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Loss of Wiz Function Affects Methylation Pattern in Palate Development and Leads to Cleft Palate
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SYSNO ASEP 0544116 Document Type J - Journal Article R&D Document Type Journal Article Subsidiary J Článek ve WOS Title Loss of Wiz Function Affects Methylation Pattern in Palate Development and Leads to Cleft Palate Author(s) Buková, Ivana (UMG-J)
Szczerkowska, Katarzyna (UMG-J)
Procházková, Michaela (UMG-J)
Beck, Inken (UMG-J) RID
Procházka, Jan (UMG-J) ORCID
Sedláček, Radislav (UMG-J) RIDNumber of authors 6 Article number 620692 Source Title Frontiers in Cell and Developmental Biology. - : Frontiers Research Foundation - ISSN 2296-634X
Roč. 9, June (2021)Number of pages 9 s. Publication form Online - E Language eng - English Country CH - Switzerland Keywords Wiz ; G9a ; glp ; histone methylation ; cleft palate ; development ; craniofacial Subject RIV EB - Genetics ; Molecular Biology OECD category Developmental biology R&D Projects LQ1604 GA MŠMT - Ministry of Education, Youth and Sports (MEYS) LM2015040 GA MŠMT - Ministry of Education, Youth and Sports (MEYS) LM2018126 GA MŠMT - Ministry of Education, Youth and Sports (MEYS) ED1.1.00/02.0109 GA MŠMT - Ministry of Education, Youth and Sports (MEYS) ED2.1.00/19.0395 GA MŠMT - Ministry of Education, Youth and Sports (MEYS) EF16_013/0001789 GA MŠMT - Ministry of Education, Youth and Sports (MEYS) GA19-21696S GA ČR - Czech Science Foundation (CSF) Method of publishing Open access Institutional support UMG-J - RVO:68378050 UT WOS 000661860400001 DOI 10.3389/fcell.2021.620692 Annotation WIZ (Widely Interspaced Zinc Finger) is associated with the G9a-GLP protein complex, a key H3K9 methyltransferase suggesting a role in transcriptional repression. However, its role in embryonic development is poorly described. In order to assess the loss of function of WIZ, we generated CRISPR/Cas9 WIZ knockout mouse model with 32 nucleotide deletion. Observing the lethality status, we identified the WIZ knockouts to be subviable during embryonic development and non-viable after birth. Morphology of developing embryo was analyzed at E14.5 and E18.5 and our findings were supported by microCT scans. Wiz KO showed improper development in multiple aspects, specifically in the craniofacial area. In particular, shorter snout, cleft palate, and cleft eyelids were present in mutant embryos. Palatal shelves were hypomorphic and though elevated to a horizontal position on top of the tongue, they failed to make contact and fuse. By comparison of proliferation pattern and histone methylation in developing palatal shelves we brought new evidence of importance WIZ dependent G9a-GLP methylation complex in craniofacial development, especially in palate shelf fusion. Workplace Institute of Molecular Genetics Contact Nikol Škňouřilová, nikol.sknourilova@img.cas.cz, Tel.: 241 063 217 Year of Publishing 2022 Electronic address https://www.frontiersin.org/articles/10.3389/fcell.2021.620692/full
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