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With the aim of better defining the long-term outcomes of this group we have performed a retrospective cohort comparison of atypical demyelination versus \u2018typical\u2019 MS presentations. Twenty-seven cases with atypical presentations (both first and subsequent demyelinating events) were identified and compared with typical MS cases. Disease features analysed included relapse rates, disability severity, whole brain and lesion volumes, lesion number and distribution. Atypical cases represented 3.9% of all MS cases. There was considerable overlap in the magnetic resonance imaging (MRI) features of ADEM-like and tumefactive demyelination cases. ADEM-like cases tended to be younger but not significantly so. Atypical cases showed a trend towards higher peak expanded disability severity score (EDSS) score at the time of their atypical presentation. Motor, cranial nerve, cerebellar, cerebral and multifocal presentations were all more common in atypical cases, and less likely to present with optic neuritis. Cerebrospinal fluid (CSF) white cell counts were higher in atypical cases (<jats:italic>p<\/jats:italic>\u2009=\u20090.002). One atypical case was associated with peripheral blood myelin oligodendrocyte glycoprotein (MOG) antibodies, but subsequent clinical and radiological course was in keeping with MS. There was no difference in long-term clinical outcomes including annualised relapse rates (ARR), brain volume, lesion numbers or lesion distributions. Atypical demyelination cases were more likely to receive high potency disease modifying therapy early in the course of their illness. Despite the severity of initial illness, our cohort analysis suggests that atypical demyelination presentations do not confer a higher risk of long-term adverse outcomes.<\/jats:p>","DOI":"10.1007\/s00415-024-12349-6","type":"journal-article","created":{"date-parts":[[2024,6,11]],"date-time":"2024-06-11T12:12:20Z","timestamp":1718107940000},"page":"5275-5289","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Long-term outcomes of ADEM-like and tumefactive presentations of CNS demyelination: a case-comparison analysis"],"prefix":"10.1007","volume":"271","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-4582-1171","authenticated-orcid":false,"given":"Simon V.","family":"Arnett","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kerri","family":"Prain","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0294-9768","authenticated-orcid":false,"given":"Sudarshini","family":"Ramanathan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7292-9658","authenticated-orcid":false,"given":"Sandeep","family":"Bhuta","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7025-2276","authenticated-orcid":false,"given":"Fabienne","family":"Brilot","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9429-4307","authenticated-orcid":false,"given":"Simon A.","family":"Broadley","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2024,6,11]]},"reference":[{"key":"12349_CR1","doi-asserted-by":"publisher","first-page":"687","DOI":"10.1155\/2013\/484959","volume":"35","author":"A Gajofatto","year":"2013","unstructured":"Gajofatto A, Calabrese M, Benedetti MD, Monaco S (2013) Clinical, MRI, and CSF markers of disability progression in multiple sclerosis. 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Dr Sudarshini Ramanathan has received research funding from the National Health and Medical Research Council (Australia), the Brain Foundation (Australia), the Royal Australasian College of Physicians, the Petre Foundation, and the University of Sydney. She is supported by an NHMRC Investigator Grant (GNT2008339). She serves as a consultant on an advisory board for UCB and Limbic Neurology, and has been an invited speaker for Biogen, Excemed, and Limbic Neurology. SVA has accept financial support to attend conferences supported by Biogen. KP, SR, SB, FB report no relevant financial disclosures.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Conflicts of interest"}},{"value":"Ethics approval was sought and obtained through the Griffith University and Gold Coast Hospital and Health Service, Human Research Ethics Committees. Written informed consent was provided by all participants.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval"}}]}}