- Email: [email protected]
Giant anterior urethral diverticulum in a neonate
Journal of Pediatric Surgery (2007) 42, 735 – 736
www.elsevier.com/locate/jpedsurg
Giant anterior urethral diverticulum in a neonate Alan J. Howieson*, Gordon A. MacKinlay Department of Paediatric Surgery, Royal Hospital for Sick Children, Sciennes Road, Edinburgh EH9 1LF, UK Index words: Urethral diverticulum; Transurethral resection
Abstract Congenital anterior urethral diverticulum is an uncommon condition that tends to present in older children with signs of chronic urinary problems. A neonate presented following recurrent collapse, and cystography revealed a giant urethral diverticulum. The diverticulum was incised at cystoscopy, leading to a full recovery. D 2007 Elsevier Inc. All rights reserved.
1. Case report Congenital anterior urethral diverticulum is an uncommon condition that tends to present in older children with signs of chronic urinary problems. An unusual case affecting a neonate is presented. A 3-week-old baby had become generally unwell and lethargic. Initial examination, blood tests, and lumbar puncture were unremarkable. Metabolic and septic screens were negative. A similar problem had been experienced at 2 days of age when he required admission to the neonatal unit due to respiratory problems. Treatment at that time was empirical for sepsis, although no convincing source was demonstrated. On his second admission, it was noted that he was dribbling urine. Ultrasound examination revealed a thickwalled trabeculated bladder with normal kidneys. A micturating cystourethrogram was attempted but was unsuccessful. Cystoscopy was performed due to acute urinary retention and difficulty with catheterization. It was evident that there was an apparent false passage in the proximal part of the urethra with the passage extending toward the right groin. Initially, it was assumed to have been created during a difficult catheterization.
* Corresponding author. Tel.: +44 0131 5360662. E-mail address: [email protected] (A.J. Howieson). 0022-3468/$ – see front matter D 2007 Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2006.12.004
The catheter remained in situ for 2 weeks before a repeat cystogram. This revealed a large anterior urethral diverticulum with dilation of the proximal urethra (Figs. 1 and 2). Incision of the diverticulum using a cystoscopic approach
Fig. 1
Outline of the diverticulum with urethral catheter in situ.
736
Fig. 2 Dilation of the proximal urethra secondary to the diverticulum is noted.
was performed, and in theatre there was noted to be a lip of tissue extending into a diverticulum on the ventral aspect of the penis, just below the dilated portion of the urethra. The cystoscope was passed into the diverticulum, and the upper edge incised. The lower lip was then located and incised, completing the disruption of the diverticulum. Flow of urine was tested by compressing the bladder, which lead to a good flow of urine. In the immediate postoperative period, the baby made a full recovery and was passing urine normally at 1 year. Long-term follow-up will be required to detect any recurrence or stricture formation.
2. Discussion Congenital anterior urethral diverticulum is a rare cause of urethral obstruction in childhood and can present at any
A.J. Howieson, G.A. MacKinlay age. It is an unusual condition, as the majority of diverticula are acquired in origin. In a review article, Zia-ui-Miraj [1] stated that the embryology remains unclear, but proposed hypotheses include a deficient corpus spongiosum, sequestration of epithelial elements after closure of the urethral folds, or cystic dilation of the urethral glands. A recent article by McLellan et al [2] suggested that rupture of a dilated bulbourethal gland may account for some cases. The main clinical feature is said to be a ventral penile swelling. Urethral diverticulum can present throughout childhood with symptoms of dribbling, frequency, poor stream, and recurrent urinary tract infections [3]. Stones can also form within the diverticulum [4]. A micturating cystourethrogram is the investigation of choice, and voiding cystourethrosonography is suggested as an additional investigation [1-3]. Classically, a diverticulum is seen bulging from the wall of the urethra. A number of approaches to treatment have been advocated. Excision of the diverticulum is not necessary, as it can remove an excessive amount of tissue leading to urethral narrowing. A transurethral resection, as carried out in our case, is the treatment of choice and is a relatively simple procedure, whereby the diverticulum is incised and laid open, thus relieving the obstructive effect [2]. Both a 1stage procedure via a midline perineal incision with excision of the valvular margin around the rim of the diverticulum and a 2-stage urethroplasty have also been described [5].
References [1] Zia-ui-Miraj M. Congential anterior urethral diverticula in children. Pediatr Surg Int 1999;15:567 - 9. [2] McLellan DL, Gaston MV, Diamond DA, et al. Anterior urethral valves and diverticula in children: a result of ruptured Cowper’s duct cyst? BJU Int 2004;94:375 - 8. [3] Gupta DK, Srinivas M. Congenital anterior urethral diverticulum in children. Pediatr Surg Int 2000;16:565 - 8. [4] Dayanc M, Peker AF, Fernandes ET. Giant congenital diverticulum of the anterior urethra with stone in a Male. Pediatr Surg Int 1994;9: 144 - 5. [5] Gingell JC, Mitchell TD, Roberts M. Concealed anterior urethral diverticulum causing urinary obstruction in the male child. Br J Urol 1972;44:602 - 6.
www.elsevier.com/locate/jpedsurg
Giant anterior urethral diverticulum in a neonate Alan J. Howieson*, Gordon A. MacKinlay Department of Paediatric Surgery, Royal Hospital for Sick Children, Sciennes Road, Edinburgh EH9 1LF, UK Index words: Urethral diverticulum; Transurethral resection
Abstract Congenital anterior urethral diverticulum is an uncommon condition that tends to present in older children with signs of chronic urinary problems. A neonate presented following recurrent collapse, and cystography revealed a giant urethral diverticulum. The diverticulum was incised at cystoscopy, leading to a full recovery. D 2007 Elsevier Inc. All rights reserved.
1. Case report Congenital anterior urethral diverticulum is an uncommon condition that tends to present in older children with signs of chronic urinary problems. An unusual case affecting a neonate is presented. A 3-week-old baby had become generally unwell and lethargic. Initial examination, blood tests, and lumbar puncture were unremarkable. Metabolic and septic screens were negative. A similar problem had been experienced at 2 days of age when he required admission to the neonatal unit due to respiratory problems. Treatment at that time was empirical for sepsis, although no convincing source was demonstrated. On his second admission, it was noted that he was dribbling urine. Ultrasound examination revealed a thickwalled trabeculated bladder with normal kidneys. A micturating cystourethrogram was attempted but was unsuccessful. Cystoscopy was performed due to acute urinary retention and difficulty with catheterization. It was evident that there was an apparent false passage in the proximal part of the urethra with the passage extending toward the right groin. Initially, it was assumed to have been created during a difficult catheterization.
* Corresponding author. Tel.: +44 0131 5360662. E-mail address: [email protected] (A.J. Howieson). 0022-3468/$ – see front matter D 2007 Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2006.12.004
The catheter remained in situ for 2 weeks before a repeat cystogram. This revealed a large anterior urethral diverticulum with dilation of the proximal urethra (Figs. 1 and 2). Incision of the diverticulum using a cystoscopic approach
Fig. 1
Outline of the diverticulum with urethral catheter in situ.
736
Fig. 2 Dilation of the proximal urethra secondary to the diverticulum is noted.
was performed, and in theatre there was noted to be a lip of tissue extending into a diverticulum on the ventral aspect of the penis, just below the dilated portion of the urethra. The cystoscope was passed into the diverticulum, and the upper edge incised. The lower lip was then located and incised, completing the disruption of the diverticulum. Flow of urine was tested by compressing the bladder, which lead to a good flow of urine. In the immediate postoperative period, the baby made a full recovery and was passing urine normally at 1 year. Long-term follow-up will be required to detect any recurrence or stricture formation.
2. Discussion Congenital anterior urethral diverticulum is a rare cause of urethral obstruction in childhood and can present at any
A.J. Howieson, G.A. MacKinlay age. It is an unusual condition, as the majority of diverticula are acquired in origin. In a review article, Zia-ui-Miraj [1] stated that the embryology remains unclear, but proposed hypotheses include a deficient corpus spongiosum, sequestration of epithelial elements after closure of the urethral folds, or cystic dilation of the urethral glands. A recent article by McLellan et al [2] suggested that rupture of a dilated bulbourethal gland may account for some cases. The main clinical feature is said to be a ventral penile swelling. Urethral diverticulum can present throughout childhood with symptoms of dribbling, frequency, poor stream, and recurrent urinary tract infections [3]. Stones can also form within the diverticulum [4]. A micturating cystourethrogram is the investigation of choice, and voiding cystourethrosonography is suggested as an additional investigation [1-3]. Classically, a diverticulum is seen bulging from the wall of the urethra. A number of approaches to treatment have been advocated. Excision of the diverticulum is not necessary, as it can remove an excessive amount of tissue leading to urethral narrowing. A transurethral resection, as carried out in our case, is the treatment of choice and is a relatively simple procedure, whereby the diverticulum is incised and laid open, thus relieving the obstructive effect [2]. Both a 1stage procedure via a midline perineal incision with excision of the valvular margin around the rim of the diverticulum and a 2-stage urethroplasty have also been described [5].
References [1] Zia-ui-Miraj M. Congential anterior urethral diverticula in children. Pediatr Surg Int 1999;15:567 - 9. [2] McLellan DL, Gaston MV, Diamond DA, et al. Anterior urethral valves and diverticula in children: a result of ruptured Cowper’s duct cyst? BJU Int 2004;94:375 - 8. [3] Gupta DK, Srinivas M. Congenital anterior urethral diverticulum in children. Pediatr Surg Int 2000;16:565 - 8. [4] Dayanc M, Peker AF, Fernandes ET. Giant congenital diverticulum of the anterior urethra with stone in a Male. Pediatr Surg Int 1994;9: 144 - 5. [5] Gingell JC, Mitchell TD, Roberts M. Concealed anterior urethral diverticulum causing urinary obstruction in the male child. Br J Urol 1972;44:602 - 6.