Establishing disability weights for congenital pediatric surgical conditions: a multi-modal approach

BACKGROUND: Burden of disease (BoD) as measured by Disability-Adjusted Life Years (DALYs) is one of the criteria for priority-setting in health care resource allocation. DALYs incorporate disability weights (DWs), which are currently expert-derived estimates or non-existent for most pediatric surgical conditions. The objective of this study is to establish DWs for a subset of key pediatric congenital anomalies using a range of health valuation metrics with caregivers in both high- and low-resource settings. METHOD: We described 15 health states to health professionals (physicians, nurses, social workers, and therapists) and community caregivers in Kenya and Canada. The health states summaries were expert- and community-derived, consisting of a narrated description of the disease and a functional profile described in EQ-5D-5 L style. DWs for each health state were elicited using four health valuation exercises (preference ranking, visual analogue scale (VAS), paired comparison (PC), and time trade-off (TTO)). The PC data were anchored internally to the TTO and externally to existing data to yield DWs for each health state on a scale from 0 (health) to 1 (dead). Any differences in DWs between the two countries were analyzed. RESULTS: In total, 154 participants, matched by profession, were recruited from Kijabe, Kenya (n = 78) and Hamilton, Canada (n = 76). Overall calculated DWs for 15 health states ranged from 0.13 to 0.77, with little difference between countries (intra-class coefficient 0.97). However, DWs generated in Kenya for severe hypospadias and undescended testes were higher than Canadian-derived DWs (p = 0.04 and p < 0.003, respectively). CONCLUSIONS: We have derived country-specific DWs for pediatric congenital anomalies using several low-cost methods and inter-professional and community caregivers. The TTO-anchored PC method appears best suited for future use. The majority of DWs do not appear to differ significantly between the two cultural contexts and could be used to inform further work of estimating the burden of global pediatric surgical disease. Care should be taken in comparing the DWs obtained in the current study to the existent list of DWs because methodological differences may impact on their compatibility. ELECTRONIC SUPPLEMENTARY MATERIAL: The online version of this article (doi:10.1186/s12963-017-0125-5) contains supplementary material, which is available to authorized users.