Management of Pisa syndrome with lateralized subthalamic stimulation
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Pisa syndrome (PS) describes a postural abnormality characterized by lateral trunk flexion greater than 10° in the upright position that reverts in the supine position or with passive mobilization . PS was initially described as truncal dystonia in patients using typical neuroleptics , but it can be triggered by other drugs such as cholinergic agents or dopamine agonists. PS can also occur in neurodegenerative disorders such as Parkinson’s disease (PD) . Even though basal ganglia abnormalities associated with asymmetric paraspinal dystonia appear to be necessary for PS, other factors such as vestibular imbalance, proprioceptive disintegration, and myopathy could also play a role [4, 5].
Pisa syndrome can be reversible in early phases when a triggering factor is identified and discontinued. Otherwise, PS can become chronic, severe, and treatment resistant. In these cases, botulinum neurotoxin (BoNT) injections and deep brain stimulation (DBS) of subthalamic (STN) or...
The authors thank the patient and his family. KJL thanks the Dystonia Medical Research Foundation Canada for their 2018 Clinical Fellowship Grant.
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Conflicts of interest
On behalf of all authors, the corresponding author states that there are no relevant conflicts of interest.
The authors declare that the work documented in this manuscript has been carried out in accordance with ethical standards.
Video 1. Pisa syndrome improvement with lateralized subthalamic neuromodulation. Bilateral subthalamic stimulation was maintained at a pulse width of 60 microseconds and a frequency of 80 hertz. Amplitude of right subthalamic stimulation was maintained at 4.8 volts (V) (monopolar contact three). Right lateral truncal deviation worsened when left subthalamic voltage was increased to 4.8 V (Segment 1) and progressively improved when it was reduced to 4.0 V (Segment 2) and 3.8 V (Segment 3) (monopolar contact six). (MP4 18508 KB)