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Role of Daple in the inner ear morphogenesis

Research Project

Project/Area Number 18K16887
Research Category

Grant-in-Aid for Early-Career Scientists

Allocation TypeMulti-year Fund
Review Section Basic Section 56050:Otorhinolaryngology-related
Research InstitutionOsaka University

Principal Investigator

Ozono Yoshiyuki  大阪大学, 医学系研究科, 特任研究員 (10724768)

Project Period (FY) 2018-04-01 – 2022-03-31
Project Status Completed (Fiscal Year 2021)
Budget Amount *help
¥4,160,000 (Direct Cost: ¥3,200,000、Indirect Cost: ¥960,000)
Fiscal Year 2019: ¥2,080,000 (Direct Cost: ¥1,600,000、Indirect Cost: ¥480,000)
Fiscal Year 2018: ¥2,080,000 (Direct Cost: ¥1,600,000、Indirect Cost: ¥480,000)
Keywords内耳 / 平面細胞極性 / 細胞骨格 / 難聴 / 発生 / 蝸牛
Outline of Final Research Achievements

The V-shaped arrangement of hair bundles on cochlear hair cells is critical for auditory sensing. However, regulation of hair bundle arrangements has not been fully understood. In the present study, we found that Daple(Ccdc88c)-/- mice exhibited hearing disturbances over a broad frequency range through auditory brainstem response testing. Consistently, distorted patterns of hair bundles were detected in almost all regions, more typically in the basal region of the cochlear duct. In Daple-/- mice, apical microtubules were irregularly aggregated, and the number of microtubules attached to plasma membranes was decreased. Similar phenotypes were manifested upon nocodazole treatment in a wild type cochlea culture without affecting the microtubule structure of the kinocilium. These results indicate critical role of Daple in hair bundle arrangement through the orchestration of apical microtubule distribution, and thereby in hearing.

Academic Significance and Societal Importance of the Research Achievements

胚の発生、器官形成、生体恒常性維持には、細胞内、細胞間の極性を正しく決めるメカニズムが重要である(平面細胞極性planar cell polarity :PCP)。本研究は蝸牛のPCPにおけるDaple(Ccdc88c)の役割を明らかにし、蝸牛PCP異常下での聴覚障害や、生体におけるPCP調整のメカニズムを解明する一助になる成果を報告した。

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

    (2 results)

All 2021 2019

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

  • [Journal Article] Daple deficiency causes hearing loss in adult mice by inducing defects in cochlear stereocilia and apical microtubules2021

    • Author(s)
      Ozono Yoshiyuki、Tamura Atsushi、Nakayama Shogo、Herawati Elisa、Hanada Yukiko、Ohata Kazuya、Takagishi Maki、Takahashi Masahide、Imai Takao、Ohta Yumi、Oshima Kazuo、Sato Takashi、Inohara Hidenori、Tsukita Sachiko
    • Journal Title

      Scientific Reports

      Volume: 11 Issue: 1 Pages: 20224-20224

    • DOI

      10.1038/s41598-021-96232-8

    • Related Report
      2021 Annual Research Report
    • Peer Reviewed / Open Access
  • [Presentation] Dapleの欠損は平面細胞極性と上皮面細胞骨格の構築に異常を示し、難聴をもたらす2019

    • Author(s)
      大薗 芳之、森鼻 哲生、猪原 秀典
    • Organizer
      第29回日本耳科学会 総会・学術講演会
    • Related Report
      2019 Research-status Report

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

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