Fazio S, Lee Y L, Ji Z S, Rall S C
Gladstone Institute of Cardiovascular Disease, University of California, San Francisco 94141-9100.
J Clin Invest. 1993 Sep;92(3):1497-503. doi: 10.1172/JCI116728.
Transgenic mice were prepared that expressed a dysfunctional apo E variant, apo E (Arg-112, Cys-142), which is associated with dominant inheritance of type III hyperlipoproteinemia (type III HLP) in humans. Among eight founder mice, plasma apo E (Arg-112, Cys-142) levels varied 100-fold and directly correlated with plasma cholesterol and triglyceride levels. On a normal chow diet, mice expressing high levels (> 70 mg/dl) of the dysfunctional apo E had grossly elevated plasma lipids, with cholesterol levels of up to 410 mg/dl and triglyceride levels of up to 1,210 mg/dl. Upon agarose electrophoresis, plasma from these mice demonstrated beta-very low density lipoproteins (beta-VLDL). Mice expressing low (< 2.5 mg/dl) or intermediate (21 mg/dl) levels of the apo E variant had much less severe hyperlipidemia and did not have beta-VLDL. Although the transgenic mouse beta-VLDL were enriched in cholesteryl esters compared with normal mouse VLDL, they were not as cholesterol enriched as human beta-VLDL from type III HLP subjects. Transgenic mouse beta-VLDL injected into normal mice were cleared from plasma at a significantly slower rate than normal mouse VLDL, demonstrating the impaired catabolism of beta-VLDL. Thus, transgenic mice expressing high levels of the dysfunctional apo E (Arg-112, Cys-142) variant have many characteristics of the human type III HLP phenotype and appear to be a suitable animal model for this disorder.
制备了转基因小鼠,其表达功能失调的载脂蛋白E变体,即载脂蛋白E(精氨酸-112,半胱氨酸-142),该变体与人类III型高脂蛋白血症(III型HLP)的显性遗传相关。在八只奠基小鼠中,血浆载脂蛋白E(精氨酸-112,半胱氨酸-142)水平相差100倍,且与血浆胆固醇和甘油三酯水平直接相关。在正常的普通饮食下,表达高水平(>70mg/dl)功能失调载脂蛋白E的小鼠血浆脂质显著升高,胆固醇水平高达410mg/dl,甘油三酯水平高达1210mg/dl。在琼脂糖电泳中,这些小鼠的血浆显示出β-极低密度脂蛋白(β-VLDL)。表达低水平(<2.5mg/dl)或中等水平(21mg/dl)载脂蛋白E变体的小鼠高脂血症程度要轻得多,且没有β-VLDL。尽管与正常小鼠的极低密度脂蛋白相比,转基因小鼠的β-VLDL富含胆固醇酯,但它们不像来自III型HLP患者的人类β-VLDL那样富含胆固醇。注入正常小鼠体内的转基因小鼠β-VLDL从血浆中清除的速度明显慢于正常小鼠的极低密度脂蛋白,这表明β-VLDL的分解代谢受损。因此,表达高水平功能失调的载脂蛋白E(精氨酸-112,半胱氨酸-142)变体的转基因小鼠具有人类III型HLP表型的许多特征,似乎是这种疾病的合适动物模型。