PXD029008 is an
original dataset announced via ProteomeXchange.
Dataset Summary
Title | C. elegans TFIIH subunit GTF-2H5/TTDA is a non-essential transcription factor indispensable for DNA repairC. elegans TFIIH subunit GTF-2H5/TTDA is a non-essential transcription factor indispensable for DNA repair. |
Description | The 10-subunit TFIIH complex is vital to transcription and nucleotide excision repair. Hereditary mutations in its smallest subunit, TTDA/GTF2H5, cause a photosensitive form of the rare developmental disorder trichothiodystrophy. Some trichothiodystrophy features are thought to be caused by subtle transcription or gene expression defects. TTDA/GTF2H5 knockout mice are not viable, making it difficult to investigate TTDA/GTF2H5 in vivo function. Here we show that deficiency of C. elegans TTDA ortholog GTF-2H5 is, however, compatible with life, in contrast to depletion of other TFIIH subunits. GTF-2H5 promotes TFIIH stability in multiple tissues and is indispensable for nucleotide excision repair, in which it facilitates recruitment of TFIIH to DNA damage. Strikingly, when transcription is challenged, gtf-2H5 embryos die due to the intrinsic TFIIH fragility in absence of GTF-2H5. These results support the idea that TTDA/GTF2H5 mutations cause transcription impairment underlying trichothiodystrophy and establish C. elegans as model for studying pathogenesis of this disease. |
HostingRepository | PRIDE |
AnnounceDate | 2021-11-19 |
AnnouncementXML | Submission_2021-11-19_07:16:37.982.xml |
DigitalObjectIdentifier | |
ReviewLevel | Peer-reviewed dataset |
DatasetOrigin | Original dataset |
RepositorySupport | Unsupported dataset by repository |
PrimarySubmitter | Jeroen Demmers |
SpeciesList | scientific name: Caenorhabditis elegans; NCBI TaxID: 6239; |
ModificationList | No PTMs are included in the dataset |
Instrument | Orbitrap Fusion Lumos |
Dataset History
Revision | Datetime | Status | ChangeLog Entry |
0 | 2021-10-08 06:42:02 | ID requested | |
⏵ 1 | 2021-11-19 07:16:38 | announced | |
Publication List
Dataset with its publication pending |
Keyword List
submitter keyword: DNA repair, C. elegans, IP-MS, TFIIH, transcription |
Contact List
Jeroen Demmers |
contact affiliation | Proteomics Center, Erasmus University Medical Center Rotterdam |
contact email | j.demmers@erasmusmc.nl |
lab head | |
Jeroen Demmers |
contact affiliation | Proteomics Center, Erasmus University Medical Center, Rotterdam, The Netherlands |
contact email | j.demmers@erasmusmc.nl |
dataset submitter | |
Full Dataset Link List
Dataset FTP location
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PRIDE project URI |
Repository Record List
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[ - ]
- PRIDE
- PXD029008
- Label: PRIDE project
- Name: C. elegans TFIIH subunit GTF-2H5/TTDA is a non-essential transcription factor indispensable for DNA repairC. elegans TFIIH subunit GTF-2H5/TTDA is a non-essential transcription factor indispensable for DNA repair.