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Both mouse strains display distinct quantitative alterations in fluid consumption compared to their wild\u2010type littermates; several of these changes are diametrically opposing between the two chromosome engineered mouse models. Mice with duplication versus deletion showed longer versus shorter intervals between visits to the waterspout, generated more versus less licks per visit and had higher versus lower variability in the number of licks per lick\u2010burst as compared to their respective wild\u2010type littermates. These findings suggest that copy number variation can affect long\u2010term fluid consumption behavior in mice. Other behavioral differences were unique for either the duplication or deletion mutants; the deletion CNV resulted in increased variability of the licking rhythm, and the duplication CNV resulted in a significant slowing of the licking rhythm. Our findings document a readily quantitated complex behavioral response that can be directly and reciprocally influenced by a gene dosage effect. \u00a9 2012 Wiley Periodicals, Inc.<\/jats:p>","DOI":"10.1002\/ajmg.a.35601","type":"journal-article","created":{"date-parts":[[2012,9,18]],"date-time":"2012-09-18T16:13:13Z","timestamp":1347984793000},"page":"2807-2814","source":"Crossref","is-referenced-by-count":12,"title":["Opposing phenotypes in mice with Smith\u2013Magenis deletion and Potocki\u2013Lupski duplication syndromes suggest gene dosage effects on fluid consumption behavior"],"prefix":"10.1002","volume":"158A","author":[{"given":"Detlef H.","family":"Heck","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wenli","family":"Gu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ying","family":"Cao","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shuhua","family":"Qi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Melanie","family":"Lacaria","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"James R.","family":"Lupski","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2012,9,18]]},"reference":[{"key":"e_1_2_7_2_1","volume-title":"Inborn errors of development","author":"Bi W","year":"2008"},{"key":"e_1_2_7_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuroimage.2003.10.040"},{"key":"e_1_2_7_4_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1601-183X.2006.00293.x"},{"key":"e_1_2_7_5_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng.279"},{"key":"e_1_2_7_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1460-9568.2010.07244.x"},{"key":"e_1_2_7_7_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature07239"},{"key":"e_1_2_7_8_1","doi-asserted-by":"publisher","DOI":"10.1007\/s002130050327"},{"key":"e_1_2_7_9_1","doi-asserted-by":"publisher","DOI":"10.1093\/brain\/123.11.2203"},{"key":"e_1_2_7_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0165-5876(01)00475-X"},{"key":"e_1_2_7_11_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1399-0004.2007.00815.x"},{"key":"e_1_2_7_12_1","doi-asserted-by":"publisher","DOI":"10.1038\/sj.ejhg.5202009"},{"key":"e_1_2_7_13_1","article-title":"Searching for Potocki\u2013Lupski syndrome phenotype: A patient with language impairment and no autism","author":"Ercan\u2010Sencicek A","year":"2011","journal-title":"Brain and Development"},{"key":"e_1_2_7_14_1","doi-asserted-by":"publisher","DOI":"10.1007\/s12311-012-0355-9"},{"key":"e_1_2_7_15_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1399-0004.2007.00959.x"},{"key":"e_1_2_7_16_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-8628(19960329)62:3<247::AID-AJMG9>3.0.CO;2-Q"},{"key":"e_1_2_7_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.pediatrneurol.2005.08.018"},{"key":"e_1_2_7_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jneumeth.2005.10.023"},{"key":"e_1_2_7_19_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddn117"},{"key":"e_1_2_7_20_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1114042108"},{"key":"e_1_2_7_21_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pgen.1002713"},{"key":"e_1_2_7_22_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/dds124"},{"key":"e_1_2_7_23_1","doi-asserted-by":"publisher","DOI":"10.1017\/S1462399410001390"},{"key":"e_1_2_7_24_1","doi-asserted-by":"publisher","DOI":"10.1038\/455178a"},{"key":"e_1_2_7_25_1","unstructured":"LupskiJR.2011.Insights for autism from Charcot\u2010Marie\u2010Tooth disease: Simons foundation autism research initiative. 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