Miller C H, Graham J B, Goldin L R, Elston R C
Blood. 1979 Jul;54(1):137-45.
In classic von Willebrand's disease (vWd), assignment of the heterozygous genotype for genetic studies and diagnosis for clinical purposes (which are not exactly the same) are formidable problems. We have pointed out in the first report in this series that almost 50% of the members of two large kindred who transmitted this disease, and were therefore heterozygous, were scored as normal by the usual tests of hemostasis. This report describes how this large proportion can be significantly reduced by application of discriminant analysis. Using linear discriminants in three variables--coagulation factor VIII (VIII:C), factor-VII-related antigen (VIIIR:Ag), and the ristocetin cofactor related to factor VIII (VIIIR:WF)--we were able to classify as heterozygous more than 80% of the transmitters in the two large kindred. It was of particular interest that the four parents of two related vWd homozygotes could be scored as heterozygous by discriminant analysis even though all their laboratory tests were within the normal ranges.
在典型的血管性血友病(vWd)中,为进行遗传学研究而确定杂合子基因型以及出于临床目的进行诊断(这两者并不完全相同)都是棘手的问题。在本系列的第一篇报告中,我们指出,在两个传递这种疾病的大家系中,近50%的成员为杂合子,但通过常规止血检测却被判定为正常。本报告描述了如何通过应用判别分析显著减少这一较大比例。利用凝血因子VIII(VIII:C)、因子VIII相关抗原(VIIIR:Ag)以及与因子VIII相关的瑞斯托霉素辅因子(VIIIR:WF)这三个变量进行线性判别分析,我们能够将两个大家系中超过80%的传递者判定为杂合子。特别值得关注的是,两个相关的vWd纯合子的四位父母,尽管他们所有的实验室检测结果都在正常范围内,但通过判别分析仍可被判定为杂合子。