Dose administered and dosing interval among patients with LTIs∗
| Median (range) . | Transient LTI (n = 7) . | Persistent LTI (n = 7)† . | LTI progressing to HTI (n = 7) . |
|---|---|---|---|
| LTI development until resolution . | LTI development until follow-up . | LTI development until HTI development . | |
| Mean dose per infusion (IU/kg) | 59.2 (27.3-94.5) | 45.0 (44.9-86.5)‡ | 98.4 (81.9-104.4)§ |
| P value‖ | .0331 | .0472 | — |
| Mean dosing interval (d) | 2.6 (1.9-7.0) | 3.1 (2.2-3.8)‡ | 4.7 (1.3-6.9)§ |
| Dose per week (IU/kg per week)¶ | 175.5 (55.2-366.2) | 144.2 (104.1-280.8)‡ | 196.4 (115.3-550.9)§ |
| Median (range) . | Transient LTI (n = 7) . | Persistent LTI (n = 7)† . | LTI progressing to HTI (n = 7) . |
|---|---|---|---|
| LTI development until resolution . | LTI development until follow-up . | LTI development until HTI development . | |
| Mean dose per infusion (IU/kg) | 59.2 (27.3-94.5) | 45.0 (44.9-86.5)‡ | 98.4 (81.9-104.4)§ |
| P value‖ | .0331 | .0472 | — |
| Mean dosing interval (d) | 2.6 (1.9-7.0) | 3.1 (2.2-3.8)‡ | 4.7 (1.3-6.9)§ |
| Dose per week (IU/kg per week)¶ | 175.5 (55.2-366.2) | 144.2 (104.1-280.8)‡ | 196.4 (115.3-550.9)§ |
ITI, immune tolerance induction.
Unless otherwise specified, P > .05.
For the persistent LTI group, ITI regimen period has been excluded from the follow-up period.
n = 5; for 2 patients, all exposures occurred before LTI development, and therefore these patients were excluded from analysis.
n = 5; 1 participant developed an HTI during the pre-ITI period and therefore is not considered for this analysis. One patient initially developed an LTI; however, an HTI was detected shortly after their formal withdrawal from the study (for the reason of lack of home health care). Thus, the patient was considered to have developed an HTI but did not have on-study data related to dosing between LTI and HTI development and is excluded from this analysis.
P values were derived from 2-sided Mann-Whitney U test.
Dose per week (IU/kg per week) = total dose (IU/kg)/total time (weeks), where total time is the assessment period referenced (ie, LTI development until resolution, LTI development until follow-up, or LTI development until HTI development).