Stillbirth Working Group of Council Meeting Summary – October 20, 2022

On October 20, 2022, the NICHD Stillbirth Working Group of Council (WG) convened its first meeting. The meeting, which was held virtually, was open to the public; the video recording is available online.

WG Members Present:

Co-chairs

• Lucky Jain, M.D., Emory University
• Uma M. Reddy, M.D., M.P.H., Columbia University Irving Medical Center, representative of the American College of Obstetricians and Gynecologists

Members

• RADM Wanda D. Barfield, M.D., M.P.H., Centers for Disease Control and Prevention (CDC)
• CAPT Amanda Cohn, M.D., CDC
• Deborah Conway, M.D., University of Texas Health Science Center at San Antonio
• Carla DeSisto, Ph.D., M.P.H., CDC
• Ada Dieke, Dr.P.H., M.P.H., CDC
• Donald Dudley, M.D., University of Virginia
• Andrew Fullerton, M.P.P., March of Dimes
• Isabelle Horon, Dr.P.H., CDC
• Denise Jamieson, M.D., M.P.H., Emory University
• Stephanie Leonard, Ph.D., Stanford University
• Monica Longo, M.D., Ph.D., Maternal-Fetal Medicine Units Network, Pregnancy and Perinatology Branch, NICHD
• Jenna Nobles, Ph.D., University of Wisconsin–Madison
• Tina Pattara-Lau, M.D., Indian Health Service
• Sonja Rasmussen, M.D., M.S., Johns Hopkins School of Medicine
• Robert M. Silver, M.D., University of Utah Health Sciences Center and representative of the Society for Maternal-Fetal Medicine
• Catherine Vladutiu, Ph.D., M.P.H., Health Resources and Services Administration
• Maeve Wallace, Ph.D., M.P.H., Tulane University School of Public Health and Tropical Medicine
• Monica H. Wojcik, M.D., Boston Children’s Hospital

WG Members Absent:

• Jennita Reefhuis, Ph.D., CDC

Others Present:

• Speakers
• NICHD staff members
• Members of the public

Welcome and Opening Remarks

Diana W. Bianchi, M.D., Director, NICHD

Dr. Bianchi welcomed WG members and attendees to the first meeting of the Stillbirth Working Group of the National Advisory Child Health and Human Development (NACHHD) Council and introduced the WG’s co-chairs.

Dr. Bianchi noted that since the 1940s, improvements in obstetrical care had led to a dramatic decrease in the incidence of stillbirth but that, more recently, the decline had slowed or halted. About 20,000 stillbirths were reported in the United States in 2020. Dr. Bianchi outlined the WG’s four priority areas of focus and noted that stillbirth research falls squarely within NICHD’s mission and under major themes outlined in the institute’s 2020 strategic plan.

Understanding more about stillbirth poses a challenge, in part because stillbirth’s causes are varied and complex. Stillbirth is a rare event, so analysis is difficult if a study does not have many participants. In addition, different ways of collecting stillbirth data can make comparisons among states difficult.

NICHD advanced stillbirth research through its Stillbirth Collaborative Research Network (SCRN), which recruited participants from 59 hospitals across the United States and confirmed common causes of stillbirth, suggesting future avenues of research. Although funding ended several years ago, specimens and data from the SCRN available through the institute’s Data and Specimen Hub (DASH) are used to analyze causes of stillbirth, sometimes with technologies not previously available. At least four related articles were published in 2022. NICHD’s Maternal-Fetal Medicine Units (MFMU) Network, Global Network for Women’s and Children’s Health Research (Global Network), and Human Placenta Project have also supported stillbirth research and established a foundation for future studies.

The deliberations of the WG offer an opportunity to look more closely at the landscape of stillbirth research. The WG’s work will also consider the profound long-term consequences of stillbirth on the health of the parents and family. Dr. Bianchi expressed her appreciation to the organizers, planning staff, and WG members and said she looked forward to collecting the wisdom of its members to navigate the way forward.

Introductions

Lucky Jain, M.D., Chair, Department of Pediatrics, Emory University School of Medicine

Dr. Jain opened his remarks by recognizing the profound psychological effect of stillbirth on mothers and on the family, one reason that the topic of stillbirth prevention is so important. The example of Jackie Kennedy and the lifelong repercussions of the stillbirth she experienced is well known.

Dr. Jain introduced the WG members.

Scope of the WG

Natasha Williams, Ph.D., J.D., L.L.M., M.P.H., Chief, Legislation and Public Policy Branch, NICHD

Dr. Williams described the WG’s establishment and responsibility.

Background

Language in a fiscal year 2022 U.S. House of Representatives report describes the creation of the Task Force on Stillbirth (the TF). It was funded in the Consolidated Appropriations Act of 2022, which became law on March 15 (Public Law 117-103); HHS asked NICHD to lead the TF.

Goals

The House report says that the TF should include members from CDC, NIH, outside specialty organizations, and maternal-fetal medicine specialists. The TF is tasked with focusing on four topics:

• Current barriers to collecting data on stillbirths throughout the United States
• Communities at higher risk of stillbirth
• The psychological impact and treatment for mothers following stillbirth
• Known risk factors for stillbirth

Timeline

The TF should provide a report within a year of the act’s enactment on March 15, 2022. The WG will also meet on November 22 and December 9.

Report

A preliminary report will be presented to the NACHHD in January 2023, and the final report will be submitted to HHS in March.

Identification of Federal Activities

Moderated by Monica Longo, M.D., Ph.D., Medical Officer, Pregnancy and Perinatology Branch, NICHD

Dr. Longo introduced Sarah Glavin and her presentation on stillbirth publications and the NIH portfolio.

NIH Portfolio Analysis

Sarah Glavin, Ph.D., Deputy Director, Office of Science Policy, Reporting, and Program Analysis (OSPRA), NICHD

Dr. Glavin presented a detailed analysis of funding for and publications resulting from stillbirth research. In preparing the analysis, the OSPRA analysis team was guided by the four priority focus areas in the WG’s mandate.

The analysis examined three major questions:

• What are the characteristics of the overall research literature on stillbirth (regardless of funding source)?
• How has NIH funding contributed to the stillbirth research literature?
• What are the characteristics of the NIH grant portfolio on stillbirth?

To answer the first question, the analysts searched PubMed and Scopus for manuscripts about stillbirth. Finding out which NIH grants funded those publications helped answer the second question. Finally, the team pulled information from internal NIH portfolio tracking systems to examine the content of NIH grants without respect to publications (the third question).

NIH conducts this type of analysis often, and publications on a given research topic typically come from grants clearly aligned with that topic. For stillbirth, that is not the case. About half of the grants related to stillbirth, identified from a textual analysis of the specific aims, titles, and descriptions of public health relevance, did not produce publications about stillbirth. Ninety percent of NIH grants that produced stillbirth publications were not classified as stillbirth-related research grants.

Dr. Glavin described characteristics of the research literature on stillbirth. The team analyzed nearly 20,000 manuscripts published between 2005 and 2022. In that time, the number of publications grew substantially. NIH-funded publications generally made up about 7% of the total, although that share recently dropped below 5%. Most stillbirth articles were published in maternal-fetal medicine or obstetrics and gynecology journals. Analysis of the publications by subtopic showed that infectious disease and other stillbirth risk factors, as well as pregnancy conditions and complications such as preeclampsia, were common topics of research. Few studies examined grief after stillbirth. A similar pattern emerged in an analysis of the most-cited journals. The share of NIH-supported publications on key subtopics (infections, placenta, substance use, and obstetrical conditions) was greater than its share of all stillbirth publications, and NIH had a dramatically higher share of publications that were most frequently cited. Notably, funders worldwide demonstrated significant interest in stillbirth. A wide range of groups and agencies in the United Kingdom support stillbirth research; the studies examined in the analysis covered a wide range of risk factors, and many were preclinical investigations in mice and other models.

In the second part of her analysis, Dr. Glavin presented the results of NIH- and NICHD-funded research. Grants were grouped in three tiers:

• Tier 1: studies in which stillbirth is the sole or primary focus
• Tier 2: studies in which stillbirth is one of several adverse outcomes measured
• Tier 3: studies that do not independently measure stillbirth but that clearly have implications for stillbirth

In the NIH portfolio classification system, stillbirth is categorized under pregnancy. Coding used at NICHD allowed analysts to pull out stillbirth-related grants separately, but this was not possible NIH-wide. Natural language processing was used to pull terms from grant information NIH-wide and compare them with NICHD-funded grants. This resulted in many false positives; manual coding was required to assign grants to the appropriate tiers.

Of the Tier 1 and 2 grants analyzed, the vast majority (87%) were Tier 2 projects; stillbirth is most often studied together with other outcomes. NICHD funded about 75% of the NIH grants, a relatively high percentage for an NIH topic. About one-half of the NIH grants resulted in stillbirth publications; the figure for Tier 1 grants was two-thirds. Notably, many projects were funded through centers or networks.

NICHD’s two major Tier 1 network programs during the period examined in the analysis were the SCRN and the Prenatal Alcohol in SIDS and Stillbirth (PASS) Network. More than one-third of publications from these networks were published 10 years or more after the funding period for the networks, pointing to the value of sharing data and making them available for additional work for years after they were collected. Notable findings from the SCRN included studies documenting common causes of stillbirth and classifying those causes. SCRN research also highlighted an increasing focus on genetic causes of stillbirth and how to identify new ones. Results from PASS Network research confirmed the role of substance use in stillbirth and described patterns of substance use among pregnant women. NICHD has also supported networks with a broader focus that have generated stillbirth publications, including the MFMU Network, the Nulliparous Pregnancy Outcomes Study: Mothers-to-Be (nuMoM2b), and the Global Network.

The largest share of the other 24% of NIH-supported research is funded by the National Institute of Environmental Health Sciences. Projects examine environmental factors that affect a range of birth outcomes, including stillbirth. The National Institute of Mental Health (NIMH) has supported several projects on complicated grief, and a National Center for Complementary and Integrative Health study investigated a yoga intervention for posttraumatic stress disorder after stillbirth. The National Institute on Drug Abuse maintains a significant portfolio on substance use in pregnant women, and other institutes, including the National Heart, Lung, and Blood Institute, the National Institute of Neurological Disorders and Stroke, and the National Institute of Arthritis and Musculoskeletal and Skin Diseases, support investigations into the impact on stillbirth of the chronic conditions in their respective portfolios.

The number of Tier 3 projects dwarfs the number of projects in the other tiers. Over time, the number of Tier 3 projects funded each year has grown as the number of Tier 1 and 2 projects has remained steady. However, the stillbirth findings should not be considered incidental. Many findings in the 300 most-cited publications, which are on a variety of topics (e.g., environmental exposures, addiction, specific co-occurring conditions) come from Tier 3 projects. For example, one large cohort study looked at a population of young people who acquired type 2 diabetes; although the grant was not specifically on the topic of stillbirth, the study generated relevant findings because stillbirth in that population was included as a measure.

Discussion

Acknowledging active participation from parents and parent groups in the chat, Dr. Jain reflected on how NICHD has long valued the voices of parents and families; he vowed to explore opportunities to incorporate their input in the group’s discussions.

Dr. Longo presented questions from the chat. One was how many Tier 1 studies examined stillbirth prevention compared with bereavement support. Dr. Glavin explained that research on grief and treatment after stillbirth would almost certainly be in Tier 1; most of those projects were funded by NIMH. Compared with other topics, there was not a lot of published literature on grief after stillbirth. This is a gap the WG might want to consider addressing.

Dr. Longo asked whether it had been possible to examine articles on preventable and not-preventable stillbirth separately. Dr. Glavin said that this had not been done, because it would have required defining what is and is not preventable. Examining the projects investigating infectious disease and genetic causes of stillbirth would approximate that type of analysis.

Dr. Barfield asked whether it would be possible to dig deeper into social determinants of health (SDOH), especially stress during pregnancy, as a cause of stillbirth. Dr. Glavin said that the automated analysis was not well suited to picking up this kind of detail; additional time for hand coding would have been required. Research incorporating SDOH was visible in the results but quantifying how much existed was not possible. SDOH were frequently discussed in conjunction with health disparities, but SDOH are not entirely a disparities issue. A proper analysis would have to look at those separately. One study from the Global Network found a strong association between low socioeconomic status and stillbirth in low- and middle-income countries (PMID: 35972913).

In response to a question about other NIH funders of stillbirth grants, Dr. Glavin offered to generate a breakdown by institute of the approximately 25% of the portfolio not funded by NICHD.

Emily Price, a representative of Healthy Birthday, Inc., asked how NIH defines rare. Given that 65 infants are lost each day, stillbirth prevention organizations would disagree that stillbirth is rare. Dr. Glavin noted that although even one stillbirth is too many, stillbirth research must draw its findings from a large enough group of participants to say something statistically significant. Research on this topic needs to be done on a scale large enough that results will be useful for prevention. Dr. Jain concurred; from a scientific perspective, an incidence of less than 1% may be a small number, but the impact for a family is big.

Dr. Jain noted that stillbirth in pregnancy often occurs with no known risk factors. Although it may be often studied, substance use is seldom a causative factor. Dr. Glavin said not enough is known about risk factors. SCRN researchers were able to find a possible or probable cause of death in 77% of cases; for the remaining 23%, they concluded that there was no way of knowing. Some stillbirths may be from genetic causes that are not yet known.

Dr. Reddy added that in research published in JAMA, SCRN researchers identified risk factors for women having stillbirths and, taking all risk factors together, which women were at highest risk of stillbirth (PMID: 22166606). Even taking together pregnancy history, maternal age, baseline diseases, and other factors, SCRN researchers were able to predict only 19% of stillbirths. With the current state of knowledge, doctors still cannot predict early in pregnancy who is at highest risk.

Dr. Jain reemphasized Dr. Bianchi’s point at the outset that 20,000 stillbirths each year is a huge problem, and the perinatal research community is very concerned about the effect. Dr. Bianchi reflected on comments in the chat in which parents described being advised by their doctors about things such as how to change the cat litter to avoid toxoplasmosis but said their doctors never warned them about risks of stillbirth. It seems clear that the perspectives of families affected by stillbirth have not been captured in the literature. The comments and questions in the chat would be captured. Dr. Bianchi acknowledged the value of analyzing the literature but noted that, going forward, it will be important to take a broader perspective.Dr. Jain agreed that including parents’ perspectives in future WG discussions and the final report would be a priority.

Dr. Silver thanked the participating parents for being involved and communicating their concerns so passionately. He reiterated the parents’ appreciation for research examining individual causes of stillbirth, risk factors, and pathways and their request for an increased focus on studies that could have an immediate and direct effect on stillbirth. This work would include research on public health measures to increase awareness of fetal movement public health measures or institute more surveillance. Dr. Dudley said that most maternal-fetal medicine specialists discuss risk factors for stillbirth with their patients often. The average OB/GYN may not want to raise the topic with their patients so as not to alarm them. In addition, it is important not to equate cause of stillbirth with risk factors for stillbirth. It can be very difficult to determine cause of death, given the many risk factors that can contribute. Risk factors for and causes of stillbirth may be very different things.

Allie Felker, a parent who had gone through a stillbirth, said that based on her experience, the perinatal pathologist may not be well trained on how to do placental pathology and fetal autopsy, contributing to the problem of getting an accurate cause of death. She reported paying $400 out of pocket to have slides with her placental tissue analyzed by an expert. Parents may receive a fetal death certificate or stillbirth certificate that lists the cause of death only as “stillbirth.” Ms. Felker said that she was discouraged from getting an autopsy, the initial placental pathology was incorrect, and finding the true cause—cord compression—took considerable time and expense. Yet that cause was never recorded. This reflects a common experience among parents and suggests that the data that researchers analyze may not be accurate. In addition, although reduced fetal movement is not a medical risk factor, it is a common experience among those who had a stillbirth. Most parents also say they were not told about the importance of monitoring fetal movements, despite receiving information from their obstetrician about listeria, sudden infant death syndrome (SIDS), and other serious outcomes that occur less often.

Dr. Reddy responded to questions in the chat about the accuracy of the study’s conclusions in identifying common causes of death. Questions about the accuracy of the records were valid. Dr. Reddy said that when the SCRN launched in 2003, there was nothing about causes of stillbirth in the literature, and clinicians had no information to help them counsel families who experienced that devastating outcome. The SCRN study identified causes of death in 77% of cases based on a thorough workup, including state-of-the-art autopsies conducted by a trained perinatal pathologist, careful evaluation of the placenta, maternal lab tests, a review of medical charts, and interviews with the families affected, including questions about fetal movement. Research shows that these elements are important for determining cause of death; that is why the percentage of known causes in the SCRN study is so high (PMID: 32762929). In typical circumstances, not all these data sources are available.

Fetal Deaths in the United States

Elizabeth Gregory, Ph.D., Health Scientist, CDC, National Center for Health Statistics (NCHS)

Dr. Gregory, who manages the national fetal death file, explained that although the causes of fetal death are not well documented, investigators believe the deaths are associated with maternal infections, noncommunicable diseases, chromosomal abnormalities, and obstetric complications.

CDC’s National Survey of Family Growth (NSFG) estimated that in 2010 there were more than 1 million fetal losses (covering all gestational ages) in the United States, or about 17% of all pregnancies. The vast majority of these happened before 20 weeks of gestation. According to data from 2020, there are slightly more fetal deaths at gestational ages greater than 20 weeks (20,854) than deaths of an infant in the first year of life (19,578).

In the United States, vital statistics are one of only two sources of data on stillbirth. The other is the NSFG, which does not provide detailed information on geography, smaller populations, medical and health information, or reliable gestational age, all of which are available in data from the fetal death report. Studying these data can help determine what health conditions and risk factors affect pregnancy outcomes and identify preventable risks of fetal loss.

All states’ reporting requirements align with a definition of fetal death called the model law definition; most require reporting of fetal deaths occurring at 20 weeks of gestation or more (some states base their reporting requirement on fetal deaths at birthweights of 350 grams or more). Fetal death data are not available for deaths before 20 weeks.

Fetal death data were first collected in 1918, as part of U.S. Census data gathering. The first standard fetal death certificate for data collection was established in 1939. In 1974, Congress required the NCHS to collect and publish these data from all states. Since 1939, the U.S. Standard Report of Fetal Death form has been revised four times. The most recent revision, in 2003, expanded the medical and health information captured and modified the cause-of-death section to encourage certifiers to report more specific conditions.

The form for reporting a fetal death collects legal, demographic, medical, and health information about the family and cause of death that is crucial for understanding how to prevent stillbirth losses in the future. Since 2014, CDC has released cause of death data routinely.

There are many challenges with collecting fetal death data, including:

• Misclassification. A fetal death may be reported as a live birth or vice versa; abortions can also be reported as fetal deaths.
• Heartbreak and grief, which can make it difficult for staff to collect complete, accurate data
• Hospital staff’s unfamiliarity with or lack of access to necessary information
• Difficulty determining cause of death. This information is often unknown when the report is completed.
• Lack of resources for vital statistics
• Lack of a standardized electronic reporting system
• Small numbers of fetal deaths in individual states
• Underreporting of fetal deaths before 20 weeks of gestation
• Wide variability in data quality by jurisdiction

Compared with live births, fetal deaths reports are more likely to report “unknown” for gestational age (0.7% for fetal death reports versus 0.1% for reports of live birth), birthweight (7.1% versus 0.1%), date of first prenatal care visit (11.9% versus 3.9%), and gestational diabetes (5.6% versus 0.1%). There are wide discrepancies in reporting among jurisdictions. For example, in 2020, some jurisdictions reported level of mother’s education as “unknown” in as many as 69.8% of reports whereas others never did. There is also a wide discrepancy among jurisdictions in the percentage of reports that list cause of death as “unspecified” (from less than 15% to more than 85%), although among 49 states, Washington, D.C., and the city of New York, 43 areas met requirements to limit “unspecified” reports to no more than 50% of cases.

The 2003 revisions of the fetal death report are among CDC’s efforts to improve data quality. These changes included removing items that were not expected to return good-quality results, issuing standard definitions, promoting electronic registration systems, collecting data from the best sources, creating worksheets for the mother and the facility, and improving the section for reporting cause of death, which now gives instructions consistent with the World Health Organization’s International Statistical Classification of Diseases and Related Health Problems, Tenth Revision reporting.

Other efforts have included developing a decision tree with guidance on live births, fetal deaths, and induced terminations of pregnancy; a guide for providers and staff on filling out the facility worksheet; and an e-learning training module on best practices for filling out the report of fetal death. CDC also publishes the Medical Examiners’ and Coroners’ Handbook on Death Registration and Fetal Death to help accurately report cause of death, data that should reflect information learned from an autopsy or other postmortem examination.

CDC continues efforts to assess the impact of changes in data collection on quality, enhance training, and improve electronic reporting and data transfer. The agency publishes three standard reports on fetal death and occasional special reports (e.g., in response to the need for more timely data due to the COVID-19 pandemic). The second provisional fetal mortality report is expected later this year. CDC’s archive of fetal death data from 1982 to 2020 is available online; WONDER contains data from 2005 to 2020 (including data on causes of death for 2014–2020).

Dr. Gregory reported on fetal death data trends for 2020. Since 1990, the U.S. fetal mortality rate has declined 23% (from 7.49 fetal deaths per 1,000 live births to 5.74). In 2020, 20,854 fetal deaths at 20 weeks of gestation or more were reported. Since 2014, the rate of fetal deaths at 20 to 27 weeks of gestation has declined 6%, while the rate for deaths at 28 weeks and later has been stable. Fetal mortality rates vary by race and Hispanic origin:

• Native Hawaiian or Other Pacific Islander: 10.59
• Black: 10.34
• American Indian or Alaska Native: 7.84
• Hispanic: 4.86
• White: 4.73
• Asian: 3.93

There are also disparities among Hispanic subgroups, although the differences are not statistically significant. Fetal mortality rates are significantly higher among mothers younger than 15 or older than 44 and lowest between the ages of 25 and 34. They are also highest at the gestational ages of 20 to 23 weeks and at birthweights of less than 500 grams. Data show that fetal mortality rates are higher for twins and males and among mothers who use tobacco during pregnancy. By jurisdiction, the highest rates for fetal deaths at 24 weeks or more over a 3-year period were in Alabama, Arkansas, Mississippi, and Washington, D.C.; the lowest were in Connecticut, Iowa, Massachusetts, New Mexico, and Texas.

CDC data show that in 31.6% of cases, the cause of death is not specified. An additional 58% of cases can be attributed to the four most common causes: placental, cord, and membrane complications; maternal complications; maternal conditions unrelated to pregnancy; and congenital malformations. The frequency of these four causes varied across racial and ethnic groups.

Discussion

Dr. Barfield said it is important to note that changes to the form for reporting a fetal death have helped speed up reporting.

Dr. Longo shared questions from the chat asking what could be done to collect and extract more data to get more reliable information about causes of death. Dr. Gregory said that CDC developed the e-learning training for certifiers and medical examiners to improve the collection of the most detailed, accurate information possible. Dropping items that were poorly reported or did not seem improvable has improved the quality of the remaining items.

In response to a question from Dr. Leonard, Dr. Gregory confirmed that CDC is working with the Perinatal Quality Collaboratives to improve data collection.

Ana Vick, from PUSH for Empowered Pregnancy, talked about her personal experience of stillbirth. Initially, she did not get a cause of death for her son, but 6 years later, she was able to find out it was due to cord compression. She asked whether there was a way to update fetal death records so that her son’s death could be properly recorded and part of research. Many other parents have a similar experience and want to see something that can benefit other families come from their loss.

Dr. Jain said on behalf of the WG that in addressing its first charge—current barriers to data collection—the WG has an obligation to everyone who has experienced a stillbirth to rectify data collection problems. In subsequent WG meetings, members will discuss recommendations to ensure that the data collected nationwide are meaningful and lead to needed public health changes.

Dr. Silver noted that better mechanisms to get new and updated data into fetal records are needed. State health departments are keen to do this, but everyone is limited by what resources are available. Although there are many options for linking electronic records, doing so will require resources; parents could help by continuing to lobby for resources for state health departments so that modern technology can be used to collect and update data. Dr. Silver added that many other countries have done audits for fetal death the way maternal death audits are conducted in the United States. Typically, state health departments collaborate with volunteers to conduct maternal death audits, an approach that has been successful in other countries. Since stillbirths outnumber maternal deaths, it may not be possible to rely on volunteers to get it done. The WG should discuss audits as a possible solution to problems with data collection and quality.

Dr. Dudley asked who fills out fetal death reports at individual hospitals, whether these staffers get compensated, and whether they all take CDC’s online training. Dr. Gregory said that the answers vary by facility, but nurses, physicians, and birth registration specialists could fill out the reports. It is not clear whether they are compensated. The training is not mandatory; whether they take it depends on the hospital.

Dr. Jain asked how the quality of the data in the forms is evaluated. Dr. Gregory said a separate branch at CDC receives the data, does quality control (QC) to identify data that are out of range (e.g., height, weight), and contacts the state that reported the data to verify their accuracy. NCHS’s Reproductive Statistics Branch does a second QC review to look at data distributions and identify states that do not meet the reporting requirement that 50% or less of fields be listed as “unknown.”

Dr. Rasmussen noted that in some parents’ experiences, cause of death was not known because the evaluation was not done. She asked whether any studies had assessed how often the American College of Obstetricians and Gynecologists (ACOG) recommendations for management of stillbirth are followed. (ACOG states, “Evaluation of a stillbirth should include fetal autopsy; gross and histologic examination of the placenta, umbilical cord, and membranes; and genetic evaluation,” among other recommendations.) Dr. Rasmussen also suggested that getting payors to cover autopsies and other studies would help in reporting of cause of death. For example, analysis of genetic causes often cannot be done unless parents pay out of pocket.

Dr. Reddy agreed that lack of coverage dissuades many parents from requesting an autopsy, microarray analysis, or other tests, which often are expensive. She asked about the current status of stillbirth surveillance. Years ago, CDC added stillbirth to a birth defects registry compiling data in metropolitan Atlanta. Is that type of active surveillance, including medical chart review, still happening in Atlanta or any defined geographic area?

Dr. Barfield mentioned a study done in Iowa on links with birth defects and stillbirth registries through the Division of Birth Defects and Infant Disorders in CDC’s National Center on Birth Defects and Developmental Disabilities (NCBDDD). The work in metropolitan Atlanta was not as comprehensive. Some states include stillbirths in birth defects registries. It is also important to consider broader barriers to data. These events do not get the attention they deserve at the time of the event in terms of data collection. In addition, training for placental pathologists may not be readily available.

Dr. Jain asked what it would take to initiate an active stillbirth registry at CDC. He emphasized the need for a single information source to guide future research and preventive measures. Dr. Barfield said there had been work in this area previously, but additional resources would be needed to support data collection. Gathering this information is labor-intensive, like maternal mortality review. Because maternal mortality is less frequent, gathering data on it does not take as much time. A stillbirth registry would require a lot of input; there has been work done in this area, and the NCBDDD has experience in it.

Dr. Reddy said that although a nationwide birth defects registry, including a thorough evaluation, interviews with families, and other assessments, might not be feasible, launching one in a specific geographic area with a high stillbirth rate could yield the detailed information necessary to study racial disparities and other factors. Dr. Cohn reported that CDC had begun stillbirth data collection at two sites doing case-control risk factor studies of birth defects. However, expanding on this would require resources. Stillbirth data collection could be built into existing research infrastructure but incorporating stillbirth into ongoing collection of data on birth defects would take a few years and substantial resources. Dr. Jain noted that, compared with a birth defects registry, a stillbirth registry would be small. Dr. Cohn offered to run the numbers and report back at a future meeting. Although the numbers would be small, the registry would make it possible to do studies of risk factors and identify potential preventable causes. For each birth defects case, a control is enrolled, powering the study to understand more than would be possible with birth defects cases alone. Dr. Barfield suggested using the Pregnancy Risk Assessment Monitoring System (PRAMS) for stillbirth surveillance as another potentially promising approach. A PRAMS site in Utah is currently looking at stillbirth. This approach may allow investigators to explore SDOH factors. A recent study confirmed that women are willing to talk about their experiences.

Dr. Silver said that the Study of Associated Risks of Stillbirth (SOARS) program in Utah is taking a relatively inexpensive approach to gathering additional epidemiological data. Everyone participating in SOARS is offered an in-person consultation at no charge. It is a valuable source of information but is not well funded. The state health department can do only a subset of cases, whereas other countries can do perinatal audits on 100% of stillbirths. It would be challenging to go from the current level to 100%, but that is what the United States should aspire to. The WG should set a realistic goal for the next year as well as an ultimate goal and say what resources it would take to conduct an ideal evaluation in all cases.

Emily Price, representing Count the Kicks, suggested supporting a stillbirth prevention bundle, which has seen success in the United Kingdom, Australia, and elsewhere. Maternal mortality bundles in the United States have helped, and they could help with stillbirth, too.

Dr. Bianchi thanked Dr. Gregory for her presentation and reiterated the need for better data. She clarified that clinical recommendations are not the focus of the WG. In an effort to clarify expectations, she explained the research and translation continuum: Better data will inform research questions that will generate the evidence that providers and professional societies can use to change practice. The day’s presentations can help identify where the gaps are. For example, it will be important to consider how best to reach the people filling out death report forms. Targeting annual license renewal is one way to reach physicians, but the people filling out the forms are not just physicians; reaching everyone who might fill out a form requires thinking through how to reach and motivate the hospital registrar.

Dr. Jain asked, given that the report generated by the WG will go to Congress, whether there might be an opportunity to recommend setting up a registry, instituting prevention bundles, or conducting further analysis of interventions. Dr. Bianchi explained that the intent of the mandate and its assignment to NIH was to develop a research agenda. A registry could be part of acquiring data to shape the research agenda. A bundle is not part of the WG’s purview, although the WG could mention bundles in its report or communicate with other areas of the government that could make that kind of change. The WG’s recommendations will be most helpful if they focus on what needs to happen to get better data, including exploring options when parents are not given the opportunity to get a full examination done after a stillbirth.

Dr. Reddy noted that ACOG’s 2020 document on the management and evaluation of stillbirth contains a chart listing the necessary tests to gather evidence of cause of death. One outlying question is how to get providers to follow the recommendations. The ACOG document does not talk about stillbirth prevention directly but rather talks about risk factors associated with stillbirth. Research on ultrasound, placental function, and other types of monitoring are needed to better understand what causes stillbirth. Dr. Bianchi suggested that one way to approach improving data collection would be to work with the Centers for Medicare & Medicaid Services (CMS). Half of U.S. births are insured with Medicaid. It might be possible to work with CMS to explore coverage of the autopsy or other assessments to provide information for every woman insured through Medicaid.

Closing

On behalf of the co-chairs, Dr. Jain thanked the organizers, speakers, and attendees for their engagement. The day’s presentations set the stage for what was to come.

Ms. Vick expressed her appreciation to the WG for addressing the topic of stillbirth. She reflected on the experience of losing a baby and how different it is from looking at stillbirth through a research lens. When you have just lost your baby, it is difficult to think in terms of data and research. It is important that the person doing the autopsy be someone who was not involved in the loss, especially if parents believe they did not receive good care. She remembered being discouraged from having an autopsy done; her family was told that the autopsy might not yield much information, would change how her child looked, and would cost money. She said no to the examination initially, but not knowing the cause of death and wondering whether it happened because of something she did caused years of heavy grief. No one should have to live with that, so parents’ groups are willing to do anything they can do make it possible for every parent affected to have a chance to get this information.

Dr. Jain acknowledged how difficult stillbirth is. He reminded attendees that the congressional mandate includes a requirement to look at the psychological impact of stillbirth and treatment for mothers who lose a child. He related his own experience spending sleepless nights worrying about his daughters, who went through high-risk pregnancies. He assured attendees that the WG was treating stillbirth not just as scientists but as parents and grandparents with direct connection to the issues. Future generations are at stake.

Action Items

• The OSPRA analysis team will provide the WG a breakdown of stillbirth research funding at NIH by institute.
• Dr. Cohn will report back to the WG on the size of a potential study incorporating stillbirths into the registry of birth defects.