CFAP45 deficiency causes situs abnormalities and asthenospermia by disrupting an axonemal adenine nucleotide homeostasis module
Open accessAbstract
Axonemal dynein ATPases direct ciliary and flagellar beating via adenosine triphosphate (ATP) hydrolysis. The modulatory effect of adenosine monophosphate (AMP) and adenosine diphosphate (ADP) on flagellar beating is not fully understood. Here, we describe a deficiency of cilia and flagella associated protein 45 (CFAP45) in humans and mice that presents a motile ciliopathy featuring situs inversus totalis and asthenospermia. CFAP45-deficient cilia and flagella show normal morphology and axonemal ultrastructure. Proteomic profiling links CFAP45 to an axonemal module including dynein ATPases and adenylate kinase as well as CFAP52, whose mutations cause a similar ciliopathy. CFAP45 binds AMP in vitro, consistent with structural modelling that identifies an AMP-binding interface between CFAP45 and AK8. Microtubule sliding of dyskinetic sperm from Cfap45−/− mice is rescued with the addition of either AMP or ADP with ATP, compared to ATP alone. We propose that CFAP45 supports mammalian ciliary and flagellar beating via an adenine nucleotide homeostasis module.
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Mutations in CFAP45 cause reduced sperm motility, suggesting a defect in motile cilia.
“At the same time, mutations in CFAP45 cause reduced sperm motility, suggesting a defect in motile cilia79.”
From The inner junction protein CFAP20 functions in motile and non-motile cilia and is critical for vision (2022) · cites this paper as [79]
In humans, CFAP45 and CFAP52 are both associated with a human laterality defect but not PCD.
“Interestingly, in humans, CFAP45 and CFAP52 are both associated with a human laterality defect but not PCD79,80.”
From The inner junction protein CFAP20 functions in motile and non-motile cilia and is critical for vision (2022) · cites this paper as [79]
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- Authors
- Dougherty, G. W., Mizuno, K., Nöthe-Menchen, T., Ikawa, Y., Boldt, K., Ta-Shma, A., Aprea, I., Minegishi, K., Pang, Y.-P., Pennekamp, P., Loges, N. T., Raidt, J., Hjeij, R., Wallmeier, J., Mussaffi, H., Perles, Z., Elpeleg, O., Rabert, F., Shiratori, H., Letteboer, S. J., Horn, N., Young, S., Strünker, T., Stumme, F., Werner, C., Olbrich, H., Takaoka, K., Ide, T., Twan, W. K., Biebach, L., Große-Onnebrink, J., Klinkenbusch, J. A., Praveen, K., Bracht, D. C., Höben, I. M., Junger, K., Gützlaff, J., Cindrić, S., Aviram, M., Kaiser, T., Memari, Y., Dzeja, P. P., Dworniczak, B., Ueffing, M., Roepman, R., Bartscherer, K., Katsanis, N., Davis, E. E., Amirav, I., Hamada, H., & Omran, H.
- Affiliations
- University Hospital Münster (DE)
- RIKEN Center for Biosystems Dynamics Research (JP)
- University of Tübingen (DE)
- Mayo Clinic (US)
- Tel Aviv University (IL)
- Hebrew University of Jerusalem (IL)
- Hadassah Medical Center (IL)
- Max Planck Institute for Molecular Biomedicine (DE)
- The University of Osaka (JP)
- Radboud University Medical Center (NL)
- University of Münster (DE)
- Duke Medical Center (US)
- Soroka Medical Center (IL)
- MRC Cancer Unit (GB)
- Northwestern University (US)
- University of Alberta (CA)
- Corresponding author
- Omran, Heymut
- ORCID
- Dougherty, Gerard W.
- Mizuno, Katsutoshi
- Nöthe-Menchen, Tabea
- Boldt, Karsten
- Ta-Shma, Asaf
- Aprea, Isabella
- Minegishi, Katsura
- Pang, Yuan-Ping
- Pennekamp, Petra
- Loges, Niki T.
- Raidt, Johanna
- Hjeij, Rim
- Wallmeier, Julia
- Mussaffi, Huda
- Elpeleg, Orly
- Letteboer, Stef J.
- Young, Samuel
- Strünker, Timo
- Olbrich, Heike
- Takaoka, Katsuyoshi
- Twan, Wang Kyaw
- Biebach, Luisa
- Große-Onnebrink, Jörg
- Bracht, Diana C.
- Höben, Inga M.
- Cindrić, Sandra
- Aviram, Micha
- Kaiser, Thomas
- Memari, Yasin
- Dzeja, Petras P.
- Dworniczak, Bernd
- Ueffing, Marius
- Roepman, Ronald
- Bartscherer, Kerstin
- Katsanis, Nicholas
- Davis, Erica E.
- Amirav, Israel
- Hamada, Hiroshi
- Omran, Heymut
- Venue
- Nature Communications
- Published
- 2020-11-02
- Volume
- 11
- Issue
- 1
- Pages
- 5520-5520
- ISSN
- 2041-1723 (ISSN-L)
- PMC
- PMC7606486
- OpenAlex
- W3094715636
- Cited by
- 88
- References
- 87
- Citations by year
- 2026: 12 · 2025: 11 · 2024: 24 · 2023: 24 · 2022: 9 · 2021: 7
- FWCI
- 5.85
- Type
- journal article
- Language
- en
- Open access
- gold
- doi.org
- Locations
- Nature Communications (publishedVersion, cc-by)
- PubMed Central (publishedVersion, cc-by)
- PubMed (publishedVersion)
- KNAW research portal (Royal Academy of Art and Sciences (KNAW)) (publishedVersion, other-oa)
- Institutional Repositories DataBase (IRDB) (submittedVersion, other-oa)
- Radboud Repository (Radboud University) (submittedVersion, other-oa)
- Data Archiving and Networked Services (DANS) (submittedVersion, other-oa)
- RePEc: Research Papers in Economics (submittedVersion, cc-by)
- DOAJ (DOAJ: Directory of Open Access Journals) (submittedVersion, cc-by-sa)
- KNAW Research Portal (The Royal Netherlands Academy of Arts and Sciences) (submittedVersion, other-oa)
- Radboud Repository (Radboud University) (submittedVersion)
- Funders
- European Commission (2007–2013)
- Deutsche Forschungsgemeinschaft (CRU326, OM 6/7, BA4044, EXC-1003, OM6/8, OM6/11, EXC 1003 CIM, OM6/9, DFG CRU-326, OM6/10, BA4044/3-1)
- Japan Science and Technology Corporation (CREST)
- ZonMw (ZonMW-91216051, 91216051, 13417760)
- Max-Planck-Gesellschaft
- University of Tokyo
- Tokyo University of Agriculture
- Tokyo University of Agriculture and Technology
- National Institutes of Health (CRU326)
- Japan Society for the Promotion of Science (19K06753, 19K23756, CREST, 24113004)
- National Institute of Diabetes and Digestive and Kidney Diseases (R01DK072301)
- Core Research for Evolutional Science and Technology (13417760)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetic and Kidney Cyst Diseases
- Health Sciences › Medicine › Pulmonary and Respiratory Medicine › Cystic Fibrosis Research Advances
- Life Sciences › Neuroscience › Cellular and Molecular Neuroscience › Hereditary Neurological Disorders
- Keywords
- motile ciliopathy, situs inversus totalis, asthenozoospermia, axoneme, AMP binding, dynein ATPase, adenylate kinase, sperm motility, ciliary beating, microtubule sliding
- MeSH terms
- Exome Sequencing, Loss of Function Mutation, Adenine Nucleotides, Adolescent, Adult, Animals, Cilia, Cytoskeletal Proteins, Disease Models, Animal, DNA Mutational Analysis, Epididymis, Female, Flagella, Humans, Male, Middle Aged, Planarians, Situs Inversus, Sperm Motility, Tomography, X-Ray Computed, Mice, Knockout, Respiratory Mucosa, Mice, Asthenozoospermia, Axoneme, CRISPR-Cas Systems
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