Zimmerman T S, Abildgaard C F, Meyer D
N Engl J Med. 1979 Dec 13;301(24):1307-10. doi: 10.1056/NEJM197912133012402.
We attempted to characterize the small amounts of factor VIII-related antigen detectable in the severe recessive form of von Willebrand's disease with newly developed radioimmunoprecipitin techniques and radiocrossed immunoelectrophoresis. Previous studies have failed to demonstrate factor VIII-related antigen in most patients tested even with the highly sensitive immunoradiometric assays. Using the newer techniques, we found antigen in the plasma of six of eight patients with severe von Willebrand's disease from different kindreds. Qualitative abnormalities of the trace quantities of factor VIII-related antigen were demonstrated in five of the patients, with absence or relative decrease of the larger, less anodal forms. In addition, five distinct patterns were observed, each suggesting a different molecular abnormality. Heterozygous parents had normal to moderately decreased factor VIII-related antigen, with normal crossed immunoelectrophoretic patterns. This study suggests that severe von Willebrand's disease is a heterogeneous syndrome with various underlying molecular defects.
我们试图用新开发的放射免疫沉淀技术和放射交叉免疫电泳法来鉴定在重度隐性血管性血友病中可检测到的少量因子VIII相关抗原。以往的研究即使采用高度敏感的免疫放射分析方法,也未能在大多数受检患者中证实因子VIII相关抗原的存在。运用这些新技术,我们在来自不同家族的8例重度血管性血友病患者中的6例血浆中发现了抗原。5例患者的微量因子VIII相关抗原存在定性异常,较大的、向阳极移动较少的形式缺失或相对减少。此外,观察到5种不同的模式,每种模式提示一种不同的分子异常。杂合子父母的因子VIII相关抗原正常至中度降低,交叉免疫电泳模式正常。这项研究表明,重度血管性血友病是一种具有多种潜在分子缺陷的异质性综合征。