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Desipramine reverses remote memory deficits by activating calmodulin-CaMKII pathway in a UTX knockout mouse model of Kabuki syndrome

Desipramine reverses remote memory deficits by activating calmodulin-CaMKII pathway in a UTX knockout mouse model of Kabuki syndrome

Lei Chen ¹, Yuting Li ¹, Minggang Liu ^2,3, Zhaohui Lan ¹, Xu Zhang ¹, Xiujuan Yang ¹, Qian Zhao ¹, Shuai Wang ¹, Longyong Xu ⁴, Ying Zhou ¹, Yifang Kuang ¹, Tatsuo Suzuki ⁵, Katsuhiko Tabuchi ⁵, Eiki Takahashi ¹, Miou Zhou ⁶, Charlie Degui Chen ⁴, Tianle Xu ², Weidong Li ^1,7

Lei Chen ¹, Yuting Li ¹, Minggang Liu ^2,3

¹Bio-X Institutes, Key Laboratory for the Genetics of Development and Neuropsychiatric Disorders (Ministry of Education), Brain Health and Brain Technology Center at Global Institute of Future Technology, Institute of Psychology and Behavioral Science, Shanghai Jiao Tong University, Shanghai, China.
²Department of Anatomy and Physiology, Shanghai Jiao Tong University School of Medicine, Shanghai, China.
³Institute of Mental Health and Drug Discovery, Oujiang Laboratory, Wenzhou, Zhejiang, China.
⁴Shanghai Institutes for Biological Sciences, Chinese Academy of Sciences, Shanghai, China.
⁵Shinshu University School of Medicine, Nagano, Japan.
⁶Western University of Health Sciences, Pomona, California, USA.
⁷WLA Laboratories, World Laureates Association, Shanghai, China.

⁰Bio-X Institutes, Key Laboratory for the Genetics of Development and Neuropsychiatric Disorders (Ministry of Education), Brain Health and Brain Technology Center at Global Institute of Future Technology, Institute of Psychology and Behavioral Science, Shanghai Jiao Tong University, Shanghai, China.

General Psychiatry +

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November 4, 2024

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View abstract on PubMed

Summary

This summary is machine-generated.

This study reveals that UTX gene dysfunction impairs cognitive function in Kabuki syndrome by affecting epigenetic regulation. The drug desipramine shows potential for treating cognitive deficits in KS patients.

Area Of Science

Epigenetics
Neuroscience
Developmental Biology

Background

Kabuki syndrome (KS) is a rare genetic disorder with intellectual disability.
The UTX gene, encoding a histone demethylase, is a key risk gene for KS.
The precise role of UTX in cognitive function and potential treatments for KS remain largely unknown.

Purpose Of The Study

To investigate the regulatory role of UTX in cognitive processes.
To elucidate the molecular mechanisms behind UTX dysfunction in KS.
To identify potential therapeutic targets for KS-associated cognitive impairments.

Main Methods

Generation of UTX conditional knockout mice.
Analysis of calmodulin gene transcription and H3K27me3 demethylation.
Assessment of synaptic plasticity and memory in mice.

Main Results

UTX deletion downregulated calmodulin transcription via disrupted H3K27me3 demethylation.
UTX-knockout mice exhibited impaired long-term potentiation and memory deficits.
Administration of desipramine reversed the observed cognitive impairments.

Conclusions

UTX plays a critical role in synaptic plasticity and cognition through an epigenetic mechanism.
Desipramine represents a potential therapeutic strategy for Kabuki syndrome.
This research provides insights into the molecular basis of KS and potential treatment avenues.

Keywords:

Cognition Cognitive Neuroscience Crosses, Genetic Drug-Seeking Behavior Models, Genetic

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