Both auditory brain abnormalities correlated with hearing impairment, which affected approximately 60% of adult <i>Df1/+</i> mice and typically emerged before 6 weeks of age.<h4>Conclusions</h4>In the <i>Df1/+</i> mouse model of 22q11.2 deletion syndrome, abnormalities in central auditory excitability and auditory cortical PV<sup>+</sup> immunoreactivity correlate with hearing impairment. Here, YTHDF1 is linked to 22q11.2 deletion syndrome.