This report describes a rare case presenting with focal epilepsy and focal dystonia associated with glutamate receptor δ2 antibody. The patient was a 47-year-old male patient with neurosyphilis. He presented with an intractable focal seizure spreading from the right arm, with dystonia of the left leg. The IgG antibody of glutamate receptor δ2 was detected. Ictal and interictal SPECT suggested focal epilepsy in the left frontal cortex. Antibiotic and antiepileptic drugs were ineffective, although steroid pulse therapy effectively diminished the patient's symptoms. Inflammatory mechanisms may have contributed to this disorder.
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