Table 3.
Source | No. of participants | ITI regimen (%) | ITI Product (%) |
Age at inhibitor detection (m) | Interval ID - ITI start (m) | Age at ITI start, (m) | No. of exposure d before inhibitor administration | Pre-ITI titer | Historical peak titer | Peak titer on ITI | Outcome (%) |
||||
---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|---|
pd | r | pd+r | CS | PS | F | ||||||||||
Escobar [44] | 7 | 100 IU/kg/d | 100 | 0 | 0 | 24 (12-36) | na | 126 (60-384) | na | 17 (2.4-109) | 104.1 (75.5-265.1) | na | 28.6 | 42.9 | 28.6 |
Greninger [45] | 4 | 1400 IU/kg/wk | 100 | 0 | 0 | 14 (3.4-17.4) | 3.4 (0.1-52.3) | 18 (5.1-67.5) | na | 7.4 (4.4-197.8) | 47 (11.6-210.8) | 1216 (346-2868.6) | 0 | na | 100 |
Kurth [30] | 25 | 50 IU/kg/d (8) 100-200 IU/kg/d (92) |
100 | 0 | 0 | 10.5 (7.3-20.5) | na | 54 (35.5-98) | na | 22 (2-76) | 277.5 (122.5-769.5) | na | 32 | 20 | 48 |
Nogami [31] | 22 | ≤75 IU/kg 3x/wk (52)a; ≥90 IU/kg/d (12)a; Other (30)a |
13 | 87 | 0 | 14.4 (10.8-25.2)a | 7.2 (2.4-26.4)a | na | na | 3.9 (1.7-7.9)a | 14 (3.7-46)a | na | 71.2 | na | 28.8 |
Oldenburg [53] | 17c 2d |
<100 IU/kg/d (22)a; ≥100 IU/kg/d (78)a |
100 | 0 | 0 | na | na | 92.3 (30-184.8b)c 345.6 (298.8-391.2 b)d |
na | 20.3 (1.6-200 b)c 10 (5-15 b)d |
92 (2.1-4505 b)c 200 (100-300 b)d |
na | 36.8 | 36.8 | 26.3 |
Numbers are reported as median (interquartile range), unless stated otherwise.
CS, complete success; F, failure; IU, international unit; ITI, immune tolerance induction; kg, kilogram; na, not available; PS, partial success; pd, plasma-derived FVIII; r, recombinant FVIII; pd+r, plasma-derived and recombinant FVIII.
Both primary and rescue ITI.
Range.
Children.
Adults.