Childhood Interstitial Lung Disease clinical research: A child-parent dyad survey on research preferences and acceptability

Childhood Interstitial Lung Disease clinical research: A child-parent dyad survey on research preferences and acceptability | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Research Article Childhood Interstitial Lung Disease clinical research: A child-parent dyad survey on research preferences and acceptability Carlee M Gilbert, Andrew Bush, Kate M Bennett, Christopher Brown This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-4468802/v1 This work is licensed under a CC BY 4.0 License Status: Posted Version 1 posted You are reading this latest preprint version Abstract Despite patient and public involvement (PPI) in paediatric rare disease research being a challenge, PPI is strongly advocated for clinical research. PPI provides the opportunity for young people to support research for the future of their healthcare and improve their own understanding of their health condition, thus improving health outcomes. The aim of this child-parent dyad survey was to investigate the understanding and preferences of clinical research in 26 young people affected with childhood Interstitial Lung Disease (chILD) (age 6 to 17) and their parents. The survey results captured young people’s understanding of informed consent/assent and confirmed young people and their parents are the main decision-makers in their undertaking research. For clinical research acceptability, parents were more open for their child to participate in clinical research than young people. Parents expressed less preference for their child to participate in research that involved untested medicines and surgery, whereas young people expressed less preference to undertake research that involved untested medicines, surgery, and venepuncture. These novel findings indicate that the survey designed for this study effectively captured young people’s opinions in an online non-clinical setting and found a potential disconnect between the young person’s needs and opinions and parents’ preferences of clinical research. The development of chILD research education and a young person forum is recommended to build and foster clinician/researcher relationships. Ultimately, this will allow for building stronger PPI links and improved chILD clinical research. PPI childhood interstitial lung disease young people research preferences consent assent Introduction Over the past few decades, management and treatment for rare chronic paediatric respiratory conditions have significantly advanced 1 , 2 , 3 . Due to the nature of rare paediatric respiratory conditions, clinicians may only see a few young people within their clinics. From a small clinic setting to large international collaborative efforts, rare disease data collection for research can take time and resources. Despite paediatric clinical research being ethically and logistically challenging to fund and develop, embracing PPI can be advantageous, and sometimes compulsory, in obtaining clinical research support 4 . PPI in paediatric clinical research, which includes both children and adolescents (collectively known as “young people”) can be difficult to undertake compared to adult PPI as there are additional considerations such as differences in research methods, presenting age-appropriate information, limited times of contact, to factoring in aspects such as parental/caregiver consent 5 . The moral and pragmatic influence of PPI engagement cannot be overlooked. The most fundamental argument for PPI inclusion is the consideration of a young person’s right to allow them to be included in shaping the future of their own health 6 , 7 . Improved recruitment, research design and quality are some benefits of using PPI 8 , 9 ; however, some with PPI challenges, including time, management of PPI and lack of tools to assess the impact 10 . To support PPI inclusion, various toolkits and guidance have been made available for study planning 11 , 12 . Yet, with PPI predominately focusing on research design, planning and dissemination methods, how young people meaningfully contribute to potentially complex discussions poses a challenge 13 . A further challenge for paediatric rare disease PPI is the development of meaningful connections to undertake effective PPI. Rare disease communities are an excellent way for individuals to connect and learn about their disease type 14 . Shared experience, emotional support and understanding of the patient journey through the patient/caregiver perspective is imperative for advancing rare disease research 15 . Opportunities for rare disease communities to connect with clinicians and researchers are expanding 16 , 17 . Despite concerns about methodological rigour and ethical safety 16 , rare disease patient organisations and non-clinician researchers are using online surveys and engagement tools to conduct their own patient-driven research, thus setting their own research agenda 17 , 18 , 19 , 20 . As highlighted with successful rare disease patient-clinician collaborations, access to rare disease community groups can provide patient journey understanding to gauge important areas of research interest. The rare disease “ Alone we are rare. Together we are strong ” motto is one which encapsulates the spirit of the online community 21 . Rare disease advocates, patients and caregivers are actively willing to work with clinicians and researchers in research 22 . By using online communities, particularly those with gatekeeping facilities and are monitored by patient organisations, incorporating non-clinical research design into rare disease clinical research will make space for young people’s viewpoints and may enable better structures and relationship-building for rare disease clinical research 3 , 6 , 23 , 24 . Little PPI has been conducted into the preferences and understanding of paediatric respiratory clinical research directly aimed at young people with a rare chronic respiratory condition. The opportunity to ask young people directly about their understanding and preferences of clinical research is an important first step in commencing dialogue in this area. This study addresses this by surveying children and their parents/caregivers in an online platform using recruitment from moderated online patient communities. The aim of this novel study was to investigate the current understanding and preferences of young people and their parents in key areas of clinical research. The child-parent dyad survey topics included informed consent/assent, understanding of the decision makers of undertaking research, how young people would like research opportunities and risks communicated, and clinical acceptability of different research scenarios. Individual responses are of main study interest; however, response validity will be checked at two time points in the survey to investigate response variance between the child and parent. This will occur at the beginning of the survey within the self-health reporting section and at the end in the research acceptability section. This will be an important facet of measuring the survey’s success regarding the reliability and validity of the child survey completion instructions. Methods A structured child-parent dyad questionnaire was developed and distributed to survey the preferences and opinions of young people diagnosed with a rare paediatric respiratory condition (age 6 to 17) and their parent/caregiver on clinical research. Survey Development The survey was developed from a community-based child-parent dyad survey originally designed to understand the research preferences of young people with Interstitial Lung Disease and from parent-based research on clinical research for minors 25 ( Supplementary material ). The survey was expanded to account for understanding differing research aspects such as informed consent/assent and decision-makers in clinical research and research acceptability of different research scenarios. The survey included a self-reported and parent-reported young person health rating of “ Not Good ”, “ OK ”, “ Good” , and “ Excellent ’, which the young person and parent completed separately to understand if there were any differences in subjective/objective health. The survey was separated into 3 age groups: age 6 to 10, age 11 to 15, and age 16 to 17, and was adapted for age-appropriate readability using the Flesch-Kincaid reading ease test. Once developed, the survey was piloted with 4 child-parent dyads. Two child-parent dyads included young people (ages 8 and 12) with a learning disability to ensure survey accessibility. Recruitment Epidemiological data for rare paediatric respiratory conditions is gathering pace 26 , 27 , 28 , 29 . However, this is not fully understood across the range of rare to ultra-rare conditions with challenges in small data 30 , 31 . Due to this, coupled with the study being exploratory in nature and the open online recruitment method, there was no sampling frame to calculate desired response rates. The survey was distributed to patient organisation and rare disease community group s online platforms between September and December 2021. Three age-specific surveys were offered between the age groups where they would be directed to the survey platform Qualtrics to undertake the survey. A participant information sheet was provided on the survey, which was to be read by both the young person and the parent. The consent process was via a confirmatory tick box on the online survey and varied between age groups. For age groups 6 to 10 and 11 to 15, the consent process comprised of the young person assenting to participate and the parent consenting for their, and their child’s, participation. For the age group 16 to 17, the consent process comprised the young person and parent consenting individually. Procedure Before commencing the survey, instructions were made available for the parent to read. Young people completed the first part of the survey with the parent present, and then the parent completed the second part of the survey. This information was extracted from the survey when the raw data was collected to retain response anonymity. Statistical Analysis Data was summarised as continuous and categorical variables with counts and percentages. Wilcoxon Signed-Ranks test was used to assess the group variation and compare the level of research acceptability between the young person and parent groups. All statistical inferences were made at a 5% significance level and performed using SPSS Version 27 (SPSS Inc., Chic, IL, USA.). Results Data Screening The initial dataset included 56 child-parent dyad survey responses. 30 of these responses were excluded from the analysis due to incomplete data and non-chILD conditions. Descriptive Statistics Table 1 presents the demographic characteristics of the final young person’s sample. The 26 responses comprised 15 males (57.7%), and 11 females (42.3%). A Wilcoxon Signed-rank test indicated a highly significant difference in young person health reporting between the young persons and parents (Z = 2.236, p = 0.25, r = .44), with the young person group indicating better health rating than the parents. Table 1 Descriptive Statistics for the Final Dataset (n = 26) Number/(% or SD) Gender of young person completing survey Male 15 (57.7%) Female 11 (42.3%) Age of young person completing survey (mean, S.D.) 10.6 (3.44) Age of parent completing survey (mean, S.D.) 39.9 (6.50) No. of young people separated into age groups Age 6 to 10 16 (61.5%) Age 11 to 15 6 (23.1%) Age 16 to 17 4 (15.4%) Reported respiratory diagnoses* Bronchiolitis Obliterans 7 LIP and FB 1 NEHI 8 Protein Surfactant Dysfunction 3 Unknown/Undeclared 7 Young person's Health rating Young person reported Parent reported Not Good 1 (3.8%) 2 (7.7%) OK 5 (19.2%) 5 (19.2%) Good 12 (46.2%) 14 (53.8%) Excellent 8 (30.8%) 5 (19.2%) LIP : Lymphoid Interstitial Pneumonia; FB : Follicular Bronchiolitis; NEHI : Neuroendocrine Cell Hyperplasia of Infancy Young People’s Awareness and Preference of Informed consent/assent Table 2 presents the young people’s responses to their awareness of consent and informed consent within clinical research. Most of the young people were not aware of what consent means in clinical research (68.8%; n = 15). For informed assent, 23 young people (87.5%) were not aware of the term ‘informed assent’ for clinical research. Table 2 Young person response to consent and informed assent (n = 26) Age Group 6–10 11–15 16–17 Total Do you know what consent is in clinical research? Yes 2 1 2 5 (12.5%) I have heard of it, but I don't know what it means 3 2 1 6 (18.8%) No 11 3 1 15 (68.8%) Do you know what informed assent is in clinical research? Yes 1 0 1 2 (6.3%) I have heard of it, but I don't know what it means 1 0 0 1 (6.3%) No 14 6 3 23 (87.5%) For the multiple option survey question, “ If a doctor or researcher asked you to participate in clinical research and you would like to, how would you like to agree to that?) ”, 65.4% ( n = 17) of young people indicated a preference for verbal confirmation. This was followed by a secondary preference of the clinical research team to provide a document for a parent to read and sign (53.8%; n = 14). Understanding Decision Makers in Clinical Research Young people were asked who they believed were the main decision-makers to allow them to participate in clinical research. The largest response was for young people who felt this was a joint decision between them and their parent/s (46.2%; n = 12). For the parent section on who the main decision-makers in their child participating in clinical research, this included a scenario of disease severity. Table 3 presents the options of potential decision makers and scenario severity types. For the “ Non-life threatening: condition will clear up over time ”, this was a close split between parents viewing clinical research participation as the decision between the child-parent (46.2%; n = 12), and the option of the young person, parent, and clinician (42.3%; n = 11). With the “ Non-life threatening: will lead to chronic disability ” scenario, there was a small shift in the responses, with the majority parental response being the young person, parent/s, and clinician to be the main decision-makers in allowing the young person to participate in clinical research (46.2%; n = 12). For the final scenario of “ Life-threatening: Progressive and with no current cure ”, there was a further small shift in responses, with most parents maintaining that the main decision-makers for their child participating in clinical research was to be agreed by the young person, parent/s, and clinician (46.2%; n = 12). Table 3 Parental response of main decision maker/s for the young person to participate in clinical research with responses dependant on condition severity scenario Condition severity Non-life-threatening: condition will clear up over time Non-life-threatening: will lead to chronic disability Life-threatening: progressive and no current cure Young person Parent/s Young person and parent/s Clinician Young person, parent/s, and clinician 0 (0%) 0 (0%) 0 (0%) 3 (11.5%) 4 (15.4%) 4 (15.4%) 12 (46.2%) 10 (38.5%) 9 (34.6%) 0 (0%) 0 (0%) 1 (3.8%) 11 (42.3%) 12 (46.2%) 12 (46.2%) Clinical Research Communication – Opportunities and Risk In the survey section on how the young people would like to learn about clinical research opportunities, the young people had a higher preference to learn about research opportunities verbally compared to written formats. Young people indicated that the main preferred communicator of these opportunities was their parent(s) (73.1%; n = 19) followed by their doctors (65.4%; n = 17). Similarly, this was reflected when asked who the young people would like to communicate with if they had any questions about the clinical research, their parent/s were the main preference (76.9%; n = 20). Table 4 expands upon this further with young people’s and parental preferences for communication with doctors and research scientists. In the young people survey section, the highest Likert score for their doctors to explain clinical research to them was “ extremely important ” (38.5%; n = 10). With the parents, the importance of doctors explaining the clinical research was rated as “ extremely important ” (76.9%; n = 20). On the question of explaining the risks of clinical research, for young people this was rated as “ extremely important ” (76.9%; n = 20) and for the parent (100%; n = 26). For the parent response of clinical risk being directly discussed with the young person, this rating is varied, with most responses being “ extremely important ” (50.0%; n = 13). In terms of preference for the scientists who are conducting the clinical research to stay in touch with the young people who participate in their research, young people rated this as “ very important’ (46.2%; n = 12), whereas parents rated this “ extremely important ” (69.7%; n = 23). Table 4 Young person and Parent Likert responses of importance for doctors to explain the clinical research, clinical research risk, and contact with research scientists Not very important Slightly important Moderately important Very important Extremely important Young Person Survey Section How important is it for the doctors to explain the clinical research to you? 0 (0.0%) 2 (7.7%) 9 (34.6%) 5 (19.2%) 10 (38.5%) How important is it for doctors to explain the risks of their research to you? 1 (3.8%) 0 (0.0%) 3 (11.5%) 2 (7.7%) 20 (76.9%) How important is it for the scientists to stay in touch with you about their research? 2 (7.7%) 2 (7.7%) 2 (7.7%) 12 (46.2%) 8 (30.8%) Parent Survey Section How important is it for the doctors to explain the clinical research to you? 0 (0.0%) 0 (0.0%) 1 (3.8%) 5 (19.2%) 20 (76.9%) How important is it for the doctors to explain the clinical research to your child? 1 (3.8%) 0 (0.0%) 5 (19.2%) 7 (26.9%) 13 (50.5%) How important is it for doctors to explain the risks of their research to you? 0 (0.0%) 0 (0.0%) 0 (0.0%) 0 (0.0%) 26 (100%) How important is it for doctors to explain the risks of their research to your child? 2 (7.7%) 0 (0.0%) 7 (26.9%) 7 (26.9%) 10 (38.5%) How important is it for the scientists to stay in touch with you about their research? 1 (3.8%) 1 (3.8%) 0 (0.0%) 6 (23.1%) 18 (69.2%) Research Acceptability This survey section asked young people and their parents about the preferred types of clinical research they feel the child would be willing to take part in. Wilcoxon Signed-ranks test indicated response variance between the young people and the parents, highlighting parents were significantly more comfortable for their child to participate in the clinical research scenarios compared to the young person (Z = 3.48, p < .001, r = .68). From the young person’s responses (Table 5 ), the most acceptable form of clinical research would be cheek swabs (88.5%; n = 23). This was followed by using devices such as a smartphone/smartwatch to measure their own health or illness (84.6%; n = 22) and answering questions about their body (88.6%; n = 22). Similar to the young person's main preferences, for parents the most accepted is a cheek swab sample (100%; n = 26). This was followed by allowing the child to answer questions about their own body (96.2%; n = 25), to then using a tech device (92.3%; n = 24) and pin prick blood sample (92.3%; n = 24). In terms of research acceptability, both the young people and parents reported swallowing an untested medicine (young person (7.7%; n = 2) parent (19.2%; n = 5) and inhaling an untested medicine (young person (15.4%; n = 4) parent (23.1%; n = 6) being the least acceptable research type. Table 5 Young person and parent responses for the survey section on research acceptability Young Person Response (n = 33) Parent Response (n = 33) Swallowing a medicine that has not been tested before 2 (7.7%) 5 (19.2%) Swallowing a medicine that has been tested before 17 (65.4%) 17 (65.4%) Inhaling a medicine that has not been tested before 4 (15.4%) 6 (23.1%) Inhaling a medicine that has been tested before 18 (69.2%) 16 (62.5%) Taking and testing my blood by a needle 11 (42.3%) 19 (73.1%) Taking and testing my blood by a pin prick on my finger 14 (53.8%) 24 (92.3%) Using a cheek swab to collect my cells 23 (88.5%) 26 (100%) Having surgery that may help or fix my lung condition 10 (38.5%) 12 (46.2%) Using a tech device such as a smartphone or smartwatch that measures me/my illness 22 (84.6%) 24 (92.3%) Answering personal questions about my body 22 (84.6%) 25 (96.2%) Answering questions about my private thoughts 14 (53.8%) 20 (76.9%) For the survey section on the use of discarded samples, Table 6 highlights the young person and parent responses of their feelings about those samples being reused for clinical research. For the sputum sample majority response, 53.8% ( n = 14) of young people were happy for researchers to use these samples for their clinical research without their permission. Conversely for parents, 57.7% ( n = 15) would like permission to be sought if these samples were used for research. For discarded tissue/biopsy samples, young people (53.8%; n = 14) and parents (65.4%, n = 17) requested permission for use of these discarded sample types. Table 6 Young person and parent responses for the survey section on the use of discarded sputum and tissue/biopsy samples for clinical research use Sputum Tissue Young Person Parent Young Person Parent I would be happy for scientists to use these samples for their research with my permission 10 (38.5%) 15 (57.7%) 14 (53.8%) 17 (65.4%) I would be happy for scientists to use these samples for their research - my permission is not needed 14 (53.8%) 11 (42.3%) 10 (38.5%) 9 (34.6%) I would not be happy for scientists to use these for their research 2 (7.7%) 0 (0.0%) 2 (7.7%) 0 (0.0%) The survey section asked if young people would like to be rewarded for participating in clinical research. The main response was 43.3% ( n = 11) of young people who stated that they were unsure. Discussion 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 level 25 . 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 research 32 : 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 research 32 . However, for voluntary research, there is a dependence on the clinician’s knowledge and expertise to inform a decision to participate 33 . 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 research 34 , 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 child 36 . 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. Conclusion This study successfully delivered an online child-parent dyad survey within chILD non-clinical population community. Several novel findings include the exploration of young people’s understanding of consent and decision-making in participating in clinical research and the want for ongoing communication of research opportunities and risks between the young people, parents, and the clinical team. Regarding research acceptability, parents were more accepting of allowing the child to participate in clinical search compared to the young person. Untested medicines, surgery and venepuncture were the least acceptable forms of clinical research for young people, with parents having different opinions of research acceptability than young people. This highlights a potential disconnect between the young person’s needs and the parent’s expectations of the young person participating in clinical research. Much of this information collected may be confirmatory in terms of current protocols and ethical research standards; however, giving young people who are directly affected by an ultra-rare respiratory condition such as chILD, a voice in future research will allow for understanding their needs in their chronic health journey. Providing ongoing communication, engagement, and education with young people on clinical research and developing a forum where young people can freely express their own needs and opinions will allow for a fair framework of PPI in chILD research. Abbreviations ChILD Childhood Interstitial Lung Disease PPI Patient and Public Involvement Declarations Author Contributions C Gilbert contributed to the conceptualisation, methodology, analysis, and writing (original) K M Bennett contributed to the writing (review and editing) C A Brown contributed to the project supervision and writing (reviewing and editing) A Bush contributed to the writing (review and editing) Acknowledgements N/A Funding None Data Availability The data that supports the analysis and review of this study is available from the corresponding author upon request. Ethical approval and consent to participate Ethical approval was granted from The University of Liverpool – Central University Ethics Committee C (Reference 10077). Triple informed consent method used: Young person consented to participate, Parent consented to participate, and Parent consented for the young person to participate. All participants were informed of the study information, risks, benefits and data management. Consent for publication Not applicable Competing interests Not applicable Conflict of Interest The authors declare no competing interests References Batshaw ML, Groft SC, Krischer JP. Research into rare diseases of childhood. JAMA. 2014;311(17):1729–30. Vece TJ, Wambach JA, Hagood JS. Childhood rare lung disease in the 21st century:-omics technology advances accelerating discovery. Pediatr Pulmonol. 2020;55(7):1828–37. Hopkinson NS, Lenney W, Langton-Hewer S, Bennett J, Swingwood E, Hughes A, Whittamore A, Woolnough S. Children’s charter for lung health. Thorax. 2022;77(1):11–2. Roennow A, Sauvé M, Welling J, Riggs RJ, Kennedy AT, Galetti I, Brown E, Leite C, Gonzalez A, Guiraud AP, Houÿez F. Collaboration between patient organisations and a clinical research sponsor in a rare disease condition: learnings from a community advisory board and best practice for future collaborations. 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Parents’ attitudes towards and perceptions of involving minors in medical research from the Japanese perspective. BMC Med Ethics. 2018;19:1–8. McCarthy C, Lara Gallego B, Trapnell BC, McCormack FX. Epidemiology of rare lung diseases: the challenges and opportunities to improve research and knowledge. Rare diseases epidemiology: Update and overview. 2017:419 – 42. Scotet V, L’hostis C, Férec C. The changing epidemiology of cystic fibrosis: incidence, survival and impact of the CFTR gene discovery. Genes. 2020;11(6):589. Mirra V, Werner C, Santamaria F. Primary ciliary dyskinesia: an update on clinical aspects, genetics, diagnosis, and future treatment strategies. Front Pead. 2017;5:135. Ferraro VA, Zanconato S, Zamunaro A, Carraro S. Children’s interstitial and diffuse lung diseases (chILD) in 2020. Children. 2020;7(12):280. Mitani AA, Haneuse S. Small data challenges of studying rare diseases. JAMA Netw open. 2020;3(3):e201965. Hickey GL, Grant SW, Dunning J, Siepe M. Statistical primer: sample size and power calculations—why, when and how? Eur J Cardiothorac Surg. 2018;54(1):4–9. Luchtenberg M, Maeckelberghe E, Locock L, Powell L, Verhagen AE. Young people's experiences of participation in clinical trials: reasons for taking part. Am J Bioeth. 2015;15(11):3–13. Dekking SA, van der Graaf R, van Delden JJ. Strengths and weaknesses of guideline approaches to safeguard voluntary informed consent of patients within a dependent relationship. BMC Med. 2014;12:1–1. Grady C, Wiener L, Abdoler E, Trauernicht E, Zadeh S, Diekema DS, Wilfond BS, Wendler D. Assent in research: the voices of adolescents. J Adolesc Health. 2014;54(5):515–20. Unguru Y, Sill AM, Kamani N. The experiences of children enrolled in pediatric oncology research: implications for assent. Pediatrics. 2010;125(4):e876–83. Sattoe JN, van Staa A, Moll HA, On Your Own Feet Research Group [email protected] . The proxy problem anatomized: child-parent disagreement in health related quality of life reports of chronically ill adolescents. Health Qual Life Outcomes. 2012;10:1–3. Additional Declarations No competing interests reported. Supplementary Files CHOICESSurveyV.1BMC.docx Cite Share Download PDF Status: Posted Version 1 posted You are reading this latest preprint version Research Square lets you share your work early, gain feedback from the community, and start making changes to your manuscript prior to peer review in a journal. As a division of Research Square Company, we’re committed to making research communication faster, fairer, and more useful. We do this by developing innovative software and high quality services for the global research community. Our growing team is made up of researchers and industry professionals working together to solve the most critical problems facing scientific publishing. Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-4468802","acceptedTermsAndConditions":true,"allowDirectSubmit":true,"archivedVersions":[],"articleType":"Research Article","associatedPublications":[],"authors":[{"id":315766695,"identity":"95177f8e-5afe-4bc6-bb4c-b605948653c4","order_by":0,"name":"Carlee M Gilbert","email":"data:image/png;base64,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","orcid":"","institution":"University of Liverpool","correspondingAuthor":true,"prefix":"","firstName":"Carlee","middleName":"M","lastName":"Gilbert","suffix":""},{"id":315766696,"identity":"d6d50c06-cf87-45de-9d00-445b9f16f445","order_by":1,"name":"Andrew Bush","email":"","orcid":"","institution":"Imperial College London","correspondingAuthor":false,"prefix":"","firstName":"Andrew","middleName":"","lastName":"Bush","suffix":""},{"id":315766697,"identity":"7ea1f480-7e5e-4467-be6a-1b7470a9a039","order_by":2,"name":"Kate M Bennett","email":"","orcid":"","institution":"University of Liverpool","correspondingAuthor":false,"prefix":"","firstName":"Kate","middleName":"M","lastName":"Bennett","suffix":""},{"id":315766698,"identity":"6aaf4d87-4e23-4e7f-ad10-1f3e0c15494a","order_by":3,"name":"Christopher Brown","email":"","orcid":"","institution":"University of Liverpool","correspondingAuthor":false,"prefix":"","firstName":"Christopher","middleName":"","lastName":"Brown","suffix":""}],"badges":[],"createdAt":"2024-05-23 19:42:40","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-4468802/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-4468802/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":68141001,"identity":"4423a9b7-4bed-419d-bafa-355b7964930b","added_by":"auto","created_at":"2024-11-04 04:54:07","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":699920,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-4468802/v1/189196c2-4a04-49ed-9d78-f721daf19023.pdf"},{"id":58992633,"identity":"a58facf3-a85a-42d3-86f2-2af3d4798c71","added_by":"auto","created_at":"2024-06-25 05:24:44","extension":"docx","order_by":1,"title":"","display":"","copyAsset":false,"role":"supplement","size":35221,"visible":true,"origin":"","legend":"","description":"","filename":"CHOICESSurveyV.1BMC.docx","url":"https://assets-eu.researchsquare.com/files/rs-4468802/v1/661617a409ea73ddd76097fb.docx"}],"financialInterests":"No competing interests reported.","formattedTitle":"Childhood Interstitial Lung Disease clinical research: A child-parent dyad survey on research preferences and acceptability","fulltext":[{"header":"Introduction","content":"\u003cp\u003eOver the past few decades, management and treatment for rare chronic paediatric respiratory conditions have significantly advanced\u003csup\u003e\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e,\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e,\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e\u003c/sup\u003e. Due to the nature of rare paediatric respiratory conditions, clinicians may only see a few young people within their clinics. From a small clinic setting to large international collaborative efforts, rare disease data collection for research can take time and resources. Despite paediatric clinical research being ethically and logistically challenging to fund and develop, embracing PPI can be advantageous, and sometimes compulsory, in obtaining clinical research support\u003csup\u003e\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e\u003c/sup\u003e. PPI in paediatric clinical research, which includes both children and adolescents (collectively known as \u0026ldquo;young people\u0026rdquo;) can be difficult to undertake compared to adult PPI as there are additional considerations such as differences in research methods, presenting age-appropriate information, limited times of contact, to factoring in aspects such as parental/caregiver consent\u003csup\u003e\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e\u003c/sup\u003e.\u003c/p\u003e \u003cp\u003eThe moral and pragmatic influence of PPI engagement cannot be overlooked. The most fundamental argument for PPI inclusion is the consideration of a young person\u0026rsquo;s right to allow them to be included in shaping the future of their own health\u003csup\u003e\u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e,\u003cspan citationid=\"CR7\" class=\"CitationRef\"\u003e7\u003c/span\u003e\u003c/sup\u003e. Improved recruitment, research design and quality are some benefits of using PPI\u003csup\u003e\u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e,\u003cspan citationid=\"CR9\" class=\"CitationRef\"\u003e9\u003c/span\u003e\u003c/sup\u003e; however, some with PPI challenges, including time, management of PPI and lack of tools to assess the impact\u003csup\u003e\u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e\u003c/sup\u003e. To support PPI inclusion, various toolkits and guidance have been made available for study planning\u003csup\u003e\u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e,\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e\u003c/sup\u003e. Yet, with PPI predominately focusing on research design, planning and dissemination methods, how young people meaningfully contribute to potentially complex discussions poses a challenge\u003csup\u003e\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e\u003c/sup\u003e.\u003c/p\u003e \u003cp\u003eA further challenge for paediatric rare disease PPI is the development of meaningful connections to undertake effective PPI. Rare disease communities are an excellent way for individuals to connect and learn about their disease type\u003csup\u003e\u003cspan citationid=\"CR14\" class=\"CitationRef\"\u003e14\u003c/span\u003e\u003c/sup\u003e. Shared experience, emotional support and understanding of the patient journey through the patient/caregiver perspective is imperative for advancing rare disease research\u003csup\u003e\u003cspan citationid=\"CR15\" class=\"CitationRef\"\u003e15\u003c/span\u003e\u003c/sup\u003e. Opportunities for rare disease communities to connect with clinicians and researchers are expanding\u003csup\u003e\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e, \u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e\u003c/sup\u003e. Despite concerns about methodological rigour and ethical safety\u003csup\u003e\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e\u003c/sup\u003e, rare disease patient organisations and non-clinician researchers are using online surveys and engagement tools to conduct their own patient-driven research, thus setting their own research agenda\u003csup\u003e\u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e,\u003cspan citationid=\"CR18\" class=\"CitationRef\"\u003e18\u003c/span\u003e,\u003cspan citationid=\"CR19\" class=\"CitationRef\"\u003e19\u003c/span\u003e,\u003cspan citationid=\"CR20\" class=\"CitationRef\"\u003e20\u003c/span\u003e\u003c/sup\u003e.\u003c/p\u003e \u003cp\u003eAs highlighted with successful rare disease patient-clinician collaborations, access to rare disease community groups can provide patient journey understanding to gauge important areas of research interest. The rare disease \u0026ldquo;\u003cem\u003eAlone we are rare. Together we are strong\u003c/em\u003e\u0026rdquo; motto is one which encapsulates the spirit of the online community\u003csup\u003e\u003cspan citationid=\"CR21\" class=\"CitationRef\"\u003e21\u003c/span\u003e\u003c/sup\u003e. Rare disease advocates, patients and caregivers are actively willing to work with clinicians and researchers in research\u003csup\u003e\u003cspan citationid=\"CR22\" class=\"CitationRef\"\u003e22\u003c/span\u003e\u003c/sup\u003e. By using online communities, particularly those with gatekeeping facilities and are monitored by patient organisations, incorporating non-clinical research design into rare disease clinical research will make space for young people\u0026rsquo;s viewpoints and may enable better structures and relationship-building for rare disease clinical research\u003csup\u003e\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e,\u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e,\u003cspan citationid=\"CR23\" class=\"CitationRef\"\u003e23\u003c/span\u003e,\u003cspan citationid=\"CR24\" class=\"CitationRef\"\u003e24\u003c/span\u003e\u003c/sup\u003e.\u003c/p\u003e \u003cp\u003eLittle PPI has been conducted into the preferences and understanding of paediatric respiratory clinical research directly aimed at young people with a rare chronic respiratory condition. The opportunity to ask young people directly about their understanding and preferences of clinical research is an important first step in commencing dialogue in this area. This study addresses this by surveying children and their parents/caregivers in an online platform using recruitment from moderated online patient communities. The aim of this novel study was to investigate the current understanding and preferences of young people and their parents in key areas of clinical research. The child-parent dyad survey topics included informed consent/assent, understanding of the decision makers of undertaking research, how young people would like research opportunities and risks communicated, and clinical acceptability of different research scenarios. Individual responses are of main study interest; however, response validity will be checked at two time points in the survey to investigate response variance between the child and parent. This will occur at the beginning of the survey within the self-health reporting section and at the end in the \u003cspan refid=\"Sec12\" class=\"InternalRef\"\u003eresearch acceptability\u003c/span\u003e section. This will be an important facet of measuring the survey\u0026rsquo;s success regarding the reliability and validity of the child survey completion instructions.\u003c/p\u003e "},{"header":"Methods","content":"\u003cp\u003eA structured child-parent dyad questionnaire was developed and distributed to survey the preferences and opinions of young people diagnosed with a rare paediatric respiratory condition (age 6 to 17) and their parent/caregiver on clinical research.\u003c/p\u003e\u003ch2\u003eSurvey Development\u003c/h2\u003e\u003cp\u003eThe survey was developed from a community-based child-parent dyad survey originally designed to understand the research preferences of young people with Interstitial Lung Disease and from parent-based research on clinical research for minors\u003csup\u003e\u003cspan citationid=\"CR25\" class=\"CitationRef\"\u003e25\u003c/span\u003e\u003c/sup\u003e (\u003cem\u003eSupplementary material\u003c/em\u003e). The survey was expanded to account for understanding differing research aspects such as informed consent/assent and decision-makers in clinical research and research acceptability of different research scenarios. The survey included a self-reported and parent-reported young person health rating of \u0026ldquo;\u003cem\u003eNot Good\u003c/em\u003e\u0026rdquo;, \u0026ldquo;\u003cem\u003eOK\u003c/em\u003e\u0026rdquo;, \u0026ldquo;\u003cem\u003eGood\u0026rdquo;\u003c/em\u003e, and \u0026ldquo;\u003cem\u003eExcellent\u003c/em\u003e\u0026rsquo;, which the young person and parent completed separately to understand if there were any differences in subjective/objective health.\u003c/p\u003e \u003cp\u003eThe survey was separated into 3 age groups: age 6 to 10, age 11 to 15, and age 16 to 17, and was adapted for age-appropriate readability using the Flesch-Kincaid reading ease test. Once developed, the survey was piloted with 4 child-parent dyads. Two child-parent dyads included young people (ages 8 and 12) with a learning disability to ensure survey accessibility.\u003c/p\u003e \u003cdiv id=\"Sec3\" class=\"Section2\"\u003e \u003ch2\u003eRecruitment\u003c/h2\u003e \u003cp\u003eEpidemiological data for rare paediatric respiratory conditions is gathering pace\u003csup\u003e\u003cspan citationid=\"CR26\" class=\"CitationRef\"\u003e26\u003c/span\u003e,\u003cspan citationid=\"CR27\" class=\"CitationRef\"\u003e27\u003c/span\u003e,\u003cspan citationid=\"CR28\" class=\"CitationRef\"\u003e28\u003c/span\u003e,\u003cspan citationid=\"CR29\" class=\"CitationRef\"\u003e29\u003c/span\u003e\u003c/sup\u003e. However, this is not fully understood across the range of rare to ultra-rare conditions with challenges in small data\u003csup\u003e\u003cspan citationid=\"CR30\" class=\"CitationRef\"\u003e30\u003c/span\u003e,\u003cspan citationid=\"CR31\" class=\"CitationRef\"\u003e31\u003c/span\u003e\u003c/sup\u003e. Due to this, coupled with the study being exploratory in nature and the open online recruitment method, there was no sampling frame to calculate desired response rates.\u003c/p\u003e \u003cp\u003eThe survey was distributed to patient organisation and rare disease community group s online platforms between September and December 2021. Three age-specific surveys were offered between the age groups where they would be directed to the survey platform Qualtrics to undertake the survey. A participant information sheet was provided on the survey, which was to be read by both the young person and the parent. The consent process was via a confirmatory tick box on the online survey and varied between age groups. For age groups 6 to 10 and 11 to 15, the consent process comprised of the young person assenting to participate and the parent consenting for their, and their child\u0026rsquo;s, participation. For the age group 16 to 17, the consent process comprised the young person and parent consenting individually.\u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec4\" class=\"Section2\"\u003e \u003ch2\u003eProcedure\u003c/h2\u003e \u003cp\u003eBefore commencing the survey, instructions were made available for the parent to read. Young people completed the first part of the survey with the parent present, and then the parent completed the second part of the survey. This information was extracted from the survey when the raw data was collected to retain response anonymity.\u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec5\" class=\"Section2\"\u003e \u003ch2\u003eStatistical Analysis\u003c/h2\u003e \u003cp\u003eData was summarised as continuous and categorical variables with counts and percentages. Wilcoxon Signed-Ranks test was used to assess the group variation and compare the level of research acceptability between the young person and parent groups. All statistical inferences were made at a 5% significance level and performed using SPSS Version 27 (SPSS Inc., Chic, IL, USA.).\u003c/p\u003e \u003c/div\u003e"},{"header":"Results","content":"\u003cdiv id=\"Sec7\" class=\"Section2\"\u003e \u003ch2\u003eData Screening\u003c/h2\u003e \u003cp\u003eThe initial dataset included 56 child-parent dyad survey responses. 30 of these responses were excluded from the analysis due to incomplete data and non-chILD conditions.\u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec8\" class=\"Section2\"\u003e \u003ch2\u003eDescriptive Statistics\u003c/h2\u003e \u003cp\u003eTable\u0026nbsp;\u003cspan refid=\"Tab1\" class=\"InternalRef\"\u003e1\u003c/span\u003e presents the demographic characteristics of the final young person\u0026rsquo;s sample. The 26 responses comprised 15 males (57.7%), and 11 females (42.3%). A Wilcoxon Signed-rank test indicated a highly significant difference in young person health reporting between the young persons and parents (Z\u0026thinsp;=\u0026thinsp;2.236, p\u0026thinsp;=\u0026thinsp;0.25, r\u0026thinsp;=\u0026thinsp;.44), with the young person group indicating better health rating than the parents.\u003c/p\u003e \u003cp\u003e \u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"Yes\" id=\"Tab1\" border=\"1\"\u003e \u003ccaption language=\"En\"\u003e \u003cdiv class=\"CaptionNumber\"\u003eTable 1\u003c/div\u003e \u003cdiv class=\"CaptionContent\"\u003e \u003cp\u003e\u003cem\u003eDescriptive Statistics for the Final Dataset (n\u0026thinsp;=\u0026thinsp;26)\u003c/em\u003e\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"4\"\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c3\" colnum=\"3\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c4\" colnum=\"4\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colspan=\"2\" nameend=\"c2\" namest=\"c1\"\u003e\u0026nbsp;\u003c/th\u003e \u003cth align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003eNumber/(% or SD)\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c2\" namest=\"c1\"\u003e \u003cp\u003eGender of young person completing survey\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c2\" namest=\"c1\"\u003e \u003cp\u003eMale\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e15 (57.7%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c2\" namest=\"c1\"\u003e \u003cp\u003eFemale\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e11 (42.3%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c2\" namest=\"c1\"\u003e \u003cp\u003eAge of young person completing survey (mean, S.D.)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e10.6 (3.44)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c2\" namest=\"c1\"\u003e \u003cp\u003eAge of parent completing survey (mean, S.D.)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e39.9 (6.50)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c2\" namest=\"c1\"\u003e \u003cp\u003eNo. of young people separated into age groups\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c2\" namest=\"c1\"\u003e \u003cp\u003eAge 6 to 10\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e16 (61.5%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c2\" namest=\"c1\"\u003e \u003cp\u003eAge 11 to 15\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e6 (23.1%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c2\" namest=\"c1\"\u003e \u003cp\u003eAge 16 to 17\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e4 (15.4%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c2\" namest=\"c1\"\u003e \u003cp\u003eReported respiratory diagnoses*\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c2\" namest=\"c1\"\u003e \u003cp\u003eBronchiolitis Obliterans\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e7\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c2\" namest=\"c1\"\u003e \u003cp\u003eLIP and FB\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c2\" namest=\"c1\"\u003e \u003cp\u003eNEHI\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e8\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c2\" namest=\"c1\"\u003e \u003cp\u003eProtein Surfactant Dysfunction\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e3\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c2\" namest=\"c1\"\u003e \u003cp\u003eUnknown/Undeclared\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e7\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eYoung person's Health rating\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c3\" namest=\"c2\"\u003e \u003cp\u003eYoung person reported\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003eParent reported\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eNot Good\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c3\" namest=\"c2\"\u003e \u003cp\u003e1 (3.8%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e2 (7.7%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eOK\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c3\" namest=\"c2\"\u003e \u003cp\u003e5 (19.2%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e5 (19.2%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eGood\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c3\" namest=\"c2\"\u003e \u003cp\u003e12 (46.2%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e14 (53.8%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eExcellent\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c3\" namest=\"c2\"\u003e \u003cp\u003e8 (30.8%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e5 (19.2%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003ctfoot\u003e \u003ctr\u003e\u003ctd colspan=\"4\"\u003e\u003cem\u003eLIP\u003c/em\u003e: Lymphoid Interstitial Pneumonia; \u003cem\u003eFB\u003c/em\u003e: Follicular Bronchiolitis; \u003cem\u003eNEHI\u003c/em\u003e: Neuroendocrine\u003c/td\u003e\u003c/tr\u003e \u003ctr\u003e\u003ctd colspan=\"4\"\u003eCell Hyperplasia of Infancy\u003c/td\u003e\u003c/tr\u003e \u003c/tfoot\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec9\" class=\"Section2\"\u003e \u003ch2\u003eYoung People\u0026rsquo;s Awareness and Preference of Informed consent/assent\u003c/h2\u003e \u003cp\u003eTable\u0026nbsp;\u003cspan refid=\"Tab2\" class=\"InternalRef\"\u003e2\u003c/span\u003e presents the young people\u0026rsquo;s responses to their awareness of consent and informed consent within clinical research. Most of the young people were not aware of what consent means in clinical research (68.8%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;15). For informed assent, 23 young people (87.5%) were not aware of the term \u0026lsquo;informed assent\u0026rsquo; for clinical research.\u003c/p\u003e \u003cp\u003e \u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"Yes\" id=\"Tab2\" border=\"1\"\u003e \u003ccaption language=\"En\"\u003e \u003cdiv class=\"CaptionNumber\"\u003eTable 2\u003c/div\u003e \u003cdiv class=\"CaptionContent\"\u003e \u003cp\u003e\u003cem\u003eYoung person response to consent and informed assent (n\u0026thinsp;=\u0026thinsp;26)\u003c/em\u003e\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"8\"\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c3\" colnum=\"3\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c4\" colnum=\"4\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c5\" colnum=\"5\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c6\" colnum=\"6\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c7\" colnum=\"7\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c8\" colnum=\"8\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e\u0026nbsp;\u003c/th\u003e \u003cth align=\"left\" colspan=\"5\" nameend=\"c6\" namest=\"c2\"\u003e \u003cp\u003eAge Group\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colspan=\"2\" nameend=\"c8\" namest=\"c7\"\u003e\u0026nbsp;\u003c/th\u003e \u003c/tr\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e\u0026nbsp;\u003c/th\u003e \u003cth align=\"left\" colspan=\"2\" nameend=\"c3\" namest=\"c2\"\u003e \u003cp\u003e6\u0026ndash;10\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colspan=\"2\" nameend=\"c5\" namest=\"c4\"\u003e \u003cp\u003e11\u0026ndash;15\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colspan=\"2\" nameend=\"c7\" namest=\"c6\"\u003e \u003cp\u003e16\u0026ndash;17\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c8\"\u003e \u003cp\u003eTotal\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eDo you know what consent is in clinical research?\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c6\" namest=\"c5\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c8\" namest=\"c7\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eYes\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c6\" namest=\"c5\"\u003e \u003cp\u003e2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c8\" namest=\"c7\"\u003e \u003cp\u003e5 (12.5%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eI have heard of it, but I don't know what it means\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c6\" namest=\"c5\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c8\" namest=\"c7\"\u003e \u003cp\u003e6 (18.8%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eNo\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e11\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c6\" namest=\"c5\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c8\" namest=\"c7\"\u003e \u003cp\u003e15 (68.8%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colspan=\"3\" nameend=\"c3\" namest=\"c1\"\u003e \u003cp\u003e\u003cem\u003eDo you know what informed assent is in clinical research?\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c6\" namest=\"c5\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c8\" namest=\"c7\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eYes\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e0\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c6\" namest=\"c5\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c8\" namest=\"c7\"\u003e \u003cp\u003e2 (6.3%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eI have heard of it, but I don't know what it means\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e0\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c6\" namest=\"c5\"\u003e \u003cp\u003e0\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c8\" namest=\"c7\"\u003e \u003cp\u003e1 (6.3%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eNo\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e14\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c4\" namest=\"c3\"\u003e \u003cp\u003e6\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c6\" namest=\"c5\"\u003e \u003cp\u003e3\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colspan=\"2\" nameend=\"c8\" namest=\"c7\"\u003e \u003cp\u003e23 (87.5%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e \u003cp\u003eFor the multiple option survey question, \u0026ldquo;\u003cem\u003eIf a doctor or researcher asked you to participate in clinical research and you would like to, how would you like to agree to that?)\u003c/em\u003e\u0026rdquo;, 65.4% (\u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;17) of young people indicated a preference for verbal confirmation. This was followed by a secondary preference of the clinical research team to provide a document for a parent to read and sign (53.8%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;14).\u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec10\" class=\"Section2\"\u003e \u003ch2\u003eUnderstanding Decision Makers in Clinical Research\u003c/h2\u003e \u003cp\u003eYoung people were asked who they believed were the main decision-makers to allow them to participate in clinical research. The largest response was for young people who felt this was a joint decision between them and their parent/s (46.2%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;12).\u003c/p\u003e \u003cp\u003eFor the parent section on who the main decision-makers in their child participating in clinical research, this included a scenario of disease severity. Table\u0026nbsp;\u003cspan refid=\"Tab3\" class=\"InternalRef\"\u003e3\u003c/span\u003e presents the options of potential decision makers and scenario severity types. For the \u0026ldquo;\u003cem\u003eNon-life threatening: condition will clear up over time\u003c/em\u003e\u0026rdquo;, this was a close split between parents viewing clinical research participation as the decision between the child-parent (46.2%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;12), and the option of the young person, parent, and clinician (42.3%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;11). With the \u0026ldquo;\u003cem\u003eNon-life threatening: will lead to chronic disability\u003c/em\u003e\u0026rdquo; scenario, there was a small shift in the responses, with the majority parental response being the young person, parent/s, and clinician to be the main decision-makers in allowing the young person to participate in clinical research (46.2%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;12). For the final scenario of \u0026ldquo;\u003cem\u003eLife-threatening: Progressive and with no current cure\u003c/em\u003e\u0026rdquo;, there was a further small shift in responses, with most parents maintaining that the main decision-makers for their child participating in clinical research was to be agreed by the young person, parent/s, and clinician (46.2%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;12).\u003c/p\u003e \u003cp\u003e \u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"Yes\" id=\"Tab3\" border=\"1\"\u003e \u003ccaption language=\"En\"\u003e \u003cdiv class=\"CaptionNumber\"\u003eTable 3\u003c/div\u003e \u003cdiv class=\"CaptionContent\"\u003e \u003cp\u003e\u003cem\u003eParental response of main decision maker/s for the young person to participate in clinical research with responses dependant on condition severity scenario\u003c/em\u003e\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"4\"\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c3\" colnum=\"3\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c4\" colnum=\"4\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e\u0026nbsp;\u003c/th\u003e \u003cth align=\"left\" colspan=\"3\" nameend=\"c4\" namest=\"c2\"\u003e \u003cp\u003e\u003cspan type=\"Underline\" class=\"Underline\" name=\"Emphasis\"\u003eCondition severity\u003c/span\u003e\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e\u003cb\u003eNon-life-threatening: condition will clear up over time\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e\u003cb\u003eNon-life-threatening: will lead to chronic disability\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e\u003cb\u003eLife-threatening: progressive and no current cure\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\" morerows=\"4\" rowspan=\"5\"\u003e \u003cp\u003eYoung person\u003c/p\u003e \u003cp\u003eParent/s\u003c/p\u003e \u003cp\u003eYoung person and parent/s\u003c/p\u003e \u003cp\u003eClinician\u003c/p\u003e \u003cp\u003eYoung person, parent/s, and clinician\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e0 (0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e0 (0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0 (0%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e3 (11.5%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e4 (15.4%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e4 (15.4%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e12 (46.2%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e10 (38.5%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e9 (34.6%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e0 (0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e0 (0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1 (3.8%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e11 (42.3%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e12 (46.2%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e12 (46.2%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec11\" class=\"Section2\"\u003e \u003ch2\u003eClinical Research Communication \u0026ndash; Opportunities and Risk\u003c/h2\u003e \u003cp\u003eIn the survey section on how the young people would like to learn about clinical research opportunities, the young people had a higher preference to learn about research opportunities verbally compared to written formats. Young people indicated that the main preferred communicator of these opportunities was their parent(s) (73.1%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;19) followed by their doctors (65.4%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;17). Similarly, this was reflected when asked who the young people would like to communicate with if they had any questions about the clinical research, their parent/s were the main preference (76.9%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;20).\u003c/p\u003e \u003cp\u003eTable\u0026nbsp;\u003cspan refid=\"Tab4\" class=\"InternalRef\"\u003e4\u003c/span\u003e expands upon this further with young people\u0026rsquo;s and parental preferences for communication with doctors and research scientists. In the young people survey section, the highest Likert score for their doctors to explain clinical research to them was \u0026ldquo;\u003cem\u003eextremely important\u003c/em\u003e\u0026rdquo; (38.5%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;10). With the parents, the importance of doctors explaining the clinical research was rated as \u0026ldquo;\u003cem\u003eextremely important\u003c/em\u003e\u0026rdquo; (76.9%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;20).\u003c/p\u003e \u003cp\u003eOn the question of explaining the risks of clinical research, for young people this was rated as \u0026ldquo;\u003cem\u003eextremely important\u003c/em\u003e\u0026rdquo; (76.9%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;20) and for the parent (100%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;26). For the parent response of clinical risk being directly discussed with the young person, this rating is varied, with most responses being \u0026ldquo;\u003cem\u003eextremely important\u003c/em\u003e\u0026rdquo; (50.0%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;13).\u003c/p\u003e \u003cp\u003eIn terms of preference for the scientists who are conducting the clinical research to stay in touch with the young people who participate in their research, young people rated this as \u0026ldquo;\u003cem\u003every important\u0026rsquo;\u003c/em\u003e (46.2%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;12), whereas parents rated this \u0026ldquo;\u003cem\u003eextremely important\u003c/em\u003e\u0026rdquo; (69.7%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;23).\u003c/p\u003e \u003cp\u003e \u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"Yes\" id=\"Tab4\" border=\"1\"\u003e \u003ccaption language=\"En\"\u003e \u003cdiv class=\"CaptionNumber\"\u003eTable 4\u003c/div\u003e \u003cdiv class=\"CaptionContent\"\u003e \u003cp\u003e\u003cem\u003eYoung person and Parent Likert responses of importance for doctors to explain the clinical research, clinical research risk, and contact with research scientists\u003c/em\u003e\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"6\"\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c3\" colnum=\"3\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c4\" colnum=\"4\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c5\" colnum=\"5\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c6\" colnum=\"6\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e\u0026nbsp;\u003c/th\u003e \u003cth align=\"left\" colname=\"c2\"\u003e \u003cp\u003eNot very important\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c3\"\u003e \u003cp\u003eSlightly important\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c4\"\u003e \u003cp\u003eModerately important\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c5\"\u003e \u003cp\u003eVery important\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c6\"\u003e \u003cp\u003eExtremely important\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e \u003cp\u003eYoung Person Survey Section\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/th\u003e \u003cth align=\"left\" colname=\"c3\"\u003e\u0026nbsp;\u003c/th\u003e \u003cth align=\"left\" colname=\"c4\"\u003e\u0026nbsp;\u003c/th\u003e \u003cth align=\"left\" colname=\"c5\"\u003e\u0026nbsp;\u003c/th\u003e \u003cth align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eHow important is it for the doctors to explain the clinical research to you?\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e0 (0.0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e2 (7.7%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e9 (34.6%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e5 (19.2%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e10 (38.5%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eHow important is it for doctors to explain the risks of their research to you?\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1 (3.8%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e0 (0.0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e3 (11.5%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e2 (7.7%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e20 (76.9%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eHow important is it for the scientists to stay in touch with you about their research?\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e2 (7.7%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e2 (7.7%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e2 (7.7%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e12 (46.2%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e8 (30.8%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cb\u003eParent Survey Section\u003c/b\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eHow important is it for the doctors to explain the clinical research to you?\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e0 (0.0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e0 (0.0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e1 (3.8%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e5 (19.2%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e20 (76.9%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eHow important is it for the doctors to explain the clinical research to your child?\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1 (3.8%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e0 (0.0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e5 (19.2%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e7 (26.9%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e13 (50.5%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eHow important is it for doctors to explain the risks of their research to you?\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e0 (0.0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e0 (0.0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0 (0.0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e0 (0.0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e26 (100%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eHow important is it for doctors to explain the risks of their research to your child?\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e2 (7.7%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e0 (0.0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e7 (26.9%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e7 (26.9%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e10 (38.5%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eHow important is it for the scientists to stay in touch with you about their research?\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1 (3.8%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e1 (3.8%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e0 (0.0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e6 (23.1%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003e18 (69.2%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e \u003c/div\u003e \u003cdiv id=\"Sec12\" class=\"Section2\"\u003e \u003ch2\u003eResearch Acceptability\u003c/h2\u003e \u003cp\u003eThis survey section asked young people and their parents about the preferred types of clinical research they feel the child would be willing to take part in. Wilcoxon Signed-ranks test indicated response variance between the young people and the parents, highlighting parents were significantly more comfortable for their child to participate in the clinical research scenarios compared to the young person (Z\u0026thinsp;=\u0026thinsp;3.48, \u003cem\u003ep\u003c/em\u003e\u0026thinsp;\u0026lt;\u0026thinsp;.001, r\u0026thinsp;=\u0026thinsp;.68).\u003c/p\u003e \u003cp\u003eFrom the young person\u0026rsquo;s responses (Table\u0026nbsp;\u003cspan refid=\"Tab5\" class=\"InternalRef\"\u003e5\u003c/span\u003e), the most acceptable form of clinical research would be cheek swabs (88.5%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;23). This was followed by using devices such as a smartphone/smartwatch to measure their own health or illness (84.6%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;22) and answering questions about their body (88.6%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;22). Similar to the young person's main preferences, for parents the most accepted is a cheek swab sample (100%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;26). This was followed by allowing the child to answer questions about their own body (96.2%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;25), to then using a tech device (92.3%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;24) and pin prick blood sample (92.3%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;24). In terms of research acceptability, both the young people and parents reported swallowing an untested medicine (young person (7.7%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;2) parent (19.2%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;5) and inhaling an untested medicine (young person (15.4%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;4) parent (23.1%; \u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;6) being the least acceptable research type.\u003c/p\u003e \u003cp\u003e \u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"Yes\" id=\"Tab5\" border=\"1\"\u003e \u003ccaption language=\"En\"\u003e \u003cdiv class=\"CaptionNumber\"\u003eTable 5\u003c/div\u003e \u003cdiv class=\"CaptionContent\"\u003e \u003cp\u003e\u003cem\u003eYoung person and parent responses for the survey section on research acceptability\u003c/em\u003e\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"3\"\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c3\" colnum=\"3\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e\u0026nbsp;\u003c/th\u003e \u003cth align=\"left\" colname=\"c2\"\u003e \u003cp\u003eYoung Person Response\u003c/p\u003e \u003cp\u003e(n\u0026thinsp;=\u0026thinsp;33)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c3\"\u003e \u003cp\u003eParent Response\u003c/p\u003e \u003cp\u003e(n\u0026thinsp;=\u0026thinsp;33)\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eSwallowing a medicine that has not been tested before\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e2\u003c/p\u003e \u003cp\u003e(7.7%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e5\u003c/p\u003e \u003cp\u003e(19.2%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eSwallowing a medicine that has been tested before\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e17\u003c/p\u003e \u003cp\u003e(65.4%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e17\u003c/p\u003e \u003cp\u003e(65.4%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eInhaling a medicine that has not been tested before\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e4\u003c/p\u003e \u003cp\u003e(15.4%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e6\u003c/p\u003e \u003cp\u003e(23.1%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eInhaling a medicine that has been tested before\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e18\u003c/p\u003e \u003cp\u003e(69.2%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e16\u003c/p\u003e \u003cp\u003e(62.5%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eTaking and testing my blood by a needle\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e11\u003c/p\u003e \u003cp\u003e(42.3%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e19\u003c/p\u003e \u003cp\u003e(73.1%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eTaking and testing my blood by a pin prick on my finger\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e14\u003c/p\u003e \u003cp\u003e(53.8%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e24\u003c/p\u003e \u003cp\u003e(92.3%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eUsing a cheek swab to collect my cells\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e23\u003c/p\u003e \u003cp\u003e(88.5%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e26\u003c/p\u003e \u003cp\u003e(100%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eHaving surgery that may help or fix my lung condition\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e10\u003c/p\u003e \u003cp\u003e(38.5%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e12\u003c/p\u003e \u003cp\u003e(46.2%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eUsing a tech device such as a smartphone or smartwatch that measures me/my illness\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e22\u003c/p\u003e \u003cp\u003e(84.6%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e24\u003c/p\u003e \u003cp\u003e(92.3%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eAnswering personal questions about my body\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e22\u003c/p\u003e \u003cp\u003e(84.6%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e25\u003c/p\u003e \u003cp\u003e(96.2%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eAnswering questions about my private thoughts\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e14\u003c/p\u003e \u003cp\u003e(53.8%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e20\u003c/p\u003e \u003cp\u003e(76.9%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e \u003cp\u003eFor the survey section on the use of discarded samples, Table\u0026nbsp;\u003cspan refid=\"Tab6\" class=\"InternalRef\"\u003e6\u003c/span\u003e highlights the young person and parent responses of their feelings about those samples being reused for clinical research. For the sputum sample majority response, 53.8% (\u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;14) of young people were happy for researchers to use these samples for their clinical research without their permission. Conversely for parents, 57.7% (\u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;15) would like permission to be sought if these samples were used for research.\u003c/p\u003e \u003cp\u003eFor discarded tissue/biopsy samples, young people (53.8%; n\u0026thinsp;=\u0026thinsp;14) and parents (65.4%, n\u0026thinsp;=\u0026thinsp;17) requested permission for use of these discarded sample types.\u003c/p\u003e \u003cp\u003e \u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"Yes\" id=\"Tab6\" border=\"1\"\u003e \u003ccaption language=\"En\"\u003e \u003cdiv class=\"CaptionNumber\"\u003eTable 6\u003c/div\u003e \u003cdiv class=\"CaptionContent\"\u003e \u003cp\u003e\u003cem\u003eYoung person and parent responses for the survey section on the use of discarded sputum and tissue/biopsy samples for clinical research use\u003c/em\u003e\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"5\"\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c3\" colnum=\"3\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c4\" colnum=\"4\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c5\" colnum=\"5\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e\u0026nbsp;\u003c/th\u003e \u003cth align=\"left\" colspan=\"2\" nameend=\"c3\" namest=\"c2\"\u003e \u003cp\u003eSputum\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colspan=\"2\" nameend=\"c5\" namest=\"c4\"\u003e \u003cp\u003eTissue\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e\u0026nbsp;\u003c/th\u003e \u003cth align=\"left\" colname=\"c2\"\u003e \u003cp\u003eYoung Person\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c3\"\u003e \u003cp\u003eParent\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c4\"\u003e \u003cp\u003eYoung Person\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c5\"\u003e \u003cp\u003eParent\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eI would be happy for scientists to use these samples for their research with my permission\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e10 (38.5%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e15 (57.7%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e14 (53.8%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e17 (65.4%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eI would be happy for scientists to use these samples for their research - my permission is not needed\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e14 (53.8%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e11 (42.3%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e10 (38.5%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e9\u003c/p\u003e \u003cp\u003e(34.6%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003e\u003cem\u003eI would not be happy for scientists to use these for their research\u003c/em\u003e\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e2\u003c/p\u003e \u003cp\u003e(7.7%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003e0\u003c/p\u003e \u003cp\u003e(0.0%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003e2\u003c/p\u003e \u003cp\u003e(7.7%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003e0\u003c/p\u003e \u003cp\u003e(0.0%)\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e \u003cp\u003eThe survey section asked if young people would like to be rewarded for participating in clinical research. The main response was 43.3% (\u003cem\u003en\u003c/em\u003e\u0026thinsp;=\u0026thinsp;11) of young people who stated that they were unsure.\u003c/p\u003e \u003c/div\u003e"},{"header":"Discussion","content":"\u003cp\u003eThis 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\u0026rsquo;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\u0026rsquo;s and parent\u0026rsquo;s perspectives to help with future research planning and design. These included the understanding of young people\u0026rsquo;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.\u003c/p\u003e \u003cp\u003eAcross 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 \u0026lsquo;informed consent\u0026rsquo; increased with education level\u003csup\u003e\u003cspan citationid=\"CR25\" class=\"CitationRef\"\u003e25\u003c/span\u003e\u003c/sup\u003e. 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.\u003c/p\u003e \u003cp\u003eFor 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.\u003c/p\u003e \u003cp\u003e 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.\u003c/p\u003e \u003cp\u003eIn 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 \u0026lsquo;network of exchange\u0026rsquo; theory of paediatric clinical research describes the balance by clinicians in promoting clinical research\u003csup\u003e\u003cspan citationid=\"CR32\" class=\"CitationRef\"\u003e32\u003c/span\u003e\u003c/sup\u003e: 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 research\u003csup\u003e\u003cspan citationid=\"CR32\" class=\"CitationRef\"\u003e32\u003c/span\u003e\u003c/sup\u003e. However, for voluntary research, there is a dependence on the clinician\u0026rsquo;s knowledge and expertise to inform a decision to participate\u003csup\u003e\u003cspan citationid=\"CR33\" class=\"CitationRef\"\u003e33\u003c/span\u003e\u003c/sup\u003e. 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 research\u003csup\u003e\u003cspan citationid=\"CR34\" class=\"CitationRef\"\u003e34\u003c/span\u003e,\u003cspan citationid=\"CR35\" class=\"CitationRef\"\u003e35\u003c/span\u003e\u003c/sup\u003e; 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.\u003c/p\u003e \u003cp\u003eIn 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.\u003c/p\u003e \u003cdiv id=\"Sec14\" class=\"Section2\"\u003e \u003ch2\u003eStrengths and Limitations\u003c/h2\u003e \u003cp\u003eStudy 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 \u0026lsquo;proxy\u0026rsquo; problem, where parents are more likely to report their child as having poorer health than the child\u003csup\u003e\u003cspan citationid=\"CR36\" class=\"CitationRef\"\u003e36\u003c/span\u003e\u003c/sup\u003e.\u003c/p\u003e \u003cp\u003eThe 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.\u003c/p\u003e \u003c/div\u003e"},{"header":"Conclusion","content":"\u003cp\u003eThis study successfully delivered an online child-parent dyad survey within chILD non-clinical population community. Several novel findings include the exploration of young people\u0026rsquo;s understanding of consent and decision-making in participating in clinical research and the want for ongoing communication of research opportunities and risks between the young people, parents, and the clinical team. Regarding research acceptability, parents were more accepting of allowing the child to participate in clinical search compared to the young person. Untested medicines, surgery and venepuncture were the least acceptable forms of clinical research for young people, with parents having different opinions of research acceptability than young people. This highlights a potential disconnect between the young person\u0026rsquo;s needs and the parent\u0026rsquo;s expectations of the young person participating in clinical research.\u003c/p\u003e \u003cp\u003eMuch of this information collected may be confirmatory in terms of current protocols and ethical research standards; however, giving young people who are directly affected by an ultra-rare respiratory condition such as chILD, a voice in future research will allow for understanding their needs in their chronic health journey. Providing ongoing communication, engagement, and education with young people on clinical research and developing a forum where young people can freely express their own needs and opinions will allow for a fair framework of PPI in chILD research.\u003c/p\u003e"},{"header":"Abbreviations","content":"\u003cdiv class=\"DefinitionList\"\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003e\u003cem\u003eChILD\u003c/em\u003e\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eChildhood Interstitial Lung Disease\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003e\u003cem\u003ePPI\u003c/em\u003e\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003ePatient and Public Involvement\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003c/div\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003cstrong\u003eAuthor Contributions\u0026nbsp;\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eC Gilbert contributed to the conceptualisation, methodology, analysis, and writing (original)\u003c/p\u003e\n\u003cp\u003eK M Bennett contributed to the writing (review and editing)\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eC A Brown contributed to the project supervision and writing (reviewing and editing)\u003c/p\u003e\n\u003cp\u003eA Bush contributed to the writing (review and editing)\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAcknowledgements\u0026nbsp;\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eN/A\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eFunding\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eNone\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eData Availability\u0026nbsp;\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe data that supports the analysis and review of this study is available from the corresponding author upon request.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eEthical approval and consent to participate\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eEthical approval was granted from The University of Liverpool \u0026ndash; Central University Ethics Committee C (Reference 10077). Triple informed consent method used: Young person consented to participate, Parent consented to participate, and Parent consented for the young person to participate. All participants were informed of the study information, risks, benefits and data management. \u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConsent for publication\u0026nbsp;\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eNot applicable\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eCompeting interests\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eNot applicable\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConflict of Interest\u0026nbsp;\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe authors declare no competing interests\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\u003cli\u003e\u003cspan\u003eBatshaw ML, Groft SC, Krischer JP. Research into rare diseases of childhood. 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Assent in research: the voices of adolescents. J Adolesc Health. 2014;54(5):515\u0026ndash;20.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eUnguru Y, Sill AM, Kamani N. The experiences of children enrolled in pediatric oncology research: implications for assent. Pediatrics. 2010;125(4):e876\u0026ndash;83.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eSattoe JN, van Staa A, Moll HA, On Your Own Feet Research Group [email protected]. The proxy problem anatomized: child-parent disagreement in health related quality of life reports of chronically ill adolescents. Health Qual Life Outcomes. 2012;10:1\u0026ndash;3.\u003c/span\u003e\u003c/li\u003e\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":true,"highlight":"","institution":"","isAcceptedByJournal":false,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true},"keywords":"PPI, childhood interstitial lung disease, young people, research preferences, consent, assent","lastPublishedDoi":"10.21203/rs.3.rs-4468802/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-4468802/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003eDespite patient and public involvement (PPI) in paediatric rare disease research being a challenge, PPI is strongly advocated for clinical research. PPI provides the opportunity for young people to support research for the future of their healthcare and improve their own understanding of their health condition, thus improving health outcomes. The aim of this child-parent dyad survey was to investigate the understanding and preferences of clinical research in 26 young people affected with childhood Interstitial Lung Disease (chILD) (age 6 to 17) and their parents. The survey results captured young people\u0026rsquo;s understanding of informed consent/assent and confirmed young people and their parents are the main decision-makers in their undertaking research. For clinical research acceptability, parents were more open for their child to participate in clinical research than young people. Parents expressed less preference for their child to participate in research that involved untested medicines and surgery, whereas young people expressed less preference to undertake research that involved untested medicines, surgery, and venepuncture. These novel findings indicate that the survey designed for this study effectively captured young people\u0026rsquo;s opinions in an online non-clinical setting and found a potential disconnect between the young person\u0026rsquo;s needs and opinions and parents\u0026rsquo; preferences of clinical research. The development of chILD research education and a young person forum is recommended to build and foster clinician/researcher relationships. Ultimately, this will allow for building stronger PPI links and improved chILD clinical research.\u003c/p\u003e","manuscriptTitle":"Childhood Interstitial Lung Disease clinical research: A child-parent dyad survey on research preferences and acceptability","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2024-06-25 05:24:39","doi":"10.21203/rs.3.rs-4468802/v1","editorialEvents":[{"type":"communityComments","content":0}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"101bbfaf-30ac-4093-87eb-bc477228717d","owner":[],"postedDate":"June 25th, 2024","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"posted","subjectAreas":[],"tags":[],"updatedAt":"2024-11-04T04:53:54+00:00","versionOfRecord":[],"versionCreatedAt":"2024-06-25 05:24:39","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-4468802","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-4468802","identity":"rs-4468802","version":["v1"]},"buildId":"qtupq5eGEP_6zYnWcrvyt","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}