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体外多谱(13)C NMR 检测成年小鼠脑丙酮酸脱氢酶缺乏症的皮质代谢。

Cortical metabolism in pyruvate dehydrogenase deficiency revealed by ex vivo multiplet (13)C NMR of the adult mouse brain.

机构信息

Department of Neurology and Neurotherapeutics, The University of Texas Southwestern Medical Center, Dallas, TX 75390, USA.

出版信息

Neurochem Int. 2012 Dec;61(7):1036-43. doi: 10.1016/j.neuint.2012.07.020. Epub 2012 Aug 3.

Abstract

The pyruvate dehydrogenase complex (PDC), required for complete glucose oxidation, is essential for brain development. Although PDC deficiency is associated with a severe clinical syndrome, little is known about its effects on either substrate oxidation or synthesis of key metabolites such as glutamate and glutamine. Computational simulations of brain metabolism indicated that a 25% reduction in flux through PDC and a corresponding increase in flux from an alternative source of acetyl-CoA would substantially alter the (13)C NMR spectrum obtained from brain tissue. Therefore, we evaluated metabolism of [1,6-(13)C(2)]glucose (oxidized by both neurons and glia) and [1,2-(13)C(2)]acetate (an energy source that bypasses PDC) in the cerebral cortex of adult mice mildly and selectively deficient in brain PDC activity, a viable model that recapitulates the human disorder. Intravenous infusions were performed in conscious mice and extracts of brain tissue were studied by (13)C NMR. We hypothesized that mice deficient in PDC must increase the proportion of energy derived from acetate metabolism in the brain. Unexpectedly, the distribution of (13)C in glutamate and glutamine, a measure of the relative flux of acetate and glucose into the citric acid cycle, was not altered. The (13)C labeling pattern in glutamate differed significantly from glutamine, indicating preferential oxidation of [1,2-(13)C]acetate relative to [1,6-(13)C]glucose by a readily discernible metabolic domain of the brain of both normal and mutant mice, presumably glia. These findings illustrate that metabolic compartmentation is preserved in the PDC-deficient cerebral cortex, probably reflecting intact neuron-glia metabolic interactions, and that a reduction in brain PDC activity sufficient to induce cerebral dysgenesis during development does not appreciably disrupt energy metabolism in the mature brain.

摘要

丙酮酸脱氢酶复合物(PDC)是完全氧化葡萄糖所必需的,对大脑发育至关重要。尽管 PDC 缺乏与严重的临床综合征有关,但对于其对底物氧化或关键代谢物(如谷氨酸和谷氨酰胺)的合成的影响知之甚少。脑代谢的计算模拟表明,PDC 通量减少 25%,同时来自替代乙酰辅酶 A 来源的通量增加,这将大大改变从脑组织获得的(13)C NMR 谱。因此,我们评估了成年小鼠大脑皮质中 [1,6-(13)C(2)]葡萄糖(由神经元和神经胶质共同氧化)和 [1,2-(13)C(2)]乙酸盐(绕过 PDC 的能量来源)的代谢,轻度和选择性缺乏脑 PDC 活性的小鼠是一种可行的模型,可重现人类疾病。在清醒的小鼠中进行静脉输注,并通过(13)C NMR 研究脑组织提取物。我们假设 PDC 缺乏的小鼠必须增加大脑中来自乙酸盐代谢的能量比例。出乎意料的是,谷氨酸和谷氨酰胺中(13)C 的分布(衡量乙酸盐和葡萄糖进入柠檬酸循环的相对通量)没有改变。谷氨酸中(13)C 的标记模式与谷氨酰胺明显不同,表明正常和突变小鼠大脑中一个易于识别的代谢区域优先氧化 [1,2-(13)C]乙酸盐相对于 [1,6-(13)C]葡萄糖,可能是神经胶质。这些发现表明,在 PDC 缺乏的大脑皮质中保持了代谢分隔,可能反映了神经元-神经胶质代谢相互作用的完整性,并且在发育过程中足以诱导大脑发育不良的脑 PDC 活性降低不会明显破坏成熟大脑中的能量代谢。

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