PUBLICATION
Tollip-deficient zebrafish display no abnormalities in development, organ morphology or gene expression in response to lipopolysaccharide
- Authors
- Wolińska-Nizioł, L., Romaniuk, K., Wojciechowska, K., Surga, K., Kamaszewski, M., Szudrowicz, H., Miączyńska, M.
- ID
- ZDB-PUB-220506-4
- Date
- 2022
- Source
- FEBS Open Bio 12(8): 1453-1464 (Journal)
- Registered Authors
- Miaczynska, Marta
- Keywords
- CRISPR/Cas9, Gaucher disease, LPS, Tollip, innate immunity, zebrafish
- MeSH Terms
-
- Animals
- Gene Expression
- Lipopolysaccharides*
- Lysosomes
- Mammals
- Zebrafish*/genetics
- PubMed
- 35506194 Full text @ FEBS Open Bio
Citation
Wolińska-Nizioł, L., Romaniuk, K., Wojciechowska, K., Surga, K., Kamaszewski, M., Szudrowicz, H., Miączyńska, M. (2022) Tollip-deficient zebrafish display no abnormalities in development, organ morphology or gene expression in response to lipopolysaccharide. FEBS Open Bio. 12(8):1453-1464.
Abstract
Tollip is a multifunctional adaptor protein implicated in innate immunity, lysosomal trafficking/autophagy of protein aggregates and various signaling processes in mammalian models. To verify evolutionary conservation of these functions, we used CRISPR/Cas9 editing to construct a zebrafish line bearing a stable tollip knockout. In contrast to previously reported tollip morphants, Tollip-deficient fish display normal development until adulthood, are fertile and have no apparent physiological defects. When challenged with lipopolysaccharide (LPS), inflammatory gene expression is unaffected. Moreover, Tollip deficiency does not aggravate swimming deficiency resulting from lysosomal dysfunction and proteotoxicity in a fish model of Gaucher disease. Thus, individual functions of Tollip may be organism-specific or manifest only upon certain conditions/challenges or disease backgrounds.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping