A pair of monozygotic twins discordant for symptoms of Asperger syndrome was evaluated at the age of 13.45 years using psychometric, morphometric, behavioural, and functional imaging methods. The lower-functioning twin had a smaller brain overall, a smaller right cerebellum, and a disproportionately large left frontal lobe, and manifested almost no differential activation between distractors of high and low-congruence with target visual stimuli. The higher-functioning twin manifested a typically autistic pattern of anterior deactivation and posterior hyperactivation in response to incongruent distractors, overlaid with a typically normal pattern of activation of superior frontal cortex. The morphometric results are consistent with known correlations between brain structure and behaviour in autism, and the physiological results suggest correspondences between structure and function.