Purpose: Acute carbon monoxide (CO) poisoning can lead to delayed neuropsychiatric sequelae (DNS). This study investigated the associations between brain magnetic resonance imaging (MRI) lesions and cognitive domain deficits measured using the Seoul Neuropsychological Screening Battery (SNSB) in patients with DNS. Methods: We retrospectively reviewed consecutive patients diagnosed with DNS after acute CO poisoning between 2011 and 2020 at a single tertiary emergency department. All patients underwent brain MRI and SNSB assessment. Lesions were classified according to location: globus pallidus, periventricular white matter (PVWM), deep white matter, cerebral cortex, and centrum semiovale. Cognitive impairment was evaluated across five SNSB domains. The primary outcome was the association between MRI lesions and the number of impaired cognitive domains. Results: Medical records from 16 patients were reviewed. The mean age was 39.2±12.1 years, and 13 patients (81.2%) were male. Frontal/executive impairment was the most common deficit (75.0%), followed by visuospatial impairment (25.0%), memory impairment (25.0%), language impairment (18.8%), and attention impairment (6.2%). PVWM lesions were associated with impairment in the language (φ=0.71, p<0.05), visuospatial (φ=0.86, p<0.01), and memory domains (φ=0.86, p<0.01); however, the small subgroup size limited the precision of these estimates. Total MRI lesion burden (ρ=0.54, p=0.033) and CO exposure duration (ρ=0.56, p=0.032) were correlated with the number of impaired cognitive domains. Conclusion: Structural lesion burden on MRI appeared to be associated with the extent of functional cognitive impairment in patients with DNS. Patients with high lesion burden, defined as lesions in ≥2 regions, showed a greater mean number of impaired domains than those with low lesion burden. These findings should be interpreted as exploratory because of the small sample size.