BackgroundThe Infant Motor Profile (IMP) is an appropriate tool to assess and monitor infant motor behaviour over time. Infants at very high risk (VHR) due to a lesion of the brain generally show impaired motor development. They may grow into or out of their neurodevelopmental deficit. AimsEvaluate associations between IMP-trajectories, summarised by IMP-scores in early infancy and rates of change, and functional and cognitive outcome at school-age in VHR-children. Study designLongitudinal study. Subjects31 VHR-children, mainly due to a brain lesion, who had multiple IMP-assessments during infancy, were re-assessed at 7–10 years (school-age). Outcome measuresFunctional outcome was assessed with the Vineland-II, cognition with RAKIT 2. Associations between IMP-trajectories and outcome were tested by multivariable linear regression analyses. ResultsWhen corrected for sex, maternal education and follow-up age, initial scores of total IMP, variation and performance domains, as well as their rates of change were associated with better functional outcome (unstandardised coefficients [95% CI]): 36.44 [19.60–53.28], 33.46 [17.43–49.49], 16.52 [7.58–25.46], and 513.15 [262.51–763.79], 356.70 [148.24–565.15], and 269 [130.57–407.43], respectively. Positive rates of change in variation scores were associated with better cognition at school-age: 34.81 [16.58–53.03]. ConclusionOur study indicated that in VHR-children IMP-trajectories were associated with functional outcome at school-age, and to a minor extent also with cognition. Initial IMP-scores presumably reflect the effect of an early brain lesion on brain functioning, whereas IMP rate of change reflects whether infants are able to grow into or out of their initial neurodevelopmental deficit.