Objective: The purpose of the study was to compare the psychological adjustment of caregivers of children with brain tumors who are on-treatment with caregivers of children who are off-treatment.Methods: Data were collected from 90 participants: 47 (52.2%) caregivers of children undergoing active treatment (on-treatment) and 43 (47.8%) caregivers of children off-treatment on measures of global psychological distress and illness and caregiving related distress.Results: Results revealed that the two groups differed significantly in their reported symptoms of general psychological distress, with the off-treatment caregivers reporting significantly lower levels of general distress. However, off-treatment caregivers continued to experience elevated levels of uncertainty and caregiving burden related to their child’s illness.Conclusions: There is significant evidence suggesting that the burden of caring for a child with a brain tumor is ongoing, continuing well into the off-treatment period. These results also suggest that the psychosocial functioning of these caregivers is best assessed using measures designed specifically to evaluate illness-related psychosocial functioning (e.g. the Parent Experience of Child Illness, Impact on Family Scale). Copyright (c) 2008 John Wiley & Sons, Ltd.