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PFD Week 2016
Case Report: Laparoscopic and Vulvar Resection of ...
Case Report: Laparoscopic and Vulvar Resection of a Congenital Prepubic Sinus
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Laparoscopic and vulvar resection of a congenital prepubic sinus. We have no financial disclosures. A 16-year-old female presented with one and a half weeks of clitoral and left labial swelling. She was admitted for possible labial abscess and started on IV antibiotics. She denied any trauma, fever, chills, or drainage. She had an unremarkable past medical history. On examination, there was significant edema of the left mons, clitoris, and left labium magus. A fluctuant mass was palpable under the mons tracking to the left labium magus. There was no erythema or drainage. The remainder of her exam, including labia minora, right labium magus, and hymen, were normal. An ultrasound revealed a three and a half centimeter fluid collection in the vulvar soft tissues. An MRI was performed. Bilobar fluid collections were noted with prepubic and retropubic components. There was no connection to the urethra or bladder. A sinus tract was thought to be present connecting the fluid collections. Based on the imaging, the patient was diagnosed with a congenital prepubic sinus. There was associated stranding indicating an infection. The patient was taken to the operating room for an ultrasound guided incision and drainage. Findings were consistent with a five centimeter superior labial abscess, with no apparent communication to the retropubic component. Packing was placed and antibiotics were continued. Three weeks postoperatively, the patient's swelling had resolved and the plan was for observation with possible future resection. Three months later, however, the patient again presented with labial swelling and pain. A CT and MRI were performed. CT revealed smaller fluid collections, but still consistent with prepubic and retropubic sinuses. There was no associated infection. A congenital prepubic sinus is a rare congenital anomaly of the external genitalia. In many cases, patients present with discharge from an infection of the sinus tract as the only symptom. There are currently two theories regarding the origin of this disorder. The first theory is that the sinus is the result of an anomalous anterior abdominal wall closure. This theory is favored when patients present with other closure defects, such as an infallicile, bladder extra feet, and epispadius. The second theory is that this is a variant of dorsal urethral duplication. Between the patient's images, we can see two fluid collections located prepubically and retropubically. They appear to be midline structures with a possible tract between them. The tract appears to run directly under the pubis. Patency of this tract was unclear, and it should be noted that the previously infected prepubic component did not clinically seem to communicate. There were no other abnormalities present. Given the patient's recurrence of symptoms, a laparoscopic and vulvar resection was attempted. Because the mass was not palpable prior to incision, it was injected with saline in order to enhance identification. A left labial incision was then performed. Dense tissue consistent with the previously infected multiloculated cystic structure was encountered and systematically resected, starting at the cephalad pole and working downward, just to the left of the corpus of the clitoris and directly under the left-sided crura. Care was taken to avoid vital structures innervating the clitoris, particularly the anterior nerve, a branch of the pudendal nerve. A stitch was then placed at the base of the sinus tract. The sinus tract was then excised. The prepubic portion of the tract, which appeared not to communicate with the retropubic portion, remained tagged. Attention was then turned to the laparoscopic approach to the retropubic portion. The bladder was backfilled and the retropubic space was entered. Care was taken to avoid vital structures, such as the obturator neurovascular bundle laterally, and the urethra medially. The normally easy dissection of areolar tissue, as seen laterally, becomes more dense and fibrous at the midline, where the cyst is encountered. The sinus tract was then transected. The sinus tract was then transected. The sinus tract was then transected just below the pubic bone and removed. A stent was then used to ensure that the residual tissue did not contain a communicating tract to the prepubic sinus. The remainder of the sinus tract was then excised and cauterized. The peritoneum was then closed with PDS suture. A cystoscopy revealed bilateral ureteral jets and no evidence of injury to the urethra or bladder. The deep tissue from the vulvar incision was then closed with 2-ovicral interrupted stitches. The skin was then closed with vertical mattress stitches of 4-ovicral. Postoperatively, the patient did well without any evidence of infection or fluid collection. It is also interesting to note that a longstanding complaint of irritative voiding and urinary frequency was significantly improved. Final pathology revealed a prepubic sinus tract with squamous mucosa and a retropubic sinus tract with squamous and transitional epithelium with underlying smooth muscle. The presence of smooth muscle with both squamous and transitional epithelium favors the theory that the congenital prepubic sinus in this case is a variant of dorsal urethral duplication. This duplication, however, appeared blind at both ends. In 2001, Huang et al. reported on the excisions of a congenital prepubic sinus in 5 patients. Nearly all specimens demonstrated squamous and transitional epithelium with smooth muscle bundles, further supporting the theory of dorsal urethral duplication. In summary, congenital prepubic sinus can present later in life. Minimally invasive techniques can be used to successfully resect the tract in its entirety. Histology of these specimens can help determine the origin of this condition. For more information, visit www.fema.gov
Video Summary
In this video, a 16-year-old female presented with clitoral and left labial swelling. After being admitted and undergoing imaging, she was diagnosed with a congenital prepubic sinus with an infection. She underwent an ultrasound-guided incision and drainage, but three months later, the swelling and pain recurred. A laparoscopic and vulvar resection was attempted to remove the sinus tract. The procedure went well, with no complications or recurrence postoperatively. Final pathology revealed that the sinus tract had squamous and transitional epithelium with smooth muscle, supporting the theory of dorsal urethral duplication. This video provides insights into the presentation, diagnosis, and treatment of congenital prepubic sinus. No financial disclosures were given. For more information, visit www.fema.gov.
Asset Subtitle
Kavita Mishra, MD
Keywords
16-year-old female
clitoral swelling
labial swelling
congenital prepubic sinus
ultrasound-guided incision and drainage
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