PUBLICATION

Neu1 deficiency induces abnormal emotional behavior in zebrafish

Authors
Ikeda, A., Komamizu, M., Hayashi, A., Yamasaki, C., Okada, K., Kawabe, M., Komatsu, M., Shiozaki, K.
ID
ZDB-PUB-210701-5
Date
2021
Source
Scientific Reports   11: 13477 (Journal)
Registered Authors
Shiozaki, Kazuhiro
Keywords
none
MeSH Terms
  • Animals
  • Behavior, Animal*
  • Emotions*
  • Gene Knockout Techniques
  • Neuraminidase/deficiency*
  • Neuraminidase/metabolism
  • Social Behavior*
  • Zebrafish*
  • Zebrafish Proteins/deficiency*
  • Zebrafish Proteins/metabolism
PubMed
34188220 Full text @ Sci. Rep.
Abstract
NEU1 sialidase hydrolyzes sialic acids from glycoconjugates in lysosomes. Deficiency of NEU1 causes sialidosis with symptoms including facial dysmorphism, bone dysplasia, and neurodegeneration. However, the effects of NEU1 deficiency on emotional activity have not been explored. Here, we conducted the behavioral analysis using Neu1-knockout zebrafish (Neu1-KO). Neu1-KO zebrafish showed normal swimming similar to wild-type zebrafish (WT), whereas shoaling was decreased and accompanied by greater inter-fish distance than WT zebrafish. The aggression test showed a reduced aggressive behavior in Neu1-KO zebrafish than in WT zebrafish. In the mirror and 3-chambers test, Neu1-KO zebrafish showed more interest toward the opponent in the mirror and multiple unfamiliar zebrafish, respectively, than WT zebrafish. Furthermore, Neu1-KO zebrafish also showed increased interaction with different fish species, whereas WT zebrafish avoided them. In the black-white preference test, Neu1-KO zebrafish showed an abnormal preference for the white region, whereas WT zebrafish preferred the black region. Neu1-KO zebrafish were characterized by a downregulation of the anxiety-related genes of the hypothalamic-pituitary-adrenal axis and upregulation of lamp1a, an activator of lysosomal exocytosis, with their brains accumulating several sphingoglycolipids. This study revealed that Neu1 deficiency caused abnormal emotional behavior in zebrafish, possibly due to neuronal dysfunction induced by lysosomal exocytosis.
Genes / Markers
Figures
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Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping