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230
Fig. 13.4 On the left side is the nished Free-Graft NU.Neighboring skin is sutured to the free
edges around the circumference of the graft to create a natural indented umbilical appearance. On
the right is the long-term appearance of neo-umblilicus
Fig. 13.5 S-Flap
NU.Two 2cm horizontal
U-shaped aps are created
by a vertically oriented
S-shaped incision centered
on the intersection of the
midsagittal line and the
level of the iliac crest
E. Roth et al.
13 Common Adjuvant Surgeries inExstrophy/Epispadias Care
231
a b
Fig. 13.6 Finished S-Flap NU. (a) The U-aps are opposed to each other after the apex of each
ap is anchored to the rectus fascia. The SP tube is located between the two aps. The aps are
approximated to each other, leaving two horizontal incisions above and below the NU.Once the
NU reaches a desired depth, the horizontal incisions are closed with interrupted sutures. (b) Longterm appearance of the S-ap
Fig. 13.7 Rhomboid Flap
NU.The ap is shifted
laterally with its left border
running along the
midsagittal line. Incisions
are made along the green
line, preparing the ap for
mobilization while also
preserving blood supply. A
close-up photo showing the
incision that outlines the
rhomboid ap. There is a
malecot catheter exiting
the site as a
suprapubic tube
232
ab
Fig. 13.8 Rotated
Rhomboid ap. The
rhomboid ap (superior
and inferior borders are
marked by green lines) is
rotated in a counterclockwise manner (blue
arrow) about the SP tube,
resulting in the
subcutaneous surface of
the ap facing outwards,
the epidermis centrally
oriented towards the
SP tube
E. Roth et al.
Fig. 13.9 Finished Rhomboid NU.Intra-operative appearance. Long-term appearance
References
1. Barroso U, Jednak R, Barthold JS, Gonzalez R.A technique for constructing an umbilicus and
concealed catheterizable stoma. BJU Int. 2001;117:87.
2. Borenstein SH, etal. Effect of subspecialty training and volume on outcome after pediatric
inguinal hernia repair. J Pediatr Surg. 2005;40(1):75–80.
3. Braga LHP, Lorenzo AJ, Jrearz R, Bagli DJ, Salle JLP. Bilateral ureteral reimplantation at
primary bladder exstrophy closure. J Urol. 2010;183(6):2337–41. https://doi.org/10.1016/j.
juro.2010.02.032.
4. Canning DA, Gearhart JP, Peppas DS, Jeffs RD.The cephalotrigonal reimplant in bladder neck
reconstruction for patients with exstrophy or epispadias. J Urol. 1993;150(1):156–8. https://
doi.org/10.1016/s0022- 5347(17)35421- 6.
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5. Cervellione MR, Kyriazis I, Dickson AP.Construction of a natural looking inverted umbilicus
for bladder exstrophy. J Urol. 2008;180:1869–72.
6. Cohen SJ.The Cohen reimplantation technique. Birth Defects Orig Artic Ser. 1977;13:391–5.
7. Connolly JA, etal. Prevalence and repair of inguinal hernias in children with bladder exstrophy. J Urol. 1995;154(5):1900–1.
8. Cook AJ, Farhat WA, Cartwright LM, Khoury AE, Pippi Salle JL.Simplied Mons Plasty: a
new technique to improve cosmesis in females with the exstrophy-epispadias complex. J Urol.
2005;173:2117–20.
9. Dickson AP. The management of bladder exstrophy: the Manchester experience. J Pediatr
Surg. 2014;49(2):244–50. https://doi.org/10.1016/j.jpedsurg.2013.11.031.
10. Ein SH, Njere I, Ein A.Six thousand three hundredsixty-one pediatric inguinal hernias: a
35-year review. J Pediatr Surg. 2006;41(5):980–6.
11. Ellison JS, etal. Impact of concomitant hernia repair at the time of complete primary repair of
bladder exstrophy. J Pediatr Urol. 2016;12(4):211.e1–5.
12. Feyaerts A, Mure PY, Jules JA, Morel-Journel N, Mouriquand P.Umbilical reconstruction in
patient with exstrophy: the kangaroo pouch technique. J Urol. 2001;2026:165.
13. Garat JM, de la Peña E, Caffaratti J, Villavicencio H.Prevention of vesicoureteral reux at
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2004;36(2):211–2. https://doi.org/10.1023/b:urol.0000034675.34977.b4.
14. Grady RW, Mitchell ME.Complete primary repair of exstrophy. J Urol. 1999;162(4):1415–20.
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15. Hanna MK.Reconstruction of the umbilicus during functional closure of bladder exstrophy.
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18. Lavien G, etal. Impact of pelvic osteotomy on the incidence of inguinal hernias in classic bladder exstrophy. J Pediatr Surg. 2014;49(10):1496–9.
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233
Continent Anal Urinary Diversions
inBladder Exstrophy
JacquelineP.Morin andV.RamaJayanthi
Introduction
Despite the evolution of reconstructive procedures over the last several decades,
bladder exstrophy continues to present the pediatric urologist with notable challenges. The ultimate goals of exstrophy repair remain to achieve satisfactory cosmetic results, adequate sexual function and fertility, and socially acceptable urinary
continence with protection of renal function. The latter has improved over time with
the development of novel surgical techniques and high-volume centers, with continence rates boasted as high as 70%; however, contemporary analyses of outcomes
continue to demonstrate continence rates as low as 16.7% after primary repair [1, 2].
As there is no standard denition of “continence” in the literature, it is likely that a
smaller percentage of patients described as dry may be truly continent, i.e., voiding
volitionally with periods of dryness >3hours [2, 3]. In fact, one systematic analysis
of the literature found that only 68% of papers dene what it means to be “continent,” and most papers include those that require clean intermittent catheterization
(CIC) within their denition of continence [3]. Therefore, it is difcult to discern
the true rate of dry patients who void volitionally.
Most patients required multiple procedures, and many continue to suffer with
life-altering urinary leakage despite these efforts [1, 4, 5]. These patients undergo an
average of 2.3 surgeries, upwards of 8 operations for some [6]. It is also postulated
that multiple reconstructive procedures of the bladder and bladder neck may compromise bladder urodynamics from previously healthy and safe to low capacity and
noncompliant [7]. Continence issues have been found to have a negative impact on
self-esteem, mental health, and social interaction for these patients [8]. Furthermore,
bladder neck reconstruction has been associated with additional risks of bladder
14
J. P. Morin (*) · V. R. Jayanthi
Department of Pediatric Urology, Nationwide Children’s Hospital, Columbus, OH, USA
e-mail: rama.jayanthi@nationwidechildrens.org
© The Author(s), under exclusive license to Springer Nature
Switzerland AG 2025
A. R. Shukla, R. S. Joshi (eds.), Bladder Exstrophy and Epispadias,
https://doi.org/10.1007/978-3-031-91238-2_14
235
236
J. P. Morin and V. R. Jayanthi
outlet obstruction, urethral leakage, pyelonephritis, bladder stones, and bladder perforation [9, 10]. Children who have already undergone sphincteroplasty, bladder
augmentations, or additional reconstructive surgeries may not be ideal candidates
for further reconstruction [11]. As such, it may be prudent to seek alternative options
for continence in a manner that minimizes overall risks.
In patients who continue to suffer with urinary leakage despite extensive reconstruction, an option to achieve dryness is the continent anal urinary diversion
(CAUD), of which several surgical methods have been developed over the years.
Additionally, a CAUD may be an option for primary reconstruction in cases of bladder exstrophy as well, depending on the goals of the family. The objective of this
chapter is to explore the CAUD options and outcomes in patients with history of
bladder exstrophy who have failed primary continence reconstruction or who may
be candidates for a primary CAUD.
Ureterosigmoidostomy
The ureterosigmoidostomy (US) is the original method of CAUD, during which the
ureters are tunneled into the sigmoid colon, thus diverting urine into the rectum
[12]. If the patient has normal anal tone, they are likely to achieve dryness through
this continence structure. This technique was initially introduced in 1852 and was a
very popular method of urinary diversion for many years [13]. The US fell out of
favor in the 1950s with the development of the ileal conduit, as it proved to be associated with a variety of life-threatening conditions such as pyelonephritis, renal
function deterioration, electrolyte imbalances, and malignancy development [12,
13]. Despite this fact, the US does remain an option for the select patient.
There are many attractive aspects of the US.The procedure itself is relatively
straightforward with a short operative time [14]. The procedure offers a method of
bladder management that does not include a stoma (with its associated complications often obviating surgical revision), require external appliances, bags, or
indwelling catheters, and may be aesthetically preferred by patients and families
[14]. Continence rates in the US are excellent. Gobet etal. reported complete continence in 82% of patients by age 20 [10]. Stockle etal. reported daytime continence
rates of 97.4%, with complete continence rates as high as 92.3% sustained for
>10years with preserved renal function [15]. Patients report improved quality of
life (QOL), and many would select the procedure if given the opportunity again,
despite managing complications [10, 13].
Risks in the US can be common and serious. It should be noted that as the ureters
are implanted into an intact sigmoid, urinary storage pressures may be elevated,
which may result in urinary/fecal incontinence and retrograde reux with potential
upper urinary tract deterioration [11]. Some may require revision surgery to another
urinary diversion due to urinary stone formation, colorectal anastomotic stricture
formation, recurrent UTIs, pelvic organ prolapse, or malignancy development [10,
16]. The majority of patients (60%) will not demonstrate an increase in creatinine;
however, mild to moderate renal insufciency by glomerular ltration rate
14 Continent Anal Urinary Diversions inBladder Exstrophy
237
estimations only is reported in most patients, with some experiencing additional
renal deterioration that requires additional surgery to protect the upper tracts [10,
14, 15]. Pyelonephritis has been reported in 16% of patients [17]. Electrolyte aber-
rations may occur as a result of absorption of urinary components. Hyperchloremic
metabolic acidosis is the most common electrolyte abnormality and may lead to
hypokalemia and chronic diarrhea [14]. This is more common in patients with baseline renal insufciency [14]. Severe and untreated metabolic acidosis can lead to
osteomalacia [14]. Hyperammonemia due to rectal absorption of urinary ammonia
can occur and, in severe cases, lead to encephalopathy/neurotoxicity, which can be
life-threatening if not treated in a timely manner [18]. Urolithiasis, urinary/fecal
stulas, proctitis, bowel leaks, and hernias may occur but are rarer complications [14].
Arguably the most sinister risk is that of colonic adenocarcinoma. Patients with
US have a 100–550-fold increased risk of developing colonic adenocarcinoma as
compared to the general population unless performed prior to age 25, which portends a 7000-fold increased risk [19]. The exact pathogenesis of malignancy development is unclear, but several theories hypothesize this may be due to chronic
inammation and irritation from mixing fecal and urinary composites. In particular,
nitrosamines produced by fecal ora may be activated by the urine, leading to carcinogenesis [20, 21]. The latency period for adenocarcinoma development is an
average of 20–25years following US and is often asymptomatic until advanced [19].
Regardless, the US may be a viable rst option, with conversion to an alternate
diversion several years later. Care must be taken to resect the ureterocolonic anastomoses at the time of conversion, as adenocarcinoma development at the site of ureteral stumps has been documented following diversion revision [19, 22].
Interestingly, patients who have developed malignancies often request a new ureterosigmoid anastomosis as opposed to a different type of diversion, attesting to the
QOL that this rectal diversion may offer [23].
The Sigma-Rectum Pouch (Mainz Pouch II)
An alternative colonic diversion known as the sigma-rectum pouch (SRP), or Mainz
pouch II, was introduced in the early 1990s by Fisch and Hohenfellner [24]. This
procedure was primarily used to manage adults with bladder cancer but also was
applied to children with bladder exstrophy. The Mainz group had signicant experience with classic ureterosigmoidostomy but developed SRP in an effort to reduce
the incidence of high-pressure complications such as pyelonephritis, renal injury,
and incontinence. Rather than simply anastomosing the ureters to the sigmoid colon
as with the US, the SRP involves detubularizing the rectosigmoid colon to construct
a reservoir in which the ureters are tunneled in an anti-reuxing manner [24]. Antireuxing anastomoses are created to help prevent episodes of pyelonephritis and
upper tract damage. This diversion allows for a low-pressure, high-capacity reservoir to store urine. In theory, this procedure is superior to the US as it allows for
longer storage of urine, thereby reducing urinary frequency/urgency.
238
J. P. Morin and V. R. Jayanthi
Like the US, the SRP is a straightforward procedure of relatively short operative
duration. Continence rates for patients with SRP far exceed anything ever demonstrated with a traditional bladder neck repair. Pahernik etal. described 100% daytime continence and 92% nighttime control in 38 children, the majority of whom
had history of bladder exstrophy [25]. Hanna etal. described 61 patients who had
failed initial closure. Sixteen ultimately underwent an SRP, and all were continent
[26]. Hussain etal. wrote on 33 adults with exstrophy who underwent SRP, and all
were dry during the day, but 2 had some nighttime bedwetting [27].
As with the US, there are some risks/complications of the SRP.Complications
include ureterocolonic anastomotic strictures (4–7%), pyelonephritis despite the
anti-reuxing anastomoses (8%), and hydronephrosis [28]. Hyperchloremic metabolic acidosis can occur after SRP, with approximately 60% of patients requiring
medical management with alkalizing agents [28].
Certainly the biggest concern with SRP is the theoretical risk of colonic malignancy; however, to date, there have been few, if any, reported cases of tumors developing after this form of rectal diversion. This may simply be due to the fact that the
procedure has been in existence for only a few decades, but it will be important for
long-term studies to be published. Regardless, acknowledging long-term concerns
about malignancy, short-term data as above show the children managed with SRP
have markedly improved continence rates compared to traditional bladder neck
repairs.
The Cologne Pouch
The Cologne Pouch (CP) is another method of urinary diversion that again uses the
anal sphincter as the primary continence mechanism, as with the US and SRP; however, it aims to separate the evacuation of urine and feces. This is accomplished by
fashioning a U-shaped segment of sigmoid colon, with one end connected to the
rectum. The bladder plate is then attached to the proximal vertex of the “U” [29].
The ureters remain attached to the bladder plate, so no ureterocolonic anastomoses
are required. This technique eliminates the risk of ureterocolonic anastomotic strictures that can complicate the US and SRP.
Continence rates are excellent in this procedure as well (100%), with separate
urinary and fecal evacuation in 81.5% of patients [29]. This allows for reduced urinary and fecal urgency/frequency due to reduced mixing of the two excretory products. There is only a single series published on the CP [29]. In this cohort of patients,
only 7.4% complained of voiding frequency [29]. Similar to the US and SRP, metabolic acidosis may ensue, and patients may require bicarbonate supplementation
(55.6%) [29]. As there are no separate ureteral anastomoses performed, there is a
reduced risk of both VUR and no ureterocolonic stricture formation, and as such,
there may be a reduced incidence of UTIs or upper tract damage [29]. Perhaps due
to its relatively recent development as a surgical technique, no malignancies have
yet been reported in this series of patients who have undergone CP, with a mean
follow-up of 8.3years (range 1.9–17.4years) [29].
14 Continent Anal Urinary Diversions inBladder Exstrophy
239
A modication of this technique has been published where the bladder is augmented with small bowel and the augmented portion of the bladder is anastomosed
to the sigmoid colon via a small bowel chimney [30]. As with both this technique
and the CP, these methods exemplify the concept of a system whereby the urine is
diverted to a pouch separate from the sigmoid that functions as a reservoir, allowing
safe urinary storage while minimizing coalescence of feces and urine.
Conclusion
In conclusion, rectal diversion is a viable option for continent urinary diversion for
patients with bladder exstrophy either as a primary or a salvage procedure. These
procedures boast excellent continence rates and encouraging QOL measurements
but are not often at the forefront of exstrophy management. While this is likely due
to historical data warning of a variety of risks and complications, those negative
implications of a rectal diversion may be balanced with improved QOL and more
contemporary techniques and data. Indeed, continence outcomes far exceed what is
attainable through more traditional means and these patients often require far fewer
surgeries than an exstrophy patient who follows the traditional surgical pathways.
This fact suggests that it is worthwhile to explore rectal diversions as an option for
either primary or salvage surgical repair for patients, depending on the family’s
level of risk aversion, social situation, and overall QOL goals. Some families may
elect for this approach due to greater continence success rates, the ability to avoid
intermittent catheterization, and the more “normal” day-to-day lifestyle.
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J. P. Morin and V. R. Jayanthi