| Autor: |
Dobbelaere, Jeroen, Su, Tiffany Y, Erdi, Balazs, Schleiffer, Alexander, Dammermann, Alexander |
| Předmět: |
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| Zdroj: |
EMBO Journal; Aug2023, Vol. 42 Issue 16, p1-29, 29p |
| Abstrakt: |
Cilia are cellular projections that perform sensory and motile functions in eukaryotic cells. A defining feature of cilia is that they are evolutionarily ancient, yet not universally conserved. In this study, we have used the resulting presence and absence pattern in the genomes of diverse eukaryotes to identify a set of 386 human genes associated with cilium assembly or motility. Comprehensive tissue‐specific RNAi in Drosophila and mutant analysis in C. elegans revealed signature ciliary defects for 70–80% of novel genes, a percentage similar to that for known genes within the cluster. Further characterization identified different phenotypic classes, including a set of genes related to the cartwheel component Bld10/CEP135 and two highly conserved regulators of cilium biogenesis. We propose this dataset defines the core set of genes required for cilium assembly and motility across eukaryotes and presents a valuable resource for future studies of cilium biology and associated disorders. Synopsis: Cilia are evolutionarily highly ancient structures, thought to date back to the last common ancestor of all eukaryotes. Here, a combination of comparative genomics and functional analysis in Drosophila and C. elegans was used to define and characterize the core set of genes involved in cilium assembly and motility across eukaryotes.Phylogenetic profiling defines a core set of 386 genes associated with cilium assembly and motility.Comprehensive tissue‐specific depletion and mutant analysis confirms ciliary association of novel genes in Drosophila and C. elegans and reveals candidates for further study.CABLES1, CCDC6, and TEX9 are identified as novel components of a basal body module that is recruited by the centriolar cartwheel component Bld10/CEP135.MAPK15 and ELMOD are highly conserved regulators of multiple steps in the initiation of ciliogenesis. [ABSTRACT FROM AUTHOR] |
| Databáze: |
Complementary Index |
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