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CASE REPORT
Year : 2019  |  Volume : 24  |  Issue : 2  |  Page : 138-140
 

Hypospadiac urethral duplication- A rare variant and novel use of snodgrass urethroplasty


Department of Pediatric Surgery, All India Institute of Medical Sciences, Jodhpur, Rajasthan, India

Date of Web Publication1-Mar-2019

Correspondence Address:
Dr. Rahul Saxena
Department of Pediatric Surgery, All India Institute of Medical Sciences, Jodhpur, Rajasthan
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaps.JIAPS_62_18

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   Abstract 


Urethral duplication is a spectrum of congenital anomalies that is more common in males, but very little is known about the possible reconstruction methods and their outcome. Hypospadiac urethral duplication is a variety treated with onlay preputial flap. We report a 3-year-old patient of hypospadiac duplication who was treated with a single-stage urethroplasty. An excellent cosmetic and functional outcome were achieved by dorsal to ventral urethra-urethrostomy and novel application of tubularized incised plate technique for urethroplasty.


Keywords: Tubularized incised plate, urethral duplication, urethra-urethrostomy


How to cite this article:
Saxena R, Pathak M, Sinha A. Hypospadiac urethral duplication- A rare variant and novel use of snodgrass urethroplasty. J Indian Assoc Pediatr Surg 2019;24:138-40

How to cite this URL:
Saxena R, Pathak M, Sinha A. Hypospadiac urethral duplication- A rare variant and novel use of snodgrass urethroplasty. J Indian Assoc Pediatr Surg [serial online] 2019 [cited 2019 May 24];24:138-40. Available from: http://www.jiaps.com/text.asp?2019/24/2/138/253347





   Introduction Top


Urethral duplication is a rare congenital anomaly with various clinical presentations and is more common in males.[1] Its embryology is not clear, and various hypothesis is proposed in the literature. The sagittal duplication is more common than coronal duplication.[2] The various sagittal duplications identified are prepubic sinuses, epispadiac, hypospadiac, spindle urethra, and Y duplications. The management of these cases has to be individualized according to the anomaly and the prognosis is usually good but depends on a successful reconstruction. We are describing a case of hypospadiac urethral duplication treated by the single-stage urethroplasty without using flap; utilizing the novel application of tubularized incised plate (TIP) technique for urethroplasty.


   Case Report Top


A 3-year-old male child presented to our outpatient clinic with a history of the passage of single urine stream from under the surface of penis. On clinical examination, urethral meatus was located at the mid-penile region, there was no chordee and prepuce was ventrally deficient. There were no associated congenital anomalies and patient was prepared for urethroplasty. Since our case was diagnosed incidentally on operation table, we did not have a preoperative retrograde urethrogram, but the management and outcome remain the same.

Intraoperatively, on careful clinical examination under general anesthesia, another small caliber urethral meatus was noted at the glandular tip. Infant feeding tube (6 Fr) was passed through the orthotropic meatus. It went smoothly for 3 cm from meatus, then with difficulty (fitting snugly) to the proximal penile region. The extent of the dorsal urethra was just proximal to hypospadiac urethral opening, which was evident on laying open the dorsal urethra. On calibrating the hypospadiac meatus 7 Fr infant feeding tube went smoothly into the bladder. These clinical findings confirmed the urethral duplication [Figure 1].
Figure 1: Hypospadiac urethral duplication. Dorsal orthotopic meatus and ventral proximal penile meatus

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A 6 Fr infant feeding tube passed through the orthotopic meatus. Infant feeding tube was felt at the urethral plate of the ventral hypospadiac urethra. An incision was made in the midline of the urethral plate from corona to hypospadiac opening and another midline incision made at the floor of the urethral plate just like Snodgrass urethroplasty to lay open the orthotopic dorsal urethra till the proximal penile region [Figure 2] leading to a wide and deep urethral plate. Proximally, the hypospadiac opening was anastomosed to the proximal end of the orthotopic urethra, and the distal urethral plate was tubularized up to corona over 6 Fr feeding tube with interrupted vicryl 6-0 suture which was covered by the local dartos and skin was closed in the midline. The urethra in glans region and meatus was normal. The postoperative cosmesis and function were excellent and patient passes a single urine stream from the tip of glans.
Figure 2: Division of ventral wall of dorsal orthotopic urethra in midline

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In 1-year follow-up, the patient passes urine with good stream without any straining and never had any episode of urinary tract infection (UTI). The meatus is slit-like and there is no evidence of metal or neourethral stenosis, so the anatomical study was not considered. Postoperative ultrasound to look for any other associated anomaly or hydroureteronephrosis was done, which was found to be normal.


   Discussion Top


Urethral duplication is a rare congenital anomaly and includes a variety of different duplication anomalies with variable presentation.[1],[2] It is more common in males and has various classifications.[3],[4] Although hypospadic urethral duplication is reported in the previous literature, very few of them discussed their management. On-lay preputial flap was used for repair by previous authors, probably because at that time it was more commonly used method in hypospadias repair also. Currently, tubularized incised plate is the most commonly used method of urethroplasty and based on it we have described a novel use of Snodgrass TIP urethroplasty, which can be applied to various hypospadiac anatomies with slight modification.

In 1975, Williams and Kenawi divided urethral duplications into sagittal and collateral categories.[4] The more common sagittal duplications are classified as the epispadic, hypospadic, spindle urethra, and Y-duplication groups, with the epispadic and hypospadic urethral duplications being further subdivided into complete, incomplete, and abortive varieties.[4]

The most commonly used classification for urethral duplication is that proposed by Effman, which classifies sagittal urethral duplications into three types [Table 1].[3]
Table 1: Effman classification of urethral duplication

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Our case was abortive hypospadiac type according to Williams and Kenawi and Type II A1, according to Effman's Classification.

The clinical features vary according to anatomical type. Many patients are asymptomatic and prepubic sinuses usually have mucus discharge only. The most common presenting symptom is double-urine stream, others being voiding dysfunction, UTI, hypospadias and urinary retention with urosepsis, which may require vesicostomy.[1],[2],[5] Duplication can also be identified as an incidental finding while epispadias repair, hypospadias repair, or other surgery like in our case.[1] In Type IIA2, Y-type duplication can be diagnosed by the characteristic finding of urinary dribbling from the penile tip and voiding through the anus.[1] Our patient presented to us with the single-urine stream from hypospadiac urethral opening.

To identify the anatomy, patients must be evaluated by a voiding cystourethrogram, and retrograde injection of contrast material into both channels in the anteroposterior and oblique planes. The upper urinary tract should be evaluated by ultrasound or intravenous pyelogram. Urethrocystoscopy may be required to confirm the radiological findings and recognizing the functional urethra.[1],[2],[3],[5],[6]

Management is dependent on the anatomy of the urethral duplication. Only symptomatic patients or those with a significant cosmetic problem need surgical treatment and it is important to recognize and preserve functional urethra.[1],[2],[5]

The hypospadiac duplications have usually been treated by marsupialization of the dorsal orthotopic urethra, ventral-to-dorsal urethro-urethrostomy and penile island flap onlay repair to cover dorsal open urethra.

In our patient, we widened the urethral plate and tubularized it with single midline suture line. The onlay repair of hypospadiac duplication has two stitch lines and thus carries more risk of fistula formation and is more time consuming. Proximal hypoplastic end of the dorsal urethra was widened by urethro-urethral anastomosis, and the distal dorsal urethra was widened by midline incision to achieve a tension-free urethral tube. Although our suture line extended from proximal penile location to corona because glans and meatus were appropriate in our patient, we can extend it up to glans and create neo-meatus with this technique. We covered the neo-urethra with local dartos, but we can also add barrier layer of dorsal prepuce or tunica vaginalis if required.


   Conclusion Top


Urethral duplication is a rare entity with a wide anatomical spectrum. Novel application of tubularized incised plate technique in hypospadiac duplication can simplify its management with excellent results.

Consent

This paper complies with the guidelines for human studies. The informed written consent was obtained from the parents of the child to publish the data and clinical photographs.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understand that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Salle JL, Sibai H, Rosenstein D, Brzezinski AE, Corcos J. Urethral duplication in the male: Review of 16 cases. J Urol 2000;163:1936-40.  Back to cited text no. 1
    
2.
Mane SB, Obaidah A, Dhende NP, Arlikar J, Acharya H, Thakur A, et al. Urethral duplication in children: Our experience of eight cases. J Pediatr Urol 2009;5:363-7.  Back to cited text no. 2
    
3.
Effmann EL, Lebowitz RL, Colodny AH. Duplication of the urethra. Radiology 1976;119:179-85.  Back to cited text no. 3
    
4.
Williams DI, Kenawi MM. Urethral duplications in the male. Eur Urol 1975;1:209-15.  Back to cited text no. 4
    
5.
Podesta ML, Medel R, Castera R, Ruarte AC. Urethral duplication in children: Surgical treatment and results. J Urol 1998;160:1830-3.  Back to cited text no. 5
    
6.
Onofre LS, Gomes AL, Leão JQ, Leão FG, Cruz TM, Carnevale J, et al. Urethral duplication – A wide spectrum of anomalies. J Pediatr Urol 2013;9:1064-71.  Back to cited text no. 6
    


    Figures

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    Tables

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