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Development of highly precise diagnostic markers for pediatric neoplasms

Research Project

Project/Area Number 15K10931
Research Category

Grant-in-Aid for Scientific Research (C)

Allocation TypeMulti-year Fund
Section一般
Research Field Pediatric surgery
Research InstitutionKanagawa Children's Medical Center (Clinical Research Institute)

Principal Investigator

Tanaka Mio  地方独立行政法人神奈川県立病院機構神奈川県立こども医療センター(臨床研究所), 臨床研究所, 医長 (60565232)

Co-Investigator(Kenkyū-buntansha) 永原 則之  日本医科大学, 医学部, 准教授 (10208043)
片山 映  日本医科大学, 医学部, 助教 (10333113)
田中 祐吉  地方独立行政法人神奈川県立病院機構神奈川県立こども医療センター(臨床研究所), 臨床研究所, 臨床研究所長 (50420691)
Research Collaborator GOTO hiroaki  
KITAGAWA norihiko  
Project Period (FY) 2015-10-21 – 2019-03-31
Project Status Completed (Fiscal Year 2018)
Budget Amount *help
¥4,680,000 (Direct Cost: ¥3,600,000、Indirect Cost: ¥1,080,000)
Fiscal Year 2017: ¥1,430,000 (Direct Cost: ¥1,100,000、Indirect Cost: ¥330,000)
Fiscal Year 2016: ¥1,690,000 (Direct Cost: ¥1,300,000、Indirect Cost: ¥390,000)
Fiscal Year 2015: ¥1,560,000 (Direct Cost: ¥1,200,000、Indirect Cost: ¥360,000)
Keywords小児腫瘍 / 診断マーカー / 病理診断 / 融合遺伝子 / 免疫組織学的検査 / 免疫組織学的 / 腎明細胞肉腫 / alpha-internexin / ALK融合遺伝子 / 炎症性筋線維芽細胞性腫瘍 / 先天性嚢胞性腺腫様奇形 / FLIT / internexin alpha
Outline of Final Research Achievements

The purpose of this study was to develop highly precise diagnostic markers of pediatric neoplasms, especially renal and pulmonary tumors, for pathological diagnosis. For renal tumor, we have detected alpha-internexin (INA) as a candidate for diagnostic marker of clear cell sarcoma of the kidney (CCSK), which is one of common malignant pediatric renal tumor. In this study, we showed high sensitivity and specificity of INA as a marker for CCSK among other pediatric renal and extrarenal tumors. BCOR-ITD is recently reported gene abnormality which is often detected in CCSKs. We achieved detection of BCOR-ITD from formalin-fixed-paraffin-embedded tissue of CCSK, some of which were stored over 40 years. For pulmonary tumors, we detected ALK-related fusion gene from several infantile pulmonary lesions, and established new subtype of inflammatory myofibroblastic tumor occurring specifically in neonatal/ infantile lung.

Academic Significance and Societal Importance of the Research Achievements

Alpha-internexinは市販の抗体を用いた免疫染色が可能であり、しばしば病理診断が困難である腎明細胞肉腫に対して簡便で特異性の高いマーカーが見出されたことの意義は大きい。また、BCOR-ITDを相当長期の保存期間を経た検体からも検出可能であることを示し得たことは、同腫瘍のアーカイブを用いた検討の可能性を広げたという点で重要な意義がある。乳児型肺炎症性筋線維芽細胞性腫瘍の組織所見と遺伝子異常を明らかにしたことは、診断不能とされていた新生児・乳児の肺病変の一部が同症と診断されることで適切な治療法の選択がなされるという点で意義がある。

Report

(5 results)
  • 2018 Annual Research Report   Final Research Report ( PDF )
  • 2017 Research-status Report
  • 2016 Research-status Report
  • 2015 Research-status Report
  • Research Products

    (7 results)

All 2018 2017 2016 2015

All Journal Article (3 results) (of which Int'l Joint Research: 1 results,  Peer Reviewed: 3 results,  Open Access: 2 results) Presentation (3 results) (of which Invited: 1 results) Book (1 results)

  • [Journal Article] Outcome of renal tumors registered in Japan Wilms Tumor Study-2 (JWiTS-2): A report from the Japan Children's Cancer Group (JCCG)2018

    • Author(s)
      Koshinaga Tsugumichi、Takimoto Tetsuya、Oue Takaharu、Okita Hajime、Tanaka Yukichi、Nozaki Miwako、Tsuchiya Kunihiko、Inoue Eisuke、Haruta Masayuki、Kaneko Yasuhiko、Fukuzawa Masahiro
    • Journal Title

      Pediatric Blood & Cancer

      Volume: 65 Issue: 7

    • DOI

      10.1002/pbc.27056

    • Related Report
      2018 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Inflammatory myofibroblastic tumors of the lung carrying a chimeric A2M-ALK gene: report of 2 infantile cases and review of the differential diagnosis of infantile pulmonary lesions2017

    • Author(s)
      Tanaka Mio、Kohashi Kenichi、Kushitani Kei、Yoshida Misa、Kurihara Sho、Kawashima Masumi、Ueda Yuka、Souzaki Ryota、Kinoshita Yoshiaki、Oda Yoshinao、Takeshima Yukio、Hiyama Eiso、Taguchi Tomoaki、Tanaka Yukichi
    • Journal Title

      Human Pathology

      Volume: 66 Pages: 177-182

    • DOI

      10.1016/j.humpath.2017.06.013

    • Related Report
      2017 Research-status Report
    • Peer Reviewed / Open Access
  • [Journal Article] The clinical phenotype of YWHAE-NUTM2B/E positive pediatric clear cell sarcoma of the kidney.2016

    • Author(s)
      Gooskens SL, Kenny C, Lazaro A, O'Meara E, van Tinteren H, Spreafico F, Vujanic G, Leuschner I, Coulomb-L'Hermine A, Perotti D, de Camargo B, Bergeron C, Acha Garcia T, Tanaka M, Pieters R, Pritchard-Jones K, Graf N, van den Heuvel-Eibrink MM, O'Sullivan MJ.
    • Journal Title

      Genes Chromosomes Cancer.

      Volume: 55 Issue: 2 Pages: 143-147

    • DOI

      10.1002/gcc.22320

    • Related Report
      2016 Research-status Report
    • Peer Reviewed / Open Access / Int'l Joint Research
  • [Presentation] DICER1症候群の概要と最近の知見2018

    • Author(s)
      田中水緒、吉田美沙、田中祐吉
    • Organizer
      日本病理学会
    • Related Report
      2018 Annual Research Report
    • Invited
  • [Presentation] 小児腎腫瘍の捺印標本における腎明細胞肉腫とその他の組織型との比較検討2018

    • Author(s)
      関谷元幹、長谷川由香里、田中水緒、田中祐吉
    • Organizer
      日本臨床細胞学会
    • Related Report
      2018 Annual Research Report
  • [Presentation] 甲状腺濾胞癌の発生を契機に生殖細胞のDICER1遺伝子異常を同定した2症例2015

    • Author(s)
      吉田 美沙, 村岡 枝里香, 関 正史, 吉田 健一, 浜之上 聡, 武山 絵里子, 田中 水緒, 後藤 裕明, 宮野 悟, 小川 誠司, 滝田 順子, 田中 祐吉
    • Organizer
      第57回日本小児血液・がん学会学術集会
    • Place of Presentation
      甲府富士屋ホテル、甲府市
    • Year and Date
      2015-11-27
    • Related Report
      2015 Research-status Report
  • [Book] 小児腫瘍組織カラーアトラス 第7巻2017

    • Author(s)
      日本病理学会小児腫瘍組織分類委員会
    • Total Pages
      128
    • Publisher
      金原出版
    • ISBN
      9784307170710
    • Related Report
      2017 Research-status Report

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Published: 2015-10-21   Modified: 2020-03-30  

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