Abstract
While several studies have explored parents' perceptions of their children's participation in research, very few studies have described the children's own perceptions of their participation in research. The aim of this study was to describe children's perceptions of their participation in a comprehensive longitudinal clinical study. Semi-structured qualitative interviews were conducted with 17 children aged seven participating in the Copenhagen Prospective Study on Asthma in Childhood. The interviews were audiotaped, transcribed and analysed using the template analysis method. The children rated their experiences with venepunctures on a Wong-Baker faces scale. The regular visits to the hospital and comprehensive clinical tests did not seem to have affected the children negatively. In particular, the children would happily engage themselves in medical tests mimicking play (moving, walking, running or playing with a computer). A majority of the children, however, disapproved of the venepunctures and some even refused to have it done. The results of this study indicate that participation in even comprehensive paediatric research can be a positive experience to the participants, with the most popular tests being those that required active participation from the children.
Introduction
‘A new era for research in children is about to begin, and should be welcomed.’ These words from a Lancet editorial 1 anticipated new legislation from the European Commission requiring testing of all new medicines in children. While this new legislation is arguably a step forward in terms of developing evidence-based therapies for children, the ethical dilemmas of paediatric research need to be discussed as an integral part of the implementation of this new legislation. 2 To inform such discussions, it is relevant to investigate how parents and children perceive their participation in research. While several studies have explored parents' perceptions of their children's participation in research, 3–7 very few studies have described the children's own perceptions of their participation in research. 8,9 Children's perceptions may, however, be quite different from what adults think they are. Studies have shown that the results of parent, nurse and child ratings of children's pain during venepuncture do vary although they are correlated, 10 and that parents tend to underestimate their children's concerns about research participation. 11 Such findings highlight the need for investigating the perceptions of children, and to include their perspective in discussions of ethical issues in paediatric research. Children's perceptions of research, however, may vary according to various factors such as age, state of health, and whether or not they are regular hospital attendees. Consequently, children cannot be considered a homogenous group, and discussions of ethics in paediatric research need to carefully consider the specific group of children participating in the research.
In this study, we report the perceptions of children participating in a comprehensive longitudinal, clinical birth cohort study on asthma. We chose to study children participating in the Copenhagen Prospective Study on Asthma in Childhood (COPSAC), because this is one of the most comprehensive longitudinal observational clinical studies in paediatrics. 12–14 The aim of this study was to describe what children think about participating in this particular study on asthma and how they perceive the specific procedures and tests.
Methods
Design
Children still participating in COPSAC (see Box 1) by the age of seven were eligible for this study. Twenty-four mothers of children who had completed the scheduled visit at the age of seven were invited consecutively by letter for this study. Twenty-one of them agreed to let their children participate in this study. Qualitative interviews were conducted with 21 children participating in the COPSAC study. The children of the remaining three did not participate: two did not reply and one child did not wish to participate according to her mother.
COPSAC is a single-centre longitudinal clinical birth cohort study of children of asthmatic mothers. Objective assessments begin at birth, with scheduled visits every six months and when acute symptoms manifest. Clinical outcomes comprise asthma, eczema, allergic rhinitis, allergy, lung function, bronchial responsiveness, and identification of airway bacteria and virus as respiratory episodes. A total of 411 infants of asthmatic mothers were enrolled at the age of one month. At the age of five years, 77% of the infants were still participating in the study. The COPSAC study has previously been described in detail
12–14
and is only reviewed here to illustrate the comprehensive tests required by the participating children:
Daily symptom recordings in diaries; Allergy skin test at age six months and at 1.5, four and six years; Urine samples every other year; Exhaled nitric oxide (FeNO) is measured at one month of age by an offline technique and from age five years by an online technique; Infant lung function is measured by the raised volume rapid thoraco-abdominal compression (RVRTC) technique during sedation at one month of age; Lung function measurement (specific airway resistance; sRaw) by whole body plethysmography is conducted with the child sitting in a closed box and breathing in a face mask for approximately three minutes. Measurements were attempted every six months from age two years; Lung function measurement by forced spirometry with the child being coached to perform maximal forced expiratory manoeuvres every six months from age five years. Infant bronchial responsiveness to methacholine successfully determined by the Raised Volume Rapid Thoraco-abdominal Compression (RVRTC) technique during sedation at one month of age; Test for bronchial responsiveness by cold, dry-air hyperventilation for four minutes, where the child is coached to hyperventilate at maximal rate by a computer animation biofeedback showing the ventilation rate on the screen and defining the target rate with measurements of sRaw in whole body plethysmography before and after hyperventilation at four and six years; Bronchial responsiveness to methacholine challenge measured by spirometry at age six years. Test for bronchial responsiveness by exercise challenge (submaximal exercise by running on a treadmill for 6 minutes) by age seven years; Reversibility of lung function measured by repeated lung function after inhalation of bronchodilator at two, three and five years; Physical activity assessed by accelerometer computer worn on the ankle for four weeks by the child by age five years; Test for nasal patency by ultrasonic measurements; glass tube directed at the nostrils before and after alpha-adrenergic vasoconstrictor at age five years; Nasal mucosal smear by curettage of the lower concha by age five years; Bone density measured by oscillometry by age one, two and three years; Body composition measured by Dexa-scan by age six years; Venepuncture by age six months, and at 1.5, four, six and seven years.The COPSAC study
The interview took place on average 45 days (median 37, range 1–131) after the scheduled visit at the COPSAC clinic. The children were interviewed at home by the first author (AG), a bioethicist who was not otherwise involved in the COPSAC study. The children decided whether they wanted a parent present (14 were interviewed on their own, and 7 were interviewed with a parent present). Pilot interviews were conducted with the first three children using a semi-structured interview guide. 15 Following an analysis of those three interviews, we decided to include a series of photos from the COPSAC clinic (photos of the equipment for measuring lung function, venepunctures and exercise challenge test) and a Wong-Baker faces scale to rate the children's experiences with venepunctures. 16,17 In the following 18 interviews, the children were presented with the photos and questioned about their experiences with the various tests of the study as well as their overall experiences with the visits to the COPSAC clinic. The children were also asked to rate their latest experience with venepuncture on the faces scale. The interviews lasted between 15 and 35 minutes and were tape-recorded, transcribed and analysed using a textual computer package (ATLAS.ti, Scientific Software Development GmbH, Berlin, Germany), and the method of template analysis as described by Crabtree and Miller 18 and King. 19 One tape was defective and omitted from subsequent analyses. The study was approved by the regional research ethics committee (KF 11 308242).
Participants
Twenty-one children participated in this study. Six of the children exhibited no symptoms of asthma or atopic dermatitis, 10 were diagnosed with asthma or atopic dermatitis and five were diagnosed with both asthma and atopic dermatitis. Six girls and 15 boys participated. Three of the children participated in the pilot interviews and one interview was omitted from the final analysis, leaving a total of 17 children in the final analysis.
Results
The qualitative interviews left us with the impression that the children generally felt comfortable about their participation in the COPSAC study; some even said that they enjoyed the visits to the COPSAC clinic and others that they looked forward to the visits. Many of the children spontaneously mentioned that the exercise challenge or the various lung function tests were great fun. Although some of the children found the face mask somewhat annoying to wear, many talked enthusiastically about their achievements on the treadmill in terms of running fast or running a long distance. What was it like to run on the treadmill? It was fun, it was the most fun thing of the whole COPSAC. Is that right? I thought it would be tough and hard? Yes, it was tough and hard too (said without concern) – I think it's fun and hard. The good thing about it all is that it's hard. Why? For me it's fun when it's hard. Otherwise it's way too easy. Do you like something that's a bit of a challenge? Yes, and I ran for the whole six minutes.
(i6, boy diagnosed with asthma)
Many of the children said they enjoyed the lung function tests due to the computer-animated games (bowling or girl flying in a balloon basket). It seemed that the most popular tests were those that required active participation from the children. Do you like to visit the COPSAC clinic? Yes. Why? Because it's fun. It's fun to run around and play with that and all sorts of things. You mean with the princess castle and the toys? Nooo! It's fun to play with those machines (computers), where you can bowl and all that stuff with the girl (in balloon basket) and draw lines (coloured circles on the screen) and all that. After I had been running, I had to run in there and blow, and then run back, and blow again, and then have some medicine, and then blow, and then that was it! That was a whole lot, but was it fun? Yes!
(i20, healthy boy)
None of the children seemed to be afraid of, or intimidated by, the various lung function tests although some did find the repeated breathing exercises a bit tedious. When asked whether they would allow the door of the whole body plethysmography box to be closed, some of them simply replied ‘yes, why not?’ Others explained that they did not mind because the box had windows and you could hear the nurse speaking from outside through a loudspeaker inside the box.
The only contentious procedure was the venepuncture. A majority of 15 children disliked venepunctures due to either pain, anxiety or a feeling of uneasiness over the visible blood. Eight of the children said they coped with the venepunctures by using various distraction techniques to avoid looking at the needle, i.e. they would study various objects or posters in the room or play with toys. Can you see what this is? (shows girl a picture of the blood test equipment) Yes, I remember that, and I don't like it much! Why not? It hurts a bit at the end, when it really starts to press your arm. It's because you get a feeling like you have a lump in there, and it just … it doesn't hurt at first, but it hurts a lot at the end, because it feels like you've got a great big lump in your arm. And then there's the blood that comes out, that's really disgusting to look at. What do you do, then? Well, I don't look at it. I look at the door. There's a picture of a gnome there.
(i8, girl diagnosed with atopic dermatitis)
The last time they did it, I found some stickers in the meantime, so I didn't look. So you sat and looked at the stickers? Mm, and I found a cow. A cow? A cow with three little balls for the eyes and tail. And what could you do with that? You had to try and get all the balls into those … I see, so you were thinking about that? Yes. And then it didn't hurt as much? No.
(i18, healthy girl)
A minority of five children, however, did not find the venepunctures painful. Can you see what this is? (shows the girl a picture of the blood test equipment) Yes (laughs a bit), that's for when I get a needle in my arm. I don't mind that, I'm good at it. I never felt it was annoying or anything. You weren't afraid of the needle? No, I'm not afraid of the needle. What do you do, when you have to have the needle? I don't do anything, I just sit and wait. I sit as quiet as a mouse, because if I panic, they might stick the needle in wrong and that wouldn't be good, would it? Can you feel it when they give you the injection? Yes, a bit. But that's all. Do you look, or do you look away? I look at it! What about when the blood comes out? How do you feel about that? I don't mind, I'm not afraid of blood!
(i12, girl diagnosed with asthma)
Four of the children said that they had refused the venepuncture at their latest visit to the clinic (this finding was corroborated by their parents after the interview). They said they were afraid of the needle and that they expected it to be painful. Can you see what this is? (shows a picture of the blood test equipment) It's a needle, I cried! You cried? It's not nice, not at all! Does it hurt? Yes. They tried to pull my arm over to the needle, but they couldn't, I'm way too strong! You just held your arm close to your body, because you weren't going to have that? I did like this (holds arm close to body). And what did they do then? They tried to pull my arm, all the time, two strong women, the doctors. And what did you say? I didn't say anything, I just cried! Did they give up in the end? Yes, they gave up. I'm not going to get the needle. Now they know I'm stronger than them, they won't even try! Is it because the needle hurts? Yes, it hurts a lot, because there's a sort of long tube as well. Are you afraid to see the blood? No, it's because this thing (points at the tube) has to be full up, and I hate that, because the needle really hurts when they stick it in. It sucks, you know, like a catfish. That sucks too, it sticks onto the glass. And that's not nice? No.
(i21, boy diagnosed with atopic dermatitis)
Fourteen children (those who did not refuse the venepuncture) rated their experience with the venepuncture at the latest visit on the faces scale (ratings from 0 to 5, with 0 for not unpleasant and 1–5 for increasing unpleasantness) resulting in a mean value of 1.3 (range 0–4). Thirteen children (including 3 of the dissenters) had memories of earlier venepunctures at the clinic, and they all said that it was worse when they were younger. Those who could recall earlier experiences of venepunctures were asked to rate them, resulting in a mean value of 4.2 (range 0–5).
Discussion
This study describes young children's experiences of participating in a very comprehensive clinical study comprising approximately 15 visits of typically 2–3 hours duration over a period of seven years since birth. This interview study with seven-year-old children looking back leaves the overall impression that participation in research can be a positive experience. They talked with enthusiasm about the tests that were fun and their achievements on the treadmill or with the biofeedback computer programs. Our study thus supports the suggestion that creating a child-friendly, positive or even ‘fun’ environment at the investigation unit can make the experience a positive one, rather than the one perceived to be painful and traumatic. 20 The children in our study liked to be challenged to a reasonable extent by the various tests requiring their active participation. Many of the research procedures seemed to be interesting almost per se but the most popular tests were those that required active participation from the children (running the treadmill and the lung function tests that included the various biofeedback computer programs). It seems that children happily engage themselves in medical tests that mimic what they do when they play: moving, walking, running or playing with a computer. This point is relevant to discussions on how children may be motivated to participate in research.
Our results accord with a previous study of older school children reporting that 89% of the children participating in a non-therapeutic study of growth hormones said it had been a learning experience, and 97% of the children also said they would consider volunteering again if they were asked to participate in a future study. 21 Clinical studies, then, may provide children with a positive introduction to the health-care environment through education and experience. 20 The mean value of 1.3 on a scale from 0 (best) to 5 (worst) describing the children's rating of the venepuncture at their latest visit is comparable to the value of 1.5 found in a study using the same Wong-Baker faces scale on 75 children aged between five and 12 years who underwent venepuncture. 17 We found a striking difference between the children's ratings of their most recent venepuncture (mean = 1.3) compared with the ratings of their memories of earlier venepunctures (mean = 4.2). Other studies have shown that younger children report more pain in connection with venepunctures. 10,22–24 However, there may be some lack of reliability in the children's ability to recall earlier venepunctures due to the amount of time that had elapsed between these venepunctures and the interviews.
While the mean rating of only 1.3 indicates that this procedure is not so bad after all, the vast majority of children in our study disapproved of venepunctures and four of the children blankly refused to give blood at their most recent visit to the COPSAC clinic. In a previous report, 8 46% of the children who decided not to participate in the study did so because of worries about venepuncture. However, several studies have shown how distraction techniques, 25 preparation of the children, 26 EMLA cream 27 or the presence of a parent 28 may reduce the distress related to venepunctures. Future studies with children intending to include venepunctures should consult this literature, and describe to the Institutional Review Board/Ethics Committee how they intend to reduce the distress of venepunctures.
Considering the rather comprehensive test program of the COPSAC study, it is remarkable that venepuncture was the only contentious procedure. The most likely reason for this appears to be that many of the tests in the COPSAC study mimic play and games for children at that particular age. This is good news for researchers who are willing to make the effort to turn the various tests and procedures of future studies into a pleasant and interesting experience for the participants.
In this study, we have attempted to record children's authentic voices about their participation in research. Arguably, there are a range of potential sources of bias in data obtained from qualitative interviews with children. 29–32 In this study, the presence or absence of a parent may have influenced the way the child communicated. However, we sought to increase the validity of the interview data by focusing on creating a pleasant and relaxed atmosphere and a good rapport with the child prior to the interview. We let the child choose the location of the interview, whether or not to have a parent present, and we had conversations about their pets, toys or other issues that naturally came up, sometimes also during the interviews. The photographs of the clinic's equipment helped to engage the children in the interview process. To minimize suggestibility, we mainly used open-ended questions.
The relatively large differences in the time that elapsed between the clinic visits and the interviews is another potential bias of this study, although we do not know how this variation may have influenced the data. Arguably, a shorter time interval would have resulted in more reliable data.
The generalizability of this study may be limited due to the small sample size. The specific sample, however, represents both sexes and healthy children as well as children suffering from asthma and atopic dermatitis. While many of the children in this study are healthy, they have attended the COPSAC clinic regularly since birth. It is relevant to question whether the findings in this study would hold true for children who are less familiar with the health-care setting. To qualify the ethical debate on children as research subjects, studies of the perceptions of different groups of children are needed.
The majority of parents who participated in our previous study of parents' experiences with the COPSAC study said that they would withdraw from the study if their children responded negatively to the visits to the clinic. 3 Consequently, it is not a surprising finding that the children who are still participating in the COPSAC study do not seem to be troubled by their experiences. The fact that at the age of five years a majority of 77% were still participating in the COPSAC study also supports our impression that the children generally feel comfortable about their participation.
Conclusion
The main implication of this study for future paediatric research is that participation in even comprehensive paediatric research can be a positive experience for the participants, with the most popular tests being those that require active participation from the children. When designing the various tests and procedures of a study involving children, researchers should make an effort to mimic the play and games of the relevant age group. The creation of a friendly and stimulating environment at the research unit is likely to be beneficial. Finally, parents and researchers should pay close attention to the children's reactions during the tests, since procedures that they assume to be painful or unpleasant may not necessarily be so to the children and vice versa.
Footnotes
Acknowledgements
This study was supported by The Danish Paediatric Asthma Centre and Aase & Ejnar Danielsens Fund; the funders had no involvement in this work. We thank the children who participated in this study as well as their families; Lene Toft Jensen and Malene Starup Stage for helping to access the COPSAC database; Dorthe Goldschmidt for suggesting that we do this study; Eva Gulløv for advice on interviewing children; and Flemming Binderup Gammelgaard for review and comments on the manuscript.