This is the first report on generalised dystonia beneficially treated by olanzapine. A 63-year-old Taiwanese man with intractable general dystonia was treated with olanzapine starting at a dose of 2.5 mg/day to the maximal dose of 15 mg/day. Clinically, there was slow, but steady, improvement in his dystonia and disability after olanzapine treatment. Evaluation of his dystonia 3 months later revealed 70% improvement on dystonic movement and 60% on disability on the Burke-Fahn-Marsden Dystonia Scale. The result suggests that treatment with olanzapine can be an alternative therapy for patients with generalised dystonia.