The DOI matches its Crossref record; 58 of the 58 references it cites were found in a registry or an index.
Cdk5rap2 regulates centrosome function and chromosome segregation in neuronal progenitors
Open accessAbstract
Microcephaly affects ∼1% of the population and is associated with mental retardation, motor defects and, in some cases, seizures. We analyzed the mechanisms underlying brain size determination in a mouse model of human microcephaly. The Hertwig's anemia (an) mutant shows peripheral blood cytopenias, spontaneous aneuploidy and a predisposition to hematopoietic tumors. We found that the an mutation is a genomic inversion of exon 4 of Cdk5rap2, resulting in an in-frame deletion of exon 4 from the mRNA. The finding that CDK5RAP2 human mutations cause microcephaly prompted further analysis of Cdk5rap2an/an mice and we demonstrated that these mice exhibit microcephaly comparable to that of the human disease, resulting from striking neurogenic defects that include proliferative and survival defects in neuronal progenitors. Cdk5rap2an/an neuronal precursors exit the cell cycle prematurely and many undergo apoptosis. These defects are associated with impaired mitotic progression coupled with abnormal mitotic spindle pole number and mitotic orientation. Our findings suggest that the reduction in brain size observed in humans with mutations in CDK5RAP2 is associated with impaired centrosomal function and with changes in mitotic spindle orientation during progenitor proliferation.
Overview
The claims and statistics of this paper haven’t been checked yet.
Run the check — free, no account needed. It reads the open-access text and checks the sources it cites.
Integrity signals
Registry
Cites
Statistics
Verification
Claims the paper makes
The claims and statistics of this paper haven’t been checked yet.
Run the check — free, no account needed. It reads the open-access text and checks the sources it cites.
Self-check
Percentages, sums and reported differences re-computed from the paper’s own numbers.
The self-check needs the paper’s full text.
Citations
Cites
The cell biology of neurogenesis
Götz, M., & Huttner, W. B. (2005) · Nature Reviews Molecular Cell Biology · doi:10.1038/nrm1739 · Cited by 2,130
Neurons derived from radial glial cells establish radial units in neocortex
Noctor, S. C., Flint, A. C., Weissman, T. A., Dammerman, R. S., & Kriegstein, A. R. (2001) · Nature · doi:10.1038/35055553 · Cited by 2,016
Evolution of the neocortex: a perspective from developmental biology
Rakic, P. (2009) · Nature reviews. Neuroscience · doi:10.1038/nrn2719 · Cited by 1,565
Autoradiographic Study of Cell Migration during Histogenesis of Cerebral Cortex in the Mouse
ANGEVINE, J. B., & SIDMAN, R. L. (1961) · Nature · doi:10.1038/192766b0 · Cited by 1,530
Regulation of Cerebral Cortical Size by Control of Cell Cycle Exit in Neural Precursors
Chenn, A., & Walsh, C. A. (2002) · Science · doi:10.1126/science.1074192 · Cited by 1,431
Self-citationDoublecortin Is a Microtubule-Associated Protein and Is Expressed Widely by Migrating Neurons
Gleeson, J. G., Lin, P. T., Flanagan, L. A., & Walsh, C. A. (1999) · Neuron · doi:10.1016/s0896-6273(00)80778-3 · Cited by 1,340
Self-citationEnglund, C., Fink, A., Lau, C., Pham, D., Daza, R. A. M., Bulfone, A., Kowalczyk, T., & Hevner, R. F. (2005) · The Journal of Neuroscience · doi:10.1523/jneurosci.2899-04.2005 · Cited by 1,261
Rakic, P. (1974) · Science · doi:10.1126/science.183.4123.425 · Cited by 1,161
Tbr1 regulates differentiation of the preplate and layer 6
Hevner, R. F., Shi, L., Justice, N., Hsueh, Y.-P., Sheng, M., Smiga, S., Bulfone, A., Goffinet, A. M., Campagnoni, A. T., & Rubenstein, J. L. R. (2001) · Neuron · doi:10.1016/s0896-6273(01)00211-2 · Cited by 857
Chenn, A., & McConnell, S. K. (1995) · Cell · doi:10.1016/0092-8674(95)90035-7 · Cited by 766
Patterns of neural stem and progenitor cell division may underlie evolutionary cortical expansion
Kriegstein, A., Noctor, S., & Martínez-Cerdeño, V. (2006) · Nature Reviews Neuroscience · doi:10.1038/nrn2008 · Cited by 738
ASPM is a major determinant of cerebral cortical size
Bond, J., Roberts, E., Mochida, G. H., Hampshire, D. J., Scott, S., Askham, J. M., Springell, K., Mahadevan, M., Crow, Y. J., Markham, A. F., et al. (2002) · Nature Genetics · doi:10.1038/ng995 · Cited by 581
Self-citationA centrosomal mechanism involving CDK5RAP2 and CENPJ controls brain size
Bond, J., Roberts, E., Springell, K., Lizarraga, S., Scott, S., Higgins, J., Hampshire, D. J., Morrison, E. E., Leal, G. F., Silva, E. O., et al. (2005) · Nature Genetics · doi:10.1038/ng1539 · Cited by 572
Self-citationAsymmetric Inheritance of Mother Versus Daughter Centrosome in Stem Cell Division
Yamashita, Y. M., Mahowald, A. P., Perlin, J. R., & Fuller, M. T. (2007) · Science · doi:10.1126/science.1134910 · Cited by 560
Nieto, M., Monuki, E. S., Tang, H., Imitola, J., Haubst, N., Khoury, S. J., Cunningham, J., Gotz, M., & Walsh, C. A. (2004) · Journal of Comparative Neurology · doi:10.1002/cne.20322 · Cited by 549
Self-citationCharacterization of Foxp2 and Foxp1 mRNA and protein in the developing and mature brain
Ferland, R. J., Cherry, T. J., Preware, P. O., Morrisey, E. E., & Walsh, C. A. (2003) · Journal of Comparative Neurology · doi:10.1002/cne.10654 · Cited by 484
Self-citationAsymmetric centrosome inheritance maintains neural progenitors in the neocortex
Wang, X., Tsai, J.-W., Imai, J. H., Lian, W.-N., Vallee, R. B., & Shi, S.-H. (2009) · Nature · doi:10.1038/nature08435 · Cited by 475
Woods, C. G., Bond, J., & Enard, W. (2005) · The American Journal of Human Genetics · doi:10.1086/429930 · Cited by 431
Misson, J.-P., Edwards, M. A., Yamamoto, M., & Caviness, V. S. (1988) · Developmental Brain Research · doi:10.1016/0165-3806(88)90121-6 · Cited by 422
Aspm specifically maintains symmetric proliferative divisions of neuroepithelial cells
Fish, J. L., Kosodo, Y., Enard, W., Pääbo, S., & Huttner, W. B. (2006) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.0604066103 · Cited by 421
The Aurora kinase family in cell division and cancer
Vader, G., & Lens, S. M. A. (2008) · Biochimica et Biophysica Acta (BBA) - Reviews on Cancer · doi:10.1016/j.bbcan.2008.07.003 · Cited by 417
Shibata, T., Yamada, K., Watanabe, M., Ikenaka, K., Wada, K., Tanaka, K., & Inoue, Y. (1997) · The Journal of Neuroscience · doi:10.1523/jneurosci.17-23-09212.1997 · Cited by 407
The Aurora kinases: Role in cell transformation and tumorigenesis
Katayama, H., Brinkley, W. R., & Sen, S. (2003) · Cancer and Metastasis Reviews · doi:10.1023/a:1023789416385 · Cited by 398
Role of Intermediate Progenitor Cells in Cerebral Cortex Development
Pontious, A., Kowalczyk, T., Englund, C., & Hevner, R. F. (2008) · Developmental Neuroscience · doi:10.1159/000109848 · Cited by 378 · cited 1×
“Apical progenitors give rise to deep layer neurons and basal progenitors, whereas basal progenitors primarily produce superficial layer neurons (Pontious et al., 2008).”
BackgroundMitotic Spindle Regulation by Nde1 Controls Cerebral Cortical Size
Feng, Y., & Walsh, C. A. (2004) · Neuron · doi:10.1016/j.neuron.2004.09.023 · Cited by 357
Self-citationBultje, R. S., Castaneda-Castellanos, D. R., Jan, L. Y., Jan, Y.-N., Kriegstein, A. R., & Shi, S.-H. (2009) · Neuron · doi:10.1016/j.neuron.2009.07.004 · Cited by 348
Oncogenes and tumour suppressors take on centrosomes
Fukasawa, K. (2007) · Nature Reviews Cancer · doi:10.1038/nrc2249 · Cited by 312
Zimmerman, W. C., Sillibourne, J., Rosa, J., & Doxsey, S. J. (2004) · Molecular Biology of the Cell · doi:10.1091/mbc.e03-11-0796 · Cited by 285
Fong, K.-W., Choi, Y.-K., Rattner, J. B., & Qi, R. Z. (2008) · Molecular Biology of the Cell · doi:10.1091/mbc.e07-04-0371 · Cited by 283
Yingling, J., Youn, Y. H., Darling, D., Toyo-oka, K., Pramparo, T., Hirotsune, S., & Wynshaw-Boris, A. (2008) · Cell · doi:10.1016/j.cell.2008.01.026 · Cited by 272
Role of Sox2 in the development of the mouse neocortex
Bani-Yaghoub, M., Tremblay, R. G., Lei, J. X., Zhang, D., Zurakowski, B., Sandhu, J. K., Smith, B., Ribecco-Lutkiewicz, M., Kennedy, J., Walker, P. R., et al. (2006) · Developmental Biology · doi:10.1016/j.ydbio.2006.03.007 · Cited by 271
The Role of Intermediate Progenitor Cells in the Evolutionary Expansion of the Cerebral Cortex
Martínez-Cerdeño, V., Noctor, S. C., & Kriegstein, A. R. (2006) · Cerebral Cortex · doi:10.1093/cercor/bhk017 · Cited by 271
The neocortex: An overview of its evolutionary development, structural organization and synaptology
Nieuwenhuys, R. (1994) · Anatomy and Embryology · doi:10.1007/bf00187291 · Cited by 270
Transcriptional Regulation of Cortical Neuron Migration by POU Domain Factors
McEvilly, R. J., de Diaz, M. O., Schonemann, M. D., Hooshmand, F., & Rosenfeld, M. G. (2002) · Science · doi:10.1126/science.1067132 · Cited by 270
Sanada, K., & Tsai, L.-H. (2005) · Cell · doi:10.1016/j.cell.2005.05.009 · Cited by 251
Mitotic spindle rotation and mode of cell division in the developing telencephalon
Haydar, T. F., Ang, E., & Rakic, P. (2003) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.0437969100 · Cited by 237
Timothy, L. M., Li, K., Kao, L.-R., & Kaufman, T. C. (1999) · Development · doi:10.1242/dev.126.13.2829 · Cited by 231
The centrosome in neuronal development
Higginbotham, H. R., & Gleeson, J. G. (2007) · Trends in Neurosciences · doi:10.1016/j.tins.2007.04.001 · Cited by 217
Cep68 and Cep215 (Cdk5rap2) are required for centrosome cohesion
Graser, S., Stierhof, Y.-D., & Nigg, E. A. (2007) · Journal of Cell Science · doi:10.1242/jcs.020248 · Cited by 217
Verde, I., Pahlke, G., Salanova, M., Zhang, G., Wang, S., Coletti, D., Onuffer, J., Jin, S.-L. C., & Conti, M. (2001) · Journal of Biological Chemistry · doi:10.1074/jbc.m006546200 · Cited by 217
Caviness, V. S. (2003) · Cerebral Cortex · doi:10.1093/cercor/13.6.592 · Cited by 187
Neurogenesis and asymmetric cell division
Zhong, W., & Chia, W. (2008) · Current Opinion in Neurobiology · doi:10.1016/j.conb.2008.05.002 · Cited by 163
CTIP1 and CTIP2 are differentially expressed during mouse embryogenesis
Leid, M., Ishmael, J. E., Avram, D., Shepherd, D., Fraulob, V., & Dollé, P. (2004) · Gene Expression Patterns · doi:10.1016/j.modgep.2004.03.009 · Cited by 156
Comparative aspects of cortical neurogenesis in vertebrates
Cheung, A. F. P., Pollen, A. A., Tavare, A. N., DeProto, J., & Molnár, Z. (2007) · Journal of Anatomy · doi:10.1111/j.1469-7580.2007.00769.x · Cited by 139
Neue Mutationen und Koppelungsgruppen bei der Hausmaus
Hertwig, P. (1942) · Molecular Genetics and Genomics · doi:10.1007/bf01741984 · Cited by 136
Heuer, J. G., Li, K., & Kaufman, T. C. (1995) · Development · doi:10.1242/dev.121.11.3861 · Cited by 135
Distribution of Group-specific Components (Gc) in the Sera of Native Africans
Hirschfeld, J., & Sonnet, J. (1961) · Nature · doi:10.1038/192766a0 · Cited by 134
Venkatram, S., Tasto, J. J., Feoktistova, A., Jennings, J. L., Link, A. J., & Gould, K. L. (2004) · Molecular Biology of the Cell · doi:10.1091/mbc.e03-10-0728 · Cited by 119
Cloning of three novel neuronal Cdk5 activator binding proteins
Ching, Y. P., Qi, R. Z., & Wang, J. H.-C. (2000) · Gene · doi:10.1016/s0378-1119(99)00499-0 · Cited by 111
Identification of Neural Outgrowth Genes using Genome-Wide RNAi
Sepp, K. J., Hong, P., Lizarraga, S. B., Liu, J. S.-H., Mejia, L. A., Walsh, C. A., & Perrimon, N. (2008) · PLoS Genetics · doi:10.1371/journal.pgen.1000111 · Cited by 106
Self-citationZhang, J., & Megraw, T. L. (2007) · Molecular Biology of the Cell · doi:10.1091/mbc.e07-05-0474 · Cited by 104
Molecular evolution of the brain size regulator genes CDK5RAP2 and CENPJ
Evans, P. D., Vallender, E. J., & Lahn, B. T. (2006) · Gene · doi:10.1016/j.gene.2006.02.019 · Cited by 86
Spindle regulation in neural precursors of flies and mammals
Buchman, J. J., & Tsai, L. (2007) · Nature reviews. Neuroscience · doi:10.1038/nrn2058 · Cited by 61
γ-Tubulin Plays an Essential Role in the Coordination of Mitotic Events
Prigozhina, N. L., Oakley, C. E., Lewis, A. M., Nayak, T., Osmani, S. A., & Oakley, B. R. (2003) · Molecular Biology of the Cell · doi:10.1091/mbc.e03-06-0405 · Cited by 59
Lee, L., Debono, C. A., Campagna, D. R., Young, D. C., Moody, D. B., & Fleming, M. D. (2006) · Journal of Investigative Dermatology · doi:10.1038/sj.jid.5700511 · Cited by 49
Self-citationTelencephalon enlargement by the convergent evolution of expanded subventricular zones
Striedter, G. F., & Charvet, C. J. (2008) · Biology Letters · doi:10.1098/rsbl.2008.0439 · Cited by 32
High incidence, early onset of histiocytic sarcomas in mice with Hertwig's anemia
Barker, J. E., Deveau, S. A., Compton, S. T., Fancher, K. S., & Eppig, J. T. (2005) · Experimental Hematology · doi:10.1016/j.exphem.2005.06.021 · Cited by 6
Self-citation[Molecular genetics of lissencephaly and microcephaly].
Mochida, G. H. (2008) · PubMed · Cited by 5
Cited by
Cerebral organoids model human brain development and microcephaly
Lancaster, M. A., Renner, M., Martin, C.-A., Wenzel, D., Bicknell, L. S., Hurles, M. E., Homfray, T., Penninger, J. M., Jackson, A. P., & Knoblich, J. A. (2013) · Nature · doi:10.1038/nature12517 · Cited by 5,372
Development and Evolution of the Human Neocortex
Lui, J. H., Hansen, D. V., & Kriegstein, A. R. (2011) · Cell · doi:10.1016/j.cell.2011.06.030 · Cited by 1,342
The Brazilian Zika virus strain causes birth defects in experimental models
Cugola, F. R., Fernandes, I. R., Russo, F. B., Freitas, B. C., Dias, J. L. M., Guimarães, K. P., Benazzato, C., Almeida, N., Pignatari, G. C., Romero, S., et al. (2016) · Nature · doi:10.1038/nature18296 · Cited by 1,326
Taverna, E., Götz, M., & Huttner, W. B. (2014) · Annual Review of Cell and Developmental Biology · doi:10.1146/annurev-cellbio-101011-155801 · Cited by 759
Expression Analysis Highlights AXL as a Candidate Zika Virus Entry Receptor in Neural Stem Cells
Nowakowski, T. J., Pollen, A. A., Di Lullo, E., Sandoval-Espinosa, C., Bershteyn, M., & Kriegstein, A. R. (2016) · Cell Stem Cell · doi:10.1016/j.stem.2016.03.012 · Cited by 529
Metadata
- Authors
- Lizarraga, S. B., Margossian, S. P., Harris, M. H., Campagna, D. R., Han, A.-P., Blevins, S., Mudbhary, R., Barker, J. E., Walsh, C. A., & Fleming, M. D.
- Venue
- Development
- Publisher
- The Company of Biologists
- Published
- 2010-06-01
- Volume
- 137
- Issue
- 11
- Pages
- 1907-1917
- ISSN
- 0950-1991 (ISSN-L)
- 1477-9129
- OpenAlex
- W2019695320
- Cited by
- 273
- References
- 58
- FWCI
- 6.58
- Type
- journal article
- Language
- en
- Open access
- green
About these checks
Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.
Statistics and arithmetic are recomputed, not judged.
Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.
What counts as a claim ›How we measure confidence ›How we check this ›
Are you an author, or do you see a problem with this page? Request a review
Data sources
Bibliographic data: Semantic Scholar, OpenAlex, Crossref.
Contains information from the Semantic Scholar Academic Graph Datasets, made available under the ODC Attribution License (ODC-BY 1.0).
Semantic Scholar’s data is described in: Kinney, R., et al. (2023). The Semantic Scholar Open Data Platform. arXiv:2301.10140.
