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Blood, 1 September 2000, Vol. 96, No. 5, pp. 1698-1702
CLINICAL OBSERVATIONS, INTERVENTIONS, AND THERAPEUTIC TRIALS
Immune tolerance induction in hemophilia patients with
inhibitors: costly can be cheaper
Alan B. Colowick,
Rhonda L. Bohn,
Jerry Avorn, and
Bruce M. Ewenstein
From the Department of Medicine, Divisions of
Hematology and Pharmacoepidemiology and Pharmacoeconomics, Brigham and
Women's Hospital, Harvard Medical School, Boston, MA.
The development of inhibitory antibodies to factor VIII (FVIII)
occurs in approximately 30% to 40% of patients with severe hemophilia
A. Management options for patients with inhibitor include eradicating
it via immune tolerance induction (ITI) or treating bleeding episodes
with large quantities of hemostatic agents. ITI is costly, approaching
$1 million for the average 5-year-old, but if successful results in
improved clinical outcomes. We constructed a decision analysis using
the Markov process to model expected clinical outcomes and costs over a
lifetime for a typical 5-year-old hemophiliac with high inhibitor
levels. Estimates of relevant variables were based on a thorough review
of the medical literature. Outcomes modeled included total lifetime
costs as well as life expectancy. The decision analytic model revealed
that the ITI strategy was associated with an increase in projected life
expectancy of 4.6 years. Total estimated lifetime costs for the ITI
strategy were approximately $1.7 million less per patient. Sensitivity analyses over clinically and economically reasonable ranges did not
change these findings. The insight that ITI can achieve an improved
clinical outcome while being cost-saving is not reflected in many
current treatment regimens. This example also illustrates that
expensive therapy for patients with a chronic disease may be cost
effective when analyzed from a societal perspective over the patient's
lifetime. This finding has important policy implications for medical
decision makers at many levels and reinforces the need to undertake
pharmacoeconomic analyses and choose therapies from a long-term,
societal perspective.
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