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Management of Ureterocele? The Search for the Holy Grail THERE are some questions in medicine that defy answers and only generate more questions. Pediatric urology is not immune to these types of dilemmas. Despite a plethora of literature describing the management of ureteroceles, treatment algorithms that suit all patients and satisfy all surgeons remain elusive. The decision to treat (and when to treat) is difficult as every patient is unique. The surgeon must consider a constellation of factors including single vs duplex system ureteroceles, ectopic vs orthotopic ureterocele, symptomatic (urinary tract infection, pain, stones, hematuria) vs asymptomatic presentation (prenatal hydronephrosis, incidental finding), vesicoureteral reflux (VUR) (ipsilateral/contralateral), severity of hydroureteronephrosis and renal function/scarring. Moreover, parental preferences have a role in the decision to incise, remove or reconstruct. Given all of these variables, it is not surprising that we are still in search of the best answerdthe Holy Grail. In 1978 Snyder and Johnston recommended open lower tract reconstruction for all ureteroceles.1 They acknowledged that in the emergent setting of a septic child, endoscopic incision would be a reasonable temporizing measure. It was not until 8 years later that Tank advocated the benefit of routine transurethral incision (TUI).2 Despite not always providing definitive treatment, the goal of the procedure was to relieve obstruction. Since TUI provided definitive treatment in 50% of his series, he recommended starting with an incision in all cases to relieve the obstruction, temporize the patient and allow normalization of the ureter for future reconstruction when indicated. In 2000, 2 series from the Children’s Hospital of Philadelphia promoted the benefits of endoscopic incision. For intravesical single system ureteroceles, incision frequently eliminated the need for open surgery altogether. In others, primarily in duplex systems with ectopic ureteroceles, the incision decompressed the ureter, for many rendering tapering of the ureter unnecessary during ureteral reimplantation.3,4 In other series the majority of duplex systems were managed with TUI alone
0022-5347/15/1932-0001/0 THE JOURNAL OF UROLOGY® © 2015 by AMERICAN UROLOGICAL ASSOCIATION EDUCATION
AND
RESEARCH, INC.
with spontaneous resolution of inherent or de novo VUR.5,6 While some of the controversy in ureterocele management is due to the inherent heterogeneity of the disease, much of the controversy stems from illdefined aims of surgery. Coming back to the basics, what is the primary goal of the surgery? In the acute setting of obstruction and pyelonephritis, an endoscopic incision or puncture has become a standard approach. The conundrum is what to do after the initial puncture. What determines the need for future interventions or adjunct procedures? How and when is the collapsed ureterocele with VUR treated? Does it necessarily need any further treatment? Historically the mere presence of VUR was an accepted indication to reconstruct the lower urinary tract. It was believed that the radiographic diagnosis of VUR put children at risk for urinary tract infections and renal deterioration. But is this assumption still accepted? It is these secondary questions that need clarification to answer the original question. Does all reflux need to be repaired? What if it is only low grade reflux? Is reflux without associated infection harmful? Is iatrogenic reflux after ureterocele puncture different than congenital primary reflux? Does contralateral reflux need to be corrected? Are there long-term sequelae such as voiding dysfunction or urinary incontinence in untreated patients? Further studies are needed to help answer these questions with enough certainty to develop evidence-based recommendations. In this issue of The Journal Sander et al (page 000) assess several end points of endoscopic incision of ureteroceles at a single institution, including “improvement in hydronephrosis, de novo reflux into ureterocele moiety, cure (defined as improved hydronephrosis and no VUR postoperatively), and need for secondary surgery.”7 They present data on 83 children (98 ureteroceles) who underwent TUI for indications ranging from fever and sepsis to hydronephrosis or acute renal failure and bladder outlet obstruction.
http://dx.doi.org/10.1016/j.juro.2014.11.077 Vol. 193, 1-2, February 2015 Printed in U.S.A.
www.jurology.com Dochead: Editorials
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MANAGEMENT OF URETEROCELE
Akin to other series,4,8 this study revealed that the majority of patients with single system ureteroceles do well after endoscopic incision or puncture alone. They reported an 87.4% success rate, defined as improvement or resolution of hydronephrosis. Moreover, de novo VUR developed in only 5 of the 18 patients with single system ureteroceles who underwent preoperative and postoperative voiding cystourethrograms. Similar to previous studies, it was the patients with duplex system ureteroceles who presented a more difficult management dilemma.3e5,7,8 Sander et al found that de novo VUR developed in 43.7% and 68.7% of intravesical and extravesical duplex systems, respectively. Additionally, 42 of 57 patients with duplex system ureterocele underwent a second surgery for a nonfunctioning moiety or VUR. Surgery for de novo VUR was more common in cases of extravesical ureteroceles than for intravesical ureteroceles that had been treated with TUI. Beyond who did vs who did not undergo a secondary surgery, the authors raise a salient point in their discussion. Perhaps the requirement for undergoing reconstructive surgery should change. With improved understanding of reflux, primary as well as secondary, perhaps most of the children in this and previous studies who underwent reconstructive surgery for reflux may have been treated with endoscopic incision alone. Like the authors, we believe that in the absence of infection, low grade
VUR associated with contralateral or even ipsilateral ureteroceles may be a benign condition that does not place the renal unit at any higher risk. Ultimately the optimal management of ureteroceles remains controversial, and begins with the same algorithm used in all patients, namely evaluation of the history, physical examination, imaging and discussion with the family. There are many parents who would take even a 19% chance of avoiding a more invasive surgery rather than a 100% chance of undergoing a major reconstruction,7 while others would want a single decisive surgery from the outset. It is becoming evident that not all patients necessarily benefit from preemptive postureterocele puncture reconstruction. What remains to be determined is who benefits and who does not. Well-designed prospective studies using active vs observatory post-puncture protocols are needed and our surgical recommendations should reflect these findings. Until such studies are available, the best treatment option for ureteroceles will continue to be the subject of speculation. Yogi Berra may have said it best: “I wish I had an answer to that because I’m tired of answering that question.” Dana A. Weiss Division of Pediatric Urology The Children’s Hospital of Philadelphia Philadelphia, Pennsylvania
REFERENCES 1. Snyder HM and Johnston JH: Orthotopic ureteroceles in children. J Urol 1978; 119: 543. 2. Tank ES: Experience with endoscopic incision and open unroofing of ureteroceles. J Urol 1986; 136: 241. 3. Hagg MJ, Mourachov PV, Snyder HM et al: The modern endoscopic approach to ureterocele. J Urol 2000; 163: 940. 4. Cooper CS, Passerini-Glazel G, Hutcheson JC et al: Long-term followup of endoscopic incision
Dochead: Editorials
of ureteroceles: intravesical versus extravesical. J Urol 2000; 164: 1097. 5. Adorisio O, Elia A, Landi L et al: Effectiveness of primary endoscopic incision in treatment of ectopic ureterocele associated with duplex system. Urology 2011; 77: 191. 6. Di Renzo D, Ellsworth PI, Caldamone AA et al: Transurethral puncture for ureteroceleewhich factors dictate outcomes? J Urol 2010; 184: 1620.
7. Sander JC, Bilgutay AN, Stanasel I et al: Outcomes of endoscopic incision for the treatment of ureterocele in children at a single institution. J Urol 2015; 193: xxx.
8. Husmann D, Strand B, Ewalt D et al: Management of ectopic ureterocele associated with renal duplication: a comparison of partial nephrectomy and endoscopic decompression. J Urol 1999; 162: 1406.
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Management of Ureterocele? The Search for the Holy Grail THERE are some questions in medicine that defy answers and only generate more questions. Pediatric urology is not immune to these types of dilemmas. Despite a plethora of literature describing the management of ureteroceles, treatment algorithms that suit all patients and satisfy all surgeons remain elusive. The decision to treat (and when to treat) is difficult as every patient is unique. The surgeon must consider a constellation of factors including single vs duplex system ureteroceles, ectopic vs orthotopic ureterocele, symptomatic (urinary tract infection, pain, stones, hematuria) vs asymptomatic presentation (prenatal hydronephrosis, incidental finding), vesicoureteral reflux (VUR) (ipsilateral/contralateral), severity of hydroureteronephrosis and renal function/scarring. Moreover, parental preferences have a role in the decision to incise, remove or reconstruct. Given all of these variables, it is not surprising that we are still in search of the best answerdthe Holy Grail. In 1978 Snyder and Johnston recommended open lower tract reconstruction for all ureteroceles.1 They acknowledged that in the emergent setting of a septic child, endoscopic incision would be a reasonable temporizing measure. It was not until 8 years later that Tank advocated the benefit of routine transurethral incision (TUI).2 Despite not always providing definitive treatment, the goal of the procedure was to relieve obstruction. Since TUI provided definitive treatment in 50% of his series, he recommended starting with an incision in all cases to relieve the obstruction, temporize the patient and allow normalization of the ureter for future reconstruction when indicated. In 2000, 2 series from the Children’s Hospital of Philadelphia promoted the benefits of endoscopic incision. For intravesical single system ureteroceles, incision frequently eliminated the need for open surgery altogether. In others, primarily in duplex systems with ectopic ureteroceles, the incision decompressed the ureter, for many rendering tapering of the ureter unnecessary during ureteral reimplantation.3,4 In other series the majority of duplex systems were managed with TUI alone
0022-5347/15/1932-0001/0 THE JOURNAL OF UROLOGY® © 2015 by AMERICAN UROLOGICAL ASSOCIATION EDUCATION
AND
RESEARCH, INC.
with spontaneous resolution of inherent or de novo VUR.5,6 While some of the controversy in ureterocele management is due to the inherent heterogeneity of the disease, much of the controversy stems from illdefined aims of surgery. Coming back to the basics, what is the primary goal of the surgery? In the acute setting of obstruction and pyelonephritis, an endoscopic incision or puncture has become a standard approach. The conundrum is what to do after the initial puncture. What determines the need for future interventions or adjunct procedures? How and when is the collapsed ureterocele with VUR treated? Does it necessarily need any further treatment? Historically the mere presence of VUR was an accepted indication to reconstruct the lower urinary tract. It was believed that the radiographic diagnosis of VUR put children at risk for urinary tract infections and renal deterioration. But is this assumption still accepted? It is these secondary questions that need clarification to answer the original question. Does all reflux need to be repaired? What if it is only low grade reflux? Is reflux without associated infection harmful? Is iatrogenic reflux after ureterocele puncture different than congenital primary reflux? Does contralateral reflux need to be corrected? Are there long-term sequelae such as voiding dysfunction or urinary incontinence in untreated patients? Further studies are needed to help answer these questions with enough certainty to develop evidence-based recommendations. In this issue of The Journal Sander et al (page 000) assess several end points of endoscopic incision of ureteroceles at a single institution, including “improvement in hydronephrosis, de novo reflux into ureterocele moiety, cure (defined as improved hydronephrosis and no VUR postoperatively), and need for secondary surgery.”7 They present data on 83 children (98 ureteroceles) who underwent TUI for indications ranging from fever and sepsis to hydronephrosis or acute renal failure and bladder outlet obstruction.
http://dx.doi.org/10.1016/j.juro.2014.11.077 Vol. 193, 1-2, February 2015 Printed in U.S.A.
www.jurology.com Dochead: Editorials
EDI 5.2.0 DTD JURO12011_proof 13 November 2014 3:10 pm EO:
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115 116 117 118 119 120 121 122 123 124 125 126 127 128 129 130 131 132 133 134 135 136 137 138 139 140 141 142 143 144 145 146 147 148 149 150 151 152 153 154 155 156 157 158 159 160 161 162 163 164 165 166 167
MANAGEMENT OF URETEROCELE
Akin to other series,4,8 this study revealed that the majority of patients with single system ureteroceles do well after endoscopic incision or puncture alone. They reported an 87.4% success rate, defined as improvement or resolution of hydronephrosis. Moreover, de novo VUR developed in only 5 of the 18 patients with single system ureteroceles who underwent preoperative and postoperative voiding cystourethrograms. Similar to previous studies, it was the patients with duplex system ureteroceles who presented a more difficult management dilemma.3e5,7,8 Sander et al found that de novo VUR developed in 43.7% and 68.7% of intravesical and extravesical duplex systems, respectively. Additionally, 42 of 57 patients with duplex system ureterocele underwent a second surgery for a nonfunctioning moiety or VUR. Surgery for de novo VUR was more common in cases of extravesical ureteroceles than for intravesical ureteroceles that had been treated with TUI. Beyond who did vs who did not undergo a secondary surgery, the authors raise a salient point in their discussion. Perhaps the requirement for undergoing reconstructive surgery should change. With improved understanding of reflux, primary as well as secondary, perhaps most of the children in this and previous studies who underwent reconstructive surgery for reflux may have been treated with endoscopic incision alone. Like the authors, we believe that in the absence of infection, low grade
VUR associated with contralateral or even ipsilateral ureteroceles may be a benign condition that does not place the renal unit at any higher risk. Ultimately the optimal management of ureteroceles remains controversial, and begins with the same algorithm used in all patients, namely evaluation of the history, physical examination, imaging and discussion with the family. There are many parents who would take even a 19% chance of avoiding a more invasive surgery rather than a 100% chance of undergoing a major reconstruction,7 while others would want a single decisive surgery from the outset. It is becoming evident that not all patients necessarily benefit from preemptive postureterocele puncture reconstruction. What remains to be determined is who benefits and who does not. Well-designed prospective studies using active vs observatory post-puncture protocols are needed and our surgical recommendations should reflect these findings. Until such studies are available, the best treatment option for ureteroceles will continue to be the subject of speculation. Yogi Berra may have said it best: “I wish I had an answer to that because I’m tired of answering that question.” Dana A. Weiss Division of Pediatric Urology The Children’s Hospital of Philadelphia Philadelphia, Pennsylvania
REFERENCES 1. Snyder HM and Johnston JH: Orthotopic ureteroceles in children. J Urol 1978; 119: 543. 2. Tank ES: Experience with endoscopic incision and open unroofing of ureteroceles. J Urol 1986; 136: 241. 3. Hagg MJ, Mourachov PV, Snyder HM et al: The modern endoscopic approach to ureterocele. J Urol 2000; 163: 940. 4. Cooper CS, Passerini-Glazel G, Hutcheson JC et al: Long-term followup of endoscopic incision
Dochead: Editorials
of ureteroceles: intravesical versus extravesical. J Urol 2000; 164: 1097. 5. Adorisio O, Elia A, Landi L et al: Effectiveness of primary endoscopic incision in treatment of ectopic ureterocele associated with duplex system. Urology 2011; 77: 191. 6. Di Renzo D, Ellsworth PI, Caldamone AA et al: Transurethral puncture for ureteroceleewhich factors dictate outcomes? J Urol 2010; 184: 1620.
7. Sander JC, Bilgutay AN, Stanasel I et al: Outcomes of endoscopic incision for the treatment of ureterocele in children at a single institution. J Urol 2015; 193: xxx.
8. Husmann D, Strand B, Ewalt D et al: Management of ectopic ureterocele associated with renal duplication: a comparison of partial nephrectomy and endoscopic decompression. J Urol 1999; 162: 1406.
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