Men’s Health and Reproductive Health Research Center, Shahid Beheshti University of Medical Sciences, Tehran, Iran Jalil Hosseini & Alimohammad Mirjalili Andrology Research Center, Yazd Reproductive Science Institute, Shahid Sadoughi University of Medical Sciences, Yazd, Iran Saeid Abouei

Urinary tract duplication is a fairly uncommon congenital disease. The various varieties of this phenomenon that have been recorded around the world are covered in this article as well. The mechanism causing this oddity is still unknown, though.

Our clinic received a referral for a 45-year-old Persian male who was complaining of recurring urinary tract infections. He had previously undergone surgery to correct penoscrotal hypospadias in childhood, as well as obstructive and uncomfortable symptoms in adolescence.

A double urethra and stenosis in the proximal bulbar urethra were found on his most recent voiding cystourethrogram and retrograde urethrogram, respectively. He underwent surgery to remove the double urethra, and one month later he was observed. He didn't complain of urine incontinence or recurrence.

An extremely uncommon congenital disease is urethral duplication. This phenomenon has already been described in several different forms [1]. Although there have been a few reports of it in women [2], it mostly affects men. It is still unknown how this illness started [3]. Here, we present a case of urethral duplication in a 45-year-old male who also had urethral stenosis and a urethral tract infection (UTI).

Our clinic received a referral for a 45-year-old Persian male suffering from recurring urinary tract infections. In childhood, he experienced penoscrotal hypospadias, which was treated; in adolescence, he experienced obstructive and uncomfortable symptoms. The patient reported having recurrent urinary tract infections UTIs, purulent urethral discharge, fever, and positive urine culture for Escherichia coli. Due to bulbar tract stenosis, he was also frequently referred to other clinics and underwent urethra dilatation.

His most recent combined voiding cystourethrogram (VCUG) and retrograde urethrogram (RUG) revealed a double urethra in the dorsal portion of the main urethra and stenosis in the proximal bulbar urethra.

The mucosa was quite pale during main urethral cystoscopy, and the penile and bulbar urethrae did not have the second urethra's opening apparent. The proximal bulbar urethra's mucosa was also constricted and pallid. The second urethra's entrance was evident on flexible cystoscopy at 12 o'clock, 3 cm from the verumontanum in the bulbar urethra. The patient's primary urethra was located dorsally, and the guidewire was initially inserted. It was then moved along the penis until it was 5 cm from the tip.

With a double urethra diagnosis, the patient was brought into the operating room and put under general anaesthesia while lying in the lithotomy position. The urethra was then dissected from one side following the creation of a longitudinal incision in the perineal region. It was removed from the corpus cavernosum and the dorsal surface due to stenosis and adhesions brought on by prior operations. The urethral duplication was then separated from the primary urethra by palpating the guidewire from the dorsal side of the urethra and drawing a plane between the two urethras. It was taken out of the urethra's proximal opening, and a 4-0 Vicryl suture was used to close the aperture.

Due to the full closure of the distal urethra in the dorsal region, it was dissected and the secondary double urethra was surgically removed. Following surgery, the patient was monitored for a full month. To guarantee urine flow and avoid restenosis, Foley 18 F and 16 F cystostomy catheters were placed. With 4-0 Vicryl suture, the skin and fascia were finally healed.After the surgical site had been wrapped and the patient's vital signs had stabilised, the case was brought to the recovery ward. Regular urine analysis (UA) and urine culture (UC) tests were carried out while the patient was in the hospital, and all of them were normal. The hospital released the patient. He didn't complain of urine incontinence or recurrence. One month after being discharged, the patient's UA and UC were found to be normal, and RUG showed no signs of urine leakage.

Genitourinary system disorders affect about 10% of babies. Double urethra is one of these uncommon anomalies for which no conclusive solution has yet been discovered. Double urethra typically appears in the sagittal plane and is separated into dorsal and ventral groups depending on where the ectopic urethra is located. In contrast to other abnormalities, this one affects men more frequently than women and is infrequently linked to other congenital malformations. Anywhere along the penis midline, epispadias meatus can form [4]. In our situation, penoscrotal hypospadias and the main urethra were linked, and the condition was treated as a child with scrotal skin flaps. However, before hypospadias surgery, a second urethra was not discovered.

The patient was treated for prostatitis and urethra stricture on multiple occasions and currently experiences obstructive and irritating symptoms in the urine as well as alternating UTIs. The patient's issue was not resolved by any of the treatments, though. Urinary cystoscopy and dilatation were done to ease urethral stricture due to repeated relapse, however it persisted even after the antibiotics were withdrawn. The patient was then sent to a specialised facility, where VCUG and RUG tests were used to confirm the diagnosis of urethral duplication. The Effman classification, which categorises urethral duplication into three categories, is the most popular categorization [5, 6].

The type of urethral duplication in our patient was II-A2, which is less common than other types. The majority of urethral duplication patients have no symptoms or frequently urinate when they visit urology clinics. In other circumstances, patients learn about the anomaly in their later years; in our case, the anomaly was discovered almost at middle age (45 years old). To ascertain the architecture, urethra function, and any accompanying abnormalities in the event of urethral duplication, imaging techniques should be required. In these situations, identifying and maintaining a functional urethra are crucial. In many instances, the functional urethra is ectopic and the urethra in its usual place may be hypoplastic.

The functional urethra has a verumontanum that is normal, a good sphincter mechanism, and enough urine volume to completely empty the bladder. In many instances, VCUG can reveal both ureters, as well as a potentially larger and more pronounced functioning urethra. When the lateral urethra is hidden by hypoplasia, RUG may be necessary [7]. To evaluate the upper urinary system, one can also employ an intravenous pyelogram (IVP) or ultrasound technology.

This study demonstrates the many classification schemes, varieties of double urethras, and methods and approaches, which primarily require surgery. Additionally, surgery may be utilised when there are serious cosmetic issues. Surgery is restricted in this kind of urethral duplication to prevent harm to the patient's incontinence external sphincter. Sagittal urethral duplication can be fixed surgically using a variety of methods. The patient's anatomy can be altered to treat type II-A lesions by ventral-to-dorsal ureterostomy or by removing the urethra with or without urethroplasty [8]. As previously said, total urethral excision is extremely delicate due to the possibility of harming the external sphincter.

For type II-A2 lesions, one or two stages of urethroplasty may be necessary if the functional urethras are inadequately calibre or have severe hypoplasia.

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Mirjalili Alimohammad . Hypospadias-related missed urethral duplication, post-hypospadias repair urethral stenosis, and recurring UTIs in an adult: a case report. Insights of Clinical and Medical Images 2022.