Feedback of trial results to participants: a survey of clinicians' and patients' attitudes and experiences.

PURPOSE: There is growing interest in the provision of trial results to trial participants. However, there are a number of gaps in the research base relating to the closure of clinical trials and feedback of results to participants.
METHODS: The aim of this research was to explore the practice of feeding back trial results to trial participants and to identify best practice in this area. Postal questionnaires were sent to members of the UK National Cancer Research Institute Clinical Studies Groups (NCRI CSG) and to patients over the age of 18 years who completed trial treatment (located in one Cancer Network) during a 16-month period (April 07-July 08).
RESULTS: 145 NCRI CSG member surveys and 81 patient questionnaires were returned. The vast majority of all respondents supported the idea of offering results to trial participants. However, NCRI members and trial participants differed in their opinions about the timing and method for the provision of results.
CONCLUSION: The results provide an insight into the views of these groups in relation to desire for results and practical aspects of results feedback which should inform further investigations into trial management and the practice of feedback of trial results.

Background

There is growing interest in the provision of trial results to trial participants. A review by Shalowitz and Miller (2008) examined 28 studies which explored the practice of results feedback and reported overall that there was considerable evidence that those who took part in trials would like to receive the results of those trials (for examples of these studies see Partridge et al., 2003; Moutel et al., 2005; Fernandez et al., 2007). In addition, there was some evidence that those responsible for clinical trials supported the return of results to those who took part (for examples of these studies see Di Blasi et al., 2002; Fernandez et al., 2003; Partridge et al., 2004). While there appears to be a desire to receive results and an increasing acceptance of the necessity of providing them, there remains a lack of information in relation to the actual practice of results feedback and the barriers and facilitators relating to results feedback.

This paper presents the findings from a piece of work that was designed to survey both clinicians and patients involved in a broad spectrum of UK cancer trials, about their views of, attitudes towards, current practice in and experiences of, the feedback of trial results. The knowledge base developed provides an insight into the views of these groups in relation to desire for results and practical aspects of results feedback which should inform further investigations into trial management and the practice of feedback of trial results.

Methods

The aim of this research was to explore the practice of feeding back trial results to those who take part in cancer trials. A number of objectives were developed in order to clarify this aim. These were:

To investigate attitudes towards the feedback of trial results

To identify barriers and facilitators associated with trial results feedback

To identify areas of best practice

To identify future areas of research in relation to effective and appropriate feedback of trial results.

In order to meet this aim and these objectives it was identified that a postal questionnaire survey targeting both clinicians and trial participants was required. The questionnaires were developed on the basis of a targeted literature review relating to feedback of trial results. Drawing on the findings of this review, survey items were generated and refined by the research team; efforts were made to construct items that could be administered to both NCRI and patient respondents (in parallel forms). Draft versions of each questionnaire were piloted with NCRI members and a patient forum (for NCRI and patient formats respectively), and items were adjusted on the basis of this feedback. The survey included closed and open questions and short and long answer formats to elicit responses about the ethical, practical, and attitudinal issues associated with feedback of trial results to study participants. Relevant ethical and NHS Trust approvals were obtained for the survey work.

In order to target clinicians involved in clinical trials the survey was sent to members of the 21 UK National Cancer Research Institute Clinical Studies Groups (NCRI CSG), including 15 cancer-site specific groups and five generic groups and the Consumer Liaison Group. The survey was distributed at group meetings or posted/emailed out to those not present. The patient survey was sent to patients over the age of 18 years (located in one Cancer Network) who had completed a range of identified trial treatments (i.e. chemotherapy, radiotherapy, hormone therapy, surgery) during a 16-month period (April 07–July 08). A questionnaire pack was mailed to eligible patients via the network trials staff at each of the participating hospitals who managed the trial databases and checked patients were eligible for the study. Questionnaires were returned to the research team via pre-paid envelopes.

Data analysis

Responses were summarised using descriptive analyses in SPSS. The Fisher exact test and Mann–Whitney U test were used to compare NCRI and patient responses. This paper presents a selection of the findings from these questionnaires.

Results

Respondent characteristics

Table 1 shows characteristics of survey respondents. In total, 145 NCRI CSG member surveys were returned, representing 32.5% of the target population (446); 120 returns were from professional members of the NCRI CSGs and 25 were from consumer members. Most professional member respondents (80.5%) had experience of being the principal investigator on a clinical trial. A minority of professional and consumer members had experience of serving on an ethics committee. A further 81 patient surveys were returned representing 52% of the target population (157).

Table 1

Characteristics of survey respondents.

Characteristic	NCRI CSG members			Patients (total N = 81)
	Professional (total N = 120)	Consumer (total N = 25)	Combined (total N = 145)
Gender	Valid n = 114	Valid n = 25	Valid n = 139	Valid n = 80
% Female	36.8	64.0	41.7	37.5
% Male	63.2	36.0	58.3	62.5
Age	Valid n = 112	Valid n = 25	Valid n = 137	Valid n = 80
Mean (SD)	47.8 (7.2)	56.0 (9.4)	49.3 (8.3)	63.9(11.3)
Served on ethics committee?	Valid n = 114	Valid n = 25	Valid n = 139
% Yes	25.4	8.0	22.3	Na
Been PI on a clinical trial?	Valid n = 113	Valid n = 25	Valid n = 138
% Yes	80.5	0.0	65.9	Na

Should trial results be offered?

Both surveys included the question ‘should trial participants be offered a lay summary of results?’ The principle of offering results was broadly supported by both groups of survey respondents. Of the 141 NCRI CSG members who responded to this question, the majority (134; 95%) indicated that trial participants should be offered a lay summary of the trial’s results. Only two respondents (1.4%) indicated that trial participants should not be offered a summary of results. Of the 79 patients who responded to this question; the majority (78; 98.7%) indicated that trial participants should be offered a lay summary of trial results. One respondent (1.3%) stated that trial results should not be offered to participants. There was no significant difference between NCRI and patient respondents in responses to this question (Fisher’s exact test, p = .25).

Managing the provision of feedback

Both surveys explored various alternatives for when and how to communicate results. Respondents were asked to select all options that they supported (by ticking appropriate boxes) and to specify (by free text response) the person/method for information delivery that they considered to be best. Table 2 presents response frequencies/proportions by respondent type.

Table 2

Support for different options around management of feedback provision.

	NCRI respondent support (1)	Patient respondent support (2)	Difference (=1–2)	95% CI (diff)
				Lower	Upper
Timing of results feedback
Results should be given to patients at a point after the whole trial has closed (all data collected)a	89.4% (127/142)	44.3% (35/79)	45.1%*	32.6%	56.3%
A summary of ‘results to date’ should be given to patients at the point at which they leave the study	23.2% (33/142)	51.9% (41/79)	−28.7%*	−41.0%	−15.4%
Regular updates of the trial’s progress should be provided to patients who wish to receive them	42.3% (60/142)	58.2% (46/79)	−16.0%*	−28.9%	−2.2%
Method of results feedback
Results are given to patients by their clinician, face-to-face	49.0% (70/143)	58.0% (47/81)	−9.1%	−22.0%	4.5%
Results are given to patients by a nurse, face-to-face	35.0% (50/143)	22.2% (18/81)	12.7%*	0.2%	23.9%
Results are given to patients by a member of the research team, face-to-face	37.8% (54/143)	44.4% (36/81)	−6.7%	−19.9%	6.5%
Results are posted to patients when they are available	46.2% (66/143)	45.7% (37/81)	0.5%	−12.9%	13.7%
Results are emailed to patients when they are available	21.0% (30/143)	18.5% (15/81)	2.5%	−9.0%	12.6%
Patients receive a letter to say results are available, and are given a telephone number to request them	49.7% (71/143)	9.9% (8/81)	39.8%*	28.1%	49.2%
Patients receive a letter to say results are available online, and are given a link to the appropriate web address	48.3% (69/143)	21.0% (17/81)	27.3%*	14.4%	38.3%

*Significant difference at p < .05. Note Difference shows the difference in proportions between sub-groups .95% confidence intervals for these differences were calculated using Newcombe methods (Newcombe, 1998). Where confidence intervals do not cross 0%, proportional differences are statistically significant at p < .05.

This response category included more specific sub-options for NCRI respondents, but not patient respondents.

The most frequently endorsed option amongst NCRI CSG members for when to share results with participants was at a point after the whole trial has closed (selected by 127 respondents; representing 89.4% of those responding to the question). Within this broad category (post-closure), the time of publication was the most frequently supported time-point for feedback provision (101; 71.1%). In contrast to this, the most frequently selected option amongst patients for when to share results with participants was to provide regular updates of the trial’s progress, (46; 58.2%). However, a substantial proportion of NCRI CSG member respondents (60; 42.3%) also endorsed a system of regular information updates.

Examination of responses by respondent type indicated that NCRI and patient respondents differed significantly in their preferences around timing of results feedback (Fisher’s exact tests, all ps < .001). Patient respondents were more likely than NCRI respondents to endorse both (1) feedback of ‘results to date’ to individuals as they end trial participation and (2) regular information updates regarding trial progress. It may be that NCRI respondents had greater concerns about releasing information before it is finalised and/or the practical demands of providing interim/regular information updates.

None of the options presented for how to distribute results to participants were endorsed by a majority of NCRI CSG members. The most frequently supported option, selected by 71 (49.7%) NCRI CSG member respondents, was for patients to receive a letter which indicates that results are available and provides a number to call to request them. Patient respondents most frequently selected that results should be given to patients by their doctor, face-to-face (47; 58.0%).

Formal comparisons of NCRI and patient responses indicated that respondents significantly differed in their perception of the acceptability of three distribution methods. The option of notifying patients by letter that results can be requested by phone was less popular with patient respondents than NCRI respondents (Fisher’s exact test, p < .001). Similarly, patient respondents were less supportive of a parallel option – notifying patients by letter that results can be accessed online – as compared with NCRI respondents (Fisher’s exact test, p < .001). NCRI respondents were more supportive of face-to-face provision of results by a nurse, as compared with patient respondents (Fisher’s exact test, p = .050). No other significant differences were observed with regard to supported distribution methods.

Offering results to next of kin

NCRI CSG members and patients were both asked to consider whether results should be offered to the next of kin of deceased patients. Of 143 NCRI CSG members who responded to this question, 76 (53.1%) indicated that the next of kin should be offered results. Of the 81 patient responses to this question, 56 (69.1%) indicated that patient’s next of kin should be offered results. Forty one NCRI CSG member respondents (28.7%) indicated that next of kin should not be offered results, and 26 (18.2%) selected the ‘don’t know’ response option. Eighteen patient respondents (22.2%) indicated that next of kin should not be offered results and seven (8.6%) selected the ‘don’t know’ response option. Formal comparison indicated that response proportions differed between respondents (Fisher’s exact test, p = .043) with patient respondents being more supportive of offering results to next of kin.

Attitudes towards results feedback

Both surveys contained a section to assess the attitude of respondents towards a variety of statements; responses were provided on a 5-point ordinal scale, with high scores indicating greater agreement with the statement. The NCRI CSG member and patient responses are presented as box plots in Fig. 1.

Fig. 1

Box plots of statement agreement by respondent type. *Significant difference at p < .05. Note. For the box plots, error bars show the minimum and maximum values, boxes show the interquartile range, and horizontal lines show the median.

Mann-Whitney U tests were carried out for each statement, comparing NCRI member and patient agreement. Patient respondents tended towards stronger agreement with the statements ‘most patients want to know the results of trials in which they have taken part’ (p < .001), ‘most patients understand the potential effects and implications of receiving trial results’ (p < .001), ‘there should be an informed consent process around the offer of trial results’ (p < .001) and ‘routinely offering trial results would increase patient trust in professionals’ (p < .001).

Possible disadvantages of offering feedback

Free text questions on both surveys asked respondents to comment on the possible disadvantages of offering trial feedback. As a broad category, negative psychological impact on patients and carers was the most frequently reported potential drawback of offering/providing results: present in 109/135 NCRI CSG sources (80.7%) and 34/67 patient sources (50.7%). Within this category, effects of negative results/bad news (e.g., of side effects or being in the inferior arm) were the dominant concern. Twelve NCRI CSG respondents (8.8%) and twenty five patient respondents (39.6%) indicated that they perceived no real disadvantages to offering feedback.

Facilitating the practice of results feedback

Respondents were asked to suggest factors that would help to facilitate the practice of feeding back results to participants (free text response). Within the NCRI CSG member responses the most frequently identified facilitator of feedback practice was to introduce the concept at the point of trial entry (41/125 sources, 32.8%) such that informed consent about feedback (and identification of associated preferences) can be obtained at an early stage. A process of normalisation/cultural shift (such that feedback becomes an expectation) was identified as a facilitator by 23 respondents (18.4%); the same number of respondents identified extra resources as a facilitator. A system for data-tracking and long-term follow-up (with implicit resource implications) was identified as facilitating by 16 respondents (12.8%). Fifteen respondents (12%) suggested that compulsion (by ethics regulators or funders) would be most effective.

The patient questionnaire asked ‘what do you think would help to encourage the practice of offering results to trial participants?’ Many respondents (24/53, 45.3%) did not answer the question posed, instead they provided a variety of responses ranging from how trial participants themselves might be encouraged to ask for results, to statements of belief about the rights of all participants to be offered results. Four respondents (7.5%) expressed uncertainty over the question, one wrote “Don’t understand the question. If it is part of the procedure to offer results why do researchers need encouraging? It is part of the task, part of the job, isn’t it?” Twelve responses did relate to ways of encouraging the practice of offering results and suggested that, offering results should be made a routine part of clinical trials (4/53, 7.5%), helping staff to understand why patients want feedback (4/53, 7.5%), the introduction of a financial incentive for research centres to provide feedback (3/53, 5.7%) and the provision of extra staff to cope with the additional workload (1/53, 1.9%).

Personal experience of being offered trial results

The patient survey asked trial participants if they had been offered the opportunity to receive feedback about the trial in which they had taken part. A large majority (68/79, 86.1%) indicated that they had not been asked, 8.9% (7/79) indicated that they had been asked and 5.1% (4/79) were unsure. Two participants did not answer this question. Respondents who indicated that they had been offered the opportunity to receive results were subsequently asked to specify (by free text response) who had asked them, what had they been told, and whether there was anything else they wanted to know about the trial. Eight trial participants provided further information about trial feedback that they had received. The feedback had been offered to them by either a doctor, a nurse or other member of the research team. One individual commented that the consent form that they signed at the start of the trial asked whether the participant wanted to receive feedback after the trial. Several individuals had yet to receive any information about the trial and only one individual stated that there was nothing else they still wanted to know in relation to the trial.

All respondents were asked if they wanted to know the results of the trial in which they took part. A substantial majority (69/78, 88.5%) indicated that they did want to know the results of the trial in which they had taken part. 5.1% (4/78) did not want to know the results and 6.4% (5/78) were unsure. Respondents who indicated that they would like to receive trial results were subsequently asked to specify (by free text response) what in particular they would like to know. The responses indicated that some individuals desired personal results, others wanted to know the overall findings and many wanted to know both of these things. A closed response question also asked respondents about the kind of trial feedback that they would like to receive. 70.4% (57/81) indicated that they would want a summary of overall findings and 69.1% (56/81) indicated that they would like personalised feedback with individual implications (respondents could endorse more than one response option). Cross tabulating responses to this question: 35 respondents (43.2%) indicated that they would want both overall and individualised findings; 22 respondents (27.2%) would want overall but not individualised findings; and 21 respondents (25.9%) would want individualised but not overall findings.

All respondents were then asked ‘if the results of your trial did not show any benefit, would you want to be given a summary of the (negative) results? The majority of respondents (68/81, 84.0%) indicated that in this situation they still wanted to receive the trial results. 9.9% (8/81) did not want negative results and 6.2% (5/81) were unsure.

Discussion

Consistent with previous studies (for example Partridge et al., 2005; Fernandez et al., 2009; Sood et al., 2009) this research identified that a majority of patients desire results feedback. Similarly the study also identified a high level of support for feedback from those involved in running clinical trials. 95% of NCRI group member respondents expressed support for the concept of offering trial results to participants. This is a more emphatic expression of support than previous studies exploring investigator support for results communication have identified (Partridge et al., 2004; Rigby and Fernandez, 2005; Di Blasi et al., 2002) and may reflect a growing awareness of the desires and rights of trial participants. Despite this it appears that translating the concept of offering results into the practice of offering results is proving difficult and in this survey over 80% of trial participant respondents reported they had not been offered any feedback of trial results.

The survey data from NCRI members indicated that despite supporting the concept of offering trial results clinicians continue to have concerns about the implications and practicalities of offering trial results. Over 80% of NCRI group member respondents reported concern over the potential negative psychological impact on participants of receiving trial results. 50.7% of patient respondents also indicated that they were aware that results feedback might be distressing or painful, however, many patient respondents indicated that they wanted the results despite this and some suggested that the satisfaction of knowing more about the trial was more important to them than the potential distress that hearing bad news might cause. Several studies have examined whether participants who have received results found them distressing, frightening or alarming. There is some evidence to suggest that receiving results can cause distress for a minority of individuals however this does not appear to consistently translate into regret about receiving results (Dixon-Woods et al., 2006; Partridge et al., 2005, 2009; Schulz et al., 2003). Further research is required which specifically examines the psychological burden of receiving trial results.

With regard to the practicalities of results feedback the findings from this survey also highlight a number of differences in the beliefs and attitudes of NCRI members and trial participants. In particular there were noticeable differences in the attitudes of NCRI members and patients towards the timing and mode of feedback delivery.

Timing of results feedback

Whilst NCRI members showed a preference for providing feedback after trial closure, the patient respondents were more likely to endorse the provision of results at the end of an individual’s trial participation and/or a system of regular trial updates. This may reflect a tendency for clinicians to consider trial results as the published findings where as patients may consider that ‘results’ include other aspects of the trial such as finding out which arm of the trial they were allocated to or being told how the trial is progressing in terms of recruitment and retention. A study conducted by Fernandez et al. (2007), in which potential trial participants were asked to indicate when they thought results should be shared with participants, notes only a single individual (out of 40) selected ‘while the study is still going on’. The majority acknowledged the need for peer-review before disclosure (60%), but did not want “to be the last to know” (Fernandez et al., 2007, p. 441). Whilst in Richard et al.’s (2003) study of genetic breast cancer, several women stated that the reason they wished to know results was in order to discover whether they had the BRCA1/2 gene for breast cancer. In other studies respondents wanted to know whether their trial arm allocation had affected their own health (Dixon-Woods et al., 2006). In our survey trial participants, along with expressing a desire for overall findings, also indicated a desire for personal information relating to their treatment and for practical information about the trial, such as how many people had been recruited. It may therefore be important to ensure that participants of trials are not only offered the overall findings, but that they are also offered the more personal and practical information that they may desire during the course of the trial.

Mode of results feedback

Existing literature highlights that investigators perceive the likely resource cost of offering/providing results to be a barrier to the return of results (Partridge et al., 2004; MacNeil and Fernandez, 2007). The survey data collected in this study appears to have reflected this concern with NCRI members expressing a preference for written feedback. In contrast the patients surveyed expressed a desire for face-to-face feedback, placing value on receiving the results in person, from someone who they knew from the trial and of whom they could ask questions. However previous research has demonstrated that receiving results in written from can be acceptable to trial participants (Partridge et al., 2009) and it has been suggested that receiving results in written form was thought by some to be preferable to personal contact because they could study written material at length and in private, without fear of forgetting the content of a verbal communication (Dixon-Woods et al., 2006). Mancini et al. (2010) have recently explored the use of the internet as a means to communicate trial results and demonstrated that greater accuracy in the understanding of trial results existed amongst those who had received the results via the internet, in comparison to a control group (Mancini et al., 2010). In studies where respondents have not been given results, postal results were also acceptable to many, but a substantial proportion wanted to be given results face-to-face (Johnson et al., 2008; Partridge et al., 2003; Kirschen et al., 2006), especially where results had negative implications (Fernandez et al., 2009). Further research is needed to identify a method or methods for providing results which meet the requirements of both clinicians and patients.

Finally, we acknowledge a number of limitations of this work. First, the response rate for the patient survey was over 50%. More disappointing was the return rate of just over 32% for the NCRI CSG survey. In addition this work concentrated on feedback of results in relation to cancer trials in the UK. It is acknowledged that further work into other health conditions and cultural backgrounds is required in order to establish a greater understanding of the issues associated with the practice of feeding back trial results in other populations.

Conclusion

There is growing support for the idea of communicating overall results to participants after a research study has ended. What is still unknown is the best and most effective way to do this (which, as Shalowitz and Miller (2008) note, is also likely to vary according to the type of trial and its outcomes) and the potential harm and benefits that such disclosure might elicit. Further research is required. We support the suggestions of Shalowitz and Miller (2008) that most informative would be research showing the resource costs and effects on patients of various feedback alternatives, as well as outcomes in terms of satisfaction, understanding, and trial perceptions/behavioural intentions, including willingness to participate again and/or recommend trials to others. Related feasibility work would help to answer questions about the data management required to track patients after the end of active participation in order to be able to return results to those who wish to receive them.

Conflict of Interest

None declared.