Mihaylov Vassil A, Horvath Anelia D, Savov Alexey S, Kurshelova Elina F, Paskaleva Ivanka D, Goudev Assen R, Stoilov Ivaylo R, Ganev Varban S
Department of Chemistry and Biochemistry, Medical University of Sofia, 2 Zdrave Str., Sofia, 1431, Bulgaria.
University Hospital for Obstetrics and Gynaecology, Medical University of Sofia, 2 Zdrave Str., Sofia, 1431, Bulgaria.
J Hum Genet. 2004;49(4):173-176. doi: 10.1007/s10038-004-0127-6. Epub 2004 Mar 10.
Familial hypercholesterolemia (FH) is a common, autosomal dominant disorder of lipid metabolism, caused by defects in the receptor-mediated uptake of LDL (low-density lipoproteins) due to mutations in the LDL receptor gene ( LDLR). Mutations underlying FH in Bulgaria are largely unknown. The aim of the present study was to provide information about the spectrum of point mutations in LDLR in a sample of 45 Bulgarian patients with severe hypercholesterolemia. Exons 3, 4, 6, 8, 9, and 14, previously shown to be mutational hot spots in LDLR, were screened using PCR-single-strand conformation polymorphism (SSCP). Samples with abnormal SSCP patterns were sequenced. Three different, hitherto undescribed point mutations (367T>A, 377T>A, 917C>A) and two previously described mutations (858C>A and 1301C>T) in eight unrelated patients were identified; four of the detected point mutations being missense mutations and one, a nonsense mutation. One of the newly described point mutations (917C>A) is a base substitution at a nucleotide position, at which two other different base substitutions have already been reported. Thus, all three possible base substitutions at this nucleotide position have been detected, making it a hot spot for point mutations causing FH. This is the first such mutational hot spot described in exon 6 of LDLR.
家族性高胆固醇血症(FH)是一种常见的常染色体显性脂质代谢紊乱疾病,由低密度脂蛋白受体基因(LDLR)突变导致受体介导的低密度脂蛋白(LDL)摄取缺陷引起。保加利亚FH的潜在突变情况 largely unknown。本研究的目的是提供45例保加利亚严重高胆固醇血症患者样本中LDLR点突变谱的信息。使用聚合酶链反应-单链构象多态性(PCR-SSCP)技术对先前已显示为LDLR突变热点的外显子3、4、6、8、9和14进行筛查。对SSCP模式异常的样本进行测序。在8例无亲缘关系的患者中鉴定出3种不同的、迄今未描述的点突变(367T>A、377T>A、917C>A)和2种先前描述的突变(858C>A和1301C>T);检测到的点突变中有4个为错义突变,1个为无义突变。新描述的点突变之一(917C>A)是在一个核苷酸位置的碱基替换,此前已报道过另外两种不同的碱基替换。因此,在该核苷酸位置已检测到所有三种可能的碱基替换,使其成为导致FH的点突变热点。这是在LDLR外显子6中首次描述的此类突变热点。