Study of motile ciliogenesis in vertebrates using Hoatzin knockout mouse

2020 Fiscal Year Final Research Report

• Project/Area Number: 18K06824
• Research Category: Grant-in-Aid for Scientific Research (C)
• Allocation Type: Multi-year Fund
• Section: 一般
• Review Section: Basic Section 48010:Anatomy-related
• Research Institution: University 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
• Keywords: 運動繊毛

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.

Free Research Field

細胞生物学

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。