One case of periprosthetic infection after artificial joint replacement caused by Coxiella burnetii was reported. The patient was admitted to hospital due to joint pain and sinus formation after artificial knee replacement. Through medical history, physical examination, imaging examination and gene detection, it was confirmed that the infection around the prosthesis was caused by Coxiella burnetii. The patient was treated with two-stage revision surgery combined with sensitive antibiotics. Through literature review, a total of 9 cases of Coxiella burnetii infection after artificial joint replacement were reported, including 6 cases of hip joint and 3 cases of knee joint; 4 cases had clear pathogen exposure history; only one case was found protheses loosening on imaging; 7 cases with positive IgG antibody against Coxiella burnetii; 7 cases were positive for gene detection; 8 cases were negative in etiological examination, and 1 case was not cultured. All patients were treated with doxycycline and hydroxychloroquine. The serological index of 1 case without Q fever symptom turned negative after treatment. 1 case with multiple system organ dysfunction relieved symptoms after treatment, and serological antibody indicators and nuclear medicine results turned negative. All 7 patients were cured after secondary revision surgery. Coxiella burnetii infection after total joint arthroplasty is rare and lacks typical imaging manifestations and clinical features. The diagnosis of Coxiella burnetii infection after total joint arthroplasty mainly depends on gene detection and serological detection. Two-stage revision surgery combined with sufficient and long-term sensitive antibiotics is the main method of treatment.