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The natural history of factor VIII:C inhibitors in patients with hemophilia
A: a national cooperative study. II. Observations on the initial
development of factor VIII:C inhibitors
CW McMillan, SS Shapiro, D Whitehurst, LW Hoyer, AV Rao and J Lazerson
University of Connecticut School of Medicine.
During a 4-year multicenter cooperative study of acquired factor VIII
inhibitors in persons with hemophilia A, new inhibitors were detected in 31
of 1,306 patients who entered the study without an inhibitor or the history
of an inhibitor. The incidence of new inhibitors was eight per 1,000
patient-years of observation. The factor VIII:C level before inhibitor
development was less than or equal to 0.03 U/mL in 29 individuals and 0.06
U/mL and 0.07 U/mL in the remaining two. Factor VIII:Ag levels were
measured in 27 individuals and were less than 0.03 U/mL in 23 and 0.05 to
0.11 U/mL in the remaining four. Maximum inhibitor levels ranged from 1.0
to 9,044 Bethesda U/mL. In seven patients under the age of 20, relatively
weak inhibitors (none higher than 4.3 Bethesda U/mL) were detected on only
a single occasion despite continued factor VIII challenge. In the other 24
patients with inhibitors detected on multiple occasions, 50% had appeared
by age 20 and 71% by age 30. Seventeen of the 31 inhibitors, including 12
of 15 with maximum values greater than 10 Bethesda U/mL, developed within
75 exposure days to factor VIII.
Volume 71,
Issue 2,
pp. 344-348,
02/01/1988
Copyright © 1988 by The American Society of Hematology
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