Dematiaceous fungi are a cause of a variety of human infections, including phaeohyphomycosis, that may affect patients with solid organ or bone marrow transplants. Exophiala jeanselmei, the most common cause of the pheomycotic cyst/subcutaneous phaeohyphomycosis in the United States, has been shown to cause disease in transplant recipients. We report a lung-transplant patient with relapsing and invasive E. jeanselmei phaeohyphomycosis, who previously had a pheomycotic cyst excised and treated with oral fluconazole. The patient was subsequently treated with re-excision and an 8-month course of oral itraconazole without relapse as to date.