Abstract Background Oriental theileriosis is a tick-borne, protozoan disease of cattle caused by members of the Theileria orientalis -complex. Recent outbreaks of this disease in eastern Australia have caused major concerns to the dairy and beef farming communities, but there are no published studies of the economic impact of this disease. On a farm in Victoria, Australia, we assessed whether oriental theileriosis has an impact on milk production and reproductive performance in dairy cows. Methods Blood samples collected from all 662 cows on the farm were tested using an established molecular test. For individual cows, milk production and reproductive performance data were collected. A clinical assessment of individual cows was performed. Based on clinical findings and molecular test results, the following groups of cows were classified: group 1, with cardinal clinical signs of oriental theileriosis and molecular test-positive for T. orientalis ; group 2, with mild or suspected signs of theileriosis and test-positive; group 3, with no clinical signs and test-positive; and group 4, with no clinical signs and test-negative. Milk production and reproductive performance data for groups 1, 2 and 3 were each compared with those for group 4 using linear and logistic regression analyses, respectively. Results At 100 days of lactation, group 1 cows produced significantly less milk (288 l; P = 0.001), milk fat (16.8 kg; P