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Rheumatology 1999; 38: 1007-1009
© 1999 British Society for Rheumatology

Case Report

Long-lasting remission and successful treatment of acquired factor VIII inhibitors using cyclophosphamide in a patient with systemic lupus erythematosus

F. Trotta, G. Bajocchi, R. La Corte, S. Moratelli1 and L.-Y. Sun2

Division of Rheumatology, Arcispedale S. Anna,
1 Centre for the Study of Thrombosis and Haemostasis, University of Ferrara, Italy and
2 Department of Immunology, Gu Lou Hospital, Nanjing, China

Correspondence to: G. Bajocchi, Divisione di Reumatologia, Arcispedale S. Anna, Cozso Giovecca 203, 44100 Ferrara, Italy.

Acquired deficiency of clotting factor VIII (FVIII) is a rare bleeding diathesis seldom encountered in systemic lupus erythematosus (SLE). Reduction of FVIII activity by autoantibodies can cause potentially life-threatening situations. Herein, an SLE patient with a positive lupus anticoagulant (LAC) test who abruptly developed metrorrhagia 4 yr after diagnosis is reported. Coagulation tests revealed FVIII activity reduced to 3% and a prolonged aPTT. FVIII inhibitor(s) were found to be as high as 3.0 Bethesda Units. Plasmapheresis, immunoglobulins, prednisolone and FVIII plasma concentrates induced the cessation of metrorrhagia, but the clotting tests were barely improved. One month later, extensive ecchymosis appeared and worsened, despite re-administration of the previous therapy. Pulse cyclophosphamide followed by oral administration was then started with normalization of coagulation parameters and long-lasting disease remission.

KEY WORDS: Systemic lupus erythematosus, Factor VIII, Acquired deficiency, Metrorrhagia, Treatment, Cyclophosphamide.


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