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Open Access Case report

Reversible Pisa syndrome associated to subdural haematoma: case-report

Pasquale Marchione12*, Aldo Spallone1, Marcella Valente2, Cristiano Giannone1, Floriana De Angelis1 and Giuseppe Meco13

Author Affiliations

1 Department of Clinical Neurosciences, Neurological Center of Latium – Institute of Neurosciences, Via Patrica 15, 00178 Rome, Italy

2 Department of Medical and Surgical Sciences and Biotechnologies – Section of Neurology, Sapienza, University of Rome, Viale dell’Università 30, 00185 Rome, Italy

3 Parkinson’s Centre and Research Centre of Social Diseases (CIMS), Department of Neurology and Psychiatry, Sapienza University of Rome, Viale dell’Università 30, 00185 Rome, Italy

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BMC Neurology 2014, 14:149  doi:10.1186/1471-2377-14-149

Published: 14 August 2014

Abstract

Background

Pisa Syndrome or Pleurothotonus is a relatively rare truncal dystonia, characterized by tonic flexion of the trunk and head to one side with slight rotation of the body. Since frequently associated to specific drugs such as antipsychotics and cholinesterase inhibitors or to Parkinson Disease, a pathophysiological role of cholinergic-dopaminergic imbalance has been suggested. We report here the first case of Pisa Syndrome due to an extracerebral pathology as subdural haematoma.

Case presentation

A hypertensive patient was admitted to Our Department for subacute onset of tonic flexion and slight rotation of the trunk associated to progressive motor deficit in left upper limb after a mild head trauma without loss of consciousness occurred around three month before. No previous or current pharmacological interventions with antidepressant, neuroleptic or anticholinergic drugs were anamnestically retrieved. Familiar and personal history was negative for neurological disorders other than acute cerebrovascular diseases. Acutely performed cerebral MRI with DWI showed a voluminous right subdural haematoma with mild shift of median line. After surgical evacuation, both motor deficit and truncal dystonia were dramatically resolved. At one-year follow up, the patient did not develop any extrapyramidal and cognitive signs or symptoms.

Conclusions

According to many Authors, the occurrence of truncal dystonia during several pharmacologic treatments and neurodegenerative disorders (such as Alzheimer disease and parkinsonian syndromes) supported the hypothesis that a complex dysregulation of multiple neurotransmitter systems are involved. We suggest a possible role of basal ganglia compression in pathogenesis of truncal dystonia by means of thalamo-cortical trait functional disruption and loss of proprioceptive integration. A further contribution of the subcortical structure displacement that alters motor cortex connectivity to basal ganglia may be postulated.

Keywords:
Pisa Syndrome; Subdural Haematoma; Parkinsonism; Cholinergic-dopaminergic imbalance; Proprioceptive integration; Motor cortex connectivity