Case Presentation
A woman in her early 40s sought medical attention due to persistent hyperarousal and bladder discomfort, symptoms she had been experiencing for approximately 2.5 years. The onset of these debilitating issues could be traced back to a horseback riding accident three years prior. During the incident, a forceful landing on the saddle resulted in a perineal hematoma, confirmed through computed tomography. Crucially, imaging ruled out pelvic fractures or spinal cord damage, suggesting a localized trauma event. The temporal link between the pelvic trauma and the emergence of her symptoms strongly indicated a causal relationship.
Initially, the perineal pain and hematoma resolved within a few months, allowing the patient to resume sexual activity. However, shortly after intercourse, she developed a urinary tract infection and was treated with antibiotics. This marked the beginning of new pelvic symptoms, characterized by vaginal and bladder pressure and discomfort, accompanied by unwanted arousal. These symptoms became a daily burden, persisting despite further antibiotic courses and anti-inflammatory treatments.
The impact of these symptoms on the patient’s life was profound. Sleep disturbances and the inability to exercise due to exercise-induced arousal episodes, coupled with uncomfortable urinary urgency and pelvic pressure, significantly diminished her quality of life. Fearful of exacerbating her discomfort, she avoided sexual intimacy. Behavioral modifications, including masturbation, proved ineffective in alleviating the persistent arousal.
Her medical history included anxiety, irritable bowel syndrome, human papillomavirus infection, hypothyroidism, and a resolved ovarian cyst. Over the preceding 2.5 years, she had consulted over 20 physicians and undergone extensive urological evaluations and treatments, highlighting the complexity and frustrating nature of her condition.
Diagnostic Procedures
Urodynamic testing was conducted on two separate occasions, both yielding normal results and excluding overactive bladder or urgency issues as the primary cause.
Five cystoscopy procedures were performed, revealing no signs of inflammation, Hunner ulcers, or mucosal abnormalities, further ruling out typical bladder pathologies. Bladder instillations also failed to provide any symptomatic relief.
Lifestyle and Alternative Therapies
The patient explored dietary modifications, including diets recommended for interstitial cystitis and the Paleo diet, but experienced no improvement in her symptoms.
Pelvic floor physical therapy revealed an extremely high-tone pelvic floor. While some transient, mild relief was noted, the core symptoms persisted. Acupuncture was attempted, resulting in reduced anxiety levels but no discernible improvement in arousal or urgency.
Pharmacological Interventions
Current medications included escitalopram (20 mg/d) for anxiety management, which effectively addressed her anxiety but did not impact the arousal and urgency symptoms. Pregabalin, initiated two months prior and titrated up to 300 mg/d (with plans to reach 600 mg/d), provided minimal symptom improvement. Gabapentin was previously discontinued due to lack of efficacy.
Ongoing Treatments
At the time of evaluation, the patient was actively engaged in functional medicine, acupuncture, physical therapy, and psychiatric care, reflecting a multi-faceted approach to managing her complex condition.
Clinical Evaluation Leading to PGAD Diagnosis
A focused physical examination revealed no abnormalities of the clitoral hood, labia majora/minora, or interlabial sulci. The vulvar vestibule appeared normal and healthy. No urethral prolapse was evident. Mild erythema and pain upon palpation with cotton swab testing were noted in the periurethral glands.
Pelvic floor muscle examination indicated moderate discomfort, high tonicity, and uncontrolled contractions in the pubococcygeus, iliococcygeus, and puborectalis muscles bilaterally. Sensation remained intact bilaterally.
Deep palpation of the Alcock (pudendal) canal bilaterally elicited a pudendal nerve trigger of the patient’s symptoms. This finding was crucial in confirming a Pgad Diagnosis with a pudendal trigger.
Management Strategy: Pudendal Nerve Block
Based on the confirmed pudendal nerve trigger, treatment for PGAD was initiated. It was determined that the high-tone pelvic floor dysfunction was secondary to pudendal nerve irritation and misfiring. A bilateral pudendal nerve block was proposed, with potential follow-up intravaginal onabotulinumtoxinA injections if the pelvic floor dysfunction responded favorably to the nerve block.
Procedure: Bilateral Pudendal Nerve Block Details
The patient was positioned in the lithotomy position in the procedure suite. A bilateral pudendal nerve block was performed using 10 mL of 0.25% lidocaine injected into both the right and left Alcock canals.1 The procedure was well-tolerated without complications. The patient was instructed to walk for 30 minutes post-procedure to assess symptom changes.
Upon re-evaluation after 30 minutes, in the lithotomy position and with Alcock canal palpation, a significant reduction in symptoms (greater than 50%) was reported.
To enhance the therapeutic effect, the decision was made to augment the block with the corticosteroid triamcinolone. A total of 50 mg of triamcinolone (25 mg per Alcock canal) was injected bilaterally. At a 2-week follow-up, the patient reported a sustained reduction in PGAD symptoms exceeding 50%. While intravaginal onabotulinumtoxinA injection was offered, the patient was satisfied with her current symptom resolution. A 3-month follow-up was scheduled for reassessment and potential further interventions if needed.
Discussion on PGAD and Pudendal Nerve Involvement
Persistent Genital Arousal Disorder (PGAD) is characterized by distressing symptoms of physiological sexual arousal, typically genital, occurring without subjective sexual desire. This arousal can be prolonged, lasting hours or days, or even become constant, and typically is not relieved by orgasm. Leiblum and Nathan’s seminal case series in 2001 2 highlighted the distressing, intrusive, unwanted, and sometimes painful nature of PGAD, often leading to significant distress, feelings of shame and isolation, and even suicidal ideation. Accurate pgad diagnosis is therefore critical for patient well-being.
The etiology of PGAD remains complex and poorly understood. It is likely multifactorial, possibly involving central and peripheral dysregulation, vascular changes, meningeal cysts like Tarlov cysts, pharmacological factors, and psychological contributions.3 The lack of large-scale studies and the prevalence of case reports underscore the limited knowledge regarding the frequency and specific causes of PGAD. Often, standard assessments fail to reveal any objective abnormalities.
In this particular case, the patient’s PGAD was strongly linked to pelvic trauma, with the pudendal nerve trigger clearly identified. Targeting this trigger with a pudendal nerve block resulted in effective initial therapy.4 It is crucial to recognize that, similar to conditions like interstitial cystitis, PGAD management often requires ongoing, multi-faceted approaches rather than a single definitive cure. This case highlights the importance of considering pudendal nerve involvement in pgad diagnosis and treatment, particularly in patients with a history of pelvic trauma or pain.
References
[1] Filler AG, Haynes J, Ribe CA, et al. Anatomic relationships of the pudendal canal and associated nerves relevant to pudendal nerve entrapment. Clin Anat. 2005;18(8):537-545.
[2] Leiblum SR, Nathan SG. Persistent sexual arousal syndrome: a newly discovered pattern of female sexuality. J Sex Marital Ther. 2001;27(5):365-380.
[3] Goldstein I, Komisaruk BR, Brody S, et al. Persistent genital arousal disorder in women: a proposed conceptual framework. J Sex Med. 2018;15(2):173-182.
[4] Bohren MO, Wagenlehner FM, Gasser TC, Bachmann A, Kessler TM. Pudendal nerve block in chronic pelvic pain syndrome. Eur Urol. 2016;70(4):625-632.
