Challenges and Opportunities in Using Residual Newborn Screening Samples for Translational Research: Workshop Summary (2010)

As Sharon Kardia observed, people tend not to know much about newborn screening programs. They also know very little about how residual dried blood spots are used after they are collected and tested. The fact that newborn screening has occurred largely “under the radar” is a vulnerability, Kardia said. Many researchers will forego studies using residual newborn screening samples if the pressure from the public gets too high. Yet the research opportunities are so great that efforts need to be made to bridge

the divide between the research community and the public. According to Kardia, educating the public about the public health infrastructure will be necessary so that people will be informed about the collection of data and the way that this information is used on their behalf.

The parents and others who have raised questions about this topic are not necessarily anti-science, Kelly Edwards noted. The Havasupai people were deeply interested in health research that was going to benefit their community. Many of the Texas parents involved in the lawsuits were also supportive of research. They were angry because they were not asked before samples were used outside of their original scope or intentions.

Edwards participated in a working group of the Trust, Integrity, and Ethics in Science (TIES) Project, which has convened representatives from a number of industries that require the public’s trust (Yarborough et al., 2009). Rebuilding trust that has been lost involves two basic actions, she said: fostering multiple types of relationships and developing accountability practices that exceed those required by external regulators. In particular, in building relationships it is important to engage the public proactively rather than in response to an adverse event. “Be out there early and often so they know you and you are a trusted face.” Then, if something does happen, people will be willing to listen to what you have to say.

Screening programs need to direct their educational efforts to healthcare providers as well as the public. “The average pediatrician in his or her office today and the average obstetrician knows little, if anything, about this program,” Alan Fleischman said. Pediatricians may appear to be somewhat unconcerned about initial positive results from newborn screening because they know that true positives are relatively rare. One consequence is that health-care providers are unprepared when they encounter a negative response about screening programs from patients. Fleischman added that ways need to be found to provide education to health-care providers without burdening obstetric or midwife professionals.

State legislators, some of whom have expressed considerable suspicion and distrust about newborn screening programs, should be another target for education, Fleischman said. But Anne-Marie Comeau countered by noting that legislators are responding to what they see as pressure from constituencies. Education that is targeted at legislators, unless complemented by public education that defuses political pressure, may not do the job, she said.

Only eight of the states whose laws and regulations could be identified require that information provided to parents regarding newborn screening programs discuss the storage and use of residual dried blood spots, said

Alissa Johnson in her review of state policies and programs. Any education for parents should cover not just how the samples are used but the screening program in general, Michele Caggana said. “When we talk to parents and they get an understanding of the entire program, it really does help them to say, ‘Okay, this is helping my child, it’s helping other kids.’” Many different forms of parent education are possible, Kardia said. Public service announcements can be effective and new ways of educating community members about the process of obtaining consent offer great promise. Caggana observed that one effective approach is to describe state policy in a very brief form in either a brochure for parents or on the screening form itself. In New York State, the brochure that parents receive has contact information and gives parents a number to call if they want to opt out of any research use or have a screening specimen destroyed. Kenneth Pass added that one-time programs are not effective. Education has to be a continuing process, he said, “because the babies are continuing to come.”

Caggana also pointed out that coverage of newborn screening programs in the media can have both positive and negative consequences. Media coverage has helped the New York State Department of Health’s Wadsworth Center review and formalize its policies, she said. “It helped us to write things down and make sure that we were doing things to the best of our abilities. And it opened dialogues between the parents in our state and the screening program.” Caggana always makes sure to invite parents to visit when they call her department and parent groups, genetic foundations, and advocacy groups have all expressed interest in visiting the facility.

As an example of a system that could generate considerable good will among parents, Caggana pointed to a familiar problem: getting a child’s immunization chart for a school, summer camp, or extracurricular activity. If parents had access to the immunization registry in New York State, they could go online, print out their child’s chart, and deliver it wherever it was needed. “[Parents] would not have to call the pediatrician, go over there, drop the form off, go back, and pick it up. On that very simple level, this could only result in a useful thing for parents.” Kardia added that such a system would also help parents understand the public health system by involving them directly with it. This is important because parents have many ideas about newborn screening that are seriously off base. “When we went to these town halls, the first half of the conversation was about the conspiracy theories that they had,” Kardia said. “They were deep and they were wide, and they were getting nods from across the room in terms of what was happening and why it was happening. It was a huge wake-up for me.” If parents had access to their children’s immunizations, Kardia said, it might generate a lot of social capital.

Ann Waldo urged that the benefits of newborn screening programs be made more transparent, with more visible and tangible links to parents.

Perhaps a newborn screening program could send information directly into an individual’s public health record, or maybe a blood spot could be reserved indefinitely for possible future uses that could improve an individual’s health.

Comeau agreed that providing the public with services that they see as beneficial would be useful to newborn screening programs. But she cautioned that the first priority of these programs still needs to be newborn screening.

Kardia asked where the money will come from for education. Ensuring that research can continue requires education, but finding funds for education is difficult. Several speakers pointed out that newborn screening programs have a common interest with research institutions, universities, biobanks, and other organizations in assuring and explaining the trustworthiness of the research enterprise. Partnerships directed toward broad public education campaigns could have specific benefits for newborn screening programs.

The states may need some help from the federal government to set up the necessary infrastructure for communication, Fleischman said. Resources are not easily obtained within the states to offer educational programs, enhance transparency, and strengthen oversight.

In addition, new and creative approaches to education are needed. For instance, Facebook or Google ads could let people know about the existence of newborn screening, since so many parents do not know that their babies were screened. Baby expos could be used to reach out to prospective and new parents. Viral networks, social networking sites, and other new media can enhance transparency and education. “It will be much, much easier than trying to tell us something with billboards,” Sharon Terry commented.

Parents have a limited motivation to absorb this information, Comeau said, particularly when the information concerns a program that will find only rare instances of harmful conditions. “To spend a lot of money and time designing specific newborn screening [educational materials], I think all of us have been through that at some level, and I am just not sure about [its effectiveness].”

Clayton said that the period of labor and delivery is the wrong time to try to educate pregnant women about newborn screening. A much better time would be during pregnancy. Obstetricians may object that they are too busy to provide such education, but “that’s nonsense,” Clayton said. “If we’re going to talk with parents or potential parents about newborn

screening, the time to do it is when they’re prepared to listen, which is not in the 24 to 48 hours postpartum.” And even prenatal education is not enough, Comeau added, because 20 to 30 percent of pregnant women do not access prenatal services.

The time lines for education and for research are different, Terry pointed out. Research can take a long time, whereas decisions about newborn screening often have to be made quickly. “How do we both keep up the energy and the urgency but also temper it, knowing that we need to be mindful and thoughtful?” Terry asked. The approach she urged is one of tempered urgency. Virtual communities might accelerate social change, but the “people part” of change still tends to be slow.

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