ArticleCell reports2019
A CEP104-CSPP1 Complex Is Required for Formation of Primary Cilia Competent in Hedgehog Signaling.
Article in Cell reports, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 35 papers.
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Who cites it
35 citing papers in PubMed, 56 citations in OpenAlex.
- Unraveling the molecular details of centriole elongation.Trends open · 2026Article
- Intron Retention as a Homeostatic State Variable for Drug Response and Recovery: Lessons from Depression for Broader Applications.International journal of molecular sciences · 2026Review
- Clinical, genetic and bioinformatic analysis of Saudi families with Joubert syndrome and related disorders.Human genomics · 2026Article
- Development of Glioblastoma Multiforme in Patients With Human Papillomavirus-Positive Oropharyngeal Squamous Cell Carcinoma: A Case Series.Cancer reports (Hoboken, N.J.) · 2026Article
- Broadening horizons: new links between cilia and heart development and disease.Frontiers in cardiovascular medicine · 2026Review
- A protein complex in the extreme distal tip of vertebrate motile cilia controls their organization, length, and function.Nature communications · 2025Article
- Primary cilia function as hubs for signal transduction.Cell & bioscience · 2025Review
- Article
- Kinase activity of DYRK family members is required for regulating primary cilium length, stability and morphology.Communications biology · 2025Article
- NetStart 2.0: prediction of eukaryotic translation initiation sites using a protein language model.BMC bioinformatics · 2025Article
- The Mn-motif protein MAP6d1 assembles ciliary doublet microtubules.Nature communications · 2025Article
- CCDC66 regulation of cytoskeleton and cilia stability is important for signaling and epithelial organization.PLoS biology · 2025Article
- A protein complex in the extreme distal tip of vertebrate motile cilia controls their organization, length, and function.bioRxiv : the preprint server for biology · 2025Article
- A network of interacting ciliary tip proteins with opposing activities imparts slow and processive microtubule growth.Nature structural & molecular biology · 2025Article
- Axonemal microtubule dynamics in the assembly and disassembly of cilia.Biochemical Society transactions · 2025Review
- Integrative bioinformatics approach identifies novel drug targets for hyperaldosteronism, with a focus on SHMT1 as a promising therapeutic candidate.Scientific reports · 2025Article
- CSPP1 stabilizes microtubules by capping both plus and minus ends.Journal of molecular cell biology · 2024Article
- Novel compound heterozygous variants in theFrontiers in pediatrics · 2024Article
- Cep104 is a component of the centriole distal tip complex that regulates centriole growth and contributes to Drosophila spermiogenesis.Current biology : CB · 2023Article
- An RFX transcription factor regulates ciliogenesis in the closest living relatives of animals.Current biology : CB · 2023Article
Corrections and comments
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Authors and funding
16 authors at 7 institutions in 5 countries.
Funding
Abstract
CEP104 is an evolutionarily conserved centrosomal and ciliary tip protein. CEP104 loss-of-function mutations are reported in patients with Joubert syndrome, but their function in the etiology of ciliopathies is poorly understood. Here, we show that cep104 silencing in zebrafish causes cilia-related manifestations: shortened cilia in Kupffer's vesicle, heart laterality, and cranial nerve development defects. We show that another Joubert syndrome-associated cilia tip protein, CSPP1, interacts with CEP104 at microtubules for the regulation of axoneme length. We demonstrate in human telomerase reverse transcriptase-immortalized retinal pigmented epithelium (hTERT-RPE1) cells that ciliary translocation of Smoothened in response to Hedgehog pathway stimulation is both CEP104 and CSPP1 dependent. However, CEP104 is not required for the ciliary recruitment of CSPP1, indicating that an intra-ciliary CEP104-CSPP1 complex controls axoneme length and Hedgehog signaling competence. Our in vivo and in vitro analyses of CEP104 define its interaction with CSPP1 as a requirement for the formation of Hedgehog signaling-competent cilia, defects that underlie Joubert syndrome.
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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.