Pleurothotonus

Abstract: Long-term administration of antipsychotics occasionally produces persistent dystonia of the trunk, a disorder known as Pisa syndrome (or pleurothotonus). The development of Pisa syndrome is most commonly associated with prolonged treatment with antipsychotics; however, it has also been reported, although less frequently, in patients who are receiving other medications (such as cholinesterase inhibitors and antiemetics), in those not receiving medication (idiopathic Pisa syndrome) and in those with neurodegenerative disorders.

Abstract: Background Pisa syndrome is usually regarded as a rare adverse event of neuroleptic medication. However, its frequency and predisposing factors have yet to be defined. Here, we investigated risk factors of Pisa syndrome occurring in a large population of psychiatric patients surveyed during a multicenter drug safety project. Method Twenty episodes of Pisa syndrome were documented in 17 patients within a population of 45,000 psychiatric patients monitored by a multicenter drug safety surveillance project (Projekt zur Uberwachung der Arzneimittelsicherheit in der Psychiatrie) between 1990 and 1997. All results were related to the epidemiologic data provided for this population and systematically analyzed regarding history of medication, current medication, comedication, and clinical course. Results A constellation of putative risk factors was found in the majority of patients: previous treatment with classical neuroleptics, combined pharmacologic treatment, female gender, old age, and the presence of an organic brain disorder. Given these risk factors, Pisa syndrome was also documented with atypical neuroleptic drugs such as clozapine, olanzapine, and sertindole. Conclusion We conclude that Pisa syndrome is a very rare adverse event occurring with neuroleptic treatment. In patients exhibiting the reported constellation of risk factors, neuroleptic drugs should be administered with particular caution.

Abstract: Dystonic lateroflexion of the trunk, also referred to as Pisa syndrome, pleurothotonus or a lean to the side, was originally described in association with prior exposure to neuroleptics. However, axial deformities (Pisa syndrome, camptocormia and antecollis) are also well recognised but poorly understood features of multiple system atrophy or late-stage Parkinson’s disease. Here, we report on three patients with longstanding Parkinson’s disease who, 4–9 years after a left pallidotomy, developed a Pisa syndrome to the right. The first patient, now 72 years old, was diagnosed with Parkinson’s disease at age 44 years, after initially presenting with pain in his right arm and leg. The right side always remained the more affected and the dyskinesias that developed after 4 years of levodopa treatment were also more pronounced on the right side. Because of progressive motor fluctuations, a left-sided pallidotomy was performed after 17 years of disease, which resulted in abolition of the right-sided dyskinesias and an improvement in the tremor and rigidity on the right. Eight years after the pallidotomy, 25 years after disease onset, he gradually developed a lean to the right, which showed some diurnal fluctuation and responded modestly to dopaminergic treatment. When “on”, he still remains independent for most daily activities. Parkinson’s disease dementia has recently been diagnosed. In the second patient, now 63 years old, Parkinson’s disease was diagnosed at the age of 47 years when he …

Abstract: Three Alzheimer's disease patients with Pisa syndrome were presented. These patients were treated with cholinesterase inhibitors within a post-marketing surveillance study. The clinical and pharmacological histories of these patients suggest a relation between cholinesterase inhibitors and the pleurothotonus. The diffuse use of cholinesterase inhibitors in the treatment of dementia may cause an increasing number of Pisa syndrome cases.