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Hyperkinetic Lingual Movements Resulting from Epileptogenesis: A 13-Patient Cohort Study on Lingual Seizures
Published online by Cambridge University Press: 14 April 2025
Abstract
Objectives:
Lingual seizures are rare hyperkinetic tongue movements with significant clinical implications due to their epileptogenic origin. Despite their diagnostic value, these seizures are often underrecognized, particularly when electroencephalographic (EEG) findings are inconclusive. This study aims to characterize their clinical features, EEG patterns, imaging findings and underlying causes, emphasizing the need for increased awareness and improved diagnosis.
Methods:
A retrospective review identified patients with isolated lingual seizures or those with additional motor involvement. Data on demographics, seizure characteristics, EEG findings, imaging results and underlying causes were collected and analyzed. Seizures were classified based on the International League Against Epilepsy (ILAE) 2017 framework to refine their clinical and diagnostic profiles.
Results:
Thirteen patients were identified: 11 with focal-aware and 1 with focal-unaware seizures. Seven had epilepsia partialis continua, and five experienced frequent seizures. Seizure involvement was limited to the tongue in four cases, extended to cranial muscles in seven and affected the tongue, cranial and extremity muscles in two. Significant ictal EEG findings were noted in only three patients with extensive motor involvement. However, nine patients had acute cerebral lesions, associated with glial tumors, encephalitis, chronic gliosis or cortical hemorrhage.
Conclusions:
This study provides a detailed characterization of lingual seizures, highlighting their clinical, electrophysiological and imaging features. Given their rarity and underdiagnosis, our findings offer valuable guidance for clinicians, underscoring the importance of improved recognition and diagnostic strategies for this distinct seizure type.
Résumé
Les crises linguales sont des mouvements hyperkinétiques rares de la langue ayant des implications cliniques significatives en raison de leur origine épileptogène. Malgré leur valeur diagnostique, ces crises sont souvent sous-estimées, en particulier lorsque les résultats obtenus dans le cadre d’un électroencéphalogramme (EEG) ne sont pas concluants. Cette étude vise donc à définir les caractéristiques cliniques et les causes sous-jacentes de ces crises, de même que les résultats consécutifs à un EEG et les observations effectuées au moyen d’un examen d’IRM, en soulignant la nécessité d’une prise de conscience accrue et d’un meilleur diagnostic.
Une étude rétrospective a permis d’identifier les patients présentant des crises linguales isolées ou une atteinte motrice supplémentaire. Leurs données démographiques, les caractéristiques et les causes sous-jacentes de leurs crises, leurs résultats obtenus lors d’un EEG et les observations effectuées au moyen d’un examen d’IRM ont été recueillis et analysés. Les crises ont ensuite été classées selon le cadre de l’International League Against Epilepsy (ILAE) 2017 afin d’affiner le profil clinique et diagnostique des patients.
Au total, treize d’entre eux ont été identifiés : onze avec des crises focales conscientes et un avec des crises focales non conscientes. De ce nombre, sept d’entre eux souffraient d’épilepsie partielle continue et cinq avaient des crises fréquentes. Les crises se limitaient à la langue dans quatre cas, s’étendaient aux muscles crâniens dans sept cas et touchaient la langue, les muscles crâniens et les muscles des extrémités dans deux cas. Des résultats significatifs lors d’un EEG en phase ictale ont été notés chez seulement trois patients présentant une atteinte motrice étendue. Cela dit, neuf patients présentaient des lésions cérébrales aiguës associées à des tumeurs gliales, des encéphalites, des glioses chroniques ou des hémorragies corticales.
Cette étude a fourni une caractérisation détaillée des crises linguales en soulignant leurs caractéristiques cliniques, électrophysiologiques et en lien avec des examens d’IRM. Compte tenu de leur rareté et de leur sous-diagnostic, nos résultats offrent des conseils précieux aux cliniciens et soulignent l’importance d’une meilleure reconnaissance et de stratégies de diagnostic pour ce type de crise distinct.
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- © The Author(s), 2025. Published by Cambridge University Press on behalf of Canadian Neurological Sciences Federation
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