ArticleMolecular biology of the cell2016
Arl13b regulates Shh signaling from both inside and outside the cilium.
Article in Molecular biology of the cell, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Cited by 54 papers.
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Who cites it
54 citing papers in PubMed, 81 citations in OpenAlex.
- Article
- Functions and trafficking mechanisms of RIC-8 inMolecular biology of the cell · 2026Article
- The nociceptor primary cilium.Brain : a journal of neurology · 2026Review
- Cell-specific roles for the conserved Galpha chaperone RIC-8 in cilia biology.bioRxiv : the preprint server for biology · 2026Article
- ARL13B-Cerulean rescues Arl13b-null mouse from embryonic lethality and reveals a role for ARL13B in spermatogenesis.Journal of cell science · 2025Article
- Ciliary ARL13B Is Essential for Vision and Morphogenesis of Cone Outer Segments.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2025Article
- ARL13B-Cerulean rescuesbioRxiv : the preprint server for biology · 2025Article
- Knocking out ARL13B completely abolishes primary ciliogenesis in cell lines.Scientific reports · 2025Article
- Biallelic TXNDC15 variants associated with Joubert syndrome-related molar tooth sign and forebrain malformation.Journal of human genetics · 2025Article
- A cell-autonomous role for primary cilium-mediated signaling in long-range commissural axon guidance.Development (Cambridge, England) · 2024Article
- Molecular and structural perspectives on protein trafficking to the primary cilium membrane.Biochemical Society transactions · 2024Review
- Thalamic Neuron Resilience during Osmotic Demyelination Syndrome (ODS) Is Revealed by Primary Cilium Outgrowth and ADP-ribosylation factor-like protein 13B Labeling in Axon Initial Segment.International journal of molecular sciences · 2023Article
- Article
- Ciliary ARL13B inhibits developmental kidney cystogenesis in mouse.Developmental biology · 2023Article
- Interactions between TULP3 tubby domain and ARL13B amphipathic helix promote lipidated protein transport to cilia.Molecular biology of the cell · 2023Article
- Trisomy 21 induces pericentrosomal crowding delaying primary ciliogenesis and mouse cerebellar development.eLife · 2023Article
- The role of a ciliary GTPase in the regulation of neuronal maturation of olfactory sensory neurons.Development (Cambridge, England) · 2023Article
- Arl13b controls basal cell stemness properties and Hedgehog signaling in the mouse epididymis.Cellular and molecular life sciences : CMLS · 2022Article
- Loss of ARL13 impedes BBSome-dependent cargo export from Chlamydomonas cilia.The Journal of cell biology · 2022Article
- Phylogenetic profiling and cellular analyses of ARL16 reveal roles in traffic of IFT140 and INPP5E.Molecular biology of the cell · 2022Article
Corrections and comments
- Erratum issuedCorrection for Arl13b regulates Shh signaling from both inside and outside the cilium.2017 · Ivanova, Anna I [corrected to Ivanova, Anna A]
Authors and funding
6 authors at 2 institutions in 2 countries.
Funding
Abstract
The regulatory GTPase Arl13b localizes to primary cilia, where it regulates Sonic hedgehog (Shh) signaling. Missense mutations in ARL13B can cause the ciliopathy Joubert syndrome, while the mouse null allele is embryonic lethal. We used mouse embryonic fibroblasts as a system to determine the effects of Arl13b mutations on Shh signaling. We tested a total of seven different mutants, three JS-causing variants, two point mutants predicted to alter guanine nucleotide handling, one that disrupts cilia localization, and one that prevents palmitoylation and thus membrane binding, in assays of transcriptional and non-transcriptional Shh signaling. We found that mutations disrupting Arl13b's palmitoylation site, cilia localization signal, or GTPase handling altered the Shh response in distinct assays of transcriptional or non-transcriptional signaling. In contrast, JS-causing mutations in Arl13b did not affect Shh signaling in these same assays, suggesting these mutations result in more subtle defects, likely affecting only a subset of signaling outputs. Finally, we show that restricting Arl13b from cilia interferes with its ability to regulate Shh-stimulated chemotaxis, despite previous evidence that cilia themselves are not required for this non-transcriptional Shh response. This points to a more complex relationship between the ciliary and non-ciliary roles of this regulatory GTPase than previously envisioned.
Identifiers
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.