The socioeconomic burden of patients affected by hemophilia with inhibitors

Lucia S D'Angiolella, Paolo A Cortesi, Angiola Rocino, Antonio Coppola, Hamisa J Hassan, Adele Giampaolo, Luigi P Solimeno, Alessandra Lafranconi, Mariangela Micale, Sveva Mangano, Giacomo Crotti, Federica Pagliarin, Giancarlo Cesana, Lorenzo G Mantovani

Research output: Contribution to journalReview article

Abstract

Hemophilia is associated with a high financial burden on individuals, healthcare systems, and society. The development of inhibitors significantly increases the socioeconomic burden of the diseases. This study aimed to review and describe the burden of hemophilia with inhibitors, providing a reference scenario to assess the impact of new products in the real word. Two systematic literature reviews were performed to collect data on (i) health economics and (ii) health-related quality of life evidences in hemophilic patients with inhibitors. The costs associated with patients with hemophilia and inhibitors are more than 3 times greater than the costs incurred in those without inhibitors, with an annual cost per patient that can be higher than €1 000 000. The costs of bypassing agents account for the large majority of the total healthcare direct costs for hemophilia treatment. The quality of life is more compromised in patients with hemophilia and inhibitors compared to those without inhibitors, in particular the physical domains, whereas mental domains were comparable to that of the general population. The development of an inhibitor has a high impact on costs and quality of life. New treatments have the potential to change positively the management and socioeconomic burden of hemophilia with inhibitors.

Original languageEnglish
Pages (from-to)435-456
Number of pages22
JournalEuropean Journal of Haematology
Volume101
Issue number4
DOIs
Publication statusPublished - Oct 2018

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Hemophilia A
Costs and Cost Analysis
Quality of Life
Health Care Costs
Economics
Delivery of Health Care
Health
Therapeutics
Population

Keywords

  • Blood Coagulation
  • Cost of Illness
  • Health Care Costs
  • Hemophilia A/blood
  • Hemophilia B/blood
  • Humans
  • Isoantibodies/blood
  • Quality of Life
  • Socioeconomic Factors
  • Surveys and Questionnaires

Cite this

The socioeconomic burden of patients affected by hemophilia with inhibitors. / D'Angiolella, Lucia S; Cortesi, Paolo A; Rocino, Angiola; Coppola, Antonio; Hassan, Hamisa J; Giampaolo, Adele; Solimeno, Luigi P; Lafranconi, Alessandra; Micale, Mariangela; Mangano, Sveva; Crotti, Giacomo; Pagliarin, Federica; Cesana, Giancarlo; Mantovani, Lorenzo G.

In: European Journal of Haematology, Vol. 101, No. 4, 10.2018, p. 435-456.

Research output: Contribution to journalReview article

D'Angiolella, LS, Cortesi, PA, Rocino, A, Coppola, A, Hassan, HJ, Giampaolo, A, Solimeno, LP, Lafranconi, A, Micale, M, Mangano, S, Crotti, G, Pagliarin, F, Cesana, G & Mantovani, LG 2018, 'The socioeconomic burden of patients affected by hemophilia with inhibitors', European Journal of Haematology, vol. 101, no. 4, pp. 435-456. https://doi.org/10.1111/ejh.13108
D'Angiolella, Lucia S ; Cortesi, Paolo A ; Rocino, Angiola ; Coppola, Antonio ; Hassan, Hamisa J ; Giampaolo, Adele ; Solimeno, Luigi P ; Lafranconi, Alessandra ; Micale, Mariangela ; Mangano, Sveva ; Crotti, Giacomo ; Pagliarin, Federica ; Cesana, Giancarlo ; Mantovani, Lorenzo G. / The socioeconomic burden of patients affected by hemophilia with inhibitors. In: European Journal of Haematology. 2018 ; Vol. 101, No. 4. pp. 435-456.
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AU - Cortesi, Paolo A

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AU - Coppola, Antonio

AU - Hassan, Hamisa J

AU - Giampaolo, Adele

AU - Solimeno, Luigi P

AU - Lafranconi, Alessandra

AU - Micale, Mariangela

AU - Mangano, Sveva

AU - Crotti, Giacomo

AU - Pagliarin, Federica

AU - Cesana, Giancarlo

AU - Mantovani, Lorenzo G

N1 - © 2018 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.

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AB - Hemophilia is associated with a high financial burden on individuals, healthcare systems, and society. The development of inhibitors significantly increases the socioeconomic burden of the diseases. This study aimed to review and describe the burden of hemophilia with inhibitors, providing a reference scenario to assess the impact of new products in the real word. Two systematic literature reviews were performed to collect data on (i) health economics and (ii) health-related quality of life evidences in hemophilic patients with inhibitors. The costs associated with patients with hemophilia and inhibitors are more than 3 times greater than the costs incurred in those without inhibitors, with an annual cost per patient that can be higher than €1 000 000. The costs of bypassing agents account for the large majority of the total healthcare direct costs for hemophilia treatment. The quality of life is more compromised in patients with hemophilia and inhibitors compared to those without inhibitors, in particular the physical domains, whereas mental domains were comparable to that of the general population. The development of an inhibitor has a high impact on costs and quality of life. New treatments have the potential to change positively the management and socioeconomic burden of hemophilia with inhibitors.

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