PUBLICATION

Increased anxiety was found in serpini1 knockout zebrafish larval

Authors
Han, S., Fei, F., Sun, S., Zhang, D., Dong, Q., Wang, X., Wang, L.
ID
ZDB-PUB-201120-78
Date
2020
Source
Biochemical and Biophysical Research Communications   534: 1013-1019 (Journal)
Registered Authors
Wang, Xu
Keywords
Anxiety, Axon defect, Mutant zebrafish model, Neurodegeneration, serpini1
MeSH Terms
  • Animals
  • Anxiety/genetics*
  • Anxiety Disorders/genetics*
  • CRISPR-Cas Systems
  • Disease Models, Animal
  • Gene Knockout Techniques
  • Humans
  • Larva/genetics
  • Neuropeptides/genetics*
  • Serpins/genetics*
  • Transcriptome
  • Zebrafish/genetics*
  • Zebrafish Proteins/genetics*
PubMed
33168193 Full text @ Biochem. Biophys. Res. Commun.
Abstract
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
Figures
Show all Figures
Expression
Phenotype
Mutation and Transgenics
Human Disease / Model Data
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping
Errata and Notes