Relapsing MOG-IgG-associated diseases coexisting with anti-NMDAR encephalitis: a case report and literature review

Wang, Mengmeng; Tan, Jun; Zhou, Zhiqiang; Wang, Yihan; Bako, Samira Yerima; Yang, Yaoqiang; Lin, Yingbin

doi:10.31083/j.jin2103082

Cited by 9 publications

(15 citation statements)

References 30 publications

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“…Finally, some case reports have shown that virus infection may be associated to the production of double antibodies in MNOS patients (10,38). Wang et al believed that virus invasion cause damage to the blood-brain barrier exposed to MOG ags and produced corresponding antibodies, leading to increased damage to the bloodbrain barrier, NMDAR ags exposure, and NMDAR abs production (21). This is similar to the mechanism of virus induced MOG-AD hypothesis proposed by Cao et al in the context of dual antibody positivity (19).…”

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

confidence: 78%

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

confidence: 78%

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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show abstract

“…The MRI of patients with MNOS overlapped more with MOGAD [ 25 , 28 ], and had a higher proportion of supratentorial lesions [ 17 , 28 ], with frequent involvement of the cortex, subcortical white matter, and basal ganglia regions. The results of a study of 58 double antibody-positive patients also showed that the cortex was the most common MRI change and found that the leptomeninges were more likely to be violated in patients with MNOS [ 25 ].…”

Section: Discussionmentioning

confidence: 99%

“…Finally, some case reports have shown that virus infection may be associated to the production of double antibodies in MNOS patients [ 7 , 38 ]. Wang et al believed that virus invasion cause damage to the blood–brain barrier exposed to MOG ags and produced corresponding antibodies, leading to increased damage to the blood–brain barrier, NMDAR ags exposure, and NMDAR abs production [ 28 ]. This is similar to the mechanism of virus induced MOG-AD hypothesis proposed by Cao et al in the context of dual antibody positivity [ 16 ].…”

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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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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“… 3 This finding implies a difference in the immunopathogenic mechanisms involved in the development of each entity, such that AQP4-IgG + NMOSD is more attributable to a defect in immune tolerance, while MOGAD is more likely to be an aberrant autoimmune response to certain triggers, such as infection, vaccination, or malignancy. 3 …”

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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Relapsing MOG-IgG-associated diseases coexisting with anti-NMDAR encephalitis: a case report and literature review

Cited by 9 publications

References 30 publications

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

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

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

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

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