ArticleJCI insight2021
An early endothelial cell-specific requirement for Glut1 is revealed in Glut1 deficiency syndrome model mice.
Article in JCI insight, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 23 papers.
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Who cites it
23 citing papers in PubMed, 36 citations in OpenAlex.
- On the Origin of the Brain Semi-Heavy Water Deuterium MR Signal Following Administration of Deuterated Metabolic Substrate: A Cautionary Tale.Magnetic resonance in medicine · 2026Article
- Glycolysis-driven cancer-associated fibroblasts shape T-Cell exclusion via the CXCL16-CXCR6 Axis: a metabolic-chemokine mechanism in tumor immunity.Journal of translational medicine · 2026Review
- A therapeutic role for a regulatory GLUT1-associated lncRNA in GLUT1-deficient mice.The Journal of clinical investigation · 2026Article
- Brain endothelial cell-derived extracellular vesicles (c-BEEVs) as a promising biomarker for brain vascular pathology and cognitive decline.Nature aging · 2026Article
- Toward a working definition of ketogenic diet resistance in GLUT1 deficiency syndrome.Epileptic disorders : international epilepsy journal with videotape · 2025Review
- Methyl-beta-cyclodextrin and myriocin alleviate blood-brain barrier impairment in septic rats.Histochemistry and cell biology · 2025Article
- Transgenic expression of the human Glucose Transporter1 (GLUT1) gene locus reduces disease burden in Glut1 deficiency syndrome model mice.Neurobiology of disease · 2025Article
- Article
- Harnessing BDNF Signaling to Promote Resilience in Aging.Aging and disease · 2024Review
- Lower GLUT1 and unchanged MCT1 in Alzheimer's disease cerebrovasculature.Journal of cerebral blood flow and metabolism : official journal of the International Society of Cerebral Blood Flow and Metabolism · 2024Article
- Gain-of-function mutations of TRPV4 acting in endothelial cells drive blood-CNS barrier breakdown and motor neuron degeneration in mice.Science translational medicine · 2024Article
- Brain microvascular endothelial cell metabolism and its ties to barrier function.Vitamins and hormones · 2024Article
- Epileptiform activity in brain organoids derived from patient with Glucose Transporter 1 Deficiency Syndrome.Frontiers in neuroscience · 2024Article
- Systemic Reduction of Glut1 Normalizes Retinal Dysfunction, Inflammation, and Oxidative Stress in the Retina of Spontaneous Type 2 Diabetic Mice.The American journal of pathology · 2023Article
- Muscle-Specific Ablation of Glucose Transporter 1 (GLUT1) Does Not Impair Basal or Overload-Stimulated Skeletal Muscle Glucose Uptake.Biomolecules · 2022Article
- Single unit analysis and wide-field imaging reveal alterations in excitatory and inhibitory neurons in glioma.Brain : a journal of neurology · 2022Article
- Advances in brain barriers and brain fluids research in 2021: great progress in a time of adversity.Fluids and barriers of the CNS · 2022Article
- One Molecule for Mental Nourishment and More: Glucose Transporter Type 1-Biology and Deficiency Syndrome.Biomedicines · 2022Review
- Generation of Cortical Brain Organoid with Vascularization by Assembling with Vascular Spheroid.International journal of stem cells · 2022Article
- Metabolic Transporters in the Peripheral Nerve-What, Where, and Why?Neurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics · 2021Review
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Authors and funding
15 authors at 5 institutions in 2 countries.
Funding
Abstract
Paucity of the glucose transporter-1 (Glut1) protein resulting from haploinsufficiency of the SLC2A1 gene arrests cerebral angiogenesis and disrupts brain function to cause Glut1 deficiency syndrome (Glut1 DS). Restoring Glut1 to Glut1 DS model mice prevents disease, but the precise cellular sites of action of the transporter, its temporal requirements, and the mechanisms linking scarcity of the protein to brain cell dysfunction remain poorly understood. Here, we show that Glut1 functions in a cell-autonomous manner in the cerebral microvasculature to affect endothelial tip cells and, thus, brain angiogenesis. Moreover, brain endothelial cell-specific Glut1 depletion not only triggers a severe neuroinflammatory response in the Glut1 DS brain, but also reduces levels of brain-derived neurotrophic factor (BDNF) and causes overt disease. Reduced BDNF correlated with fewer neurons in the Glut1 DS brain. Controlled depletion of the protein demonstrated that brain pathology and disease severity was greatest when Glut1 scarcity was induced neonatally, during brain angiogenesis. Reducing Glut1 at later stages had mild or little effect. Our results suggest that targeting brain endothelial cells during early development is important to ensure proper brain angiogenesis, prevent neuroinflammation, maintain BDNF levels, and preserve neuron numbers. This requirement will be essential for any disease-modifying therapeutic strategy for Glut1 DS.
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