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  • Clinical course, therapeutic responses and outcomes in relapsing MOG antibody-associated demyelination

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    Author(s)
    Ramanathan, Sudarshini
    Mohammad, Shekeeb
    Tantsis, Esther
    Nguyen, Tina Kim
    Merheb, Vera
    Fung, Victor SC
    White, Owen Bruce
    Broadley, Simon
    Lechner-Scott, Jeannette
    Vucic, Steve
    Henderson, Andrew PD
    Barnett, Michael Harry
    Reddel, Stephen W
    Brilot, Fabienne
    Dale, Russell C
    Griffith University Author(s)
    Broadley, Simon
    Year published
    2018
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    Abstract
    Objective: 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 ...
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    Objective: 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.
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    Journal Title
    Journal of Neurology, Neurosurgery and Psychiatry
    Volume
    89
    Issue
    2
    DOI
    https://doi.org/10.1136/jnnp-2017-316880
    Copyright Statement
    © 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.
    Subject
    Biomedical and clinical sciences
    Psychology
    Publication URI
    http://hdl.handle.net/10072/377228
    Collection
    • Journal articles

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