Structural and Lipidomic Alterations of Striatal Myelin in 16p11.2 Deletion Mouse Model of Autism Spectrum Disorder
Autor: | Jun Ju, Xiuyan Yang, Jian Jiang, Dilong Wang, Yumeng Zhang, Xiaofeng Zhao, Xiaoyi Fang, Huanquan Liao, Lei Zheng, Shupeng Li, Sheng-Tao Hou, Liyang Liang, Yihang Pan, Huiliang Li, Ningning Li |
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Jazyk: | angličtina |
Rok vydání: | 2021 |
Předmět: |
Ceramide
striatum autism spectrum disorder 16p11.2 deletion Neurosciences. Biological psychiatry. Neuropsychiatry Striatum Biology Cellular and Molecular Neuroscience chemistry.chemical_compound Myelin mental disorders lipid metabolism medicine Original Research Ceramide synthase 2 Wild type Lipid metabolism medicine.disease Cell biology myelin medicine.anatomical_structure chemistry nervous system Autism spectrum disorder Sphingomyelin Neuroscience RC321-571 |
Zdroj: | Frontiers in Cellular Neuroscience, Vol 15 (2021) Frontiers in Cellular Neuroscience |
ISSN: | 1662-5102 |
Popis: | Myelin abnormalities have been observed in autism spectrum disorder (ASD). In this study, we seek to discover myelin-related changes in the striatum, a key brain region responsible for core ASD features, using the 16p11.2 deletion (16p11.2±) mouse model of ASD. We found downregulated expression of multiple myelin genes and decreased myelin thickness in the striatum of 16p11.2± mice versus wild type controls. Moreover, given that myelin is the main reservoir of brain lipids and that increasing evidence has linked dysregulation of lipid metabolism to ASD, we performed lipidomic analysis and discovered decreased levels of certain species of sphingomyelin, hexosyl ceramide and their common precursor, ceramide, in 16p11.2± striatum, all of which are major myelin components. We further identified lack of ceramide synthase 2 as the possible reason behind the decrease in these lipid species. Taken together, our data suggest a role for myelin and myelin lipids in ASD development. |
Databáze: | OpenAIRE |
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