Some cases suggestive of a tardive dyskinesia (TD)-induced esophageal movement disorder have been reported. We represent a patient with severe orofacial dyskinesia, an oesophageal movement disorder, and aerophagia following long-term neuroleptic treatment. This 40-year-old mentally retarded man showed signs of oral dyskinesia over 20 years ago, with extension to his limbs and trunk within the last 5 years. He also demonstrated noisy air gulping every several seconds. An abdominal radiograph revealed a big magenblase and dilatated, air-filled segments of large and small intestine without a niveaus. Oesophageal contrastradiography revealed evidence of an oral dyskinesia involving the esophageal musculature and a dilated oesophagus (maximal width: 4.5 cm). Although the oesophageal dilatation was similar to that in patients with a spindle type of achalasia, the oesophageal-cardio junction was sometimes open completely, differing from conditions associated with achalasia. A polygraphic study revealed frequent high-amplitude bursts of microvibration, concurrent with the gulping. The signs of aerophagia and laryngeal dyskinesia decreased 6 days following clonazepam administration. Thus, we suggested that tardive dyskinesia is associated with the laryngeal and oesophageal-induced aerophagia. This is the first report of aerophagia associated with neuroleptic-induced tardive syndrome.