ArticleHuman molecular genetics2014
Genome-wide alteration of 5-hydroxymethylcytosine in a mouse model of fragile X-associated tremor/ataxia syndrome.
Article in Human molecular genetics, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 39 papers.
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Who cites it
39 citing papers in PubMed, 57 citations in OpenAlex.
- Integrative transcriptome-wide association analyses reveal PRKCG-linked GABAergic dysfunction in Fragile X-associated tremor/ataxia syndrome.Nature communications · 2026Article
- Molecular Pathogenesis and Peripheral Monitoring of Adult Fragile X-Associated Syndromes.International journal of molecular sciences · 2021Review
- Ten-eleven translocation protein 1 modulates medulloblastoma progression.Genome biology · 2021Article
- Proteins in DNA methylation and their role in neural stem cell proliferation and differentiation.Cell regeneration (London, England) · 2021Review
- DNA methylation methods: Global DNA methylation and methylomic analyses.Methods (San Diego, Calif.) · 2021Review
- Tet1 Regulates Astrocyte Development and Cognition of Mice Through Modulating GluA1.Frontiers in cell and developmental biology · 2021Article
- Severe Acute Respiratory Syndrome Coronavirus 2 (SARS-CoV-2) Infection: Triggering a Lethal Fight to Keep Control of the Ten-Eleven Translocase (TET)-Associated DNA Demethylation?Pathogens (Basel, Switzerland) · 2020Review
- Age-related DNA hydroxymethylation is enriched for gene expression and immune system processes in human peripheral blood.Epigenetics · 2020Article
- Diverse and dynamic DNA modifications in brain and diseases.Human molecular genetics · 2019Review
- Decreased global DNA hydroxymethylation in neural tube defects: Association with polycyclic aromatic hydrocarbons.Epigenetics · 2019Article
- Genome-Wide Alteration of 5-Hydroxymethylcytosine in Hypoxic-Ischemic Neonatal Rat Model of Cerebral Palsy.Frontiers in molecular neuroscience · 2019Article
- Tuina Massage Improves Cognitive Functions of Hypoxic-Ischemic Neonatal Rats by Regulating Genome-Wide DNA Hydroxymethylation Levels.Evidence-based complementary and alternative medicine : eCAM · 2019Article
- Active NMolecular cell · 2018Article
- Differentially Methylated Genes in Saliva are linked to Childhood Stress.Scientific reports · 2018Article
- The Dynamic DNA Demethylation during Postnatal Neuronal Development and Neural Stem Cell Differentiation.Stem cells international · 2018Article
- Ten-eleven translocation 2 interacts with forkhead box O3 and regulates adult neurogenesis.Nature communications · 2017Article
- Article
- Fragile X-Associated Tremor/Ataxia Syndrome: From Molecular Pathogenesis to Development of Therapeutics.Frontiers in cellular neuroscience · 2017Review
- New Insights into 5hmC DNA Modification: Generation, Distribution and Function.Frontiers in genetics · 2017Review
- Brain structure and intragenic DNA methylation are correlated, and predict executive dysfunction in fragile X premutation females.Translational psychiatry · 2016Article
Corrections and comments
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Authors and funding
8 authors at 3 institutions in 1 country.
Funding
Abstract
Fragile X-associated tremor/ataxia syndrome (FXTAS) is a late-onset neurodegenerative disorder in which patients carry premutation alleles of 55-200 CGG repeats in the FMR1 gene. To date, whether alterations in epigenetic regulation modulate FXTAS has gone unexplored. 5-Hydroxymethylcytosine (5hmC) converted from 5-methylcytosine (5mC) by the ten-eleven translocation (TET) family of proteins has been found recently to play key roles in neuronal functions. Here, we undertook genome-wide profiling of cerebellar 5hmC in a FXTAS mouse model (rCGG mice) and found that rCGG mice at 16 weeks showed overall reduced 5hmC levels genome-wide compared with age-matched wild-type littermates. However, we also observed gain-of-5hmC regions in repetitive elements, as well as in cerebellum-specific enhancers, but not in general enhancers. Genomic annotation and motif prediction of wild-type- and rCGG-specific differential 5-hydroxymethylated regions (DhMRs) revealed their high correlation with genes and transcription factors that are important in neuronal developmental and functional pathways. DhMR-associated genes partially overlapped with genes that were differentially associated with ribosomes in CGG mice identified by bacTRAP ribosomal profiling. Taken together, our data strongly indicate a functional role for 5hmC-mediated epigenetic modulation in the etiology of FXTAS, possibly through the regulation of transcription.
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