biostudies-literatureUnknown52(1)Zhang YQ<h4>Background and hypothesis</h4>Large-scale whole exome sequencing (WES) analyses have implicated rare protein-truncating variants (PTVs) in the AKAP11 gene contributing to schizophrenia risk. Previous studies reported alterations of EEG characteristics and synaptic proteome in Akap11 mutant mice. We hypothesize that synaptic dysfunction contributes to AKAP11 deficiency in the pathogenesis of schizophrenia.<h4>Study design</h4>We generated an Akap11 knockout mouse and employed a series of behavioral evaluations, neuronal sparse labeling assays, electron microscopy, and immunoprecipitation mass spectrometry (IP-MS) to elucidate the impacts of Akap11 on schizophrenia-relevant phenotypes.<h4>Study results</h4>Our behavioral paradigm evaluations revealed that Akap11 deficient mice exhibited Schizophrenia bulletinsbaf055https://www.ebi.ac.uk/biostudies/studies/S-EPMC12809822biostudies-literatureSchizophrenia-Related Synaptic Dysfunction and Abnormal Sensorimotor Gating in Akap11-Deficient Mice.PMC12809822Li CGuo YYin MYXiao XLi SWZhang YQCai XZhang QChang HLi MMa GWang LfalseSchizophrenia-Related Synaptic Dysfunction and Abnormal Sensorimotor Gating in Akap11-Deficient Mice.<h4>Background and hypothesis</h4>Large-scale whole exome sequencing (WES) analyses have implicated rare protein-truncating variants (PTVs) in the AKAP11 gene contributing to schizophrenia risk. Previous studies reported alterations of EEG characteristics and synaptic proteome in Akap11 mutant mice. We hypothesize that synaptic dysfunction contributes to AKAP11 deficiency in the pathogenesis of schizophrenia.<h4>Study design</h4>We generated an Akap11 knockout mouse and employed a series of behavioral evaluations, neuronal sparse labeling assays, electron microscopy, and immunoprecipitation mass spectrometry (IP-MS) to elucidate the impacts of Akap11 on schizophrenia-relevant phenotypes.<h4>Study results</h4>Our behavioral paradigm evaluations revealed that Akap11 deficient mice exhibited 2026-01-01T00:00:00Z2026 Jan2026-06-06T15:58:18.555Z2026-06-02T03:10:43.365ZS-EPMC128098224040841910.1093/schbul/sbaf055