We describe a 63-year-old man who presented with painful malodorous lesions in the perianal, perineal and scrotal regions. Following definitive diagnosis of paracoccidioidomycosis, he was treated initially with trimethoprim/sulphamethoxazole, but there was no clinical improvement. He then received terbinafine (Lamisil) 250 mg twice daily for 6 months. There was rapid resolution of all lesions and complete relief of symptoms, without any associated side-effects. The patient remains clinically well and without any evidence of infection 2 years after discontinuation of terbinafine treatment.