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原发性乳糜微粒血症中的清除缺陷:组织脂蛋白脂肪酶活性研究

Clearance defects in primary chylomicronemia: a study of tissue lipoprotein lipase activities.

作者信息

Berger G M

出版信息

Metabolism. 1986 Nov;35(11):1054-61. doi: 10.1016/0026-0495(86)90043-0.

Abstract

Tissue and postheparin lipoprotein lipase (LPL) activities were assayed in a heterogeneous group of eight chylomicronemic subjects ranging in age from 15 years to 55 years. Three patients, presenting with the classical genetic and clinical features of type I hyperlipoproteinemia, had virtually absent adipose tissue LPL activity and markedly reduced muscle LPL activity (between 5% and 20% of normal). A fourth patient, with a similar but more benign lipoprotein and clinical phenotype, showed reduced adipose tissue LPL activity (10% of control) but retained essentially normal muscle enzyme. This patient represents a variant form of familial LPL deficiency. Two of the four remaining patients presented with typical features of adult type V hyperlipoproteinemia associated with familial hypertriglyceridemia (type IV and V phenotypes) in first-degree relatives. Adipose tissue LPL activities were 25% to 35% of the control mean in these patients, but muscle activities were normal or elevated. A third patient had suggestively similar tissue enzyme levels, but a family study could not be carried out. The eighth patient presented with a brittle type V phenotype, normolipidemia in the two first-degree relatives available for study and normal lipolytic activity in adipose tissue, muscle, and postheparin plasma assayed against a 14C-triolein substrate. An oral fat load in this patient, however, led to a marked but transient increase in light scattering suggesting defective clearance. Mixing experiments in vitro using a chylomicron substrate strongly suggested an extrinsic defect of lipolysis due to the inhibitory effect of excess very low density lipoprotein peptides, presumably apo C-III.(ABSTRACT TRUNCATED AT 250 WORDS)

摘要

对8名年龄在15岁至55岁之间的乳糜微粒血症患者进行了组织和肝素后脂蛋白脂肪酶(LPL)活性测定。3名患者具有I型高脂蛋白血症的典型遗传和临床特征,其脂肪组织LPL活性几乎缺失,肌肉LPL活性显著降低(为正常水平的5%至20%)。第四名患者具有相似但更良性的脂蛋白和临床表型,其脂肪组织LPL活性降低(为对照的10%),但肌肉酶基本正常。该患者代表家族性LPL缺乏的一种变异形式。其余4名患者中有2名表现出成人V型高脂蛋白血症的典型特征,一级亲属中有家族性高甘油三酯血症(IV型和V型表型)。这些患者的脂肪组织LPL活性为对照平均值的25%至35%,但肌肉活性正常或升高。第三名患者的组织酶水平提示相似,但无法进行家族研究。第八名患者表现出脆性V型表型,可用于研究的两名一级亲属血脂正常,以14C-三油酸甘油酯为底物测定的脂肪组织、肌肉和肝素后血浆中的脂解活性正常。然而,该患者口服脂肪负荷后导致光散射显著但短暂增加,提示清除缺陷。使用乳糜微粒底物进行的体外混合实验强烈提示,由于过量极低密度脂蛋白肽(可能是载脂蛋白C-III)的抑制作用,脂解存在外在缺陷。(摘要截短于250字)

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