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Searching for new diagnostic markers for Hirschsprung disease using tissue clearing methods

Research Project

Project/Area Number 18K08601
Research Category

Grant-in-Aid for Scientific Research (C)

Allocation TypeMulti-year Fund
Section一般
Review Section Basic Section 55010:General surgery and pediatric surgery-related
Research InstitutionJuntendo University

Principal Investigator

Miyahara Katsumi  順天堂大学, 大学院医学研究科, 技術員 (00420844)

Co-Investigator(Kenkyū-buntansha) 藤原 なほ  順天堂大学, 医学部, 准教授 (20589543)
山高 篤行  順天堂大学, 医学部, 教授 (40200703)
田中 奈々  順天堂大学, 医学部, 准教授 (50530656)
Project Period (FY) 2018-04-01 – 2022-03-31
Project Status Completed (Fiscal Year 2021)
Budget Amount *help
¥4,420,000 (Direct Cost: ¥3,400,000、Indirect Cost: ¥1,020,000)
Fiscal Year 2021: ¥1,300,000 (Direct Cost: ¥1,000,000、Indirect Cost: ¥300,000)
Fiscal Year 2020: ¥1,300,000 (Direct Cost: ¥1,000,000、Indirect Cost: ¥300,000)
Fiscal Year 2019: ¥910,000 (Direct Cost: ¥700,000、Indirect Cost: ¥210,000)
Fiscal Year 2018: ¥910,000 (Direct Cost: ¥700,000、Indirect Cost: ¥210,000)
Keywordsヒルシュスプルング病 / 腸管神経系 / 組織透明化 / 蛍光イメージング / エンドセリンB受容体 / 神経堤細胞 / 疾患モデルマウス / 透明化組織イメージング / バイオマーカー / モデルマウス
Outline of Final Research Achievements

We revealed that the decrease of the molecule related of the stability and the development process of blood vessel formation in the gut of Hirschsprung disease model mice. It was suggested that the responsible gene of this disease may cause both abnormal development of vascular structure as well as abnormal development of enteric nervous system. This result may provide that the new understanding of pathogenesis and the new pathological diagnosis for Hirschsprung disease.

Academic Significance and Societal Importance of the Research Achievements

ヒルシュスプルング病の確定診断の際に診断に難渋するケースにしばしば遭遇し、また根治には外科的治療が必要であり、その治療と診断のために新しい発生病態を解明することは重要である。
今回の結果で可能性が示された原因遺伝子により神経系の発生の異常とともに血管の形成の異常が引き起こされる可能性は新しい発生機序の解明になるとともに、術後の合併症の原因ともなりえると考えており、本疾患の新しい病態の理解と診断のための一助となることが期待される。

Report

(5 results)
  • 2021 Annual Research Report   Final Research Report ( PDF )
  • 2020 Research-status Report
  • 2019 Research-status Report
  • 2018 Research-status Report
  • Research Products

    (9 results)

All 2022 2021 2020 2019 2018

All Journal Article (8 results) (of which Int'l Joint Research: 1 results,  Peer Reviewed: 8 results,  Open Access: 4 results) Presentation (1 results)

  • [Journal Article] In vitro investigation of the differentiation of enteric neural crest-derived cells following transplantation of aganglionic gut in a mouse model2022

    • Author(s)
      Fujiwara Naho、Miyahara Katsumi、Nakazawa-Tanaka Nana、Akazawa Chihiro、Yamataka Atsuyuki
    • Journal Title

      Pediatric Surgery International

      Volume: 38 Issue: 5 Pages: 755-759

    • DOI

      10.1007/s00383-022-05105-2

    • Related Report
      2021 Annual Research Report
    • Peer Reviewed
  • [Journal Article] Influence of aging on the peripheral nerve repair process using an artificial nerve conduit2021

    • Author(s)
      Kaneko Ayaka、Naito Kiyohito、Nakamura Shinji、Miyahara Katsumi、Goto Kenji、Obata Hiroyuki、Nagura Nana、Sugiyama Yoichi、Kaneko Kazuo、Ishijima Muneaki
    • Journal Title

      Experimental and Therapeutic Medicine

      Volume: 21 Issue: 2

    • DOI

      10.3892/etm.2020.9599

    • Related Report
      2021 Annual Research Report
    • Peer Reviewed
  • [Journal Article] DPP4 inhibitor reinforces cell junction proteins in mouse model of short bowel syndrome.2020

    • Author(s)
      Sueyoshi R, Miyahara K, Nakazawa-Tanaka N, Fujiwara N, Ochi T, Yamataka A.
    • Journal Title

      pediatric surgery international

      Volume: 36(1) Issue: 1 Pages: 49-55

    • DOI

      10.1007/s00383-019-04571-5

    • Related Report
      2019 Research-status Report
    • Peer Reviewed / Open Access
  • [Journal Article] Decreased expression of β1 integrin in enteric neural crest cells of the endothelin receptor B null mouse model.2020

    • Author(s)
      Nakazawa-Tanaka N, Miyahara K, Fujiwara N, Ochi T, Sueyoshi R, Nojiri S, Akazawa C, Urao M, Yamataka A.
    • Journal Title

      pediatric surgery international

      Volume: 36(1) Issue: 1 Pages: 43-48

    • DOI

      10.1007/s00383-019-04578-y

    • Related Report
      2019 Research-status Report
    • Peer Reviewed / Open Access
  • [Journal Article] Endothelin receptor B affects the perfusion of newborn intestine: possible mechanism of necrotizing enterocolitis development2019

    • Author(s)
      Miyake H, Seo S, Fujiwara N, Miyahara K, Lee C, Li B, Chen Y, Yamataka A, Pierro A.
    • Journal Title

      pediatric surgery international

      Volume: 35(12) Issue: 12 Pages: 1339-1343

    • DOI

      10.1007/s00383-019-04559-1

    • Related Report
      2019 Research-status Report
    • Peer Reviewed / Open Access / Int'l Joint Research
  • [Journal Article] Role of FBXW7 in the quiescence of gefitinib-resistant lung cancer stem cells in EGFR-mutant non-small cell lung cancer2019

    • Author(s)
      Hidayat M, Mitsuishi Y, Takahashi F, Tajima K, Yae T, Miyahara K, Hayakawa D, Winardi W, Ihara H, Koinuma Y, Wirawan A, Nurwidya F, Kato M, Kobayashi I, Sasaki S, Takamochi K, Hayashi T, Suehara Y, Moriyama M, Moriyama H, Habu S, Takahashi K.
    • Journal Title

      Bosnian Journal of Basic Medical Sciences

      Volume: 8;19(4) Pages: 355-367

    • DOI

      10.17305/bjbms.2019.4227

    • Related Report
      2019 Research-status Report
    • Peer Reviewed / Open Access
  • [Journal Article] Innervation of the entire internal anal sphincter in a mouse model of Hirschsprung's disease: a first report.2019

    • Author(s)
      Takeda M, Miyahara K, Sueyoshi R, Arakawa A, Lane GJ, Yamataka A.
    • Journal Title

      Pediatric Surgery International

      Volume: 35(2) Issue: 2 Pages: 209-214

    • DOI

      10.1007/s00383-018-4397-z

    • Related Report
      2018 Research-status Report
    • Peer Reviewed
  • [Journal Article] Compromised vitality of spermatozoa after contact with colonic mucosa in mice: implications for fertility in colon vaginoplasty patients.2019

    • Author(s)
      Kosaka S, Takeda M, Ochi T, Miyahara K, Nakamura E, Tada N, Lane GJ, Yamataka A.
    • Journal Title

      Pediatric Surgery International

      Volume: 35(1) Issue: 1 Pages: 71-75

    • DOI

      10.1007/s00383-018-4377-3

    • Related Report
      2018 Research-status Report
    • Peer Reviewed
  • [Presentation] ヒルシュスプルング病モデルマウス腸管神経細胞におけるアセチル化チューブリンの発現異常2018

    • Author(s)
      宮原 克, 田中 奈々, 藤原 なほ, 栗原 秀剛, 山高 篤行
    • Organizer
      第107回日本病理学会総会
    • Related Report
      2018 Research-status Report

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Published: 2018-04-23   Modified: 2023-01-30  

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