Vesicocutaneous fistula: continent vesicostomy, an easier route for comfortable clean intermittent catheterization

Journal of Pediatric Urology (2020) 16, 354.e1e354.e8

Department of Pediatric Surgery, Chang Gung Children’s Hospital, Chang Gung University, School of Medicine, No.5, Fuxing Street, Guishan District, Taoyuan City, 33305, Taiwan * Corresponding author. Department of Pediatric Surgery, Chang Gung General Hospital, Chang Gung University, College of Medicine, No.5, Fusing Street, Gueishan Township, Taoyuan County 333, Taoyuan, Taiwan. Tel.: þ886 3 3281200x8227; fax: þ886 3 3285056. [email protected] (P. -Y. Chang) Keywords Neurogenic bladder; Clean intermittent catheterization; Vesicostomy; Bladder drainage; Urinary diversion; Mitrofanoff Received 27 June 2019 Accepted 4 February 2020 Available online 11 February 2020

Vesicocutaneous fistula: continent vesicostomy, an easier route for comfortable clean intermittent catheterization Cynthia Sze-Ya Ting, Pei-Yeh Chang * Summary Introduction Clean intermittent catheterization (CIC) through the urethra is the treatment of choice for patients with neurogenic bladder (NGB) or other etiologies that lead to incomplete bladder emptying. However, urethral catheterization can be problematic. Vesicocutaneous fistula (VCF) is a continent catheterizable channel with a low rate of complications. The aim of the study was to evaluate the safety and effectiveness of VCF as a route for CIC. Material and methods The authors retrospectively reviewed patients who underwent creation of the VCF for bladder drainage from November 2001 to December 2017. Demographics, indication for VCF, pre-operative and postoperative laboratory/radiologic studies, incidence of febrile urinary tract infection (UTI), and adherence to CIC through VCF were examined. Results Vesicocutaneous fistula was created in a total of 20 patients (nine males and 11 females; median age, 13.2 years [range: 3.8 monthse22.8 years]). The median follow-up time was 30.5 months (range: 5.9 monthse16.9 years). The underlying etiologies that resulted in NGB included spina bifida (n Z 10), cerebral palsy (n Z 2), caudal regression syndrome (n Z 2), and others (n Z 6). Before creation of the VCF, 13 patients (65%) had either grade 3 unilateral or bilateral hydronephrosis as per the Society for

Fetal Urology grading system. Thirteen patients (65%) had experienced at least one febrile UTI the year before the creation of the VCF. At the last follow-up, renal function was improved or stabilized in 14 patients (70%). Fifteen patients (75%) had experienced no febrile UTI in the last 1 year. Upper urinary tract dilatation resolved or improved in 10 patients (77%). The VCF continence rate was 88%. In this study, bladder augmentation or the Mitrofanoff procedure was not performed. During maturation, nine patients (45%) had granuloma; five of those cases subsided within 2 years without any intervention. Five patients had VCF stricture, and only one required revision surgery (5%). Discussion The VCF continence rate was comparable with that of the Mitrofanoff procedure. Adherence to CIC through VCF lowered the rate of UTI and preserved the upper urinary tract. Bladder emptying by CIC through VCF provided the same benefits as those of the Mitrofanoff procedure: extra privacy, social independence, and reduction of parental burden. Although a long maturation stage of 6 months was required, the rate of major complications was low. Most complications were conservatively manageable and seldom required revision surgery. Conclusions Vesicocutaneous fistula is a continent catheterizable conduit, an alternative option for bladder management in patients with NGB who cannot undergo urethral CIC smoothly.

https://doi.org/10.1016/j.jpurol.2020.02.004 1477-5131/ª 2020 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved.

Vesicocutaneous fistula Summary Table

354.e2

Outcomes of CIC through VCF.

Variables CKD stage Stage 1 Stage 2 Stage 3 Stage 4 Stage 5 n/a SFU grade Normal Grade 1e2 Grade 3e4 n/a UTI episode(s) per year 0 1e2 >3 n/a

Before creation of the VCF, number of patients (%)

After creation of the VCF (last f/u), number of patients (%)

7 3 8 0 2 0

(35) (13) (40) (0) (10) (0)

7 1 5 2 0 5

(35) (5) (25) (10) (0) (25)a

0 (0) 7 (35) 13 (65) 0 (0)

5 6 8 1

(25) (30) (40) (5)

7 8 5 0

14 (70) 3 (15) 2 (10) 1 (5)b

(35) (40) (25) (0)

CIC, clean intermittent catheterization; CKD, chronic kidney disease; f/u, follow-up; n/a, not available; SFU, Society for Fetal Urology; UTI, urinary tract infection; VCF, vesicocutaneous fistula. a We did not routinely check the level of creatinine if the CKD stage before creation of the VCF was <1. b One patient was lost to follow-up for >1 year; therefore, the data of UTI episode(s) in the last 1 year were not available.

Introduction Unlike vesicostomy, vesicocutaneous fistula (VCF) is a continent fistula tract connecting the bladder and abdominal wall. Vesicocutaneous fistula is easy to perform and could serve as a continent route for clean intermittent catheterization (CIC), which is a cornerstone of treatment for patients with neurogenic bladder (NGB). Care of children with NGB has significantly advanced over the last few decades. A comprehensive treatment strategy has been applied to maintain a low-pressure bladder and to prevent urinary tract infection (UTI) and renal scaring [1]. As per the guidelines established by the Centers for Disease Control and Prevention (USA) [2], CIC, anticholinergic medications, and botulinum injections are the first-line treatment options for children with NGB to reduce the risk of urinary tract deterioration (UTD). Bladder augmentation can be tried after failure of anticholinergic medications and/or botulinum injections to improve bladder compliance. Vesicostomy can be an alternative to CIC when continence is not required or CIC will cause problems as in infants and young children. Continent stoma such as the Mitrofanoff appendicovesicostomy can replace CIC when CIC is difficult or associated with complications. CIC through the urethra is characterized by significant disadvantages. Performing CIC multiple times per day could be a parental burden. Even in experienced hands, CIC can result in pain, trauma, and urethral stricture. On the other hand, Mitrofanoff appendicovesicostomy is a complex procedure associated with a high risk of morbidity. The authors previously published their preliminary results [3] of applying VCF as a continent bladder drainage

access in patients with incomplete bladder emptying. The aim of this study was to examine the safety and effectiveness of VCF as a route for CIC.

Methods and patients After obtaining approval from the institutional review board, the authors retrospectively reviewed all patients who underwent creation of a VCF at their hospital between November 01, 2001, and December 31, 2017. Indications for VCF included patients who were unable to establish CIC through the urethra owing to pain, stricture, or mechanical obstruction. Patients in whom VCF was used as a temporary bladder diversion or patients with incomplete pre-operative data and inadequate length of follow-up were excluded. Age, gender, indications, diagnoses, age at operation, complications, creatinine level, imaging findings on a renal echogram, and urodynamic studies before creation of the VCF and at last follow-up were extracted from the medical records of the patients.

Operative method The operation procedure was previously published [3] and demonstrated in Video 1. The patient was placed in the supine position and subjected to general anesthesia. The area from the umbilicus to the genital area was sterilized. A Foley tube was inserted through the urethra, and normal saline (50e100 ml) was injected through the Foley catheter to distend the bladder. A 2.0-cm transverse skin incision equidistant between the umbilicus and the pubic

354.e3 bone was typically made. However, the length of the incision may vary as per the size of the bladder. Occasionally, NGB could be trabeculated and shrunken; in those cases, the incision should be lower than designated. When reaching the rectus sheath, a 2.0-cm transverse incision was made, and four stay stitches were placed at four angles of the rectus sheath. The bladder was located, and a 0.5-cm incision was performed at its dome. A Foley was inserted through the bladder incision. The following is the supplementary data related to this article: Supplementary video related to this article can be found at https://doi.org/10.1016/j.jpurol.2020.02.004 The muscle layer was sutured with the stay stitches of the rectus sheath. The main difference between vesicostomy and VCF is the sparing of the mucosal layer of the bladder with VCF. There was no need to separate the mucosa layer from the muscle, but it should be ensured to avoid including the mucosa when placing the sutures. The skin layers were closed with two 5-0 Dexon stitches subcuticularly, each one on the left and right side of the skin incision. Meticulous skin closure was avoided, which may have resulted in VCF stenosis.

Vesicocutaneous fistula maturation The maturation program has been standardized. The nurse practitioner at the pediatric urology department thoroughly instructed the families before surgery. After creation of the VCF, the families were provided a manual (Supplement 1) containing a timetable and were asked to keep a catheter diary. The maturation program is a stepwise shifting from the indwelling catheter to intermittent catheterization. Patients were treated with the indwelling catheter for the first 3 weeks after creation of the VCF. Intermittent catheterization was initiated at week 4. The catheter was removed for 15 min (tube-free time) every 2 h. The tubefree time was increased by 5 min on a weekly basis (e.g., tube-free time for 20 min every 2 h at week 5). Consequently, at month 4, the tube-free time was 1.5 h every 2 h. At months 5 and 6, CIC was performed every 2 h, and there was no need for tube indwelling after CIC. The duration of maturation was 6 months. During maturation, the indwelling catheter was maintained through the night. After this period, the interval of CIC was evaluated based on the residual urine volume and bladder capacity. Typically, CIC every 2 h is required for the majority of the authors’ patients (Video 2). Once matured, the VCF will not close spontaneously, even without an indwelling catheter. The following is the supplementary data related to this article: Supplementary video related to this article can be found at https://doi.org/10.1016/j.jpurol.2020.02.004 Patients who experienced frequent UTIs before creation of the VCF may continue to suffer from such infections during maturation. In these cases, the patients received treatment with antibiotics until resolution of the infection. Prophylactic treatment with antibiotics is administered to patients at high risk of UTD and UTI.

C.S.-Y. Ting, P.-Y. Chang

Shift from vesicostomy to VCF The main difference between a vesicostomy and VCF is that the latter has no mucosal layer and requires secondary reepithelialization. Vesicostomy is incontinent, whereas VCF is continent. Two patients in this study shifted from vesicostomy to VCF at the ages of 4.2 and 15.3 years. First, the vesicostomy was taken down by dissecting the plane between the bladder and the abdominal wall. The bladder mucosa was separated from the muscle layer by pushing it down. Four stitches were placed at 3, 6, 9, and 12 o’clock around the opening of the former vesicostomy. The stitches should only include the muscle layer of the bladder. Fixing the bladder muscle with the rectus sheath with 4e6 stitches is sufficient. A 12-Fr Foley catheter was inserted through the VCF. The maturation program was the same as that for a primary VCF.

Follow-up Patients were followed up each month during VCF maturation. Six months after creation of the VCF, it was often suggested to perform CIC every 2 h in patients with residual urine volume >100 ml. Patients were followed up every 3e6 months for the first year and subsequently on an annual basis to determine whether their condition was stable. Ultrasound scans were conducted once or twice per year. Blood tests and urine analyses were performed in patients who exhibited symptoms of UTI. Other urology imaging scans were arranged at the clinician’s discretion. Oral prophylactic treatment with antibiotics was administered in patients with Society for Fetal Urology (SFU) grade 3e4 urinary tract dilatation or those who recovered from frequent febrile UTIs until resolution. Improvement was defined as improved hydronephrosis and hydroureter on renal ultrasound, decreased frequency of febrile UTI, or improved renal function as per the estimated glomerular filtration rate.

Evaluation of continence The level of continence was evaluated using a questionnaire at least 12 months after creation of the VCF. The authors only assessed continence in patients aged >3 years. The patients and their caregivers were asked whether they (1) experienced leakage from the VCF during voiding, coughing, laughing, or anytime of the day; (2) experienced leakage in between voiding and catheterization; and (3) required a pad or diaper during anytime of the day owing to leakage from the VCF. Negative answers to these questions indicated that the VCF was continent. In cases of suspected incontinence, the level of leakage was further assessed and recorded.

Results A total of 332 patients with dysfunctional bladder were treated at the study institution during the study period. Twenty-eight VCFs were created during the study period. Seven patients with temporary use of VCF were excluded,

Vesicocutaneous fistula

354.e4

including 3 grade V bilateral vesicoureteral reflux, 2 urethral strictures, 1 posterior urethral valve, and 1 urethral duplication. One patient with NGB was lost to follow-up three months after creation of the VCF and was also excluded. After exclusion, 20 patients underwent creation of the VCF at the median age of 13.2 years (range: 3.8 monthse22.8 years), with a median follow-up time of 30.5 months (range: 5.9 monthse16.9 years). None of the patients had undergone bladder augmentation before creation of the VCF. As shown in Table 1, the underlying etiologies that resulted in dysfunctional bladders included spina bifida (n Z 10), cerebral palsy (n Z 2), caudal regression syndrome (n Z 2), posterior urethral valve (n Z 1), anorectal malformation (n Z 1), non-neurogenic NGB (n Z 1), and others (n Z 3). All patients were diagnosed with dysfunctional bladder and treated with urethral CIC and medication before creation of the VCF, except two patients. These two patients had undergone prior vesicostomy at the ages of 11 months and 3.7 years. Subsequently, on parental request, they were shifted to VCF at the age of 15.3 and 4.2 years,

Table 1

Patient demographics before creation of the VCF.

Patient subgroups Sex Male Female Diagnosis Spina bifida Caudal regression Cerebral palsy ARM PUV Myelitis NNNB Spinal cord injury Sacrococcygeal teratoma Operative age <5 years 5e10 years >10 years CKD stage Stage 1 Stage 2 Stage 3 Stage 4 Stage 5 SFU grade Grade 1e2 Grade 3e4 UTI episode per year 0 1e2 >3

Number of patients (%) 9 (45) 11 (55) 10 (50) 2 (10) 2 (10) 1 (5) 1 (5) 1 (5) 1 (5) 1 (5) 1 (5) 4 (20) 4 (20) 12 (60) 7 3 8 0 2

(35) (13) (40) (0) (10)

7 (35) 13 (65) 7 (35) 8 (40) 5 (25)

VCF, vesicocutaneous fistula; ARM, anorectal malformation; CKD, chronic kidney disease; NNNB, non-neurogenic neurogenic bladder; PUV, posterior urethral valve; SFU, Society for Fetal Urology; UTI, urinary tract infection.

respectively. Anticholinergics were prescribed for only one patient with evidence of detrusor overactivity for 6 months before creation of the VCF. Before creation of the VCF, the median level of serum creatinine was 0.92 mg/dl (range: 0.24e4.2). As described in Table 2, thirteen patients had either SFU grade 3 unilateral or bilateral hydronephrosis. Thirteen patients (65%) had at least one febrile UTI within 1 year before creation of the VCF. In these patients, the VCF was used as a continent conduit for intermittent catheterization for bladder emptying. The indications for VCF were both UTD and recurrent febrile UTIs (eight patients), UTIs (five patients), UTD (three patients), both UTD and parental request (two patients), and parental request (two patients). Only 10 patients were able to cooperate and complete the urodynamic study before creation of the VCF. The urodynamic data were summarized in Table 3. All participants had either incomplete relaxing or non-relaxing sphincters. Five patients had normal detrusor activity, two patients had detrusor hyperactivity, and three patients had detrusor underactivity. Eight patients had either poor or fair detrusor compliance. Only two patients presented with good compliance on the cystometry. The median residual urine volume was 185 ml (range: 70e400 ml). There were no acute postoperative complications. During the maturation period, four patients had difficulty inserting the catheter. A smaller catheter was applied for 2 weeks. Afterward, the fistula tract was gradually dilated to the optimal size. Other complications were granuloma and VCF stricture. Nine patients (45%) had granuloma; in five of those, the granuloma subsided in 2 years without any intervention. Three of the four patients with nonsubsided granuloma were followed up for <2 years. As summarized in Table 4, five patients had VCF stricture (25%) during or after the maturation program. One patient required revision surgery (5%) 2.2 years after VCF creation. The other strictures were managed with conservative treatment. Four patients missed the assessment for continence. Of the remaining 16 patients, two patients with NGB secondary to spina bifida reported minor leakage (<10 ml) once to twice per week after laughing or exercise. The continence rate for the VCF was 88% (14 of 16 patients). One year after creation of the VCF, seven patients who were UTI free before creation of the VCF did not experience febrile UTI thereafter. Five patients had a more number of UTIs within 1 year after VCF creation than the year before VCF creation. At follow-up, 15 patients (75%) had experienced no febrile UTIs in the last year. Among the 13 patients who had at least one UTI annually before VCF creation, the frequency of UTI decreased to zero in seven patients (54%). Three patients had one episode of febrile UTI requiring admission, and the other two patients had four and six episodes of febrile UTIs in the last year. More aggressive treatments may be indicated in these patients. At the latest follow-up, five patients continued to receive prophylactic treatment with antibiotics. At the latest follow-up, the median level of serum creatinine was 0.9 mg/dl (range: 0.22e3.72 mg/dl). The renal function was improved or stabilized in 14 patients (70%) compared with that recorded before VCF creation.

354.e5

Table 2

Outcome parameters before and after VCF creation.

No.Etiology of NGB GenderAge Operative Length of Creatinine (mg/dL), Creatinine CKD stage, CKD HN (R/L), HN (R/ UTIa, 1 year UTI in Bladder (years)age f/u before creation of (mg/dL), before creation stage, before creation L), last before creation the last management (years) (months) the VCF last f/u of the VCF last f/u of the VCF f/u of the VCF 1 year at last f/u 1 2 3 4 5 6 7 8 9 10

Spina Spina Spina Spina Spina Spina Spina Spina Spina Spina

bifida bifida bifida bifida bifida bifida bifida bifida bifida bifida

11 Caudal regression 12 Caudal regression 13 Cerebral palsy 14 Cerebral palsy 15 ARM 16 PUV 17 Myelitis 18 NNNB 19 Spinal cord injury

M M M M F F F M M F

5.6 7.0 9.7 10.8 11.2 11.5 14.9 18.7 21.3 23.5

0.3 5.8 8.8 4.2 5.1 5.6 12.0 15.3 20.1 20.7

64.8 15.0 11.2 79.5 73.6 72.3 27.0 38.9 10.2 37.7

0.34 0.24 1.03 0.52 2.14 0.38 0.49 4.20 1.58 0.57

0.28 x 0.72 0.46 1.65 0.34 0.44 3.72 1.55 0.44

3B 1 3B 3A 5 2 1 5 3A 1

1 x 3A 1 4 1 1 4 3A 1

N/1 N/4 4/4 4/4 3/3 4/4 1/N 4/4 3/3 x

N/N N/3 3/3 N/1 N/1 N/N 1/N 3-4/3-4 x N/N

1 0 3 2 5 5 1 1 0 0

1 0 4 0 0 0 0 0 0 0

F

14.6

13.9

8.4

0.73

0.41

1

x

4/3

4/3

3

0

CIC q2h self CIC q2h CIC q1e2h self CIC q2h CIC q2h self CIC CIC self CIC q2h self CIC q2h self CIC q3h, night IC CIC q1-2h

M

19.2

14.4

57.6

1.36

1.49

2

2

1/4

1/4

3

6

self CIC q2h

M

16.7

15.3

18.0

1.0

x

3A

x

4/4

3/3

1

x

F

28.6

22.4

44.3

1.21

1.03

3A

3A

1/4

1

M M F F

16.7 20.1 18.6 18.2

15.3 15.6 14.0 0.3

5.9 57.1 55.8 202.4

0.93 1.77 0.26 x

x 2.59 x 0.59

1 3A 1 1

x 3B x 1

N/N 4/4 x x

N/ 1 invisible N/N 0 4/4 0 1/1 0 1/N 0

0 0 0 0

M

3.9

1.9

23.3

0.25

0.22

2

1

1/N

1/N

1

1

17.7

12.1

66.6

0.92

1.03

3A

3A

4/4

3/3

2

0

Only night IC Poor adherent to CIC

ARM, anorectal malformation; CIC, clean intermittent catheterization; CKD, chronic kidney disease; f/u, follow-up; HN, hydronephrosis (as per the SFU grading system); IC, indwelling catheter; NGB, neurogenic bladder; NNNB, non-neurogenic neurogenic bladder; PUV, posterior urethral valve; SFU, Society for Fetal Urology; US, ultrasound; UTI, urinary tract infection; VCF, vesicocutaneous fistula; L, left; R, right; M, male; F, female; N, normal; x, data not available. a The frequency of UTIs was defined as febrile UTIs and is presented here as the number of episodes in (1) the year before creation of the VCF, (2) the year after creation of the VCF, and (3) the latest year.

C.S.-Y. Ting, P.-Y. Chang

20 Sacrococcygeal F teratoma

CIC q1h

M F F M M F M F M F 2 5 6 8 9 10 12 14 16 20

354.e6 VCF, vesicocutaneous fistula; MUCP, maximum, urethral closure pressure, RU, residual urine; BC, bladder capacity; VCF, vesicocutaneous fistula; x, data not available; M, male; F, female.

0.77 x x x 0.72 0.31 x x x x 142 x x x 199 255 x x x x 194 163 287 524 285 167 552 277 370 5.8 5.1 5.6 15.3 20.1 20.7 14.4 22.4 15.6 12.1

Hyperactivity Underactivity Normoactivity Hyperactivity Normoactivity Normoactivity Underactivity Normoactivity Underactivity Normoactivity

Fair Poor Poor Poor Good Fair Good Fair Fair Poor

Incomplete relaxing Incomplete relaxing Incomplete relaxing Incomplete relaxing Non-relaxing Incomplete relaxing Incomplete relaxing Incomplete relaxing Incomplete relaxing Incomplete relaxing

x 12 x x x 60 92 71 66 28

78 190 150 200 400 70 160 190 200 180

x 0.98 0.92 0.70 0.76 0.25 0.96 0.34 0.72 0.49

x 109 74 x x 144 80 x x x

RU/BC BC RU RU/BC BC RU

7.0 11.2 11.5 18.7 21.3 23.5 19.2 28.6 20.1 17.7

Detrusor activity Operative age (years) Age (years) Gender No.

Table 3

Urodynamic data before and after creation of the VCF.

Compliance

Sphincter

MUCP (cmH2O)

Before creation of the VCF

After creation of the VCF

Vesicocutaneous fistula Table 4

Complications of VCF.

Type of complications

Number of patients (%)

Granuloma Stricture Revision due to stricture Granuloma lasts > 2 years

9 5 1 1

(45) (25) (5) (5)

VCF, vesicocutaneous fistula.

As described in Table 2, among the 13 patients with SFU grade 3 hydronephrosis before VCF creation, upper tract dilatation had resolved or improved in 10 patients (77%) at follow-up. At the last follow-up, eight patients continued to have SFU grade 3 hydronephrosis. All patients in this study continued to undergo CIC through the VCF. None of the patients in this study were subjected to VCF takedown. Moreover, bladder augmentation and the Mitrofanoff procedure were not performed.

Discussion Although CIC aims to prevent UTIs and preserve renal function in patients with impaired bladder emptying, UTI is also the most common complication in patients who undergo CIC [4]. Bacterial colonization is a constant problem in patients undergoing CIC and poses a risk factor for UTI [5]. Recent studies have revealed the presence of UTIs caused by antimicrobial-resistant bacteria in pediatric patients undergoing CIC [6]. The reported rate of UTI in patients who undergo CIC for bladder emptying ranges widely owing to different diagnostic criteria, various methods of evaluation, and different CIC techniques. The overall rate of UTI among patients undergoing CIC is 25% [7]. Intermittent catheterization has been associated with repeated urethral trauma and strictures [8], presented as persistent urethral bleeding [8] and false passage [9]. Repeated trauma has been found to result in urethral stricture [10,11], and, in some cases, the change of the approach to bladder management is made owing to repeated trauma [12]. These complications affect quality of life [13], lead to perceived barriers, and result in low adherence to CIC [14]. For patients with difficulties in performing CIC through the urethra, or for wheelchair-bound patients, a continent catheterizable conduit (CCC), either the Mitrofanoff or Monti procedure, acts as another route for CIC without patient transfer or exposure of the genital area. Continent catheterizable conduit provides extra privacy and social independence and decreases parental burden [1]. As per the literature, both methods achieved continence rates of >90% [15e17]. However, CCC also has wide ranges of rates of complications [15], such as stomal stenosis (7e24%), stone formation (8e98%), and stricture (2e16%). The reported rate of surgical revision ranged from 7% to 39%. Leslie et al. [18] conducted a retrospective study of 169 patients who underwent CCC in 2011. At least one subsequent surgical revision was performed in 67 patients (40%). Based on a 15-year follow-up period, they concluded that no specific factor predicted the likelihood of complications.

354.e7 The complexity of this procedure potentially contributes to the high rate of complications. Despite the advancement of technology, limited improvement to the complication rate associated with CCC has been achieved [17,19]. Stomal stenosis is a serious complication of CCC because CIC is suspended and a revision surgery is often required. In the literature, the incidence of stomal stenosis and stricture ranged from 2% to 24% [15]. In 2017, Faure et al. [20] reported a 50% incidence of stomal stenosis, with 33 of 54 (61%) patients requiring revision surgery. Multiple factors contribute to stomal stenosis and stricture, most likely owing to ischemia and scarring [16]. In VCF, a flap is not necessary; therefore, there is no risk of tissue ischemia. Most VCF strictures were minor and could be managed with conservative treatment. In this series, although the stricture rate was 25%, only one of 20 patients (5%) had VCF stricture requiring revision surgery. Peristomal granuloma is the most common complication of VCF, yet this complication is minor. Bladder emptying by CIC through a VCF can be performed through a granuloma. Granuloma was often found to subside within 2 years, and there was no need for intervention as long as CIC could still be performed. Possible mechanism of continence in VCF was increased resistance by (1) the re-epithelized surface of the fistula tract and (2) tract length and valve-like function provided by (3) the rectus muscle along the tract. The investigator attempted two different procedures to create a small fistula tract connecting the bladder and abdominal wall: (1) stitches fixing the bladder mucosa to the rectus sheath and (2) stitches fixing only the muscle layer of the bladder to the rectus sheath. After the same maturation program, the former method yielded similar outcomes to those of an incontinent vesicostomy. Although a formal controlled trial has not been conducted, the authors avoided sutures with the mucosa layer and hypothesized that a secondary reepithelialization was partially responsible for the continent mechanism. Peristomal granuloma developed in nearly half (9/20) of the patients in this study. It was originally thought that granuloma formation played a role as a valve that provided continence. However, most cases of granuloma subsided in 2 years without any intervention, and these tracts remained continent. The nature of the re-epithelialized surface of the tract, rather than the granulation tissue, contributes to continence. The authors suspect that the uneven surface of secondary healing provides continence by increasing the resistance. The length of the tract may also contribute to continence by increasing resistance. The authors measured the tract length of the VCF in nine patients. The average tract length, measured at a median of 57.1 months (range: 11.2e79.5) after creation of the VCF, was 36 mm (range: 18e51 mm). Seven of nine patients (78%) had a VCF tract length >30 mm. Notably, tract length was also correlated with age and body weight. Further investigation is warranted to clarify whether a shorter tract is associated with lack of continence. The rectus muscle was along the fistula tract may act as a valve. However, more studies need to be conducted to identify factors that contribute to continence in VCF.

C.S.-Y. Ting, P.-Y. Chang In partial-thickness wounds, epithelialization occurs after approximately 8e10 days [21]. In full-thickness wounds, such as VCF, the secondary healing process may last several months. At first, the authors removed the indwelling catheter 2 months after operation and initiated CIC. However, VCF stricture and obliteration occurred. The authors subsequently increased the duration of the indwelling catheter; however, patients developed UTIs after 2 months. Therefore, the catheter was removed for several minutes every 2 h from week 4 after creation of the VCF and gradually shifted to intermittent catheterization within 6 months. In the preliminary stage, the VCF also serves as a temporary bladder diversion before formal urology reconstruction, such as in cases of urethral duplication and posterior urethral valve. However, because the maturation period of VCF is long and labor-intensive, the authors subsequently determined that the temporary use of a VCF in these cases was not justified, and these patients differed from those who required a conduit for long-term CIC. From then on, the authors do not use VCF for temporary bladder diversion. Nocturnal bladder emptying alone may improve renal function, increase bladder compliance, and decrease the incidence of UTI [22]. The indwelling catheter was maintained in place at night during maturation. After complete maturation, the patients could choose whether they preferred an indwelling catheter through the night. Some patients only have the indwelling catheter at night without CIC during the daytime. This is a personal choice of the patient, as long as the bladder has low pressure without UTD. There are several limitations that warrant mention aside from the retrospective design of this study. Owing to the limited size of the study population, the authors were unable to perform a randomized control trial comparing VCF with urethral CIC or VCF with the Mitrofanoff procedure. In addition, further assessment of the urodynamics after VCF creation is needed. Finally, the quality of life of patients was not formally assessed in this study.

Conclusion The authors found that VCF, which is easy to perform without the need of a flap, can be used as a CCC. Most complications were conservatively manageable and seldom required revision surgery. Use of a VCF for alternative bladder management can be considered in patients in whom urethral CIC cannot be performed.

Author statements Acknowledgments The authors would like to thank Uni-edit (www.uni-edit. net) for editing and proofreading this manuscript.

Ethical approval None sought.

Vesicocutaneous fistula

354.e8

Funding This research did not receive any specific grant from funding agencies in the public, commercial, or not-forprofit sectors.

[11]

[12]

Competing interests None declared.

[13]

Submission declaration The abstract of this manuscript has been orally presented at the 21st AsiaePacific Association of Pediatric Urologists (APAPU 2019) in Perth and at the 6th World Congress of the World Federation of Associations of Pediatric Surgeons (WOFAPS 2019) in Qatar.

[14]

References

[16]

[1] de Jong TP, Chrzan R, Klijn AJ, Dik P. Treatment of the neurogenic bladder in spina bifida. Pediatr Nephrol 2008; 23(6):889e96. [2] Gould CV, Umscheid CA, Agarwal RK, Kuntz G, Pegues DA. Guideline for prevention of catheter-associated urinary tract infections 2009. Infect Control Hosp Epidemiol 2010;31(4): 319e26. [3] Yang W, Chang PY, Lai JY, Cheng CH, Tseng MH. Vesico-cutaneous fistula: a simple method for continent urinary diversion. J Pediatr Urol 2014;10(6):1227e31. [4] Wyndaele JJ, Brauner A, Geerlings SE, Bela K, Peter T, Bjerklund-Johanson TE. Clean intermittent catheterization and urinary tract infection: review and guide for future research. BJU Int 2012;110(11 Pt C):E910e7. [5] Bakke A, Digranes A. Bacteriuria in patients treated with clean intermittent catheterization. Scand J Infect Dis 1991;23(5): 577e82. [6] Hiyama Y, Takahashi S, Uehara T, Hashimoto J, Nishinaka K, Kitamura H, et al. Emergence of antimicrobial-resistant uropathogens isolated from pediatric patients with cystitis on daily clean intermittent catheterization. J Infect Chemother : official journal of the Japan Society of Chemotherapy 2015; 21(10):703e6. [7] Chaudhry R, Balsara ZR, Madden-Fuentes RJ, Wiener JS, Routh JC, Seed P, et al. Risk factors associated with recurrent urinary tract infection in neurogenic bladders managed by clean intermittent catheterization. Urology 2017;102:213e8. [8] Webb RJ, Lawson AL, Neal DE. Clean intermittent selfcatheterisation in 172 adults. Br J Urol 1990;65(1):20e3. [9] Di Benedetto P. Clean intermittent self-catheterization in neuro-urology. Eur J Phys Rehabil Med 2011;47(4):651e9. [10] Krebs J, Wollner J, Pannek J. Urethral strictures in men with neurogenic lower urinary tract dysfunction using intermittent

[15]

[17]

[18]

[19]

[20]

[21]

[22]

catheterization for bladder evacuation. Spinal Cord 2015; 53(4):310e3. Cornejo-Davila V, Duran-Ortiz S, Pacheco-Gahbler C. Incidence of urethral stricture in patients with spinal cord injury treated with clean intermittent self-catheterization. Urology 2017;99:260e4. Afsar SI, Yemisci OU, Cosar SN, Cetin N. Compliance with clean intermittent catheterization in spinal cord injury patients: a long-term follow-up study. Spinal Cord 2013;51(8): 645e9. Pinder B, Lloyd AJ, Nafees B, Elkin EP, Marley J. Patient preferences and willingness to pay for innovations in intermittent self-catheters. Patient Prefer Adherence 2015;9: 381e8. Lim SW, Lee HE, Davis M, Park K. Perceived barriers and difficulties of intermittent catheterization: in Korean patients with spinal dysraphism and their parents. Neurourol Urodyn 2016;35(3):395e9. Harris CF, Cooper CS, Hutcheson JC, Snyder 3rd HM. Appendicovesicostomy: the mitrofanoff procedure-a 15-year perspective. J Urol 2000;163(6):1922e6. Clark T, Pope JCt, Adams m C, Wells N, Brock 3rd JW. Factors that influence outcomes of the Mitrofanoff and Malone antegrade continence enema reconstructive procedures in children. J Urol 2002;168(4 Pt 1):1537e40. discussion 1540. Thomas JC, Dietrich MS, Trusler L, DeMarco RT, Pope JCt, Brock 3rd JW, et al. Continent catheterizable channels and the timing of their complications. J Urol 2006;176(4 Pt 2): 1816e20. discussion 1820. Leslie B, Lorenzo AJ, Moore K, Farhat WA, Bagli DJ, Pippi Salle JL. Long-term followup and time to event outcome analysis of continent catheterizable channels. J Urol 2011; 185(6):2298e302. Jacobson DL, Thomas JC, Pope Jt, Tanaka ST, Clayton DB, Brock 3rd JW, et al. Update on continent catheterizable channels and the timing of their complications. J Urol 2017; 197(3 Pt 2):871e6. Faure A, Cooksey R, Bouty A, Woodward A, Hutson J, O’Brien M, et al. Bladder continent catheterizable conduit (the Mitrofanoff procedure): long-term issues that should not be underestimated. J Pediatr Surg 2017; 52(3):469e72. Sorg H, Tilkorn DJ, Hager S, Hauser J, Mirastschijski U. Skin wound healing: an update on the current knowledge and concepts. Eur Surg Res 2017;58(1e2):81e94. Koff SA, Mutabagani KH, Jayanthi VR. The valve bladder syndrome: pathophysiology and treatment with nocturnal bladder emptying. J Urol 2002;167(1):291e7.

Appendix A. Supplementary data Supplementary data to this article can be found online at https://doi.org/10.1016/j.jpurol.2020.02.004.