ArticlePLoS genetics2022
Single cell transcriptomics reveals dysregulated cellular and molecular networks in a fragile X syndrome model.
Article in PLoS genetics, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 16 papers.
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Who cites it
16 citing papers in PubMed, 34 citations in OpenAlex.
- Article
- Suppression of astrocyte BMP signaling improves molecular signatures and functional deficits in a fragile X syndrome mouse model.Nature communications · 2026Article
- Article
- Reprogramming of neuronal genome function and phenotype by astrocytes.bioRxiv : the preprint server for biology · 2026Article
- GABAergic neurons exhibit subtype-specific changes in the developing somatosensory cortex of a rat model of Fragile X Syndrome.Frontiers in neuroscience · 2026Article
- Mapping human brain cell type origin and diseases through single-cell transcriptomics.Translational psychiatry · 2025Review
- Translational modulator ISRIB alleviates synaptic and behavioral phenotypes in Fragile X syndrome.iScience · 2024Article
- Cochlear Nucleus Transcriptome of a Fragile X Mouse Model Reveals Candidate Genes for Hyperacusis.The Laryngoscope · 2024Article
- Glial dysregulation in the human brain in fragile X-associated tremor/ataxia syndrome.Proceedings of the National Academy of Sciences of the United States of America · 2023Article
- Fragile X mental retardation protein coordinates neuron-to-glia communication for clearance of developmentally transient brain neurons.Proceedings of the National Academy of Sciences of the United States of America · 2023Article
- Impaired synaptic incorporation of AMPA receptors in a mouse model of fragile X syndrome.Frontiers in molecular neuroscience · 2023Article
- CPEB3-dependent increase in GluA2 subunits impairs excitatory transmission onto inhibitory interneurons in a mouse model of fragile X.Cell reports · 2022Article
- Reversal of behavioural phenotype by the cannabinoid-like compound VSN16R in fragile X syndrome mice.Brain : a journal of neurology · 2022Article
- Transmembrane Protein Ttyh1 Maintains the Quiescence of Neural Stem Cells Through CaFrontiers in cell and developmental biology · 2021Article
- FMRP links optimal codons to mRNA stability in neurons.Proceedings of the National Academy of Sciences of the United States of America · 2020Article
- High-Resolution Mapping of Multiway Enhancer-Promoter Interactions Regulating Pathogen Detection.Molecular cell · 2020Article
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Authors and funding
3 authors at 1 institution in 1 country.
Funding
Abstract
Despite advances in understanding the pathophysiology of Fragile X syndrome (FXS), its molecular basis is still poorly understood. Whole brain tissue expression profiles have proved surprisingly uninformative, therefore we applied single cell RNA sequencing to profile an FMRP deficient mouse model with higher resolution. We found that the absence of FMRP results in highly cell type specific gene expression changes that are strongest among specific neuronal types, where FMRP-bound mRNAs were prominently downregulated. Metabolic pathways including translation and respiration are significantly upregulated across most cell types with the notable exception of excitatory neurons. These effects point to a potential difference in the activity of mTOR pathways, and together with other dysregulated pathways, suggest an excitatory-inhibitory imbalance in the Fmr1-knock out cortex that is exacerbated by astrocytes. Our data demonstrate that FMRP loss affects abundance of key cellular communication genes that potentially affect neuronal synapses and provide a resource for interrogating the biological basis of this disorder.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the Socratic graph.