Q fever is a globally widespread zoonosis, which has a large number of animal reservoirs. In humans, Q fever is asymptomatic in 60% of cases but may appear in the form of acute or chronic disease. The disease is mostly asymptomatic in animals but may be a cause of abortions or stillbirths. The aim of this study was to present and analyze the epidemiological characteristics of Q fever in Srem district, in the Autonomous Province of Vojvodina, Republic of Serbia. We conducted a descriptive study using data from the registries of infectious diseases of the Institutes of Public Health of Vojvodina and Institute of Public Health Sremska Mitrovica in the period 2011-2020. A total of 76 human cases of Q fever were registered in Srem district over ten-year period. The majority of patients (89.47%; N = 68) were registered in municipalities of Sremska Mitrovica and Šid. One third of patients were hospitalized (32.89%; N = 25). Males were 3.2 times more affected than females (M : F = 3.22 : 1). Working age population (20–59 years) were most affected (82.89%; N = 63) with a significantly higher incidence compared to the age category 0-19 years (p < 0.05). The majority of cases were registered in February (60.53%; N = 46). Probable routes of transmission of Coxiella burnetii were: direct contact with animals and the airborne route of transmission (50.00% vs. 47.37%). The highest seroprevalence of Q fever recorded in goats (55.54%) and sheep (11.88%) in the municipality of Šid, which is connected to outbreak in Kukujevci 2017. The high seroprevalence in goats, recorded in Inđija (22.87%) was not accompanied by Q fever in humans. Q fever still represents a significant problem of both veterinary and human medicine and therefore continuous disease monitoring and more efficient cooperation of the veterinary and human health care sectors is necessary.