Expression of MeCP2 in olfactory receptor neurons is developmentally regulated and occurs before synaptogenesis
Abstract
Rett syndrome, a neurodevelopmental disorder hypothesized to be due to defective neuronal maturation, is a result of mutations in the mecp2 gene encoding the transcriptional repressor methyl-CpG binding protein (MeCP2). We utilized the olfactory system, which displays postnatal neurogenesis, as a model to investigate MeCP2 expression during development and after injury. MeCP2 expression increased postnatally, localizing to mature olfactory receptor neurons (ORNs) and sustentacular supporting cells. The timing of MeCP2 expression was defined by using detergent ablation (to remove the ORNs) and unilateral olfactory bulbectomy (to remove the ORN target), both of which increase neurogenesis. MeCP2 expression in the ORNs reached prelesioning levels as cells matured after ablation, whereas expression was not completely restored after bulbectomy, in which functional synaptogenesis cannot occur. Thus, MeCP2 expression correlates with the maturational state of ORNs, and precedes synaptogenesis. Identifying the time window of MeCP2 expression should help further clarify the biological defects in Rett syndrome.
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In the central nervous system, MeCP2 is expressed at low levels prenatally, but progressively increases during neuronal maturation and synaptogenesis, reaching its peak in mature, post-migratory neurons.
“In the central nervous system (CNS), MeCP2 is expressed at low levels prenatally, but progressively increases during neuronal maturation and synaptogenesis reaching its peak in mature, post-migratory neurons, suggesting a role for MeCP2 in maintaining neuronal maturation, activity and plasticity [54–57].”
From Rett syndrome: a neurological disorder with metabolic components (2018) · cites this paper as [54]
MeCP2 has a role in maintaining neuronal maturation, activity and plasticity.
“In the central nervous system (CNS), MeCP2 is expressed at low levels prenatally, but progressively increases during neuronal maturation and synaptogenesis reaching its peak in mature, post-migratory neurons, suggesting a role for MeCP2 in maintaining neuronal maturation, activity and plasticity [54–57].”
From Rett syndrome: a neurological disorder with metabolic components (2018) · cites this paper as [54]
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- Authors
- Cohen, D. R. S., Matarazzo, V., Palmer, A. M., Tu, Y., Jeon, O. K.-H., Pevsner, J., & Ronnett, G. V.
- Affiliations
- Johns Hopkins University (US)
- Kennedy Krieger Institute (US)
- Corresponding author
- Ronnett, Gabriele V
- ORCID
- Pevsner, Jonathan
- Venue
- Molecular and Cellular Neuroscience
- Publisher
- Elsevier BV
- Published
- 2003-04
- Volume
- 22
- Issue
- 4
- Pages
- 417-429
- ISSN
- 1044-7431 (ISSN-L)
- OpenAlex
- W2165372186
- Cited by
- 119
- References
- 0
- Citations by year
- 2025: 4 · 2024: 4 · 2023: 1 · 2022: 4 · 2021: 2 · 2020: 4 · 2019: 4 · 2018: 4 · 2017: 1 · 2016: 4 · 2015: 3 · 2014: 8 · 2013: 7 · 2012: 8
- FWCI
- 5.81
- Type
- journal article
- Language
- en
- Open access
- closed
- Locations
- Molecular and Cellular Neuroscience (publishedVersion)
- PubMed (publishedVersion)
- Funders
- National Institutes of Health
- National Institute on Deafness and Other Communication Disorders
- National Institute of Neurological Disorders and Stroke
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
- Life Sciences › Neuroscience › Developmental Neuroscience › Neurogenesis and neuroplasticity mechanisms
- Life Sciences › Neuroscience › Sensory Systems › Olfactory and Sensory Function Studies
- Keywords
- MeCP2, methyl-CpG binding proteins, Rett syndrome, olfactory sensory neurons, olfactory bulb, synaptogenesis, neuronal maturation, postnatal neurogenesis, olfactory bulbectomy, sustentacular cells, transcriptional repressor
- MeSH terms
- Animals, Cell Communication, Cell Differentiation, Chromosomal Proteins, Non-Histone, Cues, DNA-Binding Proteins, Female, Genes, Regulator, Immunohistochemistry, Male, Mice, Inbred BALB C, Mutation, Neuroglia, Olfactory Bulb, Olfactory Mucosa, Repressor Proteins, Synapses, Rett Syndrome, Rats, Sprague-Dawley, Olfactory Receptor Neurons, Gene Expression Regulation, Developmental, Neural Cell Adhesion Molecules, Growth Cones, Models, Animal, Mice, Rats, Methyl-CpG-Binding Protein 2
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