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Development and desensitization therapy of high-response factor VIII inhibitors with severe allergic reaction in a moderate hemophilia A patient
Neutralizing antibodies (inhibitors) against factor VIII/IX (FVIII/FIX) poses a serious and challenging complication in the hemophilia treatment. Allergic reaction is more common in hemophilia B and always companion with FIX inhibitors, but it is rare in hemophilia A (HA). So far only few cases demo...
Autores principales: | , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
SAGE Publications
2021
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7917874/ https://www.ncbi.nlm.nih.gov/pubmed/33626954 http://dx.doi.org/10.1177/2058738420980259 |
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author | Liu, Guoqing Chen, Zhenping Wu, Wenman Wu, Runhui |
author_facet | Liu, Guoqing Chen, Zhenping Wu, Wenman Wu, Runhui |
author_sort | Liu, Guoqing |
collection | PubMed |
description | Neutralizing antibodies (inhibitors) against factor VIII/IX (FVIII/FIX) poses a serious and challenging complication in the hemophilia treatment. Allergic reaction is more common in hemophilia B and always companion with FIX inhibitors, but it is rare in hemophilia A (HA). So far only few cases demonstrated FVIII-specific allergic response in hemophilia A. Coexistence of allergic reactions with inhibitors was contraindicated for immune tolerance induction (ITI) regimen which is the only proven therapy to eliminate inhibitor. We report a rare case of a 11-year-old boy with moderate HA who developed high titer inhibitor and severe allergic reaction to both plasma derived and recombinant FVIII concentrates. Inhibitor was eliminated with the use of prednisone. Further desensitization protocol by administering rFVIII of increasing does from 0.01 IU/kg to 40 IU/kg with a pre-determined time schedule allowed patient tolerance to the normal dose and infusion time to FVIII. |
format | Online Article Text |
id | pubmed-7917874 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2021 |
publisher | SAGE Publications |
record_format | MEDLINE/PubMed |
spelling | pubmed-79178742021-03-11 Development and desensitization therapy of high-response factor VIII inhibitors with severe allergic reaction in a moderate hemophilia A patient Liu, Guoqing Chen, Zhenping Wu, Wenman Wu, Runhui Int J Immunopathol Pharmacol Case Report Neutralizing antibodies (inhibitors) against factor VIII/IX (FVIII/FIX) poses a serious and challenging complication in the hemophilia treatment. Allergic reaction is more common in hemophilia B and always companion with FIX inhibitors, but it is rare in hemophilia A (HA). So far only few cases demonstrated FVIII-specific allergic response in hemophilia A. Coexistence of allergic reactions with inhibitors was contraindicated for immune tolerance induction (ITI) regimen which is the only proven therapy to eliminate inhibitor. We report a rare case of a 11-year-old boy with moderate HA who developed high titer inhibitor and severe allergic reaction to both plasma derived and recombinant FVIII concentrates. Inhibitor was eliminated with the use of prednisone. Further desensitization protocol by administering rFVIII of increasing does from 0.01 IU/kg to 40 IU/kg with a pre-determined time schedule allowed patient tolerance to the normal dose and infusion time to FVIII. SAGE Publications 2021-02-24 /pmc/articles/PMC7917874/ /pubmed/33626954 http://dx.doi.org/10.1177/2058738420980259 Text en © The Author(s) 2021 https://creativecommons.org/licenses/by-nc/4.0/ This article is distributed under the terms of the Creative Commons Attribution-NonCommercial 4.0 License (https://creativecommons.org/licenses/by-nc/4.0/) which permits non-commercial use, reproduction and distribution of the work without further permission provided the original work is attributed as specified on the SAGE and Open Access pages (https://us.sagepub.com/en-us/nam/open-access-at-sage). |
spellingShingle | Case Report Liu, Guoqing Chen, Zhenping Wu, Wenman Wu, Runhui Development and desensitization therapy of high-response factor VIII inhibitors with severe allergic reaction in a moderate hemophilia A patient |
title | Development and desensitization therapy of high-response factor VIII
inhibitors with severe allergic reaction in a moderate hemophilia A
patient |
title_full | Development and desensitization therapy of high-response factor VIII
inhibitors with severe allergic reaction in a moderate hemophilia A
patient |
title_fullStr | Development and desensitization therapy of high-response factor VIII
inhibitors with severe allergic reaction in a moderate hemophilia A
patient |
title_full_unstemmed | Development and desensitization therapy of high-response factor VIII
inhibitors with severe allergic reaction in a moderate hemophilia A
patient |
title_short | Development and desensitization therapy of high-response factor VIII
inhibitors with severe allergic reaction in a moderate hemophilia A
patient |
title_sort | development and desensitization therapy of high-response factor viii
inhibitors with severe allergic reaction in a moderate hemophilia a
patient |
topic | Case Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7917874/ https://www.ncbi.nlm.nih.gov/pubmed/33626954 http://dx.doi.org/10.1177/2058738420980259 |
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