Electroconvulsive Therapy for Severe Refractory Tardive Dystonia Without Active Depression: A Case Report.

backgroundTardive dystonia is a disabling tardive syndrome associated with exposure to dopamine receptor-blocking agents. Responses to pharmacotherapy are often limited, and evidence supporting electroconvulsive therapy (ECT) remains sparse. CASE PRESENTATION: A woman in her early 60s with recurrent major depressive disorder (in full remission at presentation) developed progressive cervical-truncal dystonia with tremor-like involuntary movements after years of fluctuating psychotropic regimens that included exposure to antipsychotic medication. Neurologic evaluation, including brain magnetic resonance imaging and electroencephalography, was unremarkable, and no structural, metabolic, or neurodegenerative cause was identified from available records. Multiple pharmacologic strategies-including anticholinergics, benzodiazepines, and valbenazine-provided insufficient control, and she became wheelchair-dependent. During a recent psychiatric admission, she received ECT primarily for refractory tardive dystonia. Severity was tracked using the Abnormal Involuntary Movement Scale (AIMS), which was 27 at admission and 25 immediately before ECT. After benzodiazepines were discontinued, she underwent an acute course of bitemporal ECT over approximately 6 weeks under propofol (50 mg) and succinylcholine (50 mg) anesthesia, with a recorded stimulus charge of 100 mC and pulse width of 1.0 ms. Seizure duration on electroencephalography ranged from 25 to 85 s (motor 25-59 s when recorded), and the device/local-record postictal suppression index was 1-2. Motor symptoms improved progressively; by the middle of the course, she no longer required a wheelchair and ambulated independently. The AIMS score decreased to 14 later in the course and remained 14 at completion of the acute course. However, dystonia gradually recurred after ECT discontinuation.

conclusionECT may provide clinically meaningful short-term improvement in severe refractory tardive dystonia even in the absence of active depressive symptoms; relapse after acute treatment may occur, and continuation strategies may be required.