Rubinstein-Taybi Syndrome (RSTS) is recognized with features such as broad thumbs, broad big toes palate problems, psychomotor growth deficiency and facial dysmorphism. Impaired oral and dental hygiene about oral care, crowding teeth, malocclusion, multiple caries and talon cusps are frequently observed. 27-year-old patient diagnosed as having RSTS, was admitted to the psychiatry clinic because of psychiatric complaints, high fever and general deterioration. Significant improvement was observed in all complaints after caries and oral hygiene treatment. A 27-year-old female patient was diagnosed with RSTS. When she was 15 years old, aggressive behaviors, deterioration in impulse control, insomnia, mood disorders, visual hallucination and agitation were observed and significant improvement was observed after valproic acid 500 mg/day and risperidone 2 mg/day treatment. 5 months ago increase in fever, difficulty in walking and speaking, nausea, shivering, agitation attacks and fatigue was observed. Her fever was 38-39 and he was admitted to university hospital infection diseases clinic regarding fever etiology. Although detailed evaluations such as cranial MR, abdomen BT abdomen USG, routine biochemistry, blood and urine blood culture and peripheral smear were carried out during 2 months admission, she was discharged from the clinic as a result of ongoing high fever and clinic complaints with “fever of unknown origin” diagnosis. She was admitted to psychiatry clinic because it was evaluated that patients who could not be treated in clinical symptoms could display psychiatric behaviors such as agitation attacks and insomnia. The treatment of the patient who was taking valproic acid 500 mg/day and risperidone 2 mg/day until the admittance was changed as risperidone 2 mg/day, lorazepam 2 mg/day and propranolol 40 mg/day on the first day of admission. During the first week of admittance, significant improvement was not observed in her general medical situation and psychiatric complaints. Because of the evaluation carried out in our clinic regarding routine oral and dental hygiene, tooth extraction was carried out because of dental caries. After 5 days from tooth extraction, high fever disappeared dramatically; decrease in agitations and attacks were observed. On the 2nd week of admittance, her medical treatment was organized as lorazepam 1 mg/day and propranolol 20 mg/day because it was evaluated that clinical improvement was related with tooth infection. On the 20th day of her admittance, the patient was discharged thanks to general improvement in her situation. On oral appearance of RSTS multiple dental anomalies and dental caries are observed. In these patients, rates of increased dental caries differ at the rate of 15-36% and oral hygiene is very important for treatment and tracking of patients. In our case, dramatic improvement was observed in all complaints after treating dental caries in our patient, who was having high fever, deterioration in general situation and psychiatric complaints as a result of infection caused by dental caries. General physical examination is very important because patients have trouble in expressing their complaints. With this case, it was aimed to express the importance of oral care and hygiene in patients whose cognitive functions are impaired.