Söhngen D, Köster W, Kuntz B M, Glück S, Schneider W
Medizinische Klinik und Poliklinik, Heinrich-Heine Universität Düsseldorf.
Klin Wochenschr. 1990 Nov 9;68(21):1071-5. doi: 10.1007/BF01649308.
During the last ten years we observed three non-hemophilic patients with factor(F) VIII: C inhibitors (2 women aged 68 and 80 and a man aged 51). In all three cases, a sudden bleeding tendency was observed shortly after an injury or surgery. Coagulation tests showed a prolonged aPTT and a decreased F VIII: C level. Other deficiencies of blood-clotting factors and acquired or hereditary von Willebrand's disease could be excluded. Therapy with F VIII: C concentrate, cryoprecipitate, or fresh-frozen plasma did not produce the expected increase in F VIII: C. Measurement of F VIII: C inhibitor levels (Bethesda Units, BU) revealed values in the range between 9 and 64 BU. The two patients subjected to long-term therapy with a combination of prednisone (initially 2-3 mg/kg BW) and azathioprine (2-3 mg/kg BW) responded positively; the F VIII: C concentration increased. The third patient, treated only with a low dose of prednisone (30 mg/day), did not show any reaction at all. Since hereditary hemophilia A could be excluded, the inhibitors apparently were acquired. Malignant tumors did not appear. In conclusion, long-term therapy of an acquired F VIII: C inhibitor with a combination of prednisone and azathioprine may lead to complete disappearance of the inhibitor, normalization of the coagulation tests, and complete remission of the bleeding tendency.
在过去十年中,我们观察到三名非血友病患者出现了凝血因子(F)VIII:C抑制剂(两名女性,年龄分别为68岁和80岁,一名男性,年龄为51岁)。在所有三例病例中,受伤或手术后不久均突然出现出血倾向。凝血试验显示活化部分凝血活酶时间(aPTT)延长,F VIII:C水平降低。可以排除其他凝血因子缺乏以及获得性或遗传性血管性血友病。使用F VIII:C浓缩剂、冷沉淀或新鲜冰冻血浆进行治疗并未使F VIII:C出现预期的升高。F VIII:C抑制剂水平(贝塞斯达单位,BU)的测量结果显示值在9至64 BU之间。两名接受泼尼松(初始剂量为2 - 3 mg/kg体重)和硫唑嘌呤(2 - 3 mg/kg体重)联合长期治疗的患者反应良好;F VIII:C浓度升高。第三名患者仅接受低剂量泼尼松(30 mg/天)治疗,未显示任何反应。由于可以排除遗传性血友病A,这些抑制剂显然是获得性的。未出现恶性肿瘤。总之,使用泼尼松和硫唑嘌呤联合对获得性F VIII:C抑制剂进行长期治疗可能会导致抑制剂完全消失、凝血试验正常化以及出血倾向完全缓解。