A novel RNA binding nuclear protein that interacts with the fragile X mental retardation (FMR1) protein
Abstract
Silenced expression of the FMR1 gene is responsible for the fragile X syndrome. The FMR1 gene codes for an RNA binding protein (FMRP), which can shuttle between the nucleus and the cytoplasm and is found associated to polysomes in the cytoplasm. By two-hybrid assay in yeast, we identified a novel protein interacting with FMRP: nuclear FMRP interacting protein (NUFIP). NUFIP mRNA expression is strikingly similar to that of the FMR1 gene in neurones of cortex, hippocampus and cerebellum. At the subcellular level, NUFIP colocalizes with nuclear isoforms of FMRP in a dot-like pattern. NUFIP presents a C2H2 zinc finger motif and a nuclear localization signal, but has no homology to known proteins and shows RNA binding activity in vitro. NUFIP does not interact with the FMRP homologues encoded by the FXR1 and FXR2 genes. Thus, these results indicate a specific nuclear role for FMRP.
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The CYFIP1-FMRP interaction was initially identified by two-hybrid screening in yeast.
“The CYFIP1-FMRP interaction, initially identified by two-hybrid screening in yeast (Bardoni et al., 1999; Schenck et al., 2001), was confirmed by GST pull-down and immunoprecipitation both in mouse and fly cell lines and tissues (Schenck et al., 2001, 2003; De Rubeis et al., 2013).”
From New insights into the regulatory function of CYFIP1 in the context of WAVE- and FMRP-containing complexes (2017) · cites this paper as Bardoni et al., 1999
The Results and Discussion describe a yeast two-hybrid screen for FMRP interactors, so the screening method is supported. But the paper identifies NUFIP—not CYFIP1—as the novel interactor found by that screen. The cited paper therefore does not establish the claimed CYFIP1–FMRP identification. (LLM, conf=0.84)
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Cites
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FMR1 and the fragile X syndrome: Human genome epidemiology review
Crawford, D. C., Acuña, J. M., & Sherman, S. L. (2001) · Genetics in Medicine · doi:10.1097/00125817-200109000-00006 · Cited by 711 · see citing works
Zalfa, F., Giorgi, M., Primerano, B., Moro, A., Di Penta, A., Reis, S., Oostra, B., & Bagni, C. (2003) · Cell · doi:10.1016/s0092-8674(03)00079-5 · Cited by 676 · see citing works
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From mRNP trafficking to spine dysmorphogenesis: the roots of fragile X syndrome
Bagni, C., & Greenough, W. T. (2005) · Nature Reviews Neuroscience · doi:10.1038/nrn1667 · Cited by 475 · see citing works
The Pathophysiology of Fragile X Syndrome
Penagarikano, O., Mulle, J. G., & Warren, S. T. (2007) · Annual Review of Genomics and Human Genetics · doi:10.1146/annurev.genom.8.080706.092249 · Cited by 420 · see citing works
Schenck, A., Bardoni, B., Langmann, C., Harden, N., Mandel, J.-L., & Giangrande, A. (2003) · Neuron · doi:10.1016/s0896-6273(03)00354-4 · Cited by 316 · see citing works
Self-citationDarnell, J. C., Fraser, C. E., Mostovetsky, O., Stefani, G., Jones, T. A., Eddy, S. R., & Darnell, R. B. (2005) · Genes & Development · doi:10.1101/gad.1276805 · Cited by 287 · see citing works
Chen, L., Yun, S.-W., Seto, J., Liu, W., & Toth, M. (2003) · Neuroscience · doi:10.1016/s0306-4522(03)00406-8 · Cited by 160 · see citing works
Myrick, L. K., Deng, P.-Y., Hashimoto, H., Oh, Y. M., Cho, Y., Poidevin, M. J., Suhl, J. A., Visootsak, J., Cavalli, V., Jin, P., et al. (2015) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.1423094112 · Cited by 133 · see citing works
FMR1 premutation and full mutation molecular mechanisms related to autism
Hagerman, R., Au, J., & Hagerman, P. (2011) · Journal of Neurodevelopmental Disorders · doi:10.1007/s11689-011-9084-5 · Cited by 85 · see citing works
Abekhoukh, S., Sahin, H. B., Grossi, M., Zongaro, S., Maurin, T., Madrigal, I., Kazue-Sugioka, D., Raas-Rothschild, A., Doulazmi, M., Carrera, P., et al. (2017) · Disease Models & Mechanisms · doi:10.1242/dmm.025809 · Cited by 59 · see citing works
“The CYFIP1-FMRP interaction, initially identified by two-hybrid screening in yeast (Bardoni et al., 1999; Schenck et al., 2001), was confirmed by GST pull-down and immunoprecipitation both in mouse and fly cell lines and tissues (Schenck et al., 2001, 2003; De Rubeis et al., 2013).”
Self-citation
- Authors
- Bardoni, B., Schenck, A., & Louis Mandel, J.
- Affiliations
- Centre National de la Recherche Scientifique (FR)
- Venue
- Human Molecular Genetics
- Publisher
- Oxford University Press (OUP)
- Published
- 1999-12-01
- Volume
- 8
- Issue
- 13
- Pages
- 2557-2566
- ISSN
- 0964-6906 (ISSN-L)
- 1460-2083
- OpenAlex
- W2118593615
- Cited by
- 136
- References
- 2
- Citations by year
- 2025: 2 · 2024: 2 · 2023: 1 · 2022: 7 · 2021: 3 · 2020: 5 · 2019: 2 · 2018: 6 · 2017: 2 · 2016: 1 · 2015: 3 · 2014: 9 · 2013: 5 · 2012: 1
- FWCI
- 4.77
- Type
- journal article
- Language
- en
- Open access
- closed
- Locations
- Human Molecular Genetics (publishedVersion)
- PubMed (publishedVersion)
- HAL (submittedVersion)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › RNA Research and Splicing
- Life Sciences › Neuroscience › Cellular and Molecular Neuroscience › Genetic Neurodegenerative Diseases
- Keywords
- FMRP, fragile X syndrome, FMR1 gene, RNA-binding proteins, protein-protein interaction, nuclear localization, C2H2 zinc finger, FXR1, FXR2, neuronal gene expression
- MeSH terms
- Amino Acid Sequence, Animals, Brain, Cell Nucleus, Fluorescent Antibody Technique, Fragile X Syndrome, HeLa Cells, Humans, Intellectual Disability, Molecular Sequence Data, Nerve Tissue Proteins, Nuclear Proteins, Recombinant Proteins, Yeasts, Gene Expression, Zinc Fingers, RNA-Binding Proteins, In Situ Hybridization, Microscopy, Confocal, COS Cells, Two-Hybrid System Techniques, Mice, Fragile X Mental Retardation Protein, Fragile X Messenger Ribonucleoprotein 1
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