Department of Women's and Children's Health, Karolinska Vagen 37A, 171 76, Stockholm, Sweden. email@example.com. Absolute Antibody Ltd., Wilton Centre, Redcar, TS10 4RF, UK. Department of Women's and Children's Health, Karolinska Vagen 37A, 171 76, Stockholm, Sweden. Department of Learning, Informatics, Management and Ethics, Karolinska Institutet, Stockholm, Sweden. Division of Neurology, Department of Medicine, The Ottawa Hospital, and Ottawa Hospital Research Institute, University of Ottawa, Ottawa, Canada. Department of Neuropediatrics and Muscle Disorders, Medical Centre-University of Freiburg, Faculty of Medicine, Freiburg, Germany. Children's Hospital of Eastern Ontario Research Institute, University of Ottawa, Ottawa, Canada.
OBJECTIVES The objective of our study was to conduct a systematic literature review of economic costs (henceforth costs) associated with myasthenia gravis (MG). METHODS We searched MEDLINE (through PubMed), CINAHL, Embase, PsycINFO, and Web of Science for studies reporting costs of MG published from inception up until March 18, 2020, without language restrictions. Two reviewers independently screened records for eligibility,
extracted the data, and assessed included studies for risk of bias using the Newcastle-Ottawa Scale. Costs were inflated and converted to 2018 United States dollars ($). RESULTS The search identified 16 articles for data extraction and synthesis. Estimates of costs of MG were found for samples from eight countries spanning four continents (Europe, North America, South America, and Asia). Across studies, the mean per-patient annual direct medical cost of illness was estimated at between $760 and $28,780, and cost per hospitalization between $2550 and $164,730. The indirect cost of illness was estimated at $80 and $3550. Costs varied considerably by patient characteristics, and drivers of the direct medical cost of illness included intravenous immunoglobulin and plasma exchange, myasthenic crisis, mechanical ventilatory support, and hospitalizations. CONCLUSIONS We show that the current body of literature of costs of MG is sparse, limited to a few geographical settings and resource categories, mostly dated, and subject to non-trivial variability, both within and between countries. Our synthesis will help researchers and decision-makers identify gaps in the local health economic context of MG and inform future cost studies and economic evaluations in this patient population.