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. 2022 Feb;25(2):276-287.
doi: 10.1016/j.jval.2021.08.002. Epub 2021 Sep 4.

Health State Utilities for Sickle Cell Disease: A Catalog Prepared From a Systematic Review

Boshen Jiao  1 , Anirban Basu  2 , Scott Ramsey  3 , Joshua Roth  4 , M A Bender  5 , Dalyna Quach  6 , Beth Devine  7
Affiliations

Health State Utilities for Sickle Cell Disease: A Catalog Prepared From a Systematic Review

Boshen Jiao et al. Value Health. 2022 Feb.

Abstract

Objectives: Sickle cell disease (SCD) is a complex, chronic condition that impairs health-related quality of life of affected individuals and their caregivers. As curative therapies emerge, comprehensive cost-effectiveness models will inform their value. These models will require descriptions of health states and their corresponding utility values that accurately reflect health-related quality of life over the disease trajectory. The objectives of this systematic review were to develop a catalog of health state utility (HSU) values for SCD, identify research gaps, and provide future directions for preference elicitation.

Methods: Records were identified through searches of PubMed and Embase, Tufts Medical Center Cost-Effectiveness Analysis Registry, reference lists of relevant articles, and consultation with SCD experts (2008-2020). We removed duplicate records and excluded ineligible studies. For included studies, we summarized the study characteristics, methods used for eliciting HSUs, and HSU values.

Results: Five studies empirically elicited utilities using indirect methods (EQ-5D) (n = 3) and Short Form-6 Dimension (n = 2); these represent health states associated with general SCD (n = 1), SCD complications (n = 2), and SCD treatments (n = 3). Additionally, we extracted HSUs from 7 quality-adjusted life-years-based outcome research studies. The HSU among patients with general SCD without specifying complications ranged from 0.64 to 0.887. Only 36% of the HSUs used in the quality-adjusted life-year-based outcomes research studies were derived from individuals with SCD. No study estimated HSUs in caregivers.

Conclusions: There is a dearth of literature of HSUs for use in SCD models. Future empirical studies should elicit a comprehensive set of HSUs from individuals with SCD and their caregivers.

Keywords: cost-effectiveness analysis; health state utility; sickle cell disease; systematic review.

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Figures

Figure 1.
Figure 1.
Preferred Reporting Items for Systematic Reviews and Meta-Analyses flow diagram of studies included in this systematic review and reasons for exclusion.
Figure 2.
Figure 2.
Distribution of sources of the health state utilities used in the outcome research studies. SCD indicates sickle cell disease.
See this image and copyright information in PMC

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