期刊
BIOCHEMICAL AND BIOPHYSICAL RESEARCH COMMUNICATIONS
卷 534, 期 -, 页码 1013-1019出版社
ACADEMIC PRESS INC ELSEVIER SCIENCE
DOI: 10.1016/j.bbrc.2020.10.048
关键词
serpini1; Mutant zebrafish model; Anxiety; Axon defect; Neurodegeneration
资金
- Science and Technology Commission of Shanghai Municipality [13441902600]
Serpini1, the gene encoding neuroserpin, is associated with the development and normal function of the nervous system, and mutations can cause familial encephalopathy. In a zebrafish model, serpini1 deficiency resulted in anxiety-like behavior and defects in motoneuron axon extension, as well as affected expression of neurodegeneration-related genes.
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. (C) 2020 Elsevier Inc. All rights reserved.
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