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A 47-year-old female was admitted to our hospital complaining of macrohematuria. The patient had a history of von Recklinghausen's disease. Her skin showed multiple cafe-au-lait spots and neurofibromatosis. Thorough examinations were done. Urine cytology was positive. Intravenous pyelography and cystography demonstrated an irregular wall of the bladder. A computerized tomographic scan demonstrated a 7 cm nodular mass. Cystoscopy revealed a papillary tumor on the right lateral and anterior wall of the bladder. She was diagnosed as having a bladder tumor in von Recklinghausen's neurofibromatosis. Total cystectomy was performed. Histopathological diagnosis was transitional cell carcinoma with squamous cell carcinoma (Grade III, pT3N2N0). Fifty three cases of von Recklinghausen's disease in the literature were accompanied with malignancy.
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