Original Paper
Acta Haematol 1999;102:42–46
Factor VIII Inhibitors in Patients with
Hemophilia A
Hale Ören Is ¸ ın Yaprak Gülersu I
˙
rken
Departments of Pediatric Hematology, Dokuz Eylül University, Faculty of Medicine, and SSK Tepecik Hospital,
Izmir, Turkey
Received: August 21, 1998
Accepted: March 22, 1999
Dr. Hale Ören
Dokuz Eylül University, Faculty of Medicine
Department of Pediatric Hematology
TR–35340 Balçova, I
˙
zmir (Turkey)
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Key Words
Factor VIII W Hemophilia A W Inhibitors
Abstract
In 58 hemophilia A patients aged 1–18 years (mean 9.5 B
4.7 years), the prevalence of inhibitors was found to be
27% by the Bethesda method in November 1995. Inhibi-
tor activity was not detected in any of 14 patients with
mild hemophilia while it was present in 9 of 27 (33%)
patients with moderate, and 7 of 17 (41%) with severe
disease. During follow-up, the inhibitors were transient
in 10 of 16 patients (17%) and the prevalence of inhibi-
tors was 10% at the end of the study. Our study has dem-
onstrated that the patients’ age, factor VIII (F VIII) coagu-
lant activity levels, type of F VIII replacement therapy,
and frequency of F VIII administration affect inhibitor
development, and these factors should be considered in
the follow-up of hemophiliacs.
Introduction
One of the most serious problems in the treatment of
patients with hemophilia A is the development of inhibi-
tors of factor (F) VIII [1–3]. F VIII inhibitors are anti-
bodies that develop in patients with hemophilia A in
response to F VIII contained in various blood products,
mostly neutralizing F VIII coagulant activity [2]. Since F
VIII inhibitors make treatment of bleeding episodes
much more difficult, it is important to evaluate patients’
inhibitor levels. Some of the factors thought to affect
inhibitor development include the severity of the hemo-
philia, the type and duration of clotting factor therapy,
age and genetics of the patients [3, 4].
Development of inhibitors of F VIII occurs in about
5–10% of all patients with hemophilia A, but in 10–26% of
severely affected individuals [4–8]. Treatment status of the
patients affects inhibitor induction; since their immune
systems have not yet been exposed to clotting factor thera-
py, previously untreated patients show a higher rate of in-
hibitor development than previously treated patients [9].
Also type of F VIII replacement therapy affects inhibitor
formation [10]. In recent prospective studies the frequency
of inhibitors in severe hemophilia A has varied from 28 to
52% in patients using intermediate-purity concentrates
[11, 12]. In studies using recombinant F VIII concentrates,
inhibitors formed in 19–24% of previously untransfused
patients with severe hemophilia [6, 7, 13], and in studies
using monoclonally purified plasma-derived concentrates,
inhibitors developed in 6–18% [14, 15]. It has been shown
that one third of the inhibitors will develop by age 4 years
and more than 50% will develop by age 9 years [16, 17].