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      A narrative review of the economic burden of myelin oligodendrocyte glycoprotein antibody-associated disease and analogous conditions

      systematic-review

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          Abstract

          Background

          Myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) is an inflammatory demyelinating disease of the central nervous system with a serious, debilitating presentation, including residual disability after relapses.

          Objectives

          To evaluate the economic impacts of MOGAD and analogous conditions, including direct costs, indirect costs and cost drivers.

          Design

          Systematic literature search and narrative review.

          Data sources and methods

          Search strings were designed to capture any study reporting health economic impacts of MOGAD or analogous autoimmune diseases of the nervous system. The costs of diagnostic tests and short- and long-term interventions were considered, and studies from both patient and institutional (public and private) perspectives were included. Searches were conducted using medical subject headings (MeSH) in PubMed in July 2023. Retrieved publications were screened initially based on title and abstract, then based on the full text. Data were extracted manually; findings are reported descriptively. All cost data were adjusted to 2024 US Dollars using the CCEMG-EPPI-Centre Cost Converter.

          Results

          Results from 40 studies of MOGAD and analogous autoimmune neurological conditions were extracted. In the only study that included patients with MOGAD (a cost investigation from Germany in which 166 patients had neuromyelitis optica spectrum disorder and 46 had MOGAD), the mean annualised cost of illness was $94,688 (direct medical costs 43%, direct non-medical costs 34%, indirect costs 23%). Across the conditions assessed, the annual total cost of illness per patient ranged widely, from $3,690 to $507,117 (among studies that reported types of cost, the range for direct costs was $1,981–$148,388; for indirect costs, $0–$942,707). The study that included patients with MOGAD identified the need for care, number of acute attacks, unemployment and disability as independent predictors of cost. Additional cost drivers (from all the conditions) included treatments (e.g., intravenous immunoglobulin), hospitalisation, disease severity, relapses and refractory disease.

          Conclusion

          Our search identified only one study that specifically examined costs associated with MOGAD. Results from this and studies of analogous autoimmune conditions suggest that inflammatory demyelinating diseases of the central nervous system including MOGAD are costly for the individual patient and place considerable burden on healthcare systems. Further evidence is needed for increased insight into the economic burden of MOGAD.

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          Most cited references67

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          The PRISMA 2020 statement: an updated guideline for reporting systematic reviews

          The Preferred Reporting Items for Systematic reviews and Meta-Analyses (PRISMA) statement, published in 2009, was designed to help systematic reviewers transparently report why the review was done, what the authors did, and what they found. Over the past decade, advances in systematic review methodology and terminology have necessitated an update to the guideline. The PRISMA 2020 statement replaces the 2009 statement and includes new reporting guidance that reflects advances in methods to identify, select, appraise, and synthesise studies. The structure and presentation of the items have been modified to facilitate implementation. In this article, we present the PRISMA 2020 27-item checklist, an expanded checklist that details reporting recommendations for each item, the PRISMA 2020 abstract checklist, and the revised flow diagrams for original and updated reviews.
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            MOG encephalomyelitis: international recommendations on diagnosis and antibody testing

            Over the past few years, new-generation cell-based assays have demonstrated a robust association of autoantibodies to full-length human myelin oligodendrocyte glycoprotein (MOG-IgG) with (mostly recurrent) optic neuritis, myelitis and brainstem encephalitis, as well as with acute disseminated encephalomyelitis (ADEM)-like presentations. Most experts now consider MOG-IgG-associated encephalomyelitis (MOG-EM) a disease entity in its own right, immunopathogenetically distinct from both classic multiple sclerosis (MS) and aquaporin-4 (AQP4)-IgG-positive neuromyelitis optica spectrum disorders (NMOSD). Owing to a substantial overlap in clinicoradiological presentation, MOG-EM was often unwittingly misdiagnosed as MS in the past. Accordingly, increasing numbers of patients with suspected or established MS are currently being tested for MOG-IgG. However, screening of large unselected cohorts for rare biomarkers can significantly reduce the positive predictive value of a test. To lessen the hazard of overdiagnosing MOG-EM, which may lead to inappropriate treatment, more selective criteria for MOG-IgG testing are urgently needed. In this paper, we propose indications for MOG-IgG testing based on expert consensus. In addition, we give a list of conditions atypical for MOG-EM (“red flags”) that should prompt physicians to challenge a positive MOG-IgG test result. Finally, we provide recommendations regarding assay methodology, specimen sampling and data interpretation.
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              Diagnosis of myelin oligodendrocyte glycoprotein antibody-associated disease: International MOGAD Panel proposed criteria

                Author and article information

                Contributors
                URI : https://loop.frontiersin.org/people/2860584/overviewRole: Role: Role: Role: Role: Role: Role:
                Role: Role: Role: Role: Role:
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                URI : https://loop.frontiersin.org/people/3014071/overviewRole: Role: Role: Role: Role: Role: Role:
                Journal
                Front Neurol
                Front Neurol
                Front. Neurol.
                Frontiers in Neurology
                Frontiers Media S.A.
                1664-2295
                30 May 2025
                2025
                : 16
                : 1506465
                Affiliations
                [1] 1Centre for Applied Health Economics, School of Medicine and Dentistry, Griffith University , Gold Coast, QLD, Australia
                [2] 2UCB Inc. , Morrisville, NC, United States
                Author notes

                Edited by: Omid Mirmosayyeb, University at Buffalo, United States

                Reviewed by: Thanos Tsaktanis, University of Erlangen Nuremberg, Germany

                Mohammad Yazdan Panah, Shahrekord University of Medical Sciences, Iran

                Roberto Carlos Lyra Da Silva, Rio de Janeiro State Federal University, Brazil

                *Correspondence: Leonard Lee, leonard.lee@ 123456griffith.edu.au
                Article
                10.3389/fneur.2025.1506465
                12164171
                40520606
                e230ceb0-626c-4dd1-ab69-cadc33f9e473
                Copyright © 2025 Lee, Byrnes, Hope and Kim.

                This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

                History
                : 05 October 2024
                : 09 April 2025
                Page count
                Figures: 3, Tables: 4, Equations: 0, References: 73, Pages: 35, Words: 14160
                Funding
                The author(s) declare that financial support was received for the research and/or publication of this article. This study was funded by UCB. The funder was not involved in the study design, collection, analysis, interpretation of data, the writing of this article, or the decision to submit it for publication.
                Categories
                Neurology
                Systematic Review
                Custom metadata
                Multiple Sclerosis and Neuroimmunology

                Neurology
                central nervous system,economic burden,disease costs,inflammatory demyelinating disease,mogad

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