A 21-year-old male college student, presented with convulsive status epilepticus, with history of TS since age of 15 years following new-onset seizures, with subsequent diagnosis of TS in his mother, elder sister and his 3-year-old son. He had been seizure-free on Oxcarbazepine for past 2 years, when he started its unsupervised self-taper, leading to breakthrough seizure and current admission. He was treated with continuation of Oxcarbazepine and intravenous Levetiracetam load. The latter had to be switched to Phenytoin due to secondary hyperactive delirium. Brain imaging revealed cerebral tubers, including in right insula, with subependymal periventricular nodules. He did not have convulsive seizures during the hospital stay but had episodic ‘drop attacks’ with bradycardia (30 beats/ minute) 3 days post-admission. Echo ruled out any structural cardiac lesion. Electroencephalogram (EEG) confirmed the bradycardia as ictal. He improved with Valproic acid addition and Phenytoin rapid taper  and was discharged on post-admission day 6.

TS presents commonly with seizures and cardiac manifestations like arrhythmias. It is very uncommon for such patients to have ictal bradyarrhythmia. Insular lesions have been reported to cause arrhythmias including bradycardia but have not been reported to have ictal bradyarrhythmia in TS to the best of our knowledge.

Our case is a very rare presentation of TS in the form of ictal bradycardia from a right insular lesion. It highlights the need to be vigilant about this potentially dangerous complication in TS cases with normal cardiac function.