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 accessAbstract
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.
Overview
Integrity signals
Registry
Cites
Statistics
Verification
Claims the paper makes
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?
In the citing paper
“A previous study showed that the binding of MeCP2 to non-CpG methylates DNA sequences is critical for BDNF expression. This process influenced the onset timing for Rett syndrome.64”
From Epigenetic regulation in major depression and other stress-related disorders: molecular mechanisms, clinical relevance and therapeutic potential (2023) · cites this paper as [64]
In the citing paper
“In neurons, intragenic 5mCH quantitatively modulates gene expression by interacting with MECP2 and recruiting NCoR corepressor complex to chromatin (37–40).”
From Dynamic DNA methylation: In the right place at the right time (2018) · cites this paper as [38]
Citations
Cites
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-citationDNA 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-citationA 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
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
Cited by
Sorted by citations
Items 101–200 of 304
Structures of CTCF–DNA complexes including all 11 zinc fingers
Yang, J., Horton, J. R., Liu, B., Corces, V. G., Blumenthal, R., Zhang, X., & Cheng, X. (2023) · Nucleic Acids Research · doi:10.1093/nar/gkad594 · Cited by 39
CpG and Non-CpG Methylation in the Diet–Epigenetics–Neurodegeneration Connection
Fuso, A., & Lucarelli, M. (2019) · Current Nutrition Reports · doi:10.1007/s13668-019-0266-1 · Cited by 37
The Role of MeCP2 in Regulating Synaptic Plasticity in the Context of Stress and Depression
Sánchez-Lafuente, C. L., Kalynchuk, L. E., Caruncho, H. J., & Ausió, J. (2022) · Cells · doi:10.3390/cells11040748 · Cited by 41
Ehinger, Y., Bruyère, J., Panayotis, N., Abada, Y.-S. K., Borloz, E., Matagne, V., Scaramuzzino, C., Vitet, H., Delatour, B., Saidi, L., et al. (2020) · EMBO Molecular Medicine · doi:10.15252/emmm.201910889 · Cited by 39
Gene × environment interactions in autism spectrum disorders
Keil, K. P., & Lein, P. J. (2022) · Current topics in developmental biology/Current Topics in Developmental Biology · doi:10.1016/bs.ctdb.2022.11.001 · Cited by 38
Karaca, K. G., Brito, D. V. C., Zeuch, B., & Oliveira, A. M. M. (2018) · Neurobiology of Learning and Memory · doi:10.1016/j.nlm.2018.02.010 · Cited by 35
MeCP2 Levels Regulate the 3D Structure of Heterochromatic Foci in Mouse Neurons
Ito, A., Baker, S. A., Sillitoe, R. V., Sun, Y., Zhou, J., Ono, Y., Iwakiri, J., Yuzaki, M., & Zoghbi, H. Y. (2020) · Journal of Neuroscience · doi:10.1523/jneurosci.1281-19.2020 · Cited by 38
Self-citationExcitation and Inhibition Imbalance in Rett Syndrome
Li, W. (2022) · Frontiers in Neuroscience · doi:10.3389/fnins.2022.825063 · Cited by 40
Self-citationEmerging Insights into the Distinctive Neuronal Methylome
Clemens, A. W., & Gabel, H. W. (2020) · Trends in Genetics · doi:10.1016/j.tig.2020.07.009 · Cited by 36
An RNA interference screen identifies druggable regulators of MeCP2 stability
Lombardi, L. M., Zaghlula, M., Sztainberg, Y., Baker, S. A., Klisch, T. J., Tang, A. A., Huang, E. J., & Zoghbi, H. Y. (2017) · Science Translational Medicine · doi:10.1126/scitranslmed.aaf7588 · Cited by 35
Self-citationDNA Epigenetics in Addiction Susceptibility
Kaplan, G., Xu, H., Abreu, K., & Feng, J. (2022) · Frontiers in Genetics · doi:10.3389/fgene.2022.806685 · Cited by 34
Exploring the complexity of MECP2 function in Rett syndrome
Liu, Y., Whitfield, T. W., Bell, G. W., Guo, R., Flamier, A., Young, R. A., & Jaenisch, R. (2025) · Nature reviews. Neuroscience · doi:10.1038/s41583-025-00926-1 · Cited by 39
MeCP2 binds to methylated DNA independently of phase separation and heterochromatin organisation
Pantier, R., Brown, M., Han, S., Paton, K. M., Meek, S. E., Montavon, T., Shukeir, N., McHugh, T., Kelly, D. A., Hochepied, T., et al. (2024) · Nature Communications · doi:10.1038/s41467-024-47395-1 · Cited by 37
Optical Trapping Nanometry of Hypermethylated CPG-Island DNA
Pongor, C. I., Bianco, P., Ferenczy, G. G., Kellermayer, R., & Kellermayer, M. (2017) · Biophysical Journal · doi:10.1016/j.bpj.2016.12.029 · Cited by 33
Sanfeliu, A., Hokamp, K., Gill, M., & Tropea, D. (2019) · Frontiers in Psychiatry · doi:10.3389/fpsyt.2019.00278 · Cited by 35
Dosage-sensitive genes in autism spectrum disorders: From neurobiology to therapy
Javed, S., Selliah, T., Lee, Y., & Huang, W. (2020) · Neuroscience & Biobehavioral Reviews · doi:10.1016/j.neubiorev.2020.08.009 · Cited by 32
Mammalian DNA methylome dynamics: mechanisms, functions and new frontiers
Wei, A., & Wu, H. (2022) · Development · doi:10.1242/dev.182683 · Cited by 34
Plasticity at the DNA recognition site of the MeCP2 mCG-binding domain
Lei, M., Tempel, W., Chen, S., Liu, K., & Min, J. (2019) · Biochimica et Biophysica Acta (BBA) - Gene Regulatory Mechanisms · doi:10.1016/j.bbagrm.2019.194409 · Cited by 32
Genome-wide distribution of linker histone H1.0 is independent of MeCP2
Ito, A., Yamalanchili, H. K., Shao, Y., Baker, S. A., Heckman, L. D., Lavery, L. A., Kim, J., Lombardi, L. M., Sun, Y., Liu, Z., et al. (2018) · Nature Neuroscience · doi:10.1038/s41593-018-0155-8 · Cited by 32
Self-citationZheng, Z., Ambigapathy, G., & Keifer, J. (2017) · eLife · doi:10.7554/elife.25384 · Cited by 31
Chen, L., Zhou, A. S., & West, A. E. (2017) · Journal of Neurophysiology · doi:10.1152/jn.00067.2017 · Cited by 35
Self-citationRecent advances in understanding synaptic abnormalities in Rett syndrome
Johnston, M. V., Blue, M. E., & Naidu, S. (2015) · F1000Research · doi:10.12688/f1000research.6987.1 · Cited by 30
Uncovering the Role of the Methylome in Dementia and Neurodegeneration
Klein, H., & Jager, P. L. D. (2016) · Trends in Molecular Medicine · doi:10.1016/j.molmed.2016.06.008 · Cited by 31
Nuclear receptor corepressors in intellectual disability and autism
Kong, Y., Zhou, W., & Sun, Z. (2020) · Molecular Psychiatry · doi:10.1038/s41380-020-0667-y · Cited by 29
Decoding the non‐coding genome: elucidating genetic risk outside the coding genome
Barr, C. L., & Misener, V. L. (2015) · Genes Brain & Behavior · doi:10.1111/gbb.12269 · Cited by 29
The distinct methylation landscape of maturing neurons and its role in Rett syndrome pathogenesis
Lavery, L. A., & Zoghbi, H. Y. (2019) · Current Opinion in Neurobiology · doi:10.1016/j.conb.2019.08.001 · Cited by 29
Self-citationFlanking sequence preference modulates de novo DNA methylation in the mouse genome
Mallona, I., Ilie, I. M., Karemaker, I. D., Butz, S., Manzo, M., Caflisch, A., & Baubec, T. (2020) · Nucleic Acids Research · doi:10.1093/nar/gkaa1168 · Cited by 33
Developmental remodelling of non-CG methylation at satellite DNA repeats
Ross, S. E., Angeloni, A., Geng, F., de Mendoza, A., & Bogdanović, O. (2020) · Nucleic Acids Research · doi:10.1093/nar/gkaa1135 · Cited by 27
Genome-wide redistribution of MeCP2 in dorsal root ganglia after peripheral nerve injury
Manners, M. T., Ertel, A., Tian, Y., & Ajit, S. K. (2016) · Epigenetics & Chromatin · doi:10.1186/s13072-016-0073-5 · Cited by 26
Challenges for Epigenetic Research in Inflammatory Bowel Diseases
Kellermayer, R. (2017) · Epigenomics · doi:10.2217/epi-2016-0155 · Cited by 26
DNA methylation analysis in constitutional disorders: Clinical implications of the epigenome
Schenkel, L. C., Rodenhiser, D. I., Ainsworth, P., Paré, G., & Sadiković, B. (2016) · Critical Reviews in Clinical Laboratory Sciences · doi:10.3109/10408363.2015.1113496 · Cited by 25
Madrid, A., Borth, L., Hogan, K. J., Hariharan, N., Papale, L. A., Alisch, R. S., & Iskandar, B. J. (2020) · Epigenetics · doi:10.1080/15592294.2020.1786320 · Cited by 24
Emerging physiological and pathological roles of MeCP2 in non-neurological systems
Wang, J., Xiao, Y., Liu, C., Huang, Y., Petersen, R. B., Zheng, L., & Huang, K. (2021) · Archives of Biochemistry and Biophysics · doi:10.1016/j.abb.2021.108768 · Cited by 23
MeCP2 and transcriptional control of eukaryotic gene expression
Akhtar, G. B., Buist, M., & Rastegar, M. (2022) · European Journal of Cell Biology · doi:10.1016/j.ejcb.2022.151237 · Cited by 24
Bajikar, S. S., Zhou, J., O’Hara, R. A., Tirumala, H. P., Durham, M. A., Trostle, A. J., Dias, M., Shao, Y., Chen, H., Wang, W., et al. (2024) · Neuron · doi:10.1016/j.neuron.2024.11.006 · Cited by 25
Self-citationBuist, M., Tobgy, N. E., Shevkoplyas, D., Genung, M., Sher, A. A., Pejhan, S., & Rastegar, M. (2022) · Cells · doi:10.3390/cells11091442 · Cited by 24
Riedmann, C., & Fondufe‐Mittendorf, Y. N. (2016) · Scientific Reports · doi:10.1038/srep33186 · Cited by 22
Chen, M., Li, S., Srinivasasainagendra, V., Sharma, M., Li, Z., Tiwari, H. K., Tollefsbol, T. O., & Li, Y. (2022) · Carcinogenesis · doi:10.1093/carcin/bgac009 · Cited by 41
Torres, O. V., Jayanthi, S., McCoy, M. T., & Cadet, J. L. (2017) · The International Journal of Neuropsychopharmacology · doi:10.1093/ijnp/pyx105 · Cited by 21
Schmitt, I., Evert, B. O., Sharma, A., Khazneh, H., Murgatroyd, C., & Wüllner, U. (2024) · Molecular Neurobiology · doi:10.1007/s12035-024-03974-3 · Cited by 20
Downs, J. A., & Leonard, H. (2016) · Wiener Medizinische Wochenschrift · doi:10.1007/s10354-016-0494-6 · Cited by 23
Epigenetic and epitranscriptomic regulation of axon regeneration
Cheng, Y., Song, H., Ming, G., & Weng, Y. (2023) · Molecular Psychiatry · doi:10.1038/s41380-023-02028-9 · Cited by 21
Pehli̇van, D., Bengtsson, J. D., Bajikar, S. S., Grochowski, C. M., Lun, M. Y., Gandhi, M., Jolly, A., Trostle, A. J., Harris, H. K., Suter, B., et al. (2024) · Genome Medicine · doi:10.1186/s13073-024-01411-7 · Cited by 22
Self-citationTranscriptomic and Epigenomic Landscape in Rett Syndrome
Marano, D., Fioriniello, S., D’Esposito, M., & Ragione, F. D. (2021) · Biomolecules · doi:10.3390/biom11070967 · Cited by 24
Disruption of AT-hook 1 domain in MeCP2 protein caused behavioral abnormality in mice
Xu, M., Song, P., Huang, W., He, R., He, Y., Zhou, X., Gu, Y., Pan, S., & Hu, Y. (2017) · Biochimica et Biophysica Acta (BBA) - Molecular Basis of Disease · doi:10.1016/j.bbadis.2017.10.022 · Cited by 18
Effects of early-life exposure to THIP on phenotype development in a mouse model of Rett syndrome
Zhong, W., Johnson, C. M., Wu, Y., Cui, N., Xing, H., Zhang, S., & Jiang, C. L. (2016) · Journal of Neurodevelopmental Disorders · doi:10.1186/s11689-016-9169-2 · Cited by 18
Mouse models as a tool for discovering new neurological diseases
Tan, Q., & Zoghbi, H. Y. (2018) · Neurobiology of Learning and Memory · doi:10.1016/j.nlm.2018.07.006 · Cited by 18
Self-citationProteins in DNA methylation and their role in neural stem cell proliferation and differentiation
Sun, J., Yang, J., Miao, X., Loh, H. H., Pei, D., & Zheng, H. (2021) · Cell Regeneration · doi:10.1186/s13619-020-00070-4 · Cited by 19
Buist, M., Fuss, D., & Rastegar, M. (2021) · Biomolecules · doi:10.3390/biom11081253 · Cited by 18
Enzymology of Mammalian DNA Methyltransferases
Jurkowska, R. Z., & Jeltsch, A. (2022) · Advances in experimental medicine and biology · doi:10.1007/978-3-031-11454-0_4 · Cited by 18
The Role of MicroRNAs in Cerebellar Development and Autism Spectrum Disorder During Embryogenesis
Constantin, L. (2016) · Molecular Neurobiology · doi:10.1007/s12035-016-0220-9 · Cited by 19
Fagiolini, M., Patrizi, A., LeBlanc, J. J., Jin, L., Maezawa, I., Sinnett, S. E., Gray, S. J., Molholm, S., Foxe, J. J., Johnston, M. V., et al. (2020) · Neuroscience · doi:10.1016/j.neuroscience.2020.04.037 · Cited by 16
Self-citationMoore, J. R., Nemera, M. T., D’Souza, R. D., Hamagami, N., Clemens, A. W., Beard, D. C., Urman, A., Razia, Y., Mendoza, V. R., Law, T. E., et al. (2025) · Nature Neuroscience · doi:10.1038/s41593-025-01947-w · Cited by 21
Ivy, A. S., & Standridge, S. (2021) · Seminars in Pediatric Neurology · doi:10.1016/j.spen.2021.100881 · Cited by 16
Bajikar, S. S., Sztainberg, Y., Trostle, A. J., Tirumala, H. P., Wan, Y., Harrop, C. L., Bengtsson, J. D., Carvalho, C. M. B., Pehlivan, D., Suter, B., et al. (2024) · Human Molecular Genetics · doi:10.1093/hmg/ddae135 · Cited by 16
Self-citationTan, H. K., Wu, C.-S., Li, J., Tan, Z. H., Hoffman, J. R., Fry, C. J., Yang, H. H., Ruscio, A. D., & Tenen, D. G. (2019) · Nucleic Acids Research · doi:10.1093/nar/gkz520 · Cited by 15
Developmental Accumulation of Gene Body and Transposon Non-CpG Methylation in the Zebrafish Brain
Ross, S. E., Hesselson, D., & Bogdanović, O. (2021) · Frontiers in Cell and Developmental Biology · doi:10.3389/fcell.2021.643603 · Cited by 16
Jin, Y., Su, K., Kong, H. E., Ma, W., Wang, Z., Li, Y., Li, R., Allen, E. G., Wu, H., & Jin, P. (2022) · Human Molecular Genetics · doi:10.1093/hmg/ddac189 · Cited by 16
Chen, Y., Wu, X., Hu, H., Yang, S.-N., Zhang, Z., Fu, G.-L., Zhang, C., Li, Z., Wu, F., Si, K.-W., et al. (2023) · Neurobiology of Disease · doi:10.1016/j.nbd.2023.106346 · Cited by 15
Interaction of methyl-CpG-binding protein 2 (MeCP2) with distinct enhancers in the mouse cortex
Mishra, G. P., Sun, E. X., Chin, T., Eckhardt, M., Greenberg, M. E., & Stroud, H. (2024) · Nature Neuroscience · doi:10.1038/s41593-024-01808-y · Cited by 15
Functional conservation of MBD proteins: MeCP2 and Drosophila MBD proteins alter sleep
Gupta, T., Morgan, H. R., Bailey, J. A., & Certel, S. J. (2016) · Genes Brain & Behavior · doi:10.1111/gbb.12314 · Cited by 15
Gomes, A. R., Fernandes, T. G., Cabral, J. M. S., & Diogo, M. M. (2021) · International Journal of Molecular Sciences · doi:10.3390/ijms22073751 · Cited by 15
A novel pathogenic mutation of MeCP2 impairs chromatin association independent of protein levelsPreprint
Zhou, J., Cattoglio, C., Shao, Y., Tirumala, H. P., Vetralla, C., Bajikar, S. S., Li, Y., Chen, H., Wang, Q., Wu, Z., et al. (2023) · Genes & Development · doi:10.1101/gad.350733.123 · Cited by 15
Self-citationMarballi, K. K., & MacDonald, J. L. (2021) · Neurochemistry International · doi:10.1016/j.neuint.2021.105076 · Cited by 15
Absence of MeCP2 binding to non-methylated GT-rich sequences in vivo
Connelly, J. C., Cholewa-Waclaw, J., Webb, S., Steccanella, V., Waclaw, B., & Bird, A. P. (2020) · Nucleic Acids Research · doi:10.1093/nar/gkaa102 · Cited by 14
Sbardella, D., Tundo, G. R., Cunsolo, V., Grasso, G., Cascella, R., Caputo, V., Santoro, A. M., Milardi, D., Pecorelli, A., Ciaccio, C., et al. (2020) · Biochimica et Biophysica Acta (BBA) - Molecular Basis of Disease · doi:10.1016/j.bbadis.2020.165793 · Cited by 15
MeCP2 represses the activity of topoisomerase IIβ in long neuronal genes
Nettles, S. A., Ikeuchi, Y., Lefton, K. B., Abbasi, L., Erickson, A., Agwu, C., Papouin, T., Bonni, A., & Gabel, H. W. (2023) · Cell Reports · doi:10.1016/j.celrep.2023.113538 · Cited by 15
MeCP2 interacts with the super elongation complex to regulate transcription
Sonn, J. Y., Kim, W., Iwanaszko, M., Aoi, Y., Li, Y., Qi, G., Parkitny, L., Brissette, J. L., Weiner, L., Botas, J., et al. (2025) · Science Advances · doi:10.1126/sciadv.adt5937 · Cited by 14
Self-citationExceptional epigenetics in the brain
Luo, C., & Ecker, J. R. (2015) · Science · doi:10.1126/science.aac5832 · Cited by 12
Fuso, A. (2018) · Epigenomes · doi:10.3390/epigenomes2040022 · Cited by 13
Cytosine methylation regulates DNA bendability depending on the curvature
Yeou, S., Hwang, J., Yi, J., Kim, C., Kim, S. K., & Lee, N. K. (2022) · Chemical Science · doi:10.1039/d1sc07115g · Cited by 13
MeCP2 regulates Gdf11, a dosage-sensitive gene critical for neurological function
Bajikar, S. S., Anderson, A. G., Zhou, J., Durham, M. A., Trostle, A. J., Wan, Y., Liu, Z., & Zoghbi, H. Y. (2023) · eLife · doi:10.7554/elife.83806 · Cited by 14
Self-citationParadoxical physiological responses to propranolol in a Rett syndrome patient: a case report
Santosh, P., Bell, L., Lievesley, K., Singh, J., & Fiori, F. (2016) · BMC Pediatrics · doi:10.1186/s12887-016-0734-3 · Cited by 13
Epigenetics in rare neurological diseases
Roberts, C.-T., Arezoumand, K. S., Shahib, A. K., Davie, J., & Rastegar, M. (2024) · Frontiers in Cell and Developmental Biology · doi:10.3389/fcell.2024.1413248 · Cited by 16
A novel mutation R190H in the AT-hook 1 domain of MeCP2 identified in an atypical Rett syndrome
Zhou, X., Liao, Y., Xu, M., Ji, Z., Xu, Y., Zhou, L., Wei, X., Hu, P., Han, P., Yang, F., et al. (2017) · Oncotarget · doi:10.18632/oncotarget.18955 · Cited by 11
Zhao, D., Lin, M., Pedrosa, E., Lachman, H. M., & Zheng, D. (2017) · BMC Genomics · doi:10.1186/s12864-017-4261-x · Cited by 12
ME-Class2 reveals context dependent regulatory roles for 5-hydroxymethylcytosine
Schlosberg, C., Wu, D. Y., Gabel, H. W., & Edwards, J. R. (2019) · Nucleic Acids Research · doi:10.1093/nar/gkz001 · Cited by 13
Identification of Novel MeCP2 Cancer-Associated Target Genes and Post-Translational Modifications
Castro‐Piedras, I., Vartak, D., Sharma, M., Pandey, S., Casas, L., Molehin, D., Rasha, F. A., Fokar, M., Nichols, J., Almodóvar, S., et al. (2020) · Frontiers in Oncology · doi:10.3389/fonc.2020.576362 · Cited by 12
Mecp2 Mediates Experience-Dependent Transcriptional Upregulation of Ryanodine Receptor Type-3
Torres, R. F., Hidalgo, C., & Kerr, B. (2017) · Frontiers in Molecular Neuroscience · doi:10.3389/fnmol.2017.00188 · Cited by 11
Activity-induced MeCP2 phosphorylation regulates retinogeniculate synapse refinement
Tzeng, C. P., Whitwam, T., Boxer, L. D., Li, E., Silberfeld, A., Trowbridge, S. K., Mei, K., Lin, C., Shamah, R., Griffith, E. C., et al. (2023) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.2310344120 · Cited by 9
Krishnaraj, R. R., Haase, F., Coorey, B., Luca, E., Wong, I., Boyling, A., Ellaway, C., Christodoulou, J., & Gold, W. A. (2019) · Human Mutation · doi:10.1002/humu.23887 · Cited by 12
Rao, S., Chen, Y., Hong, T., He, Z., Guo, S., Lai, H., Guo, G., Du, Y., & Zhou, X. (2016) · Analytical Chemistry · doi:10.1021/acs.analchem.6b02625 · Cited by 11
Choi, G. T., Lee, S., Yoo, S., & Tae, J. (2025) · International Journal of Molecular Sciences · doi:10.3390/ijms26178277 · Cited by 10
Chia, J. Y., Tan, W. S., Ng, C. L., Hu, N., Foo, H. L., & Ho, K. L. (2016) · Scientific Reports · doi:10.1038/srep31210 · Cited by 10
Tabe-Bordbar, S., & Sinha, S. (2023) · BMC Genomics · doi:10.1186/s12864-023-09498-9 · Cited by 10
Keifer, J. (2017) · Genes · doi:10.3390/genes8020069 · Cited by 10
Heffel, M. G., Zhou, J., Zhang, Y., Lee, D.-S., Hou, K., Pastor-Alonso, O., Abuhanna, K. D., Schmitt, A. D., Li, T., Haeussler, M., et al. (2022) · bioRxiv (Cold Spring Harbor Laboratory) · doi:10.1101/2022.10.07.511350 · Cited by 10
Zhang, X., Blumenthal, R., & Cheng, X. (2024) · Nucleic Acids Research · doi:10.1093/nar/gkae832 · Cited by 9
Multiple epigenetic biomarkers for evaluation of students' academic performance
Lee, L., Su, M., Cho, Y., Lee‐Chen, G., Yeh, T., & Chang, C. (2019) · Genes Brain & Behavior · doi:10.1111/gbb.12559 · Cited by 8
Striatal ZBTB16 Is Associated With Cognitive Deficits in Alzheimer Disease Mice
Lee, S.-J., Kim, T. K., Choi, J. E., Kim, H.-S., & Im, H. (2022) · International Neurourology Journal · doi:10.5213/inj.2244254.127 · Cited by 9
Mykins, M., Layo-Carris, D. E., Dunn, L. R., Skinner, D. W., McBryar, A. H., Perez, S., Shultz, T. R., Willems, A. R., Lau, B. Y. B., Hong, T., et al. (2023) · Journal of Neuroscience Research · doi:10.1002/jnr.25190 · Cited by 9
Embryonic Stem Cell-Derived Neurons as a Model System for Epigenome Maturation during Development
Martin, S., Poppe, D., Olova, N. N., O’Leary, C. J., Ivanova, E., Pflueger, J., Dechka, J., Simmons, R. K., Cooper, H. M., Reik, W., et al. (2023) · Genes · doi:10.3390/genes14050957 · Cited by 9
The role of prenatal choline and its impact on neurodevelopmental disorders
Mujica-Coopman, M. F., Paules, E. M., & Trujillo‐Gonzalez, I. (2024) · Frontiers in Nutrition · doi:10.3389/fnut.2024.1463983 · Cited by 9
The methyl-CpG-binding protein 2 inhibits cGAS-associated signaling
Chamma, H., Guha, S., Eloiflin, R.-J., Augereau, A., Hars, P. L., Schüssler, M., Messaoud-Nacer, Y., Salma, M., McKellar, J., Re, J., et al. (2025) · Nature Communications · doi:10.1038/s41467-025-65713-z · Cited by 7
Multi‐omics in MECP2 duplication syndrome patients and carriers
Pascual‐Alonso, A., Xiol, C., Smirnov, D., Kopajtich, R., Prokisch, H., & Armstrong, J. (2024) · European Journal of Neuroscience · doi:10.1111/ejn.16389 · Cited by 8
Rett Syndrome: A Tale of Altered Genetics, Synaptic Plasticity, and Neurodevelopmental Dynamics
Oluigbo, D. (2023) · Cureus · doi:10.7759/cureus.41555 · Cited by 8
MeCP2 regulates telencephalic development in human cerebral organoids
Tenreiro, M. F., Mohana-Borges, R., Sánchez-Sánchez, S. M., Dias, Â. R. M., Blanch, R., & Muotri, A. R. (2025) · Cell Reports · doi:10.1016/j.celrep.2025.116670 · Cited by 8
Epigenetic Regulation and Neurodevelopmental Disorders: From MeCP2 to the TCF20/PHF14 Complex
Dominguez, G., Wu, Y., & Zhou, J. (2024) · Genes · doi:10.3390/genes15121653 · Cited by 11
Good, K., Kalani, L., Vincent, J. B., & Ausió, J. (2025) · Biochemistry and Cell Biology · doi:10.1139/bcb-2024-0237 · Cited by 6
Leveraging the genetic basis of Rett syndrome to ascertain pathophysiology
Yang, H., Li, K., Han, S. K., Zhou, A., & Zhou, Z. (2018) · Neurobiology of Learning and Memory · doi:10.1016/j.nlm.2018.11.006 · Cited by 6
Metadata
- 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)
- 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
- 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