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Background The aim of this study was to determine the economic burden and health-related quality of life (HRQOL) of patients with Spinal Muscular Atrophy (SMA) and their caregivers in Spain. Methods This was a cross-sectional and retrospective study of patients diagnosed with SMA in Spain. We adopted a bottom up, prevalence approach design to study patients with SMA. The patient’s caregivers completed an anonymous questionnaire regarding their socio-demographic characteristics, use of healthcare services and non-healthcare services. Costs were estimated from a societal perspective (including healthcare costs and non-healthcare costs), and health-related quality of life (HRQOL) was assessed using the EQ-5D questionnaire. The main caregivers also answered a questionnaire on their characteristics and on their HRQOL. Results A total of 81 caregivers of patients with different subtypes of SMA completed the questionnaire. Based on the reference unitary prices for 2014, the average annual costs per patient were € 33,721. Direct healthcare costs were € 10,882 (representing around 32.3% of the total cost) and the direct non-healthcare costs were € 22,839 (67.7% of the total cost). The mean EQ-5D social tariff score for patients was 0.16, and the mean score of the EQ-5D visual analogue scale was 54. The mean EQ-5D social tariff score for caregivers was 0.49 and their mean score on the EQ-5D visual analogue scale was 69. Conclusion The results highlight the burden that SMA has in terms of costs and decreased HRQOL, not only for patients but also for their caregivers. In particular, the substantial social/economic burden is mostly attributable to the high direct non-healthcare costs.

Background The economic value attributed by users of health services in public health systems can be useful in planning and evaluation. This value can differ from the perspectives of Willingness to Pay (WTP) and Willingness to Accept [Compensation] (WTA). Our objective was to study the perceptions of the patient about the service provided by the family physician by means of the WTA/WTP ratio. Methods An economic evaluation study by the Contingent Valuation Method was designed. Interviews were conducted with 451 subjects at six health centres (four urban and two rural) in areas with different socioeconomic characteristics. A payment card was used to measure the WTP and WTA. Other characteristics of the subject or service that could influence these responses were collected. An explicative model was constructed to study the WTA/WTP relationship. Results Four hundred and four subjects (89.6%) expressed a WTP and WTA different from zero. The WTA/WTP quotient showed a median of 1.55 (interquartile range 1-3.08) and a mean of 3.30 (IC 95%: 2.84-3.75). The WTA/WTP ratio increases with age and in low-income areas. It decreases in professional groups with more specialized activities, with growing family income, and in the chronically ill. Other characteristics related to the perception of state of health, accessibility to the service, satisfaction, or perception of risk were not explicative. Conclusions Subjects who were older and had a less favourable socioeconomic situation expressed a higher WTA/WTP ratio when valuing the visit to the family physician. These characteristics could identify a profile of "aversion to loss" with respect to this service.