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溶酶体酸性脂肪酶缺乏症,一种罕见病症:哥伦比亚首例儿科患者报告

Lysosomal Acid Lipase Deficiency, a Rare Pathology: The First Pediatric Patient Reported in Colombia.

作者信息

Botero Verónica, Garcia Victor H, Gomez-Duarte Catalina, Aristizabal Ana M, Arrunategui Ana M, Echeverri Gabriel J, Pachajoa Harry

机构信息

Department of Pediatric Gastroenterology and Hepatology, Valle del Lili Foundation, Cali, Valle del Cauca, Colombia.

Center for Research on Advanced Surgery and Transplants (CICAT), Icesi University, Cali, Valle del Cauca, Colombia.

出版信息

Am J Case Rep. 2018 Jun 9;19:669-672. doi: 10.12659/AJCR.908808.

Abstract

BACKGROUND Lysosomal acid lipase deficiency is a rare genetic metabolic lipid storage disease, with a high morbidity, and mortality, in children and adults. It is characterized by a mutation in the LIPA gene that causes an alteration of lipid metabolism, resulting in deposits of cholesterol esters and triglycerides in organs such as the liver, blood vessels, and gastrointestinal tract. Lysosomal acid lipase deficiency is predominantly caused by the mutation c.894G>A, seen in approximately 50-70% of patients. Our objective is to report the first pediatric case of lysosomal acid lipase deficiency in a pediatric patient in Colombia. CASE REPORT The patient is a 14-year-old boy with isolated hepatomegaly since 6 years of age without a family history of dyslipidemia. In the pediatric control, laboratory exams revealed dyslipidemia, and a hepatic biopsy was performed, revealing severe fibrosis with septation and grade 3 microvesicular steatosis (>75%). He was referred to our center and was suspected to have lysosomal acid lipase deficiency. Enzymatic activity was measured, showing absent activity. Confirmatory diagnosis with genetic sequencing showed a pathological homozygous mutation of c.894G>A. CONCLUSIONS Lysosomal acid lipase deficiency can manifest as early- or late-onset, with variable and severe signs and symptoms. The late-onset form has a broad spectrum of manifestations with mild symptoms, leading to under-diagnosis, which increases the actual disease burden. Early diagnosis is essential to initiate enzyme replacement therapy, since the natural disease course can be changed. More studies should be conducted in Latin America to evaluate the prevalence of the disease.

摘要

背景

溶酶体酸性脂肪酶缺乏症是一种罕见的遗传性代谢性脂质贮积病,在儿童和成人中发病率和死亡率都很高。其特征是LIPA基因突变导致脂质代谢改变,致使胆固醇酯和甘油三酯在肝脏、血管和胃肠道等器官中沉积。溶酶体酸性脂肪酶缺乏症主要由c.894G>A突变引起,约50%-70%的患者存在该突变。我们的目的是报告哥伦比亚首例小儿溶酶体酸性脂肪酶缺乏症患儿病例。病例报告:该患者是一名14岁男孩,自6岁起出现孤立性肝肿大,无血脂异常家族史。在儿科检查中,实验室检查发现血脂异常,并进行了肝活检,结果显示严重纤维化伴分隔以及3级微泡性脂肪变性(>75%)。他被转诊至我们中心,怀疑患有溶酶体酸性脂肪酶缺乏症。测量酶活性,结果显示活性缺失。基因测序确诊显示存在c.894G>A病理性纯合突变。结论:溶酶体酸性脂肪酶缺乏症可表现为早发型或晚发型,体征和症状多样且严重。晚发型表现形式广泛,症状较轻,易导致诊断不足,从而增加了实际疾病负担。早期诊断对于启动酶替代治疗至关重要,因为这可以改变疾病的自然病程。拉丁美洲应开展更多研究以评估该疾病的患病率。

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