The emerging significance of splicing in vertebrate development.
| Autor: | Olthof AM; Physiology and Neurobiology Department, University of Connecticut, Storrs, CT 06269, USA.; Department of Cellular and Molecular Medicine, University of Copenhagen, Copenhagen 2200, Denmark., White AK; Physiology and Neurobiology Department, University of Connecticut, Storrs, CT 06269, USA., Kanadia RN; Physiology and Neurobiology Department, University of Connecticut, Storrs, CT 06269, USA.; Institute for Systems Genomics, University of Connecticut, Storrs, CT 06269, USA. |
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| Jazyk: | angličtina |
| Zdroj: | Development (Cambridge, England) [Development] 2022 Oct 01; Vol. 149 (19). Date of Electronic Publication: 2022 Sep 30. |
| DOI: | 10.1242/dev.200373 |
| Abstrakt: | Splicing is a crucial regulatory node of gene expression that has been leveraged to expand the proteome from a limited number of genes. Indeed, the vast increase in intron number that accompanied vertebrate emergence might have aided the evolution of developmental and organismal complexity. Here, we review how animal models for core spliceosome components have provided insights into the role of splicing in vertebrate development, with a specific focus on neuronal, neural crest and skeletal development. To this end, we also discuss relevant spliceosomopathies, which are developmental disorders linked to mutations in spliceosome subunits. Finally, we discuss potential mechanisms that could underlie the tissue-specific phenotypes often observed upon spliceosome inhibition and identify gaps in our knowledge that, we hope, will inspire further research. Competing Interests: Competing interests The authors declare no competing or financial interests. (© 2022. Published by The Company of Biologists Ltd.) |
| Databáze: | MEDLINE |
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