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Published in: Acta Neurologica Belgica 1/2021

01-02-2021 | Azathioprine | Original article

Clinical spectrum, treatment and outcome of myelin oligodendrocyte glycoprotein (MOG) antibody-associated disease in children: a tertiary care experience

Authors: Hepsen Mine Serin, Sanem Yilmaz, Erdem Simsek, Seda Kanmaz, Cenk Eraslan, Gul Aktan, Hasan Tekgul, Sarenur Gokben

Published in: Acta Neurologica Belgica | Issue 1/2021

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Abstract

Anti-myelin oligodendrocyte glycoprotein antibodies have been associated with a wide range of clinical presentations including monophasic and relapsing disease courses. Lack of a definitive marker for predicting further relapses and the final diagnoses complicates the clinical follow-up and treatment decisions for patients with the first episode. This study retrospectively analyzed the clinical spectrum, treatment protocols and outcome of nine children with MOG antibody-associated demyelinating disease. Diagnoses at first presentation were acute disseminated encephalomyelitis (ADEM) in six cases (67%), optic neuritis in two cases (22%), and clinically isolated syndrome in one case (11%). The disease remained monophasic in five (56%) cases. All cases with a monophasic disease course were negative for anti-MOG antibody titers in the third month. The initial diagnosis of all relapsing cases was ADEM. Three of the four cases with a relapsing disease course were available for anti-MOG antibody testing at the third month and all were positive, however, antibody titers at the sixth month were inconsistent. Cases with a relapsing disease course had no further attacks after monthly intravenous immunoglobulin treatment. Relapsing disease course is not rare in childhood MOG-antibody associated demyelinating disease. Monthly IVIG treatment may be a good alternative for the long-term treatment of relapsing cases with a low side effect profile. Anti-MOG antibody serostatus at remission periods should be interpreted cautiously. Further studies are needed to better understand and predict the clinical course of pediatric patients with MOG-antibody associated diseases.
Literature
1.
go back to reference Ketelslegers IA, Van Pelt DE, Bryde S et al (2015) Anti-MOG antibodies plead against MS diagnosis in an acquired demyelinating syndromes cohort. MultScler 21:1513–1520 Ketelslegers IA, Van Pelt DE, Bryde S et al (2015) Anti-MOG antibodies plead against MS diagnosis in an acquired demyelinating syndromes cohort. MultScler 21:1513–1520
3.
go back to reference O’Connor KC, McLaughlin KA, De Jager PL et al (2007) Self-antigen tetramers discriminate between myelin autoantibodies to native or denatured protein. Nat Med 13:211–217CrossRef O’Connor KC, McLaughlin KA, De Jager PL et al (2007) Self-antigen tetramers discriminate between myelin autoantibodies to native or denatured protein. Nat Med 13:211–217CrossRef
4.
go back to reference Ramanathan S, Dale RC, Brilot F (2016) Anti-MOG antibody: the history, clinical phenotype, and pathogenicity of a serum biomarker for demyelination. Autoimmun Rev 15:307–324CrossRef Ramanathan S, Dale RC, Brilot F (2016) Anti-MOG antibody: the history, clinical phenotype, and pathogenicity of a serum biomarker for demyelination. Autoimmun Rev 15:307–324CrossRef
5.
go back to reference Tegetmeyer H, Merkenschlager A (2017) Anti-myelin oligodendrocyte glycoprotein antibodies in paediatric patients with optic neuritis (in German). KlinMonblAugenheilkd 234:1243–1249 Tegetmeyer H, Merkenschlager A (2017) Anti-myelin oligodendrocyte glycoprotein antibodies in paediatric patients with optic neuritis (in German). KlinMonblAugenheilkd 234:1243–1249
6.
go back to reference Hacohen Y, Absoud M, Deiva K et al (2015) Myelin oligodendrocyte glycoprotein antibodies are associated with a non-MS course in children. NeurolNeuroimmunolNeuroinflamm 2:e81 Hacohen Y, Absoud M, Deiva K et al (2015) Myelin oligodendrocyte glycoprotein antibodies are associated with a non-MS course in children. NeurolNeuroimmunolNeuroinflamm 2:e81
7.
go back to reference Hennes E-M, Baumann M, Schanda K et al (2017) Prognostic relevance of MOG antibodies in children with an acquired demyelinating syndrome. Neurology 89:900–908CrossRef Hennes E-M, Baumann M, Schanda K et al (2017) Prognostic relevance of MOG antibodies in children with an acquired demyelinating syndrome. Neurology 89:900–908CrossRef
8.
go back to reference Konuskan B, Yildirim M, Gocmen R et al (2018) Retrospective analysis of children with myelin oligodendrocyte glycoprotein antibody-related disorders. MultSclerRelatDisord 26:1–7 Konuskan B, Yildirim M, Gocmen R et al (2018) Retrospective analysis of children with myelin oligodendrocyte glycoprotein antibody-related disorders. MultSclerRelatDisord 26:1–7
9.
go back to reference Hacohen Y, Wong YY, Lechner C et al (2018) Disease course and treatment responses in children with relapsing myelin oligodendrocyte glycoprotein antibody-associated disease. JAMANeurol 75:478–487 Hacohen Y, Wong YY, Lechner C et al (2018) Disease course and treatment responses in children with relapsing myelin oligodendrocyte glycoprotein antibody-associated disease. JAMANeurol 75:478–487
11.
go back to reference Wingerchuk DM, Banwell B, Bennett JL et al (2015) International consensus diagnostic criteria for neuromyelitis optica spectrum disorders. Neurology 85:177–189CrossRef Wingerchuk DM, Banwell B, Bennett JL et al (2015) International consensus diagnostic criteria for neuromyelitis optica spectrum disorders. Neurology 85:177–189CrossRef
12.
go back to reference Krupp LB, Tardieu M, Amato MP et al (2013) International Pediatric Multiple Sclerosis Study Group criteria for pediatric multiple sclerosis and immune-mediated central nervous system demyelinating disorders: revisions to the 2007 definitions. MultScler 19:1261–1267 Krupp LB, Tardieu M, Amato MP et al (2013) International Pediatric Multiple Sclerosis Study Group criteria for pediatric multiple sclerosis and immune-mediated central nervous system demyelinating disorders: revisions to the 2007 definitions. MultScler 19:1261–1267
14.
go back to reference Ramanathan S, Mohammad S, Tantsis E et al (2018) Clinical course, therapeutic responses and outcomes in relapsing MOG antibody-associated demyelination. J NeurolNeurosurg Psychiatry 89:127–137CrossRef Ramanathan S, Mohammad S, Tantsis E et al (2018) Clinical course, therapeutic responses and outcomes in relapsing MOG antibody-associated demyelination. J NeurolNeurosurg Psychiatry 89:127–137CrossRef
15.
go back to reference Baumann M, Sahin K, Lechner C et al (2015) Clinical and neuroradiological differences of paediatric acute disseminating encephalomyelitis with and without antibodies to the myelin oligodendrocyte glycoprotein. J NeurolNeurosurg Psychiatry 86:265–272CrossRef Baumann M, Sahin K, Lechner C et al (2015) Clinical and neuroradiological differences of paediatric acute disseminating encephalomyelitis with and without antibodies to the myelin oligodendrocyte glycoprotein. J NeurolNeurosurg Psychiatry 86:265–272CrossRef
16.
go back to reference Jarius S, Kleiter I, Ruprecht K et al (2016) MOG-IgG in NMO and related disorders: a multicenter study of 50 patients. Part 3: brainstem involvement—frequency, presentation and outcome. J Neuroinflamm 13:281CrossRef Jarius S, Kleiter I, Ruprecht K et al (2016) MOG-IgG in NMO and related disorders: a multicenter study of 50 patients. Part 3: brainstem involvement—frequency, presentation and outcome. J Neuroinflamm 13:281CrossRef
17.
go back to reference Fernandez-Carbonell C, Vargas-Lowy D, Musallam A et al (2016) Clinical and MRI phenotype of children with MOG antibodies. MultScler 22:174–184 Fernandez-Carbonell C, Vargas-Lowy D, Musallam A et al (2016) Clinical and MRI phenotype of children with MOG antibodies. MultScler 22:174–184
18.
go back to reference Baumann M, Grams A, Djurdjevic T et al (2018) MRI of the first event in pediatric acquired demyelinating syndromes with antibodies to myelin oligodendrocyte glycoprotein. J Neurol 265:845–855CrossRef Baumann M, Grams A, Djurdjevic T et al (2018) MRI of the first event in pediatric acquired demyelinating syndromes with antibodies to myelin oligodendrocyte glycoprotein. J Neurol 265:845–855CrossRef
19.
go back to reference dos Passos GR, Oliveira LM, da Costa BK et al (2018) MOG-IgG-associated optic neuritis, encephalitis, and myelitis: lessons learned from neuromyelitis optica spectrum disorder. Front Neurol 9:217CrossRef dos Passos GR, Oliveira LM, da Costa BK et al (2018) MOG-IgG-associated optic neuritis, encephalitis, and myelitis: lessons learned from neuromyelitis optica spectrum disorder. Front Neurol 9:217CrossRef
20.
go back to reference Marignier R, Bernard-Valnet R, Giraudon P et al (2013) Aquaporin-4 antibody-negative neuromyelitis optica: distinct assay sensitivity-dependent entity. Neurology 80:2194–2200CrossRef Marignier R, Bernard-Valnet R, Giraudon P et al (2013) Aquaporin-4 antibody-negative neuromyelitis optica: distinct assay sensitivity-dependent entity. Neurology 80:2194–2200CrossRef
21.
go back to reference Yan Y, Li Y, Fu Y et al (2016) Autoantibody to MOG suggests two distinct clinical subtypes of NMOSD. Sci China Life Sci 59:1270–1281CrossRef Yan Y, Li Y, Fu Y et al (2016) Autoantibody to MOG suggests two distinct clinical subtypes of NMOSD. Sci China Life Sci 59:1270–1281CrossRef
22.
go back to reference Duignan S, Wright S, Rossor T et al (2018) Myelin oligodendrocyte glycoprotein and aquaporin-4 antibodies are highly specific in children with acquired demyelinating syndromes. Dev Med Child Neurol 60:958–962CrossRef Duignan S, Wright S, Rossor T et al (2018) Myelin oligodendrocyte glycoprotein and aquaporin-4 antibodies are highly specific in children with acquired demyelinating syndromes. Dev Med Child Neurol 60:958–962CrossRef
23.
go back to reference Dale RC, Tantsis EM, Merheb V et al (2014) Antibodies to MOG have a demyelination phenotype and affect oligodendrocyte cytoskeleton. NeurolNeuroimmunolNeuroinflamm 1:e12 Dale RC, Tantsis EM, Merheb V et al (2014) Antibodies to MOG have a demyelination phenotype and affect oligodendrocyte cytoskeleton. NeurolNeuroimmunolNeuroinflamm 1:e12
24.
go back to reference Hyun J, Woodhall MR, Kim S et al (2017) Longitudinal analysis of myelin oligodendrocyte glycoprotein antibodies in CNS inflammatory diseases. J NeurolNeurosurg Psychiatry 88:811–817CrossRef Hyun J, Woodhall MR, Kim S et al (2017) Longitudinal analysis of myelin oligodendrocyte glycoprotein antibodies in CNS inflammatory diseases. J NeurolNeurosurg Psychiatry 88:811–817CrossRef
26.
go back to reference Jarius S, Ruprecht K, Kleiter I 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 Neuroinflamm 13:279CrossRef Jarius S, Ruprecht K, Kleiter I 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 Neuroinflamm 13:279CrossRef
29.
go back to reference López-Chiriboga AS, Majed M, Fryer J, Dubey D et al (2018) Association of MOG-IgGserostatus with relapse after acute disseminated encephalomyelitis and proposed diagnostic criteria for MOG-IgG-associated disorders. JAMANeurol 75:1355 López-Chiriboga AS, Majed M, Fryer J, Dubey D et al (2018) Association of MOG-IgGserostatus with relapse after acute disseminated encephalomyelitis and proposed diagnostic criteria for MOG-IgG-associated disorders. JAMANeurol 75:1355
Metadata
Title
Clinical spectrum, treatment and outcome of myelin oligodendrocyte glycoprotein (MOG) antibody-associated disease in children: a tertiary care experience
Authors
Hepsen Mine Serin
Sanem Yilmaz
Erdem Simsek
Seda Kanmaz
Cenk Eraslan
Gul Aktan
Hasan Tekgul
Sarenur Gokben
Publication date
01-02-2021
Publisher
Springer International Publishing
Published in
Acta Neurologica Belgica / Issue 1/2021
Print ISSN: 0300-9009
Electronic ISSN: 2240-2993
DOI
https://doi.org/10.1007/s13760-020-01499-9

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