Schaefer E J
Arteriosclerosis. 1984 Jul-Aug;4(4):303-22. doi: 10.1161/01.atv.4.4.303.
This review assesses current knowledge of the clinical, genetic, and biochemical features of familial high density lipoprotein (HDL) deficiency syndromes. The focus is on HDL deficiency states occurring in the absence of severe hypertriglyceridemia or lecithin/cholesterol acyltransferase deficiency. Specific entities falling within this category include Tangier disease, familial HDL deficiency with planar xanthomas, familial apolipoprotein A-I and C-III deficiency (formerly known as apolipoprotein A-I absence), familial deficiency of apolipoprotein A-I and C-III, fish-eye disease, familial hypoalphalipoproteinemia, and apolipoprotein A-I variants (apo A-I Milano, apo A-I Marburg, apo A-I Giessen, and apo A-I Munster 1-3). Diffuse corneal opacification and premature coronary artery disease are common features in many of these kindreds. No striking clinical abnormalities have been noted in patients with currently known apolipoprotein A-I variants, possibly because these subjects are heterozygotes for their respective defects. The HDL deficiency in many of these disorders has been associated with abnormalities or deficiencies of apolipoprotein A-I. Further research will undoubtedly define the defects in all the disorders that have been described, uncover new mutations, as well as provide additional insights into the precise relationship between HDL deficiency and atherosclerosis.
本综述评估了家族性高密度脂蛋白(HDL)缺乏综合征的临床、遗传和生化特征的现有知识。重点是在无严重高甘油三酯血症或卵磷脂/胆固醇酰基转移酶缺乏的情况下发生的HDL缺乏状态。属于这一类别的特定疾病包括Tangier病、伴有扁平黄色瘤的家族性HDL缺乏、家族性载脂蛋白A-I和C-III缺乏(以前称为载脂蛋白A-I缺乏)、载脂蛋白A-I和C-III家族性缺乏、鱼眼病、家族性低α脂蛋白血症以及载脂蛋白A-I变异体(载脂蛋白A-I米兰、载脂蛋白A-I马尔堡、载脂蛋白A-I吉森和载脂蛋白A-I明斯特1-3)。弥漫性角膜混浊和早发性冠状动脉疾病是许多这些家族的常见特征。在目前已知的载脂蛋白A-I变异体患者中未发现明显的临床异常,可能是因为这些个体是各自缺陷的杂合子。许多这些疾病中的HDL缺乏与载脂蛋白A-I的异常或缺乏有关。毫无疑问,进一步的研究将明确所有已描述疾病中的缺陷,发现新的突变,并提供对HDL缺乏与动脉粥样硬化之间精确关系的更多见解。