Abstract
Background:
Increasingly long and complex informed consents have yielded studies demonstrating comparatively low participant understanding and satisfaction with traditional face-to-face approaches. In parallel, interest in electronic consents for clinical and research genomics has steadily increased, yet limited data are available for trio-based genomic discovery studies. We describe the design, development, implementation, and validation of an electronic iConsent application for trio-based genomic research deployed to support genomic studies of cerebral palsy.
Methods:
iConsent development incorporated stakeholder perspectives including researchers, patient advocates, institutional review board members, and genomic data-sharing considerations. The iConsent platform integrated principles derived from prior electronic consenting research and elements of multimedia learning theory. Participant understanding was assessed in an interactive teachback format. The iConsent application achieved nine of ten proposed desiderata for effective patient-focused electronic consenting for genomic research.
Results:
Overall, participants demonstrated high understanding and retention of key human subjects considerations. Enrollees reported high levels of satisfaction with the iConsent, and we found that participant understanding, iConsent clarity, privacy protections, and study goal explanations were associated with overall satisfaction.
Conclusions:
Although opportunities exist to optimize iConsent, we show that such an approach is feasible, can satisfy multiple stakeholder requirements, and can realize high participant satisfaction and understanding while increasing study reach.
Keywords: informed consent, multimedia, ethics, research, stakeholder participation, understanding, genomics
INTRODUCTION
Interest in the design and implementation of electronic informed consent platforms for both clinical care and biomedical research has been steadily increasing (Moore et al. 2017). Numerous challenges to obtaining truly informed genomic consents exist (Rego et al. 2020). Nevertheless, there is both growing interest in and utilization of electronic platforms for genomic research, although a number of important questions remain (Norton et al. 2024).
Electronic consent use in clinical research and biobank studies
Over time, the need for full disclosure has yielded increasingly long and complex traditional consents (Beardsley, Jefford, and Mileshkin 2007). Historically, when adapted to electronic formats, the long-form paper format is simply migrated to a tablet or similar device and reviewed by research coordinators during in person discussions. Research participants often do not fully comprehend information presented during the informed consent process (Flory and Emanuel 2004; Nishimura 2013). The updated Common Rule for human subjects research indicates key information presented in a “concise and focused” manner will be most valuable to potential participants in deciding whether or not to join a clinical research study (Menikoff, Kaneshiro, and Pritchard 2017). Participant satisfaction with traditional long-form consent approaches is often relatively low but can be improved with use of simplified, straightforward language (Jefford and Moore, 2008). In contrast, a biobanking study comparing electronic applications vs. long-form consents found that electronic consenting required not only significantly less staff time but was associated with improved participant understanding of key elements of the informed consent (McCarty et al. 2015).
Electronic consents may also improve recruitment and retention in clinical research studies (Boutin et al. 2016). This may be mediated in part by reducing access barriers for rural participants and by mitigating cultural and literacy barriers via the use of optional explanatory material (Lawrence et al. 2020). Assessments of stakeholders’ perspectives have also indicated that electronic frameworks can facilitate personalization and longitudinal interactions with research participants (De Sutter et al. 2020). Prior work has indicated that satisfaction is highly connected with ease of use and overall participant experience. Mobile phone-supported electronic consents show a high degree of participant satisfaction and engagement (Robins et al. 2020), while a biobank electronic consent study demonstrated that testers had a positive experience with the portal but reacted negatively to an extensive identification verification process (Thiel et al. 2015).
Electronic consent use in clinical genomic studies
Electronic informed consent has recently been expanded beyond biobanking studies to clinical genomic research by the Clinical Sequencing Evidence-Generating Research (CSER) consortium. The majority of participants found the approach acceptable and valued the access to testing that the study provided (Kraft et al. 2021). CSER also assessed participants’ perspectives regarding electronic consenting for gene panel-based testing for eleven conditions estimated to affect 1–2% of the general population. Investigators found that participants’ decision to join the study was related to genetic self-efficacy, limited concerns about genetic screening, trust in the researchers, and website user-friendliness (Cadigan et al. 2017).
Electronic consent use in genomic discovery research
Prior electronic consent studies conducted regarding residual neonatal blood spot analysis for broad research use, including genomic studies (Rothwell et al. 2017), demonstrated high overall participant satisfaction with electronic consent formats (Rothwell et al. 2020). Users have also highly rated the clarity and conciseness of information presented in electronic formats (Rothwell et al. 2020). Electronic consent utilization is also associated with higher participant understanding of most key study concepts compared to long-form consents (Rothwell et al. 2014), particularly if teachback questions are employed to reinforce key study concepts (Tait and Voepel-Lewis 2014).
Based on the foundational evidence summarized above, electronic consents have now been designed and implemented for several major ongoing genomic research studies, including the Australian Genomics study (Haas et al. 2021), Simons Foundation’s SPARK study, the Autism Speaks MSSNG initiative, and the National Institutes of Health’s All of Us study (Doerr et al. 2021). The All of Us study recently found that more than 95% of participants recognized the voluntary nature of the research study and were able to distinguish it from medical care.
Hurdles to electronic consents
Although electronic consents for genomic discovery research are already being utilized, several important gaps in the field remain. In the context of discovery-based genomic studies (particularly those using trio-based designs), participant satisfaction and understanding of key human subjects research elements have not been assessed. Nevertheless, recognizing the inherent potential of electronic consents, Parra-Calderón and colleagues proposed ten key elements (desiderata) for the successful utilization of an electronic consent platform for genomic research (Table 1) (Boutin et al. 2016; Haas et al. 2021; Parra-Calderon et al. 2018; Tenenbaum et al. 2016; Williams et al. 2015; Kaye et al. 2015; Budin Ljonse et al. 2017; Maxwell et al. 2017; Grady 2015; HEW 2014; CBCP Working Group 2017), although the challenges of implementing these elements have not previously been directly addressed.
Table 1.
Desiderata for Developing a Digital Genomic Informed Consent a
| Key Subject Areas and iConsent Goals | Desiderata Integration in iConsent |
|---|---|
| Participant engagement: Secure user interface enables continued and individualized consent management and engagement after enrollment b |
|
| Personalized participation: Transparency through provision of information at recruitment c |
|
| Participant information services: Ability to monitor each researcher’s access to data sets and biological samples over time d |
|
| Healthcare integration: For improved prevention, treatment, and care-deliverye |
|
| Genomic literacy: Facilitate understanding of precision medicine, genomic information, and implications of results f |
|
| Incidental findings: Bioethical principle of Autonomy supports individual’s right to know and to not know g |
|
| Digital accessibility: iConsent accessible from all possible electronic formats h |
|
| Identity management: iConsent employs a trusted identity management process i |
|
| Audit trail: Ability to reliably track consent status j |
|
| Security and privacy: Transparent protections k |
|
Desiderata derived from Parra-Calderón, et al. (2018)
Sources: Kaye et al. 2015; Budin-Ljøsne et al. 2017
Source: Maxwell et al. 2017
Sources: Grady 2015; HEW 2014
Sources: Kaye et al. 2015; Grady 2015
Sources: Kaye et al.; Maxwell et al. 2017; CBCP Working Group 2017
Sources: Boutin et al. 2016; Maxwell et al. 2017
Sources: Budin-Ljøsne et al. 2017
Sources: Boutin et al. 2016; CBCP Working Group 2017
Family trio (mother, father, and affected child)-based genomic study designs are widely utilized, as trio designs can help detect causative de novo mutations present in affected offspring but not found in either parent. De novo mutations are important contributors to neurodevelopmental disorders such as cerebral palsy (Jin et al. 2020). However, such designs can present unique challenges for electronic consents and have not been well-studied. By definition, three individuals must be enrolled to yield a complete trio. Trio members typically include both affected and unaffected family members. Trios often include both adults and children, and each member of the trio (or their legally authorized representative) must individually decide to participate in order for the full trio to be enrolled.
To address existing gaps, we sought to incorporate desiderata while utilizing concepts established by prior research. Accordingly, we describe the design, development, implementation and validation of a self-contained electronic iConsent application for trio-based genomic discovery research. This application is currently being utilized to enroll interested families in the Genetic Causes of Cerebral Palsy (GCCP) study conducted within the Cerebral Palsy Research Network (CPRN; www.cprn.org). We incorporated researcher, stakeholder, Institutional Review Board (IRB), and genomic data-sharing perspectives and assessed understanding and satisfaction among participants as well as factors driving these outcomes.
METHODS
The GCCP iConsent was developed to support our ongoing study of cerebral palsy (CP) genomics. The study is being conducted at participating sites within the CP Research Network. Phoenix Children’s / University of Arizona College of Medicine – Phoenix serves as the genomics hub for this study. The study utilizes the CPRN’s clinical registry (Hurvitz et al. 2020), which comprises >7,500 individuals with CP from centers across North America.
Participant driven enrollment in the GCCP study (overseen by the Phoenix Children’s central IRB) using iConsent provides a secure means of obtaining informed consent, which triggers sample collection via saliva kits mailed to the family’s home as a source of genomic DNA. After saliva samples are obtained for the trio, genomic sequencing (exome/ genome) and analysis are performed. Phoenix Children’s IRB reviewed and approved this study as the single IRB (sIRB). Informed consent was obtained from all participants.
The CP Research Network utilizes dedicated clinical documentation templates integrated within participating sites’ electronic medical record. Phenotypic data is entered by clinicians caring for patients in real time, and data is captured and securely transferred to the data coordinating center at the University of Pittsburgh (Gross et al. 2020). Participation in the CPRN clinical registry thus provides essential phenotypic data. Privacy-preserving records linkage may then be used to link clinical and genomic sequencing datasets, effectively connecting phenotype with genotype. Although participant level phenotypic data were not included in our analysis, these de-identified data were obtained for the trio-based genomic study through collaboration with the CPRN clinical registry.
Since our study design is based on mother-father-child trios, both biological parents are invited to participate given the high rate of de novo mutations in CP (Jin et al. 2020). This necessitated that the individual with CP, their biological mother, and their biological father each individually enroll. Under Arizona state law, legally authorized representatives are required to provide consent for those under the age of 18 and express permission is required for children 8–17 with the intellectual capacity to provide assent (as indicated by their legally authorized representative).
As we began the design of the iConsent application, needs assessments were conducted utilizing the Engage2020 Action Catalogue decision support tool (2020). We adopted a community-based participatory action design framework incorporating multiple stakeholder perspectives. Iterative consensus-building occurred using mixed methods approaches, including user committees and deliberative forums. Study information was provided to potential participants using an interactive webpage design (WordPress) and study data were collected and maintained in a secure Research Electronic Data Capture (REDCap; Vanderbilt University) environment housed at the University of Arizona (Harris et al. 2009).
We used an embedded series of teachback questions to assess initial participant understanding of core human subjects research concepts at the time of enrollment. We then deployed a follow-up survey to assess enrollees’ overall retention by re-presenting teachback questions (Supplemental Table 1). We assessed satisfaction in the same survey, as well as factors that could influence satisfaction. As prior work identified trust as a primary factor influencing participation in CP genomic research studies (Wilson et al. 2021), we assessed participant trust using the Hall Trust in Biomedical Research (H-TBR) scale, short form (Hall et al. 2006).
Statistical methods
Understanding scores were calculated by summing the number of correct responses out of 5 and summarized by respondent type (mother, father, or proband). The distribution of initial and follow-up scores were compared by respondent type using two-tailed Wilcoxon rank sum tests. Individual survey question responses were tabulated by respondent type and compared using chi-square tests.
To evaluate pairwise differences in Understanding vs. retention (defined as continued understanding at the time of follow-up using the same questions initially presented) scores among the subset of individuals who completed both the initial and follow-up surveys, the understanding score was stratified by respondent type and analyzed using Wilcoxon signed rank tests.
Kruskal-Wallis tests were performed to assess the relationship between participant satisfaction and other factors including understanding, retention, participant trust, and measures of participants’ impressions of study clarity¸ privacy protections, explanations of study goals, and the adequacy of information provided regarding study participation. As the study was primarily designed to characterize responses by group, a formal sample size analysis was not appropriate. All GCCP study participants at least 18 years old and able to make independent medical decisions were invited to complete the initial survey. Of those, participants who returned a saliva sample were invited to complete the follow-up survey. All analyses were performed in SAS version 9.4 (SAS Institute, Inc).
RESULTS
I. Foundational principles, needs assessment, and design
Researcher perspective
A design committee was first assembled from research team members, which identified key study goals and essential iConsent functions. Fundamentally, the application needed to facilitate enrollment while preserving participant understanding and maximizing satisfaction (here captured by the user experience). We also needed to generate metadata to connect members of the mother-father-child trio, coordinate sample collection, and generate unique identifiers to link de-identified participant phenomic and genomic data since these were being collected using different protocols at different sites. From a practical standpoint, our platform needed to be a) self-contained; b) professional in appearance (both inviting and secure); and c) user-friendly. We thus sought to balance competing needs – the need to generate a robust data set with the need to limit enrollee burden to facilitate participation. Instilling participant trust was another primary goal of the study team.
As we conceptualized iConsent, we sought to address existing gaps in the field. Specifically, we sought to determine if a trio-based genomic research iConsent application yielding high participant understanding and satisfaction could be developed. We also set out to identify specific factors influencing understanding and satisfaction to illuminate subsequent quality improvement efforts.
With these principles in mind, we developed early study mock-ups, scrutinizing language and written communication heavily at these stages. We sought to explain the study opportunity, risks, and potential benefits to prospective participants using simplified language in an engaging, succinct format. Once the original study outline had been established, we incorporated key elements related to community perspectives, human subjects protections, and data-sharing needs to further refine our study framework (Supplemental Figure 1).
II. Iterative development, incorporating stakeholder perspectives
Community perspective
We incorporated perspectives from CP community and advocacy leaders by partnering with the CPRN community advisory committee. This stakeholder group included individuals with CP as well as parents of a child with CP to represent potential participants. We solicited feedback through a brief survey, inviting impressions of early mock-ups. While comments and suggestions varied among group members, several important themes emerged to optimize the user experience and thus overall satisfaction (Table 2). Community feedback prompted us to create a more intuitive and easily navigable iConsent user interface. Recommendations for clear and concise language were also incorporated before presentation to our IRB.
Table 2.
Incorporated Recommendations from Community Stakeholders, IRB members, and Mayer’s Multimedia Learning Theory
| Topic | Incorporated elements |
|---|---|
| Language | Ensure language is concise, consistent, and understandable to target audience |
| User experience | Revise graphical user interface to be more intuitive |
| Stress key study elements | Emphasize key aspects of the study via placement and presentation |
| Clarify trio enrollment study design | Emphasize that all three family members need to individually enroll in order for the family unit to participate (given that key information often needs to be presented multiple times in different ways) |
| Assent as appropriate | Explain to parent why their child needs to provide assent |
| Communication of study information | Balance brevity with full disclosure and detailed study information through the employment of ‘More Info’ tabs within the application Optimize language to ensure it is at a 6th grade reading level |
| Risk/benefit consideration | Convey what participation entails, ensuring potential benefits and/or risks of participation are outlined before informed consent is obtained |
| Comprehension | Ensure teachback questions assess understanding of key aspects of participation |
| Documentation of informed consent | Provide copies of electronically signed, time stamped informed consents to enrolled participants |
| Coherence | Concise language was used |
| Signaling | Cues oriented users to essential material (bold typeface, 1-2-3, etc.) |
| Redundancy | Reiteration was limited to emphasizing key concepts |
| Spatial contiguity | Text was grouped for user-friendliness; bullet points employed |
| Temporal contiguity | Related text and images were presented together |
| Segmenting | User-paced pages partitioned material; ability to pause and return |
| Pretraining | Introductory video provided study overview |
| Modality | Graphics and narration were utilized |
| Multimedia | Text, graphics, video, and human voice were integrated |
| Personalization | Conversational style employed throughout |
| Voice | Human voice-over used in video narration |
| Image | Unnecessary images were avoided to focus on key content |
| Convergence between community stakeholder and IRB recommendations with concepts of Mayer’s Multimedia Learning Theory was noted, especially relating to language use and presentation of information. Early IRB and stakeholder engagement yielded valuable suggestions with actionable solutions, while integration of Mayer’s principles further advanced communication targets prioritized in community feedback. | |
Principles derived from Mayer’s Cognitive Theory of Multimedia Learning (2008)
IRB perspective
As electronic consents had not previously been utilized at our institution, we partnered with our IRB to ensure integration of robust human subjects protections. We relied on established principles that provision of informed consent requires decision-making capacity, voluntariness, adequate information, and understanding (Rego et al. 2020). We presented mock-ups via a series of consensus-building deliberative forums with IRB leaders, yielding further refinements. IRB leaders emphasized that key informed consent concepts needed to be presented in sufficient detail to be understood by study participants. They were enthusiastic about the use of embedded teachback questions to ensure understanding and retention of key study concepts for the iConsent to represent a valid alternative to traditional in-person, long-form based informed consent. Important concepts derived from these discussions are outlined in Table 2.
Data-sharing considerations
Worldwide, data-sharing concepts have become an important part of genomic research, as combining cohorts can increase statistical power and facilitate discovery. As data-sharing has been a priority topic for the National Institutes of Health (NIH) and has become an integral part of federally-funded genomic research in the United States, we sought to ensure alignment with the latest NIH recommendations (2023). We incorporated current guidelines for precise informed consent (Menikoff, Kaneshiro, and Pritchard 2017) to maintain transparency and facilitate trust between researchers and study participants while still enabling high-level data-sharing for genomic discovery (Robinson et al. 2013). This resulted in additional meetings with IRB leaders and several additional rounds of revisions, including the incorporation of broad data and sample sharing for research use as an intrinsic aspect of study participation. Informed consent language was expanded to detail data and sample sharing, informational content was added, and interactive teachback questions were modified to promote participant understanding.
III. iConsent implementation
Our iConsent platform, employing a rich media format developed in a WordPress environment, presented study information and requirements for participation (Supplemental Figure 2). We incorporated multimedia elements given evidence that this improves participant experiences (Synnot et al. 2014). We used a combination of stock photos and photographs submitted to both the LifeShots competition (sponsored by the American Academy of Cerebral Palsy & Developmental Medicine) and CPRN Photo Contest after obtaining appropriate permissions. We produced a short animation (Video Animation, Inc) providing an overview of the study that encapsulated crucial aspects of participation in under 2 minutes (Supplementary Video). The second portion of the application utilized REDCap to securely collect participant personally identifying information.
Content development
We sought to satisfy human subjects regulations while balancing study population needs and research goals. We incorporated elements of cognitive load theory (Lavie et al. 2004) and reduced individual options to lessen participant burden. We then incorporated elements of Mayer’s multimedia learning theory (Table 2) (Mayer 2008), which has been reported to improve both understanding and satisfaction (Gesualdo et al. 2021). We anticipated that participant preferences for study information would vary, but anticipated that presenting too much information in too short a time frame would reduce understanding. Therefore, we identified crucial human subjects research elements that represented core participant-facing material (see Table 2) and then provided “More Info” tabs for those interested in obtaining more detail. Additional community perspectives at this stage directed efforts to balance thoroughness and brevity to yield clear, concise language (Grady et al. 2017).
Final review and integration
After incorporating these changes, the research team internally re-reviewed the iConsent. We confirmed suitability of the application’s content for launch via a final review with IRB leaders. These measures ensured that critical concepts identified during iterative development were faithfully incorporated and concerns were addressed and recommendations integrated to the best of our ability. In cases where recommendations were at odds (i.e., remove a specific element vs. retain that element), decisions were reached by the study team in a consensus-building fashion.
After extensive internal beta testing, we launched the iConsent in March 2021. Potential participants were invited to participate in the study by email, study brochure, study poster, or direct mailing if they received care at a participating center (AI duPont Hospital for Children, Colorado Children’s, Nationwide Children’s, Phoenix Children’s, Seattle Children’s, St. Louis Children’s, or the University of Texas – Houston). We tracked iConsent completion rate (full trio enrollments) using an internal tracking system and developed an automated text messaging tool to reduce the proportion of incomplete trios. We adapted our iConsent to iOS, receiving approval to deploy the app from Apple, Inc. and launched iConsent for iPad August 2022.
IV. iConsent validation
User experience and implementation
Of the 561 families contacted, 458 (81.6%) returned at least one family member survey. Data from 821 participants was available to assess understanding (380 fathers, 420 mothers, and 21 probands). Responses to teachback questions indicated good overall understanding of key human subjects considerations, with a mean score = 4.1 ± 1.1 (out of 5), including the voluntary nature of participation, potential risks of participation, study goals, and privacy considerations (Figure 1, Supplemental Figure 3). However, we noted that many participants, particularly mothers, answered Question 1 “Do I have to participate in the genetic causes of CP study?” incorrectly (Supplemental Table 2). We solicited qualitative feedback from those that answered incorrectly, allowing us to identify a common theme – many parents interpreted this as an inquiry as to whether their participation was needed to complete the trio. Consequently, we rephrased Question 1 to read “Is it my choice to be part of the Genetic Causes of CP Study?” This led to a higher correct response rate (99.2% for the rephrased question vs. 56.8% for the initial question; p <0.0001).
Figure 1.
Overall Comparison of Initial (Understanding) and Follow-up (Retention) Scores
a Score represents the number of correct answers out of 5 questions
Follow-up Survey:
After participating families enrolled in the study and provided saliva samples, they received a follow-up survey. Nearly forty-two percent of enrolled families (142/340) responded to the follow-up survey. The majority of respondents (>90% in each instance) indicated that they were satisfied with their experience (assessed by ease of use of the iConsent), that they were given adequate information about the study to make an informed decision to participate, that the study goals were clear, that the iConsent was clear, and that privacy protections were sufficient (Figure 2).
Figure 2.
Overall Participant Satisfaction and Responses for Key Elements Impacting Satisfaction. Darker shades represent higher degrees of Satisfaction
We assessed participant retention by comparing initial teachback scores to follow-up survey teachback scores (the same questions were asked in both instances). We found a small but statistically significant dropoff in retention (mean initial understanding = 4.1 ± 1.1 vs. mean retention = 3.9 ± 1.0; p<0.0001 by Wilcoxon signed rank test). However, retention remained relatively high even after a mean of 101.7 days ± 116.1 had elapsed since initial enrollment. We assessed the potential impact of the modified Question 1 via a sensitivity analysis, which demonstrated that this change did not substantially impact our overall interpretations (Supplementary Methods and Results).
Finally, we turned our attention to the factors that may have influenced overall satisfaction. We found that study understanding, iConsent clarity, privacy protections, and appropriate explanations of study goals were associated with participant satisfaction (Figure 3, Supplemental Table 3). The adequacy of information presented was equivocally associated with satisfaction (p = 0.0552). Conversely, no relationship was found between retention of key concepts and overall satisfaction. Furthermore, no association between participant trust and satisfaction was evident.
Figure 3.
Association Between Study Elements and Overall Satisfaction Scores
a Satisfaction score was scored from strongly disagree (-2) to strongly agree (2) on the survey question “I had an easy time signing up for the Genetic Causes of CP study.”
b Comprehension score was defined as the number of comprehension questions answered correctly on the initial survey out of 5 possible.
c Retention score was defined as the number of comprehension questions answered correctly on the follow-up survey.
d Clarity was scored from strongly disagree (-2) to strongly agree (2) on the survey question “When I joined the study, I understood what I had to do to sign up.”
e Adequacy of Information was scored from not nearly enough (-2) to way too much (2) based on the statement “The amount of information I was given about the study was _________.”
f Privacy was scored as yes (1) or no (0) on the survey question “Are the privacy protections for the study strong enough?”
g Study goal was scored from strongly disagree (-2) to strongly agree (2) on the survey question “When I signed up for the study, I understood that the researchers are trying to find changes in my family's DNA that cause cerebral palsy.”
- “Doctors who do medical research care only about what is best for each patient.” (Agree)
- “Doctors tell their patients everything they need to know about being in a research study.” (Agree)
- “Medical researchers treat people like "guinea pigs." (Disagree)
- “I completely trust doctors who do medical research.” (Agree)
DISCUSSION
We describe the design, development, implementation, and validation of a fully self-contained electronic iConsent application suitable for multi-institutional recruitment of mother-father-child trios to participate in genomic discovery-based research. Although our study was targeted to families with a child with CP, the iConsent was designed to be modular enough to readily adapt to other applications. We incorporated elements identified as important by broad stakeholder representation, including the research team, CP community, IRB, and federal agencies. Our findings of high participant understanding and retention as assessed by teachback questions served as a key validation of our approach. We also implemented nine of ten desiderata previously proposed for electronic genomic research consents (Table 1). We did not incorporate participant information management into iConsent design as we deemed it impractical to notify participants of each instance their data were utilized, particularly if their data became part of a large-scale de-identified genomic database as is typical for current genomics research.
We developed and validated our application as a standalone tool to increase access to research while preserving participant understanding and satisfaction. More than 95% of participants indicated that they felt the iConsent was clear, and >90% of participants also reported that it contained an adequate amount of information for them to make an informed decision. These findings suggest that the multimedia structure adopted and learning principles utilized were effective in delivering digestible content, as indicated by high overall participant understanding scores.
Given our study design, we were unable to formally compare our outcomes to traditional paper-based in-person consenting, representing a potential limitation of our findings. Accessibility may be somewhat lesser for those of lower socioeconomic status as our application requires a device with internet connection and is currently only available in English, although a Spanish language format is in development. Inherent with any electronic method is the loss of direct person-to-person contact, which must be acknowledged and could disproportionately impact some members of society. As SES, education level, and literacy were not the foci of this study, data on these metrics was not collected. Nevertheless, as prior studies have indicated, participant decision aids may be useful in addressing potential barriers to participation in the future (Gillies et al. 2015).
Our prior work with stakeholders (families with CP) suggested the most important potential factors influencing willingness to participate in genomic research included (i) a clear understanding of study goals; (ii) adequacy of privacy protections; and (iii) trust in biomedical researchers (Wilson et al. 2021). Here, when we surveyed participants who had already enrolled in CP genomic research, we confirmed that understanding study goals and sufficient privacy protections were both associated with overall satisfaction, while participant trust was not identified as a factor influencing satisfaction. This may reflect subtle differences in families’ trust of biomedical researchers in general vs. those focused on salient topics (in this case, CP). It is possible that a certain threshold of trust is needed for participants to enroll. These findings could also indicate that trust does not influence behavior as much as it may reflect existing attitudes.
We found that participant understanding, iConsent clarity, privacy protections, and study goal explanations all contributed to overall satisfaction. Our findings indicate these elements should be considered to design effective electronic consents for genomic research. Future research will include opportunities to optimize the presentation of human subjects research concepts and further improve understanding and retention. Planned iConsent revisions are anticipated to further enhance the user experience via a personalized study dashboard and to facilitate ongoing communication with the study team using text messaging and automated updates to further align researchers and participants for mutually beneficial clinical research studies.
Supplementary Material
Supplemental Figure 1. Stakeholder Perspectives. Integrated needs analysis incorporating researchers, IRB, community, and data-sharing perspectives.
Supplemental Figure 2. Electronic iConsent App Representative Image
Supplemental Figure 3. Distribution of Initial (Understanding) and Follow-up (Retention) Scores
Supplemental Table 2. Overall Initial and Follow-up Question Responses – Breakdown
Supplemental Table 3. Association Between Study Elements and Overall Satisfaction Scores (Expanded Data)
Supplemental Table 1. Follow-up Survey Questions
Supplementary Methods and Results. Sensitivity Analysis and Comparison of Initial and Follow-up Scores to Survey Question 1
Supplementary Video. Brief Animated Study Overview
Acknowledgments
We are grateful to the families within the cerebral palsy community who have supported this work, particularly members of the CP Research Network community advisory council who provided critical feedback in the initial stages of this work. We appreciate the contributions of Jacob Kean to initial discussions of the iConsent and the input of Steve Esquivel regarding design considerations for the iConsent. We particularly wish to thank Bill Lewis, who provided crucial logistical and technical feedback and support for this project from conceptualization through implementation.
Funding Statement
MCK was supported by the National Institute of Neurological Disorders and Stroke (NINDS) (1R01 NS106298 and NS127108).
Declaration of Interest
Paul Gross is President and CEO of the CP Research Network, which contributed to the funding of this project. Mr. Gross personally made financial contributions (donations) to CPRN to support this work, but receives no financial compensation related to either. Dr. Noritz has consulted for Abbott Nutrition, unrelated to this project. Dr. Shrader receives research funding from NIH and serves on the National Advisory Board for Medical Rehabilitative Research for NIH/NICHD.
Data Availability
Participant informed consent authorizes sharing of de-identified data with researchers for research purposes only. Anonymized survey data will be made available upon reasonable request to the corresponding author.
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Associated Data
This section collects any data citations, data availability statements, or supplementary materials included in this article.
Supplementary Materials
Supplemental Figure 1. Stakeholder Perspectives. Integrated needs analysis incorporating researchers, IRB, community, and data-sharing perspectives.
Supplemental Figure 2. Electronic iConsent App Representative Image
Supplemental Figure 3. Distribution of Initial (Understanding) and Follow-up (Retention) Scores
Supplemental Table 2. Overall Initial and Follow-up Question Responses – Breakdown
Supplemental Table 3. Association Between Study Elements and Overall Satisfaction Scores (Expanded Data)
Supplemental Table 1. Follow-up Survey Questions
Supplementary Methods and Results. Sensitivity Analysis and Comparison of Initial and Follow-up Scores to Survey Question 1
Supplementary Video. Brief Animated Study Overview
Data Availability Statement
Participant informed consent authorizes sharing of de-identified data with researchers for research purposes only. Anonymized survey data will be made available upon reasonable request to the corresponding author.