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.
|
Authors | Sha Han, Fei Fei, Shaoyang Sun, Dongyang Zhang, Qiang Dong, Xu Wang, Liang Wang |
Journal | Biochemical and biophysical research communications
(Biochem Biophys Res Commun)
Vol. 534
Pg. 1013-1019
(01 01 2021)
ISSN: 1090-2104 [Electronic] United States |
PMID | 33168193
(Publication Type: Journal Article, Research Support, Non-U.S. Gov't)
|
Copyright | Copyright © 2020 Elsevier Inc. All rights reserved. |
Chemical References |
- Neuropeptides
- Serpins
- Zebrafish Proteins
|
Topics |
- 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)
- Neuroserpin
|