The DOI matches its Crossref record; 4 of the 4 references it cites were found in a registry or an index.
Chromatin remodelling complex dosage modulates transcription factor function in heart development
Open accessAbstract
Dominant mutations in cardiac transcription factor genes cause human inherited congenital heart defects (CHDs); however, their molecular basis is not understood. Interactions between transcription factors and the Brg1/Brm-associated factor (BAF) chromatin remodelling complex suggest potential mechanisms; however, the role of BAF complexes in cardiogenesis is not known. In this study, we show that dosage of Brg1 is critical for mouse and zebrafish cardiogenesis. Disrupting the balance between Brg1 and disease-causing cardiac transcription factors, including Tbx5, Tbx20 and Nkx2–5, causes severe cardiac anomalies, revealing an essential allelic balance between Brg1 and these cardiac transcription factor genes. This suggests that the relative levels of transcription factors and BAF complexes are important for heart development, which is supported by reduced occupancy of Brg1 at cardiac gene promoters in Tbx5 haploinsufficient hearts. Our results reveal complex dosage-sensitive interdependence between transcription factors and BAF complexes, providing a potential mechanism underlying transcription factor haploinsufficiency, with implications for multigenic inheritance of CHDs. Inherited congenital heart defects are prevalent in the human population, but the molecular mechanisms are poorly understood. In this article, deficiency in the chromatin remodelling factor, Brg1, is shown to alter cardiac development in both mouse and zebrafish laboratory models.
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Cites
Smyth, G. K. (2004) · Statistical Applications in Genetics and Molecular Biology · doi:10.2202/1544-6115.1027 · Cited by 11,829
Efficient in vivo manipulation of mouse genomic sequences at the zygote stage.
Lakso, M., Pichel, J. G., Gorman, J. R., Sauer, B., Okamoto, Y., Lee, E., Alt, F. W., & Westphal, H. (1996) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.93.12.5860 · Cited by 1,125
Chromatin remodelling during development
Ho, L., & Crabtree, G. R. (2010) · Nature · doi:10.1038/nature08911 · Cited by 1,114
Lessard, J., Wu, J. I., Ranish, J. A., Wan, M., Winslow, M. M., Staahl, B. T., Wu, H., Aebersold, R., Graef, I. A., & Crabtree, G. R. (2007) · Neuron · doi:10.1016/j.neuron.2007.06.019 · Cited by 735
Cited by
CRISPR Interference Efficiently Induces Specific and Reversible Gene Silencing in Human iPSCs
Mandegar, M. A., Huebsch, N., Frolov, E. B., Shin, E., Truong, A., Olvera, M. P., Chan, A. H., Miyaoka, Y., Holmes, K., Spencer, C. I., et al. (2016) · Cell Stem Cell · doi:10.1016/j.stem.2016.01.022 · Cited by 525
“The dosage of transcription factors plays a significant role during development and organogenesis (McFadden et al., 2005; Takeuchi et al., 2011).”
Goetze, J. P., Bruneau, B. G., Ramos, H. R., Ogawa, T., de Bold, M. K., & de Bold, A. J. (2020) · Nature Reviews Cardiology · doi:10.1038/s41569-020-0381-0 · Cited by 531
Self-citationMyocardial Polyploidization Creates a Barrier to Heart Regeneration in Zebrafish
González-Rosa, J. M., Sharpe, M., Field, D., Soonpaa, M. H., Field, L. J., Burns, C. E., & Burns, C. G. (2018) · Developmental Cell · doi:10.1016/j.devcel.2018.01.021 · Cited by 264
Delgado-Olguín, P., Huang, Y., Li, X., Christodoulou, D., Seidman, C. E., Seidman, J. G., Tarakhovsky, A., & Bruneau, B. G. (2012) · Nature Genetics · doi:10.1038/ng.1068 · Cited by 259
Self-citationChen, A. F., Parks, B., Kathiria, A. S., Ober-Reynolds, B., Goronzy, J. J., & Greenleaf, W. J. (2022) · Nature Methods · doi:10.1038/s41592-022-01461-y · Cited by 139
Metadata
- Authors
- Takeuchi, J. K., Lou, X., Alexander, J. M., Sugizaki, H., Delgado-Olguín, P., Holloway, A. K., Mori, A. D., Wylie, J. N., Munson, C., Zhu, Y., Zhou, Y.-Q., Yeh, R.-F., Henkelman, R. M., Harvey, R. P., Metzger, D., Chambon, P., Stainier, D. Y. R., Pollard, K. S., Scott, I. C., & Bruneau, B. G.
- Venue
- Nature Communications
- Published
- 2011-02-08
- Volume
- 2
- Issue
- 1
- Pages
- 187-187
- ISSN
- 2041-1723 (ISSN-L)
- OpenAlex
- W2114430445
- Cited by
- 208
- References
- 4
- FWCI
- 7.64
- Type
- journal article
- Language
- en
- Open access
- hybrid
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