Hmx1 regulates urfh1 expression in the craniofacial region in zebrafish
H6 family homeobox 1 (HMX1) regulates multiple aspects of craniofacial development as it is widely expressed in the eye, peripheral ganglia and branchial arches. Mutations in HMX1 are linked to an ocular defect termed Oculo-auricular syndrome of Schorderet-Munier-Franceschetti (MIM #612109). We iden...
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description | H6 family homeobox 1 (HMX1) regulates multiple aspects of craniofacial development as it is widely expressed in the eye, peripheral ganglia and branchial arches. Mutations in HMX1 are linked to an ocular defect termed Oculo-auricular syndrome of Schorderet-Munier-Franceschetti (MIM #612109). We identified UHRF1 as a target of HMX1 during development. UHRF1 and its partner proteins actively regulate chromatin modifications and cellular proliferation. Luciferase assays and in situ hybridization analyses showed that HMX1 exerts a transcriptional inhibitory effect on UHRF1 and a modification of its expression pattern. Overexpression of hmx1 in hsp70-hmx1 zebrafish increased uhrf1 expression in the cranial region, while mutations in the hmx1 dimerization domains reduced uhrf1 expression. Moreover, the expression level of uhrf1 and its partner dnmt1 was increased in the eye field in response to hmx1 overexpression. These results indicate that hmx1 regulates uhrf1 expression and, potentially through regulating the expression of factors involved in DNA methylation, contribute to the development of the craniofacial region of zebrafish. |
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Mutations in HMX1 are linked to an ocular defect termed Oculo-auricular syndrome of Schorderet-Munier-Franceschetti (MIM #612109). We identified UHRF1 as a target of HMX1 during development. UHRF1 and its partner proteins actively regulate chromatin modifications and cellular proliferation. Luciferase assays and in situ hybridization analyses showed that HMX1 exerts a transcriptional inhibitory effect on UHRF1 and a modification of its expression pattern. Overexpression of hmx1 in hsp70-hmx1 zebrafish increased uhrf1 expression in the cranial region, while mutations in the hmx1 dimerization domains reduced uhrf1 expression. Moreover, the expression level of uhrf1 and its partner dnmt1 was increased in the eye field in response to hmx1 overexpression. These results indicate that hmx1 regulates uhrf1 expression and, potentially through regulating the expression of factors involved in DNA methylation, contribute to the development of the craniofacial region of zebrafish.</description><identifier>ISSN: 1932-6203</identifier><identifier>EISSN: 1932-6203</identifier><identifier>DOI: 10.1371/journal.pone.0245239</identifier><identifier>PMID: 33465110</identifier><language>eng</language><publisher>United States: Public Library of Science</publisher><subject>Binding sites ; Biology and Life Sciences ; Cell cycle ; Cell division ; Cell migration ; Danio rerio ; Deoxyribonucleic acid ; Developmental stages ; DNA ; DNA binding proteins ; DNA methylation ; DNMT1 protein ; Editing ; Electronic mail ; Embryogenesis ; Embryonic development ; Embryonic growth stage ; Embryos ; Epigenetics ; Experiments ; Face ; Gene expression ; Genetic aspects ; Genetic research ; Heat shock ; Hindbrain ; Homeobox genes ; Homeotic genes ; Hybridization ; Intestine ; Mammalian cells ; Medicine and Health Sciences ; Mutation ; Neural crest ; Neural tube ; Ophthalmology ; Physiological aspects ; Research and Analysis Methods ; Retina ; Reviews ; Transcription ; Transcription factors ; Transfection ; Vertebrates ; Zebra fish ; Zebrafish</subject><ispartof>PloS one, 2021-01, Vol.16 (1), p.e0245239-e0245239</ispartof><rights>COPYRIGHT 2021 Public Library of Science</rights><rights>2021 El Fersioui et al. This is an open access article distributed under the terms of the Creative Commons Attribution License: http://creativecommons.org/licenses/by/4.0/ (the “License”), which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License.</rights><rights>2021 El Fersioui et al 2021 El Fersioui et al</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c692t-92af5b65043170b27e4d681d393f53a9ad735de36f2ac27225da8bed6680ee113</citedby><cites>FETCH-LOGICAL-c692t-92af5b65043170b27e4d681d393f53a9ad735de36f2ac27225da8bed6680ee113</cites><orcidid>0000-0002-6064-927X</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC7815118/pdf/$$EPDF$$P50$$Gpubmedcentral$$Hfree_for_read</linktopdf><linktohtml>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC7815118/$$EHTML$$P50$$Gpubmedcentral$$Hfree_for_read</linktohtml><link.rule.ids>230,314,723,776,780,860,881,2095,2914,23846,27903,27904,53769,53771,79346,79347</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/33465110$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><contributor>Schubert, Michael</contributor><creatorcontrib>El Fersioui, Younes</creatorcontrib><creatorcontrib>Pinton, Gaëtan</creatorcontrib><creatorcontrib>Allaman-Pillet, Nathalie</creatorcontrib><creatorcontrib>Schorderet, Daniel F</creatorcontrib><title>Hmx1 regulates urfh1 expression in the craniofacial region in zebrafish</title><title>PloS one</title><addtitle>PLoS One</addtitle><description>H6 family homeobox 1 (HMX1) regulates multiple aspects of craniofacial development as it is widely expressed in the eye, peripheral ganglia and branchial arches. Mutations in HMX1 are linked to an ocular defect termed Oculo-auricular syndrome of Schorderet-Munier-Franceschetti (MIM #612109). We identified UHRF1 as a target of HMX1 during development. UHRF1 and its partner proteins actively regulate chromatin modifications and cellular proliferation. Luciferase assays and in situ hybridization analyses showed that HMX1 exerts a transcriptional inhibitory effect on UHRF1 and a modification of its expression pattern. Overexpression of hmx1 in hsp70-hmx1 zebrafish increased uhrf1 expression in the cranial region, while mutations in the hmx1 dimerization domains reduced uhrf1 expression. Moreover, the expression level of uhrf1 and its partner dnmt1 was increased in the eye field in response to hmx1 overexpression. These results indicate that hmx1 regulates uhrf1 expression and, potentially through regulating the expression of factors involved in DNA methylation, contribute to the development of the craniofacial region of zebrafish.</description><subject>Binding sites</subject><subject>Biology and Life Sciences</subject><subject>Cell cycle</subject><subject>Cell division</subject><subject>Cell migration</subject><subject>Danio rerio</subject><subject>Deoxyribonucleic acid</subject><subject>Developmental stages</subject><subject>DNA</subject><subject>DNA binding proteins</subject><subject>DNA methylation</subject><subject>DNMT1 protein</subject><subject>Editing</subject><subject>Electronic mail</subject><subject>Embryogenesis</subject><subject>Embryonic development</subject><subject>Embryonic growth stage</subject><subject>Embryos</subject><subject>Epigenetics</subject><subject>Experiments</subject><subject>Face</subject><subject>Gene expression</subject><subject>Genetic 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urfh1 expression in the craniofacial region in zebrafish</atitle><jtitle>PloS one</jtitle><addtitle>PLoS One</addtitle><date>2021-01-19</date><risdate>2021</risdate><volume>16</volume><issue>1</issue><spage>e0245239</spage><epage>e0245239</epage><pages>e0245239-e0245239</pages><issn>1932-6203</issn><eissn>1932-6203</eissn><abstract>H6 family homeobox 1 (HMX1) regulates multiple aspects of craniofacial development as it is widely expressed in the eye, peripheral ganglia and branchial arches. Mutations in HMX1 are linked to an ocular defect termed Oculo-auricular syndrome of Schorderet-Munier-Franceschetti (MIM #612109). We identified UHRF1 as a target of HMX1 during development. UHRF1 and its partner proteins actively regulate chromatin modifications and cellular proliferation. Luciferase assays and in situ hybridization analyses showed that HMX1 exerts a transcriptional inhibitory effect on UHRF1 and a modification of its expression pattern. Overexpression of hmx1 in hsp70-hmx1 zebrafish increased uhrf1 expression in the cranial region, while mutations in the hmx1 dimerization domains reduced uhrf1 expression. Moreover, the expression level of uhrf1 and its partner dnmt1 was increased in the eye field in response to hmx1 overexpression. These results indicate that hmx1 regulates uhrf1 expression and, potentially through regulating the expression of factors involved in DNA methylation, contribute to the development of the craniofacial region of zebrafish.</abstract><cop>United States</cop><pub>Public Library of Science</pub><pmid>33465110</pmid><doi>10.1371/journal.pone.0245239</doi><tpages>e0245239</tpages><orcidid>https://orcid.org/0000-0002-6064-927X</orcidid><oa>free_for_read</oa></addata></record> |
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subjects | Binding sites Biology and Life Sciences Cell cycle Cell division Cell migration Danio rerio Deoxyribonucleic acid Developmental stages DNA DNA binding proteins DNA methylation DNMT1 protein Editing Electronic mail Embryogenesis Embryonic development Embryonic growth stage Embryos Epigenetics Experiments Face Gene expression Genetic aspects Genetic research Heat shock Hindbrain Homeobox genes Homeotic genes Hybridization Intestine Mammalian cells Medicine and Health Sciences Mutation Neural crest Neural tube Ophthalmology Physiological aspects Research and Analysis Methods Retina Reviews Transcription Transcription factors Transfection Vertebrates Zebra fish Zebrafish |
title | Hmx1 regulates urfh1 expression in the craniofacial region in zebrafish |
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