OBJECTIVE To report a case of probable domperidone-induced tardive dyskinesia and withdrawal psychosis in an elderly woman. METHODS A 75-year-old woman was first assessed for cognitive decline and personality changes. On clinical examination, diffuse choreoathetoid movements were noted. Chronic domperidone use seemed the most likely cause for the movement disorder and was abruptly discontinued. Within a few days, she developed a severe psychotic syndrome with Capgras delusions; the movement disorder continued unabated. Both the movement disorder and psychotic symptoms promptly remitted with risperidone treatment. CONCLUSIONS Domperidone has, on rare occasions, been associated with acute extrapyramidal syndromes, especially in young children, but, to our knowledge, this is the first case of domperidone-induced tardive dyskinesia and withdrawal psychosis, adverse effects associated with chronic central nervous system dopaminergic blockade. Domperidone is a dopamine D₂ receptor antagonist that does not usually cross the blood-brain barrier. However, blood-brain barrier alterations occur with aging and dementia, which may explain central nervous system penetration of domperidone in our patient, diagnosed with probable frontotemporal dementia. The association was rated as probable on the Naranjo probability scale. CONCLUSIONS Chronic use of domperidone may, on rare occasions, induce neuropsychiatric syndromes similar to those seen with the use of antipsychotics. This may be more likely in situations in which the blood-brain barrier is damaged, as in vascular and degenerative dementias.