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Neuroanatomical correlates of inter\u2010individual variation in cognitive ability have been described in health, but are not well\u2010characterized in SCA. Here, we modeled relationships between general cognitive ability (estimated using full\u2010scale IQ [FSIQ] from Wechsler scales) and regional estimates of SA and CT (from structural MRI scans) in both aneuploid (28 XXX, 55 XXY, 22 XYY, 19 XXYY) and typically\u2010developing euploid (79 XX, 85 XY) individuals. Results indicated widespread decoupling of normative anatomical\u2013cognitive relationships in SCA: we found five regions where SCA significantly altered SA\u2013FSIQ relationships, and five regions where SCA significantly altered CT\u2013FSIQ relationships. The majority of areas were characterized by the presence of positive anatomy\u2010IQ relationships in health, but no or slightly negative anatomy\u2010IQ relationships in SCA. Disrupted anatomical\u2013cognitive relationships generalized from the full cohort to karyotypically defined subcohorts (i.e., XX\u2010XXX; XY\u2010XYY; XY\u2010XXY), demonstrating continuity across multiple supernumerary SCA conditions. As the first direct evidence of altered regional neuroanatomical\u2013cognitive relationships in supernumerary SCA, our findings shed light on potential genetic and structural correlates of the cognitive phenotype in SCA, and may have implications for other neurogenetic disorders.<\/jats:p>","DOI":"10.1002\/ajmg.c.31795","type":"journal-article","created":{"date-parts":[[2020,6,9]],"date-time":"2020-06-09T06:57:00Z","timestamp":1591685820000},"page":"493-505","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":12,"title":["Sex chromosome aneuploidy alters the relationship between neuroanatomy and cognition"],"prefix":"10.1002","volume":"184","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-9412-6357","authenticated-orcid":false,"given":"Allysa","family":"Warling","sequence":"first","affiliation":[{"name":"Developmental Neurogenomics Unit, Human Genetics Branch National Institute of Mental Health  Bethesda Maryland USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Siyuan","family":"Liu","sequence":"additional","affiliation":[{"name":"Developmental Neurogenomics Unit, Human Genetics Branch National Institute of Mental Health  Bethesda Maryland USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kathleen","family":"Wilson","sequence":"additional","affiliation":[{"name":"Developmental Neurogenomics Unit, Human Genetics Branch National Institute of Mental Health  Bethesda Maryland USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ethan","family":"Whitman","sequence":"additional","affiliation":[{"name":"Developmental Neurogenomics Unit, Human Genetics Branch National Institute of Mental Health  Bethesda Maryland USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Fran\u00e7ois M.","family":"Lalonde","sequence":"additional","affiliation":[{"name":"Developmental Neurogenomics Unit, Human Genetics Branch National Institute of Mental Health  Bethesda Maryland USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Liv S.","family":"Clasen","sequence":"additional","affiliation":[{"name":"Developmental Neurogenomics Unit, Human Genetics Branch National Institute of Mental Health  Bethesda Maryland USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jonathan D.","family":"Blumenthal","sequence":"additional","affiliation":[{"name":"Developmental Neurogenomics Unit, Human Genetics Branch National Institute of Mental Health  Bethesda Maryland USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Armin","family":"Raznahan","sequence":"additional","affiliation":[{"name":"Developmental Neurogenomics Unit, Human Genetics Branch National Institute of Mental Health  Bethesda Maryland USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2020,6,8]]},"reference":[{"key":"e_1_2_8_2_1","unstructured":"Ad\u2010Dab'bagh Y. 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