法布里病的神经病变和疼痛

Vijay Krishna Medala, Nurcan Üçeyler
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摘要

法布里病(FD)是一种由α-半乳糖苷酶 A(GLA)基因突变引起的多器官溶酶体储积症。致病性 GLA 基因突变会导致酶活性受损甚至丧失,从而导致细胞和组织中的鞘脂类物质(如球糖基甘油酰胺)堆积。大多数 FD 患者会出现可诱发的疼痛,主要是刺痛和灼痛,这种疼痛通常在幼儿时期就已开始。虽然小纤维病理被认为是 FD 疼痛的基础,但其潜在的分子机制还不十分清楚。本综述总结了 FD 神经病变和神经性疼痛的临床特征,介绍了当前的治疗方案,并概述了实验和人体模型系统对导致小纤维病理学和 FD 相关疼痛的病理机制的最新发现。
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Neuropathy and pain in Fabry disease
Fabry disease (FD) is a multiorgan lysosomal storage disorder caused by mutations in the alfa-galactosidase A (GLA ) gene. Pathogenic GLA mutations lead to impaired or even lost enzyme activity, which causes the accumulation of sphingolipids, e.g., globotriaosylceramide, in cells and tissues. The majority of FD patients experience triggerable pain, mainly acral and burning, which often begins in early childhood. While small fiber pathology is assumed to be the basis of FD pain, the underlying molecular mechanisms are not well understood. This review summarizes the clinical characteristics of neuropathy and neuropathic pain in FD, presents current treatment options, and gives an overview of the latest findings from experimental and human model systems on the pathomechanisms contributing to small fiber pathology and FD-associated pain.
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