OBJECTIVE To describe a previously unreported complication (severe perineal pain) after bladder reconstruction and enterocystoplasty in patients with bladder exstrophy. PATIENTS AND METHODS The notes were reviewed retrospectively for four patients (two boys and two girls) with classical bladder exstrophy who had severe penile or perineal pain after bladder reconstruction. They were all continent and using intermittent catheterization. A range of conservative management failed and all patients subsequently required excision of their native bladders between 1997 and 2000. RESULTS All four patients had perineal or penile pain which began 4 months to 8 years after bladder augmentation. Investigations included plain abdominal X‐ray, renal and bladder ultrasonography, computerized tomography of the pelvis, video‐urodynamics and cysto‐urethroscopy. When therapeutic interventions such as more frequent bladder washouts, analgesic and anticholinergic drugs, and cystolithotomy (two patients) were unsuccessful in alleviating the symptoms, all had their native bladder excised. Histological examination of the excised tissue showed neither normal urothelium nor enteric mucosa at the margins of the excision; two patients already had squamous metaplasia within what represented the bladder, and in the others squamous epithelium was present amongst the enteric mucosa. All four children were pain‐free with a follow‐up of 2–6 years. CONCLUSION All four patients developed severe referred bladder pain that was probably secondary to the abnormal retained bladder remnants. Cystectomy cured the pain and may also have removed a potential site of future malignant tumour.