ABSTRACTBackground: Cortical lesions in status epilepticus have been reported but the underlying mechanisms are poorly elucidated. Case summary: We report on a female patient (75 years) with a history of alcohol abuse who presented with complex partial status epilepticus and lateralized epileptiform discharges in the left frontal and temporal regions in EEG. While cranial magnetic resonance imaging (MRI) showed left hippocampal T2-hyperintensity and diffusion restriction, cerebrospinal fluid was normal and revealed no limbic encephalitis-related antibodies. Following treatment with levitiracetam, seizures ceased and the patient was dismissed. Nine months later, she was readmitted with generalized status epilepticus. Cranial MRI now showed hippocampal diffusion restriction and T2 hyperintensity, but in the right hemisphere, as well as atrophy and partial gliotic transformation of the initially affected left hippocampus. Discussion: Although hippocampal damage due to antibody-negative limbic encephalitis cannot be ruled out, our observation of subsequent bilateral hippocampal diffusion restriction with gliotic transformation may demonstrate permanent seizure-induced structural brain damage and underlines the importance of further research to elucidate the effects of prolonged epileptic discharges on cerebral structural integrity.