Autoimmune neurology: Co-occurrence of anti-NMDAR encephalitis and anti-MOG associated disease, report of a case

Caparó-Zamalloa, César; Álvarez-Toledo, Kelvin; Yamunaque-Chunga, César; Castro‐Suárez, Sheila; Guevara-Silva, Erik; Osorio-Marcatinco, Víctor; Meza‐Vega, Maria

doi:10.1016/j.jneuroim.2021.577663

Cited by 5 publications

(5 citation statements)

References 14 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…Overlap syndromes with the occurrence of MOG-IgG in patients with NMDAR encephalitis are a known but rare phenomenon and have been reported only in few patients [3][4][5][6][7][8][9][10][11][12][13][15][16][17][18][19][20][21]. The present case provides a longterm follow-up over more than 5 years with detailed, longitudinally collected, clinical and radiological findings as well as antibody dynamics.…”

Section: Discussionmentioning

confidence: 71%

See 1 more Smart Citation

Anti-NMDA receptor encephalitis and MOG-associated demyelination – a case report with long-term follow-up and a systematic review

et al. 2022

View full text Add to dashboard Cite

Background Overlap syndromes of anti-NMDA receptor encephalitis and MOG-mediated demyelination have been reported. In this case we provide a long-term longitudinal follow-up of clinical and imaging characteristics as well as of antibody dynamics. Case presentation We report a 32-year-old male patient who presented with psychosis, decreased consciousness and movement disorders and was tested positive for anti-NMDA receptor antibodies. Forty-four months after symptom onset and diagnosis of autoimmune encephalitis, he suffered from relapse. At this time, the patient developed anti-MOG and anti-Caspr2 antibodies. Treatment with plasmapheresis, steroids and rituximab eventually led to substantial clinical and radiological improvement. Anti-Caspr2 antibodies persisted, anti-NMDA receptor antibodies decreased, while anti-MOG antibodies turned negative again. Conclusion We provide long-term longitudinal follow-up of a patient with anti-NMDA receptor encephalitis who developed triple antibody positivity at the time of relapse. Antibody dynamics were associated with clinical disease course.

show abstract

Section: Discussionmentioning

confidence: 71%

“…Overlap syndromes of these two disease entities have been reported, i.e. either MOG immunoglobulin G (IgG) and demyelinating aspects have been found in patients with anti-NMDAR encephalitis [3][4][5][6][7][8][9][10][11][12][13][14][15][16][17][18][19][20][21], or vice versa NMDAR IgG in patients with demyelinating CNS disease [22].…”

Section: Introductionmentioning

confidence: 99%

Anti-NMDA receptor encephalitis and MOG-associated demyelination – a case report with long-term follow-up and a systematic review

et al. 2022

View full text Add to dashboard Cite

show abstract

“…Fifty-three relevant studies were identified after title and abstract screening. After reading the full texts and reviewing the references of the retrieved articles, 35 studies were finally included in the qualitative synthesis, of which 14 were retrospective studies [ 11 , 15 , 16 , 18 , 19 , 20 , 21 , 22 , 23 , 24 , 25 , 26 , 27 , 28 ] and 21 were case reports [ 12 , 13 , 29 , 30 , 31 , 32 , 33 , 34 , 35 , 36 , 37 , 38 , 39 , 40 , 41 , 42 , 43 , 44 , 45 , 46 , 47 ]. A total of 113 patients (46 males and 67 females) were reported to show the coexistence of MOG-IgG and neuronal or glial antibodies in these 35 studies.…”

Section: Resultsmentioning

confidence: 99%

Coexistence of Myelin Oligodendrocyte Glycoprotein Immunoglobulin G and Neuronal or Glial Antibodies in the Central Nervous System: A Systematic Review

et al. 2022

View full text Add to dashboard Cite

Background: Myelin oligodendrocyte glycoprotein immunoglobulin G (MOG-IgG) has been considered a diagnostic marker for patients with demyelinating disease, termed “MOG-IgG associated disorder” (MOGAD). Recently, the coexistence of MOG-IgG and other neuronal or glial antibodies has attracted extensive attention from clinicians. In this article, we systematically review the characteristics of MOG-IgG-related antibody coexistence syndrome. Methods: Two authors independently searched PubMed for relevant studies published before October 2021. We also manually searched the references of each related article. The appropriateness of the included studies was assessed by reading the titles, abstracts, and full texts if necessary. Results: Thirty-five relevant publications that met our inclusion criteria were finally included, of which fourteen were retrospective studies and twenty-one were case reports. A total of 113 patients were reported to show the coexistence of MOG-IgG and neuronal or glial antibodies. Additionally, 68.14% of patients were double positive for MOG-IgG and N-Methyl-D-Aspartate Receptor-IgG (NMDAR-IgG), followed by 23.01% of patients who were double positive for MOG-IgG and aquaporin4-IgG (AQP4-IgG). Encephalitis was the predominant phenotype when MOG-IgG coexisted with NMDAR-IgG, probably accompanied by imaging features of demyelination. Patients with dual positivity for MOG-IgG and AQP4-IgG experienced more severe disease and more frequent relapses. The coexistence of MOG-IgG and antibodies other than NMDAR-IgG and AQP4-IgG was extremely rare, and the clinical presentations were diverse and atypical. Except for patients who were double positive for MOG-IgG and AQP4-IgG, most patients with multiple antibodies had a good prognosis. Conclusions: MOG-IgG may coexist with neuronal or glial antibodies. Expanded screening for neuronal or glial antibodies should be performed in patients with atypical clinical and radiological features.

show abstract

“… 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%

“…This overlap could be categorized as one of the following chronological phenotypes: (1) cases that start with a presentation of anti-NMDAR-EN who develop demyelinating events consistent with MOGAD later; (2) cases with a known diagnosis of MOGAD who later present with anti-NMDAR-EN; and (3) cases of autoimmune encephalitis with concurrent positive MOG-IgG and anti-NMDAR-IgG, demonstrating that antibodies against NMDAR and MOG can be simultaneously detected in one patient. 10 , 54 One possible explanation could be an immunological activation (e.g. an infection) driving the production of different subclasses of neuronal autoantibodies (anti-MOG-IgG, anti-NMDAR-IgG), resulting in the development of two different diseases.…”

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

View full text Add to dashboard Cite

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.

show abstract

Autoimmune neurology: Co-occurrence of anti-NMDAR encephalitis and anti-MOG associated disease, report of a case

Cited by 5 publications

References 14 publications

Anti-NMDA receptor encephalitis and MOG-associated demyelination – a case report with long-term follow-up and a systematic review

Anti-NMDA receptor encephalitis and MOG-associated demyelination – a case report with long-term follow-up and a systematic review

Coexistence of Myelin Oligodendrocyte Glycoprotein Immunoglobulin G and Neuronal or Glial Antibodies in the Central Nervous System: A Systematic Review

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

Contact Info

Product

Resources

About