Identification of novel fish sialidase genes responsible for KDN-cleaving activity

Shiozaki, Kazuhiro; Uezono, Keiya; Hirai, Go; Honda, Akinobu; Minoda, Masaya; Wakata, Ryuta

doi:10.1007/s10719-020-09948-6

Cited by 5 publications

(1 citation statement)

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“…Most emotional related-genes are known to be conserved across vertebrates, and several methodologies of behavioral analysis have been established in fish 22 , 23 . In addition, recent studies have revealed that neu1 genes are conserved in fish species, with their enzymatic properties 24 – 27 . Fish Neu1 is known to be localized at the lysosome and its enzymatic properties, such as optimal pH and substrate specificity, have been reported to be shared among fish species.…”

Section: Introductionmentioning

confidence: 99%

Neu1 deficiency induces abnormal emotional behavior in zebrafish

Ikeda

Komamizu

Hayashi

et al. 2021

Sci Rep

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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.

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