Abstract
Background
Anti-myelin oligodendrocyte glycoprotein antibody-associated disorders (MOGAD) are new emerging diseases with heterogeneous course, treatment, response, and prognosis.
Case report
We herein present 2 cases with antibodies to MOG, one with a cerebellar/brainstem monophasic syndrome which partially improved after treatment, and the other with an optic neuritis onset then relapsed with cortical encephalitis and presented a subsequent complete recovery. We further discuss elements possibly associated with disease heterogeneity and influencing treatment choices.
Conclusions
MOGAD is an extremely variable disease which can relapse and accumulate disability over time. An early diagnosis and correct timely treatment is fundamental to improve clinical outcome.
References
Jarius S et al (2016) MOG-IgG in NMO and related disorders: a multicenter study of 50 patients. Part 1: frequency, syndrome specificity, influence of disease activity, long-term course, association with AQP4-IgG, and origin. J Neuroinflammation 13:1–16
Cobo-Calvo A, Ruiz A, Maillart E, Audoin B, Zephir H, Bourre B, Ciron J, Collongues N, Brassat D, Cotton F, Papeix C, Durand-Dubief F, Laplaud D, Deschamps R, Cohen M, Biotti D, Ayrignac X, Tilikete C, Thouvenot E, Brochet B, Dulau C, Moreau T, Tourbah A, Lebranchu P, Michel L, Lebrun-Frenay C, Montcuquet A, Mathey G, Debouverie M, Pelletier J, Labauge P, Derache N, Coustans M, Rollot F, de Seze J, Vukusic S, Marignier R (2018) Clinical spectrum and prognostic value of CNS MOG autoimmunity in adults: the MOGADOR study. Neurology 90:e1858–e1869. https://doi.org/10.1212/WNL.0000000000005560
Jurynczyk M, Messina S, Woodhall MR, Raza N, Everett R, Roca-Fernandez A, Tackley G, Hamid S, Sheard A, Reynolds G, Chandratre S, Hemingway C, Jacob A, Vincent A, Leite MI, Waters P, Palace J (2017) Clinical presentation and prognosis in MOG-antibody disease: a UK study. Brain 140:3128–3138. https://doi.org/10.1093/brain/awx276
Mariotto S, Ferrari S, Monaco S, Benedetti MD, Schanda K, Alberti D, Farinazzo A, Capra R, Mancinelli C, de Rossi N, Bombardi R, Zuliani L, Zoccarato M, Tanel R, Bonora A, Turatti M, Calabrese M, Polo A, Pavone A, Grazian L, Sechi GP, Sechi E, Urso D, Delogu R, Janes F, Deotto L, Cadaldini M, Bianchi MR, Cantalupo G, Reindl M, Gajofatto A (2017) Clinical spectrum and IgG subclass analysis of anti-myelin oligodendrocyte glycoprotein antibody-associated syndromes: a multicenter study. J Neurol 264:2420–2430. https://doi.org/10.1007/s00415-017-8635-4
López-Chiriboga AS, Majed M, Fryer J, Dubey D, McKeon A, Flanagan EP, Jitprapaikulsan J, Kothapalli N, Tillema JM, Chen J, Weinshenker B, Wingerchuk D, Sagen J, Gadoth A, Lennon VA, Keegan BM, Lucchinetti C, Pittock SJ (2018) Association of MOG-IgG serostatus with relapse after acute disseminated encephalomyelitis and proposed diagnostic criteria for MOG-IgG-associated disorders. JAMA Neurol 75:1355. https://doi.org/10.1001/jamaneurol.2018.1814
Salama S, Khan M, Levy M, Izbudak I (2019) Radiological characteristics of myelin oligodendrocyte glycoprotein antibody disease. Mult Scler Relat Disord 29:15–22. https://doi.org/10.1016/j.msard.2019.01.021
Budhram A, Mirian A, le C, Hosseini-Moghaddam SM, Sharma M, Nicolle MW (2019) Unilateral cortical FLAIR-hyperintense lesions in anti-MOG-associated encephalitis with seizures (FLAMES): characterization of a distinct clinico-radiographic syndrome. J Neurol 266:2481–2487. https://doi.org/10.1007/s00415-019-09440-8
Jarius S, Paul F, Aktas O, Asgari N, Dale RC, de Seze J, Franciotta D, Fujihara K, Jacob A, Kim HJ, Kleiter I, Kümpfel T, Levy M, Palace J, Ruprecht K, Saiz A, Trebst C, Weinshenker BG, Wildemann B (2018) MOG encephalomyelitis: international recommendations on diagnosis and antibody testing. J Neuroinflammation 15:134. https://doi.org/10.1186/s12974-018-1144-2
Spadaro M, Gerdes LA, Krumbholz M, Ertl-Wagner B, Thaler FS, Schuh E, Metz I, Blaschek A, Dick A, Brück W, Hohlfeld R, Meinl E, Kümpfel T (2016) Autoantibodies to MOG in a distinct subgroup of adult multiple sclerosis. Neurol - Neuroimmunol Neuroinflammation 3. https://doi.org/10.1212/nxi.0000000000000257
Chen JJ, Flanagan EP, Jitprapaikulsan J, López-Chiriboga A(S)S, Fryer JP, Leavitt JA, Weinshenker BG, McKeon A, Tillema JM, Lennon VA, Tobin WO, Keegan BM, Lucchinetti CF, Kantarci OH, McClelland CM, Lee MS, Bennett JL, Pelak VS, Chen Y, VanStavern G, Adesina OOO, Eggenberger ER, Acierno MD, Wingerchuk DM, Brazis PW, Sagen J, Pittock SJ (2018) Myelin oligodendrocyte glycoprotein antibody–positive optic neuritis: clinical characteristics, radiologic clues, and outcome. Am J Ophthalmol 195:8–15. https://doi.org/10.1016/j.ajo.2018.07.020
Ogawa R, Nakashima I, Takahashi T, Kaneko K, Akaishi T, Takai Y, Sato DK, Nishiyama S, Misu T, Kuroda H, Aoki M, Fujihara K (2017) MOG antibody-positive, benign, unilateral, cerebral cortical encephalitis with epilepsy. Neurol Neuroimmunol NeuroInflammation 4:e322. https://doi.org/10.1212/NXI.0000000000000322
Mariotto S, Monaco S, Peschl P, Coledan I, Mazzi R, Höftberger R, Reindl M, Ferrari S (2017) MOG antibody seropositivity in a patient with encephalitis: beyond the classical syndrome. BMC Neurol 17. https://doi.org/10.1186/s12883-017-0971-6
Cobo-Calvo A et al (2019) Usefulness of MOG-antibody titres at first episode to predict the future clinical course in adults. J Neurol 266:806–815. https://doi.org/10.1007/s00415-018-9160-9
Mariotto S, Gajofatto A, Batzu L, Delogu R, Sechi GP, Leoni S, Pirastru MI, Bonetti B, Zanoni M, Alberti D, Schanda K, Monaco S, Reindl M, Ferrari S (2019) Relevance of antibodies to myelin oligodendrocyte glycoprotein in CSF of seronegative cases. Neurology. https://doi.org/10.1212/WNL.0000000000008479
Mariotto S, Ferrari S, Gastaldi M, Franciotta D, Sechi E, Capra R, Mancinelli C, Schanda K, Alberti D, Orlandi R, Bombardi R, Zuliani L, Zoccarato M, Benedetti MD, Tanel R, Calabria F, Rossi F, Pavone A, Grazian L, Sechi GP, Batzu L, Murdeu N, Janes F, Fetoni V, Fulitano D, Stenta G, Federle L, Cantalupo G, Reindl M, Monaco S, Gajofatto A (2019) Neurofilament light chain serum levels reflect disease severity in MOG-Ab associated disorders. J Neurol Neurosurg Psychiatry 90:1293–1296. https://doi.org/10.1136/jnnp-2018-320287
Author information
Authors and Affiliations
Corresponding author
Ethics declarations
Conflict of interest
None.
Ethical approval
None.
Additional information
Publisher’s note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Rights and permissions
About this article
Cite this article
Maniscalco, G.T., Allegorico, L., Alfieri, G. et al. Anti-MOG-associated demyelinating disorders: two sides of the same coin. Neurol Sci 42, 1531–1534 (2021). https://doi.org/10.1007/s10072-020-04892-7
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s10072-020-04892-7