Schistosomiasis, a waterborne trematode infestation, is one of the most widespread parasitic diseases in the world. It occurs in well-defined endemic geographical areas. Schistosomiasis of the appendix was first described by Turner in 1909, and has been reported from endemic areas. However, appendicular schistosomiasis in travelers has not been reported in the English literature. We describe an Israeli traveler with acute appendicitis as the presenting symptom of schistosomiasis, 2 years after a visit to Africa.