Caenorhabditis elegans
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PROVIDER: PRJNA133943 | ENA |
REPOSITORIES: ENA
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Project description:Cilia are ubiquitous cell surface projections that modulate various sensory- and motility based processes and are implicated in a growing number of multi-organ genetic disorders termed ciliopathies. As new components required for cilium biogenesis and function remain unidentified, we sought to further define and validate the transcriptional targets of the ciliogenic C. elegans RFX transcription factor DAF-19. To this end, transcriptional profiling of daf-19 mutants (which do not form cilia) and wild-type animals was performed using selectively staged embryos where ciliogenesis occurs in most ciliated sensory neurons (CSNs). Statistical comparisons between the two populations revealed 881 differentially regulated genes with 1.5-fold change or greater. A subset of these was confirmed by quantitative RT-PCR. Transgenic worms expressing transcriptional-GFP fusions revealed CSN-specific expression patterns for 9 of 12 candidate genes. We show that two uncharacterized candidate genes, which we term dyf-17 and dyf-18 because their corresponding mutants display dye-filling (Dyf) defects, are important for ciliogenesis. DYF-17 localizes at the base of cilia and interestingly, is specifically required for building the distal segment of sensory cilia. DYF-18 is an evolutionarily conserved CDK-7/CCRK-related serine-threonine kinase that is necessary for the proper function of intraflagellar transport (IFT), a process critical for cilium biogenesis. Together, our comparative microarray study identifies targets of the evolutionarily conserved RFX transcription factor, DAF-19, providing a rich dataset from which to uncover—in addition to DYF-17 and DYF-18—cellular components important for cilium formation and function. 4 daf-19,daf12; 4 daf-12; 4 WT
2011-07-25 | E-GEOD-25633 | biostudies-arrayexpress
Project description:Cilia are ubiquitous cell surface projections that modulate various sensory- and motility based processes and are implicated in a growing number of multi-organ genetic disorders termed ciliopathies. As new components required for cilium biogenesis and function remain unidentified, we sought to further define and validate the transcriptional targets of the ciliogenic C. elegans RFX transcription factor DAF-19. To this end, transcriptional profiling of daf-19 mutants (which do not form cilia) and wild-type animals was performed using selectively staged embryos where ciliogenesis occurs in most ciliated sensory neurons (CSNs). Statistical comparisons between the two populations revealed 881 differentially regulated genes with 1.5-fold change or greater. A subset of these was confirmed by quantitative RT-PCR. Transgenic worms expressing transcriptional-GFP fusions revealed CSN-specific expression patterns for 9 of 12 candidate genes. We show that two uncharacterized candidate genes, which we term dyf-17 and dyf-18 because their corresponding mutants display dye-filling (Dyf) defects, are important for ciliogenesis. DYF-17 localizes at the base of cilia and interestingly, is specifically required for building the distal segment of sensory cilia. DYF-18 is an evolutionarily conserved CDK-7/CCRK-related serine-threonine kinase that is necessary for the proper function of intraflagellar transport (IFT), a process critical for cilium biogenesis. Together, our comparative microarray study identifies targets of the evolutionarily conserved RFX transcription factor, DAF-19, providing a rich dataset from which to uncover—in addition to DYF-17 and DYF-18—cellular components important for cilium formation and function.
2011-07-26 | GSE25633 | GEO
Project description:Intraflagellar transport (IFT) is a highly conserved mechanism for motor-driven transport of cargo inside cilia. However, the mechanism of selective transport of cargo to cilia and entry across the diffusion barrier is not well understood. WDR35/IFT121 is a component of the IFT-A complex, best known for ciliary retrograde transport. Small Wdr35 mutant cilia are formed but fail to enrich in diverse classes of ciliary membrane proteins. Whilst the assembly of the IFT-B complex is unaffected, these exhibit retrograde trafficking defects in Wdr35 mutants. In contrast, the IFT-A peripheral components are degraded and core components remain stuck at the cilia base. The deep sequence homology and structural similarity of WDR35 and other IFT-As to the coatomer COPI proteins α and ß’, and accumulation of electron-light vesicles around Wdr35 mutant cilia, suggest that WDR35 is required to form coated vesicles delivering cargo from the Golgi necessary for cilia elongation. This is the first insitu proof towards a novel coatomer function for WDR35 and likely other IFT-A proteins in transporting ciliary membrane proteins from the Golgi.
2021-05-03 | PXD022652 | Pride
Project description:The mechanisms of how highly conserved intraflagellar transport (IFT) protein complexes direct ciliary entry remain unknown. Exploring human ciliopathy genes, including monogenic obesity syndrome genes, has been a rich source of critical ciliary mechanisms. Using AP-MS purification of interactors of the CEP19 monogenic obesity gene, we identify factors and the first known mechanism for triggering ciliary entry of IFT complexes. CEP19 binds the CEP350 and FOP centriolar components and the highly conserved RABL2 GTPase. We discovered that the CEP19-RABL2 complex is first recruited by the centriolar CEP350/FOP complex, concurrently activates nucleotide exchange to capture and release intraflagellar transport B holocomplexes from a large centriolar pool to intraflagellar transport. The CEP19 network of interacting proteins regulates ciliary entry and ciliation, providing a new example of ciliary mechanisms deficient in monogenic obesity syndromes.
2017-06-21 | PXD006757 | Pride
Project description:Cilia are dynamic antennae that sense the extracellular environment adjusting their length to maintain homeostasis. Mutant mouse models for the lysine demethylase KDM3A (JMJD1A, JMHD2A) share phenotypic features with human ciliopathies, including obesity and metabolic syndrome. Here we show that cilia of Kdm3a mutants are unstable with dysregulated length ranges accumulating intraflagellar transport proteins (IFTs) at the ciliary tip. RNA sequencing and mass-spectrometry identified actin cytoskeleton as the most miss-regulated feature during the ciliary cycle of KDM3A null cells and revealed that IFT81 contains Nε-methylated lysines in vivo to which recombinant KDM3A binds without subsequent demethylation. Mutations in these IFT81 methyl-lysine residues however stabilize KDM3A null cilia and potentiate ciliogenesis surpassing wild type cells; a synergism phenocopied by wild type cultures through the simultaneous destabilization of the actin cytoskeleton when over-expressing IFT81 lysine-mutants. Our work reveals that ciliogenesis requires the coordinated release of cytoskeletal constrains and the presence of intraflagellar transport proteins which KDM3A integrates aided by post-translationally modifiable lysine residues in IFT81.
2018-07-11 | PXD004334 | Pride
Project description:Cilia are required for many sensory and motility related functions and are involved in growing number of genetic disorders termed ciliopathies. DAF-19, RFX type transcription factor controls expression of many cilia structure and function related genes. To this end, transcriptional profiling of daf-19 mutants (which do not form cilia) and wild-type animals was performed using selectively staged larval stages where cilia mature and maintained, structurally as well as functionally.
2018-01-31 | GSE96068 | GEO
Project description:Cilia are microtubule-based hair-like organelles propelling locomotion and extracellular liquid flow or sensing environmental stimuli. As cilia are diffusion barrier-gated subcellular compartments, their protein components are thought to come from the cell body through intraflagellar transport or diffusion. Here we show that cilia locally synthesize proteins to maintain their ultrastructure and functions. Multicilia of mouse ependymal cells were abundant in ribosomal proteins, translation initiation factors, and RNA, including 18S rRNA and tubulin mRNA. The cilia actively generated nascent peptides, including those of tubulin. FMRP, an RNA-binding component of messenger ribonucleoprotein granules critical for local translation, was concentrated in ciliary central lumen. Its depletion by RNAi impaired ciliary nascent peptide production and induced multicilia degeneration. Expression of exogenous FMRP, but not an isoform tethered to mitochondria, rescued the degeneration defects. Therefore, local translation defects in cilia might contribute to the pathology of ciliopathies and other diseases such as Fragile X syndrome.
2021-10-19 | GSE179935 | GEO
Project description:By screening for genes possessing canonical X-box sequences in promoters of three Caenorhabditis species, namely C. elegans, C. briggsae and C. remanei, we identified 93 genes (including known X-box regulated genes) that encode putative components of ciliated neurons in C. elegans and are subject to the same regulatory control. For many of these genes, restricted anatomical expression in ciliated cells was confirmed, and control of transcription by the ciliogenic DAF-19 RFX transcription factor was demonstrated by comparative transcriptional profiling of daf-19(+) and daf-19(-) animals. Keywords: Microarry analysis; DAF-19; X-box motif; C. elegans; transcription; comparative genomics
2006-12-21 | GSE6563 | GEO
Project description:Osteoblast-derived intraflagellar transport 140 suppresses insulin sensitivity
| PRJNA739651 | ENA
Project description:To check cdk-12 and DAF-16 dependent gene expression change in different genetic backgrounds. We have collected the synchronized late L4 worms and isolated the total RNA and did the mRNA sequencing.
2023-08-25 | E-MTAB-11189 | biostudies-arrayexpress