{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,2]],"date-time":"2026-07-02T18:22:05Z","timestamp":1783016525396,"version":"3.54.6"},"reference-count":23,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2022,3,3]],"date-time":"2022-03-03T00:00:00Z","timestamp":1646265600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Public Health"],"abstract":"<jats:sec><jats:title>Background<\/jats:title><jats:p>Myasthenia gravis (MG) is a chronic autoimmune disorder, which is characterized by fatigable muscle weakness with frequent ocular signs and\/or generalized muscle fatigue, and occasionally associated with thymoma. MG patients and their families face a significant socio-economic burden. This population is often experiencing unemployment, unwilling job transfers and decreased income.<\/jats:p><\/jats:sec><jats:sec><jats:title>Objective<\/jats:title><jats:p>This study aimed to estimate the annual costs from a societal perspective in a triple dimension of direct health care costs, direct non-health care costs (formal and informal care) and labor productivity losses in MG patients from Bulgaria, as well as to identify the main clinical and demographical cost drivers.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>A bottom-up, cross-sectional, cost-of-illness analysis of 54 adult MG patients was carried out in 2020. To collect data on demographic characteristics, health resource utilization, informal care and productivity losses, questionnaires were administered to and completed by patients.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results and Conclusion<\/jats:title><jats:p>Median annual costs of MG in Bulgaria were 4,047 EUR per patient. Direct costs slightly outweighed indirect costs, with drugs cost item having the biggest monetary impact. Despite the zero-inflated median, hospitalizations also influenced the direct costs by an estimated amount of 1,512 EUR in the 3rd quartile. Social services and professional caregiver costs were found to be almost missing, with the vast majority of patients reporting reliance on informal caregivers. Severe generalized disease, disease crises, and recurrent infections were confirmed as statistically significant cost driving factors. There were no severe generalized MG patients in the bottom quartile of the total costs distribution. It should be noted that in both cases of crises or infections, the overall increase in the total costs was mainly due to higher indirect costs observed. Reliance on family members as informal caregivers is routine among Bulgarian MG patients. This phenomenon is likely due to the lack of access to appropriate social services. Moreover, it is directly related with higher disease burden and significant inequalities. There is a need for further research on MG in Bulgaria in order to design targeted health policies that meet the needs and expectations of these patients.<\/jats:p><\/jats:sec>","DOI":"10.3389\/fpubh.2022.822909","type":"journal-article","created":{"date-parts":[[2022,3,3]],"date-time":"2022-03-03T06:24:20Z","timestamp":1646288660000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":20,"title":["Socio-Economic Burden of Myasthenia Gravis: A Cost-of-Illness Study in Bulgaria"],"prefix":"10.3389","volume":"10","author":[{"given":"Valentina","family":"Ignatova","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kostadin","family":"Kostadinov","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Evguenia","family":"Vassileva","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Naira","family":"Muradyan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Georgi","family":"Stefanov","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Georgi","family":"Iskrov","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rumen","family":"Stefanov","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2022,3,3]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","first-page":"874680","DOI":"10.1155\/2012\/874680","article-title":"Myasthenia gravis: a review","volume":"2012","author":"Jayam Trouth","year":"2012","journal-title":"Autoimmune Dis."},{"key":"B2","doi-asserted-by":"publisher","first-page":"90","DOI":"10.1016\/j.jaut.2013.12.011","article-title":"Myasthenia gravis: a comprehensive review of immune dysregulation and etiological mechanisms","volume":"52","author":"Berrih-Aknin","year":"2014","journal-title":"J Autoimmun."},{"key":"B3","doi-asserted-by":"publisher","first-page":"776","DOI":"10.3389\/fimmu.2020.00776","article-title":"Autoimmune pathology in myasthenia gravis disease subtypes is governed by divergent mechanisms of immunopathology","volume":"11","author":"Fichtner","year":"2020","journal-title":"Front Immunol."},{"key":"B4","doi-asserted-by":"publisher","first-page":"16","DOI":"10.1212\/WNL.55.1.16","article-title":"Myasthenia gravis: recommendations for clinical research standards. Task Force of the Medical Scientific Advisory Board of the Myasthenia Gravis Foundation of America","volume":"55","author":"Jaretzki","year":"2000","journal-title":"Neurology."},{"key":"B5","unstructured":"The Portal for Rare Diseases and Orphan Drugs. Myasthenia Gravis2014"},{"key":"B6","doi-asserted-by":"publisher","first-page":"46","DOI":"10.1186\/1471-2377-10-46","article-title":"A systematic review of population based epidemiological studies in myasthenia gravis","volume":"10","author":"Carr","year":"2010","journal-title":"BMC Neurol."},{"key":"B7","doi-asserted-by":"publisher","first-page":"693","DOI":"10.1002\/mus.26689","article-title":"Factors associated with acute exacerbations of myasthenia gravis","volume":"60","author":"Gummi","year":"2019","journal-title":"Muscle Nerve."},{"key":"B8","doi-asserted-by":"publisher","first-page":"77","DOI":"10.1186\/s12883-017-0857-7","article-title":"Clinical predictors for the prognosis of myasthenia gravis","volume":"17","author":"Wang","year":"2017","journal-title":"BMC Neurol."},{"key":"B9","doi-asserted-by":"publisher","first-page":"114","DOI":"10.1212\/WNL.0000000000011124","article-title":"International consensus guidance for management of myasthenia gravis: 2020 update","volume":"96","author":"Narayanaswami","year":"2021","journal-title":"Neurology."},{"key":"B10","doi-asserted-by":"publisher","first-page":"98","DOI":"10.1007\/s00415-009-5279-z","article-title":"The relationship between health, disability and quality of life in myasthenia gravis: results from an Italian study","volume":"257","author":"Leonardi","year":"2010","journal-title":"J Neurol."},{"key":"B11","doi-asserted-by":"publisher","first-page":"e013278","DOI":"10.1136\/bmjopen-2016-013278","article-title":"Social disadvantages associated with myasthenia gravis and its treatment: a multicentre cross-sectional study","volume":"7","author":"Nagane","year":"2017","journal-title":"BMJ Open."},{"key":"B12","doi-asserted-by":"publisher","first-page":"252","DOI":"10.3389\/fphar.2016.00252","article-title":"Health economic data in reimbursement of new medical technologies: importance of the socio-economic burden as a decision-making criterion","volume":"7","author":"Iskrov","year":"2016","journal-title":"Front Pharmacol."},{"key":"B13","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1007\/s10198-016-0780-7","article-title":"Social\/economic costs and health-related quality of life in patients with rare diseases in Europe","volume":"17","author":"L\u00f3pez-Bastida","year":"2016","journal-title":"Eur J Health Econ."},{"key":"B14","doi-asserted-by":"publisher","first-page":"283","DOI":"10.1007\/978-3-319-67144-4_17","article-title":"Cost-of-illness in rare diseases","volume":"1031","author":"Linertov\u00e1","year":"2017","journal-title":"Adv Exp Med Biol."},{"key":"B15","volume-title":"Methods for the Economic Evaluation of Health Care Programmes","author":"Drummond","year":"2015"},{"key":"B16","doi-asserted-by":"publisher","first-page":"400","DOI":"10.1002\/gps.353","article-title":"Estimating the costs of informal care for people with Alzheimer's disease: methodological and practical challenges","volume":"16","author":"McDaid","year":"2001","journal-title":"Int J Geriatr Psychiatry."},{"key":"B17","doi-asserted-by":"publisher","first-page":"715","DOI":"10.1007\/s40273-020-00912-8","article-title":"Economic costs of myasthenia gravis: a systematic review","volume":"38","author":"Landfeldt","year":"2020","journal-title":"Pharmacoeconomics."},{"key":"B18","first-page":"1","article-title":"Bulgaria: health system review","volume":"20","author":"Dimova","year":"2018","journal-title":"Health Syst Transit."},{"key":"B19","doi-asserted-by":"publisher","first-page":"1109","DOI":"10.1007\/s10072-014-1659-y","article-title":"Predictors of outcome of myasthenic crisis","volume":"35","author":"Kalita","year":"2014","journal-title":"Neurol Sci."},{"key":"B20","doi-asserted-by":"publisher","first-page":"37","DOI":"10.4103\/0028-3886.15050","article-title":"Myasthenic crisis: clinical features, complications and mortality","volume":"53","author":"Murthy","year":"2005","journal-title":"Neurol India."},{"key":"B21","doi-asserted-by":"publisher","first-page":"1253","DOI":"10.1212\/WNL.48.5.1253","article-title":"Myasthenic crisis: clinical features, mortality, complications, and risk factors for prolonged intubation","volume":"48","author":"Thomas","year":"1997","journal-title":"Neurology."},{"key":"B22","doi-asserted-by":"publisher","first-page":"475","DOI":"10.1007\/s40273-019-00768-7","article-title":"Spillover effects on caregivers' and family members' utility: a systematic review of the literature","volume":"37","author":"Wittenberg","year":"2019","journal-title":"Pharmacoeconomics."},{"key":"B23","doi-asserted-by":"publisher","first-page":"24","DOI":"10.36865\/2020.v11i4.125","volume":"11","author":"Ivanov","year":"2020","journal-title":"Redk Boles Lek Siraci."}],"container-title":["Frontiers in Public Health"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fpubh.2022.822909\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2022,3,3]],"date-time":"2022-03-03T06:24:27Z","timestamp":1646288667000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fpubh.2022.822909\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,3,3]]},"references-count":23,"alternative-id":["10.3389\/fpubh.2022.822909"],"URL":"https:\/\/doi.org\/10.3389\/fpubh.2022.822909","relation":{},"ISSN":["2296-2565"],"issn-type":[{"value":"2296-2565","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,3,3]]},"article-number":"822909"}}