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Objective Young children with congenital visual impairment (VI) are at increased risk of behavioral vulnerabilities. Studies on ‘at risk’ populations suggest that frontal EEG asymmetry may be associated with behavioral risk. We investigated frontal asymmetry at 1 year (Time 1), behavior at 2 years (Time 2) and their longitudinal associations within a sample of infants with VI. Frontal asymmetry in the VI sample at 1 year was also compared cross-sectionally to an age-matched typically sighted (TS) group. Methods At Time 1, 22 infants with VI and 10 TS infants underwent 128-channel EEG recording. Frontal asymmetry ratios were calculated from power spectral density values in the alpha frequency band. At Time 2, Achenbach Child Behavior Checklist data was obtained for the VI sample. Results 63.6% of the VI sample and 50% of the TS sample showed left frontal asymmetry; no significant difference in frontal asymmetry was found between the two groups. 22.7% of the VI sample had subclinical to clinical range ‘internalizing’ behavior difficulties. Greater left frontal asymmetry at one year was significantly associated with greater emotionally reactive scores at two years within the VI sample ( r = 0.50, p = 0.02). Conclusions Left frontal asymmetry correlates with later behavior risk within this vulnerable population. Significance These findings make an important first contribution regarding the utility of frontal EEG asymmetry as a method to investigate risk in infants with VI. [ABSTRACT FROM AUTHOR]