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Introduction: Persistent genital arousal disorder/genito-pelvic dysesthesia (PGAD/GPD) is a rare and distressing condition characterized by persistent, intrusive genital arousal occurring independently of sexual desire or stimulation. Different studies have found an association with dysuria, elevated urinary urgency and increased urinary frequency. In this report, we describe the successful interdisciplinary management of a case of PGAD/GPD due to a Tarlov cyst (TC) at the level of S2 on the right side, alongside a review of the literature focusing on neuroscientific aspects.

Methods: A 46-year-old female Kurdish patient presented with severe PGAD/GPD symptoms, including uncontrollable and involuntary orgasms of excessive number (up to 200 per day), associated with bilateral lower limb paresthesia and profound impairment of quality of life. PGAD/GPD was diagnosed according to the consensus criteria of the International Society for the Study of Women’s Sexual Health (ISSWSH).

Magnetic resonance imaging (MRI) of the sacrum revealed a right-sided TC at the level of S2 compressing the corresponding nerve root.

Following interdisciplinary evaluation, microsurgical resection of the TC at the level of S2 was performed with intraoperative neurophysiological monitoring.

Results: Postoperatively, the patient experience improvement of PGAD/GPD symptoms. At two- and four-month follow-up, she remained symptom-free, with MRI confirming complete cyst removal and no residual sacral nerve compression. Quality of life and daily functioning improved substantially.

Conclusion: This case supports a peripheral neurogenic mechanism of PGAD/GPD related to sacral nerve root compression by a TC. Other neurogenic causes of PGAD/GPD besides TC described in the literature include epilepsy (especially in the right temporal lobe), cerebral space-occupying lesions and pudendal neuralgia. PGAD/GPD has also been discussed as a phenotypic variation of overactive bladder syndrome.