Background: Amyloidosis is a tissue protein deposition. Urethral amyloidosis is rare. Clinical presentation includes hematuria, obstructive urinary symptoms, urethritis-like symptoms, urethral discharge, or a palpable penile/urethral mass. The diagnosis is made by urethro-cystoscopy and biopsy. Case Presentation: We report the case of a 38-year-old man with a medical history of recurrent urinary tract infections, urethral discharge, and a palpable bulbar urethral mass. Urethral biopsies confirmed the urethral amyloidosis. The treatment was endoscopic urethral dilatation. Conclusion: Urethral amyloidosis is a rare condition and a challenging diagnosis. Urethroscopy is the best method for diagnosis. Biopsies should be done to exclude malignancy.
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