{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,18]],"date-time":"2026-09-18T04:57:25Z","timestamp":1789707445548,"version":"4.0.1"},"reference-count":77,"publisher":"Oxford University Press (OUP)","issue":"9","license":[{"start":{"date-parts":[[2020,4,21]],"date-time":"2020-04-21T00:00:00Z","timestamp":1587427200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/journals\/pages\/open_access\/funder_policies\/chorus\/standard_publication_model"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["1R01MH107515"],"award-info":[{"award-number":["1R01MH107515"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000001","name":"National Science Foundation","doi-asserted-by":"publisher","award":["DGE-1745038"],"award-info":[{"award-number":["DGE-1745038"]}],"id":[{"id":"10.13039\/100000001","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100008152","name":"Autism Science Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100008152","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2020,6,3]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Gtf2ird1 and Gtf2i are two transcription factors (TFs) among the 28 genes deleted in Williams syndrome, and prior mouse models of each TF show behavioral phenotypes. Here we identify their genomic binding sites in the developing brain and test for additive effects of their mutation on transcription and behavior. GTF2IRD1 binding targets were enriched for transcriptional and chromatin regulators and mediators of ubiquitination. GTF2I targets were enriched for signal transduction proteins, including regulators of phosphorylation and WNT. Both TFs are highly enriched at promoters, strongly overlap CTCF binding and topological associating domain boundaries and moderately overlap each other, suggesting epistatic effects. Shared TF targets are enriched for reactive oxygen species-responsive genes, synaptic proteins and transcription regulators such as chromatin modifiers, including a significant number of highly constrained genes and known ASD genes. We next used single and double mutants to test whether mutating both TFs will modify transcriptional and behavioral phenotypes of single Gtf2ird1 mutants, though with the caveat that our Gtf2ird1 mutants, like others previously reported, do produce low levels of a truncated protein product. Despite little difference in DNA binding and transcriptome-wide expression, homozygous Gtf2ird1 mutation caused balance, marble burying and conditioned fear phenotypes. However, mutating Gtf2i in addition to Gtf2ird1 did not further modify transcriptomic or most behavioral phenotypes, suggesting Gtf2ird1 mutation alone was sufficient for the observed phenotypes.<\/jats:p>","DOI":"10.1093\/hmg\/ddaa070","type":"journal-article","created":{"date-parts":[[2020,4,13]],"date-time":"2020-04-13T15:12:11Z","timestamp":1586790731000},"page":"1498-1519","source":"Crossref","is-referenced-by-count":35,"title":["Functions of\n                    <i>Gtf2i<\/i>\n                    and\n                    <i>Gtf2ird1<\/i>\n                    in the developing brain: transcription, DNA binding and long-term behavioral consequences"],"prefix":"10.1093","volume":"29","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0858-3242","authenticated-orcid":false,"given":"Nathan D","family":"Kopp","sequence":"first","affiliation":[{"name":"Department of Genetics, Washington University School of Medicine, St. Louis, MO 63110, USA"},{"name":"Department of Psychiatry, Washington University School of Medicine, St. Louis, MO 63110, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kayla R","family":"Nygaard","sequence":"additional","affiliation":[{"name":"Department of Genetics, Washington University School of Medicine, St. Louis, MO 63110, USA"},{"name":"Department of Psychiatry, Washington University School of Medicine, St. Louis, MO 63110, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yating","family":"Liu","sequence":"additional","affiliation":[{"name":"Department of Genetics, Washington University School of Medicine, St. Louis, MO 63110, USA"},{"name":"Department of Psychiatry, Washington University School of Medicine, St. Louis, MO 63110, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katherine B","family":"McCullough","sequence":"additional","affiliation":[{"name":"Department of Genetics, Washington University School of Medicine, St. Louis, MO 63110, USA"},{"name":"Department of Psychiatry, Washington University School of Medicine, St. Louis, MO 63110, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Susan E","family":"Maloney","sequence":"additional","affiliation":[{"name":"Department of Psychiatry, Washington University School of Medicine, St. Louis, MO 63110, USA"},{"name":"Intellectual and Developmental Disabilities Research Center, Washington University School of Medicine, St. Louis, MO 63110, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Harrison W","family":"Gabel","sequence":"additional","affiliation":[{"name":"Department of Neuroscience, Washington University School of Medicine, St Louis, MO 63110, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joseph D","family":"Dougherty","sequence":"additional","affiliation":[{"name":"Department of Genetics, Washington University School of Medicine, St. Louis, MO 63110, USA"},{"name":"Department of Psychiatry, Washington University School of Medicine, St. Louis, MO 63110, USA"},{"name":"Intellectual and Developmental Disabilities Research Center, Washington University School of Medicine, St. Louis, MO 63110, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2020,4,21]]},"reference":[{"key":"2021042100031249400_ref1","doi-asserted-by":"crossref","first-page":"3","DOI":"10.1007\/s00439-010-0827-2","article-title":"Copy number variants at Williams\u2013Beuren syndrome 7q11.23 region","volume":"128","author":"Merla","year":"2010","journal-title":"Hum. 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Neurosci."},{"key":"2021042100031249400_ref4","doi-asserted-by":"crossref","first-page":"28","DOI":"10.1002\/aur.169","article-title":"Haploinsufficiency of Gtf 2i, a gene deleted in Williams syndrome, leads to increases in social interactions","volume":"4","author":"Sakurai","year":"2011","journal-title":"Autism Res."},{"key":"2021042100031249400_ref5","doi-asserted-by":"crossref","first-page":"6481","DOI":"10.1093\/hmg\/ddu368","article-title":"Heterozygous deletion of the Williams\u2013Beuren syndrome critical interval in mice recapitulates most features of the human disorder","volume":"23","author":"Segura-Puimedon","year":"2014","journal-title":"Hum. Mol. Genet."},{"key":"2021042100031249400_ref6","doi-asserted-by":"crossref","first-page":"50","DOI":"10.1002\/emmm.200900003","article-title":"Induced chromosome deletions cause hypersociability and other features of Williams\u2013Beuren syndrome in mice","volume":"1","author":"Li","year":"2009","journal-title":"EMBO Mol. 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Chem."},{"key":"2021042100031249400_ref27","doi-asserted-by":"crossref","first-page":"E677","DOI":"10.1073\/pnas.1416674112","article-title":"Genome-wide targeting of the epigenetic regulatory protein CTCF to gene promoters by the transcription factor TFII-I","volume":"112","author":"Pe\u00f1a-Hern\u00e1ndez","year":"2015","journal-title":"PNAS"},{"key":"2021042100031249400_ref28","doi-asserted-by":"crossref","first-page":"132","DOI":"10.1038\/ng.3169","article-title":"7q11.23 dosage-dependent dysregulation in human pluripotent stem cells affects transcriptional programs in disease-relevant lineages","volume":"47","author":"Adamo","year":"2015","journal-title":"Nat. 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Genet."},{"key":"2021042100031249400_ref33","doi-asserted-by":"crossref","first-page":"1008","DOI":"10.1016\/j.cell.2013.10.031","article-title":"Integrative functional genomic analyses implicate specific molecular pathways and circuits in autism","volume":"155","author":"Parikshak","year":"2013","journal-title":"Cell"},{"key":"2021042100031249400_ref34","doi-asserted-by":"publisher","DOI":"10.1101\/531210","article-title":"Variation across 141, 456 human exomes and genomes reveals the spectrum of loss-of-function intolerance across human protein-coding genes","author":"Karczewski","year":"2019","journal-title":"bioRxiv"},{"key":"2021042100031249400_ref35","doi-asserted-by":"crossref","first-page":"4715","DOI":"10.1074\/jbc.M109.086660","article-title":"Negative autoregulation of GTF2IRD1 in Williams-Beuren syndrome via a novel DNA binding mechanism","volume":"285","author":"Palmer","year":"2010","journal-title":"J. Biol. Chem."},{"key":"2021042100031249400_ref36","doi-asserted-by":"crossref","first-page":"2588","DOI":"10.1110\/ps.04747604","article-title":"Comparison of TFII-I gene family members deleted in Williams-Beuren syndrome","volume":"13","author":"Hinsley","year":"2004","journal-title":"Protein Sci."},{"key":"2021042100031249400_ref37","article-title":"Global analysis of gene expression in the developing brain of Gtf 2ird1 knockout mice","volume":"6","author":"O\u2019Leary","year":"2011","journal-title":"PLoS One"},{"key":"2021042100031249400_ref38","doi-asserted-by":"crossref","first-page":"1184","DOI":"10.1126\/science.1116142","article-title":"GTF2IRD1 in craniofacial development of humans and mice","volume":"310","author":"Tassabehji","year":"2005","journal-title":"Science"},{"key":"2021042100031249400_ref39","doi-asserted-by":"publisher","first-page":"1691","DOI":"10.1038\/mt.2015.130","article-title":"Intracisternal Gtf 2i gene therapy ameliorates deficits in cognition and synaptic plasticity of a mouse model of Williams\u2013Beuren syndrome","volume":"23","author":"Borralleras","year":"2015","journal-title":"Mol. Ther."},{"key":"2021042100031249400_ref40","doi-asserted-by":"crossref","first-page":"291","DOI":"10.1207\/S15326942DN231&2_13","article-title":"Anxiety, fears, and phobias in persons with Williams syndrome","volume":"23","author":"Dykens","year":"2003","journal-title":"Dev. Neuropsychol."},{"key":"2021042100031249400_ref41","doi-asserted-by":"crossref","first-page":"181","DOI":"10.1073\/pnas.0811531106","article-title":"Essential functions of the Williams-Beuren syndrome-associated TFII-I genes in embryonic development","volume":"106","author":"Enkhmandakh","year":"2009","journal-title":"PNAS"},{"key":"2021042100031249400_ref42","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0049283","article-title":"SUMOylation of GTF2IRD1 regulates protein partner interactions and ubiquitin-mediated degradation","volume":"7","author":"Widagdo","year":"2012","journal-title":"PLoS One"},{"key":"2021042100031249400_ref43","doi-asserted-by":"crossref","DOI":"10.1523\/ENEURO.0016-14.2015","article-title":"Disruption of Src is associated with phenotypes related to Williams-Beuren syndrome and altered cellular localization of TFII-I","volume":"2","author":"Sinai","year":"2015","journal-title":"eNeuro"},{"key":"2021042100031249400_ref44","doi-asserted-by":"crossref","first-page":"481","DOI":"10.3389\/fnmol.2018.00481","article-title":"Common CHD8 genomic targets contrast with model-specific transcriptional impacts of CHD8 haploinsufficiency","volume":"11","author":"Wade","year":"2019","journal-title":"Front. 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Neurosci."},{"key":"2021042100031249400_ref45","doi-asserted-by":"crossref","first-page":"15536","DOI":"10.1073\/pnas.242566899","article-title":"Transcriptional profiling of a mouse model for Rett syndrome reveals subtle transcriptional changes in the brain","volume":"99","author":"Tudor","year":"2002","journal-title":"PNAS"},{"key":"2021042100031249400_ref46","doi-asserted-by":"crossref","first-page":"1224","DOI":"10.1126\/science.1153252","article-title":"MeCP2, a key contributor to neurological disease, activates and represses transcription","volume":"320","author":"Chahrour","year":"2008","journal-title":"Science"},{"key":"2021042100031249400_ref47","doi-asserted-by":"crossref","first-page":"1151","DOI":"10.1016\/j.cell.2017.09.047","article-title":"Early-life gene expression in neurons modulates lasting epigenetic states","volume":"171","author":"Stroud","year":"2017","journal-title":"Cell"},{"key":"2021042100031249400_ref48","doi-asserted-by":"crossref","first-page":"831","DOI":"10.1016\/j.neuron.2018.09.027","article-title":"Neonatal Tbr 1 dosage controls cortical layer 6 connectivity","volume":"100","author":"Fazel Darbandi","year":"2018","journal-title":"Neuron"},{"key":"2021042100031249400_ref49","doi-asserted-by":"crossref","first-page":"322","DOI":"10.1038\/85899","article-title":"A mouse Mecp 2-null mutation causes neurological symptoms that mimic Rett syndrome","volume":"27","author":"Guy","year":"2001","journal-title":"Nat. Genet."},{"key":"2021042100031249400_ref50","doi-asserted-by":"crossref","first-page":"216","DOI":"10.1016\/j.ajhg.2015.05.019","article-title":"Symmetrical dose-dependent DNA-methylation profiles in children with deletion or duplication of 7q11.23","volume":"97","author":"Strong","year":"2015","journal-title":"Am. J. Hum. Genet."},{"key":"2021042100031249400_ref51","doi-asserted-by":"crossref","first-page":"61","DOI":"10.1186\/1471-2350-11-61","article-title":"Essential role of the N-terminal region of TFII-I in viability and behavior","volume":"11","author":"Lucena","year":"2010","journal-title":"BMC Med. Genet."},{"key":"2021042100031249400_ref52","doi-asserted-by":"crossref","first-page":"203","DOI":"10.1016\/S0166-4328(01)00401-6","article-title":"Hippocampal cytotoxic lesion effects on species-typical behaviours in mice","volume":"132","author":"Deacon","year":"2002","journal-title":"Behav. Brain Res."},{"key":"2021042100031249400_ref53","doi-asserted-by":"crossref","first-page":"112","DOI":"10.1016\/j.nbd.2006.12.009","article-title":"Contribution of CYLN2 and GTF2IRD1 to neurological and cognitive symptoms in Williams syndrome","volume":"26","author":"Hagen","year":"2007","journal-title":"Neurobiol. Dis."},{"key":"2021042100031249400_ref54","first-page":"478","article-title":"Detection of an atypical 7q11.23 deletion in Williams syndrome patients which does not include the STX1A and FZD3 genes","volume":"36","author":"Botta","year":"1999","journal-title":"J. Med. Genet."},{"key":"2021042100031249400_ref55","doi-asserted-by":"crossref","first-page":"2482","DOI":"10.1101\/gad.11.19.2482","article-title":"A multifunctional DNA-binding protein that promotes the formation of serum response factor\/homeodomain complexes: identity to TFII-I","volume":"11","author":"Grueneberg","year":"1997","journal-title":"Genes Dev."},{"key":"2021042100031249400_ref56","doi-asserted-by":"crossref","first-page":"1252304","DOI":"10.1126\/science.1252304","article-title":"Neuronal activity promotes oligodendrogenesis and adaptive myelination in the mammalian brain","volume":"344","author":"Gibson","year":"2014","journal-title":"Science"},{"key":"2021042100031249400_ref57","doi-asserted-by":"crossref","first-page":"2114","DOI":"10.1093\/bioinformatics\/btu170","article-title":"Trimmomatic: a flexible trimmer for Illumina sequence data","volume":"30","author":"Bolger","year":"2014","journal-title":"Bioinformatics"},{"key":"2021042100031249400_ref58","doi-asserted-by":"crossref","first-page":"421","DOI":"10.1093\/bioinformatics\/bty648","article-title":"Scaling read aligners to hundreds of threads on general-purpose processors","volume":"35","author":"Langmead","year":"2019","journal-title":"Bioinformatics"},{"key":"2021042100031249400_ref59","doi-asserted-by":"crossref","first-page":"R137","DOI":"10.1186\/gb-2008-9-9-r137","article-title":"Model-based analysis of ChIP-Seq (MACS)","volume":"9","author":"Zhang","year":"2008","journal-title":"Genome Biol."},{"key":"2021042100031249400_ref60","doi-asserted-by":"crossref","first-page":"841","DOI":"10.1093\/bioinformatics\/btq033","article-title":"BEDTools: a flexible suite of utilities for comparing genomic features","volume":"26","author":"Quinlan","year":"2010","journal-title":"Bioinformatics"},{"key":"2021042100031249400_ref61","doi-asserted-by":"crossref","first-page":"139","DOI":"10.1093\/bioinformatics\/btp616","article-title":"Edge R: a Bioconductor package for differential expression analysis of digital gene expression data","volume":"26","author":"Robinson","year":"2010","journal-title":"Bioinformatics"},{"key":"2021042100031249400_ref62","doi-asserted-by":"crossref","first-page":"576","DOI":"10.1016\/j.molcel.2010.05.004","article-title":"Simple combinations of lineage-determining transcription factors prime cis-regulatory elements required for macrophage and B cell identities","volume":"38","author":"Heinz","year":"2010","journal-title":"Mol. 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