Hyalohyphomycosis caused by Paecilomyces has rarely been described among solid organ recipients. Its management is elusive without an established consensus concerning antifungal therapy. Herein we have reported a case of extensive cellulitis caused by Paecilomyces lilacinus observed in a 48-year-old kidney transplanted woman with hepatitis C. Kidney transplantation from a cadaveric donor was performed in October 2006 with an uneventful early course except for posttransplant diabetes mellitus and a reversible acute rejection episode. Cutaneous nodular and verrucous lesions of the left leg appeared in August 2007. In a few weeks, these lesions become ulcerated, hemorrhagic, and painful. The diagnosis was made on the basis of microbiologic culture and histological examination. There was no improvement in the skin lesions after 6 weeks treatment with itraconazole, but voriconazole yielded a good response within the first 2 weeks. There was a good tolerance to antifungal therapy; graft function and liver tests remained normal. We concluded that an increasing emerging of fungal infections is observed with the introduction of more powerful immunosuppressive drugs. Diagnosis and management of such infections is elusive. Preventive measures should be considered including the adaptation of immunosuppressive therapy among at-risk patients especially those with hepatitis C virus infection and diabetics.