- Email: [email protected]
Urethral diverticulum in a female child
Journal of Pediatric Urology (2007) 3, 148e150
CASE REPORT
Urethral diverticulum in a female child Parshotam K. Gera a,*, Kim Matthews b, Grahame Smith c a
Department of Urology, The Children Hospital at Westmead, Locked bag 4001, NSW 2145, Sydney, Australia b The Children Hospital at Westmead, Locked bag 4001, NSW 2145, Sydney, Australia c Department of Urology, The Children Hospital at Westmead, Locked bag 4001, NSW 2145, Sydney, Australia Received 5 April 2006; accepted 19 April 2006 Available online 12 June 2006
KEYWORD Urethral diverticulum
Abstract A urethral diverticulum in a female arises from the wall of the urethra and consists mainly of fibrous tissue lined with epithelium. It is uncommon, and usually presents between decades 3 and 5. Most patients present with non-specific, refractory, lower urinary tract symptoms, unrelated to the diverticulum size or number. The diverticulum may also present as a mass at the introitus. We report a case of urethral diverticulum in a female child that presented as an introital mass. ª 2006 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved.
Introduction A urethral diverticulum in a female arises from the wall of the urethra and consists mainly of fibrous tissue lined with epithelium. A urethral diverticulum is uncommon and usually presents between decades 3 and 5. The most commonly accepted mechanism of female urethral diverticulum formation in adults implicates periurethral glands. Obstruction of the periurethral gland duct causes retention of secretions, followed by infection * Corresponding author. Tel.: þ61 4 1912 5486; fax: þ61 8 9340 8693. E-mail address: [email protected] (P.K. Gera).
within the occluded gland that results in abscess formation [1]. The abscess subsequently ruptures into the urethral lumen forming the diverticulum. Alternatively, evidence for the congenital origin of urethral diverticulum is based on a few paediatric case reports [2e4]. Most patients present with non-specific, refractory, lower urinary tract symptoms, unrelated to the diverticulum size or number. They undergo extensive evaluation and empirical treatments before the correct diagnosis is established. The diverticulum may also present as a mass at the introitus. We report a case of urethral diverticulum in a female child that presented as an introital mass.
1477-5131/$30 ª 2006 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.jpurol.2006.04.006
Urethral diverticulum in a female child
149
Case report A 14-year-old female presented with a 1-week history of a mass between her urethral meatus and vaginal opening. The mass was painful to touch and she had discomfort during voiding. There was a history of a decrease in the size of the mass from time to time, associated with a serosanguineous discharge. An ultrasound showed echogenic material in the mass and the bladder was normal. Examination and aspiration of cyst under anaesthesia revealed 2 ml of yellow pus. Cystoscopic examination of the urethra and bladder was normal. Pus culture revealed a light growth of Candida glabrate. Treatment with broad-spectrum antibiotics and antifungals was undertaken for 10 days. A recurrence of the swelling was noted after 1 month. The patient was taken to theatre for excision of the mass. A transverse vaginal incision was made and the vaginal wall was separated from the anterior surface of the mass. The posterior wall of the mass was in continuity with the posterior wall of urethra. The swelling was excised, leaving a longitudinal urethral defect. This was repaired with Vicryl 7-0 and then the vaginal epithelium was closed transversely with 6/0 Vicryl. Fig. 1 gives preoperative and intraoperative views of the urethral diverticulum. Histopathology demonstrated a urethral diverticulum with acute inflammation (Fig. 2). Examination under anaesthesia and cystoscopy was repeated 6 weeks after surgery and revealed normal urethral and vaginal orifices. A small distal urethral fistula was noted, approximately 3 mm proximal to the meatus. This was opened in continuity with the urethral meatus.
Figure 1 Preoperative (A); and intraoperative (B) views of urethral diverticulum.
Discussion Urethral diverticula are believed to arise from infection of periurethral glands [1]. Although they have been described in infants, the vast majority are thought to be acquired and may be a site of infection, stone formation or rarely carcinoma or endometriosis. By definition a urethral diverticulum is a sac-like protrusion or pocket that is continuous with the lumen of the urethra and contains all the layers of urethra. According to Williams and Woodard [5] some cases of children’s urethral diverticula may be remnants of an ectopic ureterocele. Silk and Lebowitz [6] had another theory that they might be the result of an abortive attempt at formation of a second urethra. The presenting symptoms of urethral diverticulum vary considerably Table 1 [3,4,7]. The symptoms
Figure 2 Urethral diverticulum lined by transitional epithelium, with stroma and loose fibrous connective tissue (20 magnification).
150 Table 1
P.K. Gera et al. Reported cases of urethral diverticulum
Case
Age
Presentation
Management
1. [3]
Neonate
Planned excision at 2 years
2. [4]
17 months
3. [4]
13 years
4. [4]
At birth
5. [4]
Neonate
Fluctuant mass at vaginal introitus Bleeding mass from lower rim of urethra 1.5-cm cystic mass just proximal to urethral meatus and anterior vaginal wall Mass protruding from vagina obscuring urethral meatus Bulging vaginal mass obscuring urethral meatus, with compression cloudy liquid from urethra Mass on anterior vaginal wall Urinary incontinence with UTI
6. [4]
At birth
7. [7]
5 years
Surgical excision by vaginal approach Surgical excision by vaginal approach
Regressed completely at 3 weeks
Regressed completely at 2 months
vaginal wall), ectopic ureterocele, vaginal wall inclusion cyst, periurethral or vaginal fibroma or myoma, haemangioma, and urethral varices. A VCUG is considered to be a useful diagnostic tool [8,9]. Various treatments have been advocated for symptomatic urethral diverticulum. Marshall [4] advocated conservative treatment for asymptomatic diverticulum in children. Endoscopic procedures are useful in diverticula situated in the distal urethra, creating a wide-mouthed diverticulum that hopefully drains freely. Lapides [9] transurethrally saucerized diverticula by opening the diverticula into the urethral lumen with a knife electrode, especially in females with previous surgical intervention and multiple recurrent diverticula. We preferred transvaginal excision of the diverticulum [10] in our case. To prevent recurrence it is essential to excise completely the communication to the urethra. All pockets of multiloculated diverticulum should be removed. Care should also be taken to completely close the urethral defect without tension to prevent pseudodiverticulum. In our case we had good functional as well as cosmetic results with a transvaginal approach.
References Regressed at 5 weeks Associated ectopic ureter Surgical excision
are neither related to the size nor the number of diverticula but are probably dictated by the size and patency of the diverticular opening and the amount of inflammatory response associated with recurrent UTI. A high index of suspicion for the possibility of a urethral diverticulum is essential in making the diagnosis. The differential diagnosis of a periurethral mass includes Skene’s gland abscess (lateral to urethral meatus), Gartner’s duct cyst (anterior lateral
[1] Huffman JW. The detailed anatomy of the periurethral ducts in the adult human female. Am J Obstet Gynecol 1948;55:86. [2] Johnson CM. Diverticula and cyst of the female urethra. J Urol 1938;39:506. [3] Glassman TA, Weinerth JL, Glenn JF. Neonatal female urethral diverticulum. Urology 1975;5(2):249e51. [4] Marshall S. Urethral diverticula in young girls. Urology 1981;7:243e5. [5] Williams DI, Woodard JR. Problems in the management of ectopic ureteroceles. J Urol 1964;92:635e52. [6] Silk MR, Lebowitz JM. Anterior urethral diverticulum. J Urol 1969;101:66e7. [7] Orikasa S, Metoki R, Ishikawa H, Arai M. Congenital urethral and vesical diverticula allied to blind ureters. Urology 1990;35:137e41. [8] Zimmern PE. The role of voiding cystourethroscopy in the evaluation of the female lower urinary tract. In: Paulson DF, editor. Problems in Urology, vol. 5. Philadelphia: J.B. Lippencott Co.; 1991. p. 23e41. [9] Lapides J. Transurethral treatment of urethral diverticula in women. J Urol 1979;121:736e8. [10] Moore TD. Diverticulum of female urethra: an improved technique of surgical excision. J Urol 1952;68:611.
CASE REPORT
Urethral diverticulum in a female child Parshotam K. Gera a,*, Kim Matthews b, Grahame Smith c a
Department of Urology, The Children Hospital at Westmead, Locked bag 4001, NSW 2145, Sydney, Australia b The Children Hospital at Westmead, Locked bag 4001, NSW 2145, Sydney, Australia c Department of Urology, The Children Hospital at Westmead, Locked bag 4001, NSW 2145, Sydney, Australia Received 5 April 2006; accepted 19 April 2006 Available online 12 June 2006
KEYWORD Urethral diverticulum
Abstract A urethral diverticulum in a female arises from the wall of the urethra and consists mainly of fibrous tissue lined with epithelium. It is uncommon, and usually presents between decades 3 and 5. Most patients present with non-specific, refractory, lower urinary tract symptoms, unrelated to the diverticulum size or number. The diverticulum may also present as a mass at the introitus. We report a case of urethral diverticulum in a female child that presented as an introital mass. ª 2006 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved.
Introduction A urethral diverticulum in a female arises from the wall of the urethra and consists mainly of fibrous tissue lined with epithelium. A urethral diverticulum is uncommon and usually presents between decades 3 and 5. The most commonly accepted mechanism of female urethral diverticulum formation in adults implicates periurethral glands. Obstruction of the periurethral gland duct causes retention of secretions, followed by infection * Corresponding author. Tel.: þ61 4 1912 5486; fax: þ61 8 9340 8693. E-mail address: [email protected] (P.K. Gera).
within the occluded gland that results in abscess formation [1]. The abscess subsequently ruptures into the urethral lumen forming the diverticulum. Alternatively, evidence for the congenital origin of urethral diverticulum is based on a few paediatric case reports [2e4]. Most patients present with non-specific, refractory, lower urinary tract symptoms, unrelated to the diverticulum size or number. They undergo extensive evaluation and empirical treatments before the correct diagnosis is established. The diverticulum may also present as a mass at the introitus. We report a case of urethral diverticulum in a female child that presented as an introital mass.
1477-5131/$30 ª 2006 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.jpurol.2006.04.006
Urethral diverticulum in a female child
149
Case report A 14-year-old female presented with a 1-week history of a mass between her urethral meatus and vaginal opening. The mass was painful to touch and she had discomfort during voiding. There was a history of a decrease in the size of the mass from time to time, associated with a serosanguineous discharge. An ultrasound showed echogenic material in the mass and the bladder was normal. Examination and aspiration of cyst under anaesthesia revealed 2 ml of yellow pus. Cystoscopic examination of the urethra and bladder was normal. Pus culture revealed a light growth of Candida glabrate. Treatment with broad-spectrum antibiotics and antifungals was undertaken for 10 days. A recurrence of the swelling was noted after 1 month. The patient was taken to theatre for excision of the mass. A transverse vaginal incision was made and the vaginal wall was separated from the anterior surface of the mass. The posterior wall of the mass was in continuity with the posterior wall of urethra. The swelling was excised, leaving a longitudinal urethral defect. This was repaired with Vicryl 7-0 and then the vaginal epithelium was closed transversely with 6/0 Vicryl. Fig. 1 gives preoperative and intraoperative views of the urethral diverticulum. Histopathology demonstrated a urethral diverticulum with acute inflammation (Fig. 2). Examination under anaesthesia and cystoscopy was repeated 6 weeks after surgery and revealed normal urethral and vaginal orifices. A small distal urethral fistula was noted, approximately 3 mm proximal to the meatus. This was opened in continuity with the urethral meatus.
Figure 1 Preoperative (A); and intraoperative (B) views of urethral diverticulum.
Discussion Urethral diverticula are believed to arise from infection of periurethral glands [1]. Although they have been described in infants, the vast majority are thought to be acquired and may be a site of infection, stone formation or rarely carcinoma or endometriosis. By definition a urethral diverticulum is a sac-like protrusion or pocket that is continuous with the lumen of the urethra and contains all the layers of urethra. According to Williams and Woodard [5] some cases of children’s urethral diverticula may be remnants of an ectopic ureterocele. Silk and Lebowitz [6] had another theory that they might be the result of an abortive attempt at formation of a second urethra. The presenting symptoms of urethral diverticulum vary considerably Table 1 [3,4,7]. The symptoms
Figure 2 Urethral diverticulum lined by transitional epithelium, with stroma and loose fibrous connective tissue (20 magnification).
150 Table 1
P.K. Gera et al. Reported cases of urethral diverticulum
Case
Age
Presentation
Management
1. [3]
Neonate
Planned excision at 2 years
2. [4]
17 months
3. [4]
13 years
4. [4]
At birth
5. [4]
Neonate
Fluctuant mass at vaginal introitus Bleeding mass from lower rim of urethra 1.5-cm cystic mass just proximal to urethral meatus and anterior vaginal wall Mass protruding from vagina obscuring urethral meatus Bulging vaginal mass obscuring urethral meatus, with compression cloudy liquid from urethra Mass on anterior vaginal wall Urinary incontinence with UTI
6. [4]
At birth
7. [7]
5 years
Surgical excision by vaginal approach Surgical excision by vaginal approach
Regressed completely at 3 weeks
Regressed completely at 2 months
vaginal wall), ectopic ureterocele, vaginal wall inclusion cyst, periurethral or vaginal fibroma or myoma, haemangioma, and urethral varices. A VCUG is considered to be a useful diagnostic tool [8,9]. Various treatments have been advocated for symptomatic urethral diverticulum. Marshall [4] advocated conservative treatment for asymptomatic diverticulum in children. Endoscopic procedures are useful in diverticula situated in the distal urethra, creating a wide-mouthed diverticulum that hopefully drains freely. Lapides [9] transurethrally saucerized diverticula by opening the diverticula into the urethral lumen with a knife electrode, especially in females with previous surgical intervention and multiple recurrent diverticula. We preferred transvaginal excision of the diverticulum [10] in our case. To prevent recurrence it is essential to excise completely the communication to the urethra. All pockets of multiloculated diverticulum should be removed. Care should also be taken to completely close the urethral defect without tension to prevent pseudodiverticulum. In our case we had good functional as well as cosmetic results with a transvaginal approach.
References Regressed at 5 weeks Associated ectopic ureter Surgical excision
are neither related to the size nor the number of diverticula but are probably dictated by the size and patency of the diverticular opening and the amount of inflammatory response associated with recurrent UTI. A high index of suspicion for the possibility of a urethral diverticulum is essential in making the diagnosis. The differential diagnosis of a periurethral mass includes Skene’s gland abscess (lateral to urethral meatus), Gartner’s duct cyst (anterior lateral
[1] Huffman JW. The detailed anatomy of the periurethral ducts in the adult human female. Am J Obstet Gynecol 1948;55:86. [2] Johnson CM. Diverticula and cyst of the female urethra. J Urol 1938;39:506. [3] Glassman TA, Weinerth JL, Glenn JF. Neonatal female urethral diverticulum. Urology 1975;5(2):249e51. [4] Marshall S. Urethral diverticula in young girls. Urology 1981;7:243e5. [5] Williams DI, Woodard JR. Problems in the management of ectopic ureteroceles. J Urol 1964;92:635e52. [6] Silk MR, Lebowitz JM. Anterior urethral diverticulum. J Urol 1969;101:66e7. [7] Orikasa S, Metoki R, Ishikawa H, Arai M. Congenital urethral and vesical diverticula allied to blind ureters. Urology 1990;35:137e41. [8] Zimmern PE. The role of voiding cystourethroscopy in the evaluation of the female lower urinary tract. In: Paulson DF, editor. Problems in Urology, vol. 5. Philadelphia: J.B. Lippencott Co.; 1991. p. 23e41. [9] Lapides J. Transurethral treatment of urethral diverticula in women. J Urol 1979;121:736e8. [10] Moore TD. Diverticulum of female urethra: an improved technique of surgical excision. J Urol 1952;68:611.