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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Veröffentlicht in:PloS one 2021-01, Vol.16 (1), p.e0245239-e0245239
Hauptverfasser: El Fersioui, Younes, Pinton, Gaëtan, Allaman-Pillet, Nathalie, Schorderet, Daniel F
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Pinton, Gaëtan
Allaman-Pillet, Nathalie
Schorderet, Daniel F
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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1932-6203
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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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