Unexpected pain with electrocortical stimulation in a teenager with temporal encephalocele

Temporal lobe encephalocele has emerged as a potentially unrecognized cause of drug-resistant temporal lobe epilepsy (TLE) that can be effectively treated with epilepsy surgery. Here we present a case in which a 17-year-old male with drug-resistant epilepsy and left temporal encephalocele underwent workup for epilepsy surgery, and experienced unexpected pain with electrocortical stimulation. Stimulation of stereo-EEG electrodes in the left temporal pole resulted in severe, unilateral left-sided facial pain due to inadvertent stimulation of the trigeminal nerve. Stereo-EEG showed seizure onset adjacent to encephalocele with no involvement of mesial temporal structures. A temporal pole resection sparing the mesial temporal structures and repair of the sphenoid bone defect was performed. The patient experienced post-operative seizure freedom, with no loss of language function or sensory deficits in the distribution of the trigeminal nerve. This case highlights temporal encephalocele as a potentially overlooked cause of TLE and underscores the anatomical proximity of the trigeminal nerve to the temporal pole, an important consideration for surgical planning.