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Family Spillover Effects in Pediatric Cost-Utility Analyses

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Abstract

Background

Childhood illness can impose significant costs and health strains on family members, but these are not routinely captured by pediatric economic evaluations. This review investigated how family “spillover effects” related to costs and health outcomes are considered in pediatric cost-utility analyses (CUAs).

Methods

We reviewed pediatric CUAs published between 2000 and 2015 using the Tufts Medical Center Cost-effectiveness Analysis (CEA) Registry and the Pediatric Economic Database Evaluation (PEDE) Registry. We selected studies conducted from the societal perspective and included in both registries. We investigated how frequently family spillover was incorporated into analyses, and how the inclusion of spillover health effects and costs changed CUA results.

Results

We found 142 pediatric CUAs meeting inclusion criteria. Of those, 105 (72%) considered either family spillover costs (n = 98 time costs, n = 33 out-of-pocket costs, n = 2 caregiver healthcare costs) or health outcomes (n = 15). Twenty-four studies included 43 pairs of incremental cost-effectiveness ratios (ICERs) with and without spillover. In 19 pairs of ICERs, adding spillover changed the ICER enough to cross a common cost-effectiveness threshold (i.e., $50,000/QALY, $100,000/QALY, $150,000/QALY; values are in 2016 US$). Incorporating spillover generally made interventions more cost-effective (n = 18; 42%), or did not change CUA results enough to cross a threshold (n = 24; 56%). Including family spillover reduced ICERs by 31% ($40,000/QALY) on average.

Conclusion

Most pediatric CUAs conducted from a societal perspective include family costs but fewer include family health effects. Inclusion of family spillover effects tends to make CUA results more favorable. Future pediatric CUAs should aim to more fully incorporate the family burden of illness.

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Fig. 1

Source: Authors’ calculation based on data from: 1 Bos et al. 2003 [60]; 2 Coyle and Barbeau [61]; 3–8 Barton et al. [62]; 9–11 Coco [63]; 12–14 Goossens et al. [30]; 15 Bergman et al. [64]; 16 Luce et al. [65]; 17–20 Bilcke et al. [28]; 21 Martin et al. [33]; 22 Shim and Galvani [37]; 23 Talbird et al. [66]; 24 Tan et al. [67]; 25 Rozenbaum et al. [35]; 26 Sato et al. [36]; 27–28 Perez-Rubio et al. [34]; 29–32 Coyle et al. [29]; 33–34 Hoshi et al. [68]; 35 Suwantika et al. [38]; 36 Itzler et al. [32]; 37 Imaz et al. [31]; 38 Shiragami et al. [69]; 39 Al Awaidy et al. [27]; 40–41 van der Schans et al. [25]; 42–43 Schawo et al. [26]

Fig. 2

Source: Authors’ calculation based on data from 2 studies: 1–2 Coyle et al. [29]; 3–4 Bilcke et al. [28]

Fig. 3

Source: Authors’ calculation based on data from 17 studies: 1 Bos et al. [60]; 2 Coyle and Barbeau [61]; 3–8 Barton et al. [62]; 9–10 Coco [63]; 11–13 Goossens et al. [30]; 14 Bergman et al. [64]; 15 Luce et al. [65]; 16 Martin et al. [33]; 17 Talbird et al. [66]; 18 Tan et al. [67]; 19 Sato et al. [36]; 20–21 Hoshi et al. [68]; 22 Suwantika et al. [38]; 23 Shiragami et al. [69]; 24–25 van der Schans et al. [25]; 26–27 Bilcke et al. [28]; 28–29 Coyle et al. [29]

Fig. 4

Source: Authors’ calculation based on data from 2 studies: 1 Itzler et al. [32]; 2–3 Bilcke et al. [28]

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Data Availability Statement

In a supplementary document (Online Supplementary Material), we have included all abstracted data for a subset of 24 of the studies in our review that were used to recalculate incremental cost effectiveness ratios with and without spillover included. Abstracted data from remaining studies are available in the referenced literature.

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Acknowledgements

We would like to acknowledge Ashley Leech, PhD for her comments during the literature review process, and Peter Neumann, ScD for his comments on the manuscript.

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Authors and Affiliations

  1. Center for the Evaluation of Value and Risk in Health, Institute for Clinical Research and Health Policy Studies, Tufts Medical Center, 800 Washington St, #63, Boston, MA, 02111, USA

    Tara A. Lavelle, Brittany N. D’Cruz, Babak Mohit & Pei-Jung Lin

  2. Program of Child Health Evaluative Sciences, The Hospital for Sick Children Research Institute, Toronto, ON, Canada

    Wendy J. Ungar & Kate Tsiplova

  3. Institute for Health Policy, Management and Evaluation, University of Toronto, Toronto, ON, Canada

    Wendy J. Ungar

  4. Child Health Evaluation and Research Unit, Department of Pediatrics, University of Michigan, Ann Arbor, MI, USA

    Lisa A. Prosser

  5. Global Health Economics Outcomes Research and Epidemiology, Shire, Lexington, MA, USA

    Montserrat Vera-Llonch

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  1. Tara A. Lavelle
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Contributions

Study concept and design: Lavelle, Lin, D’Cruz, Prosser, Ungar, Mohit, Vera-Llonch; data acquisition: Tsiplova, Lin, D’Cruz, and Lavelle; drafting publication: Lavelle, Lin, D’Cruz; all authors participated in analysis or interpretation of data as well as preparation and critical revision of the manuscript.

Corresponding author

Correspondence to Tara A. Lavelle.

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Funding

This study was supported by research funding from Shire Pharmaceuticals to Tufts Medical Center.

Conflict of interest

Montserrat Vera-Llonch is an employee of Shire and owns stock/options. Tara Lavelle, Brittany D’Cruz, and Pei-Jung Lin were funded by a grant from Shire during this work. Wendy Ungar reports receiving a consulting fee from Tufts Medical Center for her contribution to this work. Babak Mohit reports receiving funds from Bayer to support his post-doctoral fellowship. Pei-Jung Lin reports personal fees from Avanir and Otsuka, outside of the scope of this work. Lisa Prosser and Kate Tsiplova report no conflicts of interest.

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Lavelle, T.A., D’Cruz, B.N., Mohit, B. et al. Family Spillover Effects in Pediatric Cost-Utility Analyses. Appl Health Econ Health Policy 17, 163–174 (2019). https://doi.org/10.1007/s40258-018-0436-0

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