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葡萄糖转运缺陷模型中的癫痫发作与氧化还原挽救

Seizure and redox rescue in a model of glucose transport deficiency.

作者信息

Coggan Jay S, Shichkova Polina, Markram Henry, Keller Daniel

机构信息

Blue Brain Project, EPFL: École Polytechnique Fédérale de Lausanne, Geneva, Switzerland.

Biognosys AG, Schlieren, Switzerland.

出版信息

PLoS Comput Biol. 2025 Apr 4;21(4):e1012959. doi: 10.1371/journal.pcbi.1012959. eCollection 2025 Apr.

DOI:10.1371/journal.pcbi.1012959
PMID:40184423
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12002639/
Abstract

Disruptions of energy supply to the brain are associated with many neurodegenerative pathologies and are difficult to study due to numerous interlinked metabolic pathways. We explored the effects of diminished energy supply on brain metabolism using a computational model of the neuro-glia-vasculature ensemble, in the form of a neuron, an astrocyte and local blood supply. As a case study, we investigated the glucose transporter type-1 deficiency syndrome (GLUT1-DS), a childhood affliction characterized by impaired glucose utilization and associated with phenotypes including seizures. Compared to neurons, astrocytes exhibited markedly higher metabolite concentration variabilities for all but a few redox species. This effect could signal a role for astrocytes in absorbing the shock of blood nutrient fluctuations. Redox balances were disrupted in GLUT1-DS with lower levels of reducing equivalent carriers NADH and ATP. The best non-glucose nutrient or pharmacotherapies for re-establishing redox normalcy involved lactate, the keto-diet (β-hydroxybutyrate), NAD and Q10 supplementation, suggesting a possible glucose sparing mechanism. GLUT1-DS seizures resulted from after-discharge neuronal firing caused by post-stimulus ATP reductions and impaired Na+/K+-ATPase, which can be rescued by restoring either normal glucose or by relatively small increases in neuronal ATP.

摘要

大脑能量供应中断与许多神经退行性疾病相关,并且由于众多相互关联的代谢途径而难以研究。我们使用神经-神经胶质-血管系统的计算模型,以神经元、星形胶质细胞和局部血液供应的形式,探索了能量供应减少对脑代谢的影响。作为一个案例研究,我们调查了1型葡萄糖转运体缺乏综合征(GLUT1-DS),这是一种儿童期疾病,其特征是葡萄糖利用受损,并伴有癫痫发作等表型。与神经元相比,除了少数氧化还原物质外,星形胶质细胞对所有代谢物的浓度变异性都明显更高。这种效应可能表明星形胶质细胞在吸收血液营养波动冲击方面发挥作用。在GLUT1-DS中,氧化还原平衡被打破,还原当量载体NADH和ATP水平降低。恢复氧化还原正常的最佳非葡萄糖营养或药物疗法包括乳酸、生酮饮食(β-羟基丁酸)、NAD和Q10补充,这表明可能存在一种葡萄糖节约机制。GLUT1-DS癫痫发作是由刺激后ATP减少和Na+/K+-ATP酶受损导致的放电后神经元放电引起的,通过恢复正常葡萄糖或相对小幅增加神经元ATP可以挽救这种情况。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/22f9/12002639/4ed9ea0c3032/pcbi.1012959.g005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/22f9/12002639/7fc70fa3170a/pcbi.1012959.g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/22f9/12002639/01fbb7e0fe29/pcbi.1012959.g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/22f9/12002639/ba61c630f54c/pcbi.1012959.g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/22f9/12002639/bd3a412819ab/pcbi.1012959.g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/22f9/12002639/4ed9ea0c3032/pcbi.1012959.g005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/22f9/12002639/7fc70fa3170a/pcbi.1012959.g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/22f9/12002639/01fbb7e0fe29/pcbi.1012959.g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/22f9/12002639/ba61c630f54c/pcbi.1012959.g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/22f9/12002639/bd3a412819ab/pcbi.1012959.g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/22f9/12002639/4ed9ea0c3032/pcbi.1012959.g005.jpg

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本文引用的文献

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Mechanistic multiscale modelling of energy metabolism in human astrocytes reveals the impact of morphology changes in Alzheimer's Disease.人类星形胶质细胞能量代谢的机制多尺度建模揭示了阿尔茨海默病中形态变化的影响。
PLoS Comput Biol. 2023 Sep 20;19(9):e1011464. doi: 10.1371/journal.pcbi.1011464. eCollection 2023 Sep.
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Combination of triheptanoin with the ketogenic diet in Glucose transporter type 1 deficiency (G1D).三庚酸甘油酯联合生酮饮食治疗葡萄糖转运蛋白 1 缺乏症(G1D)。
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Uncommon use of intermittent glucose administration for infrequent non-epileptic paroxysmal events in GLUT1-DS.
间歇性葡萄糖给药在GLUT1缺陷综合征(GLUT1-DS)中用于罕见的非癫痫性阵发性事件的不常见应用。
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Correlation between blood glucose and cerebrospinal fluid glucose levels in patients with differences in glucose metabolism.糖代谢存在差异的患者血糖与脑脊液葡萄糖水平之间的相关性
Front Neurol. 2023 Apr 27;14:1103026. doi: 10.3389/fneur.2023.1103026. eCollection 2023.
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Neurons require glucose uptake and glycolysis in vivo.神经元在体内需要葡萄糖摄取和糖酵解。
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