Introduction: Urethral stricture is a common urological condition, resulting from trauma or venereal infections. The aim of our study was to report a rare case of squamous cell carcinoma of the penis and pseudoepitheliomatous hyperplasia (PEH) of scrotal skin, on top of repeatedly managed urethral stricture which was of unknown aetiology.

Methods: A Medline search of publications studying the association of urethral stricture with penile cancer was done.

Results: Two case reports were identified that described two occurrences, which were separated by a few months.

Conclusion: Repeated management of urethral stricture with visual urethrotomy or urethral dilation may result in a chronic inflammatory status, predisposing to PEH and squamous cell carcinoma of the genital organs.