Economic Burden and Quality-of-Life Effects of Chronic Lymphocytic Leukemia: A Systematic Review of the Literature
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Chronic lymphocytic leukemia (CLL) is the most prevalent type of leukemia in the Western hemisphere. The disease affects quality of life (QOL) and poses an economic burden on patients, payers, and society. The objective of this review was to quantify the economic burden and quality-of-life effects and identify the gaps that should be addressed by future research.
Free-text and subject heading searches in MEDLINE, EMBASE, the Cochrane Library, the University of York Centre for Reviews and Dissemination Database, and the Web of Science Core Collection database were conducted to identify observational and interventional studies reporting costs and/or quality-of-life effects published up to 2 October 2015. Studies were included irrespective of whether they were conducted prospectively or retrospectively. The focus population consisted of adult patients aged 18 years or older affected by any stage of CLL. Studies were included regardless of whether the underlying population was treated at baseline or not. Risk of bias was assessed using a quality checklist developed by the Effective Public Health Practice Project for (randomized) controlled trials, cohort studies, and cross-sectional studies. Economic evaluations were rated using a checklist developed by Stuhldreher et al. (Int J Eat Disord 45:476–91, 2012).
From 2451 records identified, 27 studies were found to be eligible for inclusion. Studies were heterogeneous with respect to methodology, perspective, and data used. Annual direct costs per person ranged from US$4491 in Germany to US$43,913 in the USA. The share of costs attributable to drug treatment varied between 26.2 and 79 %. Indirect costs amounted to US$4208. Severity of disease was a predictor for quality of life, whereas differences by age and sex were mainly present in subdomains. Comparisons of treated and untreated populations resulted in an increase of quality of life in favor of treated populations in the long-term perspective. Differences between treatments were small. Consequently, cost effectiveness in decision–analytic models did not depend on whether quality of life or survival are used to describe the benefits of treatment.
Although the quantity and the quality of health economic and quality-of-life evidence have substantially increased, there is still a need for studies that take a patient or societal perspective. Factors that influence costs and the quality of life of patients seem to be well-established, while longitudinal lifetime cost studies at the population level are still scarce.
KeywordsChronic Lymphocytic Leukemia Alemtuzumab Chlorambucil Bendamustine Chronic Lymphocytic Leukemia Patient
Compliance with Ethical Standards
SF, CRB, and TS did not receive specific funding for this study. SF, CRB, and TS declare that they have no conflicting interests, commercial, personal, political, intellectual or otherwise, relating to this manuscript.
No relevant financial relationship exists.
SF, CRB, and TS designed the study, carried out the methodological framework, collected the data, and reviewed the studies. SF, CRB, and TS contributed to the writing, interpretation of the results, and revision of the manuscript.
- 3.Orphanet. Prevalence of rare diseases: bibliographic data. 2014 May. Report No.: 1. http://www.orpha.net/orphacom/cahiers/docs/GB/Prevalence_of_rare_diseases_by_alphabetical_list.pdf. Accessed 1 Nov 2015.
- 4.European Commission. European Commission Regulation (EC) no. 141/2000 of the European Parliament and the Council of 16 December 1999 on orphan medicinal products. 1999 Dec 16. http://ec.europa.eu/health/files/eudralex/vol-1/reg_2000_141_cons-2009-07/reg_2000_141_cons-2009-07_en.pdf. Accessed 11 Dec 2015.
- 5.US Government Publishing Office. US Code § 360bb—designation of drugs for rare diseases or conditions. 2011 Jan 7. https://www.gpo.gov/fdsys/granule/USCODE-2010-title21/USCODE-2010-title21-chap9-subchapV-partB-sec360bb/content-detail.html. Accessed 11 Dec 2015.
- 6.Howlader N, Noone A, Krapcho M, Garshell J, Miller D, Altekruse S, et al., editors. SEER cancer statistics review, 1975-2011. Bethesda: National Cancer Institute: 2014. http://seer.cancer.gov/csr/1975_2011. Accessed 11 Dec 2015.
- 11.National Cancer Institute. SEER cancer statistics. Bethesda: National Cancer Institute; 2008.Google Scholar
- 18.Higgins JP, Green S, editors. Cochrane handbook for systematic reviews of interventions version 5.1.0 [updated March 2011]. The Cochrane Collaboration; 2011. http://www.cochrane-handbook.org. Accessed 1 Nov 2015.
- 26.Eichhorst BF, Busch R, Obwandner T, Kuhn-Hallek I, Herschbach P, Hallek M, et al. Health-related quality of life in younger patients with chronic lymphocytic leukemia treated with fludarabine plus cyclophosphamide or fludarabine alone for first-line therapy: a study by the German CLL Study Group. J Clin Oncol. 2007;25:1722–31.CrossRefPubMedGoogle Scholar
- 37.Mulligan SP, Karlsson K, Stromberg M, Jonsson V, Gill D, Hammerstrom J, et al. Cladribine prolongs progression-free survival and time to second treatment compared to fludarabine and high-dose chlorambucil in chronic lymphocytic leukemia. Leuk Lymphoma. 2014. http://informahealthcare.com/doi/abs/10.3109/10428194.2014.893306. Accessed 1 Nov 2015.
- 46.De Wreede LC, Watson M, van Os M, Milligan D, van Gelder M, Michallet M, et al. Improved relapse-free survival after autologous stem cell transplantation does not translate into better quality of life in chronic lymphocytic leukemia: lessons from the randomized European Society for Blood and Marrow Transplantation-Intergroup study. Am J Hematol. 2014;89:174–80.CrossRefPubMedGoogle Scholar
- 47.Husereau D, Drummond M, Petrou S, Carswell C, Moher D, Greenberg D, et al. Consolidated Health Economic Evaluation Reporting Standards (CHEERS)—explanation and elaboration: a report of the ISPOR Health Economic Evaluation Publication Guidelines Good Reporting Practices Task Force. Value Health. 2013;16:231–50.CrossRefPubMedGoogle Scholar
- 48.Fayers P, Aaronson N, Bjordal K, Groenvold M, Curran D, Bottomley A, et al. The EORTC QLQ-C30 Scoring Manual. 3rd ed. Brussels: European Organisation for Research and Treatment of Cancer; 2001.Google Scholar
- 49.Dapueto JJ, Francolino C, Servente L, Chang C-H, Gotta I, Levin R, et al. Evaluation of the Functional Assessment of Cancer Therapy-General (FACT-G) Spanish version 4 in South America: classic psychometric and item response theory analyses. Health Qual Life Outcomes. 2003;1:32.CrossRefPubMedPubMedCentralGoogle Scholar