Journal of Pediatric Urology (2014) 10, 1227e1231
Vesico-cutaneous fistula: A simple method for continent urinary diversion W. Yang a, P.-Y. Chang a,*, J.-Y. Lai a, C.-H. Cheng b, M.-H. Tseng b a Department of Pediatric Surgery, Chang Gung Children’s Hospital, Chang Gung University, College of Medicine, Linkou, Taiwan b Department of Pediatrics, Chang Gung Children’s Hospital, Chang Gung University, College of Medicine, Linkou, Taiwan
Received 3 March 2014; accepted 5 July 2014
Available online 22 July 2014
KEYWORDS Urinary diversion; Neurogenic bladder; Vesical fistula; Urethral stricture
Abstract Introduction: Patients with lower urinary tract anomalies or neurogenic disorders often suffer from voiding difficulties. Clean intermittent catheterization (CIC) is effective for bladder drainage; however, this is often painful. Transurethral catheterization is also impossible in patients with urethral stricture. A Mitrofanoff conduit may solve some of these problems, but a few disadvantages have been reported, including: difficult surgical techniques and frequent operative complications. A vesicostomy is easy to perform but persistent urine leak over the abdomen and diaper rash can be annoying. A better way to achieve continent urinary diversion is indicated. Method: Between December 01 1998 and December 31 2013, six patients underwent a vesicocutaneous fistula for CIC. The etiologies included urethral stricture (n Z 2) and neurogenic bladder (n Z 4). The fistula was created at the bladder dome with only the muscle layer of the bladder sutured to the skin. A Foley catheter was left in place for at least two weeks to prevent stoma stricture. After removing the Foley catheter, regular CIC from the fistula was performed every 2 h during the daytime with a Fr. 10e12 feeding tube, depending on the patient’s age. Further stenting during the night in the first six months was necessary to prevent early closure of the fistula. Patients were followed with periodic renal ultrasonography, blood tests and urinalysis in the outpatient department. Results: Follow-up ranged from 6 months to 16 years. All patients showed improvements in hydronephrosis. Decreased UTI frequency was seen in five patients. Renal function was normal in five patients, whilst the other suffered from chronic renal failure preoperatively. Only one patient had occasional mild urine leakage from the stoma at night, which was once in two weeks. No patient experienced painful or difficult catheterization and CIC becomes easy, even by young children.
* Corresponding author. Department of Pediatric Surgery, Chang Gung Children’s Hospital, Chang Gung University, College of Medicine, Linkou, No. 5, Fusing St, Gueishan Township, Taoyuan County 333, Taiwan. Tel.: þ886 3 3281200; fax: þ886 3 3285056. E-mail addresses: [email protected] (W.Yang), [email protected] (P.-Y.Chang), [email protected] (J.-Y.Lai), pednephcheng@ cgmh.org.tw (C.-H.Cheng), [email protected] (M.-H.Tseng). http://dx.doi.org/10.1016/j.jpurol.2014.07.002 1477-5131/ª 2014 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved.
1228
W. Yang et al. Conclusions: The vesico-cutaneous fistula is a simple, effective and tolerable method for CIC. It may be a substitute for or a transition to a Mitrofanoff conduit in some patients. ª 2014 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved.
Introduction The optimal treatment for children with neurogenic bladder and other urinary difficulties has not been established. Renal function deterioration may be related to VUR, hydronephrosis and recurrent UTI. End-stage renal disease can develop if a child is left untreated [1]. Clean intermittent catheterization (CIC) has been used widely in patients with neurogenic bladder (NB). Although a low complication rate and good tolerance of CIC have been reported after long-term follow-up, bleeding, urethral stricture or a false tract can still occur [2,3]. A subgroup of patients can suffer from recurrent UTI, even under regular CIC and prophylactic antibiotic treatment [4]. In patients with lower urinary tract anomalies, such as urethral stricture, performing CIC is impossible. A urinary diversion with a Mitrofanoff conduit has disadvantages, including: difficult surgical techniques and a high rate of operative complications. A traditional vesicostomy, although preserving upper urinary tract function, is often difficult for patients to accept due to the resulting urinary incontinence and risk of diaper dermatitis [5,6]. In the present study, a simple and nearly continent vesico-cutaneous fistula (VCF) was performed instead of a traditional vesicostomy in children who were under the CIC program. The present study reports on the experiences of six cases.
peritoneal reflection could be seen. After pushing away the peritoneum and the perivesical fat, the bladder was grasped at the dome and pulled through the wound. The bladder was opened and evacuated through a 0.5 cm longitudinal incision. Four to six stitches were used to fasten the outer layer of the bladder wall to the Scarpa’s fascia and the skin; the mucosa should not be sutured to the skin. A French 12 Foley catheter was left in place for two weeks in order to prevent early closure of the VCF. After removing the Foley catheter, regular CIC from the fistula was performed every 2 h during the daytime with a 10e12 Fr. feeding tube, depending on the patient’s age. The tube was inserted as a stent and left open for drainage at night for the first six months after the VCF had been created. Once the fistula had stabilized, no further stenting was required after the first six months.
Follow-up Patients were followed with periodic renal ultrasonography, blood tests and urinalysis in the outpatient department. VCUG, urodynamic studies and magnetic resonance urography (MRU) were performed as needed. Improvement was defined as improved hydronephrosis and hydroureter on renal echo, decreased frequency of febrile UTI or improving creatinine level. The severity of hydronephrosis was evaluated using the Society for Fetal Urology grading system [7].
Materials and methods After obtaining Institutional Review Board approval, a retrospective chart review of all patients who underwent a VCF between December 01 1998 and December 31 2013 was conducted.
Patients Sixty-six neurogenic bladder patients were treated at the present hospital between December 1998 and December 2013. Five of these patients underwent VCF procedures. Indications included: recurrent UTI, despite regular CIC from the urethra and prophylactic antibiotics (n Z 3), and worsening hydronephrosis and renal function (n Z 2). For patients who suffered from neurogenic bladder, a bladder capacity exceeding 150 ml should be proved during CIC. Another two urethral strictures were recruited due to difficulty with CIC from the urethra.
Operation method A 2.0 cm incision was made at the midpoint between the umbilicus and the pubic bone. The underlying fascia was incised and the rectus was retracted laterally until the
Results Four males and three females underwent the procedure; one was excluded as lost to follow-up. The characteristics and results of the six patients are described in Table 1. The mean age at surgery was 7.17 years (range 18 dayse22 years). Two patients (33.3%) had congenital urethral strictures. Four patients suffered from bladder dysfunction for the following reasons: meningomyelocele (n Z 2), cerebral palsy (n Z 1) and non-neurogenic neurogenic bladder (Hinman syndrome, n Z 1). Although CIC and prophylactic antibiotics were used for all patients with neurogenic bladders, febrile UTI that needed hospitalization for treatment (more than two episodes in six months) remained a common problem. All of the patients who underwent the operation showed improved hydronephrosis in the follow-up renal echo assessments. The frequency for febrile UTI declined and the nocturnal diuresis decreased gradually during the first six months of nocturnal stenting. One patient suffered from chronic renal failure and had a creatinine level around 7.0e8.0 mg/dl before the operation. The creatinine level improved to 5.0 mg/dl after three-months of regular CIC from the VCF. The rest of the
A simple method for continent urinary diversion Table 1
1229
Patients and outcomes.
Patient
1
2
3
4
5
6
Gender Diagnosis Age at surgery
M US1 18 days
M US 13 years
F NGB-12 4 months
F NGB3 22 years
F NGB 5 years
Preoperative hydronephrosis (left/right)a Postoperative hydronephrosis (left/right)a Complications
1/3
3/3
3/3
3/1
M NGB 1st: 2 years 8 months 2nd: 3 years 2 months 3/3
1/1
1/0
1/0
2/1
1e2/1e2
1/1
None
None
None
Fistula stenosis needed 2nd operation
None
16 years (fistula closure at 7 years old)
8 months
Urine leak at night, once in 2 weeks 11 years
7 months
9 months
3 months
Follow-up
2/2
Abbreviations: US (urethral stricture); NGB-1(neurogenic bladder of unknown cause); NGB (neurogenic bladder). The SFU system was graded 0-4 (0 Z no hydronephrosis; 1 Z visualized only renal pelvis; 2 Z plus a few caliceal dilatation; 3 Z all calyceal dilatation; 4 Z plus parenchymal thinning). a The severity of hydronephrosis was evaluated according to the Society for Fetal Urology grading system.
patients had creatinine levels below 1 mg/dl before the operation and these remained stable afterward. The mean follow-up was 4.8 years (range 3 months-16 years). Two of them had been followed for more than 10 years. No UTI episode was seen in the female who had a non-neurogenic neurogenic bladder over the past three years. The male who suffered from urethra duplication and stricture had his fistula closed at seven-years old, after completed treatment for a urethral stricture. He is now 16-years old and the one or two episodes of UTI that he has had in the last year were managed with oral antibiotics alone. No prophylactic antibiotic was given to either of these patients. All of the patients could keep dry during the day and no bag or pad was needed (Fig. 1). One patient (16.7%) underwent surgical revision of the VCF because the patient’s mother was unwilling to perform CIC via the VCF after the Foley catheter was removed. After explaining the process clearly, regular CIC was performed smoothly after the second operation; no complications have occurred since. One patient (16.7%) suffered from occasional urine leakage at night (once in two weeks). The leakage was easily managed by covering the fistula with a pad at night. Two complications occurred in two of the six patients (33.3%), but both were minor and could be managed readily. No patient suffered from dermatitis or prolapse.
Discussion Deterioration of upper urinary tract function in children who have abnormal lower urinary tract function can be as high as 70% [8]. The renal failure rate is 40e50% in these patients and rises steeply with age [1]. The patients often benefit from regular CIC and this should start at an early age [9,10]. However, approximately 10% of these patients suffer from decreased upper urinary tract function [11]. Furthermore, the CIC technique is relatively difficult for young or mentally
disabled children. As children grows up, CIC can be challenging for the caregiver and the patient due to rigid posture and greater body weight. When the child reaches school age, a continent catheterizable stoma (such as a Mitrofanoff) is more socially acceptable than CIC [12]. Previous studies have shown that in patients with difficulties in CIC from the urethra, deteriorated upper urinary tract function or recurrent UTI, that a vesicostomy is a possible solution in 70e90% of cases [5,6]. However, a continent catheterizable stoma needs more-complex surgical techniques and has a high complication rate. A study by Kavanagh et al. revealed that 39% needed further operations for stomal injection of bulking agents (e.g. dextranomer hyaluronic acid) for leakage, stomal revision for stenosis or prolapse, stones and repair of an augment rupture [13]. Moreover, if the patient requires further augmentation, adhesion related to a continent catheterizable stoma will likely result in a more difficult surgery and a potentially higher complication rate. Although a vesicostomy is effective in protecting the upper urinary tract, it is not continent and the rate of complications, such as prolapse, stenosis and dermatitis can be as high as 38% [5]. A modified technique of vesicostomy using a gastrostomy button has been described, which could be used as a continent urinary stoma for regular CIC. Early results in several studies have been encouraging [14,15]; however, the patient must change the button regularly, which has caused some discomfort and inconvenience. The device is not always available and is associated with higher medical costs. Other surgical modifications have also been attempted, based on the concept of a vesicostomy. A ‘mini-vesicostomy’ has been described for CIC in patients suffering from posterior urethral valve issues; it maintains bladder cycling and protects the upper urinary tract. However, minor leakage from the stoma occurs when the child is crying or playing [16]. An epithelialized neobladder-cutaneous fistula resulting in a
1230
W. Yang et al.
Figure 1
The appearance of the vesico-cutaneous fistula: dry with no dermatitis or mucosa prolapse.
continent and easily catheterizable abdominal stoma was reported as an incidental result [17]. Similar to the reported neobladder-cutaneous fistula, a VCF is a readily catheterizable stoma with good function. Therefore, a new concept was proposed: CIC via a VCF. Dryness, with no pads or bags to cover the fistula, was achieved in all patients (n Z 6) during the daytime with regular CIC every 2e3 h. There was no stenosis if regular CIC was performed; however, caregiver compliance is important for success. Experience has shown that the
parents’ compliance with CIC every 2 h was good. This could be because CIC via a VCF is easy, does not cause discomfort and each session can be completed in about 5 min. In addition, the patients were suffering from frequent UTIs with CIC every 3e4 h before the operation, so their parents were willing to perform CIC more frequently to decrease the chance of UTI. It is believed that the fistula is kept dry via multiple mechanisms. First, patients were chosen who had large bladder capacities, so there was sufficient volume to store
Figure 2 a VCF: only a small opening was made at the bladder dome and only the outer layer of bladder wall was fixed to the skin. b The Blocksom vesicostomy: the fascia is fixed to the bladder wall with a larger opening to prevent stenosis; the bladder is matured as a stoma.
A simple method for continent urinary diversion the urine between catheterizations; the bladder volume was estimated with a VCUG and renal echo. A bladder capacity exceeding 150 ml was required for patients who suffered from neurogenic bladder. Second, the fistula was made at the bladder dome, thus reducing the possibility of prolapse or leakage Fig. 2. In addition, the diameter of the fistula is small and the pressure of the abdominal wall muscles closes the tract to the bladder easily. However, these mechanisms might be not strong enough to maintain dryness in daytime activities. Therefore, regular daytime CIC is also necessary to keep the patient dry. At nighttime, when the patients sleep and relax, the anti-reflux mechanisms may be sufficient. Besides, for young patients, the parents will perform CIC for them before sleep and right after waking up. The interval between the two CICs was only 6e8 h. In older patients, they also restrict fluid intake at night after dinner. The fistula will close if regular CIC is no longer needed. The VCF is effective in protecting the upper urinary tract. All of the patients showed improved hydronephrosis, and 83.3% (n Z 5) showed decreased UTI frequency. No dermatitis was found in any patient. If there is a need for further surgery, such as augmentation or a Mitrofanoff, the presence of the fistula should not increase its difficulty. The present study was limited due to the small number of cases and that most of the patients were followed for less than one year. Although these initial results are encouraging, further prospective investigation and a longer duration follow-up are needed to reach more definite conclusions Fig 2.
Conclusions The vesico-cutaneous fistula is a simple, effective and tolerable method for CIC. It may be a substitute for, or a transition before, a Mitrofanoff conduit in some patients.
Conflict of interest/funding statement The authors have no conflicts of interest or funding relevant to this article.
References [1] Lawrenson R, Wyndaele JJ, Vlachonikolis I, Farmer C, Glickman S. Renal failure in patients with neurogenic lower urinary tract dysfunction. Neuroepidemiology 2001;20: 138e43.
1231 [2] Lindehall B, Abrahamsson K, Hja ¨lma ¨s K, Jodal U, Olsson I, Sille ´n U. Complications of clean intermittent catheterization in boys and young males with neurogenic bladder dysfunction. J Urol 2004;172:1686e8. [3] Lindehall B, Abrahamsson K, Jodal U, Olsson I, Sille ´n U. Complications of clean intermittent catheterization in young females with myelomeningocele: 10 to 19 years of followup. J Urol 2007;178:1053e5. [4] Lee MW, Greenfield SP. Intractable high-pressure bladder in female infants with spina bifida: clinical characteristics and use of vesicostomy. Urology 2005;65:568e71. [5] Prudente A, Reis LO, Franca Rde P, Miranda M, D’Ancona CA. Vesicostomy as a protector of upper urinary tract in long-term follow-up. Urol J 2009;6:96e100. [6] Morrisroe SN, O’Connor RC, Nanigian DK, Kurzrock EA, Stone AR. Vesicostomy revisited: the best treatment for the hostile bladder in myelodysplastic children? BJU Int 2005;96: 397e400. [7] Fernbach SK, Maizels M, Conway JJ. Ultrasound grading of hydronephrosis: introduction to the system used by the society for fetal urology. Pediatr Radiol 1993;23:478e80. [8] Bauer SB, Hallett M, Khoshbin S, Lebowitz RL, Winston KR, Gibson S, et al. Predictive value of urodynamic evaluation in newborns with myelodysplasia. J Am Med Assoc 1984;252: 650e2. [9] Dik P, Klijn AJ, van Gool JD, de Jong-de Vos van Steenwijk CC, de Jong TP. Early start to therapy preserves kidney function in spina bifida patients. Eur Urol 2006;49:908e13. [10] Lertsithichai P, Roongreungsilp U, Kochakarn W, RatanaOlarn K. Follow-up of long-term treatment with clean intermittent catheterization for neurogenic bladder in children. Asian J Surg 2004;27:134e6. [11] Edelstein RA, Bauer SB, Kelly MD, Darbey MM, Peters CA, Atala A, et al. The long-term urological response of neonates with myelodysplasia treated proactively with intermittent catheterization and anticholinergic therapy. J Urol 1995;154: 1500e4. [12] Mingin GC, Baskin LS. Surgical management of the neurogenic bladder and bowel. Int Braz J Urol 2003;29:53e61. [13] Milliken I, Munro NP, Subramaniam R. Cystostomy button for bladder drainage in children. J Urol 2007;178:2604e6. [14] Hitchcock RJ, Sadiq MJ. Button vesicostomy: a continent urinary stoma. J Pediatr Urol 2007;3:104e8. [15] Haider N, Subramaniam R. Endoscopic insertion of cystostomy button for bladder drainage in children. J Pediatr Urol 2008;4: 457e9. [16] Nanda M, Bawa M, Narasimhan KL. Mini-vesicostomy in the management of PUV after valve ablation. J Pediatr Urol 2012; 8:51e4. [17] Pinte ´r A, Vajda P, Kroowand L, Farkas A. Can epithelized neobladder-cutaneous fistula provide urinary continence? Int J Urol 2005;12:101e3.
Vesico-cutaneous fistula: A simple method for continent urinary diversion W. Yang a, P.-Y. Chang a,*, J.-Y. Lai a, C.-H. Cheng b, M.-H. Tseng b a Department of Pediatric Surgery, Chang Gung Children’s Hospital, Chang Gung University, College of Medicine, Linkou, Taiwan b Department of Pediatrics, Chang Gung Children’s Hospital, Chang Gung University, College of Medicine, Linkou, Taiwan
Received 3 March 2014; accepted 5 July 2014
Available online 22 July 2014
KEYWORDS Urinary diversion; Neurogenic bladder; Vesical fistula; Urethral stricture
Abstract Introduction: Patients with lower urinary tract anomalies or neurogenic disorders often suffer from voiding difficulties. Clean intermittent catheterization (CIC) is effective for bladder drainage; however, this is often painful. Transurethral catheterization is also impossible in patients with urethral stricture. A Mitrofanoff conduit may solve some of these problems, but a few disadvantages have been reported, including: difficult surgical techniques and frequent operative complications. A vesicostomy is easy to perform but persistent urine leak over the abdomen and diaper rash can be annoying. A better way to achieve continent urinary diversion is indicated. Method: Between December 01 1998 and December 31 2013, six patients underwent a vesicocutaneous fistula for CIC. The etiologies included urethral stricture (n Z 2) and neurogenic bladder (n Z 4). The fistula was created at the bladder dome with only the muscle layer of the bladder sutured to the skin. A Foley catheter was left in place for at least two weeks to prevent stoma stricture. After removing the Foley catheter, regular CIC from the fistula was performed every 2 h during the daytime with a Fr. 10e12 feeding tube, depending on the patient’s age. Further stenting during the night in the first six months was necessary to prevent early closure of the fistula. Patients were followed with periodic renal ultrasonography, blood tests and urinalysis in the outpatient department. Results: Follow-up ranged from 6 months to 16 years. All patients showed improvements in hydronephrosis. Decreased UTI frequency was seen in five patients. Renal function was normal in five patients, whilst the other suffered from chronic renal failure preoperatively. Only one patient had occasional mild urine leakage from the stoma at night, which was once in two weeks. No patient experienced painful or difficult catheterization and CIC becomes easy, even by young children.
* Corresponding author. Department of Pediatric Surgery, Chang Gung Children’s Hospital, Chang Gung University, College of Medicine, Linkou, No. 5, Fusing St, Gueishan Township, Taoyuan County 333, Taiwan. Tel.: þ886 3 3281200; fax: þ886 3 3285056. E-mail addresses: [email protected] (W.Yang), [email protected] (P.-Y.Chang), [email protected] (J.-Y.Lai), pednephcheng@ cgmh.org.tw (C.-H.Cheng), [email protected] (M.-H.Tseng). http://dx.doi.org/10.1016/j.jpurol.2014.07.002 1477-5131/ª 2014 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved.
1228
W. Yang et al. Conclusions: The vesico-cutaneous fistula is a simple, effective and tolerable method for CIC. It may be a substitute for or a transition to a Mitrofanoff conduit in some patients. ª 2014 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved.
Introduction The optimal treatment for children with neurogenic bladder and other urinary difficulties has not been established. Renal function deterioration may be related to VUR, hydronephrosis and recurrent UTI. End-stage renal disease can develop if a child is left untreated [1]. Clean intermittent catheterization (CIC) has been used widely in patients with neurogenic bladder (NB). Although a low complication rate and good tolerance of CIC have been reported after long-term follow-up, bleeding, urethral stricture or a false tract can still occur [2,3]. A subgroup of patients can suffer from recurrent UTI, even under regular CIC and prophylactic antibiotic treatment [4]. In patients with lower urinary tract anomalies, such as urethral stricture, performing CIC is impossible. A urinary diversion with a Mitrofanoff conduit has disadvantages, including: difficult surgical techniques and a high rate of operative complications. A traditional vesicostomy, although preserving upper urinary tract function, is often difficult for patients to accept due to the resulting urinary incontinence and risk of diaper dermatitis [5,6]. In the present study, a simple and nearly continent vesico-cutaneous fistula (VCF) was performed instead of a traditional vesicostomy in children who were under the CIC program. The present study reports on the experiences of six cases.
peritoneal reflection could be seen. After pushing away the peritoneum and the perivesical fat, the bladder was grasped at the dome and pulled through the wound. The bladder was opened and evacuated through a 0.5 cm longitudinal incision. Four to six stitches were used to fasten the outer layer of the bladder wall to the Scarpa’s fascia and the skin; the mucosa should not be sutured to the skin. A French 12 Foley catheter was left in place for two weeks in order to prevent early closure of the VCF. After removing the Foley catheter, regular CIC from the fistula was performed every 2 h during the daytime with a 10e12 Fr. feeding tube, depending on the patient’s age. The tube was inserted as a stent and left open for drainage at night for the first six months after the VCF had been created. Once the fistula had stabilized, no further stenting was required after the first six months.
Follow-up Patients were followed with periodic renal ultrasonography, blood tests and urinalysis in the outpatient department. VCUG, urodynamic studies and magnetic resonance urography (MRU) were performed as needed. Improvement was defined as improved hydronephrosis and hydroureter on renal echo, decreased frequency of febrile UTI or improving creatinine level. The severity of hydronephrosis was evaluated using the Society for Fetal Urology grading system [7].
Materials and methods After obtaining Institutional Review Board approval, a retrospective chart review of all patients who underwent a VCF between December 01 1998 and December 31 2013 was conducted.
Patients Sixty-six neurogenic bladder patients were treated at the present hospital between December 1998 and December 2013. Five of these patients underwent VCF procedures. Indications included: recurrent UTI, despite regular CIC from the urethra and prophylactic antibiotics (n Z 3), and worsening hydronephrosis and renal function (n Z 2). For patients who suffered from neurogenic bladder, a bladder capacity exceeding 150 ml should be proved during CIC. Another two urethral strictures were recruited due to difficulty with CIC from the urethra.
Operation method A 2.0 cm incision was made at the midpoint between the umbilicus and the pubic bone. The underlying fascia was incised and the rectus was retracted laterally until the
Results Four males and three females underwent the procedure; one was excluded as lost to follow-up. The characteristics and results of the six patients are described in Table 1. The mean age at surgery was 7.17 years (range 18 dayse22 years). Two patients (33.3%) had congenital urethral strictures. Four patients suffered from bladder dysfunction for the following reasons: meningomyelocele (n Z 2), cerebral palsy (n Z 1) and non-neurogenic neurogenic bladder (Hinman syndrome, n Z 1). Although CIC and prophylactic antibiotics were used for all patients with neurogenic bladders, febrile UTI that needed hospitalization for treatment (more than two episodes in six months) remained a common problem. All of the patients who underwent the operation showed improved hydronephrosis in the follow-up renal echo assessments. The frequency for febrile UTI declined and the nocturnal diuresis decreased gradually during the first six months of nocturnal stenting. One patient suffered from chronic renal failure and had a creatinine level around 7.0e8.0 mg/dl before the operation. The creatinine level improved to 5.0 mg/dl after three-months of regular CIC from the VCF. The rest of the
A simple method for continent urinary diversion Table 1
1229
Patients and outcomes.
Patient
1
2
3
4
5
6
Gender Diagnosis Age at surgery
M US1 18 days
M US 13 years
F NGB-12 4 months
F NGB3 22 years
F NGB 5 years
Preoperative hydronephrosis (left/right)a Postoperative hydronephrosis (left/right)a Complications
1/3
3/3
3/3
3/1
M NGB 1st: 2 years 8 months 2nd: 3 years 2 months 3/3
1/1
1/0
1/0
2/1
1e2/1e2
1/1
None
None
None
Fistula stenosis needed 2nd operation
None
16 years (fistula closure at 7 years old)
8 months
Urine leak at night, once in 2 weeks 11 years
7 months
9 months
3 months
Follow-up
2/2
Abbreviations: US (urethral stricture); NGB-1(neurogenic bladder of unknown cause); NGB (neurogenic bladder). The SFU system was graded 0-4 (0 Z no hydronephrosis; 1 Z visualized only renal pelvis; 2 Z plus a few caliceal dilatation; 3 Z all calyceal dilatation; 4 Z plus parenchymal thinning). a The severity of hydronephrosis was evaluated according to the Society for Fetal Urology grading system.
patients had creatinine levels below 1 mg/dl before the operation and these remained stable afterward. The mean follow-up was 4.8 years (range 3 months-16 years). Two of them had been followed for more than 10 years. No UTI episode was seen in the female who had a non-neurogenic neurogenic bladder over the past three years. The male who suffered from urethra duplication and stricture had his fistula closed at seven-years old, after completed treatment for a urethral stricture. He is now 16-years old and the one or two episodes of UTI that he has had in the last year were managed with oral antibiotics alone. No prophylactic antibiotic was given to either of these patients. All of the patients could keep dry during the day and no bag or pad was needed (Fig. 1). One patient (16.7%) underwent surgical revision of the VCF because the patient’s mother was unwilling to perform CIC via the VCF after the Foley catheter was removed. After explaining the process clearly, regular CIC was performed smoothly after the second operation; no complications have occurred since. One patient (16.7%) suffered from occasional urine leakage at night (once in two weeks). The leakage was easily managed by covering the fistula with a pad at night. Two complications occurred in two of the six patients (33.3%), but both were minor and could be managed readily. No patient suffered from dermatitis or prolapse.
Discussion Deterioration of upper urinary tract function in children who have abnormal lower urinary tract function can be as high as 70% [8]. The renal failure rate is 40e50% in these patients and rises steeply with age [1]. The patients often benefit from regular CIC and this should start at an early age [9,10]. However, approximately 10% of these patients suffer from decreased upper urinary tract function [11]. Furthermore, the CIC technique is relatively difficult for young or mentally
disabled children. As children grows up, CIC can be challenging for the caregiver and the patient due to rigid posture and greater body weight. When the child reaches school age, a continent catheterizable stoma (such as a Mitrofanoff) is more socially acceptable than CIC [12]. Previous studies have shown that in patients with difficulties in CIC from the urethra, deteriorated upper urinary tract function or recurrent UTI, that a vesicostomy is a possible solution in 70e90% of cases [5,6]. However, a continent catheterizable stoma needs more-complex surgical techniques and has a high complication rate. A study by Kavanagh et al. revealed that 39% needed further operations for stomal injection of bulking agents (e.g. dextranomer hyaluronic acid) for leakage, stomal revision for stenosis or prolapse, stones and repair of an augment rupture [13]. Moreover, if the patient requires further augmentation, adhesion related to a continent catheterizable stoma will likely result in a more difficult surgery and a potentially higher complication rate. Although a vesicostomy is effective in protecting the upper urinary tract, it is not continent and the rate of complications, such as prolapse, stenosis and dermatitis can be as high as 38% [5]. A modified technique of vesicostomy using a gastrostomy button has been described, which could be used as a continent urinary stoma for regular CIC. Early results in several studies have been encouraging [14,15]; however, the patient must change the button regularly, which has caused some discomfort and inconvenience. The device is not always available and is associated with higher medical costs. Other surgical modifications have also been attempted, based on the concept of a vesicostomy. A ‘mini-vesicostomy’ has been described for CIC in patients suffering from posterior urethral valve issues; it maintains bladder cycling and protects the upper urinary tract. However, minor leakage from the stoma occurs when the child is crying or playing [16]. An epithelialized neobladder-cutaneous fistula resulting in a
1230
W. Yang et al.
Figure 1
The appearance of the vesico-cutaneous fistula: dry with no dermatitis or mucosa prolapse.
continent and easily catheterizable abdominal stoma was reported as an incidental result [17]. Similar to the reported neobladder-cutaneous fistula, a VCF is a readily catheterizable stoma with good function. Therefore, a new concept was proposed: CIC via a VCF. Dryness, with no pads or bags to cover the fistula, was achieved in all patients (n Z 6) during the daytime with regular CIC every 2e3 h. There was no stenosis if regular CIC was performed; however, caregiver compliance is important for success. Experience has shown that the
parents’ compliance with CIC every 2 h was good. This could be because CIC via a VCF is easy, does not cause discomfort and each session can be completed in about 5 min. In addition, the patients were suffering from frequent UTIs with CIC every 3e4 h before the operation, so their parents were willing to perform CIC more frequently to decrease the chance of UTI. It is believed that the fistula is kept dry via multiple mechanisms. First, patients were chosen who had large bladder capacities, so there was sufficient volume to store
Figure 2 a VCF: only a small opening was made at the bladder dome and only the outer layer of bladder wall was fixed to the skin. b The Blocksom vesicostomy: the fascia is fixed to the bladder wall with a larger opening to prevent stenosis; the bladder is matured as a stoma.
A simple method for continent urinary diversion the urine between catheterizations; the bladder volume was estimated with a VCUG and renal echo. A bladder capacity exceeding 150 ml was required for patients who suffered from neurogenic bladder. Second, the fistula was made at the bladder dome, thus reducing the possibility of prolapse or leakage Fig. 2. In addition, the diameter of the fistula is small and the pressure of the abdominal wall muscles closes the tract to the bladder easily. However, these mechanisms might be not strong enough to maintain dryness in daytime activities. Therefore, regular daytime CIC is also necessary to keep the patient dry. At nighttime, when the patients sleep and relax, the anti-reflux mechanisms may be sufficient. Besides, for young patients, the parents will perform CIC for them before sleep and right after waking up. The interval between the two CICs was only 6e8 h. In older patients, they also restrict fluid intake at night after dinner. The fistula will close if regular CIC is no longer needed. The VCF is effective in protecting the upper urinary tract. All of the patients showed improved hydronephrosis, and 83.3% (n Z 5) showed decreased UTI frequency. No dermatitis was found in any patient. If there is a need for further surgery, such as augmentation or a Mitrofanoff, the presence of the fistula should not increase its difficulty. The present study was limited due to the small number of cases and that most of the patients were followed for less than one year. Although these initial results are encouraging, further prospective investigation and a longer duration follow-up are needed to reach more definite conclusions Fig 2.
Conclusions The vesico-cutaneous fistula is a simple, effective and tolerable method for CIC. It may be a substitute for, or a transition before, a Mitrofanoff conduit in some patients.
Conflict of interest/funding statement The authors have no conflicts of interest or funding relevant to this article.
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