缺乏星形胶质细胞稳态支持有助于杜氏肌营养不良的脑损伤。

IF 3.8 3区 医学 Q2 BIOCHEMISTRY & MOLECULAR BIOLOGY
Dariusz C Gorecki, Abdulsamie M Patel, Joanna Pomeroy, Alexei Verkhratsky
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引用次数: 0

摘要

除了它们的肌肉功能外,肌营养不良蛋白(DMD基因的蛋白质产物)也在中枢神经系统(CNS)中发挥作用。在杜氏肌营养不良症(DMD)中,进行性肌肉退化与认知和行为障碍有关,三分之一的患者严重,并大大增加了总体临床负担。这些神经精神异常的机制是复杂的,只是部分理解。特异性营养不良蛋白在各种脑细胞及其特化中具有重要的功能,在整个大脑发育和成熟过程中,大脑各区域的多样性进一步增加。影响全长肌营养不良蛋白(Dp427)的突变导致疾病,而额外的较短亚型(Dp140和Dp71)的丢失会加剧临床表现。截断型肌营养不良蛋白在健康大脑中强烈表达,因此被广泛研究,但大多数表现为神经精神异常的DMD患者没有影响这些亚型的突变。当考虑到全长肌营养不良蛋白时,异常无疑不仅涉及神经元,还涉及星形胶质细胞,其中缺乏肌营养不良蛋白会减少增殖,破坏神经递质调节,突触稳定性和神经血管完整性。星形细胞营养不良蛋白的缺乏损害谷氨酸清除,导致与多种神经精神表现相关的兴奋性毒性和神经元高兴奋性。这篇综述评估了肌营养不良蛋白在星形胶质细胞中的作用,它对突触功能障碍的贡献,以及与其他神经发育障碍的相似之处。了解星形胶质细胞内平衡缺陷导致DMD可以导致探索新的治疗策略,包括星形胶质细胞介导的神经递质调节,以减轻神经精神缺陷。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Deficient Astrocyte Homeostatic Support Contributes to Brain Impairment in Duchenne Muscular Dystrophy.

In addition to their muscle functions, dystrophins -the protein products of the DMD gene - also play a role in the central nervous system (CNS). In Duchenne muscular dystrophy (DMD), the progressive muscle degeneration, is associated with cognitive and behavioural impairments, which are severe in one-third of patients and substantially increase the overall clinical burden. The mechanisms of these neuropsychiatric abnormalities are complex and only partially understood. Specific dystrophins are functionally important in various brain cells and at their specialisations, with further diversity occurring throughout brain development and maturation across brain regions. Mutations affecting the full-length dystrophins (Dp427) cause the disease, while the additional loss of shorter isoforms (Dp140 and Dp71) can exacerbate the clinical presentation. Truncated dystrophins are strongly expressed in the healthy brain and therefore were investigated extensively, but most of DMD patients presenting with neuropsychiatric abnormalities do not have mutations affecting these isoforms. When the full-length dystrophins are considered, the abnormalities unquestionably involve not only neurones but also astrocytes, where absence of dystrophins reduces proliferation, disrupts neurotransmitter regulation, synaptic stability, and neurovascular integrity. Deficiency of astrocytic dystrophins impairs glutamate clearance, leading to excitotoxicity and neuronal hyperexcitability linked to multiple neuropsychiatric manifestations. This review evaluates evidence on dystrophin's role in astrocytes, its contribution to synaptic malfunction, and parallels with other neurodevelopmental disorders. Understanding that the deficient astrocyte homeostasis contributes to DMD can lead to the exploration of novel therapeutic strategies involving astrocyte-mediated neurotransmitter regulation to mitigate neuropsychiatric deficits.

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来源期刊
Neurochemical Research
Neurochemical Research 医学-神经科学
CiteScore
7.70
自引率
2.30%
发文量
320
审稿时长
6 months
期刊介绍: Neurochemical Research is devoted to the rapid publication of studies that use neurochemical methodology in research on nervous system structure and function. The journal publishes original reports of experimental and clinical research results, perceptive reviews of significant problem areas in the neurosciences, brief comments of a methodological or interpretive nature, and research summaries conducted by leading scientists whose works are not readily available in English.
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