Ictal Whistling: A Rare Musical Automatism in the Setting of Parahippocampal Focal Cortical Dysplasia
Patrick Hartnett1, Jaideep Kapur1, Ifrah Zawar1
1Neurology, University of Virginia School of Medicine
Background:
Musical automatisms are subtype of mimic automatisms, and include ictal singing, humming, and whistling. There is a paucity of data on ictal whistling, the rarest form, with only 8 cases currently reported. Most have been reported to be a manifestation of temporal lobe epilepsy. However, the exact localization within the temporal lobe remains unclear. Here we describe a unique case of ictal whistling in the setting of left para-hippocampal gyrus dysplasia and amygdala hippocampal enlargement.
Results:
A 64-year-old right-handed female presented with a 20-year history of frequent episodes of uncontrollable whistling, right hand manual automatisms, verbal repetition often with religious content and internal religiosity, and impaired awareness.
Epilepsy monitoring unit admission demonstrated focal seizures arising from the left anterior temporal region. MRI demonstrated cortical dysplasia in the left para-hippocampal gyrus and left amygdala and hippocampal enlargement. At 6 month follow up, she remained seizure-free on lamotrigine monotherapy.
Conclusions:
Ictal whistling has been described as a manifestation of either dominant or nondominant temporal lobe epilepsy. A nondominant hemisphere release phenomenon has also been suggested. However, further localization within the temporal lobe remains unclear. Our patient’s MRI findings, EEG findings, and semiologic features of mystic aura are consistent with dominant temporal lobe onset, in keeping with previous cases of ictal whistling. Interestingly, to our knowledge the neuro-imaging findings of our case may represent the first instance of further localization of ictal whistling within the temporal lobe. This phenomenon in our patient was seen in the setting of left parahippocampal gyrus dysplasia along with amygdala and hippocampal enlargement implying that ictal whistling likely localizes to the antero-mesial temporal structures.