Abstract Details

Title Ictal Whistling: A Rare Musical Automatism in the Setting of Parahippocampal Focal Cortical Dysplasia

Topic Epilepsy/Clinical Neurophysiology (EEG)

Presentation(s) P3 - Poster Session 3 (5:30 PM-6:30 PM)

Poster/Presentation
Number 1-004

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.

Objective NA

Design/Methods NA

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.

Authors/Disclosures
Patrick Hartnett, MD PRESENTER	Dr. Hartnett has nothing to disclose.
Jaideep Kapur, MD, PhD (UVA Neurology)	The institution of Dr. Kapur has received personal compensation in the range of $500-$4,999 for serving on a Scientific Advisory or Data Safety Monitoring board for Marinus. Dr. Kapur has received personal compensation in the range of $5,000-$9,999 for serving as an officer or member of the Board of Directors for Robert Wood Johnson Foundation . Dr. Kapur has received personal compensation in the range of $500-$4,999 for serving as an Editor, Associate Editor, or Editorial Advisory Board Member for Elsevier. The institution of Dr. Kapur has received research support from NIH. Dr. Kapur has received intellectual property interests from a discovery or technology relating to health care. Dr. Kapur has a non-compensated relationship as a Chair, Board North America with International League against Epilepsy that is relevant to AAN interests or activities. Dr. Kapur has a non-compensated relationship as a Board of Directors with American Epilepsy Society that is relevant to AAN interests or activities.
Ifrah Zawar, MD (Cleveland Clinic)	The institution of Dr. Zawar has received research support from Alzheimer's association. The institution of Dr. Zawar has received research support from American epilepsy society .