Background: The aim of this study is to summarize and improveknowledge regarding a Candida fracture-related infection (CFRI) through asystematic review on the topic, accompanied by a case report.Methods: A systematic review and meta-analysis based on PRISMAstatement were conducted on the CFRI topic. The following combined search termswere used to explore PubMed, Cochrane, and the Embase database: "fungalinfection", "candida", "fracture related infection", "boneinfection", "orthopedic infection", "internal fixation","post-traumatic infection", and "osteomyelitis".Results: Out of 1514 records, only 5 case reports matched theselection criteria and were included. Moreover, a new case of CFRI, notpreviously described, was reported in this paper and reviewed.The main risk factors for CFRI were open wounds (three cases) andimmunodeficiency (three cases).Initial improvement of clinical and laboratory signs of infection was notedin all cases. In the available short-term follow-up (mean 12.1 months;range 3-42), the reoperation rate was 33.3 %.Using a strategy based on extensive debridement/resection methods andprolonged systemic antifungal therapy (mean 8.8 weeks; range 6-18), four of sixcases (66.6 %) were cured. Bone union occurred in three out of six cases.Conclusion: There is very low-quality evidence availableregarding CFRI. Candida infections in surgically treated fractures are rarebut difficult-to-treat events, with a slow onset, unspecific symptoms orsigns, and a significant relapse risk; therefore, they still represent acurrent diagnostic challenge. The existing fracture-related infectiontreatment algorithm combined with long-term systemic antifungal therapy hasan anecdotal value and needs more extensive studies to be validated.