{"doi":"10.1038/s42003-021-02875-8","title":"C. elegans TFIIH subunit GTF-2H5/TTDA is a non-essential transcription factor indispensable for DNA repair","abstract":"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.","journal":"Communications Biology","year":2021,"id":183699,"datarank":0.0,"base_score":0.0,"endowment":0.0,"self_citation_contribution":0.0,"citation_network_contribution":0.0,"self_endowment_contribution":0.0,"citer_contribution":0.0,"corpus_percentile":null,"corpus_rank":null,"citation_count":13,"citer_count":0,"citers_with_citation_signal":0,"citers_with_endowment":0,"datacite_reuse_total":0,"is_dataset":false,"is_dataset_confidence":0.9561,"is_data_producer":false,"deposit_databanks":null,"is_oa":true,"file_count":0,"downloads":0,"has_version_chain":false,"published_date":"2021-01-01","fair_score":null,"fair_percentile":null,"algorithm_id":"datarank_citation_only_1hop_v6","ranking_scope":"data_only","authors":[{"id":737537,"name":"Melanie van der Woude","orcid":"0000-0003-2043-8563","position":1,"is_corresponding":false},{"id":348802,"name":"Carlota Davó-Martínez","orcid":"0000-0002-7182-6921","position":2,"is_corresponding":false},{"id":737538,"name":"Dick H. W. Dekkers","orcid":"0000-0001-7057-6257","position":3,"is_corresponding":false},{"id":686191,"name":"Mariangela Sabatella","orcid":"0000-0002-9177-4946","position":4,"is_corresponding":false},{"id":502863,"name":"Jeroen Demmers","orcid":"0000-0002-8757-9611","position":5,"is_corresponding":false},{"id":664931,"name":"Wim Vermeulen","orcid":"0000-0003-3616-734X","position":6,"is_corresponding":false},{"id":686193,"name":"Hannes Lans","orcid":"0000-0003-4417-5358","position":7,"is_corresponding":false},{"id":686192,"name":"Karen L. Thijssen","orcid":"0000-0002-6136-5164","position":0,"is_corresponding":true}],"reference_count":88,"raw_metadata":null,"created_at":"2026-07-18T23:48:22.008011Z","pmid":"34824371","pmcid":null,"fwci":null,"citation_percentile":null,"influential_citations":0,"oa_status":null,"license":null,"views":0,"total_file_size_bytes":0,"version_count":0,"fair_f":null,"fair_a":null,"fair_i":null,"fair_r":null,"fair_zscore":null,"fair_rationale":null,"fair_model":null,"fair_agent_version":null,"fair_fulltext_source":null,"fair_has_llm":null,"fair_computed_at":null,"clinical_trials":[],"software_tools":[],"db_accessions":[],"linked_datasets":[],"topics":[]}