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球样细胞脑白质营养不良中的脱髓鞘和神经退行性变机制。

Mechanisms of demyelination and neurodegeneration in globoid cell leukodystrophy.

机构信息

Hunter James Kelly Research Institute, Buffalo, New York, USA.

Department of Biochemistry, Jacobs School of Medicine and Biomedical Sciences, State University of New York at Buffalo, Buffalo, New York, USA.

出版信息

Glia. 2021 Oct;69(10):2309-2331. doi: 10.1002/glia.24008. Epub 2021 Apr 14.

Abstract

Globoid cell leukodystrophy (GLD), also known as Krabbe disease, is a lysosomal storage disorder causing extensive demyelination in the central and peripheral nervous systems. GLD is caused by loss-of-function mutations in the lysosomal hydrolase, galactosylceramidase (GALC), which catabolizes the myelin sphingolipid galactosylceramide. The pathophysiology of GLD is complex and reflects the expression of GALC in a number of glial and neural cell types in both the central and peripheral nervous systems (CNS and PNS), as well as leukocytes and kidney in the periphery. Over the years, GLD has garnered a wide range of scientific and medical interests, especially as a model system to study gene therapy and novel preclinical therapeutic approaches to treat the spontaneous murine model for GLD. Here, we review recent findings in the field of Krabbe disease, with particular emphasis on novel aspects of GALC physiology, GLD pathophysiology, and therapeutic strategies.

摘要

球形细胞脑白质营养不良(GLD),也称 Krabbe 病,是一种溶酶体贮积病,可导致中枢和周围神经系统广泛脱髓鞘。GLD 是由溶酶体水解酶半乳糖脑苷脂酶(GALC)的功能丧失性突变引起的,该酶可分解髓鞘鞘脂半乳糖脑苷脂。GLD 的病理生理学较为复杂,反映了 GALC 在中枢和周围神经系统(CNS 和 PNS)以及外周血白细胞和肾脏中的多种神经胶质细胞和神经元中的表达。多年来,GLD 引起了广泛的科学和医学兴趣,特别是作为研究基因治疗和新型临床前治疗方法的模型系统,用于治疗 GLD 的自发性鼠模型。在这里,我们综述了 Krabbe 病领域的最新发现,特别强调了 GALC 生理学、GLD 病理生理学和治疗策略的新方面。

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