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GM1神经节苷脂贮积症:临床、分子及治疗方面的综述

GM1 gangliosidosis: review of clinical, molecular, and therapeutic aspects.

作者信息

Brunetti-Pierri Nicola, Scaglia Fernando

机构信息

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX 77030, USA.

Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX 77030, USA; Texas Children's Hospital, Houston, TX 77030, USA.

出版信息

Mol Genet Metab. 2008 Aug;94(4):391-396. doi: 10.1016/j.ymgme.2008.04.012. Epub 2008 Jun 3.

Abstract

GM(1) gangliosidosis is a lysosomal storage disorder due to deficiency of the beta-galactosidase enzyme. This deficiency results in accumulation of GM(1) gangliosides and related glycoconjugates in the lysosomes leading to lysosomal swelling, cellular damage, and organ dysfunction. The disease is lethal in the infantile and juvenile forms. To date, up to 102 mutations distributed along the beta-galactosidase gene (GLB1) have been reported. This review gives an overview of the clinical and molecular findings in patients with GM(1) gangliosidosis. Furthermore, it describes therapeutic approaches which are currently under investigation in animal models of the disease.

摘要

GM(1)神经节苷脂贮积症是一种由于β-半乳糖苷酶缺乏引起的溶酶体贮积病。这种缺乏导致GM(1)神经节苷脂和相关糖缀合物在溶酶体中蓄积,从而导致溶酶体肿胀、细胞损伤和器官功能障碍。该疾病在婴儿型和青少年型中是致命的。迄今为止,已报道了沿β-半乳糖苷酶基因(GLB1)分布的多达102种突变。本综述概述了GM(1)神经节苷脂贮积症患者的临床和分子研究结果。此外,还描述了目前正在该疾病动物模型中研究的治疗方法。

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