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Discovery of Vertebrate-Specific Factor that Processes Flagellar Glycolytic Enolase During Motile Ciliogenesis

38 Pages Posted: 9 Sep 2019 Sneak Peek Status: Review Complete

See all articles by Keishi Narita

Keishi Narita

University of Yamanashi

Hiroaki Nagatomo

University of Yamanashi

Hiroko Kozuka-Hata

University of Tokyo - Medical Proteomics Laboratory

Masaaki Oyama

University of Tokyo - Medical Proteomics Laboratory

Sen Takeda

University of Yamanashi - Department of Anatomy and Cell Biology

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Abstract

Motile cilia and flagella require ATP for their formation and function. Although glycolytic enzymes have been found in flagellar proteomes, it remains unknown how they translocate into these motile organelles. Here, we characterized Hoatzin, a functionally non-annotated gene found only in vertebrates. Their expression pattern suggested a functional association with motile cilia and flagella. The knockout mouse developed hydrocephalus and male infertility in an autosomal recessive manner. In the null mutant, the ependymal cilia frequently showed disorganized axoneme, causing reduced motility, and the spermatid flagella collapsed during cytodifferentiation. Interestingly, HOATZ was found to be associated with various proteins, including a flagellar glycolytic enzyme, ENO4. In the null mutant testis, the immature form of ENO4 accumulated in abnormal cytoplasmic puncta of developing spermatids. Overall, these data indicate that HOATZ is a cytoplasmic factor that plays a critical role in motile ciliogenesis and flagellogenesis in vertebrates by mediating the maturation of ENO4.

Keywords: motile cilia, flagella, enolase, male infertility, Hydrocephalus

Suggested Citation

Narita, Keishi and Nagatomo, Hiroaki and Kozuka-Hata, Hiroko and Oyama, Masaaki and Takeda, Sen, Discovery of Vertebrate-Specific Factor that Processes Flagellar Glycolytic Enolase During Motile Ciliogenesis (September 6, 2019). ISCIENCE-D-19-00918. Available at SSRN: https://ssrn.com/abstract=3449276 or http://dx.doi.org/10.2139/ssrn.3449276
This is a paper under consideration at Cell Press and has not been peer-reviewed.

Keishi Narita (Contact Author)

University of Yamanashi

Hiroaki Nagatomo

University of Yamanashi ( email )

Yamanashi
Japan

Hiroko Kozuka-Hata

University of Tokyo - Medical Proteomics Laboratory ( email )

4-6-1 Shirokanedai
Minato-ku
Tokyo, 108-8639
Japan

Masaaki Oyama

University of Tokyo - Medical Proteomics Laboratory ( email )

4-6-1 Shirokanedai
Minato-ku
Tokyo, 108-8639
Japan

Sen Takeda

University of Yamanashi - Department of Anatomy and Cell Biology ( email )

Japan

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