ArticleCell death & disease2014
Mutation of a Nopp140 gene dao-5 alters rDNA transcription and increases germ cell apoptosis in C. elegans.
Article in Cell death & disease, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers.
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Who cites it
18 citing papers in PubMed, 28 citations in OpenAlex.
- rRNA intermediates associate with nucleolar reshaping in C. elegans.Nucleic acids research · 2026Article
- RNA Pol I shapes meiotic chromatin, germline H3K4me3 dynamics, and oogenesis independent of ribosome assembly.Cell reports · 2026Article
- ENY2 transcription and export complex 2 subunit deficiency induces nucleolar stress to inhibit tumor progression through NPM1/MDM2/p53-dependent and -independent responses.Cellular oncology (Dordrecht, Netherlands) · 2026Article
- RNA Pol I activity is required for meiotic chromatin organization and the H3K4me3 gradient essential for oogenesis, independent of ribosome synthesis.bioRxiv : the preprint server for biology · 2025Article
- Phenotypic tolerance for rDNA copy number variation within the natural range of C. elegans.PLoS genetics · 2025Article
- DRMY1 promotes robust morphogenesis in Arabidopsis by sustaining the translation of cytokinin-signaling inhibitor proteins.Developmental cell · 2024Article
- Neuronal IL-17 controlsProceedings of the National Academy of Sciences of the United States of America · 2024Article
- Substantial rDNA copy number reductions alter timing of development and produce variable tissue-specific phenotypes in C. elegans.Genetics · 2023Article
- Antisense ribosomal siRNAs inhibit RNA polymerase I-directed transcription in C. elegans.Nucleic acids research · 2021Article
- An essential role for the piRNA pathway in regulating the ribosomal RNA pool in C. elegans.Developmental cell · 2021Article
- Single-Molecule Tracking of Chromatin-Associated Proteins in theThe journal of physical chemistry. B · 2021Article
- Nucleolar stress inBiology open · 2020Article
- Inhibitor of Growth 4 (ING4) is a positive regulator of rRNA synthesis.Scientific reports · 2019Article
- The Nopp140 gene in Drosophila melanogaster displays length polymorphisms in its large repetitive second exon.Molecular genetics and genomics : MGG · 2019Article
- Developmental dynamics of gene expression and alternative polyadenylation in the Caenorhabditis elegans germline.Genome biology · 2018Article
- Genetic control of nucleolar size: An evolutionary perspective.Nucleus (Austin, Tex.) · 2016Review
- Nuclear bodies: the emerging biophysics of nucleoplasmic phases.Current opinion in cell biology · 2015Review
- Nucleolar stress with and without p53.Nucleus (Austin, Tex.)Review
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Authors and funding
9 authors at 3 institutions in 1 country.
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No grant is acknowledged in the PubMed record.
Abstract
Human diseases of impaired ribosome biogenesis resulting from disruption of rRNA biosynthesis or loss of ribosomal components are collectively described as 'ribosomopathies'. Treacher Collins syndrome (TCS), a representative human ribosomopathy with craniofacial abnormalities, is attributed to mutations in the tcof1 gene that has a homologous gene called nopp140. Previous studies demonstrated that the dao-5 (dauer and aged animal overexpression gene 5) of Caenorhabditis elegans is a member of nopp140 gene family and plays a role in nucleogenesis in the early embryo. Here, we established a C. elegans model for studying Nopp140-associated ribosomopathy. A null dao-5 mutant ok542 with a semi-infertile phenotype showed a delay in gonadogenesis, as well as a higher incidence of germline apoptosis. These phenotypes in dao-5(ok542) are likely resulted from inefficient rDNA transcription that was observed by run-on analyses and chromatin immunoprecipitation (ChIP) assays measuring the RNA Pol I occupancy on the rDNA promoter. ChIP assays further showed that the modifications of acetylated histone 4 (H4Ac) and dimethylation at the lysine 9 of histone 3 (H3K9me2) around the rDNA promoter were altered in dao-5 mutants compared with the N2 wild type. In addition, activated CEP-1 (a C. elegans p53 homolog) activity was also linked to the loss of DAO-5 in terms of the transcriptional upregulation of two CEP-1 downstream effectors, EGL-1 and CED-13. We propose that the dao-5 mutant of C. elegans can be a valuable model for studying human Nopp140-associated ribosomopathy at the cellular and molecular levels.
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