Reversible Pisa syndrome caused by chronic subdural hematoma in a patient with Parkinson's disease: a case report

Abstract

Background: Pisa syndrome (PS), characterized by lateral trunk flexion, is quite common in patients with Parkinson's disease (PD). Patients with PS are older and have a significantly longer disease duration, more severe motor phenotype, ongoing combined treatment with levodopa and dopamine agonists, and higher levodopa equivalent daily dose. We describe here, to the best of our knowledge, the first case of a woman with PD who developed acute-onset PS caused by chronic subdural hematoma (CSDH).

Case presentation: A 70-year-old woman developed acute-onset lateral flexion of her trunk to the left side while standing, and she was admitted to our hospital. One month before, she had a mild head trauma with loss of consciousness. At 65 years of age, she noticed difficulty with walking and clumsiness with her hands. She was diagnosed as having PD (Hoehn and Yahr stage 2) and levodopa was initiated. Her symptoms were markedly improved. At 67 years of age, she developed orthostatic hypotension and was treated sequentially with fluids, compression stockings, and midodrine. Urgently performed brain computed tomography (CT) showed a CSDH in the right hemisphere resulting in a marked compression of the hemisphere. After surgical evacuation, her PS disappeared. She has fully recovered to her preoperative level of function.

Conclusion: The present case provides a valuable insight, that is, the mesial frontal lobe and its connections from the posterior parietal cortex play crucial roles in maintaining the body schema and in the pathophysiology of PS. This case suggests that CSDH should be considered when clinicians examine acute-onset PS, even in patients with neurodegenerative disorders such as PD. Appropriate patient triage and timely neurosurgical intervention should be considered.

Keywords: Body schema; Chronic subdural hematoma; Frontal lobe; Parkinson’s disease; Pisa syndrome.

Fig. 1

Tonic flexion of the trunk to the left was present in the standing position before surgical intervention (a). Presurgical brain CT showing voluminous subdural hematoma in the right convexity with marked compression of the right hemisphere (b). Postsurgical brain CT carried out four days after the operation showing improvement of the right subdural hematoma (c). Seven days after hematoma evacuation, tonic flexion of the trunk disappeared (d)