Abstract
Sperm motility is driven by motile cytoskeletal elements in the tail, called axonemes. The structure of axonemes consists of 9 + 2 microtubules, molecular motors (dyneins), and their regulatory structures. Axonemes are well conserved in motile cilia and flagella through eukaryotic evolution. Deficiency in the axonemal structure causes defects in sperm motility, and often leads to male infertility. It has been known since the 1970s that, in some cases, male infertility is linked with other symptoms or diseases such as Kartagener syndrome. Given that these links are mostly caused by deficiencies in the common components of cilia and flagella, they are called “immotile cilia syndrome” or “primary ciliary dyskinesia,” or more recently, “ciliopathy,” which includes deficiencies in primary and sensory cilia. Here, we review the structure of the sperm flagellum and epithelial cilia in the human body, and discuss how male fertility is linked to ciliopathy.
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Abbreviations
- AK:
-
Adenylate kinase
- ARMC:
-
Armadillo repeat containing
- BB:
-
Basal body
- BBS:
-
Bardet–Biedl syndrome
- CEP:
-
Centrosomal protein
- CCDC:
-
Coiled-coil domain-containing
- CP:
-
Central pair apparatus
- DC:
-
Docking complex
- DNAAF:
-
Dynein axonemal assembly factor
- DNAH:
-
Dynein, axonemal, heavy chain
- DNAI:
-
Dynein, axonemal, intermediate chain
- DNALI:
-
Dynein, axonemal, light intermediate chain
- DYNC2:
-
Dynein, cytoplasmic 2
- DYX1C1:
-
Dyslexia susceptibility 1 candidate 1
- FAP:
-
Flagella-associated protein
- FBB:
-
Flagellar basal body
- FoxJ1:
-
Forkhead box J1
- HC:
-
Heavy chain
- HEATR:
-
HEAT-repeat containing
- IC:
-
Intermediate chain
- IAD:
-
Inner arm dynein
- IDA:
-
Inner dynein arm
- IFT:
-
Intraflagellar transport
- IMT:
-
Intramanchette transport
- Iqcg:
-
IQ motif containing G
- LC:
-
Light chain
- LRRC:
-
Leucine-rich repeat containing
- MFN:
-
Mitofusin
- MIA:
-
Modifier of inner arms
- MKS:
-
McKusick–Kaufman syndrome
- MNS1:
-
Meiosis-specific nuclear structural 1
- N-DRC:
-
Nexin-dynein regulatory complex
- NPHP:
-
Nephronophthisis
- OAD:
-
Outer arm dynein
- ODA:
-
Outer dynein arm
- ODF:
-
Outer dense fiber
- OFD:
-
Oral-facial-digital syndrome
- PACRG:
-
Parkin co-regulated gene
- PCD:
-
Primary ciliary dyskinesia
- Pcdp1:
-
Primary ciliary dyskinesia protein 1
- PCP:
-
Planar cell polarity
- PF:
-
Paralyzed flagella
- PIH:
-
Protein interacting with HSP90
- PKD:
-
Polycystic kidney disease
- RFX:
-
Regulatory factor X
- RPGR:
-
Retinitis pigmentosa GTPase regulator
- RS:
-
Radial spoke
- SPAG:
-
Sperm-associated antigen
- Spef2:
-
Sperm flagellar protein 2
- TXNDC:
-
Thioredoxin domain containing
- TZ:
-
Transition zone
- VDAC3:
-
Voltage-dependent anion channel 3
- XLRP:
-
X-linked retinitis pigmentosa
- ZMYND:
-
Zinc-finger, MYND-type containing protein
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Acknowledgments
This work was supported in part by Grants-in-aid #15H01201 for Scientific Research on Innovative Areas and #22370023 for Scientific Research (B) from the Ministry of Education, Culture, Sports, Science, and Technology of Japan (MEXT). Studies with marine invertebrates cited in this paper were supported by the members of the Onagawa Field Research Center, Tohoku University; International Coastal Research Center, AORI, The University of Tokyo; and staff of the National Bioresource Project.
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Inaba, K., Mizuno, K. Sperm dysfunction and ciliopathy. Reprod Med Biol 15, 77–94 (2016). https://doi.org/10.1007/s12522-015-0225-5
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DOI: https://doi.org/10.1007/s12522-015-0225-5