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Psychiatry"],"published-print":{"date-parts":[[2024,10]]},"abstract":"<jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>\n                      Kabuki syndrome (KS) is a rare developmental disorder characterised by multiple congenital anomalies and intellectual disability.\n                      <jats:italic>UTX<\/jats:italic>\n                      (ubiquitously transcribed tetratricopeptide repeat, X chromosome), which encodes a histone demethylase, is one of the two major pathogenic risk genes for KS. Although intellectual disability is a key phenotype of KS, the role of\n                      <jats:italic>UTX<\/jats:italic>\n                      in cognitive function remains unclear. Currently, no targeted therapies are available for KS.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Aims<\/jats:title>\n                    <jats:p>\n                      This study aimed to investigate how\n                      <jats:italic>UTX<\/jats:italic>\n                      regulates cognition, to explore the mechanisms underlying\n                      <jats:italic>UTX<\/jats:italic>\n                      dysfunction and to identify potential molecular targets for treatment.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>\n                      We generated\n                      <jats:italic>UTX<\/jats:italic>\n                      conditional knockout mice and found that\n                      <jats:italic>UTX<\/jats:italic>\n                      deletion downregulated calmodulin transcription by disrupting H3K27me3 (trimethylated histone H3 at lysine 27) demethylation.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      <jats:italic>UTX<\/jats:italic>\n                      \u2010knockout mice showed decreased phosphorylation of calcium \/ calmodulin\u2010dependent protein kinase II, impaired long\u2010term potentiation and deficit in remote contextual fear memory. These effects were reversed by an Food and Drug Administration\u2010approved drug desipramine.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>\n                      Our results reveal an epigenetic mechanism underlying the important role of\n                      <jats:italic>UTX<\/jats:italic>\n                      in synaptic plasticity and cognitive function, and suggest that desipramine could be a potential treatment for KS.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1136\/gpsych-2023-101430","type":"journal-article","created":{"date-parts":[[2024,10,30]],"date-time":"2024-10-30T11:24:33Z","timestamp":1730287473000},"update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":0,"title":["Desipramine reverses remote memory deficits by activating calmodulin\u2010CaMKII 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