Economic Analysis of an Ambulatory Care Service Delivery Model for Hemophilia a Patients; A Cost Minimization Study

Main Article Content

Abolfazl Sadeghi
Majid Davari
Zahra Gharibnaseri
Roya Ravanbod
Peyman Eshghi


Aim: This study aimed to perform an economic evaluation of Hemophilia ambulatory service delivery model (HASDM) comparing to the traditional home-episodic treatment model.

Study Design: Tehran university of medical science, department pharmacoeconomics and pharmaceutical administration, between Jun 2016 and September 2018.

Methods: A cost-minimization analysis (CMA) was conducted for evaluating potential savings of HASDM in comparison to the traditional home-episodic treatment model. The main cost of regular episodic service delivery, basic arm, consists of the cost of recombinant factor VIII (FVIII). In the comparator arm, HASDM, the costs of HASDM for 1660 hemophilia A patients (HAPs) in Tehran were calculated. One-way sensitivity analysis was done to investigate the robustness of the results and to investigate the impact of uncertainty in the percentage of mistakes in bleeding sensation.

Results: There were 1660 patients with severe Hemophilia A (PWSHA) in Tehran in 2018. The mean utilization of annual per patient FVIII was 44814 international units (IUs) in Iran. The total annual cost of FVIII concentrate for 1660 hemophilic patients in Tehran was estimated at $ 11,001,816. The cost of running HASDM, personal, and equipment is equal to $ 580,956. The cost of FVIII in HASDM would be $ 4,004,661. Therefore, the total cost of HASDM is estimated at $ 4,585,617. The amount of savings was $ 6,416,199. Sensitivity analysis indicated the robustness of the results up to 94.64% of the variation in the model parameters.

Conclusions: HASDM, compared to episodic model, can save 58.32% of the funding for controlling bleeding in HAPs annually. This can save more than 38 times of HAPs annual cost over their lifetime.

Hemophilia A, factor VIII, economic evaluation, cost-minimization analysis, efficiency.

Article Details

How to Cite
Sadeghi, A., Davari, M., Gharibnaseri, Z., Ravanbod, R., & Eshghi, P. (2020). Economic Analysis of an Ambulatory Care Service Delivery Model for Hemophilia a Patients; A Cost Minimization Study. Journal of Pharmaceutical Research International, 31(6), 1-6.
Original Research Article


Valleix S, et al. Skewed X-chromosome inactivation in monochorionic diamniotic twin sisters results in severe and mild hemophilia A. Blood. 2002;100(8):3034-3036.

Kang H-S, et al. Treatment and characteristics related to patients with hemophilia. Journal of Muscle and Joint Health. 2006;13(2):119-129.

Dorgalaleh A, Dadashizadeh G, Bamedi T. Hemophilia in Iran. Hematology. 2016; 21(5):300-310.

Mehdizadeh M, et al. Occurrence of haemophilia in Iran. 2009;15(1):348-351.

Unim B, et al. Haemophilia A: Pharmaco-economic review of prophylaxis treatment versus on-demand; 2015.

Gharibnaseri Z, et al. Health care resource utilization and cost of care for haemophilia A and B patients in Iran. 2016;54(1):122-126.

Radossi P, et al. Intra-articular rifamycin in haemophilic arthropathy. Haemophilia. 2003;9(1):60-63.

Oldenburg J. Blood coagulation and hemorrhagic diathesis. Pharm Unserer Zeit. 2006;35:20-28.

Rosendaal F. Definitions in hemophilia, recommendation of the scientific sub committee on factor VIII and factor IX of the scientific and standardization committee of the international society on thrombosis and haemostasis factor VII and factor IX subcommittee; 2001.

Meyers RD, et al. The social and economic impact of hemophilia--a survey of 70 cases in Vermont and New Hampshire. American Journal of Public Health. 1972;62(4):530-535.

Ceponis A. et al. Rapid musculoskeletal ultrasound for painful episodes in adult haemophilia patients. 2013;19(5):790- 798.

Davari M. Sadeghi A. et al. An efficient and effective online ambulatory service model for hemophilia-A patients; an introduction to a novel service delivery model for developing countries; Under evaluation; 2018.

Duncan N, et al. A haemophilia disease management programme targeting cost and utilization of specialty pharma-ceuticals. Haemophilia. 2014;20(4):519-526.

Valentino L, et al. Healthcare resource utilization among haemophilia A patients in the United States. Haemophilia. 2012; 18(3):332-338.

Zappa S, et al. Treatment trends for haemophilia A and haemophilia B in the United States: Results from the 2010 practice patterns survey. Haemophilia. 2012;18(3):e140-e153.

O’Hara J, et al. The cost of severe haemophilia in Europe: The CHESS study. Orphanet Journal of Rare Diseases. 2017; 12(1):106.

Gouw S, van den Berg H, le Cessie S, et al. Treatment characteristics and the risk of inhibitor development: A multi- center cohort study among previously untreated patients with severe hemophilia A. J Thromb Haemost. 2007;5:1383– 1390.

Gouw S, van der Bom J, Marijke van den Berg H. Treatment-related risk factors of inhibitor development in previously untreated patients with hemophilia A: The CANAL cohort study. Blood. 2007;109: 4648–4654.

Maclean P, Richards M, Williams et al. Treatment related factors and inhibitor development in children with severe haemophilia A. Haemophilia. 2011;17: 282–287.

Ragni M, Ojeifo O, Feng J, et al. Risk factors for inhibitor formation in haemo-philia: A prevalent case-control study. Haemophilia. 2009;15:1074–1082.