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ASHG Panelists Discuss Interest in Sequencing-based Newborn Screening

BOSTON (GenomeWeb Daily News) – While panelists at a session at the American Society of Human Genetics annual meeting on Saturday said that the public is cautiously interested in including whole-exome or whole-genome sequencing as part of newborn screening efforts, there are concerns regarding privacy as well as the technologies' ability to meet the technical criteria required for such public health screening.

Newborn screening is "internationally recognized as a public health tool and is immediately useful for the infant," said Olaf Bodamer, a medical geneticist at the University of Miami Miller School of Medicine, at the ASHG session.

Recently, the tests — typically conducted by analytical chemists — included on those panels have expanded and could incorporate even more diseases if either whole-exome or whole-genome sequencing is adopted.

The US National Institutes of Health has funded four studies to the tune of $25 million to study the medical and ethical implications, among others, of sequencing the exomes or genomes of newborns at birth.

Parents are generally interested in knowing what information genome sequencing may provide about their children's risk of disease, according to a study presented by Aaron Goldenberg, a bioethicist at Case Western Reserve University. They examined the baseline interest of parents in the US of genome sequencing as part of newborn screening.

He and his colleagues surveyed some 1,540 parents with at least one child under the age of 18 at home. Half of the survey population was asked whether they'd be interested in a genomic sequencing screen as part of the mandatory state-based newborn screening process. The other half was asked whether they'd be interested in genomic sequencing screening conducted at the pediatrician's office.

Both sets of parents exhibited similar rates of interest —74 percent and 70 percent, respectively, said they were interested or somewhat interested — in genome sequencing as part of newborn screening.

"There was clearly a high level of parental interest," Goldenberg said.

A number of factors correlated with the parents' interest in genome sequencing-based newborn screening, such as education level, plans to have another child, or having a child with multiple health issues.

The respondents also indicated that their interest was influenced by the accuracy of the test, whether it would help prevent or decrease the risk of disease, and privacy. Indeed, when Goldenberg and his colleagues asked how interested parents would be in a state-based genome sequencing newborn screening program that saved de-identified samples for research purposes, interest declined — just under 65 percent said they'd be interested in such a program. He and his colleagues published this work in Genetics in Medicine in June.

A similar study conducted in Canada, and presented by Yvonne Bombard from the University of Toronto, examined the wider public's view of genome sequencing as a newborn screening tool and found some reservations.

She and her colleagues surveyed some 1,213 people from across Canada about their willingness to participate in the current newborn screening programs or in a hypothetical genome sequencing-based newborn screening program and about parental responsibility to do so.

While Goldenberg's study aimed to establish a baseline attitude and gave a limited overview of genome sequencing and what it could find, Bombard and her colleagues included an educational component in their survey about sequencing technologies.

They found that respondents were less likely to choose newborn screening with a genomic component, and were less likely to consider parents who opted out of such screening irresponsible.

"The willingness to participate in newborn screening decreased for genome sequencing compared to current technologies," Bombard said, adding that the "integration of genome sequencing into newborn screening might reduce participation."

The tools themselves might not be quite ready to be put to such a task, the University of Miami's Bodamer added. In general, he said that screening technologies have to meet certain technical criteria, such as having high-throughput and multiplex capabilities, short turnaround times, high specificities and sensitivities, stable and automated platforms, and low per-sample costs.

The University of Utah's Jeff Botkin, a medical ethicist, added that the tools have to be able to handle, samples from, for example, the 4 million babies that are born each year in the US, or the 700 born each day in New York or the 1,500 born each day in California.

Botkin also argued that the cost could quickly climb too high. Assuming a cost of $1,000 per genome, that would translate to $4 billion a year for sequencing, he said, even before adding in the analysis and follow-up testing costs.

"Genomic technologies … are not ready yet," Bodamer said.

Further, Botkin noted, screening programs traditionally have focused on conditions whose early identification would benefit the child.

An expanded panel of diseases that would easily be able to be included in a sequencing-based test — such as for diabetes or cancer or cardiovascular disease risk — would shift away from that traditional approach and lead to ambiguous information of uncertain benefit, Botkin added, also saying that there is not much evidence that genetic data is more sensitive or specific than current testing approaches.

Currently, Bodamer noted, there is also a lack of good evidence for the inclusion of a number of diseases on the newborn panel that are tested through traditional means.

"I think sequencing millions of asymptomatic babies is probably not the best use of our resources," Botkin said, later adding that "sequencing makes enormous sense as a research tool."