Abstract

Background: Forced normalization is the development of psychiatric symptoms in a patient experiencing remission of intractable seizures. The mechanism of this phenomenon is unknown. We present a complex case of Lennox Gastaut syndrome that experiences forced normalization after vagus nerve stimulation (VNS). Methods: This case details a 31-year-old male with seizures since early childhood. The patient has intractable epilepsy and failed AEDs, VNS, and a partial callosotomy. Results: The patient was in remission from 2-12 years old, when seizures returned at a frequency of 2-5 per day. He has multiple types of seizures including drop attacks, absences, and tonic-clonic seizures. Patient experienced status epilepticus multiple times. Twelve AEDs were failed before VNS was started in 2010, which helped curb the severity of seizures and the potential for clusters. Forced normalization developed over the course of treatment with VNS. The patient behavior was characterized by aggression, paranoia, and hallucinations. VNS was turned off late in 2010 and then re-started in January of 2011. Patient proceeded to cycle between several days of seizures without psychiatric symptoms and several days of psychosis without seizures. Conclusions: Vagus nerve stimulation gave way to forced normalization, characterized here as aggressive behaviour and psychosis. Forced normalization is seen commonly after epilepsy surgery, but rarely following VNS.

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