Seroprevalence and clinical phenotype of MOG-IgG-associated disorders in Sri Lanka

Jitprapaikulsan, Jiraporn; Aravinthan, M.; Wijesekera, J.; Ranawaka, Udaya K.; Riffsy, M.; Paramanathan, Thakshan; Sagen, Jessica; Fryer, James P.; Schmeling, John E.; Majed, Masoud; Flanagan, Eoin P.; Pittock, Sean J.

doi:10.1136/jnnp-2018-320243

Cited by 24 publications

(37 citation statements)

References 5 publications

(6 reference statements)

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“…Although our study was not designed to evaluate the frequency of MOG-Ab among patients starting with a first ADS, we observed herein that the frequency of MOG-Ab in adults is probably higher than previously thought, according to recent studies. 24,25 An age-dependent clinical phenotype is a typical feature in MOGAD characterized by an ADEM phenotype in children, whereas ON or TM is more frequently found in adults. 2,3,[5][6][7][8][9][10] Interestingly, we found here that from early ages, MOGAD follows a shift in the clinical disease presentation; patients aged <10 years displayed an ADEM phenotype, which was still representative in children aged 10-18 years, but showed an abrupt decline in adult subgroups toward a marginal representation.…”

Section: Discussionmentioning

confidence: 99%

Clinical Features and Risk of Relapse in Children and Adults with Myelin Oligodendrocyte Glycoprotein Antibody–Associated Disease

Cobo‐Calvo

Ruiz

Rollot

et al. 2020

Annals of Neurology

130

134

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Objective The main objective was to compare clinical features, disease course, and myelin oligodendrocyte glycoprotein (MOG) antibody (Ab) dynamics between children and adults with MOG‐Ab–associated disease (MOGAD). Methods This retrospective multicentric, national study included 98 children and 268 adults with MOGAD between January 2014 and September 2019. Cox regression model for recurrent time‐to‐event data and Kaplan–Meier curves for time to antibody negativity were performed for the objectives. Results Isolated optic neuritis was the most frequent clinical presentation in both children (40.8%) and adults (55.9%, p = 0.013), and acute disseminated encephalomyelitis syndrome was more frequent in children (36.7% vs 5.6%, p < 0.001). Compared to adults, children displayed better recovery (Expanded Disability Status Scale ≥ 3.0 at last follow‐up reached only by 10 of 97 [10.3%] vs 66/247 [26.7%], p < 0.001). In the multivariate analysis, adults were at higher risk of relapse than children (hazard ratio = 1.41, 95% confidence interval [CI] = 1.12–1.78, p = 0.003). At 2 years, 64.2% (95% CI = 40.9–86.5) of nonrelapsing children became MOG‐Ab negative compared to 14.1% (95% CI = 4.7–38.3) of relapsing children (log‐rank p < 0.001), with no differences observed in adults (log‐rank p = 0.280). Interpretation MOGAD patients differ in the clinical presentation at onset, showing an age‐related shift in the clinical features across age groups. Compared to children, adults have a higher risk of relapse and worse functional recovery. Finally, children with monophasic disease become MOG‐Ab negative earlier than relapsing children, but this is not true in adults. Considering these differences, management and treatment guidelines should be considered independently in children and adults. ANN NEUROL 2021;89:30–41

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Section: Discussionmentioning

confidence: 99%

Clinical Features and Risk of Relapse in Children and Adults with Myelin Oligodendrocyte Glycoprotein Antibody–Associated Disease

Cobo‐Calvo

Ruiz

Rollot

et al. 2020

Annals of Neurology

130

134

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“…Of the adults, 6.5% were MOG-antibody positive vs. 2.6% for AQP4 antibody, while in children, 21.1% were positive for MOG antibody as compared to 1.9% for AQP4 antibody (76). Similarly, one study in Sri Lanka, in collaboration with the Mayo Clinic, also reported more MOG-antibody-positive cases (126 patients) than AQP4-antibody-positive cases (36 patients) (77). On the other hand, MOG-antibody-associated disease was relatively uncommon in the non-Caucasian population in Rio de Janeiro (Brazil) (70).…”

Section: Mog-antibody-associated Disease: Prevalence and Incidencementioning

confidence: 97%

Epidemiology of Neuromyelitis Optica Spectrum Disorder and Its Prevalence and Incidence Worldwide

Hor¹,

et al. 2020

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Neuromyelitis optica spectrum disorder (NMOSD) is an uncommon inflammatory disease of the central nervous system, manifesting clinically as optic neuritis, myelitis, and certain brain and brainstem syndromes. Cases clinically diagnosed as NMOSD may include aquaporin 4 (AQP4)-antibody-seropositive autoimmune astrocytopathic disease, myelin oligodendrocyte glycoprotein (MOG)-antibody-seropositive inflammatory demyelinating disease, and double-seronegative disease. AQP4-antibody disease has a high female-to-male ratio (up to 9:1), and its mean age at onset of ~40 years is later than that seen in multiple sclerosis. For MOG-antibody disease, its gender ratio is closer to 1:1, and it is more common in children than in adults. Its clinical phenotypes differ but overlap with those of AQP4-antibody disease and include acute disseminated encephalomyelitis, brainstem and cerebral cortical encephalitis, as well as optic neuritis and myelitis. Double-seronegative disease requires further research and clarification. Population-based studies over the past two decades report the prevalence and incidence of NMOSD in different populations worldwide. One relevant finding is the varying prevalence observed in different racial groups. Consistently, the prevalence of NMOSD among Whites is ~1/100,000 population, with an annual incidence of <1/million population. Among East Asians, the prevalence is higher, at ~3.5/100,000 population, while the prevalence in Blacks may be up to 10/100,000 population. For MOG-antibody disease, hospital-based studies largely do not observe any significant racial preponderance so far. This disorder comprises a significant proportion of NMOSD cases that are AQP4-antibody-seronegative. A recent Dutch nationwide study reported the annual incidence of MOG-antibody disease as 1.6/million population (adult: 1.3/million, children: 3.1/million). Clinical and radiological differences between AQP4-antibody and MOG-antibody associated diseases have led to interest in the revisions of NMOSD definition and expanded stratification based on detection of a specific autoantibody biomarker. More population-based studies in different geographical regions and racial groups will be useful to further inform the prevalence and incidence of NMOSD and their antibody-specific subgroups. Accessibility to AQP4-antibody and MOG-antibody testing, which is limited in many centers, is a challenge to overcome. Environmental and genetic studies will be useful accompaniments to identify other potential pathogenetic factors and specific biomarkers in NMOSD.

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“…In the latter study, an increase in median EDSS score with observation time was reported. Conversely, among patients from the UK cohort [6], only 7% had a final EDSS score ≥4, and in a study of Sri Lankan patients [2], 6% had a final EDSS score ≥6 (Table 1). However, these studies had relatively short observation periods.…”

Section: Introductionmentioning

confidence: 99%

The long‐term outcome of MOGAD: An observational national cohort study of 61 patients

Deschamps

Pique

Ayrignac

et al. 2021

Euro J of Neurology

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Background and objective The prognosis in myelin oligodendrocyte glycoprotein (MOG) antibody‐associated disease (MOGAD) is a matter of debate. Our aim was to assess the long‐term outcomes of patients with MOGAD. Methods We retrospectively analysed the clinical and paraclinical data of patients from the French nationwide observatory study NOMADMUS who tested positive for MOG antibodies (MOG‐IgG) and who had clinical follow‐up of at least 8 years from their first episode. Results Sixty‐one patients (median [range] age at onset 27 [3–69] years), with a median (mean; range) follow‐up of 177 (212.8; 98–657) months, were included. Among 58 patients with a relapsing course, 26.3% relapsed in the first year after onset. Of the 61 patients, 90.2% experienced at least one episode of optic neuritis. At last visit, the median (mean; range) Expanded Disability Status Scale (EDSS) score was 1 (2.12; 0–7.5), 12.5% had an EDSS score ≥6 and 37.5% had an EDSS score ≥3. Of 51 patients with final visual acuity (VA) data available, 15.7% had VA ≤0.1 in at least one eye and 25.5% had VA ≤0.5 in at least one eye. Bilateral blindness (VA ≤0.1) was present in 5.9% of patients. Finally, 12.5% of patients presented bladder dysfunction requiring long‐term urinary catheterization. No factor associated significantly with a final EDSS score ≥3 or with final VA ≤0.1 was found. Conclusion Overall long‐term favourable outcomes were achieved in a majority of our patients, but severe impairment, in particular visual damage, was not uncommon.

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Seroprevalence and clinical phenotype of MOG-IgG-associated disorders in Sri Lanka

Cited by 24 publications

References 5 publications

Clinical Features and Risk of Relapse in Children and Adults with Myelin Oligodendrocyte Glycoprotein Antibody–Associated Disease

Clinical Features and Risk of Relapse in Children and Adults with Myelin Oligodendrocyte Glycoprotein Antibody–Associated Disease

Epidemiology of Neuromyelitis Optica Spectrum Disorder and Its Prevalence and Incidence Worldwide

The long‐term outcome of MOGAD: An observational national cohort study of 61 patients

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