The DOI matches its Crossref record; 40 of the 40 references it cites were found in a registry or an index.
Experience-Dependent Retinogeniculate Synapse Remodeling Is Abnormal in MeCP2-Deficient Mice
Open accessAbstract
Mutations in MECP2 underlie the neurodevelopmental disorder Rett syndrome (RTT). One hallmark of RTT is relatively normal development followed by a later onset of symptoms. Growing evidence suggests an etiology of disrupted synaptic function, yet it is unclear how these abnormalities explain the clinical presentation of RTT. Here we investigate synapse maturation in Mecp2-deficient mice at a circuit with distinct developmental phases: the retinogeniculate synapse. We find that synapse development in mutants is comparable to that of wild-type littermates between postnatal days 9 and 21, indicating that initial phases of synapse formation, elimination, and strengthening are not significantly affected by MeCP2 absence. However, during the subsequent experience-dependent phase of synapse remodeling, the circuit becomes abnormal in mutants as retinal innervation of relay neurons increases and retinal inputs fail to strengthen further. Moreover, synaptic plasticity in response to visual deprivation is disrupted in mutants. These results suggest a crucial role for Mecp2 in experience-dependent refinement of synaptic circuits.
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“MECP2 has recently been shown to be required for experience-dependent synaptic remodelling of the retinogeniculate synapse85.”
From Activity-dependent neuronal signalling and autism spectrum disorder (2013) · cites this paper as [85]
In the citing paper
“In Mecp2 knockout mice, the initial development and refinement of the retinogeniculate circuit proceeds without impairment, during a phase of development that does not depend on visual experience.”
From Activity-dependent neuronal signalling and autism spectrum disorder (2013) · cites this paper as [85]
In the citing paper
“during a late stage of development when visual experience is required for modulation and maintenance of the retinogeniculate circuit, the synapses that form on the neurons within this circuit are abnormal in Mecp2 knockout mice, resulting in impaired synaptic plasticity on visual deprivation85.”
From Activity-dependent neuronal signalling and autism spectrum disorder (2013) · cites this paper as [85]
In the citing paper
“It is of interest to note that at the retinogeniculate synapse of MeCP2 -/y mice, the initial phase of synapse formation proceeds normally (between P9 and P21) but the subsequent experiencedependent phase of synapse remodeling goes awry (58).”
From MeCP2 regulates the timing of critical period plasticity that shapes functional connectivity in primary visual cortex (2015) · cites this paper as [58]
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Rett syndrome is caused by mutations in X-linked MECP2, encoding methyl-CpG-binding protein 2
Amir, R. E., Van den Veyver, I. B., Wan, M., Tran, C. Q., Francke, U., & Zoghbi, H. Y. (1999) · Nature Genetics · doi:10.1038/13810 · Cited by 4,990
MeCP2, a Key Contributor to Neurological Disease, Activates and Represses Transcription
Chahrour, M., Jung, S. Y., Shaw, C., Zhou, X., Wong, S. T. C., Qin, J., & Zoghbi, H. Y. (2008) · Science · doi:10.1126/science.1153252 · Cited by 1,823
A mouse Mecp2-null mutation causes neurological symptoms that mimic Rett syndrome
Guy, J., Hendrich, B., Holmes, M., Martin, J. E., & Bird, A. (2001) · Nature Genetics · doi:10.1038/85899 · Cited by 1,558
Lewis, J. D., Meehan, R. R., Henzel, W. J., Maurer-Fogy, I., Jeppesen, P., Klein, F., & Bird, A. (1992) · Cell · doi:10.1016/0092-8674(92)90610-o · Cited by 1,371
Derepression of BDNF Transcription Involves Calcium-Dependent Phosphorylation of MeCP2
Chen, W. G., Chang, Q., Lin, Y., Meissner, A., West, A. E., Griffith, E. C., Jaenisch, R., & Greenberg, M. E. (2003) · Science · doi:10.1126/science.1086446 · Cited by 1,232
Dysfunction in GABA signalling mediates autism-like stereotypies and Rett syndrome phenotypes
Chao, H.-T., Chen, H., Samaco, R. C., Xue, M., Chahrour, M., Yoo, J., Neul, J. L., Gong, S., Lu, H.-C., Heintz, N., et al. (2010) · Nature · doi:10.1038/nature09582 · Cited by 1,168
Zhou, Z., Hong, E. J., Cohen, S., Zhao, W.-N., Ho, H.-Y. H., Schmidt, L., Chen, W. G., Lin, Y., Savner, E., Griffith, E. C., et al. (2006) · Neuron · doi:10.1016/j.neuron.2006.09.037 · Cited by 877
Postnatal neurodevelopmental disorders: meeting at the synapse?
Zoghbi, H. Y. (2003) · Science · doi:10.1126/science.1089071 · Cited by 691
Neuronal MeCP2 Is Expressed at Near Histone-Octamer Levels and Globally Alters the Chromatin State
Skene, P. J., Illingworth, R. S., Webb, S., Kerr, A. R. W., James, K. D., Turner, D. J., Andrews, R., & Bird, A. P. (2010) · Molecular Cell · doi:10.1016/j.molcel.2010.01.030 · Cited by 688
Dani, V. S., Chang, Q., Maffei, A., Turrigiano, G. G., Jaenisch, R., & Nelson, S. B. (2005) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.0506071102 · Cited by 639
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Lee, H.-M., Kuijer, M. B., Blanes, N. R., Clark, E. P., Aita, M., Galiano Arjona, L., Kokot, A., Sciaky, N., Simon, J. M., Bhatnagar, S., et al. (2020) · Figshare · doi:10.6084/m9.figshare.13219490
Lee, H.-M., Kuijer, M. B., Blanes, N. R., Clark, E. P., Aita, M., Galiano Arjona, L., Kokot, A., Sciaky, N., Simon, J. M., Bhatnagar, S., et al. (2020) · Figshare · doi:10.6084/m9.figshare.13219496
Tinano, F. R., Meireles, C. G., Macedo, D. B., Boris, N. P., Pereira, S. A., Magnuson, M., Abreu, A. P., Carroll, R. S., Latronico, A. C., & Kaiser, U. B. (2026) · Journal of the Endocrine Society · doi:10.1210/jendso/bvag086
Metadata
- Authors
- Noutel, J., Hong, Y. K., Leu, B., Kang, E., & Chen, C.
- Affiliations
- Boston Children's Hospital (US)
- Corresponding author
- Chen, Chinfei
- Venue
- Neuron
- Publisher
- Elsevier BV
- Published
- 2011-04
- Volume
- 70
- Issue
- 1
- Pages
- 35-42
- ISSN
- 0896-6273 (ISSN-L)
- PMC
- PMC3082316
- OpenAlex
- W2068111240
- Cited by
- 103
- References
- 40
- Citations by year
- 2026: 1 · 2025: 5 · 2024: 2 · 2023: 5 · 2022: 5 · 2021: 7 · 2020: 12 · 2019: 7 · 2018: 3 · 2017: 10 · 2016: 9 · 2015: 4 · 2014: 10 · 2013: 5 · 2012: 13
- FWCI
- 6.59
- Type
- journal article
- Language
- en
- Open access
- bronze
- doi.org
- Licence
- Locations
- Neuron (publishedVersion)
- PubMed (publishedVersion)
- Europe PMC (PubMed Central) (acceptedVersion)
- PubMed Central (acceptedVersion)
- Funders
- National Institutes of Health
- National Institute of Neurological Disorders and Stroke (R01NS070300)
- Eunice Kennedy Shriver National Institute of Child Health and Human Development (R21HD058196, P30HD018655, P01 HD-18655)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
- Life Sciences › Neuroscience › Cellular and Molecular Neuroscience › Neuroscience and Neuropharmacology Research
- Health Sciences › Medicine › Ophthalmology › Retinal Development and Disorders
- Keywords
- Rett syndrome, MeCP2, retinogeniculate synapses, synaptic remodeling, synaptic plasticity, visual deprivation, retinal innervation, relay neurons, synaptic development, neurodevelopmental disorders
- MeSH terms
- Animals, Darkness, Female, Geniculate Bodies, Mice, Inbred C57BL, Synaptic Transmission, Neuronal Plasticity, Retina, Sensory Deprivation, Synapses, Visual Perception, Rett Syndrome, Mice, Knockout, Mice, Methyl-CpG-Binding Protein 2
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