Active involvement of patients in the health sector is growing [18, 19]. Patient involvement affects procedures and decisions regarding health policy [20], treatment [21], research [18, 22], health technology assessments [23], genetic testing [24], and scientific advisory processes [19]. Patient involvement is thought to make health systems more user-friendly and information more accessible. Patient empowerment also involves respecting their rights and voices [25]. For these reasons, informed consent considerations cannot be ignored. Informed consent represents a very fundamental tool to achieve these goals; development of an efficient procedure that respects these principles is critical.
As per law and ethics, in many countries, physicians must collect informed consent from each patient or patient’s representative (e.g. parent or guardian) before enrolment in a study or initiating treatment [3, 4]. This rule also applies to biobanks, as the biological material they collect is for scientific research purposes. Although the informational phase usually results in patients merely signing the informed consent form, this stage should also involve truly educating patients so that they can make informed decisions [1]. Despite growing interest in patients’ understanding of research in which they are enrolled, participants’ awareness and attitudes towards biological material donated for research are not well-studied [2]. Moreover, failing to properly inform patients about the purposes of human tissue sampling and use has caused a reduction of public confidence in biobanks, which can only be recovered by a transparency policy [26]. Issues raised in this investigation bear on both scientific and ethical perspectives [27]; we aimed to improve understanding, awareness and involvement of CRO-Biobank participants through a multisource information approach.
As no standard methodology for evaluating the informed consent process exists [28], we developed a self-administered questionnaire to assess patients’ information and satisfaction, and a final assessment test on the biobank purposes.
A review by Flory et al. evaluated effects of different interventions to improve participants’ understanding of informed consent for research, including multimedia interventions, enhanced consent forms, extended discussion, and miscellaneous methods. The review showed that these information sources do not consistently improve participants’ understanding, especially for less-educated patients. It appears that the only effective source of information is a one-on-one session where a study team member spends extra time explaining the study to the participant [1]. Our multisource information procedure included this type of approach among others; we added the support of a biobank nurse or a biologist to the physician to provide more explanation in simple language, particularly for less educated patients [15]. Most participants of this study were satisfied with the quality of the information received during the enrolment, as they rated each CRO-Biobank source of information “very understandable” (brochure, physician, nurse/biologist) or “fairly understandable” (enhanced informed consent form), although 56.1% expressed the wish for further information. Education level was associated with understanding: more educated participants rated sources of information as more understandable. This is in accordance with other studies [1]. Less-educated participants might, nevertheless, be poorly skilled in filling in the questionnaire, which could lead to underestimation of their understanding of the information [1]. Another biobank set a multisource approach to communication based on newsletters, external advisory boards, and focus group discussions in order to provide on-going feedback [10]. Participants expressed their desire to be informed only about changes in protocols or informed consent procedures; this result emphasizes the need for constant communication on these topics to maintain participants’ understanding, confidence, and trust.
Our study gave us insight into patients’ knowledge of biobanks prior to their participation therein. Only 36.5% were aware of the existence of biobanks: this percentage dropped to 25.8% in less-educated patients. Most participants who knew of biobanks before entering the study had found information through our Institute, which led to an overestimation of the percentage. Indeed, our goal of improving patients’ knowledge is not limited to the present study but is part of our biobank policy. The informational material can be found in various places around the Institute (i.e., display cases, stands), on-line, and through a dedicated phone line. The results of the study indicate that our efforts were successful for 30.7% of our patients (compared to 4.7% who answered that they had been informed by other hospitals). The information provided by our Institute was more effective than television (21.3%), which is the main source of information for less educated patients, and newspapers/magazines (17.6%), or the Internet (6.6%).
The most noteworthy result was that, with our multisource information approach, more than 99% of participants correctly answered questions about the purposes of biobanks. Moreover, our results showed that, unlike most studies, our approach allowed the information to be widely understood by less educated participants [1]. A survey among participants in another biobank, like ours, indicated that participants were motivated by a “pragmatic attitude” to contribute to research advancements; knowing what prompts individuals to participate helps define how to strengthen their involvement and awareness.
A novel aspect of this study is to investigate how patients preferred to be informed. Most preferred multiple sources of information. More educated patients preferred a website, while less educated patients opted for having information provided by their general practitioner.
The study showed wide awareness among participants; 95.5% of patients were aware of their involvement in a biobank (as all participants donated biological samples to our biobank, the question: “Did you donate biological material to CRO-Biobank?” was meant to see whether they were aware of their participation). These data are in accordance with studies by Toccaceli et al. (89.9%) [2] and Nakayama et al. (92%) [29], but in contrast to those of Moutel et al. (0%) [15].
The protection of personal rights in biobanks is a topic of great interest [30, 31], and studies of public perception of biobanks have increased [30, 32, 33]. In contrast, we investigated the participants’ perceptions of this topic, rather than public perception. More than 98% of patients felt respected in their privacy and desires, and most rated the enhanced informed consent forms as “fairly understandable” to “very understandable”; these results represent an important aspect of the quality of our system and our intention to make patients feel part of the research.
Cancer patients face their diseases in personal ways; their views on participating in research or biobank projects vary, and can be different from the general population. For this reason, we directly investigated their understanding and awareness rather than perform a general population survey. Such surveys among cancer patients remain scarce but some articles have been published over the years [34–39]. In particular, Mancini et al. assessed cancer patients’ understanding about a biobanking informed consent process in the framework of a routine “opt-in” scenario [37]. Their results showed that 61.5% of patients who remembered giving consent and 31.5% of patients who declared not to have given consent, actually had given it. Moreover 41.3% of patients understood that consent implied giving access to their medical data. Scarce consensus exist about cancer patients’ reasons for participating in a biobank. In our experience, patients mainly contribute to aid cancer research (in accordance with the survey of Huber et al.) [39] and secondarily, to support care or for moral obligation; in other studies they contribute hoping for personal benefit [38, 39], because of societal welfare or as an act of benevolence [38].
Nowadays, the need to collect informed consent for biobanking is somewhat controversial. Some authors proposed “opt-out” [40] or “opt-out-plus” procedures [36]. Whereas participants in opt-in procedures explicitly express their consent, in opt-out procedures, inaction signifies consent, and participants must affirmatively decline to take part, either orally or in writing. In some countries, such as Denmark, France and Belgium, this option is considered a sufficient consent modality for the use of residual tissue for research [40]. The surveys by Vermeulen et al. of cancer patients support adoption of opt-plus procedure, in which patients are informed about the possibility to opt-out, both verbally and by means of a leaflet [35, 36]. In contrast, when Mancini et al. questioned cancer patients about their attitudes concerning informed consent, most felt that biobank research should require patients’ signed consent. Opting-in is the method suggested for including people in clinical research [41]; in most countries, it is the only allowed solution.
Regardless of the type of procedure that is chosen, the concept of informed consent is based on respect for participants’ autonomy and their right to control their medical care and research participation. Our system fulfils this condition from both ethical and legal points of view.
Although our informed consent procedure was designed and developed in an institutional biobank, its features can be easily implemented at low cost by other institutions thanks to its simplicity, which represents its major strength. Moreover it can be applied in several fields, from clinical practice to research, to improve patients’ awareness and understanding of information relevant to the informed consent process. Resources needed to apply this system are the following: (a) an initial investment of time to enhance the informed consent form (see Figure 1) and to format the graphic layout of the patient’s information sheet to create a brochure, a poster, and a web page; (b) a meeting to train staff about the project; and (c) involvement of a trained biologist/nurse who can give participants further information in plain language, and answer any of their questions, before the collection of samples site or through a phone line (estimated time ≤ 15 minutes for each participant). The last is the only resource that could lead to sizeable financial outlay— the cost of a nurse’s services if one is not already available. For our program, a research nurse (whose job was to collect samples and interview patients for our biobank) was trained with no additional cost to our institution.
Among the limitations of the study, we must consider potential bias due to participants who did not return the questionnaire, or who refused to answer some questions. Although we do not have the detailed demographical data regarding non-responders, the percentage of them was approximately the same distributed by department, pathology, sex and age. We assessed patients’ perception of the quality of each source of information, but this parameter does not indicate the real quality of the sources. As a starting point for future investigations of this topic, we suggest developing an assessment test to be given after having informed patients through each source. Further studies are also needed to determine whether, after a longer period (e.g. one year), participants still retain the disclosed information. The multisource information approach could also be optimized, for example by verifying which sources are the most efficient and excluding the others. To enhance the understanding of less educated participants, the results of our study suggest involving their general practitioners as a new source of information, as this was frequently requested by patients in the questionnaire.
