ArticleHuman molecular genetics2009
Zebrafish Tsc1 reveals functional interactions between the cilium and the TOR pathway.
Article in Human molecular genetics, 2009. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 55 papers.
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
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Who cites it
55 citing papers in PubMed, 84 citations in OpenAlex.
- A loss of primary cilia by a reduction in mTOR signaling correlates with age-related deteriorations in condylar cartilage.GeroScience · 2024Article
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- Esrrγa regulates nephron and ciliary development by controlling prostaglandin synthesis.Development (Cambridge, England) · 2023Article
- Review
- Testicular miRNAs and tsRNAs provide insight into gene regulation during overwintering and reproduction of Onychostoma macrolepis.Fish physiology and biochemistry · 2022Article
- Islet cilia and glucose homeostasis.Frontiers in cell and developmental biology · 2022Review
- Role of TSC1 in physiology and diseases.Molecular and cellular biochemistry · 2021Review
- Functional aspects of primary cilium in signaling, assembly and microenvironment in cancer.Journal of cellular physiology · 2021Review
- Zebrafish Cancer Predisposition Models.Frontiers in cell and developmental biology · 2021Review
- Zebrafish Models for Human Skeletal Disorders.Frontiers in genetics · 2021Review
- Ppargc1a Controls Ciliated Cell Development by Regulating Prostaglandin Biosynthesis.Cell reports · 2020Article
- Crosstalk of Hedgehog and mTORC1 Pathways.Cells · 2020Review
- Myogenin is an essential regulator of adult myofibre growth and muscle stem cell homeostasis.eLife · 2020Article
- Protein phosphatase 1α interacts with a novel ciliary targeting sequence of polycystin-1 and regulates polycystin-1 trafficking.FASEB journal : official publication of the Federation of American Societies for Experimental Biology · 2019Article
- Imbalanced mitochondrial function provokes heterotaxy via aberrant ciliogenesis.The Journal of clinical investigation · 2019Article
- Prenatal Neuropathologies in Autism Spectrum Disorder and Intellectual Disability: The Gestation of a Comprehensive Zebrafish Model.Journal of developmental biology · 2018Review
- Review
- TSC1 and TSC2 regulate cilia length and canonical Hedgehog signaling via different mechanisms.Cellular and molecular life sciences : CMLS · 2018Article
- Fibrous cephalic plaques in tuberous sclerosis complex.Journal of the American Academy of Dermatology · 2018Article
- Cell volume changes contribute to epithelial morphogenesis in zebrafish Kupffer's vesicle.eLife · 2018Article
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Authors and funding
3 authors at 1 institution in 1 country.
Funding
Abstract
The cell surface organelle called the cilium is essential for preventing kidney cyst formation and for establishing left-right asymmetry of the vertebrate body plan. Recent advances suggest that the cilium functions as a sensory organelle in vertebrate cells for multiple signaling pathways such as the hedgehog and the Wnt pathways. Prompted by kidney cyst formation in tuberous sclerosis complex (TSC) patients and rodent models, we investigated the role of the cilium in the TSC-target of rapamycin (TOR) pathway using zebrafish. TSC1 and TSC2 genes are causal for TSC, and their protein products form a complex in the TOR pathway that integrates environmental signals to regulate cell growth, proliferation and survival. Two TSC1 homologs were identified in zebrafish, which we refer to as tsc1a and tsc1b. Morpholino knockdown of tsc1a led to a ciliary phenotype including kidney cyst formation and left-right asymmetry defects. Tsc1a was observed to localize to the Golgi, but morpholinos against it, nonetheless, acted synthetically with ciliary genes in producing kidney cysts. Consistent with a role of the cilium in the same pathway as Tsc genes, the TOR pathway is aberrantly activated in ciliary mutants, resembling the effect of tsc1a knockdown. Moreover, kidney cyst formation in ciliary mutants was blocked by the Tor inhibitor, rapamycin. Surprisingly, we observed elongation of cilia in tsc1a knockdown animals. Together, these data suggest a signaling network between the cilium and the TOR pathway in that ciliary signals can feed into the TOR pathway and that Tsc1a regulates the length of the cilium itself.
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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.