Low-Grade Papillary Urothelial Carcinoma of the Bladder Presenting as Persistent Genital Arousal Disorder/Genito-Pelvic Dysesthesia: A Case Report.

Persistent genital arousal disorder/genito-pelvic dysesthesia (PGAD/GPD) is a rare and distressing condition characterized by unwanted genital arousal without associated sexual desire. While cases of PGAD/GPD in association with interstitial cystitis, urethritis, urinary tract infection, and urethral diverticulum have been described, bladder cancer has not yet been reported. To our knowledge, the present report describes the first known case of PGAD/GPD as the initial presentation of low-grade papillary urothelial carcinoma of the bladder in a young female. Case Presentation: A 26 year-old healthy female presented with a 3-month history of constant, distressing clitoral engorgement, throbbing, and pain in her vulvar vestibule that began during masturbation. She also noted dysuria, urinary frequency, urgency, intermittent hematuria, and nocturia two to three times per night, pelvic floor hypertonicity, and restless legs. She denied any smoking history but did report a history of occasional resin exposure while making jewelry as a hobby. She underwent a bladder ultrasound that revealed a 7 × 5 × 6-mm nonmobile lesion in the left posterior bladder wall, lateral to the ureteral orifice. Cystoscopy and pathology revealed low-grade papillary (Ta) urothelial carcinoma of the bladder, and transurethral resection of bladder tumor was performed. Symptoms had completely resolved 3 months after the procedure, with continued use of topical lidocaine gel applied to the clitoris and pelvic floor physical therapy. Discussion: Our postulate to account for the constellation of symptoms tumor-induced chronic irritation of the pelvic nerve resulting in reflexive bladder hypermotility, clitoral engorgement, and bowel dysfunction. In addition, central cross-sensitization at the level of the sacral spinal cord could activate the postsynaptic neurons that normally respond to pudendal nerve afferent activity, resulting in clitoral and vestibular dysesthesia, reflexive pelvic floor hypertonicity via postsynaptic pudendal nerve efferents, and reflexive restless legs via postsynaptic sciatic nerve efferents. Further investigation is warranted to better understand the relationship between bladder tumors, PGAD/GPD, and related viscero-visceral and viscero-somatic reflex activity.