Abstract
Background
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.
Methods
We
present the case of a 15 year old with seizures characterized by agitation and
coprolalia which was medically intractable.
Results
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.
Conclusions
Ictal
coprolalia is a rare presentation of ictal speech. We review the various forms
of ictal speech and their value in localizing seizure onset.
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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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