MAP9/MAPH-9 supports axonemal microtubule doublets and modulates motor movement
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Abstract
Microtubule doublets (MTDs) comprise an incomplete microtubule (B-tubule) attached to the side of a complete cylindrical microtubule. These compound microtubules are conserved in cilia across the tree of life; however, the mechanisms by which MTDs form and are maintained in vivo remain poorly understood. Here, we identify microtubule-associated protein 9 (MAP9) as an MTD-associated protein. We demonstrate that C. elegans MAPH-9, a MAP9 homolog, is present during MTD assembly and localizes exclusively to MTDs, a preference that is in part mediated by tubulin polyglutamylation. We find that loss of MAPH-9 causes ultrastructural MTD defects, including shortened and/or squashed B-tubules with reduced numbers of protofilaments, dysregulated axonemal motor velocity, and perturbed cilia function. Because we find that the mammalian ortholog MAP9 localizes to axonemes in cultured mammalian cells and mouse tissues, we propose that MAP9/MAPH-9 plays a conserved role in regulating ciliary motors and supporting the structure of axonemal MTDs.
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Zhang, Y., He, M., & Pan, J. (2025) · Biochemical Society Transactions · doi:10.1042/bst20240688 · Cited by 11 · see citing works
Takahashi, K., Kwok, J., Sato, Y., Aguirre, G. D., & Miyadera, K. (2023) · Frontiers in Cellular Neuroscience · doi:10.3389/fncel.2023.1226603 · Cited by 8 · see citing works
Native molecular architectures of centrosomes in C. elegans embryosPreprint
Tollervey, F., Rios, M. U., Zagoriy, I., Woodruff, J. B., & Mahamid, J. (2024) · bioRxiv (Cold Spring Harbor Laboratory) · doi:10.1101/2024.04.03.587742 · Cited by 6 · see citing works
Alpha-tubulin tails regulate axoneme differentiation
Li, M., Chen, Z., Guo, Z., Wang, Y., Chai, Y., Li, W., & Ou, G. (2025) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.2414731122 · Cited by 5 · see citing works
Structure and mechanism of microtubule stabilization and motor regulation by MAP9Preprint
Çetin, B., Taheri, A., Gölcük, M., Monroy, B. Y., Fernandes, J., Ori-McKenney, K. M., Gür, M., Nogales, E., & Yıldız, A. (2025) · bioRxiv (Cold Spring Harbor Laboratory) · doi:10.1101/2025.11.17.688911 · Cited by 5 · see citing works
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Ng, R., Magescas, J., & Feldman, J. L. (2025) · bioRxiv (Cold Spring Harbor Laboratory) · doi:10.1101/2025.11.17.688953 · Cited by 2 · see citing works
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Self-citationEditorial: Primary cilia as therapeutic targets
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A cytoskeletal scaffold promotes motile cilia assembly by regulating transition-zone integrityPreprint
Park, H., Choi, M., Cheung, H. O.-L., Zhang, Y., Makino, S., Yoshikawa, Y., Qi, H., Liu, Z., Lan, G., Fu, G., et al. (2025) · bioRxiv (Cold Spring Harbor Laboratory) · doi:10.1101/2025.01.10.632296 · Cited by 1 · see citing works
NEKL-4 regulates microtubule stability and mitochondrial health in C. elegans ciliated neuronsPreprint
Power, K. M., Nguyen, K. C. Q., Silva, A., Singh, S. M., Hall, D. H., Rongo, C., & Barr, M. M. (2024) · bioRxiv (Cold Spring Harbor Laboratory) · doi:10.1101/2024.02.14.580304
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Molecular Mechanisms Underlying the Construction of Ciliary Doublet Microtubules
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Structure of the mammalian ciliary transition zone microtubule doubletPreprint
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- Authors
- Tran, M. V., Khuntsariya, D., Fetter, R. D., Ferguson, J. W., Wang, J. T., Long, A. F., Cote, L. E., Wellard, S. R., Vázquez-Martínez, N., Sallee, M. D., Genova, M., Magiera, M. M., Eskinazi, S., Lee, J. D., Peel, N., Janke, C., Stearns, T., Shen, K., Lansky, Z., Magescas, J., & Feldman, J. L.
- Affiliations
- Stanford University (US)
- Czech Academy of Sciences (CZ)
- Howard Hughes Medical Institute (US)
- Centre National de la Recherche Scientifique (FR)
- The College of New Jersey (US)
- Stanford Medicine (US)
- Corresponding author
- Magescas, Jérémy
- Feldman, Jessica L.
- ORCID
- Venue
- Developmental Cell
- Publisher
- Elsevier BV
- Published
- 2024-01
- Volume
- 59
- Issue
- 2
- Pages
- 199-210.e11
- ISSN
- 1534-5807 (ISSN-L)
- PMC
- PMC11385174
- OpenAlex
- W4390414505
- Cited by
- 18
- References
- 91
- Citations by year
- 2026: 3 · 2025: 7 · 2024: 6 · 2023: 2
- FWCI
- 2.99
- Type
- journal article
- Language
- en
- Open access
- bronze
- doi.org
- Licence
- www.elsevier.com/tdm/userlicense/1.0/ (tdm)
- www.elsevier.com/legal/tdmrep-license (tdm)
- www.elsevier.com/open-access/userlicense/1.0/ (vor, 387 d)
- doi.org/10.15223/policy-017 (stm-asf)
- doi.org/10.15223/policy-037 (stm-asf)
- doi.org/10.15223/policy-012 (stm-asf)
- doi.org/10.15223/policy-029 (stm-asf)
- doi.org/10.15223/policy-004 (stm-asf)
- Locations
- Developmental Cell (publishedVersion)
- bioRxiv (Cold Spring Harbor Laboratory) (acceptedVersion)
- SSRN Electronic Journal (acceptedVersion)
- PubMed (publishedVersion)
- PubMed Central (acceptedVersion)
- HAL (submittedVersion, other-oa)
- PubMed Central (submittedVersion)
- Funders
- European Molecular Biology Organization
- Fondation Vaincre Alzheimer
- Agence Nationale de la Recherche (ANR-20-CE13-0011)
- American Heart Association
- Damon Runyon Cancer Research Foundation (DRG-2428-21)
- National Institutes of Health (1S10OD01227601, F32GM142181, K99GM131024, K99GM135489, R01GM133950, R01GM136902, R01NS082208, R15GM114727, R15GM135886, R35GM130286, T32GM007276)
- Howard Hughes Medical Institute
- National Center for Research Resources (P40 OD010440)
- Grantová Agentura České Republiky (19-27477X)
- Fondation pour la Recherche Médicale (DEQ20170336756)
- Institut Curie
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetic and Kidney Cyst Diseases
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Cell Biology › Microtubule and mitosis dynamics
- Health Sciences › Medicine › Pulmonary and Respiratory Medicine › Cystic Fibrosis Research Advances
- Keywords
- doublet microtubules, axoneme, cilia, tubulin polyglutamylation, microtubule-associated proteins, Caenorhabditis elegans, ciliary ultrastructure
About these checks
Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.
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