Background: Although the breakage of a Sachse's knife blade is already a rare event while performing optical internal urethrotomy, a double failure appears to be at the same time a unique and a challenging complication to manage since no reference has emerged from literature review. Case Presentation: A male patient, 80 years of age, underwent retreatment of recurrent urethral stricture that occurred after transurethral resection of the prostate. The latter was complicated by severe intraoperative urethrorrhagia; this is the reason he was transferred from another institution where at first a suprapubic cystostomy was carried out, followed by urethral recanalization through internal urethrotomy and finally he underwent intracavernous Sachse's knife blade discharge. Preoperative evaluation included combined retrograde and voiding urethrography and CT to evaluate the complete resolution of the urethral stenosis and to establish the correct location of the blades inside the corpus cavernosum. A transperineal approach to the left corpus cavernosum was carried out with manageable removal of the foreign body and postoperative assessment showed no early considerable complications for the patient. Conclusion: To our knowledge, this is the first case of double breakage of Sachse's knife blade performing optical internal urethrotomy reported in literature. Although it may appear to be an easy procedure, close attention to its execution must always be paid to prevent major complications. A transperineal approach has proven to be effective and safe without creating any further outcomes to the patient.