A case of Q fever in a sheep producer was detected by a surveillance system in North Dakota in 1993, when Q fever was not reportable. This is the first officially documented case in the state. To estimate the prevalence of Coxiella burnetii infection and identify associated risk factors, we conducted a study covering the whole state. A total of 17 cases were identified among 496 sheep producers, their family members, and hired helpers. The number of sheep raised was a good predictor of C. burnetii infection. Lambing outdoors and frequent physical contacts with sheep during lambing were associated with a higher risk, but petting dogs was correlated with a lower risk. We conclude that C. burnetii infection is prevalent among sheep producers in North Dakota. As the result, Q fever became a reportable disease in North Dakota.