Increased anxiety was found in serpini1 knockout zebrafish larval
- Han, S., Fei, F., Sun, S., Zhang, D., Dong, Q., Wang, X., Wang, L.
- Biochemical and Biophysical Research Communications 534: 1013-1019 (Journal)
- Registered Authors
- Wang, Xu
- Anxiety, Axon defect, Mutant zebrafish model, Neurodegeneration, serpini1
- MeSH Terms
- Anxiety Disorders/genetics*
- CRISPR-Cas Systems
- Disease Models, Animal
- Gene Knockout Techniques
- Zebrafish Proteins/genetics*
- 33168193 Full text @ Biochem. Biophys. Res. Commun.
Han, S., Fei, F., Sun, S., Zhang, D., Dong, Q., Wang, X., Wang, L. (2020) Increased anxiety was found in serpini1 knockout zebrafish larval. Biochemical and Biophysical Research Communications. 534:1013-1019.
Serpini1, which encodes neuroserpin, has been implicated in the development and normal function of the nervous system. Mutations in serpini1 cause familial encephalopathy, a rare neurodegenerative disorder characterized with neuroserpin inclusion bodies. However, function of neuroserpin in the nervous system is not fully understood. In this study, we generated a novel serpini1 mutant zebrafish model to investigate the loss of function of neuroserpin. Serpini1- deficient mutation was created with the CRISPR/Cas9 technique. No severe morphological characteristics were found in serpini1- deficient zebrafish. Serpini1-/- zebrafish larvae did not cause locomotor defects but displayed anxiety-like behavior. Extension of motoneurons axon defect was observed in serpini1-/- zebrafish. Furthermore, RNA-sequencing analysis revealed that loss of serpini1 resulted in affected expression of neurodegeneration-related genes.
Genes / Markers
Mutation and Transgenics
Human Disease / Model Data
Sequence Targeting Reagents
Engineered Foreign Genes
Errata and Notes