Fascioliasis is a food-borne anthropozoonotic disease caused by Fasciola hepatica that affects multiple hosts, including humans. We herein report the first case of human fascioliasis in the state of Santa Catarina, Brazil. A 57-year-old female patient complaining of abdominal pain was admitted to the hospital for a clinical investigation. The diagnosis of F. hepatica was confirmed by ultrasound and indirect enzyme-linked immunosorbent assay. Authorities of the Northern coast of Santa Catarina were notified to investigate other cases and risk factors for contamination. The disease is also prevalent in cattle, which could pose as a potential route for infection.