“Telling” and assent: Parents’ attitudes towards children's participation in a birth cohort study

1.1 Parent's proxy consent and children's informed assent in birth cohort studies

Several countries have implemented longitudinal birth cohort studies; national projects in UK have been going on since the 1940s (ie, the 1946, 1958 and 1970 Birth Cohort, and 2000 Millennium Cohort Study), and recently, Denmark, Norway and Japan have implemented their own large-scale studies, each with around 100 000 participants. These studies usually follow children from the foetal or neonatal stage until they are teenagers or adults and study the possible long-term health consequences of maternal lifestyle, genetic factors and early exposure to environmental contaminants and other substances.1, 2

Key ethical issues in birth cohort studies involve the following: (i) parents’ initial proxy consent, which is related to recruitment and enrolment, and (ii) investigators’ subsequent obtaining of informed assent—defined as “affirmative agreement to participate in research”—from those children capable of providing it.3 While pregnant mothers may give consent by proxy, once children become older it is ethically important to provide them with the opportunity to express their own opinions and involve them in the decision-making process, depending on their level of comprehension.4-6 Moreover, after children mature, investigators must again obtain informed consent from adolescents.

1.2 Parents’ influence on children's decision making in birth cohort studies

There have been studies regarding parent's proxy consent, most of which focus on pregnant women's incentives/disincentives to give consent, and attitudes towards children's future participation in birth cohort studies.7-10 Garg et al10 show that some mothers demonstrate discomfort when making proxy consent on behalf of their children and voice concern over possible psychological harm, anticipating their children's feelings as they mature. Regarding children's assent, Goodenough et al11 interview children about their perceptions of participating in a specific birth cohort study (the Avon Longitudinal Study of Parents and Children: ALSPAC, UK) and find that some children as well as parents misinterpret their involvement or role in the study.

Although these studies clarified pregnant mothers’ attitudes and children's perceptions towards the studies, fewer studies focus on “informed” stages of assent, or the process by which children are given information about the study in which they participate. Helgesson12 raises a question of “how to inform children” and suggests that information should be shared in advance of their capacity for full decision making in ways that extend beyond verbal descriptions: drawings, photographs or video recordings.

Besides media or communication tools, who shares information with children is also important. When the participants are adults, informed consent must be sought by an individual who has no prior relationship with the study participants;13 such roles are usually expected to be performed by clinical research coordinators, research nurses, counsellors or other health-care professionals.14-16

When the participants are children, as in birth cohort studies, parents—who often gave consent by proxy before or shortly after the child is born—may be involved in the explanation process before investigators or health-care professionals seek informed assent. Since children are especially susceptible to parents’ influence, children's decisions in this case cannot be understood as autonomous, but rather as a transformation of the parent-child interaction.17

We can assume that at some point, children will be told that they were recruited into a longitudinal cohort study. How children learn this information may influence their attitudes towards the study and their future decisions concerning whether to continue cooperating. However, despite the importance of how children are informed, little work has been done on parents’ role and influence on their children's decision making.

1.3 “Telling” as the premise for informed assent

Based on the above information, we propose a framework for parents to convey information to their children called “telling,” which was originally used for adopted and donor-conceived children. These children have the “right to know” their origin, and parents’ ability to “tell” their family story is a way for children to learn the information in an age-appropriate manner. Parents are increasingly recommended to openly talk with their children,18-20 and the process of “telling the story” about a child's birth is an ongoing one that starts when children are very young and is repeated daily as they grow older.21

The same method of “telling” can be applied to birth cohort studies: this is another situation in which children have to be told something that was already determined before their birth. As in the case of adoption, parents should be the initial storyteller, prior to interactions or conversations with physicians/investigators.

Thus, the purpose of this study was to confirm parents’ attitudes towards “telling” their children about study participation in a birth cohort study. This article presents our qualitative analysis of parents whose children participated in the “Japan Environment and Children's Study” (JECS), a large-scale, nationwide birth cohort study which we will later describe in detail.

2.1 Case study

The Japanese Ministry of the Environment funded a large-scale, nationwide birth cohort study in 2011. Called the “Japan Environment and Children's Study” (JECS), it follows 100 000 children from pre-birth to age 13.22 Fifteen Regional Centers across Japan were responsible for recruitment and participant follow-up, and from January 2011 to March 2014, they recruited women in the stages of early pregnancy at obstetric facilities and/or local government offices that issued mother-child health handbooks.23

The 2015 “Ethical Guidelines for Medical and Health Research Involving Human Subjects” enforced by the Japanese government requires investigators to endeavour to obtain informed assent from minors under 16 years old who are considered capable of expressing their intentions.24 The JECS is the only nationwide birth cohort study conducted since the rule were put in place. Parents were informed when they gave initial consent that, at some point in the research process, children would be asked to confirm their intentions to continue as research participants.

2.2 Study area and design

We conducted our research with “JECS Yamanashi,” located at the Koshin-Yamanashi Regional Center (University of Yamanashi) in the Yamanashi prefecture, which itself is located near the centre of Honshu (the main island of Japan), 2 hours’ drive from Tokyo. The participants of JECS Yamanashi were recruited from 5 cities: Kofu, Chuo, Koshu, Yamanashi and Fujiyoshida. Study participants included 4474 children, 4630 mothers and 3040 fathers.25 At the time, the entire population of the Yamanashi prefecture was 857 690, with a 2011 total number of live births of 6412;26 participants thus included over 50% of newborns in the 5 cities during the 3-year recruitment period. Besides the investigators, there were 7 research coordinators involved in the study who were in charge of recruitment, collecting questionnaires, conducting health check-ups, monitoring air quality and managing participant events.

We adopted a mix of quantitative and qualitative methods in this study; we conducted a small, face-to-face survey and followed this with in-depth interviews. The survey mainly consisted of closed-ended questions, which allowed us to comprehensively collect answers. Using the data collected from our survey, we next conducted in-depth interviews, which consisted of open-ended questions, in order to better understand the reasons for respondents’ answers. To find participants for the interviews, we handed invitation letters to respondents and later followed up with both them and some of their partners. We adopted a “maximum variation sampling” strategy27, 28 in order to include a wide range of extremes in demographics (including occupation, number of children, children's ages and children's genders), motivations for participating and degree of interest in the JECS.

Our research proposal was approved in advance by the research ethics committees of the University of Tokyo, the University of Yamanashi and the National Institute for Environmental Studies (NIES).

2.3 Data collection

Three interviewers (IR, ES and KM) conducted the face-to-face survey at the event booth for JECS Yamanashi participants during community events on 22 and 23 August 2015. Each survey took about 10 minutes, and we asked 17 closed-ended questions and 2 open-ended questions on topics such as parents’ motivations for participating in the JECS and their attitudes towards telling their children. We then recorded the answers on questionnaire forms. We surveyed mothers and fathers separately, although we numbered the questionnaires to reciprocally compare couples’ answers in our analysis.

One of 2 interviewers (IR and/or ES) carried out each in-depth interview between October and November 2015. Interviews took place either in a University of Yamanashi meeting room or in participants’ homes. When both parents of a child were interviewed, the mother and father were interviewed individually. We obtained written informed consent from each interviewee. Each interview took about 1.5 hours, and they were all recorded. We asked 20 open-ended questions on a variety of topics, including parents’ motivations for participating in the JECS; who held the major responsibility for childcare; whether they had talked about the JECS with their partner, family or friends; and their attitude towards providing personal information. We also provided information about the JECS study schedules for the next 13 years and about the new informed assent rule, which enabled parents to imagine what and how they would tell their child about their study participation at each development stage, as well as how their child would react.

2.4 Data analysis

Survey data were simply aggregated, and transcripts of the in-depth interviews were coded using Qualitative Data Analysis Software MAXQDA10 (VERBI GmbH). IR generated most of the codes and categories and held several meetings with ES and KM to verify the coding. The final data analysis examined the following categories: parents’ attitudes towards the necessity of disclosing study participation to their child, parents’ willingness to disclose, who and with whom parents planned to tell, what and how to tell their children and various background information, including why parents participated in the research and what kind of benefits they thought this would give them and their children. Parents’ communication style emerged though the analysis, and this article categorizes these styles into “telling” categories and elaborates on each.

3.1 Sample characteristics

In total, 44 mothers and 23 fathers (including 22 couples), whose children were aged approximately 9 months to 4 years, participated in our survey (Table 1). About half of the fathers were also JECS participants. Eleven mothers and 3 fathers participated in our in-depth interviews (Table 2). All 3 fathers we interviewed were also JECS participants and the partners of women who were also interviewed. Their children were aged from 10 months to 3 years, 3 months.

3.2 Reasons for participating in the study

Parents described various motivations for participating in the study. Common reasons included free health check-ups, an interest in the JECS study, a desire to do something good for their child and hope that the study would solve an environmental or health problem. It is noteworthy that several informants mentioned that they participated in the JECS because “I just happened to be recruited in the hospital” or “I was talked into it by a midwife.”

Face-to-face communication before a child's birth seemed to build friendly relations between mothers and research coordinators, and such interaction sometimes continued even after the child was born. Parents were pleased that the research coordinators still remembered them when they met again at events, which eventually led investigators and the JECS study to gain more credibility in parents’ eyes.

3.3 Personal benefits of participation for parents and children

Our informants discovered that there were various advantages to participating in the study, both for themselves and for their children: parents received blood tests and could access some of their results (ie, allergy testing), they considered their children's growth and reflected on everyday habits through answering questionnaires, families were sent newsletters with interesting statistical data or useful information about health and the environment, and there was some monetary compensation or small gifts for their participation. Many family members, especially mothers, had many favourable comments regarding the events held by JECS Yamanashi. For mothers, these events were not only fun, but also a way to build a community among the “JECS moms.” Some informants even said they were reluctant to talk about the JECS with mothers who did not participate, saying that “those who did not have rights to participate” would “envy me,” since enrolment was limited to a certain time period and geographical area.

Some parents hoped that becoming a member of a national study would be a good opportunity for their children to foster altruism. They seemed to find significance in the educational value of study participation.

3.4 Parents’ attitudes towards “telling”

In our survey (N = 44 for mothers, N = 23 for fathers, including 22 couples), 72.7% of mothers and 60.8% of fathers believed it was necessary to tell children of their participation in the research study. Although no parents had told their children at the time of the interviews, 81.8% of mothers and 43.4% of fathers said they would tell their children at some point in the future. 38.6% of mothers and 43.5% of fathers preferred to tell the children with their partners or family, and 25.0% of mothers and 8.7% of fathers preferred to tell them on their own. In contrast, only 15.9% of mothers and 13.0% of fathers preferred to have professionals get involved in the early stages of telling. Therefore, it seems that they valued communication within the family. In general, mothers had positive and active attitudes towards telling their children, while fathers showed more unmotivated attitudes or expected their partner (both as children's mothers and as the JECS participants) to play major roles in telling. When comparing the answers of the 22 couples, 8 couples stated a different preference for whether to tell their children and who would be responsible for doing so.

3.5 What and how to tell? Three communication styles

One of the major questions our study tried to answer is how parents planned on telling their children about participation in the study. Although none of the parents had told children yet, since they were too young at the time, the interview process led them to imagine their grown-up children and anticipate the long-term future. By analysing the recorded narratives, we found that communication styles seemed to vary depending on parents’ perception of the JECS study's risks and benefits. We categorized their answers into 3 communication styles. Twelve parents, who predicted that the study would benefit their children, preferred “directive telling,” which we further divided into (i) “empowered telling”, and (ii) “persuasive telling.” Only 2 parents, who weighed the possible benefits with the potential risk, preferred (iii) “non-directive telling.”

4.1 Study limitations and future directions for research

There were some limitations in our sampling, including the method and the number of informants. Quantitative research with a large number of participants from other Regional Centers is needed to verify our pilot qualitative research; this will also allow us to consider parents’ attributes, the differences between mothers and fathers, and children's gender. We also have to consider the social and cultural context, since parent-child relationships may be different in Japan than in other countries. Similarities and differences in parental views or education policies should be explored though an international comparative study of birth cohort studies. The next question would be whether and how investigators should try to speak with children independently from parents when the children grow up.

The most crucial point is that since the children were so young when we conducted our research, we were unable to discuss any actual “telling” experiences; future research that explores how parents actually tell their children is vital for testing our temporary hypotheses. One of the important ethical and legal considerations in the JECS protocol is its policy informed assent; the following agenda may be used to conduct additional research regarding participants’ attitudes and “telling” strategy over the long-term. Children's attitudes must be respected and investigated;11, 32 it is important to investigate how children act on information and the changing dynamics between parents and children as they mature.