|Year : 2011 | Volume
| Issue : 3 | Page : 81-87
Bladder exstrophy: An overview of the surgical management
Department of Pediatric Surgery, All India Institute of Medical Sciences, New Delhi, India
|Date of Web Publication||4-Aug-2011|
Department of Pediatric Surgery, All India Institute of Medical Sciences, New Delhi - 110 029
Source of Support: None, Conflict of Interest: None
| Abstract|| |
Background: The surgical management of urinary bladder exstrophy is challenging. This paper describes the personal experience in a tertiary care hospital over a period exceeding a quarter of a century. Methods: During the period 1984-2010, 248 patients of the epispadias-exstrophy complex have been treated. The cases of classical bladder exstrophy (n = 210) form the basis of this paper. The stages/procedures used in the surgical reconstruction of bladder exstrophy included bladder closure with anterior abdominal wall reconstruction, bladder neck repair, ureteric reimplantation, epispadias repair and augmentation colocystoplasty in various combinations. Some of these patients had their initial operations done prior to 1984 or in other hospitals. Evaluation methods included, amongst others, clinical evaluation and urodynamic assessment. Eight patients opted out of treatment; 15 patients underwent permanent urinary diversion by either ureterosigmoidostomy or colon conduit. The remaining 187 patients were treated with bladder reconstruction, and of these, 132 patients have had at least one attempt at bladder neck reconstruction with 56 of these patients having undergone an augmentation colocystoplasty. Results: A total of 105 patients had socially acceptable continence: 57 from the bladder neck reconstruction group and 48 from the bladder augmentation group. Further attempts at continence surgery have been offered to the inadequately continent patients. Conclusions: Surgical management of bladder exstrophy demands patience and perseverance. It is possible to provide all patients with socially acceptable continence with bladder neck division and catheterizable continent stoma as the last resort. Urodynamic assessment has emerged as an essential tool in the follow-up evaluation of these patients. Anticholinergic medication with imipramine or oxybutinin is a useful adjunct in the overall management.
Keywords: Anticholinergic medication, bladder augmentation, bladder closure, bladder neck repair, epispadias repair, ureteric reimplantation, urinary bladder exstrophy, urinary continence, urodynamics
|How to cite this article:|
Bhatnagar V. Bladder exstrophy: An overview of the surgical management. J Indian Assoc Pediatr Surg 2011;16:81-7
| Introduction|| |
The multiplicity of the procedures available to close an exstrophied urinary bladder testifies to the fact that it is one of the most difficult challenges to the pediatric surgeon/pediatric urologist. The aim of primary closure is to repair the bladder and anterior abdominal wall, resulting in a cosmetically acceptable urinary reservoir which, if necessary, can be operated upon at a later date in order to achieve a dry patient capable of evacuating the bladder at will or with assistance. The role of pelvic osteotomy to achieve closure of the abdominal wall and in the achievement of continence is controversial. However, the pelvic brim needs to be completed so that the closed bladder can be retained in a recessed pelvic position and also to provide an angulation between the bladder and the urethra. This paper describes the experience at a tertiary care hospital.
| Materials and Methods|| |
During the period 1984-2010, 248 patients of exstrophy-epispadias complex have undergone surgical reconstruction. Of these, 36 patients were treated for epispadias and 2 patients were treated for variants of the bladder exstrophy. The remaining 210 patients of classical bladder exstrophy form the basis of this communication. Fourteen of these patients had undergone the initial bladder closure prior to 1984 and the remaining reconstruction procedures were carried out subsequently and 23 patients had undergone the bladder closure in other hospitals and were then referred for the subsequent reconstruction.
The operative techniques for the various stages of treatment have been published and demonstrated at various live operative workshops before and are described briefly.
Endotracheal tube general anesthesia with muscle relaxants and caudal morphine is administered. In older patients, an epidural catheter is placed for postoperative pain relief. The patient is placed in the supine position and a thorough hand scrub with chlorohexidine and povidone iodine is given.
A circumferential incision is made around the exstrophied bladder just outside the mucocutaneous junction. Cephalad, a triangular piece of skin, is excised, and caudad, the incision, is carried on to the urethral plate on the epispadic shaft. The incision is deepened from the cephalad extent and a plane is created between the peritoneum and the bladder wall. This allows an accurate dissection of the bladder muscle off the edge of the rectus abdominus on either side.
The ligaments of the bladder are divided. The urethral plate is then dissected off the corpora cavernosa using bipolar diathermy forceps till the corpora are clearly visible arching on to the pubic rami. An intersymphysial band of tissue is now seen and this is divided from the symphysis and mobilized for a short distance so that it can be brought to the midline. The distance between the mobilized urethral plate and the distal end of the incision on the epispadic shaft is measured and equal lengths of paraexstrophy flaps are mobilized on either side in cases with a short urethral plate (paraexstrophy flaps have not been used since 2006). A 3:1 length: width ratio of the paraexstrophy flaps is maintained. The corpora are then dissected off the inferior pubic rami till the vascular bundle.
The two paraexstrophy flaps are sutured to each other in the midline in two layers using 6/0 or 7/0 polydioxanone (PDS) sutures and the suture line is continued to approximate the proximal free edge of the paraexstrophy flaps to the urethral plate. Using the same suture material, the lateral edges of the paraexstrophy flaps are brought together in the midline and sutured to each other over a 5-F infant feeding tube. The paraexstrophy flaps are thus tubularized.
Both the ureters are cannulated with 5-F infant feeding tubes which are anchored at the ureteric orifices with 4/0 chromic catgut. The free edges of the bladder are trimmed so that a layer of healthy mucosa and muscle is available for closure which is achieved by bringing the cephalad portion down and suturing it to the caudad portion in a curved transverse suture line with 5/0 polyglactin sutures in two layers. The ureteric stents are brought out from either extremes of this suture line and they are anchored to the bladder wall with 4/0 chromic catgut. A 12-F malecot catheter is placed in the anterior wall of the bladder and anchored separately to the urethral catheter and the bladder wall with 4/0 chromic catgut.
The intersymphysial band of tissue is closed over the junction of the tubularized paraexstrophy flaps and the bladder with 3/0 polyglactin sutures. The corpora cavernosa are sutured to each other in the midline with 3/0 polyglactin sutures.
The technique of abdominal wall closure entails detachment of the rectus abdominis muscle from the superior pubic ramus on either side and suturing to each other in the midline with 2/0 polyglactin sutures. The muscles are then pulled caudad and re-anchored at the pubic symphysis. This completes the pelvic brim. The ureteric stents are brought out by lateral stab incisions, the malecot catheter is brought out in the midline, and a corrugated red rubber drain is placed in the space between the bladder and the abdominal wall through a stab incision. The urethral catheter is anchored at the glans and the ureteric stents and malecot catheter are anchored at the skin with 4/0 silk sutures. The subcutaneous tissue and skin are closed in layers. A simple dressing is applied to the wound. 
The ureteric stents are removed after 2 weeks and the bladder catheters 3 weeks after surgery. Skin sutures are removed between the 10 th and 14 th postoperative days. Adjunctive procedures include: bladder closure with simultaneous herniotomy or herniorrhaphy, bladder closure with bladder neck reconstruction, bladder closure with ureteric reimplantation.
In the postoperative period till bladder neck reconstruction (which is usually 1 year later), these patients are followed up in Pediatric Urology Clinic with regular assessment of the upper and lower urinary tract. Micturating cystourethrogram is done 1 year later to note the bladder capacity, reflux status and bladder residue, if any. Radionuclide renography and ultrasonography are used for the assessment of upper tract function. Urine cultures are done to confirm urinary tract infection (UTI). Care is taken to ensure that there is no outlet obstruction.
Bladder neck repair
This procedure is undertaken at least 1 year after primary closure of the bladder exstrophy. Surgical exposure is achieved by a vertical midline incision in the scar of the previous operation. The bladder is dissected free from the parieties and the peritoneum and mobilized up to the external urinary meatus. The bladder is then opened vertically right up to the external meatus. The region of the posterior urethra including the verumontanum is thus exposed. The region of the bladder neck can be identified as the most proximal part of the vertical folds of mucosa which run down towards the verumontanum from just below the ureteric orifices. The ureteric orifices are cannulated with 5-F infant feeding tubes which are anchored at the respective ureteric orifice with 4/0 chromic catgut. The aim of this operation is to tubularize the posterior urethra from the region of the bladder neck up to the external meatus. This is achieved by making two parallel incisions through all the layers on either side of the verumontanum from the region of the bladder neck up to the most distal part of the opened out bladder. The distance between the two incisions is equal to the circumference of a 6-10 F infant feeding tube depending on the size of the patient. The two triangular flaps, one on each side of the part to be tubularized, are preserved along with the mucosa. The tubularization is begun distally and performed with 6/0 PDS continuous suture; the mucosa and muscle are sutured in separate layers over a 6-10 F feeding tube. After the tubularization is completed up to the region of the bladder neck, this feeding tube is replaced with a 5-F feeding tube. The suture line is continued proximally to stitch the two triangular flaps to each other in the midline. The ureteric catheters are brought out by stab incisions in the lateral wall of the bladder; the left ureteric catheter is brought out from the right side and the right ureteric catheter is brought out from the left side. Both the ureteric catheters are anchored to the outer bladder wall also. A 12-14 F malecot catheter is introduced into the bladder by a stab incision in the region of the fundus. The flower of the malecot catheter is sutured to the tip of the catheter in the urethra with one 4/0 chromic catgut suture. The bladder is then closed transversely in two layers-mucosa and the muscle, with 5/0 continuous vicryl sutures. The malecot catheter is anchored to the bladder wall with 4/0 chromic catgut suture. The midline abdominal wound is closed in layers. The urethral catheter is anchored at the external urethral meatus and the malecot catheter is brought out through the main incision wound and anchored to the skin. The ureteric catheters are brought out through separate stab incisions on either side and anchored to the skin. The extravesical space is drained by a corrugated red rubber drain. A simple dressing is applied.
The bladder is irrigated with normal saline for the first 24 hours after operation if there is bleeding from the mucosa. The corrugated drain is removed on the 4 th day. The ureteric catheters are removed after at least 2 weeks, the malecot catheter at the end of 3 weeks and the urethral catheter at the end of 4 weeks.
Adjunctive procedures include primary bladder closure, epispadias repair, ureteral reimplantation and bladder augmentation. Postoperatively, in addition to renal biochemistry, the upper tracts are monitored with radionuclide renography and ultrasound scans, and the bladder is evaluated with micturating cystourethrography and ultrasonography for size, shape, residual urine and vesico-ureteral reflux. Urodynamic studies are also done. 
Preoperative urine cultures are performed to rule out a UTI. Bowel preparation is done with oral PEGLEC and rectal washes. Broad-spectrum intravenous antibiotics are administered preoperatively.
Detubularized sigmoid colocystoplasty is usually performed as an additional procedure. The procedure consists of isolation of a 15-cm loop of sigmoid colon and restoration of sigmoid continuity. The isolated loop is thoroughly cleansed with povidone iodine and slit open longitudinally between the tenia coli. The colonic patch is then sutured transversely to form a pouch which is then sutured to the generously opened fundus of the native bladder.
The patient and the parents are counselled and explained regarding the need for clean intermittent catheterization in the postoperative period. Adjunctive procedures include primary bladder closure, bladder neck reconstruction, bladder neck closure, ureteral reimplantation, catheterizable stoma, cystolithotomy and epispadias repair. Postoperative, slow saline irrigation of the reservoir is continued for 48 hours. Care is taken to ensure patency of the catheters in the postoperative period. The patients are followed up with an MCU and radioisotope renogram study 6 months following surgery. Prophylactic antibiotics are continued in the postoperative period. 
Preferably, this is performed after all other procedures. A major portion of the epispadias repair is achieved already during bladder closure. In effect, what remains is the most distal part of the urethra and the glans. This repair is achieved with simple tubularization and de-epithelialization and approximation of the glans. The corpora are sutured to each other over the urethral tube.
Ureteric reimplantation is performed by an extravesical mobilization of the ureters and then by either the Cohen or Leadbetter-Politano technique depending upon the bladder size. The indications for reimplantation include worsening of upper tract dilatation/grade of reflux, high intravesical pressures in the presence of reflux, recurrent pyelonephritis and the requirement of clean intermittent catheterization to empty the bladder.
These techniques have been in use for over the past three decades. Currently, a rigid staging of the reconstruction is not followed, thus allowing flexibility in combining stages depending upon the state of the tissues.
The author has not performed pelvic osteotomy in any patient. However, 16 patients had undergone either a posterior or an anterior iliac osteotomy as part of the primary treatment prior to referral.
Primary permanent urinary diversion
Primary diversion is seldom performed at our center. Only 13 patients have undergone primary urinary diversion by either an ureterosigmoidostomy (2 patients) or an ileal/colon conduit (11 patients) for bladders which were not amenable to closure even by staged procedures. In addition, two patients had undergone colon conduit prior to referral for external genitalia reconstruction.
Assessment of continence
The assessment of continence was graded into four groups. Group I: Dry during day and night for >2 hours, no stress incontinence. Group II: Dry during day for >1.5 hours, occasional wetting at night (<2 episodes/week). Group III: Dry during day for <1 hour, daily nocturnal wetting and stress incontinence. Group IV: Continuously wet. Patients with group II continence are treated with oral imipramine or oxybutinin. Patients with group III continence are given a trial of oral imipramine or oxybutinin, and if they respond, then bladder augmentation is performed only if the bladder is small in capacity or if the intravesical pressures are high on urodynamic evaluation. A repeat bladder neck reconstruction is performed in all cases with group IV continence. Failure to improve to group I or II continence will require bladder neck division, bladder augmentation and an appendico-vesicostomy for clean intermittent catheterization. The ultimate goal of treatment is to put all patients in group I with or without bladder neck division, bladder augmentation and appendico-vesicostomy.
Artificial slow fill cystometry was performed 1 year after the bladder neck reconstruction and again 1 year after the augmentation colocystoplasty using the Phoenix Griffon machine (Albyn Medical, Dingwall, UK) and software (an interactive computer-based program). The parameters studied included maximum cystometric capacity (MCC), expected capacity (EC; 16 × age + 70 ml), compliance ("20 below" capacity) (20BC), end fill pressure (EFP), unstable/mass contractions (UC/MC) and voiding and residual volume (RV).
| Results|| |
Eight patients opted out of treatment after they were counseled regarding the need for multiple operations and long-term follow-up. Three patients developed chronic renal failure and one patient died in the postoperative period following augmentation colocystoplasty. Almost all patients have had at least one follow-up visit in the last 5 years. Patients who have completed staged reconstruction and are well adjusted with either spontaneous voiding or clean intermittent catheterization with or without bladder augmentation tend not to visit the clinic for long periods of time. The oldest patient on follow-up is 38 years of age.
There were 27 girls. The median age at primary bladder closure was 36 months (range 5 days-156 months). Only 20 patients were operated in the newborn period; however, 21 patients who had been operated in the newborn period in other hospitals were referred at a later age for re-do bladder closure following complete dehiscence. Of the remaining patients, 67 were under 1 year of age and 16 patients presented for treatment after 10 years of age.
Significant squamous metaplasia and/or cystitis cystica changes were seen in the majority of children. A majority of the late presenters were children from low socioeconomic status who gave a history of recurrent trauma/ulceration and bleeding from the exposed bladder plate. Twenty-five patients referred with bladder dehiscence had 1 or more attempts at closure elsewhere. The bladder plate was uniformly non-pliable in all these children.
Inguinal hernia was the commonest associated finding in more than 50% patients; one other patient required surgical treatment for congenital laryngeal stenosis.
The average hospital stay was 25 days following primary bladder closure, 28 days following bladder neck reconstruction (with or without additional procedures) and 32 days following augmentation colocystoplasty (with or without additional procedures).
Wound healing was good, with a linear scar, in the majority of patients; dehiscence was seen in 20 (10.5%) children. In the majority, the dehiscence was limited to lower third of the abdominal incision; wound infection was the primary etiological factor. Only four patients who underwent bladder closure in this hospital required redo bladder closure. Two patients had early ureteric stent obstruction or inadvertent removal. In another patient, there was persistent oozing from the abdominal closure which led to a hematoma and secondary infection. Bladder outlet obstruction was seen in four children who required calibration/dilatation under anesthesia. These children had recurrent episodes of UTI and showed significant residue on ultrasonography/MCU. Recurrent UTIs were seen in 24 patients. Twenty-three patients developed bladder calculi after surgery, including augmentation colocystoplasty. Renal scarring was seen on dimercapto succinic acid (DMSA) scans in 12 patients on follow-up. These included patients who had recurrent episodes of urinary tract infection with documented vesico-ureteric reflux on micturating cystourethrogram. Upper tract assessment by Diethylenetriamine Penta-acetic Acid (DTPA) renography showed normal or non-obstructive hydroureteronephrosis in all patients.
Bladder neck reconstruction has been performed in 132 patients and 56 of these have subsequently undergone bladder augmentation for either a small capacity bladder or high intravesical pressures. Of the 76 patients who have undergone bladder neck reconstruction without bladder augmentation, 37 are fully continent (group I), 20 have group II continence, 12 have group III continence and 7 are still incontinent. Of the 56 patients who have undergone bladder augmentation following or with bladder neck reconstruction, 46 are continent (group I) (9 patients void spontaneously and the remaining 37 patients require clean intermittent catheterization). Of the remaining 10 patients, 2 have group II continence, 4 have group III continence and 4 are incontinent. Of the 37 patients on clean intermittent catheterization, only 3 have undergone bladder neck division and appendico-vesicostomy; the remaining patients are well adjusted on catheterization via the native urethra.
Augmentation colocystoplasty has been done in 56 patients. Urodynamic improvement occurred in all patients; the mean maximum capacity showed a sevenfold increase, the mean compliance showed a 6.6 times increase and the mean volume at detrusor pressure 20 cm of H 2 O showed a 7.9-fold increase. The biochemical changes and bone mineral density changes have been reported in another communication.
The ureters have been reimplanted in 67 patients: in 4 patients along with bladder closure, in 40 patients along with bladder neck reconstruction, and in the remaining either as a separate procedure or along with bladder augmentation.
| Discussion|| |
Division and mobilization of the urethral plate allows the bladder to be pushed back so that it lies comfortably in a recessed position in the pelvis. The raw area thus created is bridged by the paraexstrophy flaps which also serve to provide an extra 2-3 cm of urethra. The length of the urethra has been reported to be an important determinant of success in the achievement of continence after repair of bladder exstrophy.  Tubularization of the paraexstrophy flaps can be safely done without compromising the vascularity if the mobilization and tissue handling is done carefully and the length: width ratio is meticulously maintained. In these patients, it serves the same purpose as epispadias repair in providing a certain amount of outlet resistance which in turn allows the bladder capacity to develop. Better bladder capacity has been shown to develop if epispadias repair precedes bladder neck reconstruction.  Vascular compromise resulting in sloughing off of the paraexstrophy flaps has not occurred in any of our patients, neither has any patient developed a refractory stricture. However, this complication of paraexstrophy flaps has been reported before.  A mild degree of narrowing may occur at the junction of the paraexstrophy flaps and the bladder but this responds very well to one or two dilatations. However, in the recent past, patients with paraexstrophy flaps have experienced difficulties in self-catheterization. Hence, currently, the use of paraexstrophy flaps has been restricted to only those with severe shortening of the urethral plate.
Closure of the bladder by bringing the cephalad portion down and suturing it in a curved transverse manner allows a peritoneum covered dome of bladder to reside in the pelvis, and since the suture line is at the bottom of the bladder, it does not adhere to the wound in the parieties. Both these factors allow the bladder to grow in capacity upward into the pelvis. Lack of adhesions with the parieties helps in making the dissection of the bladder easier during subsequent surgeries for bladder neck reconstruction, re-implantation of ureters for correction of vesico-ureteral reflux and augmentation colocystoplasty.
The role of pelvic osteotomy in the overall management of patients with urinary bladder exstrophy has been controversial. Pelvic osteotomy is not essential for repair of the anterior abdominal wall in bladder exstrophy patients. Equally good cosmetic results have been achieved without pelvic osteotomy.  Clinical experience has shown that even with different types of pelvic osteotomy, the closure of pubic symphysis does not remain intact over a period of time. ,, Hence, what is really important is the completion of the pelvic brim with or without osteotomy.  Pelvic osteotomy may also call for participation by other departments, particularly orthopedics, and this may cause logistical problems at times. There is no common agreement on the type of osteotomy which is required as is evident from the many different types of pelvic osteotomies which have been described. ,,,, A high degree of morbidity is also associated with the pelvic osteotomy and the restrictive dressings which are applied subsequently. In the procedure which has been described, the morbidity is much less, dressings are not restrictive and the patient is encouraged to adopt comfortable postures in bed and later move about even with the catheters. Cosmetically, the results have been good with this procedure for primary bladder closure. In addition, pelvic osteotomy may be the cause of serious complications. 
The approximation of pubic bones was once considered to be essential for the achievement of continence in bladder exstrophy patients. However, our experience and that of others has shown that good urinary continence can be achieved even without pelvic osteotomy. , A report has even suggested that although there may be a possibility of patellofemoral instability in exstrophy bladder patients with unapproximated pelvis, all the patients were active with regard to daily life and sport related activities. 
Bladder neck reconstruction has been done by a modification of the Young-Dees-Leadbetter technique. The triangular flaps of the original operation are incorporated into the bladder instead of de-epithelializing them and using them to buttress the bladder neck repair. This not only helps in utilizing all bladder tissue for enhancing the bladder capacity, but also improves the results of surgery with regard to urinary continence. 
The assessment of continence has a multidimensional approach. We have used a very practical grading based on how long the patient can remain dry and this essentially establishes socially acceptable continence. A number of patients get adjusted to a higher frequency of micturition in order to remain dry and they would not accept bladder augmentation for the sole purpose of increasing the dry period. However, a more objective method of assessing the results of surgery is the computer-based urodynamic evaluation. Even before bladder neck reconstruction, urodynamic assessment can be predictive for detrusor function and the ability of the bladder to increase in size without high intravesical pressures and also the application of anticholinergic therapy to enhance bladder volume.  Following bladder neck reconstruction, urodynamic assessment provides an objective correlation with the clinical assessment of continence. It also helps in planning pharmacotherapy for elimination of uninhibited detrusor contractions, improving bladder compliance and reducing intravesical pressures. All these factors help in improving continence. ,
Bladder augmentation is the final option in the surgical management of bladder exstrophy for a variety of reasons. The augmentation can be done with the use of ureters or parts of the gastrointestinal tract. We have preferred sigmoid colon because it is nearest to the bladder, the vascularity and tissues are reliable and it offers the best options keeping in mind the bladder dynamics.  The most important contribution of bladder augmentation, apart from the increase in bladder capacity, is the increase in dry periods, relief of intravesical pressure which then protects the upper tracts from pressure-related damage and the maintenance of separate fecal and urinary streams. Hence, we advocate the liberal use of bladder augmentation in the management of bladder exstrophy. Clean intermittent catheterization is frequently required in these patients to empty the bladder. Although this may be resisted initially, counselling and sharing experience with patients who have had this procedure before helps greatly in the final acceptance and practice. A stoma on the abdomen is usually not accepted and hence, we perform it only in situations when catheterizing the native urethra is not possible or if the bladder neck has to be divided due to failed repeated attempts at bladder neck reconstruction.
Thus, the strategy in the management of bladder exstrophy is to convert the exstrophied bladder patch into a continent reservoir which can be periodically emptied either spontaneously or with assistance and at the same time to preserve the upper tracts. This can be achieved in one stage or in multiple stages. The procedures available for the reconstruction are bladder closure, bladder neck repair, epispadias repair, ureteric reimplantation, bladder augmentation, bladder neck division and a catheterizable continent stoma. These procedures performed individually or in strategic combinations and stages are fully capable of achieving the goals of management, with a little help from medication to relax the detrusor. There is no reason why every patient of bladder exstrophy cannot be rendered dry. It requires patience and perseverance from both the surgeon and the patient. The importance of record keeping, documentation and long-term follow-up cannot be overemphasized.
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