The complexity of the cilium: spatiotemporal diversity of an ancient organelle
Open accessAbstract
Cilia are microtubule-based appendages present on almost all vertebrate cell types where they mediate a myriad of cellular processes critical for development and homeostasis. In humans, impaired ciliary function is associated with an ever-expanding repertoire of phenotypically-overlapping yet highly variable genetic disorders, the ciliopathies. Extensive work to elucidate the structure, function, and composition of the cilium is offering hints that the `static' representation of the cilium is a gross oversimplification of a highly dynamic organelle whose functions are choreographed dynamically across cell types, developmental, and homeostatic contexts. Understanding this diversity will require discerning ciliary versus non-ciliary roles for classically-defined `ciliary' proteins; defining ciliary protein-protein interaction networks within and beyond the cilium; and resolving the spatiotemporal diversity of ciliary structure and function. Here, focusing on one evolutionarily conserved ciliary module, the intraflagellar transport system, we explore these ideas and propose potential future studies that will improve our knowledge gaps of the oversimplified cilium and, by extension, inform the reasons that underscore the striking range of clinical pathologies associated with ciliary dysfunction.
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The set of signalling components within the cilium is dynamic and closely coupled to the cell's differentiation state and microenvironment.
“Emerging evidence indicates that the constellation of signalling components within the cilium is also dynamic and closely coupled to the differentiation state and microenvironment of the cell 3.”
From Cellular signalling by primary cilia in development, organ function and disease (2019) · cites this paper as [3]
The Concluding remarks describe the cilium as a dynamic organelle whose proteins interact in changing functional networks. “Unique spatiotemporal roles of ciliary proteins” links ciliation and Hedgehog signalling to differentiation and describes cilia restricted to particular extracellular-matrix zones. These passages do not establish that the specific set of signalling components within a cilium changes in close response to both differentiation state and microenvironment. (LLM, conf=0.82)
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The keratin-binding protein Albatross regulates polarization of epithelial cells
Sugimoto, M., Inoko, A., Shiromizu, T., et al. (2008) · The Journal of Cell Biology · doi:10.1083/jcb.200803133 · Cited by 46 · see citing works
Characterization of the human homologue of the mouse Tg737 candidate polycystic kidney disease gene
Schrick, J. J., Onuchic², L. F., Reeders, S. T., Korenberg, J. R., Chen, X.-N., Moyer, J. H., Wilkinson, J. E., & Woychik, R. P. (1995) · Human Molecular Genetics · doi:10.1093/hmg/4.4.559 · Cited by 45 · see citing works
Contribution of the TTC21B gene to glomerular and cystic kidney diseases
Bullich, G., Vargas, I., Trujillano, D., et al. (2016) · Nephrology Dialysis Transplantation · doi:10.1093/ndt/gfv453 · Cited by 37 · see citing works
The T cell IFT20 interactome reveals new players in immune synapse assembly
Galgano, D., Onnis, A., Pappalardo, E., et al. (2017) · Journal of Cell Science · doi:10.1242/jcs.200006 · Cited by 37 · see citing works
Regulation of Vesicular Traffic at the T Cell Immune Synapse: Lessons from the Primary Cilium
Finetti, F., Onnis, A., & Baldari, C. T. (2014) · Traffic · doi:10.1111/tra.12241 · Cited by 35 · see citing works
CRISPRスクリーンはモルフォゲンSonicH edgehogに対する標的細胞の感受性を調節する遺伝子を明らかにする【Powered by NICT】
Ganesh, V. P., Jennifer, H., Bhaven, B. P., et al. (2018) · Developmental Cell · Cited by 31 · see citing works
Structure and motility of primary cilia in the follicular epithelium of the human thyroid
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Iwanaga, T., Hozumi, Y., & Takahashi‐Iwanaga, H. (2011) · Biomedical Research · doi:10.2220/biomedres.32.225 · Cited by 23 · see citing works
Nuclear roles for cilia-associated proteins
McClure‐Begley, T. D., & Klymkowsky, M. W. (2017) · Cilia · doi:10.1186/s13630-017-0052-x · Cited by 22 · see citing works
Comparative study of the primary cilia in thyrocytes of adult mammals
Utrilla, J. C., Gordillo-Martínez, F., Gómez‐Pascual, A., Fernández-Santos, J. M., Garnacho, C., Vázquez‐Román, V., Morillo-Bernal, J., García‐Marín, R., Jiménez-García, A., & Martín‐Lacave, I. (2015) · Journal of Anatomy · doi:10.1111/joa.12360 · Cited by 13 · see citing works
Localization of Protein Kinase NDR2 to Peroxisomes and Its Role in Ciliogenesis
Abe, S. E., Nagai, T., Masukawa, M. M. H., Okumoto, K., Homma, Y., Fujiki, Y., & Mizuno, K. (2017) · Journal of Biological Chemistry · doi:10.1074/jbc.m117.775916 · Cited by 13 · see citing works
Thyroid-specific inactivation of KIF3A alters the TSH signaling pathway and leads to hypothyroidism
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Is ciliary Hedgehog signalling dispensable in the kidneys?
Otto, E. A. (2018) · Nature Reviews Nephrology · doi:10.1038/s41581-018-0014-6 · Cited by 2 · see citing works
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Cellular signalling by primary cilia in development, organ function and disease
Anvarian, Z., Mykytyn, K., Mukhopadhyay, S., Pedersen, L. B., & Christensen, S. T. (2019) · Nature Reviews Nephrology · doi:10.1038/s41581-019-0116-9 · Cited by 845 · see citing works
- Authors
- Heydeck, W., Fievet, L., Davis, E. E., & Katsanis, N.
- Affiliations
- Duke Medical Center (US)
- Centre National de la Recherche Scientifique (FR)
- Duke University (US)
- Corresponding author
- Katsanis, Nicholas
- Venue
- Current Opinion in Cell Biology
- Publisher
- Elsevier BV
- Published
- 2018-12
- Volume
- 55
- Pages
- 139-149
- ISSN
- 0955-0674 (ISSN-L)
- PMC
- PMC6269220
- OpenAlex
- W2888222789
- Cited by
- 25
- References
- 100
- Citations by year
- 2025: 1 · 2024: 4 · 2023: 2 · 2022: 2 · 2021: 2 · 2020: 8 · 2019: 4 · 2018: 1
- FWCI
- 0.52
- Type
- journal article
- Language
- en
- Open access
- green
- www.ncbi.nlm.nih.gov
- Locations
- Current Opinion in Cell Biology (publishedVersion)
- PubMed (publishedVersion)
- Europe PMC (PubMed Central) (acceptedVersion)
- PubMed Central (acceptedVersion)
- HAL (submittedVersion)
- Funders
- Foundation for the National Institutes of Health (GM121317, HD042601, DK072301)
- 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
- cilia, ciliopathies, intraflagellar transport, ciliary dysfunction
- MeSH terms
- Animals, Cilia, Humans, Time Factors, Organelles, Proteomics, Protein Interaction Maps
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.
Statistics and arithmetic are recomputed, not judged.
Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.
What counts as a claim ›How we measure confidence ›How we check this ›
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