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Hemarthrosis as acute presentation of acquired hemophilia in a patient with systemic lupus erythematosus: successful treatment and long-lasting remission

Porru, Giovanni and Mura, Valentina and Piga, Matteo and Ibba, Valentina and Vacca, Alessandra and Cauli, Alberto and Passiu, Giuseppe and Targhetta, Roberto and Marongiu, Fabio and Mathieu, Alessandro (2008) Hemarthrosis as acute presentation of acquired hemophilia in a patient with systemic lupus erythematosus: successful treatment and long-lasting remission. Clinical Rheumatology, Vol. 27 (12), p. 1581-1584. eISSN 1434-9949. Article.

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DOI: 10.1007/s10067-008-0993-y

Abstract

Hemorrhagic events due to production of antibodies directed against coagulation factors are rarely observed in systemic lupus erythematosus (SLE). We report the case of a patient with clinically quiescent SLE who developed factor VIII inhibitor in acquired hemophilia presenting as hemarthrosis. Initial treatment with FVII, FVIII and FIX plasma concentrate, metilprednisolone and immunoglobulins i.v. were started but new hemorrhagic manifestation occurred. Plasma exchange was also administered, but it was discontinued early due to partial efficacy. In addition, pulse cyclophosphamide 0.5 g/m2 was started. Eight weeks later, FVIII and FIX activity returned within normal ranges, FVIII and FIX inhibitors decreased significantly and hemorrhagic manifestations disappeared. The rare occurrence of acquired hemophilia due to the presence of anti-factor VIII antibodies associated to SLE, which was reviewed, might explain the lack of therapeutic guide-lines; indeed therapeutic options are available but the outcome in each single patient is not predictable.

Item Type:Article
ID Code:4675
Status:Published
Refereed:Yes
Uncontrolled Keywords:Acquired hemophilia, combined treatment, hemarthrosis, systemic lupus erythematosus
Subjects:Area 06 - Scienze mediche > MED/16 Reumatologia
Divisions:001 Università di Sassari > 03 Istituti > Patologia speciale medica e metodologia clinica
Publisher:Springer London
eISSN:1434-9949
Deposited On:13 Oct 2010 09:16

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