title: Economic Burden and Quality-of-Life Effects of Chronic Lymphocytic Leukemia: A Systematic Review of the Literature.
abstract: BACKGROUND
  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.
  
  
  METHODS
  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).
  
  
  RESULTS
  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.
  
  
  CONCLUSIONS
  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.
authors.affiliation: Hamburg Center for Health Economics, Universität Hamburg, Esplanade 36, 20354, Hamburg, Germany.
authors.forename: Simon
authors.initials: S
authors.lastname: Frey
authors.affiliation: Hamburg Center for Health Economics, Universität Hamburg, Esplanade 36, 20354, Hamburg, Germany.|Center for Gerontology and Health Care Research, School of Public Health, Brown University, Providence, RI, USA.
authors.forename: Carl R
authors.initials: CR
authors.lastname: Blankart
authors.affiliation: Hamburg Center for Health Economics, Universität Hamburg, Esplanade 36, 20354, Hamburg, Germany. tom.stargardt@wiso.uni-hamburg.de.
authors.forename: Tom
authors.initials: T
authors.lastname: Stargardt
mesh_terms: D017281:Cost of Illness; D003362:Cost-Benefit Analysis; D006801:Humans; D015451:Leukemia, Lymphocytic, Chronic, B-Cell / Q000188:drug therapy / Q000191:economics* / Q000523:psychology; D011788:Quality of Life; D019057:Quality-Adjusted Life Years