Pulai J I, Latour M A, Kwok P Y, Schonfeld G
Department of Internal Medicine, Washington University School of Medicine, St. Louis, MO 63110, USA.
Atherosclerosis. 1998 Feb;136(2):289-95. doi: 10.1016/s0021-9150(97)00222-0.
Familial hypobetalipoproteinemia is an autosomal co-dominant disorder, which in a minority of cases is due to a truncation producing mutation in the apoB gene. We have identified an apoB mutation in a 40-year old hypobetalipoproteinemic man with Type II diabetes mellitus. Immunoblotting of plasma revealed a major band for apoB-100 and a minor band with estimated size between apoB-52 and apoB-55. The proband's 75-year old father with Type II diabetes and a non-diabetic daughter also possessed the truncated protein. Direct sequencing of the amplified fragment of genomic DNA revealed a C-->T transition at nt 7692 in exon 26 of the apoB gene. This substitution yielded a premature stop codon at residue 2495 and abolished a BsaI restriction endonuclease site. The identical mutation has been described previously; however, the genotypes and ancestors of the kindred were different, suggesting that the mutation may have occurred independently. The majority of apoB-55 was eluted as particles smaller than LDL-sized apoB-100, and floated mostly between the LDL and HDL density range. It is worth noting that despite the presence of Type II diabetes, both the proband and his father have very low plasma lipid levels and neither have any clinically manifest macrovascular complications.
家族性低β脂蛋白血症是一种常染色体共显性疾病,少数情况下是由于载脂蛋白B(apoB)基因发生截短突变所致。我们在一名患有2型糖尿病的40岁低β脂蛋白血症男性中鉴定出一种apoB突变。血浆免疫印迹显示apoB - 100有一条主要条带,以及一条估计大小在apoB - 52和apoB - 55之间的次要条带。先证者75岁患有2型糖尿病的父亲和一个非糖尿病女儿也拥有这种截短蛋白。对基因组DNA扩增片段进行直接测序,发现在apoB基因第26外显子的第7692位核苷酸处发生了C→T转换。这种替换在第2495位残基处产生了一个提前终止密码子,并消除了一个BsaI限制性内切酶位点。之前已经描述过相同的突变;然而,该家族的基因型和祖先不同,这表明该突变可能是独立发生的。大多数apoB - 55以小于低密度脂蛋白(LDL)大小的apoB - 100的颗粒形式洗脱,并且大多漂浮在LDL和高密度脂蛋白(HDL)密度范围之间。值得注意的是,尽管存在2型糖尿病,但先证者及其父亲的血浆脂质水平都非常低,且均无任何临床明显的大血管并发症。