Loss of Wiz Function Affects Methylation Pattern in Palate Development and Leads to Cleft Palate

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...
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Autor*in:	Ivana Bukova [verfasserIn] Katarzyna Izabela Szczerkowska [verfasserIn] Michaela Prochazkova [verfasserIn] Inken M. Beck [verfasserIn] Jan Prochazka [verfasserIn] Radislav Sedlacek [verfasserIn]

Format:	E-Artikel
Sprache:	Englisch

Erschienen:	2021

Schlagwörter:	Wiz G9a/GLP histone methylation cleft palate development craniofacial

Übergeordnetes Werk:	In: Frontiers in Cell and Developmental Biology - Frontiers Media S.A., 2014, 9(2021)
Übergeordnetes Werk:	volume:9 ; year:2021

Links:	Link aufrufen Link aufrufen Link aufrufen Journal toc

DOI / URN:	10.3389/fcell.2021.620692

Katalog-ID:	DOAJ059191619

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spelling	10.3389/fcell.2021.620692 doi (DE-627)DOAJ059191619 (DE-599)DOAJcf311487e87e48b8b2855482f96f3e19 DE-627 ger DE-627 rakwb eng QH301-705.5 Ivana Bukova verfasserin aut Loss of Wiz Function Affects Methylation Pattern in Palate Development and Leads to Cleft Palate 2021 Text txt rdacontent Computermedien c rdamedia Online-Ressource cr rdacarrier 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. Wiz G9a/GLP histone methylation cleft palate development craniofacial Biology (General) Katarzyna Izabela Szczerkowska verfasserin aut Michaela Prochazkova verfasserin aut Inken M. Beck verfasserin aut Inken M. Beck verfasserin aut Jan Prochazka verfasserin aut Radislav Sedlacek verfasserin aut In Frontiers in Cell and Developmental Biology Frontiers Media S.A., 2014 9(2021) (DE-627)770398138 (DE-600)2737824-X 2296634X nnns volume:9 year:2021 https://doi.org/10.3389/fcell.2021.620692 kostenfrei https://doaj.org/article/cf311487e87e48b8b2855482f96f3e19 kostenfrei https://www.frontiersin.org/articles/10.3389/fcell.2021.620692/full kostenfrei https://doaj.org/toc/2296-634X Journal toc kostenfrei GBV_USEFLAG_A SYSFLAG_A GBV_DOAJ GBV_ILN_11 GBV_ILN_20 GBV_ILN_22 GBV_ILN_23 GBV_ILN_24 GBV_ILN_39 GBV_ILN_40 GBV_ILN_62 GBV_ILN_63 GBV_ILN_65 GBV_ILN_69 GBV_ILN_70 GBV_ILN_73 GBV_ILN_74 GBV_ILN_95 GBV_ILN_105 GBV_ILN_110 GBV_ILN_151 GBV_ILN_161 GBV_ILN_170 GBV_ILN_213 GBV_ILN_230 GBV_ILN_285 GBV_ILN_293 GBV_ILN_602 GBV_ILN_2003 GBV_ILN_2014 GBV_ILN_4012 GBV_ILN_4037 GBV_ILN_4112 GBV_ILN_4125 GBV_ILN_4126 GBV_ILN_4249 GBV_ILN_4305 GBV_ILN_4306 GBV_ILN_4307 GBV_ILN_4313 GBV_ILN_4322 GBV_ILN_4323 GBV_ILN_4324 GBV_ILN_4325 GBV_ILN_4338 GBV_ILN_4367 GBV_ILN_4700 AR 9 2021
allfields_unstemmed	10.3389/fcell.2021.620692 doi (DE-627)DOAJ059191619 (DE-599)DOAJcf311487e87e48b8b2855482f96f3e19 DE-627 ger DE-627 rakwb eng QH301-705.5 Ivana Bukova verfasserin aut Loss of Wiz Function Affects Methylation Pattern in Palate Development and Leads to Cleft Palate 2021 Text txt rdacontent Computermedien c rdamedia Online-Ressource cr rdacarrier 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. Wiz G9a/GLP histone methylation cleft palate development craniofacial Biology (General) Katarzyna Izabela Szczerkowska verfasserin aut Michaela Prochazkova verfasserin aut Inken M. Beck verfasserin aut Inken M. Beck verfasserin aut Jan Prochazka verfasserin aut Radislav Sedlacek verfasserin aut In Frontiers in Cell and Developmental Biology Frontiers Media S.A., 2014 9(2021) (DE-627)770398138 (DE-600)2737824-X 2296634X nnns volume:9 year:2021 https://doi.org/10.3389/fcell.2021.620692 kostenfrei https://doaj.org/article/cf311487e87e48b8b2855482f96f3e19 kostenfrei https://www.frontiersin.org/articles/10.3389/fcell.2021.620692/full kostenfrei https://doaj.org/toc/2296-634X Journal toc kostenfrei GBV_USEFLAG_A SYSFLAG_A GBV_DOAJ GBV_ILN_11 GBV_ILN_20 GBV_ILN_22 GBV_ILN_23 GBV_ILN_24 GBV_ILN_39 GBV_ILN_40 GBV_ILN_62 GBV_ILN_63 GBV_ILN_65 GBV_ILN_69 GBV_ILN_70 GBV_ILN_73 GBV_ILN_74 GBV_ILN_95 GBV_ILN_105 GBV_ILN_110 GBV_ILN_151 GBV_ILN_161 GBV_ILN_170 GBV_ILN_213 GBV_ILN_230 GBV_ILN_285 GBV_ILN_293 GBV_ILN_602 GBV_ILN_2003 GBV_ILN_2014 GBV_ILN_4012 GBV_ILN_4037 GBV_ILN_4112 GBV_ILN_4125 GBV_ILN_4126 GBV_ILN_4249 GBV_ILN_4305 GBV_ILN_4306 GBV_ILN_4307 GBV_ILN_4313 GBV_ILN_4322 GBV_ILN_4323 GBV_ILN_4324 GBV_ILN_4325 GBV_ILN_4338 GBV_ILN_4367 GBV_ILN_4700 AR 9 2021
allfieldsGer	10.3389/fcell.2021.620692 doi (DE-627)DOAJ059191619 (DE-599)DOAJcf311487e87e48b8b2855482f96f3e19 DE-627 ger DE-627 rakwb eng QH301-705.5 Ivana Bukova verfasserin aut Loss of Wiz Function Affects Methylation Pattern in Palate Development and Leads to Cleft Palate 2021 Text txt rdacontent Computermedien c rdamedia Online-Ressource cr rdacarrier 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. Wiz G9a/GLP histone methylation cleft palate development craniofacial Biology (General) Katarzyna Izabela Szczerkowska verfasserin aut Michaela Prochazkova verfasserin aut Inken M. Beck verfasserin aut Inken M. Beck verfasserin aut Jan Prochazka verfasserin aut Radislav Sedlacek verfasserin aut In Frontiers in Cell and Developmental Biology Frontiers Media S.A., 2014 9(2021) (DE-627)770398138 (DE-600)2737824-X 2296634X nnns volume:9 year:2021 https://doi.org/10.3389/fcell.2021.620692 kostenfrei https://doaj.org/article/cf311487e87e48b8b2855482f96f3e19 kostenfrei https://www.frontiersin.org/articles/10.3389/fcell.2021.620692/full kostenfrei https://doaj.org/toc/2296-634X Journal toc kostenfrei GBV_USEFLAG_A SYSFLAG_A GBV_DOAJ GBV_ILN_11 GBV_ILN_20 GBV_ILN_22 GBV_ILN_23 GBV_ILN_24 GBV_ILN_39 GBV_ILN_40 GBV_ILN_62 GBV_ILN_63 GBV_ILN_65 GBV_ILN_69 GBV_ILN_70 GBV_ILN_73 GBV_ILN_74 GBV_ILN_95 GBV_ILN_105 GBV_ILN_110 GBV_ILN_151 GBV_ILN_161 GBV_ILN_170 GBV_ILN_213 GBV_ILN_230 GBV_ILN_285 GBV_ILN_293 GBV_ILN_602 GBV_ILN_2003 GBV_ILN_2014 GBV_ILN_4012 GBV_ILN_4037 GBV_ILN_4112 GBV_ILN_4125 GBV_ILN_4126 GBV_ILN_4249 GBV_ILN_4305 GBV_ILN_4306 GBV_ILN_4307 GBV_ILN_4313 GBV_ILN_4322 GBV_ILN_4323 GBV_ILN_4324 GBV_ILN_4325 GBV_ILN_4338 GBV_ILN_4367 GBV_ILN_4700 AR 9 2021
allfieldsSound	10.3389/fcell.2021.620692 doi (DE-627)DOAJ059191619 (DE-599)DOAJcf311487e87e48b8b2855482f96f3e19 DE-627 ger DE-627 rakwb eng QH301-705.5 Ivana Bukova verfasserin aut Loss of Wiz Function Affects Methylation Pattern in Palate Development and Leads to Cleft Palate 2021 Text txt rdacontent Computermedien c rdamedia Online-Ressource cr rdacarrier 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. Wiz G9a/GLP histone methylation cleft palate development craniofacial Biology (General) Katarzyna Izabela Szczerkowska verfasserin aut Michaela Prochazkova verfasserin aut Inken M. Beck verfasserin aut Inken M. Beck verfasserin aut Jan Prochazka verfasserin aut Radislav Sedlacek verfasserin aut In Frontiers in Cell and Developmental Biology Frontiers Media S.A., 2014 9(2021) (DE-627)770398138 (DE-600)2737824-X 2296634X nnns volume:9 year:2021 https://doi.org/10.3389/fcell.2021.620692 kostenfrei https://doaj.org/article/cf311487e87e48b8b2855482f96f3e19 kostenfrei https://www.frontiersin.org/articles/10.3389/fcell.2021.620692/full kostenfrei https://doaj.org/toc/2296-634X Journal toc kostenfrei GBV_USEFLAG_A SYSFLAG_A GBV_DOAJ GBV_ILN_11 GBV_ILN_20 GBV_ILN_22 GBV_ILN_23 GBV_ILN_24 GBV_ILN_39 GBV_ILN_40 GBV_ILN_62 GBV_ILN_63 GBV_ILN_65 GBV_ILN_69 GBV_ILN_70 GBV_ILN_73 GBV_ILN_74 GBV_ILN_95 GBV_ILN_105 GBV_ILN_110 GBV_ILN_151 GBV_ILN_161 GBV_ILN_170 GBV_ILN_213 GBV_ILN_230 GBV_ILN_285 GBV_ILN_293 GBV_ILN_602 GBV_ILN_2003 GBV_ILN_2014 GBV_ILN_4012 GBV_ILN_4037 GBV_ILN_4112 GBV_ILN_4125 GBV_ILN_4126 GBV_ILN_4249 GBV_ILN_4305 GBV_ILN_4306 GBV_ILN_4307 GBV_ILN_4313 GBV_ILN_4322 GBV_ILN_4323 GBV_ILN_4324 GBV_ILN_4325 GBV_ILN_4338 GBV_ILN_4367 GBV_ILN_4700 AR 9 2021
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container_title	Frontiers in Cell and Developmental Biology
authorswithroles_txt_mv	Ivana Bukova @@aut@@ Katarzyna Izabela Szczerkowska @@aut@@ Michaela Prochazkova @@aut@@ Inken M. Beck @@aut@@ Jan Prochazka @@aut@@ Radislav Sedlacek @@aut@@
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callnumber-first	Q - Science
author	Ivana Bukova
spellingShingle	Ivana Bukova misc QH301-705.5 misc Wiz misc G9a/GLP misc histone methylation misc cleft palate misc development misc craniofacial misc Biology (General) Loss of Wiz Function Affects Methylation Pattern in Palate Development and Leads to Cleft Palate
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topic_title	QH301-705.5 Loss of Wiz Function Affects Methylation Pattern in Palate Development and Leads to Cleft Palate Wiz G9a/GLP histone methylation cleft palate development craniofacial
topic	misc QH301-705.5 misc Wiz misc G9a/GLP misc histone methylation misc cleft palate misc development misc craniofacial misc Biology (General)
topic_unstemmed	misc QH301-705.5 misc Wiz misc G9a/GLP misc histone methylation misc cleft palate misc development misc craniofacial misc Biology (General)
topic_browse	misc QH301-705.5 misc Wiz misc G9a/GLP misc histone methylation misc cleft palate misc development misc craniofacial misc Biology (General)
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title	Loss of Wiz Function Affects Methylation Pattern in Palate Development and Leads to Cleft Palate
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title_full	Loss of Wiz Function Affects Methylation Pattern in Palate Development and Leads to Cleft Palate
author_sort	Ivana Bukova
journal	Frontiers in Cell and Developmental Biology
journalStr	Frontiers in Cell and Developmental Biology
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author_browse	Ivana Bukova Katarzyna Izabela Szczerkowska Michaela Prochazkova Inken M. Beck Jan Prochazka Radislav Sedlacek
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title_sort	loss of wiz function affects methylation pattern in palate development and leads to cleft palate
callnumber	QH301-705.5
title_auth	Loss of Wiz Function Affects Methylation Pattern in Palate Development and Leads to Cleft Palate
abstract	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.
abstractGer	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.
abstract_unstemmed	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.
collection_details	GBV_USEFLAG_A SYSFLAG_A GBV_DOAJ GBV_ILN_11 GBV_ILN_20 GBV_ILN_22 GBV_ILN_23 GBV_ILN_24 GBV_ILN_39 GBV_ILN_40 GBV_ILN_62 GBV_ILN_63 GBV_ILN_65 GBV_ILN_69 GBV_ILN_70 GBV_ILN_73 GBV_ILN_74 GBV_ILN_95 GBV_ILN_105 GBV_ILN_110 GBV_ILN_151 GBV_ILN_161 GBV_ILN_170 GBV_ILN_213 GBV_ILN_230 GBV_ILN_285 GBV_ILN_293 GBV_ILN_602 GBV_ILN_2003 GBV_ILN_2014 GBV_ILN_4012 GBV_ILN_4037 GBV_ILN_4112 GBV_ILN_4125 GBV_ILN_4126 GBV_ILN_4249 GBV_ILN_4305 GBV_ILN_4306 GBV_ILN_4307 GBV_ILN_4313 GBV_ILN_4322 GBV_ILN_4323 GBV_ILN_4324 GBV_ILN_4325 GBV_ILN_4338 GBV_ILN_4367 GBV_ILN_4700
title_short	Loss of Wiz Function Affects Methylation Pattern in Palate Development and Leads to Cleft Palate
url	https://doi.org/10.3389/fcell.2021.620692 https://doaj.org/article/cf311487e87e48b8b2855482f96f3e19 https://www.frontiersin.org/articles/10.3389/fcell.2021.620692/full https://doaj.org/toc/2296-634X
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author2	Katarzyna Izabela Szczerkowska Michaela Prochazkova Inken M. Beck Jan Prochazka Radislav Sedlacek
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doi_str	10.3389/fcell.2021.620692
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up_date	2024-07-03T22:15:48.062Z
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