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Significance Using ribosome profiling of cilia regeneration in green algae Chlamydomonas, we uncovered that ciliary genes undergo an early transcriptional activation and late translational suppression, the pattern of which guided… Click to show full abstract
Significance Using ribosome profiling of cilia regeneration in green algae Chlamydomonas, we uncovered that ciliary genes undergo an early transcriptional activation and late translational suppression, the pattern of which guided us to identify serine palmitoyltransferase (SPT) as an essential regulator for ciliogenesis. We found that SPT-product ceramides interact with intraflagellar transport particle proteins, connecting the ciliary membrane with the axoneme, a bundle of microtubules and associated proteins forming the core of cilia. SPT mutations from patients with hereditary sensory neuropathy disrupted cilia, which could be rescued by sphingolipid supplementation, implying an effective therapeutic strategy.
Journal Title: Proceedings of the National Academy of Sciences of the United States of America
Year Published: 2022
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