Cited by 103Genetics and Neurodevelopmental DisordersNeuroscience and Neuropharmacology ResearchRetinal Development and Disorders

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Experience-Dependent Retinogeniculate Synapse Remodeling Is Abnormal in MeCP2-Deficient Mice

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Noutel, Joao, Hong, Y. Kate, Leu, Byunghee, Kang, Erin, Chen, Chinfei·2011·Neuron

DOI 10.1016/j.neuron.2011.03.001PMC PMC3082316

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Abstract

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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Authors: Noutel, J., Hong, Y. K., Leu, B., Kang, E., & Chen, C.
Affiliations: Boston Children's Hospital (US)
Corresponding author: Chen, Chinfei
ORCID: Hong, Y. Kate
Kang, Erin
Chen, Chinfei
Venue: Neuron
Publisher: Elsevier BV
Published: 2011-04
Volume: 70
Issue: 1
Pages: 35-42
ISSN: 0896-6273 (ISSN-L)
DOI: 10.1016/j.neuron.2011.03.001
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: www.elsevier.com/tdm/userlicense/1.0/ (tdm)
www.elsevier.com/open-access/userlicense/1.0/ (vor, 838 d)
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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Experience-Dependent Retinogeniculate Synapse Remodeling Is Abnormal in MeCP2-Deficient Mice

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