Pisa syndrome (PS) is a type of dystonia of rare occurrence, first described in 1972 as an adverse effect of neuroleptic agents. It is used to describe a postural abnormality that includes trunk flexion in the coronal plane and axial rotation, which improves in the supine position.

In this work, we aim to conduct a brief review of Pisa Syndrome aetiology, pathophysiology and treatment.

A non-systematic search was conducted through the PubMed database for “pisa syndrome”. Articles were screened for relevant information on PS aetiology, pathophysiology and treatment.

Pisa syndrome has been associated as an adverse effect of multiple drugs from different classes, mainly antipsychotics, dopaminergic agents and cholinesterase inhibitors. The underlying mechanisms are not yet fully understood. Nevertheless, one of the most consensual hypothesis considers PS as a consequence of a cholinergic-dopaminergic imbalance that can be caused by antipsychotic treatment. Some factors have been associated with increased risk for developing PS such as old age and polypharmacy. PS appears to be better treated with the reduction or interruption of the agent(s) associated with its onset.

Despite its low incidence, Pisa syndrome can occur as a side effect of a number of different medications and the identification of the trigger-drug is fundamental so it can be reduced or interrupted in order to treat this condition.

No significant relationships.

Pisa Syndrome or pleurothotonus is a form of dystonia and often can arise as a side effect of antipsychotic treatment conditioning high morbidity and limiting management options. Despite the fact that the precise mechanism remains unclear, a neurochemical imbalance in dopaminergic and cholinergic transmission but also in serotoninergic and noradrenergic transmission can be a possible pathophysiologic mechanism, which can lead to changes in the axial axis with abnormal posture and marked lateral trunk flexion and abnormal gait.

Regarding a clinical case, the authors intend to review the relevant and current literature on the relationship between psychotropic drugs and Pisa Syndrome.

Description of a clinical case by consulting databases of current and scientifically relevant articles.

The clinical case reports a 48-year-old woman with a history of HIV and Substance Use Disorder, hospitalized for unspecific behavioral changes, characterized by mood changes, self-referential, persecutory and somatic delusional ideas, and delusions of the control of thought. She was medicated with antipsychotics and mood stabilizers, with subsequent development of an acute-onset dystonic condition, characterizing the Pisa Syndrome. In this context, the dose of antipsychotics was lowered and anticholinergics were introduced, with progressive improvement of the clinical picture.

Pisa Syndrome, previously seen as a rare adverse effect, can occur as a dystonic reaction related to the use of psychotropic drugs, so its use should be judicious. Further studies are needed to understand the extent of this association and its pathophysiological mechanisms in order to guide more rigorous therapeutic lines.

No significant relationships.