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Preliminary evidence suggests that the corpus callosum may be dysmorphic in orofacial clefting; however, this midline brain structure has not been systematically assessed in this population. The goal of the present study was to carry out a morphometric assessment of the corpus callosum and its relationship to cognitive performance in a well\u2010characterized patient cohort with orofacial cleft. Midline brain images were obtained from previously collected MRI scans of 24 CL\/P subjects and 40\u2010adult\u2010male controls. Eight landmarks on the corpus callosum were digitized on each image and their <jats:italic>x<\/jats:italic>,<jats:italic>y<\/jats:italic> coordinate locations saved. A geometric morphometrics analysis was applied to the landmark coordinate data to test for shape differences across groups. The relationship between corpus callosum shape and IQ was explored with nonparametric correlation coefficients. Results revealed significant differences in mean corpus callosum shape between CL\/P cases and controls (<jats:italic>P<\/jats:italic>\u2009=\u20090.029). The CL\/P corpus callosum was characterized by increased overall convexity resulting from a superior and posterior displacement. Within CL\/P cases, increased corpus callosum shape dysmorphology was moderately correlated with reduced performance IQ (r\u2009=\u20090.546). These results provide additional evidence that midline brain changes may be an important part of the orofacial cleft phenotype. \u00a9 2013 Wiley Periodicals, Inc.<\/jats:p><\/jats:sec>","DOI":"10.1002\/ajmg.a.35835","type":"journal-article","created":{"date-parts":[[2013,3,27]],"date-time":"2013-03-27T04:09:50Z","timestamp":1364357390000},"page":"1002-1007","source":"Crossref","is-referenced-by-count":16,"title":["Corpus Callosum Shape Is Altered in Individuals With Nonsyndromic Cleft Lip and Palate"],"prefix":"10.1002","volume":"161","author":[{"given":"Seth M.","family":"Weinberg","sequence":"first","affiliation":[{"name":"Center for Craniofacial and Dental Genetics, Department of Oral Biology School of Dental Medicine, University of Pittsburgh Pittsburgh, Pennsylvania"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Trish E.","family":"Parsons","sequence":"additional","affiliation":[{"name":"Center for Craniofacial and Dental Genetics, Department of Oral Biology 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