A Sixteen Year Retrospective of Stillbirth Assessment

Richard M. Pauli, M.D., Ph.D.

Origins
It is often hard for me to conceive that the Wisconsin Stillbirth Service Program (WiSSP) is soon to enter its third decade. Most of you who have helped maintain it were likely uninvolved in the early years. Some of you were children yourselves! This In Depth, then, is a way for us to describe for you the history of the Program, to summarize what we have learned, and to speculate about what the future might hold.

My personal involvement began with the death and birth of our son, Zachary, in 1979. Although trained in pediatrics and medical genetics, I was, before that, only vaguely aware of the occurrence of stillbirth, and had no understanding either of its personal impact on families or of the role that etiologic assessment could play in helping families experiencing the death of a baby. In addition to personally grieving our loss, I set out to learn what was known about the causes of stillbirth (particularly since no cause was discovered to explain Zachary’s death). While there was some exceedingly insightful litera-ture concerning parental grieving (much of it at that time from England rather than this country), I was surprised, and a bit appalled, at how little had the causes of stillbirth been studied. Furthermore, it was clear that those studies that did exist were selective, anecdotal and/or biased, and so gave no true reflection of the causes of intrauterine death in general.

Shortly thereafter our family moved to Madison. As a young, enthusiastic and naive Assistant Professor, one of the tasks I set upon was to develop a model for stillbirth evaluation. With the encouragement of colleagues at the Clinical Genetics Center, the rudiments of the model we still use were generated: a non-hierarchical, protocol-driven, University-community collaborative program. By 1981 it was ready to go.

Then came the ‘time of troubles’. Initially we sought funding for an experimental model program (involving around 10-30 hospitals) with an emphasis on research design and rigorous analysis of outcomes. A proposal to the National Institutes of Health was summarily rejected as unworkable and bound to fail. Application to the March of Dimes met a similar fate — I was not only told that I could never expect local hospitals to collaborate on such an endeavor but was asked why I was wasting my time on such a ‘dull, backwater’ issue when I could be doing something exciting like fetal surgery!

Establishing the Wisconsin Stillbirth Project
Fortunately, an alternative approach was found. The research focus was transformed into a primarily clinical service focus (both endeavors were always meant to be inextricably linked) and funding was obtained through the Maternal and Child Health competitive block grant process in 1982-83.

Then the hard work began. Protocols were generated, not too dissimilar to those now in use. What seemed to be a never ending round of continuing education trips began. I visited the larger hospitals around the State while genetics counselors (particularly Casey Reiser, who is now Associate Director of the Program, and Ruth Lebovitz, who is now a pediatrician herself) travelled to many of the smaller birthing sites. Through those first few years (phase 1 and phase 2 of what we then termed the Stillbirth Project) we were again and again impressed by both the goodwill of virtually all of the nurses and physicians with whom we met and their (sometimes reluctant) willingness to accept that the University had something of value to offer.

An unmet need was clearly present. Referrals began. Diagnoses were generated. Families were helped. The Project’s success was best measured by the continued use of the protocols and the continued referrals. Soon we reached a steady state of about 100-110 assessments each year — in the 30-50% range of compliance for the part of the State we had committed to serve. Compliance of 100% would have been a lot more satisfying, but at least we knew that over time many families had received information and help who otherwise would not.

Project to program
I had always intended that this would be a model project — a project with a defined beginning and a defined end! Sometime in the late 80s we all recognized that the need was not going to go away, so how could we arbitrarily discontinue offering the service? So long as some method of funding could be found, we decided to persevere — and at some moment, now only vaguely recalled, the time-delimited "Project" became the ongoing Wisconsin Stillbirth Service Program. So, too, there was a recommitment to our primary role of helping families and the professionals who help care for them, and so the Wisconsin Stillbirth Project became the Wisconsin Stillbirth Service Program.

Lesson from 16 years of stillbirth assessment
In reviewing the core service component of WiSSP, I think that three lessons stand out. First, University-community collaboration is possible and desirable. The Wisconsin Idea — "I shall never rest content until the boundaries of the University are coextensive with the boundaries of the State" — is not a particularly well rewarded point of view at the University right now. Certainly it is not in great favor at the Medical School where far more emphasis is placed on bottom line, financial needs. Nonetheless, I hope that this sense of a larger community can somehow persist until the current convulsions of academic medicine subside. Secondly, the WiSSP model for stillbirth assessment has worked. Since its inception, 1723 referrals have been made to us. Compliance for each component of the protocol remains at or near 90%. Diagnoses have been established in 43% of those referred and, of all referrals, 27% were found to have a fetal diagnosis (i.e. birth defect or genetic process that explains why the baby died). Finally, it became evident that if we only offered diagnostic services on a case by case, family by family basis, then we could not possibly reach or help the tens of thousands of other families experiencing stillbirth throughout the country nor could we hope that other health care providers could assume the needed role of local expert.

Expanding the concept of a service program
Beginning in 1988 we recognized that use of alternative media (rather than only face to face continuing education) was essential. Our first, rather primitive, continuing education videotape was produced at that time (it was little more than a recording of a lecture but seemed better than nothing). With the help of a small grant from the Perinatal Foundation (which also has been crucial in other educational activities we have undertaken) a more professional, five part videotape series was generated in 1995 and remains in use in hospitals throughout Wisconsin; copies have also been used in various locales throughout the world.

This newsletter was begun in 1994. From the beginning we wanted it to be substantive and to contain information that would be of enduring value to those helping to care for families who experience stillbirth. While I think that it has succeeded, we remain disappointed that its readership remains as limited as it is (about 300 subscriptions) and that we receive precious little feedback from our readers.

WiSSP was only a bit behind the curve in recognizing that we are in the age of web-surfing. Here, too, our intent was ambitious — a factually substantive website that could serve the needs both of health care providers and of families. www.wisc.edu/wissp has had about 8000 hits from 55 countries since its inception in October 1997.

Last year also saw publication of two brochures that have been distributed throughout the State and nationally. We saw a need for fact based brochures, to com-plement bereavement based materials that have been available for some time. These brochures include "Helping when the Least Expected Happens" for the general public, and "When Your Baby Is Stillborn" for parents who have experienced the stillbirth of an infant. You can preview these two brochures on our webpage.

More mundane service and education activities continue, including developing and maintaining a lending library of materials pertaining to parental bereavement (with a complete annotated bibliography), and maintaining a library of all scientific/medical literature on stillbirth.

Measures of benefit are harder with such activities than for direct patient care. We are throwing out information in the wind and hoping that it somehow reaches someone who needs it. While we continue to try to measure the effects of various outreach activities, over time I have become less wed to the need for direct demonstrations of benefit.

Exporting the idea
Ideally all families should have available a stillbirth assessment program. My greatest disappointment to date is an inability to successfully export our ideas nationwide. First, it requires one or more committed individuals with expertise in genetics/pathology/obstetrics who can act as the organizers as well as the interpreters of the gathered data. Secondly, changes in health care financing are making it always harder to generate a new, non-remunerative program in any setting. Because of the latter, we are currently developing an approximation of a cost benefit analysis that may be sufficiently convincing that managed care organizations will be forced to fund etiologic assessment of stillborns on its basis. Third, resistance will be met unless stillbirth evaluation is viewed as an ordinary, expected part of standard care. In this state that is, to a large extent, a reality, I think. Elsewhere more forceful initiatives are needed. The American College of Medical Genetics will have a guideline for care forthcoming soon (it had better, since I am chair of the committee writing it!).

What has been learned
The research arm of WiSSP has generated data (published in 6 peer reviewed articles) that have helped define the manner in which stillborn babies should be evaluated and the tremendous heterogeneity of cause of stillbirth that is present (see publication list, below). We have, I think, demonstrated that the NIH and March of Dimes were wrong — collaborative assessments can be done even on a rather large geographic and institutional scale. I suspect that many in the bereavement community have a different, fuller view of stillbirth through contacts with WiSSP (as we have learned enormously from them).

What next?
From a scientific perspective I anticipate major advances in our understanding of intrauterine death over the next decade. I suspect these may come from three avenues of investigation. First, there are a host of lethal genetic processes in other mammals that have been incompletely investigated. Inquiry into these processes using developing molecular technology may yield insights applicable to human stillbirth, too. Secondly, we still don’t know how much (or how little) newly recognized, novel genetic mechanisms in humans, such as microdeletions, uniparental disomy, etc., contribute to intrauterine death; I suspect these may be more important than is currently demonstrable. Third, I can not imagine that progress will not be made in both understanding non-genetic causes of stillbirth and in generating prevention strategies.

I am far less sanguine about whether assessment, counseling and research regarding stillbirth will be viewed as a funding priority in any sector, public or private. And I don’t know what to do about that...

Publications to date of the Wisconsin Stillbirth Service Program:

Greb AE, Pauli RM, Kirby RS: Accuracy of fetal death reports: Comparison with data from an independent stillbirth assessment program. Am j pub health 77:1202-1206, 1987.

Luebke HJ, Reiser CA, Pauli RM: Fetal disruptions: assessment of frequency, heterogeneity and embryologic mechanisms in a population referred to a community-based stillbirth assessment program. Am j med genet 36:56-72, 1990.

Pauli RM, Reiser CA, Lebovitz RM, Kirkpatrick SJ: The Wisconsin Stillbirth Service Program: I. Establishment and assessment of a community based program for etiologic investigation of intrauterine deaths. Am j med genet 50:116-134, 1994.

Pauli RM, Reiser CA: The Wisconsin Stillbirth Service Program: II. Analyses of diagnoses and diagnostic categories in the first 1000 referrals. Am j med genet 50:135-153, 1994.

Pauli RM: Lower mesodermal defects — a common cause of fetal and early neonatal death. Am j med genet 50:154-172, 1994.

Dasgupta NR, Pauli RM, Horton VK, Reiser CA: Validation of radiographic criteria for the diagnosis of Down syndrome in stillborn infants. Am j med genet 72:347-350, 1997.

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