C. elegans TFIIH subunit GTF-2H5/TTDA is a non-essential transcription factor indispensable for DNA repair

Hereditary mutations in TTDA/GTF2H5 cause a photosensitive form of the rare developmental disorder trichothiodystrophy, however the development of models has been hampered by mutations being lethal. Thijssen et al. show that deficiency of C. elegans TTDA ortholog GTF-2H5 is, compatible with life, in...

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Main Authors: Karen L. Thijssen, Melanie van der Woude, Carlota Davó-Martínez, Dick H. W. Dekkers, Mariangela Sabatella, Jeroen A. A. Demmers, Wim Vermeulen, Hannes Lans
Format: Article
Language:English
Published: Nature Portfolio 2021-11-01
Series:Communications Biology
Online Access:https://doi.org/10.1038/s42003-021-02875-8
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author Karen L. Thijssen
Melanie van der Woude
Carlota Davó-Martínez
Dick H. W. Dekkers
Mariangela Sabatella
Jeroen A. A. Demmers
Wim Vermeulen
Hannes Lans
author_facet Karen L. Thijssen
Melanie van der Woude
Carlota Davó-Martínez
Dick H. W. Dekkers
Mariangela Sabatella
Jeroen A. A. Demmers
Wim Vermeulen
Hannes Lans
author_sort Karen L. Thijssen
collection DOAJ
description Hereditary mutations in TTDA/GTF2H5 cause a photosensitive form of the rare developmental disorder trichothiodystrophy, however the development of models has been hampered by mutations being lethal. Thijssen et al. show that deficiency of C. elegans TTDA ortholog GTF-2H5 is, compatible with life, in contrast to depletion of other TFIIH subunits and thus propose that this model could be used for studying the pathogenesis of trichothiodystrophy.
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spelling doaj.art-aa9b284b2d79417c957d8200b15428232022-12-21T23:08:51ZengNature PortfolioCommunications Biology2399-36422021-11-014111210.1038/s42003-021-02875-8C. elegans TFIIH subunit GTF-2H5/TTDA is a non-essential transcription factor indispensable for DNA repairKaren L. Thijssen0Melanie van der Woude1Carlota Davó-Martínez2Dick H. W. Dekkers3Mariangela Sabatella4Jeroen A. A. Demmers5Wim Vermeulen6Hannes Lans7Department of Molecular Genetics, Oncode Institute, Erasmus MC Cancer Institute, Erasmus University Medical CenterDepartment of Molecular Genetics, Oncode Institute, Erasmus MC Cancer Institute, Erasmus University Medical CenterDepartment of Molecular Genetics, Oncode Institute, Erasmus MC Cancer Institute, Erasmus University Medical CenterProteomics Center, Erasmus University Medical CenterDepartment of Molecular Genetics, Oncode Institute, Erasmus MC Cancer Institute, Erasmus University Medical CenterProteomics Center, Erasmus University Medical CenterDepartment of Molecular Genetics, Oncode Institute, Erasmus MC Cancer Institute, Erasmus University Medical CenterDepartment of Molecular Genetics, Oncode Institute, Erasmus MC Cancer Institute, Erasmus University Medical CenterHereditary mutations in TTDA/GTF2H5 cause a photosensitive form of the rare developmental disorder trichothiodystrophy, however the development of models has been hampered by mutations being lethal. Thijssen et al. show that deficiency of C. elegans TTDA ortholog GTF-2H5 is, compatible with life, in contrast to depletion of other TFIIH subunits and thus propose that this model could be used for studying the pathogenesis of trichothiodystrophy.https://doi.org/10.1038/s42003-021-02875-8
spellingShingle Karen L. Thijssen
Melanie van der Woude
Carlota Davó-Martínez
Dick H. W. Dekkers
Mariangela Sabatella
Jeroen A. A. Demmers
Wim Vermeulen
Hannes Lans
C. elegans TFIIH subunit GTF-2H5/TTDA is a non-essential transcription factor indispensable for DNA repair
Communications Biology
title C. elegans TFIIH subunit GTF-2H5/TTDA is a non-essential transcription factor indispensable for DNA repair
title_full C. elegans TFIIH subunit GTF-2H5/TTDA is a non-essential transcription factor indispensable for DNA repair
title_fullStr C. elegans TFIIH subunit GTF-2H5/TTDA is a non-essential transcription factor indispensable for DNA repair
title_full_unstemmed C. elegans TFIIH subunit GTF-2H5/TTDA is a non-essential transcription factor indispensable for DNA repair
title_short C. elegans TFIIH subunit GTF-2H5/TTDA is a non-essential transcription factor indispensable for DNA repair
title_sort c elegans tfiih subunit gtf 2h5 ttda is a non essential transcription factor indispensable for dna repair
url https://doi.org/10.1038/s42003-021-02875-8
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