Drug-Resistant Psychosis in a Tuberous Sclerosis Patient Treated With Electroconvulsive Therapy

J ECT. 2026 May 27. doi: 10.1097/YCT.0000000000001285. Online ahead of print.

ABSTRACT

Tuberous sclerosis complex (TSC) leads to the development of severe neuropsychiatric disorders (TAND), which do not respond to treatment (1). This paper focuses on a 17-year-old male with TSC and uncontrolled epilepsy, which started when he was 1 year old. The surgical removal of his left frontal tuber successfully stopped his seizures (2). Afterwards, his psychiatric condition deteriorated into a treatment-resistant psychosis, which caused him to experience severe agitation and hallucinations. This inverse pattern-gaining seizure control but losing psychiatric stability led us to question whether forced normalization might be the underlying cause. (3,4). Electroconvulsive therapy (ECT) was chosen because pharmacotherapy did not produce a satisfactory response. This decision was made even though the patient had multiple risk factors, including a large postsurgical cortical defect, remaining cortical tubers, and a previous history of status epilepticus (5,6). He received 12 inpatient ECT sessions (10 unilateral, 2 bilateral) and 6 outpatient sessions (18 total), and his post-ECT maintenance was gradually tapered from every 2 weeks to once every 2 months, which successfully eliminated his psychotic symptoms. He experienced no neurological complications. This treatment experience demonstrates that ECT provides a safe method to treat severe TAND psychosis or Force Normalization (FN) psychosis, which does not respond to other treatments, even for patients who have brain structure changes following neurosurgical procedures (4,5).

PMID:42218830 | DOI:10.1097/YCT.0000000000001285