Taylor R, Jeenah M, Seed M, Humphries S
Charing Cross Sunley Research Centre, London.
J Med Genet. 1988 Oct;25(10):653-9. doi: 10.1136/jmg.25.10.653.
We have studied four different restriction fragment length polymorphisms (RFLPs) for the LDL receptor gene, detected using the restriction enzymes StuI, PvuII, ApaLI, and NcoI, in normal subjects and in patients with familial hypercholesterolaemia (FH) from London. Significant linkage disequilibrium was detected between all four RFLPs. Used together they give a polymorphism information content (PIC) of greater than 0.7 which makes them useful for studying the inheritance of the LDL receptor gene in more than 70% of families with FH. The NcoI and ApaLI RFLPs were found to be the most useful, giving a combined PIC value of 0.6. The allele frequencies of all four polymorphisms were compared in the normal and FH groups and the frequency of the rarer N2 allele of the NcoI RFLP was found to be significantly higher in the FH group. This suggests that a mutation has occurred on the rare NcoI N2 allele and that it may be making a significant contribution to the defects causing FH in this patient group. We have also used these RFLPs to look for evidence that variation at the LDL receptor gene locus contributes to the determination of cholesterol levels in the normal population. People with different RFLP genotypes do not have significantly different levels of serum total or LDL cholesterol. At present we have no evidence that variation at this locus may be determining cholesterol levels in the non-FH population.
我们研究了低密度脂蛋白受体基因的四种不同限制性片段长度多态性(RFLP),使用限制性内切酶StuI、PvuII、ApaLI和NcoI进行检测,研究对象为正常受试者以及来自伦敦的家族性高胆固醇血症(FH)患者。在所有这四种RFLP之间均检测到显著的连锁不平衡。它们一起使用时,多态性信息含量(PIC)大于0.7,这使得它们对于研究超过70%的FH家族中低密度脂蛋白受体基因的遗传情况很有用。发现NcoI和ApaLI RFLP最为有用,其组合PIC值为0.6。比较了正常组和FH组中所有四种多态性的等位基因频率,发现FH组中NcoI RFLP较罕见的N2等位基因频率显著更高。这表明在罕见的NcoI N2等位基因上发生了突变,并且它可能对导致该患者群体FH的缺陷有重大贡献。我们还使用这些RFLP来寻找证据,以证明低密度脂蛋白受体基因位点的变异对正常人群胆固醇水平的决定有影响。具有不同RFLP基因型的人血清总胆固醇或低密度脂蛋白胆固醇水平没有显著差异。目前我们没有证据表明该位点的变异可能决定非FH人群的胆固醇水平。