21/05/2023
Triplication of ureter in a 3.5-year-old: a case report
Professor Dr. Mustafa Zmarai and Dr. Mohammad Shafiqi
Additional article information
Abstract
We report a 3.5-year-old boy who presented with dysurea and left flank pain. Radiological workup revealed a left ureteral duplication. This presentation was opposite of what we saw at operation which was a triplication of collecting system. Early diagnosis is important to avoid complications and future renal damage.
Introduction
Ureteral triplication is a rare congenital anomaly of the upper urinary tract. We present a 3.5-year old child with a triplication of ureter on the left side. Due to lack of specific clinical signs, the mainstay of diagnosis remains a radiological one which can direct the best treatment strategy.
Case presentation
A 3.5 year old Afghan boy from Afghanistan presented with dysurea with flank pain. The clinical examination of the patient was normal with normal developmental assessment. Urine analysis showed infection and culture was positive for E-coli. Routine blood tests including full blood count, serum urea and electrolytes were normal. There was no family history of relevance.
The patient underwent ultrasound examination of the kidneys that showed evidence of left duplex kidney with dilatation of the calyces of the upper moiety.
Intravenous urography (IVU) (Figure 1) reported double collecting system with severe hydronephrosis of upper pole moiety that at operation was appeared that it was wrong and there were triplication of collecting system.
The patient was then hospitalized and planned for surgical intervention.
At operation was seen that there is a triple collecting system that the upper one had severe hydronephrosis with dilated ureter and the other two ureters were normal, therefore a left partial nephrectomy surgery was done with removal of left dilated ureter. While hospitalized she was treated with antibiotic and pain medications.
Patient was discharged home three days later after surgery with no complications and is currently under surveillance. Family members were also screened by ultrasound examination which was normal.
Discussion
Ureteral duplication occurs in 0.3-0.8% of the population, but ureteral triplication has been very rarely described [1]. Ureteral triplication is a developmental abnormality of the ureteral bud originating from the Wolffian duct at the 5th week of embryological life. The ureteral bud arises from the distal part of the wollfian duct after 4 weeks of fetal development [2]. It grows dorsally at first and cranially later and makes contact with the metanephros and the distal end differentiates into the renal pelvis and the major and minor calyces during the 6th-8th weeks. In triplication of the ureter, three ureteral buds could arise independently from the mesonephric duct or from early fission of one or more ureteral buds to join the metanephros [2]. Smith [3] classified ureteral triplication into 4 types as follows: Complete ureteral triplication (35%), incomplete triplication (21%), trifid ureters (31%), double ureter, and one bifurcated (9%).
Our case was Smith type 1 triplication. Triplication is more common in females and on the left side [2]. Presenting symptoms are renal colic, recurrent UTI, and urinary incontinence [2,4]. In addition, patients with triplication may encounter symptoms and signs of reflux, obstruction, ureterocele, or ectopia, similar to those with duplication anomalies [1]. The most frequently encountered urological anomalies associated with ureteral triplication are: contralateral duplication (37%), ureteral ectopia (28%), renal dysplasia (8%) and reflux [2]. Treatment is best tailored to individual cases and depends on the extent of the clinical problem i.e. presence of obstruction, extravesical ureteral ectopia, ureterocele or VUR. Transurethral incision of ureterocele effectively relieves the obstruction but may result in VUR necessitating ureteral re-implantation at a later stage. Other forms of surgical intervention require partial nephrectomy with aspiration of ureterocele in the first instance with re-implantation of ureter at a later stage [5].
Ureteral triplication is a rare congenital anomaly that requires high index of suspicion in order to diagnose. Due to lack of specific clinical signs, radiological examination remains the mainstay of diagnosis and can direct the best treatment strategy. This case emphasizes the importance of complete anatomic and functional evaluation of the urinary tract in the management of a child with urinary symptoms.
Abbreviations
IVU intravenous urography
MCUG micturating cystourethrogram
VUR vesicoureteric reflux
Department of pediatric surgery, Indira Gandhi Institute of Chlid Health, Kabul, Afghanistan
Corresponding author.
Professor Dr. Mustafa Zmarai; Dr. Mohammad Shafiai
References
1. Kudela G, Koszutski T, Mikosinski M, Utrata W. Ureteral triplication: report of four cases. Eur J Paediatr Surg. 2006;16:279–281. doi: 10.1055/s-2006-924338. [PubMed] [CrossRef] [Google Scholar]
2. Sivrikaya A, Cay A, Imamoglu M, Sarihan H. A case of ureteral triplication associated with ureteropelvic junction obstruction. Int Urol Nephrol. 2007;39:755–757. doi: 10.1007/s11255-006-9087-4. [PubMed] [CrossRef] [Google Scholar]
3. Smith I. Triplicate ureter. Br J Surg. 1946;34:182. doi: 10.1002/bjs.18003413409. [PubMed] [CrossRef] [Google Scholar]
4. Perkins PJ, Kroovand RL, Evans AT. Ureteral triplication. Radiology. 1973;108:533. [PubMed] [Google Scholar]
5. Gosalbez R, Jr, Gosalbez R, Piro C, Martin JA, Jimenez A. Ureteral triplication and ureterocele: report of 3 cases and review of the literature. Urology. 1991;145:105–108. [PubMed] [Google Scholar]
