Case Report: Overlapping Multiple Sclerosis With Anti-N-Methyl-D-Aspartate Receptor Encephalitis: A Case Report and Review of Literature

Huang, Ying; Qian, Wang; Zeng, Silin; Zhang, Yaqing; Zou, Lina; Fu, Xuejun; Xu, Qianhui

doi:10.3389/fimmu.2020.595417

Cited by 18 publications

(16 citation statements)

References 19 publications

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“…These finding suggest the existence of two simultaneously active immune mechanisms at the same time. We found two cases of overlapping syndrome in our study, one with MS [18], and the other with a positive MOG antibody. Demyelination-related diseases, including AQP-4 antibody and MOG antibody-related and MS, may be a potential cause of anti-NMDAR encephalitis.…”

Section: Discussionsupporting

confidence: 50%

Clinical features, treatments, and outcomes of patients with anti-N-methyl-D-aspartate encephalitis—a single-center, retrospective analysis in China

Zhu

Wang

et al. 2021

Front Biosci (Landmark Ed)

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Introduction 3. Materials and methods 3.1 Study subjects 3.2 Data collection 3.3 Assessment of prognosis 3.4 Statistical analysis 4. Results 4.1 General characteristics and clinical features 4.2 Ancillary examination 4.3 Diagnosis and treatment 4.4 Prognosis and relapse 5. Discussion 6. Conclusions 7. Author contributions 8. Ethics approval and consent to participate 9. Acknowledgment 10. Funding 11. Conflict of interest 12. References

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Section: Discussionsupporting

confidence: 50%

Clinical features, treatments, and outcomes of patients with anti-N-methyl-D-aspartate encephalitis—a single-center, retrospective analysis in China

Zhu

Wang

et al. 2021

Front Biosci (Landmark Ed)

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“…Some are positive for MOG-or AQP4-antibodies, while others remain seronegative. [32][33][34] Other patients have anti-NMDAR encephalitis that overlaps with MS. [35][36][37][38][39][40][41][42][43] In our patient, brain MRI was initially unremarkable and demyelinating lesions were first found 4 years after the onset of NMDAR-encephalitis and showed imaging features more typical of MS than NMOSD or MOG-antibody-associated disease. The mechanisms that trigger overlapping NMDAR-encephalitis and demyelination are unclear and the pathogenesis may be heterogeneous.…”

Section: Discussionmentioning

confidence: 55%

Neuropathological Variability within a Spectrum of NMDAR‐Encephalitis

Zrzavy

Endmayr

Bauer

et al. 2021

Annals of Neurology

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Objective: To describe the neuropathological features of N-methyl-D-aspartate receptor (NMDAR)-encephalitis in an archival autopsy cohort. Methods: We examined four autopsies from patients with NMDAR-encephalitis; two patients were untreated, three had comorbidities: small cell lung cancer, brain post-transplant lymphoproliferative disease (PTLD), and overlapping demyelination. Results: The two untreated patients had inflammatory infiltrates predominantly composed of perivascular and parenchymal CD3 + /CD8 À T cells and CD79a + B cells/plasma cells in basal ganglia, amygdala, and hippocampus with surrounding white matter. The hippocampi showed a significant decrease of NMDAR-immunoreactivity that correlated with disease severity. The patient with NMDAR-encephalitis and immunosuppression for kidney transplantation developed a brain monomorphic PTLD. Inflammatory changes were compatible with NMDAR-encephalitis. Additionally, plasma cells accumulated in the vicinity of the necrotic tumor along with macrophages and activated microglia that strongly expressed pro-inflammatory activation markers HLA-DR, CD68, and IL18. The fourth patient developed demyelinating lesions in the setting of a relapse 4 years after NMDAR-encephalitis. These lesions exhibited the hallmarks of classic multiple sclerosis with radially expanding lesions and remyelinated shadow plaques without complement or immunoglobulin deposition, compatible with a pattern I demyelination. Interpretation: The topographic distribution of inflammation in patients with NMDAR-encephalitis reflects the clinical symptoms of movement disorders, abnormal behavior, and memory dysfunction with inflammation dominantly observed in basal ganglia, amygdala, and hippocampus, and loss of NMDAR-immunoreactivity correlates with disease severity. Co-occurring pathologies influence the spatial distribution, composition, and intensity of inflammation, which may modify patients' clinical presentation and outcome.

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“…Undeniably, the differential diagnosis of multiple intracranial lesions is a challenging task, especially when the clinical features are neither specific nor unique, or laboratory tests do not correspond to clinical symptoms. We reviewed all case reports on MS and anti-NMDAR encephalitis, and the results of the literature review, containing the first case from England ( 21), along with one from Japan (22), two from Germany (8) (23), three from England (24) (25), one from Turkey (26), one from Austria (19), and one from China (27), are presented in Table 2. Through collation of previous cases, we found that only two male patient out of 10 had co-existence of anti-NMDAR encephalitis and MS, a proportion higher than the male-to-female ratio in MS (28) and anti-NMDAR encephalitis (29).…”

Section: Anti-nmdar Encephalitis and Multiple Sclerosismentioning

confidence: 99%

“…In conclusion, neurologists should notice that anti-neuronal antibodies in demyelinating disorders can lead to atypical symptoms and ambiguous diagnosis, which might result in the inaccurate and untimely treatment of patients and affect the prognosis and quality of life of patients (36). Additionally, the molecular mimicry and the breakdown of immunologic tolerance toward NMDAR released following neuronal damage have been described as possible hypothesis for pathogenesis, but the mechanisms connecting anti-NMDAR antibody with demyelinating disorders remain to be further explored (27).…”

Section: Anti-nmdar Encephalitis and Multiple Sclerosismentioning

confidence: 99%

Case Report: Antibodies to the N-Methyl-D-Aspartate Receptor in a Patient With Multiple Sclerosis

et al. 2021

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The association between multiple sclerosis and anti-N-Methyl-D-Aspartate receptor encephalitis is limited to merely a few case reports, and the exploration of the pathogenic mechanisms underlying the overlap of these two disease entities is very limited. Therefore, case reports and literature review on N-Methyl-D-aspartate receptor antibody in patients with multiple sclerosis are unusual and noteworthy. A young female had the first episode of paresthesia and motor symptoms with positive anti-N-Methyl-D-Aspartate receptor antibody and recovered after immunotherapy, and at the first relapse, the patient developed disorders of consciousness with positive anti-N-Methyl-D-Aspartate receptor antibody, findings of magnetic resonance imaging showed features of autoimmune encephalitis, which was also controlled by immunotherapy. At the second relapse, anti-N-Methyl-D-Aspartate receptor antibody turned negative while oligoclonal bands presented positive, and findings of magnetic resonance imaging showed features of multiple sclerosis. Afterwards, we followed the patient after receiving disease modifying treatment to monitor the efficacy and safety of teriflunomide. Based on literature review, demyelinating diseases patients with anti-neuronal antibody have complex, diverse and atypical symptoms; therefore, high attention and increased alertness are necessary for neurologists. Conclusively, anti-neuronal antibody may present in many neuroinflammatory conditions, and diagnostic criteria should be used with caution if the clinical presentation is atypical, and neurologists should not rely excessively on laboratory tests to diagnose neurological diseases. Timely and comprehensive examination and consideration as well as early standardized treatment are the key factors to reduce patient recurrence and obtain a good prognosis.

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Case Report: Overlapping Multiple Sclerosis With Anti-N-Methyl-D-Aspartate Receptor Encephalitis: A Case Report and Review of Literature

Cited by 18 publications

References 19 publications

Clinical features, treatments, and outcomes of patients with anti-N-methyl-D-aspartate encephalitis—a single-center, retrospective analysis in China

Clinical features, treatments, and outcomes of patients with anti-N-methyl-D-aspartate encephalitis—a single-center, retrospective analysis in China

Neuropathological Variability within a Spectrum of NMDAR‐Encephalitis

Case Report: Antibodies to the N-Methyl-D-Aspartate Receptor in a Patient With Multiple Sclerosis

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