The impact of improving haemophilia A management within the Spanish National Healthcare System: a social return on investment analysis

Inmaculada Soto,Teresa Martín Lorenzo,Beatriz Gil,Álvaro Hidalgo-Vega,Daniel-Aníbal García-Diego,José Mateo,María Merino,Elena Ruiz-Beato,Yoana Ivanova,Paulina Maravilla-Herrera

doi:10.1186/s12913-021-07447-4

Abstract

BackgroundHaemophilia A (HA) has been associated with poor health-related quality of life and a large economic burden, accentuated by severity, arthropathy, and inhibitors. To meet global standards of care, the management of HA should align with the principles of care outlined by the World Federation of Haemophilia. The aims of the present study were to establish a set of proposals to improve HA management within the Spanish National Health System (SNHS) and to estimate the impact its hypothetical implementation would generate from a clinical, healthcare, economic, and social perspective.MethodsA multidisciplinary group of experts agreed on a set of 15 proposals to improve HA management within the SNHS. Thereafter, a forecast-type Social Return on Investment analysis was carried out to estimate the impact of implementing this set of proposals within the SNHS over a one-year timeframe, in relation to the required investment.ResultsThis study estimated that the implementation of the complete set of 15 proposals would require a total investment of 2.34 M€ and have a total impact of 14.60 M€. Accordingly, every euro invested in the complete set of 15 proposals would yield a social return of €6.23 (€3.37 in the worst-case scenario and €9.69 in the best-case scenario) of both tangible and intangible nature in similar proportions (45.71 and 54.29%, respectively).ConclusionsThese results can be used to inform policy and practice such that interventions that may potentially improve current public health challenges associated with the management of HA may be implemented.

Highlights

Haemophilia A (HA) has been associated with poor health-related quality of life and a large economic burden, accentuated by severity, arthropathy, and inhibitors
The Multidisciplinary Expert Committee (MEC) agreed on a set of 15 proposals to improve HA management within the Spanish National Health System (SNHS) (Table 1)
Establishing the impact To determine the proportion of each outcome that could be ascribed to the activities within each proposal and avoid over claiming, outcomes were individually adjusted by removing deadweight, attribution, and displacement from their total value

Summary

Introduction

Haemophilia A (HA) has been associated with poor health-related quality of life and a large economic burden, accentuated by severity, arthropathy, and inhibitors. While patients with mild HA (baseline level of circulating factor VIII 5 to 40 IU/dL or 5 to 40% of normal) rarely experience bleeding unless subjected to major trauma or surgery, patients with moderate (1 to 5 IU/dL or 1 to 5% of normal) or severe HA (< 1 IU/dL or < 1% of normal) more commonly experience spontaneous bleeding into joints, muscles, and internal organs throughout their lifetime [1, 2]. Haemophilia has been associated with impaired health-related quality of life (HRQoL) and a large economic burden [6, 7], further accentuated by increased severity [8, 9], the development of inhibitors [9,10,11], and the presence of arthropathy [5, 12], among others [7]

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