Cited by 304Genetics and Neurodevelopmental DisordersEpigenetics and DNA MethylationGenomics and Chromatin Dynamics

The DOI matches its Crossref record; 31 of the 31 references it cites were found in a registry or an index.

MeCP2 binds to non-CG methylated DNA as neurons mature, influencing transcription and the timing of onset for Rett syndrome

Open access

Chen, Lin, Chen, Kaifu, Lavery, Laura A., Baker, Steven Andrew, Shaw, Chad A., Li, Wei, Zoghbi, Huda Y.·2015·Proceedings of the National Academy of Sciences

DOI 10.1073/pnas.1505909112PMC PMC4418849

Open this paper in CoChat — free account

Abstract

Significance: Decades of research have not deciphered the mechanism by which methyl-CpG binding protein 2 (MeCP2) regulates transcription and why Rett symptoms manifest 1 to 2 y after birth. We hypothesized that the temporal dynamics of MeCP2 binding might provide an answer. We developed mice with an EGFP-tagged MeCP2 allele to identify high-resolution MeCP2 binding profiles in the adult mouse brain. Using genomic binding profiles, methylation maps, and mRNA deep-sequencing data, we found MeCP2 binds to non-CG methylation (mCH, not mCG) to regulate expression of genes altered in mouse models of MeCP2 disorders. These data and the parallel timing of mCH and MeCP2 postnatal accumulation suggest MeCP2 binds mCH as neurons mature to regulate gene expression, offering an explanation for the delayed onset of Rett.

Integrity signals

Registry

Crossref · 10.1073/pnas.1505909112

Cites

31 of 31 references verified0 citation errors · 0 not checkable

Statistics

Recomputing p-values needs the paper’s full text.

Claims the paper makes

We couldn’t retrieve this paper’s full text, so its claims and self-check weren’t run. The registry and citation checks still apply.

Claims other papers make about this one

Each is checked against this paper’s own text: does it say what the citing paper says it does?

Initial sequencing and comparative analysis of the mouse genome
Mouse Genome Sequencing Consortium (2002) · Nature · doi:10.1038/nature01262 · Cited by 7,187
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
Self-citation
DNA methylation landscapes: Provocative insights from epigenomics
Suzuki, M. M., & Bird, A. (2008) · Nature Reviews Genetics · doi:10.1038/nrg2341 · Cited by 3,039
Methylated DNA and MeCP2 recruit histone deacetylase to repress transcription
Jones, P. L., Veenstra, G. J. C., Wade, P. A., Vermaak, D., Kass, S. U., Landsberger, N., Strouboulis, J., & Wolffe, A. P. (1998) · Nature Genetics · doi:10.1038/561 · Cited by 2,791
Global epigenomic reconfiguration during mammalian brain development
Lister, R., Mukamel, E. A., Nery, J. R., Urich, M., Puddifoot, C. A., Johnson, N. D., Lucero, J., Huang, Y., Dwork, A. J., Schultz, M. D., et al. (2013) · Science · doi:10.1126/science.1237905 · Cited by 1,830
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
Self-citation
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
Purification, sequence, and cellular localization of a novel chromosomal protein that binds to Methylated DNA
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
Reversal of Neurological Defects in a Mouse Model of Rett Syndrome
Guy, J., Gan, J., Selfridge, J., Cobb, S., & Bird, A. (2007) · Science · doi:10.1126/science.1138389 · Cited by 1,230
MeCP2 binds to 5hmC enriched within active genes and accessible chromatin in the nervous system
Mellén, M., Ayata, P., Dewell, S., Kriaucionis, S., & Heintz, N. (2012) · Cell · doi:10.1016/j.cell.2012.11.022 · Cited by 943

Show all 31 references

Sorted by citations

The DNA methyltransferase family: a versatile toolkit for epigenetic regulation
Lyko, F. (2017) · Nature Reviews Genetics · doi:10.1038/nrg.2017.80 · Cited by 1,510
MOFA+: a statistical framework for comprehensive integration of multi-modal single-cell data
Argelaguet, R., Arnol, D., Bredikhin, D., Deloro, Y., Velten, B., Marioni, J. C., & Stegle, O. (2020) · Genome Biology · doi:10.1186/s13059-020-02015-1 · Cited by 1,091
Dynamic DNA methylation: In the right place at the right time
Luo, C., Hajkova, P., & Ecker, J. R. (2018) · Science · doi:10.1126/science.aat6806 · Cited by 668
Activity-Regulated Transcription: Bridging the Gap between Neural Activity and Behavior
Yap, E.-L., & Greenberg, M. E. (2018) · Neuron · doi:10.1016/j.neuron.2018.10.013 · Cited by 706
CpG and non-CpG methylation in epigenetic gene regulation and brain function
Jang, H., Shin, W., Lee, J., & Do, J. (2017) · Genes · doi:10.3390/genes8060148 · Cited by 427
DNA methylation in human diseases
Jin, Z., & Liu, Y. (2018) · Genes & Diseases · doi:10.1016/j.gendis.2018.01.002 · Cited by 422
Cerebral Organoids Recapitulate Epigenomic Signatures of the Human Fetal Brain
Luo, C., Lancaster, M. A., Castanon, R., Nery, J. R., Knoblich, J. A., & Ecker, J. R. (2016) · Cell Reports · doi:10.1016/j.celrep.2016.12.001 · Cited by 370
Epigenetic mechanisms in neurogenesis
Yao, B., Christian, K. M., He, C., Jin, P., Ming, G., & Song, H. (2016) · Nature reviews. Neuroscience · doi:10.1038/nrn.2016.70 · Cited by 406
DNA methylation atlas of the mouse brain at single-cell resolution
Liu, H., Zhou, J., Tian, W., Luo, C., Bartlett, A., Aldridge, A., Lucero, J., Osteen, J. K., Nery, J. R., Chen, H., et al. (2021) · Nature · doi:10.1038/s41586-020-03182-8 · Cited by 289
Non-CG Methylation in the Human Genome
He, Y., & Ecker, J. R. (2015) · Annual Review of Genomics and Human Genetics · doi:10.1146/annurev-genom-090413-025437 · Cited by 280
DNA methylation in mammalian development and disease
Smith, Z. D., Hetzel, S., & Meissner, A. (2024) · Nature Reviews Genetics · doi:10.1038/s41576-024-00760-8 · Cited by 310
Loss of Endometrial Plasticity in Recurrent Pregnancy Loss
Lucas, E. S., Dyer, N. P., Murakami, K., Lee, Y. H., Chan, Y., Grimaldi, G., Muter, J., Brighton, P. J., Moore, J. D., Patel, G., et al. (2015) · Stem Cells · doi:10.1002/stem.2222 · Cited by 234
Epigenetic regulation in major depression and other stress-related disorders: molecular mechanisms, clinical relevance and therapeutic potential
Yuan, M., Yang, B., Rothschild, G., Mann, J. J., Sanford, L. D., Tang, X., Huang, C., Wang, C., & Zhang, W. (2023) · Signal Transduction and Targeted Therapy · doi:10.1038/s41392-023-01519-z · Cited by 246
“64 Although the importance of non-CpG methylation in the nervous system has been demonstrated, whether it contributes to the pathophysiology of MDD and other stress-related disorders remains poorly investigated and requires further study.”
Background
MECP2 disorders: from the clinic to mice and back
Lombardi, L. M., Baker, S. A., & Zoghbi, H. Y. (2015) · Journal of Clinical Investigation · doi:10.1172/jci78167 · Cited by 221
Self-citation
Allosteric control of mammalian DNA methyltransferases – a new regulatory paradigm
Jeltsch, A., & Jurkowska, R. Z. (2016) · Nucleic Acids Research · doi:10.1093/nar/gkw723 · Cited by 219
Early-Life Gene Expression in Neurons Modulates Lasting Epigenetic States
Stroud, H., Su, S. C., Hrvatin, S., Greben, A. W., Renthal, W. R., Boxer, L. D., Nagy, M. A., Hochbaum, D. R., Kinde, B., Gabel, H. W., et al. (2017) · Cell · doi:10.1016/j.cell.2017.09.047 · Cited by 193
Mammalian DNA methyltransferases: new discoveries and open questions
Gowher, H., & Jeltsch, A. (2018) · Biochemical Society Transactions · doi:10.1042/bst20170574 · Cited by 183
Spatiotemporal DNA methylome dynamics of the developing mouse fetus
He, Y., Hariharan, M., Gorkin, D. U., Dickel, D. E., Luo, C., Castanon, R., Nery, J. R., Lee, A. Y., Zhao, Y., Huang, H., et al. (2020) · Nature · doi:10.1038/s41586-020-2119-x · Cited by 165
5-hydroxymethylcytosine accumulation in postmitotic neurons results in functional demethylation of expressed genes
Mellén, M., Ayata, P., & Heintz, N. (2017) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.1708044114 · Cited by 151
Neuropathology of suicide: Recent findings and future directions
Lutz, P.-E., Mechawar, N., & Turecki, G. (2017) · Molecular Psychiatry · doi:10.1038/mp.2017.141 · Cited by 158
Rett Syndrome: Crossing the Threshold to Clinical Translation
Katz, D. M., Bird, A., Coenraads, M., Gray, S. J., Menon, D. U., Philpot, B. D., & Tarquinio, D. C. (2016) · Trends in Neurosciences · doi:10.1016/j.tins.2015.12.008 · Cited by 150
MeCP2 recognizes cytosine methylated tri-nucleotide and di-nucleotide sequences to tune transcription in the mammalian brain
Lagger, S., Connelly, J. C., Schweikert, G., Webb, S., Selfridge, J., Ramsahoye, B. H., Yu, M., DeSousa, D., Seiser, C., He, C., et al. (2017) · PLoS Genetics · doi:10.1371/journal.pgen.1006793 · Cited by 146
Methylation across the central dogma in health and diseases: new therapeutic strategies
Liu, R., Zhao, E., Yu, H., Yuan, C., Abbas, M. N., & Cui, H. (2023) · Signal Transduction and Targeted Therapy · doi:10.1038/s41392-023-01528-y · Cited by 155
Epigenetic Etiology of Intellectual Disability
Iwase, S., Bérubé, N. G., Zhou, Z., Kasri, N. N., Battaglioli, E., Scandaglia, M., & Barco, Á. (2017) · Journal of Neuroscience · doi:10.1523/jneurosci.1840-17.2017 · Cited by 145
Single-cell DNA methylome and 3D multi-omic atlas of the adult mouse brain
Liu, H., Zeng, Q., Zhou, J., Bartlett, A., Wang, B.-A., Berube, P., Tian, W., Kenworthy, M., Altshul, J., Nery, J. R., et al. (2023) · Nature · doi:10.1038/s41586-023-06805-y · Cited by 139
Epigenetic Regulations in Neuropsychiatric Disorders
Kuehner, J. N., Bruggeman, E. C., Wen, Z., & Yao, B. (2019) · Frontiers in Genetics · doi:10.3389/fgene.2019.00268 · Cited by 144
DNA methylation in the gene body influences MeCP2-mediated gene repression
Kinde, B., Wu, D. Y., Greenberg, M. E., & Gabel, H. W. (2016) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.1618737114 · Cited by 118
S-Adenosyl Methionine and Transmethylation Pathways in Neuropsychiatric Diseases Throughout Life
Gao, J., Cahill, C. M., Huang, X., Roffman, J. L., Lamon‐Fava, S., D, M. F. M., Mischoulon, D., & Rogers, J. T. (2018) · Neurotherapeutics · doi:10.1007/s13311-017-0593-0 · Cited by 142
The Role of Epigenetic Mechanisms in the Regulation of Gene Expression in the Nervous System
Cholewa-Waclaw, J., Bird, A. P., von Schimmelmann, M. J., Schaefer, A., Yu, H., Song, H., Madabhushi, R., & Tsai, L. (2016) · Journal of Neuroscience · doi:10.1523/jneurosci.2492-16.2016 · Cited by 137
DDM1 and Lsh remodelers allow methylation of DNA wrapped in nucleosomes
Lyons, D. B., & Zilberman, D. (2017) · eLife · doi:10.7554/elife.30674 · Cited by 121
Characterization of human mosaic Rett syndrome brain tissue by single-nucleus RNA sequencing
Renthal, W. R., Boxer, L. D., Hrvatin, S., Li, E., Silberfeld, A., Nagy, M. A., Griffith, E. C., Vierbuchen, T., & Greenberg, M. E. (2018) · Nature Neuroscience · doi:10.1038/s41593-018-0270-6 · Cited by 122
Biotin tagging of MeCP2 in mice reveals contextual insights into the Rett syndrome transcriptome
Johnson, B. S., Zhao, Y.-T., Fasolino, M., Lamonica, J. M., Kim, Y. J., Georgakilas, G. K., Wood, K. H., Bu, D. D., Cui, Y., Goffin, D., et al. (2017) · Nature Medicine · doi:10.1038/nm.4406 · Cited by 120
Exercise-induced endothelial Mecp2 lactylation suppresses atherosclerosis via the Ereg/MAPK signalling pathway
Wang, Y., Chen, L., Zhang, M., Li, X., Yang, X., Huang, T., Ban, Y., Li, Y., Li, Q., Zheng, Y., et al. (2023) · Atherosclerosis · doi:10.1016/j.atherosclerosis.2023.05.009 · Cited by 127
Self-citation
MeCP2 Represses the Rate of Transcriptional Initiation of Highly Methylated Long Genes
Boxer, L. D., Renthal, W. R., Greben, A. W., Whitwam, T., Silberfeld, A., Stroud, H., Li, E., Yang, M. G., Kinde, B., Griffith, E. C., et al. (2019) · Molecular Cell · doi:10.1016/j.molcel.2019.10.032 · Cited by 115
IRF2BPL Is Associated with Neurological Phenotypes
Marcogliese, P. C., Shashi, V., Spillmann, R. C., Stong, N., Rosenfeld, J. A., Koenig, M. K., Martínez-Agosto, J. A., Herzog, M. R., Chen, A. H., Dickson, P., et al. (2018) · The American Journal of Human Genetics · doi:10.1016/j.ajhg.2018.07.006 · Cited by 115
Substantial DNA methylation differences between two major neuronal subtypes in human brain
Kozlenkov, A., Wang, M., Roussos, P., Rudchenko, S., Barbu, M., Bibikova, M. V., Klotzle, B., Dwork, A. J., Zhang, B., Hurd, Y. L., et al. (2015) · Nucleic Acids Research · doi:10.1093/nar/gkv1304 · Cited by 130
Chromatin Regulation of Neuronal Maturation and Plasticity
Gallegos, D. A., Chan, U., Chen, L., & West, A. E. (2018) · Trends in Neurosciences · doi:10.1016/j.tins.2018.02.009 · Cited by 98
Self-citation
The Interplay between DNA Methylation, Folate and Neurocognitive Development
Irwin, R. E., Pentieva, K. D., Cassidy, T., Lees‐Murdock, D. J., McLaughlin, M., Prasad, G., McNulty, H., & Walsh, C. P. (2016) · Epigenomics · doi:10.2217/epi-2016-0003 · Cited by 98
MeCP2: A Critical Regulator of Chromatin in Neurodevelopment and Adult Brain Function
Karaca, K. G., Brito, D. V. C., & Oliveira, A. M. M. (2019) · International Journal of Molecular Sciences · doi:10.3390/ijms20184577 · Cited by 103
RNA sequencing and proteomics approaches reveal novel deficits in the cortex of Mecp2-deficient mice, a model for Rett syndrome
Pacheco, N. L., Heaven, M. R., Holt, L. M., Crossman, D. K., Boggio, K. J., Shaffer, S. A., Flint, D. L., & Olsen, M. L. (2017) · Molecular Autism · doi:10.1186/s13229-017-0174-4 · Cited by 90
Dynamic DNA methylation in the brain: a new epigenetic mark for experience-dependent plasticity
Tognini, P., Napoli, D., & Pizzorusso, T. (2015) · Frontiers in Cellular Neuroscience · doi:10.3389/fncel.2015.00331 · Cited by 94
Structural basis for the ability of MBD domains to bind methyl-CG and TG sites in DNA
Liu, K., Xu, C., Lei, M., Yang, A. W. H., Loppnau, P., Hughes, T. R., & Min, J. (2018) · Journal of Biological Chemistry · doi:10.1074/jbc.ra118.001785 · Cited by 84
DNA Methylation and Adult Neurogenesis
Jobe, E. M., & Zhao, X. (2016) · Brain Plasticity · doi:10.3233/bpl-160034 · Cited by 85
Rett Syndrome and MECP2 Duplication Syndrome: Disorders of MeCP2 Dosage
Collins, B. E., & Neul, J. L. (2022) · Neuropsychiatric Disease and Treatment · doi:10.2147/ndt.s371483 · Cited by 87
Loss of function of NCOR1 and NCOR2 impairs memory through a novel GABAergic hypothalamus–CA3 projection
study, D., Zhou, W., He, Y., Rehman, A. U., Kong, Y., Hong, S., Ding, G., Yalamanchili, H. K., Wan, Y., Paul, B., et al. (2019) · Nature Neuroscience · doi:10.1038/s41593-018-0311-1 · Cited by 83
Invited Review: Epigenetics in neurodevelopment
Salinas, R. D., Connolly, D. R., & Song, H. (2020) · Neuropathology and Applied Neurobiology · doi:10.1111/nan.12608 · Cited by 90
Developmental Dynamics of Rett Syndrome
Feldman, D., Banerjee, A., & Sur, M. (2016) · Neural Plasticity · doi:10.1155/2016/6154080 · Cited by 80
MeCP2 Represses Enhancers through Chromosome Topology-Associated DNA Methylation
Clemens, A. W., Wu, D. Y., Moore, J. R., Christian, D. L., Zhao, G., & Gabel, H. W. (2019) · Molecular Cell · doi:10.1016/j.molcel.2019.10.033 · Cited by 78
DNMT3A Haploinsufficiency Results in Behavioral Deficits and Global Epigenomic Dysregulation Shared across Neurodevelopmental Disorders
Christian, D. L., Wu, D. Y., Martin, J. R., Moore, J. R., Liu, Y. R., Clemens, A. W., Nettles, S. A., Kirkland, N. M., Papouin, T., Hill, C. A., et al. (2020) · Cell Reports · doi:10.1016/j.celrep.2020.108416 · Cited by 84
Global DNA methylation remodeling during direct reprogramming of fibroblasts to neurons
Luo, C., Lee, Q. Y., Wapinski, O., Castanon, R., Nery, J. R., Mall, M., Kareta, M. S., Cullen, S. M., Goodell, M. A., Chang, H. Y., et al. (2019) · eLife · doi:10.7554/elife.40197 · Cited by 76
MeCP2 regulates gene expression through recognition of H3K27me3
Lee, W., Kim, J., Yun, J., Ohn, T., & Gong, Q. (2020) · Nature Communications · doi:10.1038/s41467-020-16907-0 · Cited by 75
The Crucial Role of DNA Methylation and MeCP2 in Neuronal Function
Fasolino, M., & Zhou, Z. (2017) · Genes · doi:10.3390/genes8050141 · Cited by 76
Placenta and fetal brain share a neurodevelopmental disorder DNA methylation profile in a mouse model of prenatal PCB exposure
Laufer, B. I., Neier, K. E., Valenzuela, A. E., Yasui, D. H., Schmidt, R. J., Lein, P. J., & LaSalle, J. M. (2022) · Cell Reports · doi:10.1016/j.celrep.2022.110442 · Cited by 73
Elevating expression of MeCP2 T158M rescues DNA binding and Rett syndrome–like phenotypes
Lamonica, J. M., Kwon, D. Y., Goffin, D., Fenik, P., Johnson, B. S., Cui, Y., Guo, H., Veasey, S. C., & Zhou, Z. (2017) · Journal of Clinical Investigation · doi:10.1172/jci90967 · Cited by 71
Rett syndrome: disruption of epigenetic control of postnatal neurological functions
Pohodich, A. E., & Zoghbi, H. Y. (2015) · Human Molecular Genetics · doi:10.1093/hmg/ddv217 · Cited by 76
Self-citation
Differential landscape of non-CpG methylation in embryonic stem cells and neurons caused by DNMT3s
Lee, J. H., Park, S., & Nakai, K. (2017) · Scientific Reports · doi:10.1038/s41598-017-11800-1 · Cited by 73
The Role of DNMT Methyltransferases and TET Dioxygenases in the Maintenance of the DNA Methylation Level
Davletgildeeva, A. T., & Kuznetsov, N. A. (2024) · Biomolecules · doi:10.3390/biom14091117 · Cited by 76
Nucleosome positioning: resources and tools online
Teif, V. B. (2015) · Briefings Bioinform. · doi:10.1093/bib/bbv086 · Cited by 66
CNS-wide Sexually Dimorphic Induction of the Major Histocompatibility Complex 1 Pathway With Aging
Mangold, C. A., Masser, D. R., Stanford, D. R., Bixler, G. V., Pisupati, A., Giles, C. B., Wren, J. D., Ford, M. M., Sonntag, W. E., & Freeman, W. M. (2016) · The Journals of Gerontology Series A · doi:10.1093/gerona/glv232 · Cited by 69
Autism genetics – an overview
Yin, J., & Schaaf, C. P. (2016) · Prenatal Diagnosis · doi:10.1002/pd.4942 · Cited by 78
Misregulation of alternative splicing in a mouse model of Rett syndrome
Li, R., Dong, Q., Yuan, X., Zeng, X., Gao, Y., Chiao, C., Li, H., Zhao, X., Keles, S., Wang, Z., et al. (2016) · PLOS Genetics · doi:10.1371/journal.pgen.1006129 · Cited by 67
Sequence features accurately predict genome-wide MeCP2 binding in vivo
Rube, H. T., Lee, W., Hejna, M., Chen, H., Yasui, D. H., Hess, J. F., LaSalle, J. M., Song, J. S., & Gong, Q. (2016) · Nature Communications · doi:10.1038/ncomms11025 · Cited by 64
Neuroepigenetic mechanisms in disease
Christopher, M. A., Kyle, S. M., & Katz, D. J. (2017) · Epigenetics & Chromatin · doi:10.1186/s13072-017-0150-4 · Cited by 69
MECP2 directly interacts with RNA polymerase II to modulate transcription in human neurons
Liu, Y., Flamier, A., Bell, G. W., Diao, A. J., Whitfield, T. W., Wang, H., Wu, Y., Schulte, F., Friesen, M., Guo, R., et al. (2024) · Neuron · doi:10.1016/j.neuron.2024.04.007 · Cited by 64
Caloric restriction mitigates age-associated hippocampal differential CG and non-CG methylation
Hadad, N., Unnikrishnan, A., Jackson, J. A., Masser, D. R., Otalora, L., Stanford, D. R., Richardson, A. G., & Freeman, W. M. (2018) · Neurobiology of Aging · doi:10.1016/j.neurobiolaging.2018.03.009 · Cited by 66
DNA methylation and de-methylation using hybrid site-targeting proteins
Lei, Y., Huang, Y., & Goodell, M. A. (2018) · Genome biology · doi:10.1186/s13059-018-1566-2 · Cited by 67
Linking genetics to epigenetics: The role of folate and folate‐related pathways in neurodevelopmental disorders
Lintas, C. (2019) · Clinical Genetics · doi:10.1111/cge.13421 · Cited by 62
Antisense oligonucleotide therapy in a humanized mouse model of MECP2 duplication syndrome
Shao, Y., Sztainberg, Y., Wang, Q., Bajikar, S. S., Trostle, A. J., Wan, Y., Jafar‐Nejad, P., Rigo, F., Liu, Z., Tang, J., et al. (2021) · Science Translational Medicine · doi:10.1126/scitranslmed.aaz7785 · Cited by 61
Self-citation
MicroRNA-29 is an essential regulator of brain maturation through regulation of CH methylation
Swahari, V., Nakamura, A., Hollville, É., Stroud, H., Simon, J. M., Ptacek, T., Beck, M. V., Flowers, C., Guo, J., Plestant, C., et al. (2021) · Cell Reports · doi:10.1016/j.celrep.2021.108946 · Cited by 64
New Insights Into the Epigenetic Regulation of Inflammatory Bowel Disease
Xu, J., Xu, H., Yang, M., Liang, Y.-J., Peng, Q., Zhang, Y., Tian, C., Wang, L., Yao, J., Nie, Y., et al. (2022) · Frontiers in Pharmacology · doi:10.3389/fphar.2022.813659 · Cited by 62
Loss of MeCP2 in the rat models regression, impaired sociability and transcriptional deficits of Rett syndrome
Veeraragavan, S., Wan, Y.-W., Connolly, D. R., Hamilton, S. M., Ward, C. S., Soriano, S., Pitcher, M. R., McGraw, C. M., Huang, S. G., Green, J. R., et al. (2016) · Human Molecular Genetics · doi:10.1093/hmg/ddw178 · Cited by 59
Enzymology of Mammalian DNA Methyltransferases
Jurkowska, R. Z., & Jeltsch, A. (2016) · Advances in experimental medicine and biology · doi:10.1007/978-3-319-43624-1_5 · Cited by 60
MeCP2 Deficiency in Neuroglia: New Progress in the Pathogenesis of Rett Syndrome
Jin, X.-R., Chen, X.-S., & Xiao, L. (2017) · Frontiers in Molecular Neuroscience · doi:10.3389/fnmol.2017.00316 · Cited by 59
Non-CG methylation and multiple histone profiles associate child abuse with immune and small GTPase dysregulation
Lutz, P.-É., Chay, M.-A., Pacis, A., Chen, G. G., Aouabed, Z., Maffioletti, E., Théroux, J., Grenier, J., Yang, J., Aguirre, M., et al. (2021) · Nature Communications · doi:10.1038/s41467-021-21365-3 · Cited by 59
Neuronal non-CG methylation is an essential target for MeCP2 function
Tillotson, R., Cholewa-Waclaw, J., Chhatbar, K., Connelly, J. C., Kirschner, S. A., Webb, S., Koerner, M. V., Selfridge, J., Kelly, D. A., Sousa, D. D., et al. (2021) · Molecular Cell · doi:10.1016/j.molcel.2021.01.011 · Cited by 56
Turning over DNA methylation in the mind
Lister, R., & Mukamel, E. A. (2015) · Frontiers in Neuroscience · doi:10.3389/fnins.2015.00252 · Cited by 62
Non-CpG methylation by DNMT3B facilitates REST binding and gene silencing in developing mouse hearts
Zhang, D., Wu, B., Wang, P., Wang, Y., Lu, P., Nechiporuk, T., Floß, T., Greally, J. M., Zheng, D., & Zhou, B. (2016) · Nucleic Acids Research · doi:10.1093/nar/gkw1258 · Cited by 56
MiR-218 targets MeCP2 and inhibits heroin seeking behavior
Yan, B., Hu, Z., Yao, W., Le, Q., Xu, B., Liu, X., & Ma, L. (2017) · Scientific Reports · doi:10.1038/srep40413 · Cited by 56
5-Hydroxymethylcytosine-mediated active demethylation is required for mammalian neuronal differentiation and function
Stoyanova, E. I., Riad, M. H., Rao, A., & Heintz, N. (2021) · eLife · doi:10.7554/elife.66973 · Cited by 57
The effects of cytosine methylation on general transcription factors
Jin, J., Lian, T., Gu, C., Yu, K., Gao, Y. Q., & Su, X. (2016) · Scientific Reports · doi:10.1038/srep29119 · Cited by 55
Retention of Mitochondria in Mature Human Red Blood Cells as the Result of Autophagy Impairment in Rett Syndrome
Sbardella, D., Tundo, G. R., Campagnolo, L., Valacchi, G., Orlandi, A., Curatolo, P., Borsellino, G., D’Esposito, M., Ciaccio, C., Cesare, S. D., et al. (2017) · Scientific Reports · doi:10.1038/s41598-017-12069-0 · Cited by 55
Target specificity of mammalian DNA methylation and demethylation machinery
Ravichandran, M., Jurkowska, R. Z., & Jurkowski, T. P. (2017) · Organic & Biomolecular Chemistry · doi:10.1039/c7ob02574b · Cited by 62
Glial Dysfunction in MeCP2 Deficiency Models: Implications for Rett Syndrome
Kahanovitch, U., Patterson, K. C., Hernandez, R. D., & Olsen, M. L. (2019) · International Journal of Molecular Sciences · doi:10.3390/ijms20153813 · Cited by 53
FoxG1 regulates the formation of cortical GABAergic circuit during an early postnatal critical period resulting in autism spectrum disorder-like phenotypes
Miyoshi, G., Ueta, Y., Natsubori, A., Hiraga, K., Osaki, H., Yagasaki, Y., Kishi, Y., Yanagawa, Y., Fishell, G., Machold, R. P., et al. (2021) · Nature Communications · doi:10.1038/s41467-021-23987-z · Cited by 56
Sex differences in Mecp2-mutant Rett syndrome model mice and the impact of cellular mosaicism in phenotype development
Ribeiro, M. C., & MacDonald, J. L. (2020) · Brain Research · doi:10.1016/j.brainres.2019.146644 · Cited by 55
Absence of genomic hypomethylation or regulation of cytosine-modifying enzymes with aging in male and female mice
Hadad, N., Masser, D. R., Logan, S., Wronowski, B., Mangold, C. A., Clark, N. W., Otalora, L., Unnikrishnan, A., Ford, M. M., Giles, C. B., et al. (2016) · Epigenetics & Chromatin · doi:10.1186/s13072-016-0080-6 · Cited by 48
Allele-specific non-CG DNA methylation marks domains of active chromatin in female mouse brain
Keown, C. L., Berletch, J. B., Castanon, R., Nery, J. R., Distèche, C. M., Ecker, J. R., & Mukamel, E. A. (2017) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.1611905114 · Cited by 47
Structural Basis of MeCP2 Distribution on Non-CpG Methylated and Hydroxymethylated DNA
Sperlazza, M. J., Bilinovich, S. M., Sinanan, L. M., Javier, F. R., & Williams, D. C. (2017) · Journal of Molecular Biology · doi:10.1016/j.jmb.2017.04.009 · Cited by 49
Detecting and interpreting DNA methylation marks
Ren, R., Horton, J. R., Zhang, X., Blumenthal, R., & Cheng, X. (2018) · Current Opinion in Structural Biology · doi:10.1016/j.sbi.2018.06.004 · Cited by 50
Losing Dnmt3a dependent methylation in inhibitory neurons impairs neural function by a mechanism impacting Rett syndrome
Lavery, L. A., Ure, K., Wan, Y., Luo, C., Trostle, A. J., Wang, W., Jin, H., Lopez, J., Lucero, J. D., Durham, M. A., et al. (2020) · eLife · doi:10.7554/elife.52981 · Cited by 53
Self-citation
MeCP2 and Chromatin Compartmentalization
Schmidt, A., Zhang, H., & Cardoso, M. C. (2020) · Cells · doi:10.3390/cells9040878 · Cited by 51
MeCP2 and the enigmatic organization of brain chromatin. Implications for depression and cocaine addiction
Ausió, J. (2016) · Clinical Epigenetics · doi:10.1186/s13148-016-0214-5 · Cited by 49
Non-cell Autonomous OTX2 Homeoprotein Regulates Visual Cortex Plasticity Through Gadd45b/g
Apulei, J., Kim, N., Testa, D., Ribot, J., Morizet, D., Bernard, C., Jourdren, L., Blugeon, C., Nardo, A. A. D., & Prochiantz, A. (2018) · Cerebral Cortex · doi:10.1093/cercor/bhy108 · Cited by 51
MeCP2-induced heterochromatin organization is driven by oligomerization-based liquid–liquid phase separation and restricted by DNA methylation
Zhang, H., Romero, H., Schmidt, A., Gagova, K., Qin, W., Bertulat, B., Lehmkuhl, A., Milden, M., Eck, M., Meckel, T., et al. (2022) · Nucleus · doi:10.1080/19491034.2021.2024691 · Cited by 44
Negative Allosteric Modulation of mGluR5 Partially Corrects Pathophysiology in a Mouse Model of Rett Syndrome
Tao, J., Wu, H., Coronado, A. A., de Laittre, E. A., Osterweil, E. K., Zhang, Y., & Bear, M. F. (2016) · Journal of Neuroscience · doi:10.1523/jneurosci.0672-16.2016 · Cited by 46
Disruption of MeCP2–TCF20 complex underlies distinct neurodevelopmental disorders
Zhou, J., Hamdan, H., Yalamanchili, H. K., Pang, K., Pohodich, A. E., Lopez, J., Shao, Y., Osés-Prieto, J. A., Li, L., Kim, W., et al. (2022) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.2119078119 · Cited by 44
Self-citation
Apparent bias toward long gene misregulation in MeCP2 syndromes disappears after controlling for baseline variations
Raman, A. T., Pohodich, A. E., Wan, Y., Yalamanchili, H. K., Lowry, W. E., Zoghbi, H. Y., & Liu, Z. (2018) · Nature Communications · doi:10.1038/s41467-018-05627-1 · Cited by 42
Self-citation
MECP2 and the biology of MECP2 duplication syndrome
D’Mello, S. R. (2021) · Journal of Neurochemistry · doi:10.1111/jnc.15331 · Cited by 42
DNA Methylation Atlas of the Mouse Brain at Single-Cell Resolution
Liu, H., Zhou, J., Tian, W., Luo, C., Bartlett, A., Aldridge, A., Lucero, J., Osteen, J. K., Nery, J. R., Chen, H., et al. (2020) · doi:10.1101/2020.04.30.069377 · Cited by 290
Genomic insights into MeCP2 function: A role for the maintenance of chromatin architecture
Connolly, D. R., & Zhou, Z. (2019) · Current Opinion in Neurobiology · doi:10.1016/j.conb.2019.07.002 · Cited by 40

Authors: Chen, L., Chen, K., Lavery, L. A., Baker, S. A., Shaw, C. A., Li, W., & Zoghbi, H. Y.
Affiliations: Texas Children's Hospital (US)
Dan L Duncan Comprehensive Cancer Center (US)
Department of Molecular and Human Genetics,
Howard Hughes Medical Institute (US)
ORCID: Chen, Lin
Chen, Kaifu
Lavery, Laura A.
Baker, Steven Andrew
Shaw, Chad A.
Zoghbi, Huda Y.
Venue: Proceedings of the National Academy of Sciences
Publisher: National Academy of Sciences
Published: 2015-04-13
Volume: 112
Issue: 17
Pages: 5509-5514
ISSN: 0027-8424 (ISSN-L)
1091-6490
DOI: 10.1073/pnas.1505909112
PMC: PMC4418849
OpenAlex: W2166288018
Cited by: 301
References: 31
Citations by year: 2026: 20 · 2025: 21 · 2024: 25 · 2023: 20 · 2022: 23 · 2021: 23 · 2020: 28 · 2019: 21 · 2018: 24 · 2017: 38 · 2016: 37 · 2015: 13
FWCI: 25.51
Type: journal article
Language: en
Update notices: correction (2015-04-27)
Open access: green
www.ncbi.nlm.nih.gov
Locations: Proceedings of the National Academy of Sciences (publishedVersion)
Europe PMC (PubMed Central) (publishedVersion)
PubMed Central (publishedVersion)
PubMed (publishedVersion)
Funders: Howard Hughes Medical Institute
National Human Genome Research Institute (R01HG007538, HG007538)
National Institute of Neurological Disorders and Stroke (T32NS043124, R01NS057819)
Eunice Kennedy Shriver National Institute of Child Health and Human Development (U54HD083092, P30HD024064)
Topics: Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Epigenetics and DNA Methylation
Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Genomics and Chromatin Dynamics
Keywords: MeCP2, Rett syndrome, non-CG DNA methylation, neuronal gene expression, transcriptional regulation, DNA methylation, brain-derived neurotrophic factor, brain development, delayed symptom onset, epigenetic regulation
MeSH terms: Animals, Disease Models, Animal, DNA, Gene Expression Regulation, Male, Mice, Transgenic, Neurons, Transcription, Genetic, Rett Syndrome, DNA Methylation, Brain-Derived Neurotrophic Factor, Mice, Methyl-CpG-Binding Protein 2

About these checks

Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.

Statistics and arithmetic are recomputed, not judged.

Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.

What counts as a claim ›How we measure confidence ›How we check this ›

Are you an author, or do you see a problem with this page? Request a review

MeCP2 binds to non-CG methylated DNA as neurons mature, influencing transcription and the timing of onset for Rett syndrome

Abstract

Overview

Integrity signals

Verification

Claims the paper makes

Claims other papers make about this one

Citations

Cites

Cited by

Related

Metadata

Record

About these checks