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J Ped Surg Case Reports (2015) 192e193 Contents lists available at ScienceDirect Journal of Pediatric Surgery CASE REPORTS journal homepage: www.jpscasereports.com Blood spotting on underpants: Case report of urethral prolapse in a pre-pubertal Chinese girl Hei Yi Wong*, Siu Yan Tsui, Wai Cheung Mou, Kim Hung Lee Division of Pediatric Surgery and Urology, Department of Surgery, Prince of Wales Hospital, Shatin, New Territories, Hong Kong, China a r t i c l e i n f o a b s t r a c t Article history: Received 16 November 2014 Accepted March 2015 Urethral prolapse is a rare urological condition with non-specific clinical manifestations which is mostly seen in pre-pubertal black girls and postmenopausal woman The exact etiology still remains unknown We herein present a case report of urethral mucosa prolapse in a year-old Chinese pre-pubertal girl Ó 2015 The Authors Published by Elsevier Inc This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/) Key words: Urethral prolapse Per vaginal bleeding Urogenital bleeding Urethral prolapse is a rare pediatric urological problem with an incidence of in 3000 It is more common amongst pre-pubertal black girls and postmenopausal women It occurs when the urethral mucosa protrudes beyond the urethral meatus spontaneously, causing vascular congestion of the prolapsed tissue [1,2] The primary presentation is “vaginal bleeding” It is essential to distinguish this uncommon clinical entity from other causes of urogenital bleeding, to avoid misdiagnosis and unnecessary investigations We herein present the first case report of urethral prolapse in pre-pubertal girl in this locality (Fig 1) A separate vaginal opening was identified Diagnosis of urethral prolapse was made and surgical resection was performed in view of active symptoms A Foley’s catheter was inserted into the bladder and the prolapsed tissue was subsequently excised quadrant by quadrant Mucosalemucosal anastomosis was then performed (Fig 2) The Foley’s catheter was removed on post-operative day one and the girl was discharged on post-operative day two with Sitz bath and adequate oral analgesia She returned for assessment on postoperative day with no immediate urinary complication; and remain well months after the operation without any long term sequale Case Discussion A healthy year-old girl complained of 1-week history of blood spotting on underwear and also noted to have a genital mass There was no history of genital trauma or urinary symptoms She was initially admitted to a hospital in China for workup with the provisional diagnosis of vaginal tumor She came to Hong Kong to seek second opinion and was admitted to our surgical unit via the Accident and Emergency Department On physical examination, a doughnut-shaped mass with contact bleeding was identified within the labium but further examination was limited due to struggling Blood tests were unremarkable In view of limited information from bedside examination, examination under anesthesia was performed A 0.5 cm circumferential edematous mucosa surrounding the urethral opening was revealed Urethral prolapse is a rare condition in children, especially in pre-pubertal girls It can either be partial or complete Some may be complicated by bleeding, urine retention or vascular thrombosis The exact cause is still unknown, yet disproportionate growth in childhood has been proposed to be a provoking factor Reports have shown that children with higher body mass indices tend to be more prone to have urethral prolapse [3] In our patient, she has a body weight of 97th percentile but a body height of 50th percentile only Inadequate periurethral supporting tissue, weakness of the submucosal tissue and high intra-abdominal pressure are other proposed provoking factors Perineal trauma, including sexual abuse, has to be ruled out in all cases for it can possibly lead to urethral prolapse [4,5] Other differential diagnoses include ureterocele prolapse, vulvovaginitis and rhabdomyosarcoma It is essential to recognize this condition and to have an accurate diagnosis, whereas minimizing unnecessary investigations These patients primarily present with bleeding from urogenital region * Corresponding author E-mail address: hywongb@yahoo.com.hk (H.Y Wong) 2213-5766/Ó 2015 The Authors Published by Elsevier Inc This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/) http://dx.doi.org/10.1016/j.epsc.2015.03.006 H.Y Wong et al / J Ped Surg Case Reports (2015) 192e193 193 Fig Post-anastomotic outcome with no apparent prolapse Foley catheter has been temporarily removed for better delineation of mucosal-mucosal anastomosis Fig Appearance of prolapse on examination under general anesthesia, note the edematous mucosa and some may rarely be accompanied with urinary symptoms including dysuria or urinary retention Diagnosis of urethral prolapse is made by identification of the urethral meatus, and separation from vaginal opening Circular type of urethral prolapse is a soft circular mass of tissue completely surrounding the urethral meatus [3,5] Examination can be done at bedside or in some cases examination under anesthesia would be more appropriate Management of urethral prolapse has been controversial Conservative treatment consists of bed rest, topical estrogen and antibiotics, Sitz bath etc Some would adopt initial conservative treatment and if lacking response or recurrence is noted, then surgical resection would be performed [6] Recent literature has been advocating the conservative treatment strategy However, Ballohey et al have shown that surgery remains the primary strategy for patients with significant symptoms including pain and dysuria Surgical resection is a safe modality with low recurrence rate [2,7] Possible complications would include meatal stenosis and dysuria Surgery begins with insertion of Foley’s catheter and then application of stay sutures over the prolapsed tissue Then excision is done quadrant by quadrant, followed by anastomosis in single-layer, interrupted manner [7] The Foley’s catheter is usually removed within 1e3 days after operation Though being an uncommon entity, through this case illustration, the importance of clinical awareness and application of appropriate management have been demonstrated Surgical resection would still be the recommendation for all symptomatic patients Conflict of interest No conflict of interest to declare References [1] Hillyer S, Mooppan U, Kim H, Gulmi F Diagnosis and treatment of urethral prolapse in children: experience with 34 cases Urology 2009;73:1008e11 [2] Shurtleff BT, Barone JG Urethral prolapse: four quadrant excisional technique J Pediatr Adolesc Gynecol 2002;15:209e11 [3] Rudin JE, Geldt VG, Alecseev EB Prolapse of urethral mucosa in white female children: experience with 58 cases J Pediatr Surg 1997;32:423e5 [4] Johnson CF Prolapse of the urethra: confusion of clinical and anatomic characteristics with sexual abuse Pediatrics 1991;87:722e5 [5] Vunda A, Vandertuin L, Gervaix A Urethral prolapse: an overlooked diagnosis of urogenital bleeding in pre-menarche girls J Pediatr 2011;158:682e3 [6] Richardson DA, Hajj SN, Herbst AL Medical treatment of urethral prolapse in children Obstet Gynecol 1982;59:69e74 [7] Ballouhey Q, Galinier P, Gryn A, Grimaudo A, Pienkowski C, Fourcase L Benefits of primary surgical resection for symptomatic urethral prolapse in children J Pediatr Urol 2014;10:94e7 ... urinary retention Diagnosis of urethral prolapse is made by identification of the urethral meatus, and separation from vaginal opening Circular type of urethral prolapse is a soft circular mass of. .. surrounding the urethral meatus [3,5] Examination can be done at bedside or in some cases examination under anesthesia would be more appropriate Management of urethral prolapse has been controversial... mucosal-mucosal anastomosis Fig Appearance of prolapse on examination under general anesthesia, note the edematous mucosa and some may rarely be accompanied with urinary symptoms including dysuria or urinary
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