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


Autoři: Akane Morohoshi aff001;  Haruhiko Miyata aff001;  Keisuke Shimada aff001;  Kaori Nozawa aff001;  Takafumi Matsumura aff001;  Ryuji Yanase aff004;  Kogiku Shiba aff004;  Kazuo Inaba aff004;  Masahito Ikawa aff001
Působiště autorů: Research Institute for Microbial Diseases, Osaka University, Osaka, Japan aff001;  Graduate School of Medicine, Osaka University, Osaka, Japan aff002;  Graduate School of Pharmaceutical Sciences, Osaka University, Osaka, Japan aff003;  Shimoda Marine Research Center, University of Tsukuba, Shizuoka, Japan aff004;  The Institute of Medical Science, The University of Tokyo, Tokyo, Japan aff005
Vyšlo v časopise: Nexin-Dynein regulatory complex component DRC7 but not FBXL13 is required for sperm flagellum formation and male fertility in mice. PLoS Genet 16(1): e32767. doi:10.1371/journal.pgen.1008585
Kategorie: Research Article
doi: 10.1371/journal.pgen.1008585

Souhrn

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.

Klíčová slova:

Cilia – Flagella – Microtubules – Sperm – Spermatids – Spermatogenesis – Sperm head – Flagellar motility


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Genetika Reprodukční medicína

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