BACKGROUND Perineal hernia is a protrusion of intra-abdominal viscera through a defect in the pelvic floor and is a rare but challenging complication after extensive abdominoperineal surgery. There have been small series published after colorectal exenteration, but no cases have been reported after radical cystectomy and urethrectomy. METHODS A 68 years old woman developed an anterior perineal hernia, with no vaginal prolapse, after an anterior exenteration for bladder cancer. A perineal approach with the use of a synthetic polypropylene mesh was chosen to resolve the condition. After 6 months of follow-up, the patient has no symptoms or recurrence of the anterior perineal hernia. CONCLUSIONS To our knowledge, this case is the first report of perineal hernia after radical urethrocystectomy. Although being a case report, this article describes a potential and challenging complication after extensive anterior pelvic surgery, that could increase its incidence in the future. Literature review shows that whether perineal, abdominal or combined approach is chosen, surgery must respect hernia repair principles.