Background: Urethral duplication is a rare congenital malformation mainly affecting men and boys. Although a number of theories have been proposed to describe this condition, the actual mechanism of this disorder is still not clear. The most frequent anomaly occurs in the sagittal plane, in which the duplicated urethra is in either dorsal or ventral position in relation to the orthotopic urethra. The therapeutic management of these conditions is complicated and depends on the presence of symptoms as well as the type of anomaly.

Case Description: We present a case of urethral duplication in a 2 years old male child. The malformation was characterised by the presence of the meatus in the dorsal penoscrotal and accompanied by the sign of infection in it. Urethrography showed contrast stopped filling 2 cm from the meatus in the dorsal penoscrotal with a total dead-end impression as an accessory urethra and there was no abnormalities in the principal urethra looked from the contrast filled the penile urethra, bulbous urethra, prostatic urethra, and bladder. That was the form of Effmann Type IA urethral duplication. The dorsal accessory urethra was excised entirely without complication and he remains symptom-free eight months after surgery.

Conclusion: In summary, urethral duplication is a rare clinical condition. It has many variants that were classified by Effman. Management depends on the anatomy of the duplication involved and the patient’s clinical symptoms.

Coleman RA, Winkle DC, Borzi PA. Urethral duplication: Cases of ventral and dorsal complete duplication and review of the literature. J Pediatr Urol. 2010;6:188–91.

Ismail A, Al Rayes T, Alsalihi M, Abbas T. Female double urethra: A case report. J Pediatr Surg. 2012;47:e29–31.

Abouzeid AA, Safoury HS, Mohammad SA, el-Naggar O, Zaki AM, Hassan TA, et al. The double urethra: Revisiting the surgical classification. Ther Adv Urol. 2015;7:76–84.

Lopes RI, Giron AM, Mello MF, Neto CMB, dos Santos J, Moscardi PRM, et al. Urethral duplication type influences on the complications rate and number of surgical procedures. Int Braz J Urol. 2017;43:1144–51.

Haleblian G, Kraklau D, Wilcox D, Duffy P, Ransley P, Mushtaq I. Y-type urethral duplication in the male. BJU Int. 2006;97:597–602.

Arena S, Arena C, Scuderi MG, Sanges G, Arena F, Di Benedetto V. Urethral duplication in males: Our experience in ten cases. Pediatr Surg Int. 2007;23:789–94.

Woodhouse C, Williams D. Duplications of the lower urinary tract in children. Br J Urol. 1979;51:481–7.

Das S, Brosman S. Duplication of the male urethra. J Urol. 1977;117:452–4.

Sánchez M, Vellibre R, Castelo J, Arias M, Sarmiento R, Costa A. A new case of male Y-type urethral duplication and review of literature. J Pediatr Surg. 2006;41:e69-71.

Casselman J, Williams D. Duplication of the urethra. Acta Urol Belg. 1966;34:535–41.

Rica P, Holder T, Ashcraft K. Congenital posterior urethral perineal fistula: a case report. J Urol. 1978;199:416–7.

Alanee S, Gupta P, Gleich P, Shukla AR. Complete urethral duplication: Description of surgical approach mimicking distal epispadias repair. J Pediatr Urol [Internet]. 2012;8:343–7.

Palmer LS, Palmer JS. Management of Abnormalities of the External Genitalia in Boys. In: Campbell-Walsh Urology Eleventh Edition. 2016. p. 3379–80.

Onofre LS, Gomes AL, Leão JQDS, Leão FG, Cruz TMA, Carnevale J. Urethral duplication - A wide spectrum of anomalies. J Pediatr Urol. 2013;9:1064–71.

Williams D, Kenawi M. Urethral duplication in the male. Eur Urol. 1975;1:209–15.

Effmann E, Lebowitz R, Colodny A. Duplication of the urethra. Radiology. 1976;119:179–85.

Maitama HY, Tella UM, Mbibu HN. Urethral duplication: Case report and literature review. Ann Afr Med. 2012;11:186.

Podesta M, Medel R, Castera R, Ruarte A. Urethra duplication in children: Surgical treatment and results. J Urol. 1998;160:1830–3.

Wagner J, Carr M, Bauer S, AH C, Retik A, Hendren W. Congenital posterior urethral perineal fistulae: A unique form of urethral duplication. Urology. 1996;48:277–80.

Slavov C, Donkov I, Popov E. Case of duplication of the urethra in an adult male, presenting with symptoms of bladder outlet obstruction. Eur Urol. 2007;52:1249–51.

Stephens F. Abnormal Embryology-cloacal dysgenesis. Congenital Malformations of the Urinary Tract. Part 1, Chapter 2. Vol. 5, New York: Praeger Scientific. p. 1983. 15–52.

Lima M, Destro F, Maffi M, Persichetti Proietti D, Ruggeri G. Practical and functional classification of the double urethra: A variable, complex and fascinating malformation observed in 20 patients. J Pediatr Urol [Internet]. 2017;13:42.e1-42.e7.

Okur MH, Aydogdu B, Arslan S, Basuguy E, Zeytun H, Arslan MŞ. Type II A2 duplication of urethra in an 8-month-old male child: A case report. Pediatr Urol Case Reports. 2015;2:17–22.

Baid M, Dutta A. Urethral duplication in a 15-year-old: case report with review of the literature. Rev Urol. 2014;16:149–51.

Erdil H, Mavi A, Erdil S, Gumusburun E. Urethral Duplication. Acta Med Okayama. 2003;57:91–3.

Gozar H, Prișcă R, Derzsi Z. Two Cases of Dorsal Urethral Duplication. Acta Medica Marisiensis. 2013;59:52–4.

Cicek T, Kizilkan Y, Coban G, Kosan M. Urethral duplication: A rare cause of recurrent urinary infection. J Pak Med Assoc. 2015;65:90–2.

Middleton AJ, Melzer R. Duplicated urethra: an anomaly best repaired. Urology. 1992;39:538–42.

Salle J, Sibai H, Rosenstain D, Brzezinski A, Corcos J. Urethral duplication in the male: review of 16 cases. J Urol. 2000;163:1936–40.