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Reactions of participants to the results of a randomised controlled trial: exploratory study
Claire Snowdon a Medical Statistics Unit, London School of
Hygiene and Tropical Medicine, University of London, London WC1E 7HT, b National Perinatal
Epidemiology Unit, University of Oxford, Radcliffe Infirmary, Oxford
OX2 6HE
Correspondence to: Ms C Snowdon, Centre for Family Research,
University of Cambridge, Cambridge CB2 3RF
CMS1000{at}cam.ac.uk
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Objectives: To assess views of parents of babies who
participated in a neonatal trial, about feedback of trial results.
Design: Qualitative analysis of interviews.
Setting: Parents' homes.
Subjects: Parents of 24 surviving babies enrolled in
a UK randomised controlled trial comparing ventilatory support by
extracorporeal membrane oxygenation with conventional management.
Main outcome measures: Views about contents of
results, reactions to results, effect of hindsight, and importance of
feedback.
Results: Information about mortality was well
understood by the parents but morbidity was less clearly reported. Even
when the content was emotionally exacting, the information was still
wanted as it removed uncertainty; provided an endpoint to difficult
events; promoted further discussion within couples; and acknowledged
their contribution to answering an important clinical question.
Conclusions: Feedback of trial results to
participants should be a consideration of researchers, but a careful
approach is required. This study was based on a highly selective group
of parents within a particularly sensitive trial. More research is
needed to assess the extent to which these results can be generalised
to other trials or to groups such as bereaved parents.
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In recent years there has been a demand for the feedback of results of medical research to participants.1-5 There are, however, practical and ethical factors to consider as results can be complicated, alarming, and distressing. Two studies examined participants' responses to results in sensitive situations where the results could induce feelings of guilt or fear. 6 7
Epidemiological research into risk factors for paediatric brain tumours indicated an association with parental behaviour or lifestyle, or both.6 Despite concerns about the impact on parents a questionnaire based study showed that mothers said that they understood the results and viewed feedback as important. The authors concluded that feedback was not emotionally exacting, but a poor response rate undermined the validity of their findings.
When treatment is randomised feedback may be particularly problematic. While randomisation is appropriate at the start of a trial at closure uncertainty should (ideally) be resolved. Trial participants who did not receive what was shown to be the best treatment may with hindsight feel deprived or placed at risk. In a trial of surgery to lower cholesterol after myocardial infarction, where mortality was higher in the control group, a quality of life assessment showed no detrimental effects of feedback.7
Although these studies suggest that feedback of sensitive results is not problematic closer examination is needed. Our study describes the reactions of a sample of parents of surviving babies to the communication of results of a neonatal trial.
United Kingdom collaborative trial of extracorporeal membrane
oxygenation
The trial compared two methods of ventilatory support for
critically ill neonates with acute respiratory failure. At
randomisation the babies were already receiving ventilatory support by
conventional management in a neonatal intensive care unit. Conventional
management was compared with oxygenation of the blood by an external
circuit (extracorporeal membrane oxygenation) in one of five specialist
centres. Neonatal extracorporeal membrane oxygenation was only
available in the trial as it was an unevaluated treatment.
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Communication of results to the parents
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Extract from letter to parents of surviving babies
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Study design
Eight parents (seven mothers and one father) of seven babies in
the trial participated in a pilot study by telephone (table 1). These
parents had already taken part in a general study of parental views of
the trial.10 As a result of the pilot study minor
modifications were made to the questions.
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Content
Before discussing the letter giving the results of the trial the
parents were asked if they had thought about what the trial would show.
In nine of the 14 interviews with parents of babies allocated to
extracorporeal membrane oxygenation the parents said they thought that
the treatment their baby received would be the better treatment.
Although their views were based mainly on personal experience other
reasons included confidence in the medical staff and knowledge of the
use of this approach in the United States. Their babies survived and
they attributed this to extracorporeal membrane oxygenation. One
couple, Rose and Liam, drew the same conclusion although for different
reasons. Their son had complications with his treatment, and at times
Rose believed he was suffering because of their decision to include him
in the trial. She thought extracorporeal membrane oxygenation a
difficult and risky treatment, justified only by its success.
I don't knowa way of making sure before going straight into
it."
In four interviews the parents of babies who were treated
conventionally had thought that conventional management would be the
better treatment. When they received the results they were
"surprised" (Lorna, Helen, and Bill) and "quite shocked"
(Gail), mostly because their babies had survived. Janet was the only
parent who thought this treatment would be the more effective because
"they had used [conventional management] for a long time now."
In 23 of the 24 interviews at least one parent correctly described
improved rates of survival or said that extracorporeal membrane
oxygenation was shown to be the better treatment. The similarity in
health status of the surviving babies in both groups at one year was
less clearly described, with parents in only four interviews accurately
stating there were no major differences. In eight interviews the
parents thought that the babies treated conventionally had more
problems than those treated by extracorporeal membrane oxygenation. In
three interviews parents stated that there were more side effects from
extracorporeal membrane oxygenation. Eileen said:
"It was quite a shock to find out how many disabilities you can get
from ECMO."
Level of information
It is difficult to pitch information at a level suitable for
everyone. While some parents found it difficult to understand the
letter others thought it "wasn't enough" (Adam) and was
"sketchy" (Lee), and some wanted more information. Bill and Frank
wanted to see "graphs" and "figures." Ellen and Adam, Shirley
and Hugh, and Rose and Liam had sought further details through
published material.8 Rose said:
Reactions to results
Pleasure
Six women described the pleasure they felt on reading the results.
They were "over the moon" (Liz), "pleased" (Julie and Sandra),
"very pleased" (Tina and Mary), and "glad" (Hilary). They felt
a great personal involvement in the trial, and there was a sense of
having made an important contribution. Hilary took pleasure in being
"part of helping other children have the benefit of getting the best
treatment that was possible." Hilary and Liz expressed relief that
the trial had come to an early close, ending the trial situation for
parents and establishing extracorporeal membrane oxygenation as an
established treatment.
Feeling lucky
The parents of babies who were treated conventionally could find
the information that babies in this treatment group were less likely to
survive "rather sobering." In eight of 14 interviews the parents
repeatedly described themselves as lucky. The results emphasised how
different things might have been:
"Statistically she shouldn't be here, and there's no doubt about
that ... when we picked it up and examined the
statistics it wasit was a shock to realise that ... she had no realreal sort of chance. So it was a miracle." (Bill)
Feeling lucky was not limited to parents of babies who were treated conventionally as the thought that their baby's life depended on a random process could be generally disconcerting. Tina felt they were fortunate to be in the right place at the right time:
"It's not so much we were lucky to get ECMO because we were taking part in the trial. We were lucky to be able to take part in that trial! The crazy thing is that if the hospital Freddy has been born in [wasn't] in the trial, there would have been this life saving treatment that we would never even have heard of."
Eileen and Jerry, Rose, and Andrea felt lucky that their baby was unaffected by the side effects they thought were associated with extracorporeal membrane oxygenation.
Feeling upset
A major concern in feeding back results to participants is that
the content may be distressing or alarming. In 10 interviews (parents
of four babies treated by extracorporeal membrane oxygenation and six
conventionally) parents described degrees of upset. Some were
"shocked" (Sophie, Eileen, Liam, Bill, and Jim). Hilary said it was
"quite upsetting" and "shook us up." John said "it brought it
all back to us again." The results "bothered" Moira, Lorna was
"a bit down," and Valerie was upset by the reference to the babies
who had died. Although the letter pointed out how few health problems
there were among surviving babies in both treatment groups, Eileen
worried about her son's current and future health:
"When we got them and we read about how many disabilities there were,
I then panicked about Joshua's eye sight, was his hearing okay, were
his lungs working? And I phoned up the consultant and I had to go down
therejust for peace of mind."
In eight interviews parents of babies treated conventionally said their child had had less chance of survival. Lorna said:
"I was a bit down, actually thinking he had the poorer of the two.
You know I was thinking, oh it could have been a lot different. We were
lucky that he managed on that type of treatment when the ECMO was
probablywould probably have been a better course of treatment."
Shirley, like Ellen and Adam, said she was upset not for herself but for others:
"They're saying that ECMO worked and all the rest of it, and quite a
high percentage survived. But if you sit there and you count how many
children actually did dieI find it very upsetting for the parents."
Ellen and Adam sought further information from a published paper.8 The letter had not worried them but they were upset by the paper:
Adam: "That was ... the only time that actually I think since being at the hospital that that actually hit home again."
Ellen: "Yeah."
Interviewer: "Would you have preferred not to have had that information then?"
Ellen: "No."
Adam: "No. We prefer to have as much information as we can get."
Parents often said they believed they would have viewed the results differently if their child had had a poor outcome. Jo and Frank's son has long term complications. Although they did not say the results were upsetting Frank regretted not insisting on extracorporeal membrane oxygenation for his child. He knew that he could not have changed the allocation but still thought he should have "fought tooth and nail."
Effect of hindsight
Few parents said the results had affected their view of the trial,
randomisation, their decision, or their doctor. Although some parents
suggested that randomisation seemed unfair there was also recognition
of the difficulties where there is uncertainty over treatment. Tina was
aware of the need for the trial but saddened by the process. Liam and
Angela expressed similar opinions:
obviously a
... needed experiment etc, but if ECMO would have
saved the baby, thenwell, there's just nowhat's the word I'm
looking for? No, no justification for the trial."
Neil still thought that randomisation was helpful, and took a
philosophical approach:
"His recovery was obviously delayed by the treatment he had [but
if] he had had to move, something could have happened while he was
being moved from hospital to hospital. So there's no guarantee that
even if he had ended up having ECMO that everything would have turned
out all right."
Importance of the results
Sending the results to parents informed them of the findings and,
as recommended by the Royal College of Physicians,12
acknowledged and thanked them for their help. It was clear that the
parents felt strongly they should be sent the trial results out of
courtesy and for further information. Bill described "a need to
know" and Andrea thought it would be "unfair" if parents were not
told the outcome. The results were important to Angela as they
confirmed she was right in wanting extracorporeal membrane oxygenation
for her son:
it's almost as if you don't know
what you think you know, and then when you get the confirmation it's
yes! yes! I feel more sure about myself now. Yes, I was right. All my
instincts were right."
Jim saw the information as important, even if it was emotionally
difficult news, drawing on the enormity of the parents' part in the
process:
"If you're asking somebody to take part in a trial, there's no
point sort of holding the hard facts back from anybody. If you're
wanting people to put other people's lives in their hands
... you've got to come back with figures and say:
`Look, this one was better than that one' ... if
we'd nae gotten them I think we'd be quite, sort of, annoyed."
Although the chance to contribute to research was not the main reason
for most parents participating, it was highly valued by some parents.
There was often a sense of personal investment in the trial, and the
letter made parents feel "included" (Julie), "important"
(Lorna) and "part of it" (Mary). Sophie pointed out that as she and
Ray had been involved in the trial for three years she wanted to know
the results "otherwise how do you know you've been of any use."
Only Frank questioned why he had requested the results. He thought he
had not taken the decision seriously and had simply ticked a box on the
questionnaire. He said: "Don't know why I said send them
... cos there's nothing I can do with them." His
wife, Jo, thought the results would have some value and interest for
their child at a later stage. This was a commonly held view. For most
of the parents the letter was part of a collection of mementos of their
baby's traumatic start to life. The results also marked the end of a
stressful period.
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The parents in this study thought that the participants of a trial should have access to its results. For these parents the letter giving the results had been important for reasons in addition to providing information: it recalled an emotional time, sometimes prompting them to re-examine and discuss their experiences; it provided tangible information on a previously uncertain situation and clarified and rounded off a stressful period; and it acknowledged the important and valuable contribution they had made in answering a medical question.
The parents were surprised that results are not commonly sent to participants. They had often wanted more details about the trial and at the end of the interviews had frequently asked how the other parents felt. Some parents took the time to read a clinical paper8; protecting the parents from potentially distressing knowledge would have seemed paternalistic and possibly insulting. Although some parents were upset all reiterated that they wanted to know the results; they emphasised the need for a careful approach to communication of this information.
The results from this trial are particularly sensitive, and feedback was discussed with the steering group, an advisory meeting of voluntary groups, and the trial and study team. Of particular concern was the possible impact on bereaved parents especially when babies were treated conventionally. The original study was designed to include bereaved parents. When it became apparent that not all parents who requested results had received them a dilemma arose. Telephoning these parents to ask about receipt of results seemed insensitive, and there was concern that even a carefully worded letter might cause distress. It was therefore decided to contact only the parents of surviving babies. For future studies it is important to establish in advance the procedures for contacting vulnerable groups.
It should not, however, be assumed that bereaved parents would have only viewed the results negatively. Firstly, these parents would have chosen to see the findings and, like the parents of surviving babies, may have used the results to talk to their families, or may have found relief in a resolution to their uncertainty over treatment. Secondly, these parents may have valued the contribution their baby made to the welfare of future families. Mary, a parent of one of the surviving children, was clear that when her family agreed to join the trial part of the decision was to make sense of their loss if their child died.
We suggest a cautious approach to feedback even in seemingly harmless circumstances. Only a few parents appreciated the news that there were few lasting effects of either treatment for survivors. Instead some parents felt that surviving children who were treated conventionally were at a disadvantage, and others that extracorporeal membrane oxygenation had worrying side effects.
It is crucial that only participants opting to receive the results of a trial are contacted. Although all the parents of surviving babies who returned their initial postal questionnaire chose to receive the results, three of the 38 bereaved parents who replied specifically did not want this information. It is clearly important that participants do not receive results without their permission. It could be argued that those who request results cannot know what they are agreeing to at the time; it is only when information is obtained that it is possible to say whether it is helpful or harmful. For this reason it is important that more research, especially with vulnerable groups, should be carried out.
Conclusions
As this study included only those parents who requested results
this research was unlikely to show many parents who saw the results as
irrelevant or unimportant. It may, however, have highlighted confusion
and distress, and largely this was not the case.
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Acknowledgments |
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We thank the parents for their time and information; Sally Roberts (Centre for Family Research) and Carole Harris (National Perinatal Epidemiology Unit) for practical assistance; and Ann Johnson, Hazel McHaffie, and Fiona Lockett (a parental support group representative from the extracorporeal membrane oxygenation trial steering committee) who commented on the communication of results to bereaved parents.
Contributors: CS, JG, and DE initiated the study, discussed core ideas, planned the study design and fieldwork, participated in the analysis, and wrote the paper. CS conducted all the interviews and the textual analysis; she will act as guarantor for the paper.
Funding: The study was funded by the Department of Health and developed from research funded by the Wellcome Trust. The National Perinatal Epidemiology Unit is funded by the Department of Health.
Conflict of interest: None.
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(Accepted 6 March 1998)
© BMJ 1998
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