This study is the first to collect the understanding and preferences of young people and their parents affected by a rare chronic respiratory condition, childhood Interstitial Lung Disease. The survey investigated the topics of understanding clinical research aspects such as consent/assent, enquiring who the young person’s decision-makers are in participating in clinical research, how research opportunities and risk are communicated and research acceptability preferences. This insight into clinical research allows clinicians and researchers to understand the young person’s and parent’s perspectives to help with future research planning and design. These included the understanding of young people’s understanding of consent, along with both young people and the parents agreeing that they are the main decision-makers for the young person to participate in clinical research. Alongside this, parents and young people felt that communication of research opportunities, risks and ongoing contact with clinicians and researchers, if the young person does participate in clinical research, was very important to them. Regarding research acceptability, parents were more accepting of the young person participating in clinical research compared to the young person.
Across the young person age groups, verbal consent was the preferred method of providing consent/assent to participate in clinical research; although the need for continued education across all age groups for explaining age-related informed consent/assent concepts is evident, as a portion of the young people across all age groups were not aware of the consent process. This is supportive of other literature on paediatric consent where understanding of ‘informed consent’ increased with education level25. When looking at who is the main decision maker of the young people participating in clinical research, most of the child-parent dyads agreed that it was both the young person and the parent/caregiver.
For clinical research communication, young people predominately preferred learning about research opportunities from their parent/caregivers directly. However, when communicating with clinicians, young people expressed they would prefer face-to-face communication with clinicians to relay information such as research opportunities, answer any questions relating to the research, and explain research risks. There was interest from the young people to find out about research opportunities from the research scientists directly, along with staying in touch with the researchers if the young person did participate in their research. This may indicate personal interest or investment in any research the young person may participate in.
Regarding parental preferences for clinical research communication, parents indicated that when communicating clinical research risk, it was more important for the parent to know this information than the young person directly. Possible factors for this may be age, where the parent feels the child may be too young to understand such information, or alternatively, the child if knowing the information or risks may then be wary to undertake the clinical research. Due to the small sample size, age stratification to understand this further was not possible; therefore, further exploration of this is recommended in future research, such as opening the recruitment to other online communities and respiratory conditions and keeping the survey open for a longer period for fuller data collection.
In the section on research acceptability, there were some diverging young person and parent responses, particularly in the acceptability of providing blood via pin prick or venepuncture. It is suggested that further qualitative exploration of this may be beneficial as there may be mechanisms at play, such as fear or lack of education when giving blood. One additional consideration is how young people may feel pressured into clinical research. The ‘network of exchange’ theory of paediatric clinical research describes the balance by clinicians in promoting clinical research32: on one hand to not be overly helpful as this may be deemed coercive, yet, be governed by the Declaration of Helsinki to focus on informed consent by either an independent professional or emphasise the voluntary nature of participating in clinical research32. However, for voluntary research, there is a dependence on the clinician’s knowledge and expertise to inform a decision to participate33. For young people the role of the parent is one of education, support, and protection as to understanding voluntary research participation and avoidance of experiencing pressure. Yet, if these findings show differing expectations between the parent and their child, then it may expose a space of pressure and/or negotiation to participate in clinical research. Young people have discussed experiencing this pressure when agreeing to participate in research34,35; therefore, finding strategies for young people to engage in open dialogue and safe space sharing with an independent advocate may be a solution to alleviate any potential pressures.
In the section on the clinical research use of discarded sputum or tissue/biopsy samples, there was a difference between the permitted and non-permitted use of samples for both the young person and the parent where there was a preference for towards parental consent to use both sample types, where the young people preferred consent for use of the tissue/biopsy samples. Further exploration into the use of discarded samples may be beneficial as biomedical specimens within paediatric research are scarce; this will allow for debate in this ethically challenging area.
Strengths and Limitations
Study feasibility and discriminant validity for response bias was not a study aim due to difficulty assessing sample size due to recruitment uncertainty. However, within the survey two-time point responses indicated response variance between the young person and the parent (self-health report and research acceptability). To support this variance further, the health reporting variance between the young person and parent may also indicate the ‘proxy’ problem, where parents are more likely to report their child as having poorer health than the child36.
The child-parent dyad survey format was found to be suitable for online distribution, and pilot feedback highlighted the survey was interesting and thought-provoking for the young people to think about their respiratory condition. However, recruitment from online community groups may have resulted in response bias, and so the results may be influenced by those who are more positive-minded towards clinical research. The small sample size did not allow for inference in clinical research preferences between age groups, the chILD type and subjective health rating.