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Continuous infusion of factor IX concentrate to induce immune tolerance in two patients with haemophilia B

Tengborn, Lilian LU and Berntorp, Erik LU (1998) In Haemophilia 4(1). p.56-59
Abstract
Two patients with haemophilia B and high-responding inhibitor to factor IX were subjected to immune tolerance induction according to the Malmo protocol, including high dosage of factor IX, cyclophosphamide and intravenous gammaglobulin. In one of the patients the treatment was preceded by extracorporeal protein A adsorption. Both patients had previously been subjected to immune tolerance induction without success and as an attempt to improve the tolerance induction regimen and lower cost, factor IX was given as continuous infusion, with a dose of around 300 units per kg body weight daily for 3 weeks. The inhibitor level declined in one of the patients but tolerance was not achieved. In the second patient the inhibitor level remained high.... (More)
Two patients with haemophilia B and high-responding inhibitor to factor IX were subjected to immune tolerance induction according to the Malmo protocol, including high dosage of factor IX, cyclophosphamide and intravenous gammaglobulin. In one of the patients the treatment was preceded by extracorporeal protein A adsorption. Both patients had previously been subjected to immune tolerance induction without success and as an attempt to improve the tolerance induction regimen and lower cost, factor IX was given as continuous infusion, with a dose of around 300 units per kg body weight daily for 3 weeks. The inhibitor level declined in one of the patients but tolerance was not achieved. In the second patient the inhibitor level remained high. Despite the failure of the treatment in these two cases, we propose that the constant antigen load provided by the continuous infusion of modern, safe, purified factor IX concentrate may theoretically be of greater benefit in immune tolerance induction than the varying load resulting from intermittent infusions. Larger study materials are needed to show if this is so. (Less)
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author
organization
publishing date
type
Contribution to journal
publication status
published
subject
keywords
continuous infusion, factor IX inhibitor, Haemophilia B, immune tolerance induction, protein A adsorption
in
Haemophilia
volume
4
issue
1
pages
56 - 59
publisher
Federation of European Neuroscience Societies and Blackwell Publishing Ltd
external identifiers
  • pmid:9873867
  • scopus:0031598555
ISSN
1351-8216
DOI
10.1046/j.1365-2516.1998.00142.x
language
English
LU publication?
yes
id
571f7c01-68e2-4eed-bb11-73fbb2f38f8a (old id 1113243)
date added to LUP
2008-07-14 15:16:25
date last changed
2017-01-01 04:52:45
@article{571f7c01-68e2-4eed-bb11-73fbb2f38f8a,
  abstract     = {Two patients with haemophilia B and high-responding inhibitor to factor IX were subjected to immune tolerance induction according to the Malmo protocol, including high dosage of factor IX, cyclophosphamide and intravenous gammaglobulin. In one of the patients the treatment was preceded by extracorporeal protein A adsorption. Both patients had previously been subjected to immune tolerance induction without success and as an attempt to improve the tolerance induction regimen and lower cost, factor IX was given as continuous infusion, with a dose of around 300 units per kg body weight daily for 3 weeks. The inhibitor level declined in one of the patients but tolerance was not achieved. In the second patient the inhibitor level remained high. Despite the failure of the treatment in these two cases, we propose that the constant antigen load provided by the continuous infusion of modern, safe, purified factor IX concentrate may theoretically be of greater benefit in immune tolerance induction than the varying load resulting from intermittent infusions. Larger study materials are needed to show if this is so.},
  author       = {Tengborn, Lilian and Berntorp, Erik},
  issn         = {1351-8216},
  keyword      = {continuous infusion,factor IX inhibitor,Haemophilia B,immune tolerance induction,protein A adsorption},
  language     = {eng},
  number       = {1},
  pages        = {56--59},
  publisher    = {Federation of European Neuroscience Societies and Blackwell Publishing Ltd},
  series       = {Haemophilia},
  title        = {Continuous infusion of factor IX concentrate to induce immune tolerance in two patients with haemophilia B},
  url          = {http://dx.doi.org/10.1046/j.1365-2516.1998.00142.x},
  volume       = {4},
  year         = {1998},
}