A novel approach to immune tolerance induction in haemophilia A with factor VIII inhibitor

Haemophilia ◽  
2018 ◽  
Vol 25 (1) ◽  
pp. e48-e50 ◽  
Author(s):  
Courtney D. Thornburg ◽  
Jonathan Ducore
Keyword(s):  
Factor Viii ◽  
Immune Tolerance ◽  
Tolerance Induction ◽  
Factor Viii Inhibitor ◽  
Haemophilia A ◽  
Immune Tolerance Induction ◽  
Novel Approach ◽  
Viii Inhibitor

Reduction of Factor VIII Inhibitor Titers During Immune Tolerance Induction With Recombinant Factor VIII-Fc Fusion Protein

2016 ◽  
Vol 63 (5) ◽  
pp. 922-924 ◽  
Author(s):  
Charles L. Groomes ◽  
David M. Gianferante ◽  
Gary D. Crouch ◽  
Dina S. Parekh ◽  
David W. Scott ◽  
...  
Keyword(s):  
Fusion Protein ◽  
Factor Viii ◽  
Immune Tolerance ◽  
Tolerance Induction ◽  
Recombinant Factor Viii ◽  
Factor Viii Inhibitor ◽  
Immune Tolerance Induction ◽  
Fc Fusion Protein ◽  
Viii Inhibitor

The long-term course of factor VIII inhibitors in patients with congenital haemophilia A without immune tolerance induction

2011 ◽  
Vol 105 (01) ◽  
pp. 59-65 ◽  
Author(s):  
Camila Caram ◽  
Roberta Grazielle de Souza ◽  
Júlio Carepa de Sousa ◽  
Tatiana Araújo Pereira ◽  
Ana Maria do Amaral Cerqueira ◽  
...  
Keyword(s):  
Confidence Interval ◽  
Factor Viii ◽  
Immune Tolerance ◽  
Tolerance Induction ◽  
Clotting Factor ◽  
Haemophilia A ◽  
Immune Tolerance Induction ◽  
Inhibitor Titre ◽  
Very High

SummaryThe development of alloantibodies that inhibit or neutralise the function of factor VIII is considered the most serious complication of the treatment of congenital haemophilia A. In order to describe their course without immune tolerance induction (ITI), we documented data on all performed inhibitor tests with dates as well as on clotting factor infusions of all consecutive patients who were treated in our centre between 1993 and 2006. Patients were tested every 7.1 months (95% confidence interval [CI], 6.6–7.8). A ‘sustained negative inhibitor status’ was defined as consistent non-positive inhibitor measurements for two years or longer. A total of 60/486 (12%) patients tested had a positive inhibitor titre in two or more occasions. Most of the patients (56%) with a maximum inhibitor titre of < 5 Bethesda unit (BU)/ml (named “low titre inhibitor”) developed a sustained negative inhibitor status. Among patients with high (5–9.9 BU/ml) and very high (≥ 10 BU/ ml) inhibitor titres, the proportions were 50% and 3%, respectively. Our findings suggest that ITI might not be needed for all patients with non-transient inhibitors, especially when their maximum inhibitor titre is below 10 BU/ml. Further studies in countries where ITI is not available are needed to examine predictors of the natural sustained negative inhibitor status.

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