Structural Analysis and Spatiotemporal Expression of <i>Atxn1</i> Genes in Zebrafish Embryos and Larvae

Zebrafish have come into focus to model cerebellar diseases such as spinocerebellar ataxias (SCAs), which is caused by an expansion of translated CAG repeats in several unrelated genes. In spinocerebellar ataxia type 1 (SCA1), gain-of-function in the mutant ATXN1 contributes to SCA1’s neuropathy. Hu...

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Autores principales:	Franz Vauti, Viktoria Vögele, Isabel Deppe, Susanne T. Hahnenstein, Reinhard W. Köster
Formato:	article
Lenguaje:	EN
Publicado:	MDPI AG 2021
Materias:	zebrafish spinocerebellar ataxia SCA ataxin-1 <i>atxn1a</i> <i>atxn1b</i> Biology (General) QH301-705.5 Chemistry QD1-999
Acceso en línea:	https://doaj.org/article/67cb7b997c3047139be18c6bebeb2011
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spelling	oai:doaj.org-article:67cb7b997c3047139be18c6bebeb20112021-11-11T16:49:35ZStructural Analysis and Spatiotemporal Expression of <i>Atxn1</i> Genes in Zebrafish Embryos and Larvae10.3390/ijms2221113481422-00671661-6596https://doaj.org/article/67cb7b997c3047139be18c6bebeb20112021-10-01T00:00:00Zhttps://www.mdpi.com/1422-0067/22/21/11348https://doaj.org/toc/1661-6596https://doaj.org/toc/1422-0067Zebrafish have come into focus to model cerebellar diseases such as spinocerebellar ataxias (SCAs), which is caused by an expansion of translated CAG repeats in several unrelated genes. In spinocerebellar ataxia type 1 (SCA1), gain-of-function in the mutant ATXN1 contributes to SCA1’s neuropathy. Human ATXN1 and its paralog ATXN1L are chromatin-binding factors, act as transcriptional repressors, and have similar expression patterns. However, little is known about <i>atxn1</i> genes in zebrafish. Recently, two family members, <i>atxn1a</i> and <i>atxn1b</i>, were identified as duplicate orthologs of <i>ATXN1</i>, as was a<i>txn1l</i>, the ortholog of <i>ATXN1L</i>. In this study, we analyzed the phylogenetic relationship of the <i>atxn1</i> family members in zebrafish, compared their genetic structures, and verified the predicted transcripts by both RT-PCR and whole-mount in situ hybridization. All three genes, <i>atxn1a</i>, <i>atxn1b</i>, and <i>atxn1l</i>, show overlapping, but also distinct, expression domains during embryonic and larval development. While <i>atxn1a</i> and <i>atxn1l</i> display similar spatiotemporal embryonic expression, <i>atxn1b</i> expression is initiated during the onset of brain development and is predominantly expressed in the cerebellum throughout zebrafish development. These results provide new insights into <i>atxn1</i> genes and their expression patterns in zebrafish during embryonic and late-larval development and may contribute importantly to future experiments in disease modeling of SCAs.Franz VautiViktoria VögeleIsabel DeppeSusanne T. HahnensteinReinhard W. KösterMDPI AGarticlezebrafishspinocerebellar ataxiaSCAataxin-1<i>atxn1a</i><i>atxn1b</i>Biology (General)QH301-705.5ChemistryQD1-999ENInternational Journal of Molecular Sciences, Vol 22, Iss 11348, p 11348 (2021)
institution	DOAJ
collection	DOAJ
language	EN
topic	zebrafish spinocerebellar ataxia SCA ataxin-1 <i>atxn1a</i> <i>atxn1b</i> Biology (General) QH301-705.5 Chemistry QD1-999
spellingShingle	zebrafish spinocerebellar ataxia SCA ataxin-1 <i>atxn1a</i> <i>atxn1b</i> Biology (General) QH301-705.5 Chemistry QD1-999 Franz Vauti Viktoria Vögele Isabel Deppe Susanne T. Hahnenstein Reinhard W. Köster Structural Analysis and Spatiotemporal Expression of <i>Atxn1</i> Genes in Zebrafish Embryos and Larvae
description	Zebrafish have come into focus to model cerebellar diseases such as spinocerebellar ataxias (SCAs), which is caused by an expansion of translated CAG repeats in several unrelated genes. In spinocerebellar ataxia type 1 (SCA1), gain-of-function in the mutant ATXN1 contributes to SCA1’s neuropathy. Human ATXN1 and its paralog ATXN1L are chromatin-binding factors, act as transcriptional repressors, and have similar expression patterns. However, little is known about <i>atxn1</i> genes in zebrafish. Recently, two family members, <i>atxn1a</i> and <i>atxn1b</i>, were identified as duplicate orthologs of <i>ATXN1</i>, as was a<i>txn1l</i>, the ortholog of <i>ATXN1L</i>. In this study, we analyzed the phylogenetic relationship of the <i>atxn1</i> family members in zebrafish, compared their genetic structures, and verified the predicted transcripts by both RT-PCR and whole-mount in situ hybridization. All three genes, <i>atxn1a</i>, <i>atxn1b</i>, and <i>atxn1l</i>, show overlapping, but also distinct, expression domains during embryonic and larval development. While <i>atxn1a</i> and <i>atxn1l</i> display similar spatiotemporal embryonic expression, <i>atxn1b</i> expression is initiated during the onset of brain development and is predominantly expressed in the cerebellum throughout zebrafish development. These results provide new insights into <i>atxn1</i> genes and their expression patterns in zebrafish during embryonic and late-larval development and may contribute importantly to future experiments in disease modeling of SCAs.
format	article
author	Franz Vauti Viktoria Vögele Isabel Deppe Susanne T. Hahnenstein Reinhard W. Köster
author_facet	Franz Vauti Viktoria Vögele Isabel Deppe Susanne T. Hahnenstein Reinhard W. Köster
author_sort	Franz Vauti
title	Structural Analysis and Spatiotemporal Expression of <i>Atxn1</i> Genes in Zebrafish Embryos and Larvae
title_short	Structural Analysis and Spatiotemporal Expression of <i>Atxn1</i> Genes in Zebrafish Embryos and Larvae
title_full	Structural Analysis and Spatiotemporal Expression of <i>Atxn1</i> Genes in Zebrafish Embryos and Larvae
title_fullStr	Structural Analysis and Spatiotemporal Expression of <i>Atxn1</i> Genes in Zebrafish Embryos and Larvae
title_full_unstemmed	Structural Analysis and Spatiotemporal Expression of <i>Atxn1</i> Genes in Zebrafish Embryos and Larvae
title_sort	structural analysis and spatiotemporal expression of <i>atxn1</i> genes in zebrafish embryos and larvae
publisher	MDPI AG
publishDate	2021
url	https://doaj.org/article/67cb7b997c3047139be18c6bebeb2011
work_keys_str_mv	AT franzvauti structuralanalysisandspatiotemporalexpressionofiatxn1igenesinzebrafishembryosandlarvae AT viktoriavogele structuralanalysisandspatiotemporalexpressionofiatxn1igenesinzebrafishembryosandlarvae AT isabeldeppe structuralanalysisandspatiotemporalexpressionofiatxn1igenesinzebrafishembryosandlarvae AT susannethahnenstein structuralanalysisandspatiotemporalexpressionofiatxn1igenesinzebrafishembryosandlarvae AT reinhardwkoster structuralanalysisandspatiotemporalexpressionofiatxn1igenesinzebrafishembryosandlarvae
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Structural Analysis and Spatiotemporal Expression of <i>Atxn1</i> Genes in Zebrafish Embryos and Larvae

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