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.

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.

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.

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.