PUBLICATION
The zebrafish cohesin protein Sgo1 is required for cardiac function and eye development
- Authors
- Kamel, S.M., Broekman, S., Tessadori, F., van Wijk, E., Bakkers, J.
- ID
- ZDB-PUB-220312-4
- Date
- 2022
- Source
- Developmental Dynamics : an official publication of the American Association of Anatomists 251(8): 1357-1367 (Journal)
- Registered Authors
- Bakkers, Jeroen, van Wijk, Erwin
- Keywords
- Sgo1, cohesinopathy, heart defect, retinal defect, shugoshin, zebrafish
- MeSH Terms
-
- Animals
- Cell Cycle Proteins/physiology
- Centromere*/metabolism
- Chromatids/metabolism
- Chromosomal Proteins, Non-Histone/genetics
- Zebrafish*/genetics
- PubMed
- 35275424 Full text @ Dev. Dyn.
Citation
Kamel, S.M., Broekman, S., Tessadori, F., van Wijk, E., Bakkers, J. (2022) The zebrafish cohesin protein Sgo1 is required for cardiac function and eye development. Developmental Dynamics : an official publication of the American Association of Anatomists. 251(8):1357-1367.
Abstract
Background Cohesinopathies is a term that refers to/covers rare genetic diseases caused by mutations in the cohesin complex proteins. The cohesin complex is a multi-protein complex that facilitates different aspects of cell division, gene transcription, DNA damage repair and chromosome architecture. Shugoshin proteins prevent the cohesin complex from premature dissociation from chromatids during cell division. Patients with a homozygous missense mutation in SGO1, which encodes for Shugoshin1, have problems with normal pacing of the heart and gut.
Results To study the role of shugoshin during embryo development, we mutated the zebrafish sgo1 gene. Homozygous sgo1 mutant embryos display various phenotypes related to different organs, including a reduced heart rate accompanied by reduced cardiac function. In addition, sgo1 mutants are vision-impaired as a consequence of structurally defective and partially non-functional photoreceptor cells. Furthermore, the sgo1 mutants display reduced food intake and early lethality.
Conclusion We have generated a zebrafish model of Sgo1 that showed its importance during organ development and function. This article is protected by copyright. All rights reserved.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping