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In the brain, Dnmt3a is the sole \u2018writer\u2019 of atypical non-CpG methylation (mCH), and MeCP2 is the only known \u2018reader\u2019 for mCH. We asked if MeCP2 is the sole reader for Dnmt3a dependent methylation by comparing mice lacking either protein in GABAergic inhibitory neurons. Loss of either protein causes overlapping and distinct features from the behavioral to molecular level. Loss of Dnmt3a causes global loss of mCH and a subset of mCG sites resulting in more widespread transcriptional alterations and severe neurological dysfunction than MeCP2 loss. These data suggest that MeCP2 is responsible for reading only part of the Dnmt3a dependent methylation in the brain. Importantly, the impact of MeCP2 on genes differentially expressed in both models shows a strong dependence on mCH, but not Dnmt3a dependent mCG, consistent with mCH playing a central role in the pathogenesis of Rett Syndrome.<\/jats:p>","DOI":"10.7554\/elife.52981","type":"journal-article","created":{"date-parts":[[2020,3,11]],"date-time":"2020-03-11T09:00:12Z","timestamp":1583917212000},"source":"Crossref","is-referenced-by-count":54,"title":["Losing Dnmt3a dependent methylation in inhibitory neurons impairs neural function by a mechanism impacting Rett syndrome"],"prefix":"10.7554","volume":"9","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-7153-0266","authenticated-orcid":true,"given":"Laura A","family":"Lavery","sequence":"first","affiliation":[{"name":"Jan and Dan Duncan Neurological Research Institute at Texas Children\u2019s Hospital, Houston, United States"},{"name":"Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, United 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