| Summary: | Fungal prosthetic joint infections (PJIs), despite the fact that they are rare, represent a devastating complication. Such infections in revised knee arthroplasties pose a unique surgical and medical challenge. A rare case of <i>Candida parapsilosis</i> PJI in revised knee arthroplasty is reported. Furthermore, a thorough review of all published fungal PJIs cases in revised knee arthroplasties is provided. A 72-year-old female with total knee replacement surgery due to osteoarthritis 10 years ago, followed by two revision surgeries six and two years ago due to aseptic loosening, presented with signs and symptoms of septic loosening of the knee components. Resection arthroplasty and cement-spacer placement was performed and periprosthetic tissue cultures yielded <i>Candida parapsilosis</i>. The patient was commenced on proper antifungal treatment (AFT) for six months and then the second stage of the revision surgery was performed successfully. From 2000 to 2022, a total of 46 patients with median age 69 years [interquartile range (IQR = 10)], suffering fungal PJI occurring in revised knee arthroplasty have been reported. The median time from initial arthroplasty to symptoms’ onset was 12 months (IQR = 14). Cultures of local material (52.2%) and histology (6.5%) were the reported diagnostic method, while <i>Candida</i> species were the most commonly isolated fungi. Regarding surgical management, two-stage revision arthroplasty (TSRA) was performed in most cases (54.3%), with median time-interval of six months (IQR = 6) between the two stages. Regarding AFT, fluconazole was the preferred antifungal compound (78.3%), followed by voriconazole and amphotericin B (19.6% each). The median duration of AFT was five months (IQR = 4.5). Infection’s outcome was successful in 38 cases (82.6%). Fungal PJIs, especially in revised knee arthroplasties, are devastating complications. A combination of AFT and TSRA seems to be the treatment of choice. TSRA in these cases poses a special challenge, since major bone defects may be present. Therapeutic procedures remain unclear, thus additional research is needed.
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