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PubMed2000

Penile reconstruction combined with bladder closure in the management of classic bladder exstrophy: illustration of technique.

OBJECTIVES: Interest has increased in combining procedures during reconstruction of bladder exstrophy in an effort to reduce the number of procedures required for reconstruction and to improve results. This study illustrates our technique of reconstruction and summarizes our current experience with the combination of epispadias repair and bladder closure during initial reconstruction or following prior failed bladder closure. METHODS: Twenty-four boys with classic bladder exstrophy underwent combined bladder closure and epispadias repair. The mean patient age was 20 months, and 18 boys had a prior failed closure. Osteotomies were performed in all patients. RESULTS: No instances of bladder prolapse or dehiscence were noted on follow-up. Urethrocutaneous fistula developed in 7 patients. Eventual bladder neck reconstruction has been performed in 11 boys (6 boys are dry day and night, 3 are dry during the day with occasional wet episodes at night, 2 have required follow-up bladder augmentation and continent diversion for persistent incontinence), and 1 boy had augmentation at the same time as bladder neck reconstruction. Twelve boys are awaiting adequate capacity for bladder neck reconstruction, and 1 is awaiting bladder augmentation and continent stoma construction. CONCLUSIONS: Epispadias closure can be safely combined with bladder closure in select patients with classic bladder exstrophy. Complication rates and cosmesis approximate that achieved with staging the two procedures. This achievement represents strict patient selection and attentive follow-up.

Abnormalities, Multiple↗

PubMed2003

Pelvic and extremity immobilization after bladder exstrophy closure: complications and impact on success.

OBJECTIVES: A successful initial bladder closure is critical to the development of adequate bladder capacity and ultimate continence among patients with bladder exstrophy. Although secure pelvic fixation is essential to success, the various methods of pelvic and extremity immobilization and their impact on surgical outcome have not previously been examined. METHODS: A review of the approved bladder exstrophy database at our institution identified 194 patients for whom data were available regarding initial bladder closure and the method of pelvic immobilization. The records were reviewed with respect to age at bladder closure, use/type of osteotomy, method of pelvic immobilization, complications related to the immobilization technique, and surgical outcome. RESULTS: Of 194 patients identified, initial exstrophy closure failed in 86. Of these 86 patients, the initial closure failed in 80 at another hospital who were referred to our institution for additional treatment. Of the 114 initially closed at this institution, 6 failed. The success rates for initial and second closures were highest after osteotomy (75% versus 38% and 81.5% versus 27%, respectively), and when the patients were immobilized with an external fixator and 6 to 8 weeks of modified Buck's traction with osteotomy (96%) or 4 to 6 weeks of modified Bryant's traction without osteotomy (61%). The spica cast and "mummy wrapping" immobilization techniques were less effective and were associated with significant complications. CONCLUSIONS: These results demonstrate that a successful exstrophy bladder closure depends on the proper use of the osteotomy and effective postoperative immobilization of the pelvis. Our best outcomes have been achieved using lower extremity traction (with or without external fixation of the pelvis).

Age Factors↗

PubMed1980

A family study of bladder exstrophy.

The families of 102 index patients with bladder exstrophy treated at The Hospital for Sick Children, Great Ormond Street were studied in an attempt to arrive at an empirical risk of recurrence for sibs. Of the 102 patients, 89 had complete exstrophy, eight had partial exstrophy (variant), and five had exstrophy of the cloaca. In all they had 162 sibs, none of whom had bladder exstrophy. The risk to sibs, in this study and from reports by surgeons of sib pairs in their consecutive series, is low and probably less than 1%. There is a suggestion of an increase in central nervous system malformation in sibs as well as in the index patients.

Bladder Exstrophy↗

PubMed1997

Spinal abnormalities in classic bladder exstrophy.

OBJECTIVE: To determine the frequency and clinical sequelae of significant spinal malformations in children born with classic bladder exstrophy. PATIENTS AND METHODS: All patients evaluated or treated for classic bladder exstrophy at this institution were reviewed retrospectively. Radiographs or reports pertinent to the spine were retrieved and reviewed with a paediatric radiologist and all vertebral abnormalities categorized. Clinical charts of those with spinal anomalies were reviewed to determine any clinical neurological disorders associated with the radiographic findings. RESULTS: Of 423 patients with classic bladder exstrophy who were identified, 299 had radiographs or reports available for adequate review. Of these, 34 (11%) normal variants, e.g. spina bifida occulta and lumbarization or sacralization of vertebrae, were identified. Abnormalities of spinal curvature were identified in eight patients (2.7%), all with uncomplicated scoliosis. Spinal dysraphism was diagnosed in 12 patients (4%) and included myelomeningocele, lipomeningocele, scimitar sacrum, posterior laminal defects in two or more vertebrae, vertebral fusion and hemivertebrae. The one patient with myelomeningocele had clinical neurological dysfunction, giving an overall incidence of 0.3%. CONCLUSIONS: Spinal anomalies, excluding normal variants, occur in children born with classic bladder exstrophy at a rate of about 6.7%. The incidence of this association is much less than that for cloacal exstrophy. Although rare, neurological dysfunction can occur in the case of spinal dysraphism. Paediatric urologists and neurologists should be aware of this significant difference between patients with classic bladder and cloacal exstrophy to properly diagnose, evaluate and treat the attendant neurological problems.

Adolescent↗

PubMed2001

The dilemma of prenatal diagnosis of bladder exstrophy: a case report and a review of the literature.

Bladder exstrophy is a very rare congenital anomaly characterized by the exteriorization of the viscera on the abdominal surface, low insertion of the umbilical cord, divergent pubic rami and abnormal exterior genitalia. We report a case of prenatal diagnosis of fetal bladder exstrophy in which the urachus resembled the normal bladder. Considering that non-visualization of the bladder is one of the main findings in cases of bladder exstrophy, special attention should be given to fetal lower abdominal cystic images in order to differentiate them from a normally positioned fetal bladder. A review of the literature together with a sonographic study of the urachus is provided.

Abortion, Therapeutic↗

PubMed1984

Urethral reconstruction in boys with classical bladder exstrophy.

A total of 24 boys with classical bladder exstrophy underwent initial urethral reconstruction at our hospital between 1975 and 1982. Penile reconstruction in male patients with classical bladder exstrophy includes penile lengthening, release of the dorsal chordee and reconstruction of the urethra. The former 2 procedures are performed during the primary bladder closure and urethroplasty usually follows bladder neck reconstruction. A modified Young urethroplasty was done in 22 of the 24 patients. Preputial pedicle grafts of free full thickness skin grafts were used for urethroplasty in 2 boys with insufficient penile skin. Fistulas requiring surgical revision developed after urethroplasty in 21 per cent of the patients. A prior osteotomy was associated with a decreased fistula rate. The cosmetic and preliminary functional results of the penile reconstruction were assessed by parental interviews. The definitive assessment of the penile reconstruction will be determined when these boys reach sexual maturity.

Bladder Exstrophy↗

PubMed2005

Surgical repair of bladder exstrophy in the modern era: contemporary practice patterns and the role of hospital case volume.

PURPOSE: Bladder exstrophy is a rare condition, and data are lacking regarding practice patterns in its surgical management. We used a large nationwide database to investigate practice patterns of bladder exstrophy repair. MATERIALS AND METHODS: We used the Nationwide Inpatient Sample (1988 to 2000) to identify patients who underwent surgical repair of bladder exstrophy (International Classification of Disease-9 code 578.6). We analyzed factors affecting practice patterns and outcomes. Hospital volume was based on caseload during the highest volume year of study participation (high volume 5 or more, mid volume 3 to 4 and low volume less than 3 cases). RESULTS: We identified 407 cases. Approximately half of the patients (53.2%) were hospitalized within 24 hours of birth, although 28% of patients were older than 1 year. Of the patients 54% were male. Exstrophy repair is extremely resource intensive. In this series mean length of hospital stay (LOS) was 24.6 +/- 22.8 days, and mean inflation adjusted hospital charges were 62,302 dollars (median 39,978 dollars). High volume hospitals (HVHs) had lower hospital charges (37,370 dollars) than mid volume (51,778 dollars) or low volume hospitals (LVHs, 50,474 dollars, p = 0.0095). On multivariate regression HVHs had lower charges even after controlling for other significant predictors, including LOS (p <0.0001). Patients at HVHs were more likely to undergo osteotomy (p = 0.007). Six patients died after exstrophy repair (1.5%), all of whom had been born prematurely (p <0.0001). Although death was more likely at LVHs, this was due to the fact that more patients at LVHs were born prematurely (4.2% at HVHs vs 5.9% at mid volume hospitals and 11.1% at LVHs, p = 0.027). CONCLUSIONS: Bladder exstrophy repair carries a high risk of morbidity and is resource intensive. Variations between high and low volume hospitals in practice patterns and case mix may contribute to observed differences in resource use, LOS and clinical outcomes.

Bladder Exstrophy↗

PubMed1997

Bladder exstrophy: evaluation of factors leading to continence with spontaneous voiding after staged reconstruction.

PURPOSE: We performed a long-term retrospective review of patients with bladder exstrophy to evaluate the results of staged surgical reconstruction in regard to urinary continence, spontaneous voiding and preservation of the upper urinary tract. MATERIALS AND METHODS: We reviewed the charts of 42 boys and 15 girls treated at Hôpital St. Joseph for bladder exstrophy between 1965 and 1995. All patients underwent staged repair, including bladder augmentation in 7 (12%) and secondary urinary diversion in 13 (23%). Criteria for good outcome in terms of continence are defined and factors influencing outcomes are reviewed. RESULTS: A total of 38 patients (67%) achieved good or acceptable urinary continence (22 or 39% and 16 or 28%, respectively). Outcome was poor in 19 patients, including 13 (23%) who underwent secondary urinary diversion. Bladder stones, which developed in 13 patients (23%), were the most common long-term complication of bladder exstrophy reconstruction. Ten patients who underwent pelvic osteotomies ultimately had better continence and 9 are dry. Bladder neck reconstruction was performed at a later age overall (mean 10 years). Repeat bladder neck reconstruction was generally associated with poor results. The technique of bladder neck reconstruction did not appear to influence outcome. CONCLUSIONS: A carefully planned surgical reconstruction for bladder exstrophy can lead to satisfactory long-term urinary continence in most patients. Factors contributing to successful results include early bladder closure, pelvic osteotomy, adequate bladder neck reconstruction with bladder neck suspension in girls, and a motivated child and family. Alternatives to surgical reconstruction should be discussed. Ultimate predictors of outcome in bladder exstrophy repair are difficult to ascertain.

Adolescent↗

PubMed1997

Orthopaedic long term aspects of bladder exstrophy.

This study evaluates long term orthopaedic aspects of children with bladder exstrophy who were operated on using different techniques and at different ages. Data were accumulated from 20 patients with an age range of 2 to 29 years (average, 13 years). Fourteen patients underwent pelvic osteotomy. Interviews and physical examinations confirmed that, in the long term, children with classical bladder exstrophy do not have significant orthopaedic problems or disability, whether or not they underwent pelvic osteotomy. Radiographic imaging showed normal hip joint configuration with marked pubic diastasis. There were no clinical problems associated with the diastasis. Pelvic computed tomography studies in 7 patients showed marked remodeling of the femora and acetabula. Radiographs of the spine showed a curve in 7 (47%) of the patients, but in only 3 cases was the curve larger than 10 degrees. Pelvic osteotomy is indicated during surgical correction of bladder exstrophy to facilitate closure of the abdominal wall to prevent postoperative wound dehiscence and possibly achieve better urinary control in older age. However, there is no clear indication for pelvic osteotomy from an orthopaedic point of view.

Adolescent↗

PubMed2005

Mucocolpos associated with bladder exstrophy: a case report.

INTRODUCTION: We present the first reported case demonstrating an association of mucocolpos and bladder exstrophy. MATERIALS: A term baby girl presented with bladder exstrophy and underwent a successful primary bladder closure and ureteral reimplantation. Subsequently, she presented with a fluid-filled pelvic mass which was found to be a mucocolpos. RESULTS: Surgical drainage of the collection was required. CONCLUSION: Mucocolpos should be considered in the differential diagnosis of a pelvic mass in a patient with bladder exstrophy.

Bladder Exstrophy↗

PubMed2004

Complete repair of bladder exstrophy in boys: can hypospadias be avoided?

INTRODUCTION: Complete repair of classic bladder exstrophy in male newborns has been successful with minimal morbidity. However, the technique may create hypospadias in some cases. We have recently adopted a modification to obtain an orthotopic meatus in bladder exstrophy boys. MATERIALS AND METHODS: Between November 1998 and December 2002 complete repair of classic bladder exstrophy was carried out in 27 boys. Complete penile disassembly was performed in 22 boys including 4 newborns and 18 older children; mean age 3+/-2 years old. Modified disassembly was used in the last 5 boys including 4 newborns and a 9-month-old boy. During repair of epispadias, the dissection starts on the ventral aspect of the penis as usual. The urethral plate is separated from both corpora cavernosa to allow ventral transposition of the plate. In the complete disassembly technique, the urethral plate is completely separated from both hemiglans. In this modification, while the urethral plate is completely separated from both corpora cavernosa, the extreme distal end of the urethral plate remains attached to the distal ends of both hemiglans. Thus, when the plate is tubularized with fine interrupted sutures the meatus ends up at the tip of the glans penis. The symphysis pubis is re-approximated as usual and corpora cavernosa are approximated dorsally in the midline. RESULTS: There was no major complication. Mean follow-up was 43+/-7 and 7+/-4 months for complete and modified disassembly groups respectively. Of the 22 boys, who underwent complete disassembly, 15 (68%) ended up with hypospadias and 7 (32%) had an orthotopic meatus. Modified disassembly has resulted in an excellent cosmetic appearance and orthotopic meatus in the 5 boys. Penile length was not shorter than boys who underwent complete disassembly. Parents of the 5 boys noticed normal straight morning erection. CONCLUSION: Although complete penile disassembly allows ventral placement of the urethra, hypospadias is created in approximately two thirds of the cases. When the extreme distal end of the urethral plate remains attached to the distal ends of both hemiglans (modified disassembly), orthotopic meatus can be obtained in all boys. With modified disassembly, posterior mobilization of the bladder and urethra does not result in corporal angulations or shortening because of the proportionate inward movement of the corpora that accompanies symphyseal approximation. The modification is feasible in newborns and infants. These short-term results may obviate the need for later penile reconstructive procedure.

Bladder Exstrophy↗

PubMed2003

Seven new cases of familial isolated bladder exstrophy and epispadias complex (BEEC) and review of the literature.

Clustering of the bladder exstrophy and epispadias complex (BEEC) has been described in 29 families. To explore the genetic contribution we located new families through the German and Austrian Bladder Exstrophy Support Group and the Association for the Bladder Exstrophy Community (predominantly US and Canada). We report six new families with two occurrences of BEEC, one family where the proband is the product of a consanguineous union, and four discordant twin pairs. In conjunction with the published clinical and epidemiological reports this collection suggests that there is a significant genetic predisposition for susceptibility to the BEEC. It also highlights the importance of self-referral groups for recruiting families for multicenter collaborative research efforts to identify susceptibility loci.

Abnormalities, Multiple↗

PubMed2005

Acetabular coverage in bladder exstrophy: role of 3-dimensional computed tomography.

We studied the acetabular coverage in bladder exstrophy patients and normal control children. The study included 13 patients with bladder exstrophy, who were divided according to their ages into group I below 5 years, group II between 5 and 10 years, and group III above 10 years of age. Four normal children served as controls, 2 were below 5 years, one was 7 and one was 12 years old. We measured the acetabular coverage (AC) and the center-edge-angle (CEA) by utilising the reconstructed 3-dimensional CT images. The mean right and left AC angles were 82 +/- 12 and 82 +/- 20 degrees in group-I, 107 +/- 15 and 103 +/- 17 in group-II and 119 +/- 24 and 116 +/- 22 in group-lII, respectively (p < 0.05). The mean right and left CEA angles were 33 +/- 4 and 32 +/- 4 degrees in group-I, 30 +/- 4 and 29 +/- 5 in group-II and 25 +/- 2 and 24 +/- 1 in group-III, respectively (p < 0.05). In controls, the mean right and left AC angles were 151 +/- 2 and 153 +/- 2 degrees for children below 5 years, 166 and 165 degrees in the 7 year-old child and 180 and 180 degrees in the 12 year-old child. The CEA angle ranged from 15 to 25 degrees. Acetabular coverage in bladder exstrophy patients is deficient during the first 5 years of life. Although it gradually improves over time, it does not reach to normal value.

Acetabulum↗

PubMed2005

Complete primary repair of bladder exstrophy in children presenting late and those with failed initial closure: single center experience.

PURPOSE: Complete primary repair (CPR) of bladder exstrophy using Mitchell's technique gained wide popularity. We present a single center experience with CPR in 30 children with bladder exstrophy presenting late or after failed initial closure. MATERIALS AND METHODS: Between November 1998 and November 2003, 45 patients underwent CPR of bladder exstrophy using Mitchell's technique. All 22 boys and 8 girls presenting beyond the age of 1 year were evaluated. Of the 30 patients 19 (63%) had a history of failed exstrophy closure. Mean patient age at surgery was 3.2 years (range 1 to 8). Bilateral anterior iliac osteotomies were performed in all children. Ultrasound was performed before surgery and 3 months thereafter in all patients. Voiding cystourethrography was obtained at 3 months and then annually. Continence was defined as dry intervals of 3 hours or more. RESULTS: Mean followup is 40 months (range 5 to 64). Concomitant intestinal bladder augmentation was performed in 5 children (17%). The repair resulted in hypospadias in 17 of 22 boys (77%). Following catheter removal 7 patients (23%) had suprapubic urine leakage that ceased spontaneously in all. Early postoperative hydronephrosis was present in 19 of the 30 children (63%) and resolved spontaneously in all. Six patients (20%) had febrile urinary tract infection that was treated conservatively. Vesicoureteral reflux was present in 23 children (68%). Of the 5 patients treated with concomitant bladder augmentation 2 are continent, 2 underwent bladder neck closure and 1 underwent bladder neck reconstruction (BNR). All 5 patients are currently dry. The remaining 25 patients had a mean bladder capacity of 90 ml (range 30 to 200) with continence in 6 children (24%). Continence was achieved in 3 of 6 girls (50%) versus 3 of 19 boys (16%). Five patients underwent BNR with ileocystoplasty in 4. The remaining 14 patients are awaiting BNR with or without bladder augmentation. CONCLUSIONS: CPR of bladder exstrophy is feasible in children presenting late or after failed initial closure. Concomitant intestinal bladder augmentation was required in 17% of our patients. The procedure resulted in hypospadias in 77% of the boys. Continence was achievable in 50% of the girls without subsequent bladder neck surgery. On the other hand, most boys (84%) will require BNR with or without augmentation.

Bladder Exstrophy↗

PubMed1996

Creation of a model of bladder exstrophy in the fetal lamb.

PURPOSE: To our knowledge we describe the first reproducible large animal model to mimic the congenital birth defect of classic bladder exstrophy. MATERIALS AND METHODS: Eight male and 15 female fetal sheep underwent in utero surgical creation of classic bladder exstrophy. The fetus was then replaced within the amniotic sac and allowed to come to term. A sham procedure was performed on 16 male fetuses. RESULTS: After the creation of bladder exstrophy 53% of the female and 62% of the male fetuses survived to full term and through delivery. Of the male fetuses that underwent sham surgery 62% also survived through delivery. Lambs with exstrophy were born with the bladder wall in continuity with the abdominal wall and exposed to the environment. The sham operated male lambs had a well healed abdominal scar. CONCLUSIONS. To our knowledge we have devised the first reproducible large animal:model of bladder exstrophy. This model will be useful in studying bladder development and composition in the patient with exstrophy. It may also serve as a useful tool in developing innovative forms of surgical treatment for such patients.

Animals↗

PubMedDate not supplied

[Bladder exstrophy: a neonatal emergency. II: Urinary diversion].

The goal of the therapy involving the children with exstrophy of bladder must include urinary continence, protection of renal function and a good quality of life. Primary closure of the bladder would seem to be the ideal solution, but when it is impossible or fails, ureterosigmoidostomy can provide satisfactory solution. We have treated 15 selected exstrophied patients with ureterosigmoidostomy. The average length of follow up was 8 years and 9 months (range is from 19 years to 18 months). All patients have normal renal function; in 13 patients upper urinary tract is normal and two have a moderate ureteral dilatation. In 13 patients the fecal and urinary continence is complete, in two it's acceptable. We report radiological and manometric studies of the rectum and sigma after ureterosigmoidostomy. The long term results are excellent; it is mandatory to have correct indications, meticulous care in the operative technique producing a long submucosal tunnel with direct anastomosis of the ureter to the bowel, post operative management and follow up care.

Adolescent↗

PubMed1987

The importance of a successful initial bladder closure in the surgical management of classical bladder exstrophy: analysis of 144 patients treated at the Johns Hopkins Hospital between 1975 and 1985.

The staged functional closure of classical bladder exstrophy has produced improved results for many urologists involved in the surgical management of this congenital anomaly. To determine which factors are most important for achieving a successful outcome (urinary continence with preservation of normal renal function) the 144 patients treated at The Johns Hopkins Hospital between 1975 and 1985 were reviewed. Of these patients 51 were managed entirely at our institution, while 93 had had the initial surgical treatment elsewhere. All patients who had undergone primary bladder closure and bladder neck reconstruction were divided into 2 groups: group 1--patients who had a successful initial bladder closure (an exstrophied bladder that is converted into a complete epispadias without wound infection, dehiscence or any degree of bladder prolapse on the first attempt) and group 2--children whose initial bladder closure was not successful. Both groups were analyzed with respect to bladder capacity at the time of bladder neck reconstruction, time required for the bladder to become sufficiently large for bladder neck reconstruction, urinary continence rate, and interval between bladder neck reconstruction and achievement of urinary continence. Patients in group 1 had the largest bladders at the time of bladder neck reconstruction (mean capacity 79 cc, p equals 0.03), shortest intervals between primary closure and bladder neck reconstruction (mean 3.5 years, p equals 0.006), highest urinary continence rate (92 per cent, p equals 0.002), and the shortest interval between bladder neck reconstruction and achievement of urinary continence (mean 1.5 years, p equals 0.18). These findings suggest that a successful initial bladder closure is an important factor for obtaining a larger bladder more quickly and for achieving a high urinary continence rate in patients with classical bladder exstrophy undergoing the staged functional bladder closure.

Bladder Exstrophy↗

PubMed2001

A population based analysis of continence outcomes and bladder exstrophy.

PURPOSE: Whereas the literature on bladder exstrophy is replete with outcomes of specific continence surgical procedures in highly select patients, there are no data on the outcomes related to continence for a complete exstrophy population, which is more comprehensive with respect to a variety of surgical procedures performed to achieve continence. To provide urologists and patients with an overview of potential continence outcomes devoid of any selection bias, we report on a comprehensive exstrophy population, focusing on the various procedures required for urinary continence. MATERIALS AND METHODS: We reviewed the charts of all patients with bladder and cloacal exstrophy who underwent a staged repair to achieve urinary continence at a single institution between 1988 and 1998. Urinary continence was then correlated to the types of surgical procedures, and subgroup analysis for predictors of urinary continence was performed. The type of bladder neck reconstruction allowed subgrouping cases into group 1-bladder neck reconstruction only, group 2-bladder neck reconstruction with augmentation and/or appendicovesicostomy and group 3-bladder neck closure. RESULTS: Of the 43 patients identified 26 were male, 4 had cloacal exstrophy and 3 had complex exstrophy variants with ectopic hindgut and spina bifida. Groups 1 to 3 comprised 9, 15 and 19 patients with urinary continence rates of 56%, 67% and 100%, respectively. The age at which patients became continent was delayed in groups 2 and 3 (8.2 and 8.7 years, respectively) compared to group 1 (4.8). Of all the potential variants measured gender was the strongest predictor of continence with 94% of females versus 69% of males achieving it. Of the males those with (57%) compared to those without (83%) a history of bladder neck stenosis or paraexstrophy flaps had worse continence. Repeat bladder neck reconstruction was only successful in 23% of patients. CONCLUSIONS: All patients can be rendered continent but many may achieve this successful outcome by other procedures following initial bladder neck reconstruction. When managing failed bladder neck reconstruction, the type of surgical repair chosen may need to address the need for enhanced bladder storage and the issue of potential bladder augmentation. The advances made in the treatment of the epispadiac urethra may now facilitate clean intermittent catheterization. Earlier recognition of the need for adjunctive storage procedures in addition to bladder neck reconstruction may facilitate the timing of providing enhanced continence, independence and self-esteem, and do so with fewer operative procedures. We speculate that the current complete urethral and bladder repair in newborns will add further to the storage functions of the native bladder tissues and improve the potential of achieving more effective bladder outlet control.

Bladder Exstrophy↗