BACKGROUND: Patients affected with persistent genital arousal disorder report unprovoked, excessive, and unremitting genital arousal unrelated to sexual desire. Those afflicted experience severe physical and psychological distress with significant feelings of shame, guilt, and frustration. Definitive treatment options for this disorder are limited. CASE: We present the case of a 32-year-old woman, gravida 1 para 1, with persistent genital arousal disorder after treatment of an uncomplicated urinary tract infection while in a military-deployed environment. After numerous treatment modalities failed, she responded to an implanted sacral neuromodulator. CONCLUSION: Persistent genital arousal disorder may be the result of upregulated sacral nerve pathways. We report the use of sacral neuromodulation using unique program settings, which may be an effective alternative in the treatment of this distressing disorder.