Primary Cilia Signaling Promotes Axonal Tract Development and Is Disrupted in Joubert Syndrome-Related Disorders Models
Open accessAbstract
Appropriate axonal growth and connectivity are essential for functional wiring of the brain. Joubert syndrome-related disorders (JSRD), a group of ciliopathies in which mutations disrupt primary cilia function, are characterized by axonal tract malformations. However, little is known about how cilia-driven signaling regulates axonal growth and connectivity. We demonstrate that the deletion of related JSRD genes, Arl13b and Inpp5e, in projection neurons leads to de-fasciculated and misoriented axonal tracts. Arl13b deletion disrupts the function of its downstream effector, Inpp5e, and deregulates ciliary-PI3K/AKT signaling. Chemogenetic activation of ciliary GPCR signaling and cilia-specific optogenetic modulation of downstream second messenger cascades (PI3K, AKT, and AC3) commonly regulated by ciliary signaling receptors induce rapid changes in axonal dynamics. Further, Arl13b deletion leads to changes in transcriptional landscape associated with dysregulated PI3K/AKT signaling. These data suggest that ciliary signaling acts to modulate axonal connectivity and that impaired primary cilia signaling underlies axonal tract defects in JSRD.
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The mother centriole synthesizes and releases acetylated microtubules for transport down the axon and the primary cilia, and modulation of primary cilia signaling such as AKT and AC3 controls axonal behavior.
“The mother centriole synthesizes and releases acetylated microtubules for transport down the axon and the primary cilia, and modulation of primary cilia signaling such as AKT and AC3 controls axonal behavior [1, 26].”
From Abnormal accumulation of extracellular vesicles in hippocampal dystrophic axons and regulation by the primary cilia in Alzheimer's disease (2023) · cites this paper as [26]
The Results section reports that manipulating ciliary ACIII and AKT signaling changes axonal growth-cone behavior. Neither the supplied passages nor searches of our copy support the separate assertion that the mother centriole synthesizes and releases acetylated microtubules for transport down the axon and primary cilium. The indexed passages have no page numbers. (LLM, conf=0.88)
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“The mother centriole synthesizes and releases acetylated microtubules for transport down the axon and the primary cilia, and modulation of primary cilia signaling such as AKT and AC3 controls axonal behavior [1, 26].”
BackgroundHTR6 and SSTR3 targeting to primary cilia
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The developing brain by trimester
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The primary cilia are associated with the axon initial segment in neurons
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The Role of Primary Cilia-Associated Phosphoinositide Signaling in Development
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- Authors
- Guo, J., Otis, J. M., Suciu, S. K., Catalano, C., Xing, L., Constable, S., Wachten, D., Gupton, S., Lee, J., Lee, A., Blackley, K. H., Ptacek, T., Simon, J. M., Schurmans, S., Stuber, G. D., Caspary, T., & Anton, E. S.
- Affiliations
- University of North Carolina at Chapel Hill (US)
- Emory University (US)
- University of Calgary (CA)
- University of Bonn (DE)
- University of Washington (US)
- Corresponding author
- Guo, Jiami
- Caspary, Tamara
- Anton, E.S.
- ORCID
- Venue
- Developmental Cell
- Publisher
- Elsevier BV
- Published
- 2019-12
- Volume
- 51
- Issue
- 6
- Pages
- 759-774.e5
- ISSN
- 1534-5807 (ISSN-L)
- PMC
- PMC6953258
- OpenAlex
- W2995406279
- Cited by
- 100
- References
- 119
- Citations by year
- 2026: 7 · 2025: 12 · 2024: 21 · 2023: 15 · 2022: 15 · 2021: 18 · 2020: 10 · 2019: 1
- FWCI
- 6.61
- Type
- journal article
- Language
- en
- Open access
- bronze
- doi.org
- Licence
- Locations
- Developmental Cell (publishedVersion)
- PubMed (publishedVersion)
- PubMed Central (acceptedVersion)
- Open Repository and Bibliography (University of Liège) (submittedVersion, other-oa)
- MPG.PuRe (Max Planck Society) (submittedVersion)
- Funders
- National Institute of Neurological Disorders and Stroke
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetic and Kidney Cyst Diseases
- Life Sciences › Neuroscience › Cellular and Molecular Neuroscience › Axon Guidance and Neuronal Signaling
- Health Sciences › Medicine › Pulmonary and Respiratory Medicine › Cystic Fibrosis Research Advances
- Keywords
- Joubert syndrome-related disorders, ciliopathies, ARL13B, INPP5E, PI3K/Akt signaling, axon fasciculation, axon guidance, optogenetics, chemogenetics, projection neurons, axonal connectivity
- MeSH terms
- Abnormalities, Multiple, Animals, Axons, Cerebellum, Cilia, Disease Models, Animal, Eye Abnormalities, Mutation, Retina, Mice, Kidney Diseases, Cystic, Neurogenesis
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