The Maintenance of Tolerance After Successful Immune Tolerance Induction in Hemophilia A and B: The North American Registry
Bibliographic record
Abstract
The North American Immune Tolerance Registry (NAITR) was initiated with the goal of determining, by questionnaire, immune tolerance (ITT) practices in hemophilia treatment centers in Canada and the United States. Sixty-eight centers (40%) responded. Of the 130 registry subjects with hemophilia A who completed ITT, 93 (72%) achieved tolerance. Of the 11 completed ITT courses in patients with hemophilia B, 4 (36%) were successful. Maintenance therapy was defined as any clotting factor regimen administrated subsequent to the patient achieving the treating physician's criteria for successful immune tolerance. Seventy-five (81%) of 93 individuals in the hemophilia A cohort who successfully achieved tolerance were maintained on a regular (prophylactic factor VIII (FVIII) regimen for a variable time period post-ITT. The median dose used was 150 units/kg/week (range: 17-700). Forty-eight (64%) subjects remained tolerant while receiving regular doses of FVIII for a median observation period of 13 months (range 0-129 months). Of 27 patients whose maintenance therapy had been stopped, 17 (68%) remained tolerant over a median period of 19 months (range 1-54 months) and 9 relapsed. Among the relapses, 3 occurred after maintenance therapy was stopped; 6 were noted on prophylactic FVIII at a median time of 11 months (range 2-61 months). The definition of tolerance was reviewed for the 9 subjects who relapsed and was defined by a normal recovery and survival in only 1/9 patients. Among the 11 hemophilia B subjects in the cohort who completed tolerance, ' had a successful outcome. Four individuals were placed on maintenance regimens of 25-100 units FIX/kg/day and all remained tolerant.
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How this classification was reachedexpand
Full frame distilled prediction
Teacher imitationNot calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.
Codex and Gemma teacher scores by category
| Category | Codex | Gemma |
|---|---|---|
| Metaresearch | 0.000 | 0.000 |
| Meta-epidemiology (narrow) | 0.000 | 0.000 |
| Meta-epidemiology (broad) | 0.000 | 0.000 |
| Bibliometrics | 0.000 | 0.000 |
| Science and technology studies | 0.000 | 0.002 |
| Scholarly communication | 0.000 | 0.000 |
| Open science | 0.000 | 0.000 |
| Research integrity | 0.000 | 0.000 |
| Insufficient payload (model declined to judge) | 0.000 | 0.000 |
Machine scores (provisional)
The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.
Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.
score_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from itClassification
machine, unvalidatedMachine predicted; a candidate call from one teacher head, not a consensus.
How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".