Wdfy3 regulates glycophagy, mitophagy, and synaptic plasticity
| العنوان: | Wdfy3 regulates glycophagy, mitophagy, and synaptic plasticity |
|---|---|
| المؤلفون: | Nathaniel Satriya, Eleonora Napoli, Konstantinos Zarbalis, Alexios A Panoutsopoulos, Kira Sterling, Cecilia R Giulivi, P.E. Kysar, Denise M. Imai, Bradley Shibata, David N. Ruskin |
| المصدر: | J Cereb Blood Flow Metab Journal of cerebral blood flow and metabolism : official journal of the International Society of Cerebral Blood Flow and Metabolism, vol 41, iss 12 |
| بيانات النشر: | SAGE Publications, 2021. |
| سنة النشر: | 2021 |
| مصطلحات موضوعية: | Scaffold protein, Glycogenolysis, brain, Autism, Intellectual and Developmental Disabilities (IDD), 1.1 Normal biological development and functioning, Clinical Sciences, Autophagy-Related Proteins, Mice, Transgenic, Haploinsufficiency, Mitochondrion, Cardiorespiratory Medicine and Haematology, Transgenic, 03 medical and health sciences, chemistry.chemical_compound, Mice, 0302 clinical medicine, Mitophagy, Animals, 030304 developmental biology, Adaptor Proteins, Signal Transducing, 0303 health sciences, Neurology & Neurosurgery, Neuronal Plasticity, Glycogen, electron microscopy, Glycophagy, Chemistry, Autophagy, Signal Transducing, Neurosciences, Gluconeogenesis, Adaptor Proteins, Brain, Original Articles, Cell biology, Brain Disorders, Mitochondria, Mental Health, Neurology, Synaptic plasticity, Neurological, synapses, Neurology (clinical), Cardiology and Cardiovascular Medicine, 030217 neurology & neurosurgery |
| الوصف: | Autophagy is essential to cell function, as it enables the recycling of intracellular constituents during starvation and in addition functions as a quality control mechanism by eliminating spent organelles and proteins that could cause cellular damage if not properly removed. Recently, we reported on Wdfy3’s role in mitophagy, a clinically relevant macroautophagic scaffold protein that is linked to intellectual disability, neurodevelopmental delay, and autism spectrum disorder. In this study, we confirm our previous report that Wdfy3 haploinsufficiency in mice results in decreased mitophagy with accumulation of mitochondria with altered morphology, but expanding on that observation, we also note decreased mitochondrial localization at synaptic terminals and decreased synaptic density, which may contribute to altered synaptic plasticity. These changes are accompanied by defective elimination of glycogen particles and a shift to increased glycogen synthesis over glycogenolysis and glycophagy. This imbalance leads to an age-dependent higher incidence of brain glycogen deposits with cerebellar hypoplasia. Our results support and further extend Wdfy3’s role in modulating both brain bioenergetics and synaptic plasticity by including glycogen as a target of macroautophagic degradation. |
| اللغة: | English |
| URL الوصول: | https://explore.openaire.eu/search/publication?articleId=doi_dedup___::5d50ee5f28f8ab6ab09d229c73f48d35 https://europepmc.org/articles/PMC8669292/ |
| Rights: | OPEN |
| رقم الانضمام: | edsair.doi.dedup.....5d50ee5f28f8ab6ab09d229c73f48d35 |
| قاعدة البيانات: | OpenAIRE |
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