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The best interests of the child and the return of results in genetic research: International comparative perspectives

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Abstract

Background Paediatric genomic research raises particularly challenging questions on whether and under what circumstances to return research results. In the paediatric context, decision-making is guided by the best interests of the child framework, as enshrined in the 1989 international Convention on the Rights of the Child. According to this Convention, rights and responsibilities are shared between children, parents, researchers, and the state. These "relational" obligations are further complicated in the context of genetic research. Discussion A comparative review of international, regional and national documents on the return of research results reveals that there is a dearth of normative documents in the paediatric context. The best interests of the child framework is increasingly complicated by a growing appreciation of pediatric autonomy and the development thereof; parental rights (particularly when parents are affected by the genomic information of their children); and the right not to know. Summary This comparative analysis reveals that policy-makers and legislators have responded to the above challenges in different ways. Nevertheless, in Europe as well as in Canada, there is an emerging trend towards making the return of certain results mandatory in the paediatric context, should this course of action prove to be in the best interests of the child.
D E B A T E Open Access
The best interests of the child and the return of
results in genetic research: international
comparative perspectives
Man H Zawati
*
, David Parry and Bartha Maria Knoppers
Abstract
Background: Paediatric genomic research raises particularly challenging questions on whether and under what
circumstances to return research results. In the paediatric context, decision-making is guided by the best interests
of the child framework, as enshrined in the 1989 international Convention on the Rights of the Child. According to
this Convention,rights and responsibilities are shared between children, parents, researchers, and the state. These
relationalobligations are further complicated in the context of genetic research.
Discussion: A comparative review of international, regional and national documents on the return of research
results reveals that there is a dearth of normative documents in the paediatric context. The best interests of the
child framework is increasingly complicated by a growing appreciation of pediatric autonomy and the
development thereof; parental rights (particularly when parents are affected by the genomic information of their
children); and the right not to know.
Summary: This comparative analysis reveals that policy-makers and legislators have responded to the above
challenges in different ways. Nevertheless, in Europe as well as in Canada, there is an emerging trend towards
making the return of certain results mandatory in the paediatric context, should this course of action prove to be
in the best interests of the child.
Keywords: Return of research results, Best interests of the child, Paediatrics, Genomic research, Convention on the
Rights of the Child, Childrens rights
Background
An individual research resultis defined as a finding
concerning an individual research participant that has
potential health or reproductive importance or personal
utility and is discovered in the course of research [] in
meeting the projects research aims[1]. In recent years,
discussions surrounding the topic of the return of such
results in genetic research have mainly focused on whether
individual findings should be returned to participants and,
if so, under what conditions [2]. The increasing availability
and affordability of next-generation sequencing has made
this debate all the more pressing [3].
That being said, little attention has been given to the
challenging issue of return of results in paediatric genetic
research, or to the complex dynamics of decision-making
in this context. Indeed, while the best interests of the
child(BIC) framework remains the foundation for
decisions concerning children in research, these decisions
are shared between 1) parents, who, as a matter of law,
are authorized to speak for their children, guide their
actions and determine their healthcare; 2) researchers,
who have an obligation to conduct research according to
professional norms; 3) children, whose views become
increasingly important as they mature; and finally, 4) the
State, which has the power to intervene in cases of neglect
and to legislate on matters relating to the participation of
children in research. In the case of paediatric genetics,
where research results may not be actionable during
childhood or may have implications for parents, siblings
and other relatives, the best interests of the child
framework is all the more complicated. What role do
the aforementioned stakeholders play in the decision
* Correspondence: man.zawati@mcgill.ca
Centre of Genomics and Policy, McGill University, 740 Dr. Penfield Avenue,
Suite 5200, Montreal, Quebec H3A 0G1, Canada
© 2014 Zawati et al.; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative
Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and
reproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain
Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article,
unless otherwise stated.
Zawati et al. BMC Medical Ethics 2014, 15:72
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to return paediatric research findings? What are the
general principles that guide such decision-making
processes? What are the responsibilities of the various
actors involved?
Despite the fact that children have been involved
in medical research for hundreds of years, pediatric
participation has been consistently overlooked in
guidelines and policy-related documents [4]. In recent
years, however, the international community has reached
a consensus [4] on the importance of including children
in research. Accordingly, this text will undertake a
comparative analysis of international norms relating
to the return of results in paediatric genomic research.
First, this text will examine general principles as enshrined
in the 1989 Convention on the Rights of the Child [5]
(CRC) (Section II.A). Second, it will review principles in
regional (European) normative documents as well as
professional norms (Section II.B). These norms, when
adopted by professional bodies, often play a role in
determining standard of care. Findings from Sections
II.A and II.B will allow us to comprehend how these
principles and professional norms are applied to the
return of research results in paediatrics for select common
and civil law countries (UK, France, the Netherlands,
Spain and Canada)
a
(Section II.C). Finally, we return to
our international comparison in order to critically evaluate
the future of the return of results in the context of
paediatric research (Section II.D).
Methodology
The international documents referenced in this text were
collected using the HumGen International Database
(http://www.humgen.org), a database of international,
national, and regional guidelines and policies specific
to human genetic research. In order to narrow our
search, the keywords minor/childand communication
of resultswere used to identify those documents that
were most relevant and important. Keywords such as
research resultor incidental findingswere not avail-
able. All organizations were selected, and no limitations
were set as to jurisdiction. This provided a large selection
of documents. Only English documents dating from 1990
to 2013 were queried. This search generated 145 results,
of which approximately 20 were selected based on their
level of relevance to this text.
Case law was retrieved using Quicklaw, a legal database.
For the purposes of our research, the keywords research
and best interests of the childwere used. This search
generated 933 results; these results were then narrowed
based on their relevance to the best interests of the child in
medicine. Legal doctrine was retrieved using HeinOnline, a
database of legal journals. The keywords return of results,
genetic researchand childrenwere used to facilitate the
search process. This search yielded 25 results.
Discussion
International norms: general principles
There is a dearth of international normative literature
on return of results in the paediatric context [6]. Existing
policies and/or guidelines are primarily concerned with
adult-level participation, and are extremely broad in
both scope and application [4]. That being said, however,
several important principles can be distilled from these
normative documents that are highly relevant to the issue
of return of results in paediatric research. For example,
UNESCOs 1997 Universal Declaration on the Human
Genome and Human Rights sets out [t]he right of each
individual to decide whether or not to be informed of
the results of genetic examination and the resulting
consequences should be respected[7], despite the fact
that it does not explore the nature of this right in the
paediatric context.
In international law, the BIC is the central ethos for
decisions concerning children participating in genomic
research. Consequently, the CRC can be viewed as a
cornerstonedocument; with the dual distinction of
(1) having been unanimously approved by the UNs
General Assembly; and (2) being the most universally rati-
fied UN treaty to date [8], the CRC offers a comprehensive
statement on paediatric human rights. Article 3 of the
CRC enshrines the BIC as a general principle, which has
since been reinforced by a number of international
normative documents.
The CRC also enshrines other important paediatric
rights. Notably, for our purposes, these include the childs
right to be heard[5] and right to enjoy the highest
attainable standard of health[5]. Though the CRC is not
specific to paediatric research (and, in fact, this paper
acknowledges that research is fundamentally different
from clinical care), the driving spirit of this convention
can be applied to the debate surrounding paediatric
research. In point of fact, since the CRC came into force,
scholarship on this convention has focused on the rights
of autonomy and participation in the paediatric context,
and decisions affecting these rights [9].
Importantly, the rights spelled out in the CRC exist
independently of [] parents and are enforceable by
children over against their parents through the medium
of the state[10]. This is significant both because it
has altered the rights and responsibilities of those
who share in decision-making surrounding children (i.e.,
parents, researchers, children, and the State), but also
because children are seen to have independent legal
rights (albeit usually exercised via a court-appointed
tutor). Moreover, this duality begs the question of how to
balance those rights and responsibilities under the
BIC framework. For the purposes of this text, we will
use the CRCs definition of child, which refers to
every human being below the age of eighteen years
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unless under the law applicable to the child, majority
is attained earlier[5].
The best interests of the child: definition and scope
Article 3 of the CRC states:
In all actions concerning children, whether undertaken
by public or private social welfare institutions, courts of
law, administrative authorities or legislative bodies, the best
interests of the child shall be a primary consideration [5].
While this article emphasizes the paramountcy of the
BIC, it is necessarily broad. Nonetheless, the CRC provides
some indication of the principles overall scope. Indeed, this
article establishes the BIC as a general principle guiding
the interpretation of the entire CRC. In other words, when
taking measures under article 24 (the right of the child to
the enjoyment of the highest attainable standard of health)
for example, States Parties to the CRC must consider
whether a decision or policy satisfies the requirements
outlined in article 3.
However, the interpretation of article 3 calls for a
nuanced approach, with particular attention to wording.
The use of the term primaryimplies that the BIC
principle is not the only factor to be considered in the
actions of institutions, authorities and administration
[11]. Additionally, article 3 refers to the BIC as a primary
(and not the primary) consideration. Therefore, the BIC
should be among the first aspects to be considered
andgiven considerable weight in all decisions affect-
ing children[12]. The World Medical Associations
Declaration of Ottawa on Child Health [13] adopts a
similar approach: [t]he best interests of the child shall be
the primary consideration in the provision of health care
[13] (emphasis added).
In 2008, the Council of Europes Commissioner for
Human Rights tentatively defined the BIC principle as the
sum total of the norms in the CRC[12]. Thus, a decision
is held to be in the best interests of the child if it most
effectively applies the rights contained in the CRC. It
requires the use of good and reasonable options[14] that
safeguard the rights of children to the maximum extent
possible [15]. Furthermore, the BIC principle requires that
children be placed at the centre of the decision-making
process and that policies and strategies be adapted to
childrensrightsandneeds[16].
If the BIC principle is to inform paediatric genomic
research, then it must favour the most effective application
of the rights of the child. This means utilizing all good and
reasonable optionsto safeguard and indeed assist in the
exercise of the rights of paediatric participants. However,
the substantive content of these rights and how they are
balanced against the best interests of the child remains an
open question in genomic research. After all, though the
CRC states that the BIC is a primary consideration, it is
not the only consideration children possess other rights
that must coexist with the BIC principle. We now turn to
some of these other rights.
The right to be heard: elements to consider
The UN Committee on the Rights of the Child (UN
Committee) views the right to be heard as a general
principle, guiding the interpretation and implementation of
all other paediatric rights [11]. Article 12, which codified
this right for the first time, reads:
1. States Parties shall assure to the child who is capable
of forming his or her own views the right to express those
views freely in all matters affecting the child, the views of
the child being given due weight in accordance with the
age and maturity of the child [5].
Unlike article 3, this provision contains several specific
criteria that must also be satisfied during the course of
paediatric decision-making. Recall also that this right
exists independent of parents, and consequently can
be exercised independently of same [10].
As soon as children become capable of forming their
own views, they should be given the opportunity to express
them. As children mature, they should be granted a higher
degree of participation. In this same vein, the Organization
for Economic Cooperation and DevelopmentsGuidelines
for Human Biobanks and Genetic Research Databases
recommends finding ways in which minor[s] can play a
more active role’‘in light of [their] age and autonomy[17].
This view appears to be supported by the UN Committee
[11] and by previous versions of article 3, which included
the right of the child to be heard in relation to his or her
best interests[18].
The right to be heard rests on the evaluation of a
childs mental capacity. In this regard, the UN Committee
refers to an obligation of States Parties to assess the
capacity of the child to form an autonomous opinion
to the greatest extent possible[11]. The Committee
insists, however, that there be a presumption that
children have the capacity to form their own views
[11]. Thus, the right to be heard should not be linked
to the childs age, but to the acquisition of necessary
intellectual, cognitive, social, and emotional skills. Children
do not need to fully understand a complex issue (such as
genomic research) to be able to express their opinions. As
mentioned earlier, States Parties can determine the age of
majority by law, and may also prescribe a specific age for
participation in research.
Article 12 also requires that children be able to express
their views freely. This freedom of expression comprises
three criteria. First, children should be able to decide
whether or not to exercise their rights [11]. Second, the
right to be heard should be exercised without undue
influence or pressure from family members and/or society.
Third, children must have access to and receive complete
information in accordance with articles 13 (freedom to
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seek information) and 17 (access to information) of the
CRC [19]. According to the UN Committee:
Physicians and health-care facilities should provide
clear and accessible information to children on their rights
concerning their participation in paediatric research and
clinical trials. They have to be informed about the
research, so that their informed consent can be obtained
in addition to other procedural safeguards [11].
Also, childrens views must be given due weight in
accordance with their age and maturity.
b
In the context of
adolescent health, the UN Committee even recommends
that sufficiently mature adolescents be given the right to
consent to their own health care, and that parents should
only be informed if such disclosure proves to be in the
best interests of the child [20]. The UN Committee did
not address the issue of whether a greater degree of
maturityis required for participation in research.
Whereas clinical care provides therapeutic benefit to
individual patients, research is intended to produce
generalizable results for society as a whole. In view of the
foregoing, it may be possible to infer a higher degree of
maturity for paediatric participation in research the
better to avoid therapeutic misconception.
Finally, while article 18 of the CRC provides that
States Parties shall use their best efforts to ensure
recognition of the principle that both parents have
common responsibilities for the upbringing and develop-
ment of the child,the same article specifies that the best
interests of the child will be their basic concern[5]. There
is thus potential for conflict between, on the one hand, the
responsibility (of parents, the state, and researchers) to
protect the best interests of the child; and on the other
hand, the childs right to be heard and to participate in the
decision-making process.
In Mabon v Mabon [21], an English case on the right
(of an adolescent) to be heard in custody proceedings,
the court is instructive on this conflict and on article 12
of the CRC. Lord Justice Thorpe writes we must, in the
case of articulate teenagers, accept that the right to
freedom of expression and participation outweighs the
paternalistic judgment of welfare[21]. This passage
suggests that a broader interpretation of the BIC is
emerging in light of article 12. An interpretation of
the BIC that is restricted to an objective standard is
no longer sufficient. Instead, the BIC must include space
for the maturing child to exercise his or her rights as
enshrined in the CRC.
Under the CRC, the implication for paediatric genomic
research is that a maturing child has a say in whether he
or she participates in research, and whether his or her
results are to be returned. In some cases the childs best
interests are paramount, while in others, these interests
may be overridden. This duality imposes unique respon-
sibilities on researchers to ascertain the childs wishes,
the weight of these wishes relative to the childs age and
maturity, and the lengths to which paediatric participant
rights must be safeguarded. Likewise, States Parties must
consider these rights when designing legislative regimes
surrounding paediatric genomic research.
Right to the enjoyment of the highest attainable standard
of health
The right of the child to the enjoyment of the highest
attainable standard of health[5] is another important
principle for the issue of return of research results in the
field of paediatrics. Indeed, article 24(2) of the CRC requires
that States pursue full implementation of this right and
carry out appropriate measures: ‘…b) to ensure the
provision of necessary medical assistance and health care to
all children with emphasis on the development of primary
health care[5].
The majority of case law on the childs right to health
pertains to immigration litigation, and is therefore
largely irrelevant to paediatric research. That being said,
in a 2003 General Comment on HIV/AIDS and the rights
of the child, the UN Committee associated the childs
right to health with States Parties ensuring that
HIV/AIDS research programmes include specific studies
that contribute to effective prevention, care, treatment
and impact reduction for children[16].
While the UN Committee is currently drafting a
General Comment on the childs right to health, the
Human Rights Council recently adopted a resolution
[22] that briefly discusses this right. In article 37 of the
Resolution, the Council calls upon states to take all
necessary measures to ensure the right of the child to
life, survival and the enjoyment of the highest attainable
standard of physical and mental health is promoted and
protected. To the extent that the decision to return
research results that are clinically significant, useful and
actionable during childhood can result in preventative and/
or therapeutic treatment, a connection may be drawn
between the right to the highest attainable standard of
health and the return of research results. Though the right
to the highest attainable standard of health is predicated
upon clinical action, the decision to return such research
results is sufficiently tied to clinical care that it merits
theoretical inclusion. Indeed, a number of international
norms on research generally have associated the return of
results with the health and quality of life of participants [4].
Regional norms
In Europe, childrens rights are increasingly grounded in
international principles (such as those discussed above),
and as a result, they shed some light on the issues of
paediatric research, return of results and the right not
to know.
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The best interests of the child
The European Convention on Human Rights [23], a treaty
aimed at protecting human rights and fundamental
freedoms in Europe, does not specifically address the
rights of children. Consequently, the European Court
of Human Rights (ECtHR)mustincreasinglyreferto
the CRC and interpretations thereof in cases involving
children [24].
In a landmark custody case on the interpretation of
the BIC, the ECtHR described the BIC as a principle
requiring primary consideration in decision-making
concerning children.
c
The Court added that the principle
could, depending on the nature and seriousness of the
situation, override parental interests. While the Court
decided that parental interest remains a factor when
balancing the various interests at stake, it insisted that
this does not entitle parents to take measures that would
harm the childs health and development. The BIC
will depend on a variety of individual circumstances,
in particular his age and level of maturity, the presence or
absence of his parents and his environment and his
experiences[25]. In addition, when adjudicating a case
concerning children, the Court must conduct an in-depth
examination of the entire family situation and of a whole
series of factors, in particular of a factual, emotional,
psychological, material and medical nature, and make
a balanced and reasonable assessment of the respective
interests of each person, with a constant concern for
determining what the best solution would be for the
()child[25].
Although, prima facie, parental custody is a world
apart from genetic research, the BIC principle is also
applicable to the return of results in genomic research. In
point of fact, according to Ross LF et al.:
In deciding whether a child should undergo [genetic
testing], the focus must be on the childs medical best
interests; however, parents and guardians may also consider
the potential psychosocial benefits and harms to the child
and the extended family. Extending consideration beyond
the childsmedicalbestinterestnotonlyacknowledgesthe
traditional deference given to parents about how they raise
their children, but also recognizes that the interest of
a child is embedded in and dependent on the interests of
a family unit [26].
Thus, the BIC framework sits in [] a web of moral,
legal, medical or social policies about duties to people
who cannot make decisions for themselves, including
[] acceptable thresholds of care[14]. Consequently,
researchers must not only defer to the BIC principle in
the context of return of results, but must also evaluate
this principle in light of competing interests. In Europe,
this deference entails an assessment of the multiple factors
identified by the ECtHR, where the weight attached to each
factor necessarily depends on the context. Nevertheless, in
such analysis, considerations other than the BIC should be
considered. We now turn to these.
The Childs right to be heard: autonomy vs. Parental
authority
The childs right to autonomy is often juxtaposed with
parental rights (i.e.parental authority). Interestingly,
both of these rights are derived from the same source
article 8 ECHR on the [r]ight to respect for private and
family life[23]. Other conventions have also addressed
these rights. Chapter II (articles 5 to 9) of the Convention
on Human Rights and Biomedicine (CHRB) [27],also
known as the Oviedo Convention, addresses the issue of
informed consent. In the case of a minor who lacks
capacity, article 6(2) states that consent can only be
given by an authorised representative and that the
opinion of the minor shall be taken into consideration as
an increasingly determining factor in proportion to his or
her age and degree of maturity.
Similar to the UN CommitteesGeneral Comment on
the right to be heard, the Explanatory Report to the CHRB
mentions that in certain situations which take account of
the nature and seriousness of the intervention as well as
the minors age and ability to understand, the minors
opinion should increasingly carry more weight in the final
decision[28]. This is all the more pertinent given article
27 of the Council of EuropesAdditional Protocol on
Biomedical Research, which reads:
If research gives rise to information of relevance to the
current or future health or quality of life of research
participants, this information must be offered to them.
That shall be done within a framework of health care or
counselling. In communication of such information, due
care must be taken in order to protect confidentiality and
to respect any wish of a participant not to receive
such information [29].
According to the Explanatory Report, the word offered
implies that participants must consent to or refuse any
future communication of information prior to participation
in research. This indicates that participants have a choice
when the results become available.
Several cases on the interpretation of the European
instruments described above have explored the interplay
between the childs right to autonomy and parental
authority. Unfortunately, none of these cases specifically
address the issue of research. However, given the fact that
research is intended to produce generalizable results for
society as a whole, the bar for return of results in this
context is arguably higher than in the context of clinical
care, where doctor-patient interests are aligned and
treatment is intended to provide therapeutic benefit to
individual patients.
The early 1988 ECtHR case of Nielsen v Denmark [30]
involved a childs detention in a psychiatric hospital at
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his mothers request.
d
The key issue was whether this
was a lawful exercise of the mothers parental authority
under article 8. The Court held that the exercise of
parental rights constitutes a fundamental element of
family life[30]. However, the Court went on to add,
the rights of the holder of parental authority cannot
be unlimited and that it is incumbent on the State to
provide safeguards against abuse[30].
The Nielsen case is important because it strongly
reaffirms the principle that parents are authorized to
make healthcare decisions on behalf of their children.
Although the Court recognized the existence of limitations
to parental authority, it did not adequately define these
limitations. A discussion on this topic was held nearly
twenty years later in R (on the Application of Axon) v
Secretary of State for Health [31].
Axon, a UK court decision, discussed the limits on
parental rights as articulated in article 8 of the ECHR
in terms of the childs right to autonomy. The case
involved a mother who challenged a UK health policy
allowing physicians to prescribe birth control to adolescent
women without informing parents. Upholding the policy,
Justice Silberman said that parental authority dwindles as
their child gets older and is able to understand the
consequence of different choices and then to make
decisions relating to them[31].
In other words, the childs right to autonomy increases
with maturity and eventually trumps parental authority.
This is because parental rights are, in reality, duties, and
extend only so far as needed for parents to discharge
their responsibilities relating to the childs upbringing
and development.
e
As the child becomes more and more
capable of guiding their own development, parental
responsibilities naturally diminish. Herein is the limiting
principle as alluded to by the ECtHR in Nielsen.
The implications of Nielsen and Axon for genomic
research are unclear, but some potential themes emerge.
While parents have authority to make decisions concern-
ing their childs participation in research and as concerns
return of results, that authority dwindlesas the child
matures. Thus the researcher should be conscious of the
childs increasing autonomy in decision-making and take
steps to safeguard it where appropriate.
That being said, it is important to note that Nielsen
was a case of psychiatric confinement and Axon involved
the parentsright to be informed as to their childs use
of birth control.
The right not to know
Article 10 of the CHRB concerns the right not to know
information about ones health.
f
Article 10 states:
Everyone is entitled to know any information collected
about his or her health. However, the wishes of individuals
not to be so informed shall be observed.
In exceptional cases, restrictions may be placed by law
on the exercise of the rights contained in paragraph 2 in
the interests of the patient [27].
The right not to know can be restricted in certain
circumstances when it is in the best interests of the
patient or on an exceptional basis [27], as provided in
article 26(1). As the Explanatory Report points out,
these exceptions would allow for informing patients of a
predisposition to disease if this information could enable
individuals to take potentially effective (preventive)
measures[28], even if the person has expressed the
wish not to know this information. Similarly,the possibility
for prevention of the risk to [a] third party might []
warrant his or her right taking precedence over the
patients right to privacy, [] and as a result the right not
to know[28]. Again, the BIC is a primary consider-
ation, but not the only consideration. In such cases,
the Explanatory Report suggests that internal law balance
the rights. As will be explored in Section 4, some
European countries have chosen to enact legislation
allowing an exception to privacy in the context of medical
research whenever there is a serious risk to the health or
life of a family member.
As mentioned earlier, article 27 of the Additional Protocol
stipulates that information of relevance to current or future
health or quality of life must be offered to the partici-
pants themselves, and that any wish not to receive such
information must be respected as part of the duty of
care [29]. This article, however, does not address the
parentchild relationship, as parents can refuse to receive
information pertaining to their childrens health.
Nevertheless, professional societies such as the European
Society of Human Genetics (ESHG) have adopted more
specific guidance, providing that parents are responsible to
inform their children about their genetic risks[32]. Indeed,
a 2012 ESHG Principles for Good Practice in Paediatric
Biobanks goes further: [t]he right of parents to receive or
not receive genetic information about their children is
limited[33]. The limited nature of parental authority
mirrors the Axon case [31] where the childs developing
autonomy reached a tipping point at which it trumped the
parental right to know. Interestingly, the ESHG adds that
[i]n the rare case that information about a preventable or
treatable early-onset disease is found, [parents] should be
notified regardless of their wishes providing the findings
are subject to assessment of clinical validity and utility[33]
(emphasis added). This more specific guidance on the
return of results in the paediatric context protects the childs
best interests in the context of health care and research, and
is echoed in the positions of several countries.
Interestingly, the Whole Genome Sequencing in Health
Care: Recommendations of the European Society of
Human Genetics [34] is not only concerned with the
childs right to autonomy, but also with the preservation
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of his or her future autonomy in the context of return of
results. Specifically, the ESHG is concerned that the
childs right to be informed (or to refuse information)
will be undermined as he or she matures. However, the
Recommendations also stress that there must be balance
between the childs interests and autonomy and the
parental rights and needs (not) to receive information that
may be in the interest of their (future) family[34]. Finally,
some authors have suggested that research results should
be discussed during the informed consent process and
that researchers have a duty to return results when the
criteria of scientific validity, clinical utility, and medical
actionability are met [35]. The parentsright not to know
cannot be used to override the duty owed to the child.
National norms
g
France
In France, parents are vested with decision-making
authority on all matters relating to health care until such
time as children reach the age of majority (i.e., eighteen
years of age). Parents are responsible for the health and
security of their children, and under French law, parental
authority must always be exercised in the best interests of
the child [36]. Moreover, since the ratification of the
CRC by the French Parliament in 1990, parental
decision-making must also respect the fundamental
principles outlined therein.
h
The Code de la santé publique [37] has detailed a
number of provisions relating to the participation of
children in biomedical research. While consent must be
given by both parents [37],
i
children must be consulted
and their views taken into account, according to their
level of maturity [37] and in conformity with the
right to be heard, as found in the CRC. Accordingly,
section L-1122-2 of the CSP requires researchers to
obtain paediatric consent and to respect the childs
consent or refusal, as the case may be.
The new Loi Jardé [38] (adopted but not yet in force)
specifically addresses the return of research results.
These new provisions would mandate the provision of feed-
back during baseline assessment (i.e., upon recruitment)
(Loi Jardé L1121-1) as well as feedback of general research
results (Loi Jardé L1121-1). Moreover, participants have a
right to information, in the course of and at the conclusion
of the research project, concerning their health, in the form
of a written document given to individuals who have con-
sented to receive said information (e.g. guardians/parents)
[38]. Section L1131-1-1 alsostipulatesthatdonorswho
participate in secondary genetic research can consent (or
refuse) to be informed of serious genetic anomalies.
Although parents are responsible for consenting to
the return of individual research results concerning
the health of their children, they can also refuse to be
informed, as long as their refusal respects the requirement
of section 3711 of the Civil Code i.e., that this decision
be in the best interests of the child. In any case, clinician-
researchers have a duty to protect the best interests of
the child [37,39], and as a consequence, may choose to
implement policies that would see the return of paediatric
results even where parental refusal has already been
exercised [38].
United Kingdom
In the UK, the 1985 Gillick case [40] is a clinical and
jurisprudential landmark for the principle that the
capacity of a minor to consent to medical care should
be determined on a case-by-case basis. According to
the majority of the Court, it must be determined
whether the child has sufficient understanding and
intelligence to fully comprehend what is involved in a
proposed intervention.
In the UK, the statutory age of presumed maturity
(in the context of medical care) is age sixteen [41];
however, this age may be lower in particular treatment
situations. It is important that the age of consent for
medical care not be confused with the age of consent for
research, including the choice to receive (or not) research
results and incidental findings. Moreover, it cannot be
presumed that ethics review committees and UK courts
would judge that the threshold for capacity to consent to
research is the same for treatment, especially considering
the risks and benefits of the latter.
When children are deemed to be incapable of consent,
parents (or legal guardians) must consent to health care
or medical research on behalf of their children. In such
cases, parents are presumed to have acted in the best
interests of their children [42]. The BIC is determined
according to childrens wishes, their capacity to understand
their options and the consequences of these options, their
emotional and physical needs, their parentswishes and the
need to minimise restrictions on future autonomy, among
other factors [42,43]. Children should be involved as much
as possible in the decision-making process and their views
should be taken into account.
j
Where parents and health
professionals disagree on what is in the best interests of the
child, the health professional may seek the opinion of the
court. In such cases, the courts have traditionally followed
the opinion of the physician [44]. In Re MM (Medical
Treatment) [45], for example, the parents of a child suffer-
ing from primary immunodeficiency requested that their
son continue with a programme of immunostimulant ther-
apy and not receive replacement immunoglobin intraven-
ously, as suggested by his physicians. Although the parties
eventually came to an agreement during the course of the
hearing, the Court nonetheless laid out the extent to which
it would have been prepared to intervene in this case:
Although the parentsobjections to the particular
treatment recommended by the doctors were rational
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objections, the court would have been prepared to
override them had that been necessary, because the
evidence that this treatment was in the childsbest
interests was overwhelming [45].
In 1994, the Clinical Genetics Society issued a report
on the genetic testing of children [46]. Though this
report was not specifically aimed at genetic research,
it recommended that genetic testing be limited to
childhood onset disorders where useful medical interven-
tion exists. In contrast, testing for adult-onset disorders
and carrier status (having reproductive significance only)
should generally be deferred until adulthood. The
recommendations required that any decisions concerning
immediate testing or deferral should be made on the basis
of the best interests of the child, and should also involve
a discussion between the parents and genetics health
professionals.
Recent guidance from the British Society for Human
Genetics on whole genome sequencing limits itself to
questioning whether UK law would restrict evidence of
best interests to a narrow clinical context[44]. The PHG
Foundations 2011 review of the implications of whole
genome sequencing states that increased involvement of
minors in decision-making through assent is standard
practice, and identifies re-consent at age of majority as an
emerging practice [47]. Finally, as in France, medical
deontology in the United Kingdom dictates that parental
refusal should be overridden in cases where the communi-
cation of health information would be in the best interests
of the child [44].
Spain
Similarly to the Loi Jardé in France, the Spanish Law 14/
2007 of 3 July 2007 on Biomedical Research contains provi-
sions specific to the communication of research results.
Section 4(5) states that everyone has a right to be informed
of his or her genetic data or other data of a personal nature
obtained during the course of research. This same right
exists for retrospective (secondary) tissue research. The
corollary of this right is an obligation for researchers to
inform participants of relevant health information arising
from research [48]. That being said, participants could
refuse to receive such information in conformity with the
1997 Oviedo Convention by exercising their right not to
know under article 4(5) of the 2007 law, which states that:
There shall be an observance of the right of the person
not to know that data, which is referred to in the former
paragraph, including unexpected findings that could
arise. Nonetheless, when this information, according to
the criteria of the doctor in charge, is necessary in order to
avoid serious damage to his health or that of his biological
family members, a close family member or a representative
shall be informed, after consulting with the clinical ethics
committee, if it exists [48].
In short, while no specific mention is made of minors, it
would seem that parental refusal could also be overturned
if certain legal conditions were met.
The Netherlands
The Medical Research Involving Human Subjects Act
governs research in the Netherlands [49]. According to
section 4, the legal age of consent is 18. The participation
of a minor (under the age of 18) in research would require
the written consent of parents or legal representatives, if
applicable [49].
As for the issue of return of research results, there
are no legal provisions on the communication of these
results in Dutch law. That said, the 2011 Code Goed
Gebruik of the Federation of Dutch Medical Scientific
Societies recommends that results be returned where:
1) there is a real risk of a serious condition; 2) a
professionally-recognised treatment can be offered to
the donor; 3) the donor is known to carry the risk factor;
4) disclosure would allow treatment of his or her condi-
tion; and, 5) the donor has consented [50]. Similar to the
Spanish 2007 Law on Biomedical Research, the Dutch
Code of Conduct also recommends the return of results if
doing so might benefit a family member.
Canada
The second edition of the Tri-Council Policy Statement
(TCPS 2) apan-Canadiannormativedocument
governing research requires that research ethics
committees ensure, among other things, that researchers
seek and maintain consent from authorized third parties
in accordance with the best interests of the persons
concerned[51], when individuals who lack the capacity to
consent are participating in research. In Canada, the
definition of the BIC principle is statutorily enshrined in
provincial legislation. In the province of Quebec [52], for
example, Article 33 of the Civil Code of Quebec states:
Every decision concerning a child shall be taken in
light of the child's interests and the respect of his rights.
Consideration is given, in addition to the moral, intellectual,
emotional and physical needs of the child, to the child's
age, health, personality and family environment, and to the
other aspects of his situation[53]. In British Columbia,
Article 37(2) of the Family Law Act stipulates that,
concerning guardianship:
To determine what is in the best interests of a
child, all of the child's needs and circumstances must
be considered, including the following: (a) the child's
health and emotional well-being; (b) the child's views,
unless it would be inappropriate to consider them; (c)
the nature and strength of the relationships between
the child and significant persons in the child's life; (d) the
history of the child's care; (e) the child's need for stability,
given the child's age and stage of development; (f )
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the ability of each person who is a guardian or seeks
guardianship of the child, or who has or seeks parental
responsibilities, parenting time or contact with the child,
to exercise his or her responsibilities; (g) the impact
of any family violence on the child's safety, security
or well-being, whether the family violence is directed
toward the child or another family member; (h)
whether the actions of a person responsible for family
violence indicate that the person may be impaired in
his or her ability to care for the child and meet the
child's needs; (i) the appropriateness of an arrangement
that would require the child's guardians to cooperate on
issues affecting the child, including whether requiring
cooperation would increase any risks to the safety, security
or well-being of the child or other family members; (j) any
civil or criminal proceeding relevant to the child's safety,
security or well-being [54].
Last, in Manitoba, Article 2(1) of The Child and Family
Services Act holds that:
The best interests of the child shall be the paramount
consideration of the director, an authority, the children's
advocate, an agency and a court in all proceedings under
this Act affecting a child, other than proceedings to
determine whether a child is in need of protection,
and in determining best interests the child's safety and
security shall be the primary considerations. After that, all
other relevant matters shall be considered, including (a)
the child's opportunity to have a parentchild relationship
as a wanted and needed member within a family structure;
(b) the mental, emotional, physical and educational needs
of the child and the appropriate care or treatment, or
both, to meet such needs; (c) the child's mental, emotional
and physical stage of development; (d) the child's sense of
continuity and need for permanency with the least
possible disruption; (e) the merits and the risks of any plan
proposed by the agency that would be caring for the child
compared with the merits and the risks of the child
returning to or remaining within the family; (f) the views
and preferences of the child where they can reasonably be
ascertained; (g) the effect upon the child of any delay in
the final disposition of the proceedings; and (h) the child's
cultural, linguistic, racial and religious heritage [55].
In AC v Manitoba (Director of Child and Family Services),
the Supreme Court of Canada further described the BIC as
operating according to a:sliding scale of scrutiny, with
the childs views becoming increasingly determinative
depending on his or her maturity. The more serious
the nature of the decision and the more severe its
potential impact on life or health, the greater the degree of
scrutiny required [] The more a court is satisfied that a
child is capable of making a truly mature and independent
decision on his or her own behalf, the greater the weight
that must be given to his or her views when a court is
exercising its discretion [] If, after a careful analysis
of the young persons ability to exercise mature and
independent judgment, the court is persuaded that
the necessary level of maturity exists, the young persons
views ought to be respected [56].
As for the return of individual findings to participants,
the TCPS 2 created an ethical obligation for researchers to
return what it describes as materialincidental findings
or findings interpreted as having significant welfare
implications for the participant, whether health-related,
psychological or social[51]. According to the TCPS 2,
researchers are also required to develop a plan for
managing information that may be revealed through their
genetic research, submit such a plan to their research
ethics committees, and advise prospective participants of
the plan to manage their information [51]. Participants
should be provided with an opportunity to make informed
choices on whether they wish to receive information about
themselves, and be given the opportunity to express their
preference on whether information emanating from the
research project can be shared with biological relatives [51].
This preference, however, may be subject to overriding
considerationssuch as information on life-threatening
conditions that can be prevented or treated.
The Centre of Genomics and Policy and the Mother
Infant Child and Youth Network, in collaboration with
the Ethics Office of the Canadian Institutes of Health
Research, have adopted more specific guidance in their
2012 Best Practices for Health Research involving Children
and Adolescents [4].According to this document:
Generally, researchers should respect the wishes of the
competent adolescent, or those of the parents, if the
minor is not competent to consent, regarding the return
of research results. However, individual results and
incidental findings should be communicated 1) if they are
scientifically valid, 2) if they have significant implications
for the health of the child or adolescent, and 3) if there is
a means of prevention or treatment available during
childhood or adolescence [4].
The Best Practices later specify that when the research
involves school-age minors capable of assent, the
information should also be delivered to them with the
agreement of their parents, and in a manner appropriate
to their development, level of understanding and degree
of maturity[4].
Rights, responsibilities and the best interests of the child
The foregoing discussion has raised several questions on
the meaning of the BIC and its relation to other laws
and rights, both under the CRC and elsewhere. Is it in
the best interests of the child to allow him or her to
subjectivelychoose a course of action that may result in
adverse or suboptimal consequences? Or is it in the
childs best interests to impose an objectivelyfavourable
course of action that would override his or her wishes?
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In such cases, how should the right to be heard factor
into the decision-making process?
Some have accused the BIC principle of being overly
paternalistic and have questioned whether it is, in fact, at
odds with childrens rights and respect for their autonomy
[8]. Consider the comments of Diana Bryant, Chief Justice
of Australias Family Court:
If childrens immutable rights are in issue we should
say so, rather than attempting, as I think we have done,
to quietly subsume human rights considerations under
the rubric of best interests[8].
There is thus a growing appreciation of the fact that a
careful balance must be struck between adequate welfare
protection of a still-developing child and the childs
other rights in particular, his or her right to autonomy
and self-determination with due regard for age and
maturity. That being said, at what point does the opinion
of the minor become the only opinion that should be
taken into account [57]? The answer to this question
can only be determined on a case-by-case basis.
In contrast, other commentators have called the BIC
overly individualistic, arguing that achilds interests cannot
be completely distinguished from those of his or her
parents, but are always intertwined with those of the
parents and siblings[58]. Interests other than those
solely of the child can, and should, come into play
where decisions affecting the child may potentially
impact the family unit. The childs interests are intimately
connected with those of his or her family a fact that
should not be ignored.
Others have argued that the childs right to be heard
in article 12 of the CRC should be understood as a
thresholdrequirement [59]. Once the child is mature
enough to make a decision on any matter affecting his or
her interests, he or she has the right to do so. Parental
authority cannot be used to outweigh the childs
autonomy once that threshold of maturity has been
reached (i.e., a Gillick-like test on a case-by-case basis).
This issue raises a further question: whose responsibility
is it to ensure the protection of paediatric rights? Whereas
in the past, parents were viewed as custodians[10] of
paediatric rights, with the advent of the CRC, the scope of
actors sharing in that responsibility and their marge de
manoeuvre was substantially widened. Commenting on
the advent of the CRC, Reading et al. write:
One of the far-reaching consequences of the [CRC]
is that it makes the child an individual with rights
and not just a passive recipient, and hence the child
has the right to actively participate at all levels of
decision-making. The traditional association between the
state, the family, and the child could be conceptualised as
a series of concentric circles with the child at the centre.
The [CRC] implies that this association should now
be understood to be triangular in which the state has
a direct responsibility to the child to promote her or
his rights [60].
To this triangular structure, we suggest adding a fourth
actor the researcher in the context of paediatric
research. Indeed, the researchers responsibilities to the
child participant could be said to exist independently of
those of the parents or State.
k
This new structure
raises questions as to the nature of State and researcher
responsibilities towards the child and how far they must
go in discharging those responsibilities. As demonstrated,
different regional and national norms have approached
these issues in different ways.
Genomic research and the question of return of results
in the paediatric context give rise to new challenges for
the BIC principle and the rights of the child. The arrival
of next-generation sequencing has challenged whether the
BIC principle and the childsanticipatoryautonomy rights
or right to an open future(whereby decision-making
concerning genetic screening of adult-onset disorders must
be deferred until adulthood) continue to offer a suitable
(or even workable) framework [47,61]. Likewise, by
examining the entire human genome (as opposed to
discrete sections), whole genome sequencing has changed
the nature of genetic testing and increased the possibility
of incidental findings, thereby potentially pre-empting
the childs right to defer test-related decisions until
adulthood [47,61,62].
With this in mind, it is interesting to note the controversy
surrounding the more radical position of the American
College of Medical Genetics and Genomics (ACMG) in its
Recommendations for Reporting of Incidental Findings in
Clinical Exome and Genome Sequencing,publishedon19
March 2013 [63]. While an in-depth discussion is beyond
the scope of this paper, the Recommendations mandated
that laboratories test for 57 reportable genetic conditions
during whole genome sequencing, irrespective of the ages
of participants or their wishes. Shortly thereafter, the
ACMG published a clarification re-affirming this position
and imposing an overall obligation to report pathogenic
variations for these 57 genes irrespective of choice
[64]. In justifying this, the ACMG pointed to the potential
implications for parents in cases where actionable
genetic predispositions are discovered through genomic
sequencing of related children (e.g. in cases concerning
the BRCA1 gene). The ACMGs perspective challenges
the BIC in favour of a broader, more family-centric view,
and seemingly brings the parentsinterests in direct
conflict with those of the child.
Summary
Our comparative analysis of international, regional and
national norms pertaining to the return of research results
has revealed a dearth of normative documents specifically
addressing the issue of return of results in paediatric
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research. Though the best interests of the child principle
guides both international and regional normative
paradigms, other principles, such as the right to auton-
omy, the right to be heard, the right to enjoy the highest
attainable standard of health and the right not to know,
further delimit the parameters of paediatric-specific
approaches to the question of return of research results in
paediatric genomic research. Today, the challenge is to
determine the content and scope of the BIC frame-
work in the context of the increasingly recognised right to
autonomy. The childs future autonomy (upon reaching
the age of maturity) is also an important consideration.
Nevertheless, genomic research raises the possibility that
the childs parents may also have an interest in the research
results, an issue that must likewise be considered.
National guidelines and laws differ in their approach
to the return of results as well as on the role and obliga-
tions of the various stakeholders involved in the decision-
making process. However, a trend towards making the
return of results obligatory if this return is in the best
interests of the child (during childhood) is clearly tangible.
Moreover, based on laws and jurisprudential interpreta-
tions, parental refusal of results may be overridden if doing
so would be in the childs best interests. Given that next-
generation sequencing technologies may reveal clinically
significant health information of importance to the child
prior to maturity, the duty to protect children from harm
and promote their health interests lends to the appli-
cation of a more proactive approach, one that would
mandate return irrespective of parental preferences
under certain conditions. Return of paediatric results,
however, is not akin to the duty to actively seek out
or hunt for findings.
Finally, given that research is increasingly international
and collaborative, initiatives fostering discussion between
members of the paediatric research community should be
encouraged (see for example the position on the return of
results of the International Paediatric Research Platform
of the Public Population Project in Genomics and Society
[65]). Indeed, the sharing of policies, tools and ideas
at the international level will not only ensure the efficient
mobilisation of knowledge, but will also set the stage for
uptake by the paediatric community to benefit those who
need it most the children.
Endnotes
a
For a review of US policies, see Predictive Genetic
Testing of Children and the Role of the Best Interest
Standard[66]. For a review of US law, see The Legal
Risks of Returning Results of Genomics Research[67].
b
The United Nations Committee on the Rights of the
Child defines maturity as the capacity of the child to
express his or her views on issues in a reasonable and
independent manner[11].
c
The case of Neulinger and Shuruk v Switzerland [25]
was brought by a mother who had unlawfully removed
her son from Israel in 2005 and returned to her native
Switzerland. The father sought the return of his son
despite having shown a lack of interest in the boy.
The mother argued that she could not return her son
to Israel because she feared the risk of prosecution
and conviction for the abduction. Her son would
therefore have to go back alone. The mother argued
that the father had not seen his son in the last two
years and that returning the son would not be in the
best interests of the child. The Grand Chamber of
the Court considered that since the boy had been in
Switzerland during the proceedings (7 years) and was
well integrated, it would not be in his best interests
to be returned to Israel without his mother [68].
d
A mother sought confinement of her 12 year old son
in a psychiatric hospital because it was clear he did
not want to live with herand the father did not have
custody rights. The hospital accepted the mothers wishes
as the holder of parental authority despite the lack of an
adequate diagnosis. The child challenged the lawfulness of
the confinement through his father.
e
Axon confirmed this principle from the earlier English
case Gillick v West Norfolk and Wisbech Area Health
Authority [40] which, although similar to Axon, did not
refer to the ECHR. See discussion of the United Kingdom
for an examination of the Gillick case.
f
See also Chapter VII of the Additional Protocol [29].
The Explanatory Report on the Convention mentions that
a personsright to knowencompasses all information
collected about his or her health, whether it be a diagnosis,
prognosis or any other relevant fact[28].
g
This section is based on BM Knoppers, A Rioux and
MH Zawati [3].
h
Treaties that have been ratified by the French Parliament
are automatically integrated in domestic law. These
treaties are in theory considered to be hierarchically
superior and to prevail over domestic legislation in
accordance with section 55 of Frances Constitution of 4
October 1958.
i
However, if the research project meets three conditions:
1) negligible risks, 2) the minor is not participating to
research as a healthy volunteer and 3) it is impracticable
to obtain the other parents consent in time, the consent
of one of the parents is sufficient
j
The involvement of the child appears to be an import-
ant principle as it is mentioned in most laws and guide-
lines [69].
k
While on paediatric oncology, on this point see
Hens [33,57].
Competing interests
The authors declare that they have no competing interests.
Zawati et al. BMC Medical Ethics 2014, 15:72 Page 11 of 13
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Authorscontributions
MHZ and DP were responsible for data analysis with intellectual contribution
from BMK. MHZ, DP and BMK drafted the article. All authors contributed to
the conception and design of the paper, interpretation of data, critical
revisions contributing to the intellectual content and approval of the final
version of the manuscript.
Acknowledgments
The authors would like to thank Eliza Cohen from the Centre of Genomics
and Policy for her excellent research assistance. The authors would also like
to acknowledge the financial support of the NIH Grant #1R21HG006612-01
and the Mother, Infant, Child and Youth Research Network of Canada
(MICYRN).
Received: 10 February 2014 Accepted: 29 September 2014
Published: 4 October 2014
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doi:10.1186/1472-6939-15-72
Cite this article as: Zawati et al.:The best interests of the child and the
return of results in genetic research: international comparative
perspectives. BMC Medical Ethics 2014 15:72.
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