We examined the reliability, validity and factor structure of the Eye Contact Avoidance Scale (ECAS), a new 15-item screening tool designed to measure eye contact avoidance in individuals with fragile X syndrome (FXS). Internal consistency of the scale was acceptable to excellent and convergent validity with the Social Responsiveness Scale, Second Edition (SRS-2) and the Anxiety, Depression, and Mood Scale (ADAMS) was good. Boys with a comorbid ASD diagnosis obtained significantly higher scores on the ECAS compared to boys without ASD, when controlling for communication ability. A confirmatory factor analysis indicated that a two-factor model (avoidance and aversion) provided an excellent fit to the data. The ECAS appears to be a promising reliable and valid tool that could be employed as an outcome measure in future pharmacological/behavioral treatment trials for FXS.