PUBLICATION
Zebrafish as a Model to Study Cohesin and Cohesinopathies
- Authors
- Muto, A., Schilling, T.F.
- ID
- ZDB-PUB-161101-11
- Date
- 2017
- Source
- Methods in molecular biology (Clifton, N.J.) 1515: 177-196 (Chapter)
- Registered Authors
- Schilling, Tom
- Keywords
- Chromatin, Cohesin, Cohesinopathies, Transcriptional regulation, Zebrafish
- MeSH Terms
-
- Animals
- Cell Cycle Proteins/deficiency
- Cell Cycle Proteins/genetics*
- Chromosomal Proteins, Non-Histone/deficiency
- Chromosomal Proteins, Non-Histone/genetics*
- Chromosome Segregation/genetics
- Disease Models, Animal
- Gene Expression Regulation, Developmental
- Humans
- Mice
- Morpholinos/genetics
- Zebrafish/genetics
- Zebrafish Proteins/genetics*
- PubMed
- 27797080 Full text @ Meth. Mol. Biol.
Citation
Muto, A., Schilling, T.F. (2017) Zebrafish as a Model to Study Cohesin and Cohesinopathies. Methods in molecular biology (Clifton, N.J.). 1515:177-196.
Abstract
The cohesin protein complex regulates multiple cellular events including sister chromatid cohesion and gene expression. Several distinct human diseases called cohesinopathies have been associated with genetic mutations in cohesin subunit genes or genes encoding regulators of cohesin function. Studies in different model systems, from yeast to mouse have provided insights into the molecular mechanisms of action of cohesin/cohesin regulators and their implications in the pathogenesis of cohesinopathies. The zebrafish has unique advantages for embryonic analyses and quantitative gene knockdown with morpholinos during the first few days of development, in contrast to knockouts of cohesin regulators in flies or mammals, which are either lethal as homozygotes or dramatically compensated for in heterozygotes. This has been particularly informative for Rad21, where a role in gene expression was first shown in zebrafish, and Nipbl, where the fish work revealed tissue-specific functions in heart, gut, and limbs, and long-range enhancer-promoter interactions that control Hox gene expression in vivo. Here we discuss the utility of the zebrafish in studying the developmental and pathogenic roles of cohesin.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping