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CASE REPORT
Year : 2009  |  Volume : 25  |  Issue : 1  |  Page : 134-136
 

Penile shaft sinus: A sequalae of circumcision in urethral duplication


Paediatric Surgery Unit, University of Ilorin Teaching Hospital, Ilorin, P. O. Box 5291, Ilorin, Ilorin-240 001, Kwara, Nigeria

Correspondence Address:
Lukman O Abdur-Rahman
Padiatric Surgery Unit, Department of Surgery, University of Ilorin Teaching Hospital, Ilorin, P. O. Box 5291, Ilorin, 240 001
Nigeria
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-1591.45554

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   Abstract 

Urethral duplication (UD) is rare congenital anomalies with varied presentation. Careful clinical evaluation of children by specialist would enhance diagnosis, adequate management and reduce occurrence of complication. We present a 12-year-old boy with chronic post circumcision ventral penile sinus that was successfully managed for urethral duplication.


Keywords: Circumcision, penile sinus, urethral duplication


How to cite this article:
Abdur-Rahman LO, Nasir AA, Agboola JO, Adeniran JO. Penile shaft sinus: A sequalae of circumcision in urethral duplication. Indian J Urol 2009;25:134-6

How to cite this URL:
Abdur-Rahman LO, Nasir AA, Agboola JO, Adeniran JO. Penile shaft sinus: A sequalae of circumcision in urethral duplication. Indian J Urol [serial online] 2009 [cited 2019 Oct 16];25:134-6. Available from: http://www.indianjurol.com/text.asp?2009/25/1/134/45554



   Introduction Top


Urethral duplication (UD) is a rare congenital anomaly with varied clinical presentations and treatment. The embryologic development of UD is still poorly understood and theories have shown that it is due to cloaca membrane/genital tubercle and urogenital sinus anomalies. [1]

All patients should be thoroughly evaluated because of strong association with other congenital anomalies and significant surgical management challenge this may present which may lead to unpredictable outcome. [2],[3]

We report a case of incomplete UD in a 12-year-old boy who presented with a post-circumcision penile sinus.


   Case Report Top


A 12-year-old boy who presented with recurrent sero-mucoid discharge from a wound on the ventral surface of the penis. This occurred post circumcision done at home by a 'nurse' on the sixth day of live because of a distal penile swelling. Pregnancy and delivery were uneventful. The wound refused to heal in spite of repeated dressings and use of antibiotics. He had good urinary stream and normal developmental milestone. No history of recurrent urinary tract infections. He was a healthy looking young boy with adequate penile size; there was an ulcer with a background sinus and serous fluid discharge from it [Figure 1]. The urethral meatus was normally sited and adequate in size. A diagnosis of penile sinus to rule out urethral duplication was entertained. He had a micturating cystourethrogram [Figure 2] which showed normal bladder and single urethral channel. A penile sinogram showed a shallow tract that is not communicating with the orthotopic urethral. Examination under anaesthesia using a lubricated size 6 French gauge feeding tube passed via the sinus went through a long tract up to the floor of the pelvis but ended blindly [Figure 3]. This tract coursed initially in the ventral surface but later to the right lateral aspect of the orthotopic urethral from midshaft to the bulbar urethra. The whole tract was excised and histology confirmed an urothelium. Postoperative period was uneventful and patient has remained well [Figure 4].


   Discussion Top


The occurrence of UD presents a great challenge in its identification and management because of its rarity and its variants. [4],[5] A little over 200 cases have been published world wide including a case reported by Okeke et al. [6] from Nigeria. Innes Williams [7] described a morphologic classification in which urethral duplication could be; epispadial UD, hypospadial UD, spindle urethras, bifid urethras with accessory preanal branch and collateral duplication and Effmann et al. [8] classification described the variants of complete and incomplete UD with proximal, distal or non communicating orthotopic urethral. Our patient fits into the Innes William's bifid urethras with accessory preanal branch and Effmann's incomplete type IA UD.

In spite of warnings to practitioners against circumcision in cases of congenital abnormalities of the penis especially in cases of hypospadias and epispadias, this patient had his circumcision done within the first week of live because of the abnormal swelling noticed on the ventrum of the distal penile /preputial skin which left a residual sinus. In our environment, circumcision practice is by the untrained (traditional circumcisionists) and junior cadre health workers usually in late childhood between 5 and 12 years when children are gathered in groups for them to be initiated into manhood.

The differential diagnoses are congenital anterior urethrocutaneous fistula, [9] excised urethral diverticulum from urethral valve and post circumcision urethrocutaneous fistula. The micturating cystourethrogram and penile sinogram done outlined separate channels between the orthotopic urethra and the duplicate urethra however, it was shallow and did not get to the depth of the tract which was later demonstrated at operation by a probe. This buttress the fact that infusion of contrast under pressure and fluoroscopy (not available in our centre) including examination under anaesthsia would assist in demonstrating the tract.

The danger in poor demonstration of tract is the possibility of inadequate and improper preparation for surgery leaving a residual tract or damage to adjoining tissues. Paediatric urethrocystoscopy for better definition of the UD was not available at our centre. Antibiotic and wound dressing treatment are ineffective, and other treatment such as diathermocoagulation or the injection of caustic substance into the accessory duct have been condemned and abandoned. [5] The surgical treatment would depend on the type of urethral duplication and associated malformation. All effort should be made to preserve the sphincter.

In conclusion, urethral duplication, though rare, have good prognosis if adequately managed. Pre-emptive circumcision should be avoided in patients with any penile malformation and patient should be referred to a trained surgeon.

 
   References Top

1.Berrocal T, Lopez- Pereira P, Arjonilla A, Gutiιrrez J. Anomalies of the distal ureter, bladder and urethral in children: Embryologic, radiologic and pathologic features. Radiographics 2002;22:1139-64.  Back to cited text no. 1    
2.Bartone FF. The urethral. In: O'Donnell B, Koff SA, editors. Pediatric urology. 3 rd ed. Oxford: Butterworth-Heinemann; 1997.  Back to cited text no. 2    
3.Halebian G, Kraklan D, Wilcox D, Duffy P, Ransley P, Mushtaq I. Y-type Urethral duplication in the male. Br J Urol Int 2006;97:597-602.  Back to cited text no. 3    
4.Merriot T, Weber D, Steyeart H, Alessandrini P. Urethral sagittal duplication in children: Report of 7 cases. Prog Urol 2002;12:77-83.  Back to cited text no. 4    
5.Abbate B, Centonze N, Dante DA. Urethral duplication in pediatric age: A case report. Pediatr Med Chir 2002;24:387-9.  Back to cited text no. 5    
6.Okeke LI, Aisuodionoe-Shadrach OI, Adekanye AO. Urethral Duplication with a perineal opening in a four-year-old boy. J Natl Med Assoc 2006;98:284-6.  Back to cited text no. 6    
7.Williams DI, Kenawi MM. Urethral duplication in the male. Eur Urol 1975;1:209-15.  Back to cited text no. 7    
8.Effmann EL, Lebowitz RL, Colodny AH. Duplication of the urethral. Radiology 1976;119:179-85.  Back to cited text no. 8    
9.Merriot T, Pankevych T, Chaumoitre k, Alessandrini P. Congenital anterior urethrocutaneous fistula associated with duplication. Pediatr Surg Int 2003;19:744-5.  Back to cited text no. 9    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]

This article has been cited by
1 Urethral duplication: Case report and literature review
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2 Urethral duplication: Experience of four cases
Ramareddy, R.S. and Alladi, A. and Siddappa, O.S.
Journal of Indian Association of Pediatric Surgeons. 2012; 17(3): 111-115
[Pubmed]



 

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    Abstract
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