Sandfly fever is an infectious disease transmitted to people through sandfly bites. It usually takes three days and causes chills, high fever, headache, nausea-vomiting and myalgia. The causative agent, namely sandfly fever virus (SFV), is a member of the Bunyaviridae family, Phlebovirus genus. Toscana virus (TOSV) is a serotype of SFV, as so Sicilian and Naples viruses. Seroprevalence studies have demonstrated that SFV infections which have mild symptoms or asymptomatic, can be overcome. Studies concerning TOSV infections in Turkey are limited to a small number of regional seroprevalence surveys, blood-donor screening studies and detection of viral RNA in previously collected cerebrospinal fluid samples of suspected meningoentephalitis patients in whom no causative agents were identified. In this report from Turkey, the first acute case of TOSV infection diagnosed in a patient with HIV seropositivity, was presented. A 42-year-old male patient was admitted to Numune Research and Training Hospital Adana, Turkey with high fever, headache and malaise. The patient who lived in an area near to a forest in Istanbul, had no contact history with ticks, mosquitoes and other animals. He stated that he had had the symptoms before arriving to Adana. The patient was hospitalized due to leucopenia, anemia, and thrombocytopenia accompanying high fever. Serum samples were sent to National Arbovirus and Viral Zoonotic Diseases Unit of the Turkish Public Health Institute, for the detection of Crimean-Congo haemorrhagic fever (CCHF) virus and SFV. Western Blot test was run to confirm the presence of anti-HIV antibodies detected twice with ELISA. In the following days, the patient's fever and symptoms decreased, and thrombocyte levels increased. Although CCHF virus PCR and ELISA IgM tests as well as SFV IgM and IgG immunofluorescence antibody (IFA) tests were negative, real time reverse transcriptase PCR test yielded a positive result for TOSV. SFV IgG antibodies against Toscana and Naples viruses were found to be positive in the serum sample collected at the end of a three-week follow-up. Even though TOSV infection is usually known to have an asymptomatic clinical course, it may rarely lead to serious manifestations like meningoencephalitis. In our country where SFV is endemic, TOSV should be considered in the differential diagnosis of patients presenting with high fever and meningoencephalitis symptoms.