Abstract

Abstract Introduction Buccal mucosal grafts (BGM) are widely used in male urethral reconstruction and have more recently been reported for female urethral reconstruction. We report the first case of using a buccal mucosa graft (BMG) to cover the excised 12:00 vestibule in a 19 year old woman with lifelong persistent genital arousal disorder (PGAD) presumed secondary to congenital neuro-proliferative vestibulodynia. She presented to our clinic at 17 and underwent lysis of clitoral adhesions, excision of the core 12:00 region with primary closure, and complete vestibulectomy with vaginal advancement flap. Her vestibular specimen positively stained for CD117 and PGP9.5 consistent with excess mast cells and nerves, respectively. Post-operatively, there was marked reduction of vestibular pain in the 1:00 to 11:00 region posteriorly, however she had persistent PGAD and significant residual tenderness to cotton-tipped swab testing at the 12:00 region. Her PGAD was medically managed successfully with tramadol, clonazepam and low dose zolpidem. Desiring to eliminate the need for systemic medications, the patient returned to the clinic at age 19. Again, local anesthesia applied to the clitoris and 12:00 vestibule region resulted in clinically significant PGAD symptom reduction and 12:00 vestibulodynia. We discussed repeat clitoral lysis and re-excision of the 12:00 region with BMG reconstruction. Objective To report on a novel technique using BMG for 12:00 region vestibular reconstruction in one woman with distressing vestibulodynia and PGAD. Methods Pre-operative counseling included discussion of routine surgical risks, benefits, alternatives, with additional possible complications including failure of the procedure to relieve PGAD or vestibulodynia, graft contracture, buccal harvest site contracture, sexual dysfunction, and genital disfigurement. The patient gave consent for the procedure and underwent a repeat lysis of clitoral adhesions and 12:00 regional subepithelial vestibulectomy. The defect measured 2.5 cm x 2.5 cm. A non-tubularized onlay patch of buccal mucosa was harvested and used for vestibular reconstruction. The procedures were performed by a single surgeon. The patient was discharged the same day. Results The patient’s post-operative course was notable for acute urinary retention in the recovery room. She was discharged with a foley catheter and passed a voiding trial on post-op day 1. Her pain was well-controlled with non-opioid medications and she was able to resume a soft food diet immediately after surgery. At post-op week 5, the clitoral area, buccal harvest site, and graft site were healed without complication. The patient reports that her 12:00 pain to touch has resolved. Her PGAD has decreased slightly, although she has not yet attempted to discontinue oral medications. Conclusions For this patient, regional vestibulectomy with BMG to the 12:00 region of the vestibule appears to have been a safe and feasible technique for refractory neuroproliferative vestibulodynia. Given that the vestibule is of endodermal origin derived from the urogenital sinus, and neuroproliferation may represent a congenital anomaly, there is a rationale to replace urogenital sinus endoderm with a non-urogenital sinus endoderm such as buccal mucosa. Pre-operative counseling should include surgery specific adverse events like mouth opening restriction and genital disfigurement. Longer follow up and larger studies are needed. Disclosure No

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