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28 Gynecology inBladder Exstrophy
437
have suggested that CBE patient risk of prolapse is independent of parity [35].
Minimal data currently exist regarding the gynecologic outcomes of CBE patients
into menopause and beyond. It is likely that evolving research will also help to
clarify concerns in this patient population.
Multiple surgical techniques have been proposed to treat pelvic organ prolapse in
CBE, though no one approach has been determined to be the standard of care [35].
For patients who desire future childbearing, uterine preservation procedures have
been proposed, such as the Manchester-Fothergill and Elevate Posterior techniques,
though little literature exists on the outcomes of these repairs after pregnancy is
achieved or the pregnancy outcomes themselves [35, 40].
Many nonsurgical techniques, such as pessaries, have been suggested. Given differences in bony and soft tissue structure, many commonly used pessaries may not
easily stay in place in patients with CBE. Finding the appropriate pessary may
require a clinician well-versed in the many pessary options available. The role of a
general gynecologist, urogynecologist, or urologist specialized in female pelvic
medicine and reconstructive surgery may provide a natural transition into adulthood
for the pediatric bladder exstrophy patient and allow for early discussion of multiple
options available for management of pelvic organ prolapse in the context of the
patient’s life stage and desire for future pregnancy and/or uterine conservation.
Fertility
As more patients with CBE desire to become parents, increasing research has
focused on fertility and pregnancy. Still, data remains limited. Because ovarian
development is not affected, hormonal development is typical in patients with CBE,
as noted above. Infertility, dened as failure to achieve pregnancy in 12months of
unprotected intercourse in women younger than 35years or 6months if older than
35years, affects up to 15% of couples in the general population [41]. A series of six
patients desiring pregnancy reported conception without assisted reproductive technology in 5/6 patients [42]. Another series reported 100% of women with CBE who
desired pregnancy were able to achieve pregnancy spontaneously; however, this
series followed only two women, one who conceived twice and the other once [43].
A British series reported 28 women with BE attempting pregnancy, of which 19
women (66%) were able to conceive, although only 4 (21%) conceived spontaneously within 1 year. Tubal infertility was noted as a contributory factor in most
cases, consistent with the multiple previous surgeries and likelihood of subsequent
adhesive disease that may affect fallopian tube function. Only 5 (26%) sought
assisted reproductive technology, of which 4 underwent IVF and only one was successful, and 1 underwent ovulation induction. As this series was performed in the
UK, they noted that the low uptake of assisted reproductive technology and success
rate of IVF (25%) may not be consistent with practices of patients with tubal factor
infertility in other parts of the world [10].
438
L. Yu et al.
Contraception andRoutine Gynecologic Care
Patients should be counseled that, in the absence of specic anatomic considerations that would prevent pregnancy, people with CBE can become pregnant and
that contraception should be used if a patient is having penile-vaginal sex and pregnancy is not desired. Unplanned pregnancy has been reported in a patient with bladder exstrophy who thought conception could not occur due to the anomaly [44]. In
addition, despite higher rates of infertility, many people with CBE who become
pregnant will do so spontaneously [10, 45]. Finally, there is data that young patients
with medically complex conditions, including vaginoplasty and pelvic reconstruction, seek additional guidance regarding sexual and reproductive health beyond
what is typically provided in their care, and over half expressed anxiety regarding
sexual relationships [30, 46]. Obstetrician/gynecologists knowledgeable about the
patients’ prior medical and surgical history may help facilitate these questions and
concerns.
CBE patients should be counseled regarding contraception with similar frequency and considerations to patients without CBE.Considerations when choosing
a form of hormonal contraception include renal dysfunction, hypertension, uterine
anomalies, and degree of pelvic organ prolapse. Patients should undergo surveillance for cervical cancer per routine guidelines. Of note, cervical cancer screening
in a patient with didelphys uterus will require a swab of each cervix, as cervical
dysplasia may present differently, and a Pap examination and/or HPV testing of one
cervix may be insufcient to identify and appropriately triage cervical cancer risk
[47]. HPV vaccination should be recommended in concordance with routine recommendations. Additional routine gynecologic care, such as STI screening and evaluation of vaginal discharge, should be provided to patients by a provider who is
familiar with the patients’ medical and surgical history and with recommendations
for gynecologic evaluation. False positive urine pregnancy testing in the setting of
augmentation cystoplasty has been reported in both male and female patients; if
urine testing does not support clinical ndings, conrmation with serum beta-hCG
levels should be sought [48].
Obstetric Considerations
Regarding the pregnancy itself, many case series have reported successful pregnancy in women with CBE.Specic concerns for CBE patients include the increased
risks of pelvic organ prolapse, difculty with bladder emptying (both via catheterizable channel and urethra), increased constipation, and risks to renal function. A
series of patients with seven total pregnancies reported uterine prolapse in 7, acute
pyelonephritis in 1, prolapsed ileal conduit in 1, and transient urinary incontinence
in 1 [42]. In patients with chronic hypertension, additional maternal risks include
pulmonary edema, myocardial infarction, preeclampsia, placental abruption, cesarean delivery, postpartum hemorrhage, and gestational diabetes, while fetal risks
28 Gynecology inBladder Exstrophy
439
include increased fetal growth restriction, preterm birth, perinatal mortality, and
congenital anomalies [49].
Mullerian anomalies, when present, are associated with known fertility and
obstetric risks.
Having a Mullerian anomaly increases risks of preterm birth, primary cesarean
delivery, fetal malpresentation, and premature rupture of membranes [50–52].
Patients with cloacal exstrophy have unique challenges, including bowel management, that may be in addition to the challenges faced by CBE patients in pregnancy
[8]. A history of multiple prior abdominal surgeries for most patients with CBE of
childbearing age adds additional challenge when planning for mode of delivery. Wu
etal. reported a case of pregnancy in unrepaired CBE; these patients, though rare,
pose even more unique perinatal management challenges [53].
A preconception visit with a maternal-fetal medicine physician is recommended
for planning both for the pregnancy and medical optimization of underlying health
conditions prior to conception, and use of contraception until pregnancy is desired
to minimize risk of unplanned pregnancy. During pregnancy, close surveillance
with a maternal-fetal medicine specialist, a urologist, and a nephrologist are helpful
to optimize maternal health. Fetal development can be closely monitored via serial
ultrasounds throughout the pregnancy. Antenatal consultation with an anesthesiologist may also be recommended. The availability of spinal anesthesia may be limited
due to comorbidities involving the spine.
Techniques for cesarean delivery in patients with bladder reconstructions have
highlighted the proximity of the uterus to the reconstructed genitourinary tract [54]
(Fig.28.1). Historically, patients with CBE were recommended to deliver via scheduled cesarean delivery at a location and time when urologic specialists were available for participation. Clemetson reviewed 65 pregnancies in 46 women from 1924
Fig. 28.1 Schematic of a
pregnant patient with an
augmentation cystoplasty
and catheterizable
channel [54]
440
L. Yu et al.
to 1958, noting increased incidences of premature labor, malpresentation (suspected
to be due to poor anterior support of the pelvic oor), obstruction, or vaginal stenosis precluding vaginal delivery. They also noted a 50% incidence of subsequent
pelvic organ prolapse [55]. A series of 40 women reported that 11 patients had 14
pregnancies; those with prior urinary diversion were delivered vaginally to minimize the risk of additional surgery, while those with functional bladder closures
received cesarean sections to eliminate stress on the pelvic oor or risk of injury to
the bladder neck [42]. A series of 57 pregnancies in BE reported 3 sets of twins in
19 patients, with a total of 34 live births, 21 miscarriages, 1 termination, and 4 stillbirths or neonatal deaths. 42% of these patients experienced worsening prolapse
symptoms, often managed by bedrest and ring pessary. All live births were delivered
by cesarean, including 2 planned vaginal deliveries that resulted in cesarean section;
3 cesareans were performed earlier than scheduled due to premature labor or fetal
distress [10]. Dy etal. reported a series of 12 women with 22 pregnancies, resulting
in 14 live births, with 100% planned cesarean deliveries using a paramedian incision and the same surgical team. Of this group, 9 had undergone bladder augmentation, 11 required catheterization for bladder emptying, and 5 had catheterizable
stomas (2 Hemi-Kock pouches, 3 Mitrofanoffs, one of which abandoned the tract
due to recurrent stones). Six required urethral catheterization, and one woman
voided through the urethra with an articial urinary sphincter. Mullerian anatomy
was known in nine of the 12 women, with 6 having a bicornuate uterus, one with a
unicornuate uterus, and two women with typical uterine anatomy. Of the seven
women with Mullerian anomaly, three had documented spontaneous abortions or
termination of pregnancy, and four had live births. All patients in this group had
umbilical or right lower quadrant stomas, and a vertical paramedian skin incision
was used to avoid the augmented bladder and catheterizable channel, with a classical uterine incision then used for hysterotomy to avoid the reconstructed urinary
tract [56] (Fig.28.2).
Given the need for coordination and the risks of emergent cesarean delivery after
a failed attempt at vaginal delivery, many centers continue to recommend a scheduled cesarean delivery. Emphasis has been placed on the importance of a surgical
plan and an experienced, well-resourced team. However, Deans etal. reported the
case of a singleton pregnancy delivered via emergency cesarean at 30weeks with
difculty delivering the fetus due to adhesions from bladder reconstruction, resulting in neonatal demise despite an experienced surgical and neonatal team [10].
Additionally, cesarean delivery has its own risks, with increased risk in the CBE
patient, including altered anatomy and post-surgical adhesions from prior surgeries.
Vaginal delivery has been described; in one case, Duhrssen (cervical) incisions were
made to facilitate a successful late preterm (36 4/7weeks) delivery in a patient with
CBE and uterine prolapse at the time of delivery, a technique that has been used on
other laboring patients with uterine prolapse [57]. At present, delivery is typically
28 Gynecology inBladder Exstrophy
Fig. 28.2 A suggested
surgical approach to
Cesarean delivery. A
paramedian skin incision
contralateral to the
catheterizable stoma is
used to access the uterus,
which is then entered using
a classical uterine incision.
Patients in this study had
umbilical or right lower
quadrant (RLQ)
stomas [56]
441
recommended at early term unless an alternative indication for earlier delivery is
present, ideally in a scheduled fashion with a urologist familiar with the patient’s
anatomy present for consultation as needed.
Delivery planning conversations, including both mode and time of delivery,
should be initiated early in pregnancy and should involve shared decision-making
with patients, urologists, and maternal-fetal medicine specialists. Dy et al. have
argued that three components are necessary to optimize pregnancy outcomes in
patients with CBE: (1) a smooth transition of care from a pediatric to adult multidisciplinary team, (2) an understanding of reconstructed urological anatomy, and (3) a
planned delivery surgical approach [56]. Multidisciplinary groups have made recommendations on additional pregnancy management in the CBE patient throughout
the patient’s pregnancy and postpartum course, summarized here in Table28.1.
Additional studies will help to clarify recommendations for specic patient
situations.
442
L. Yu et al.
Table 28.1
Preconception 1. Establish routine gynecologic care (Pap smear, menstruation management,
First trimester
Second/third
trimester
Obstetric care recommendations in patients with bladder exstrophy
contraception).
2. Emphasize that pregnancies should be planned. Infertility workup should
be undertaken if no spontaneous conception after 6–12months for patients
<35years of age and earlier if >35years of age. Single embryo transfer
only if invitro fertilization is required.
3. Evaluate renal function, urinary bacterial prole, and infection history.
4. Discuss commitment to frequent surveillance schedule.
5. Discuss practical challenges of pregnancy with EEC, planned cesarean
delivery at term.
6. Access information including operative notes about reconstructed anatomy,
location, and path of catheterization channels.
1. Early ultrasound conrmation of gestation, identication of multiple
pregnancies, and ensuring patients are well counseled on pregnancy
options.
2. Shared prenatal care between MFM and urology teams, ensuring obstetric
care and delivery planning for a location with access to surgical and
urologic services and comprehensive neonatal services.
3. Baseline urine specimen and culture, compare pathogen type and burden to
prior positive cultures. Baseline labs to include creatinine and glomerular
ltration rate. Consider renal ultrasound if not recently performed.
4. Treat positive urine cultures if new pathogens, symptomatic, and/or
increased colony count compared to baseline. Asymptomatic bacteriuria is
common and, in the context of a reconstructive bladder, should not be
treated. Persistent urinary tract infection should prompt investigation for
urological complications such as obstruction or stones. Occasionally
prophylactic antibiotics may be needed.
1. Monitor for signs and symptoms of lower and upper urinary tract
obstruction—image upper tracts if needed.
2. Catheterization difculties arise due to malpositioning of conduits or
channels, thus there may be a need to modify catheterization angles,
positioning, change catheter size, or type. Some women may prefer to have
an indwelling catheter either on free drainage or with a “ip ow tap” and
may require a longer catheter if the Mitrofanoff channel becomes
stretched.
3. Close observation of regular blood pressure and urinary dipstick
monitoring according to standard antenatal protocols.
4. Observation of changes in bladder control; counsel patients that some
incontinence may develop but is typically improved postpartum.
5. Develop a surgical approach plan, and place it in the patient’s medical
record.
(continued)
28 Gynecology inBladder Exstrophy
443
Table 28.1
Delivery
planning
Postpartum 1. Postoperative length of stay usually 3 or 4days.
Adapted from Deans [10] and Dy [56]
(continued)
1. Vaginal delivery may be considered based on the complexity of anatomical
reconstruction and the number of past laparotomies.
2. Plan elective cesarean for 37–39weeks with MFM, urology, and
obstetrical anesthesia teams.
3. Delivery in full operating room suite with neonatal care available.
4. Regional anesthesia is provided by the obstetrical anesthesia team.
5. Consider upper paramedian/supraumbilical incision contralateral to stoma
with classical uterine incision (vertical hysterotomy).
6. Consider the use of a exible O retractor (Alexis) for circumferential
retraction of abdominal contents.
7. Dene the course of the ureters and their implantation into the
reconstructed bladder.
8. Inated Foley catheter balloon for dening the boundaries of the
reconstructed bladder intraoperatively.
2. Monitor for return to pre-pregnancy catheterization regimen, continence,
and predisposition toward UTIs.
3. Follow-up for late complications of pregnancy including increased risk of
pregnancy-related hypertensive diseases.
4. Counsel patients on future increased risk in subsequent pregnancies and
iatrogenic late preterm delivery due to repeat CD following classical incision.
Conclusions
The role of gynecologic care in the patient with CBE spans from infancy to
adulthood. A multidisciplinary approach to the bladder exstrophy patient can
optimize patient outcomes at each step of the process. Gynecologic care should
be incorporated early and proactively into pediatric care for patients with
CBE.Continued evaluation and management of menstruation, sexual function,
fertility, pregnancy care, and pelvic organ prolapse should be included in longterm CBE follow-up.
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