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Eliciting Preferences for Prioritizing Treatment of Rare Diseases: the Role of Opportunity Costs and Framing Effects

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Abstract

Background

Understanding societal preferences regarding resource allocation in the health sector has gained importance as countries increasingly base reimbursement decisions on economic evaluations. Preference elicitation using surveys, a common practice in the health sector, is subject to a range of framing effects.

Objective

This research investigates the importance of (theoretically relevant) opportunity costs and (theoretically irrelevant) framing effects on stated preferences for prioritizing treatment of rare (orphan) diseases.

Methods

We elicited preferences from Norwegians, aged 40–67, using simple trade-off exercises. Respondents were randomised to different opportunity costs of the rare disease or to different framings of the trade-off exercises.

Results

Respondents were quite sensitive to the visual presentation of the choice problem, and, to a lesser extent, to focusing and labelling effects. Elicited preferences varied little in response to large changes in opportunity costs, suggesting scope-insensitivity among respondents.

Conclusions

Preferences for prioritizing treatment of rare diseases elicited using trade-off exercises are insensitive to (theoretically relevant) opportunity costs, but sensitive to (theoretically irrelevant) framing effects.

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Notes

  1. We exclude from the current discussion two additional survey versions: The first tested whether a slight modification to the wording of the survey used in Desser [15] had an effect on responses. The second served as a bridge between survey versions V2 and V5 by explicitly providing total numbers of patients treated on the slide bar used for allocation choices in V2, as this information was provided in the random bar chart used in V5 (see below). There were no significant differences in the responses in either case. A complete set of all versions of the current survey is available upon request.

  2. This represents the total number of respondents for the nine survey versions to which individuals were actually randomized. Because we exclude two survey versions from our discussion here, we report on a total sample of 2,137.

  3. TNS Gallup is unable to track how many of the 5,593 individuals who were invited to participate actually received the email invitation.

  4. This information is available because all respondents completed the allocation question for the ‘equal-cost’ scenario. See ESM 3.

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Acknowledgements

The authors thank John Cairns, Ivar Sønbø Kristiansen, Eline Aas, participants at the Health Economics Bergen/Health Economics Research Network at University of Oslo (HEB/HERO) Workshop, 2012, two anonymous reviewers for their insightful comments, and Chris McCabe for his suggestion of a version in which the common disease was more costly. This research was supported by a grant from the Norwegian Research Council. ASD, JAO and SG each report no conflicts of interest. ASD designed the survey, conducted the statistical analysis and drafted the paper and revisions. JAO and SG made important contributions to survey design, interpretation of results and improving the paper. All authors take responsibility for the work. ASD is the overall guarantor.

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Authors and Affiliations

  1. Department of Health Management and Health Economics, University of Oslo, PO Box 1089, Blindern, 0317, Oslo, Norway

    Arna S. Desser & Sverre Grepperud

  2. Norwegian Knowledge Centre for the Health Services, PO Box 7004, St Olavs Plass, 0130, Oslo, Norway

    Arna S. Desser

  3. Department of Community Medicine, University of Tromsø, 9019, Tromsø, Norway

    Jan Abel Olsen

Authors
  1. Arna S. Desser
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  2. Jan Abel Olsen
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  3. Sverre Grepperud
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Correspondence to Arna S. Desser.

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Desser, A.S., Olsen, J.A. & Grepperud, S. Eliciting Preferences for Prioritizing Treatment of Rare Diseases: the Role of Opportunity Costs and Framing Effects. PharmacoEconomics 31, 1051–1061 (2013). https://doi.org/10.1007/s40273-013-0093-y

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