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原发性脂蛋白脂肪酶活性缺乏症:法裔加拿大人群的临床研究。

Primary lipoprotein-lipase-activity deficiency: clinical investigation of a French Canadian population.

作者信息

Gagné C, Brun L D, Julien P, Moorjani S, Lupien P J

机构信息

Department of Medicine, Centre hospitalier de l'université Laval, Ste-Foy, PQ.

出版信息

CMAJ. 1989 Feb 15;140(4):405-11.

Abstract

We examined 56 French Canadians, aged 1 week to 54 years, from eastern Quebec who were referred to the Laval University Lipid Research Centre and in whom coincidental finding (in 46% of the cases), abdominal pain (in 32%) or family screening (in 22%) led to the diagnosis of primary lipoprotein-lipase-activity deficiency (familial hyperchylomicronemia). Half of the patients had one or more of the following signs: lipemia retinalis, eruptive xanthomas, splenomegaly and hepatomegaly; the plasma triglyceride concentrations were significantly higher (greater than 40 mmol/L) among these patients than among those without clinical signs (mean 21.7 [standard deviation 13.5] mmol/L). The prevalence rate of this disorder was 30 times higher than the previously published rate and was highest in the counties of Charlevoix and Saguenay-Lac-St-Jean (200 and 100 cases per million respectively) because of the distinct demographic history of these areas. Because of a founder effect an autosomal recessive gene involved in lipoprotein-lipase expression or activation has probably been disseminated among this isolated French Canadian population.

摘要

我们对56名来自魁北克东部、年龄从1周大到54岁的法裔加拿大人进行了检查,这些人被转诊至拉瓦尔大学脂质研究中心,其中,偶然发现(46%的病例)、腹痛(32%)或家族筛查(22%)导致了原发性脂蛋白脂肪酶活性缺乏症(家族性高乳糜微粒血症)的诊断。一半的患者有以下一种或多种体征:视网膜脂血症、疹性黄瘤、脾肿大和肝肿大;这些患者的血浆甘油三酯浓度显著高于无临床体征的患者(大于40 mmol/L)(无临床体征患者的血浆甘油三酯浓度平均为21.7[标准差13.5]mmol/L)。这种疾病的患病率比之前公布的患病率高30倍,在沙勒沃伊县和萨格奈-圣让湖地区最高(分别为每百万人口200例和100例),原因是这些地区独特的人口统计学历史。由于奠基者效应,一个与脂蛋白脂肪酶表达或激活有关的常染色体隐性基因可能在这个与世隔绝的法裔加拿大人群中传播开来。

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