Schistosomiasis, an infection with the three anthropophilic species of Schistosoma, is endemic throughout wide areas of the tropics and subtropics with an estimated rate of over 200 million people infected worldwide. Whereas symptoms and signs of vesical and gastrointestinal forms of the infection are recognized readily, cutaneous manifestations are still a challenging diagnosis particularly in Western countries. A case is described of a 34-year-old Caucasian pregnant woman who presented to our department and was diagnosed with a cutaneous schistosomiasis involvement of the perianal region. Shistosoma haematobium was shown to be present in the lesion by histopathology and was considered to be the causative organism of the disease. Treatment with a course of oral praziquantel in a dose of 40mg/kg allowed resolution of the symptoms.