ObjectiveTo clarify the characteristic of secondary cerebral amyloidoma which is relapsing in one year after seven years gamma knife radiosurgery and review relevant literature.MethodsTo analyze the clinical manifestation, preoperative and postoperative MRI imaging, inter-ictal and ictal electroencephalogram (EEG) and histopathological evaluation.ResultsThe patient suffered from epilepsy (mainly autonomic seizure and global tonic-clonic seizure) at the age of 22 and took a gamma knife radiosurgery for right medial temporal epilepsy as the refractory seizures occurred at the age of 36. In her 43 and 44 years’ old, she suddenly found left hemiplegia and mental retardation, the MRI showed right frontal and parietal space-occupying lesion and relapsed after the partial excision respectively, the inter-ictal and ictal EEG displayed persistent slow wave in the right hemisphere and spike wave located in the right posterior temporal and central-parietal, after the surgery, we found amyloid in the histopathological evaluation.ConclusionOne of the delayed complications of gamma knife radiosurgery is secondary cerebral amyloidoma, and partial excision may induced relapsing easily.