Primary Cilia in the Developing and Mature Brain
Open accessAbstract
Primary cilia were the largely neglected nonmotile counterparts of their better-known cousin, the motile cilia. For years these nonmotile cilia were considered evolutionary remnants of little consequence to cellular function. Fast forward 10 years and we now recognize primary cilia as key integrators of extracellular ligand-based signaling and cellular polarity, which regulate neuronal cell fate, migration, differentiation, as well as a host of adult behaviors. Important future questions will focus on structure-function relationships, their roles in signaling and disease and as areas of target for treatments.
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Treatment of mouse hippocampal neurons with somatostatin stimulates recruitment of endogenous beta-arrestin into SSTR3-positive cilia, leading to a rapid beta-arrestin-2-dependent decrease in ciliary SSTR3.
“Treatment of mouse hippocampal neurons with the SSTR3 ligand, somatostatin, stimulates recruitment of endogenous β-arrestin into SSTR3-positive cilia, leading to a rapid β-arrestin-2-dependent decrease in the ciliary SSTR3 167”
From Cellular signalling by primary cilia in development, organ function and disease (2019) · cites this paper as [167]
The section “Primary Cilia in the Adult Nervous System: Neurogenesis and Signaling?” states that SSTR3 is localized to neuronal cilia. It does not describe somatostatin treatment, β-arrestin recruitment, or a rapid β-arrestin-2-dependent decrease in ciliary SSTR3 (searched for somatostatin, SSTR3, and β-arrestin). (LLM, conf=0.86)
Experimental — accuracy not yet measuredCiliary localization of SSTR3 is not affected in beta-arrestin-2-deficient hippocampal neurons.
“ciliary localization of SSTR3 is not affected in β-arrestin-2-deficient hippocampal neurons167.”
From Cellular signalling by primary cilia in development, organ function and disease (2019) · cites this paper as [167]
The section ‘Primary Cilia in the Adult Nervous System: Neurogenesis and Signaling?’ states that SSTR3 localizes to neuronal cilia (page not provided). It does not address β-arrestin-2-deficient hippocampal neurons or whether SSTR3 localization is unaffected in them; searches for those terms found no such result. (LLM, conf=0.86)
Experimental — accuracy not yet measuredNeuronal cilia are enriched for certain GPCRs and downstream effector proteins.
“Neuronal cilia are enriched for certain GPCRs and downstream effector proteins 152, 166, 167”
From Cellular signalling by primary cilia in development, organ function and disease (2019) · cites this paper as [167]
The sections on special sensory neurons and neuronal migration describe GPCRs in neuronal cilia, including olfactory GPCRs and ciliary receptors needed for guidance-cue responses (page not provided). The copy also describes ciliary signaling, but the located passages do not establish that neuronal cilia are enriched for downstream effector proteins. (LLM, conf=0.81)
Experimental — accuracy not yet measuredThe primary cilium is important in neuronal cells.
“Numerous studies have suggested the importance of the primary cilium in neuronal cells (Guemez-Gamboa et al. 2014).”
From Astrocyte-specific inhibition of the primary cilium suppresses C3 expression in reactive astrocyte (2024) · cites this paper as Guemez-Gamboa et al. 2014
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Genetically encoded calcium indicator illuminates calcium dynamics in primary cilia
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Amador‐Arjona, A., Elliott, J., Miller, A., Ginbey, A., Pazour, G. J., Enikolopov, G., Roberts, A. J., & Terskikh, A. V. (2011) · Journal of Neuroscience · doi:10.1523/jneurosci.1062-11.2011 · Cited by 117 · cited 2× · see citing works
“, 2003), and loss of ciliary genes (Kif3a, Ift88, or Ftm) leads to defective hippocampal neurogenesis due to premature cellcycle exit (Amador-Arjona et al., 2011; Han et al., 2008).”
Gene therapy rescues cilia defects and restores olfactory function in a mammalian ciliopathy model
McIntyre, J. C., Davis, E. E., Joiner, A. M., Williams, C. L., Tsai, I.-C., Jenkins, P. M., McEwen, D. P., Zhang, L., Escobado, J., Thomas, S., et al. (2012) · Nature Medicine · doi:10.1038/nm.2860 · Cited by 113 · see citing works
Gorivodsky, M., Mukhopadhyay, M., Wilsch‐Bräuninger, M., Phillips, M., Teufel, A., Kim, C., Malik, N., Huttner, W. Β., & Westphal, H. (2008) · Developmental Biology · doi:10.1016/j.ydbio.2008.09.019 · Cited by 113 · see citing works
Molecular components of signal amplification in olfactory sensory cilia
Hengl, T., Kaneko, H., Dauner, K., Vocke, K., Frings, S., & Möhrlen, F. (2010) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.0909032107 · Cited by 109 · cited 1× · see citing works
“a cAMP-mediated depolarization, whereas photoreceptor outer segments mediate phototransduction via the rhodopsin family of GPCR, producing a cGMP-mediated hyperpolarization (Hengl et al., 2010).”
DISC1 Regulates Primary Cilia That Display Specific Dopamine Receptors
Marley, A., & von Zastrow, M. (2010) · PLoS ONE · doi:10.1371/journal.pone.0010902 · Cited by 106 · see citing works
Carter, C. S., Vogel, T. W. A., Zhang, Q., Seo, S., Swiderski, R. E., Moninger, T. O., Cassell, M. D., Thedens, D. R., Keppler‐Noreuil, K. M., Nopoulos, P., et al. (2012) · Nature Medicine · doi:10.1038/nm.2996 · Cited by 105 · see citing works
Cruz, C., Ribes, V., Kutejova, E., Cayuso, J., Lawson, V. J., Norris, D. P., Stevens, J. L., Davey, M. G., Blight, K., Bangs, F. K., et al. (2010) · Development · doi:10.1242/dev.051714 · Cited by 103 · see citing works
Leptin resistance is a secondary consequence of the obesity in ciliopathy mutant mice
Berbari, N. F., Pasek, R. C., Malarkey, E. B., Yazdi, S. M. Z., McNair, A. D., Lewis, W. R., Nagy, T. R., Kesterson, R. A., & Yoder, B. K. (2013) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.1210192110 · Cited by 98 · see citing works
Development and distribution of neuronal cilia in mouse neocortex
Arellano, J. I., Guadiana, S. M., Breunig, J. J., Rakic, P., & Sarkisian, M. R. (2012) · Journal of Comparative Neurology · doi:10.1002/cne.22793 · Cited by 97 · cited 2× · see citing works
“neurons did not alter their migration but did induce axonal outgrowth and connectivity defects (Arellano et al., 2012).”
A systems-biology approach to understanding the ciliopathy disorders
Lee, J. E., & Gleeson, J. G. (2011) · Genome Medicine · doi:10.1186/gm275 · Cited by 96 · see citing works
Self-citationAssociation of common variants in the Joubert syndrome gene (AHI1) with autism
Alvarez Retuerto, A. I., Cantor, R. M., Gleeson, J. G., Ustaszewska, A., Schackwitz, W. S., Pennacchio, L. A., & Geschwind, D. H. (2008) · Human Molecular Genetics · doi:10.1093/hmg/ddn291 · Cited by 96 · see citing works
Self-citationDrack, A. V., Dumitrescu, A. V., Bhattarai, S., Gratie, D., Stone, E. M., Mullins, R. F., & Sheffield, V. C. (2011) · Investigative Ophthalmology & Visual Science · doi:10.1167/iovs.11-8544 · Cited by 95 · see citing works
Ttc21b is required to restrict sonic hedgehog activity in the developing mouse forebrain
Stottmann, R. W., Tran, P. V., Turbé-Doan, A., & Beier, D. R. (2009) · Developmental Biology · doi:10.1016/j.ydbio.2009.08.023 · Cited by 90 · see citing works
Barker, A., Thomas, R., & Dawe, H. R. (2013) · Organogenesis · doi:10.4161/org.27375 · Cited by 88 · cited 1× · see citing works
“MKS is an early lethal disorder, genetically overlapping with JBTS and BBS, with occipital encephalocele, cystic kidney, and polydactyly (Barker et al., 2013).”
Simons, D. L., Boye, S. L., Hauswirth, W. W., & Wu, S. M.-S. (2011) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.1019222108 · Cited by 87 · see citing works
RTTN Mutations Link Primary Cilia Function to Organization of the Human Cerebral Cortex
Kia, S. K., Verbeek, E., Engelen, E., Schot, R., Poot, R. A., de Coo, I. F. M., Lequin, M. H., Poulton, C. J., Pourfarzad, F., Grosveld, F. G., et al. (2012) · The American Journal of Human Genetics · doi:10.1016/j.ajhg.2012.07.008 · Cited by 81 · see citing works
A Simple Cell-Based Assay Reveals That Diverse Neuropsychiatric Risk Genes Converge on Primary Cilia
Marley, A., & von Zastrow, M. (2012) · PLoS ONE · doi:10.1371/journal.pone.0046647 · Cited by 76 · see citing works
Hedgehog-Stimulated Chemotaxis Is Mediated by Smoothened Located Outside the Primary Cilium
Bijlsma, M. F., Damhofer, H., & Roelink, H. (2012) · Science Signaling · doi:10.1126/scisignal.2002798 · Cited by 75 · see citing works
Morbid Obesity Resulting from Inactivation of the Ciliary Protein CEP19 in Humans and Mice
Shalata, A., Ramirez, M. C. M., Desnick, R. J., Priedigkeit, N., Buettner, C., Lindtner, C., Mahroum, M., Abdul‐Ghani, M. A., Dong, F., Arar, N., et al. (2013) · The American Journal of Human Genetics · doi:10.1016/j.ajhg.2013.10.025 · Cited by 73 · cited 1× · see citing works
“Moreover, Cep19 knockout mice are morbidly obese, hyperphagic, glucose intolerant, and insulin resistant (Shalata et al., 2013).”
Compartmentalized Calcium Signaling in Cilia Regulates Intraflagellar Transport
Collingridge, P. W., Brownlee, C., & Wheeler, G. L. (2013) · Current Biology · doi:10.1016/j.cub.2013.09.059 · Cited by 68 · see citing works
Chang, M., & Ong, A. C. M. (2011) · Nephron Clinical Practice · doi:10.1159/000334166 · Cited by 67 · cited 1× · see citing works
“However, human clinical trials have shown only limited success to date (Chang and Ong, 2012; Torres et al., 2012).”
Interaction of PACAP with Sonic hedgehog reveals complex regulation of the Hedgehog pathway by PKA
Niewiadomski, P., Zhujiang, A., Youssef, M., & Waschek, J. A. (2013) · Cellular Signalling · doi:10.1016/j.cellsig.2013.07.012 · Cited by 65 · see citing works
Chhin, B., Négre, D., Merrot, O., Pham, J. T., Tourneur, Y., Ressnikoff, D., Jaspers, M., Jorissen, M., Cosset, F., & Bouvagnet, P. (2009) · PLoS Genetics · doi:10.1371/journal.pgen.1000422 · Cited by 63 · see citing works
Berbari, N. F., Kin, N. W., Sharma, N., Michaud, E. J., Kesterson, R. A., & Yoder, B. K. (2011) · Developmental Biology · doi:10.1016/j.ydbio.2011.09.001 · Cited by 63 · see citing works
Baker, K., Northam, G. B., Chong, W. K., Banks, T., Beales, P. L., & Baldeweg, T. (2010) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.33773 · Cited by 61 · see citing works
Developmental changes in frequency of the ciliary somatostatin receptor 3 protein
Stanić, D., Malmgren, H., He, H., Scott, L., Aperia, A. C., & Hökfelt, T. G. M. (2008) · Brain Research · doi:10.1016/j.brainres.2008.10.024 · Cited by 55 · see citing works
Mockel, A., Obringer, C., Hakvoort, T. B. M., Seeliger, M. W., Lamers, W. H., Stoetzel, C., Dollfus, H., & Marion, V. (2012) · Journal of Biological Chemistry · doi:10.1074/jbc.m112.386821 · Cited by 52 · see citing works
Greene, V. B., Kremer, S., Stoetzel, C., Christmann, D., Schuster, C., Durand, M., Verloès, A., Sigaudy, S., Holder‐Espinasse, M., Godet, J., et al. (2011) · Clinical Genetics · doi:10.1111/j.1399-0004.2011.01688.x · Cited by 45 · see citing works
Tubby-Like Protein 1 (Tulp1) Is Required for Normal Photoreceptor Synaptic Development
Grossman, G., Pauer, G. J. T., Narendra, U., & Hagstrom, S. A. (2009) · Advances in experimental medicine and biology · doi:10.1007/978-1-4419-1399-9_11 · Cited by 10 · 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
hESC-Derived Thalamic Organoids Form Reciprocal Projections When Fused with Cortical Organoids
Xiang, Y., Tanaka, Y., Cakir, B., Patterson, B., Kim, K.-Y., Sun, P., Kang, Y.-J., Zhong, M., Liu, X., Patra, P., et al. (2019) · Cell Stem Cell · doi:10.1016/j.stem.2018.12.015 · Cited by 485 · see citing works
“The CBC showed a high expression of genes involved in cilium development, which is essential for brain development and function (Guemez-Gamboa et al., 2014).”
BackgroundSynthetic Analyses of Single-Cell Transcriptomes from Multiple Brain Organoids and Fetal Brain
Tanaka, Y., Çakır, B., Xiang, Y., Sullivan, G. J., & Park, I. (2020) · Cell Reports · doi:10.1016/j.celrep.2020.01.038 · Cited by 238 · see citing works
“Cell clusters represented by cilium (cilia-bearing cell [CBC]) and BMP signaling (BMP-related cell [BRC]), which are crucial for early neurogenesis and survival (Guemez-Gamboa et al., 2014), were observed both in fetal brain and in all hCOs (Figure 2D).”
BackgroundCPAP promotes timely cilium disassembly to maintain neural progenitor pool
Gabriel, E., Wason, A., Ramani, A., Gooi, L. M., Keller, P., Pozniakovsky, A., Poser, I., Noack, F., Telugu, N. S., Calegari, F., et al. (2016) · The EMBO Journal · doi:10.15252/embj.201593679 · Cited by 238 · see citing works
“The cilium is the primary microtubule-based organelle that is dynamically regulated, with assembly occurring during cell cycle exit, and disassembly coinciding with cell cycle re-entry (Jackson, 2011; Kim et al , 2011; Paridaen et al , 2013; Guemez-Gamboa et al , 2014).”
Takata, A., Matsumoto, N., & Kato, T. (2017) · Nature Communications · doi:10.1038/ncomms14519 · Cited by 184 · see citing works
“…adult human tissues 44 and plays a key role in stabilization of neuronal cilia 44 , whose contribution to various neural functions including nervous system development and adult neurogenesis 45 , as well as possible involvement in the pathophysiology of schizophrenia 46,47 , have been reported.”
A serotonergic axon-cilium synapse drives nuclear signaling to alter chromatin accessibility
Sheu, S.-H., Upadhyayula, S., Dupuy, V., Pang, S., Deng, F., Wan, J., Walpita, D., Pasolli, H. A., Houser, J., Sanchez-Martinez, S., et al. (2022) · Cell · doi:10.1016/j.cell.2022.07.026 · Cited by 162 · see citing works
Primary Cilia Signaling Shapes the Development of Interneuronal Connectivity
Guo, J., Otis, J. M., Higginbotham, H., Monckton, C., Cheng, J., Asokan, A., Mykytyn, K., Caspary, T., Stuber, G. D., & Anton, E. S. (2017) · Developmental Cell · doi:10.1016/j.devcel.2017.07.010 · Cited by 136 · see citing works
“The critical role of primary cilia in brain development and function is evident in human ciliopathies (Guemez-Gamboa et al., 2014; Guo et al., 2015), where disrupted cilia function lead to brain malformations and behavioral deficits.”
Developmental disruptions underlying brain abnormalities in ciliopathies
Guo, J., Higginbotham, H., Li, J., Nichols, J., Hirt, J., Ghukasyan, V., & Anton, E. S. (2015) · Nature Communications · doi:10.1038/ncomms8857 · Cited by 140 · see citing works
“progenitor formation and organization (1), radial glial progenitor and intermediate progenitor proliferation (2), multipolar-to-bipolar transition of newborn”
Understanding Language from a Genomic Perspective
Graham, S. A., & Fisher, S. E. (2015) · Annual Review of Genetics · doi:10.1146/annurev-genet-120213-092236 · Cited by 135 · see citing works
Elevating Growth Factor Responsiveness and Axon Regeneration by Modulating Presynaptic Inputs
Zhang, Y., Williams, P. R., Jacobi, A., Wang, C., Goel, A., Hirano, A. A., Brecha, N. C., Kerschensteiner, D., & He, Z. (2019) · Neuron · doi:10.1016/j.neuron.2019.04.033 · Cited by 130 · see citing works
“Despite the documented role of primary cilia as a signaling center during development (Guemez-Gamboa et al., 2014; Hilgendorf et al., 2016), their role in mature neurons has not been well characterized (Siljee et al.”
Park, S. M., Lim, J. S., Ramakrishina, S., Kim, S. H., Kim, W. K., Lee, J., Kang, H.-C., Reiter, J. F., Kim, D. S., Kim, H. (., et al. (2018) · Neuron · doi:10.1016/j.neuron.2018.05.039 · Cited by 114 · see citing works
Mechanisms of PINK1, ubiquitin and Parkin interactions in mitochondrial quality control and beyond
Bayne, A. N., & Trempe, J. (2019) · Cellular and Molecular Life Sciences · doi:10.1007/s00018-019-03203-4 · Cited by 113 · see citing works
The Centrosome, a Multitalented Renaissance OrganellePreprint
Vertii, A., Hehnly, H., & Doxsey, S. (2016) · Cold Spring Harbor Perspectives in Biology · doi:10.1101/cshperspect.a025049 · Cited by 108 · see citing works
Planar Organization of Multiciliated Ependymal (E1) Cells in the Brain Ventricular Epithelium
Ohata, S., & Álvarez-Buylla, A. (2016) · Trends in Neurosciences · doi:10.1016/j.tins.2016.05.004 · Cited by 101 · see citing works
Guo, J., Otis, J. M., Suciu, S. K., Catalano, C., Xing, L., Constable, S., Wachten, D., Gupton, S., Lee, J., Lee, A., et al. (2019) · Developmental Cell · doi:10.1016/j.devcel.2019.11.005 · Cited by 100 · see citing works
“A diagnostic feature of many ciliopathies is axonal tract defects (Green et al., 2018; Guadiana et al., 2013; Guemez-Gamboa et al., 2014; Hildebrandt et al., 2011; Reiter and Leroux, 2017; Sarkisian and Guadiana, 2015; Parisi, 2019; Poretti et al., 2007).”
BackgroundBaluška, F., Miller, W. B., & Reber, A. S. (2022) · Biological Journal of the Linnean Society · doi:10.1093/biolinnean/blac005 · Cited by 97 · see citing works
SHARP: hyperfast and accurate processing of single-cell RNA-seq data via ensemble random projectionPreprint
Wan, S., Kim, J., & Won, K. (2020) · Genome Research · doi:10.1101/gr.254557.119 · Cited by 91 · see citing works
“Further, the random matrix that we used for SHARP was highly sparse, whereas the one used by Halko et al. (2011) was not.”
MethodInfluentialHuang, Y., Dai, Y., Li, M., Guo, L., Cao, C., Huang, Y.-T., Ma, R., Qiu, S., Su, X., Zhong, K., et al. (2021) · The Science of The Total Environment · doi:10.1016/j.scitotenv.2021.149043 · Cited by 85 · see citing works
Composition, organization and mechanisms of the transition zone, a gate for the cilium
Park, K., & Leroux, M. R. (2022) · EMBO Reports · doi:10.15252/embr.202255420 · Cited by 83 · see citing works
Mapping of neuronal and glial primary cilia contactome and connectome in the human cerebral cortex
Wu, J. Y., Cho, S.-J., Descant, K. D., Li, P. H., Shapson-Coe, A., Januszewski, M., Berger, D. R., Meyer, C., Casingal, C. R., Huda, A., et al. (2023) · Neuron · doi:10.1016/j.neuron.2023.09.032 · Cited by 81 · see citing works
Bowie, E., & Goetz, S. C. (2020) · eLife · doi:10.7554/elife.51166 · Cited by 77 · see citing works
“and patterning of neural progenitors (Guemez-Gamboa et al., 2014), the migration and laminar placement of interneurons (Higginbotham et al.”
BackgroundNeuronal and astrocytic primary cilia in the mature brain
Sterpka, A., & Chen, X. (2018) · Pharmacological Research · doi:10.1016/j.phrs.2018.10.002 · Cited by 80 · see citing works
“Accordingly, primary cilia regulate cell division [7], development [4,8,9], and tissue regeneration [10].”
BackgroundInfluentialAn Olfactory Cilia Pattern in the Mammalian Nose Ensures High Sensitivity to Odors
Challis, R. C., Tian, H., Wang, J., He, J., Jiang, J., Chen, X., Yin, W., Connelly, T., Ma, L., Yu, C. R., et al. (2015) · Current Biology · doi:10.1016/j.cub.2015.07.065 · Cited by 79 · see citing works
He, Y.-X., Luo, X., Zhou, B., Hu, T., Meng, X., Audano, P., Kronenberg, Z., Eichler, E., Jin, J., Guo, Y., et al. (2019) · Nature Communications · doi:10.1038/s41467-019-12174-w · Cited by 70 · see citing works
mTORC1 signaling and primary cilia are required for brain ventricle morphogenesis
Foerster, P., Daclin, M., Shihavuddin, A., Faucourt, M., Boletta, A., Genovesio, A., & Spassky, N. (2016) · Development · doi:10.1242/dev.138271 · Cited by 74 · see citing works
Electron Microscopic Reconstruction of Functionally Identified Cells in a Neural Integrator
Vishwanathan, A., Daie, K., Ramirez, A. D., Lichtman, J. W., Aksay, E. R. F., & Seung, H. S. (2017) · Current Biology · doi:10.1016/j.cub.2017.06.028 · Cited by 74 · see citing works
Neocortex expansion in development and evolution — from cell biology to single genes
Wilsch‐Bräuninger, M., Florio, M., & Huttner, W. Β. (2016) · Current Opinion in Neurobiology · doi:10.1016/j.conb.2016.05.004 · Cited by 73 · see citing works
“Inhibition of PDGFD–PDGFRb signalling in human cortical slice culture prevented normal cell cycle progression of RG. Conversely, ectopic expression of PDGFRb in developing mouse neocortex increased RG abundance and subventricular displacement.”
BackgroundInfluentialConvergent and Divergent Mechanisms of Epileptogenesis in mTORopathies
Nguyen, L. H., & Bordey, A. (2021) · Frontiers in Neuroanatomy · doi:10.3389/fnana.2021.664695 · Cited by 69 · see citing works
Advances in Dyslexia Genetics—New Insights Into the Role of Brain Asymmetries
Paracchini, S., Diaz, R., & Stein, J. F. (2016) · Advances in genetics · doi:10.1016/bs.adgen.2016.08.003 · Cited by 68 · see citing works
Karunakaran, K. B., Chaparala, S., Lo, C. W., & Ganapathiraju, M. K. (2020) · Scientific Reports · doi:10.1038/s41598-020-72024-4 · Cited by 56 · see citing works
Ohata, S., Herranz‐Pérez, V., Nakatani, J., Boletta, A., García‐Verdugo, J. M., & Álvarez-Buylla, A. (2015) · Journal of Neuroscience · doi:10.1523/jneurosci.0686-15.2015 · Cited by 66 · see citing works
Genome-Wide Association Study of Susceptibility Loci for Radiation-Induced Brain Injury
Wang, T., Shen, G., Chen, M., Zhang, J., Sun, Y. H., He, J., Xue, W., Li, X., Huang, S., Zheng, X., et al. (2018) · JNCI Journal of the National Cancer Institute · doi:10.1093/jnci/djy150 · Cited by 65 · see citing works
“Increasing evidence has indicated that primary cilia plays important roles in coordinating a variety of cellular signaling pathways in neuronal cell and regulate cell migration, differentiation, as well as a host of adult behaviors (34).”
BackgroundHuang, Y., Guo, L., Cao, C., Ma, R., Huang, Y.-T., Zhong, K., Gao, H., Huang, Y., & Bu, Q. (2022) · The Science of The Total Environment · doi:10.1016/j.scitotenv.2022.157047 · Cited by 64 · see citing works
Shortened primary cilium length and dysregulated Sonic hedgehog signaling in Niemann-Pick C1 disease
Canterini, S., Dragotto, J., Dardis, A., Zampieri, S., Stefano, M. E. D., Mangia, F., Erickson, R. P., & Fiorenza, M. T. (2017) · Human Molecular Genetics · doi:10.1093/hmg/ddx118 · Cited by 63 · see citing works
“In fact, recent studies have linked defects of genes encoding for ciliary proteins to various cerebellar disorders (64), laying the basis for a coherent definition and classification of human ciliopathies.”
Tereshko, L., Gao, Y., Cary, B. A., Turrigiano, G. G., & Sengupta, P. (2021) · eLife · doi:10.7554/elife.65427 · Cited by 60 · see citing works
The essential role of primary cilia in cerebral cortical development and disorders
Liu, S., Trupiano, M. X., Simon, J. M., Guo, J., & Anton, E. S. (2021) · Current topics in developmental biology/Current Topics in Developmental Biology · doi:10.1016/bs.ctdb.2020.11.003 · Cited by 59 · see citing works
Trisomy 21 Represses Cilia Formation and Function
Galati, D. F., Sullivan, K. D., Pham, A. T., Espinosa, J. M., & Pearson, C. G. (2018) · Developmental Cell · doi:10.1016/j.devcel.2018.07.008 · Cited by 59 · see citing works
Matsumoto, M., Sawada, M., García‐González, D., Herranz‐Pérez, V., Ogino, T., Nguyen, H. B., Thai, T. Q., Narita, K., Kumamoto, N., Ugawa, S., et al. (2019) · Journal of Neuroscience · doi:10.1523/jneurosci.1503-19.2019 · Cited by 61 · see citing works
“The primary cilium is a tiny cellular protrusion that contains signaling receptors on its membrane (Guemez-Gamboa et al., 2014; Malicki and Johnson, 2017), and regulates neural stem cell proliferation (Breunig et al.”
BackgroundKasioulis, I., Das, R. M., & Storey, K. G. (2017) · eLife · doi:10.7554/elife.26215 · Cited by 59 · see citing works
“Mediators of the mitogenic Sonic hedgehog pathway are processed into activated forms in the primary cilium (Guemez-Gamboa et al., 2014; Kim et al., 2009) and so this may be a further way in which cell biological mechanisms associated with delamination link this process to cell state change.”
BackgroundWaugh, M. G. (2015) · Biochimica et Biophysica Acta (BBA) - Molecular and Cell Biology of Lipids · doi:10.1016/j.bbalip.2015.02.002 · Cited by 57 · see citing works
Primary cilia and autophagic dysfunction in Huntington’s disease
Kaliszewski, M., Knott, A. B., & Bossy-Wetzel, E. (2015) · Cell Death & Differentiation · doi:10.1038/cdd.2015.80 · Cited by 55 · see citing works
“Present in neurons, astrocytes and progenitors, these structures have an important role in neurodevelopment–– acting in neuronal homeostasis, differentiation and survival.(71) Neurological defects associated with so-called ciliopathies,”
BackgroundXu, Y., Cao, J., Huang, S., Feng, D., Zhang, W., Zhu, X., & Yan, X. (2015) · PLoS ONE · doi:10.1371/journal.pone.0124378 · Cited by 56 · see citing works
“Defects in cilia assembly and function result in severe disorders called ciliopathies, including infertility, retinal degeneration, hydrocephalus, polycystic kidney, and situs inversus [2,4,5].”
BackgroundMartin, L., Kaci, N., Estibals, V., Goudin, N., Garfa-Traore, M., Benoist-Lasselin, C., Dambroise, E., & Legeai-Mallet, L. (2018) · Human Molecular Genetics · doi:10.1093/hmg/ddx374 · Cited by 51 · see citing works
“The PC is chemosensitive and controls diverse signaling pathways important in cartilage growth plate development and tissue homeostasis (21,22), including hedgehog (23), Wnt/b-catenin (24), Pdgf (25), cAMP/PKA signaling and cell cycle progression (26).”
BackgroundRethinking the cilia hypothesis of hydrocephalus
Duy, P. Q., Greenberg, A. B. W., Butler, W. E., & Kahle, K. T. (2022) · Neurobiology of Disease · doi:10.1016/j.nbd.2022.105913 · Cited by 51 · see citing works
Beck, T. N., Nicolas, E., Kopp, M. C., & Golemis, E. A. (2014) · Oncoscience · doi:10.18632/oncoscience.64 · Cited by 50 · see citing works
“Further interactions between NEDD9 and AURKA in G0/G1 cells regulate integrity of the cell cilium [84], a structure important for brain patterning and function [85, 86] that is organized around a microtubule-based axoneme.”
BackgroundJurisch‐Yaksi, N., Wachten, D., & Gopalakrishnan, J. (2024) · Trends in Neurosciences · doi:10.1016/j.tins.2024.03.004 · Cited by 50 · see citing works
Diagnostic Approach to Cerebellar Hypoplasia
Accogli, A., Addour-Boudrahem, N., & Srour, M. (2021) · The Cerebellum · doi:10.1007/s12311-020-01224-5 · Cited by 46 · see citing works
The cilium: a cellular antenna with an influence on obesity risk
Mariman, E. C. M., Vink, R., Roumans, N. J. T., Bouwman, F. G., Schrander-Stumpel, C. T. R. M., Aller, E. E. J. G., van Baak, M. A., & Wang, P. (2016) · British Journal Of Nutrition · doi:10.1017/s0007114516002282 · Cited by 46 · see citing works
Shohayeb, B., Ho, U. Y., Yeap, Y. Y. C., Parton, R. G., Millard, S. S., Xu, Z., Piper, M., & Ng, D. C. H. (2019) · Human Molecular Genetics · doi:10.1093/hmg/ddz281 · Cited by 45 · see citing works
MECP2 mutations affect ciliogenesis: a novel perspective for Rett syndrome and related disorders
Frasca, A., Spiombi, E., Palmieri, M., Albizzati, E., Valente, M. M., Bergo, A., Leva, B., Kilstrup‐Nielsen, C., Bianchi, F., Di Carlo, V., et al. (2020) · EMBO Molecular Medicine · doi:10.15252/emmm.201910270 · Cited by 46 · see citing works
“Primary cilium is a non-motile microtubule-based organelle that grows from a centrosome-derived structure, termed the basal body, and protrudes from the cell surface of almost every quiescent or differentiated mammalian cell, including neurons (Guemez-Gamboa et al, 2014).”
BackgroundKang, G. M., Han, Y., Ko, H. W., Kim, J., Oh, B., Kwon, I., & Kim, M. (2015) · Journal of Biological Chemistry · doi:10.1074/jbc.m115.639468 · Cited by 45 · see citing works
Primary cilia modulate TLR4-mediated inflammatory responses in hippocampal neurons
Baek, H., Shin, H. J., Kim, J.-J., Shin, N., Kim, S., Yi, M.-H., Zhang, E., Hong, J., Kang, J. W., Kim, Y., et al. (2017) · Journal of Neuroinflammation · doi:10.1186/s12974-017-0958-7 · Cited by 43 · see citing works
“Primary cilia have well-established roles in the regulation of many signaling pathways [34].”
BackgroundRab7 regulates primary cilia disassembly through cilia excision
Wang, G., Hu, H.-B., Chang, Y., Huang, Y., Song, Z.-Q., Zhou, S., Chen, L., Zhang, Y.-C., Wu, M., Tu, H.-Q., et al. (2019) · The Journal of Cell Biology · doi:10.1083/jcb.201811136 · Cited by 45 · see citing works
Congenital Diseases of DNA Replication: Clinical Phenotypes and Molecular Mechanisms
Schmit, M., & Bielinsky, A. (2021) · International Journal of Molecular Sciences · doi:10.3390/ijms22020911 · Cited by 43 · see citing works
Ciliary Dyneins and Dynein Related Ciliopathies
Antony, D., Brunner, H. G., & Schmidts, M. (2021) · Cells · doi:10.3390/cells10081885 · Cited by 41 · see citing works
“Dysfunction of primary cilia results in multiple developmental defects including cyst formations, fibrosis, skeletal abnormalities, craniofacial defects [4] neural tube defects, cerebellar vermis hypoplasia and photoreceptor defects [28,29].”
BackgroundPrimary cilia TRP channel regulates hippocampal excitability
Vien, T. N., Ta, M. C., Kimura, L. F., Onay, T., & DeCaen, P. G. (2023) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.2219686120 · Cited by 33 · see citing works
“Primary cilia are proposed chemosensory antennae in the CNS—receiving neuro-modulatory inputs through activation of enriched G-protein-coupled receptors (3–6).”
Primary Cilia Dysfunction in Neurodevelopmental Disorders beyond Ciliopathies
Karalis, V., Donovan, K. E., & Şahin, M. (2022) · Journal of Developmental Biology · doi:10.3390/jdb10040054 · Cited by 39 · see citing works
Respiratory motile cilia dysfunction in a patient with cranioectodermal dysplasia
Li, Y., Garrod, A. S., Madan‐Khetarpal, S., Sreedher, G., McGuire, M., Yagi, H., Klena, N., Gabriel, G. C., Khalifa, O., Zahid, M., et al. (2015) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.37133 · Cited by 40 · see citing works
“The finding of extra-axial CSF, brain dysplasia, and small optic nerve have been described in other ciliopathies [Baker and Beales, 2013] and may reflect the combined effects of primary and motile cilia defects [McIntyre et al., 2012; Guemez-Gamboa et al., 2014].”
BackgroundGuadiana, S. M., Parker, A. K., Filho, G. F., Sequeira, A. R., Semple‐Rowland, S. L., Shaw, G., Mandel, R. J., Foster, T. C., Kumar, A., & Sarkisian, M. R. (2016) · Frontiers in Aging Neuroscience · doi:10.3389/fnagi.2016.00127 · Cited by 39 · see citing works
“They are believed to sense and respond to a variety of chemical changes in the local extracellular environment like small ‘‘cellular antennae’’ (for review see: Louvi and Grove, 2011; Guemez-Gamboa et al., 2014; Sarkisian and Guadiana, 2015).”
BackgroundCilia in hereditary cerebral anomalies
Thomas, S., Boutaud, L., Reilly, M. L., & Benmerah, A. (2019) · Biology of the Cell · doi:10.1111/boc.201900012 · Cited by 42 · see citing works
“…(MIM #249000; MKS) syndromes where almost 13 common genes have so far been reported, supporting the notion that MKS represents the extreme lethal form of JBTS. Developmental brain anomalies are among the characteristic and frequent ciliopathy-associated manifestations [Guemez-Gamboa et al., 2014].”
BackgroundGli2 Rescues Delays in Brain Development Induced by Kif3a Dysfunction
Chen, J.-L., Chang, C.-H., & Tsai, J. (2017) · Cerebral Cortex · doi:10.1093/cercor/bhx356 · Cited by 39 · see citing works
“2013) in the primary cilium, an antenna-like protrusion from the cell surface of many cell types, including NEPs, RGCs, and cortical neurons (Dahl 1963; Arellano et al. 2002; Higginbotham et al. 2013; Guemez-Gamboa et al. 2014).”
BackgroundCiliary kinesins beyond IFT: Cilium length, disassembly, cargo transport and signalling
Reilly, M. L., & Benmerah, A. (2019) · Biology of the Cell · doi:10.1111/boc.201800074 · Cited by 40 · see citing works
“Central nervous system defects are also observed in ciliopathies highlighting the important role of cilia during brain development [Guemez-Gamboa et al., 2014; Thomas et al., manuscript in preparation).”
BackgroundFinn, R., Evans, C. C., & Lee, L. (2014) · Neuroscience · doi:10.1016/j.neuroscience.2014.07.029 · Cited by 36 · see citing works
“The immotile primary cilia found on other cell types, which have a 9+0 microtubule structure and a mechanosensory function, are unaffected in PCD patients (Berbari et al., 2009; Guemez-Gamboa et al., 2014).”
BackgroundBrain Dysplasia Associated with Ciliary Dysfunction in Infants with Congenital Heart Disease
Panigrahy, A., Lee, V., Ceschin, R. C., Zuccoli, G., Beluk, N., Khalifa, O., Votava‐Smith, J. K., DeBrunner, M. G., Munoz, R. A., Domnina, Y. A., et al. (2016) · The Journal of Pediatrics · doi:10.1016/j.jpeds.2016.07.041 · Cited by 36 · see citing works
“[42, 43] We note Pde2a, a dual specificity phosphodiesterase [51] known to mediate long-term potentiation and synaptic plasticity [52] was recovered among cilia related genes causing CHD in our mouse mutagenesis screen.”
BackgroundLoss of Rsph9 causes neonatal hydrocephalus with abnormal development of motile cilia in mice
Zou, W., Lv, Y., Liu, Z. I., Xia, P., Li, H., & Jiao, J. (2020) · Scientific Reports · doi:10.1038/s41598-020-69447-4 · Cited by 37 · see citing works
Ma, R., Kutchy, N. A., & Hu, G. (2021) · Biological Psychiatry · doi:10.1016/j.biopsych.2021.06.009 · Cited by 37 · see citing works
Wang, L., Guo, Q., Acharya, S., Zheng, X., Huynh, V., Whitmore, B., Yimit, A., Malhotra, M., Chatterji, S., Rosin, N., et al. (2024) · Nature Neuroscience · doi:10.1038/s41593-024-01726-z · Cited by 33 · see citing works
“Primary cilia serve as a primary mediator for canonical SHH (cSHH) signaling transduction in most mammalian cell types, but their role in mediating cSHH signaling in astrocytes has not been examined 12,29,43 .”
Xiao, H., Zhang, T., Li, C., Cao, Y. B., Wang, L., Chen, H., Li, S., Guan, C., Hu, J., Chen, D., et al. (2022) · eLife · doi:10.7554/elife.73614 · Cited by 37 · see citing works
Mapping GPR88-Venus illuminates a novel role for GPR88 in sensory processing
Ehrlich, A. T., Semache, M., Bailly, J., Wojcik, S., Arefin, T. M., Colley, C., Gouill, C. L., Gross, F., Lukasheva, V., Hogue, M., et al. (2017) · Brain Structure and Function · doi:10.1007/s00429-017-1547-3 · Cited by 36 · see citing works
“…in barrel cortex function and associated sensorimotor behavior (Ferezou et al. 2007; Manita et al. 2015; Schneider et al. 2014; Zhang et al. 2014; Guo et al. 2014), and L6 projections to the thalamus are known to filter and feedback sensory inputs to the cortex (Crandall et al. 2015; Cudeiro and…”
Background5-HT6R null mutatrion induces synaptic and cognitive defects
Sun, Z., Wang, B., Chen, C., Li, C., & Zhang, Y. (2021) · Aging Cell · doi:10.1111/acel.13369 · Cited by 35 · see citing works
Magnani, D., Morlé, L., Hasenpusch‐Theil, K., Paschaki, M., Jacoby, M., Schurmans, S., Durand, B., & Theil, T. (2015) · Human Molecular Genetics · doi:10.1093/hmg/ddv021 · Cited by 35 · see citing works
A neural stem cell paradigm of pediatric hydrocephalus
Duy, P. Q., RAKIC, P. T., Alper, S. L., Robert, S. M., Kundishora, A. J., Butler, W. E., Walsh, C. A., Šestan, N., Geschwind, D. H., Jin, S. C., et al. (2022) · Cerebral Cortex · doi:10.1093/cercor/bhac341 · Cited by 32 · see citing works
Muthusamy, N., Sommerville, L. J., Moeser, A. J., Stumpo, D. J., Sannes, P. L., Adler, K. B., Blackshear, P. J., Weimer, J. M., & Ghashghaei, H. T. (2015) · Aging Cell · doi:10.1111/acel.12354 · Cited by 32 · see citing works
“Primary cilia have emerged as important sensors of circulating proteins in the CSF (Guemez-Gamboa et al., 2014).”
Unique spatiotemporal requirements for intraflagellar transport genes during forebrain development
Snedeker, J., Schock, E. N., Struve, J. N., Chang, C., Cionni, M., Tran, P. V., Brugmann, S. A., & Stottmann, R. W. (2017) · PLoS ONE · doi:10.1371/journal.pone.0173258 · Cited by 31 · see citing works
The Sonic Hedgehog signaling pathway regulates inferior alveolar nerve regeneration
Yamada, Y., Ohazama, A., Maeda, T., & Seo, K. (2018) · Neuroscience Letters · doi:10.1016/j.neulet.2017.12.051 · Cited by 33 · see citing works
Heterogeneous clinical phenotypes and cerebral malformations reflected by rotatin cellular dynamics
Vandervore, L. V., Schot, R., Kasteleijn, E., Oegema, R., Stouffs, K., Gheldof, A., Grochowska, M. M., van der Sterre, M. L. T., van Unen, L., Wilke, M., et al. (2019) · Brain · doi:10.1093/brain/awz045 · Cited by 32 · see citing works
Diverged morphology changes of astrocytic and neuronal primary cilia under reactive insults
Sterpka, A., Yang, J., Strobel, M., Zhou, Y., Pauplis, C., & Chen, X. (2020) · Molecular Brain · doi:10.1186/s13041-020-00571-y · Cited by 30 · see citing works
“including cognitive impairment [12], obesity [13], developmental disorders [14], and glioblastomas [15, 16].”
BackgroundHartwig, C. L., Monis, W. J., Chen, X., Dickman, D. K., Pazour, G. J., & Faúndez, V. (2017) · Developmental Neurobiology · doi:10.1002/dneu.22542 · Cited by 29 · see citing works
“Cilia are required for G-protein-coupled receptor dependent signaling as well as sonic hedgehog- and planar cell polarity-dependent neuronal specification and tissue morphogenesis (Guemez-Gamboa et al., 2014).”
BackgroundInfluentialMice with a conditional deletion of Talpid3 (KIAA0586) – a model for Joubert syndrome
Bashford, A. L., & Subramanian, V. (2019) · The Journal of Pathology · doi:10.1002/path.5271 · Cited by 27 · see citing works
“The primary cilium is an antenna-like non-motile structure which protrudes from the cell and consists of a basal body (mother centriole) and an axoneme [1,2].”
BackgroundBrain‐specific functions of the endocytic machinery
Camblor‐Perujo, S., & Kononenko, N. L. (2021) · FEBS Journal · doi:10.1111/febs.15897 · Cited by 29 · see citing works
Analysis of cilia structure and function in zebrafish
Leventea, E., Hazime, K. S., Zhao, C., & Malicki, J. J. (2016) · Methods in cell biology · doi:10.1016/bs.mcb.2016.04.016 · Cited by 30 · see citing works
Sonic hedgehog signalling pathway contributes in age-related disorders and Alzheimer’s disease
Parashar, A., jha, D., Mehta, V., Chauhan, B., Ghosh, P., Deb, P. K., Jaiswal, M., & Prajapati, S. K. (2024) · Ageing Research Reviews · doi:10.1016/j.arr.2024.102271 · Cited by 28 · see citing works
Impaired hypocretin/orexin system alters responses to salient stimuli in obese male mice
Tan, Y. C., Hang, F., Liu, Z.-W., Stoiljković, M., Wu, M., Tu, Y., Han, W., Lee, A. M., Kelley, C., Hajós, M., et al. (2020) · Journal of Clinical Investigation · doi:10.1172/jci130889 · Cited by 26 · see citing works
Kazatskaya, A., Kuhns, S., Lambacher, N. J., Kennedy, J. E., Brear, A. G., McManus, G., Sengupta, P., & Blacque, O. E. (2017) · Genetics · doi:10.1534/genetics.117.300383 · Cited by 27 · see citing works
“These includemost postmitotic vertebrate cells,whichpossess an immotile or primary cilium that senses and transduces environmental cues (Goetz et al. 2009; Drummond 2012; Ko 2012; Guemez-Gamboa et al.2014).”
BackgroundEmerging Roles of Primary Cilia in Glioma
Sarkisian, M. R., & Semple‐Rowland, S. L. (2019) · Frontiers in Cellular Neuroscience · doi:10.3389/fncel.2019.00055 · Cited by 27 · see citing works
Guen, V. J., Edvardson, S., Fraenkel, N., Fattal‐Valevski, A., Jalas, C., Anteby, I. I., Shaag, A., Dor, T., Gillis, D., Kerem, E., et al. (2017) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.38506 · Cited by 26 · see citing works
Nardo, A. D., Lenoël, I., Winden, K. D., Rühmkorf, A., Modi, M. E., Barrett, L. B., Ercan-Herbst, E., Venugopal, P., Behne, R., Lopes, C. A. M., et al. (2020) · Cell Reports · doi:10.1016/j.celrep.2020.107780 · Cited by 26 · see citing works
“In the CNS, ciliopathies are associated with severe neurodevelopmental outcomes including brain malformations, ASD, and intellectual disability (Bettencourt-Dias et al., 2011; Guemez-Gamboa et al., 2014).”
McKnight, I., Hart, C., Park, I., & Shim, J. W. (2020) · Experimental Neurology · doi:10.1016/j.expneurol.2020.113523 · Cited by 24 · see citing works
“SHH is highly expressed in neural stem cells of the developing cortex, including the radial glia, intermediate progenitors, and outer radial astrocytes (Britto et al., 2002; Guemez-Gamboa et al., 2014; Han et al., 2008).”
BackgroundThe Primary Cilium and Neuronal Migration
Stoufflet, J., & Caillé, I. (2022) · Cells · doi:10.3390/cells11213384 · Cited by 25 · see citing works
“T e third, and now prove to be true, hypothesis was that the PC is a key cellular sensory org nelle [6,7].”
BackgroundPrimary Cilia–An Underexplored Topic in Major Mental Illness
Pruski, M., & Lang, B. (2019) · Frontiers in Psychiatry · doi:10.3389/fpsyt.2019.00104 · Cited by 24 · see citing works
Guarnieri, G., Sarchielli, E., Comeglio, P., Herrera-Puerta, E., Piaceri, I., Nacmias, B., Benelli, M., Kelsey, G., Maggi, M., Gallina, P., et al. (2020) · International Journal of Molecular Sciences · doi:10.3390/ijms21176128 · Cited by 24 · see citing works
“Indeed, recent studies have implicated primary cilia formation in the mechanism regulating neurogenesis both during fetal development [45] and in the adult brain [46].”
BackgroundIshii, S., Sasaki, T., Mohammad, S., Hwang, H. M., Tomy, E., Somaa, F. A., Ishibashi, N., Okano, H., RAKIC, P. T., Hashimoto‐Torii, K., et al. (2020) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.2012482118 · Cited by 23 · see citing works
“Primary cilia are known to play a pivotal role in early normal brain development (28) by regulating the signal transduction of key molecular pathways such as Hedgehog and Wnt signaling (29).”
BackgroundLoss of Primary Cilia Results in the Development of Cancer in the Murine Thyroid Gland.
Lee, J., Yi, S., Chang, J. Y., Kim, J. T., Sul, H. J., Park, K. C., Zhu, X., Cheng, S.-Y., Kero, J. T., Kim, J., et al. (2019) · Europe PMC (PubMed Central) · doi:10.14348/molcells.2018.0430 · Cited by 24 · see citing works
Centrioles are amplified in cycling progenitors of olfactory sensory neurons
Ching, K., & Stearns, T. (2020) · PLoS Biology · doi:10.1371/journal.pbio.3000852 · Cited by 24 · see citing works
Noh, M., Oh, S., Kim, Y.-E., Cha, S. J., Sung, W., Oh, K., Park, Y., Mun, J. Y., Ki, C., Nahm, M., et al. (2025) · Molecular Neurodegeneration · doi:10.1186/s13024-025-00848-7 · Cited by 23 · see citing works
“Primary cilia are microtubule-based, non-motile organelles found in most mammalian cell types, including neurons and astrocytes [18].”
Ciliary biology intersects autism and congenital heart disease
Teerikorpi, N., McCluskey, K. E., Bader, E., Lasser, M. C., Wang, S., Nguyen, C. H., Schmidt, J. D., Kostyanovskaya, E., Sun, N., Dea, J., et al. (2025) · Development · doi:10.1242/dev.204295 · Cited by 22 · see citing works
McKenzie, C. W., Preston, C. C., Finn, R., Eyster, K. M., Faustino, R. S., & Lee, L. (2018) · Scientific Reports · doi:10.1038/s41598-018-31743-5 · Cited by 23 · see citing works
Loss of p73 in ependymal cells during the perinatal period leads to aqueductal stenosis
Fujitani, M., Sato, R., & Yamashita, T. (2017) · Scientific Reports · doi:10.1038/s41598-017-12105-z · Cited by 23 · see citing works
Primary Ciliary Signaling in the Skin—Contribution to Wound Healing and Scarring
Hosio, M., Jaks, V., Lagus, H., Vuola, J., Ogawa, R., & Kankuri, E. M. (2020) · Frontiers in Cell and Developmental Biology · doi:10.3389/fcell.2020.578384 · Cited by 22 · see citing works
“Defects in cilia, as seen in the case of ciliopathies, also affect the nervous system (Guemez-Gamboa et al., 2014).”
BackgroundThe tubulin code specializes neuronal cilia for extracellular vesicle release
Akella, J. S., & Barr, M. M. (2020) · Developmental Neurobiology · doi:10.1002/dneu.22787 · Cited by 21 · see citing works
“In the nervous system, a single primary cilium is observed on neuronal cell bodies and astrocyte glia, while ependymal cells lining the brain have multiple motile cilia (Brailov et al., 2000; Guemez-Gamboa et al., 2014; Händel et al., 1999; Rieke et al., 1987).”
Background
- Authors
- Guemez-Gamboa, A., Coufal, N. G., & Gleeson, J. G.
- Affiliations
- Howard Hughes Medical Institute (US)
- Corresponding author
- Gleeson, Joseph G.
- Venue
- Neuron
- Publisher
- Elsevier BV
- Published
- 2014-05
- Volume
- 82
- Issue
- 3
- Pages
- 511-521
- ISSN
- 0896-6273 (ISSN-L)
- PMC
- PMC4104280
- OpenAlex
- W2074800621
- Cited by
- 296
- References
- 99
- Citations by year
- 2026: 22 · 2025: 29 · 2024: 21 · 2023: 26 · 2022: 27 · 2021: 22 · 2020: 28 · 2019: 34 · 2018: 17 · 2017: 21 · 2016: 23 · 2015: 22 · 2014: 4
- FWCI
- 3.09
- Type
- journal article
- Language
- en
- Open access
- bronze
- doi.org
- Licence
- Locations
- Neuron (publishedVersion)
- PubMed (publishedVersion)
- Europe PMC (PubMed Central) (acceptedVersion)
- PubMed Central (acceptedVersion)
- Funders
- University of California Institute for Mexico and the United States
- National Institute of Neurological Disorders and Stroke (R13NS053097, R01NS048453, R01NS052455)
- 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 › Developmental Neuroscience › Neurogenesis and neuroplasticity mechanisms
- Keywords
- primary cilia, brain development, neuronal polarity, extracellular signaling, neuronal cell fate, neuronal migration, neuronal differentiation
- MeSH terms
- Animals, Brain, Brain Diseases, Cell Movement, Cilia, Humans, Signal Transduction, Cell Polarity
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