Electroconvulsive therapy‐associated Pisa syndrome in clozapine treatment

Abstract

PISA SYNDROME is a rare type of truncal dystonia. It is characterized by sustained tonic flexion of the trunk with the head to one side. There have been several reports indicating that this movement side-effect is related to the use of classical or atypical antipsychotics.1 There have been no reports, however, of this movement disorder occurring in the patients who receive electroconvulsive therapy (ECT). We here report a schizophrenic patient whose refractory psychosis did not respond to 400 mg/day of clozapine and who developed Pisa syndrome when she received additional ECT. A 57-year-old female patient had chronic and treatment-refractory schizophrenia for 30 years. She had no past medical or surgical history, and there was no family history of psychiatric disorders. The patient has been hospitalized in the chronic ward at Armed Forces Beitou Hospital, Taipei, since 2003. On 4 April 2006 the patient's symptoms of paranoid thoughts, auditory hallucinations, and violent behavior became exacerbated and she was transferred to the acute ward. The patient was initially treated with clozapine and the dose was gradually titrated up to 400 mg/day, but her psychosis and violence did not respond to the clozapine therapy. As a result, the patient had her first bilateral ECT on 21 April 2006. After the patient had received the 10th ECT she was noted to have symptoms of sustained tonic flexion of her head and trunk, tilting to the right side. Due to the likelihood of ECT-associated Pisa syndrome, her ECT was discontinued. The clozapine dose was reduced from 400 mg/day down to 25 mg/day. Biperiden 2 mg three times a day was prescribed. On this regimen the patient's dystonic symptoms gradually remitted, and clozapine 300 mg/day was then resumed. Subsequent brain computed tomography and electroencephalography showed no abnormalities, and the patient remained in a relatively stable condition on clozapine. The present case suggests that not only classical or atypical antipsychotics can cause Pisa syndrome,1 but also ECT can lead to this unusual movement side-effect. Risk factors for developing Pisa syndrome during neuroleptic therapy include combined pharmacological treatment, old age, female gender, and the presence of organic brain disorder.1 In the present patient, potential risk factors for developing Pisa syndrome included (i) a relatively high dose of clozapine, (ii) old age, (iii) female gender, and (iv) bilateral ECT. Reductions in dose or discontinuation of antipsychotics, and use of anticholinergic agents have been suggested to resolve neuroleptic-associated Pisa syndrome.1 The present patient's Pisa syndrome gradually improved when ECT was stopped, clozapine was tapered and anticholinergic medication was added. We thus think that the Pisa syndrome in the present patient may have been triggered by ECT in the presence of clozapine therapy. Dopaminergic–cholinergic imbalances have been suggested to play a role in the development of antipsychotic-induced Pisa syndrome.1 This raises the possibility that such imbalances are also involved in the underlying mechanisms of ECT-associated Pisa syndrome. Last, we would like to advise that antipsychotic dose should be reduced before use of ECT in the treatment of refractory schizophrenic patients who have risk factors for Pisa syndrome.