There are conflicting reports regarding the efficacy of trihexyphenidyl, an anticholinergic drug, for treatment of dystonia in cerebral palsy. The author hypothesized that trihexyphenidyl may be more effective in specific subgroups and performed a retrospective analysis of 31 children (8.2 ± 5.8 years) with dystonia following treatment with high-dose trihexyphenidyl (>0.5 mg/kg/day). Main outcome measure was extent of motor improvement calculated according to the body areas affected. Most (21/31) caregivers reported improvement in 1 or more areas, mainly arm, hand, and oromotor function. Improvement was greater in children without spasticity (P = .02) and in those with higher cognitive function (P = .02). While a third of caregivers (10/31) reported tone reduction, and half (15/31) noted overall functional improvement. Side effects were transient, with the exception of hyperopia (n = 1), and occurred less frequently in children with a history of prematurity (P = .02). In summary, trihexyphenidyl is effective particularly in absence of spasticity and in children with higher cognitive abilities.