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ACMG Panel Discusses Potential, Pitfalls of Precision Medicine Following Newborn Screening

SALT LAKE CITY — Personalized medicine has the potential to address issues of access and equity in healthcare but also the ability to exacerbate them, according to a panel at this year's American College of Medical Genetics and Genomics annual meeting.

"There is so much excitement" surrounding genome sequencing, said Luca Brunelli, professor of pediatrics and director of the neonatal genomics program at the University of Utah, during a meeting session on personalized medicine on Friday. "There is the idea that somehow genomic technologies can do more than cure the single patient; they can really mend these inequities that we have in healthcare."

The panel discussed the potential and pitfalls of genomic and personalized medicine in the context of newborn screening. Brunelli noted that, just a few years ago, there was a lot of enthusiasm as to how genomic tools could be used in the NICU, as rapid and ultra-rapid sequencing were being used to suss out conditions affecting ill infants. Now, he said, that excitement has shifted toward newborn genomic screening.

"But I think we should step back for a second and ask whether newborn genomic screening is by itself sufficient to be the key to saving lives, reducing disparities across the life course," Brunelli said.

In the US, newborn screening varies state by state, though most states test for dozens of rare conditions that affect infants, ranging from amino acid disorders to cystic fibrosis and hearing loss, and there has been increasing interest in folding in genome sequencing to complement those efforts.

Panel member Amy Brower, co-principal investigator of the Newborn Screening Translational Research Network (NBSTRN) at the National Institute of Child Health and Human Development (NICHD), said, though, that screening to identify infants with rare conditions, or at risk of them, is just one part of the process. Many of the conditions screening programs search for are associated with lifelong care, which needs to be accessible, she said.

Brunelli further cited two sociologists who examined the effect of state-mandated screening on infant health outcomes and mortality in the 1960s, 1970s, and 1980s. That analysis, he said, found that implementing screening programs had no effect on infant mortality but instead led to greater health inequities. That changed, he said, when Medicaid was overlaid atop newborn screening efforts.

"Having access to healthcare … is really critical for newborn screening to have a positive effect on children. Without it, you are not reaching your goal," he added.

For newborn genetic screening in particular to have a positive effect on all children, medical genetics needs to be inclusive, said panel member James Tabery, a professor of philosophy at the University of Utah. Participants in genome-wide association studies are predominantly white, he said, as an analysis in Nature in 2016 found that 81 percent of GWAS samples were from individuals of European ancestry.

The field is aware of that disparity, and researchers often note that increased sample diversity is needed, Tabery noted. But scientists need to be mindful of the strained history different groups have with the medical profession and need to work to earn communities' trust.

Tabery also said that acquiring diverse data does not necessarily mean that benefits, like drugs, will then flow to those communities. "Even if you get diverse data, it isn't like you just snap your fingers and suddenly you've got cures and treatments for the people who are in that dataset," he said.

Any disease-linked variants have to be prioritized for follow-up, which may be influenced by social and other factors and exacerbate inequalities, he said. As an example, he pointed to sickle cell disease, which predominantly affects Black individuals and is considered the first molecular disease identified, and cystic fibrosis, which predominantly affects white individuals.

"Black Americans were happy to participate in sickle cell research," he said, but did not benefit from as much investment in disease research, even though the disease was discovered early. "When you go out to the world and try to turn [a genetic disease] into actual treatments, cystic fibrosis fares much better because white people for all sorts of historical reasons have a lot more money than Black people," he said.

Bowser added that both sickle cell disease and cystic fibrosis are included on most newborn screens. Data collected from these has shown that there are differences in health outcomes across the US depending on access to care, quality treatment, and family support for these two conditions.

Further, even if drugs are developed and approved for rare genetic disorders, they can be prohibitively expensive, added Tabery. For instance, Novartis' Zolgensma (onasemnogene abeparvovec-xioi) was approved by the US Food and Drug Administration in 2019 for children with a certain form of spinal muscular atrophy. The treatment was announced with a list price of $2.1 million.

According to Tabery, high prices are often attributed to inefficiencies in the drug development process. But he also argued that the high prices are a direct result of how patient populations are fragmented for personalized medicine. This, he said, creates market segmentation and leads to increased costs.

"[If you have] a patient population of people with lung cancer, and rather than giving them all the same thing, you're going to chop it up in the 15 different genetic groups and give them 15 different things, the cost of each of those things is going to necessarily go up," he said.

Brunelli added that these questions of accessibility and equity are ones that the genomic community needs to address as sequencing is folded into newborn screening.

"If you just think about the discovery, the ultra-rapid whole genomes, the latest treatment, genomic treatment for that disease, you are just looking at part of the puzzle," he said.

"We need to find a way to make … children, all children, better, not just a few," he added.

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