Primary and rescue immune tolerance induction in children and adults: a multicentre international study with a VWF-containing plasma-derived FVIII concentrate

Haemophilia : the Official Journal of the World Federation of Hemophilia
J OldenburgE Santagostino

Abstract

Most studies on immune tolerance induction (ITI) therapy in haemophilia A patients are focused on primary ITI in children. Here we report on the ITI outcome in a large retrospective cohort, including adults and patients with rescue ITI, treated with a pdFVIII/VWF concentrate. Retrospective data from haemophilic patients (FVIII< 2%) with inhibitors from 22 centres in Spain, Italy and Germany, who underwent primary or rescue ITI with pdFVIII/VWF concentrate, were collected. Complete success (CS), partial success (PS) and failure were defined based on the criteria of the consensus recommendations of the 2006 International ITI Workshop. A total of 41 cases of primary ITI (32 children and 9 adults) and 19 cases of rescue ITI (17 children and 2 adults) were evaluated. Success (CS+PS) rate of 87% was achieved in primary ITI and 74% in the higher risk profile of rescue ITI. Eight of nine (85%) patients with poorest prognosis (three or more of the known risk factors of poor response to ITI) achieved success (CS+PS). CS of 100% was observed in eight primary ITI patients with titre at start of ITI ≤2.5 BU and inhibitor peak ≤25 BU. The favourable response rates in primary and rescue ITI in children and in adult patients, even in the prese...Continue Reading

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Citations

Jul 24, 2015·Haemophilia : the Official Journal of the World Federation of Hemophilia·W KreuzUNKNOWN ObsITI study group and the ObsITI committee
Jun 3, 2015·Haemophilia : the Official Journal of the World Federation of Hemophilia·L A ValentinoUNKNOWN International Immune Tolerance Induction Study Investigators
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Feb 13, 2019·Haemophilia : the Official Journal of the World Federation of Hemophilia·Louis M Aledort
Mar 14, 2019·Haemophilia : the Official Journal of the World Federation of Hemophilia·Louis AledortCindy Leissinger
Feb 21, 2019·Haemophilia : the Official Journal of the World Federation of Hemophilia·Elena SantagostinoVíctor Jiménez-Yuste
Jan 28, 2015·Haemophilia : the Official Journal of the World Federation of Hemophilia·R Kruse-JarresJ A Kaye
May 10, 2019·British Journal of Haematology·Guy Young
Apr 30, 2019·Haemophilia : the Official Journal of the World Federation of Hemophilia·Manuel CarcaoUNKNOWN Future of Immunotolerance Treatment Group
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Oct 5, 2021·Expert Opinion on Emerging Drugs·Hande Kizilocak, Guy Young
Feb 21, 2019·Acta Haematologica·Elena SantagostinoManuel Carcao

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