Bombay Hospital Journal] Case Reports[Contents][Home][Archives][Search][Books][Feedback]

CONGENITAL ANTERIOR URETHRAL DIVERTICULUM IN
A CASE OF OPERATED HYPOSPADIAS


Bharat B More*, Swaroop R Potdar**,Vishal V Sawant*, Tejaswini A Kamat*,V S Waingankar***,
M M Nadkarni****

*Senior Registrar; ***Honorary Associate Professor, Department of Paediatric Surgery; **Senior Registrar; ****Head of Department, Department of Radiology, Bai Jerbai Wadia Hospital for Children, Parel, Mumbai 400 012, India.

We report a case of congenital anterior urethral diverticulum in a three and a half-year-old boy. The patient being operated for proximal penile hypospadias makes this case more interesting, as we had to differentiate it from an acquired diverticulum.

INTRODUCTION

Diverticuli of the male anterior urethra are uncommon. Majority is acquired and approximately ten per cent are congenital. Though urethral diverticulum is a known complication of hypospadias repair, congenital diverticulum associated with it has not been reported to our knowledge.

CASE REPORT

A three and a half year old boy came with a penoscrotal swelling while passing urine since three months; difficulty in voiding and post void dribbling of urine since two months. The swelling would typically increase during voiding and gradually decrease on its own after sometime. He had proximal penile hypospadias with chordee. Chordee correction was done at two and a half years of age and second stage tube repair was done after an interval of six months. Postoperative period was uneventful and suprapubic cystostomy was removed on the thirteenth day.

On examination there was a soft, fluctuant, cystic swelling measuring 4x4 cm in the midline of the scrotum next to the penoscrotal junction. The swelling was clinically proximal to the reconstructed urethra. On applying pressure there was dribbling of urine from the reconstructed meatus. There was no meatal stenosis. A no. 8 infant feeding tube could be passed easily.

An ultrasound examination was done with an 11 MHz linear probe (GE LOGIC 500 MD). An anechoic well-defined swelling with low level internal echoes and a regular wall was seen at base of the penis. The nipple of the swelling was seen to be dipping into the spongiosum. Both the testes were seen separately and were normal. The child was asked to void and a sudden increase in the size of the swelling was noted. Its communication with the anterior urethra could be demonstrated. A voiding cystourethrogram revealed the diverticulum and confirmed the ultrasound findings.

On Cystoscopy, there was a mucosal diverticulum arising from penile urethra well proximal to the neo-urethra. It contained flakes and a small mulberry stone. Urethra proximal and distal to the diverticulum was normal; reconstructed urethra was of good size and could be demarcated from the proximal urethra. Bladder was normal except for debris.

Fig. 1a
Fig. 1a : Congenital Anterior Urethral diverticulum in a case of operated hypospadias.


Fig. 1b
Fig. 1b : Schematic representation. High-resolution ultrasonography done from ventral aspect of penis showing cystic structure filing up during micturition. Communication with the anterior urethra is seen.


On exploration there was a single diverticulum in the proximal anterior urethra lined by mucosa. The distal extent of the diverticulum was one cm proximal to the neourethra, which was normal. Diverticulectomy and urethroplasty with suprapubic cystostomy was done. Histopathology of the wall showed stratified epithelium and few muscle fibers confirming its congenital nature.

DISCUSSION

Anterior urethral diverticula may be congenital or acquired and are common causes of urologic complaints. Most cases are acquired and are secondary to infection, trauma or distal obstruction. Acquired urethral diverticuli are seen frequently following repair of proximal hypospadias and have been reported as a late complication in as many as 3-5% of boys undergoing onlay island flap or tube repair. A diverticulum may occur if the neourethra is too wide and/or too long or if meatal stenosis develops.[1] Sometimes a leak from the inner suture line may create a pool that subsequently reepithelises as a diverticulum. Classically acquired diverticula are found to be lined by septic granulation tissue and the walls usually contain no smooth muscle fibres.[2] Rarely they are lined by stratified epithelium particularly in chronic lesions. Congenital diverticuli account for 10-20% of total number of patients with this disorder. Important causes of congenital urethral diverticula are anterior urethral valve and congenital megalourethra. Signs and symptoms vary depending upon aetiology of the diverticulum and include weak stream, post void dribbling, dysuria and infection. The diverticulum is usually obvious during micturition.[3] Voillemier had proposed that they originated from a partial lack of spongiosum development and represented an incomplete hypospadias.[4] Although an association with hypospadias has been discounted, 2 cases of congenital diverticulum of anterior urethra owing to the absence of corpus spongiosum development in association with aplasia of the abdominal wall musculature have been reported.[5] Preoperative evaluation should include either a retrograde urethrogram or voiding cystourethrogram and cystourethroscopy for proper assessment. Surgical management depends upon the aetiology the diverticulum.[6] Large diverticulum requires open diverticulectomy and urethroplasty.


REFERENCES
  1. Duckett JW, Baskin LS. Hypospadias. James A O’Neill, Jr. et al. editors. Pediatric Surgery Fifth Edition. Mosby. 1998; 1778.
  2. Wickman MB, Engel RME. Case of anterior urethral diverticulum in male quadriplegic. J Urol 1976; 116 : 118.
  3. Virinder Mohan, Gupta SK, Joseph Cherian, Tripathi VNP, Sharma BB. Urethral diverticulum in male subjects : Report of 5 cases. J Urol 1980; 132 : 592-94.
  4. Voillemier LD. Traite des Maladies des Voies Urinaires V Mason and Fils. Cited by Boissonnat and Duhamel. 1868.
  5. Boissonnat P, Duhamel B. Congenital anomaly of the anterior urethra associated with aplasia of the abdominal wall in a male infant. Brit J Urol 1962; 34 : 59.
  6. Stephen A Ortlip, Ricardo Gonzales, Richard D. Williams Diverticula of the male urethra. J Urol 1980; 350-55.

To Section TOC

Sponsor-Dr. Reddy's Lab