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Study of motile ciliogenesis in vertebrates using Hoatzin knockout mouse

Research Project

Project/Area Number 18K06824
Research Category

Grant-in-Aid for Scientific Research (C)

Allocation TypeMulti-year Fund
Section一般
Review Section Basic Section 48010:Anatomy-related
Research InstitutionUniversity of Yamanashi

Principal Investigator

Narita Keishi  山梨大学, 大学院総合研究部, 准教授 (50452131)

Project Period (FY) 2018-04-01 – 2021-03-31
Project Status Completed (Fiscal Year 2020)
Budget Amount *help
¥4,290,000 (Direct Cost: ¥3,300,000、Indirect Cost: ¥990,000)
Fiscal Year 2020: ¥1,430,000 (Direct Cost: ¥1,100,000、Indirect Cost: ¥330,000)
Fiscal Year 2019: ¥1,430,000 (Direct Cost: ¥1,100,000、Indirect Cost: ¥330,000)
Fiscal Year 2018: ¥1,430,000 (Direct Cost: ¥1,100,000、Indirect Cost: ¥330,000)
Keywords運動繊毛 / 上衣細胞 / RNA-seq / 多繊毛形成 / 繊毛 / 解糖系 / エノラーゼ / ATP / 分娩停止 / 不妊症 / 水頭症
Outline of Final Research Achievements

To understand the molecular function of HOATZIN, a novel protein involved in motile ciliogenesis in vertebrates, HOATZIN-interacting proteins were enriched from testis lysate by immunoprecipitation and identified by shotgun proteomics. The identified molecules included tubulins, heat shock proteins, and glycolytic enzymes. The subsequent analyses suggested that HOATZIN might function as a molecular chaperone to process ENO4, one of the glycolytic enzymes, into mature form.
Transmission electron microscopy of immature multiciliated epithelia from wild-type mouse trachea, oviduct and brain ventricle, strongly suggested that cytosolic ciliogenesis, an “extraordinary” mode of ciliogenesis described only in elongating spermatids, takes place in these epithelia.
Characterization of mouse CFAP70 and its Chlamydomonas homolog FAP70 revealed that it is a novel axoneme-binding protein that localizes at the base of the outer dynein arm and regulates ciliary motility.

Academic Significance and Societal Importance of the Research Achievements

これらの研究成果の学術的意義は脳の恒常性維持、呼吸器の免疫機能、そして生殖に極めて重要な役割を果たしている運動繊毛がどのようにして作られ機能しているかについて、その一端を明らかにしたという点にある。社会的意義としては運動繊毛の異常が原因で生じる水頭症や呼吸器感染症、そして不妊の原因解明と根本的治療法開発につながるという点が挙げられる。

Report

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

    (11 results)

All 2021 2020 2019 2018

All Journal Article (7 results) (of which Int'l Joint Research: 1 results,  Peer Reviewed: 6 results,  Open Access: 5 results) Presentation (4 results) (of which Int'l Joint Research: 1 results)

  • [Journal Article] Ultrastructural evidence for an unusual mode of ciliogenesis in mouse multiciliated epithelia2020

    • Author(s)
      Narita Keishi、Takeda Sen
    • Journal Title

      Microscopy

      Volume: 70 Issue: 3 Pages: 308

    • DOI

      10.1093/jmicro/dfaa074

    • Related Report
      2020 Annual Research Report
    • Peer Reviewed / Open Access
  • [Journal Article] Discovery of a Vertebrate-Specific Factor that Processes Flagellar Glycolytic Enolase during Motile Ciliogenesis2020

    • Author(s)
      Narita Keishi、Nagatomo Hiroaki、Kozuka-Hata Hiroko、Oyama Masaaki、Takeda Sen
    • Journal Title

      iScience

      Volume: 23 Issue: 4 Pages: 100992-100992

    • DOI

      10.1016/j.isci.2020.100992

    • Related Report
      2019 Research-status Report
    • Peer Reviewed / Open Access
  • [Journal Article] Hypercholesterolemia negatively influences morphology and molecular markers of epithelial cells within the choroid plexus in rabbits2020

    • Author(s)
      Obata Fumiko、Narita Keishi
    • Journal Title

      Fluids and Barriers of the CNS

      Volume: 17 Issue: 1 Pages: 13-13

    • DOI

      10.1186/s12987-020-0175-0

    • Related Report
      2019 Research-status Report
    • Peer Reviewed / Open Access
  • [Journal Article] Dynamic changes in ultrastructure of the primary cilium in migrating neuroblasts in the postnatal brain2019

    • Author(s)
      Matsumoto Mami、Sawada Masato、Garcia-Gonzalez Diego、Herranz-Perez Vicente、Ogino Takashi、Bang Nguyen Huy、Quynh Thai Truc、Narita Keishi、Kumamoto Natsuko、Ugawa Shinya、Saito Yumiko、Takeda Sen、Kaneko Naoko、Khodosevich Konstantin、Monyer Hannah、Garcia-Verdugo Jose Manuel、Ohno Nobuhiko、Sawamoto Kazunobu
    • Journal Title

      The Journal of Neuroscience

      Volume: 39 Issue: 50 Pages: 9967

    • DOI

      10.1523/jneurosci.1503-19.2019

    • Related Report
      2019 Research-status Report
    • Peer Reviewed / Open Access / Int'l Joint Research
  • [Journal Article] Arima syndrome caused by CEP290 specific variant and accompanied with pathological cilium; clinical comparison with Joubert syndrome and its related diseases2018

    • Author(s)
      Itoh M, Ide S, Iwasaki Y, Saito T, Narita K, Dai H, Yamakura S, Furue T, Kitayama H, Maeda K, Takahashi E, Matsui K, Goto YI, Takeda S, Arima M
    • Journal Title

      Brain & development

      Volume: 40 Issue: 4 Pages: 259

    • DOI

      10.1016/j.braindev.2017.11.002

    • Related Report
      2018 Research-status Report
    • Peer Reviewed
  • [Journal Article] CFAP70 is a novel axoneme-binding protein that localizes at the base of the outer dynein arm and regulates ciliary motility2018

    • Author(s)
      Shamoto N, Narita K. Kubo T, Oda T, Takeda S.
    • Journal Title

      Cells

      Volume: 7 Issue: 9 Pages: 1-12

    • DOI

      10.3390/cells7090124

    • Related Report
      2018 Research-status Report
    • Peer Reviewed / Open Access
  • [Journal Article] Transport Across the Choroid Plexus: How to Culture Choroid Plexus Cells and Establish a Functional Assay System2018

    • Author(s)
      Takeda Sen、Narita Keishi
    • Journal Title

      Neuromethods

      Volume: 142 Pages: 163-173

    • DOI

      10.1007/978-1-4939-8946-1_9

    • ISBN
      9781493989454, 9781493989461
    • Related Report
      2018 Research-status Report
  • [Presentation] Ultrastructural evidence for an unusual mode of ciliogenesis in mouse multiciliated epithelia2021

    • Author(s)
      Narita Keishi
    • Organizer
      第126回 日本解剖学会
    • Related Report
      2020 Annual Research Report
  • [Presentation] Hoatzin is a vertebrate-specific gene involved in motile ciliogenesis by mediating the processing of flagellar glycolytic enzyme2019

    • Author(s)
      Keishi Narita, Hiroaki Nagatomo, Hiroko Kozuka-Hata, Masaaki Oyama, and Sen Takeda
    • Organizer
      Cold Spring Harbor Asia meeting
    • Related Report
      2019 Research-status Report
    • Int'l Joint Research
  • [Presentation] Functional characterization of a novel cilia-related gene, Hoatzin, unveils the presence of distinct, tissue-specific mechanisms for motile ciliogenesis2018

    • Author(s)
      Keishi Narita, Hiroaki Nagatomo, Sen Takeda
    • Organizer
      日本細胞生物学会
    • Related Report
      2018 Research-status Report
  • [Presentation] 新規外腕ダイニン調節分子CFAP70のドメイン解析2018

    • Author(s)
      成田 啓之, 社本 憲俊, 久保 智広, 小田 賢幸, 竹田 扇
    • Organizer
      日本解剖学会
    • Related Report
      2018 Research-status Report

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Published: 2018-04-23   Modified: 2022-01-27  

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