Persistent genital arousal disorder/genito-pelvic dysesthesia with severe psychiatric comorbidity and treatment refractoriness: a case report

Introduction

Persistent genital arousal disorder (PGAD), also termed genito-pelvic dysesthesia (GPD), is a spontaneous, intrusive and unwanted condition characterised by persistent, unwanted sensations of genital arousal, including throbbing, pressure, and hypersensitivity, occurring in the absence of sexual desire and not relieved by orgasm. Although there is no defined pathophysiology, proposed mechanisms include serotonergic dysregulation, peripheral neuropathy (including pudendal nerve involvement), pelvic floor dysfunction, and psychological sexual trauma. Due to the absence of established diagnostic criteria and the lack of evidence-based treatment guidelines, we present a case of a complex, treatment-refractory PGAD/GPD which was associated with severe psychiatric morbidity, multiple hospital admissions, and high suicidality.

Aims

To describe the clinical course, psychiatric comorbidity, treatment exposures, and outcomes of a severe, treatment-refractory case of PGAD/GPD, and to discuss pharmacological and interventional management challenges within a multidisciplinary framework.

Methods

Retrospective single-patient case report based on review of the electronic health record, including psychiatric assessments, medication exposures, procedures, and clinical outcomes. Descriptive analysis only. No statistical analysis was applicable given the single-patient design.

Results

A woman with longstanding major depressive disorder presented with PGAD/GPD symptoms of 13 years duration prior to diagnosis, characterised by persistent genital pressure, throbbing, and hypersensitivity occurring independently of sexual desire. During 10 years follow-up, she required 11 inpatient psychiatric admissions, including 1 intensive care unit admissions for self-injurious behaviour and active suicidality. She was trialled on 25 pharmacological agents across serotonergic, noradrenergic, dopaminergic, and voltage-gated sodium channel-modulating mechanisms. Partial, transient symptom reduction was observed with duloxetine 120 mg/day during 3 years approximately , clomipramine 75 mg/day during, and eslicarbazepine acetate 800 mg/twice a day during 3 years. other agents were discontinued due to intolerance or acute symptom exacerbation. Consistent with the known procholinergic and proserotonergic receptor profile of clomipramine — and the potential for serotonergic agents to paradoxically worsen genital hypersensitivity via 5-HT2-mediated pathways — treatment response was partial and inconsistent. Bilateral pudendal nerve blocks n=2 yielded no clinically meaningful benefit, with documented symptom severity unchanged at 4 years post-procedure (between 2019-2022). Throughout follow-up, marked functional impairment persisted with no sustained remission achieved.

Conclusions

This case illustrates PGAD/GPD as a highly distressing, disabling condition that can markedly worsen affective symptoms and suicidality risk, with substantial impairment in quality of life. Management is frequently refractory and requires iterative, individualised balancing of symptom control against iatrogenic exacerbation and medication side effects, ideally within a multidisciplinary biopsychosocial framework incorporating psychiatric, neurological, gynaecological, and pain medicine input. Given the limited evidence base predominantly case reports and small series incompletely understood and likely heterogeneous mechanisms, systematic reporting of cases and prospective studies using patient-reported outcomes are needed to guide more effective, and safer treatments. Limitations include the retrospective single-patient design and reliance on routine clinical documentation; findings are hypothesis-generating and not generalisable.

References

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