AbstractBackgroundAs research moves from understanding the natural history of frontotemporal dementia (FTD) to therapeutic trials, reliable and valid psychological measures are required to assess the efficacy of treatments. Current psychometric tests have only been able to identify subtle changes in genetic FTD around 5 years before the onset of symptoms suggesting more sensitive tasks are required.MethodThe aim of this study was to investigate whether an anti‐saccade eye‐tracking task could detect impaired executive function in a cohort of individuals that are at‐risk of developing genetic FTD. A total of 18 mutation carriers (MCs) and 20 mutation negative control participants completed the task, being instructed to look in the opposite direction to the red dot when it appeared on the screen. There were eight trials in the horizontal direction at 5 and 8 degrees of visual angle (°VA) and four trials in the vertical condition at 5°VA. The number of correct and self‐corrected anti‐saccades was calculated and a linear regression, covarying for sex, was used to assess performance between mutation carriers and controls.ResultOf the 18 mutation carriers, 8 had expansions in chromosome 9 open reading frame 72, 6 had progranulin mutations and 5 had mutations in microtubule‐associated protein tau. The mean age for the mutation carriers group overall was 33.2 years, and 34.3 for the control group. The mutation carriers had significantly fewer correct anti‐saccades than the control group when the stimuli was presented in the vertical direction (MCs: mean = 1.44, SD = 1.29; controls: 2.32, 0.95; p = 0.016). There were no significant differences observed in the number of correct anti‐saccades in the horizontal direction, nor in the number of self‐corrected anti‐saccades made in either direction.ConclusionThis preliminary study suggests that early executive function deficits can be detected presymptomatically in FTD using an anti‐saccade eye‐tracking task. It is possible that tasks like this, using eye‐tracking with very simple instructions, may be able to help assess the efficacy of therapeutic trials in the near future.