{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,19]],"date-time":"2026-10-19T16:55:42Z","timestamp":1792428942722,"version":"4.5.0"},"reference-count":75,"publisher":"Rockefeller University Press","issue":"5","license":[{"start":{"date-parts":[[2019,8,29]],"date-time":"2019-08-29T00:00:00Z","timestamp":1567036800000},"content-version":"vor","delay-in-days":868,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001691","name":"JSPS","doi-asserted-by":"publisher","award":["JSPS 25221309"],"award-info":[{"award-number":["JSPS 25221309"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100009619","name":"Japan Agency for Medical Research and Development","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100009619","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001700","name":"Ministry of Education, Culture, Sports, Science and Technology","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100001700","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["rupress.org"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2017,5,1]]},"abstract":"<jats:p>Abnormal epigenetic regulation can cause the nervous system to develop abnormally. Here, we sought to understand the mechanism by which this occurs by investigating the protein complex cohesin, which is considered to regulate gene expression and, when defective, is associated with higher-level brain dysfunction and the developmental disorder Cornelia de Lange syndrome (CdLS). We generated conditional Smc3-knockout mice and observed greater dendritic complexity and larger numbers of immature synapses in the cerebral cortex of Smc3+\/\u2212 mice. Smc3+\/\u2212 mice also exhibited more anxiety-related behavior, which is a symptom of CdLS. Further, a gene ontology analysis after RNA-sequencing suggested the enrichment of immune processes, particularly the response to interferons, in the Smc3+\/\u2212 mice. Indeed, fewer synapses formed in their cortical neurons, and this phenotype was rescued by STAT1 knockdown. Thus, low levels of cohesin expression in the developing brain lead to changes in gene expression that in turn lead to a specific and abnormal neuronal and behavioral phenotype.<\/jats:p>","DOI":"10.1084\/jem.20161517","type":"journal-article","created":{"date-parts":[[2017,4,13]],"date-time":"2017-04-13T14:10:17Z","timestamp":1492092617000},"page":"1431-1452","update-policy":"https:\/\/doi.org\/10.1084\/jem.crossmarkpolicy","source":"Crossref","is-referenced-by-count":61,"title":["Decreased cohesin in the brain leads to defective synapse development and anxiety-related behavior"],"prefix":"10.1084","volume":"214","author":[{"given":"Yuki","family":"Fujita","sequence":"first","affiliation":[{"name":"Department of Molecular Neuroscience, Graduate School of Medicine, Osaka University, Osaka 565-0871, Japan 1"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Koji","family":"Masuda","sequence":"additional","affiliation":[{"name":"Research Center for Epigenetic Disease, Institute for Molecular and Cellular Biosciences, The University of Tokyo, Tokyo 113-0032, Japan 7"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Masashige","family":"Bando","sequence":"additional","affiliation":[{"name":"Research Center for Epigenetic Disease, Institute for Molecular and Cellular Biosciences, The University of Tokyo, Tokyo 113-0032, Japan 7"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ryuichiro","family":"Nakato","sequence":"additional","affiliation":[{"name":"Research Center for Epigenetic Disease, Institute for Molecular and Cellular Biosciences, The University of Tokyo, Tokyo 113-0032, Japan 7"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yuki","family":"Katou","sequence":"additional","affiliation":[{"name":"Research Center for Epigenetic Disease, Institute for Molecular and Cellular Biosciences, The University of Tokyo, Tokyo 113-0032, Japan 7"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Takashi","family":"Tanaka","sequence":"additional","affiliation":[{"name":"Department of Molecular Neuroscience, Graduate School of Medicine, Osaka University, Osaka 565-0871, Japan 1"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Masahiro","family":"Nakayama","sequence":"additional","affiliation":[{"name":"Department of Pathology, Osaka Medical Center and Research Institute for Maternal and Child Health, Osaka 594-1101, Japan 8"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Keizo","family":"Takao","sequence":"additional","affiliation":[{"name":"Life Science Research Center, University of Toyama, Toyama 930-0194, Japan 9"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tsuyoshi","family":"Miyakawa","sequence":"additional","affiliation":[{"name":"Life Science Research Center, University of Toyama, Toyama 930-0194, Japan 9"},{"name":"Division of Systems Medical Science, Institute for Comprehensive Medical Science, Fujita Health University, Aichi 470-1192, Japan 10"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tatsunori","family":"Tanaka","sequence":"additional","affiliation":[{"name":"Laboratory of Molecular Neuropharmacology, Graduate School of Pharmaceutical Sciences, Osaka University, Osaka 565-0871, Japan 2"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yukio","family":"Ago","sequence":"additional","affiliation":[{"name":"Laboratory of Molecular Neuropharmacology, Graduate School of Pharmaceutical Sciences, Osaka University, Osaka 565-0871, Japan 2"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6548-4016","authenticated-orcid":true,"given":"Hitoshi","family":"Hashimoto","sequence":"additional","affiliation":[{"name":"Laboratory of Molecular Neuropharmacology, Graduate School of Pharmaceutical Sciences, Osaka University, Osaka 565-0871, Japan 2"},{"name":"Division of Bioscience, Institute for Datability Science, Osaka University, Osaka 565-0871, Japan 3"},{"name":"iPS Cell-based Research Project on Brain Neuropharmacology and Toxicology, Graduate School of Pharmaceutical Sciences, Osaka University, Osaka 565-0871, Japan 4"},{"name":"Molecular Research Center for Children\u2019s Mental Development, United Graduate School of Child Development, Osaka University, Osaka 565-0871, Japan 5"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katsuhiko","family":"Shirahige","sequence":"additional","affiliation":[{"name":"Research Center for Epigenetic Disease, Institute for Molecular and Cellular Biosciences, The University of Tokyo, Tokyo 113-0032, Japan 7"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4559-7018","authenticated-orcid":true,"given":"Toshihide","family":"Yamashita","sequence":"additional","affiliation":[{"name":"Department of Molecular Neuroscience, Graduate School of Medicine, Osaka University, Osaka 565-0871, Japan 1"},{"name":"Graduate School of Frontier Biosciences, Osaka University, Osaka 565-0871, Japan 6"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"291","published-online":{"date-parts":[[2017,4,13]]},"reference":[{"key":"2023072603543612500_bib1","doi-asserted-by":"publisher","first-page":"195","DOI":"10.1097\/00005072-199503000-00006","article-title":"Selective dendritic alterations in the cortex of Rett syndrome","volume":"54","author":"Armstrong","year":"1995","journal-title":"J. Neuropathol. Exp. Neurol."},{"key":"2023072603543612500_bib2","doi-asserted-by":"publisher","first-page":"1013","DOI":"10.1097\/00005072-199811000-00003","article-title":"Decreased dendritic branching in frontal, motor and limbic cortex in Rett syndrome compared with trisomy 21","volume":"57","author":"Armstrong","year":"1998","journal-title":"J. Neuropathol. Exp. Neurol."},{"key":"2023072603543612500_bib3","doi-asserted-by":"publisher","first-page":"671","DOI":"10.1111\/j.1365-2788.2007.00977.x","article-title":"The behavioural phenotype of Cornelia de Lange Syndrome: A study of 56 individuals","volume":"51","author":"Basile","year":"2007","journal-title":"J. Intellect. Disabil. Res."},{"key":"2023072603543612500_bib4","doi-asserted-by":"publisher","first-page":"520","DOI":"10.1002\/ana.410200413","article-title":"Dendritic atrophy in children with Down\u2019s syndrome","volume":"20","author":"Becker","year":"1986","journal-title":"Ann. Neurol."},{"key":"2023072603543612500_bib5","first-page":"133","article-title":"Growth and development of the brain in Down syndrome","volume":"373","author":"Becker","year":"1991","journal-title":"Prog. Clin. Biol. Res."},{"key":"2023072603543612500_bib6","doi-asserted-by":"publisher","first-page":"333","DOI":"10.1136\/adc.81.4.333","article-title":"Behavioural phenotype of Cornelia de Lange syndrome","volume":"81","author":"Berney","year":"1999","journal-title":"Arch. Dis. Child."},{"key":"2023072603543612500_bib7","doi-asserted-by":"publisher","first-page":"551","DOI":"10.1038\/nrn3992","article-title":"From the genetic architecture to synaptic plasticity in autism spectrum disorder","volume":"16","author":"Bourgeron","year":"2015","journal-title":"Nat. Rev. Neurosci."},{"key":"2023072603543612500_bib8","doi-asserted-by":"publisher","first-page":"248","DOI":"10.1002\/mrdd.20039","article-title":"Designing mouse behavioral tasks relevant to autistic-like behaviors","volume":"10","author":"Crawley","year":"2004","journal-title":"Ment. Retard. Dev. Disabil. Res. Rev."},{"key":"2023072603543612500_bib9","doi-asserted-by":"publisher","first-page":"7793","DOI":"10.1523\/JNEUROSCI.0577-10.2010","article-title":"Delayed stabilization of dendritic spines in fragile X mice","volume":"30","author":"Cruz-Mart\u00edn","year":"2010","journal-title":"J. Neurosci."},{"key":"2023072603543612500_bib10","doi-asserted-by":"publisher","first-page":"698","DOI":"10.1101\/gad.13.6.698","article-title":"The boundaries of the silenced HMR domain in Saccharomyces cerevisiae","volume":"13","author":"Donze","year":"1999","journal-title":"Genes Dev."},{"key":"2023072603543612500_bib11","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1007\/s00412-006-0072-6","article-title":"Roles of the sister chromatid cohesion apparatus in gene expression, development, and human syndromes","volume":"116","author":"Dorsett","year":"2007","journal-title":"Chromosoma."},{"key":"2023072603543612500_bib12","doi-asserted-by":"publisher","first-page":"771","DOI":"10.1016\/j.neubiorev.2005.03.017","article-title":"Recent advances in animal models of chronic antidepressant effects: The novelty-induced hypophagia test","volume":"29","author":"Dulawa","year":"2005","journal-title":"Neurosci. Biobehav. Rev."},{"key":"2023072603543612500_bib13","doi-asserted-by":"publisher","first-page":"423","DOI":"10.1038\/nn.2514","article-title":"Dnmt1 and Dnmt3a maintain DNA methylation and regulate synaptic function in adult forebrain neurons","volume":"13","author":"Feng","year":"2010","journal-title":"Nat. Neurosci."},{"key":"2023072603543612500_bib14","doi-asserted-by":"publisher","first-page":"e766","DOI":"10.1038\/tp.2016.31","article-title":"Deletion of JMJD2B in neurons leads to defective spine maturation, hyperactive behavior and memory deficits in mouse","volume":"6","author":"Fujiwara","year":"2016","journal-title":"Transl. Psychiatry."},{"key":"2023072603543612500_bib15","doi-asserted-by":"publisher","first-page":"47","DOI":"10.1016\/S0092-8674(01)80008-8","article-title":"A direct link between sister chromatid cohesion and chromosome condensation revealed through the analysis of MCD1 in S. cerevisiae","volume":"91","author":"Guacci","year":"1997","journal-title":"Cell."},{"key":"2023072603543612500_bib16","doi-asserted-by":"publisher","first-page":"773","DOI":"10.1016\/j.neuron.2004.05.003","article-title":"Altered cortical synaptic morphology and impaired memory consolidation in forebrain-specific dominant-negative PAK transgenic mice","volume":"42","author":"Hayashi","year":"2004","journal-title":"Neuron."},{"key":"2023072603543612500_bib17","doi-asserted-by":"publisher","first-page":"289","DOI":"10.1002\/ajmg.1320410306","article-title":"Analysis of neocortex in three males with the fragile X syndrome","volume":"41","author":"Hinton","year":"1991","journal-title":"Am. J. Med. Genet."},{"key":"2023072603543612500_bib18","doi-asserted-by":"publisher","first-page":"e132","DOI":"10.1093\/nar\/gkv670","article-title":"Biological chromodynamics: A general method for measuring protein occupancy across the genome by calibrating ChIP-seq","volume":"43","author":"Hu","year":"2015","journal-title":"Nucleic Acids Res."},{"key":"2023072603543612500_bib19","doi-asserted-by":"publisher","first-page":"233","DOI":"10.1002\/dvg.20212","article-title":"Temporal regulation of Cre recombinase activity in neural stem cells","volume":"44","author":"Imayoshi","year":"2006","journal-title":"Genesis."},{"key":"2023072603543612500_bib20","doi-asserted-by":"publisher","first-page":"1153","DOI":"10.1038\/nn.2185","article-title":"Roles of continuous neurogenesis in the structural and functional integrity of the adult forebrain","volume":"11","author":"Imayoshi","year":"2008","journal-title":"Nat. Neurosci."},{"key":"2023072603543612500_bib21","doi-asserted-by":"publisher","first-page":"338","DOI":"10.1038\/ng.3229","article-title":"Germline gain-of-function mutations in AFF4 cause a developmental syndrome functionally linking the super elongation complex and cohesin","volume":"47","author":"Izumi","year":"2015","journal-title":"Nat. Genet."},{"key":"2023072603543612500_bib22","doi-asserted-by":"publisher","first-page":"R36","DOI":"10.1186\/gb-2013-14-4-r36","article-title":"TopHat2: Accurate alignment of transcriptomes in the presence of insertions, deletions and gene fusions","volume":"14","author":"Kim","year":"2013","journal-title":"Genome Biol."},{"key":"2023072603543612500_bib23","doi-asserted-by":"crossref","first-page":"4530","DOI":"10.1523\/JNEUROSCI.22-11-04530.2002","article-title":"Interferon gamma induces retrograde dendritic retraction and inhibits synapse formation","volume":"22","author":"Kim","year":"2002","journal-title":"J. Neurosci."},{"key":"2023072603543612500_bib24","doi-asserted-by":"publisher","first-page":"1118","DOI":"10.2353\/ajpath.2006.060024","article-title":"Interferon-gamma plays protective roles in sodium arsenite-induced renal injury by up-regulating intrarenal multidrug resistance-associated protein 1 expression","volume":"169","author":"Kimura","year":"2006","journal-title":"Am. J. Pathol."},{"key":"2023072603543612500_bib25","doi-asserted-by":"publisher","first-page":"823","DOI":"10.1038\/nrn3618","article-title":"Temporal fate specification and neural progenitor competence during development","volume":"14","author":"Kohwi","year":"2013","journal-title":"Nat. Rev. Neurosci."},{"key":"2023072603543612500_bib26","doi-asserted-by":"publisher","first-page":"1088","DOI":"10.3791\/1088","author":"Komada","year":"2008","journal-title":"J. Vis. Exp."},{"key":"2023072603543612500_bib27","doi-asserted-by":"publisher","first-page":"33","DOI":"10.1007\/978-1-4939-0805-9_4","article-title":"Chromatin immunoprecipitation protocol for mammalian cells","volume":"1164","author":"Komata","year":"2014","journal-title":"Methods Mol. Biol."},{"key":"2023072603543612500_bib28","doi-asserted-by":"publisher","first-page":"631","DOI":"10.1038\/ng1364","article-title":"Cornelia de Lange syndrome is caused by mutations in NIPBL, the human homolog of Drosophila melanogaster Nipped-B","volume":"36","author":"Krantz","year":"2004","journal-title":"Nat. Genet."},{"key":"2023072603543612500_bib29","doi-asserted-by":"publisher","first-page":"357","DOI":"10.1038\/nmeth.1923","article-title":"Fast gapped-read alignment with Bowtie 2","volume":"9","author":"Langmead","year":"2012","journal-title":"Nat. Methods."},{"key":"2023072603543612500_bib30","doi-asserted-by":"publisher","first-page":"524","DOI":"10.1038\/nrm3393","article-title":"Navigating the epigenetic landscape of pluripotent stem cells","volume":"13","author":"Li","year":"2012","journal-title":"Nat. Rev. Mol. Cell Biol."},{"key":"2023072603543612500_bib31","doi-asserted-by":"publisher","first-page":"5657","DOI":"10.1093\/nar\/gkq346","article-title":"Genome-wide DNA methylation analysis in cohesin mutant human cell lines","volume":"38","author":"Liu","year":"2010","journal-title":"Nucleic Acids Res."},{"key":"2023072603543612500_bib32","doi-asserted-by":"publisher","first-page":"1986","DOI":"10.1101\/gad.12.13.1986","article-title":"Identification of Xenopus SMC protein complexes required for sister chromatid cohesion","volume":"12","author":"Losada","year":"1998","journal-title":"Genes Dev."},{"key":"2023072603543612500_bib33","doi-asserted-by":"publisher","first-page":"275","DOI":"10.1016\/j.bbrc.2004.06.139","article-title":"Lineage-specific cell disruption in living mice by Cre-mediated expression of diphtheria toxin A chain","volume":"321","author":"Matsumura","year":"2004","journal-title":"Biochem. Biophys. Res. Commun."},{"key":"2023072603543612500_bib34","doi-asserted-by":"publisher","first-page":"729","DOI":"10.1038\/nn1696","article-title":"Hippocampal neurogenesis is not required for behavioral effects of environmental enrichment","volume":"9","author":"Meshi","year":"2006","journal-title":"Nat. Neurosci."},{"key":"2023072603543612500_bib35","doi-asserted-by":"publisher","first-page":"35","DOI":"10.1016\/S0092-8674(01)80007-6","article-title":"Cohesins: Chromosomal proteins that prevent premature separation of sister chromatids","volume":"91","author":"Michaelis","year":"1997","journal-title":"Cell."},{"key":"2023072603543612500_bib36","doi-asserted-by":"publisher","first-page":"1694","DOI":"10.1016\/j.cub.2015.05.017","article-title":"Esco1 acetylates cohesin via a mechanism different from that of Esco2","volume":"25","author":"Minamino","year":"2015","journal-title":"Curr. Biol."},{"key":"2023072603543612500_bib37","doi-asserted-by":"publisher","first-page":"787","DOI":"10.1016\/j.cell.2007.01.028","article-title":"Beyond the sequence: cellular organization of genome function","volume":"128","author":"Misteli","year":"2007","journal-title":"Cell."},{"key":"2023072603543612500_bib38","doi-asserted-by":"publisher","first-page":"522","DOI":"10.1093\/cercor\/bhj177","article-title":"A transcriptional enhancer that directs telencephalon-specific transgene expression in mouse brain","volume":"17","author":"Mitsui","year":"2007","journal-title":"Cereb. Cortex."},{"key":"2023072603543612500_bib39","doi-asserted-by":"crossref","first-page":"5239","DOI":"10.1523\/JNEUROSCI.21-14-05239.2001","article-title":"Hyperactivity and intact hippocampus-dependent learning in mice lacking the M1 muscarinic acetylcholine receptor","volume":"21","author":"Miyakawa","year":"2001","journal-title":"J. Neurosci."},{"key":"2023072603543612500_bib40","doi-asserted-by":"publisher","first-page":"287","DOI":"10.1111\/j.1601-1848.2004.00076.x","article-title":"Sociability and preference for social novelty in five inbred strains: an approach to assess autistic-like behavior in mice","volume":"3","author":"Moy","year":"2004","journal-title":"Genes Brain Behav."},{"key":"2023072603543612500_bib41","doi-asserted-by":"publisher","first-page":"419","DOI":"10.1016\/j.bbrc.2008.03.103","article-title":"Neuron-specific recombination by Cre recombinase inserted into the murine tau locus","volume":"370","author":"Muramatsu","year":"2008","journal-title":"Biochem. Biophys. Res. Commun."},{"key":"2023072603543612500_bib42","doi-asserted-by":"publisher","first-page":"528","DOI":"10.1038\/ng1779","article-title":"X-linked Cornelia de Lange syndrome owing to SMC1L1 mutations","volume":"38","author":"Musio","year":"2006","journal-title":"Nat. Genet."},{"key":"2023072603543612500_bib43","doi-asserted-by":"publisher","first-page":"1841","DOI":"10.1002\/ajmg.a.34014","article-title":"Investigation of autistic features among individuals with mild to moderate Cornelia de Lange syndrome","volume":"158A","author":"Nakanishi","year":"2012","journal-title":"Am. J. Med. Genet. A."},{"key":"2023072603543612500_bib44","doi-asserted-by":"publisher","first-page":"589","DOI":"10.1111\/gtc.12058","article-title":"DROMPA: Easy-to-handle peak calling and visualization software for the computational analysis and validation of ChIP-seq data","volume":"18","author":"Nakato","year":"2013","journal-title":"Genes Cells."},{"key":"2023072603543612500_bib45","doi-asserted-by":"publisher","first-page":"1170","DOI":"10.1038\/ncb2349","article-title":"Cohesin: a catenase with separate entry and exit gates?","volume":"13","author":"Nasmyth","year":"2011","journal-title":"Nat. Cell Biol."},{"key":"2023072603543612500_bib46","doi-asserted-by":"publisher","first-page":"595","DOI":"10.1146\/annurev.biochem.74.082803.133219","article-title":"The structure and function of SMC and kleisin complexes","volume":"74","author":"Nasmyth","year":"2005","journal-title":"Annu. Rev. Biochem."},{"key":"2023072603543612500_bib47","doi-asserted-by":"publisher","first-page":"63","DOI":"10.1016\/0091-3057(91)90590-X","article-title":"Evaluation of marble-burying behavior as a model of anxiety","volume":"38","author":"Njung\u2019e","year":"1991","journal-title":"Pharmacol. Biochem. Behav."},{"key":"2023072603543612500_bib48","doi-asserted-by":"publisher","first-page":"27","DOI":"10.1016\/j.neuron.2014.11.011","article-title":"Genomic perspectives of transcriptional regulation in forebrain development","volume":"85","author":"Nord","year":"2015","journal-title":"Neuron."},{"key":"2023072603543612500_bib49","doi-asserted-by":"publisher","first-page":"12","DOI":"10.1186\/1756-6606-6-12","article-title":"Synaptosomal-associated protein 25 mutation induces immaturity of the dentate granule cells of adult mice","volume":"6","author":"Ohira","year":"2013","journal-title":"Mol. Brain."},{"key":"2023072603543612500_bib50","doi-asserted-by":"publisher","first-page":"239","DOI":"10.1016\/j.devcel.2007.12.009","article-title":"Cell-type-specific TEV protease cleavage reveals cohesin functions in Drosophila neurons","volume":"14","author":"Pauli","year":"2008","journal-title":"Dev. Cell."},{"key":"2023072603543612500_bib51","volume-title":"The Mouse Brain in Stereotaxic Coordinates.","author":"Paxinos","year":"1997"},{"key":"2023072603543612500_bib52","first-page":"895","article-title":"The growth of non-pyramidal neurons in the primary motor cortex of man: a Golgi study","volume":"12","author":"Prinz","year":"1997","journal-title":"Histol. Histopathol."},{"key":"2023072603543612500_bib53","doi-asserted-by":"publisher","first-page":"W307","DOI":"10.1093\/nar\/gkr378","article-title":"g:Profiler--a web server for functional interpretation of gene lists (2011 update)","volume":"39","author":"Reimand","year":"2011","journal-title":"Nucleic Acids Res."},{"key":"2023072603543612500_bib54","doi-asserted-by":"publisher","first-page":"1155","DOI":"10.1007\/s10803-009-0730-7","article-title":"Social anxiety in Cornelia de Lange syndrome","volume":"39","author":"Richards","year":"2009","journal-title":"J. Autism Dev. Disord."},{"key":"2023072603543612500_bib55","doi-asserted-by":"crossref","first-page":"577","DOI":"10.1093\/genetics\/152.2.577","article-title":"Nipped-B, a Drosophila homologue of chromosomal adherins, participates in activation by remote enhancers in the cut and Ultrabithorax genes","volume":"152","author":"Rollins","year":"1999","journal-title":"Genetics."},{"key":"2023072603543612500_bib56","doi-asserted-by":"publisher","first-page":"289","DOI":"10.1007\/BF00687814","article-title":"Adult fragile X syndrome. Clinico-neuropathologic findings","volume":"67","author":"Rudelli","year":"1985","journal-title":"Acta Neuropathol."},{"key":"2023072603543612500_bib57","doi-asserted-by":"publisher","first-page":"227","DOI":"10.1016\/j.devcel.2007.11.001","article-title":"piggyBac-based mosaic screen identifies a postmitotic function for cohesin in regulating developmental axon pruning","volume":"14","author":"Schuldiner","year":"2008","journal-title":"Dev. Cell."},{"key":"2023072603543612500_bib58","doi-asserted-by":"publisher","first-page":"272","DOI":"10.1038\/nrg3682","article-title":"Transcriptional enhancers: From properties to genome-wide predictions","volume":"15","author":"Shlyueva","year":"2014","journal-title":"Nat. Rev. Genet."},{"key":"2023072603543612500_bib59","doi-asserted-by":"publisher","first-page":"3300","DOI":"10.3791\/3300","article-title":"T-maze forced alternation and left-right discrimination tasks for assessing working and reference memory in mice","author":"Shoji","year":"2012","journal-title":"J. Vis. Exp."},{"key":"2023072603543612500_bib60","doi-asserted-by":"publisher","first-page":"50871","DOI":"10.3791\/50871","article-title":"Contextual and cued fear conditioning test using a video analyzing system in mice","author":"Shoji","year":"2014","journal-title":"J. Vis. Exp."},{"key":"2023072603543612500_bib61","first-page":"387","article-title":"Dendritic organization in the neurons of the visual and motor cortices of the cat","volume":"87","author":"Sholl","year":"1953","journal-title":"J. Anat."},{"key":"2023072603543612500_bib62","doi-asserted-by":"publisher","first-page":"272","DOI":"10.1038\/nature13714","article-title":"Regulation of RNA polymerase II activation by histone acetylation in single living cells","volume":"516","author":"Stasevich","year":"2014","journal-title":"Nature."},{"key":"2023072603543612500_bib63","doi-asserted-by":"publisher","first-page":"104","DOI":"10.3791\/104","article-title":"Light\/dark transition test for mice","author":"Takao","year":"2006","journal-title":"J. Vis. Exp."},{"key":"2023072603543612500_bib64","doi-asserted-by":"publisher","first-page":"11","DOI":"10.1186\/1756-6606-1-11","article-title":"Impaired long-term memory retention and working memory in sdy mutant mice with a deletion in Dtnbp1, a susceptibility gene for schizophrenia","volume":"1","author":"Takao","year":"2008","journal-title":"Mol. Brain."},{"key":"2023072603543612500_bib65","doi-asserted-by":"publisher","first-page":"131","DOI":"10.1016\/S0387-7604(89)80082-8","article-title":"Dendrites, dementia and the Down syndrome","volume":"11","author":"Takashima","year":"1989","journal-title":"Brain Dev."},{"key":"2023072603543612500_bib66","doi-asserted-by":"publisher","first-page":"343","DOI":"10.1016\/j.tins.2008.03.005","article-title":"Chromatin and nuclear architecture in the nervous system","volume":"31","author":"Takizawa","year":"2008","journal-title":"Trends Neurosci."},{"key":"2023072603543612500_bib67","doi-asserted-by":"publisher","first-page":"19","DOI":"10.1186\/1756-6606-2-19","article-title":"Abnormal social behavior, hyperactivity, impaired remote spatial memory, and increased D1-mediated dopaminergic signaling in neuronal nitric oxide synthase knockout mice","volume":"2","author":"Tanda","year":"2009","journal-title":"Mol. Brain."},{"key":"2023072603543612500_bib68","doi-asserted-by":"publisher","first-page":"361","DOI":"10.1007\/s00213-009-1466-y","article-title":"Marble burying reflects a repetitive and perseverative behavior more than novelty-induced anxiety","volume":"204","author":"Thomas","year":"2009","journal-title":"Psychopharmacology (Berl.)."},{"key":"2023072603543612500_bib69","doi-asserted-by":"publisher","first-page":"636","DOI":"10.1038\/ng1363","article-title":"NIPBL, encoding a homolog of fungal Scc2-type sister chromatid cohesion proteins and fly Nipped-B, is mutated in Cornelia de Lange syndrome","volume":"36","author":"Tonkin","year":"2004","journal-title":"Nat. Genet."},{"key":"2023072603543612500_bib70","doi-asserted-by":"publisher","first-page":"46","DOI":"10.1038\/nbt.2450","article-title":"Differential analysis of gene regulation at transcript resolution with RNA-seq","volume":"31","author":"Trapnell","year":"2013","journal-title":"Nat. Biotechnol."},{"key":"2023072603543612500_bib71","doi-asserted-by":"publisher","first-page":"660","DOI":"10.1038\/nn1689","article-title":"Gene expression changes and molecular pathways mediating activity-dependent plasticity in visual cortex","volume":"9","author":"Tropea","year":"2006","journal-title":"Nat. Neurosci."},{"key":"2023072603543612500_bib72","doi-asserted-by":"publisher","first-page":"4","DOI":"10.3389\/neuro.08.004.2008","article-title":"Mice lacking the kf-1 gene exhibit increased anxiety- but not despair-like behavior","volume":"2","author":"Tsujimura","year":"2008","journal-title":"Front. Behav. Neurosci."},{"key":"2023072603543612500_bib73","doi-asserted-by":"publisher","first-page":"1056","DOI":"10.1016\/S1474-4422(09)70262-5","article-title":"Epigenetic mechanisms in neurological diseases: Genes, syndromes, and therapies","volume":"8","author":"Urdinguio","year":"2009","journal-title":"Lancet Neurol."},{"key":"2023072603543612500_bib74","doi-asserted-by":"publisher","first-page":"796","DOI":"10.1038\/nature06634","article-title":"Cohesin mediates transcriptional insulation by CCCTC-binding factor","volume":"451","author":"Wendt","year":"2008","journal-title":"Nature."},{"key":"2023072603543612500_bib75","doi-asserted-by":"publisher","first-page":"e17317","DOI":"10.1371\/journal.pone.0017317","article-title":"Synaptic E3 ligase SCRAPPER in contextual fear conditioning: extensive behavioral phenotyping of Scrapper heterozygote and overexpressing mutant mice","volume":"6","author":"Yao","year":"2011","journal-title":"PLoS One."}],"container-title":["Journal of Experimental Medicine"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/rupress.org\/jem\/article-pdf\/214\/5\/1431\/1757693\/jem_20161517.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/rupress.org\/jem\/article-pdf\/214\/5\/1431\/1757693\/jem_20161517.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,7,26]],"date-time":"2023-07-26T03:59:00Z","timestamp":1690343940000},"score":1,"resource":{"primary":{"URL":"https:\/\/rupress.org\/jem\/article\/214\/5\/1431\/163\/Decreased-cohesin-in-the-brain-leads-to-defective"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2017,4,13]]},"references-count":75,"journal-issue":{"issue":"5","published-print":{"date-parts":[[2017,5,1]]}},"URL":"https:\/\/doi.org\/10.1084\/jem.20161517","relation":{},"ISSN":["0022-1007","1540-9538"],"issn-type":[{"value":"0022-1007","type":"print"},{"value":"1540-9538","type":"electronic"}],"subject":[],"published":{"date-parts":[[2017,4,13]]}}}