A mouse model of ATRX deficiency with cognitive deficits and autistic traits.

ABSTRACT:

Background

ATRX is an ATP-dependent chromatin remodeling protein with essential roles in safeguarding genome integrity and modulating gene expression. Deficiencies in this protein cause ATR-X syndrome, a condition characterized by intellectual disability and an array of developmental abnormalities, including features of autism. Previous studies demonstrated that deleting ATRX in mouse forebrain excitatory neurons postnatally resulted in male-specific memory deficits, but no apparent autistic-like behaviours.

Methods

We generated mice with an earlier embryonic deletion of ATRX in forebrain excitatory neurons and characterized their behaviour using a series of memory and autistic-related paradigms.

Results

We found that mutant mice displayed a broader spectrum of impair

SUBMITTER: Quesnel KM 

PROVIDER: S-EPMC10644498 | biostudies-literature | 2023 Nov

REPOSITORIES: biostudies-literature