In diffuse idiopathic skeletal hyperostosis (DISH), recurrence of hyperostosis after surgical removal is common. While both non-steroidal anti-inflammatory drugs (NSAIDs) and radiotherapy have been used in the prevention of heterotopic ossification, only NSAIDs have been applied for prophylaxis in DISH. As a previously undescribed prophylaxis of recurrence in DISH, we report a case treated with local radiotherapy.