ArticleMolecular and cellular biology2003
Fibrillarin is essential for early development and required for accumulation of an intron-encoded small nucleolar RNA in the mouse.
Article in Molecular and cellular biology, 2003. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 62 papers.
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Who cites it
62 citing papers in PubMed, 114 citations in OpenAlex.
- Muscular dystrophy-associated lamin variants disrupt cellular organization through a nucleolar-ribosomal axis.Science advances · 2026Article
- Muscular dystrophy-associated lamin variants disrupt cellular organization through a nucleolar-ribosomal axis controlling cytoplasmic macromolecular crowding.bioRxiv : the preprint server for biology · 2025Article
- SNORD88B promotes proliferation and invasion of trophoblast by regulating G3BP1 m6 A modification and alternative splicing.Cellular and molecular life sciences : CMLS · 2025Article
- ZNHIT3 Regulates Translation to Ensure Cell Lineage Differentiation in Mouse Preimplantation Development.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2025Article
- Emerging roles for the nucleolus in development and stem cells.Development (Cambridge, England) · 2025Review
- Ribosome biogenesis rate, a parameter of sensitivity to chemotherapeutic drugs inhibiting rRNA synthesis.Oncology reviews · 2025Review
- Fibrillarin homologs regulate translation in divergent cell lineages during planarian homeostasis and regeneration.The EMBO journal · 2024Article
- Decoding the molecular script of 2'-O-ribomethylation: Implications across CNS disorders.Heliyon · 2024Review
- 2'-RNA (New York, N.Y.) · 2024Review
- Fibrillarin promotes homologous recombination repair by facilitating the recruitment of recombinase RAD51 to DNA damage sites.Journal of Zhejiang University. Science. B · 2023Article
- A snoRNA-tRNA modification network governs codon-biased cellular states.Proceedings of the National Academy of Sciences of the United States of America · 2023Article
- Fallopian tube lesions as potential precursors of early ovarian cancer: a comprehensive proteomic analysis.Cell death & disease · 2023Article
- rRNA transcription is integral to phase separation and maintenance of nucleolar structure.PLoS genetics · 2023Article
- RG/RGG repeats in the C. elegans homologs of Nucleolin and GAR1 contribute to sub-nucleolar phase separation.Nature communications · 2022Article
- Low level of Fibrillarin, a ribosome biogenesis factor, is a new independent marker of poor outcome in breast cancer.BMC cancer · 2022Article
- Selective translation of epigenetic modifiers affects the temporal pattern and differentiation of neural stem cells.Nature communications · 2022Article
- Systematic mapping of rRNA 2'-O methylation during frog development and involvement of the methyltransferase Fibrillarin in eye and craniofacial development in Xenopus laevis.PLoS genetics · 2022Article
- Identification of aberrantly methylated differentially expressed genes and pro-tumorigenic role of KIF2C in melanoma.Frontiers in genetics · 2022Article
- RNA 2Frontiers in immunology · 2022Article
- U8 variants on the brain: a small nucleolar RNA and human disease.RNA biology · 2022Review
2 more citing papers are in PubMed but not listed here.
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
4 authors at 2 institutions in 1 country.
Funding
Abstract
Fibrillarin, a protein component of C/D box small nucleolar ribonucleoproteins (snoRNPs), directs 2'-O-methylation of rRNA and is also involved in other aspects of rRNA processing. A gene trap screen in embryonic stem (ES) cells resulted in an insertion mutation in the fibrillarin gene. This insertion generated a fusion protein that contained the N-terminal 132 amino acids of fibrillarin fused to a beta-galactosidase-neomycin phosphotransferase reporter. As a result, the N-terminal GAR domain was present in the fusion protein but the methyltransferase-like domain was missing. The ES cell line with the targeted fibrillarin allele was transmitted through the mouse germ line, creating heterozygous animals. Western blot analyses showed a reduction in fibrillarin protein levels in the heterozygous knockout animals. Animals homozygous for the mutation were inviable, and massive apoptosis was observed in early Fibrillarin(-/-) embryos, showing that fibrillarin is essential for development. Fibrillarin(+/-) live-born mice displayed no obvious growth defect, but heterozygous intercrosses revealed a reduced ratio of +/- to +/+ mice, showing that some of the Fibrillarin heterozygous embryos die in utero. Analyses of tissue samples and cultured embryonic fibroblasts showed no discernible alteration in pre-rRNA processing or the level of the U3 snoRNA. However, the level of the intron-encoded box C/D snoRNA U76 was clearly reduced. This suggests a high requirement for snoRNA synthesis during an early stage in development.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.