Bluetongue (BT) is a viral disease that affects ruminants and is transmitted by midges of the genus Culicoides spp. The seroprevalence, the clinical form and the occurrence rates significantly differ in relation to several factors such as bluetongue virus (BTV) serotype, host species, breed susceptibility, specific previous exposure, vector ecology, husbandry and health status. Following the 2001-2006 BTV2 and BTV16 epidemics in central Italy, a new epidemic caused by BTV1 occurred in 2013-2015 causing 398 outbreaks in a susceptible population of about 1 million ruminants. The present study assessed the BTV1 seroprevalence in the sheep population of central Italy by conducting two cross-sectional surveys, in the proximity of and within BT outbreak farms. A total of 2,984 sheep from 437 farms were sampled. The animal-level prevalence was 19% (95% CI: 17-21%), the between-herd prevalence was 46% (95% CI: 41-51%) and the within-herd prevalence was 21% (95% CI: 16-26%). Risk factors were investigated by logistic regression models. Living on a farm where an outbreak occurred and the number of outbreaks in proximity of the farm were identified as risk factors, while herd size was identified as a protective factor. This study represents the first BT survey in southern Europe and reports valuable findings on BTV epidemiology. Despite intensive virus circulation, the estimated seroprevalences were low. The assessment of the population immunity level is crucial for defining an efficient vaccination strategy and for predicting the impact of future virus circulation. In view of the low seroprevalence detected albeit an extensive BTV1 circulation, the population immunity was likely to be inadequate in preventing new BTV1 epidemics. Moreover, considering the recurrent introduction of new serotypes from North Africa and the Balkans, the control of multi-serotype BTV infections will continue to present a challenge in the near future.