Opsoclonus myoclonus syndrome in a postpartum period

Adhikari, Srijan; Thuringer, Amanda; Maali, Laith; Jassam, Yasir

doi:10.1016/j.msard.2021.102862

Cited by 10 publications

(9 citation statements)

References 8 publications

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“…However, at no point should sustained remission be considered equivalent to permanent remission, as relapses are possible even several years later and have been reported decades later 53 with specific stresses such as pregnancy. 54 Relapses reportedly occur in over half of cases in 1 series. 10 Long-term sequelae including cognitive impairment and speech problems may be seen in up to 75% of patients but may potentially be mitigated by earlier and more aggressive immunotherapy.…”

Section: Side Effectsmentioning

confidence: 95%

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Diagnosis and Management of Opsoclonus-Myoclonus-Ataxia Syndrome in Children

Rossor

Yeh

Khakoo

et al. 2022

Neurol Neuroimmunol Neuroinflamm

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Background and ObjectivesOpsoclonus-myoclonus-ataxia syndrome (OMAS) is a rare disorder of the nervous system that classically presents with a combination of characteristic eye movement disorder and myoclonus, in addition to ataxia, irritability, and sleep disturbance. There is good evidence that OMAS is an immune-mediated condition that may be paraneoplastic in the context of neuroblastoma. This syndrome may be associated with long-term cognitive impairment, yet it remains unclear how this is influenced by disease course and treatment. Treatment is largely predicated on immune suppression, but there is limited evidence to indicate an optimal regimen.MethodsFollowing an international multiprofessional workshop in 2004, a body of clinicians and scientists comprising the International OMS Study group continued to meet biennially in a joint professionals and family workshop focusing on pediatric OMAS. Seventeen years after publication of the first report, a writing group was convened to provide a clinical update on the definitions and clinical presentation of OMAS, biomarkers and the role of investigations in a child presenting with OMAS, treatment and management strategies including identification and support of long-term sequelae.ResultsThe clinical criteria for diagnosis were reviewed, with a proposed approach to laboratory and radiologic investigation of a child presenting with possible OMAS. The evidence for an upfront vs escalating treatment regimen was reviewed, and a treatment algorithm proposed to recognize both these approaches. Importantly, recommendations on monitoring of immunotherapy response and longer-term follow-up based on an expert consensus are provided.DiscussionOMAS is a rare neurologic condition that can be associated with poor cognitive outcomes. This report proposes an approach to investigation and treatment of children presenting with OMAS, based on expert international opinion recognizing the limited data available.

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Section: Side Effectsmentioning

confidence: 95%

Section: Immune Treatment Strategies In Omasmentioning

confidence: 99%

Diagnosis and Management of Opsoclonus-Myoclonus-Ataxia Syndrome in Children

Rossor

Yeh

Khakoo

et al. 2022

Neurol Neuroimmunol Neuroinflamm

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“… 68 , 75 Given the overall rarity of double positive cases based on the results of the larger studies, and considering the superior specificity of the AQP4-IgG test compared to the MOG-IgG test, and that a reliable cell-based-assay (CBA) for detecting MOG-IgG has been available just recently (2017), there is a high possibility that most of the double positive cases may likely be AQP4-IgG + NMOSD with false positive MOG-IgG test results, especially in reports made before this time with using enzyme-linked immunosorbent assay (ELISA). 68 , 75 …”

Section: Discussionmentioning

confidence: 99%

Autoimmune diseases and cancers overlapping with myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD): A systematic review

Molazadeh

Bose

Lotan

et al. 2022

Multiple Sclerosis Journal - Experimental, Translational and Cl

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Background Myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) has various similarities with AQP4-IgG-seropositive Neuromyelitis Optica Spectrum Disorder (AQP4-IgG + NMOSD) in terms of clinical presentations, magnetic resonance imaging (MRI) findings, and response to treatment. But unlike AQP4-IgG + NMOSD, which is known to coexist with various autoimmune diseases and cancers, an association of MOGAD with these conditions is less clear. Methods We conducted a systematic search in PubMed, Scopus, Web of Science, and Embase based on the preferred reporting items for systematic reviews and meta-analysis (PRISMA). Duplicates were removed using Mendeley 1.19.8 (USA production) and the citations were uploaded into Covidence systematic review platform for screening. Results The most common autoimmune disease overlapping with MOGAD was anti-N-Methyl-D-Aspartate receptor encephalitis (anti-NMDAR-EN), followed by autoimmune thyroid disorders, and the most common autoantibody was antinuclear antibody (ANA), followed by AQP4-IgG (double-positive MOG-IgG and AQP4-IgG). A few sporadic cases of cancers and MOG-IgG-associated paraneoplastic encephalomyelitis were found. Conclusion Unlike AQP4-IgG + NMOSD, MOGAD lacks clustering of autoimmune diseases and autoantibodies associated with systemic and organ-specific autoimmunity. Other than anti-NMDAR-EN and perhaps AQP4-IgG + NMOSD, the evidence thus far does not support the need for routine screening of overlapping autoimmunity and neoplasms in patients with MOGAD.

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“…Twenty-two papers were finally excluded because of lack of sufficient clinical data provided or unconfirmed MOGAD diagnosis (eFigure 1 in online supplemental file 1). 11–32 As no randomised controlled clinical trials were found, only uncontrolled observational studies were included in the meta-analysis.…”

Section: Methodsmentioning

confidence: 99%

Efficacy and safety of rituximab in myelin oligodendrocyte glycoprotein antibody-associated disorders compared with neuromyelitis optica spectrum disorder: a systematic review and meta-analysis

Spagni

Sun

Monte

et al. 2022

J Neurol Neurosurg Psychiatry

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BackgroundRituximab (RTX) efficacy in patients with myelin oligodendrocyte glycoprotein (MOG) antibody-associated disorders (MOGADs) is still poorly understood, though it appears to be lower than in aquaporin-4-IgG-positive neuromyelitis optica spectrum disorders (AQP4-IgG+NMOSDs). The aim of this systematic review and meta-analysis is to assess the efficacy and safety profile of RTX in patients with MOGAD and to compare RTX efficacy between MOGAD and AQP4-IgG+NMOSD.MethodsWe searched original English-language articles published between 2012 and 2021 in MEDLINE, Cochrane, Central Register of Controlled Trials and clinicaltrials.gov, reporting data on RTX efficacy in patients with MOGAD. The main outcome measures were annualised relapse rate (ARR) and Expanded Disability Status Scale (EDSS) score mean differences (MDs) after RTX. The meta-analysis was performed with a random effects model. Covariates associated with the outcome measures were analysed with a linear meta-regression.ResultsThe systematic review included 315 patients (138 women, mean onset age 26.8 years) from 32 studies. Nineteen studies (282 patients) were included in the meta-analysis. After RTX, a significant decrease of ARR was found (MD: −0.92, 95% CI –1.24 to –0.60, p<0.001), markedly different from the AQP4-IgG+NMOSD (MD: −1.73 vs MOGAD −0.92, subgroup difference testing: Q=9.09, p=0.002). However, when controlling for the mean ARR pre-RTX, this difference was not significant. After RTX, the EDSS score decreased significantly (MD: −0.84, 95% CI −1.41 to –0.26, p=0.004). The frequency of RTX-related adverse events was 18.8% (36/192) and overall RTX-related mortality 0.5% (1/192).ConclusionsRTX showed effective in MOGAD, although to a lesser extent than in AQP4-IgG+NMOSD, while the safety profile warrants some caution in its prescription. Randomised-controlled trials are needed to confirm these findings and provide robust evidence to improve treatment strategies in patients with MOGAD.PROSPERO registration numberCRD42020175439.

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Opsoclonus myoclonus syndrome in a postpartum period

Cited by 10 publications

References 8 publications

Diagnosis and Management of Opsoclonus-Myoclonus-Ataxia Syndrome in Children

Diagnosis and Management of Opsoclonus-Myoclonus-Ataxia Syndrome in Children

Autoimmune diseases and cancers overlapping with myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD): A systematic review

Efficacy and safety of rituximab in myelin oligodendrocyte glycoprotein antibody-associated disorders compared with neuromyelitis optica spectrum disorder: a systematic review and meta-analysis

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