D-cycloserine (DCS) is an anti-tuberculosis medication that has been utilized for years for drug-resistant tuberculosis. DCS works via a centrally acting mechanism which can cause neurotoxic adverse effects which has limited its use. This centrally acting mechanism also allows for DCS to be utilized for various neuropsychiatric purposes. Our patient was on high-dose DCS for autism spectrum disorder and presented to the emergency department (ED) with a seizure. The seizure episode was managed with both anti-epileptics and pyridoxine. With increasing novel use of this older medication, it is imperative for ED clinicians to be aware of the different management strategies that may be required when a patient presents with a neurotoxic effect, specifically seizures, secondary to DCS.
Keywords: cycloserine; emergency medicine; seizures.