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
  • Chromatids/metabolism
  • Centromere*/metabolism
  • Zebrafish*/genetics
  • Cell Cycle Proteins/physiology
  • Animals
  • Chromosomal Proteins, Non-Histone/genetics
(all 6)
PubMed
35275424 Full text @ Dev. Dyn.
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
Figures
Figure Gallery (4 images)
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Expression
Phenotype
Fish Conditions Stage Phenotype Figure
sgo1hu13334/+ (TL)standard conditionsProtruding-mouth
sgo1hu13334/+ (TL)standard conditionsProtruding-mouth
sgo1hu13334/+ (TL)standard conditionsProtruding-mouth
sgo1hu13334/+ (TL)standard conditionsDay 5
sgo1hu13334/+ (TL)standard conditionsDay 5
sgo1hu13334/+ (TL)standard conditionsDay 5
sgo1hu13334/+ (TL)standard conditionsDay 5
sgo1hu13334/+ (TL)standard conditionsDay 5
sgo1hu13334/+ (TL)standard conditionsDay 5
sgo1hu13334/+ (TL)standard conditionsDay 5
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Mutations / Transgenics
Allele Construct Type Affected Genomic Region
hu13334
    		Small Deletion
    1 - 1 of 1
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    Human Disease / Model
    No data available
    Sequence Targeting Reagents
    Target Reagent Reagent Type
    sgo1CRISPR2-sgo1CRISPR
    1 - 1 of 1
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    Fish
    Fish
    sgo1hu13334/hu13334 (TL)
    sgo1hu13334/+ (TL)
    TL
    1 - 3 of 3
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    Antibodies
    Orthology
    No data available
    Engineered Foreign Genes
    No data available
    Mapping
    No data available
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    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.