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While previous studies have examined brain connectivity in AS, the specifics of white matter structural changes have remained unclear. In this study, we utilized advanced diffusion MRI techniques to investigate the microstructural abnormalities of white matter for AS patients. A total of 30 AS patients and 19 age\u2010 and sex\u2010matched healthy controls were included in the study. We used metrics derived from both fixel\u2010based analysis (FBA) and diffusion tensor imaging to compare the white matter microstructure differences between AS patients and healthy controls. The results indicate that patients with AS have white matter microstructural differences throughout the whole brain, particularly in the corticospinal tract, arcuate fasciculate, and corpus callosum. FBA\u2010derived metrics demonstrated greater specificity and sensitivity than tensor\u2010based measures. Subsequently, we extracted six fiber tracts with significant differences from the FBA analysis and conducted tract\u2010based statistics, including parieto\u2010occipital pontine, anterior commissure, arcuate fasciculate, corticospinal tract, splenium of corpus callosum, and isthmus of corpus callosum. In all six fiber tracts, we found that AS patients with a higher frequency of seizures exhibited more white matter alterations. Overall, this study provides new insights into the structural differences in AS and their association with clinical symptoms, highlighting the extensive white matter differences and their potential impact on patient outcomes.<\/jats:p>","DOI":"10.1002\/aur.70160","type":"journal-article","created":{"date-parts":[[2025,12,24]],"date-time":"2025-12-24T07:45:23Z","timestamp":1766562323000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Microstructural White Matter Alterations in Angelman Syndrome: A Fixel\u2010Based Analysis"],"prefix":"10.1002","volume":"19","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-2126-2785","authenticated-orcid":false,"given":"Lei","family":"Wei","sequence":"first","affiliation":[{"name":"Institute of Science and Technology for Brain\u2010Inspired Intelligence Fudan University  Shanghai China"},{"name":"CAS Key Laboratory of Computational Biology, Shanghai Institute of Nutrition and 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