Objective To determine the psychological consequences for parents of children with Down's syndrome of having received a false negative result on prenatal screening. Design Comparison of adjustment of parents who received a false negative result with that of parents not offered a test and those who declined a test. Setting Parents were interviewed in their own homes. Participants Parents of 179 children with Down's syndrome (mean age 4 (range 2-6) years). Main outcome measures Anxiety, depression, parenting stress, attitudes towards the child, and attributions of blame for the birth of the affected child. Results Overall, regardless of screening history, parents adjusted well to having a child with Down's syndrome. Compared with mothers who declined a test, mothers in the false negative group had higher parenting stress (mean score 81.2 v 71.8, P = 0.016, 95% confidence interval for the difference 1.8 to 17.0) and more negative attitudes towards their children (124.9 v 134.2, P = 0.009, -16.2 to -2.4). Fathers in the false negative group had higher parenting stress test scores (77.8 v 70.0, P = 0.046, 1.5 to 14.2) than fathers not offered a test Mothers in the false negative group were more likely to blame others for the outcome than mothers who had not been offered the test (28% v 13%, P = 0.032, 3% to 27%). Mothers and fathers in the false negative group were more likely to blame others for this outcome than parents who had declined a test (mothers 28% v 0%, P = 0.001, 19% to 37%; fathers 27% v 0%, P = 0.004, 17% to 38%). Blaming others was associated with poorer adjustment for mothers and fathers. Conclusions A false negative result on prenatal screening seems to have a small adverse effect on parental adjustment evident two to six years after the birth of an affected child.