Background/Objective: To report a case of keratinizing squamous metaplasia of the bladder treated with radical cystectomy.Design: Case report and discussion of management options.Methods: Keratinizing squamous metaplasia of the bladder is a rare entity that can result from chronic irritative stimuli involving the bladder. It is considered a premalignant condition associated with invasive squamous cell carcinoma. A case report is presented describing the diagnosis and management of keratinizing squamous metaplasia of the bladder in a tetraplegic man with a chronic indwelling urinary catheter.Results: Radical cystectomy with an Indiana continent reservoir was performed after cystoscopy with biopsy confirmed keratinizing squamous metaplasia. Final pathology revealed focal erosion and diffuse keratinizing squamous metaplasia of the bladder with prostatic adenocarcinoma as an incidental finding. Conclusions:Patients with spinal cord injury who use indwelling catheters for bladder management are at higher risk of developing keratinizing squamous metaplasia. Surveillance for early detection of this entity is recommended. Prophylactic cystectomy is sometimes warranted; however, observation and frequent cystoscopic surveillance to identify potential malignant transformation can be an alternative strategy. An interdisciplinary approach is recommended before consideration of bladder resection.