Nashan D, Sunderkötter C, Hamm H, Krukenmeyer J, Bonsmann G
Universitäts-Hautklinik, Münster.
Hautarzt. 1995 May;46(5):339-42. doi: 10.1007/s001050050263.
A 61-year-old patient with life-threatening angioneurotic oedema was found to have an acquired C1-inhibitor (C1-INH) deficiency. In addition to lowered serum levels of C1-INH (both protein concentration and enzymatic activity), C2, C4 and CH50, which are characteristic for the hereditary form of angioneurotic oedema, markedly lowered C1q was found, which is typical for the acquired form. There were no antibodies against C1-INH. Repeated thorough examination disclosed no neoplasm, though the presence of neoplasm has often been reported to be associated with the acquired C1-INH deficiency. However, the patient showed persistent paraproteinaemia and paraproteinuria and developed livedo reticularis. Treatment with danazol resulted in a rise of the complement fraction levels and cessation of angioneurotic oedema. Paraproteinaemia and livedo reticularis persisted unchanged.
一名61岁患有危及生命的血管性水肿的患者被发现存在获得性C1抑制物(C1-INH)缺乏。除了血清C1-INH水平降低(蛋白质浓度和酶活性均降低)外,血管性水肿遗传性形式所特有的C2、C4和CH50也降低,同时还发现C1q显著降低,这是获得性形式的典型表现。不存在抗C1-INH抗体。反复全面检查未发现肿瘤,尽管经常有报道称肿瘤的存在与获得性C1-INH缺乏有关。然而,该患者持续存在副蛋白血症和副蛋白尿,并出现网状青斑。用达那唑治疗导致补体成分水平升高,血管性水肿停止。副蛋白血症和网状青斑持续不变。