To the Editor: Tardive dystonia syndromes are well known complications of prolonged treatment with antipsychotic medications, especially the classic ones. Pisa syndrome, a sustained dystonia of the trunk and head to one side, is considered as an atypical form of tardive dystonia and sometimes can be acute.1 Several cases have also been reported in patients taking acetylcholinesterase inhibitors (ACEIs) for at least 1 month.2 We report a patient suffering from Alzheimer’s disease (AD) who presented with acute Pisa syndrome after administration of a single donepezil dose.