PUBLICATION
Characterization of genetic loss-of-function of Fus in zebrafish
- Authors
- Lebedeva, S., de Jesus Domingues, A.M., Butter, F., Ketting, R.F.
- ID
- ZDB-PUB-161130-2
- Date
- 2017
- Source
- RNA Biology 14(1): 29-35 (Journal)
- Registered Authors
- Ketting, René
- Keywords
- CRISPR-Cas9, Danio rerio, Fus, genetic knockout, morpholino, zebrafish
- Datasets
- GEO:GSE85554
- MeSH Terms
-
- RNA-Binding Protein FUS/genetics*
- RNA-Binding Protein FUS/metabolism
- Gene Targeting
- RNA, Guide, Kinetoplastida
- Gene Knockout Techniques
- Base Sequence
- Proteome
- Binding Sites
- Zebrafish/genetics*
- Zebrafish/metabolism
- 3' Untranslated Regions
- Animals
- Transcriptome
- Genetic Background
- Brain/metabolism
- Genotype
- CRISPR-Cas Systems
- Alleles
- Exons
- PubMed
- 27898262 Full text @ RNA Biol.
Citation
Lebedeva, S., de Jesus Domingues, A.M., Butter, F., Ketting, R.F. (2017) Characterization of genetic loss-of-function of Fus in zebrafish. RNA Biology. 14(1):29-35.
Abstract
The RNA-binding protein FUS is implicated in transcription, alternative splicing of neuronal genes and DNA repair. Mutations in FUS have been linked to human neurodegenerative diseases such as ALS (amyotrophic lateral sclerosis). We genetically disrupted fus in zebrafish (Danio rerio) using the CRISPR-Cas9 system. The fus knockout animals are fertile and did not show any distinctive phenotype. Mutation of fus induces mild changes in gene expression on the transcriptome and proteome level in the adult brain. We observed a significant influence of genetic background on gene expression and 3'UTR usage, which could mask the effects of loss of Fus. Unlike published fus morphants, maternal zygotic fus mutants do not show motoneuronal degeneration and exhibit normal locomotor activity.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping