Hindawi Case Reports in Urology Volume 2018, Article ID 3186060, 5 pages https://doi.org/10.1155/2018/3186060 Case Report Adult Bilateral Ureteroceles Presenting with Lower Urinary Tract Symptoms and Acute Urinary Retention Alexis Rompré-Brodeur and Sero Andonian Division of Urology, McGill University Health Center, 1001 Decarie Boulevard, Montreal, QC, Canada H4A 3J1 Correspondence should be addressed to Sero Andonian; sero.andonian@muhc.mcgill.ca Received 25 February 2018; Accepted 3 May 2018; Published 26 June 2018 Academic Editor: Giorgio Carmignani Copyright © 2018 Alexis Rompré-Brodeur and Sero Andonian. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. Ureterocele is a well-known pathologic entity in the pediatric urology population but remains a diagnostic and treatment challenge in the adult population. Adult ureteroceles remain a diagnostic challenge for the adult urologist. Its prevalence is estimated between 1/500 and 1/4000 patients with a wide variety of clinical presentations. We present the case of a 30-year-old female patient who presented with severe lower urinary tract symptoms (LUTS) and acute urinary retention secondary to prolapsing bilateral singlesystem orthotopic ureteroceles. She was successfully treated with transurethral unroofing of her bilateral ureteroceles and she is currently asymptomatic. This case represents the first reported case of bilateral ureteroceles presenting with severe LUTS and subsequent urinary retention from the prolapse of one of the ureteroceles. We provide a review of the most recent case series of adult ureteroceles and their outcomes. Transurethral unroofing of the ureterocele is a safe and minimally invasive approach for this disease. 1. Introduction Ureterocele is a well-known pathologic entity in the pediatric urology population but remains a diagnostic and treatment challenge in adult population. Its prevalence is estimated between 1/500 and 1/4000 patients with a wide variety of clinical presentations described [1]. We present a case of a 30-year-old female patient with bilateral single-system ureteroceles and secondary urinary retention successfully treated endoscopically. Her case constitutes the first reported adult case of bilateral ureteroceles causing bladder outlet obstruction and urinary retention. 2. Case Presentation A 30-year-old female was referred to the urology clinic for severe lower urinary tract symptoms (LUTS) in addition to suspicion of a urethral mass. Her symptoms started two years earlier after the indwelling urethral catheter was removed following her Caesarian section. She noticed that a vestibular mass protruded at the level of her urethra and she reported increasing need to strain to void. In addition, this vestibular mass was very painful, and it required manual reduction to allow for her to void. LUTS got progressively worse over the course of the two years. In addition to the dysuria, she had intermittent hematuria, frequency of every hour as well as nocturia 3 to 4 times per night. Her personal past medical history was only positive for a remote appendectomy and a caesarean section. The patient had been previously worked up by her obstetrician since the LUTS appeared after her caesarean delivery. She had repeated urine analyses and cultures that were negative. Abdominal ultrasonography demonstrated the presence of two cystic lesions in the bladder of 2.5cm and 9mm in diameter. The kidneys did not show any hydronephrosis nor hydroureter. Her blood work, including renal function, was unremarkable. Examination in lithotomy position revealed a very sensitive erythematous mucosa protruding from the urethra. Patient had to manually reduce the vestibular mass in the sitting position so that she could undergo cystoscopic examination under local anesthesia. Otherwise, it was too painful for the patient. Once reduced, cystoscopy was performed and demonstrated the presence of two large ureteroceles. The left ureterocele, which was significantly larger than 2 Case Reports in Urology (a) (b) (c) (d) (e) (f) Figure 1: Preoperative (a, b, c) and postoperative (d, e, f) comparison of computed tomography (CT) scans. Preoperative triphasic CT scan images. (a) Contrast-infused axial images of the pelvis demonstrating two large bilateral intravesical cavities. (b) Axial delayed-phase images of the pelvis confirming the diagnosis of bilateral ureteroceles. (c) Coronal delayed-phase images of the pelvis provide another view of the bilateral ureteroceles. Postoperative triphasic CT scan images. (d) Contrast-infused axial images of the pelvis demonstrating the absence of intravesical cavities. (e) Axial delayed-phase images of the pelvis confirming the absence of any residual ureteroceles. (f) Coronal delayedphase images of the pelvis. the right side, demonstrated erythematous and edematous mucosa indicating that the vestibular mass previously noted by the patient and on examination was likely the wall of the left ureterocele. To rule out duplex system, a triphasic CTurogram was ordered and it confirmed the patient’s bilateral single-system ureteroceles (Figures 1(a)–1(c)). No urolithiasis was identified in both collecting systems. She was offered transurethral “unroofing” of her ureteroceles with placement of bilateral indwelling ureteral stents to reduce risk of ureteral obstruction postoperatively (Figure 2). While the patient was waiting for her elective surgery, she presented to the emergency department in acute urinary retention in addition to significant lower abdominal pain. An indwelling urethral catheter was placed to reduce the prolapsed ureterocele, decompress the bladder, and control her severe pain. Subsequently, she underwent the planned endoscopic procedure. We started with the larger and more symptomatic left ureterocele. As soon as we unroofed the ureterocele, we identified the normal ureteral orifice and placed ureteral catheter over a guidewire (Figure 2). Ureteral catheters were used to avoid injuring the back wall of the ureterocele and avoid injuring the true ureteral orifice within the ureterocele. The same procedure was repeated for the smaller right ureterocele. Once the anterior wall of the ureterocele was resected, we placed indwelling ureteral stents bilaterally. Final pathology demonstrated benign urothelium with cystitis cystica and glandularis in addition to Von Brunn’s nests. Indwelling ureteral stents were removed shortly after her operation. At 3-month follow-up, the patient was completely asymptomatic with normal voiding patterns, a normal flow study and renal function. Repeat CT-urogram demonstrated resolution of her bilateral ureteroceles without any signs of ureteral stricture, nor hydronephrosis (Figures 1(d)–1(f)). A voiding cystourethrogram was not performed given that the patient remained asymptomatic at 6 months postoperatively without any signs of vesico-ureteral reflux or urinary tract infections. 3. Discussion A ureterocele is a well-known entity among pediatric urologic population but it remains a challenge in the adult population. Most of our knowledge is based on case reports and case series. Based on autopsy studies, its prevalence in the adult population ranges between 1/500 and 1/4000 [1]. Whereas pediatric ureteroceles are often associated with duplicated collecting systems in nonorthotopic positions, adult ureteroceles are mainly reported in unilateral single systems in intravesical orthotopic positions [1, 2]. The mean age of presentation ranges from the third to the fifth decade (Table 1). Presenting symptoms vary greatly but urinary tract infections remain the most common [2]. Urinary retention secondary to prolapsing ureteroceles in adults has been reported in only seven cases. All of these occurred with unilateral prolapse of a single-system ureterocele [3–9]. This case constitutes the first reported adult case of a bilateral ureteroceles causing bladder outlet obstruction secondary to prolapse of one of the ureteroceles. Similar to pediatric populations, adult ureteroceles can contain stones at a rate of 4–39%. The main composition of theses stones is calciumoxalate and calcium-phosphate [10]. In the present case, CT scan confirmed the absence of urolithiasis. Treatment options vary but most reported cases seem to favor a low transverse incision with Collin’s knife in a “smiling” fashion, similarly to what is being advocated for the pediatric population (Table 1) [11]. Recently, Holium and KTP lasers have been used to make the incision [12, 13]. Other approaches such as simple puncture or endoscopic unroofing Mean age/Range Number of cases Bilateral Urinary retention Urolithiasis Jimenez et al. [8] 1976 31 1 0/1 1/1 0/1 Sehn et al. [7] 1981 32 1 0/1 1/1 0/1 Sandhu et al. [9] 1992 N/A 1 0/1 1/1 N/A Sekine et al. [3] 1996 40 1 0/1 1/1 0/1 Chtourou et al. [13] 2002 48.3 20 4/20 N/S 20/20 Spatafora et al. [14] 2006 18-62 15 4/15 0/15 N/S Shah et al. [9] 2008 35 16 2/16 N/S 16/16 Seibold et al. [16] 2010 48 8 1/8 0/8 5/8 Vijay et al. [11] 2011 25 26 2/26 0/26 3/26 Isen et al. [15] Westesson et al. [4] 2012 2013 47 41 5 1 /05 0/1 0/5 1/1 2/5 0/1 Sinha et al. [5] 2014 35 1 0/1 1/1 1/1 Liu et al. [10] 2015 31 30 2/30 0/30 2/30 Villagomez-Camargo et al. [6] 2015 24 1 0/1 1/1 0/1 - - 127 18/127 7/91 52/111 Authors Total Method of resection Transcutaneous puncture Transvesical excision and reimplantation N/A Transverse incision Collin’s knife Transverse incision Collin’s knife Transurethral transverse incision and percutaneous combined approach F/U imaging Complication N/A N/A N/A 0/1 N/A N/A IVU, VCUG 0/1 VCUG 1/20 with resolution at 6 months’ follow-up VCUG 2/15 low grade VUR Transverse incision Holium laser U/S, IVU, VCUG Bugbee wire elec-trode Transverse incision Collin’s knife Nephroscopic scissors Transverse incision Transurethral unroofing resection Transverse incision KTP Laser U/S, VCUG U/S, IVU and VCUG VCUG Nil 3/16 low-grade VUR at 3months, with resolution at 6 months 0/8 2/26 Low grade VUR asymptomatic 0/5 0/1 N/A 0/1 U/S, IVU, VCUG 1/30 grade I VUR with resolution at 6 months Cohen’s reimplantation Nil 0/1 - - 9/127 low grade VUR. 5/9 had documented spontaneous resolution of VUR at 6 months. Case Reports in Urology Table 1: Review of literature of adult ureteroceles. Publication year 3 4 Case Reports in Urology (a) (b) (c) (d) Figure 2: Endoscopic images during transurethral resection of bilateral ureteroceles. (a) View from the bladder neck of the left ureterocele with bullous edematous mucosa that was prolapsing. (b) After unroofing of the left ureterocele, the internal left ureteral orifice was cannulated with a hybrid nitinol-PTFE guidewire. (c) A ureteral catheter was placed over the guidewire. (d) Image of the left bladder trigone after complete resection of the left ureterocele. BN: bladder neck. UO: ureteric orifice. Ur: ureterocele wall. with the resectoscope have also been described (Table 1). Case series have reported rate of developing postoperative vesicoureteral reflux (VUR) to range from 0% to 33% in patients with low transverse “smiling” incision; most cases resolved spontaneously at 6 months of follow-up (Table 1) [14]. Our review of the current literature showed a low (7.1%) risk of postoperative VUR after endoscopic treatment with documented spontaneous resolution in half of the cases with none requiring further treatment, hence reinforcing the widespread clinical practice to investigate adult VUR only in symptomatic patients (Table 1). In our patient’s case, we opted for a transurethral unroofing technique as the ureterocele had been prolapsing and causing urinary retention. A “smiling” incision was not performed since excess remnant tissue associated with the low transverse “smiling” incision has been reported to prolapse, requiring a second procedure [15]. Therefore, in our case, the patient was cured with a single transurethral unroofing of both ureteroceles. She did not complain of any VUR symptoms. To our knowledge, this case represents the first reported case of bilateral ureteroceles presenting with severe LUTS and subsequent urinary retention from the prolapse of one of the ureteroceles. At any age of presentation, ureteroceles can be part of a complex malformation of the upper urinary tract and careful investigation is mandatory for an appropriate treatment plan. Transurethral unroofing of the ureterocele is a safe and minimally invasive approach for this disease in adults. Abbreviations BN: LUTS: UO: Ur: VUR: Bladder neck Lower urinary tract symptoms Ureteric orifice Ureterocele wall Vesicoureteral reflux. Conflicts of Interest The authors have no conflicts of interest to disclose. Case Reports in Urology References [1] C. Peter and C. Mendelsohn, “Ectopic ureter, ureterocele, and ureteral anomalies,” in Campbell-Walsh Urology, A. J. Wein, L. R. Kavoussi, A. W. Partin, and C. Peters, Eds., pp. 3075–3101, Elsevier, Philadelphia, PA, USA, 11th edition, 2016. [2] S. Halachmi and G. Pillar, “Congenital urological anomalies diagnosed in adulthood - Management considerations,” Journal of Pediatric Urology, vol. 4, no. 1, pp. 2–7, 2008. [3] H. Sekine, S.-I. Kojima, M. 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Bansal, “Endoscopic management of adult orthotopic ureterocele and associated calculi with holmium laser: Experience with 16 patients over 4 years and review of literature,” Journal of Endourology, vol. 22, no. 3, pp. 489–495, 2008. [13] C. Liu, W. Chen, C. Xie et al., “Efficacy and safety of transurethral photoselective greenlight() laser vaporization for the treatment of orthotopic ureteroceles in adults,” Photomedicine and Laser Surgery, vol. 33, no. 6, pp. 326–329, 2015. [14] M. K. Vijay, P. Vijay, A. Dutta, A. Gupta, P. Tiwari, S. Kumar et al., “The safety and efficacy of endoscopic incision of orthotopic ureterocele in adult,” Saudi Journal of Kidney Diseases and Transplantation, vol. 22, no. 6, pp. 1169–1174, 2011. [15] D. E. Coplen and J. S. Barthold, “Controversies in the management of ectopic ureteroceles,” Urology, vol. 56, no. 4, pp. 665– 668, 2000. [16] J. Seibold, S. H. Alloussi, D. Schilling, D. Colleselli, A. Stenzl, and C. Schwentner, “Minimally-invasive treatment of complicated ureteroceles in adults avoiding vesico-ureteric reflux,” Central European Journal of Urology, vol. 63, no. 2, pp. 74–76, 2010. 5 MEDIATORS of INFLAMMATION The Scientific World Journal Hindawi Publishing Corporation http://www.hindawi.com www.hindawi.com 2013 Volume 2018 Gastroenterology Research and Practice Hindawi www.hindawi.com Journal of Diabetes Research Hindawi www.hindawi.com Volume 2018 Volume 2018 Hindawi www.hindawi.com Volume 2018 Hindawi www.hindawi.com Volume 2018 International Journal of Journal of Endocrinology Immunology Research Hindawi www.hindawi.com Disease Markers Hindawi www.hindawi.com Volume 2018 Volume 2018 Submit your manuscripts at www.hindawi.com BioMed Research International PPAR Research Hindawi www.hindawi.com Hindawi www.hindawi.com Volume 2018 Volume 2018 Journal of Obesity Journal of Ophthalmology Hindawi www.hindawi.com Volume 2018 Evidence-Based Complementary and Alternative Medicine Stem Cells International Hindawi www.hindawi.com Volume 2018 Hindawi www.hindawi.com Volume 2018 Journal of Oncology Hindawi www.hindawi.com Volume 2018 Hindawi www.hindawi.com Volume 2013 Parkinson’s Disease Computational and Mathematical Methods in Medicine Hindawi www.hindawi.com Volume 2018 AIDS Behavioural Neurology Hindawi www.hindawi.com Research and Treatment Volume 2018 Hindawi www.hindawi.com Volume 2018 Hindawi www.hindawi.com Volume 2018 Oxidative Medicine and Cellular Longevity Hindawi www.hindawi.com Volume 2018