PUBLICATION
Genetic compensation by epob in pronephros development in epoa mutant zebrafish
- Authors
- She, J., Wu, Y., Lou, B., Lodd, E., Klems, A., Schmoehl, F., Yuan, Z., Noble, F.L., Kroll, J.
- ID
- ZDB-PUB-190828-5
- Date
- 2019
- Source
- Cell cycle (Georgetown, Tex.) 18(20): 2683-2696 (Journal)
- Registered Authors
- Klems, Alina, Kroll, Jens, Lodd, Elisabeth
- Keywords
- Erythropoietin, compensation, gene knockout, kidney development, zebrafish
- MeSH Terms
-
- Animals
- CRISPR-Cas Systems
- Embryo, Nonmammalian/embryology
- Embryo, Nonmammalian/metabolism
- Embryo, Nonmammalian/ultrastructure
- Erythropoietin/genetics
- Erythropoietin/metabolism*
- Gene Expression Regulation, Developmental/genetics*
- Gene Knockdown Techniques
- Gene Knockout Techniques
- Heterozygote
- Homozygote
- Microscopy, Electron
- Morpholinos/genetics
- Organogenesis/genetics*
- Pronephros/abnormalities
- Pronephros/embryology*
- Pronephros/metabolism
- Recombinant Proteins/genetics
- Zebrafish/embryology
- Zebrafish/genetics*
- Zebrafish/metabolism
- Zebrafish Proteins/genetics
- Zebrafish Proteins/metabolism*
- PubMed
- 31451030 Full text @ Cell Cycle
Citation
She, J., Wu, Y., Lou, B., Lodd, E., Klems, A., Schmoehl, F., Yuan, Z., Noble, F.L., Kroll, J. (2019) Genetic compensation by epob in pronephros development in epoa mutant zebrafish. Cell cycle (Georgetown, Tex.). 18(20):2683-2696.
Abstract
Zebrafish erythropoietin a (epoa) is a well characterized regulator of red blood cell formation. Recent morpholino mediated knockdown data have also identified epoa being essential for physiological pronephros development in zebrafish, which is driven by blocking apoptosis in developing kidneys. Yet, zebrafish mutants for epoa have not been described so far. In order to compare a transient knockdown vs. permanent knockout for epoa in zebrafish on pronephros development, we used CRISPR/Cas9 technology to generate epoa knockout zebrafish mutants and we performed structural and functional studies on pronephros development. In contrast to epoa morphants, epoa-/- zebrafish mutants showed normal pronephros structure; however, a previously uncharacterized gene in zebrafish, named epob, was identified and upregulated in epoa-/- mutants. epob knockdown altered pronephros development, which was further aggravated in epoa-/- mutants. Likewise, epoa and epob morphants regulated similar and differential gene signatures related to kidney development in zebrafish. In conclusion, stable loss of epoa during embryonic development can be compensated by epob leading to phenotypical discrepancies in epoa knockdown and knockout zebrafish embryos.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping