Brucellosis, caused by a facultative intracellular gram-negative coccobacillus, is one of the most common zoonotic infections worldwide. Very rarely, brucellosis can cause periprosthetic joint infections (PJI). In this case-based literature review, we summarize the current medical literature regarding Brucella PJI, with the aim to raise awareness among clinicians, particularly in non-endemic areas. We report a case of a woman with a 3 weeks history of increasing hip pain, who was eventually diagnosed with Brucella PJI. We searched multiple databases to identify all case reports on Brucella PJI in humans published from February, 1983 to December, 2023. A total of 42 cases from 38 published articles were retrieved and summarized, along with our case. Mean age was 65.5 years, with sex ratio nearly one. Almost all patients presented with local symptoms and 56% with systemic symptoms. Only knee (n=30) and hip (n=13) prosthetic joint were involved. If performed, culture showed positive results on synovial fluid (74%), intra- operative tissue (79%), and/or blood (38%). Serological confirmation was obtained in 97%. Antimicrobial treatment consisted of a combination of doxycycline and rifampin in most cases, with in nearly half of the cases the addition of an aminoglycoside. The majority of patients (n=24) were surgically treated with a two stage exchange arthroplasty; although other options were successful as well. Brucella PJI is a rare but potentially severe manifestation of brucellosis. Brucella PJI must be considered in patients from endemic areas, especially when another causative agent has not been isolated. If culture results remain negative, PCR or serology should be performed. To date, there is no uniform recommendation for the duration of antimicrobial therapy nor the preferred surgical procedure. Relapse is possible even with adequate therapy.