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Original Paper
Received: June 29, 2016
Accepted after revision: November 25, 2016
Published online: March 17, 2017
Fetal Diagn Ther 2017;42:210–217
DOI: 10.1159/000455024
Specialty-Based Variation in Applying
Maternal-Fetal Surgery Trial Evidence
Ryan M. Antiel a–c Alan W. Flake a Mark P. Johnson a Nahla Khalek a
Natalie E. Rintoul a John D. Lantos e Farr A. Curlin f Jon C. Tilburt b, d
Chris Feudtner a a
University of Pennsylvania Perelman School of Medicine and Children’s Hospital of Philadelphia, Philadelphia, PA,
Biomedical Ethics Program, c Department of General Surgery, and d Division of General Internal Medicine, Mayo
Clinic, Rochester, MN, e Department of Pediatrics, Children’s Mercy Hospital, Kansas City, MO, and f Trent Center for
Bioethics, Humanities, and History of Medicine, Duke University, Durham, NC, USA
Keywords
Maternal-fetal surgery · Myelomeningocele · Spina bifida ·
Prenatal diagnosis · Physician attitudes
Abstract
Introduction: The Management of Myelomeningocele Study
(MOMS) compared prenatal with postnatal surgery for fetal
myelomeningocele (MMC). We sought to understand how
subspecialists interpreted the trial results and whether their
practice has changed. Materials and Methods: Cross-sectional, mailed survey of 1,200 randomly selected maternalfetal medicine (MFM) physicians, neonatologists, and pediatric surgeons. Results: Of 1,176 eligible physicians, 670
(57%) responded. Compared to postnatal closure, 33%
viewed prenatal closure as “very favorable” and 60% as
“somewhat favorable.” Most physicians reported being
more likely to recommend prenatal surgery (69%), while
28% were less likely to recommend pregnancy termination.
In multivariable analysis, neonatologists were more likely to
report prenatal closure as “very favorable” (OR 1.6; 95% CI:
1.03–2.5). Pediatric surgeons and neonatologists were more
© 2017 S. Karger AG, Basel
E-Mail karger@karger.com
www.karger.com/fdt
likely to recommend prenatal closure (OR 2.1; 95% CI: 1.3–
3.3, and OR 2.9; 95% CI: 1.8–4.6) and less likely to recommend
termination (OR 3.8; 95% CI: 2.2–6.7, and OR 4.7; 95% CI: 2.7–
8.1). In addition, physicians with a higher tolerance for prematurity were more likely to report prenatal closure as “very
favorable” (OR 1.02; 95% CI: 1.00–1.05). Discussion: In light
of the MOMS trial, the vast majority of pediatric subspecialists and MFMs view prenatal MMC closure favorably. These
attitudes vary by specialty and risk tolerance.
© 2017 S. Karger AG, Basel
Introduction
Myelomeningocele (MMC) represents the first nonlethal prenatal diagnosis that can be managed with maternal-fetal surgery. MMC is characterized by protrusion of
the spinal cord through the open vertebrae into the amniotic fluid, frequently resulting in variable degrees of
lifelong disability. In the recent past, three options were
available: postnatal closure, postnatal palliative care, or
termination of pregnancy. Now a fourth option exists.
Ryan M. Antiel, MD
University of Pennsylvania Perelman School of Medicine
Blockley Hall, 423 Guardian Drive, Floor 14
Philadelphia, PA 19104 (USA)
E-Mail antiel @ mail.med.upenn.edu
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b
Surgeons can now perform maternal-fetal surgery and
close the MMC prenatally. This specific open maternalfetal surgery aims to mitigate damage from chronic chemical and mechanical insults in utero. Early results from
nonrandomized studies suggested that prenatal closure
could minimize or reverse hindbrain herniation and decrease the need for cerebrospinal fluid shunt for hydrocephalus [1–5].
In February 2003, with the endorsement of the American College of Obstetrics and Gynecology [6] and others
[7–9], the NIH sponsored a prospective randomized clinical trial to test the efficacy and safety of prenatal surgical
treatment. The Management of Myelomeningocele Study
(MOMS) was conducted at the Children’s Hospital of
Philadelphia, Vanderbilt University, and the University
of California, San Francisco [10]. Prenatal closure resulted in a decrease in shunt placement (40 vs. 82%), a decrease in the presence of hindbrain herniation (64 vs.
96%), as well as an increase in the ability to walk independently without orthotics upon exam (42 vs. 21%) at 30
months of age. There were, however, significant maternal
and fetal risks. The prenatal surgery group experienced
an increased incidence of chorioamniotic membrane separation (26 vs. 0%), spontaneous membrane rupture (46
vs. 8%), need for blood transfusion at cesarean delivery (9
vs. 1%), and area of uterine dehiscence at hysterotomy (10
vs. 0%). These complications led to a greater incidence of
prematurity in the prenatal surgery group (13% delivered
at less than 30 weeks’ gestation vs. 0%).
Given the tradeoffs revealed by the trial results between the potential neonatal benefits and maternal and
neonatal risks and the uncertainty of outcomes for any
given pregnant woman and fetus, some physicians urged
caution before widespread adoption of this approach
[11]. As such, MOMS illustrates some of the well-known
limitations of clinical trials that measure multiple outcomes. Patient decisions inevitably reflect preferences
and values, along with high-quality evidence [12]. In the
case of prenatally diagnosed MMC, the decision-making
process typically occurs in a stepwise manner, with obstetricians and maternal-fetal medicine (MFM) physicians
first communicating with and counseling the pregnant
woman regarding a range of options. Neonatologists and
pediatric surgeons are often involved later in the multidisciplinary process of prenatal evaluation and counseling.
We sought to clarify how the results of MOMS influenced the opinions and potential recommendations of
these three professional specialties involved in the care of
pregnant women and fetuses with MMC. We hypothe-
sized that MFM physicians, with their focus on the health
and well-being of the maternal-fetal dyad, would be less
persuaded by the MOMS results. In comparison, neonatologists and pediatric surgeons, with their focus on the
care of neonates, would be more favorably influenced by
the trial results. Since the publication of the MOMS trial
results, no empirical studies have been conducted to describe how these subspecialists have interpreted and applied the trial findings.
Applying Maternal-Fetal Surgery Trial
Evidence
Fetal Diagn Ther 2017;42:210–217
DOI: 10.1159/000455024
Materials and Methods
Study Participants and Data Collection
In the spring of 2015, we mailed a confidential, self-administered, 11-page, 32-item questionnaire titled “Emerging Issues in
Maternal-Fetal Surgery” to a random sample of 1,200 practicing
US physicians. The sample included 400 MFMs, 400 neonatologists, and 400 pediatric surgeons. These physicians were randomly
selected from member lists of the Society for Maternal-Fetal Medicine, the American Academy of Pediatrics Section on Perinatal
Pediatrics, and the American Academy of Pediatrics Section on
Surgery, respectively. Using the Tailored Design Method [13], up
to three separate mailings were sent, the first mailing including a
USD 20 bill. This study was reviewed and deemed exempt by the
Mayo Clinic Institutional Review Board.
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Survey Instrument
We reviewed the literature, consulted with content experts,
conducted physician interviews at five maternal-fetal centers, formulated questions, and then conducted cognitive interviews in
which draft survey instruments were administered to specialty colleagues whose feedback was used to revise questions further. The
primary outcome variables were physicians’ responses to a series
of questions asked after participants reviewed a table of results
from the MOMS trial (see Appendix A). First, physicians were
asked, “In your opinion, how favorable is prenatal surgery for
MMC, compared to postnatal surgery?” Response categories were
“very favorable,” “somewhat favorable,” “somewhat unfavorable,”
“unfavorable,” or “not sure.” Next, physicians were asked, “In light
of these results, please tell us whether your attitude changed regarding any of the options that are available to pregnant women
with a newly diagnosed fetal MMC. Do these results make you
more or less likely to recommend: termination of pregnancy, open
uterine prenatal surgery, and postnatal closure (no fetal surgery)?”
Response categories for each were “less likely,” “no change,” and
“more likely.”
Given that the results of the MOMS trial may not be reproducible in some centers, we asked physicians to assume their center
offered prenatal surgery and that they were reviewing their institution’s 5-year outcome data. We then asked physicians to indicate
the highest complication rate of three outcomes associated with
prenatal surgery that they would consider acceptable. Outcomes
included: rate of perinatal mortality, rate of prematurity (less than
30 weeks), and rate of uterine dehiscence.
We also examined physician demographic characteristics (age,
sex, region, specialty, and years in practice) and practice setting
type.
Results
Of the 1,200 potential respondents, 24 (2%) could not
be contacted. Of the remaining participants, 670 returned
completed surveys for a response rate of 57%. Response
rates did not differ significantly by specialty (MFM, 54%;
neonatology, 57%; pediatric surgery, 60%; p = 0.2). The
characteristics of the respondents are shown in Table 1.
Subspecialty Interpretation of Evidence and Influence
on Clinical Recommendations
As seen in Table 2, the majority of our cohort believe
that prenatal closure for MMC (compared to postnatal
closure) was favorable (33% “very favorable,” 60% “somewhat favorable”). In light of the MOMS trial results, 69%
of respondents reported that they are more likely to recommend prenatal closure, and only 4% indicated they
were less likely to recommend prenatal closure. In addition, 38% of all respondents reported they would be less
likely to recommend postnatal closure in light of trial results, and only 4% would be more likely (58% no change).
The MOMS trial appeared to influence some physicians’ attitudes about termination of pregnancy: 28% of
respondents reported that they were now less likely to recommend termination (71% reported no change). A third
of neonatologists and pediatric surgeons indicated that
they are now less likely to recommend termination (36%
and 32%, respectively), whereas 13% of MFM specialists
reported being now less likely to recommend termination.
Risk Tolerance
As seen in Table 3, physician-reported median acceptable complication rates were quite similar to the complication rates reported in the MOMS trial for perinatal
mortality (5 vs. 3%, respectively), gestational age at birth
<30 weeks (15 vs. 13%, respectively), and uterine dehiscence (10 vs. 10%, respectively). The highest acceptable
rates of complications by specialty are reported in Ta212
Fetal Diagn Ther 2017;42:210–217
DOI: 10.1159/000455024
Table 1. Characteristics of the 670 US physicians who responded
to the survey
Characteristic
Response
Specialty
Maternal-fetal medicine specialist
Neonatologist
Pediatric surgeon
Female sex
Age
<50 years
≥50 years
Years in practice, mean (SD), years
Race or ethnic group
White or Caucasian
Asian
Black or African American
Other
Region
Midwest
Northeast
South
West
Practice setting type
Solo, private
Group, private
Institutional, private
Academic
Other
Affiliation with fetal center
Location (fetal care affiliates only)
General hospital
Women’s/women and infant hospital
Children’s hospital
Freestanding clinic or outpatient facility
Other
208/388 (54)
228/397 (57)
234/391 (60)
245/659 (37)
256/653 (39)
397/653 (61)
18 (11)
523/664 (79)
88/664 (13)
27/664 (4)
26/664 (4)
168/670 (25)
141/670 (21)
235/670 (35)
126/670 (19)
21/663 (3)
152/663 (23)
95/663 (14)
345/663 (52)
50/663 (8)
402/660 (61)
107/376 (29)
99/376 (26)
132/376 (35)
28/376 (7)
10/376 (3)
Data are given as n (%), except where indicated. Percentages
may not total 100 because of rounding.
ble 3. MFM specialists were willing to accept slightly
higher average rates of perinatal mortality than neonatologists or pediatric surgeons (7.8% compared to 6.4 and
5.8%, respectively, p < 0.001).
Multivariate Analysis
In multivariate logistic regression models controlling
for age, years in practice, region, and practice setting (Table 4), self-reported affiliation with a fetal center was not
significantly associated with responses to the relevant
items. Male physicians were more likely to recommend
open maternal-fetal surgery (74 vs. 66%; odds ratio [OR]
1.69; 95% confidence interval [CI]: 1.08–2.64) and were
more likely to report that they would not recommend terAntiel/Flake/Johnson/Khalek/Rintoul/
Lantos/Curlin/Tilburt/Feudtner
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Data Management and Analysis
Responses were double entered and imported into SAS version
9.3. We used the American Association for Public Opinion Research RR2 response rate definition [14]. We used descriptive statistics and logistic regression to assess associations between physician specialty and characteristics and their attitudes toward prenatal MMC closure in light of the MOMS trial results. When
distributions allowed, we dichotomized results for ease of presentation. The Kruskal-Wallis test was used to compare the self-reported highest acceptable complication rates among specialties.
Statistical inferences were based on 2-tailed tests with significance
set at p < 0.05.
Table 2. Physicians’ opinions about the MOMS trial results
All physicians
MFMs
Neonatologists
Considering the known risks and benefits, how favorable is open prenatal surgery for MMC?
Very favorable
219 (33)
62 (31)
86 (39)
Somewhat favorable
398 (60)
127 (64)
124 (56)
Somewhat unfavorable
25 (4)
10 (5)
5 (2)
Unfavorable
7 (1)
1 (1)
5 (2)
Compared to before the MOMS trial, are you now more or less likely to recommend:
Termination of pregnancy
Less likely
182 (28)
26 (13)
No change
468 (71)
175 (86)
More likely
8 (1)
2 (1)
Prenatal MMC closure
Less likely
23 (4)
8 (4)
No change
185 (28)
81 (40)
More likely
454 (69)
115 (56)
Postnatal MMC closure (no fetal surgery)
Less likely
250 (38)
69 (34)
No change
382 (58)
123 (61)
More likely
25 (4)
10 (5)
Surgeons
71 (31)
147 (64)
10 (4)
1 (1)
81 (36)
139 (62)
4 (2)
75 (32)
154 (67)
2 (1)
9 (4)
51 (23)
165 (73)
6 (3)
53 (23)
174 (75)
92 (41)
124 (55)
8 (4)
89 (39)
135 (58)
7 (3)
Data are given as n (%). Percentages may not total 100 because of rounding. MFMs, maternal-fetal medicine specialists; MMC,
myelomeningocele.
Table 3. Physicians’ risk tolerance
Survey item
Group
All physicians
p value
MFMs
Neonatologists
Surgeons
What is the highest complication rate associated with open prenatal surgery that you would consider acceptable?
Rate of perinatal death, %
Mean (SD)
6.6 (8.6)
7.8 (9.67)
6.4 (8.1)
5.8 (8.0)
Median (IQR)
5 (3–5)
5 (3–10)
5 (2–5)
4 (2–5)
Rate of prematurity (less than 30 weeks), %
Mean (SD)
16.8 (11.0)
16.8 (9.6)
17.1 (12.3)
16.5 (10.7)
Median (IQR)
15 (10–20)
15 (10–20)
15 (10–20)
15 (10–20)
Rate of uterine dehiscence, %
Mean (SD)
9.4 (8.1)
8.6 (7.9)
10.3 (9.1)
9.0 (7.0)
Median (IQR)
10 (5–10)
10 (5–10)
10 (5–10)
10 (5–10)
<0.001
0.47
0.20
mination of pregnancy (30 vs. 24%; OR 1.91; 95% CI:
1.15–3.16).
Neonatologists, compared to MFMs, were more likely
to report prenatal closure as “very favorable” (OR 1.64;
95% CI: 1.00–2.70). Compared to MFMs, pediatric surgeons and neonatologists were more likely to recommend
prenatal closure (OR 2.11; 95% CI: 1.27–3.49, and OR
2.57; 95% CI: 1.55–4.25, respectively) and were less likely
to recommend termination (OR 4.09; 95% CI: 2.17–7.72,
and OR 4.40; 95% CI: 2.36–8.21, respectively).
The acceptable rates of complications were also significantly associated with how a physician interpreted
Applying Maternal-Fetal Surgery Trial
Evidence
Fetal Diagn Ther 2017;42:210–217
DOI: 10.1159/000455024
213
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p values are for the comparison between specialty groups and were calculated with the use of the Kruskal-Wallis test. MFMs,
maternal-fetal medicine specialists.
Table 4. Physicians’ opinions about the MOMS trial results according to physician characteristics
Age
<50 years
≥50 years
Sex
Female
Male
Affiliation with fetal center
Yes
No
Specialty
MFM
Surgeon
Neonatologist
Risk tolerance
Rate of perinatal death
Rate of prematurity
Rate of uterine dehiscence
Interpretation of
MOMS results
Recommendations after MOMS trial
Prenatal surgery for
MMC (very favorable
vs. somewhat favorable)
Less likely to recommend
termination of pregnancy
(vs. no change)
More likely to
recommend open prenatal
closure (vs. no change)
Less likely to
recommend postnatal
closure (vs. no change)
1.0 (referent)
0.73 (0.37–1.45)
1.0 (referent)
0.67 (0.31–1.44)
1.0 (referent)
1.05 (0.52–2.12)
1.0 (referent)
1.67 (0.86–3.24)
1.0 (referent)
1.35 (0.86–2.10)
1.0 (referent)
1.91 (1.15–3.16)*
1.0 (referent)
1.69 (1.08–2.64)*
1.0 (referent)
0.94 (0.61–1.44)
1.0 (referent)
0.85 (0.57–1.29)
1.0 (referent)
0.93 (0.58–1.47)
1.0 (referent)
0.93 (0.61–1.44)
1.0 (referent)
1.15 (0.77–1.73)
1.0 (referent)
0.98 (0.59–1.66)
1.64 (1.00–2.70)*
1.0 (referent)
4.09 (2.17–7.72)***
4.40 (2.36–8.21)***
1.0 (referent)
2.11 (1.27–3.49)**
2.57 (1.55–4.25)***
1.0 (referent)
1.30 (0.79–2.12)
1.43 (0.88–2.32)
0.99 (0.96–1.02)
1.02 (1.00–1.05)*
1.01 (0.99–1.03)
0.96 (0.93–0.99)*
1.05 (1.02–1.07)***
1.01 (0.98–1.04)
0.97 (0.94–1.00)
1.05 (1.02–1.08)**
1.02 (0.99–1.05)
0.98 (0.95–1.01)
1.02 (0.99–1.04)
1.03 (1.00–1.06)*
Data are given as odds ratios (95% confidence intervals). Odds ratios are from multivariate logistic regression, with adjustment for
years in practice, region of practice, practice setting type, and ethnicity/race. MMC, myelomeningocele; MFM, maternal-fetal medicine
specialist. * p < 0.05, ** p < 0.01, *** p < 0.001.
Discussion
Most physicians among these relevant specialties believe the results from the MOMS trial are “somewhat” or
“very” favorable. In light of the trial results, the majority of
surveyed subspecialists report that they have shifted to now
being more likely to recommend prenatal MMC closure,
with the greatest shift occurring among neonatologists and
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Fetal Diagn Ther 2017;42:210–217
DOI: 10.1159/000455024
pediatric surgeons. The majority of physicians report that
the MOMS trial results have not changed their likelihood
of recommending termination of pregnancy; however, a
significant minority (28%) report that they are now less
likely to recommend termination. MFM physicians were
less likely to have changed their recommendations compared to the pediatric subspecialists, likely reflecting MFMs’
commitment to nondirective or neutral counseling [15].
The interpretation and influence of the MOMS trial
evidence appears to vary by respondent characteristics.
Compared to MFMs, neonatologists interpret the MOMS
trial results more favorably. Compared to MFMs, neonatologists and pediatric surgeons are more likely to recommend prenatal MMC surgery, and both are now less likely to recommend termination of pregnancy. In addition,
physician tolerance for risk of complications was also associated with trial interpretation and influence, even
while controlling for specialty. And finally, female physicians were less likely to change their recommendations
about prenatal closure or termination of pregnancy than
were male physicians, potentially because maternal-fetal
surgery directly affects the reproductive health of women.
Antiel/Flake/Johnson/Khalek/Rintoul/
Lantos/Curlin/Tilburt/Feudtner
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and applied MOMS trial results. Physicians who would
tolerate a high rate of prematurity were more likely to
view prenatal closure as “very favorable” (OR 1.02; 95%
CI: 1.00–1.05), more likely to recommend prenatal surgery (OR 1.05; 95% CI: 1.02–1.08), and more likely to not
recommend termination (OR 1.05; 95% CI: 1.02–1.07).
Physicians who would tolerate higher rates of perinatal
death did not change in their recommendations for termination following the trial (OR 0.96; 95% CI: 0.93–0.99).
Finally, physicians who reported a higher tolerance for
uterine dehiscence were less likely to recommend postnatal surgery (OR 1.03; 95% CI: 1.00–1.06).
tions of pregnancy [31]. A European center recently reported that 76% of prenatal diagnoses of spina bifida resulted in termination, and the authors found that requests
were not correlated with the severity of disease [32]. The
results of the MOMS trial may modify this number as
perceptions surrounding open maternal-fetal surgery
change. Our results suggest, however, that the impact of
the MOMS trial results on decisions surrounding termination may depend upon who conveys those results to the
pregnant woman. MFMs are more likely than neonatologists or pediatric surgeons to discuss termination of pregnancy as an acceptable option for pregnancy management in addition to prenatal surgery for MMC.
Patients and families may be affected, directly or indirectly, by the professional and personal factors that shape
how respondents replied to the questions posed by this
survey. Specifically, prenatal counseling may be influenced by provider specialty as well as risk tolerance for
various associated complications. This study highlights
the need for balanced consultation, full disclosure, and an
open dialogue with families to optimize shared decisionmaking and mitigate unconscious bias. Ideally, all physicians would engage in explicit and open discussions about
the potential risks and benefits to both the pregnant
woman and to her fetus [30]. As pediatric subspecialists
participate in the care discussion for women who elect
prenatal surgery for MMC, they must be sensitive to, and
mindful of, issues surrounding the pregnant woman and
her family. Furthermore, while some of the individual
centers involved in the MOMS trial have replicated the
trial results following the closure of the study [33], it is
crucial for less experienced centers to be transparent with
their outcomes.
Our findings should be interpreted with four main
limitations kept in mind. First, in order to capture attitudinal changes, we used similar language to previously
published studies, including the term “recommend” [34].
While we cannot determine whether respondents’ reports of changes in their attitudes correspond to changes
in their clinical practices, including specific recommendations, ample evidence exists that attitudes towards
medical and surgical interventions influence how physicians counsel patients [35–37]. Second, while we identified specific hypothesized associations in this cross-sectional study, other factors may also be associated with
how physicians interpret trial data and form their subsequent recommendations, including professional society
statements. Previous analysis of the positions of the ethics
committees of the American Academy of Pediatrics and
the American College of Obstetricians and Gynecologists
Applying Maternal-Fetal Surgery Trial
Evidence
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DOI: 10.1159/000455024
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This study suggests that professional identity and specialty role and culture influence the interpretation and
application of trial evidence in maternal-fetal surgery.
Such a finding is consistent with other studies of how
physicians and surgeons interpret and apply medical science [15–17]. Specialty bias and differing interpretations
of evidence lead to divergent treatment recommendations and ongoing debates for conditions ranging from
prostate cancer [18, 19] to menopausal hormone therapy
[20–22] to treatment for sciatica [23–25].
Different specialty groups view certain ethical aspects
of maternal-fetal surgery differently [26]. In the specific
case of the interpretation of the MOMS trial, a concern
for maternal-fetal health, both during the affected pregnancy and from the perspective of future reproductive
health, continues to prevail among MFMs about prenatal
surgery for MMC. Historically, the care of pregnant
women carrying fetuses with anomalies was exclusively
the domain of obstetrics and MFM. With the rapid evolution of prenatal diagnosis and fetal therapy, pediatric subspecialists are now participating in multidisciplinary care
planning for those patients electing open maternal-fetal
surgery. Traditionally, pediatricians and pediatric surgeons are trained to focus on the well-being of the neonate, whereas MFMs are uniquely positioned in that they
routinely consider both the well-being of the pregnant
woman and fetus. In this specific context, these roles have
become more fluid as the fetus has become a “patient”
[27–29]. For most pregnancies, the well-being of the
pregnant woman and the fetus are fully aligned. In the
context of prenatal surgery for MMC, a divergence can
occur, as the pregnant woman assumes increased risks for
the prospect of improving her future child’s well-being.
While MFMs are also dedicated to improving neonatal
outcomes, they must simultaneously manage and balance
the risks to the pregnant woman. Furthermore, MFMs
encounter pregnant women earlier in the process of medical evaluation and decision-making, and the profession
strongly advocates for discussing all possible management options, including termination, in a nondirective
manner. This specific aspect of maternal-fetal care plan
discussion may not be applicable by the time pediatric
subspecialists enter the conversation. At the same time,
however, the majority of both MFMs and pediatric subspecialists believe that a pediatric consult should take
place prior to decision to terminate a pregnancy with spina bifida (77 vs. 82%, respectively) [30].
A systematic review of studies published between 1994
and 2004 estimated that in the US, approximately 43% of
prenatal diagnoses of spina bifida resulted in termina-
found subtle differences between the societies concerning
maternal-fetal conflicts and interventions [38]. Third,
even with the high response rate to this survey, the attitudes of physicians who did not respond to the survey
may differ from those who did respond. Fourth, not all
physicians who responded to the survey are directly involved in caring for patients and families who elect maternal-fetal surgery. Also, we were unable to survey all
types of providers who may be involved in maternal-fetal
counseling including pediatric neurosurgeons or genetic
counselors. We designed the sample frame of our study
to reflect the broader obstetric and pediatric professional
interpretation of the MOMS trial results and recommendations as the field of maternal-fetal surgery is expanding,
with seventeen centers in the US now offering or performing open prenatal surgery for spina bifida.
Notwithstanding these limitations, this study indicates
that the majority of subspecialists view the MOMS trial
results favorably, yet it also notes that the degree to which
physicians’ potential recommendations were influenced
by the MOMS results varied across specialties and by risk
tolerance. Prenatal maternal-fetal surgery for spina bifida
is demonstrative of how the use of evidence-based medicine necessarily requires consideration of trade-offs between maternal health, future reproductive health, fetal
health, and the potential well-being of the future child
[12]. The results of this study can be considered as professional societies draft and update guidelines for counseling. Since individual physician characteristics influence
the interpretation of trial outcome data, physicians should
be self-aware about their own values and risk tolerance
that are operative in their professional interpretation and
discussion of treatment options.
Acknowledgment
We thank Sarah M. Jenkins, MS, from the Department of
Health Sciences Research, Division of Biomedical Statistics and
Informatics, Mayo Clinic, Rochester, MN, USA, for her help with
the statistical analysis.
Disclosure Statement
The authors declare that they have no conflicts of interest.
Funding Sources
Supported by a grant from the Greenwall Foundation.
Appendix A
The recent Management of Myelomeningocele Study (MOMS) was a randomized controlled trial that compared prenatal and postnatal surgery for myelomeningocele (MMC). Results are below:
Fetal or neonatal outcome
Perinatal death
Prematurity <30 weeks
At 12 months
Placement of shunt
Hindbrain herniation
At 30 months
Walking independently on exam
Maternal outcome
Maternal death
Chorioamniotic separation
Spontaneous membrane rupture
Uterine dehiscence
Blood transfusion at delivery
216
Prenatal surgery
(n = 78)
Postnatal surgery
(n = 80)
p value
2 (3%)
10 (13%)
2 (2%)
0
1.00
<0.001
31 (40%)
45/70 (64%)
66 (82%)
66/69 (96%)
<0.001
<0.001
26/62 (42%)
14/67 (21%)
0.01
0
20 (26%)
36 (46%)
8/76 (10%)
7 (9%)
0
0
6 (8%)
NA
1 (1%)
Fetal Diagn Ther 2017;42:210–217
DOI: 10.1159/000455024
<0.001
<0.001
0.03
Antiel/Flake/Johnson/Khalek/Rintoul/
Lantos/Curlin/Tilburt/Feudtner
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Outcome
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