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

There is no author summary for this article yet. Authors can add summaries to their articles on ScienceOpen to make them more accessible to a non-specialist audience.

Abstract

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.

Related collections

Most cited references 145

Record: found
Abstract: found
Article: not found

Treatment and prognostic factors for long-term outcome in patients with anti-NMDA receptor encephalitis: an observational cohort study.

Maarten J. Titulaer, Lindsey McCracken, Iñigo Gabilondo … (2013)

Anti-NMDA receptor (NMDAR) encephalitis is an autoimmune disorder in which the use of immunotherapy and the long-term outcome have not been defined. We aimed to assess the presentation of the disease, the spectrum of symptoms, immunotherapies used, timing of improvement, and long-term outcome. In this multi-institutional observational study, we tested for the presence of NMDAR antibodies in serum or CSF samples of patients with encephalitis between Jan 1, 2007, and Jan 1, 2012. All patients who tested positive for NMDAR antibodies were included in the study; patients were assessed at symptom onset and at months 4, 8, 12, 18, and 24, by use of the modified Rankin scale (mRS). Treatment included first-line immunotherapy (steroids, intravenous immunoglobulin, plasmapheresis), second-line immunotherapy (rituximab, cyclophosphamide), and tumour removal. Predictors of outcome were determined at the Universities of Pennsylvania (PA, USA) and Barcelona (Spain) by use of a generalised linear mixed model with binary distribution. We enrolled 577 patients (median age 21 years, range 8 months to 85 years), 211 of whom were children (<18 years). Treatment effects and outcome were assessable in 501 (median follow-up 24 months, range 4-186): 472 (94%) underwent first-line immunotherapy or tumour removal, resulting in improvement within 4 weeks in 251 (53%). Of 221 patients who did not improve with first-line treatment, 125 (57%) received second-line immunotherapy that resulted in a better outcome (mRS 0-2) than those who did not (odds ratio [OR] 2·69, CI 1·24-5·80; p=0·012). During the first 24 months, 394 of 501 patients achieved a good outcome (mRS 0-2; median 6 months, IQR 2-12) and 30 died. At 24 months' follow-up, 203 (81%) of 252 patients had good outcome. Outcomes continued to improve for up to 18 months after symptom onset. Predictors of good outcome were early treatment (0·62, 0·50-0·76; p<0·0001) and no admission to an intensive care unit (0·12, 0·06-0·22; p<0·0001). 45 patients had one or multiple relapses (representing a 12% risk within 2 years); 46 (67%) of 69 relapses were less severe than initial episodes (p<0·0001). In 177 children, predictors of good outcome and the magnitude of effect of second-line immunotherapy were similar to those of the entire cohort. Most patients with anti-NMDAR encephalitis respond to immunotherapy. Second-line immunotherapy is usually effective when first-line treatments fail. In this cohort, the recovery of some patients took up to 18 months. The Dutch Cancer Society, the National Institutes of Health, the McKnight Neuroscience of Brain Disorders award, The Fondo de Investigaciones Sanitarias, and Fundació la Marató de TV3. Copyright © 2013 Elsevier Ltd. All rights reserved.

0 comments Cited 530 times – based on 0 reviews      Review now

Bookmark

Record: found
Abstract: found
Article: not found

Autoimmune encephalitis epidemiology and a comparison to infectious encephalitis

Divyanshu Dubey, Sean Pittock, Cecilia R. Kelly … (2018)

Objectives We evaluate incidence and prevalence of autoimmune encephalitis and compare the epidemiology of autoimmune and infectious encephalitis. Methods We performed a population-based comparative study of the incidence and prevalence of autoimmune and infectious encephalitis in Olmsted County, USA. Autoimmune encephalitis diagnosis and subgroups were defined by 2016 diagnostic criteria and infectious encephalitis diagnosis required a confirmed infectious pathogen. Age- and sex-adjusted prevalence and incidence rates were calculated. Patients with encephalitis of uncertain etiology were excluded. Results The prevalence of autoimmune encephalitis on January 1, 2014 of 13.7/100,000 was not significantly different from that of all infectious encephalitides (11.6/100,000; p=0.63) or the viral subcategory (8.3/100,000; p=0.17). The incidence rates (1995–2015) of autoimmune and infectious encephalitis were 0.8/100,000 and 1.0/100,000 person-years respectively (p=0.58). The number of relapses or recurrent hospitalizations was higher for autoimmune than infectious encephalitis (p=0.03). The incidence of autoimmune encephalitis increased over time from 0.4/100,000 person-years (1995–2005) to 1.2/100,000 person-years (2006–2015) (p=0.02), attributable to increased recognition of autoantibody-positive cases. The incidence (2.8 vs 0.7/100,000 person-years; p=0.01) and prevalence (38.3 vs 13.7/100,000; p=0.04) of autoimmune encephalitis was higher among African-Americans than Caucasians. The prevalence of specific neural autoantibodies was: myelin-oligodendrocyte-glycoprotein (MOG) (1.9/100,000); glutamic acid decarboxylase-65 (GAD65) (1.9/100,000); unclassified neural autoantibody (1.4/100,000); leucine-rich glioma-inactivated-protein-1 (LGI1) (0.7/100,000); collapsin response-mediator protein-5 (CRMP5) (0.7/100,000); N-methyl-D-aspartate-receptor (NMDAR) (0.6/100,000); anti-neuronal nuclear antibody-2 (ANNA-2/anti-Ri) (0.6/100,000) and glial fibrillary acidic protein-α (GFAPα) (0.6/100,000). Interpretation This study shows that the prevalence and incidence of autoimmune encephalitis is comparable to infectious encephalitis and its detection is increasing over time.

0 comments Cited 201 times – based on 0 reviews      Review now

Bookmark

Record: found
Abstract: found
Article: found

Is Open Access

Clinical course, therapeutic responses and outcomes in relapsing MOG antibody-associated demyelination

Sudarshini Ramanathan, Shekeeb S. Mohammad, Esther Tantsis … (2017)

Objective We characterised the clinical course, treatment and outcomes in 59 patients with relapsing myelin oligodendrocyte glycoprotein (MOG) antibody-associated demyelination. Methods We evaluated clinical phenotypes, annualised relapse rates (ARR) prior and on immunotherapy and Expanded Disability Status Scale (EDSS), in 218 demyelinating episodes from 33 paediatric and 26 adult patients. Results The most common initial presentation in the cohort was optic neuritis (ON) in 54% (bilateral (BON) 32%, unilateral (UON) 22%), followed by acute disseminated encephalomyelitis (ADEM) (20%), which occurred exclusively in children. ON was the dominant phenotype (UON 35%, BON 19%) of all clinical episodes. 109/226 (48%) MRIs had no brain lesions. Patients were steroid responsive, but 70% of episodes treated with oral prednisone relapsed, particularly at doses <10 mg daily or within 2 months of cessation. Immunotherapy, including maintenance prednisone (P=0.0004), intravenous immunoglobulin, rituximab and mycophenolate, all reduced median ARRs on-treatment. Treatment failure rates were lower in patients on maintenance steroids (5%) compared with non-steroidal maintenance immunotherapy (38%) (P=0.016). 58% of patients experienced residual disability (average follow-up 61 months, visual loss in 24%). Patients with ON were less likely to have sustained disability defined by a final EDSS of ≥2 (OR 0.15, P=0.032), while those who had any myelitis were more likely to have sustained residual deficits (OR 3.56, P=0.077). Conclusion Relapsing MOG antibody-associated demyelination is strongly associated with ON across all age groups and ADEM in children. Patients are highly responsive to steroids, but vulnerable to relapse on steroid reduction and cessation.

0 comments Cited 190 times – based on 0 reviews      Review now

Bookmark

All references

Author and article information

Contributors

Negar Molazadeh:

ORCID: https://orcid.org/0000-0003-0861-2178

Gauruv Bose

Journal

Journal ID (nlm-ta): Mult Scler J Exp Transl Clin

Journal ID (iso-abbrev): Mult Scler J Exp Transl Clin

Journal ID (publisher-id): MSO

Journal ID (hwp): spmso

Title: Multiple Sclerosis Journal - Experimental, Translational and Clinical

Publisher: SAGE Publications (Sage UK: London, England )

ISSN (Electronic): 2055-2173

Publication date (Electronic): 20 October 2022

Publication date Collection: Oct-Dec 2022

Volume: 8

Issue: 4

Electronic Location Identifier: 20552173221128170

Affiliations

[1-20552173221128170]Department of Neurology, Ringgold 2348, universityMassachusetts General Hospital; , Boston, MA, USA

[2-20552173221128170]Ringgold 1811, universityHarvard Medical School; , Boston, MA, USA

[4-20552173221128170]Brigham MS Center, Department of Neurology, Ringgold 1861, universityBrigham and Women’s Hospital; , Boston, MA, USA

[3-20552173221128170]Ringgold 1811, universityHarvard Medical School; , Boston, MA, USA

[5-20552173221128170]Division of Neurology, Department of Medicine, The Ottawa Hospital and University of Ottawa; Ottawa Hospital Research Institute, ON, Canada

[6-20552173221128170]Department of Neurology, Ringgold 2348, universityMassachusetts General Hospital; , Boston, MA, USA

[7-20552173221128170]Ringgold 1811, universityHarvard Medical School; , Boston, MA, USA

Author notes

[*]Negar Molazadeh, Neuromyelitis Optica Research Laboratory, Division of Neuroimmunology & Neuroinfectious Disease, Department of Neurology, Massachusetts General Hospital, Building 114, 16th St, Room 3150, Charlestown, MA 02129, USA. nmolazadeh@ 123456mgh.harvard.edu Twitter: http://twitter.com/NegarMowlazadeh

Author information

Negar Molazadeh https://orcid.org/0000-0003-0861-2178

Michael Levy https://orcid.org/0000-0002-7969-8346

Article

Publisher ID: 10.1177_20552173221128170

DOI: 10.1177/20552173221128170

PMC ID: 9597055

PubMed ID: 36311694

SO-VID: f10d585f-5fdb-49d4-b051-ec3b59167f35

License:

This article is distributed under the terms of the Creative Commons Attribution-NonCommercial 4.0 License ( https://creativecommons.org/licenses/by-nc/4.0/) which permits non-commercial use, reproduction and distribution of the work without further permission provided the original work is attributed as specified on the SAGE and Open Access page ( https://us.sagepub.com/en-us/nam/open-access-at-sage).

History

Date received : 28 April 2022

Date accepted : 6 September 2022

Custom metadata

typesetter ts19

cover-date October-December 2022

Keywords: myelin oligodendrocyte glycoprotein,autoimmune disease,autoantibody,neoplasm,paraneoplastic,systematic review

Data availability:

Keywords: myelin oligodendrocyte glycoprotein, autoimmune disease, autoantibody, neoplasm, paraneoplastic, systematic review

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

Read this article at

Abstract

Background

Methods

Results

Conclusion

Related collections

Genes & Diseases

Most cited references 145

Treatment and prognostic factors for long-term outcome in patients with anti-NMDA receptor encephalitis: an observational cohort study.

Autoimmune encephalitis epidemiology and a comparison to infectious encephalitis

Clinical course, therapeutic responses and outcomes in relapsing MOG antibody-associated demyelination

Author and article information

Contributors

Journal

Affiliations

Author notes

Author information

Article

History

Categories

Custom metadata

Comments

Comment on this article

Similar content 454

Cited by 9

Most referenced authors 1,462