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dc.contributor.authorRamanathan, Sudarshini
dc.contributor.authorMohammad, Shekeeb
dc.contributor.authorTantsis, Esther
dc.contributor.authorNguyen, Tina Kim
dc.contributor.authorMerheb, Vera
dc.contributor.authorFung, Victor SC
dc.contributor.authorWhite, Owen Bruce
dc.contributor.authorBroadley, Simon
dc.contributor.authorLechner-Scott, Jeannette
dc.contributor.authorVucic, Steve
dc.contributor.authorHenderson, Andrew PD
dc.contributor.authorBarnett, Michael Harry
dc.contributor.authorReddel, Stephen W
dc.contributor.authorBrilot, Fabienne
dc.contributor.authorDale, Russell C
dc.date.accessioned2019-07-05T12:32:43Z
dc.date.available2019-07-05T12:32:43Z
dc.date.issued2018
dc.identifier.issn0022-3050
dc.identifier.doi10.1136/jnnp-2017-316880
dc.identifier.urihttp://hdl.handle.net/10072/377228
dc.description.abstractObjective: We characterised the clinical course, treatment and outcomes in 59 patients with relapsing myelin oligodendrocyte glycoprotein (MOG) antibody-associated demyelination. Methods: We evaluated clinical phenotypes, annualised relapse rates (ARR) prior and on immunotherapy and Expanded Disability Status Scale (EDSS), in 218 demyelinating episodes from 33 paediatric and 26 adult patients. Results: The most common initial presentation in the cohort was optic neuritis (ON) in 54% (bilateral (BON) 32%, unilateral (UON) 22%), followed by acute disseminated encephalomyelitis (ADEM) (20%), which occurred exclusively in children. ON was the dominant phenotype (UON 35%, BON 19%) of all clinical episodes. 109/226 (48%) MRIs had no brain lesions. Patients were steroid responsive, but 70% of episodes treated with oral prednisone relapsed, particularly at doses <10 mg daily or within 2 months of cessation. Immunotherapy, including maintenance prednisone (P=0.0004), intravenous immunoglobulin, rituximab and mycophenolate, all reduced median ARRs on-treatment. Treatment failure rates were lower in patients on maintenance steroids (5%) compared with non-steroidal maintenance immunotherapy (38%) (P=0.016). 58% of patients experienced residual disability (average follow-up 61 months, visual loss in 24%). Patients with ON were less likely to have sustained disability defined by a final EDSS of ≥2 (OR 0.15, P=0.032), while those who had any myelitis were more likely to have sustained residual deficits (OR 3.56, P=0.077). Conclusion:Relapsing MOG antibody-associated demyelination is strongly associated with ON across all age groups and ADEM in children. Patients are highly responsive to steroids, but vulnerable to relapse on steroid reduction and cessation.
dc.description.peerreviewedYes
dc.languageEnglish
dc.language.isoeng
dc.publisherBMJ Group
dc.publisher.placeUnited Kingdom
dc.relation.ispartofpagefrom127
dc.relation.ispartofpageto137
dc.relation.ispartofissue2
dc.relation.ispartofjournalJournal of Neurology, Neurosurgery and Psychiatry
dc.relation.ispartofvolume89
dc.subject.fieldofresearchBiomedical and clinical sciences
dc.subject.fieldofresearchPsychology
dc.subject.fieldofresearchcode32
dc.subject.fieldofresearchcode52
dc.titleClinical course, therapeutic responses and outcomes in relapsing MOG antibody-associated demyelination
dc.typeJournal article
dc.type.descriptionC1 - Articles
dc.type.codeC - Journal Articles
dcterms.licensehttp://creativecommons.org/licenses/by/4.0/
dc.description.versionVersion of Record (VoR)
gro.rights.copyright© The Author(s) 2018. This is an Open Access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work.
gro.hasfulltextFull Text
gro.griffith.authorBroadley, Simon


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