Failure of rituximab to induce immune tolerance in a boy with severe haemophilia A and an alloimmune factor VIII antibody: a case report and review of the literature

We report the use of rituximab (MabThera®; Roche Grenzach‐Wyhlen, Germany) in a 6‐year‐old boy with severe haemophilia A and a high titre alloimmune factor VIII (FVIII) antibody, which had failed to respond to standard immune tolerance therapy. Rituximab was administered in 4 weekly doses with concu...

Full description

Saved in:
Bibliographic Details
Published in:Haemophilia : the official journal of the World Federation of Hemophilia 2006-05, Vol.12 (3), p.280-284
Main Authors: BISS, T. T., VELANGI, M. R., HANLEY, J. P.
Format: Article
Language:English
Subjects:
acquired haemophilia
alloimmune FVIII antibody
Antibodies - immunology
Antibodies, Monoclonal - therapeutic use
Antibodies, Monoclonal, Murine-Derived
Factor VII - immunology
Factor VII - therapeutic use
Factor VIIa
Factor VIII - immunology
Factor VIII - therapeutic use
haemophilia A
Hemophilia A - drug therapy
Hemophilia A - immunology
Humans
Immune Tolerance - drug effects
immune tolerance induction
Immunologic Factors - therapeutic use
Infant
Male
Recombinant Proteins - immunology
Recombinant Proteins - therapeutic use
Rituximab
Treatment Failure
Citations: Items that this one cites
Items that cite this one
Online Access:Get full text
Tags: Add Tag
No Tags, Be the first to tag this record!
Description
Summary:We report the use of rituximab (MabThera®; Roche Grenzach‐Wyhlen, Germany) in a 6‐year‐old boy with severe haemophilia A and a high titre alloimmune factor VIII (FVIII) antibody, which had failed to respond to standard immune tolerance therapy. Rituximab was administered in 4 weekly doses with concurrent high‐dose i.v. immunoglobulin (Flebogamma®; Grifols, Barcelona, Spain) followed by daily high‐dose recombinant FVIII concentrate (Recombinate®; Baxter, CA, USA). Despite a fall in CD20 positive cell count to undetectable levels the inhibitor persisted. We discuss the possible reasons for failure of immune tolerance induction and review the literature concerning the use of rituximab for this indication.
ISSN:1351-8216
1365-2516
DOI:10.1111/j.1365-2516.2006.01212.x