RSPH6A is required for sperm flagellum formation and male fertility in mice

Abbasi, Ferheen; Miyata, Haruhiko; Shimada, Keisuke; Morohoshi, Akane; Nozawa, Kaori; Matsumura, Takafumi; Xu, Zoulan; Pratiwi, Putri; Ikawa, Masahito

doi:https://doi.org/10.1242/jcs.221648

RSPH6A is required for sperm flagellum formation and male fertility in mice

Abbasi, Ferheen; Miyata, Haruhiko; Shimada, Keisuke et al.

Journal of Cell Science, 2018, 131(19), jcs.221648

アクセス数:180件（2025-04-18 01:59 集計）

固定URL: https://hdl.handle.net/11094/78569

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タイトル	RSPH6A is required for sperm flagellum formation and male fertility in mice
著者	著者名 Abbasi, Ferheen
	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 Morohoshi, Akane
	著者名 Nozawa, Kaori
	著者名 Matsumura, Takafumi
	著者名 Xu, Zoulan
	著者名 Pratiwi, Putri
	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
キーワード等	Axoneme
	CRISPR/Cas9
	Flagella
	Radial spoke protein
	Spermatozoa
抄録	The flagellum is an evolutionarily conserved appendage used for sensing and locomotion. Its backbone is the axoneme and a component of the axoneme is the radial spoke (RS), a protein complex implicated in flagellar motility regulation. Numerous diseases occur if the axoneme is improperly formed, such as primary ciliary dyskinesia (PCD) and infertility. Radial spoke head 6 homolog A (RSPH6A) is an ortholog of Chlamydomonas RSP6 in the RS head and is evolutionarily conserved. While some RS head proteins have been linkedtoPCD, littleisknown about RSPH6A. Here, weshow that mouse RSPH6A is testis-enriched and localized in the flagellum. Rsph6a knockout (KO) male mice are infertile as a result of their short immotile spermatozoa. Observation of the KO testis indicates that the axoneme can elongate but is disrupted before accessory structures are formed. Manchette removal is also impaired in the KO testis. Further, RSPH9, another radial spoke protein, disappeared in the Rsph6a KO flagella. These data indicate that RSPH6A is essential for sperm flagellar assembly and male fertility in mice. This article has an associated First Person interview with the first author of the paper.
出版者	Company of Biologists Ltd
収録物名	Journal of Cell Science
巻(号)	131(19)
ページ	jcs.221648
刊行年月	2018-10-11
言語	英語
ハンドルURL	https://hdl.handle.net/11094/78569
PISSN	00219533
eISSN	14779137
NCID	AA11720335
関連情報 (isIdenticalTo)	DOI (出版社版) https://doi.org/10.1242/jcs.221648
関連情報 (isIdenticalTo)	PMID 30185526
アクセス権	open access
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	Journal of Cell Science. 2018, 131(19), p. jcs.221648
DC.title	RSPH6A is required for sperm flagellum formation and male fertility in mice
DC.creator	Abbasi, Ferheen
	Miyata, Haruhiko
	Shimada, Keisuke
	Morohoshi, Akane
	Nozawa, Kaori
	Matsumura, Takafumi
	Xu, Zoulan
	Pratiwi, Putri
	Ikawa, Masahito
DC.publisher	Company of Biologists Ltd
DC.language' scheme='DCTERMS.RFC1766	英語
DCTERMS.issued' scheme='DCTERMS.W3CDTF	2018-10-11
DC.identifier	https://doi.org/10.1242/jcs.221648
DC.subject	Axoneme
	CRISPR/Cas9
	Flagella
	Radial spoke protein
	Spermatozoa
DCTERMS.abstract	The flagellum is an evolutionarily conserved appendage used for sensing and locomotion. Its backbone is the axoneme and a component of the axoneme is the radial spoke (RS), a protein complex implicated in flagellar motility regulation. Numerous diseases occur if the axoneme is improperly formed, such as primary ciliary dyskinesia (PCD) and infertility. Radial spoke head 6 homolog A (RSPH6A) is an ortholog of Chlamydomonas RSP6 in the RS head and is evolutionarily conserved. While some RS head proteins have been linkedtoPCD, littleisknown about RSPH6A. Here, weshow that mouse RSPH6A is testis-enriched and localized in the flagellum. Rsph6a knockout (KO) male mice are infertile as a result of their short immotile spermatozoa. Observation of the KO testis indicates that the axoneme can elongate but is disrupted before accessory structures are formed. Manchette removal is also impaired in the KO testis. Further, RSPH9, another radial spoke protein, disappeared in the Rsph6a KO flagella. These data indicate that RSPH6A is essential for sperm flagellar assembly and male fertility in mice. This article has an associated First Person interview with the first author of the paper.
DC.format	78569
citation_title	RSPH6A is required for sperm flagellum formation and male fertility in mice
citation_author	Abbasi, Ferheen
	Miyata, Haruhiko
	Shimada, Keisuke
	Morohoshi, Akane
	Nozawa, Kaori
	Matsumura, Takafumi
	Xu, Zoulan
	Pratiwi, Putri
	Ikawa, Masahito
citation_publisher	Company of Biologists Ltd
citation_language	英語
citation_date	2018-10-11
citation_journal_title	Journal of Cell Science
citation_volume	131
citation_issue	19
citation_firstpage	jcs.221648
citation_doi	https://doi.org/10.1242/jcs.221648
citation_pmid	30185526
citation_public_url	https://hdl.handle.net/11094/78569

RSPH6A is required for sperm flagellum formation and male fertility in mice

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