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
Empowering young participants in medical-scientific research
Petronella Grootens-Wiegers
Petronella Grootens-Wiegers
[dossiernummer 113203016].
Het onderzoek in dit proefschrift werd uitgevoerd aan de Universiteit Leiden, afdeling Science Communication & Society, Leiden, Nederland.
Ontwerp omslag en lay-out: Esther Scheide, www.proefschriftomslag.nl Drukwerk: Ridderprint, Ridderkerk
ISBN: 978-94-92332-15-8
© 2016 P. Grootens-Wiegers
Proefschrift
ter verkrijging van
de graad van Doctor aan de Universiteit Leiden, op gezag van de Rector Magnificus Prof.mr. C.J.J.M. Stolker,
volgens besluit van het College voor Promoties te verdedigen op dinsdag 6 december 2016
klokke 15.00 uur
door
Petronella Grootens-Wiegers
geboren op 1 oktober 1986 te Hengelo
Promotor:
Prof. Dr. J.M. van den Broek Universiteit Leiden
Copromotor:
Dr. M.C. de Vries Leids Universitair Medisch Centrum
Overige leden:
Dr. C.W.M. Dedding Vrije Universiteit Amsterdam Prof. Dr. Mr. M.E. Kalverboer Rijksuniversiteit Groningen &
Bureau Nationale Ombudsman Prof. Dr. Ir. I. Smeets Universiteit Leiden
Prof. Dr. H. Spaink Universiteit Leiden
Prof. Dr. H.F.M. te Molder Wageningen University & Research Dr. A.M.C. van Rossum Erasmus MC Sophia, Rotterdam
Dr. I.M. van Vliet Leids Universitair Medisch Centrum
Chapter 1 General Introduction
PART I – Research Information for Minors: Current Practice
Chapter 2 Research Information for Minors: Suitable Formats and Readability.
A Systematic Review
Chapter 3 Readability and Visuals in Medical Research Information Forms for Children and Adolescents
PART II – Participatory Development of Research Information for Minors Chapter 4 Comic Strips Help Children Understand Medical Research
Targeting the Informed Consent Procedure to Children’s Needs Chapter 5 Information for Minors Participating in Research Projects:
Current Practice and Evaluation of a New Format
PART III – The Child as Moral Subject
Chapter 6 ‘It was my own decision and that of someone else […] in fact, 100% of the three of us.’ Perspectives of Adolescents on Decision-Making about Clinical Research Participation
Chapter 7 Child Development and the Neuroscience of Medical Decision-Making Chapter 8 Recognizing the Capacities of Minors in Medical Decision-Making:
Not Tokenism but Engaging and Empowering Chapter 9 General Discussion
Addendum References
List of Abbreviations Nederlandse Samenvatting Samenvatting voor Kinderen Curriculum Vitae
Dankwoord Publications
7
21 23
45
73 75
93
105 107
123 147
161
183
201
203
215
221
223
227
General Introduction
The need for pediatric research
Scientific progress in pediatrics is urgently needed, as currently 45-60% of medication is prescribed off-label, meaning that the medication for children and adolescents is not tested in a clinical trial with the specific target group for which it is prescribed (Conroy et al., 2000; ‘t Jong et al., 2000; WHO, 2013). In pediatric practice this has several consequences (WHO, 2013):
- Drugs are prescribed to children, although a clinical trial has only been performed with adult patients;
- Medication is tested for one disease and subsequently prescribed for another;
- Prescribed dosages for children are not based on scientific evidence.
This situation is problematic, as the physiology of children’s developing bodies is different from that of adults (De Wildt, Tibboel, & Leeder, 2014). As a result, medication proven effective and safe in adults could be ineffective or even harmful in children. It is thus vital that more insight is gained in the physiology, disease pathology and treatment opportunities for children and adolescents. To this end the World Health Organization has issued a report on Priority Medicines for Children with a strong plea for more research with minors (WHO, 2013).
However, the possibilities to perform research with minors are more limited than for research with adults, as children are a protected research population. In the Netherlands it is currently only allowed to perform so-called therapeutic research with minors, meaning that the trial could be of direct benefit to the subject. Non-therapeutic research is possible only when a trial could not be conducted without the participation of the minor as a research subject, and provided that the risk associated with participation is negligible and the burden is minimal (Medical Research (Human Subjects) Act, 1998, Section 4).
These restrictions result in a limited scope of research that can be performed with minors, as research procedures that can advance scientific insights do not always directly benefit participants, or do place more than minimal risks and burden on the subject.
Attitudes towards involving minors in research
The dilemma of research needs on the one hand and protection on the other is addressed
in a recent Ministry of Health-commissioned advisory report on research with children,
presented in 2009 by the Doek Committee (Doek Committee, 2009). According to this report, changes need to be made in current regulations for research with minors.
Specifically, the age to decide upon research participation should be lowered (from 18 to 16 years old), and the limits restricting the level of risks and burden of research for minors should be loosened. According to the committee, these changes would lead to the much needed and desired increase in research possibilities with minors.
The report of the Doek Committee signifies a shift in the attitude towards research with minors. A previous commissioned report on research with children from the Meijers Committee (Meijers Committee, 1995) was based on a restrictive “no-unless” attitude;
no non-therapeutic research should be performed with minors, unless the research is absolutely necessary, cannot be performed with adults, and places negligent risks and minimal burden upon the young participants. This report was the basis for the current Dutch regulations. In contrast to the Meijers report, the report of the Doek Committee holds a “yes-if” attitude towards research with minors. Instead of the strict distinction between therapeutic and non-therapeutic research, the Doek report advises that interventional medical research may be performed with minors provided that there is at least a chance that the subject may benefit from participation, or that there is some direct benefit for the group to which the participant belongs. Observational medical research, which commonly does not provide direct benefits, may be carried out if it places ‘minimal’
(instead of ‘negligible’) risks and burden. Evaluation of the risks and burden of research participation should include the particular circumstances of the category of children and should be proportional to the importance of the research.
This change in attitude originates from a shift in the way minors are viewed. In the Meijers report, children under the age of 12 are considered incapacitated, and children older than 12 could possibly be deemed competent, but need to prove this in a competence assessment (Meijers Committee, 1995, p.32). The report recommends that consent for research participation should be sought from a representative, and the researcher should inform the child at the proper level of understanding (Meijers Committee, 1995, p.32).
It is thus clear that the Meijers report focuses on the incompetence and protection of minors and thereby they are assigned a passive role in the decision-making process.
The Doek report, however, claims that we should no longer view children as moral objects, i.e. objects in need of protection, as this does not do justice to the children.
Rather, we should view them as moral subjects, i.e. children are agents and have their own
conceptions and feelings (Doek Committee, 2009, p. 26). The child as a moral subject should be allowed to act according to its personal values, insight and will, and should thus be provided with the opportunity to participate in medical research and to be involved in the decision-making process about research participation (Doek Committee, 2009, p.33).
It is remarkable that in a timespan of only 15 years the attitude towards children has changed so significantly. Where does this change come from and what evidence do we have supporting this changing attitude? Is it indeed legitimate to view children as moral subjects instead of objects, and if so, what are the consequences for research practices?
Little research evidence exists concerning the role that children can and want to play in decision-making about medical-scientific research. Increased insights are necessary in order to justify the changing attitude towards children, and to understand how children can truly be involved to support best practices.
Background of this thesis
An amendment of the Dutch law based on the Doek report would facilitate an increase in medical-scientific research with children in the Netherlands, and indeed the Dutch Parliament seems to be willing to grant substantial changes in the Dutch law
1. However, a number of issues arise concerning this new attitude towards children. These concern (1) informed consent as a prerequisite for research participation, and (2) the claim that children should be considered as moral subjects.
1. Informed Consent as a prerequisite for research participation
With regard to involvement in the decision about research participation, there is a rather unique situation in the Netherlands (Lepola et al., 2016). Next to the internationally recognized distinction between adults who have the right for self-determination, and minors (and other incapacitated groups) for whom decisions need to be executed by representatives, there is a third situation when it comes to medical decision-making.
1
An amendment is at the time of writing under discussion by the Dutch Senate, to be retrieved at:
https://www.eerstekamer.nl/wetsvoorstel/33508_verrichten_van_medisch
Children from the age of 12 until 18
2are assigned a role in shared decision-making about research participation together with their parents or legal guardians
3. This means that they have an official role in the so-called informed consent process, a necessary require- ment for research participation in order to protect research participants from unethical practices. The requirements for informed consent stem from various guidelines, among which the Nuremberg Code (1949) and the Helsinki Code (originating from 1964, revised multiple times: World Medical Association, 2013). The core value of these guidelines is the protection of research participants and the ethical consideration that the interest of the research participants supersedes the interest of the research itself. All research guidelines have in common that they state that research with humans is only allowed if (1) the research setup is scientifically sound and approved by an Institutional Review Board; (2) participation is voluntary; and (3) informed consent is provided.
In the Netherlands, research participation is regulated by the Dutch law on medical research with humans (Medical Research (Human Subjects) Act, 1998), which in accor- dance with the previous advice from the Meijers Committee, holds a “no-unless” attitude towards research participation of minors: research with persons younger than 18 years old is prohibited, unless it can benefit them. Non-therapeutic research is only possible when the research cannot be conducted without minors and there is negligible risk and minimal burden (Art. 4). If research is performed with children from the age of 12, then they should provide written consent, together with their parents. In Article 6, it is specified that before asking consent for participation, information should be provided on the purpose, nature and duration of the research and the risks for the participant (Art. 6.5), and that it should be ensured that the participant can understand this information (Art 6.6). It thus follows that adequately informing young research participants is an essential requirement for obtaining informed consent, and thus for research participation. Without proper understanding of what it is that someone signs up for, we can neither speak of informed consent, nor of actual voluntary research participation.
2
At the time of writing an amendment to change the age limits for shared consent to 12-16 has been approved by the Dutch House of Representatives (Tweede Kamer) and is under discussion in the Senate (Eerste Kamer; file 33.508).
3
In the remainder of the text, whenever there is mention of ‘parents’ one can also read ‘parents or
legal guardians’.
In practice, this informed consent procedure translates into the provision of an ‘informed consent form’ consisting of several pages of written text concerning the specifics on the research, sometimes accompanied by additional oral explanation. For children in the age range of 12-18, a special form is provided next to the form that the parents receive. This special form is supposed to be directed at the level of understanding of the minors.
However, it is questionable whether the special forms indeed create comprehension, as studies on children participating in research demonstrate a lack of understanding of essential research concepts. For example, in one study, 76% of the children did not understand the risks related to participation (O'Lonergan & Forster-Harwood, 2011), 86%
reported not to understand what the doctor told them about the research (Unguru, Sill,
& Kamani, 2010) and 51% of the children was even unaware that the treatment they underwent was in fact part of clinical research (Unguru et al., 2010). Other studies have reported that children often feel excluded from doctor-patient conversations, because doctors tend to direct their explanations to the parents, and because the language is too complex and too technical (Broome, Richards, & Hall, 2001; Chappuy, Doz, Blanche, Gentet, & Tréluyer, 2008; Kodish et al., 2004; Tates, Meeuwesen, Bensing, & Elbers, 2002).
These results indicate a fundamental problem with the informed consent procedure among minors: it appears that current practice is not up to standards with the ethical intent to inform research participants at the proper level of understanding and stimulate an informed and voluntary decision.
2. The child as moral subject
According to the Doek Committee, children should be granted influence in decision-
making about medical-scientific research participation and should be allowed to act
according to their personal values, based on the notion that they should be considered
a moral subject. In the Doek report, however, there is hardly any justification for the
fundamental shift from viewing the child as an object of protection to viewing the child
as a moral subject. The main evidence that is provided for this shift is a research study
of Sokol et al. (Sokol, Chandler, & Jones, 2004). In this research an analysis is presented
of children’s moral judgements, demonstrating that children initially have an objective
view of moral responsibility (i.e. actions are either ‘good’ or ‘bad’ in itself), which then
during development changes into a subjective notion of responsibility (i.e. the intention
of an action is also considered when judging an action as ‘good’ or ‘bad’). Sokol does
not mention the concept of a ‘moral subject’ or ‘moral object’, but discusses the type
of morality that children possess. It is remarkable that such a big shift in thinking as proposed by the Doek report (that is even responsible for an amendment in the law) is not supported by a body of sound scientific evidence. It appears as if the Doek Committee has based her recommendations mostly on intuitive expert opinion.
Aims
If we are to involve more children in research, and especially if we are to involve children more in research decision-making, we need to make sure that the informed consent process is ethically sound. Given the lack of understanding among young participants in other countries, as mentioned before, it is likely that a similar problem exists in the Netherlands.
In order to guarantee an adequate informed consent process as prescribed by the Dutch law, we need to gain more insight in the quality of the current informed consent process and materials for minors in the Netherlands. In addition, the currently used information format for minors is similar to that of adults (i.e. several pages of plain text), whereas information needs and preferred communication formats differ between children and adults (Broome, 1999). Therefore, new informed consent material should be developed specifically targeted at children and adolescents, in order to achieve optimal understanding in minors and empower them to make an informed and voluntary decision about research participation.
The legitimacy of the claim that ‘children should be viewed as moral subjects’ should be
investigated. Is there evidence supporting this claim? And are children indeed capable
of decision-making in a research context? Moreover, what can we learn from children’s
experiences during the informed consent process, and what are their preferences for
involvement? In other words: what is the role that children can play, should play and want
to play in decision-making on participation in medical-scientific research. These issues
will be addressed in this thesis from a theoretical and empirical angle, in order to lead to
recommendations on involvement of minors in the informed consent process.
Research Questions
Following the above, three central research questions were formulated:
1. How can research information material be targeted to minors in order to empower them to participate in decision-making?
2. What is the role that children can, should and want to play in decision-making concerning participation in medical-scientific research?
3. Is the change in view on the status of the child in medical-scientific research (from moral object to moral subject) legitimate?
In order to answer these questions, the following subquestions will be addressed:
1. Is there scientific evidence on the quality of research information for minors?
(Chapter 2)
2. What is the quality of currently used research information material in the Netherlands? (Chapter 3)
3. What are children’s preferences and needs concerning text and supporting visuals in research information? (Chapter 3)
4. How can novel information material be adapted to optimally connect to the preferences of the end-users (i.e. children, adolescents, parents, research nurses, pediatricians)? (Chapter 4 and 5)
5. What are the perspectives of minors themselves on their role in decision-making about medical-scientific research? (Chapter 6)
6. At what age can minors be expected to be competent for medical decision-making?
(Chapter 7)
7. What view on children should we adopt in the context of participation in medical decision-making? (Chapter 8)
Target group
The study target group is “minors”, in this thesis loosely defined as children and adolescents
younger than 18 years old, in the Netherlands. In our evaluation of the competence of
minors in medical decision-making, we have looked beyond the juridical boundaries of
involving children from the age of 12, in order to focus on the (developing) capacities
and possibilities of children (Hein, Troost, & Lindeboom, 2014). For the development of novel research information material, we have focused on children in the age category from 10-14 years old, as research has indicated that this is a distinct target group in terms of understanding research information. Children younger than 10 years old seem to be incapable of understanding certain essential research concepts, whereas children older than 14 seem to have a similar understanding to that of adults (Hein, Troost & Lindeboom, 2014; Ondrusek, Abramovitch, Pencharz, & Koren, 1998; Weithorn & Campbell, 1982).
Methods
In order to gain more insight in the quality of patient information forms for minors (sub- question 1), a systematic literature search was performed. Two aspects of quality were analyzed: the effect of format on understanding and the optimal readability of text in the documents.
To address the quality of research information for children in the Netherlands (subquestion 2), text readability and the role of visuals in the forms were assessed.
In order to increase our understanding of how to optimally connect research information to children (subquestion 4 and 5), we used a process of consultation of the target group and subsequent participatory development. This participatory method was chosen based on the notion that children are the experts on their own information needs and preferences and perspectives (Dedding, Reis, Wolf, & Hardon, 2015; Lansdown, 2001).
Participation of the target group is a means to obtain a better understanding of the target group and to ensure that the final product will reach the intended audience (C. Hart & Chesson, 1998). Participation is not an on-or-off phenomenon; it can be reached at various levels of intensity. According to Hart (R.A. Hart, 1992), there are eight levels of participatory involvement, described by the so-called Ladder of Hart. The levels vary from manipulation (i.e. pretend involvement, without real implications) to shared decision-making (see box 1).
In our study, we developed research information material that is of legal status and there-
fore we adopted an initial top-down approach (i.e. without involving the target group) in
order to ensure that all the required information would be present. Subsequently, children
were involved at level 5: children were consulted about their perspective and experiences
and their input was implemented. Due to the nature of the legally mandatory content for the new information material, level 5 was the highest participatory involvement possible in this study.
Levels of participation (R.A. Hart, 1992) 8. Child-initiated; shared decisions with adults 7. Child-initiated and directed
6. Adult-initiated; shared decisions with children 5. Children are consulted and informed 4. Children are assigned but informed 3. Tokenism
2. Decoration 1. Manipulation
Box 1. The Ladder of Hart (R.A. Hart, 1992). Level 1-3 are assigned as ‘non-participation’
Children were consulted in focus groups to identify their preferences and needs for text and supporting visuals (subquestion 3). This group form of interviewing was chosen, because it reduces the influence of the interviewer and thereby increases the influence of the children on the discussion outcome (Kitzinger, 1995). In addition, in a focus group the children can respond to each other, leading to a more in-depth discussion of the topics that they themselves consider important. Based on the results, new information material was developed in co-creation: feedback from end-users on the information material was collected by surveys, interviews and focus groups (subquestion 4).
The new information material was designed in the format of a comic book. Comic books combine written text with pictures and a story-line, which makes them ideally suited for education of young patients (M. J. Green & Myers, 2010). Pictures supporting written information are a powerful tool to increase comprehension (Mayer, 2001). Considering the story-line, Barnett observed that the use of a story-format for children significantly improves comprehension when compared to a standard text-format (Barnett, Harrison, Newman, Bentley, & Cummins, 2005). Comics have already shown their potential as a useful medium in school education and can improve patient education as well (M. J.
Green & Myers, 2010; Tjiam et al., 2013).
A draft of the comic strip was developed by a science communicator in collaboration with pediatricians. The draft was presented to children participating in a clinical trial and to two school classes. Children were consulted for further development in surveys and interviews. Subsequently, the material was revised and re-evaluated in four school classes with children of varying ages and educational levels. Next, the new material was evaluated in focus groups with research nurses of 8 Dutch medical centers and children participating in a clinical trial and their parents. Based on their recommendations, a final revision was executed in order to optimize the material for use in research practice.
An empirical study using semi-structured interviews was set up to gain insight in the perspectives of minors on their role in the decision-making process (subquestion 5). In order to discuss the role that children can play in decision-making, a multidisciplinary review was drafted, combining insights from neuroscience, psychology, ethics, decision- making science and medical practice (subquestion 6). Evidence on the developing brain in children and adolescents was reviewed and related to the capacities required for medical- decision making; (1) communicating a choice, (2) understanding, (3) reasoning, and (4) appreciation (Appelbaum & Grisso, 2001), in order to assess at what age minors can be expected to be competent to be involved in the informed consent process. In addition, an ethical background review was written, discussing the way we view children in the context of medical decision-making, and to identify how the debate can be advanced by recognizing the potential of children (subquestion 7).
Outline
The body of this thesis is divided into three parts.
Part I: If we are to involve more children in research, we need to make sure that informed
consent as the prerequisite for ethical research practice is performed adequately. There-
fore, in Part I the quality of the current informed consent for children and adolescents
is examined based on a systematic literature review (Chapter 2), the analysis of Dutch
information forms for minors, and focus groups with minors on their information needs
and preferences (Chapter 3).
Part II: We will discuss how to improve informed consent practice in Part II, in which the participatory development of innovative research information material for minors is presented (Chapter 4 and 5).
Part III: Finally, the change in attitude towards children in research as described by the Doek Committee will be evaluated. In Part III the role children can, should and want to play in decision-making about medical-scientific research is discussed from the perspectives of minors themselves (Chapter 6) and from a theoretical standpoint (Chapter 7 and 8).
In the discussion (Chapter 9), the insights from this research project will be combined and
recommendations will be given on how to empower minors in decision-making about
research participation.
?
ja nee
?
ja nee
Research Information for Minors:
Current Practice
Research Information for Minors: Suitable Formats and Readability.
A Systematic Review
Grootens-Wiegers, P., De Vries, M. C., & Van den Broek, J. M. (2015).
Journal of Paediatrics and Child Health, 51(5), 505-511.
Abstract
As children age, their capacity to consent or dissent to research participation in-
creases. Numerous regulations and guidelines require that children should receive
information ‘according to their capacity of understanding’. In order to gain more
insight in the quality of patient information forms for minors, a systematic literature
search was performed. Two aspects of quality will be analyzed in this chapter: the
effect of format on understanding and the readability of text in the documents. A
systematic search was executed in PubMed, Embase and PsycINFO. Seventeen
papers on format were included. Interventions testing information formats indicate
that improvement is possible, but outcome measurement varied per study and
no apparently successful intervention was repeated. Only three readability papers
were found, all indicating a readability gap between patient information forms
and children’s actual reading level. The results indicate an urgent need for further
research on how to adequately inform minors about clinical trials.
2 Background and Objective
Informed consent and assent are a major issue in the ethics of pediatric care, especially when it concerns parents’ and children’s consent for research (Bos, Tromp, Tibboel, &
Pinxten, 2013; Committee on Bioethics, 1995). As approximately 45-60% of medication is currently prescribed off-label to minors, clinical trials among children are necessary to improve medication outcomes (WHO, 2013). This implies that patients under the age of 18 can be confronted with the choice to participate in clinical research. Depending on age, children do not always possess the legal right to consent to research participation, but can however have the mental capacity to understand the implications of participation.
Since this capacity increases as children age, the ethical ideal of respect for the developing autonomy of children in making decisions comes into play (Committee on Bioethics, 1995). Numerous regulations require that assent or consent be obtained from children capable of providing it (Office for Human Research Protections, 2009). Article 12 of the UN Convention on the Rights of the Child states that “children shall be provided with the opportunity to be heard in any judicial or administrative proceeding affecting the child directly” (Unicef, 1989). Although this statement is not specifically aimed at medical situations, it follows that children should also be heard in the decision process for clinical research. The Second Directive 2001/20/EC by the European Parliament and the Council of the European Union, indicates that “A clinical trial on minors may be undertaken only if […] the minor has received information according to its capacity of understanding” (EU, 2001). These international regulations are translated to various national regulations in different countries. For example, in the Netherlands children from the age of 12 have the right to legally co-consent, together with their parents. In other countries, including the UK and the US, laws and regulations require that research participants who are not competent to provide consent, are informed, not with the primary aim to give true informed consent but from the moral principle of respect for persons and the more pragmatic aim to create commitment of the pediatric research participant (Alderson, 2007).
Although the variation between national laws and regulations, there is a rule of thumb
for information provision and age. Children under the age of 9 are not fully capable of
understanding concepts such as aim, benefits and risks of research (Gill, 2004; Ondrusek,
et al. 1998). Children from the age of 14 appear to have an abstract understanding similar
to that of adults (Kurz, Gill, & Mjones, 2006; Ondrusek et al., 1998), thus following that
children between 9 and 14 have a developing understanding of research participation.
Therefore, these minors should be addressed in such a way that they can comprehend what is involved in research participation.
Research information is offered by means of the patient information form, often accompanied by verbal explanations from a researcher, doctor, or nurse. Contrary to verbal explanations, the patient information form itself can be taken home and read again, allowing a child to take the time to process the information. Pediatric forms therefore play a pivotal role in informing children and supporting their decision to provide assent, consent or dissent.
In order to offer more insight in the quality of written information provided to children in pediatric research, two aspects of quality will be analyzed in this chapter: the effect of format on understanding and the readability of the text in the documents.
Format
Numerous studies indicate that consenting adult research participants do not under- stand essential research aspects, such as risks involved in participation (Falagas, Korbila, Gianno poulou, Kondilis, & Peppas, 2009). Therefore, the effect of different formats, such as simplified text and multimedia use, on understanding has been studied in adults (Dunn
& Jeste, 2001; Flory & Emanuel, 2004; Palmer, Lanouette, & Jeste, 2012). However, infor- mation needs and preferred communication formats differ between children and adults (Broome, 1999). In order to gain more insight in the specific modes of communication suitable for children, a literature search was performed.
Readability
Readability analyses of adult consent documents indicate without exception a large gap between the required reading level to understand the information and the actual reading ability of research participants (Berto, Peroni, Miller, & Spagnolo, 2000; Franck & Winter, 2004; Gribble, 1999; Kass, Chaisson, Taylor, & Lohse, 2011; Ogloff & Otto, 1991; Melman, Kaplan, Caloustian, Weinberger, Smith, & Anbar, 1994; Paasche-Orlow, Taylor, & Brancati, 2003; Raich, Plomer, & Coyne, 2001; Sanders, Federico, Klass, Abrams, & Dreyer, 2009;
Sudore, Landefeld, Williams, Barnes, Lindquist, & Schillinger, 2006; Terranova, Ferro,
Carpeggiani, Recchia, Braga, et al. 2012). In addition to the use of complex terminology,
the length of patient information forms is an obstacle for understanding (Kass et al.,
2011). Even surveillance by Institutional Review Boards does not guarantee adequate
document readability (Hammerschmidt & Keane, 1992; Paasche-Orlow et al., 2003). We
2
expect a similar readability gap in pediatric information material. In order to examine this hypothesis, a systematic literature search was performed.
Methods
A. Format
Research Question
Which way of presenting research information ensures understanding, satisfaction or fear reduction for children who are asked to participate in medical scientific research?
Search string
The search was originally executed in combination with a publication restriction for high quality evidence (reviews, meta-analyses, RCTs). As only four papers were included, the search was performed without publication restriction, in order to collect all available evidence. The search strings were executed in three databases: PubMed, EMBASE and PsycINFO, on May 2, 2013. These databases were chosen as they cover most of the publications in health care and information.
In- and exclusion
Inclusion was performed independently by the main researcher (PGW) and a second reader. Papers were excluded if the study was not performed with children or was irrelevant for the research question. Subsequently, all papers were read full-text, and eventually 20 papers were included. Three papers could not be accessed in the Netherlands; two conference proceedings and one conference abstract. In total, 17 papers were included, including 4 papers from the first search. A flow chart of the inclusion process is displayed in figure 1.
B. Readability Research Question
What readability level of research information leads to understanding, satisfaction or fear
reduction for children who are asked to participate in medical scientific research?
Search string
The search was executed in combination with a publication restriction: only high quality evidence (reviews, meta-analyses, RCTs) was included. As there were no papers included, the original search string was adapted to find all papers on the readability of pediatric research information (i.e. instead of evidence on target-group based preferred readability level). The search strings were executed in three databases: PubMed, EMBASE and PsycINFO, on May 2, 2013.
Figure 1: Detailed Flow Chart Consent Formats; No Publication Limit Total of 1397 papers
found
693 excluded: not about research on consent 59 excluded: ethical discussions on consent
410 excluded: adult consent
91 excluded: pediatric parental consent 13 excluded: focus on children’s competence 70 excluded: children in research, not consent 41 excluded: not format of consent
20 papers selected for inclusion
17 papers included
3 papers: no access
Figure 1. Detailed flow chart consent formats: no publication limit.
2
In- and exclusion
Paper in- and exclusion was performed independently by the main researcher (PGW) and a second researcher. Papers from the first search were excluded in case the research was not performed with children and when the research topic was not readability of research or hospital information. After exclusion, no paper remained. A number of papers on readability were found, but these only tested readability with readability instruments, and did not triangulate outcomes with the target group.
Only one paper investigated the actual readability for the intended target group, which consisted of adults (Sudore et al., 2006). An adapted search was executed for readability analyses of pediatric information forms. Papers were excluded when there was no actual measurement (e.g. qualitative interviews without readability analysis), when the topic was parental consent or ethical discussions, and when the study was irrelevant for the research question.
Selection
A total of 811 unique papers were found. Of these, 5 papers were selected for inclusion.
A large amount of papers were excluded, because no actual measurements were reported, or the analyzed documents were intended for adults or parents (see figure 2). Two papers were excluded full-text as they did not measure pediatric documents for children as target group. As a result, 3 papers were selected in which a readability analysis of research information was performed. The included papers were used as a starting point for snowballing for more literature. However, no other papers were found that could be included in our review.
Results
A. Format
A total of seventeen papers were found, of which eight focused mainly on understanding
of research information (Burke, Abramovitch, & Zlotkin, 2005; Chappuy et al. 2008; Dorn,
Susman, & Fletcher, 1995; Fogas, Oesterheld, & Shader, 2001; Hurley & Underwood,
2002; Ondrusek et al., 1998; Tait et al., 2003; Swartling, Hansson, Ludvigsson, & Nordgren,
2011) and nine investigated the effect of adaptations of the patient information form
or process (Adcock, Hogan, Elci, & Mills, 2012; Barnett et al., 2005; Carr et al., 2012;
O'Lonergan & Forster-Harwood, 2011; Shani, Ayalon, Hammad, & Sikron, 2003; Tait, Voepel-Lewis, & Malviya, 2007; Tait, Voepel-Lewis, McGonegal, & Levine, 2012; Ulph, Townsend, & Glazebrook, 2009; Wright, Fleming, Sharma, & Battagel, 2010). Papers were graded for level of evidence as indicated in table 1; a description of the evidence per paper can be found in table 2 (both at the end of this paper).
Figure 2: Detailed Flow Chart Readability; Second Round
Total of 811 papers
found
323 excluded: not about
research on consent
73 excluded: ethical
discussions on consent
34 excluded: focus on
children’s competence
37 excluded: cultural
barriers in readability
156 excluded: consent,
but not readability
62 excluded: pediatric
parental consent
31 excluded: readability
of adult consent
91 excluded: readability
not measured
5 papers selected for
inclusion
3 papers included
2 full-text excluded:
children not target group
Figure 2. Detailed flow chart readability; second round.
2
Understanding of pediatric research information
Seven studies found that understanding of pediatric research information is related to age, where children older than 9 or 11 appeared to have better understanding than younger children (Adcock et al., 2012; Barnett et al., 2005; Burke, et al., 2005; Carr et al., 2012; Hurley & Underwood, 2002; Ondrusek et al., 1998; Shani et al., 2003; Tait et al., 2003, 2007; Tait et al., 2012; Ulph et al., 2009; Wright et al., 2010). However, others have criticized these findings as the documents were aimed at children age 14, influencing understanding in younger children (Ford, Sankey, & Crisp, 2007).
Timing of the information might also play a role in understanding, as in one study participants showed significantly higher understanding when information was offered more than 7 days after diagnosis (Chappuy et al., 2008). Five studies indicated variations between understanding of different research components. Specifically, understanding was low for research aim, right to withdraw, and possible risks and benefits (Burke et al., 2005; Ondrusek et al., 1998; Swartling, et al., 2011; Tait et al., 2003, 2007). In one study, children did not understand the word 'confidentiality', but did grasp the meaning of the concept (Hurley & Underwood, 2002). This indicates that children might be able to understand the meaning even though they do not understand the jargon. Another study found that emotional, rather than cognitive factors are predictors for understanding (Dorn et al., 1995). One study showed that pediatric participants were satisfied with their participation and with the supplied information, when interviewed 8 months after participation (Fogas, et al., 2001).
Interventions to improve information material
Studies on interventions to improve patient information showed inconsistent results. Four studies investigated the effect of a multimedia procedure (Carr et al., 2012; O'Lonergan
& Forster-Harwood, 2011; Shani et al., 2003; Tait et al., 2012). Multimedia use improved understanding in three studies (Carr et al., 2012; O'Lonergan & Forster-Harwood, 2011;
Tait et al., 2012), particularly when information was customized (Carr et al., 2012), and
appeared ineffective in one study (Shani et al., 2003). Supplemental information next to
the document did not improve understanding in two studies (Adcock et al. 2012; Wright
et al. 2010), but in one study participants did have a higher feeling of understanding
(Adcock et al., 2012). Another study investigated the effect of different presentations
of probability information, and found that children best understood pie charts and
percentages (Ulph et al., 2009). In two studies, enhanced patient information forms were
investigated (Barnett et al., 2005; Tait, Voepel-Lewis, & Malviya, 2007). In one study (Barnett et al., 2005) children preferred a story format to a standard or Q&A format;
understanding was significantly improved, and in addition, a significantly lower number of children chose to participate in the proposed research after reading this format. In another study (Tait et al., 2007) children preferred an enhanced form with bullet points, bolding, larger font size and illustrations, over a standard form. The enhanced form also lead to increased understanding.
Conclusion
Based on the reviewed papers, there is no clear conclusion as to which intervention or information format is the most successful in empowering children in decision making.
Understanding was investigated with different methods (RCTs, but also qualitative interviews). The other outcomes, satisfaction and fear reduction, were only recorded in a few studies. Interventions effective in enhancing understanding in one study, appear ineffective in another study. Also, no apparently successful intervention was repeated in a follow-up study.
B. Readability
Detailed description of the evidence in this round can be found in table 3 (at the end of this chapter).
Conclusion
The evidence on readability of pediatric information forms is scarce; only three papers
could be included for review. These papers indicate that a readability gap is present in
pediatric information forms. In one study, analyzed forms had a Flesch Reading Ease
comparable to academic journals (Tarnowski, Allen, Mayhall, & Kelly, 1990). A comparison
between research information and non-medical texts illustrates this gap: a large difference
was found for the Flesch Reading Ease (Menoni et al., 2011). The third study described
the development of research information material together with children, resulting in a
Flesch Reading Ease of 86.3 and Flesch-Kincaid Grade of 3.9 for children age 6-12 years
old (Ford et al., 2007).
2 Discussion
Information format and empowerment
There is a lack of studies on the quality of pediatric research information material; only 17 papers on format and 3 papers on readability were included. Positive effects of inter ventions were found for the use of a story format, multimedia use and presenting probabilities as pie charts or percentages (Barnett et al., 2005; Carr et al., 2012;
O'Lonergan & Forster-Harwood, 2011; Tait et al., 2012; Ulph et al., 2009). No effects were found for the use of a supplemental folder or a Q&A format (Barnett et al., 2005).
The understanding of children seems to be related to age and timing of the information provision. There is an association between research aspects and understanding: papers reported that children often are unaware of their right to withdraw and possible benefits and risks of participation (Burke et al., 2005; Ondrusek et al., 1998; Swartling et al., 2011;
Tait et al., 2003, 2007).
As different studies have used different methods, it is difficult to compare results. Further- more, the nature of the research for which consent is being asked might influence anxiety and thereby understanding, but this factor is not analyzed in the studies. Therefore, we conclude that based on the current evidence, it is difficult to draw a conclusion or provide recommendations on which formats lead to better understanding, satisfaction or lower anxiety.
This is not to say that nothing is known about appropriate ways to engage children in the consent process. Our study indicates that the literature on consent in clinical trials is very scarce. There is however a growing body of knowledge from the social sciences, which could be drawn on even though it does not meet our selection criteria for a systematic review. A key point in approaching children is the use of plain language (Alderson &
Morrow, 2011; Farrell, 2005; J.B. Green, Duncan, Barnes, & Oberklaid, 2003) and text
written in an active voice (Ford et al., 2007). In addition to written information, it is
important to offer verbal explanations and time to ask questions and check understanding
(Alderson & Morrow, 2011). A guideline for consent writers is developed in collaboration
with children (NIHR, 2014). This guideline underscores the importance of plain language,
and stimulates researchers to use colors and images to relate to children. Also, a separate
sheet could be provided next to the study-specific information, to explain what research
is (NIHR, 2014). In addition, it can be very helpful to test the information in a pilot with
children (Alderson & Morrow, 2011). The authors are currently developing research information together with children, which has provided us with interesting and valuable insights that we would otherwise not have had.
Limitations
The two systematic searches were performed in three databases (PubMed, PsycINFO, Embase) and it is possible that evidence exists which is not included in these databases.
However, we believe that the most influential studies were found with our approach.
Only a small number of papers on information format were included, therefore more applied research is required before a conclusion for pediatric consent practice can be drawn.
Conclusion and recommendations
As aging children have a growing influence in the assent/consent process, they need to be provided with information ‘according to their capacity of understanding’ (EU, 2001).
Our systematic review shows that a readability gap exists between the reading level of information material and reading ability of children. In addition, very little research has been performed on adequate formats of informing minors. Our systematic literature search did not yield many results, which in itself is a result worth reporting. The lack of studies on research information for children is remarkable, as hospitals often have targeted websites, brochures and booklets for children about specific diseases or treatments in the hospital.
It appears that efforts to target information material to children remains restricted to
´regular´ hospital matters, and does not extend to research information. This apparent lack
of research and thus attention for improving information in the consent/assent process is
concerning. The ethical and sometimes legal necessity to involve children in the decision
to participate in clinical research should create an urge to find the most optimal way
to do so. Therefore, we conclude that there is a need for better readable information
for children, and for more research and practice guidelines on informing minors about
research participation.
2
Table 1 Level of evidence
Level of evidence Intervention
A1 Systematic review of at least two independently executed studies of A2-level A2 Randomized double-blind research of good quality and sample size B Comparative research, but not with all the characteristics of A2
(patient-control research, cohort studies)
C Non-comparative research
D Expert opinion
Table 2 Evidence for for mat and understanding, with evidence levels
Author, yearStudy DesignStudy ObjectiveOutcomesLimitations Adcock, 2012Randomized crossover study Setting: local elementary school Inclusion: Children of local elementary school enrolled in
general education curriculum Enr
olled: 217 (195 intervention; 190 control) Participant characteristics: children age 7-11
Intervention: assent booklet with pictur
es and written information. Control: standard assent form Measurements: comprehension measured by 6-item quiz. Assessment
in two days: first day one type of assent, 3 days later the other (two for
ms for different studies, to prevent information crossover)
Outcomes: significant difference in perfect scores; control 34.7%, intervention 22.1% More females had a perfect score than males, perfect score increased with age Other outcomes: majority reported to understand booklet better than standard text, style may not be only factor in comprehension
Critical Appraisal: simulation study
, no standardized questionnaire Level of evidence: B Barnett, 2005
Piloted comparative intervention study Setting: school Enr olled: 342 (Q&A format 115; story format 110; standard text
117) Participant characteristics: school childr
en age 9-11 from seven different schools.
Intervention: Q&A format and story format Control: standard text format Measurements: understanding of
4 concepts (randomization, safety and ef
fectiveness, voluntariness and avenues of redress) tested by 12-item questionnaire
Outcomes: significant difference in understanding; story format most correct answers, Q&A format least understandable. Other outcomes: inclusion: Q&A 72%, story 58%, text 71%
Critical Appraisal: simulation, no standar
dized questionnaire, some questions reported difficult to read.
Sampled school had higher than average educational achievement Level of evidence: B Burke, 2005
Interviews Setting: waiting r
oom in Hospital for Sick Children in Toronto Inclusion: children and adolescents Enrolled: 251 children, 237 adults
Participant characteristics: childr
en: age 6-15; adults
Intervention: 6 information forms on 2 procedures for fractured thigh, cast or pins. Treatment options manipulated for risks and benefits, creating 'correct' or ambiguous choice for procedure. Control: two treatment options presented Measurements: interviews on
understanding of 1. ter
m 'research' 2. purpose of research in general 3. general procedure 4. cast procedure 5. pins procedure 6. benefits research 7. disadvantages research
Outcomes: 1. sign less young childr
en understood definition
(24% ages 6-10 vs. 45% 10-12 and 65% 13+, 72% adults) 2. understanding sign higher with age (73% vs. 87% vs. 93% vs. 90%) 3. no dif
ference between ages, average around
70% understanding 4. sign age ef
fect (80% vs. 92% vs. 95% vs. 96%) 5. sign age effect (78% vs. 90%, 96% and 92%) 6. sign age effect (56% vs. 75%, 82% and 76%) 7. sign age effect (64% vs. 80%, 76%, 86%) Other outcomes: younger children significantly preferred cast over pins, older children significantly preferred pins over cast. Even young children can (partly) understand research information.
Critical Appraisal: hypothetical study
, forms might have been too difficult,
as understanding among adults was not 100% Level of evidence: B
2
Author, yearStudy DesignStudy ObjectiveOutcomesLimitations Carr, 2012Structured interview and questionnaire Setting: orthodontist Inclusion: patients scheduled for orthodontic tr
eatment Enrolled: 80 patient-parents pairs Participant characteristics: children age 12-18
Intervention: A. customized slide show + presentation + consent form, B. customized slide show + consent form
Slides shows customized for risks associated with tr
eatment Consent form simplified Control: C. standard slide show and customized consent form Measurements: recall and comprehension by interview, consent domains treatment, risk and responsibility, literacy
Outcomes: group A significant higher risk recall than B and C Group B higher overall comprehension, treatment recall and risk recall than group C Other outcomes: better recall for information on consent presented early in slide show compared to later in slide show
Critical Appraisal: understanding of risks and limitations still inadequate, inter
nal reliability
of subjects in interviews ranged from slight to substantial Level of evidence:B Chappuy, 2008
Qualitative interviews Setting: childr
en in trials for cancer
or HIV Inclusion: childr
en of parents who participated in a previous study,
exclusion when <5yrs old Enr
olled: 29 children Participant characteristics: children age 8-15.
Intervention: semi-directive interview Measurements: understanding of 9 items of consent (goal, protocol, risks, direct/indirect benefits, right to withdraw, duration, alternatives, voluntariness)
Outcomes: lower understanding of procedures
(17%), duration (21%), voluntariness (21%), right to withdraw (21%) and alter
native treatments (31%)
Higher understanding of goal (62%), risks (58%), self-benefits (62%), benefits to others (58%). Understanding better when the information was offered >7 days after diagnosis. Understanding related to time and age.
Critical Appraisal: it is dif
ficult to
disentangle the understanding of a child and the interaction with the par
ents. Level of evidence: C Dorn, 1995
Interviews Setting: patients of an academic hospital Inclusion: childr
en at pediatric
dept. of hospital Enr
olled: 20 for research, 24 for obesity treatment
Participant characteristics: age 7-20 (r
esearch average age 14.6; treatment average age 13.9); 23 patients had prior experience in hospital
Intervention: interview on 12 aspects of resear
ch, global control and trait anxiety Measurements: 1. knowledge 2.
weighted knowledge (based on importance) 3. global contr
ol 4. trait
anxiety and Piagetian task for cognitive development Outcomes: 1. sign corr
elation knowledge and global control
(r=.40) 2. sign corr
elation weighted knowledge and global control (r=.38) 3. and 4. global control only or combined with anxiety best predictor for knowledge Other outcomes: knowledge of research aspects in children linked to psychological factors, rather than to cognitive factors.
Critical Appraisal: small sample size, anxiety in childr
en with life- threatening illness
not generalizable, contr
ol in obese children possibly not generalizable. Level of evidence: C
Author, yearStudy DesignStudy ObjectiveOutcomesLimitations Fogas, 2001Structured telephone interviews Setting: minimal risk study Inclusion: participants of study on medication for ADHD Enr
olled: 115 of 189 asked
Participant characteristics: childr
en age 6-19 (average 11.34;
66.1% <12) in 27.8% other psych diagnosis next to ADHD
Intervention: structured phone interview with 14 questions on participation Measurements: experience of
participation 1. voluntariness 2. accuracy consent infor
mation 3. reason
for participation 4. satisfaction with experience Outcomes: 1. 89% thought participation was voluntary 2. 80% though infor
mation was accurate
3. 47% participated for him/herself 4. 97% was satisfied with participation Other outcomes: subtest in 25 children, test-retest higher than 72%
Critical Appraisal: interviews 8 months after experience, time could influence opinions. Results har
d to generalize
because of specific sample characteristics. Level of evidence: C Hurley, 2002
Survey Setting: study of peer pr
ovocation (not medical research)
Inclusion: random subsample of 382 original participants of resear
ch Enrolled: 178
Participant characteristics: age 8-12 (74 in 2
nd grade, 52 in 4th , 52 in 6th
Intervention: post-assent questionnaire for understanding before and after debriefing Measurements: understanding of 1. voluntary assent 2. procedure 3. confidentiality
Outcomes: 1. childr en understood almost everything, but not the aim of the study. After debriefing still almost
50% did not understand aim 2. understanding of pr
ocedures before debriefing better for 4th- and 6th graders than 2nd, after debriefing correct for most children
3. poor understanding of the concept of confidentiality ndOther outcomes: debriefing: 2 graders did not understand how they were misled in the research, thth4 and 6 graders did.
Although the concept of 'confidentiality' was not understood well, most childr
en could explain who would know what they did in the research
Critical Appraisal: resear
ch was
fairly complex to understand for childr
en Level of evidence: B O'Lonergan, 2011I: Prestudy; II: RCT Setting: children's hospital or
outpatient facility Inclusion: Par
ent-child dyads,
exclusion for mental/visual/ hearing deficits or with pr
evious
experience of DXA or ultrasound Enr
olled: I. 24 parent-child dyads II. 170 parent-child dyads Participant characteristics: children age 11-14, parents
Intervention: Multimedia-information (video or animated presentation) Control: standard text Measurements: Understanding for 8 aspects (purpose, procedures, risks, direct benefit, indirect benefit, alternatives, right to withdraw, voluntariness)
Outcomes: prestudy: feedback on methods used for
adaptations RCT
: better understanding with the use of
multimedia vs. text, but still over 70% did not understand risks. All par
ents and children overestimated their own understanding. Other outcomes: all children understood the right to withdraw
Critical Appraisal: hypothetical study Level of evidence: A2
2
Author, yearStudy DesignStudy ObjectiveOutcomesLimitations
Ondrusek, 1998
Cross-sectional survey
Setting: nutrition study in Canadian hospital Inclusion: participants of nutrition study <18 Enr
olled: 18
Participant characteristics: Healthy childr
en 5-18
Intervention: survey and interview Measurements: understanding based on questionnaire and semi-structured interview
Outcomes: Understanding of aim, risks, right to withdraw and benefits was age-r
elated, these concept were not well understood when age <9.
Other outcomes: participants indicated a total of 5 different reasons for 18 participants
Critical Appraisal: different disclosures: children over 12 read the form by themselves, children under 12 were
read to. This might indicate that the readability of the form was low for children under 12. Level of evidence: C Shani, 2003
RCT Setting: 2 schools in the Negev (Bedouins, economic and social deprivation incr
eases risk of burn
wounds) Inclusion: Schools involved in bur
n wound prevention program Enrolled: 179 (2 schools, originally
3, but eliminated because of technical pr
oblems)
Participant characteristics: Childr
en age 12-13
Intervention: Information on Slides (S), video (V) or first slides then video (S+V) Measurements: survey on health beliefs before intervention and 2 months later: 1. threat 2. internal/external control 3. self-efficacy 4. sense of coherence 5. post-intervention fear reaction, knowledge improvement, safety behavior
Outcomes: no significant difference post- intervention for 1,2,3,4. 5. In S-group highest level of fear and lower perceived luck control in case of wounds, S+V group lowest within-change Other outcomes: improvement predicted by self- efficacy, fear, higher SES and woman. Health belief and demographics better predictor for effect than medium of intervention
Critical Appraisal: very specific type of infor
mation and specific sample No comparison with standar
d text information Level of evidence: B
Swartling, 2011 Qualitative interviews Setting: childr
en in a large-scale longitudinal screening Inclusion: children involved in
longitudinal study now or in the past Enr
olled: 39 Participant characteristics: children age 10-12
Intervention: 6 focus group interviews Measurements: questions on six topics (research, children&research, information&consent, data collection, research consequences, risk to get a disease)
Outcomes: All groups, regardless of experience, considered research to be important Children thought that age of 10 years would be the age when children can understand research information, and age 5-7 would be when to start informing children Children could not identify any risk related to research participation
Other outcomes: All childr
en indicated that research participation can help other people
Critical Appraisal: small sample size Level of evidence: C
Author, yearStudy DesignStudy ObjectiveOutcomesLimitations Tait, 2003Semi-structured interviews Setting: hospital/clinic Inclusion: Childr
en who assented
for participation in anesthesiology study Enr
olled: 102 Participant characteristics: children age 7-18
Measurements: interview within 24h after surgery for 8 aspects of research (purpose, protocol, risks, direct benefits, indirect benefits, freedom to withdraw, alternatives, voluntariness)
Outcomes: understanding ranged between 30.4- 89.4% childr
en older than 11 had a significant
better understanding than under 11, specifically for pr
ocedures, benefits and right to withdraw
Other outcomes: subjective understanding higher than objective understanding, childr
en under 11 had a better understanding when reading the form instead of verbal explanation
Critical Appraisal: interview after surgery, might have affected anxiety and thereby recall Level of evidence: C Tait, 2007
RCT Setting: hospital Inclusion: inpatients for tr
eatment or surgery, exclusion when unable to read, mental impairment of
serious illness Enr
olled: 392 asked, 190 included Participant characteristics: children age 7-17 (average age 11)
Intervention: Information on hypothetical study, with improved readability and clarity Control: standard information Measurements: survey for 8 research aspects (aim, procedure, risks, benefits direct/indirect, alternative, voluntariness, right to withdraw)
Outcomes: better understanding for adapted infor
mation in general, for procedure and indirect benefits. Association age-understanding stronger for standard info than for adapted info, all groups overestimate own understanding Other outcomes: understanding of research information among children is very poor
Critical Appraisal: hypothetical study
,
bias in motivation to participate, difference in
reading grades could influence understanding and is not similar to age differences Level of evidence: B Tait, 2012
Design: pilot study with qualitative interviews Setting: waiting r
oom in American
hospital Inclusion: childr
en and parents in waiting room Enrolled: 4 children, 5 unrelated parents Participant characteristics: children 8-14, parents
Intervention: prototype interactive consent program Measurements: pre-interview for baseline understanding, semi-structured
interviews for understanding (clinical trials, randomization, placebo, blinding); responses to questions embedded in the program real-time ability to respond on first attempt
Outcomes: Understanding pr
e-post per concept Clinical trials;adult 60%-80%child 25%-50% Randomization;adult 20%-60%child0%-0% Placeboadult 80%-100% child 0%-50% Blindingadult 80%-80%child 25%-50% Results not significant Correct responses to embedded questions on first attempt 90.2% for parents, 61.1% for children Other outcomes: children thought program was ‘fun to use’, liked the interactive nature and would like to receive study information in this format the next time
Critical Appraisal: pilot study
, so small
sample size Understanding not sufficient for all concepts Level of evidence: C
2
Author, yearStudy DesignStudy ObjectiveOutcomesLimitations Ulph, 2009Cross-sectional study Setting: school children in the UK, sampled for academic achievements, fr
ee school meals and percentage of ethnic
minorities Inclusion: Childr
en from representative schools in the UK Enrolled: 106 Participant characteristics: children age 7-11
Intervention: verbal labels, percentages, proportion-word, proportion-notation, top view-pie charts, mixed format condition Measurements: picture of three cups presented, with different probability
depicted, child has to select cup most likely to contain the ball
Outcomes: sign better performance by pie chart vs. percentages, proportion-notation/word and mixed format. In pie chart most children correct response (84%) and sign more certain of response, in percentages second-best response (70%)
Other outcomes: older age associated with better compr
ehension for all formats except percentages,
when using verbal labels almost half of participants asked for explanation Critical Appraisal: simulation study
,
cup game not same as in medical setting Level of evidence: B Wright, 2010
RCT Setting: orthodontist Inclusion: childr
en who started
fixed appliance therapy Enr
olled: 60 Participant characteristics: children age 12-16
Intervention: verbal + written info Control: verbal info Measurements: Survey before consult
(T1), after consent (T2) and 12 weeks after tr
eatment (T3). Measurements of: 1. anxiety, motivation, understanding 2. compliance
Outcomes: 1. no dif ference in anxiety between T1 and T2 for intervention and control group. Sign higher
motivation in intervention At T3 no dif
ferences for anxiety, understanding,
motivation 2. compliance impr
oved in intervention group, but not sign
Critical Appraisal: Very specific sample and pr
ocedure, small sample size Level of evidence: B
