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blood spotting on underpants case report of urethral prolapse in a pre pubertal chinese girl

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We herein present a case report of urethral mucosa prolapse in a 5 year-old Chinese pre-pubertal girl.. It occurs when the urethral mucosa protrudes beyond the urethral meatus spontaneou

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Blood spotting on underpants: Case report of urethral prolapse in a

pre-pubertal Chinese girl

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

Article history:

Received 16 November 2014

Accepted 8 March 2015

Key words:

Urethral prolapse

Per vaginal bleeding

Urogenital bleeding

a b s t r a c t

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 5 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/)

Urethral prolapse is a rare pediatric urological problem with an

incidence of 1 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

pri-mary 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 thefirst case report of urethral prolapse in

pre-pu-bertal girl in this locality

1 Case

A healthy 5 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

pro-visional diagnosis of vaginal tumor She came to Hong Kong to seek

second opinion and was admitted to our surgical unit via the

Ac-cident 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,

exam-ination under anesthesia was performed A 0.5 cm circumferential

edematous mucosa surrounding the urethral opening was revealed

(Fig 1) A separate vaginal opening was identified Diagnosis of ure-thral 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 post-operative day 7 with no immediate urinary complication; and remain well 9 months after the operation without any long term sequale

2 Discussion 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 ure-terocele 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).

Contents lists available atScienceDirect Journal of Pediatric Surgery CASE REPORTS

j o u rn a l h o m e p a g e : w w w j p s c a s e r e p o r t s c o m

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

J Ped Surg Case Reports 3 (2015) 192e193

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and some may rarely be accompanied with urinary symptoms

including dysuria or urinary retention Diagnosis of urethral

pro-lapse is made by identification of the urethral meatus, and

sepa-ration 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

Con-servative treatment consists of bed rest, topical estrogen and

anti-biotics, 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 illustra-tion, 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 char-acteristics 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

Fig 1 Appearance of prolapse on examination under general anesthesia, note the

edematous mucosa.

Fig 2 Post-anastomotic outcome with no apparent prolapse Foley catheter has been temporarily removed for better delineation of mucosal-mucosal anastomosis.

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