An immunosuppressed heart transplant recipient developed Exophiala jeanselmei infection on the second toe. After unsuccessful treatment with different antifungal drugs, the infection responded to a high-dose regimen of oral terbinafine (an antifungal agent not yet approved in the United States for use against the dematiaceous fungi) and warm packs. This is, to our knowledge, the only known case of successful terbinafine treatment of E. jeanselmei infection.