Schistosomiasis is one of the oldest and commonest parasitic infestations of mankind and one of the leading infestations of public health concern. Hitherto, praziquantel has been the only drug for mass eradication of Schistosomiasis. Few reports have shown possible resistance of the schistosome to praziquantel in some part of the world with global concern about the future of the drug. We report this case to illustrate progression of Schistosomiasis to surgical complication despite treatment, with bizarre presentation misleading attending physicians. Diagnosis was finally confirmed at post-operation histological examination of specimen.