{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,11]],"date-time":"2026-02-11T18:00:25Z","timestamp":1770832825745,"version":"3.50.1"},"reference-count":29,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2014,3,25]],"date-time":"2014-03-25T00:00:00Z","timestamp":1395705600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Neuropathology"],"published-print":{"date-parts":[[2014,8]]},"abstract":"<jats:p>We report clinicopathological features of a 23\u2010year\u2010old woman with <jats:styled-content style=\"fixed-case\">D<\/jats:styled-content>own syndrome (<jats:styled-content style=\"fixed-case\">DS<\/jats:styled-content>) presenting with subacute myelopathy treated with chemotherapy, including intravenous and intrathecal administration of methotrexate (<jats:styled-content style=\"fixed-case\">MTX<\/jats:styled-content>), and with allogenic bone\u2010marrow transplantation for <jats:styled-content style=\"fixed-case\">B<\/jats:styled-content> lymphoblastic leukemia. Autopsy revealed severe demyelinating vacuolar myelopathy in the posterior and lateral columns of the spinal cord, associated with macrophage infiltration, marked axonal loss and some swollen axons. Pathological changes of posterior and lateral columns were observed from the medulla oblongata to lumbar cord. Proximal anterior and posterior roots were preserved. Cerebral white matter was relatively well preserved. There were no vascular lesions or meningeal dissemination of leukemia. Longitudinal extension of cord lesions was extensive, unlike typical cases of subacute combined degeneration (<jats:styled-content style=\"fixed-case\">SACD<\/jats:styled-content>), but distribution of lesions and histological findings were similar to that of <jats:styled-content style=\"fixed-case\">SACD<\/jats:styled-content>. <jats:styled-content style=\"fixed-case\">DS<\/jats:styled-content> patients show heightened sensitivity to <jats:styled-content style=\"fixed-case\">MTX<\/jats:styled-content> because of their genetic background. Risk factors for toxic myelopathy of <jats:styled-content style=\"fixed-case\">DS<\/jats:styled-content> are discussed, including delayed clearance of <jats:styled-content style=\"fixed-case\">MTX<\/jats:styled-content> despite normal renal function, alterations in <jats:styled-content style=\"fixed-case\">MTX<\/jats:styled-content> polyglutamation and enhanced folic acid depletion due to gene dosage effects of chromosome 21. Alteration of folate metabolism and\/or vitamin <jats:styled-content style=\"fixed-case\">B<\/jats:styled-content><jats:sub>12<\/jats:sub> levels through intravenous or intrathecal administration of <jats:styled-content style=\"fixed-case\">MTX<\/jats:styled-content> might exist, although vitamin <jats:styled-content style=\"fixed-case\">B<\/jats:styled-content><jats:sub>12<\/jats:sub> and other essential nutrients were managed using intravenous hyperalimentation. To the best of our knowledge, this is the first report of an autopsy case that shows myelopathy mimicking <jats:styled-content style=\"fixed-case\">SACD<\/jats:styled-content> in a <jats:styled-content style=\"fixed-case\">DS<\/jats:styled-content> patient accompanied by <jats:styled-content style=\"fixed-case\">B<\/jats:styled-content> lymphoblastic leukemia. The case suggests a pathophysiological mechanism of <jats:styled-content style=\"fixed-case\">MTX<\/jats:styled-content>\u2010related myelopathy in <jats:styled-content style=\"fixed-case\">DS<\/jats:styled-content> patients with <jats:styled-content style=\"fixed-case\">B<\/jats:styled-content> lymphoblastic leukemia mimicking <jats:styled-content style=\"fixed-case\">SACD<\/jats:styled-content>.<\/jats:p>","DOI":"10.1111\/neup.12114","type":"journal-article","created":{"date-parts":[[2014,3,25]],"date-time":"2014-03-25T06:53:08Z","timestamp":1395730388000},"page":"414-419","source":"Crossref","is-referenced-by-count":5,"title":["Myelopathy mimicking subacute combined degeneration in a <scp>D<\/scp>own syndrome patient with methotrexate treatment for <scp>B<\/scp> lymphoblastic leukemia: Report of an autopsy case"],"prefix":"10.1111","volume":"34","author":[{"given":"Kaishi","family":"Satomi","sequence":"first","affiliation":[{"name":"Department of Pathology Division of Pediatric 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