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題名 Mice lacking cyclin-dependent kinase-like 5 manifest autistic and ADHD-like behaviors 作者 廖文霖
Jhang, Cian-Ling
Huang, Tzyy-Nan
Hsueh, Yi-Ping
Liao, Wenlin貢獻者 神科所 關鍵詞 dopamine; attention-deficit/hyperactivity disorder; autistic disorder; cyclins; phosphotransferases; mice 日期 2017 上傳時間 28-Sep-2017 17:39:26 (UTC+8) 摘要 Neurodevelopmental disorders frequently share common clinical features and appear high rate of comorbidity, such as those present in patients with attention-deficit hyperactivity disorder (ADHD) and autism spectrum disorders (ASD). While characterizing behavioral phenotypes in the mouse model of cyclin-dependent kinase-like 5 (CDKL5) disorder, a neurodevelopmental disorder caused by mutations in the X-linked gene encoding CDKL5, we found that these mice manifested behavioral phenotypes mimicking multiple key features of ASD, such as impaired social interaction and communication, as well as increased stereotypic digging behaviors. These mice also displayed hyper-locomotion, increased aggressiveness and impulsivity, plus deficits in motor and associative learning, resembling primary symptoms of ADHD. Through brain region-specific biochemical analysis, we uncovered that loss of CDKL5 disrupts dopamine synthesis and the expression of social communication-related key genes, such as forkhead-box P2 and mu-opioid receptor, in the corticostriatal circuit. Together, our findings support that CDKL5 plays a role in the comorbid features of autism and ADHD, and mice lacking CDKL5 may serve as an animal model to study the molecular and circuit mechanisms underlying autism-ADHD comorbidity. 關聯 Human Molecular Genetics 資料類型 article DOI https://doi.org/10.1093/hmg/ddx279 dc.contributor 神科所 zh_TW dc.creator (作者) 廖文霖 zh_TW dc.creator (作者) Jhang, Cian-Ling en_US dc.creator (作者) Huang, Tzyy-Nan en_US dc.creator (作者) Hsueh, Yi-Ping en_US dc.creator (作者) Liao, Wenlin en_US dc.date (日期) 2017 dc.date.accessioned 28-Sep-2017 17:39:26 (UTC+8) - dc.date.available 28-Sep-2017 17:39:26 (UTC+8) - dc.date.issued (上傳時間) 28-Sep-2017 17:39:26 (UTC+8) - dc.identifier.uri (URI) http://nccur.lib.nccu.edu.tw/handle/140.119/113135 - dc.description.abstract (摘要) Neurodevelopmental disorders frequently share common clinical features and appear high rate of comorbidity, such as those present in patients with attention-deficit hyperactivity disorder (ADHD) and autism spectrum disorders (ASD). While characterizing behavioral phenotypes in the mouse model of cyclin-dependent kinase-like 5 (CDKL5) disorder, a neurodevelopmental disorder caused by mutations in the X-linked gene encoding CDKL5, we found that these mice manifested behavioral phenotypes mimicking multiple key features of ASD, such as impaired social interaction and communication, as well as increased stereotypic digging behaviors. These mice also displayed hyper-locomotion, increased aggressiveness and impulsivity, plus deficits in motor and associative learning, resembling primary symptoms of ADHD. Through brain region-specific biochemical analysis, we uncovered that loss of CDKL5 disrupts dopamine synthesis and the expression of social communication-related key genes, such as forkhead-box P2 and mu-opioid receptor, in the corticostriatal circuit. Together, our findings support that CDKL5 plays a role in the comorbid features of autism and ADHD, and mice lacking CDKL5 may serve as an animal model to study the molecular and circuit mechanisms underlying autism-ADHD comorbidity. en_US dc.format.extent 98 bytes - dc.format.mimetype text/html - dc.relation (關聯) Human Molecular Genetics zh_TW dc.subject (關鍵詞) dopamine; attention-deficit/hyperactivity disorder; autistic disorder; cyclins; phosphotransferases; mice en_US dc.title (題名) Mice lacking cyclin-dependent kinase-like 5 manifest autistic and ADHD-like behaviors zh_TW dc.type (資料類型) article dc.identifier.doi (DOI) 10.1093/hmg/ddx279 dc.doi.uri (DOI) https://doi.org/10.1093/hmg/ddx279