Hokanson J E, Brunzell J D, Jarvik G P, Wijsman E M, Austin M A
Divisions of Metabolism, Endocrinology, and Nutrition, Department of Medicine, University of Washington, Seattle WA 98195-6426, USA.
Am J Hum Genet. 1999 Feb;64(2):608-18. doi: 10.1086/302234.
Small low-density lipoprotein (LDL) particles are a genetically influenced coronary disease risk factor. Lipoprotein lipase (LpL) is a rate-limiting enzyme in the formation of LDL particles. The current study examined genetic linkage of LDL particle size to the LpL gene in five families with structural mutations in the LpL gene. LDL particle size was smaller among the heterozygous subjects, compared with controls. Among heterozygous subjects, 44% were classified as affected by LDL subclass phenotype B, compared with 8% of normal family members. Plasma triglyceride levels were significantly higher, and high-density lipoprotein cholesterol (HDL-C) levels were lower, in heterozygous subjects, compared with normal subjects, after age and sex adjustment. A highly significant LOD score of 6.24 at straight theta=0 was obtained for linkage of LDL particle size to the LpL gene, after adjustment of LDL particle size for within-genotype variance resulting from triglyceride and HDL-C. Failure to adjust for this variance led to only a modest positive LOD score of 1.54 at straight theta=0. Classifying small LDL particles as a qualitative trait (LDL subclass phenotype B) provided only suggestive evidence for linkage to the LpL gene (LOD=1. 65 at straight theta=0). Thus, use of the quantitative trait adjusted for within-genotype variance, resulting from physiologic covariates, was crucial for detection of significant evidence of linkage in this study. These results indicate that heterozygous LpL deficiency may be one cause of small LDL particles and may provide a potential mechanism for the increase in coronary disease seen in heterozygous LpL deficiency. This study also demonstrates a successful strategy of genotypic specific adjustment of complex traits in mapping a quantitative trait locus.
小密度脂蛋白(LDL)颗粒是一种受遗传影响的冠心病危险因素。脂蛋白脂肪酶(LpL)是LDL颗粒形成过程中的一种限速酶。本研究检测了5个LpL基因存在结构突变的家系中LDL颗粒大小与LpL基因的遗传连锁关系。与对照组相比,杂合子受试者的LDL颗粒较小。在杂合子受试者中,44%被归类为受LDL亚类表型B影响,而正常家庭成员中这一比例为8%。在对年龄和性别进行调整后,与正常受试者相比,杂合子受试者的血浆甘油三酯水平显著升高,高密度脂蛋白胆固醇(HDL-C)水平降低。在对LDL颗粒大小进行因甘油三酯和HDL-C导致的基因型内方差调整后,LDL颗粒大小与LpL基因连锁在直θ=0时获得了6.24的高度显著LOD分数。未对该方差进行调整导致在直θ=0时仅得到1.54的适度阳性LOD分数。将小LDL颗粒分类为定性性状(LDL亚类表型B)仅提供了与LpL基因连锁的提示性证据(直θ=0时LOD=1.65)。因此,使用经生理协变量导致的基因型内方差调整后的定量性状对于在本研究中检测连锁的显著证据至关重要。这些结果表明,杂合子LpL缺乏可能是小LDL颗粒的一个原因,并可能为杂合子LpL缺乏中所见的冠心病增加提供潜在机制。本研究还展示了在定位数量性状基因座时对复杂性状进行基因型特异性调整的成功策略。