Economic and Humanistic Burden of Moderate and Severe Hemophilia A and B in Spain: Real-World Evidence Insights from the CHESS II Study

Author:

Peral Carmen1,De Lossada Juste Alfonso1,Lwoff Nadia1,Espinoza-Cámac Nataly2,Casado Miguel Ángel2,Burke Tom34,Alvir Jose5,Thakkar Sheena6,Ferri Grazzi Enrico3

Affiliation:

1. Pfizer S.L.U., Madrid, Spain

2. Pharmacoeconomics and Health Outcomes Research Iberia (Spain)

3. HCD Economics, Daresbury, UK

4. University of Chester, Chester, UK

5. Pfizer Inc., New York, New York, USA

6. Pfizer Inc., New York, NY, USA

Abstract

Background: Hemophilia is a congenital disorder characterized by deficiency or absence of clotting factor VIII in hemophilia A (HA) or clotting factor IX in hemophilia B (HB), resulting in frequent, repeated, and prolonged spontaneous or traumatic bleeding into joints or soft tissue. Severity is classified by the patient’s baseline level of clotting factor activity as mild (>5%-40%), moderate (1%-5%), or severe (<1%). In Spain, there is limited information on the societal economic burden of disease. Objective: To estimate the economic and humanistic burden of disease in adult patients with non-inhibitor moderate and severe HA and HB in Spain. Methods: Spanish data from the CHESS II study (2018-2020) on patients’ clinical characteristics, health-related quality of life (HRQoL) and hemophilia-related healthcare resource utilization were analyzed. Economic burden was determined by estimating condition-related annual per-patient direct (medical and nonmedical) and indirect costs, stratified according to hemophilia type and severity and presented as 2022 Euros. HRQoL was assessed via the EQ-5D-5L. Results: Of 341 patients in the Spanish CHESS II cohort, 288 patients met the inclusion criteria: 181 had HA (37% [n = 66] moderate and 63% [n=115] severe) and 107 had HB (26% [n = 28] moderate and 74% [n = 79] severe). Mean annual direct cost was higher in HB than in HA, and higher in severe than in moderate patients, resulting in an annual cost/patient of €17 251 (moderate HA), €17 796 (moderate HB), €116 767 (severe HA) and €206 996 (severe HB). The main direct cost component in all groups except moderate HA was factor replacement therapy. Mean per-patient indirect cost was €4089 (moderate HA), €797 (moderate HB), €8633 (severe HA) and €8049 (severe HB). Finally, the mean total cost (direct and indirect) for moderate and severe patients were €91 017 (HA) and €163 924 (HB). EQ-5D-5L [SD] scores were lower in patients with severe HA (0.77 [0.18]) and severe HB (0.70 [0.22]) compared with patients with moderate HA (0.81 [0.15]) and moderate HB (0.86 [0.17]). Conclusions: Independently of the type of hemophilia, greater condition severity was associated with increased costs and a decrease in HRQoL.

Publisher

The Journal of Health Economics and Outcomes Research

Reference33 articles.

1. Guías españolas para el manejo del paciente con hemofilia;M.T. Álvarez,2022

2. The impact of extended half-life factor concentrates on prophylaxis for severe hemophilia in the United States;Lynn M. Malec;American Journal of Hematology,2020

3. Effect of late prophylaxis in hemophilia on joint status: a randomized trial;M.J. Manco-Johnson;Journal of Thrombosis and Haemostasis,2017

4. Interrelationship between depression, anxiety, pain, and treatment adherence in hemophilia: results from a US cross-sectional survey;Michelle L. Witkop;Patient Preference and Adherence,2019

5. The relationship between target joints and direct resource use in severe haemophilia;Jamie O’Hara;Health Economics Review,2018

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