Acute symptomatic seizures secondary to myelin oligodendrocyte glycoprotein antibody‐associated disease

Montalvo, Mayra; Khattak, Jamal F; Redenbaugh, Vyanka; Britton, Jeffrey W.; Sánchez, Cristina Valencia; Datta, Abhigyan; Tillema, Jan‐Mendelt; Chen, John; McKeon, Andrew; Pittock, Sean J.; Flanagan, Eoin P.; Dubey, Divyanshu

doi:10.1111/epi.17424

Cited by 16 publications

(11 citation statements)

References 28 publications

(47 reference statements)

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“…Seizures are a common manifestation of AE [ 18 ], occurring in the early stages of approximately 70 percent of patients with NMDARE [ 19 ]. However, MOG-IgG has recently been associated with cortical encephalitis and seizures [ 20 ]. Seizures can arise in isolation in cases of single-antibody disorders and are also more frequently observed in male patients [ 1 , 21 , 22 ].…”

Section: Discussionmentioning

confidence: 99%

“…This study is consistent with previous results, sensitive to steroids, and has a good prognosis. Seizures caused by these autoimmune antibodies are usually self-limited or require short-term antiseizure medication and long-term antiseizure medication is not needed in most patients [ 19 , 20 ] (see Supplementary file 1 ). Some patients are prone to relapse during hormonal tapering [ 9 , 12 , 34 ], and failure to receive second-line and/or repeated first-line immunotherapy is associated with relapse [ 51 ].…”

Section: Discussionmentioning

confidence: 99%

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FLAMES overlaying anti-N-methyl-D-aspartate receptor encephalitis: a case report and literature review

Zhong,

Chen,

Liao

et al. 2024

BMC Neurol

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Background In recent years, simultaneous or sequential occurrence of MOG antibody disease and anti-NMDAR encephalitis in the same patient has been reported with increasing frequency. Scholars refer to the overlapping occurrence of these two disorders as MOG antibody disease and anti-NMDAR encephalitis overlap syndrome (MNOS). Cortical T2-weighted fluid-attenuated inversion recovery (FLAIR) -hyperintense lesions in anti-MOG-associated encephalitis with seizures (FLAMES) is a rare clinical phenotype of MOGAD in which cortical FLAIR high-signal lesions are unilateral, with little spread to the cortex and meninges bilaterally. Although cases of FLAMES have been consistently reported. However, to our knowledge, such cases of FLAMES combined with NMDARE are rare. Case presentation Here, we describe a case of FLAMES combined with anti-NMDARE. The patient was a young male, 29 years old, admitted to our hospital with isolated seizures, whose MRI showed unilateral thalamic and bilateral frontal and parietal leptomeningeal involvement. Since we were unaware of the possibility of bilateral meningo-cortical MOGAD manifestations, the case was initially diagnosed as viral encephalitis and was given antiviral therapy. The diagnosis was not clarified until anti-NMDAR-IgG and MOG-IgG positivity was detected in the cerebrospinal fluid and serum. The patient was then treated with high-dose corticosteroids and his symptoms responded well to the steroids. Therefore, this case expands the clinical spectrum of MNOS overlap syndrome. In addition, we describe the clinical features of MNOS by summarizing the existing literature and exploring the possible mechanisms of its immune response. Conclusions Our case serves as a reminder to clinicians that when patients present with atypical clinical manifestations such as seizures, consideration should be given to MNOS and conduct testing for various relevant autoantibodies (including MOG abs) and viruses in both serum and cerebrospinal fluid, as it is easy to misdiagnose the disease as other CNS diseases, such as viral meningoencephalitis. This syndrome exhibits a high responsiveness to steroids, highlighting the critical importance of recognizing the clinical and neuroimaging features of this overlap syndrome for prompt diagnosis and treatment. Furthermore, it enriches the disease spectrum of MNOS.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

FLAMES overlaying anti-N-methyl-D-aspartate receptor encephalitis: a case report and literature review

Zhong,

Chen,

Liao

et al. 2024

BMC Neurol

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“…A recent paper proposed to better characterize the seizures in the context of MOG-Ab positivity [43 ▪ ]. Unsurprisingly, seizures were mainly associated to CCE and ADEM presentations.…”

Section: Core Clinical Demyelinating Events: Classical Features and R...mentioning

confidence: 99%

Clinical characteristics of patients with myelin oligodendrocyte glycoprotein antibodies

Maillart,

Deiva,

Marignier

2024

Current Opinion in Neurology

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Purpose of review The clinical landscape associated to myelin oligodendrocyte glycoprotein antibodies (MOG-Ab) has undergone a remarkable transformation over the past two decades, primarily driven by advancements in antibody detection techniques that have enhanced both the specificity and sensitivity of assays, enabling the identification of novel clinical phenotypes. Recent findings Recent pivotal research publications, comprehensive reviews from established research groups, and most notably the first proposed international criteria for MOG-Ab associated disease (MOGAD) have substantially enriched our understanding of the clinical features associated with MOG-Ab. This review presents a comprehensive overview of the clinical characteristics of patients with MOG-Ab, systematically examining each core clinical syndrome defined by the proposed international MOGAD criteria. We incorporated recent insights and discussed potential challenges in applying these criteria across diverse clinical scenarios. Summary The proposed international MOGAD criteria provide a comprehensive, homogeneous, and specific framework for characterizing the clinical features of patients with MOG-Ab, encompassing both paediatric and adult populations. In the future, the widespread adoption of specific and reliable assays for MOG-Ab detection, complemented by the development of surrogate fluid and imaging markers, holds promise for better characterizing atypical presentations, only-cerebrospinal fluid positivity and the MOGAD “seronegative” situations.

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“…This study is consistent with previous results, sensitive to hormones, and has a good prognosis. Seizures caused by these autoimmune antibodies are usually self-limited or require short-term antiepileptic therapy and long-term antiepileptic therapy is not needed in most patients (23,24) ( see Supplementary le 1). Some patients are prone to relapse during hormonal tapering (12,15,45), and failure to receive second-line and/or repeated rst-line immunotherapy is associated with relapse(60).…”

Section: The Exact Mechanism Of the Positive Formation Of Double Anti...mentioning

confidence: 99%

Anti-NMDAR encephalitis with MOGAD overlap syndrome, isolated epilepsy, and unilateral thalamus changes: a case report and literature review

Zhong,

Chen,

Liao

et al. 2023

Preprint

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Background Anti-N-methyl-D-aspartate receptor (anti-NMDAR) encephalitis is an autoimmune brain disease mediated by antibodies and characterized by psychiatric symptoms, seizures, behavioral abnormalities, dyskinesia, and cognitive impairment. Recently, the co-existence of anti-NMDAR IgG and MOG IgG has been reported, and anti-NMDAR encephalitis has been found to occur concurrently or successively with demyelinating disease. With gradually increasing reports, the coexistence of anti-NMDAR-IgG and MOG-IgG has been noticed by scholars, and the combination of MOG antibody disease with anti-NMDAR encephalitis is called MOG antibody disease and anti-NMDAR encephalitis overlap syndrome (MNOS). MNOS is clinically rare, and due to its atypical clinical characteristics and its imaging manifestations, the disease is poorly understood, making clinical diagnosis difficult. Seizures are one of the most common atypical symptoms, and cases of mono antibody disease with bilateral thalamus symmetric involvement have also been reported, among which isolated seizures and MNOS cases of unilateral thalamus involvement have not been reported in the literature. Case presentation: A 29-year man was admitted to our hospital with isolated seizures, who had unilateral thalamic involvement and showed anti-NMDAR-IgG and MOG-IgG positivity in both cerebrospinal fluid and serum. His symptoms responded to immunotherapy and antiepileptic drugs with a good prognosis. To the best of our knowledge, this is the first case report of a double antibody-positive case with isolated seizures and unilateral thalamic involvement. Conclusions Our case is a reminder that we clinicians should think of MNOS when patients present with atypical clinical manifestations like epilepsy and test both serum and cerebrospinal fluid for multiple associated autoantibodies (including MOG antibodies) and viruses to avoid misdiagnosis, missed diagnosis, and impact on prognosis. In addition, it enriches the disease spectrum of MNOS syndrome.

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Acute symptomatic seizures secondary to myelin oligodendrocyte glycoprotein antibody‐associated disease

Cited by 16 publications

References 28 publications

FLAMES overlaying anti-N-methyl-D-aspartate receptor encephalitis: a case report and literature review

FLAMES overlaying anti-N-methyl-D-aspartate receptor encephalitis: a case report and literature review

Clinical characteristics of patients with myelin oligodendrocyte glycoprotein antibodies

Anti-NMDAR encephalitis with MOGAD overlap syndrome, isolated epilepsy, and unilateral thalamus changes: a case report and literature review

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