{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,12,14]],"date-time":"2025-12-14T15:58:05Z","timestamp":1765727885048,"version":"3.37.3"},"reference-count":35,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2020,1,14]],"date-time":"2020-01-14T00:00:00Z","timestamp":1578960000000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2020,1,14]],"date-time":"2020-01-14T00:00:00Z","timestamp":1578960000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100001809","name":"The National Natural Science Foundation of China","doi-asserted-by":"crossref","award":["71333005"],"award-info":[{"award-number":["71333005"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"crossref"}]},{"name":"The National Social Science Foundation of China","award":["15ZDC037"],"award-info":[{"award-number":["15ZDC037"]}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Orphanet J Rare Dis"],"published-print":{"date-parts":[[2020,12]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>Myasthenia gravis is a rare autoimmune neuromuscular disorder. The disorder requires long-term use of expensive medication to control clinical symptoms. This study analyzed the change in trends of total medical expenses and out-of-pocket expenses for patients with myasthenia gravis and explored the factors influencing them.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>In this retrospective study, data were derived from a survey of medical service utilization for patients insured by the Urban Basic Medical Insurance in China from 2013 to 2015. The cost data of 3347 patients with myasthenia gravis were included in this study. The baseline characteristics and medical expenses for patients with myasthenia gravis were analyzed using a descriptive method. The difference and influencing factors of the out-of-pocket ratio were analyzed from both outpatient and inpatient expenses by using the quantile regression method.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>The total expenses reimbursed by the Urban Basic Medicine Insurance for all patients with myasthenia gravis fell progressively from 73.1 to 58.7% during the study period. Patients\u2019 out-of-pocket expenses increased gradually, of which expenses within the scope of Basic Medicine Insurance increased from 14.7 to 22.6% and expenses outside of the Basic Medicine Insurance scope increased from 12.6 to 18.7%. Moreover, the panel quantile results showed a positive correlation between the<jats:italic>year of receiving treatment<\/jats:italic>and the out-of-pocket ratio. In addition to the 25th quantile of the out-of-pocket ratio among outpatients with myasthenia gravis, there were significant differences in<jats:italic>medical insurance<\/jats:italic>and<jats:italic>medical institution<\/jats:italic>among all the other quantiles. Significant regional differences were found in all quantiles of the out-of-pocket ratio, except for the 75th quantile among inpatients. Lastly,<jats:italic>age<\/jats:italic>had a negative effect on inpatients with myasthenia gravis across all quantiles, but not on outpatients.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>From 2013 to 2015, patients with myasthenia gravis\u2019s out-of-pocket expenses increased progressively. Moreover, the individual out-of-pocket ratio was affected by the year, medical insurance, medical institution, region, and age. The current medical insurance policy for the general public has a low ability to cater for patients with myasthenia gravis.<\/jats:p><\/jats:sec>","DOI":"10.1186\/s13023-019-1289-9","type":"journal-article","created":{"date-parts":[[2020,1,14]],"date-time":"2020-01-14T18:02:39Z","timestamp":1579024959000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["Out-of-pocket expenses for myasthenia gravis patients in China: a study on patients insured by basic medical insurance in China, 2013\u20132015"],"prefix":"10.1186","volume":"15","author":[{"given":"Tao-yu","family":"Lin","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Xiao-yan","family":"Zhang","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Peng-qian","family":"Fang","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9119-6595","authenticated-orcid":false,"given":"Rui","family":"Min","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2020,1,14]]},"reference":[{"issue":"7","key":"1289_CR1","doi-asserted-by":"publisher","first-page":"1109","DOI":"10.1007\/s10072-014-1659-y","volume":"35","author":"J Kalita","year":"2014","unstructured":"Kalita J, Kohat AK, Misra UK. Predictors of outcome of myasthenic crisis. Neurol Sci. 2014;35(7):1109\u201314. https:\/\/doi.org\/10.1007\/s10072-014-1659-y.","journal-title":"Neurol Sci"},{"issue":"1","key":"1289_CR2","doi-asserted-by":"publisher","first-page":"670","DOI":"10.1111\/j.1749-6632.1981.tb33765.x","volume":"377","author":"MS Cohen","year":"1981","unstructured":"Cohen MS, Younger D. Aspects of the natural history of myasthenia gravis: crisis and death. Ann N Y Acad Sci. 1981;377(1):670\u20137. https:\/\/doi.org\/10.1111\/j.1749-6632.1981.tb33765.x.","journal-title":"Ann N Y Acad Sci"},{"issue":"5","key":"1289_CR3","doi-asserted-by":"publisher","first-page":"523","DOI":"10.1097\/WCO.0b013e3283572588","volume":"25","author":"NE Gilhus","year":"2012","unstructured":"Gilhus NE. Myasthenia and the neuromuscular junction. Curr Opin Neurol. 2012;25(5):523\u20139. https:\/\/doi.org\/10.1097\/WCO.0b013e3283572588.","journal-title":"Curr Opin Neurol"},{"key":"1289_CR4","doi-asserted-by":"publisher","first-page":"241","DOI":"10.1016\/j.jns.2017.02.009","volume":"375","author":"K Stieglbauer","year":"2017","unstructured":"Stieglbauer K, Pichler R, Topakian R. 10-year-outcomes after rituximab for myasthenia gravis: efficacy, safety, costs of inhospital care, and impact on childbearing potential. J Neurol Sci. 2017;375:241\u20134. https:\/\/doi.org\/10.1016\/j.jns.2017.02.009.","journal-title":"J Neurol Sci"},{"key":"1289_CR5","doi-asserted-by":"publisher","first-page":"114","DOI":"10.1016\/j.jocn.2016.11.003","volume":"38","author":"KK Sonkar","year":"2017","unstructured":"Sonkar KK, Bhoi SK, Dubey D, Kalita J, Misra UK. Direct and indirect cost of myasthenia gravis: a prospective study from a tertiary care teaching hospital in India. J Clin Neurosci. 2017;38:114\u20137. https:\/\/doi.org\/10.1016\/j.jocn.2016.11.003.","journal-title":"J Clin Neurosci"},{"issue":"3","key":"1289_CR6","doi-asserted-by":"publisher","first-page":"363","DOI":"10.1002\/mus.22327","volume":"45","author":"JT Guptill","year":"2012","unstructured":"Guptill JT, Sharma BK, Marano A, Soucy A, Krueger A, Sanders DB. Estimated cost of treating myasthenia gravis in an insured U. S .population. Muscle Nerve. 2012;45(3):363\u20136. https:\/\/doi.org\/10.1002\/mus.22327.","journal-title":"Muscle Nerve"},{"key":"1289_CR7","volume-title":"The world health report: health systems financing: the path to universal coverage","author":"World Health Organization","year":"2010","unstructured":"World Health Organization. The world health report: health systems financing: the path to universal coverage. Geneva: World Health Organization; 2010. Available at: https:\/\/apps.who.int\/iris\/bitstream\/handle\/10665\/44371\/9789241564021_eng.pdf?sequence=1&isAllowed=y. Accessed 10 Dec 2019"},{"key":"1289_CR8","unstructured":"Commission of the European Communities. Communication from the European commission to the council, the European parliament, the economic and social committee and committee of the regions on Rare Diseases: Europe\u2019s challenges. 2008. Available at: http:\/\/ec.europa.eu\/health\/ph_threats\/non_com\/docs\/rare_com_en.pdf. Accessed 10 Dec 2019."},{"issue":"2","key":"1289_CR9","doi-asserted-by":"publisher","first-page":"81","DOI":"10.3969\/j.issn.1673-5501.2011.02.001","volume":"6","author":"M Rui","year":"2011","unstructured":"Rui M, Dingguo L, Xue Z, Lin H. Opportunities and challenges in the prevention and control of rare diseases in China. Chin J Evid Based Pediatrics. 2011;6(2):81\u20132. https:\/\/doi.org\/10.3969\/j.issn.1673-5501.2011.02.001.","journal-title":"Chin J Evid Based Pediatrics"},{"issue":"2","key":"1289_CR10","doi-asserted-by":"publisher","first-page":"179","DOI":"10.1111\/hir.12256","volume":"36","author":"SU Weerasooriya","year":"2019","unstructured":"Weerasooriya SU. The impact of orphan drug policies in treating rare diseases. Health Inf Libr J. 2019;36(2):179\u201384. https:\/\/doi.org\/10.1111\/hir.12256.","journal-title":"Health Inf Libr J"},{"key":"1289_CR11","unstructured":"Office of Inspector General, Department of Health and Human Services, The orphan drug act implementation and impact. 2001. Available at: http:\/\/oig.hhs.gov\/oei\/reports\/oei-09-00-00380.pdf. Accessed 10 Dec, 2019."},{"issue":"3","key":"1289_CR12","doi-asserted-by":"publisher","first-page":"648","DOI":"10.1093\/jlb\/lsy019","volume":"5","author":"E Harris","year":"2018","unstructured":"Harris E. Addressing the needs of Canadians with rare diseases: an evaluation of orphan drug incentives. J Law Biosci. 2018;5(3):648\u201381. https:\/\/doi.org\/10.1093\/jlb\/lsy019.","journal-title":"J Law Biosci"},{"key":"1289_CR13","volume-title":"Rare Diseases: Leaving no one behind in Universal Health Coverage","author":"Rare diseases international","year":"2019","unstructured":"Rare diseases international. Rare Diseases: Leaving no one behind in Universal Health Coverage. 2019. Available at: http:\/\/download2.eurordis.org.s3-eu-west-1.amazonaws.com\/rdi\/2019\/RDI%20UHC%20Paper%20Final%20May%202019_For%20Print.pdf. Accessed 10 Dec 2019."},{"key":"1289_CR14","volume-title":"Circular of the general office of the national health and family planning commission on the establishment of an expert committee on the diagnosis, treatment and protection of rare diseases of the state health and family planning commission","author":"NHFPC","year":"2015","unstructured":"NHFPC. Circular of the general office of the national health and family planning commission on the establishment of an expert committee on the diagnosis, treatment and protection of rare diseases of the state health and family planning commission. 2015. Available at: http:\/\/www.nhc.gov.cn\/yzygj\/s7659\/201601\/c918591a06154c1ea193fc32ba93c085.shtml. Accessed 10 Dec 2019."},{"issue":"2","key":"1289_CR15","doi-asserted-by":"publisher","first-page":"95","DOI":"10.13220\/j.cnki.jipr.2017.02.002","volume":"44","author":"F Shi","year":"2017","unstructured":"Shi F, Shuang L, Mengchun G, Shuyang Z. Rare disease research and orphan drug discovery. J Int Pharmaceutical Res. 2017;44(2):95\u2013100. https:\/\/doi.org\/10.13220\/j.cnki.jipr.2017.02.002.","journal-title":"J Int Pharmaceutical Res"},{"issue":"9845","key":"1289_CR16","doi-asserted-by":"publisher","first-page":"933","DOI":"10.1016\/S0140-6736(12)61147-7","volume":"380","author":"G Lagomarsino","year":"2012","unstructured":"Lagomarsino G, Garabrant A, Adyas A, Muga R, Otoo N. Moving towards universal health coverage: health insurance reforms in nine developing countries in Africa and Asia. Lancet. 2012;380(9845):933\u201343. https:\/\/doi.org\/10.1016\/S0140-6736(12)61147-7.","journal-title":"Lancet."},{"key":"1289_CR17","first-page":"8","volume":"0","author":"F Cheng","year":"2018","unstructured":"Cheng F, Jiajun H. A study of welfare effect on basic medical insurance policy of urban and rural residents. Welfare Res. 2018;0:8\u201315 http:\/\/kns.cnki.net\/KCMS\/detail\/detail.aspx?FileName=WERE201800003&DbName=CJFNTEMP.","journal-title":"Welfare Res"},{"key":"1289_CR18","doi-asserted-by":"publisher","first-page":"17","DOI":"10.14055\/j.cnki.33-1056\/f.20180404.016","volume":"4","author":"L Yang","year":"2018","unstructured":"Yang L, Guangfeng D, Li Y, Wenhua T. Analysis on the development trend of serious illness insurance in China\u2014\u2014based on three typical models. Health Econ Res. 2018;4:17\u20139. https:\/\/doi.org\/10.14055\/j.cnki.33-1056\/f.20180404.016.","journal-title":"Health Econ Res"},{"key":"1289_CR19","doi-asserted-by":"crossref","DOI":"10.1596\/978-92-4-151355-5","volume-title":"Tracking universal health coverage: 2017 global monitoring report","author":"World Bank","year":"2017","unstructured":"World Bank. Tracking universal health coverage: 2017 global monitoring report. 2017. Available at: http:\/\/documents.worldbank.org\/curated\/en\/640121513095868125\/Tracking-universal-health-coverage-2017-global-monitoring-report. Accessed 10 Dec 2019."},{"issue":"3","key":"1289_CR20","doi-asserted-by":"publisher","first-page":"71","DOI":"10.7664\/CHE20180320","volume":"37","author":"L Fei","year":"2018","unstructured":"Fei L, Jing Z, Ming H. Analysis on the medical insurance policy and medical insurance catalog for rare diseases in China. Chinese Health Econ. 2018;37(3):71\u20136. https:\/\/doi.org\/10.7664\/CHE20180320.","journal-title":"Chinese Health Econ"},{"issue":"10031","key":"1289_CR21","doi-asserted-by":"publisher","first-page":"1906","DOI":"10.1016\/S0140-6736(16)30418-4","volume":"387","author":"D Dong","year":"2016","unstructured":"Dong D, Wang Y. Challenges of rare diseases in China. Lancet. 2016;387(10031):1906. https:\/\/doi.org\/10.1016\/S0140-6736(16)30418-4.","journal-title":"Lancet."},{"key":"1289_CR22","volume-title":"Treatment for MG","author":"Myasthenia gravis foundtion of America","year":"2015","unstructured":"Myasthenia gravis foundtion of America. Treatment for MG. 2015. Available at: http:\/\/myasthenia.org\/WhatisMG\/TreatmentforMG.aspx. Accessed 10 Dec 2019."},{"key":"1289_CR23","doi-asserted-by":"publisher","first-page":"8","DOI":"10.13778\/j.cnki.11-3705\/c.2014.02.010","volume":"2","author":"S Fengdan","year":"2014","unstructured":"Fengdan S. Differences in the development of China\u2019s eastern, central and western regions in building a moderately prosperous society in all respects. World Survey Res. 2014;2:8\u201313. https:\/\/doi.org\/10.13778\/j.cnki.11-3705\/c.2014.02.010.","journal-title":"World Survey Res"},{"issue":"40","key":"1289_CR24","doi-asserted-by":"publisher","first-page":"4081","DOI":"10.3969\/j.issn.1007-9572.2013.12.021","volume":"16","author":"C Jirui","year":"2013","unstructured":"Jirui C, Kaijin Z, Yanghong T. Canonical correlation analysis on hospital costs and medical insurance reimbursement of common chronic disease. Chin Gen Pract. 2013;16(40):4081\u20134. https:\/\/doi.org\/10.3969\/j.issn.1007-9572.2013.12.021.","journal-title":"Chin Gen Pract"},{"key":"1289_CR25","doi-asserted-by":"publisher","first-page":"105","DOI":"10.13497\/j.cnki.is.2014.06.001","volume":"6","author":"Y Yao","year":"2014","unstructured":"Yao Y, Bin L, Gordon L, Wenbin Z. Medical insurance, household registration system and healthcare utilization\u2014evidences from CHARLS data analysis. Insurance Stud. 2014;6:105\u201316. https:\/\/doi.org\/10.13497\/j.cnki.is.2014.06.001.","journal-title":"Insurance Stud"},{"issue":"9378","key":"1289_CR26","doi-asserted-by":"publisher","first-page":"111","DOI":"10.1016\/S0140-6736(03)13861-5","volume":"362","author":"K Xu","year":"2003","unstructured":"Xu K, Evans DB, Kawabata K, Zeramdini R, Klavus J, Murray C. Household catastrophic health expenditure: a multicountry analysis. Lancet. 2003;362(9378):111\u20137. https:\/\/doi.org\/10.1016\/S0140-6736(03)13861-5.","journal-title":"Lancet."},{"key":"1289_CR27","doi-asserted-by":"publisher","first-page":"52","DOI":"10.16091\/j.cnki.cn32-1308\/c.2014.01.021","volume":"1","author":"S Shuguang","year":"2014","unstructured":"Shuguang S. Theoretical thinking and path conception of the integration of national basic medical insurance system. Academia Bimestrie. 2014;1:52\u20138. https:\/\/doi.org\/10.16091\/j.cnki.cn32-1308\/c.2014.01.021.","journal-title":"Academia Bimestrie"},{"issue":"14","key":"1289_CR28","doi-asserted-by":"publisher","first-page":"59","DOI":"10.13546\/j.cnki.tjyjc.2018.14.013","volume":"34","author":"Y Hongyan","year":"2018","unstructured":"Hongyan Y, Mengqi N, Fanjie L. Does universal health insurance effectively resist the economic risk of disease? Stat Decis. 2018;34(14):59\u201363. https:\/\/doi.org\/10.13546\/j.cnki.tjyjc.2018.14.013.","journal-title":"Stat Decis"},{"key":"1289_CR29","doi-asserted-by":"publisher","first-page":"21","DOI":"10.3782\/j.issn.1006-0863.2014.07.04","volume":"7","author":"H Wenjiong","year":"2014","unstructured":"Wenjiong H. Establish a more fair and sustainable medical security system. Chin Public Adm. 2014;7:21\u20134. https:\/\/doi.org\/10.3782\/j.issn.1006-0863.2014.07.04.","journal-title":"Chin Public Adm"},{"issue":"3","key":"1289_CR30","doi-asserted-by":"publisher","first-page":"40","DOI":"10.16152\/j.cnki.xdxbsk.2018-03-005","volume":"48","author":"Q Yulin","year":"2018","unstructured":"Yulin Q, Zhaoxi W. Universal health insurance and healthy China: foundations, links and guidance. J Northwest Univ (Philosophy and Social Sciences Edition). 2018;48(3):40\u20137. https:\/\/doi.org\/10.16152\/j.cnki.xdxbsk.2018-03-005.","journal-title":"J Northwest Univ (Philosophy and Social Sciences Edition)"},{"issue":"17","key":"1289_CR31","doi-asserted-by":"publisher","first-page":"1655","DOI":"10.13286\/j.cnki.chinhosppharmacyj.2017.17.01","volume":"37","author":"X Meijun","year":"2017","unstructured":"Meijun X, Shiwe G. Problems and strategies of rare diseases and orphan drugs in China based on a systematic literature. Chin J Hosp Pharm. 2017;37(17):1655\u201360. https:\/\/doi.org\/10.13286\/j.cnki.chinhosppharmacyj.2017.17.01.","journal-title":"Chin J Hosp Pharm"},{"issue":"1","key":"1289_CR32","doi-asserted-by":"publisher","first-page":"173","DOI":"10.1186\/s12939-018-0891-0","volume":"17","author":"X Yang","year":"2018","unstructured":"Yang X, Chen M, Du J, Wang Z. The inequality of inpatient care net benefit under integration of urban-rural medical insurance systems in China. Int J Equity Health. 2018;17(1):173. https:\/\/doi.org\/10.1186\/s12939-018-0891-0.","journal-title":"Int J Equity Health"},{"key":"1289_CR33","volume-title":"Notice on measures for the administration of special illness in the outpatient department of basic medical insurance in Chengdu","author":"Chengdu Bureau of Human Resources and Social Security","year":"2014","unstructured":"Chengdu Bureau of Human Resources and Social Security. Notice on measures for the administration of special illness in the outpatient department of basic medical insurance in Chengdu. 2014. Available at: http:\/\/gk.chengdu.gov.cn\/govInfoPub\/detail.action?id=63135&tn=6. Accessed 10 Dec 2019."},{"issue":"12","key":"1289_CR34","doi-asserted-by":"publisher","first-page":"1387","DOI":"10.4103\/0366-6999.183425","volume":"129","author":"X Xin","year":"2016","unstructured":"Xin X, Zhao L, Guan X, Shi L. Determinants and equity evaluation for health expenditure among patients with rare diseases in China. Chinese Med J-Peking. 2016;129(12):1387\u201393. https:\/\/doi.org\/10.4103\/0366-6999.183425.","journal-title":"Chinese Med J-Peking"},{"issue":"3","key":"1289_CR35","doi-asserted-by":"publisher","first-page":"40","DOI":"10.3969\/j.issn.1674-2982.2017.03.008","volume":"10","author":"A Yi","year":"2017","unstructured":"Yi A, Yi C, Yu-liang C. Study on hospitalization benefit equity of the basic medical insurance programs in China. Chin J Health Policy. 2017;10(3):40\u20136. https:\/\/doi.org\/10.3969\/j.issn.1674-2982.2017.03.008.","journal-title":"Chin J Health Policy"}],"container-title":["Orphanet Journal of Rare Diseases"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1186\/s13023-019-1289-9.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/article\/10.1186\/s13023-019-1289-9\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1186\/s13023-019-1289-9.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2022,10,11]],"date-time":"2022-10-11T08:01:22Z","timestamp":1665475282000},"score":1,"resource":{"primary":{"URL":"https:\/\/ojrd.biomedcentral.com\/articles\/10.1186\/s13023-019-1289-9"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2020,1,14]]},"references-count":35,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2020,12]]}},"alternative-id":["1289"],"URL":"https:\/\/doi.org\/10.1186\/s13023-019-1289-9","relation":{},"ISSN":["1750-1172"],"issn-type":[{"type":"electronic","value":"1750-1172"}],"subject":[],"published":{"date-parts":[[2020,1,14]]},"assertion":[{"value":"19 March 2019","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"24 December 2019","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"14 January 2020","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"Not applicable.","order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Not applicable.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare that they have no competing interests.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"13"}}