Abstract
Background
Increased survival in patients with systemic lupus erythematosus (SLE) has shifted attention towards the burden that SLE imposes upon patients, healthcare systems and society. New interventions aimed at alleviating this burden will require economic evaluation. A summary of the current evidence of the humanistic and economic burden provides a platform for such subsequent studies.
Objective
The objective of this study was to systematically review the current evidence on the humanistic and economic burden of SLE in terms of health-related quality of life (HR-QOL) and costs, and summarize the evidence on the factors found to be associated with this burden.
Methods
Relevant literature for the years 1990 to February 2011 were obtained from systematic searches of MEDLINE, EMBASE and Web of Science. Articles reporting the humanistic (preference-based outcome measures or an SLE disease-specific HR-QOL measure) or economic burden (costs) of SLE in adult populations published in English were identified. The following exclusion criteria were applied: studies specifically examining lupus nephritis, SLE not being the main condition of focus (e.g. SLE is a co-existing condition), studies focusing on diagnostics or tests (including genetics and antibodies), mixed patient groups from which SLE could not be separated, paediatric populations, case studies, abstract unavailable, and non-English language studies. Estimates of the burden in terms of either HR-QOL or costs were extracted, tabulated and reported narratively. Annual cost figures were also converted into year 2010 US dollars using the consumer price index (CPI) and the purchasing power parity (PPP) conversion factor to allow for greater comparability across studies. Evidence on the factors found to be independently associated with either HR-QOL or costs was also examined.
Results
Of the 1969 studies initially identified as being potentially relevant, 32 papers were retained for the final review. Eighteen of these presented estimates of the burden in terms of HR-QOL, and 14 in terms of the economic cost. Mean utility scores reported on preference-based measures of HR-QOL ranged from 0.6 to 0.75. Mean annual direct costs per patient ranged from US$2,214 to US$16,875, and mean annual indirect cost estimates from US$2,239 to US$35,540 (year 2010 values). Disease activity and damage, along with poor mental and physical health, were repeatedly reported to predict both reduced HR-QOL and increased costs.
Conclusions
The burden of SLE was found to be substantial, not only for patients but also for the health services that provide care for them. Treatments that are able to alleviate this burden are therefore likely to be highly valued by sufferers. After an extended period in which few therapeutic advances were made or treatments licensed, fundamental developments are finally being made. These new treatments will need to be evaluated for both clinical and cost effectiveness if their use is to be widely implemented.
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References
Nightingale AL, Farmer RDT, de Vries CS. Incidence of clinically diagnosed systemic lupus erythematosus 1992–1998 using the UK General Practice Research Database. Pharmacoepidemiol Drug Saf. 2006;15(9):656–61.
Somers EC, Thomas SL, Smeeth L, Schoonen WM, Hall AJ. Incidence of systemic lupus erythematosus in the United Kingdom, 1990–1999. Arthritis Care Res. 2007;57(4):612–8.
Ward MM. Prevalence of physician-diagnosed systemic lupus erythematosus in the United States: results from the Third National Health and Nutrition Examination Survey. J Womens Health. 2004;13(6):713–8.
Samanta A, Roy S, Feehally J, Symmons DPM. The prevalence of diagnosed systemic lupus erythematosus in whites and Indian Asian immigrants in Leicester city. UK. Br J Rheumatol. 1992;31(10):679–82.
Hopkinson ND, Doherty M, Powell RJ. The prevalence and incidence of systemic lupus erythematosus in Nottingham, UK, 1989–1990. Br J Rheumatol. 1993;32(2):110–5.
Johnson AE, Gordon C, Palmer RG, Bacon PA. The prevalence and incidence of systemic lupus erythematosus in Birmingham, England: relationship to ethnicity and country of birth. Arthritis Rheum. 1995;38(4):551–8.
Gourley IS, Patterson CC, Bell AL. The prevalence of systemic lupus erythematosus in Northern Ireland. Lupus. 1997;6(4):399–403.
LUPUS UK. So it’s lupus. Romford: LUPUS UK; 2011. http://www.lupusuk.org.uk/what-is-lupus/so-its-lupus. Accessed 29 Nov 2011.
Helmick CG, Felson DT, Lawrence RC, Gabriel S, Hirsch R, Kwoh CK, et al. Estimates of the prevalence of arthritis and other rheumatic conditions in the United States. Arthritis Rheum. 2008;58(1):15–25.
Urowitz MB, Gladman DD, AbuShakra M, Farewell VT. Mortality studies in systemic lupus erythematosus: results from a single center. III. Improved survival over 24 years. J Rheumatol. 1997;24(6):1061–5.
Jacobsen S, Petersen J, Ullman S, Junker P, Voss A, Rasmussen JM, et al. Mortality and causes of death of 513 Danish patients with systemic lupus erythematosus. Scand J Rheumatol. 1999;28(2):75–80.
Jolly M. How does quality of life of patients with systemic lupus erythematosus compare with that of other common chronic illnesses? J Rheumatol. 2005;32(9):1706–8.
Kuriya B, Gladman DD, Ibanez D, Urowitz MB. Quality of life over time in patients with systemic lupus erythematosus. Arthritis Rheum. 2008;59(2):181–5.
McElhone K, Abbott J, Teh LS. A review of health related quality of life in systemic lupus erythematosus. Lupus. 2006;15(10):633–43.
Drummond MF, Sculpher MJ, Torrance GW, O’Brien BJ, Stoddart GL. Methods for the economic evaluation of health care programmes. 3rd ed. Oxford: Oxford University Press; 2005.
Clarke AE, Penrod J, St PY, Petri MA, Manzi S, Isenberg DA, et al. Underestimating the value of women: assessing the indirect costs of women with systemic lupus erythematosus. Tri-Nation Study Group. J Rheumatol. 2000;27(11):2597–604.
Bruce IN. Re-evaluation of biologic therapies in systemic lupus erythematosus. Curr Opin Rheumatol. 2010;22(3):273–7.
Michaud K, Messer J, Choi HK, Wolfe F. Direct medical costs and their predictors in patients with rheumatoid arthritis: a three-year study of 7,527 patients. Arthritis Rheum. 2003;48(10):2750–62.
Gordon C, Clarke AE. Quality of life and economic evaluation in SLE clinical trials. Lupus. 1999;8(8):645–54.
Zhu TY, Tam LS, Li EK. Cost-of-illness studies in systemic lupus erythematosus: a systematic review. Arthritis Care Res. 2011;63(5):751–60.
National Institute for Health and Clinical Excellence. Guide to the methods of technology appraisal. London: NICE; 2008. http://www.nice.org.uk/media/B52/A7/TAMethodsGuideUpdatedJune2008.pdf. Accessed 17 Jan 2012.
Canadian Agency for Drugs and Technologies in Health. Guidelines for the economic evaluation of health technologies: Canada. 3rd ed. Ottawa: CADTH; 2006.
Ontario Ministry of Health and Long-Term Care. Ontario guidelines for economic analysis of pharmaceutical products. Toronto: Ontario Ministry of Health and Long-Term Care; 2004. http://www.health.gov.on.ca/english/providers/pub/drugs/economic/economic_mn.html. Accessed 31 Jan 2007.
Sullivan SD, Lyles A, Luce B, Grigar J. AMCP guidance for submission of clinical and economic evaluation data to support formulary listing in U.S. health plans and pharmacy benefits management organizations. J Manag Care Pharm. 2001;7(4):272–82.
Health Information and Quality Authority. Guidelines for the economic evaluation of health technologies in Ireland. Cork: Health Information and Quality Authority; 2010.
Strand V, Gladman D, Isenberg D, Petri M, Smolen J, Tugwell P. Endpoints: consensus recommendations from OMERACT IV. Lupus. 2000;9(5):322–7.
Strand V, Chu AD. Generic versus disease-specific measures of health-related quality of life in systemic lupus erythematosus. J Rheumatol. 2011;38(9):1821–3.
OECD. Consumer prices (MEI). Paris: OECD; 2011. http://stats.oecd.org/Index.aspx?DataSetCode=MEI_PRICES. Accessed 4 Oct 2011.
OECD. Purchasing power parities for GDP and related indicators. Paris: OECD; 2011. http://stats.oecd.org/Index.aspx?DataSetCode=PPPGDP. Accessed 4 Oct 2011.
Sanchez ML, McGwin G Jr, Duran S, Fernandez M, Reveille JD, Vila LM, et al. Factors predictive of overall health over the course of the disease in patients with systemic lupus erythematosus from the LUMINA cohort (LXII): use of the SF-6D. Clin Exp Rheumatol. 2009;27(1):67–71.
Fernandez M, Alarcon GS, McGwin G Jr, Sanchez ML, Apte M, Vila LM, et al. Using the Short Form 6D, as an overall measure of health, to predict damage accrual and mortality in patients with systemic lupus erythematosus: XLVII, results from a multiethnic US cohort. Arthritis Rheum. 2007;57(6):986–92.
Aggarwal R, Wilke CT, Pickard AS, Vats V, Mikolaitis R, Fogg L, et al. Psychometric properties of the EuroQol-5D and Short Form-6D in patients with systemic lupus erythematosus. J Rheumatol. 2009;36(6):1209–16.
Harrison MJ, Ahmad Y, Haque S, Dale N, Teh LS, Snowden N, et al. Construct and criterion validity of the short form-6D utility measure in patients with systemic lupus erythematosus (SLE). J Rheumatol. 2012;39(4):735–42.
Wolfe F, Michaud K, Li T, Katz RS. Chronic conditions and health problems in rheumatic diseases: comparisons with rheumatoid arthritis, noninflammatory rheumatic disorders, systemic lupus erythematosus, and fibromyalgia. J Rheum. 2010;37(2):305–15.
Luo N, Chew LH, Fong KY, Koh DR, Ng SC, Yoon KH, et al. A comparison of the EuroQol-5D and the Health Utilities Index mark 3 in patients with rheumatic disease. J Rheumatol. 2003;30(10):2268–74.
Wolfe F, Michaud K, Li T, Katz RS. EQ-5D and SF-36 quality of life measures in systemic lupus erythematosus: comparisons with rheumatoid arthritis, noninflammatory rheumatic disorders, and fibromyalgia. J Rheumatol. 2010;37(2):296–304.
Moore AD, Clarke AE, Danoff DS, Joseph L, Belisle P, Neville C, et al. Can health utility measures be used in lupus research? A comparative validation and reliability study of 4 utility indices. J Rheumatol. 1999;26(6):1285–90.
Suarez-Almazor ME, Conner-Spady B. Rating of arthritis health states by patients, physicians, and the general public: implications for cost-utility analyses. J Rheumatol. 2001;28(3):648–56.
McElhone K, Castelino M, Abbott J, Bruce IN, Ahmad Y, Shelmerdine J, et al. The LupusQoL and associations with demographics and clinical measurements in patients with systemic lupus erythematosus. J Rheumatol. 2010;37(11):2273–9.
Jolly M, Pickard SA, Mikolaitis RA, Rodby RA, Sequeira W, Block JA. LupusQoL-US benchmarks for US patients with systemic lupus erythematosus. J Rheumatol. 2010;37(9):1828–33.
Leong KP, Chong EY, Kong KO, Chan SP, Thong BY, Lian TY, et al. Discordant assessment of lupus activity between patients and their physicians: the Singapore experience. Lupus. 2010;19(1):100–6.
Clarke AE, Petri M, Manzi S, Isenberg DA, Gordon C, Senecal JL, et al. The systemic lupus erythematosus Tri-nation Study: absence of a link between health resource use and health outcome. Rheumatology. 2004;43(8):1016–24.
Panopalis P, Petri M, Manzi S, Isenberg DA, Gordon C, Senegal J-L, et al. The systemic lupus erythematosus Tri-nation study: cumulative indirect costs. Arthritis Care Res. 2007;1:64–70.
Zhu TY, Tam L-S, Lee VWY, Lee KKC, Li EK. The impact of flare on disease costs of patients with systemic lupus erythematosus. Arthritis Care Res. 2009;9:1159–67.
Zhu TY, Tam LS, Lee VW, Lee KK, Li EK. Systemic lupus erythematosus with neuropsychiatric manifestation incurs high disease costs: a cost-of-illness study in Hong Kong. Rheumatology. 2009;48(5):564–8.
Clarke AE, Esdaile JM, Bloch DA, Lacaille D, Danoff DS, Fries JF. A Canadian study of the total medical costs for patients with systemic lupus erythematosus and the predictors of costs. Arthritis Rheum. 1993;36(11):1548–59.
Pelletier EM, Ogale S, Yu E, Brunetta P, Garg J. Economic outcomes in patients diagnosed with systemic lupus erythematosus with versus without nephritis: results from an analysis of data from a US claims database. Clin Ther. 2009;31(11):2653–64.
Sutcliffe N, Clarke AE, Taylor R, Frost C, Isenberg DA. Total costs and predictors of costs in patients with systemic lupus erythematosus. Rheumatology. 2001;40(1):37–47.
Gironimi G, Clarke AE, Hamilton VH, Danoff DS, Bloch DA, Fries JF, et al. Why health care costs more in the US: comparing health care expenditures between systemic lupus erythematosus patients in Stanford and Montreal. Arthritis Rheum. 1996;39(6):979–87.
Liljas B. How to calculate indirect costs in economic evaluations. Pharmacoeconomics. 1998;13(1):1–7.
Koopmanschap MA, Vanineveld BM. Towards a new approach for estimating indirect costs of disease. Soc Sci Med. 1992;34(9):1005–10.
Koopmanschap MA, Rutten FFH, Vanineveld BM, Vanroijen L. The friction cost method for measuring indirect costs of disease. J Health Econ. 1995;14(2):171–89.
Huscher D, Merkesdal S, Thiele K, Zeidler H, Schneider M, Zink A, et al. Cost of illness in rheumatoid arthritis, ankylosing spondylitis, psoriatic arthritis and systemic lupus erythematosus in Germany. Annals Rheum Dis. 2006;65(9):1175–83.
Clarke AE, Bloch DA, Danoff DS, Esdaile JM. Decreasing costs and improving outcomes in systemic lupus erythematosus: using regression trees to develop health policy. J Rheumatol. 1994;21(12):2246–53.
Lacaille D, Clarke AE, Bloch DA, Danoff D, Esdaile JM. The impact of disease activity, treatment and disease severity on short-term costs of systemic lupus erythematosus. J Rheumatol. 1994;21(3):448–53.
Panopalis P, Yazdany J, Gillis JZ, Julian L, Trupin L, Hersh AO, et al. Health care costs and costs associated with changes in work productivity among persons with systemic lupus erythematosus. Arthritis Rheum. 2008;59(12):1788–95.
Cohen J. Statistical power analysis for the behavioural sciences. Hillsdale: Lawrence Earlbaum; 1977.
McElhone K, Abbott J, Shelmerdine J, Bruce IN, Ahmad Y, Gordon C, et al. Development and validation of a disease-specific health-related quality of life measure, the LupusQol, for adults with systemic lupus erythematosus. Arthritis Rheum. 2007;57(6):972–9.
Doward LC, McKenna SP, Whalley D, Tennant A, Griffiths B, Emery P, et al. The development of the L-QoL: a quality-of-life instrument specific to systemic lupus erythematosus. Annals Rheum Dis. 2009;68(2):196–200.
Harrison MJ, Davies LM, Bansback NJ, Ingram M, Anis AH, Symmons DP. The validity and responsiveness of generic utility measures in rheumatoid arthritis: a review. J Rheumatol. 2008;35(4):592–602.
Bansback N, Harrison M, Brazier J, Davies L, Kopec J, Marra C, et al. Health state utility values: a description of their development and application for rheumatic diseases. Arthritis Rheum. 2008;59(7):1018–26.
Carls G, Li T, Panopalis P, Wang S, Mell AG, Gibson TB, et al. Direct and indirect costs to employers of patients with systemic lupus erythematosus with and without nephritis. J Occup Environ Med. 2009;51(1):66–79.
Isenberg DA, Shortall E, Newman S. Systemic lupus erythematosus: disease activity, severity, treatment and costs. J Rheumatol. 1994;21(3):394–6.
U.S. Food and Drug Administration. FDA approves Benlysta to treat lupus. Silver Spring: FDA; 2011. http://www.fda.gov/NewsEvents/Newsroom/PressAnnouncements/ucm246489.htm. Accessed 1 Dec 2011.
National Institute for Health and Clinical Excellence. NICE consults on belimumab for systemic lupus erythematosus. London: NICE; 2011. http://www.nice.org.uk/newsroom/pressreleases/SystemicLupusBelimumabACD.jsp. Accessed 29 Nov 2011.
Clarke AE, Petri MA, Manzi S, Isenberg DA, Gordon C, Senecal JL, et al. An international perspective on the well being and health care costs for patients with systemic lupus erythematosus. Tri-Nation Study Group. J Rheumatol. 1999;26(7):1500–11.
Paisley S, Booth A, Mensinkai S. Health-related quality of life studies. In: National Library of Medicine. Etext on health technology assessment (HTA) information resources. Bethseda: NLM; 2005. http://www.nlm.nih.gov/archive/20060905/nichsr/ehta/chapter12.html. Accessed 17 Nov 2011.
Garratt A, Schmidt L, Mackintosh A, Fitzpatrick R. Quality of life measurement: bibliographic study of patient assessed health outcome measures. Br Med J. 2002;324(7351):1417–9.
Centre for Reviews and Dissemination. NHS economic evaluation database. York: University of York; 2007.
Acknowledgments
No sources of funding were used to conduct this study or prepare this article, and the authors have no conflicts of interest that are directly relevant to its content. The opinions expressed in this article are those of the authors. Dr. Mark Harrison as corresponding author acts as guarantor for the overall content.
Author Contributions
MH, RM and ND were involved in the conception, planning and completion of the systematic review, and the subsequent interpretation of the results of the review. MH, RM and ND wrote the paper and contributed to the subsequent review and revisions of drafts. MH had the idea for the paper. All authors approved the final submitted version.
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Meacock, R., Dale, N. & Harrison, M.J. The Humanistic and Economic Burden of Systemic Lupus Erythematosus. PharmacoEconomics 31, 49–61 (2013). https://doi.org/10.1007/s40273-012-0007-4
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DOI: https://doi.org/10.1007/s40273-012-0007-4