• 文献检索
  • 文档翻译
  • 深度研究
  • 学术资讯
  • Suppr Zotero 插件Zotero 插件
  • 邀请有礼
  • 套餐&价格
  • 历史记录
应用&插件
Suppr Zotero 插件Zotero 插件浏览器插件Mac 客户端Windows 客户端微信小程序
定价
高级版会员购买积分包购买API积分包
服务
文献检索文档翻译深度研究API 文档MCP 服务
关于我们
关于 Suppr公司介绍联系我们用户协议隐私条款
关注我们

Suppr 超能文献

核心技术专利:CN118964589B侵权必究
粤ICP备2023148730 号-1Suppr @ 2026

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验

作为溶酶体酸性脂肪酶缺乏症(LAL-D)表现形式的肝脏疾病和血脂异常。临床与诊断方面,以及一种新疗法。最新进展。

Liver disease and dyslipidemia as a manifestation of lysosomal acid lipase deficiency (LAL-D). Clinical and diagnostic aspects, and a new treatment. An update.

作者信息

Bay Luisa, Canero Velasco Cristina, Ciocca Mirta, Cotti Andrea, Cuarterolo Miriam, Fainboim Alejandro, Fassio Eduardo, Galoppo Marcela, Pinero Federico, Rozenfeld Paula

机构信息

Consultora, Hospital Nacional de Pediatría J. P. Garrahan, Ciudad Autónoma de Buenos Aires.

Sección Hepatología Infantil, Hospital Municipal de Niños de San Justo, Provincia de Buenos Aires.

出版信息

Arch Argent Pediatr. 2017 Jun 1;115(3):287-293. doi: 10.5546/aap.2017.eng.287.

DOI:10.5546/aap.2017.eng.287
PMID:28504497
Abstract

Lysosomal acid lipase deficiency (LAL-D) is still a little recognized genetic disease with significant morbidity and mortality in children and adults. This document provides guidance on when to suspect LAL-D and how to diagnose it. It is recommended to add lysosomal acid lipase deficiency to the List of differential diagnoses of sepsis, oncological diseases, storage diseases, persistent diarrhea, chronic malnutrition, and hemophagocytic lymphohistiocytosis. It should also be considered in young patients with dyslipidemia and atherosclerosis as well as diseases associated with fatty liver and/or hepatomegaly. LAL-D should be suspected in patients with hepatomegaly, hyperlipidemia and /or elevated transaminases found during routine checks or testing for other conditions, and in patients with cryptogenic cirrhosis. At present, there is the option of a specific enzyme replacement treatment.

摘要

溶酶体酸性脂肪酶缺乏症(LAL-D)仍是一种鲜为人知的遗传性疾病,在儿童和成人中具有较高的发病率和死亡率。本文档提供了关于何时怀疑LAL-D以及如何诊断该病的指导。建议将溶酶体酸性脂肪酶缺乏症添加到脓毒症、肿瘤性疾病、贮积病、持续性腹泻、慢性营养不良和噬血细胞性淋巴组织细胞增生症的鉴别诊断列表中。对于血脂异常和动脉粥样硬化的年轻患者以及与脂肪肝和/或肝肿大相关的疾病,也应考虑该病。在因其他病症进行常规检查或检测时发现肝肿大、高脂血症和/或转氨酶升高的患者,以及隐源性肝硬化患者中,应怀疑LAL-D。目前,有特定的酶替代治疗方案可供选择。

相似文献

1
Liver disease and dyslipidemia as a manifestation of lysosomal acid lipase deficiency (LAL-D). Clinical and diagnostic aspects, and a new treatment. An update.作为溶酶体酸性脂肪酶缺乏症(LAL-D)表现形式的肝脏疾病和血脂异常。临床与诊断方面,以及一种新疗法。最新进展。
Arch Argent Pediatr. 2017 Jun 1;115(3):287-293. doi: 10.5546/aap.2017.eng.287.
2
Lysosomal acid lipase deficiency--an under-recognized cause of dyslipidaemia and liver dysfunction.溶酶体酸性脂肪酶缺乏症——一种被低估的血脂异常和肝功能障碍的病因。
Atherosclerosis. 2014 Jul;235(1):21-30. doi: 10.1016/j.atherosclerosis.2014.04.003. Epub 2014 Apr 15.
3
Lysosomal Acid Lipase Deficiency: Could Dyslipidemia Drive the Diagnosis?
Curr Pediatr Rev. 2017;13(4):232-242. doi: 10.2174/1573396314666180111144514.
4
Lysosomal acid lipase deficiency: Expanding differential diagnosis.溶酶体酸性脂肪酶缺乏症:扩大鉴别诊断范围。
Mol Genet Metab. 2017 Jan-Feb;120(1-2):62-66. doi: 10.1016/j.ymgme.2016.11.002. Epub 2016 Nov 10.
5
Lysosomal acid lipase deficiency manifestations in children and adults: Baseline data from an international registry.溶酶体酸性脂肪酶缺乏症在儿童和成人中的表现:国际注册研究的基线数据。
Liver Int. 2023 Jul;43(7):1537-1547. doi: 10.1111/liv.15620. Epub 2023 May 24.
6
Diagnostic and therapeutic management of children with lysosomal acid lipase deficiency (LAL-D). Review of the literature and own experience.溶酶体酸性脂肪酶缺乏症(LAL-D)患儿的诊断与治疗管理。文献综述及自身经验。
Dev Period Med. 2016;20(3):212-215.
7
Molecular and clinical characterization of a series of patients with childhood-onset lysosomal acid lipase deficiency. Retrospective investigations, follow-up and detection of two novel LIPA pathogenic variants.儿童期起病溶酶体酸性脂肪酶缺乏症患者的分子和临床特征。回顾性研究、随访及两种新型 LIPA 致病性变异的检测。
Atherosclerosis. 2017 Oct;265:124-132. doi: 10.1016/j.atherosclerosis.2017.08.021. Epub 2017 Aug 26.
8
The Frequency of Lysosomal Acid Lipase Deficiency in Children With Unexplained Liver Disease.不明原因肝病儿童中溶酶体酸性脂肪酶缺乏症的频率。
J Pediatr Gastroenterol Nutr. 2019 Mar;68(3):371-376. doi: 10.1097/MPG.0000000000002224.
9
Managing Cardiovascular Risk in Lysosomal Acid Lipase Deficiency.溶酶体酸性脂肪酶缺乏症中心血管风险的管理
Am J Cardiovasc Drugs. 2017 Jun;17(3):217-231. doi: 10.1007/s40256-017-0216-5.
10
Does Lysosomial Acid Lipase Reduction Play a Role in Adult Non-Alcoholic Fatty Liver Disease?溶酶体酸性脂肪酶减少在成人非酒精性脂肪性肝病中起作用吗?
Int J Mol Sci. 2015 Nov 25;16(12):28014-21. doi: 10.3390/ijms161226085.

引用本文的文献

1
Pedigree Analysis of Nonclassical Cholesteryl Ester Storage Disease with Dominant Inheritance in a LIPA I378T Heterozygous Carrier.载脂蛋白基因 I378T 杂合突变致非经典型胆固醇酯贮积病家系分析
Dig Dis Sci. 2024 Jun;69(6):2109-2122. doi: 10.1007/s10620-024-08395-9. Epub 2024 Apr 2.
2
Metabolomic and lipidomic studies on the intervention of taurochenodeoxycholic acid in mice with hyperlipidemia.牛磺鹅去氧胆酸对高脂血症小鼠干预作用的代谢组学和脂质组学研究
Front Pharmacol. 2023 Nov 15;14:1255931. doi: 10.3389/fphar.2023.1255931. eCollection 2023.
3
A Form of Metabolic-Associated Fatty Liver Disease Associated with a Novel LIPA Variant.
一种与新型 LIPA 变异相关的代谢相关脂肪性肝病。
Arch Iran Med. 2023 Feb 1;26(2):86-91. doi: 10.34172/aim.2023.14.
4
Natural history and management of liver dysfunction in lysosomal storage disorders.溶酶体贮积症中肝功能障碍的自然史与管理
World J Hepatol. 2022 Oct 27;14(10):1844-1861. doi: 10.4254/wjh.v14.i10.1844.
5
The Emerging Battle: Lysosomal Acid Lipase Deficiency vs Familial Hypercholesterolemia in Children.新出现的较量:儿童溶酶体酸性脂肪酶缺乏症与家族性高胆固醇血症
ACG Case Rep J. 2021 Jan 13;8(1):e00516. doi: 10.14309/crj.0000000000000516. eCollection 2021 Jan.
6
Cholesteryl ester storage disease of clinical and genetic characterisation: A case report and review of literature.胆固醇酯贮积病的临床与遗传学特征:一例报告及文献复习
World J Clin Cases. 2020 May 6;8(9):1642-1650. doi: 10.12998/wjcc.v8.i9.1642.
7
Lysosomal Acid Lipase Deficiency, a Rare Pathology: The First Pediatric Patient Reported in Colombia.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
Am J Case Rep. 2018 Jun 9;19:669-672. doi: 10.12659/AJCR.908808.
8
Genetically modified mouse models to study hepatic neutral lipid mobilization.研究肝内中性脂质动员的基因修饰小鼠模型。
Biochim Biophys Acta Mol Basis Dis. 2019 May 1;1865(5):879-894. doi: 10.1016/j.bbadis.2018.06.001. Epub 2018 Jun 5.