Neu1 deficiency induces abnormal emotional behavior in zebrafish

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 co...

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Autores principales:	Asami Ikeda, Mayu Komamizu, Akito Hayashi, Chiharu Yamasaki, Keiji Okada, Momoko Kawabe, Masaharu Komatsu, Kazuhiro Shiozaki
Formato:	article
Lenguaje:	EN
Publicado:	Nature Portfolio 2021
Materias:	Medicine R Science Q
Acceso en línea:	https://doaj.org/article/9df69a038ca2444f80f9179b756c92d9
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spelling	oai:doaj.org-article:9df69a038ca2444f80f9179b756c92d92021-12-02T16:10:36ZNeu1 deficiency induces abnormal emotional behavior in zebrafish10.1038/s41598-021-92778-92045-2322https://doaj.org/article/9df69a038ca2444f80f9179b756c92d92021-06-01T00:00:00Zhttps://doi.org/10.1038/s41598-021-92778-9https://doaj.org/toc/2045-2322Abstract 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.Asami IkedaMayu KomamizuAkito HayashiChiharu YamasakiKeiji OkadaMomoko KawabeMasaharu KomatsuKazuhiro ShiozakiNature PortfolioarticleMedicineRScienceQENScientific Reports, Vol 11, Iss 1, Pp 1-15 (2021)
institution	DOAJ
collection	DOAJ
language	EN
topic	Medicine R Science Q
spellingShingle	Medicine R Science Q Asami Ikeda Mayu Komamizu Akito Hayashi Chiharu Yamasaki Keiji Okada Momoko Kawabe Masaharu Komatsu Kazuhiro Shiozaki Neu1 deficiency induces abnormal emotional behavior in zebrafish
description	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.
format	article
author	Asami Ikeda Mayu Komamizu Akito Hayashi Chiharu Yamasaki Keiji Okada Momoko Kawabe Masaharu Komatsu Kazuhiro Shiozaki
author_facet	Asami Ikeda Mayu Komamizu Akito Hayashi Chiharu Yamasaki Keiji Okada Momoko Kawabe Masaharu Komatsu Kazuhiro Shiozaki
author_sort	Asami Ikeda
title	Neu1 deficiency induces abnormal emotional behavior in zebrafish
title_short	Neu1 deficiency induces abnormal emotional behavior in zebrafish
title_full	Neu1 deficiency induces abnormal emotional behavior in zebrafish
title_fullStr	Neu1 deficiency induces abnormal emotional behavior in zebrafish
title_full_unstemmed	Neu1 deficiency induces abnormal emotional behavior in zebrafish
title_sort	neu1 deficiency induces abnormal emotional behavior in zebrafish
publisher	Nature Portfolio
publishDate	2021
url	https://doaj.org/article/9df69a038ca2444f80f9179b756c92d9
work_keys_str_mv	AT asamiikeda neu1deficiencyinducesabnormalemotionalbehaviorinzebrafish AT mayukomamizu neu1deficiencyinducesabnormalemotionalbehaviorinzebrafish AT akitohayashi neu1deficiencyinducesabnormalemotionalbehaviorinzebrafish AT chiharuyamasaki neu1deficiencyinducesabnormalemotionalbehaviorinzebrafish AT keijiokada neu1deficiencyinducesabnormalemotionalbehaviorinzebrafish AT momokokawabe neu1deficiencyinducesabnormalemotionalbehaviorinzebrafish AT masaharukomatsu neu1deficiencyinducesabnormalemotionalbehaviorinzebrafish AT kazuhiroshiozaki neu1deficiencyinducesabnormalemotionalbehaviorinzebrafish
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Neu1 deficiency induces abnormal emotional behavior in zebrafish

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