Schuster H, Keller C, Wolfram G, Zöllner N
Medizinische Poliklinik, Universität München, FRG.
Ann Nutr Metab. 1992;36(2):79-86. doi: 10.1159/000177702.
Since the cloning of the human LDL receptor (LDLR) gene, familial hypercholesterolemia (FH) can be diagnosed by recombinant DNA technology either using restriction enzyme mapping to detect major rearrangements of the gene or using restriction fragment length polymorphisms (RFLPs) and linkage analysis in family studies. Genotypes and haplotypes of four RFLPs (StuI, ApaII 5', PvuII, NcoI) were used to study the inheritance of the detective LDLR gene in three families. Diagnosis of FH based on the lipid levels alone was not possible because in these kindreds both parents exhibit elevated lipid levels. However, in two families using haplotype analysis, elevated cholesterol levels in certain relatives could be attributed to the inheritance of a defective LDRL gene and thereby distinguished from hypercholesterolemia due to familial combined hyperlipidemia. In the third family where both hypercholesterolemic parents carried a defective LDLR gene, a case of homozygous FH could be excluded in a child by demonstrating the inheritance of a normal LDLR gene.
自人类低密度脂蛋白受体(LDLR)基因克隆以来,家族性高胆固醇血症(FH)可通过重组DNA技术进行诊断,既可以使用限制酶图谱检测该基因的主要重排,也可以在家族研究中使用限制性片段长度多态性(RFLP)和连锁分析。利用四种RFLP(StuI、ApaII 5'、PvuII、NcoI)的基因型和单倍型,研究了三个家族中缺陷性LDLR基因的遗传情况。仅根据血脂水平无法诊断FH,因为在这些家族中父母双方的血脂水平均升高。然而,在两个家族中通过单倍型分析,某些亲属胆固醇水平升高可归因于缺陷性LDRL基因的遗传,从而与家族性混合性高脂血症导致的高胆固醇血症区分开来。在第三个家族中,高胆固醇血症的父母双方都携带缺陷性LDLR基因,通过证明一个孩子继承了正常的LDLR基因,排除了该孩子患纯合子FH的情况。