Cholinergic–Dopaminergic Imbalance in Pisa Syndrome

Abstract

Pisa syndrome is a rare type of truncal dystonia. Its development is associated commonly with neuroleptic treatment, but there are rare idiopathic cases or those related to neurodegenerative disorders. Recently, an association between cholinesterase inhibitors and Pisa syndrome has been described. The authors report two patients, one with Alzheimer's disease treated with risperidone and another with Parkinson's disease who presented this kind of dystonia after donepezil initiation. In the first patient the condition resolved after discontinuation of risperidone, and in the second one the condition resolved when donepezil was withdrawn. In patients with pharmacologic or degenerative dopaminergic neurotransmission disorders, cholinergic excess may induce this peculiar type of dystonia.

Pearls & Oy-sters: Pisa syndrome

“…The response to anticholinergics and reduction or withdrawal of neuroleptics suggest the possible role of cholinergic excess or imbalance between cholinergic and dopaminergic neurotransmission in the development of this rare syndrome. 6,7 The abnormality in recruitment of truncal muscles or abnormal proprioceptive motor control have also been postulated as major reasons behind Pisa syndrome in Parkinson disease. 5 The marked hyperactivity in the paraspinal muscles on the less affected side is considered another plausible cause for Pisa syndrome in Parkinson disease.…”

Acute Pisa Syndrome as a Neurological Emergency

“…4 Treatment options for Pisa syndrome are empirical and include administration of anticholinergics, agonists of GABA B receptors, benzodiazepines, botulinum toxin application, and discontinuation of the provoking agent. 5 We decided not to use anticholinergics because of the underlying Alzheimer's disease in our patient. Instead, the atypical antipsychotic quetiapine with low antagonism for D 2 receptors was administered in a low dose, resulting in significant clinical improvement.…”

Pisa syndrome in Parkinson's disease: diagnostic and management challenges

“…22 The improvement of PS with deep brain stimulation (DBS) of the pedunculopontine nucleus (PPN) suggests the contribution of the cholinergic system, and supports the hypothesis of the crucial role of dopaminergic-cholinergic imbalance in the pathophysiology of PS. 21,39 Lateral trunk deviation can also occur following pallidotomy. There are two reports of PS developing in patients after pallidotomy, at a time interval of 4-9 years after surgery.…”

“…18 Other drugs associated with PS are typical and atypical antipsychotics, tricyclic antidepressants, selective serotonin reuptake inhibitors, antiemetic drugs, lithium, benzodiazepines, cholinesterase inhibitors, and valproic acid (Table 1). [19][20][21][22][23] In this review, we will focus our attention on PS in patients with PD and provide an update on prevalence, pathophysiology, clinical manifestation, and treatment options.…”