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[卵磷脂胆固醇酰基转移酶缺乏症:一种肾脏科诊断]

[LCAT deficiency: a nephrological diagnosis].

作者信息

Boscutti Giuliano, Calabresi Laura, Pizzolitto Stefano, Boer Emanuela, Bosco Manuela, Mattei Piero Luigi, Martone Massimiliano, Milutinovic Neva, Berbecar Dorina, Beltram Elisabetta, Franceschini Guido

机构信息

SOC di Nefrologia e Dialisi, ASS2 Isontina Ospedale S. Giovanni di Dio, Gorizia, Italy.

出版信息

G Ital Nefrol. 2011 Jul-Aug;28(4):369-82.

Abstract

A genetic mendelian autosomal recessive condition of deficiency of lecithin- cholesterol acyltransferase (LCAT) can produce two different diseases: one highly interesting nephrologic picture of complete enzymatic deficiency (lecithin:cholesterol acyltransferase deficiency; OMIM ID #245900; FLD), characterized by the association of dyslipidemia, corneal opacities, anemia and progressive nephropathy; and a partial form (fish eye disease; OMIM ID #136120; FED) with dyslipidemia and progressive corneal opacities only. The diagnosis of FLD falls first of all under the competence of nephrologists, because end-stage renal disease appears to be its most severe outcome. The diagnostic suspicion is based on clinical signs (corneal opacities, more severe anemia than expected for the degree of chronic renal failure, progressive proteinuric nephropathy) combined with histology obtained by kidney biopsy (glomerulopathy evolving toward sclerosis with distinctive lipid deposition). However, the final diagnosis, starting with a finding of extremely low levels of HDL-cholesterol, requires collaboration with lipidology Centers that can perform sophisticated investigations unavailable in common laboratories. To be heterozygous for a mutation of the LCAT gene is one of the monogenic conditions underlying primary hypoalphalipoproteinemia (OMIM ID #604091). This disease, which is characterized by levels of HDL-cholesterol below the 5th percentile of those of the examined population (<28 mg/dL for Italians), has heritability estimates between 40% and 60% and is considered to be a predisposing condition for coronary artery disease. Nevertheless, some monogenic forms, and especially those associated with LCAT deficiency, seem to break the rule, confirming once more the value of a proper diagnosis before drawing prognostic conclusions from a laboratory marker. As in many other rare illnesses, trying to discover all the existing cases will contribute to allow studies broad enough to pave the way for further therapies, in this case also fostering the production by industries of the lacking enzyme by genetic engineering. Epidemiological studies, although done on selected populations such as hypoalphalipoproteinemia patients on dialysis and with the effective genetic tools of today, have been disappointing in elucidating the disease. Spreading the clinical knowledge of the disease and its diagnostic course among nephrologists seems to be the best choice, and this is the aim of our work.

摘要

卵磷脂胆固醇酰基转移酶(LCAT)缺乏的一种孟德尔常染色体隐性遗传病可引发两种不同疾病:一种是完全酶缺乏的极具研究价值的肾脏疾病表现(卵磷脂:胆固醇酰基转移酶缺乏症;OMIM编号#245900;FLD),其特征为血脂异常、角膜混浊、贫血和进行性肾病;另一种是仅伴有血脂异常和进行性角膜混浊的部分形式(鱼眼病;OMIM编号#136120;FED)。FLD的诊断首先属于肾病学家的职责范围,因为终末期肾病似乎是其最严重的后果。诊断怀疑基于临床体征(角膜混浊、比慢性肾衰竭程度预期更严重的贫血、进行性蛋白尿性肾病)以及肾脏活检获得的组织学结果(肾小球病变发展为硬化并伴有独特的脂质沉积)。然而,最终诊断始于发现极低水平的高密度脂蛋白胆固醇,这需要与脂质学中心合作,脂质学中心能够进行普通实验室无法开展的复杂检测。携带LCAT基因突变的杂合子是原发性低α脂蛋白血症(OMIM编号#604091)的单基因疾病之一。这种疾病的特征是高密度脂蛋白胆固醇水平低于所检测人群的第5百分位数(意大利人为<28mg/dL),遗传度估计在40%至60%之间,被认为是冠状动脉疾病的易患因素。然而,一些单基因形式,尤其是与LCAT缺乏相关的形式,似乎并不遵循这一规律,这再次证实了在从实验室指标得出预后结论之前进行正确诊断的价值。与许多其他罕见疾病一样,努力发现所有现有病例将有助于开展足够广泛的研究,为进一步治疗铺平道路,在这种情况下还能促进工业界通过基因工程生产缺失的酶。尽管流行病学研究是针对诸如透析的低α脂蛋白血症患者等特定人群,并运用了当今有效的基因工具,但在阐明该疾病方面一直不尽人意。在肾病学家中传播该疾病的临床知识及其诊断过程似乎是最佳选择,这也是我们工作的目标。

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