Abstract
BACKGROUND AND OBJECTIVES: Case 2 best-worst scaling (BWS-2) is an increasingly popular method to elicit patient preferences. Because BWS-2 potentially has a lower cognitive burden compared with discrete choice experiments, the aim of this study was to compare treatment preference weights and relative importance scores.
METHODS: Patients with neuromuscular diseases completed an online survey at two different moments in time, completing one method per occasion. Patients were randomly assigned to either first a discrete choice experiment or BWS-2. Attributes included: muscle strength, energy endurance, balance, cognition, chance of blurry vision, and chance of liver damage. Multinomial logit was used to calculate overall relative importance scores and latent class logit was used to estimate heterogeneous preference weights and to calculate the relative importance scores of the attributes for each latent class.
RESULTS: A total of 140 patients were included for analyses. Overall relative importance scores showed differences in attribute importance rankings between a discrete choice experiment and BWS-2. Latent class analyses indicated three latent classes for both methods, with a specific class in both the discrete choice experiment and BWS-2 in which (avoiding) liver damage was the most important attribute. Ex-post analyses showed that classes differed in sex, age, level of education, and disease status. The discrete choice experiment was easier to understand compared with BWS-2.
CONCLUSIONS: This study showed that using a discrete choice experiment and BWS-2 leads to different outcomes, both in preference weights as well as in relative importance scores, which might have been caused by the different framing of risks in BWS-2. However, a latent class analysis revealed similar latent classes between methods. Careful consideration about method selection is required, while keeping the specific decision context in mind and pilot testing the methods.
Original language | English |
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Pages (from-to) | 239-253 |
Journal | Patient-patient centered outcomes research |
Volume | 16 |
Issue number | 3 |
Early online date | 13 Feb 2023 |
DOIs | |
Publication status | Published - May 2023 |
Bibliographical note
© 2023. The Author(s).Funding
We thank all patient organizations supporting this study: Muscular Dystrophy UK, Myotonic Dystrophy Support Group; Cure DM CIC; the Lily Foundation for Mitochondrial Disorders; United Mitochondrial Disease Foundation, MitoCanada; Muscular Dystrophy Canada; Muscular Dystrophy Association; MDF; and Muscular Dystrophy New Zealand. We thank members of the Wellcome Research Centre for Mitochondrial Research who provided input and expertise into the design of this study. We thank the UK Myotonic Dystrophy Patient Registry at Newcastle University for facilitating participant recruitment and supporting with the pilot stages of the study. We thank the Newcastle University Ethics Committee for revising the ethical aspect of this project. Mito Foundation; Cure DM CIC; (Lily); (UMDF); Muscular Dystrophy Canada (MDC); Muscular Dystrophy Association (MDA); Myotonic org; MitoCanada; (MDNZ); and, via the UK Myotonic Dystrophy Patient Registry and the New Zealand Neuromuscular Disease Patient Registry.
Funders | Funder number |
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Cure DM CIC | |
MitoCanada | |
Muscular Dystrophy New Zealand | |
Myotonic Dystrophy Support Group | |
New Zealand Neuromuscular Disease Patient Registry | |
UK Myotonic Dystrophy Patient Registry | |
Muscular Dystrophy Association | |
United Mitochondrial Disease Foundation | |
Muscular Dystrophy UK | |
Muscular Dystrophy Canada | |
Newcastle University |