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Abstract Introduction Pyrenochaeta romeroi (P. romeroi) is a saprophytic fungus found in soil and plants. The fungal spores can be introduced into deeper tissues by trauma. It causes eumycetoma, which affects skin and subcutaneous tissues. Case presentation A 57-year-old South Asian man presented with a painless, nodular lesion (1 cm × 0.5 cm) on the left knee. He had had a renal transplant eight months earlier for end-stage renal failure. The patient was on tacrolimus, mycophenolate mofetil and prednisolone for immunosuppression. The lesion had progressed dramatically (to 5 cm × 5 cm) despite antibiotic treatment. The size and location of the lesion was severely affecting his quality of life, so an excision biopsy was performed. Nuclear ribosomal repeat-region sequencing confirmed the causative organism as P. romeroi. An in vitro antifungal susceptibility test demonstrated that P. romeroi was sensitive to voriconazole. Following a successful surgical removal, voriconazole was continued orally for two months. Conclusion To the best of our knowledge, we are reporting the first case of Eumycetoma caused by P. romeroi in a renal transplant recipient. Physicians should be aware of this rare fungal disease in transplant recipients. We recommend a combination of medical and surgical management in these immunosuppressed patients.