The handle http://hdl.handle.net/1887/44738 holds various files of this Leiden University dissertation
Author: Grootens-Wiegers, P.
Title: Targeted informed consent : empowering young participants in medical-scientific research
Issue Date: 2016-12-06
General Discussion
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This study was set up in response to the Ministry of Health-commissioned advisory report on the participation of minors in medical research, presented by the Doek Committee in 2009 (Doek Committee, 2009). In this report, it is argued that amendments should be made to the Dutch research regulations to allow for more medical-scientific research with minors. The need for research with minors is undisputable, as currently 45-60% of medication is prescribed off-label, i.e. is not based on evidence from clinical trials with the specific target group (WHO, 2013). Advancements in pediatric medicine can only be made by conducting clinical trials with children. However, one underlying ethical argument in the Doek report requires closer examination. According to the Doek Committee children should not be viewed as moral objects in need of protection (as is until now the foundation on which research regulations are built), but rather as moral subjects with personal opinions and feelings (Doek Committee, 2009, p.26). Accordingly, children should not be denied the chance to participate in research and should be assigned an active role in the decision-making process about research participation. Children should have the right to make decisions based on their perspective and values and personal concerns, such as the hope for a better treatment or helping others with the same disease (Doek Committee, 2009, p.33).
As little evidence is provided to back up the moral subject-argument in the Doek report, the following questions arise concerning this statement:
- Should children indeed be involved in decisions about research participation;
- To what extent are they capable of deciding;
- How should we support minors in decision-making;
- What is their perspective upon their own role in decision-making?
In this thesis we aimed to answer these questions and to contribute to an evidence-based practice of involving minors in decision-making about research participation.
In order to be capable of decision-making, understanding what is at stake in the decision is a prerequisite (Alderson, 2007; Coyne & Gallagher, 2011; Hein et al., 2012; Kuther &
Posada, 2004). To this end, first, current research information material was examined.
Next, based on this analysis, we developed new research information material in collaboration with children, in order to support them in the decision-making process.
Subsequently, we set out to gain insight in the perspectives of children themselves on their role in the decision-making process. Finally, we addressed the issue to what extent
children can and should be involved in decision-making, from an ethical as well as from a developmental point of view. In the following paragraphs, the results of these studies and the implications for research practice will be discussed, and recommendations for informing and involving minors will be provided.
Research information: current practice
In Part I the current practice with respect to research information material for minors was analyzed. We set out with a systematic literature review of available evidence about the formats and readability levels that are most effective to communicate research information to minors (Chapter 2). This analysis demonstrated a lack of evidence about how to adequately inform minors about medical-scientific research as well as an urgent need for more research in this area.
Only 20 papers could be included in our systematic literature review. The evidence on effective formats was variable; intervention methods to improve understanding were effective in one study and ineffective in another, and no apparently successful interventions were repeated in a follow-up study. In addition, the different studies used different outcome measurements, complicating a comparison between the intervention types. Although no clear conclusions can be drawn as to which format is the most successful in empowering children in decision-making, some of the included studies do however demonstrate that it is possible to improve comprehension in minors by altering the research information format (Tait et al., 2007). Positive effects were found for the use of a story format, for multimedia use and for presenting probabilities as pie charts or percentages (Barnett et al., 2005; Carr et al., 2012; O’Lonergan & Forster-Hardwood, 2011; Ulph et al., 2009; Tait et al., 2007).
The variation in measurements and outcome levels is also reported in a recent review on research information interventions for adults (Nishimura et al., 2013). This review reports positive effects of multimedia interventions, enhanced consent forms, extended discussion and test/feedback approaches (Nishimura et al., 2013). Due to the scarcity of research on interventions for minors, it is unclear whether interventions that are effective for adults are as effective for minors. It is very well possible that children have different preferences for communication formats than adults (as they generally also do in daily life), and that the nature of the research information for minors should be different from that for adults (Broome, 1999). For example, a recent study (published after our review) compared the effect of presenting information on an iPad to a traditional paper format,
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and the iPad was found to be significantly easier to follow and to be more effective for children, but not for adults (Tait, Voepel-Lewis, & Levine, 2015). Further research should clarify whether this positive effect of using interactive multimedia specifically for minors is a general trend and how it can be implemented in the process of informing minors about medical-scientific research.
Even worse than the poor availability of data about effective formats, was the evidence on readability of information for minors: only 3 papers could be included in our study. The included papers reported that this readability does not correspond with the reading level of minors; in one study the readability was even comparable to that of academic journals (Tarnowski et al., 1990).
These findings are in accordance with an extensive body of literature on research information for adults, reporting poor readability and a gap between the provided information and average reading skills in society (Jefford & Moore, 2008; Kass et al, 2011;
Malik, Kuo, Yip, & Mejia, 2014; Souza et al., 2013; Sudore et al., 2006; Terranova et al., 2012). Based on these results it is very likely that children are incapable of understanding the information provided to them, and thus cannot fully participate in the decision-making about their research participation.
In order to gain more insight in the Dutch situation concerning comprehensibility of research information, we examined informed consent forms from two major academic medical centers in the Netherlands (Chapter 3). This analysis identified and thus confirmed the presence of a readability gap between the reading skills required to understand the information and the average reading level of children. The forms were even harder to read than the level that is recommended for adult research information (CCMO, 2008b), and significantly harder to read than popular children’s novels. In addition, some forms were very lengthy and consisted mostly of pure text; only three forms contained accompanying images. The results of this study are in accordance with the papers included in the literature review, reporting poor readability (Ford et al., 2007; Tarnowski et al., 1990) and the absence of visual support (Menoni et al., 2011). With this study we have confirmed the existence of a readability gap in research information for minors in the Netherlands.
We can conclude that current informed consent practice is insufficient to render minors capable of involvement in decision-making about medical-scientific research participation.
Origin of the readability gap
The presence of a readability gap seems to be a general problem in informed consent practice, despite laws about informing research participants at the appropriate level of understanding (EU, 2001; Medical Research (Human Subjects) Act, 1998, Section 6), and oversight from institutional review boards (Kass et al., 2011; Paasche-Orlow et al., 2003).
We believe that the origin of this gap is twofold: (1) lack of insight in the target group by information providers, and (2) lack of evidence-based communication. First, information forms are generally drafted by pediatricians, researchers, or research nurses, all highly educated professionals who are experienced in reading complicated and lengthy texts.
Thus, there is a gap between information designers and information receivers to begin with (to illustrate: the average scientific article has a Flesch Reading Ease score of 20-30, whereas the average adult reading level is 65). Because the information providers are skilled readers and likely are surrounded by a highly educated environment, they may lose awareness of the specifics of the target group that they provide the information to (J.B. Green et al., 2003). In general, writers of informed consent material will target an audience that is less educated, and is likely less experienced in reading. This target group then, requires a different communication approach than the information providers are used to employ.
In addition, although information designers might at best have received some form of training on informed consent writing, they remain medical practitioners and will not easily become communication professionals. This brings us to the second cause of the readability gap: a lack of basing communication efforts on scientific evidence and theory, leading to communication based merely upon clinical intuition (J.B. Green et al., 2003;
Jaaniste, Hayes, & von Baeyer, 2007). From our readability study in Chapter 3 we can derive that currently used consent forms fail to rely upon established fundamental theories from the field of communication studies on principles for creating optimal understanding.
An important and thoroughly tested theory in this respect is the cognitive theory of multimedia learning (Mayer, 2001). According to this theory, information is processed in two distinct channels, auditory/verbal, and visual/pictorial, each with a limited processing capacity. When communication input exceeds the capacity of a processing channel, this so- called cognitive overload can reduce the amount of information taken up and understood by the receiver (Mayer & Moreno, 2003). As medical research information often covers complicated concepts and explanations of various different topics, preventing cognitive overload can be a challenge. This is even more so when only one channel is used: the lack
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of visual support in these forms shows complete neglect of the visual/pictorial channel.
When this neglect is combined with poorly readable text, it is of no surprise that several studies have shown poor understanding among research participants (Barrett, 2005;
Burke et al., 2005; Chappuy et al., 2008; Ondrusek et al., 1998; Swartling et al., 2011; Tait et al., 2007; Unguru et al., 2010).
Based on the cognitive theory of multimedia learning, we hypothesized that a promising opportunity to improve current informed consent practice is to address the visual/pictorial channel by making images an integral part of the information. This was confirmed in our evaluation with children (Chapter 3), who indicated that they were capable of reading nonfiction books which required a high reading level, provided that the text was accompanied by supporting visuals.
Evidence from communication studies shows that visuals have the potential to not only improve comprehension, but also to increase attention, recall, and adherence (Houts et al., 2001). Our results confirm this evidence and add that there are also other benefits of including visuals in information for children: it increases the initial motivation to read a text, the motivation to make an effort when complicated concepts are discussed, and gives a feeling of comprehensibility about the information (Chapter 3). All these factors can contribute to an improved understanding and appreciation of the information.
It is remarkable that the majority of informed consent documents analyzed do neither comply with readability standards, nor with evidence-based use of visual support. An important opportunity for facilitating comprehension is completely neglected, resulting in poor informed consent practice. We plea for increased awareness among research information providers about targeting the intended audience, and to exert evidence-based communication. In order to facilitate this in practice, we will present recommendations for targeting minors later in this chapter.
Development of new, evidence-based research information
In order to address the shortcomings in current research information, and to empower children in decision-making about research participation, we have developed new information material targeted at the needs of minors. Chapter 4 and 5 in Part II of this thesis describe the various developmental stages, starting with an initial top-down approach by communication experts with a medical background, followed by a bottom-
up approach with end-user participation (Houts et al., 2006). The new information material consists of two parts: (a) a comic strip explaining general research concepts, and (b) a form conveying the specifics of a research study. The latter form functions as an example of how specific research information can be developed in conjunction with the comic strip, so that they can reinforce each other. We prepared a form specifically adapted for the Sophia biobank study, in which the novel material was tested.
The material was based on evidence about effective communication methods. A comic strip format was chosen as it combines text with pictures and a storyline, which makes it an ideally suited format to communicate with young patients (M.J. Green & Myers, 2010; McNihol, 2014). Comic strips have a strong potential besides conveying factual information, as is done in current textual forms. A storyline appeals to children and is a way to create a connection between multiple topics (Barnett et al., 2005). As the reader relates to the character in the story, the information becomes more personalized and the reader is more involved in the information (McNihol, 2014). As stated above, the combination of text and pictures reduces cognitive load and offers the opportunity to explain complicated information in an accessible manner (M.J. Green & Myers, 2010;
Mayer & Moreno, 2003). Also, the text was kept as plain as possible, eliminating medical jargon and redundant explanations (J.B. Green et al., 2003; Houts et al., 2006).
On participatory development
Initially, the topics for the comic strip were chosen in a top-down approach, as the material needed to comply with the current legal requirements of informed consent, and therefore needed to contain certain topics, like voluntariness, consent, and randomization (CCMO, 2008a). Subsequently all end-users (children, parents, pediatricians, researchers and research nurses) were involved in various stages in the design, as is discussed in Chapter 4 and 5. The participation of the target group in the design is a promising means to increase insight in their information needs and perspective (Rudd, 1994; Lee et al., 2013).
Although participation is gaining popularity in various areas, such as commercial product development (where it is referred to as co-creation) and social research (Dedding, Jurrius, Moonen, & Rutjes, 2013), it is not commonly embedded in healthcare practice (Weil et al., 2015). We believe that this is an overlooked strategy for improving information provision and empowering patients in a medical context. Involving minors in information development is a solution for addressing the gap between information providers and
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receivers. It increases understanding of the target group, their preferences and interests and perspectives, and can lead to valuable insights that would not have been gained without them (Dedding et al., 2015; C. Hart & Chesson, 1998; Lansdown, 2001). This was also the case in our study: children identified elements in the information material that were inadequate or were confusing to them, even though the adult developers were convinced that these parts were clear. The children challenged us to leave our comfort zone and imagine their perception of medical experiences, by telling personal stories and sharing opinions. A powerful example of how children’s participation was essential beyond adult expertise is the recurrent request for more humor in the storyline. Initially, when adult end-users were consulted, both research nurses and parents remarked that the tone was too informal and that they had doubts concerning the funny parts of the story, ‘because informed consent is a serious matter’. However, every time we discussed the material with the children, they requested more humor, because otherwise the story would be ‘too serious’ and thereby ‘boring’, and they would not feel like reading it at all.
Another example is the remark from various adults that not all complex topics needed to be covered in so much depth, particularly the explanation of placebos was considered redundant by some. Yet, children specifically mentioned to be contented that they now understood this concept and were interested to learn more about it.
These examples indicate that without participation of the target group itself, it is impossible to gain a full understanding of their perspective. Participation is thus a vital element in solving the current issues concerning research information provision and in ensuring that information material is optimally targeted to minors.
Limitations of participatory development
Despite its merits, it may not always be possible or desirable to achieve the highest level of participation in tool development, as is also demonstrated in our study. Some stages of design may require the expertise of communication or design professionals.
This was the case for our first stage of development. It would have been interesting to start off with a participatory stage and let children have a say about the contents of the information, because it is possible that the information provided does not match with the information desired by the audience, as also became apparent during our focus group discussions with research nurses. However, since we needed to implement all legally required elements of the information, this was not possible in our project.
Also, information material designers may be inexperienced in granting influence in material development, as it is not commonly used in healthcare yet (Weil et al., 2015).
More training and guidelines are needed to support participation practice and to ensure that results are indeed implemented in the final products.
In addition, it may be impossible for research practitioners to involve children on the scale reported in this thesis, due to time limits. In order to assist those designing information material, more empirical evidence is needed about the optimal ways to target the information material to children. Aspects that need to be addressed are: which format do children prefer; is interactive multimedia use indeed effective in increasing understanding in minors (Tait et al., 2015); what is the optimal readability level for documents with supporting visuals (i.e. this level can thus be higher than for text without visuals, but how high?); more insight in the specific characteristics of effective visuals (e.g. as has been studied in a systematic approach for low-literate populations by Van Beusekom, Bos, Wolterbeek, Guchelaar, & Van den Broek, 2015) .
Finally, it should be noted that there are limits to child participation already from theory, as is indicated in the so-called Ladder of Hart (R.A. Hart, 1992). The highest achievable level of participation is that of shared decision-making. It follows that the emphasis of participatory research does not lie in fading out all influences from adults, but rather in achieving a context of teamwork in which justice is done to the input from the children.
Recommendations for informed consent material
Guidelines on writing research information and other medical information material are necessary in order to address the aforementioned readability gap. In the Netherlands, the Central Committee on Research involving Human Subjects (CCMO) provided a writing guide for research information, addressing basic writing tips on length, syntax, wording and readability (CCMO, 2008b). However, this guide is aimed at adults, and only mentions that text should be ‘further simplified’ for minors younger than 12 years old (CCMO, 2008b, p.1). No advice is provided for preparing pediatric documents.
The same is true for the ‘Second Evaluation of Research Involving Human Subjects Act’, which contains advice to write clear texts for adults, but no word is spent on the pediatric situation (Stukart et al., 2012). We will address this hiatus based on our obtained insights in how to improve information for minors. In Box 1, these insights are presented as recommendation to assist those involved in drafting information for children and adolescents. Although these recommendations are based on our research with minors,
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we can also give more general advice for all medical practitioners who communicate with patients, young and old. First, gaining insight in the target group is a valuable method for developing appropriate communication strategies. Second, the use of visuals has the potential to reduce cognitive load and can aid in comprehension, also in adult audiences.
And finally, when providing information, it is vital to evaluate whether the message has reached the receiver.
Box 1 – Recommendations for providing research information On the development process:
- Keep in mind the characteristics of the specific target group. Think about education level, interests, the target group’s need to know, right to know and desire to know;
- If possible, involve the target group in de development of the information, in order to increase a true connection and to ensure optimal interest and understanding.
On the contents:
- Check the readability of the text (through readily available online tools, e.g. Classical Readability Formula Calculator to be retrieved at http://www.lt3.ugent.be/en/tools/) and make an effort to keep the reading level as close as possible to that of the intended audience;
- Use plain language, avoid unnecessary jargon, explain were possible;
- Eliminate nonessential and redundant text in order to reduce cognitive load;
- Structure the information in order to make it more accessible to the reader and assist the reader in creating oversight; e.g. use subheadings, bullet points, boxes, and arrows;
- Use visuals to draw the reader’s attention and spark interest in reading the text;
- Use visuals in order to increase understanding and reduce cognitive load;
- Always accompany visuals by explanatory captions;
- Ensure that the supporting visuals are informative, rather than just decorative;
- Visual styles: use cartoons, or use realistic images (e.g. photographs or 3D images), unless for ‘scary’ topics (e.g. venipuncture);
- Include an element of fun if possible.
On information provision:
- Evaluate with the receivers whether they have (read and) understood the information;
- Allow the receivers to ask questions for clarifications.
Informed consent: the child as a moral subject
In Part III of this thesis we aimed to answer the question whether children can, want, and should be involved in the decision-making process, given that the informed consent procedure is adequate. In order to practice what we preach about engaging children, we have studied a tool to gain an impression of the child’s individual perspective and wishes on its involvement in the decision-making process. In addition, a literature review on the developing decision-making capacity of minors was described to answer the question to what extent minors can be involved in decisions about research participation. Lastly, a theoretical analysis was presented about the standpoint that children should be viewed as a ‘moral subject’, as written in the Doek report (Doek Committee, 2009).
Children’s perspectives
To understand to what extent minors want to be involved in decision-making, we need to look at the perspective of minors themselves. Understanding their preferences and perspectives can give new insights in how to best involve them and treat them as actors in the decision-making process (Clavering & McLaughlin, 2010; Virkki et al., 2014). To this end, an exploratory study was performed in which participants in an ongoing biobank study were consulted about their experiences and opinions of the informed consent process, as described in Chapter 6. Semi-structured interviews were performed with the use of a tentative decision guide, that was designed based on previous research by Tait (Tait et al. 2003) and Lipstein (Lipstein et al., 2013). The goal of this decision guide is to increase optimal participation practices in decision-making, and its application is twofold.
First, it can be used as a tool in research to add to the body of evidence on children’s perspectives on decision-making; and second, it could be used in clinical practice, to gauge the individual minor’s understanding, perspective and satisfaction in the decision- making process, and it may thereby aid in tailoring the situation to the individual child or adolescent.
In our pilot study (Chapter 6), the decision guide was an effective tool to gain more insight in the perspectives of the consulted adolescents. Especially the pie chart exercise, in which adolescents indicated the role division in the decision that had been made, and the preferred division for a future decision, appeared to be a fruitful starting point for a more elaborate discussion. The consulted adolescents did indeed want to be involved in decision-making about research participation, because it was a decision about their
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own body and they therefore felt entitled to be involved. However, they did not want to decide by themselves, but rather in a shared decision-making process together with their parents. The exact extent of sharing varied between individuals, ranging from wanting to discuss with the parents and then decide for themselves, to relying upon the parents to interpret the information and to propose a decision, which the adolescent then could confirm.
A surprising outcome of our exploratory study was that a large part of the adolescents were not very interested in the provided (legally mandatory) information and instead preferred to turn to their parents for an explanation about research participation. This outcome implies that in the endeavor to adequately inform children and adolescents, we might need to look beyond the information material specifically provided for minors alone. When decision-making is a shared process between parent and child, we may also need to take into account the quality of the information supplied to the parents, and examine their understanding of the research at hand. Future studies should analyze these aspects, as well as the interaction between the parents and the child, in order to gain a better understanding of exactly how children are informed by their parents, and how parents can be supported to be a reliable information supplier. Parents in our research indicated that they wanted to use the comic strip as a family conversation support; further research should aim at optimizing information material for this function. In addition, more studies investigating the perspectives of minors on their role in decision-making should be performed, in order to confirm and complement our exploratory findings and to yield more generalizable results.
The capacities of children for medical decision-making
In Chapter 7 we have presented a literature review of the developing decision-making capacities of minors. To our knowledge this is the first multidisciplinary review on this subject, combining insights from neuroscience, psychology, decision-making sciences, medical sciences, and ethics. We have demonstrated that the four capacities that are required for medical decision-making (communicating a choice; understanding;
reasoning; appreciation; Appelbaum & Grisso, 2001) are sufficiently developed around the age of 12 years old. This result is in accordance with recent empirical research among pediatric patients, in which children from around the age of 11.6 generally appeared to be competent to decide on research participation (Hein et al., 2014). The age of 12 also corresponds with current Dutch legislation for shared informed consent for research
participation (Medical Research (Human Subjects) Act, 1998). Between the age of 12-18 both child and parents have to give written consent to research participation. However, at around the same age a new phase in (brain) development begins: adolescence. This phase is characterized by an early development of the reward system in the brain, combined with a late development of the control system. As a result, decision-making capacities under emotional circumstances are diminished, and although the adolescent might possess the skills to decide, the outcome in practice might be affected. Due to the emotional factor, it is to be expected that treatment decisions are more likely to be affected than decisions about research participation. Still, the consequences of the developing brain for the informed consent process will need further empirical examination.
Factors influencing decision-making competence
Many factors are reported in the literature that can influence decision-making competence.
As already discussed, the adequacy of provided information has a major effect on the capacity of a participant to understand the issue about which a decision should be made (Hein et al., 2012). In addition, internal factors such as stress, anxiety, and coping behavior may play a role (Alderson, 1992). But also external factors may impact the role that a minor can play in the decision-making; parental support, the attitude of professionals and the doctor-patient relationship can all play a significant role in bringing out the full potential of minors, or reducing it (Alderson, 2007; Sturman, 2005). It is thus clear that decision- making competence is a developing trait that is not solely defined by age, but is also dependent upon circumstances, and may differ per situation. This makes involving minors in decision-making complex, and requires a situational perspective on the best way to involve the individual child (Coyne & Harder, 2011; Lipstein et al. 2015). More research is necessary to identify the impact of the various influencing factors in the decision-making process, and to guide best practices on involving children.
On how to view children
In Chapter 8 two common views on children and their moral status were described. First, the “child as not-yet-adult” view defines a child as vulnerable, in need of protection, and depending upon adults to act in its best interest (Unicef, 1989, Art. 3.1 and 18.1).
This view is also represented in the Meijers report, which recommends a “no-unless”
stance concerning research with children, and describes children as ‘incapable of a reasonable appreciation of their interests in the case at hand’ (Meijers Committee, 1995,
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p.32). However, this view does not do justice to the potential of children as moral agents (Alderson, 1992; Dedding, 2009; De Vries, 2015; Kuther & Posada, 2014). By defining the child as being in an inferior stage towards the ultimate goal of adulthood, we hinder it to live up to its full potential (Alderson 1992, 2007; Kuther & Posada, 2004; Martenson &
Fagerskiold, 2008).
We argue that, instead, we need to adopt the view of the “child-as-person”, with its own characteristics and perspective on the world. Although this seems somewhat similar to the attitude of the Doek Committee to view children as ‘moral subjects’, we take it a step further and plea for a more fundamental change. The ‘moral subjects’ argument by Doek is hardly based upon scientific evidence or on consultations with children, but rather appears to be based on intuitive insights from pediatric professionals. Paradoxically, this shows characteristics of the view of children as “not-yet-adults”: a standpoint about children is formulated without thoroughly consulting and involving children themselves, or relying upon evidence from research with children. Recognizing the “child-as-person”
implies engaging with children to understand their perspective, potential and needs.
It includes the awareness that children are the experts when it comes to their personal situation, preferences and goals (Dedding, 2009; C. Hart & Chesson, 1998). Children do have the potential to make reasonable decisions (Coyne & Gallagher, 2011; Hein et al., 2014). Only when we engage with children will we be able to understand how they can be involved in decision-making upon research participation, and can we make adequate guidelines for medical practice that empower children.
In other words, we applaud the dismissing of the standpoint represented in the Meijers report, that children are merely vulnerable beings in need of protection. We should allow children to participate and have a say in matters that concern them, as is also described in the Convention on the Rights of the Child (Unicef, 1989, Article 12). However, at present there is insufficient evidence to guide participation of children in the decision-making process (Clavering & McLaughlin, 2010 ; Coyne & Gallagher, 2011; Ruhe et al., 2015). We need to gain an understanding of the capabilities of children when it comes to medical decision-making and increase insights in the preferences of children for participation and the best ways to support their involvement.
Questions that need to be addressed are (among others): do children want to be involved in the entire process of decision-making, or do they prefer to be involved only in certain stages or in certain (aspects of the) decisions (Coyne & Gallagher, 2001); and, how can dynamics in conversations with the doctor and or research nurse be adapted in such a
way that the minor is adequately addressed and feels part of the conversation (Tates, Meeuwesen, Elbers, & Bensing, 2002). In this thesis, we aimed to answer the more fundamental questions about the preferred role in decision-making of minors.
Recommendations for involving children and adolescents
In order to translate our results and our experiences with children and adolescents into applicable advice for clinical practice, we provide a number of recommendations for involving children in decision-making about research participation (see Box 2). Further research should confirm, complement or specify these recommendations.
Box 2 – Recommendations for involving children in the informed consent procedure - Be aware that communication is often not targeted at children;
- Adequate information supply is a prerequisite to further involvement;
- Evaluate understanding of the information;
- Give children time and space to ask questions or propose improvements;
- Insight in the child’s perspective facilitates communication and the ability to interpret its answers and behavior;
- There are individual and situational differences in capacity and preferences, use the decision guide to assess the specific situation of the child;
- Engage with children instead of talking about them;
- Be prepared to act upon the input of the child: not only consult out of good intentions, but respect the input and implement it where possible.
Conclusion
In this thesis, the issue of involving children in decision-making about participation in medi cal research is addressed. This thesis contributes to answering the questions whether children can, should and want to be involved in decision-making. Novel insights on the role of minors in the informed consent process were collected. These insights can guide the current discussion and developments on stimulating more pediatric research and involving minors in decision-making about research participation (Doek Committee 1995; WHO, 2013). The underlying aim of our research was not in the first place to accomplish an increase in medical-scientific research with minors. Rather, given the current
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developments of allowing more pediatric research, our aim was to empower children in the decision-making process about research participation. As an ultimate consequence, this might even lead to a reduction in research inclusion, as it is unknown whether more informed and more involved minors will be more inclined to consent, or will decide to dissent. Regardless, empowering minors as potential research participants is an important ethical aspect of informed consent for medical-scientific research.
We conclude that we should adopt a view of children as persons, with whom we need to engage to understand their perspective and to be able to empower and involve them in decisions regarding their personal situations.
We have demonstrated that children from the age of 12 might be capable of participating in research decisions. However, adequate information supply is a prerequisite for this participation and our results show that current information practice is insufficient to create understanding and bring out the full potential of minors. To empower minors in informed consent practices, new research information material was designed in an innovative approach by involving children and other end-users in the development. To our knowledge, this is one of the first studies in which children were consulted about the decision-making process for research participation, and in which children are involved in the development of new material.
Next to the influence of adequate information on the capacity of minors to be involved in decision-making, there are also environmental factors that play a role. In addition, there are individual differences between minors in their preferences to be involved in the decision. Therefore, involving minors in the informed consent process requires a situational perspective and an individual approach. To this end, we have tested a tentative decision guide that may be used in practice, after further study, to guide a more targeted approach towards involving children. In addition, the decision guide may be used for future research on children’s perspectives, as our study has demonstrated the urgent need for more research on the most effective ways to support minors to participate in decision-making about research participation.
Future directions
More research is necessary to identify how the informed consent process can be further optimized. Our study has indicated a lack of evidence on how to best inform minors about medical-scientific research. We have mainly looked at the information material,
but verbal explanations and conversations also play a role; the quality and content of the verbal aspect of the consent process needs further studying. In addition, as our results suggest that minors rely on their parents for information and explanations, it is worth studying the information transfer between parents and children and the reliability of the information that parents provide. Also, other factors can influence understanding, such as anxiety, severity of the diagnosis and timing of the information. More research is necessary to identify the impact of these factors and the ways in which negative effects on understanding can be addressed. Also, more studies should be performed with children of various age categories, in order to be able to draw more generalized conclusions about the best way to present research information to minors. We plea for more evidence on the perspectives and preferences of minors in the informed consent process, and to identify ways to optimally involve and support minors in research practice. Further research should be based on recognizing the potential of children and should hold a “child-as-person”
view in approaching minors. It goes without saying that respecting children as persons entails the adoption of meaningful levels of child involvement in research studies (level 5-8 of the participation ladder) and a rejection of non-participation practices (level 1-3).
