Sunday, February 21, 2016

Ictal coprolalia

Cerin Daniel,  M. Scott Perry.   Ictal Coprolalia: A Case Report and Review of Ictal Speech as a Localizing Feature in Epilepsy.  Pediatric Neurology.  Article in press.



Recognizing ictal semiology is an essential component to localization of seizure onset, especially in intractable epilepsy where surgical therapies may be beneficial. Ictal speech can be a common component of seizure semiology, but the various forms of ictal speech may have different lateralizing and localizing value. Coprolalia is a very rare form of ictal speech.


We present the case of a 15 year old with seizures characterized by agitation and coprolalia which was medically intractable.


The patient underwent surgical evaluation including video EEG, MRI, and functional neuroimaging. Data indicated onset within the dominant frontal lobe which was further localized using stereo-electroencephalography prior to focal cortical resection.


Ictal coprolalia is a rare presentation of ictal speech. We review the various forms of ictal speech and their value in localizing seizure onset.


From the article:

Coprolalia is a form of involuntary swearing that is used out of social and emotional context. Although it occurs most commonly in individuals with Tourette syndrome, it may be present in other neurological disorders such as traumatic brain injuries, stroke, encephalitis, and, very rarely, epilepsy...

He returned to our clinic at age 12 years for evaluation of possible Tourette syndrome after being treated by mental health providers for coprolalia in the setting of oppositional defiant disorder. Careful history revealed no motor or vocal tics; however, his events were characterized by a sudden onset of downturned pursed lips (chapeau de gendarme), followed by prominent agitation with coprolalia and bilateral manual automatisms. After his coprolalia was recognized to be related to frontal lobe epilepsy, he was treated with maximum doses of oxcarbazepine, zonisamide, lacosomide, and clobazam without remission, prompting an epilepsy surgery evaluation. Presurgical testing revealed a nonlesional magnetic resonance imaging with video EEG demonstrating bifrontal interictal spikes with typical ictal events characterized by bifrontal attenuation with maximum evolution of repetitive spike discharges within the left frontal lobe. Positron emission tomography (PET) demonstrated a focal region of hypometabolism within the left mesial frontal lobe extending to the anterior cingulate gyrus, which was also a region of interictal dipole localization by magnetoencephalography. Language was localized to the left hemisphere.                         .
Given the likelihood of deep mesial frontal localization based on presurgical evaluation and the need to investigate the presence or absence of primary speech (i.e., Broca area) involvement in the epileptogenic zone, we pursued stereo-electroencephalography (SEEG) with depth electrodes over a subdural grid placement. The SEEG approach allows monitoring of deep, difficult-to-access cortical regions and can aid in delineating the epileptogenic network when more diffuse cortical regions may be involved. Depth electrodes were placed within the left anterior insula, left anterior cingulate, body of the left cingulate gyrus, Broca region, left frontal pole, and orbitofrontal surface. SEEG data of typical seizures localized onset within the mesial left frontal lobe at the appearance of typical chapeau facies, followed by involvement of the left anterior cingulate gyrus when coprolalia begins corresponding to the hypometabolic cortex demonstrated by PET. The Broca area was not involved in the ictal EEG pattern. Surgical resection of the region of PET hypometabolism including the left frontal pole and anterior cingulate gyrus was completed without complication and he has remained seizure free since surgery...

 Coprolalia, a unique form of nonpropositional ictal speech, may be a unique localizing and lateralizing feature of frontal lobe epilepsy. Nonpropositional swearing presents as overlearned and automatic expressions, whereas propositional forms of cursing present in a controlled manner, and involve both intact Broca and Wernicke areas.  Although the definitive pathophysiologic mechanism remains unknown, dysfunction in the cortical-striatal-limbic pathways has been implicated to play a role in coprolalia.

There is a very interesting video with the article.

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