Background: Candida prosthetic joint infection (PJI) is a rare complication of total knee arthroplasty (TKA). The literature regarding its diagnosis and management is scarce. Case summary: We present the case of a 79-year-old woman with history of TKA and femoral intramedullary nailing, who developed PJI caused by Candida lusitaniae. A two-stage exchange arthroplasty was performed. She underwent implant removal, debridement and placement of temporary antibiotic impregnated spacer. Postoperatively, the patient received treatment with micafungin for 24 weeks. Eleven months after the first stage, the spacer was removed and a new knee prosthesis was placed. Complete eradication of infection was demonstrated by negative culture of cement spacer, synovial fluid and periprosthetic tissue. Histology of surgical samples was unremarkable. Empiric treatment with micafungin was given for 6 weeks after placement of the new prosthesis. At 6-month follow-up, there was no evidence of recurrent infection. Conclusion: To our knowledge, this is the first reported case of Candida lusitaniae PJI.