OBJECTIVE: The 22q11.2 deletion syndrome (22q11DS) is a neurogenetic syndrome with high risk for the development of psychiatric disorder. There is interest in identifying reliable markers for measuring and monitoring socio-emotional impairments in 22q11DS during development. The current study investigated eye gaze as a potential marker during a face-processing task in children and young adolescents with 22q11DS. METHOD: Eye gaze and behavioral correlates were investigated in 26 subjects (aged 8 to 15 years) with 22q11DS during the Jane Task, which targets featural and configural face processing. Individuals with 22q11DS were compared with chronologically age-matched healthy controls and individuals with idiopathic developmental delay (DD). RESULTS: Few differences in accuracy were observed between patients with 22q11DS and DD controls; however individuals with 22q11DS spent less time on the eyes and more time on the mouths than both comparison groups. IQ predicted time on the eyes in subjects with 22q11DS, and anxiety predicted time on the eyes in DD and 22q11DS subjects. CONCLUSIONS: These results provide evidence for abnormal exploration of faces in the syndrome and suggest that time spent on the eyes may contribute to face processing difficulties and interact with anxiety levels to exacerbate socio-emotional dysfunction in affected individuals.