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The patient, a man in his 40s, exhibited progressive neuropsychiatric symptoms with predominant frontal white matter lesions and corpus callosum atrophy. Neuropathological examination revealed frontal\u2010predominant demyelination with a relatively low number of axonal spheroids compared with typical cases, as well as the absence of intracranial calcifications. In addition, prominent cerebral amyloid angiopathy was observed despite an APOE \u03b53\/\u03b53 genotype. These findings expand the clinicopathological spectrum of\n                    <jats:italic>CSF1R<\/jats:italic>\n                    \u2010related leukoencephalopathy and highlight variability in pathological features associated with different\n                    <jats:italic>CSF1R<\/jats:italic>\n                    mutations.\n                  <\/jats:p>","DOI":"10.1111\/neup.70065","type":"journal-article","created":{"date-parts":[[2026,6,9]],"date-time":"2026-06-09T06:30:15Z","timestamp":1780986615000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["The First Autopsy\u2010Proven Case of\n                    <scp>\n                      <i>CSF1R<\/i>\n                    <\/scp>\n                    \u2010Related Leukoencephalopathy Harboring the p.\n                    <scp>Cys774Arg<\/scp>\n                    Mutation"],"prefix":"10.1111","volume":"46","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-5649-6947","authenticated-orcid":false,"given":"Hiroyuki","family":"Hatsuta","sequence":"first","affiliation":[{"name":"Department of Laboratory Medicine National Center of Neurology and Psychiatry  Kodaira Tokyo Japan"},{"name":"Hatsuta Neurology Clinic  Hirakata Osaka Japan"},{"name":"Department of Neurology Osaka Metropolitan University  Osaka Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4911-9320","authenticated-orcid":false,"given":"Akitoshi","family":"Takeda","sequence":"additional","affiliation":[{"name":"Department of Neurology Osaka Metropolitan University  Osaka Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tomomi","family":"Fujita","sequence":"additional","affiliation":[{"name":"Department of Neurology Osaka Metropolitan University  Osaka 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