

    • DOI

      10.1186/s13287-017-0754-4

    • Related Report
      2017 Annual Research Report
    • Peer Reviewed / Open Access
  • [Journal Article] The HIV co-receptor CCR5 regulates osteoclast function.2017

    • Author(s)
      Lee JW, Hoshino A, Inoue K, Saitou T, Uehara S, Kobayashi Y, Ueha S, Matsushima K, Yamaguchi A, Imai Y, Iimura T.
    • Journal Title

      Nat Commun

      Volume: 8 Issue: 1 Pages: 2226-2226

    • DOI

      10.1038/s41467-017-02368-5

    • Related Report
      2017 Annual Research Report
    • Peer Reviewed / Open Access / Int'l Joint Research
  • [Journal Article] Gorlin syndrome-derived induced pluripotent stem cells are hypersensitive to Hedgehog-mediated osteogenic induction.2017

    • Author(s)
      Hasegawa D, Shino H, Onodera S, Nakamura T, Saito A, Onda T, Watanabe K, Nishimura K, Ohtaka M, Nakanishi M, Kosaki K, Yamaguchi A, Shibahara T, Azuma T
    • Journal Title

      PLos One

      Volume: 12 Issue: 10 Pages: e0186879-e0186879

    • DOI

      10.1371/journal.pone.0186879

    • Related Report
      2017 Annual Research Report
    • Peer Reviewed / Open Access
  • [Journal Article] Multi-layered mutatin in hedgehog-related genes in Gorlin syndrome may affect the phenotype.2017

    • Author(s)
      Onodera S, Saito A, Hasegawa D, Watanabe K, Morita N, Nomura T, Shibahara T, Ohba S, Yamaguchi A, Azuma T
    • Journal Title

      PLOS ONE

      Volume: 12 Issue: 9 Pages: e0184702-e0184702

    • DOI

      10.1371/journal.pone.0184702

    • Related Report
      2017 Annual Research Report
    • Peer Reviewed / Open Access
  • [Journal Article] Genetic basis of calcifying cystic odontogenic tumors.2017

    • Author(s)
      Yukimori A, Oikawa Y, Morita K, Nguyen CTK, Harada H, Yamaguchi, S, Kayamori K, Yamaguchi A, Ikeda T, Sakamoto K
    • Journal Title

      PLos One

      Volume: 12 Issue: 6 Pages: e0180224-e0180224

    • DOI

      10.1371/journal.pone.0180224

    • Related Report
      2017 Annual Research Report
    • Peer Reviewed / Open Access
  • [Journal Article] Practical whole-tooth restoration utilizing autologous bioengineered tooth germ transplantation in a postnatal canine model.2017

    • Author(s)
      Ono M, Oshima M, Ogawa M, Sonoyama W, Hara ES, Oida Y, Shinkawa S, Nakajima R , Mine A ,Hayano S, Fukumoto S , Kasugai8 S , Yamaguchi A, Tsuji T, Kuboki T
    • Journal Title

      Sci Rep

      Volume: 7 Issue: 1 Pages: 44522-44522

    • DOI

      10.1038/srep44522

    • NAID

      120006535050

    • Related Report
      2017 Annual Research Report 2016 Research-status Report
    • Peer Reviewed / Open Access
  • [Journal Article] The Bone Regeneration Model and Primary Osteoblastic Cell Culture Used in the Analysis of Ccn3 Transgenic and Knockout Mice2017

    • Author(s)
      Sakamoto K, Matsushita Y, Minamizato T, Katsuki Y, Katsube KI, Yamaguchi A
    • Journal Title

      Methods Mol Biol

      Volume: 1489 Pages: 309-324

    • Related Report
      2017 Annual Research Report 2016 Research-status Report
    • Peer Reviewed
  • [Journal Article] LAMC2 is a predictive marker for malignant progression of leukoplakia.2017

    • Author(s)
      Nguyen CTK, Okamura T, Morita K, Miki Y, Izumo T, Kayamori K, Yamaguchi A, Sakamoto K
    • Journal Title

      J Oral Pathol Med

      Volume: 46 Issue: 3 Pages: 223-231

    • DOI

      10.1111/jop.12485

    • Related Report
      2017 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Parathyroid hormone facilitates osteogenesis differently in cancellous and cortical bones.2016

    • Author(s)
      Ogura K, Iimura T, Makino Y, Sugie-Oya A, Shimomura A, Takao-Kawabata R, Ishizuya T, Moriyama K, Yamaguchi A
    • Journal Title

      Bone Reports

      Volume: 5 Pages: 7-14

    • DOI

      10.1016/j.bonr.2016.01.002

    • Related Report
      2016 Research-status Report
    • Peer Reviewed / Open Access
  • [Journal Article] NOTCH3 Is Induced in Cancer-Associated Fibroblasts and Promotes Angiogenesis in Oral Squamous Cell Carcinoma2016

    • Author(s)
      Kayamori K, Katsube K, Sakamoto K, Ohyama Y, Hirai H, Yukimori A, Ohata Y, Akashi T, Saitoh M, Harada K, Harada H, Yamaguchi A
    • Journal Title

      PLoS ONE

      Volume: 11 Issue: 4 Pages: 223-231

    • DOI

      10.1371/journal.pone.0154112

    • Related Report
      2016 Research-status Report
    • Peer Reviewed / Open Access / Acknowledgement Compliant
  • [Journal Article] KRT17 is induced in oral cancer and facilitate tumor gowth.2016

    • Author(s)
      Khanom R, Nguyen CTK, Kayamori K, Zhao X, Morita K, Yamaguchi A, Sakamoto K
    • Journal Title

      PlosOne

      Volume: 11 Issue: 8 Pages: e0161163-e0161163

    • DOI

      10.1371/journal.pone.0161163

    • Related Report
      2016 Research-status Report
    • Peer Reviewed / Open Access / Int'l Joint Research
  • [Journal Article] THBS1 is induced by TGFB1 in the cancer stroma and promotes invasion of oral squamous cell carcinoma.2016

    • Author(s)
      Pal SK, Nguyen CTK, Morita, K, Miki Y, Kayamori K, Yamaguchi A, Sakamoto K
    • Journal Title

      J Oral Pathol Med

      Volume: 45 Issue: 10 Pages: 730-739

    • DOI

      10.1111/jop.12430

    • Related Report
      2016 Research-status Report
    • Peer Reviewed / Open Access
  • [Journal Article] A ficile one-step strategy for the egeneration of conditional knockout mice to explore the role of Notch1 in oroesophageal tumrigenesis2016

    • Author(s)
      Mandasari M, Sawangarun W, Ktsube K, Kayamori K, Yamaguchi A, Sakamoto K
    • Journal Title

      Biochem Bioph Res Co

      Volume: 469 Pages: 61-767

    • Related Report
      2016 Research-status Report
    • Peer Reviewed / Open Access
  • [Presentation] THの骨形成促進作用における基礎研究の展開2017

    • Author(s)
      山口 朗
    • Organizer
      第7回骨・軟骨フロンティア
    • Related Report
      2017 Annual Research Report
    • Invited
  • [Presentation] iPS 細胞を用いた顎骨疾患病態解明へのアプローチ2017

    • Author(s)
      東 俊文、小野寺晶、齋藤 暁子、中村 貴、大庭 伸介、小崎健次郎、 山口 朗、柴原 孝彦
    • Organizer
      第59回歯科基礎医学会総会学術大会アップデートシンポジウム
    • Related Report
      2017 Annual Research Report
    • Invited
  • [Presentation] ステオネットワークの構築・維持・破綻:顎顔面骨疾患の病態解析の基盤構築(宿題報告)2016

    • Author(s)
      山口 朗
    • Organizer
      第70回NPO法人日本口腔科学会学術集会
    • Place of Presentation
      広島市
    • Related Report
      2016 Research-status Report
    • Invited
  • [Presentation] 歯学研究における基礎と臨床のシグナル伝達2016

    • Author(s)
      山口 朗
    • Organizer
      第58回歯科基礎医学会学術大会日本学術会議シンポジウム
    • Place of Presentation
      札幌市
    • Related Report
      2016 Research-status Report
    • Invited
  • [Book] 最新口腔外科学2017

    • Author(s)
      山口 朗、栢森高
    • Publisher
      医歯薬出版株式会社
    • Related Report
      2017 Annual Research Report

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Published: 2016-04-21   Modified: 2019-03-29  

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