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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 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]
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“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]
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“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]
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“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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Gene therapy rescues cilia defects and restores olfactory function in a mammalian ciliopathy model
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A systems-biology approach to understanding the ciliopathy disorders
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Interaction of PACAP with Sonic hedgehog reveals complex regulation of the Hedgehog pathway by PKA
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Cited by
Sorted by citations
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
hESC-Derived Thalamic Organoids Form Reciprocal Projections When Fused with Cortical Organoids
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“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 244
“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 243
“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 192
“…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.”
Metadata
- 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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