Abstract
IntroductionPallidal DBS is an established treatment for severe isolated dystonia. However, its use in disabling and treatment-refractory tardive syndromes (TS) including tardive dyskinesia and tardive dystonia (TD) is less well investigated and long-term data remain sparse. This observational study evaluates long-term effects of deep brain stimulation (DBS) of the globus pallidus internus (GPi) in patients with medically refractory TS.MethodsWe retrospectively analyzed a cohort of seven TD patients with bilateral GPi-DBS. Involuntary movements, dystonia and disability were rated at long-term follow-up (LT-FU) after a mean of 122 ± 33.2 SD months (range 63–171 months) and compared to baseline (BL), short-term (ST-FU; mean 6 ± 2.0 SD months) and 4-year follow-up (4y-FU; mean 45 ± 12.3 SD months) using the Abnormal Involuntary Movement Scale (AIMS) and the Burke–Fahn–Marsden Dystonia Rating Scale (BFMDRS), respectively. Quality of life and mood were evaluated using the SF36 and Beck Depression Index (BDI) questionnaires, respectively.ResultsAt LT-FU patients had improved by 73% ± 14.2 SD in involuntary movements and 90% ± 1.0 SD in dystonia. Mood had improved significantly whereas quality of life remained unchanged compared to baseline. No serious long-lasting stimulation-related adverse events (AEs) were observed. Three patients of this cohort presented without active stimulation and ongoing symptom relief at long-term follow-up after 3–10 years of continuous DBS.ConclusionPallidal DBS is a safe and effective long-term TD treatment. Even more interesting, three of our patients could stop stimulation after several years of DBS without serious relapse. Larger studies need to explore the phenomenon of ongoing symptom relief after DBS cessation.
Highlights
Pallidal deep brain stimulation (DBS) is an established treatment for severe isolated dystonia
Up to 21% of the patients treated with dopamine receptor blocking agents (DRBA) are estimated to develop tardive symptoms [1]
The longest follow-up, so far, has been reported in 14 tardive dystonia (TD) patients after 6–11 years of globus pallidus internus (GPi)-DBS with an overall 63 and 58% motor improvement measured by the Abnormal Involuntary Movement Scale (AIMS) and extrapyramidal symptom rating scale, respectively [11]
Summary
Tardive syndromes (TS) encompass a broad spectrum of abnormal involuntary movements (AIMs) of the tongue, jaw, trunk and/or extremities emerging after at least 3 months of exposure to dopamine receptor blocking agents, and after treatment with certain antiemetics and antidepressants [1]. Deep brain stimulation (DBS) of the globus pallidus internus (GPi) is an effective treatment for medically refractory dystonia and has progressively evolved into a widely available therapeutic strategy in dystonia as it reduces motor impairment and disability [4]. According to the most recent reviews, 24 single case reports and 6 rather small open-label case series reported GPi-DBS to be a safe and promising treatment option, with improvements between 30 and 90% on disease-specific scales after up to 7 years [6,7,8]. The longest follow-up, so far, has been reported in 14 TD patients after 6–11 years of GPi-DBS with an overall 63 and 58% motor improvement measured by the AIMS and extrapyramidal symptom rating scale, respectively [11]. Long-term outcome as well as information on QoL, mood and side effects of this potentially life-long therapy are of special clinical interest
Sign-in/Register to view highlights & summary Sign-in/Register to access full text options 
Free) 
Talk to us
Join us for a 30 min session where you can share your feedback and ask us any queries you have
Schedule a call
