ArticleDevelopment (Cambridge, England)2009
Cilia localization is essential for in vivo functions of the Joubert syndrome protein Arl13b/Scorpion.
Article in Development (Cambridge, England), 2009. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 94 papers.
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Who cites it
94 citing papers in PubMed, 149 citations in OpenAlex.
- ARL13B-Cerulean rescues Arl13b-null mouse from embryonic lethality and reveals a role for ARL13B in spermatogenesis.Journal of cell science · 2025Article
- Aurora-A Promotes Cell-Cycle Progression From Quiescence Through Primary Cilia Disassembly.Cancer science · 2025Article
- Primary cilium disappearance in podocytes during vertebrate phylogeny revealed by array tomography.Cell and tissue research · 2025Article
- Novel PIBF1 Pathogenic Variant in Three Siblings with Joubert Syndrome Type 33.Molecular syndromology · 2025Article
- ARL13B-Cerulean rescuesbioRxiv : the preprint server for biology · 2025Article
- Knocking out ARL13B completely abolishes primary ciliogenesis in cell lines.Scientific reports · 2025Article
- Biallelic TEDC1 variants cause a new syndrome with severe growth impairment and endocrine complications.European journal of human genetics : EJHG · 2025Article
- Proteins with proximal-distal asymmetries in axoneme localisation control flagellum beat frequency.Nature communications · 2025Article
- ARL13B controls male reproductive tract physiology through primary and Motile Cilia.Communications biology · 2024Article
- Permanent deconstruction of intracellular primary cilia in differentiating granule cell neurons.The Journal of cell biology · 2024Article
- Male germ cell-associated kinase is required for axoneme formation during ciliogenesis in zebrafish photoreceptors.Disease models & mechanisms · 2024Article
- Molecular and structural perspectives on protein trafficking to the primary cilium membrane.Biochemical Society transactions · 2024Review
- Ciliogenesis defects after neurulation impact brain development and neuronal activity in larval zebrafish.iScience · 2024Article
- Overexpression of SMYD3 Promotes Autosomal Dominant Polycystic Kidney Disease by Mediating Cell Proliferation and Genome Instability.Biomedicines · 2024Article
- Permanent deconstruction of intracellular primary cilia in differentiating granule cell neurons.bioRxiv : the preprint server for biology · 2023Article
- Article
- Dehydration Accelerates Cytogenesis and Cyst Growth in Pkd1Inflammation · 2023Article
- The Zinc Transporter SLC39A10 Plays an Essential Role in Embryonic Hematopoiesis.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2023Article
- Interactions between TULP3 tubby domain and ARL13B amphipathic helix promote lipidated protein transport to cilia.Molecular biology of the cell · 2023Article
- Hedgehog signaling regulates Wolffian duct development through the primary cilium†.Biology of reproduction · 2023Article
34 more citing papers are in PubMed but not listed here.
Corrections and comments
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Authors and funding
3 authors at 1 institution in 1 country.
Funding
Abstract
arl13b was initially cloned as the novel cystic kidney gene scorpion (sco) in zebrafish and was shown to be required for cilia formation in the kidney duct. In mouse, a null mutant of Arl13b shows abnormal ultrastructure of the cilium and defective sonic hedgehog (Shh) signaling. Importantly, a recent study linked mutations in ARL13B to a classical form of Joubert syndrome (JS), an autosomal recessive disorder characterized by a distinctive cerebellar malformation. In this study, we analyzed the zebrafish arl13b (sco) mutant and gene products in detail. We first demonstrate that Arl13b is a protein that is highly enriched in the cilium and is required for cilia formation in multiple organs in zebrafish, and that knockdown of arl13b leads to multiple cilia-associated phenotypes. We additionally show that multiple regions of Arl13b are required for its localization to the cilium. By means of rescuing experiments with a series of deletion and point mutants, we further demonstrate that the ciliary localization is crucial for the in vivo function of Arl13b. Together, these results strongly support the hypothesis that JS-related disease (JSRD) is a ciliopathy, or a disease caused by ciliary defects, and that Arl13b functions mainly through the cilium.
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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.