Nexin-Dynein regulatory complex component DRC7 but not FBXL13 is required for sperm flagellum formation and male fertility in mice

Morohoshi, Akane; Miyata, Haruhiko; Shimada, Keisuke; Nozawa, Kaori; Matsumura, Takafumi; Yanase, Ryuji; Shiba, Kogiku; Inaba, Kazuo; Ikawa, Masahito

doi:https://doi.org/10.1371/journal.pgen.1008585

Nexin-Dynein regulatory complex component DRC7 but not FBXL13 is required for sperm flagellum formation and male fertility in mice

Morohoshi, Akane; Miyata, Haruhiko; Shimada, Keisuke et al.

PLoS Genetics, 2020, 16(1), e1008585

アクセス数:261件（2025-04-18 02:10 集計）

固定URL: https://hdl.handle.net/11094/78552

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タイトル	Nexin-Dynein regulatory complex component DRC7 but not FBXL13 is required for sperm flagellum formation and male fertility in mice
著者	著者名 Morohoshi, Akane
	NRID 1000050604732 1000050604732 researchmap permalink hmiyata hmiyata Scopus Author ID 37161837000 37161837000 ORCID 0000-0003-4758-5803 0000-0003-4758-5803 著者名 Miyata, Haruhiko
	NRID 1000060779601 1000060779601 ORCID 0000-0003-3739-7163 0000-0003-3739-7163 researchmap permalink nununununu nununununu Scopus Author ID 56479808000 56479808000 著者名 Shimada, Keisuke
	著者名 Nozawa, Kaori
	著者名 Matsumura, Takafumi
	著者名 Yanase, Ryuji
	NRID 1000070533561 1000070533561 著者名 Shiba, Kogiku
	NRID 1000080221779 1000080221779 著者名 Inaba, Kazuo
	NRID 1000020304066 1000020304066 ORCID 0000-0001-9859-6217 0000-0001-9859-6217 researchmap permalink read0054910 read0054910 Scopus Author ID 16689054500 16689054500 著者名 Ikawa, Masahito
内容	Nexin-Dynein regulatory complex component DRC7 but not FBXL13 is required for sperm flagellum formation and male fertility in mice. Morohoshi A, Miyata H, Shimada K, Nozawa K, Matsumura T, et al. PLOS Genetics. 2020. 16(1) doi:10.1371/journal.pgen.1008585
抄録	Flagella and cilia are evolutionarily conserved cellular organelles. Abnormal formation or motility of these organelles in humans causes several syndromic diseases termed ciliopathies. The central component of flagella and cilia is the axoneme that is composed of the ‘9 +2’ microtubule arrangement, dynein arms, radial spokes, and the Nexin-Dynein Regulatory Complex (N-DRC). The N-DRC is localized between doublet microtubules and has been extensively studied in the unicellular flagellate Chlamydomonas. Recently, it has been reported that TCTE1 (DRC5), a component of the N-DRC, is essential for proper sperm motility and male fertility in mice. Further, TCTE1 has been shown to interact with FBXL13 (DRC6) and DRC7; however, functional roles of FBXL13 and DRC7 in mammals have not been elucidated. Here we show that Fbxl13 and Drc7 expression are testes-enriched in mice. Although Fbxl13 knockout (KO) mice did not show any obvious phenotypes, Drc7 KO male mice were infertile due to their short immotile spermatozoa. In Drc7 KO spermatids, the axoneme is disorganized and the ‘9+2’ microtubule arrangement was difficult to detect. Further, other N-DRC components fail to incorporate into the flagellum without DRC7. These results indicate that Drc7, but not Fbxl13, is essential for the correct assembly of the N-DRC and flagella.
出版者	Public Library of Science
収録物名	PLoS Genetics
巻(号)	16(1)
ページ	e1008585
刊行年月	2020-01-21
言語	英語
ハンドルURL	https://hdl.handle.net/11094/78552
PISSN	15537390
eISSN	15537404
NCID	AA12073492
関連情報 (isIdenticalTo)	DOI (出版社版) https://doi.org/10.1371/journal.pgen.1008585
関連情報 (isIdenticalTo)	PMID 31961863
アクセス権	open access
権利情報	© 2020 Morohoshi et al. This article is licensed under a Creative Commons Attribution 4.0 International License.
出版タイプ	Version of Record
カテゴリ	学術雑誌論文 Journal Article

資源タイプ	学術雑誌論文
ローカル資源タイプ	学術雑誌論文
DCTERMS.bibliographicCitation	PLoS Genetics. 2020, 16(1), p. e1008585
DC.title	Nexin-Dynein regulatory complex component DRC7 but not FBXL13 is required for sperm flagellum formation and male fertility in mice
DC.creator	Morohoshi, Akane
	Miyata, Haruhiko
	Shimada, Keisuke
	Nozawa, Kaori
	Matsumura, Takafumi
	Yanase, Ryuji
	Shiba, Kogiku
	Inaba, Kazuo
	Ikawa, Masahito
DC.publisher	Public Library of Science
DC.language' scheme='DCTERMS.RFC1766	英語
DCTERMS.issued' scheme='DCTERMS.W3CDTF	2020-01-21
DC.identifier	https://doi.org/10.1371/journal.pgen.1008585
DC.description	Nexin-Dynein regulatory complex component DRC7 but not FBXL13 is required for sperm flagellum formation and male fertility in mice. Morohoshi A, Miyata H, Shimada K, Nozawa K, Matsumura T, et al. PLOS Genetics. 2020. 16(1) doi:10.1371/journal.pgen.1008585
DCTERMS.abstract	Flagella and cilia are evolutionarily conserved cellular organelles. Abnormal formation or motility of these organelles in humans causes several syndromic diseases termed ciliopathies. The central component of flagella and cilia is the axoneme that is composed of the ‘9 +2’ microtubule arrangement, dynein arms, radial spokes, and the Nexin-Dynein Regulatory Complex (N-DRC). The N-DRC is localized between doublet microtubules and has been extensively studied in the unicellular flagellate Chlamydomonas. Recently, it has been reported that TCTE1 (DRC5), a component of the N-DRC, is essential for proper sperm motility and male fertility in mice. Further, TCTE1 has been shown to interact with FBXL13 (DRC6) and DRC7; however, functional roles of FBXL13 and DRC7 in mammals have not been elucidated. Here we show that Fbxl13 and Drc7 expression are testes-enriched in mice. Although Fbxl13 knockout (KO) mice did not show any obvious phenotypes, Drc7 KO male mice were infertile due to their short immotile spermatozoa. In Drc7 KO spermatids, the axoneme is disorganized and the ‘9+2’ microtubule arrangement was difficult to detect. Further, other N-DRC components fail to incorporate into the flagellum without DRC7. These results indicate that Drc7, but not Fbxl13, is essential for the correct assembly of the N-DRC and flagella.
DC.rights	© 2020 Morohoshi et al. This article is licensed under a Creative Commons Attribution 4.0 International License.
DC.format	78552
citation_title	Nexin-Dynein regulatory complex component DRC7 but not FBXL13 is required for sperm flagellum formation and male fertility in mice
citation_author	Morohoshi, Akane
	Miyata, Haruhiko
	Shimada, Keisuke
	Nozawa, Kaori
	Matsumura, Takafumi
	Yanase, Ryuji
	Shiba, Kogiku
	Inaba, Kazuo
	Ikawa, Masahito
citation_publisher	Public Library of Science
citation_language	英語
citation_date	2020-01-21
citation_journal_title	PLoS Genetics
citation_volume	16
citation_issue	1
citation_firstpage	e1008585
citation_doi	https://doi.org/10.1371/journal.pgen.1008585
citation_pmid	31961863
citation_public_url	https://hdl.handle.net/11094/78552

Nexin-Dynein regulatory complex component DRC7 but not FBXL13 is required for sperm flagellum formation and male fertility in mice

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