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. Author manuscript; available in PMC: 2020 Dec 1.
Published in final edited form as: J Empir Res Hum Res Ethics. 2020 Jun 18;15(5):452–464. doi: 10.1177/1556264620927583

Attitudes of mothers regarding willingness to enroll their children in research

Jane Paik Kim 1, Maryam Rostami 1, Laura Weiss Roberts 1
PMCID: PMC7606296  NIHMSID: NIHMS1589414  PMID: 32552481

Clinical research has led to tremendous contributions to and improvements in children’s health, as demonstrated by numerous breakthroughs in the areas of pediatric cancer (e.g., chimeric antigen receptor T-cell therapy for acute lymphoblastic leukemia), pediatric genetic conditions (e.g., cystic fibrosis transmembrane conductance regulator modulator therapies), and neonatology (e.g., surfactant therapies for premature infants), among others (Gardner et al., 2017; Kramer & Clancy, 2016; Lee et al., 2015; Maude et al., 2014; Ramanathan, 2006). These breakthroughs have allowed many children to thrive despite significant health threats and have improved the health of populations throughout the world.

The discovery of effective new treatments and prevention strategies rests heavily on the participation of a significant number of children with diverse illnesses and children in good health in clinical research. Such participation, in turn, relies on the engagement of parents and guardians who decide whether to enroll their children in protocols with intrinsic burdens as well as potential risks and benefits. Human subjects regulations are appropriately and necessarily strict in research involving youth because of the limited freedoms and decision-making capacity of children and the greater vulnerability of children in the research situation (Office for Human Research Protections, 2016). In the past few decades, medical decision-making has shifted from a paternalistic approach to a more patient-centered approach, and such changes have impacted research decision-making. Pediatric shared decision making, in particular, is challenged by the fact that the parent is the surrogate decision maker (Barry & Edgman-Levitan, 2012; Boland et al., 2019; Lipstein, Lindly, Anixt, Britto, & Zuckerman, 2016; Opel, 2018). Because scientific neglect of childhood illnesses is not acceptable, it is valuable to understand how parents and guardians view research that involves children, how parents and guardians perceive the risks of research that involves children, and how certain influences, such as past personal experiences with illness, shape the enrollment decision-making of parents and guardians.

Several authors have examined various issues regarding informed consent in the context of pediatric clinical investigations (e.g., phase 1 oncology, leukemia trials, and in trials involving neonates born with evidence of perinatal asphyxia; Allmark & Mason, 2006; Baker et al., 2013; Eder, Yamokoski, Wittmann, & Kodish, 2007; Leibson & Koren, 2015). Others have examined-via interviews with parents whose children were enrolled in oncology or HIV trials-the degree to which parents understand information when seeking consent, and have sought to understand factors that influence the decision-making process (Chappuy, Doz, Blanche, Gentet, Pons, & Tréluyer, 2006). There is a body of scholarly work examining ethical issues that emerge from the conduct of pediatric research, including issues related to recruitment, continuous consent, therapeutic misconception, and others (Dekking, van der Graaf, Kars, et al., 2015; Pickler & Martin, 2010). Taken together, these scholarly efforts underscore the unique and complex ethical challenges germane to the setting of pediatric research. One distinction, for example, is that even in studies involving very seriously ill adults, the use of alternative decision makers may or may not be required, whereas in research involving children, permission of an alternative decision maker in the form of a parent or guardian is typically required (Dunn et al., 2013; Kodish, 2003; Overton, Appelbaum, Fisher, Dohan, Roberts, & Dunn, 2013; Rossi, Reynolds, & Nelson, 2003).

Among the studies that have sought to investigate the determinants of parental decision-making, several factors have been identified, such as trust and the propensity to take risks (Fisher, McKevitt, & Boaz, 2011; Wiener, Viola, Wilfond, Wendler, & Grady, 2015). For example, in an assessment of parents whose children participated in a randomized, placebo-controlled trial, parents who consented had higher levels of trust and altruism, among other factors including perceived potential for enhanced care (Hoberman et al., 2013). In other work, parents of chronically and terminally ill children were prepared to take greater risks in treatment trials, but similar levels of acceptance of risk were not seen in quality of life trials (Caldwell, Butow, & Craig, 2003). Other work has examined parental willingness to enroll children as related to demographic factors, such as parental age, and other factors, such as the child’s health status and parental trust in researchers’ honesty (Leykin, Roberts, & DeRubeis, 2010; Miu, Heilman, & Houser 2008; Tait, Voepel-Lewis, & Malviya, 2003; Tarini, Goldenberg, Singer, Clark, Butchart, & Davis, 2010). Some studies show that parental demographic characteristics do not differ for parents making decisions for chronically ill children (Mack, Wolfe, Cook, Grier, Cleary, & Weeks, 2011; Rosenberg et al., 2014).

Many studies of children’s health care suggest that mothers have an important role in making health care decisions for their children (Benin, 2006; Gross & Howard, 2001; Minkovitz, O’Campo, Chen, & Grason, 2002; Pridham & Chang, 1991). Case and Paxson’s results (2001) showed that health choices and behaviors (e.g., whether the child had a routine doctor visit in the last year; whether the child visited a dentist in the last year; whether the child has a usual place for routine medical care and a usual place for sick care; whether household members smoke in the home) are disproportionately made by mothers. Phares and Compas (1992), in a review of 577 articles, found that more mothers were included in developmental psychopathology research compared to fathers. Gross and Howard (2001) explored mothers’ decision-making processes for health care and emphasized the importance of mothers’ perceptions in making decisions about health care for children. Of the very few papers that investigated the decision-making of mothers with potential vulnerabilities, such as a history of personal health issues, Tymchuk, Yokota, and Rahbar (1990) found that mothers with intellectual disabilities made hypothetical decisions that were deemed at least as good as those made by mothers from similar backgrounds without intellectual disabilities. A few qualitative studies examining the parenting experiences, identities, and experiences of social support of mothers with chronic mental illness demonstrated that mothers with chronic mental illness express a range of challenges and family support needs (Jones, Pietilä, Joronen, Simpson, Gray, & Kaunonen, 2016; Rampou, Havenga, & Madumo, 2015). Such studies provide evidence for an additional layer of stressors and illness associated influences on interpersonal resources, coping abilities, social and economic levels of function, that impact the degree of autonomy in decision making among mothers with chronic illness.

A large body of scholarly work examines factors that influence the willingness of parents to allow their children to participate in clinical research, but fewer studies focus on parents who endorse health issues themselves (Pickler & Martin, 2010; Rothmier, Lasley, & Shapiro, 2003; Tait, Voepel-Lewis, & Malviya, 2003) and their perceptions of the risk of research. The association between participants’ risk perceptions and their willingness to participate in research has been examined (Bentley & Thacker, 2004; Dunn et al., 2009; Roberts et al., 2002), in light of questions regarding the capacities of people with mental illness to assess the risks of research participation. Enrolling children in research is a distinct decision-making process, however, that necessitates its own considerations (Baek et al., 2017; Knowles, Christensen, & Bentall, 2005; Leykin, Roberts, & DeRubeis, 2010).

In this paper we focused on mothers who potentially face the decision of enrolling their children in research, specifically their perceptions of risk and the association between perceptions of risk and expressed willingness to enroll children in research. We also investigated the factors that might affect mothers’ perceptions of risk and their willingness to enroll children in research studies, such as mothers’ self-reported mental health status and the health status of children. Our first hypothesis was that perceptions of risk across the eight procedures and the expressed willingness of mothers to enroll their children in research would be negatively related. Our second hypothesis was that mothers without mental health issues with children in good health would be the most likely to identify risks associated with research procedures and thus would be the least inclined to express willingness to enroll their children in protocols that involved these research procedures. We evaluated the possible effects of mothers’ mental health statuses, as well as interactions between mothers’ mental health statuses and children’s health statuses, on mothers’ decisions regarding their children’s participation in eight different kinds of clinical research, adjusting for potential confounders such as maternal demographic characteristics and perceptions towards medical research.

Methods

Study design

We surveyed mothers in order to (1) better understand factors associated with their willingness to enroll children in research and (2) determine whether mothers of children with at least one physical or mental health issue and mothers with children without health issues hold a different set of views. We defined comparison groups based on two important factors: (A) mothers’ mental health status and (B) children’s health status.

Participants

We conducted recruitment using Amazon Mechanical Turk (MTurk) (Gillan & Daw, 2016). MTurk is a web service that allows individuals to outsource tasks that can be performed virtually to an on-demand pool of participants. “Requesters” post tasks called “human intelligence tasks” (HITs) to an online marketplace to be completed by participants. For our study, MTurk prospective participants who reported living in the United States, being at least 18 years of age, and who had an MTurk approval rating of at least 90% completed a brief screening assessment consisting of 10 questions on demographics. We utilized tools embedded in Mturk to invite prospective participants who achieved an approval rating of greater than 90% on all previous HITs for all requesters. Applying these criteria ensured quality control; we invited only participants who were attentive and likely to complete the surveys. Participants opted into the study after this initial selection process. Participants logged onto MTurk, viewed our HIT on the online marketplace, and self-selected into the study, making their own decision to participate.

An advertisement was placed on the MTurk platform for prospective participants to complete a HIT, in this case, a survey entitled “Health Information Survey for parents”. We kept the description of the study in the HIT very vague so that the HIT itself would not bias the selection of participants. The screening survey was open for few days in April 2018. Respondents were paid $0.25 for completing the screening assessment. In total, 590 individuals completed the screening assessment.

In the screening survey, respondents were queried regarding their parental status. Respondents who met our study inclusion criteria, i.e., who identified as mothers (n=206), were eligible to participate in our study. There were no exclusion or inclusion criteria related to children’s health statuses. Using the same procedure as described above, an advertisement for our survey was posted to MTurk from April 26 to May 29 of 2018, visible only to eligible study participants who met our inclusion criteria. Among the eligible participants, 126 out of 206 (61.2%) individuals successfully completed our survey. To clarify, this rate is the proportion of individuals who completed the survey among all survey respondents who were eligible. In the literature, longitudinal MTurk studies collecting follow-up data from participants (following single lags of 1–12 months) obtained response rates between 44 and 60% (Chandler, Mueller, & Paolacci, 2013). Study respondents who clicked on the advertisement were given a brief description and a weblink to our survey instrument hosted on Qualtrics (Snow, 2012). Respondents were required to read and agree to an electronic consent form before proceeding. Individuals who provided electronic consent were then lead to the 152-question survey, which was estimated to take 30 minutes to complete. Respondents were paid $6.00 for completing the survey.

Survey Instrument

Web-based surveys were hosted by Qualtrics (Snow, 2012) and electronically distributed to individuals through MTurk, as described above. The initial screening survey consisted of 8 questions regarding participants’ demographics and was estimated to take 1 to 2 minutes to complete. Our full survey is a compilation of standardized instruments and home-grown instruments used in prior works in empirical ethics (Roberts, Hammond, Warner, & Lewis, 2004; Roberts et al., 2002; Roberts & Kim, 2014, 2017) that were adapted for this specific domain.

Perceived risk and willingness to participate in eleven research projects procedures.

For this report, we analyzed responses from the section of the survey that assessed participants’ perspectives regarding research projects procedures. The “Research Projects Procedures” section of the follow-up survey queried participants’ views of 8 different research procedures (see Appendix 1 for further details). After a description of each research projects procedure, participants were asked two questions (total of 16 items). The first set of questions related to “willingness” to participate. Participants were asked to rate their answer to the question “How willing would you be to have your child participate in [procedure X]?” on a 5-point Likert scale to (1 = “Not at all willing”, 2 = “A little willing”, 3 = “Somewhat willing”, 4 = “Quite willing”, 5 = “Highly willing”). The second set of questions related to “risk” of the research projects procedure. Participants were asked to rate their answer to the question “How risky is this [procedure X] to children who participate?” on a 5-point Likert scale (1 = “Not at all risky”, 2 = “A little risky”, 3 = “Somewhat risky”, 4 = “Quite risky”, 5 = “Highly risky”).

Trust in medical researchers.

Participants were asked to rate 5 items about trust in medical researchers on a 5-point scale. These items were derived from the Trust in Medical Research scale (Mainous et al., 2006). Possible scores ranged from 5 to 25, with higher scores indicating greater trust in medical research. In addition to examining scores as a continuous variable, we also created a dichotomous variable based on exploratory data analysis, i.e., “weak to moderate trust in medical researchers” (<20) and “strong trust in medical researchers” (≥20).

Mothers’ mental health status.

Respondents were asked if they had ever been diagnosed with any psychiatric disorder, depression, mood disorder, or anxiety.

Children’s health status.

Respondents were asked if they had at least one child living with either a chronic or significant medical disorder, psychiatric disorder, intellectual disability, neurogenetic disease, or developmental delay.

Ethical approval.

The Institutional Review Board at Stanford University approved this study protocol. Before the start of the survey, all participants were provided with an electronic version of the consent form. All participants provided electronic consent before participation. Each respondent was assigned a Study ID number at random. Participants’ MTurk identification numbers were replaced with our Study ID numbers, and MTurk IDs were deleted, anonymizing the study respondents. Demographic information included self-reported history of illness, history of illness among children, gender, ethnicity, race, education, number of children, and age of youngest child. Socio-economic status was not collected.

Data Analysis

Basic descriptive statistics and frequencies were used to describe the four groups of mothers who participated in this study: (1) mothers without mental health issues with children without any health issues (MHCH), (2) mothers without mental health issues with at least one child with health issues (MHCI), (3) mothers with at least one mental health issue with children without any health issues (MICH), (4) mothers with at least one mental health issue with at least one child with health issues (MICI).

Differences among participant groups were analyzed using ANOVA and chi-squared tests. Mean ratings of domains (i.e., perceived risk and participation willingness for each research procedure) were analyzed using ANOVA tests and the effect sizes are reported. Within group differences were analyzed using repeated measure ANOVA tests. Generalized estimating equations (GEE) were used to assess (1) the association between the perceived risk of research procedures and sociodemographic factors, and (2) the association between participation willingness and perceived risk, adjusting for potential confounders. Potential confounders included the mother’s self-reported mental health status, children’s health status, the interaction between the mother’s mental health status and children’s health status, race, ethnicity, level of education, and trust in medical researchers. All statistical analyses were performed using SPSS version 25 and R version 1.0.153.

Results

Respondents (N = 126) were 35.4 years of age on average (SD = 8.04). The majority of participants identified as white (n = 104, 82.5%), and the remaining participants identified as another race (n = 22, 17.5 %) including African American, Asian, and other. A majority of participants did not identify as Hispanic or Latino (n = 111, 88.1%). Of the entire sample, 54% of mothers (n = 68) had either a college degree or graduate or professional school degree. There were no statistically significant group differences between mothers with respect to age, ethnicity, race, and education. Demographic characteristics of mothers are shown in Table 1.

Table 1.

Demographic characteristics

Mothers without mental health issues Mothers with at least one mental health issue Overall P valuea
(n=54) (n=72)
Demographics Mothers with only healthy children Mothers of children with at least one physical or mental health issueb Mothers with only healthy children Mothers of children with at least one physical or mental health issueb
(n=46) (n=8) (n=48) (n=24) (N=126)
Age, Mean (SD) 34.96 (8.21) 36.75 (6.98) 35.85 (8.10) 35.04 (8.27) 35.43 (8.04) 0.90
Ethnicity, n (%)
 Hispanic or Latino 5 (10.9%) 1 (12.5%) 4 (8.3%) 5 (20.8%) 15 (11.9%) 0.48
 Not Hispanic or Latino 41 (89.1%) 7 (87.5%) 44 (91.7%) 19 (79.2%) 111(88.1%)
Race, n (%)
 White 32 (69.6%) 8 (100%) 43 (89.6%) 21 (87.5%) 104 (82.5%) 0.10
 Non-Whitec 14 (30.4%) - 5 (10.4%) 3 (12.5%) 22 (17.5%)
Education, n (%)
 Less than high school 1 (2.2%) - - - 1 (0.8%) 0.07
 High school degree 4 (8.7%) 2 (25%) 6 (12.5%) 1 (4.2%) 13 (10.3%)
 Some College 14 (30.4%) 6 (75%) 16 (33.3%) 7 (29.2%) 43 (34.1%)
 College degree 18 (39.1%) - 21 (43.8%) 10 (41.7%) 49 (38.9%)
 Graduate or professional school 9 (19.6%) - 4 (8.3%) 6 (25%) 19 (15.1%)
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a

P values from ANOVA and Chi-square, as appropriates

b
Health conditions:
  • Chronic or significant medical disorder
  • History of psychiatric disorder
  • Intellectual disabilities
  • Neurogenetic disease
  • Developmental delays
c
Non-White includes:
  • Asian (n=7; n=0, n=4, n=2, in the 4 respective groups)
  • Black or African American (n=7, n=0, n=1, n=1, in the 4 respective groups)

Perceived risk of research procedures (overall trends):

Overall, respondents rated all eight procedures as having low to moderate risk (range of means = 1.82 to 2.93 out of 5; Table 2). On average, participants labeled “Images of your child’s head being taken” as having the highest risk (mean = 2.93 out of 5, SD = 1.2) and “Filling out questionnaires” as having the lowest risk out of the eight procedures (mean = 1.82, SD = 1.07). Although there were no significant differences between the four groups in their ratings of each procedure, the healthiest group (i.e., the MHCH group) tended to provide slightly higher ratings of perceived risk, although still within the low to moderate range (range of means = 1.89 to 3.17 out of 5; Table 2). Three of the four respondent groups differentiated between their ratings of risk of the procedures, demonstrating nuanced perceptions of risk across procedures (MHCH group range of means = [1.89–3.17], P value < 0.00; MICH group, range of means = [1.73–2.73], P value < 0.001; MICI group, range of means = [2–2.96], P value = 0.015). See Table 2.

Table 2.

Mothers’ perceptions of risk of research procedures, by personal and children’s health status

Mothers without mental health issues Mothers with at least one mental health issue Overall P valuea Cohen’s d
(n=54) (n=72)
How risky are the following research procedures for children? Mothers with only healthy children Mothers of children with at least one physical or mental health issueb Cohen’s d Mothers with only healthy children Mothers of children with at least one physical or mental health issueb Cohen’s d
(n=46) (n=8) (n=48) (n=24) (N=126)
Imaging of child’s head 3.17 (1.29) 2.63 (0.74) 0.77 2.73 (1.16) 2.96 (1.23) 0.19 2.93 (1.21) 0.29 0.446
Imaging of child’s abdomen 2.91 (1.19) 2.5 (0.53) 0.45 2.73 (1.12) 2.75 (1.03) 0.01 2.79 (1.1) 0.73 0.373
Taking a very small sample of child’s blood 2.76 (1.30) 1.75 (0.89) 0.90 2.42 (1.07) 2.58 (1.25) 0.14 2.53 (1.19) 0.13 0.843
Taking a very small sample of child’s blood for a genetic test 2.72 (1.39) 1.75 (0.89) 0.83 2.45 (1.06) 2.46 (1.10) 0.00 2.5 (1.20) 0.19 0.807
Performing a physical examination 2.64 (1.32) 1.63 (0.74) 0.95 2.42 (1.05) 2.25 (1.19) 0.15 2.42 (1.18) 0.12 0.857
Taking a very small sample of child’s saliva for a genetic test 2.33 (1.51) 1.5 (0.93) 0.66 1.98 (1.06) 2.17 (1.17) 0.17 2.11 (1.26) 0.28 0.659
Being interviewed by a research staff member 2.24 (1.19) 1.5 (0.76) 0.74 1.85 (0.97) 2 (1.25) 0.13 2(1.11) 0.19 0.668
Filling out questionnaires 1.89 (1.13) 1.25 (0.71) 0.68 1.73 (0.96) 2.04 (1.23) 0.29 1.82 (1.07) 0.28 0.736
P valuea <0.001 0.65 <0.001 0.015 <0.001
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All the questions are rated from 1–5

1 = Not at all risky, 2 = A little risky, 3 = Somewhat risky, 4 = Quite risky, 5 = Highly risky

a

P values from ANOVA/ Repeated Measure ANOVA, as appropriates

b
At least one of the following conditions:
  • Chronic or significant medical disorder
  • History of psychiatric disorder
  • Intellectual disabilities
  • Neurogenetic disease
  • Developmental delays

Association between the perceived level of risk of research procedures and demographic characteristics of the participants:

We found that the perceived level of risk of research procedures was related to a number of factors, namely trust in medical researchers and ethnicity and race (Table S1). First, perceived risk was negatively associated with mothers’ trust in medical researchers; mothers who endorsed high levels of trust in medical researchers perceived lower levels of risk on average than mothers with low to moderate trust in medical researchers (regression coefficient = −0.65, confidence interval = [−0.38, −0.92], P value < 0.001). Respondents who were Hispanic or Latino endorsed higher levels of risk for procedures on average than respondents who were not Hispanic or Latino (regression coefficient = 0.77, confidence interval = [0.24, 1.29], P value < 0.001). In comparison to participants who identified as members of minority groups, white participants endorsed a lower level of risk for procedures on average (regression coefficient = −0.44, confidence interval = [−0.027, −0.85], P value = 0.036).

Mothers’ perceptions of risk varied by mothers’ mental health status:

Among mothers who self-reported as having no mental health issues, having a child without a health issue implied a heightened sensitivity to risk, relative to mothers of children with at least one health issue (MHCH vs. MHCI = 0.49, 95% CI = [0.13, 0.84]). For mothers with mental health issues, by contrast, the health of children was not related to perceptions of risk of the research procedures (MICI vs. MICH = 0.13; 95% CI [−0.242, 0.5]; Figure 1).

Figure 1.

Figure 1.

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Overall comparisons of mothers’ perceptions of risk, by personal and children’s health status

Mothers’ willingness to enroll children in research:

Mothers expressed moderate to high levels of willingness to enroll their children in different procedures and responses differed across procedures (overall range of means: 2.76–4.02, P value < 0.001; Table 3; Figure 2). “Filling out questionnaires” had the highest rating of willingness (mean = 4.02, SD = 1.05) and was strongly endorsed. “Images of your child’s head being taken” had the lowest rating, but was still moderately endorsed (mean = 2.76, SD = 1.28).

Table 3.

Mothers’ self-reported willingness to enroll children in research, by personal and children’s health status

Mothers without mental health issues Mothers with at least one mental health issue Overall P valuea Cohen’s d
(n=54) (n=72)
How willing would you be to have your child participate in a research project that involves …? Mothers with only healthy children Mothers of children with at least one physical or mental health issueb Cohen’s d Mothers with only healthy children Mothers of children with at least one physical or mental health issueb Cohen’s d
(n=46) (n=8) (n=48) (n=24) (N=126)
Filling out questionnaires 3.96 (1.13) 4.25 (1.49) 0.22 3.98 (0.98) 4.17 (0.92) 0.20 4.02 (1.05) 0.78 0.276
Being interviewed by a research staff member 3.45 (1.34) 3.75 (1.39) 0.22 3.63 (1.10) 4.08 (0.93) 0.44 3.66 (1.19) 0.22 0.254
Taking a very small sample of child’s saliva for a genetic test 3.24 (1.38) 3.5 (1.41) 0.18 3.33 (1.12) 3.67 (1.27) 0.28 3.37 (1.26) 0.59 0.341
Performing a physical examination 2.78 (1.41) 3.5 (1.31) 0.53 3.27 (1.14) 3.63 (1.28) 0.29 3.18 (1.31) 0.05 0.654
Taking a very small sample of child’s blood for a genetic test 2.74 (1.42) 3.38 (1.41) 0.45 3.02 (1.11) 3.54 (1.10) 0.47 3.04 (1.27) 0.07 0.628
Taking a very small sample of child’s blood 2.83 (1.43) 3.38 (1.41) 0.38 2.92 (1.14) 3.5 (1.25) 0.48 3.02 (1.30) 0.16 0.421
Imaging of child’s head 2.74 (1.40) 2.63 (1.30) 0.08 2.69 (1.26) 3.13 (1.15) 0.36 2.79 (1.29) 0.55 0.388
Imaging of child’s abdomen 2.59 (1.38) 2.63 (1.30) 0.02 2.69 (1.19) 3.29 (1.20) 0.50 2.76 (1.28) 0.16 0.547
P valuea <0.001 0.45 <0.001 0.03 <0.001
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All the questions are rated from 1–5

1 = Not at all willing, 2 = A little willing, 3 = Somewhat willing, 4 = Quite willing, 5 = Highly willing

a

P values from ANOVA/ Repeated Measure ANOVA, as appropriates

b
At least one of the following conditions:
  • Chronic or significant medical disorder
  • History of psychiatric disorder
  • Intellectual disabilities
  • Neurogenetic disease
  • Developmental delays

Figure 2.

Figure 2.

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Overall comparisons of mothers’ willingness to enroll children in research, by personal and children’s health status

Interestingly, a significant difference was observed between groups with respect to moderate ratings of willingness to enroll children in research that involved “Performing a physical examination” (range of means: 2.78 – 3.63, P value = 0.05). The MICI group expressed the highest average rating of willingness (mean = 3.63, SD = 1.28) and the MHCI group expressed the lowest average rating of willingness for this item (mean = 2.78, SD = 1.41). For 7 of the 8 procedures, the MICI group expressed the highest willingness ratings, but still within a moderate to high range (range of means: 3.13–4.17 out of 5). The MHCI group provided the highest ratings for the “Filling out questionnaires” procedure (mean = 4.25, SD = 1.49).

Associations between willingness and mothers’ characteristics:

Participants’ willingness to enroll their children in pediatric research was found to be negatively related to a number of factors. Perceived risk of the research procedures was found to be negatively associated with willingness to enroll children in research procedures (regression coefficient = −0.51, confidence interval = [−0.42, −0.60], P value < 0.001). Mothers without mental health issues were less willing to have their children participate in research procedures compared to mothers with at least one mental health issue (regression coefficient = −0.90, confidence interval = [−0.06, −1.74], P value = 0.037). For mothers with mental health issues, having a child with at least one health issue implied a greater willingness to enroll their child in research, relative to other mothers who also had mental health issues but who had children with no health issues (MICI vs. MICH = 0.65; 95% CI [0.22, 1.075]). Among mothers without mental health issues, the health of their child was not statistically related to their expressed willingness to enroll their child in research (MhCh vs. MhCI = 0.56, 95% CI = [−0.25, 1.37]).

Discussion

Research involving children is needed in order to discover cures and methods to ameliorate symptoms of childhood diseases. For this reason, research involving children merits ethical engagement and support. In this study, we sought to understand influences that may shape the views of mothers regarding the participation of their children in research. We found that mothers with and without a personal history of mental illness and with and without ill children differed in their perspectives of risk across a broad range of research procedures. Mothers in all comparison groups demonstrated nuanced reasoning in their risk assessments.

Consistent with our first hypothesis, we found that mothers’ perceptions of risk were negatively associated with their willingness to enroll their children in research. That is to say, mothers were less inclined to enroll their children in research with procedures posing higher risk, and this observation held even after controlling for the health status of children and mothers, which is consistent with previous findings (Bentley & Thacker, 2004; Dunn et al., 2009; Roberts et al., 2002). We also found that perceptions of risk were related to the level of trust in researchers endorsed by mothers.

Our finding about risk and ethnicity may not be robust, given the sample size, and requires further replication. A limitation of our finding is that we were not powered to test any hypotheses regarding race/ethnicity and willingness to participate in research. Future work in this area should ensure that ample efforts are taken to recruit representative samples. Many have investigated factors related to the experiences of minority groups in clinical research (Arevalo et al., 2016; Barsdorf & Wassenaar, 2005; Ford et al., 2008; Heredia et al., 2017) and some have revealed potential barriers to participation (Ceballos et al., 2014; Scharf et al., 2010). Research engagement among minority groups remains a challenge that is deserving of emphasis, as the participation of individuals who identify as members of minority groups is direly needed for the advancement of clinical innovation.

We constructed this analysis in order to answer the questions of whether a mother’s mental health status or the health status of a mother’s child or children might influence her perceptions of risk and her willingness to enroll her child in research, and whether there might be some difference in risk perceptions and willingness based on an interaction between the mental health of the mother and the health of the child. We found that mothers of children with health issues who self-reported at least one mental health issue were more willing to enroll their children in research, compared to those with children without health issues. In terms of risk, risk ratings across groups were not different, with the exception of the ratings of mothers without mental health issues with children without health issues, who had the highest ratings of risk. These findings offer an additional perspective to the literature because past work (e.g., Caldwell et al., 2003) has suggested that parents of ill children may be very willing to take on risk in order for their children to have the opportunity to participate in research. It also has been shown in the literature that parents of children with life-threatening conditions were “less bothered by possible side effects” or viewed the risks of research less negatively (Fisher, McKevitt, & Boaz, 2011; Wiener et al., 2015). Indeed, parents’ desperation in seeking treatment for their seriously ill children has in the past been identified in the ethics literature as a vulnerability that may lead to exploitation in the context of human research (Solomon 2013).

Ethical considerations in pediatric research are unique and have received scholarly attention (Baker et al., 2013; Dekking, van der Graaf, Kars, et al., 2015a; Dekking, van der Graaf, de Vries, et al., 2015b; Wiener et al., 2015). Government regulations do not specify the assent process in pediatric research. Pediatric research and care are more closely related in some areas than adult research and care. Ethical considerations in pediatric research include role conflicts in the work of pediatric oncologists and the question of what constitutes research versus treatment (Dekking, van der Graaf, Kars, et al., 2015). In some areas, the uniqueness of the research is the complexity of the information that must be shared and the requirement for parental consent and patient assent where applicable (Baker et al., 2013). Other ethical considerations relate to improving the delicate process of consent, for example, by obtaining more direct input from adolescents when parental and adolescent views diverge and allowing more time to make a decision or using different methods to deliver information (Wiener et al., 2015).

It is still clear that risk perceptions play a key role in enrollment decision-making and thus it is possible that familiarity with health-related procedures, such as having blood drawn, being interviewed, or having images taken may play a role in influencing mothers’ decisions to enroll their children in research (Gammelgaard, Knudsen, Bisgaard, 2006). A lack of exposure to health related procedures, and a lack of health education, could thus potentially be a barrier to research participation. As demonstrated in Hoberman et al. 2013, parents of ill children who were more altruistic were also more likely to consent in research. The data from our small study helps to raise the interesting question of how maternal health status may shape the participation of children in clinical research, a topic that warrants further study.

One limitation of this study is that the size of one of the comparison groups (i.e., mothers without mental health issues with at least one child with at least one health issue) was fairly small. Another limitation worth highlighting is the relative lack of diversity in the MTurk sample, as well as the hypothetical nature of the survey. Previous investigations demonstrated that MTurk samples are not representative of the US population (Paolacci et al., 2010; Shapiro et al., 2013). Respondents were asked to respond to attitudinal items for hypothetical scenarios rather than make actual decisions regarding their own lives. It is possible that as research participants experience life situations or changes in health, their attitudes concerning research will change over time (Tromp & van de Vathort, 2019; Tsevat et al., 2018; Ward, 2010). Finally, we did not include questions regarding prior exposure to or engagement in research, prior exposure to the various procedures queried, socioeconomic status, the physical health condition of mothers, or questions regarding children’s health other than chronic medical conditions. Further research will be needed to replicate and confirm our findings. This study nonetheless adds to the pediatric research literature by offering an inferential point of view regarding parental willingness to enroll children in research as a function of risk perception, and confirming that parents of children with health issues are indeed willing to tolerate risks.

Best Practices

A better understanding of the factors that lead parents to enroll their children in clinical research could play a vital role in developing strategies to facilitate and clarify the ongoing dialogue between providers, investigators, and parents in the research enrollment process. Investigators and institutional review boards should ensure that the perspectives of parents are given proper attention and understood to the same degree that parents attempt to understand the nature and purpose of the research in which their children might participate. In efforts to increase understanding of risks that are associated with research, investigators and institutional review boards could also strive to ensure that prospective research participants are provided with practical information that helps them assess potential risks and benefits.

Research Agenda

Future work should seek to clarify degrees of willingness and degrees of certainty of parents in enrolling their children in research of different kinds. We also suggest that the paucity of data in the literature regarding informed refusal, i.e., unwillingness by fully informed, decisionally capable individuals to enter research protocols or to provide surrogate consent, represents a tremendous gap in the adult and pediatric research literature. This gap has implications for the scientific interpretation of human studies and has ethical importance as well. The results of previous studies suggested that individuals who provided consent, compared with those who did not, exhibited less uncertainty in their decision-making, were more trusting in medical researchers, had greater understanding of the research, and believed that the environment in which consent was sought was less pressured. The mental health status of the decision maker has been reported in a few studies to significantly influence the decision-making process (Leykin, Roberts, & DeRubeis 2010; Miu, Heilman, & Houser 2008) and utility judgment is itself influenced by factors such as strong emotions or incorrect prediction of future preferences (Loewenstein, O’Donoghue, & Rabin 2003). Hoberman et al. (2013) reported that parents who declined consent had a relatively higher socioeconomic status, had more anxiety about their decision, and found it harder to make their decision.

Educational Implications

In order to improve the participation of children in medical research, it is essential to educate investigators, the research team, and IRB members. Our findings suggest that mothers’ perceptions of risk of research, mothers’ trust in medical researchers, and the health status of both mothers and children may be factors that play a role in decision making. Approaches for recruitment and informed consent will be most effective if investigators and practitioners take these issues into consideration. For investigators to design ethical research protocols and for IRB members to appropriately review protocols, both need to understand the particular vulnerabilities of the populations with which they work and how these vulnerabilities may impede the ability to make autonomous decisions related to children’s research participation. Considering the importance of maternal trust in researchers, it may be helpful for investigators to spend more time speaking with parents, specifically with mothers, concerning the details of the research and the benefits and the risks of the study. This may offer mothers more reassurance and produce higher willingness to enroll their child in a study.

Advancing the understanding of pediatric health relies on educating the general public on the importance of the conduct of pediatric research, which includes education about the various types of research and the risks involved, as well as safeguards that exist to protect children in the research situation. Educational interventions prior to the recruitment of research volunteers, could enable ethically robust decision making about research participation. Such efforts, streamlined with community engagement efforts that seek to foster greater transparency and trust in research are needed prior to conducting research, not solely during the informed consent process.

Supplementary Material

1

Funding Source:

This study was funded by the Department of Psychiatry and Behavioral Sciences at Stanford University School of Medicine and by grant 1RO1MH11485601 from the National Institute of Mental Health. This content is solely the responsibility of the authors and does not represent official views of the National Institute of Mental Health.

Footnotes

Disclosure: Dr. Roberts is Editor-in-Chief of the journal Academic Medicine.

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