Post-Partum Factor VIII Inhibitors

Hauser, I. Jerome; Schneider, B.; Lechner, Klaus

doi:10.1055/s-0038-1651666

Cited by 114 publications

(24 citation statements)

References 2 publications

(2 reference statements)

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“…The final outcome of postpartum inhibitors is favorable, with a probability of disappearance of the inhibitor in the majority of cases (4,18). From our report in 1978 (4) and the present study (Table 1), the inhibitor disappeared spontaneously in 25 out of 40 cases.…”

supporting

confidence: 59%

“…Fortunately, the natural history of the hemophilic disease postpartum is characterized by a spontaneous disappearance of the factor VIII inhibitor in the majority of the cases after a mean period of 30 months (4,14,18). In acquired hemophilia A postpartum, the factor VIII inhibitor is usually of type II, with potency varying from weak to moderate (4,14).…”

Section: Discussionmentioning

confidence: 99%

“…No remission was observed in 11 cases after a follow-up period of a few to 108 months (Tables 1,2) or even 24 years in one exceptional case (14). Treatment with prednisone was found to be ineffective in acquired hemophilia postpartum (4,14,18) (Table 1 ). Our first case (4) showed gradual disappearance of factor VIII inhibitor both during and after azathioprine treatment, but this may have coincided with the spontaneous disappearance of the inhibitor after 10 months.…”

mentioning

confidence: 99%

“…The potency of the factor VIII inhibitor in acquired hemophilia A postpartum is rather low in the majority of cases, and high in only a minority (4,14,18). The median inhibitor level in 24 patients with acquired hemophilia A postpartum was rather low-20 Bethesda Units (BU) (18). In vitro inactivation of low titer factor VIII inhibitor in acquired hemophilia A postpartum usually shows an antibody of nonlinear type II factor VIII inactivation (4,14).…”

mentioning

confidence: 99%

“…Our first case (4) showed gradual disappearance of factor VIII inhibitor both during and after azathioprine treatment, but this may have coincided with the spontaneous disappearance of the inhibitor after 10 months. In a recent analysis of 51 patients with postpartum factor VIII inhibitor, the time to disappearance of the inhibitor was 8 months in 18 patients who were treated with immunosuppressives; 12 months in 23 patients who were treated with steroids; and 16 months in 10 patients who were given neither steroids nor immunosuppressive treatment ( 18). This study demonstrates that immunosuppression in acquired hemophilia A postpartum does not induce the disappearance of the factor VIII inhibitor (complete remission), but may slightly reduce the time to complete remission after discontinuation of the immunosuppressive treatment.…”

mentioning

confidence: 99%

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Acquired Hemophilia A in Women Postpartum: Clinical Manifestations, Diagnosis, and Treatment

Michiels

2000

Clin Appl Thromb Hemost

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Acquired hemophilia A in women postpartum is diagnosed by a prolonged activated partial thromboplastin time (APTT), low plasma levels of coagulant factor VIII, and the detection of an inhibitor against factor VIII in the Bethesda assay. Effective treatment of bleeding symptoms should be based upon the clinical situation and depends on the inhibitor characteristics against human and porcine factor VIII. Immunosuppression usually does not significantly affect the disappearance of the factor VIII inhibitor antibody. The natural history of acquired hemophilia postpartum is independent of immunosuppressive treatment and featured by spontaneous disappearance of the inhibitor against factor VIII in the majority of cases.

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supporting

confidence: 59%

Section: Discussionmentioning

confidence: 99%

mentioning

confidence: 99%

mentioning

confidence: 99%

mentioning

confidence: 99%

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Acquired Hemophilia A in Women Postpartum: Clinical Manifestations, Diagnosis, and Treatment

Michiels

2000

Clin Appl Thromb Hemost

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Postpartum acquired factor VIII inhibitors: Results of a survey

Solymoss

1998

Am. J. Hematol.

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Postpartum factor VIII inhibitors are rare; thus, collecting clinical and treatment data may provide valuable information in guiding patient management. This paper reports the results of a survey of United States and Canadian hemophilia centers on 14 patients with postpartum acquired factor VIII inhibitors. Patients ranged in age from 23 to 40 years. Parity at diagnosis was most frequently the first gestation. Two patients were diagnosed prepartum, but two patients only up to one year postpartum. Initial inhibitor titers ranged from two to 550 Bethesda units (BU), and rose to five to 885 BU. The median duration of the inhibitor was 27 months, with two of the 14 inhibitors unresolved at the time of the survey. A total of 80 bleeding episodes occurred, five of which were life or limb threatening. Three patients received no blood products and were not hospitalized. Of those treated for hemostasis, activated prothrombin complex concentrates and porcine factor VIII were most often reported to be effective. For immunosuppression, all patients received steroids and most received additional treatment. Steroids were reported ineffective more frequently than cyclophosphamide. No adverse fetal events were noted and there was no maternal mortality. These results underline the clinical heterogeneity of the severity of postpartum factor VIII inhibitors, and provide treatment guidelines in the absence of prospective studies.

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