Background: Schizophrenia patients have difficulty inhibiting automatic saccades. Many studies have failed to resolve whether healthy first-degree relatives share the same deficit. Measures of brain activity may be more sensitive than behavioral measures. In patients, the saccadic inhibition deficit has been related to impaired frontostriatal functioning. This study attempts to establish whether this abnormality is also present in unaffected relatives of patients. Methods: Functional brain images were acquired during prosaccades and antisaccades in 16 control subjects and 16 unaffected siblings of schizophrenia patients using an event-related functional magnetic resonance imaging design. Eye movements were measured during scanning. Results: The task activated a network of regions corresponding to the oculomotor system. Siblings and control subjects did not differ during execution of prosaccades. During antisaccades, siblings did not activate the caudate nucleus. Siblings and control subjects did not differ on the percentage of antisaccade errors. Conclusions: Siblings did not appropriately activate the striatum during antisaccades, similar to what has been reported in patients. Siblings, however, did not make significantly more errors during antisaccades, indicating that they were able to compensate for the inactive caudate. Future research is needed to assess the potential of this striatal deficit as (genetic) risk factor for schizophrenia.