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. 2020 Jun;26(7):806-814.
doi: 10.1177/1352458519845112. Epub 2019 May 16.

The clinical spectrum and incidence of anti-MOG-associated acquired demyelinating syndromes in children and adults

C L de Mol  1 , Yym Wong  1 , E D van Pelt  1 , Bha Wokke  1 , Tam Siepman  1 , R F Neuteboom  2 , D Hamann  3 , R Q Hintzen  1
Affiliations

The clinical spectrum and incidence of anti-MOG-associated acquired demyelinating syndromes in children and adults

C L de Mol et al. Mult Scler. 2020 Jun.

Abstract

Objectives: The aim of this study was to assess the Dutch nationwide incidence of myelin oligodendrocyte glycoprotein (MOG)-IgG-associated acquired demyelinating syndromes (ADS) and to describe the clinical and serological characteristics of these patients.

Methods: All serum samples for routine diagnostics from February 2014 to December 2017 were sent to the single central reference laboratory for the full-length MOG-IgG cell-based assay (CBA) in the Netherlands. Clinical data from patients known in our National ADS centre were available.

Results: A total of 1414 samples of 1277 patients were received; of these, 92 patients (7%) were MOG-IgG-seropositive. The mean incidence was 0.16/100,000 people, with higher seropositivity in children (0.31/100,000) than in adults (0.13/100,000). In MOG-IgG-positive patients at the National ADS centre (61/92, 66%), the most common presenting phenotype is acute disseminated encephalomyelitis (ADEM, 56%) in children and optic neuritis (ON, 44%) in adults. Relapsing disease occurred in 9/34 (26%) children and 11/27 (41%) adults during median follow-up of 27.5 months. Patients were tested MOG-IgG-positive >200 months after the initial attack, suggesting an extended time to first relapse (TTFR). Longitudinal analysis of MOG-IgG (25/61, 41%) showed that 67% of the monophasic patients remain seropositive and 60% in relapsing patients. Majority of seronegative patients had no relapses (89%).

Conclusion: This nationwide study shows that the overall incidence of MOG-IgG-seropositive disorders is 0.16 per 100,000 people. The distribution over the clinical phenotypes differs between adults and children. Seropositivity can be maintained over years even without clinical activity, while seronegative patients generally had no relapses.

Keywords: Acquired demyelinating syndromes; adults; anti-MOG antibodies; children; incidence; multiple sclerosis variants.

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Conflict of interest statement

Declaration of Conflicting Interests: The author(s) declared the following potential conflicts of interest with respect to the research, authorship and/or publication of this article: R.F.N. participated in trials with Sanofi and Novartis. R.Q.H. received honoraria for serving on advisory boards for Biogen Idec, Roche and Sanofi; he participated in trials with Biogen Idec, Merck-Serono, Roche, Genzyme and Novartis

Figures

Figure 1.
Figure 1.
Distribution of clinical phenotypes in adults and children: (a) phenotypes at first presentation; (b) phenotypes at last follow-up. Other (n = 7) children were diagnosed with ADEM-ON (n = 3), ADEM-TM (n = 1), ON followed by cortical phenotype (epilepsy and hemiparesis, n = 1), clinically isolated syndrome (CIS, n = 1) and relapsing remitting MS (n = 1). Striped areas: bilateral optic neuritis and longitudinal extensive transverse myelitis, respectively. ON: optic neuritis; TM: transverse myelitis; CIS: clinically isolated syndromes other than ON or TM (other CIS); NMO: neuromyelitis optica; ADEM: acute disseminated encephalomyelitis.
Figure 2.
Figure 2.
Longitudinal analysis of clinical and serological data; 25 of the 61 MOG-IgG-positive patients who were known at the National ADS centre were tested serially for MOG-IgG. Patients are classified into four groups according to their disease course (relapsing or monophasic) and the results of the last tested serum sample of that patient.
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