ABSTRACT
Urethral diverticula are very rare conditions in boys and may occur due to congenital or acquired reasons. Post-circumcision urethral diverticulum has not been previously reported in the literature. Here, we present a case of an acquired urethral diverticulum due to its unusual etiology.
Introduction
Urethral diverticulum is defined as a localized outpouching of the urethra and is very rare in boys. A urethral diverticulum may have a narrow or wide connection to the urethra and therefore may be seen in different shapes (1). A urethral diverticulum caused by circumcision has not been reported in the literature, and here, we present a case of acquired urethral diverticulum due to its unusual etiology.
Case Presentation
A 4-year-old male patient presented with the complaints of swelling on the shaft of the penis, dysuria, and post-voiding dribbling. In his past medical history, he had a circumcision procedure with plastic clamp technique six months ago and his complaints had started after this procedure. He had no urinary complaints before the procedure. After insertion of the circumcision clamp, he became unable to urinate and groin swelling was observed, thus, he was readmitted to the hospital the day after circumcision procedure. The circumcision clamp was removed immediately and a urinary catheter was inserted into the bladder. A urethral injury was suspected and he was hospitalized for one week with urinary catheter and discharged after removal of the catheter. Although the patient was able to urinate normally after discharge, penile swelling appeared later and gradually increased.
On his physical examination, there was a massive swelling on the ventral root of the penis beginning from the penoscrotal region, and urine coming out through the urethra when pressed on it (Figure 1). Ultrasonography revealed a localized subcutaneous collection measuring 33 mm in length and 20 mm in width on the ventral root of the penis. Retrograde urethrogram showed a large periurethral pouch (Figure 2). The pouch was in connection with the distal part of urethra and filled with contrast material given from external urethral meatus. In the diagnostic cystoscopy, the connection point of the urethra with the diverticular neck was noted at a distance of 1.5 cm from the external urethral meatus. Later, the diverticulum was assessed with a midline incision at the penoscrotal junction. The giant diverticulum was extending from the coronal region to the middle of the scrotum and was surrounding the ventral half of the penis body (Figure 3). The diverticulum was dissected from the surrounding tissue adhesions and was opened from its anterior aspect. A narrow stalk connection to the coronal part of the urethra was detected. Diverticulectomy was performed and the defect in the urethra was repaired with two layers. No intra- and post-operative complications occurred. The urinary catheter was removed on the postoperative 7th day and the patient urinated without any complications (Figure 4). The histopathological examination revealed a diverticulum structure with urothelial epithelium and a wall composed of fibrous connective tissue. The six-year follow-up was uneventful.
Discussion
Urethral diverticula may be congenital or acquired. Congenital diverticula may develop due to an anterior urethral valve or urethral duplication and can cause serious problems in the upper urinary tract starting from the intrauterine period similar to the posterior urethral valve (1,2).
In the etiology of acquired diverticula, urethral trauma, catheterization, instrumentation, infection, and obstruction are the main culprits. Acquired diverticula usually develop at the fixed points of the urethra, such as the penoscrotal junction and, more often, the proximal bulbous part (2).
As in our case, urethral diverticula may present with swelling on the penoscrotal region that increase during urination, dysuria, weak urine stream, and post-voiding dribbling. It may be complicated by urinary tract infection and stone development in the diverticulum (1). The diagnosis is usually confirmed by micturating cystourethrography or retrograde urethrogram. Urethral diverticulectomy with urethroplasty is the recommended treatment, as in our case (1,3).
Urethral injury and urethral fistula development are among the well-known complications of circumcision (4). The possible etiology of the urethral diverticulum in our case was thought to be an injury at the ventral aspect of the urethra due to plastic circumcision clamping. It was thought that the continuous urinary leakage from the urethral injury point and accumulation of urine in this area caused urethral epithelization and formation of the diverticulum. Plastic circumcision clamps have been increasingly applied in recent years due to their low costs and successful results (4). As with all circumcision techniques, it should be kept in mind that these plastic circumcision clamps may also cause a urethral injury and serious complications such as urethral diverticulum.