Economic burden of systemic sclerosis: systematic review
https://doi.org/10.17749/2070-4909/farmakoekonomika.2020.041
Abstract
Background. Systemic sclerosis (SSc) is a severe orphan disease, one of the systemic pathologies of connective tissue. This disease has a significant negative impact on the patient’s quality of life and has a high mortality rate. Treatment of its various complications imposes a great financial burden on the healthcare system. The difficulties of daily functioning and social adaptation and the overall burden of SSc for patients, as well as their caregivers, also contribute to the economic component of the disease.
Aim. To assess the social and economic burden of SSc.
Materials and methods. A systematic review was conducted according to the PRISMA guidelines using predefined PICO(S) criteria. The search was carried out in December 2019 using the MeSH terms in the Embase, MEDLINE / PubMed, Cochrane library databases. The publication date range was 10 years. To identify Russian-language studies, an additional search was conducted in eLIBRARY.ru and the Internet network. The evidence levels of the included studies were determined.
Results. A total of 934 studies were identified from all databases; 53 publications were selected for eligibility; 9 of which were included in the final review. There were no studies identified to assess the burden of SSc in Russia, so the evaluations were based on foreign studies. The estimates of the annual direct costs per patient with SSc for the past decade were almost similar in different countries: 5 038 Canadian dollars in Canada, 11 607 Australian dollars in Australia, 17 365-22 016 US dollars in the USA, and 1 413-17 300 Euros in Europe (an average of about 8 000 euros). The cost structure was dominated by direct medical costs for hospitalization and drug therapy and indirect costs were mostly associated with the loss of productivity and early retirement. The costs associated with the diffuse cutaneous form of SSc were statistically higher if compared to the costs for the limited form of the disease. Among the clinical manifestations of the disease, lung lesions and gastrointestinal problems made the largest contribution to the economic burden.
Conclusion. SSc is associated with significant healthcare resource use compared to the general population. The economic burden of SS has grown significantly in recent years, and this trend is global. At the same time, it is difficult to evaluate the disease costs in Russia due to a lack of information on the patient population.
About the Authors
D. L. KlabukovaRussian Federation
Daria L. Klabukova – PhD in Biological Sciences, Leading Researcher, Department of Clinical and Economic Analysis, RSCI SPIN-code: 1567-0007, 4 lit. A Karbysheva Str., Krasnogorsk 143403, Russia
V. S. Krysanova
Russian Federation
Vera S. Krysanova – Researcher, Department of Clinical and Economic Analysis, RSCI SPIN-code: 6433-2420, 4 lit. A Karbysheva Str., Krasnogorsk 143403, Russia
T. N. Ermolaeva
Russian Federation
Tatiana N. Ermolaeva – Head of Department of Clinical and Economic Analysis, 4 lit. A Karbysheva Str., Krasnogorsk 143403, Russia
M. V. Davydovskaya
Russian Federation
Maria V. Davydovskaya – MD, Dr Sci Med, Deputy Director for Science; Professor, RSCI SPIN-code: 1083-8789, 4 lit. A Karbysheva Str., Krasnogorsk 143403, Russia; 1 Ostrovityanova Str., Moscow 117997, Russia
K. A. Kokushkin
Russian Federation
Konstantin A. Kokushkin – Director, 4 lit. A Karbysheva Str., Krasnogorsk 143403, Russia
References
1. Desbois A.C., Cacoub P. Systemic sclerosis: An update in 2016. Autoimmunity Reviews. 2016; 15 (5), 417–426. https://doi.org/10.1016/j.autrev.2016.01.007.
2. Ingegnoli F., Ughi N., Mihai C. Update on the epidemiology, risk factors, and disease outcomes of systemic sclerosis. Best Pract Res Clin Rheumatol. 2018; 32 (2): 223–240. https://doi.org/10.1016/j.berh.2018.08.005.
3. Freire M., Rivera A., Sopeña B., et al. Clinical and epidemiological differences between men and women with systemic sclerosis: a study in a Spanish systemic sclerosis cohort and literature review. Clin Exp Rheumatol. 2017; 35 Suppl 106 (4): 89–97.
4. Guseva N.G. Systemic scleroderma. In the book: Sigidin Y.A., Guseva N.G., Ivanova MM. Diffuse connective tissue diseases. Moscow. 2004; 341–57.
5. Anan'eva L.P. New classification criteria for systemic scleroderma (lecture). Nauchno-prakticheskaya revmatologiya (in Russ). 2013; 51 (5): 539–44.
6. van den Hoogen F., Khanna D., Fransen J., et al. 2013 classification criteria for systemic sclerosis: an American college of rheumatology/European league against rheumatism collaborative initiative. Ann Rheum Dis. 2013; 72 (11): 1747–1755. https://doi.org/10.1136/annrheumdis-2013-204424.
7. Starovoitova M.N., Desinova O.V., Koneva O.A. Autoantibody profile in systemic scleroderma. Nauchno-prakticheskaya revmatologiya (in Russ). 2016; 54 (4): 418–23.
8. Wollheim F.A. Classification of systemic sclerosis. Visions and reality. Rheumatology . 2005; 44 (10): 1212–1216. https://doi.org/10.1093/rheumatology/keh671.
9. Elhai M., Meune C., Boubaya M., et al. Mapping and predicting mortality from systemic sclerosis. Ann Rheum Dis. 2017; 76 (11): 1897–1905. https://doi.org/10.1136/annrheumdis-2017-211448.
10. Tyndall A.J., Bannert B., Vonk M., et al. Causes and risk factors for death in systemic sclerosis: a study from the EULAR Scleroderma Trials and Research (EUSTAR) database. Ann Rheum Dis. 2010; 69 (10): 1809–15. https://doi.org/10.1136/ard.2009.114264.
11. Kowal-Bielecka O., Fransen J., Avouac J., et al. Update of EULAR recommendations for the treatment of systemic sclerosis. Ann Rheum Dis. 2017 Aug; 76 (8): 1327–1339. https://doi.org/10.1136/annrheumdis-2016-209909.
12. Nasonov E.L. Rheumatology: clinical recommendations. Edited by E.L. Nasonov. Moscow. 2017; 464 s.
13. Anan'eva L.P., Alekperov L.T., Volkov A.V., Desinova O.V., Koneva O.A., Starovoitova M.N., Sukhareva M.L. Clinical recommendations. Progressive systemic sclerosis (systemic scleroderma). Association of Rheumatologists of Russia. Ministry of Health of the Russian Federation. 2016.
14. Khanna D., Hays RD., Furst DE. Functional disability and other health-related quality-of-life domains: points to consider for clinical trials in systemic sclerosis. Rheumatology. 2017; 56 (suppl 5): 17–22. https://doi.org/10.1093/rheumatology/kex193.
15. Hudson M., Thombs B.D., Steele R., et al. Quality of life in patients with systemic sclerosis compared to the general population and patients with other chronic conditions. J Rheumatol. 2009; 36 (4): 768–772. https://doi.org/10.3899/jrheum.080281.
16. Bretterklieber A., Painsi C., Avian A., Wutte N., Aberer E. Impaired quality of life in patients with systemic sclerosis compared to the general population and chronic dermatoses. BMC Res Notes. 2014; 7: 594. https://doi.org/10.1186/1756-0500-7-594.
17. Almeida C., Almeida I., Vasconcelos C. Quality of life in systemic sclerosis. Autoimmunity Reviews. 2015; 14 (12): 1087–1096. https://doi.org/10.1016/j.autrev.2015.07.012.
18. Pellar R.E., Tingey T.M., Pope E. Patient-Reported Outcome Measures in Systemic Sclerosis (Scleroderma). Rheum Dis Clin N Am. 2016; 42 (2): 301–316. https://doi.org/10.1016/j.rdc.2016.01.003.
19. Ingegnoli F., Carmona L., Castrejon I. Systematic review of systemic sclerosis-specific instruments for the EULAR Outcome Measures Library: An evolutional database model of validated patient-reported outcomes. Semin Arthritis Rheum. 2017; 46 (5); 609–614. https://doi.org/10.1016/j.semarthrit.2016.10.002.
20. Ignatyeva V.I., Avxentyeva M.V. The analysis of methodologic characteristics of researches on social and economic burden of diseases in Russia in the frames of development of standard cost of illness methodology for the health technology assessment. FARMAKOEKONOMIKA. Modern Pharmacoeconomic and Pharmacoepidemiology. 2014; 7 (3): 3–11 (In Russ.).
21. Moher D., Liberati A., Tetzlaff J., Altman D.G. PRISMA Group. Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. PLoS M ed. 2009; 6 (7): e1000097. https://doi.org/10.1371/journal.pmed.1000097.
22. Higgins J.P.T., Green S. (editors). Cochrane Handbook for Systematic Reviews of Interventions Version 5.1.0 [updated March 2011]. The Cochrane Collaboration. 2011. [Electronic resource]. URL: https://training.cochrane.org/handbook/current/chapter-20#section-20-2-3-2. Accessed: 25.09.2019.
23. Decree of the Government of the Russian Federation of 08.28.2014 No. 871 (as amended by Decisions of the Government of the Russian Federation of 12.06.2017 N 700, of 29.10.2018 N 1283) “On approval of the Rules for the formation of lists of drugs for medical use and the minimum range of drugs required to provide medical care" (In Russ.).
24. GOST R 57525-2017. Clinical and economic research. General requirements. Approved and enforced by Order of the Federal Agency for Technical Regulation and Metrology of July 6, 2017 N 655-st. (In Russ.).
25. Morrisroe K., Stevens W., Sahhar J., et al. Quantifying the direct public health care cost of systemic sclerosis: A comprehensive data linkage study. Medicine. 2017; 96 (48): e8503. https://doi.org/10.1097/MD.0000000000008503.
26. Zhou Z., Fan Y, Tang W., et al. Economic Burden among Commercially Insured Patients with Systemic Sclerosis in the United States. J Rheumatol. 2019; 46 (8): 9 20–927. https://doi.org/10.3899/jrheum.180445.
27. López-Bastida J., Linertová R., Oliva-Moreno J., et al. Social/economic costs and health-related quality of life in patients with scleroderma in Europe. Eur J Health Econ. 2016; 17 Suppl 1: 109–117. https://doi.org/10.1007/s10198-016-0789-y.
28. Chevreul K., Brigham K.B., Gandré C., Mouthon L. BURQOL-RD Research Network. The economic burden and health-related quality of life associated with systemic sclerosis in France. Scand J Rheumatol. 2015; 44 (3): 238–246. https://doi.org/10.3109/03009742.2014.976653.
29. López-Bastida J., Linertová R., Oliva-Moreno J., et al. Social/economic costs and health-related quality of life in patients with scleroderma in Europe. Eur J Health Econ. 2016; 17 Suppl 1: 109–117. DOI: https://doi.org/10.1007/s10198-016-0789-y.
30. McCormick N., Marra C.A., Sayre E.C., Avina-Zubieta J.A. Longitudinal analysis of direct medical costs for systemic sclerosis patients: a population-based study. Arthritis Rheum. 2013; 65 (Suppl. 10): S429–30.
31. Furst D.E., Fernandes A.W., Iorga S.R., Greth W., Bancroft T. Annual medical costs and healthcare resource use in patients with systemic sclerosis in an insured population. J Rheumatol. 2012; 39 (12): 2303–2309. https://doi.org/10.3899/jrheum.120600.
32. Bernatsky S., Hudson M., Panopalis P., et al. The cost of systemic sclerosis. Arthritis Rheum. 2 009; 6 1 ( 1): 119–123. https://doi.org/10.1002/art.24086.
33. Minier T., Pentek M., Brodszky V., et al. Cost-of-illness of patients with systemic sclerosis in a tertiary care centre. Rheumatology. 2010; 49 (10): 1920–1928. https://doi.org/10.1093/rheumatology/keq165.
34. Belotti Masserini A., Zeni S., Cossutta R., Soldi A., Fantini F. Cost-of-illness in systemic sclerosis: a retrospective study of an Italian cohort of 106 patients. Reumatismo. 2003; 55: 245–55 [Italian].
35. Kim H., Cho S.K., Kim J.W, et al. An increased disease burden of autoimmune inflammatory rheumatic diseases in Korea. Semin Arthritis Rheum. 2019; S0049-0172(19)30688-2. https://doi.org/10.1016/j.semarthrit.2019.11.007.
36. Fischer A., Zimovetz E., Ling C., Esser D., Schoof N. Humanistic and cost burden of systemic sclerosis: A review of the literature. Autoimmun Rev. 2017; 16 (11): 1147–1154. https://doi.org/10.1016/j.autrev.2017.09.010.
Review
For citations:
Klabukova D.L., Krysanova V.S., Ermolaeva T.N., Davydovskaya M.V., Kokushkin K.A. Economic burden of systemic sclerosis: systematic review. FARMAKOEKONOMIKA. Modern Pharmacoeconomics and Pharmacoepidemiology. 2020;13(3):291-303. (In Russ.) https://doi.org/10.17749/2070-4909/farmakoekonomika.2020.041

This work is licensed under a Creative Commons Attribution-NonCommercial-ShareAlike 4.0 International License.