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Successful eradication of a FVIII inhibitor in a 60-year-old patient with mild haemophilia A using single-agent prednisolone.
MedLine Citation:
PMID:  22960877     Owner:  NLM     Status:  In-Data-Review    
Background: Development of FVIII inhibitors represents a major challenge in patients with mild haemophilia A (HA), because they tend to occur at an older age and classical immune tolerance induction appears to be less effective. Case report: A man (age: 60 years) with mild HA due to the missense mutation, Leu1929Arg, received a single dose of rFVIII at 35IU/kg prior to routine colonoscopy, totalling 25 lifetime exposure days. Two months later, rFVIII was infused for a traumatic hip haematoma. However, FVIII recovery was inappropriate, and a FVIII inhibitor of 19 BU with type-2 kinetics was detected, resulting in FVIII:C of <1%. Two weeks later, the patient experienced spontaneous iliopsoas bleeding. Parallel to bypassing therapy, we started single-agent immunosuppression with prednisolone at 1.5mg/kg. FVIII:C "normalized" at 10.2% after four weeks. After five months, the inhibitor titre fell to <0.4BU with sustained remission after one year of follow-up. Conclusion: In mild HA, FVIII inhibitors may share characteristic features with FVIII autoantibodies commonly observed in acquired HA. Therefore, immunosuppressive therapy alone could be successful at least in a subset of patients.
K Holstein; G Marx; B Lentz; C Bokemeyer; F Langer
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Publication Detail:
Type:  Journal Article    
Journal Detail:
Title:  Hämostaseologie     Volume:  32 Suppl 1     ISSN:  0720-9355     ISO Abbreviation:  Hamostaseologie     Publication Date:  2012  
Date Detail:
Created Date:  2012-09-10     Completed Date:  -     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  8204531     Medline TA:  Hamostaseologie     Country:  Germany    
Other Details:
Languages:  eng     Pagination:  S48-51     Citation Subset:  IM    
Dr. Katharina Holstein, Gerinnungsambulanz und Hämophiliezentrum, II. Medizinische Klinik und Poliklinik, Universitätsklinikum Hamburg-Eppendorf, Martinistr. 52, 20246 Hamburg, Germany, Tel. +49/(0)40/741052453, Fax +49/(0)40/741055193, E-mail:
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