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Successful Immunosuppressive Treatment of High-Titer Postpartum Factor VIII Inhibitor
- Joyce M. Annichino-Bizzacchi, MD;
- Tanya F. G. S. Machado, MD; and
- Valder R. Arruda, MD
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TO THE EDITOR:
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.
The Editors welcome submissions for possible publication in the Letters section. Authors of letters should:
Include no more than 300 words of text, three authors, and five references
Type with double-spacing
Send three copies of the letter, an authors' form signed by all authors, and a cover letter describing any conflicts of interest related to the contents of the letter.
Letters commenting on an Annals article will be considered if they are received within 6 weeks of the time the article was published. Only some of the letters received can be published. Published letters are edited and may be shortened; tables and figures are included only selectively. Authors will be notified that the letter has been received. If the letter is selected for publication, the author will be notified about 3 weeks before the publication date. Unpublished letters cannot be returned.
Annals welcomes electronically submitted letters.
- Copyright 2004 by the American College of Physicians
