In a recent report, Lian and colleagues [1] described the effectiveness of combined cyclophosphamide, vincristine, and prednisone (CVP) therapy after antigenic stimulation with factor VIII to eradicate factor VIII inhibitor. We report the use of this regimen in a nonhemophilic woman with high-titer postpartum antibody against factor VIII that was resistant to other treatments.

A 45-year-old nonhemophilic black woman was first seen at the hospital in September 1990. She had a 10-year history of recurrent postpartum bleeding, characterized by epistaxis, ecchymoses, hematomas, and post-traumatic or spontaneous hemarthrosis. No history of previous bleeding or familial history of bleeding diathesis was noted. Her level of inhibitor to factor VIII was 4.0 BU/mL. Bleeding was controlled only with concentrate complex prothrombin activator but not with prednisone or desmopressin infusion. Administration of CVP therapy after factor VIII concentrate infusion was well tolerated, but the patient developed alopecia and refused the second CVP cycle. Three months later, she had a remission that was complete and persisted during 2 years of follow-up.

Although rare, the occurrence of an acquired factor VIII inhibitor related to pregnancy represents the second cause of such inhibitors in nonhemophilic patients [2]. Inhibitors have generally been present in low titers, and most patients improve spontaneously or with steroid treatment alone [3].

Our patient had high-titer postpartum inhibitor lasting 11 years and was resistant to usual forms of treatment. Some investigators have observed that patients with high titers of inhibitor respond less frequently to immunosuppression [1, 4]. It appears that the disappearance of inhibitor in our patient occurred only after CVP treatment and that aggressive immunosuppressive therapeutic modalities can be tried with success.