Immunosuppression for organ transplantation results in increased susceptibility to opportunistic infections including fungal, such as Scedosporium apiospermum. Even though many reported cases of this infection had both local and systemic manifestations, majority of the systemic infections had a fatal outcome. We report a case of a 50-year-old Caucasian male with lymphocutaneous and presumed pulmonary Scedosporium infection 4 years after renal transplantation that was successfully treated with voriconazole and discontinuation of immunosuppression. He received a second transplant 3 years later in the absence of clinical evidence of S. apiospermum infection. Unfortunately, 4 months after transplantation he developed recurrence of the same infection localizing to the soft tissues. Presently this infection is under control with surgical excision and voriconazole therapy. To our knowledge this is the first reported case of recurrent S. apiospermum infection in a renal transplant recipient. We suggest prophylactic antifungal therapy in all re-transplants with this infection.