This study reports on histological findings in the temporal lobe of a 36-year-old woman who underwent a right temporal lobectomy for pharmaco-resistant complex partial seizures. Since surgery, the patient has remained seizure-free. The patient had an established diagnosis of right temporal lobe epilepsy, based on video EEG recordings of seizures, MRI hippocampal atrophy, focal interictal hypometabolism on fluoro-desoxyglucose, hypofixation of Cl1-flumazenil in PET studies, and ictal intracerebral recordings. Biopsies were studied under light- and electron microscopy. Histology showed diffuse distribution of a large number of polyglucosan bodies (PBs) in the whole right temporal lobe white matter. PBs were mostly confined to the perivascular areas and in subpial zones rarely and were observed in the most superficial cortical layers. There was some neuronal loss, especially in opercular zone T , but no other histological lesion was found. Ultrastructurally, PBs were made of filamentous and amorphous material, and were found both in intra-astrocytic processes and in axons. The presence of numerous PBs in the temporal lobe of patients with refractory temporal lobe epilepsy has been reported in 3 patients in the literature. It raises the questions whether this histological abnormality could be related to the epileptogenic process as a cause or as a consequence.