ArticlePLoS genetics2020
Cfap97d1 is important for flagellar axoneme maintenance and male mouse fertility.
Article in PLoS genetics, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 19 papers.
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Who cites it
19 citing papers in PubMed.
- Biallelic loss-of-function variants in DNAH12 cause inner dynein arm defects and sperm head abnormalities leading to male infertility.Journal of assisted reproduction and genetics · 2026Article
- Integrated environmental and genomic analysis reveals the drivers and genetic mechanisms of agro-climatic adaptation in geese.BMC genomics · 2026Article
- Doublet microtubule-associated tektins and enzymes differentially regulate sperm flagellar integrity and motility.Nature communications · 2026Article
- Ferroptosis-Mediated Hippocampal Neuronal Loss Post-mTBI: Chromatin Accessibility Profiling and Single-Nucleus Transcriptomics.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2026Article
- Age-related changes in lncRNA expression in sperm.Translational andrology and urology · 2025Article
- TMEM232 is required for the formation of sperm flagellum and male fertility in mice.Cell death & disease · 2024Article
- The lack of Tex44 causes severe subfertility with flagellar abnormalities in male mice.Cellular & molecular biology letters · 2024Article
- Novel putative causal mutations associated with fat traits in Nellore cattle uncovered by eQTLs located in open chromatin regions.Scientific reports · 2024Article
- TTC6-Mediated Stabilization of the Flagellum Annulus Ensures the Rapid and Directed Motion of Sperm.Cells · 2023Article
- A phylogenetic profiling approach identifies novel ciliogenesis genes in Drosophila and C. elegans.The EMBO journal · 2023Article
- Article
- Deficiency of theCells · 2023Article
- MRNIP interacts with sex body chromatin to support meiotic progression, spermatogenesis, and male fertility in mice.FASEB journal : official publication of the Federation of American Societies for Experimental Biology · 2022Article
- IRGC1, a testis-enriched immunity related GTPase, is important for fibrous sheath integrity and sperm motility in mice.Developmental biology · 2022Article
- Trim41 is required to regulate chromosome axis protein dynamics and meiosis in male mice.PLoS genetics · 2022Article
- AXDND1, a novel testis-enriched gene, is required for spermiogenesis and male fertility.Cell death discovery · 2021Article
- Zebrafish Motile Cilia as a Model for Primary Ciliary Dyskinesia.International journal of molecular sciences · 2021Article
- Testis-expressed protein 33 is not essential for spermiogenesis and fertility in mice.Molecular medicine reports · 2021Article
- KCTD19 and its associated protein ZFP541 are independently essential for meiosis in male mice.PLoS genetics · 2021Article
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Authors and funding
12 authors.
Funding
Abstract
The flagellum is essential for sperm motility and fertilization in vivo. The axoneme is the main component of the flagella, extending through its entire length. An axoneme is comprised of two central microtubules surrounded by nine doublets, the nexin-dynein regulatory complex, radial spokes, and dynein arms. Failure to properly assemble components of the axoneme in a sperm flagellum, leads to fertility alterations. To understand this process in detail, we have defined the function of an uncharacterized gene, Cfap97 domain containing 1 (Cfap97d1). This gene is evolutionarily conserved in mammals and multiple other species, including Chlamydomonas. We have used two independently generated Cfap97d1 knockout mouse models to study the gene function in vivo. Cfap97d1 is exclusively expressed in testes starting from post-natal day 20 and continuing throughout adulthood. Deletion of the Cfap97d1 gene in both mouse models leads to sperm motility defects (asthenozoospermia) and male subfertility. In vitro fertilization (IVF) of cumulus-intact oocytes with Cfap97d1 deficient sperm yielded few embryos whereas IVF with zona pellucida-free oocytes resulted in embryo numbers comparable to that of the control. Knockout spermatozoa showed abnormal motility characterized by frequent stalling in the anti-hook position. Uniquely, Cfap97d1 loss caused a phenotype associated with axonemal doublet heterogeneity linked with frequent loss of the fourth doublet in the sperm stored in the epididymis. This study demonstrates that Cfap97d1 is required for sperm flagellum ultra-structure maintenance, thereby playing a critical role in sperm function and male fertility in mice.
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