Background: Charles Bonnet syndrome is a rare disorder characterized by complex and recurrent visual hallucinations in elderly patients with visual pathway pa thologic defects. To date, to our knowledge, it has not been described in patien ts undergoing surgical resection for occipital lobe epilepsy due to cortical dys plasia. Objective: To describe a patient who experienced complex visual hallucin ations following resection of cortical dysplasia on the right occipital lobe and who was diagnosed as having Charles Bonnet syndrome. Patient: A 35-year-old w oman underwent surgical resection for medically intractable epilepsy caused by c ortical dysplasia involving the right occipital lobe. Results: Two months after resection of the epileptogenic zone, complex visual hallucinations in the left v isual field not associated with loss of consciousness or delusion developed in t he patient. Hallucinations persisted for more than 12 months despite treatment w ith anti-epileptic medications. During hallucination, no electrographic seizure s were recorded through long-term video-electroencephalographic monitoring. Co nclusions: Charles Bonnet syndrome may occur in a patient with occipital lobe ep ilepsy following resection of the diseased brain with a developmental malformati on. Charles Bonnet syndrome associated with surgical treatment of occipital lobe epilepsy may have been overlooked. Background: Charles Bonnet syndrome is a rare disorder characterized by complex and recurrent visual hallucinations in elderly patients with visual pathway pa thologic defects. To date, to our knowledge, it has not been described in patien ts undergoing surgical resection for occipital lobe epilepsy due to cortical dys plasia. Objective: To describe a patient who experienced complex visual hallucin ations following resection of cortical dysplasia on the right occipital lobe and who was diagnosed with Charles Bonnet syndrome. Patient: A 35-year-old w oman underwent surgical resection for medically intractable epilepsy caused by c ortical dysplasia involving the right occipital lobe. Results: Two months after resection of the epileptogenic zone, complex visual hallucinations in the left v isual field not associated with loss of consciousness or delusion developed in patients. Hallucinations persisted for more than 12 months despite treatment w ith anti-epileptic medications. Dur ing hallucination, no electrographic seizures were were recorded in long-term video-electroencephalographic monitoring. Co nclusions: Charles Bonnet syndrome may occur in a patient with occipital lobe ep ilepsy following resection of the diseased brain with a developmental malformati on. Charles Bonnet syndrome associated with surgical treatment of occipital lobe epilepsy may have been overlooked.