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In Iceland, Debate Simmers Over Imputed Genotypes, Preventive Medicine, and Public Health

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NEW YORK (GenomeWeb) – With the push of a button, DeCode Genetics CEO Kári Stefánsson claims he could identify everyone in Iceland who carries gene mutations linked to elevated cancer risk, and would be keen to work with partners in the healthcare sector to reach those people.

Yet questions about the ethics of alerting people to their cancer risk without their consent have so far frustrated DeCode Genetics' vision of using genetics to improve public health in the island country.

"Some ethicists might argue that people have the right not to know and that you shouldn't approach people because they might lose sleep over it," Stefánsson told GenomeWeb during a recent interview in Reykjavik, where DeCode is headquartered.

"I think they should be given the opportunity to lose sleep over it," he said.

"If someone gets lost in the highlands of Iceland, we dispatch hundreds of people to look for them," Stefánsson continued. "We don't say that because they haven't signed informed consent, we are not allowed to look for them. The tradition in our culture is to try to help people if there is a threat to their lives."

Stefánsson has led DeCode since he co-founded the biotechnology company 20 years ago. Though it was acquired by Amgen for $415 million in 2012, Stefánsson said that DeCode has been left to pursue its own discovery work, while contributing to Amgen's internal development programs. Much of DeCode's research has been based on its growing database, which includes the genotypes of 150,000 Icelanders genotyped on Illumina whole-genome SNP arrays, as well as 20,000 whole genomes sequenced using Illumina machines. And the output continues. Stefánsson said that, on average, DeCode sequences 2,750 whole genomes per month.

Given the genetic data that DeCode has amassed, genealogical records dating back centuries, and the country's population size of 320,000, Stefánsson said he is able to impute the genotypes of Icelanders who have not only not been genotyped, but even those who are deceased. "We can impute variants with a frequency down to .05 percent, so basically everything except extremely rare familial or de novo mutations," Stefánsson claimed.

That includes variants that might impact an individual's health, should DeCode be legally authorized to do so.

"All of our data is encrypted by the Data Protection Authority (DPA) in Iceland, and they hold the key to this," said Stefánsson. "If they would want to identify someone in our database, they could," he said. "We could basically with the push of a button find everyone in Iceland who has a mutation in one of the breast cancer genes, the BRCA2 gene."

Females who carry a mutation in BRCA2 have an 82 percent probability of developing a lethal cancer, Stefánsson noted, and their life expectancy is on average 12 years shorter. Men who carry the mutation are 3.5 times more likely to get prostate cancer, and are seven times more likely to die from it. "This is a lethal mutation and, particularly when it comes to the women, most of the risk can be mitigated by preventive surgery," said Stefánsson. "So why aren't we taking advantage of this today?"

In 2013, DeCode together with partners at Landspítali, the National University Hospital of Iceland, submitted a request to the DPA to permit DeCode to impute the genotypes of the 280,000 Icelanders who never agreed to take part in the company's research, and to link that data to hospital records. The DPA, however, declined the request, arguing that those 280,000 people had never consented to having their data imputed or having it stored.

Þórður Sveinsson, Head of the Legal Department at the DPA, told GenomeWeb that DeCode later sought permission to conduct such imputation internally and in a way that its imputations were not stored or accessible. That request was granted.

"The DPA was of the view that this was okay, because it did not lead to the retention of data of possible genotypes of people who have not consented to such processing of such data," he said.

The question of whether or not DeCode could approach people who were at heightened risk of developing a lethal cancer, had who had consented to being genotyped by the firm, though not consented to being contacted about health risks, has not yet been addressed to the DPA, Sveinsson said. He said that such a scheme would require some kind of national legislation.

Sveinsson noted that in 2014 the Icelandic parliament passed a law that authorized the minister of health to set regulations regarding when and how an individual who has taken part in a scientific research project should be informed about aspects regarding his or her health, but that the new provision does not address the issue of whether it should be undertaken with or without consent. "As of yet there have not been passed any rules on this," he said.

This lack of clarity has for the time left DeCode and the DPA alike waiting for a political resolution to the issue to arise, perhaps at an international level.

"The DPA is not the problem," said Stefánsson. "Their role is to protect privacy. Their role is not to launch new healthcare services. That is basically a political decision," he said. "But somehow we have been dragging our feet," Stefánsson continued. "I have been fighting for the use of this for years, and there is no question we will be using this within a very short period of time."

"I have a feeling that Iceland is not the only country in the world where the scientific community and healthcare community is wondering about these things," said Sveinsson. "Possibly, some international instruments will be made in this regard."

According to Barbara Prainsack, a professor in the department of social science, health, and medicine at King's College London, the ethical considerations raised by the Icelandic case are being debated at the international level, but the ability to impute the genotypes of the entire Icelandic population in the way that DeCode claims it is able to do distinguishes Iceland from other countries with large national genetic databases.

More specifically, Prainsack said it is possible to impute the genotypes of Icelanders in a way that the scientists involved in the UK Biobank project, for instance, would not be able to do, in part because the UK is more diverse, and because the biorepository's 500,000 samples are a much smaller fraction of the UK's 64 million population.

"The ethical challenge is not new," Prainsack, who specializes in bioethics, told GenomeWeb. "In clinical genetics, when you genotype family members, you can say in some cases with certainty that another family member will be a carrier of a certain dominant disease; however, you are not permitted to inform them of this without consent."

However, the "situation that Kari is dealing with is an unusual one," Prainsack added. "It begs the question whether instruments such as informed consent are relevant anymore. It would be conceivable to make an argument that there might be a moral duty for a government or public health authority to promote measures that could save lives, and could save costs without informed consent," she said.

"I am not making this argument but it is possible that this argument could be made," she added.

Prainsack suggested that one way to deal with the situation Iceland is facing could be to publicize the availability of such health information, and to have a system in place so that Icelanders, whether they had their risk assessed on the basis of genotyping or imputation, could approach DeCode directly and receive genetic counseling as well.

"It would be important that the initiative is taken by the person who decides to take the information," said Prainsack.

"Of course, if people themselves want to receive this kind of information, they can of course ask for it and the DPA would not prohibit people from accessing data on themselves," Sveinsson said. He noted that the DPA has in the past received demands from people that have been genotyped and requested their information. The DPA in these instances has given the answer that it does not oppose this information being given to them, he said.

"It is quite another issue whether or not this information should be given to people who have not expressed their will to receive it. That is of course different," Sveinsson added.

While the UK may be many years away from encountering the same issues under debate right now in Iceland, Prainsack did not rule out that it could happen there.

"If we move toward a society where people are genotyped at birth or sometime in their lives, then we have a similar situation to Iceland's," said Prainsack. "And if these databases are linked globally, the imputation becomes more accurate, and we will need to address the same questions," she said. "In a way, the Icelandic case is a pioneer for the things we all need to think about."