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Journal of Pediatric Urology (2015) xx, 1.e1e1.e6

Delayed surgical repair of posttraumatic posterior urethral distraction defects in children and adolescents: Long-term results Urology Unit, Department of Surgery, Hospital de Nin ˜os “Ricardo Gutierrez” and Associated Hospital to The University of Buenos Aires, Buenos Aires, Argentina Correspondence to: M. Podesta [email protected] (M. Podesta) Keywords Urethra; Pelvic fracture; Urethral stricture; Post-traumatic; Children Received 21 June 2014 Accepted 4 September 2014 Available online xxx

Miguel Podesta, Miguel Podesta (Jr) Summary Introduction Various surgical techniques have been proposed to treat pelvic fracture urethral distraction defects (PFUDDs) in children (Figure): primary alignment of the acute transected urethra, substitution procedures and delayed anastomosis urethroplasties (DAU) by perineal, elaborated perineal, transpubic or perineoabdominal/partial transpubic access. However, longterm follow-up of surgical correction for PFUDDS with DAU is infrequently reported in the literature. Purpose Long-term efficacy of DAU in children and adolescents with PFUDDs was evaluated. Other surgical methods used to accomplish tension-free DAU were also described. Material and methods We reviewed records of 49 male children aged 3.5e17.5 years (median 9.6) with PFUDDS who underwent DAU from 1980 to 2006. Median PFUDDs length was 3 cm (range 2e6). Six patients had prior failed treatments: anastomotic urethroplasties (5) and internal urethrotomy (1). Surgical access was transperineal in 28 cases and perineal/partial pubectomy in 21. Urethral rerouting was performed in 8 cases. Median follow-up was 6.5 years (range 5e22). Results On review median PFUDDS length in patients treated with primary cystostomy was 3 cm compared to those initially managed with urethral alignment (4 cm). Five patients treated with perineal DAU developed recurrent strictures at the anastomosis site, successfully managed with additional perineal/ partial pubectomy anastomosis (4 cases) and internal urethrotomy (1). Primary and overall success rate was 89, 7% and 100%, respectively. Urinary incontinence occurred in 9 cases. Two had overflow incontinence and performed selfcatheterization; 1 developed sphincter incontinence and required AUS placement, while 4 of 6 cases with mild stress incontinence achieved dryness at pubertal age. Retrospectively, associated bladder neck lesions at trauma time were noted in 5 patients. Three patients with erectile dysfunction before DAU remained impotent.

Discussion In children, several factors make management of PFUDDs more difficult than in adults: 1) restricted surgical access to reach a high lying proximal urethral end, 2) long distraction defects, 3) simultaneous bladder neck and membranous urethral lesions and 4) small urethral caliber. In our experience and that of others (Turner Warwick, 1989 and Ranjan, 2012), radiographic and endoscopic findings provide information on stricture features; however, the final choice of surgical exposure to restore urethral continuity is made at operative time based on PFUDD complexity. Perineal exposure usually allows performing DAU in 2 cm long PFUDDs. Ten percent of our patients treated with perineal DAU developed recurrent strictures attributed to inappropriate access selection or unrecognized PFUDD complexity. Failures were treated endoscopically (1) and by perineal/partial pubectomy anastomotic urethroplasty (4) with 100% final success. We used perineal/partial pubectomy DAU in 43% of the cases to excise pelvic scarring and bridge long urethral gaps, with urethral rerouting in 8 cases. Success rate of initial perineal and perineal/partial pubectomy anastomotic procedures was 82% and 100%, respectively. Koraitim (1997), Orabi (2008) and Ranjan (2012) reported excellent outcomes in children with either transperineal or transpubic anastomotic repair, as opposed to poor results in those undergoing substitution urethroplaties. Most reports rarely evaluate urinary incontinence after successful DAU. At the end of follow-up only 2 of our 9 initial incontinent cases remain with acceptable stress incontinence. Retrospectively, in 5 cases the original trauma comprised the bladder neck and the membranous sphincter mechanism. In our series erectile dysfunction after trauma did not change after DAU except in 1 patient who regained potency 1 year after repair. Study limitations All patients were referred after initial treatment was done elsewhere, thus they may represent the most severe PFUDDs cases. Additionally, erection dysfunction was not investigated in the kind of detail required due to patients’ age. Conclusions DAU has durable success rate for PFUDDs treatment in children with a healthy bulbar urethra. In childhood, additional surgical steps are frequently needed to achieve direct anastomotic repair.

http://dx.doi.org/10.1016/j.jpurol.2014.09.010 1477-5131/ª 2015 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved.

Please cite this article in press as: Podesta M, Podesta M, (Jr), Delayed surgical repair of posttraumatic posterior urethral distraction defects in children and adolescents: Long-term results, Journal of Pediatric Urology (2015), http://dx.doi.org/10.1016/ j.jpurol.2014.09.010

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M. Podesta, M. Podesta (Jr) completed 5e8 years follow-up and 19 patients 10e22 years. Information on trauma features and treatment modalities in the acute phase was obtained from the records of the receiving hospitals. Age at injury ranged from 2.4 to 15.7 years (median 8.1). Patient evaluation included complete medical history, renal/bladder ultrasonography and laboratory tests. Prior to DAU, all patients underwent simultaneous retrograde urethrogram and voiding cystogram in the oblique position, together with cystourethroscopy examinations under general anesthesia to evaluate location and urethral separation length, as well as bladder neck morphology.

Surgical technique

Figure Combined cystography and retrograde urethrography in a 10 year old patient with PFUDD. Note open bladder neck as patient attempts to void.

Introduction Surgical treatment of posttraumatic posterior urethral distraction defects (PPUDDs) continues to be a difficult problem to solve in urology. Various reports outline surgical techniques to treat PPUDDs in children [1e4]; however, there are very few reports of long-term follow-up of DAU in childhood [5,6]. In adults, the outcome of complete disrupted posterior urethra in association with pelvic fracture is a subprostatic urethral-distraction defect which can be repaired by 1stage perineal DAU, provided the anterior urethra is normal [6e9]. However, in children, circumferential rupture of the membranous urethra is commonly followed by a marked separation of the divided urethral ends, which often requires additional operative maneuvers to achieve tensionfree end to end anastomosis [5,10]. We reviewed long-term results in 49 consecutive children and adolescents with PPUDDs associated with pelvic fracture managed by 1-stage DAU with excision of the fibrous tissue between the separated urethral extremities. Furthermore, we describe extra surgical methods used to perform successful anastomotic repair.

Materials and methods Between 1980 and 2006, 49 consecutive children aged 3.5e17.5 years (median 9.6) with PFUDDs underwent DAU. These procedures were performed by 1 surgeon (M L P). Two patients were 15 years old, one 16, and one 17.5. Patients eligible for this study included children and adolescents with PPUDDs associated with a pelvic fracture whose anterior urethra was normal and had minimum follow-up of 5 years. All cases were referred to our hospital with a suprapubic cystostomy (SC) caused by urethral obliteration, and were followed for 5e22 years (median 6.5); 30 patients

Patients were placed in the standard lithotomy position with a beanbag under the buttocks. A perineal midline incision was made to expose and fully mobilize the bulbous urethra, which was transected at the distal margin of the urethral obliteration. Identification of the proximal end of the avulsed urethra was performed after excision of the pelvic fibrosis with the help of a descending sound passed through the suprapubic cystostomy tract and the prostatic urethra. Tension-free epithelial apposition of the healthy spatulated ends of the urethra was accomplished with interrupted absorbable suture over a soft fenestrated catheter that remained in place for 3 weeks, and was withdrawn after radiographic fenestrated catheter study confirmed a patent anastomosis. SC was removed once satisfactory micturition was achieved. If the urethral gap was too long to bridge or tension could not be taken off the anastomosis, we further mobilized the perineal urethra up to the suspensory ligament. This maneuver was combined with separation of the corporeal bodies. If the PPUDD still did not allow accurate restoration of urethral continuity, we proceeded with abdominal exposure and partial pubectomy, which permitted resection of the residual fibrosis, access to the proximal urethral end and anastomosis of the separated urethral extremities. In longer distraction defects, rerouting the urethra around the corporal bodies and through the space created after partial pubectomy allowed direct anastomosis without tension. Success criteria after urethroplasty included reestablishment of a homogenous urethral caliber, no further urethral dilatations, internal urethrotomies or additional surgical procedures. Postoperative follow-up involved clinical visits and retrograde urethrograms 1 month after DAU, repeated at 1, 3, 5, 7, 10 and 15 years thereafter. Patients with incontinence after urethroplasty underwent video urodynamic studies. Erectile dysfunction was identified in older children who were able to give information of their ability to have erections.

Ethical approval Approval was granted by the hospital ethics review board.

Please cite this article in press as: Podesta M, Podesta M, (Jr), Delayed surgical repair of posttraumatic posterior urethral distraction defects in children and adolescents: Long-term results, Journal of Pediatric Urology (2015), http://dx.doi.org/10.1016/ j.jpurol.2014.09.010

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Posttraumatic posterior urethral distraction

Results PFUDDs were the result of vehicular pedestrians accidents in 35 cases (71%), as occupants of a vehicle involved in an accident in 5 (10%), falls in 4 (8%), train accidents in 3 (6%) and farm work lesions in 2 (5%). Retrospectively, acute management comprised stabilization of the patient, followed by SC in 35 (71%) cases and primary realignment with simultaneous SC in 14. Alignment techniques varied; the most frequent methods used were sound to sound or combined antegrade tied to retrograde catheter. Six patients had prior failed treatments, 5 anastomotic urethroplasties (4 transperineally and 1 transpubically) and 1 internal urethrotomy. Retrospectively, 24 (49%) patients had multiple associated injuries: ruptured bladder in 9 (bladder neck lesions in 6), musculoskeletal injury in 8, rectal injury in 5, and chest injury in 6 and nerve avulsion in 2. On review of the operation notes, preoperative radiographic images and endoscopy findings confirmed in all cases complete disruption of the posterior urethra below the verumontanum (Fig. 1). Estimated urethral obliteration length ranged from 2 to 6 cm (median 3). Fourteen patients were recorded as having ring disruption pelvic fractures. DAU was performed transperineally in 28 patients (57%) and in 21 (43%) via a perineo-abdominal partial transpubic approach. Supracrural urethral rerouting was required in 8 of the last 21 patients. No orthopedic disability was observed after perineal/partial pubectomy exposures. In the early postoperative period, stricture recurred at the site of the anastomosis in 5 (10%) patients, who underwent perineal DAU. One of these patients had undergone a prior transpubic anastomotic procedure before referral to our institution. Four of the 5 failures developed

1.e3 complete obliterated strictures, whose length ranged from 2.8 to 3.5 cm (median 3), and 1 a short continuity stricture. The failed cases were managed successfully by repeat anastomotic repair through perineal-partial pubectomy exposure (4), with urethral rerouting in 1 case and by internal urethrotomy (1). No further urethral dilatation or additional surgical procedures were needed in these cases. Median (range) age at stricture recurrence was 8.8 years (7.4e11.2) and at internal urethrotomy and repeat urethroplasty 9.3 years (8.1e11.7). Median (range) postoperative follow-up was 10 years (6e17). Consequently, primary and overall stricture-free success rate for DAU was 90% (44/49 patients) and 100%, respectively (Fig. 2). Comparison of patients’ features and DAU outcomes between patients treated initially elsewhere with SC alone and primary realignment is depicted in Table 1. Nine patients had urinary incontinence after successful DAU: overflow incontinence secondary to an acontractile bladder in 2, mild stress incontinence in 6 and total urinary leakage in 1. Retrospectively, the first 2 patients had bucket-handle fractures and are now continent on selfcatheterization. Four of the 6 stress incontinent patients and 1 with total incontinence suffered double injuries comprising the bladder neck and the membranous urethra at the original trauma. Four patients with stress incontinence achieved dryness when they reached puberty and the totally incontinent case required artificial urinary sphincter placement. The remaining 2 patients with stress incontinence are being treated with imipramine and do not need protection from leakage. Erectile dysfunction was present preoperatively and remained unchanged after DAU in 3 patients. Retrospectively, these cases had vertical shear fractures (2) and lateral compression fracture (1). One further patient with

Figure 1 Preoperative combined cystography and retrograde urethrography. A, PFUDD in a 5-year-old boy. B, Seven-year-old patient showing severe PPUDD.

Please cite this article in press as: Podesta M, Podesta M, (Jr), Delayed surgical repair of posttraumatic posterior urethral distraction defects in children and adolescents: Long-term results, Journal of Pediatric Urology (2015), http://dx.doi.org/10.1016/ j.jpurol.2014.09.010

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M. Podesta, M. Podesta (Jr)

Figure 2 Retrograde urethrography. A, Eight-year postoperative perineal DAU. B, Twenty-two-years postoperative combined perineal/partial transpubic repair. Note the anastomosis below the verumontanum.

erectile dysfunction regained penile erections 2 years after urethroplasty.

Discussion Treatment of PPUDDs is complex and its management has evolved in recent decades. Approaches to treat PPUDDs with internal urethrotomy and dilatations are unlikely to provide a lasting cure [12]. Various surgical techniques and modifications have been reported to manage this disease: primary alignment of the acute disrupted urethra [13], substitution skin flap procedures [14] and tension-free mucosa to mucosa anastomosis repairs by perineal [7e9], elaborated perineal [10], transpubic [6] or perinealabdominal partial transpubic access [7,15]. Nerli et al. reported that 6 of 12 children undergoing primary realignment needed additional endoscopic urethrotomies, with 3 of these cases requiring urethroplasty to manage resultant stricture [13]. Although these authors Table 1 Comparison of patients’ characteristics and DAU outcomes between cases treated in the acute trauma phase with SC alone and primary realignment.

No pts (%) Median age (yrs) At injury (range) At reconstruction (range) Median cm PFUDD (range) Approach Perineal (pts) Combined/partial pubectomy (pts) Re-routing (pts) Incontinence (pts) Erectile dysfunction (pts)

SC

Primary alignment

35 (71)

14 (29)

9 (2.4e14.2) 9.8 (3.5e15)

7.4 (4.1e15.7) 8.1 (5.3e17.5)

3 (2e5)

4 (2e6)

23 12

5 9

4 5 2

1 4 1

recommended immediate realignment to shorten the distraction defect, long-term results were poor [13,16,17]. In our study, comparison outcomes between patients treated initially elsewhere with SC and with urethral realignment showed that the latter patients required less rerouting maneuvers and developed less recurrent strictures. However, there was no marked difference between urethral distraction defect length in patients treated initially with SC compared with those managed with urethral alignment (Table 1). In children, although PFUDDs pathogenesis tends to follow a similar pattern to those in adults, several factors make management of urethral ruptures more complicated. These difficulties can be related to: 1) a restricted surgical field to reach a high lying proximal urethral end surrounded by dense fibrosis, 2) long distraction defects involving the membranous urethra and the proximal bulbous urethra, 3) concomitant bladder neck and membranous urethral lesions and 4) small size urethral caliber [5,11,17]. In our experience, although radiographic and endoscopic findings provided information on stricture location and length, the decision for a combined perineal-abdominal partial pubectomy exposure was taken at the time of surgery, based on the severity of local anatomic alterations and PFUDD complexity. This concept has been previously addressed by other authors [7,9,17]. Koraitim reported excellent outcomes in children with either transperineal (93%) or transpubic anastomotic repair (91%), as opposed to a failure rate of 54% with urethroscrotal inlay procedures [5]. Orabi et al. have also shown very good results in 47 boys with PFUDDs treated by perineal anastomotic repair (40), combined inferior pubectomy (3) or transpubic approach (4) and poor results in those undergoing substitution urethroplasties [1]. In 27 children with PFUDDs managed with perineal DAU, Singla et al. reported repeat strictures in 7 patients which were successfully managed endoscopically (4) and by redo anastomotic urethroplasty (2). Thus, nowadays excellent results can be expected from 1-stage DAU in adults and in children with PFUDDs, provided the anterior urethra is healthy [9,10,17]. In our patients when PFUDDs was 2 cm long, with reduced pelvic fibrosis, we mobilized the bulbar urethra and accomplished a perineal DAU. However, early in this

Please cite this article in press as: Podesta M, Podesta M, (Jr), Delayed surgical repair of posttraumatic posterior urethral distraction defects in children and adolescents: Long-term results, Journal of Pediatric Urology (2015), http://dx.doi.org/10.1016/ j.jpurol.2014.09.010

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Posttraumatic posterior urethral distraction series 5 patients treated by this approach had recurrent strictures at the site of the anastomosis. Reasons for failure can be attributed to initial surgeon inexperience, inadequate selection of surgical access when treating long PFUDDs or performance of urethral anastomosis under tension. In these patients, median length of the urethral distraction defect was 3 cm. All failed cases were successfully repaired by internal urethrotomy (1) and by perineal-abdominal partial pubectomy anastomotic urethroplasty (4) with additional supracrural rerouting in 1 case. In 23 patients with restricted perineal access, severe pelvic fibrosis and long PPUDDs, we opted for a perinealabdominal partial pubectomy approach to achieve good exposure, excise all pelvic fibrosis and straighten the curve of the bulbar urethra [1,14]. In 8 cases supracrural urethral rerouting was required to bridge these long urethral gaps. Other authors have previously described the advantages of the combined-partial pubectomy access for long PPUDDs in children [5,11]. In our hands, intercavernosal septum division, frequently used in adult patients [9,18,19], did not shorten sufficiently the distance between the separated urethral ends to perform tension-free anastomosis in prepubertal patients. Thus, primary and overall final stricture-free success rate of these 49 patients was observed in 89.7% and 100%, respectively with median follow-up of 6.5 years (range 5e22). Primary success rate was 82% in patients who underwent perineal DAU, and 100% in those treated with perineal/partial pubectomy dissection. In our series, 9 patients developed urinary incontinence after DAU. Two patients with acontractile bladders had initially severe ring disruption pelvic fractures and needed self-catheterization to be dry. One case developed total urinary leakage and required AUS placement, while 4 of 6 cases with mild stress incontinence achieved dryness with prostatic growth at pubertal age. In contrast with our results, Orabi et al., as well as Das et al., reported excellent continence status in their series after perineal or transpubic anastomotic urethroplasty [1,3]. Similarly, Koraitim observed only 3 out of 68 patients with postoperative incontinence [5]. Urinary incontinence seems more likely related to associated bladder neck lesions and severity of pelvic fracture rather than to primary treatment or deferred urethral reconstruction. Retrospectively, in our study associated bladder neck injury occurred at the initial trauma in 4 of the 6 patients with mild stress incontinence and in 1 with total urine leakage. Lastly, absent erections following PPUDDs occurred in 4 patients; we speculated they were related to the original trauma, as this disability was present in 3 cases before and after DAU. Only 1 case regained erectile function 2 years after repair. Midline careful dissection during surgery was performed to avoid mobilization of the proximal urethral end and damage to neurovascular structures, located postero-laterally to the subprostatic urethra. Limitations in this study need to be considered. All patients in this series were referred to our hospital after initial treatment, possibly representing the most severe PPUDD cases. Furthermore, decision on primary management modality depended on the urologist who initially

1.e5 treated each patient. Six patients had failed anastomotic repairs performed elsewhere; only one of these had stricture recurrence after perineal DAU performed in our hospital. Further, the exact mechanism of erection dysfunction was not investigated in the kind of detail required because of the age of these patients.

Conclusions Our data confirm DAU ensure long-term success in the management of PFUDD in children, as long as the bulbar urethra is normal. When distraction defects are significantly long or extensive pelvic fibrosis is present, perineal/ partial pubectomy access offers an effective exposure to restore urethral continuity without tension.

Conflict of interest None.

Funding None.

References [1] Orabi S, Badawy H, Saad A, Youssef M, Hanno AJ. Post-traumatic posterior urethral stricture in children: how to achieve a successful repair. J Pediatr Urol 2008;4:290e4. [2] Singla M, Jha MS, Murugagnandam K, Srivastava A, Ansari MS, Mandhani A, et al. Posttraumatic posterior urethral strictures in children- management and intermediate-term follow-up in tertiary care center. Urology 2008;72(3):540e3. [3] Das K, Charles AR, Alladi A, Rao S, D’Cruz AJ. Traumatic posterior urethral disruption in boys: experience with the perineal/perineal-transpubic approach in ten cases. Pediatr Surg Int 2004;20:449e54. [4] Al-Rifael MA, Gaafar S, Abdel-Rahman M. Management of posterior urethral strictures secondary to pelvic fractures in children. J Urol 1991;145:353e6. [5] Koraitim MM. Posttraumatic posterior urethral strictures in children: a 20 year experience. J Urol 1997;157:641e5. [6] Patil UB. Long term results of transpubic prostatomembranous urethroplasty in children. J Urol 1986;136:286e8. [7] Turner Warwick R. Prevention of complications resulting from pelvic fracture urethral injuries e and from surgical management. Urol Clin North Am 1989;16(2):335e8. [8] Mundy AR. Urethroplasty for posterior urethral strictures. Br J Urol 1996;78:243e7. [9] Morey AF, McAninch JW. Reconstruction of posterior urethral disruption injuries: outcomes of analysis in 82 patients. J Urol 1997;157:506e10. [10] Webster GD, Ramon J. Repair of pelvic fracture posterior urethral defects using an elaborated perineal approach: experience with 74 cases. J Urol 1991;145:744e8. [11] Podesta ML. Use of the perineal and perineal-abdominal (transpubic) approach for delayed management of pelvic fracture urethral obliterative strictures in children: long-term outcome. J Urol 1998;160:160e4. [12] Hsiao KC, Baez-Trinidad L, Lendvay T, Smith EA, Broecker B, Scherz H, et al. Direct vision internal urethrotomy for the treatment of pediatric urethral strictures: analysis of 50 patients. J Urol 2003;170:952e3.

Please cite this article in press as: Podesta M, Podesta M, (Jr), Delayed surgical repair of posttraumatic posterior urethral distraction defects in children and adolescents: Long-term results, Journal of Pediatric Urology (2015), http://dx.doi.org/10.1016/ j.jpurol.2014.09.010

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1.e6 [13] Nerli RB, Koura AC, Ravish IR, Amarkhed SS, Prabha V, Alur SB. Posterior urethral injury in male children: long-term followup. J Pediatr Urol 2008;4:154e9. [14] Turner Warwick R. The repair of urethral strictures in the region of the membranous urethra. J Urol 1968;100: 303e14. [15] Koraitim MM. On the art of anastomotic posterior urethroplasty: a 27 year experience. J Urol 2005;173:135e9. [16] Husmann D. Pediatric genitourinary trauma. In: Wein AJ, Kavousi LR, Novick AC, Partin AW, Peters CA, editors.

M. Podesta, M. Podesta (Jr) Campbell e Walsh Urology. Philadelphia: Saunders Elsevier; 2007. p. 3939e45. [17] Ranjan P, Ansari MS, Singh M, Chipde SS, Singh R, Kapoor R. Post-traumatic urethral strictures in children: what have we learned over years? J Pediatr Urol 2012;8:234e9. [18] Andrich DE, O Malley KJ, Summerton DJ, Greenwell TJ, Mundy AR. The type of urethroplasty for a pelvic fracture urethral distraction defect cannot be predicted preoperatively. J Urol 2003;170:464e7. [19] Chapple CR. Urethral injury. BJU Int 2000;86:318e26.

Please cite this article in press as: Podesta M, Podesta M, (Jr), Delayed surgical repair of posttraumatic posterior urethral distraction defects in children and adolescents: Long-term results, Journal of Pediatric Urology (2015), http://dx.doi.org/10.1016/ j.jpurol.2014.09.010

Delayed surgical repair of posttraumatic posterior urethral distraction defects in children and adolescents: long-term results.

Various surgical techniques have been proposed to treat pelvic fracture urethral distraction defects (PFUDDs) in children (Figure): primary alignment ...
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