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Region-dependent differences in tonic inhibition underlie epileptic features in Angelman syndrome model mice

delete2026-06-02
delete0
PRE
AI
M
Miho Watanabe
T
Takeru Goto
R
Ryoko Miyoshi
S
Sachiko Nakakubo
Y
Yasuyoshi Hiramatsu
M
Midori Nakajima
Y
Yuki Ueda
H
Hideaki Shiraishi
A
Atsushi Manabe
P
Prithu Mondal
D
Dishary Sharmin
M
Michael Ming Poe
J
James M. Cook
K
Kiyoshi Egawa *
DOI：10.1002/epi.70307delete
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Abstract

Abstract

En 中文
Angelman syndrome (AS) is a neurodevelopmental disorder caused by loss of function of the maternally expressed UBE3A gene. Epilepsy and abnormal electroencephalographic (EEG) rhythms are key features, but their mechanisms and treatment remain unclear. Previous work showed that extrasynaptic γ-aminobutyric acid type A (GABAA) receptor-mediated tonic inhibition is reduced in cerebellar granule cells of AS model mice, contributing to motor deficits. Here, we evaluated tonic inhibition across brain regions and tested whether its dysregulation drives epilepsy, EEG abnormalities, and behavioral deficits in AS.
Keywords:
electroencephalography
GABAA receptors
mouse model
neurodevelopmental disorders
tonic inhibition

Journal

Epilepsia cover
Epilepsia
IF:
6.6
Papers:
1.1W
Citations:
3.2W

Organization

D
Dokkyo Medical University
Scholars:
3.2K
Papers: 2.3K
Citations: 1.1K
N
Nippon Medical School
Scholars:
4.8K
Papers: 4.1K
Citations: 2.7K
H
hokkaido university
Scholars:
4.1K
Papers: 1.5K
Citations: 0
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