Wadelius C, Blombäck M, Goonewardena P, Anvret M, Lindstedt M, Gustavson K H, Pettersson U
Department of Medical Genetics, University of Uppsala, Sweden.
Scand J Clin Lab Invest. 1991 Nov;51(7):625-33. doi: 10.1080/00365519109104573.
Haemophilia A is an X-linked disorder affecting 1.7/10,000 males. Carrier detection in females and prenatal diagnosis of male foetuses is greatly improved by DNA-based diagnosis. This study describes the use of the polymerase chain reaction (PCR) and allele-specific oligonucleotides (ASO) in a clinical study comprising 190 individuals in 27 families. Prenatal diagnosis was performed in eight cases. Of three male fetuses, two were found to be affected and one unaffected. It is shown that 62% of women in the Swedish population are heterozygous for the intragenic BclI or XbaI polymorphisms and consequently a majority of them (53%) can be analysed in the PCR-based format. Using three intragenic polymorphisms, a combined heterozygosity of 64% was recorded in the females. If the extragenic loci DXS52 and DXS15 were used in addition, 97% of the women could be counselled by DNA-analysis. Our study demonstrates the usefulness of PCR-based analysis of the BclI- and the XbaI-polymorphisms in genetic counselling. The counselling of women with conflicting results between coagulation data and DNA-based linkage analysis is discussed.
甲型血友病是一种X连锁疾病,每10000名男性中有1.7人患病。基于DNA的诊断极大地改善了女性携带者的检测和男性胎儿的产前诊断。本研究描述了聚合酶链反应(PCR)和等位基因特异性寡核苷酸(ASO)在一项包含27个家庭的190名个体的临床研究中的应用。对8例进行了产前诊断。在3名男性胎儿中,2例被发现患病,1例未患病。结果表明,瑞典人群中62%的女性为基因内BclI或XbaI多态性的杂合子,因此其中大多数(53%)可以采用基于PCR的方法进行分析。利用三种基因内多态性,女性的综合杂合率为64%。如果再加上基因外位点DXS52和DXS15,97%的女性可以通过DNA分析进行遗传咨询。我们的研究证明了基于PCR分析BclI和XbaI多态性在遗传咨询中的有用性。本文还讨论了凝血数据与基于DNA的连锁分析结果相互矛盾的女性的遗传咨询问题。