{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,30]],"date-time":"2026-04-30T07:24:05Z","timestamp":1777533845853,"version":"3.51.4"},"reference-count":48,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2017,12,19]],"date-time":"2017-12-19T00:00:00Z","timestamp":1513641600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100010702","name":"Azusa Pacific University","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100010702","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Brain and Behavior"],"published-print":{"date-parts":[[2018,1]]},"abstract":"Abstract<\/jats:title>Introduction<\/jats:title>Williams\u2013Beuren syndrome (WBS<\/jats:styled-content>) is a developmental disorder caused by hemizygous deletion of human chromosome 7q11.23. Hypersocial behavior is one symptom of WBS<\/jats:styled-content> and contrasts with hyposociality observed in autism spectrum disorder (ASD<\/jats:styled-content>). Interestingly, duplications of 7q11.23 have been associated with ASD<\/jats:styled-content>. The social phenotype of WBS<\/jats:styled-content> has been linked to GTF<\/jats:styled-content>2I<\/jats:italic> or general transcription factor II<\/jats:styled-content>i (TFII<\/jats:styled-content>\u2010I<\/jats:italic>).\u00a0Duplication of GTF<\/jats:styled-content>2I<\/jats:italic> has also been associated with ASD<\/jats:styled-content>.<\/jats:p><\/jats:sec>Methods<\/jats:title>We compared mice having either a deletion (Gtf2i<\/jats:italic>+\/\u2212<\/jats:italic><\/jats:sup>) or duplication (Gtf2i<\/jats:italic>+\/dup<\/jats:italic><\/jats:sup>) of Gtf2i<\/jats:italic> to wild\u2010type (Gtf2i<\/jats:italic>+\/+<\/jats:italic><\/jats:sup>) littermate controls in a series of behavioral tasks including open\u2010field activity monitoring, olfactory probes, a social choice task, social transmission of food preference, habituation\u2013dishabituation, and operant social motivation paradigms.<\/jats:p><\/jats:sec>Results<\/jats:title>In open\u2010field observations, Gtf2i<\/jats:italic>+\/\u2212<\/jats:italic><\/jats:sup> and Gtf2i<\/jats:italic>+\/dup<\/jats:italic><\/jats:sup> mice demonstrated normal activity and thigmotaxis, and surprisingly, each strain showed a significant preference for a stimulus mouse that was not observed in Gtf2i<\/jats:italic>+\/+<\/jats:italic><\/jats:sup> siblings. Both Gtf2i<\/jats:italic>+\/\u2212<\/jats:italic><\/jats:sup> and Gtf2i<\/jats:italic>+\/dup<\/jats:italic><\/jats:sup> mice demonstrated normal olfaction in buried food probes, but the Gtf2i<\/jats:italic>+\/\u2212<\/jats:italic><\/jats:sup> mice spent significantly more time investigating urine scent versus water, which was not observed in the other strains. Gtf2i<\/jats:italic>+\/\u2212<\/jats:italic><\/jats:sup> mice also spent significantly more time in nose\u2010to\u2010nose contact compared to Gtf2i<\/jats:italic>+\/+<\/jats:italic><\/jats:sup> siblings during the open\u2010field encounter of the social transmission of food preference task. In operant tasks of social motivation, Gtf2i<\/jats:italic>+\/\u2212<\/jats:italic><\/jats:sup> mice made significantly more presses for social rewards than Gtf2i<\/jats:italic>+\/+<\/jats:italic><\/jats:sup> siblings, while there was no difference in presses for the Gtf2i<\/jats:italic>+\/dup<\/jats:italic><\/jats:sup> mice.<\/jats:p><\/jats:sec>Discussion<\/jats:title>Results were remarkably consistent across testing paradigms supporting a role for GTF<\/jats:styled-content>2i<\/jats:italic> in the hypersocial phenotype of WBS<\/jats:styled-content> and more broadly in the regulation of social behavior. Support was not observed for the role of GTF<\/jats:styled-content>2i<\/jats:italic> in ASD<\/jats:styled-content>.<\/jats:p><\/jats:sec>","DOI":"10.1002\/brb3.895","type":"journal-article","created":{"date-parts":[[2017,12,19]],"date-time":"2017-12-19T07:56:42Z","timestamp":1513670202000},"source":"Crossref","is-referenced-by-count":17,"title":["Consistent hypersocial behavior in mice carrying a deletion of Gtf2i<\/i> but no evidence of hyposocial behavior with Gtf2i<\/i> duplication: Implications for Williams\u2013Beuren syndrome and autism spectrum disorder"],"prefix":"10.1002","volume":"8","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-7512-4134","authenticated-orcid":false,"given":"Loren A.","family":"Martin","sequence":"first","affiliation":[{"name":"Department of Graduate Psychology Azusa Pacific University Azusa CA USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Erica","family":"Iceberg","sequence":"additional","affiliation":[{"name":"Department of Graduate Psychology Azusa Pacific University Azusa CA USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Gabriel","family":"Allaf","sequence":"additional","affiliation":[{"name":"Department of Biology and Chemistry Azusa Pacific University Azusa CA USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2017,12,19]]},"reference":[{"key":"e_1_2_6_2_1","doi-asserted-by":"publisher","DOI":"10.1176\/appi.books.9780890425596"},{"key":"e_1_2_6_3_1","doi-asserted-by":"publisher","DOI":"10.1038\/nn.4319"},{"key":"e_1_2_6_4_1","doi-asserted-by":"publisher","DOI":"10.1086\/376565"},{"key":"e_1_2_6_5_1","doi-asserted-by":"publisher","DOI":"10.1097\/00001756-199906030-00006"},{"key":"e_1_2_6_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1566-2772(01)00008-1"},{"key":"e_1_2_6_7_1","doi-asserted-by":"publisher","DOI":"10.1097\/GIM.0b013e3180986192"},{"key":"e_1_2_6_8_1","doi-asserted-by":"publisher","DOI":"10.1161\/01.CIR.26.6.1235"},{"key":"e_1_2_6_9_1","doi-asserted-by":"publisher","DOI":"10.1038\/mt.2015.130"},{"key":"e_1_2_6_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.tics.2012.02.007"},{"key":"e_1_2_6_11_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0167024"},{"key":"e_1_2_6_12_1","doi-asserted-by":"publisher","DOI":"10.1097\/DBP.0000000000000235"},{"key":"e_1_2_6_13_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12868-014-0127-1"},{"key":"e_1_2_6_14_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.32652"},{"key":"e_1_2_6_15_1","doi-asserted-by":"publisher","DOI":"10.1007\/BF00518192"},{"key":"e_1_2_6_16_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.2006.047092"},{"key":"e_1_2_6_17_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.20416"},{"key":"e_1_2_6_18_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10803-016-2974-3"},{"key":"e_1_2_6_19_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng0993-11"},{"key":"e_1_2_6_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuropsychologia.2005.05.008"},{"key":"e_1_2_6_21_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cell.2014.05.017"},{"key":"e_1_2_6_22_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.conb.2012.12.006"},{"key":"e_1_2_6_23_1","doi-asserted-by":"publisher","DOI":"10.1017\/S0954579408000011"},{"key":"e_1_2_6_24_1","doi-asserted-by":"publisher","DOI":"10.1162\/089892900561968"},{"key":"e_1_2_6_25_1","doi-asserted-by":"publisher","DOI":"10.1002\/emmm.200900003"},{"key":"e_1_2_6_26_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10803-011-1389-4"},{"key":"e_1_2_6_27_1","first-page":"e53009","article-title":"Quantifying social motivation in mice using operant conditioning","volume":"102","author":"Martin L.","year":"2015","journal-title":"Journal of Visualized Experiments"},{"key":"e_1_2_6_28_1","doi-asserted-by":"publisher","DOI":"10.1002\/brb3.273"},{"key":"e_1_2_6_29_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2012.04.012"},{"key":"e_1_2_6_30_1","doi-asserted-by":"publisher","DOI":"10.1207\/S15326942DN1701_07"},{"key":"e_1_2_6_31_1","doi-asserted-by":"publisher","DOI":"10.1006\/brcg.2000.1232"},{"key":"e_1_2_6_32_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1601-1848.2004.00076.x"},{"key":"e_1_2_6_33_1","doi-asserted-by":"publisher","DOI":"10.3389\/fpsyg.2016.00666"},{"key":"e_1_2_6_34_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMra0903074"},{"key":"e_1_2_6_35_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41598-017-03615-x"},{"key":"e_1_2_6_36_1","doi-asserted-by":"publisher","DOI":"10.1002\/eji.200839031"},{"key":"e_1_2_6_37_1","doi-asserted-by":"publisher","DOI":"10.1002\/aur.169"},{"key":"e_1_2_6_38_1","doi-asserted-by":"publisher","DOI":"10.1177\/1362361312455704"},{"key":"e_1_2_6_39_1","unstructured":"Science A.I.f.B. 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