Pediatric Urology Secondary Reclosure in Classic Bladder Exstrophy: Challenges and Outcomes Brian M. Inouye, Heather N. Di Carlo, Ezekiel E. Young, Ali Tourchi, and John P. Gearhart OBJECTIVE
METHODS
RESULTS
CONCLUSION
To analyze the outcomes of failed classic bladder exstrophy (CBE) reclosure with and without pelvic osteotomy. Each failed CBE closure decreases the chance of eventual continence. To minimize secondary failures, most institutions use pelvic osteotomy with reclosure. Reclosure with and without osteotomy can still fail. An institutional database of 1210 exstrophy-epispadias complex patients was reviewed for CBE patients who had 2 prior failed closures with the third closure at the authors’ institution. Patient demographics, closure history, diastasis distance, bladder capacity, and outcomes were examined by chi-square tests comparing osteotomy status with first reclosure. Of 848 CBE patients, 17 met inclusion criteria: 12 with osteotomy at reclosure (group 1) and 5 without (group 2). Median time between initial closure and reclosure in the 2 groups were 6.5 months (range, 0-42 months) and 3 months (range, 0-59 months), respectively. There was no significant difference in the rate of attaining sufficient bladder capacity for bladder neck reconstruction (BNR; 100 cc) between groups 1 and 2 (42% vs 40%; P ¼ .490). Within group 1, patients receiving proper immobilization with external fixation (n ¼ 5) demonstrated a significantly greater rate of attaining sufficient bladder capacity for BNR compared with patients who did not (80% vs14%; P ¼ .023). There were no differences in the rates of attaining dryness per urethra. CBE outcomes worsen with each successive failed closure. Reclosure should be performed with osteotomy and proper immobilization to maximize the chance of sufficient capacity for BNR or augmentation cystoplasty. UROLOGY 85: 1179e1182, 2015. 2015 Elsevier Inc.
D
espite the recent advances in the management of the exstrophy-epispadias complex, failed initial closure is still extremely challenging, especially outside of centers of excellence. Primary failure jeopardizes the bladder’s ability to grow and the patient’s chance of gaining continence.1,2 Patients have a decreased chance of achieving sufficient bladder capacity for bladder neck reconstruction (BNR) and eventual continence through their urethra after reclosure.3 Pelvic osteotomy is recommended in all exstrophy reclosures.2 Even if osteotomy was performed with the primary closure, a repeat pelvic osteotomy has been shown to be safe and efficacious even though it further disrupts the pelvic rim.4,5 Performing this osteotomy at the time of reclosure allows for greater pelvic mobility for better pelvic dissection and tissue reapproximation. Reclosure with and without osteotomy can still fail, Financial Disclosure: The authors declare that they have no relevant financial interests. From the Division of Pediatric Urology, Department of Urology, James Buchanan Brady Urological Institute, Charlotte Bloomberg Children’s Hospital, The Johns Hopkins University School of Medicine, Baltimore, MD Address correspondence to: John P. Gearhart, M.D., Division of Pediatric Urology, Department of Urology, James Buchanan Brady Urological Institute, Charlotte Bloomberg Children’s Hospital, The Johns Hopkins University School of Medicine, 1800 Orleans St., Suite 7204, Baltimore, MD 21287. E-mail: [email protected] Submitted: January 19, 2015, accepted (with revisions): January 27, 2015
ª 2015 Elsevier Inc. All Rights Reserved
necessitating additional surgeries to prevent further alternations in bladder growth and achieving continence. There is a paucity of reports describing these patients with 2 failed exstrophy closures and their outcomes. Herein, the authors analyze their experience with the management of failed classic bladder exstrophy (CBE) reclosures with and without osteotomy to determine the impact of these failed closures on subsequent continence.
METHODS A prospectively maintained institutional database of 1210 exstrophy-epispadias complex patients was screened for CBE patients who had 2 prior failed closures with the third closure at the authors’ institution. After selection, patients were divided into 2 groups by their osteotomy status at their first reclosure (second closure overall): initial reclosure with osteotomy (group 1) and initial reclosure without osteotomy (group 2). Patient demographics, closure history, pelvic osteotomy history, preoperative pubic diastasis measurement, type of most recent osteotomy and pelvic immobilization, and length of followup were examined. Specific attention was paid to closure failure (defined as dehiscence, prolapse, bladder outlet obstruction, and complex vesicocutaneous fistula), bladder capacity, and outcomes after second reclosure at the authors’ institution (third closure overall). Descriptive variables are presented as median (range). Variables were analyzed using the nonparametric t test, the http://dx.doi.org/10.1016/j.urology.2015.01.029 0090-4295/15
1179
Table 1. Patient demographics and closure history Initial Reclosure þ Osteotomy Initial Reclosure Osteotomy P Value Age at primary closure (d) Age at initial reclosure (mo) Time between initial closure and reclosure (mo) Type of osteotomy at initial reclosure, n (%) Anterior innominate and vertical iliac Anterior innominate Posterior innominate Pelvic ramotomy Type of pelvic immobilization at initial reclosure, n (%) Buck traction with external fixation Bryant traction Spica cast None
2 (0-126) 8 (0-42) 6.5 (0-42) 0 8 3 1
(0) (67) (25) (8)
5 3 4 0
(42) (25) (33) (0)
3 (0-19) 3 (0-11) 3 (0-11)
.879 .799 .879
N/A
N/A
(0) (40) (0) (60)
.107
0 2 0 3
Table 2. Second reclosure specifics Initial Reclosure þ Osteotomy Age at second reclosure (mo) 22 (8-62) Presecondary reclosure diastasis (cm) 5 (3.5-5.2) Type of osteotomy at initial reclosure, n (%) Anterior innominate and vertical iliac 10 (83) Anterior innominate 2 (17) Posterior innominate 0 (0) Pelvic ramotomy 0 (0) Type of pelvic immobilization at initial reclosure, n (%) Modified Buck traction 12 (100) Bryant traction 0 (0) Spica cast 0 (0) None 0 (0) Current continent status, n (%) Completely continent 1 (8) Dry continent intervals 0 (0) Dry with urethral catheterization 1 (8) Dry with continent stoma 6 (50) Incontinent after continence procedure 0 (0) Waiting for continence procedure 4 (33) Length of follow-up (y) 5.5 (1-17)
Initial Reclosure Osteotomy
P Value
23 (13-43) 6 (5.5-8)
.799 .009
2 3 0 0
(40) (60) (0) (0)
.080
4 1 0 0
(80) (20) (0) (0)
.107
1 2 0 2 0 0 5
(20) (40) (0) (40) (0) (0) (1-22)
.188*
1.000
* Excludes patients who are waiting for continence procedure.
chi-square test, and the univariate logistic regression with a significant P value cut-off of .05. All statistical analysis was performed with Microsoft Excel 2010 (Microsoft Corp, Redmond, WA) and SPSS 16 (SPSS Inc, Chicago, IL). Pelvic x-rays were taken either the day before or on the day of osteotomy to measure the preoperative pubic diastasis. After closure, patients undergo annual gravity cystograms, the results of which determine further continence management. A bladder capacity >100 cc was deemed sufficient for BNR.
RESULTS Of 848 CBE patients, 17 met inclusion criteria: 12 patients with osteotomy at first reclosure, group 1, and 5 patients without osteotomy at initial reclosure, group 2. Table 1 describes the demographics and closure histories of the 2 groups. There was no difference between the age of the 2 groups at primary closure or initial reclosure. Furthermore, the median time between primary closure and initial reclosure was also nonsignificant (6.5 vs 3 months; P ¼ .879). No patients in group 1 had a 1180
combined anterior innominate and vertical iliac osteotomy with their initial reclosure. Instead, 8 (67%) had anterior innominate osteotomy, 3 (25%) had posterior innominate osteotomy, and 1 (8%) had a pelvic ramotomy. Five of these patients (42%) had pelvic immobilization by external fixation and Buck traction. Three (25%) other patients were immobilized in Modified Bryant traction. There was no difference between the ages of patients undergoing their second reclosure (Table 2). Group 1 patients had a significantly smaller median pubic diastasis than group 2 patients before their second reclosure (5 vs 6 cm; P ¼ .009). There was no difference in the type of osteotomy or immobilization performed on these 2 groups. After second reclosure, pelvic osteotomy, and immobilization, there was no significant difference in the rate of attaining sufficient bladder capacity for BNR between the 2 groups (42% vs 40%; P ¼ .490). Interestingly, within group 1, patients receiving external fixation and Buck traction at initial reclosure demonstrated UROLOGY 85 (5), 2015
a significantly greater rate of attaining sufficient bladder capacity for BNR compared with patients who did not (80% vs14%; P ¼ .023). Similar to the bladder capacity outcomes, there was no difference in the rate of attaining dryness per urethra between the 2 groups. Even when comparing the rate of achieving some urethral continence (completely continent, dry intervals, and continent with clean intermittent catheterization), there was still no difference between the 2 groups (17% vs 60%; P ¼ .117). Four patients in group 1 were still awaiting a continence procedure at most recent follow-up. There was no difference in the groups’ length of follow-up.
COMMENT Primary exstrophy closure failure markedly decreases the patient’s chance for achieving sufficient bladder capacity for BNR and eventual continence.1,2 If there is a failed primary closure, reclosure must be performed with osteotomy to increase its chance of succeeding.2,6 Still, there is chance for these reclosures to fail. Because of lack of studies examining the outcomes of secondary reclosures in CBE patients, the authors examined the outcomes of these patients at their exstrophy center. Furthermore, to help guide future reclosure management, patients were divided into 2 groups based on whether they received pelvic osteotomy at initial reclosure to determine if specific techniques improve their eventual outcomes. Combined anterior innominate and vertical iliac osteotomy helps support a good successful primary closure or reclosure.7,8 It is therefore interesting to note that none of the failed reclosure patients underwent this type of pelvic osteotomy. Many institutions, even centers of excellence in the treatment of CBE, do not have pediatric orthopedic surgeons on the exstrophy team that appreciate the nuances of the combined anterior innominate and vertical iliac osteotomy with proper immobilization and therefore have difficulty in managing these patients. Unfortunately, without the number of patients who underwent osteotomy and successful reclosure, one cannot compare the rates of success and fully determine the importance of osteotomy at initial reclosure in this study population. Still, patients who received an osteotomy with initial reclosure had a significantly narrower pubic diastasis at second reclosure compared with patients who did not receive osteotomy with their first reclosure. Prevention of a wide diastasis is always important because extreme pubic diastases in CBE patients require staged osteotomy that may increase the patient’s length of stay and the ultimate hospital charges incurred by the family.9,10 The percentage of patients in the 2 groups achieving sufficient bladder capacity for BNR is similar to the 50% chance previously cited in patients having failed 2 exstrophy closures.11 Without knowing the outcomes of patients who had successful initial reclosure with and without osteotomy, it is impossible to determine the UROLOGY 85 (5), 2015
impact of pelvic osteotomy on eventual bladder capacity and continence. However, in patients who underwent osteotomy, patients placed in Buck traction with external pelvic fixation had a significantly greater chance of achieving a bladder capacity sufficient for BNR. This finding demonstrates the importance of proper immobilization with osteotomy. Even though the initial reclosure failed, most likely secondary to it not being a combined anterior innominate and vertical iliac osteotomy, the eventual bladder growth demonstrates the importance of placing a child in adequate traction and immobilization after reclosure. Of course, immobilization must be accompanied by adequate pain management and sedation.12 Even with an increase in bladder capacity, patients with osteotomy and proper immobilization at initial reclosure or any group in this study demonstrated an increased chance of complete continence via their urethra. Again, these continence rates are similar to previous findings that
METHODS
RESULTS
CONCLUSION
To analyze the outcomes of failed classic bladder exstrophy (CBE) reclosure with and without pelvic osteotomy. Each failed CBE closure decreases the chance of eventual continence. To minimize secondary failures, most institutions use pelvic osteotomy with reclosure. Reclosure with and without osteotomy can still fail. An institutional database of 1210 exstrophy-epispadias complex patients was reviewed for CBE patients who had 2 prior failed closures with the third closure at the authors’ institution. Patient demographics, closure history, diastasis distance, bladder capacity, and outcomes were examined by chi-square tests comparing osteotomy status with first reclosure. Of 848 CBE patients, 17 met inclusion criteria: 12 with osteotomy at reclosure (group 1) and 5 without (group 2). Median time between initial closure and reclosure in the 2 groups were 6.5 months (range, 0-42 months) and 3 months (range, 0-59 months), respectively. There was no significant difference in the rate of attaining sufficient bladder capacity for bladder neck reconstruction (BNR; 100 cc) between groups 1 and 2 (42% vs 40%; P ¼ .490). Within group 1, patients receiving proper immobilization with external fixation (n ¼ 5) demonstrated a significantly greater rate of attaining sufficient bladder capacity for BNR compared with patients who did not (80% vs14%; P ¼ .023). There were no differences in the rates of attaining dryness per urethra. CBE outcomes worsen with each successive failed closure. Reclosure should be performed with osteotomy and proper immobilization to maximize the chance of sufficient capacity for BNR or augmentation cystoplasty. UROLOGY 85: 1179e1182, 2015. 2015 Elsevier Inc.
D
espite the recent advances in the management of the exstrophy-epispadias complex, failed initial closure is still extremely challenging, especially outside of centers of excellence. Primary failure jeopardizes the bladder’s ability to grow and the patient’s chance of gaining continence.1,2 Patients have a decreased chance of achieving sufficient bladder capacity for bladder neck reconstruction (BNR) and eventual continence through their urethra after reclosure.3 Pelvic osteotomy is recommended in all exstrophy reclosures.2 Even if osteotomy was performed with the primary closure, a repeat pelvic osteotomy has been shown to be safe and efficacious even though it further disrupts the pelvic rim.4,5 Performing this osteotomy at the time of reclosure allows for greater pelvic mobility for better pelvic dissection and tissue reapproximation. Reclosure with and without osteotomy can still fail, Financial Disclosure: The authors declare that they have no relevant financial interests. From the Division of Pediatric Urology, Department of Urology, James Buchanan Brady Urological Institute, Charlotte Bloomberg Children’s Hospital, The Johns Hopkins University School of Medicine, Baltimore, MD Address correspondence to: John P. Gearhart, M.D., Division of Pediatric Urology, Department of Urology, James Buchanan Brady Urological Institute, Charlotte Bloomberg Children’s Hospital, The Johns Hopkins University School of Medicine, 1800 Orleans St., Suite 7204, Baltimore, MD 21287. E-mail: [email protected] Submitted: January 19, 2015, accepted (with revisions): January 27, 2015
ª 2015 Elsevier Inc. All Rights Reserved
necessitating additional surgeries to prevent further alternations in bladder growth and achieving continence. There is a paucity of reports describing these patients with 2 failed exstrophy closures and their outcomes. Herein, the authors analyze their experience with the management of failed classic bladder exstrophy (CBE) reclosures with and without osteotomy to determine the impact of these failed closures on subsequent continence.
METHODS A prospectively maintained institutional database of 1210 exstrophy-epispadias complex patients was screened for CBE patients who had 2 prior failed closures with the third closure at the authors’ institution. After selection, patients were divided into 2 groups by their osteotomy status at their first reclosure (second closure overall): initial reclosure with osteotomy (group 1) and initial reclosure without osteotomy (group 2). Patient demographics, closure history, pelvic osteotomy history, preoperative pubic diastasis measurement, type of most recent osteotomy and pelvic immobilization, and length of followup were examined. Specific attention was paid to closure failure (defined as dehiscence, prolapse, bladder outlet obstruction, and complex vesicocutaneous fistula), bladder capacity, and outcomes after second reclosure at the authors’ institution (third closure overall). Descriptive variables are presented as median (range). Variables were analyzed using the nonparametric t test, the http://dx.doi.org/10.1016/j.urology.2015.01.029 0090-4295/15
1179
Table 1. Patient demographics and closure history Initial Reclosure þ Osteotomy Initial Reclosure Osteotomy P Value Age at primary closure (d) Age at initial reclosure (mo) Time between initial closure and reclosure (mo) Type of osteotomy at initial reclosure, n (%) Anterior innominate and vertical iliac Anterior innominate Posterior innominate Pelvic ramotomy Type of pelvic immobilization at initial reclosure, n (%) Buck traction with external fixation Bryant traction Spica cast None
2 (0-126) 8 (0-42) 6.5 (0-42) 0 8 3 1
(0) (67) (25) (8)
5 3 4 0
(42) (25) (33) (0)
3 (0-19) 3 (0-11) 3 (0-11)
.879 .799 .879
N/A
N/A
(0) (40) (0) (60)
.107
0 2 0 3
Table 2. Second reclosure specifics Initial Reclosure þ Osteotomy Age at second reclosure (mo) 22 (8-62) Presecondary reclosure diastasis (cm) 5 (3.5-5.2) Type of osteotomy at initial reclosure, n (%) Anterior innominate and vertical iliac 10 (83) Anterior innominate 2 (17) Posterior innominate 0 (0) Pelvic ramotomy 0 (0) Type of pelvic immobilization at initial reclosure, n (%) Modified Buck traction 12 (100) Bryant traction 0 (0) Spica cast 0 (0) None 0 (0) Current continent status, n (%) Completely continent 1 (8) Dry continent intervals 0 (0) Dry with urethral catheterization 1 (8) Dry with continent stoma 6 (50) Incontinent after continence procedure 0 (0) Waiting for continence procedure 4 (33) Length of follow-up (y) 5.5 (1-17)
Initial Reclosure Osteotomy
P Value
23 (13-43) 6 (5.5-8)
.799 .009
2 3 0 0
(40) (60) (0) (0)
.080
4 1 0 0
(80) (20) (0) (0)
.107
1 2 0 2 0 0 5
(20) (40) (0) (40) (0) (0) (1-22)
.188*
1.000
* Excludes patients who are waiting for continence procedure.
chi-square test, and the univariate logistic regression with a significant P value cut-off of .05. All statistical analysis was performed with Microsoft Excel 2010 (Microsoft Corp, Redmond, WA) and SPSS 16 (SPSS Inc, Chicago, IL). Pelvic x-rays were taken either the day before or on the day of osteotomy to measure the preoperative pubic diastasis. After closure, patients undergo annual gravity cystograms, the results of which determine further continence management. A bladder capacity >100 cc was deemed sufficient for BNR.
RESULTS Of 848 CBE patients, 17 met inclusion criteria: 12 patients with osteotomy at first reclosure, group 1, and 5 patients without osteotomy at initial reclosure, group 2. Table 1 describes the demographics and closure histories of the 2 groups. There was no difference between the age of the 2 groups at primary closure or initial reclosure. Furthermore, the median time between primary closure and initial reclosure was also nonsignificant (6.5 vs 3 months; P ¼ .879). No patients in group 1 had a 1180
combined anterior innominate and vertical iliac osteotomy with their initial reclosure. Instead, 8 (67%) had anterior innominate osteotomy, 3 (25%) had posterior innominate osteotomy, and 1 (8%) had a pelvic ramotomy. Five of these patients (42%) had pelvic immobilization by external fixation and Buck traction. Three (25%) other patients were immobilized in Modified Bryant traction. There was no difference between the ages of patients undergoing their second reclosure (Table 2). Group 1 patients had a significantly smaller median pubic diastasis than group 2 patients before their second reclosure (5 vs 6 cm; P ¼ .009). There was no difference in the type of osteotomy or immobilization performed on these 2 groups. After second reclosure, pelvic osteotomy, and immobilization, there was no significant difference in the rate of attaining sufficient bladder capacity for BNR between the 2 groups (42% vs 40%; P ¼ .490). Interestingly, within group 1, patients receiving external fixation and Buck traction at initial reclosure demonstrated UROLOGY 85 (5), 2015
a significantly greater rate of attaining sufficient bladder capacity for BNR compared with patients who did not (80% vs14%; P ¼ .023). Similar to the bladder capacity outcomes, there was no difference in the rate of attaining dryness per urethra between the 2 groups. Even when comparing the rate of achieving some urethral continence (completely continent, dry intervals, and continent with clean intermittent catheterization), there was still no difference between the 2 groups (17% vs 60%; P ¼ .117). Four patients in group 1 were still awaiting a continence procedure at most recent follow-up. There was no difference in the groups’ length of follow-up.
COMMENT Primary exstrophy closure failure markedly decreases the patient’s chance for achieving sufficient bladder capacity for BNR and eventual continence.1,2 If there is a failed primary closure, reclosure must be performed with osteotomy to increase its chance of succeeding.2,6 Still, there is chance for these reclosures to fail. Because of lack of studies examining the outcomes of secondary reclosures in CBE patients, the authors examined the outcomes of these patients at their exstrophy center. Furthermore, to help guide future reclosure management, patients were divided into 2 groups based on whether they received pelvic osteotomy at initial reclosure to determine if specific techniques improve their eventual outcomes. Combined anterior innominate and vertical iliac osteotomy helps support a good successful primary closure or reclosure.7,8 It is therefore interesting to note that none of the failed reclosure patients underwent this type of pelvic osteotomy. Many institutions, even centers of excellence in the treatment of CBE, do not have pediatric orthopedic surgeons on the exstrophy team that appreciate the nuances of the combined anterior innominate and vertical iliac osteotomy with proper immobilization and therefore have difficulty in managing these patients. Unfortunately, without the number of patients who underwent osteotomy and successful reclosure, one cannot compare the rates of success and fully determine the importance of osteotomy at initial reclosure in this study population. Still, patients who received an osteotomy with initial reclosure had a significantly narrower pubic diastasis at second reclosure compared with patients who did not receive osteotomy with their first reclosure. Prevention of a wide diastasis is always important because extreme pubic diastases in CBE patients require staged osteotomy that may increase the patient’s length of stay and the ultimate hospital charges incurred by the family.9,10 The percentage of patients in the 2 groups achieving sufficient bladder capacity for BNR is similar to the 50% chance previously cited in patients having failed 2 exstrophy closures.11 Without knowing the outcomes of patients who had successful initial reclosure with and without osteotomy, it is impossible to determine the UROLOGY 85 (5), 2015
impact of pelvic osteotomy on eventual bladder capacity and continence. However, in patients who underwent osteotomy, patients placed in Buck traction with external pelvic fixation had a significantly greater chance of achieving a bladder capacity sufficient for BNR. This finding demonstrates the importance of proper immobilization with osteotomy. Even though the initial reclosure failed, most likely secondary to it not being a combined anterior innominate and vertical iliac osteotomy, the eventual bladder growth demonstrates the importance of placing a child in adequate traction and immobilization after reclosure. Of course, immobilization must be accompanied by adequate pain management and sedation.12 Even with an increase in bladder capacity, patients with osteotomy and proper immobilization at initial reclosure or any group in this study demonstrated an increased chance of complete continence via their urethra. Again, these continence rates are similar to previous findings that
