Objectives The aim of this report is to describe a case of GAD-65 autoantibody associated epilepcy, diagnosed long before the onset of autoimmune diabetes. Case presentation This report presents a 36-year-old female with type 1 diabetes, diagnosed at the age of 26, and a cryptogenic focal epilepsy with complex partial seizures, with duration of 2–3 min and frequency of 5–6 per month, diagnosed at 16 years of age. Electroencephalography revealed epileptiform abnormalities temporally and centro-parietally on the left and temporally on the right with forward propagation on both sides. Due to the drug refractory seizures, titers of GAD-65 autoantibodies were examined (19 years after the diagnosis of epilepsy and 9 years after the diagnosis of diabetes) and were found to be elevated in serum and cerebrospinal fluid, strongly supporting its autoimmune genesis. Insulin pump therapy was used in this patient with a beneficial effect on glycemia. Conclusions Autoimmune epilepsy is a clinical entity and should be taken into consideration in patients with other autoimmune diseases, especially diabetes, and with drug refractory seizures, even preceding the onset of diabetes. Achieving stable glycemic control, including the usage of the new technologies in type 1 diabetes treatment, is vital in these cases.
