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GU schistosomiasis is primarily caused by S. haematobium. However, Schistosoma mansoni mediated GU schistosomiasis is unusual, making this a quite interesting case. Rhabdomyosarcoma (RMS) of the breast, a mesenchymal neoplasm with skeletal muscle differentiation, is an extremely rare tumour in males, with less than 30 cases published in English-language literature. We report on the first case of a male breast RMS, with an unusual ectomesenchymal/neuroectodermal component. A 55-year-old, previously healthy male, underwent a radical left ma