Treatment Bladder: By C. Granata,
of Vesico-Ureteric Reflux in Children With Neuropathic A Comparison of Surgical and Endoscopic Correction P. Buffa, E. Di Rovasenda,
G. Mattioli, Genova,
P.L. Scarsi, Italy
E. Podesta,
R Dodero,
and V. Jasonni
/3ackground/furpose: Vesico-ureteric reflux (VUR) is a common problem in children with neuropathic bladder. Lesserdegree VUR may be manageable by intermittent catheterization or by anticholinergics, but higher grades usually require surgical treatment. If left untreated, two thirds of such patients may experience deterioration of the upper renal tracts. The aim of this study was to compare the results of the STING (Subureteric Teflon Injection) technique with surgical ureteric reimplantation as treatment for VUR in neuropathic bladder. Methods: From January 1981 to December 1996,58 children with NB (81 ureters) were treated for VUR. STING and Cohen ureteroneocystotomy were performed in 40 and 41 ureters, respectively. Mean age was 4.5 years (STING) and 5.1 years (Cohen). Results: Twenty-nine of 40 refluxing ureters (72.5%) were cured by STING, whereas Cohen eradicated reflux in 39 of 41 ureters (95.5%). No complications were observed in either group. AH the ureters in which STING failed were treated successfully by Cohen ureteroneocystostomy. The 2 ureters
V
ESICOURETERAL REFLUX (VUR) is present in 15% to 50% of children with neuropathic bladder (NB). 1 Clean intermittent catheterization and anticholinergic agents can lead to spontaneous resolution in 43% to 58% of cases of mild-degree VUR.2.3 However, highergrade VUR usually requires a more aggressive treatment to avoid the risk of progressive deterioration of the renal function caused by pyelonephritic scsu-ring. Previously, ileal diversion was thought to be the most effective method of managing reflux in the NB,4 but recurrent infections, upper urinary tract deterioration, and calculi are now well-known complications of this approach.5 In 1976, Jeffs et al’ showed that ureteric reimplantation was a suitable technique in children with NB and VUR. In the following years, ureteric reimplantation was adopted in other centers and yielded increasingly good results provided that the bladder was not hyperreflexic and of acceptable compliance. 6 lo Since 1984, subureteric injection of Teflon paste (STING) has been adopted as a less invasive method of curing primary VUR. Currently, From the Departments of Paediatric Surgery and Emergency Surgery, Giannbm Gaslini Hospital for Sick Children, Geneva, Italy. Address reprint requests to C. Granata, MD, Via Pontiroli 615, 16031 Bogliasco, Geneva, Italy. Copyright o 1999 by WB. Saurlders Company 0022-3468/99/3412-0020$03.00/O 1836
still refluxing after surgical reimplantation were cured successfully by a single STING. The mean follow-up was 6.8 years in the Cohen group and 4.8 years in the STING group. During follow-up, no recurrence was observed in patients cured by open reimplantation. In the STING group, 2 previously cured ureters showed recurrence of VUR: both were treated successfully by a further STING. Conclusions: Open ureteral reimplantation is more effective than STING in correcting VUR in children with neuropathic bladder dysfunction. Nevertheless, the good success rate, the relative technical simplicity, outpatient nature, and rapid recovery point to STING as a safe and effective procedure for the initial treatment of VUR. Failure of STING does not preclude a successful open operation. J Pediatr Surg 34:1836-1838. Copyright o ‘1999 by W.B. Saunders Company. INDEX ureteric
WORDS: Neuropathic bladder, vesico-ureteric reimplantation, subureteric teflon injection.
reflux,
STING is a widespread technique, and successful results have been reported in more than 90% of cases of normal bladder.” Nevertheless, only a few reports are available on STING in the treatment of VUR in NB,3,‘2-16some of which have involved a small number of patients and a short follow-up. The aim of our study was to verify and compare the efficacy of ureteric reimplantation and STING in terms of success,long-term results, and complications by retrospectively reviewing our 15-year experience in 58 patients with 81 refluxing ureters. MATERIALS
AND
METHODS
From January 1981 to December 1996, 81 ureters in 58 children (41 girls, 17 boys; mean age, 4.8 years; range, 4 months to 16 years) with NB mainly caused by myelomeningocele underwent invasive treatment for VUR. High-grade reflux (grades III to V), breakthrough infections dming a 12- to 24.month trial of antibiotic prophylaxis, persistence of low-grade reflux despite intermittent catheterization and anticholinergics were indications for invasive correction. Grade I VUR was treated only if concomitant with a contralateral higher grade reflux. Reflux was graded according to the classification of the International Reflux Study Committee. Preoperative reflux was assessed by conventional voiding cystourethrogram (VCUG) in all patients. The underlying pattern of NB was assessed routinely and treated accordingly to allow complete bladder emptying, to improve compliance, and to abate uninhibited contractions. Patients with at least more than 60% of the expected bladder capacity and bladder compliance normal or moderately comproJournal
of Pediatric
Surgery,
Vol 34, No 12 (December),
1999: pp 1836-1838
VUR
IN THE NEUROPATHIC
BLADDER
1837
mised (compliance >6 r&/cm H20) underwent Cohen reimplantation or STING as the only surgical procedure to correct VUR. Simple STING or Cohen reimplantation were considered not adequate in patients whose bladder capacity was less than 60% of the expected value or compliance less than 6 mL/cm H20) despite anticholinergics agents. Bladder augmentation (colocistoplasty or autoaugmentation) and Cohen reimplantation were performed in these patients. Our study does not deal with this group of patients, because the aim of our study was to compare the efficacy of Cohen reimplantation and STING in subjects who did not undergo any other bladder surgical procedure. During the period from 1981 through 1990, all the patients with indications for surgical correction of VUR and a bladder with capacity and compliance normal or moderately compromised were treated by Cohen cross-trigonal ureteroneocystostomy. Since lY91, we have performed STING as first-choice treatment in patients with grade III and IV VUR or with grade II reflux not responding to antimicrobials. Exclusion criteria were severe cystitis cystica, diverticula, completely duplicated ureters. and severely compromised bladder capacity or compliance. In the case of persistence of reflux after 2 injections of Teflon paste, no further attempts were made, and reimplantation generally was performed in these patients. Cystomanometry was carried out in 2X of the 3 1 patients who underwent STING. Success or failure of treatment was evaluated by means of VCUG and ultrasound scan 3 months after the procedure; all children subsequently underwent direct radionuclide cystography at 1 and 3 years and ultrasound scan every 6 months. A procedure was considered successful only when the refluxing ureter was completely cured. Antibiotic prophylaxis was discontinued only when the S-month VCUG showed complete correction of reflux. All patients, both in the STlNG and Cohen groups. undenvent full preoperative antibiotic prophylaxis to avoid infective complications because many NBS are potentially infected.
RESULTS
STING STING was performed in 40 refluxing ureters (31 children, mean age, 4.5 years). After a single injection, reflux was cured in 24 refluxing units (60%). A second injection was carried out in 9 of the remaining 16 refluxing ureters: in 5 ureters reflux was eradicated, 2 downgraded, and 2 unaffected. Therefore, 29 of 40 units (72.5%) were cured after 1 or 2 injections. In no case was a third injection attempted. The success rate of STING was 100% in grade I, 75% in grade II, 75% in grade III, and 33% in grade IV, respectively (Table 1). The volume of teflon paste injected behind each ureteric orifice varied between 0.2 and 0.6 mL: in most cases less than 0.4 mL was required. In 6 patients with high-grade reflux, STING was atTable
1. STING
and Cohen
Reimplantation
Groups
No. Ureters (success/total/% Preoperative Reflux Grade
STING (1 or2 Injections)
Reimplantation
I
l/l
II
12/16
(75)
10/l 1 (90.9)
Ill
15/20
(75)
14114 (93.3)
113 (33.3)
12/13 (92.3)
IV V Total NOTE.
success)
Results
(100)
l/l
-
2/2 (100)
29140 (72.5)
according
to preoperative
(100)
39/41
refluxgrade.
(95.1)
tempted, but severe trabeculation with trigonal cystitis prevented identification of the ureteric orifices, and, therefore, the procedure was abandoned. Subsequently, these patients underwent ureteric reimplantation. The mean follow-up period was 4.8 years (range, 1 to 7 years). During follow-up, VUR relapsed in 2 previously cured ureters after 18 and 24 months, respectively. Recurrence of VUR was associated with recurrent breakthrough infections in both cases. In 95% of children, hospitalization lasted 1.5 days. No complications were observed, excepted mild postoperative hematuria observed in 3 patients. Moderate impairment of bladder compliance did not correlate with success or failure of STING in the 28 children in whom a urodynamic study was done. In patients with a bladder compliance less than 10 mL/cm HZ0 and more than 6 mL/cm H,O, STING was successful in 11 cases and unsuccessful in 10, whereas in patients with a compliance greater than 10 n&/cm HzO, STING was successful in 4 and unsuccessful in 3. Cohen Forty-one ureters (27 children, mean age, 5.1 years) were treated with Cohen ureteroneocystostomy. Eleven of these underwent reimplantation after failure of STING; ureteroneocystostomy was successful in all of them. Globally, of the 41 reimplanted ureters, 39 (95.1%) were cured after surgery. Cohen reimplantation failed to cure reflux in a child with bilateral moderate-severe reflux and adequate bladder compliance. A single STING corrected reflux in both ureters. The mean follow-up period was 6.8 years (range, 1 to 11 years). During follow-up, we did not observe any relapse of VUR or complications related to the Cohen procedure. The successrate of Cohen reimplantation was 100% in grade I, 90.9% in grade II, 100% in grade III, 92.3% in grade IV, and 100% in grade V. respectively (Table 1). DISCUSSION
Since the pioneering study of Jeffs et al,’ who reported an 87% success rate for ureteric reimplantation to treat VUR in 23 children with NB, several other studies have been published. In 1979 Belloli et al6 observed a cure rate of 85% in 26 reimplanted ureters. In 1981 Sullivan et al7 reported 100% success in a small series of 5 patients. In 1983, Kaplan and Firlit* observed a 96% success rate in 25 children. Subsequently, in 1986 Sidi et al9 reported 100% correction in a series of 12 patients, and in 1993, Merlini et ali0 noted 82% success. Recently, Engel et al3 described a cure rate of 83.7% in a series of 49 refluxing ureters treated with Cohen ureteroneocystostomy. Our data on Cohen reimplantation to treat of VUR in children with NB are not dissimilar from these previous results. However, it may be difficult to make a comparison,
1838
GRANATA
because the cure rate reported by some investigators is the result of a combination of different surgical techniques (Politano-Leadbetter, Cohen, Glen-Anderson, Paquin) adopted in a single series of patients. During follow-up, we did not observe any relapse in children cured by ureteric reimplantation. This finding probably is related to our choice to perform simple ureteric reimplantation exclusively in patients with adequate bladder compliance and capacity, because recurrence of VUR after ureteric reimplantation without bladder augmentation is frequent in a poorly compliant bladder.” Data on STING as an alternative treatment for VUR in NB are still scarce, although STING has been used for almost 15 years now. Early results in 1986 from Puri and Guiney12 and Kaplan et all3 showed reflux resolution in 86.7% and 70.6% of ureters, respectively. Subsequently, in 1988, Quinn et all4 observed resolution in 90% of 41 ureters, whereas in 1991, Kaminetsky and Hanna15 reported a success rate of 70% in 20 ureters. However, in most of these studies follow-up was no longer than 2 years. Recently, Misra et all6 and Engel et al3 obtained reflux resolution in 82% of 69 ureters and in 61% of 60 ureters with a mean follow-up of 4.1 and 4.5 years, respectively. Our overall cure rate of 72.5% is comparable with these in previous reports and might well have been higher if we had tried a third injection in the ureters still refluxing after two STING treatments. However, we preferred not to perform more than two STING treatments to avoid the rare, but possible, complication of obstruction at the vesico-ureteric junction. To the best of our knowledge, follow-up in our study is the longest reported in the literature so far. In our series,
ET AL
VUR relapsed in 2 previously cured ureters after 18 months and 24 months, respectively. Misra et alI6 and Engel et al3 noted recurrence of VUR in 4 (7%) of 57 previously cured ureters and in 2 (5.4%) of 37 cured ureters, respectively; recurrence was a late complication, appearing in 5 of these 6 ureters more than 3 years after the initial success with STING. Therefore, long-term follow-up is mandatory in these patients because of the risk of late recurrence. Failure of STING was more likely with high-grade VUR, whereas open ureteric reimplantation was not affected by the severity of VUR, as shown by ours and other investigators’ data. Interestingly, STING was not affected by a moderate impairment of bladder compliance; our findings confirm the similar preliminary observations by Engels et a1.3 STING yields acceptable results in patients with NB and VUR, but the excellent results obtained in selected cases with Cohen reimplantation suggest that this technique is still the benchmark treatment. However, open ureteric reimplantation is a major surgical procedure that may occasionally be difficult to perform in spina bifida patients. By contrast, STING is a quick and easy procedure, which avoids the discomfort and long hospitalization typical of an open procedure. Failure of STING does not preclude further injections and, if necessary, does not make open reimplantation less successful or more difficult to perform. These characteristics and the overall good results suggest that, in children with VUR and NB without severe impairment of capacity and compliance, STING should be considered the first-choice treatment in grade IV, grade III, and persisting grade II reflux.
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