Objective: We present a case of a patient with Parkinson’s disease who developed “malignant Deep Brain Stimulation (DBS) Withdrawal Syndrome” following the removal of the pulse generator (PG) due to infection.

Background: STN-DBS is an established therapy for advanced Parkinson’s disease, providing sustained improvement of motor symptoms. Complications such as hardware infection or technical issues may necessitate temporary cessation of stimulation. In such cases, patients may experience a syndrome known as “malignant STN-DBS withdrawal syndrome,” characterized by severe non-Levodopa responsive Parkinsonism, elevated body temperature, and rhabdomyolysis. Because early reinstalling stimulation might be impossible due to infection, the therapeutic options are limited. These may include escalating dopaminergic medication, administering Amantadine and providing intensive care support. However, optimal management remains uncertain.

Method: Case Report

Results: A 63-year old patient with akinetic-rigid Parkinson’s disease and STN-DBS since 2016 and concomitant Levodopa therapy, presented with a slowly progressive cutaneous infection, leading to its temporary removal. Despite immediate increase of Levodopa from 300mg to 2150mg per day, a rapid deterioration of the patients’ motor functions was observed to the extent of complete dependency in the activities of daily living and serious dysphagia. Elevated creatine kinase levels up to 789U/l and slightly increased body temperature (37.1°C) and C-reactive protein were noticed. Amantadine infusion and increase of Levodopa were ineffective. Only after the establishment of the novel Catechol-O-Methyltransferase (COMT) Inhibitor Opicapone a significant and persistent improvement was achieved reflected in an improvement of mobility, swallowing and self care.

Conclusion: Adding a COMT inhibitor like Opicapone improved STN-DBS withdrawal syndrome significantly. This observation might have different explanations: a) 2150mg of Levodopa were not enough and Opicapone just increased dopamine availability b) Opicapone has an additional effect on dopamine transmission beyond the sole increment of dopamine availability. Further observations are needed.

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MDS Abstracts - https://www.mdsabstracts.org/abstract/a-case-of-malignant-deep-brain-stimulation-withdrawal-syndrome-treated-with-opicapone/