Immune Tolerance Therapy for Haemophilia A Patients with Acquired Factor VIII Alloantibodies: Comprehensive Analysis of Experience at a Single Institution

M. P. Smith; K. J. Spence; E. L. Waters; R. Beresford-Webb; M. J. Mitchell; J. Cuttler; A. Alhaq; S. A. Brown; G. F. Savidge

doi:10.1055/s-0037-1614414

Thrombosis and Haemostasis, Inhaltsverzeichnis

Thromb Haemost 1999; 81(01): 35-38
DOI: 10.1055/s-0037-1614414

Review Article

Schattauer GmbH

Immune Tolerance Therapy for Haemophilia A Patients with Acquired Factor VIII Alloantibodies: Comprehensive Analysis of Experience at a Single Institution

M. P. Smith

¹From the Haemophilia Centre, Reference Centre for Haemostatic and Thrombotic Disorders, St Thomas’ Hospital, London, UK

,

K. J. Spence

¹From the Haemophilia Centre, Reference Centre for Haemostatic and Thrombotic Disorders, St Thomas’ Hospital, London, UK

,

E. L. Waters

¹From the Haemophilia Centre, Reference Centre for Haemostatic and Thrombotic Disorders, St Thomas’ Hospital, London, UK

,

R. Beresford-Webb

¹From the Haemophilia Centre, Reference Centre for Haemostatic and Thrombotic Disorders, St Thomas’ Hospital, London, UK

,

M. J. Mitchell

¹From the Haemophilia Centre, Reference Centre for Haemostatic and Thrombotic Disorders, St Thomas’ Hospital, London, UK

,

J. Cuttler

¹From the Haemophilia Centre, Reference Centre for Haemostatic and Thrombotic Disorders, St Thomas’ Hospital, London, UK

,

A. Alhaq

¹From the Haemophilia Centre, Reference Centre for Haemostatic and Thrombotic Disorders, St Thomas’ Hospital, London, UK

,

S. A. Brown

¹From the Haemophilia Centre, Reference Centre for Haemostatic and Thrombotic Disorders, St Thomas’ Hospital, London, UK

,

G. F. Savidge

¹From the Haemophilia Centre, Reference Centre for Haemostatic and Thrombotic Disorders, St Thomas’ Hospital, London, UK

› Institutsangaben

Abstract

Summary

Eleven children with severe haemophilia A associated with the IVS 22 inversion and acquired high titre neutralising antibodies to factor VIII underwent immune tolerance induction. HLA class I and high resolution class II type is detailed for each patient. A three phase approach to immune tolerance induction was used. During phase 1, which lasted a median of six weeks, patients received factor VIII 100 IU/kg twice daily. Phase 2 comprised a factor VIII dose reduction to 100 IU/kg once daily, and continued for a median duration of 14 weeks. Subsequently 10 of the 11 patients satisfied the criteria of absent factor VIII neutralising activity by the Bethesda method, and a factor VIII elimination half life of greater than 5 h, allowing progression to phase 3, a further factor VIII dose reduction to 50 IU/kg three times weekly. A model for dose reduction as factor VIII tolerance evolves, based on pharmacokinetic analysis, is described.

Volltext

Referenzen

References
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