{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,30]],"date-time":"2026-06-30T12:53:27Z","timestamp":1782824007500,"version":"3.54.5"},"reference-count":13,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2021,9,30]],"date-time":"2021-09-30T00:00:00Z","timestamp":1632960000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Neuropathology"],"published-print":{"date-parts":[[2021,12]]},"abstract":"<jats:p>Progressive multifocal leukoencephalopathy (PML) is a fatal disease caused by John Cunningham virus (JCV) infection; however, a growing number of PML patients now survive longer and achieve remission, largely due to the advent of combination antiretroviral therapy. Several reports have suggested that the pathology in such patients presents only chronic demyelination without characteristic cellular changes, being referred to as \u201cburnt\u2010out\u201d PML. On the other hand, our knowledge of \u201cburnt\u2010out\u201d PML is still substantially limited, especially in patients with non\u2010human immunodeficiency virus infection. Here, we report a case of PML associated with idiopathic CD4<jats:sup>+<\/jats:sup> lymphocytopenia (ICL) who presented with spontaneous remission and survived for 11\u2009years after onset. Notably, postmortem examination revealed surprisingly broad \u201cburnt\u2010out\u201d lesions lacking the classic histopathological findings. However, pathogenic JCV\u2010specific DNA sequences was still present in the autopsied brain tissue. This case suggests that complete remission can be achieved with a persistent presence of JCV\u2010specific pathogenic sequences, even after a catastrophic infection. Considering that there have been a few reported cases of PML with ICL with long survival, the long\u2010term survival of our case may share a favorable immunological response that is unique to a subgroup of ICL.<\/jats:p>","DOI":"10.1111\/neup.12773","type":"journal-article","created":{"date-parts":[[2021,10,1]],"date-time":"2021-10-01T05:24:57Z","timestamp":1633065897000},"page":"484-488","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":6,"title":["\u201cBurnt\u2010out\u201d progressive multifocal leukoencephalopathy in idiopathic <scp>CD4<\/scp><sup>+<\/sup> lymphocytopenia"],"prefix":"10.1111","volume":"41","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-3384-6881","authenticated-orcid":false,"given":"Tatsuya","family":"Fukumoto","sequence":"first","affiliation":[{"name":"Department of Neurology Tokushima University Graduate School of Biomedical Sciences  Tokushima Japan"},{"name":"Department of Neurology Kameda Medical 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