Prenatal Decision-Making for Myelomeningocele:
Can We Minimize Bias and Variability?
Stephen D. Brown, MDa,b, Chris Feudtner, MD, PhD, MPHc,d, Robert D. Truog, MD, MAb,e
Prenatal decision-making for myelomeningocele changed in 2011, when
MOMS (Management of Myelomeningocele Study) demonstrated the
effectiveness of intrauterine fetal surgery, thereby securing this
intervention as an alternative to either pregnancy termination or
postnatal repair.1 Although it is hoped that an evidence-based
approach to medical decisions using the MOMS results would
improve the quality of information received by pregnant patients, we
believe that unless specific steps are taken, prenatal counseling for
myelomeningocele (counseling that occurs in myriad locations across the
United States) will be persistently marred by hidden biases and excess
variability.
We believe this prenatal counseling will be affected for 3 reasons. First,
the MOMS results present tradeoffs. Rates of ventriculoperitoneal shunt
placement and ambulation were improved for children who underwent
prenatal repair but with higher rates of prematurity and respiratory
distress syndrome, as well as important maternal complications. Adding to
the uncertainty, long-term outcomes are unavailable; prognosis remains
difficult to predict for any given fetus; and posttrial complication rates
reportedly differ from trial results, with higher rates of perinatal death
(6% vs 3%) and respiratory distress syndrome (52% vs 21%) but fewer
maternal complications (pulmonary edema, 2% vs 6%; maternal
transfusion, 3.4% vs 9%).1 Women undergoing intrauterine surgery
require cesarean deliveries for all future pregnancies. The reasoning
regarding such multifaceted risk/benefit tradeoffs for obstetric and
neonatal interventions is notably difficult for both physicians and
patients.2
Second, physicians commonly have differing opinions regarding newer
interventions that have not become standard of care, based on personal
experiences and interpretations of the literature. One survey of
maternal-fetal medicine and pediatrics specialists conducted before the
MOMS results were published found no agreement regarding whether to
recommend open intrauterine surgery for a hypothetical fetal condition
similar to myelomeningocele.3 Furthermore, attitudes diverged about
appropriate reasons to consider pregnancy termination in general, the
importance of offering information about pregnancy termination when
myelomeningocele is diagnosed, and supportiveness for patients’
Departments of aRadiology, and eAnesthesia, Perioperative and Pain
Medicine, Boston Children’s Hospital, Boston, Massachusetts; bCenter
for Bioethics, Harvard Medical School, Boston, Massachusetts; and
Departments of cPediatrics and dMedical Ethics, Children’s Hospital of
Philadelphia, Philadelphia, Pennsylvania
Dr Brown conceptualized, drafted, and revised the initial
manuscript; Drs Feudtner and Truog critically reviewed and
revised the manuscript; and all authors approved the final
manuscript as submitted.
www.pediatrics.org/cgi/doi/10.1542/peds.2015-1181
DOI: 10.1542/peds.2015-1181
Accepted for publication Jun 9, 2015
Address correspondence to Stephen D. Brown, MD,
Department of Radiology, Boston Children’s Hospital, 300
Longwood Ave, Boston, MA 02115. E-mail: stephen.brown@
childrens.harvard.edu
PEDIATRICS (ISSN Numbers: Print, 0031-4005; Online, 10984275).
Copyright © 2015 by the American Academy of Pediatrics
FINANCIAL DISCLOSURE: The authors have indicated they have
no financial relationships relevant to this article to disclose.
FUNDING: No external funding.
POTENTIAL CONFLICT OF INTEREST: The authors have indicated
they have no potential conflicts of interest to disclose.
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PEDIATRICS Volume 136, number 3, September 2015
PEDIATRICS PERSPECTIVES
decisions to terminate affected
pregnancies.3,4 We do not know how
such attitudes affect counseling
proclivities or whether the MOMS
results will change them. After
publication of MOMS, experts have
continued to disagree on whether
prenatal or postnatal repair for
myelomeningocele is the better
option.5
At issue is how provider differences
influence decision-making and
outcomes for myelomeningocele,
particularly given that much of the
initial counseling is provided by
obstetricians in centers that do not
perform the prenatal surgery and
most pediatrics centers offering the
surgery do not perform pregnancy
termination. Similar heterogeneity in
clinical practice and technological
capabilities is common in obstetrics
and neonatology and is associated
with considerable outcome
variations.2 Endemic variation in the
treatment of and outcomes for
periviable newborns raises concern
that clinician biases and center
predilections influence parental
decisions in a hidden and undue
manner. We anticipate similar
dynamics regarding prenatal decisionmaking for myelomeningocele, for
which practice and outcome variation
will likely be fostered by uneven
geographic and financial access to
intrauterine surgery and pregnancy
termination, and further exacerbated
as inexperienced centers begin to offer
the surgery.
Third, potential provider biases
regarding prenatally diagnosed
myelomeningocele may be
accentuated by differences between
practice guidelines issued by the
Myelomeningocele Maternal-Fetal
Management Task Force convened by
the Eunice Kennedy Shriver Institute
of Child Health and Human
Development (MMCTF) and the
position statement on maternal-fetal
interventions issued jointly by the
American Academy of Pediatrics
(AAP) and the American College of
410
Obstetricians and Gynecologists
(ACOG).6,7 To facilitate optimal
decision-making, the MMCTF
recommends a “maternal/fetal
advocate to ensure that counseling is
nondirective,” whereas the AAP/
ACOG recommends “an independent
advocate” who “should be
nondirective in his or her support of
the woman’s decision and focus on
meeting the woman’s decisionmaking needs.” Unwittingly or not,
the language variance suggests value
differences regarding the central
focus of counseling. Furthermore,
both documents endorse nondirective
counseling, but only the AAP/ACOG
statement explicitly acknowledges
the challenge of providing unbiased
information. Both statements
prescribe that pregnancy termination
should be discussed as an option,
along with prenatal and postnatal
surgery. The AAP/ACOG adds that
centers offering fetal treatment also
bear ethical responsibility to women
considering termination to provide
“appropriate mechanisms, including
the ability and resources for
referral…” They further recommend
the availability of bereavement
support for patients who terminate
the pregnancy. The MMCTF does not
recognize such obligations, which is
an important potential source of bias.
Some pediatrics-based fetal care
centers may not recognize such
obligations as within their domain,
but further bias emerges if such
support is available for a hospital’s
adolescent patients with normal
pregnancies but not for adult
pregnant patients carrying fetuses
with congenital abnormalities. Finally,
the AAP/ACOG document specifies
that maternal-fetal medicine
specialists should direct the care of
women undergoing fetal intervention
and expresses concern that obstetric
and pediatric practitioners may
sometimes possess conflicting
perspectives. The MMCTF, which
represented clinical providers specific
to myelomeningocele, does not
stipulate who should direct the
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woman’s overall care and does not
clearly define the maternal-fetal
medicine role. In sum, the AAP/ACOG
statement more clearly supports the
pregnant woman’s central decisionmaking status. Although the proportion
of centers that subscribe to either
statement is unknown, preferential
adherence to 1 or the other may result
in considerably different counseling
across clinicians and centers.
Despite calls for nondirectiveness, the
complex conditions depicted here
leave wide openings for undue
provider and institutional influence
over patient decisions and, ultimately,
outcomes. Even if all options are
discussed, outcomes may vary for
those offered different degrees of
accessibility, information, and
support for any given decision.
Differences regarding various
counseling points may leave patients
with uncertainty regarding whether
the information has been selectively
offered, withheld, or framed favorably
or negatively and to what degree the
recommendation they received is
influenced by individual consultants’
values and beliefs or by particular
centers’ practice characteristics.
Patients may thus be uninformed
regarding the accessibility and level
of support for pregnancy termination
and prenatal or postnatal surgery at
the place where they received
consultation, as well as how these
features differ among institutions;
these factors could hamper patients’
ability to seek care elsewhere.
We therefore urge the community of
providers who diagnose, provide
counseling for, and treat
myelomeningocele prenatally and
postnatally to establish uniform
guidelines regarding the requisite
information to be offered. A
standardized counseling information
sheet should be made available that
clearly states:
1. Major treatment options for
myelomeningocele, including
prenatal surgery, postnatal survey, and termination.
BROWN et al
2. Expected outcome ranges for
each option, including potential
physical and psychosocial risks
and benefits to pregnant women
and neonates, and long-term
profiles of individuals with
myelomeningocele.
not eliminate hidden bias and
capricious variation, this practice
would reduce variation in the
information offered and help patients
make informed decisions about what
treatment to select and where to seek
care.
3. Outcome variations among
centers.
ACKNOWLEDGMENTS
4. Options the consultant’s organization offers.
5. Options recommended and why.
6. Selection criteria for patients offered prenatal surgery and how
they compare with MOMS.
7. The center’s own experience with
the surgery (if offered) and how
center outcomes compare with
MOMS.
8. What remains unknown.
9. Transparent admissions regarding providers’ limits of
knowledge and experience.
10. Referral information for questions about or services for termination or surgery if not
provided within the institution.
We do not seek standardization of
services or recommendations. The
process and content of counseling
would remain the independent
responsibility of the clinician.
A standardized information sheet
would ensure that a range of basic
information and options is covered
along with a summation of the key
literature. Although this resource will
PEDIATRICS Volume 136, number 3, September 2015
The authors thank Christine Mitchell,
RN, MS, MTS, for her review of
numerous drafts of the manuscript.
ABBREVIATIONS
AAP: American Academy of
Pediatrics
ACOG: American College of
Obstetricians and
Gynecologists
MMCTF: Myelomeningocele
Maternal-Fetal
Management Task Force
convened by the Eunice
Kennedy Shriver National
Institute of Child Health
and Human Development
MOMS: Management of
Myelomeningocele Study
2. Raju TN, Mercer BM, Burchfield DJ, Joseph
GF Jr. Periviable birth: executive summary
of a joint workshop by the Eunice
Kennedy Shriver National Institute of Child
Health and Human Development, Society
for Maternal-Fetal Medicine, American
Academy of Pediatrics, and American
College of Obstetricians and
Gynecologists. Obstet Gynecol. 2014;
123(5):1083–1096
3. Brown SD, Ecker JL, Ward JR, et al.
Prenatally diagnosed fetal conditions in
the age of fetal care: does who counsels
matter? Am J Obstet Gynecol. 2012;206(5):
409.e1–11
4. Brown SD, Donelan K, Martins Y, et al.
Does professional orientation predict
ethical sensitivities? Attitudes of
paediatric and obstetric specialists
toward fetuses, pregnant women and
pregnancy termination. J Med Ethics.
2014;40(2):117–122
5. van Lith JM, Johnson MP, Wilson RD.
Current controversies in prenatal
diagnosis 3: fetal surgery after MOMS: is
fetal therapy better than neonatal? Prenat
Diagn. 2013;33(1):13–16
REFERENCES
6. American College of Obstetricians and
Gynecologists, Committee on Ethics;
American Academy of Pediatrics,
Committee on Bioethics. Maternal-fetal
intervention and fetal care centers.
Pediatrics. 2011;128(2). Available at:
www.pediatrics.org/cgi/content/full/128/
2/e473
1. Moldenhauer JS, Soni S, Rintoul NE, et al.
Fetal myelomeningocele repair: the
post-MOMS experience at the Children’s
Hospital of Philadelphia. Fetal Diagn Ther.
2015;37(3):235–240
7. Cohen AR, Couto J, Cummings JJ, et al;
MMC Maternal-Fetal Management Task
Force. Position statement on fetal
myelomeningocele repair. Am J Obstet
Gynecol. 2014;210(2):107–111
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411
Prenatal Decision-Making for Myelomeningocele: Can We Minimize Bias and
Variability?
Stephen D. Brown, Chris Feudtner and Robert D. Truog
Pediatrics 2015;136;409; originally published online August 3, 2015;
DOI: 10.1542/peds.2015-1181
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PEDIATRICS is the official journal of the American Academy of Pediatrics. A monthly
publication, it has been published continuously since 1948. PEDIATRICS is owned, published,
and trademarked by the American Academy of Pediatrics, 141 Northwest Point Boulevard, Elk
Grove Village, Illinois, 60007. Copyright © 2015 by the American Academy of Pediatrics. All
rights reserved. Print ISSN: 0031-4005. Online ISSN: 1098-4275.
Downloaded from by guest on July 28, 2017
Prenatal Decision-Making for Myelomeningocele: Can We Minimize Bias and
Variability?
Stephen D. Brown, Chris Feudtner and Robert D. Truog
Pediatrics 2015;136;409; originally published online August 3, 2015;
DOI: 10.1542/peds.2015-1181
The online version of this article, along with updated information and services, is
located on the World Wide Web at:
/content/136/3/409.full.html
PEDIATRICS is the official journal of the American Academy of Pediatrics. A monthly
publication, it has been published continuously since 1948. PEDIATRICS is owned,
published, and trademarked by the American Academy of Pediatrics, 141 Northwest Point
Boulevard, Elk Grove Village, Illinois, 60007. Copyright © 2015 by the American Academy
of Pediatrics. All rights reserved. Print ISSN: 0031-4005. Online ISSN: 1098-4275.
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