epidemiology

Redefining Inclusion: Co-Researchers with Down Syndrome Set their Own Research Agenda

Over the past two years, Eric Rubenstein and a team of SPH researchers have supported seven adults with Down syndrome as they investigate the health issues that matter to their community.

When Eric Rubenstein, assistant professor of epidemiology, convened a group of individuals with Down syndrome to guide his research on how to improve the health and wellbeing of people with intellectual and developmental disabilities, he expected they would come up with some interesting questions for his Medicaid dataset of over 120,000 individuals with Down syndrome. However, once assembled, the team had other priorities.

“We get the team together and they were pretty much like, ‘No, Eric, we’re not interested in any of that. That’s not important to us. We want to know about how people [with Down syndrome] cope, mental health, and strategies to reduce stress’—which, as cool as my Medicaid data are, [the data] does not really have information on that,” says Rubenstein. He first formed the seven-member team to serve as an advisory board to his study DSTOTHEMAX, which aims to further understand the causes of obstructive sleep apnea, dementia, and mortality among individuals with Down syndrome.

He realized, he says, that there is often a disconnect between what researchers choose to focus on and what people with intellectual and developmental disabilities care about.

“[This] is especially important for something like Down syndrome where the big boogeyman is dementia,” he says. By age 40, most adults with Down syndrome will show signs of Alzheimer’s disease in their brains. While research into the cause and treatment of dementia is therefore crucial, Rubenstein notes that many other research questions, such as what kinds of mental health concerns people with Down syndrome face, might be overlooked because of the emphasis on this significant public health crisis. Talk to a 22-year-old with Down syndrome who just finished high school, for example, and dementia is probably not their greatest concern day-to-day, he says.

Rubenstein instead supported the team in redefining their purpose to include investigating the health issues that that they deem important to their community. The approach follows an emerging model of research known as co-research that deliberately incorporates community members at every step of the research process, rather than simply as study subjects. In 2022, together with SPH researchers, the seven individuals Rubenstein recruited for advice—Staci Christensen, Gus Edouard, Benjamin Golden, Eden Rapp, Kaethe Sigelko, Alexis Sokoloff, and Caley Versfelt—formed the country’s only public health co-research program led entirely by people with Down syndrome.

“In qualitative research especially, you bring a lot of your own personal experience into the analysis. You use yourself as a research tool. That is why it is so unique to have the co-researchers alongside us, interpreting the data in real time, because they bring their own lived experience,” says Ashley Scott, a project manager in the Department of Epidemiology who works closely with Rubenstein and the co-researchers.

The co-research team meets about once per month via Zoom, she says, since its members come from all over the country. They each bring their own experience with self-advocacy and Scott and her team provide training in health research and ethics, as well as host guest researchers and health professionals to teach research communication. Through guided brainstorming sessions, Scott and the co-researchers choose a research topic, develop research questions, and decide on a research method.

For the team’s first research project, they focused on mental health, creating an online survey on stress, anxiety, and coping mechanisms for adults with Down syndrome. Their findings are documented in a forthcoming academic paper accompanied by an easy read format for increased accessibility. The team is now conducting qualitative interviews for their second research project on what it means to be independent as adults with Down syndrome.

“Within the world of diversity, equity, and inclusion in academia, people with intellectual disabilities are not always included because of the necessary degree requirements to be part of this community,” says Rubenstein, whose interest in disability health research stems from a long history of working closely with people with intellectual and developmental disabilities as a Special Olympics coach. “We have had folks speak at the American Public Health Association [annual meeting], the MELODEM Dementia Conference, Massachusetts Down Syndrome Congress—these Down syndrome events [that] often do not have the folks with Down syndrome speaking at them. We are trying to open up more avenues for real inclusion in research teams. It is a cliché at this point, but the disability rights motto is ‘Nothing about us without us.’”

Recently, Rubenstein, Scott, and Salina Tewolde, a senior data analyst supporting DSTOTHEMAX and the co-research program, attended the fifth International Conference of the Trisomy 21 Research Society in Rome, Italy (Down syndrome is also known as Trisomy 21 in reference to the extra copy of chromosome 21 that someone with the genetic condition has in their cells). Because they could not afford to fly out their co-researchers to join them, they presented a poster about the co-research model and recorded a video of the co-researchers discussing the importance of the model to them.

While the conference focused heavily on the science of Down syndrome at the genetic and cellular levels, Rubenstein, Scott, and Tewolde shared their population health research at a special session on science and society that was intended to be more broadly accessible. The session, which was open to the public, attracted families from around Rome and fostered direct exchanges between researchers and the Down syndrome community, says Rubenstein.

In the future, he hopes to assist other researchers with starting their own co-research teams by developing a hub-and-spoke model of trainings and resources with a home base at SPH. He plans to use an Early Career Catalyst Award from idea hub, SPH’s innovation accelerator, to conduct an evaluation of the co-research model and to pave the way for additional grant applications.