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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.

Zebrafish Tsc1 reveals functional interactions between the cilium and the TOR pathway