Mohammadi-Asl, AliBahadori, Amir RezaSabzgolin, ImanDavari, AfshanRazmafrooz, MohammadTafakhori, AbbasRanji, Sara2025-05-312025-05-3120252050-090410.1002/ccr3.70444https://doi.org/10.1002/ccr3.70444https://hdl.handle.net/20.500.14517/7914Musicogenic epilepsy (ME) is a rare form of reflex epilepsy with a prevalence of 1 in 10,000,000. Recent research suggests a potential link between ME and autoimmune encephalitis, particularly involving glutamic acid decarboxylase 65-isoform (GAD65) antibodies. A 48-year-old female presented with a one-year history of music-triggered seizures. Her episodes were characterized by an initial aura followed by unresponsiveness and oral automatisms. Electroencephalography revealed abnormalities in the left anterior temporal lobe and temporal leads. Laboratory studies showed positive anti-GAD65 antibodies. The patient was treated with a combination of antiepileptic medication (Lamotrigine) and corticosteroids and intravenous immunoglobulin. This case contributes to the growing evidence supporting an association between ME and autoimmune mechanisms, particularly GAD65 antibody-mediated autoimmunity. It highlights the importance of screening autoimmune factors in ME patients and highlights the need for further research into targeted treatment strategies.eninfo:eu-repo/semantics/openAccessEncephalitisEpilepsyGad65SeizuresArticleN/AN/A135WOS:00147276470000140291565