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

被引：76

作者：

Abbasi, Ferheen ^{[1
,2
]}

Miyata, Haruhiko ^{[1
]}

Shimada, Keisuke ^{[1
]}

Morohoshi, Akane ^{[1
,2
]}

Nozawa, Kaori ^{[1
,2
,5
]}

Matsumura, Takafumi ^{[1
,3
]}

Xu, Zoulan ^{[1
,3
]}

Pratiwi, Putri ^{[1
]}

Ikawa, Masahito ^{[1
,2
,3
,4
]}

机构：

[1] Osaka Univ, Res Inst Microbial Dis, Suita, Osaka 5650871, Japan

[2] Osaka Univ, Grad Sch Med, Suita, Osaka 5650871, Japan

[3] Osaka Univ, Grad Sch Pharmaceut Sci, Suita, Osaka 5650871, Japan

[4] Univ Tokyo, Inst Med Sci, Tokyo 1088639, Japan

[5] Baylor Coll Med, Dept Pathol & Immunol, Houston, TX 77030 USA

来源：

JOURNAL OF CELL SCIENCE | 2018年 / 131卷 / 19期

基金：

美国国家卫生研究院; 比尔及梅琳达.盖茨基金会; 日本学术振兴会;

关键词：

Radial spoke protein; Axoneme; Flagella; Spermatozoa; CRISPR/Cas9; PRIMARY CILIARY DYSKINESIA; RADIAL SPOKES; MALE-INFERTILITY; CHLAMYDOMONAS FLAGELLA; MOUSE; MUTATIONS; SPERMATOZOA; TAIL; EXPRESSION; MANCHETTE;

D O I：

10.1242/jcs.221648

中图分类号：

Q2 [细胞生物学];

学科分类号：

071009 ; 090102 ;

摘要：

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 linked to PCD, little is known about RSPH6A. Here, we show 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.

引用

页数：11

共 50 条

[1] ELECTRON MICROSCOPY OF THE SPERM TAIL - RESULTS OBTAINED WITH A NEW FIXATIVE [J].