{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,10]],"date-time":"2026-06-10T16:18:53Z","timestamp":1781108333946,"version":"3.54.1"},"reference-count":43,"publisher":"Oxford University Press (OUP)","issue":"4","license":[{"start":{"date-parts":[[2023,3,27]],"date-time":"2023-03-27T00:00:00Z","timestamp":1679875200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"Time for Lyme"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023,8,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Background<\/jats:title>\n                  <jats:p>A proportion of patients with Lyme borreliosis (LB) report long-term persisting signs and symptoms, even after recommended antibiotic treatment, which is termed post-treatment Lyme disease syndrome (PTLDS). Consensus on guidance regarding diagnosis and treatment is currently lacking. Consequently, patients suffer and are left searching for answers, negatively impacting their quality of life and healthcare expenditure. Yet, health economic data on PTLDS remain scarce. The aim of this article is therefore to assess the cost-of-illness related to PTLDS, including the patient perspective.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Methods<\/jats:title>\n                  <jats:p>PTLDS patients (N\u2009=\u2009187) with confirmed diagnosis of LB were recruited by a patient organization. Patients completed a self-reported questionnaire on LB-related healthcare utilization, absence from work and unemployment. Unit costs (reference year 2018) were obtained from national databases and published literature. Mean costs and uncertainty intervals were calculated via bootstrapping. Data were extrapolated to the Belgian population. Generalized linear models were used to determine associated covariates with total direct costs and out-of-pocket expenditures.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>Mean annual direct costs amounted to \u20ac4618 (95% CI \u20ac4070\u20135152), of which 49.5% were out-of-pocket expenditures. Mean annual indirect costs amounted to \u20ac36\u00a0081 (\u20ac31\u00a0312\u201340\u00a0923). Direct and indirect costs at the population level were estimated at \u20ac19.4 and 151.5 million, respectively. A sickness or disability benefit as source of income was associated with higher direct and out-of-pocket costs.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Conclusions<\/jats:title>\n                  <jats:p>The economic burden associated with PTLDS on patients and society is substantial, with patients consuming large amounts of non-reimbursed healthcare resources. Guidance on adequate diagnosis and treatment of PTLDS is needed.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1093\/eurpub\/ckad045","type":"journal-article","created":{"date-parts":[[2023,3,27]],"date-time":"2023-03-27T17:23:43Z","timestamp":1679937823000},"page":"668-674","source":"Crossref","is-referenced-by-count":5,"title":["Cost of illness in patients with post-treatment Lyme disease syndrome in Belgium"],"prefix":"10.1093","volume":"33","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-8732-9455","authenticated-orcid":false,"given":"Ruben","family":"Willems","sequence":"first","affiliation":[{"name":"Department of Public Health and Primary Care, Interuniversity Centre for Health Economics Research (I-CHER), Ghent University , Gent, Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nick","family":"Verhaeghe","sequence":"additional","affiliation":[{"name":"Department of Public Health and Primary Care, Interuniversity Centre for Health Economics Research (I-CHER), Ghent University , Gent, Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christian","family":"Perronne","sequence":"additional","affiliation":[{"name":"Infectious Diseases Department, University Hospital Raymond Poincar\u00e9, APHP, Universit\u00e9 de Versailles Saint-Quentin\u2014Paris Saclay , Garches, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Liesbeth","family":"Borgermans","sequence":"additional","affiliation":[{"name":"Department of Public Health and Primary Care, Interuniversity Centre for Health Economics Research (I-CHER), Ghent University , Gent, Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8305-7210","authenticated-orcid":false,"given":"Lieven","family":"Annemans","sequence":"additional","affiliation":[{"name":"Department of Public Health and Primary Care, Interuniversity Centre for Health Economics Research (I-CHER), Ghent University , Gent, Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2023,3,27]]},"reference":[{"key":"2023080216365172300_ckad045-B1","doi-asserted-by":"crossref","first-page":"16090","DOI":"10.1038\/nrdp.2016.90","article-title":"Lyme borreliosis","volume":"2","author":"Steere","year":"2016","journal-title":"Nat Rev Dis Primers"},{"key":"2023080216365172300_ckad045-B2","doi-asserted-by":"crossref","first-page":"19906","DOI":"10.2807\/ese.16.27.19906-en","article-title":"Lyme borreliosis in Europe","volume":"16","author":"Rizzoli","year":"2011","journal-title":"Euro Surveill"},{"key":"2023080216365172300_ckad045-B3","first-page":"74","article-title":"An estimate of Lyme borreliosis incidence in Western Europe\u2020","volume":"39","author":"Sykes","year":"2017","journal-title":"J Public Health (Oxf)"},{"key":"2023080216365172300_ckad045-B4","doi-asserted-by":"crossref","first-page":"74","DOI":"10.3389\/fcimb.2014.00074","article-title":"Lyme and associated tick-borne diseases: global challenges in the context of a public health threat","volume":"4","author":"Perronne","year":"2014","journal-title":"Front Cell Infect Microbiol"},{"key":"2023080216365172300_ckad045-B5","doi-asserted-by":"crossref","first-page":"2385","DOI":"10.1007\/s10096-012-1580-3","article-title":"Incidence and management of presumption of Lyme borreliosis in Belgium: recent data from the sentinel network of general practitioners","volume":"31","author":"Vanthomme","year":"2012","journal-title":"Eur J Clin Microbiol Infect Dis"},{"key":"2023080216365172300_ckad045-B6","doi-asserted-by":"crossref","first-page":"452","DOI":"10.1016\/S0140-6736(18)31738-0","article-title":"Introducing EU-wide surveillance of Lyme neuroborreliosis","volume":"392","author":"Editorial","year":"2018","journal-title":"Lancet"},{"key":"2023080216365172300_ckad045-B7","doi-asserted-by":"crossref","first-page":"978","DOI":"10.5114\/aoms.2012.30948","article-title":"Lyme disease: review","volume":"8","author":"Biesiada","year":"2012","journal-title":"Arch Med Sci"},{"key":"2023080216365172300_ckad045-B8","doi-asserted-by":"crossref","first-page":"372","DOI":"10.1016\/j.tim.2013.06.005","article-title":"Illuminating the roles of the Borrelia burgdorferi adhesins","volume":"21","author":"Coburn","year":"2013","journal-title":"Trends Microbiol"},{"key":"2023080216365172300_ckad045-B9","doi-asserted-by":"crossref","first-page":"58","DOI":"10.1016\/j.ttbdis.2013.09.002","article-title":"Functional outcomes in patients with Borrelia burgdorferi reinfection","volume":"5","author":"Jares","year":"2014","journal-title":"Ticks Tick Borne Dis"},{"key":"2023080216365172300_ckad045-B10","doi-asserted-by":"crossref","first-page":"m1041","DOI":"10.1136\/bmj.m1041","article-title":"Lyme borreliosis: diagnosis and management","volume":"369","author":"Kullberg","year":"2020","journal-title":"BMJ"},{"key":"2023080216365172300_ckad045-B11","doi-asserted-by":"crossref","first-page":"79","DOI":"10.1186\/1471-2296-15-79","article-title":"Experiences of patients identifying with chronic Lyme disease in the healthcare system: a qualitative study","volume":"15","author":"Ali","year":"2014","journal-title":"BMC Fam Pract"},{"key":"2023080216365172300_ckad045-B12","doi-asserted-by":"crossref","first-page":"224","DOI":"10.3389\/fmed.2017.00224","article-title":"The clinical, symptom, and quality-of-life characterization of a well-defined group of patients with posttreatment Lyme disease syndrome","volume":"4","author":"Rebman","year":"2017","journal-title":"Front Med"},{"key":"2023080216365172300_ckad045-B13","doi-asserted-by":"crossref","first-page":"57","DOI":"10.3389\/fmed.2020.00057","article-title":"Post-treatment Lyme disease as a model for persistent symptoms in Lyme disease","volume":"7","author":"Rebman","year":"2020","journal-title":"Front Med"},{"key":"2023080216365172300_ckad045-B14","doi-asserted-by":"crossref","first-page":"291","DOI":"10.2147\/IJGM.S44114","article-title":"Review of evidence for immune evasion and persistent infection in Lyme disease","volume":"6","author":"Berndtson","year":"2013","journal-title":"Int J Gen Med"},{"key":"2023080216365172300_ckad045-B15","doi-asserted-by":"crossref","first-page":"129","DOI":"10.3390\/healthcare6040129","article-title":"Precision medicine: the role of the MSIDS model in defining, diagnosing, and treating chronic Lyme disease\/post treatment Lyme disease syndrome and other chronic illness: part 2","volume":"6","author":"Horowitz","year":"2018","journal-title":"Healthcare (Basel)"},{"key":"2023080216365172300_ckad045-B16","doi-asserted-by":"crossref","first-page":"85","DOI":"10.1016\/j.jneuroim.2012.10.011","article-title":"Increased IFN\u03b1 activity and differential antibody response in patients with a history of Lyme disease and persistent cognitive deficits","volume":"255","author":"Jacek","year":"2013","journal-title":"J Neuroimmunol"},{"key":"2023080216365172300_ckad045-B17","doi-asserted-by":"crossref","first-page":"40","DOI":"10.3389\/fcimb.2013.00040","article-title":"Borrelia burgdorferi aggrecanase activity: more evidence for persistent infection in Lyme disease","volume":"3","author":"Stricker","year":"2013","journal-title":"Front Cell Infect Microbiol"},{"key":"2023080216365172300_ckad045-B18","author":"US Department of Health and Human Services. Tick-Borne Disease Working Group","year":"2018"},{"key":"2023080216365172300_ckad045-B19","first-page":"e18703","article-title":"Post-treatment Lyme disease syndrome: need for diagnosis and treatment","volume":"13","author":"Maksimyan","year":"2021","journal-title":"Cureus"},{"key":"2023080216365172300_ckad045-B20","doi-asserted-by":"crossref","first-page":"e0210280","DOI":"10.1371\/journal.pone.0210280","article-title":"The economic burden of Lyme disease and the cost-effectiveness of Lyme disease interventions: a scoping review","volume":"14","author":"Mac","year":"2019","journal-title":"PLoS One"},{"key":"2023080216365172300_ckad045-B21","doi-asserted-by":"crossref","first-page":"1245","DOI":"10.1111\/j.1469-0691.2009.03059.x","article-title":"Neuroborreliosis\u2013an epidemiological, clinical and healthcare cost study from an endemic area in the south-east of Sweden","volume":"16","author":"Henningsson","year":"2010","journal-title":"Clin Microbiol Infect"},{"key":"2023080216365172300_ckad045-B22","doi-asserted-by":"crossref","first-page":"538","DOI":"10.1093\/eurpub\/ckw269","article-title":"The cost of Lyme borreliosis","volume":"27","author":"van den Wijngaard","year":"2017","journal-title":"Eur J Public Health"},{"key":"2023080216365172300_ckad045-B23","doi-asserted-by":"crossref","first-page":"56","DOI":"10.1016\/j.ttbdis.2014.09.004","article-title":"Epidemiology and cost of hospital care for Lyme borreliosis in Germany: lessons from a health care utilization database analysis","volume":"6","author":"Lohr","year":"2015","journal-title":"Ticks Tick Borne Dis"},{"key":"2023080216365172300_ckad045-B24","doi-asserted-by":"crossref","first-page":"2194","DOI":"10.1186\/s12889-022-14380-6","article-title":"Lyme borreliosis in Belgium: a cost-of-illness analysis","volume":"22","author":"Geebelen","year":"2022","journal-title":"BMC Public Health"},{"key":"2023080216365172300_ckad045-B25","doi-asserted-by":"crossref","first-page":"489","DOI":"10.2307\/3349924","article-title":"The economic costs of the health effects of smoking, 1984","volume":"64","author":"Rice","year":"1986","journal-title":"Milbank Q"},{"key":"2023080216365172300_ckad045-B26","doi-asserted-by":"crossref","first-page":"75","DOI":"10.2165\/00148365-200605020-00002","article-title":"How cost-of-illness studies can be made more useful for illicit drug policy analysis","volume":"5","author":"Moore","year":"2006","journal-title":"Appl Health Econ Health Policy"},{"key":"2023080216365172300_ckad045-B27","volume-title":"Methods for the economic evaluation of health care programmes","author":"Drummond","year":"2015","edition":"4th edn"},{"key":"2023080216365172300_ckad045-B28","author":"Nomenclature database","year":"2018"},{"key":"2023080216365172300_ckad045-B29","author":"Kohn","year":"2016"},{"key":"2023080216365172300_ckad045-B30","author":"FPS Health, NIHDI","year":"2017"},{"key":"2023080216365172300_ckad045-B31","author":"Van de Sande","year":"2012"},{"key":"2023080216365172300_ckad045-B32","author":"Gecommentarieerd geneesmiddelenrepertorium","year":"2018"},{"key":"2023080216365172300_ckad045-B33","first-page":"1","article-title":"De organisatie en financiering van de ziekenhuizen [Organization and financing of hospitals]","volume":"253","author":"Crommelynck","year":"2013","journal-title":"Christelijke Mutualiteit"},{"key":"2023080216365172300_ckad045-B34","author":"Verlinden","year":"2019"},{"key":"2023080216365172300_ckad045-B35","doi-asserted-by":"crossref","first-page":"257","DOI":"10.2307\/3552302","article-title":"Expanding economic costing in health care: values, gender and diversity","volume":"30","author":"Hankivsky","year":"2004","journal-title":"Can Public Policy Anal Polit"},{"key":"2023080216365172300_ckad045-B36","doi-asserted-by":"crossref","first-page":"8","DOI":"10.1016\/j.ttbdis.2018.12.007","article-title":"Combining primary care surveillance and a meta-analysis to estimate the incidence of the clinical manifestations of Lyme borreliosis in Belgium, 2015\u20132017","volume":"10","author":"Geebelen","year":"2019","journal-title":"Ticks Tick Borne Dis"},{"key":"2023080216365172300_ckad045-B37","author":"STATBEL","year":"2018"},{"key":"2023080216365172300_ckad045-B38","doi-asserted-by":"crossref","first-page":"1776","DOI":"10.1093\/cid\/civ186","article-title":"Unorthodox alternative therapies marketed to treat Lyme disease","volume":"60","author":"Lantos","year":"2015","journal-title":"Clin Infect Dis"},{"key":"2023080216365172300_ckad045-B39","first-page":"33","volume-title":"Well-Being in Belgium: Beyond Happiness and Income","author":"Cap\u00e9au","year":"2015"},{"key":"2023080216365172300_ckad045-B40","doi-asserted-by":"crossref","first-page":"705","DOI":"10.1017\/S026646230200051X","article-title":"The accuracy of self-reported healthcare resource utilization in health economic studies","volume":"18","author":"Petrou","year":"2002","journal-title":"Int J Technol Assess Health Care"},{"key":"2023080216365172300_ckad045-B41","doi-asserted-by":"crossref","first-page":"117","DOI":"10.1111\/j.1465-3362.2008.00040.x","article-title":"Why we should still estimate the costs of substance abuse even if we needn't pay undue attention to the bottom line","volume":"28","author":"Single","year":"2009","journal-title":"Drug Alcohol Rev"},{"key":"2023080216365172300_ckad045-B42","doi-asserted-by":"crossref","first-page":"831","DOI":"10.1089\/jwh.2008.1193","article-title":"Implications of gender in chronic Lyme disease","volume":"18","author":"Wormser","year":"2009","journal-title":"J Womens Health (Larchmt)"},{"key":"2023080216365172300_ckad045-B43","doi-asserted-by":"crossref","first-page":"756","DOI":"10.1186\/s12879-022-07686-8","article-title":"Non-specific symptoms and post-treatment Lyme disease syndrome in patients with Lyme borreliosis: a prospective cohort study in Belgium (2016-2020)","volume":"22","author":"Geebelen","year":"2022","journal-title":"BMC Infect Dis"}],"container-title":["European Journal of Public Health"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/eurpub\/advance-article-pdf\/doi\/10.1093\/eurpub\/ckad045\/49651431\/ckad045.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/eurpub\/article-pdf\/33\/4\/668\/51024901\/ckad045.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/eurpub\/article-pdf\/33\/4\/668\/51024901\/ckad045.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,8,2]],"date-time":"2023-08-02T16:37:37Z","timestamp":1690994257000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/eurpub\/article\/33\/4\/668\/7087174"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,3,27]]},"references-count":43,"journal-issue":{"issue":"4","published-online":{"date-parts":[[2023,3,27]]},"published-print":{"date-parts":[[2023,8,1]]}},"URL":"https:\/\/doi.org\/10.1093\/eurpub\/ckad045","relation":{},"ISSN":["1101-1262","1464-360X"],"issn-type":[{"value":"1101-1262","type":"print"},{"value":"1464-360X","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2023,8,1]]},"published":{"date-parts":[[2023,3,27]]}}}