Ictal vocalization varies from simple moaning or grunting noises to complex repetitive utterances and phrases. Ictal whistling is a rare phenomenon with intricate vocal and motor components. The semiology has not been shown to have reliable localizing or lateralizing value.

We present stereoelectroencephalography (sEEG) case of ictal whistling during mesial temporal structures involvement.

A 21-year-old right-handed man presented with seizures starting at age 10 years. His habitual seizures were of intense fear sensation, deep breathing, humming, and whistling with variable awareness. Brain MRI demonstrated left medial occipital encephalomalacia and a left middle cranial fossa arachnoid cyst. Scalp EEGs were poorly localizing, with some suggested left posterior onset. Subtraction ictal SPECT co-registered to MRI and MEG, were inconclusive. SEEG with 14 intracranial electrodes exploring the left temporal, frontal, insular, and occipital head regions was pursued to delineate the seizure onset and determine candidacy for resective surgery. Typical clinical seizures and seizures with minimal symptoms were recorded. The ictal onset localized within the left occipital encephalomalacia in the lingual and mesial occipital gyrus The sEEG data indicated involvement of the left mesial temporal region with ictal whistling.

Our case demonstrates that ictal whistling may be one of the unusual ictal vocalizations of mesial temporal involvement. This clinical vignette included multilobar hemispheric exploration and support temporal lobe localization for ictal whistling with intracranial monitoring