Progressive myelopathy in myelin oligodendrocyte glycoprotein antibody-associated disease: A new mimicker of progressive multiple sclerosis?

Marcucci, Samuel B.; Elkasaby, Mohamed; Walch, R.; Zare-Shahabadi, Ameneh; Mahammedi, Abdelkader; Abboud, Hesham; Zabeti, Aram

doi:10.1016/j.msard.2021.102964

Cited by 8 publications

(5 citation statements)

References 20 publications

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“…Progressive cases of MOGAD are rare, with only seven reported to date. [1][2][3] In this patient, the initial presentation with ADEM was in keeping with the agedependent pattern reported in other large cohorts. [4][5][6] In children with onset <10 years, the most common MOG-IgG-associated presentation is ADEM, while in adults ON and/or TM predominate.…”

Section: Discussionsupporting

confidence: 87%

Progressive myelin oligodendrocyte glycoprotein-associated demyelination mimicking leukodystrophy

et al. 2022

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Background: Myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) may be associated with relapsing disease, but clinical progression independent of relapse activity is rare. Objectives: To report progressive disease in a patient with MOGAD. Methods: A single retrospective case report. Results: At 4 years of age, the patient had a single episode of acute disseminated encephalomyelitis. She remained well until age 17 years but over the next 9 years developed progressive spastic quadriparesis, cognitive and bulbar dysfunction. Brain imaging showed a leukodystrophy-like pattern of white matter abnormality with contrast enhancement at different time points. Myelin oligodendrocyte glycoprotein (MOG)-IgG was repeatedly positive by live cell-based assay. Conclusion: Secondary progression may be a rare presentation of MOG-IgG-associated disease.

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Section: Discussionsupporting

confidence: 87%

Progressive myelin oligodendrocyte glycoprotein-associated demyelination mimicking leukodystrophy

et al. 2022

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“…Although a chronic onset has been shown to be a useful discriminator for noninflammatory myelopathy, a longer progression to nadir has been described in cases of AQP4-IgG NMOSD-LETM and MOGAD-LETM. 18,19 In our cohort, two AQP4-IgG NMOSD-LETM and two MOGAD patients had a chronic disease onset. Five dsLETM patients (18%) progressed to symptom nadir by a median of 60 days.…”

Section: Discussionmentioning

confidence: 91%

The absence of antibodies in longitudinally extensive transverse myelitis may predict a more favourable prognosis

Rocchi,

Forcadela,

Kelly

et al. 2024

Mult Scler

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Background: Isolated first episodes of longitudinally extensive transverse myelitis (LETM) have typically been associated with neuromyelitis optica spectrum disorder (NMOSD) or myelin oligodendrocyte glycoprotein antibody–associated disease (MOGAD). However, in some cases, serological testing and screening for other aetiologies are negative, a condition referred to as double seronegative longitudinally extensive transverse myelitis (dsLETM). Objective: The objective of this study was to evaluate comparative outcomes of dsLETM, MOGAD-LETM and NMOSD-LETM. Methods: Cohort study of LETM cases seen in the UK NMOSD Highly Specialised Service between January 2008 and March 2022. Results: LETM = 87 cases were identified (median onset age = 46 years (15–85); median follow-up = 46 months (1–144); 47% NMOSD-LETM = 41 (aquaporin-4 antibodies (AQP4-IgG) positive = 36), 20% MOGAD-LETM = 17 and 33% dsLETM = 29). Despite similar Expanded Disability Status Scale (EDSS) at nadir, last EDSS was higher in AQP4-IgG and seronegative NMOSD-LETM (sNMOSD) ( p = 0.006). Relapses were less common in dsLETM compared to AQP4-IgG NMOSD-LETM and sNMOSD-LETM (19% vs 60% vs 100%; p = 0.001). Poor prognosis could be predicted by AQP4-IgG (odds ratio (OR) = 38.86 (95% confidence interval (CI) = 1.36–1112.86); p = 0.03) and EDSS 3 months after onset (OR = 65.85 (95% CI = 3.65–1188.60); p = 0.005). Conclusion: dsLETM remains clinically challenging and difficult to classify with existing nosological terminology. Despite a similar EDSS at nadir, patients with dsLETM relapsed less and had a better long-term prognosis than NMOSD-LETM.

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“…International panel group for MOGAD diagnostic criteria have recommended avoiding MOG-IgG screening of all patients with CNS inflammatory demyelination, and a cautious interpretation of positive results in patients with atypical findings for MOGAD [ 2 ]. We agree that such a prudent stance is appropriate; however, since atypical manifestations including progressive myelopathy or combined peripheral neuropathy with MOG-IgG have been reported [ 11 , 12 ] and the significance of clear positivity of serum MOG-IgG in patients with atypical phenotype is yet to be elucidated, more substantial data are needed for defining the spectrum of MOGAD phenotype. MOG-IgG testing should be considered in these contexts in image-negative myelopathy patients, particularly if no better explanation is identified even though extensive work-up yields negative results.…”

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confidence: 87%

MRI-negative myelitis associated with MOG-IgG antibody: A case report and literature reviews

Yang,

Lee,

Park

2023

eNeurologicalSci

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Progressive myelopathy in myelin oligodendrocyte glycoprotein antibody-associated disease: A new mimicker of progressive multiple sclerosis?

Cited by 8 publications

References 20 publications

Progressive myelin oligodendrocyte glycoprotein-associated demyelination mimicking leukodystrophy

Progressive myelin oligodendrocyte glycoprotein-associated demyelination mimicking leukodystrophy

The absence of antibodies in longitudinally extensive transverse myelitis may predict a more favourable prognosis

MRI-negative myelitis associated with MOG-IgG antibody: A case report and literature reviews

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