Suppr超能文献

经验依赖性视顶盖突触重塑在 MeCP2 缺陷型小鼠中异常。

Experience-dependent retinogeniculate synapse remodeling is abnormal in MeCP2-deficient mice.

机构信息

Department of Neurology, F.M. Kirby Neurobiology Center, Children's Hospital, Boston, Harvard Medical School, 300 Longwood Avenue, Boston, MA 02115, USA.

出版信息

Neuron. 2011 Apr 14;70(1):35-42. doi: 10.1016/j.neuron.2011.03.001.

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.

摘要

MECP2 基因突变是导致神经发育障碍雷特综合征(RTT)的原因。RTT 的一个显著特征是相对正常的发育,然后是症状的后期发作。越来越多的证据表明突触功能障碍是其病因,但这些异常如何解释 RTT 的临床表现尚不清楚。在这里,我们研究了具有不同发育阶段的电路中的 Mecp2 缺陷型小鼠中的突触成熟:视网膜-视放射突触。我们发现,在出生后第 9 天至 21 天之间,突变体中的突触发育与野生型同窝仔相似,这表明突触形成、消除和强化的初始阶段不受 MeCP2 缺失的显著影响。然而,在随后的突触重塑的经验依赖性阶段,由于中继神经元的视网膜神经支配增加,而视网膜输入未能进一步增强,因此突变体中的电路变得异常。此外,视觉剥夺后的突触可塑性在突变体中被破坏。这些结果表明 Mecp2 在突触回路的经验依赖性细化中起着关键作用。

相似文献

1
Experience-dependent retinogeniculate synapse remodeling is abnormal in MeCP2-deficient mice.
Neuron. 2011 Apr 14;70(1):35-42. doi: 10.1016/j.neuron.2011.03.001.
2
Synaptic maturation at cortical projections to the lateral amygdala in a mouse model of Rett syndrome.
PLoS One. 2010 Jul 2;5(7):e11399. doi: 10.1371/journal.pone.0011399.
3
Activity-induced MeCP2 phosphorylation regulates retinogeniculate synapse refinement.
Proc Natl Acad Sci U S A. 2023 Oct 31;120(44):e2310344120. doi: 10.1073/pnas.2310344120. Epub 2023 Oct 23.
4
Distinct roles for spontaneous and visual activity in remodeling of the retinogeniculate synapse.
Neuron. 2006 Oct 19;52(2):281-91. doi: 10.1016/j.neuron.2006.07.007.
5
Learning and memory and synaptic plasticity are impaired in a mouse model of Rett syndrome.
J Neurosci. 2006 Jan 4;26(1):319-27. doi: 10.1523/JNEUROSCI.2623-05.2006.
7
Perineuronal net degradation rescues CA2 plasticity in a mouse model of Rett syndrome.
J Clin Invest. 2021 Aug 16;131(16). doi: 10.1172/JCI137221.
8
Early environmental enrichment moderates the behavioral and synaptic phenotype of MeCP2 null mice.
Biol Psychiatry. 2010 Apr 1;67(7):657-65. doi: 10.1016/j.biopsych.2009.12.022. Epub 2010 Feb 20.
9
Time-dependent modulation of GABA(A)-ergic synaptic transmission by allopregnanolone in locus coeruleus neurons of Mecp2-null mice.
Am J Physiol Cell Physiol. 2013 Dec 1;305(11):C1151-60. doi: 10.1152/ajpcell.00195.2013. Epub 2013 Sep 25.

引用本文的文献

1
Microglia: Mediators of experience-driven corrective neuroplasticity.
IBRO Neurosci Rep. 2025 Jun 5;19:91-100. doi: 10.1016/j.ibneur.2025.05.013. eCollection 2025 Dec.
2
Reciprocal interaction between cortical SST and PV interneurons in top-down regulation of retinothalamic refinement.
Proc Natl Acad Sci U S A. 2025 Jun 24;122(25):e2504224122. doi: 10.1073/pnas.2504224122. Epub 2025 Jun 18.
3
A novel developmental critical period of orexinergic signaling in the primary visual thalamus.
iScience. 2024 Jun 24;27(7):110352. doi: 10.1016/j.isci.2024.110352. eCollection 2024 Jul 19.
4
Activity-induced MeCP2 phosphorylation regulates retinogeniculate synapse refinement.
Proc Natl Acad Sci U S A. 2023 Oct 31;120(44):e2310344120. doi: 10.1073/pnas.2310344120. Epub 2023 Oct 23.
6
Activity-Dependent Synapse Refinement: From Mechanisms to Molecules.
Neuroscientist. 2024 Dec;30(6):673-689. doi: 10.1177/10738584231170167. Epub 2023 May 4.
7
Wild-type MECP2 expression coincides with age-dependent sensory phenotypes in a female mouse model for Rett syndrome.
J Neurosci Res. 2023 Aug;101(8):1236-1258. doi: 10.1002/jnr.25190. Epub 2023 Apr 7.
8
The molecular signals that regulate activity-dependent synapse refinement in the brain.
Curr Opin Neurobiol. 2023 Apr;79:102692. doi: 10.1016/j.conb.2023.102692. Epub 2023 Feb 17.
10
Increased Reliability of Visually-Evoked Activity in Area V1 of the MECP2-Duplication Mouse Model of Autism.
J Neurosci. 2022 Aug 17;42(33):6469-6482. doi: 10.1523/JNEUROSCI.0654-22.2022. Epub 2022 Jul 13.

本文引用的文献

1
2
Genomic imprinting of experience-dependent cortical plasticity by the ubiquitin ligase gene Ube3a.
Proc Natl Acad Sci U S A. 2010 Mar 23;107(12):5611-6. doi: 10.1073/pnas.1001281107. Epub 2010 Mar 8.
3
MeCP2 is required for normal development of GABAergic circuits in the thalamus.
J Neurophysiol. 2010 May;103(5):2470-81. doi: 10.1152/jn.00601.2009. Epub 2010 Mar 3.
4
Neuronal MeCP2 is expressed at near histone-octamer levels and globally alters the chromatin state.
Mol Cell. 2010 Feb 26;37(4):457-68. doi: 10.1016/j.molcel.2010.01.030.
5
Critical period plasticity is disrupted in the barrel cortex of FMR1 knockout mice.
Neuron. 2010 Feb 11;65(3):385-98. doi: 10.1016/j.neuron.2010.01.024.
7
LTD and LTP at the developing retinogeniculate synapse.
J Neurophysiol. 2009 Dec;102(6):3082-90. doi: 10.1152/jn.90618.2008. Epub 2009 Sep 23.
9
Ube3a is required for experience-dependent maturation of the neocortex.
Nat Neurosci. 2009 Jun;12(6):777-83. doi: 10.1038/nn.2327. Epub 2009 May 10.
10
Phosphorylation of MeCP2 at Serine 80 regulates its chromatin association and neurological function.
Proc Natl Acad Sci U S A. 2009 Mar 24;106(12):4882-7. doi: 10.1073/pnas.0811648106. Epub 2009 Feb 18.

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验