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We previously showed that mice with conditional ATRX ablation in forebrain excitatory neurons display deficits in fear memory and autism\u2010related behaviors, with some effects exhibiting sexual dimorphism. In this study, we used high\u2010resolution magnetic resonance imaging (MRI) to systematically characterize brain structural changes associated with these behavioral abnormalities. Whole\u2010brain analysis revealed male\u2010specific microcephaly, while subregional analysis identified significant reductions in hippocampal structures and increased volume of the caudal cortex in mutant animals of both sexes. We also identified structural alterations in regions retaining ATRX expression, such as the thalamus, midbrain, cerebellum, and several fiber tracts. These findings suggest that ATRX loss disrupts the coordinated development of interconnected brain regions. Overall, our results implicate impaired cortico\u2010thalamic\u2010cerebellar connectivity as a potential neural substrate underlying the autistic\u2010like behaviors observed in this mouse model, providing new insights into the neurobiological basis of ATR\u2010X syndrome.\n                  <\/jats:p>","DOI":"10.1002\/aur.70205","type":"journal-article","created":{"date-parts":[[2026,2,23]],"date-time":"2026-02-23T02:25:01Z","timestamp":1771813501000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Altered Brain Structure in an\n                    <scp>ATRX<\/scp>\n                    \u2010Deficient Mouse Model of Autism Spectrum Disorder"],"prefix":"10.1002","volume":"19","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0842-6589","authenticated-orcid":false,"given":"Katherine","family":"Quesnel","sequence":"first","affiliation":[{"name":"Department of Anatomy &amp; Cell Biology Western University  London ON Canada"},{"name":"Department of Paediatrics Western University  London ON Canada"},{"name":"Division of Genetics &amp; Development Children's Health Research Institute  London ON Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jacob","family":"Ellegood","sequence":"additional","affiliation":[{"name":"Mouse Imaging Centre The Hospital for Sick Children  Toronto ON Canada"},{"name":"Department of Medical Biophysics The University of Toronto  Toronto ON Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jason P.","family":"Lerch","sequence":"additional","affiliation":[{"name":"Mouse Imaging Centre The Hospital for Sick Children  Toronto ON Canada"},{"name":"Department of Medical Biophysics The University of Toronto  Toronto ON Canada"},{"name":"Nuffield Department of Clinical Neuroscience University of Oxford  Oxford UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1948-8715","authenticated-orcid":false,"given":"Nathalie G.","family":"B\u00e9rub\u00e9","sequence":"additional","affiliation":[{"name":"Department of Anatomy &amp; Cell Biology Western University  London ON Canada"},{"name":"Department of Paediatrics Western University  London ON Canada"},{"name":"Division of Genetics &amp; Development Children's Health Research Institute  London ON Canada"},{"name":"Department of Oncology Western University  London ON Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2026,2,22]]},"reference":[{"key":"e_1_2_10_2_1","doi-asserted-by":"publisher","DOI":"10.3389\/fnins.2022.885335"},{"key":"e_1_2_10_3_1","doi-asserted-by":"publisher","DOI":"10.1093\/ijnp\/pyy043"},{"key":"e_1_2_10_4_1","doi-asserted-by":"publisher","DOI":"10.4103\/0972\u20102327.104323"},{"key":"e_1_2_10_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.media.2007.06.004"},{"key":"e_1_2_10_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuroimage.2010.09.025"},{"key":"e_1_2_10_7_1","doi-asserted-by":"publisher","DOI":"10.1172\/JCI22329"},{"key":"e_1_2_10_8_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/9.4.539"},{"key":"e_1_2_10_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuroimage.2012.01.100"},{"key":"e_1_2_10_10_1","doi-asserted-by":"publisher","DOI":"10.5765\/jkacap.200018"},{"key":"e_1_2_10_11_1","doi-asserted-by":"publisher","DOI":"10.3389\/fnagi.2023.1195748"},{"key":"e_1_2_10_12_1","doi-asserted-by":"publisher","DOI":"10.1097\/00004728\u2010199403000\u201000005"},{"key":"e_1_2_10_13_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2007.10.016"},{"key":"e_1_2_10_14_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuroimage.2016.06.028"},{"key":"e_1_2_10_15_1","volume-title":"StatPearls","author":"Leon A. 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