Anti‐GAD65 musicogenic epilepsy: Bilateral and independent mesial temporal seizures revealed by foramen ovale electrodes

• Published in: Epilepsia open Vol. 10; no. 2; pp. 609 - 614
• Main Authors: Di Giacomo, Roberta, Maccanti, Giulia, Gnatkovsky, Vadym, Vatti, Giampaolo, Parente, Annalisa, Dominese, Ambra, Sebastiano, Davide Rossi, Doniselli, Fabio Martino, Andreetta, Francesca, Stabile, Andrea, Deleo, Francesco, Pastori, Chiara, Battaglia, Giulia, Duran, Dunja, Didato, Giuseppe, Del Sole, Angelo, Rizzi, Michele, Curtis, Marco
• Format: Journal Article
• Language: English
• Published: United States John Wiley & Sons, Inc 01.04.2025; John Wiley and Sons Inc; Wiley
• Subjects: Adult; Antibodies; antiGAD65; Autoantibodies - blood; autoimmune‐associated epilepsy; Conflicts of interest; Convulsions & seizures; Drug resistance; Electrodes; Electrodes, Implanted; Electroencephalography; Epilepsy; Epilepsy, Reflex - diagnosis; Epilepsy, Reflex - immunology; Epilepsy, Reflex - physiopathology; Epilepsy, Temporal Lobe - diagnosis; Epilepsy, Temporal Lobe - immunology; Epilepsy, Temporal Lobe - physiopathology; Female; Foramen Ovale; foramen ovale electrodes; Glutamate Decarboxylase - immunology; Humans; Immunology; Localization; Music; Musical recordings; musicogenic epilepsy; Neuropsychology; Patients; Seizures; Short; Surgery; Temporal Lobe - physiopathology; temporal lobe epilepsy; antiGAD65; autoimmune‐associated epilepsy; temporal lobe epilepsy; foramen ovale electrodes; musicogenic epilepsy
• ISSN: 2470-9239; 2470-9239
• DOI: 10.1002/epi4.13132

Musicogenic epilepsy (ME) is characterized by seizures triggered by music. The epileptogenic focus in this rare reflex epilepsy is often in the temporal lobe, although the precise localization is still unclear. A correlation between ME and the presence of GAD65 antibodies indicates a potential immunological pathogenic mechanism. We evaluated a 32‐year‐old woman with drug‐resistant temporal lobe epilepsy as a candidate for epilepsy surgery. In the absence of clear clinical lateralizing signs, video‐EEG monitoring with intracranial electrodes inserted through the foramen ovale was performed to record from the amygdalo‐hippocampal regions. The foramen ovale electrodes revealed bilateral, asynchronous, and independent seizure onsets in the mesial temporal regions triggered by music. Testing for GAD65 antibodies confirmed high‐titer positivity. The efficacy of epilepsy surgery in antiGAD65‐positive ME patients remains limited. We highlight the use of semi‐invasive recording with foramen ovale electrodes in ME, as it can reveal bilateral seizures of mesial origin that contraindicate surgery and support the consideration of immunotherapy options. Plain Language Summary Musicogenic epilepsy is a type of epilepsy in which music triggers seizures. Our understanding of its origin and cause is still limited. We assessed a patient with music‐induced seizures to see if surgery was an option. Since noninvasive tests before surgery were not clear, we used a minimally invasive method with electrodes inserted through a small opening in the skull called the foramen ovale to record the seizures. Thus, we found that the seizures started independently from both temporal lobes, contraindicating epilepsy surgery. We also found high levels of GAD65 antibodies indicating an immunological pathogenic mechanism.