Cuthbert J A, East C A, Bilheimer D W, Lipsky P E
N Engl J Med. 1986 Apr 3;314(14):879-83. doi: 10.1056/NEJM198604033141404.
In familial hypercholesterolemia, structural and functional abnormalities of the receptor for low-density lipoprotein (LDL) lead to hypercholesterolemia and premature atherosclerosis. We have developed a simplified method to identify LDL-receptor defects in peripheral-blood lymphocytes. When lymphocytes are cultured in lipoprotein-depleted medium and endogenous sterol biosynthesis is suppressed with mevinolin, mitogen-stimulated proliferation of lymphocytes is dependent on an exogenous source of cholesterol. Whereas a small concentration of supplemental LDL cholesterol (3 to 4 micrograms per milliliter) permits a maximal response in normal lymphocytes, even high concentrations (10 to 50 micrograms per milliliter) are unable to support the proliferation of lymphocytes from patients with homozygous familial hypercholesterolemia. Thus, functional LDL receptors are necessary to allow lymphocyte proliferation in these cultures. The response of lymphocytes from patients with hyperlipidemia not caused by defective LDL receptors was like that of normal cells. In contrast, the response of lymphocytes from patients with heterozygous familial hypercholesterolemia was intermediate between that of homozygotes and that of normal or hyperlipidemic controls. Our method can therefore be used to identify persons who are heterozygous for abnormalities of LDL receptors.
在家族性高胆固醇血症中,低密度脂蛋白(LDL)受体的结构和功能异常会导致高胆固醇血症和早发性动脉粥样硬化。我们已开发出一种简化方法,用于识别外周血淋巴细胞中的LDL受体缺陷。当淋巴细胞在脂蛋白缺乏的培养基中培养,且用美伐他汀抑制内源性固醇生物合成时,有丝分裂原刺激的淋巴细胞增殖依赖于外源性胆固醇来源。正常淋巴细胞在补充少量LDL胆固醇(每毫升3至4微克)时就能产生最大反应,而即使是高浓度(每毫升10至50微克)的LDL胆固醇也无法支持纯合子家族性高胆固醇血症患者淋巴细胞的增殖。因此,功能性LDL受体是这些培养物中淋巴细胞增殖所必需的。由非缺陷性LDL受体引起的高脂血症患者的淋巴细胞反应与正常细胞相似。相比之下,杂合子家族性高胆固醇血症患者的淋巴细胞反应介于纯合子与正常或高脂血症对照之间。因此,我们的方法可用于识别LDL受体异常的杂合子个体。