Tuberous sclerosis complex is a neurocutaneous disorder characterized by multiple hamartomas of brain, eyes, heart, lung, liver, kidney, and skin. While in the 1/3 of the ones having the disease, a dominant heredity is at issue, in the remaining part, the sporadic mutations have been declared. According to the settlement, number and size of the tubers in the brain, there can be neuropsychiatric symptoms in the patients from the weak situations to the heavy clinics. The psychotic disorder, which is related with the tuberous sclerosis, has been declared even being rare. The 25-year-old female patient applied to the psychiatric polyclinics with the complaints of having congestion in her throat and thinking that there are foreign substances in her stomach, trying to cut her throat with the knife. It was learned from her story that she did not eat almost anything for 1 month due to her belief that there was congestion in her throat. Even though the doctor she applied, tried to make her believe in lack of congestion, she had expressions such as “cut my throat and open the congestion” ; she claimed frogs were touring in her stomach and they disturb her. She said she wanted to get rid of the frogs from her stomach through a hole made with a knife. In her physical examination, on the face, there were symmetrical, angiofibroma-like lesions on the erythematous ground, ungual fibroma and retinal hamartoma, shagreen patch were existing. It was learned that the patient was followed with the diagnosis of tuberous sclerosis and she used valproic acid 1500 mg/g and levetiracetam 2000 mg/g. In the intelligence test, it was determined that she had functionality in the limit intelligence level. In her cranial MR examination, a great number of hamartamatous lesions were observed as being expanded in the frontal, parietal and temporal region and as having cortical and subcortical placement in the brain. It was thought that these lesions were compliant with the tuberous sclerosis. The risperidone 4 mg/g was started to the patient with the diagnosis of the atypical psychosis. In the follow-ups, the patient whose delusions regressed was discharged for continuing her polyclinic follow-ups as outpatient. The tuberous sclerosis is an autosomal dominant neurocutaneous syndrome, which is diagnosed frequently in the childhood age. The prominent neurological characteristics of the syndrome are the mental retardation, seizures and behavioral disorders. However, psychosis can rarely be observed. For this reason, each case for which the diagnosis of tuberous sclerosis has been established, should be examined in terms of the neuropsychiatric situations and should be followed.