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Review
Patients’and Members of the Public’s Wishes Regarding
Transparency in the Context of Secondary Use of Health Data:
Scoping Review
Annabelle Cumyn1,2, MDCM, MHPE; Jean-Frédéric Ménard2,3, BCL, LLB, PhD; Adrien Barton2,4, PhD; Roxanne
Dault2, MSc; Frédérique Lévesque2, MSc; Jean-François Ethier1,2, MDCM, PhD
1Département de médecine, Faculté de médecine et des sciences de la santé, Université de Sherbrooke, Sherbrooke, QC, Canada
2Groupe de recherche interdisciplinaire en informatique de la santé, Faculté des sciences/Faculté de médecine et des sciences de la santé, Université de
Sherbrooke, Sherbrooke, QC, Canada
3Faculté de droit, Université de Sherbrooke, Sherbrooke, QC, Canada
4Institut de recherche en informatique de Toulouse, Toulouse, France
Corresponding Author:
Jean-François Ethier, MDCM, PhD
Groupe de recherche interdisciplinaire en informatique de la santé
Faculté des sciences/Faculté de médecine et des sciences de la santé
Université de Sherbrooke
2500 boul. Université
Sherbrooke, QC, J1K 2R1
Canada
Phone: 1 819 346 1110 ext 74977
Email: jean-francois.ethier@usherbrooke.ca
Abstract
Background: Secondary use of health data has reached unequaled potential to improve health systems governance, knowledge,
and clinical care. Transparency regarding this secondary use is frequently cited as necessary to address deficits in trust and
conditional support and to increase patient awareness.
Objective: We aimed to review the current published literature to identify different stakeholders’ perspectives and
recommendations on what information patients and members of the public want to learn about the secondary use of health data
for research purposes and how and in which situations.
Methods: Using PRISMA-ScR (Preferred Reporting Items for Systematic Reviews and Meta-Analyses extension for Scoping
Reviews) guidelines, we conducted a scoping review using Medline, CINAHL, PsycINFO, Scopus, Cochrane Library, and
PubMed databases to locate a broad range of studies published in English or French until November 2022. We included articles
reporting a stakeholder’s perspective or recommendations of what information patients and members of the public want to learn
about the secondary use of health data for research purposes and how or in which situations. Data were collected and analyzed
with an iterative thematic approach using NVivo.
Results: Overall, 178 articles were included in this scoping review. The type of information can be divided into generic and
specific content. Generic content includes information on governance and regulatory frameworks, technical aspects, and scientific
aims. Specific content includes updates on the use of one’s data, return of results from individual tests, information on global
results, information on data sharing, and how to access one’s data. Recommendations on how to communicate the information
focused on frequency, use of various supports, formats, and wording. Methods for communication generally favored broad
approaches such as nationwide publicity campaigns, mainstream and social media for generic content, and mixed approaches for
specific content including websites, patient portals, and face-to-face encounters. Content should be tailored to the individual as
much as possible with regard to length, avoidance of technical terms, cultural competence, and level of detail. Finally, the review
outlined 4 major situations where communication was deemed necessary: before a new use of data, when new test results became
available, when global research results were released, and in the advent of a breach in confidentiality.
Conclusions: This review highlights how different types of information and approaches to communication efforts may serve
as the basis for achieving greater transparency. Governing bodies could use the results: to elaborate or evaluate strategies to
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educate on the potential benefits; to provide some knowledge and control over data use as a form of reciprocity; and as a condition
to engage citizens and build and maintain trust. Future work is needed to assess which strategies achieve the greatest outreach
while striking a balance between meeting information needs and use of resources.
(J Med Internet Res 2023;25:e45002) doi: 10.2196/45002
KEYWORDS
transparency; information; means of communication; public; patients; secondary use; health data; learning health systems
Introduction
Potential Benefits From the Secondary Use of Health
Data
In recent decades, the potential benefits of secondary use of
health data have been highlighted in many different contexts,
including health systems governance, quality improvement [1],
point-of-care decision-making [2-4], and research [5]. While a
unique, unanimously accepted definition of secondary use
remains elusive, we will operate under the premise that it covers
any use beyond the initial intent of data collection. Given the
resources required to generate health data, using them to their
fullest extent to improve health is a laudable goal. Nevertheless,
health data often describe an individual, and thus their autonomy
regarding the use of data also needs to be considered [6]. For
individuals to be able to weigh in on the use of data about them,
they at least need to be aware that data generated for a primary
reason (eg, during a consultation with a physician) have values
above and beyond the specific context for which they were
created and can therefore be used for secondary goals such as
research projects or systems policy research. As mentioned
previously, the secondary use of health data covers a broad
landscape, and the scale of expected benefits (eg, How much?
How soon? Who?) varies from one use to the other, which could
modulate how individuals react to different types of secondary
use.
Therefore, while the potential of secondary use of health data
for research purposes is seen as promising to many stakeholders
in the academic and health care worlds, trust regarding data
sharing in novel contexts should not be considered as a given
[7]. Indeed, despite substantial evidence that most individuals
support the secondary use of health data, this support is neither
unanimous nor unconditional [6,8-11]. Stakeholders including
patients and members of the public express different needs that
touch upon trust and agency [12]. Controversial initiatives such
as care data in England [13], the National Electronic Health
Record System in Australia [14], and other incidents involving
unconsented and unauthorized use of data [15-17] have
contributed to some mistrust in the population, which may then
tend to develop greater sensitivity to the risks associated with
data sharing and health data in general [7,12]. In addition, few
people seem to be aware that the secondary use of their health
data is already permissible in certain circumstances [18]. In all
these situations, some stakeholders perceived a lack of
transparency. Although transparency is unquestionably identified
as an essential element of social license regarding the secondary
use of health data, it is not always clearly defined. For example,
Kisekka et al [19] showed that information accuracy and
availability are important parameters for the adoption of
e-portals; however, an open question is which kind of
information is expected by the stakeholders and under which
modalities of communication. Furthermore, technological
advances offer new ways for transparency to become tangible
and bring forth strategies for more open communication [20].
Therefore, there is a real need to better understand the actual
expectations regarding transparency with regard to secondary
use of health data.
Potential of Learning Health Systems
The fact that a research project is targeting a common health
issue such as heart attacks does not guarantee a rapid and
substantial benefit as an important justification for valuing the
secondary use of health data. For example, in 1982, studies
identified a class of medication, namely β-blockers, as useful
in reducing mortality in the context of heart attacks.
Nevertheless, it took more than 20 years to confirm the
widespread adoption of this simple and relatively inexpensive
intervention [21].
To increase the pertinence of projects undertaken, the rapidity
at which the resulting new knowledge is put into action, and
therefore the benefits to patients, learning health systems (LHSs)
have been proposed. They are a model that embodies how
linking data from clinical care and research can create new
knowledge and improve clinical decision-making [22]. Recent
publications have reported the success of early LHSs [1,23,24],
which is encouraging. Moreover, this integration of care
delivery, research, and effective knowledge transfer can be
implemented as described in the study by Faden et al in 2013
[25], in which a new ethical contract is based on a large
engagement of all stakeholders (including patients), transparency
(both on ongoing activities and data supporting them), and
accountability to demonstrate benefits for the participants. While
communicating about secondary data use is important, it is
particularly essential in an LHS context.
Aims
This scoping review aims to describe how transparency is
portrayed by various stakeholders regarding the secondary use
of health data in a research context. Specifically, we wanted to
address the following questions: (1) What information do
patients and members of the public want to learn about the
secondary use of their health data for research purposes (what)?
(2) How do patients and members of the public want to interact
with this information (how)? and (3) In which situations (when)?
The objectives of the scoping review were elaborated by a team
composed of an expert in health informatics (J-FE), an expert
in research ethics (AC), a legal expert (J-FM), and an expert in
ethics and philosophy of science (AB).
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The study aims to capture a broad landscape of the literature to
serve as a cornerstone of the upcoming consultations with
patients and members of the public to identify the operational
requirements of a transparency portal as LHSs are deployed in
the province of Quebec. Therefore, resources specifically
targeting an LHS context were identified when available, but
the search was not restricted to only these as the LHS field is
relatively young.
Methods
Overview
We addressed our research objectives by performing a scoping
review following the 6-stage methodology framework by Levac
et al [26] (based on the framework initially proposed by Arksey
and O’Malley [27]), which we reported in accordance with the
PRISMA-ScR (Preferred Reporting Items for Systematic
Reviews and Meta-Analyses extension for Scoping Reviews)
framework [28] (the PRISMA-ScR checklist is available in
Multimedia Appendix 1). Our aim was to obtain a large
spectrum of articles, from original research to opinion papers,
law articles, and workshop reports. Our review protocol was
finalized in July 2021, but was not made publicly available.
Definition of Health Data in This Review
We defined health data as data generated either in the context
of health care, whether found in patient charts or connected
objects, or in the context of health care research to capture
perceptions about all kinds of secondary uses. In these secondary
uses, we included approved future uses of biobank data because
the literature on that matter was quite relevant to our research
question.
The Concept of Transparency in This Review
The term “transparency” characterizes a process of information
communication that follows certain norms about the amount,
type, and framing of information that is shared. For example,
the process of informing a person about access to one’s health
data by third parties through a poster in a health professional’s
office might be characterized as aiming at being transparent.
By extension, the term “principle of transparency” can refer to
a normative principle that instructs to communicate information
in a transparent way (in the first sense). Thus, the 2 senses of
the term “transparency” are closely linked. Since the 2 core
notions of transparency are its relevance and its easiness to
understand, this suggests that an investigation of transparency
should concentrate on questions such as “What information
should be conveyed?” “How should it be conveyed?” “In which
circumstances should it be conveyed?”
Search Strategy
An extensive literature search was performed for articles up to
November 2022 in the following bibliographic databases:
Medline, CINAHL, PsycINFO, Scopus, Cochrane Library, and
PubMed. The search strategy was developed with the assistance
of a university librarian and was adapted to each database (the
detailed search strategies are provided in Multimedia Appendix
2). The 5 core concepts used in the search strategy were patient,
public, citizen (and synonyms); health data, medical data, data
use, data sharing (and synonyms); attitude, view, perspective,
opinion, position (and synonyms); information, transparency,
communication, dissemination, awareness, notification,
education (and synonyms); and research, secondary use, LHS
(and synonyms). We did not have to limit the time span.
Eligibility Criteria and Screening
Two reviewers (RD and FL) independently screened the titles
and abstracts of all articles identified by the search strategy
(after removing duplicates), followed by a full-text review of
the remaining articles. To perform an in-depth and broad
research, no article was excluded based on year, type, or location
of publication. The eligibility criteria are presented in Textbox
1.
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Textbox 1. Eligibility criteria.
Inclusion criteria
•Language and article availability
•French
•English
•Full-text availability
•Article type
•Original articles
•Commentary or opinion papers
•Policy or law articles
•Ethics articles
•Population
•Reporting at least patients, members of the public or other experts’ (eg, health care professionals and researchers in various domains) point
of views or recommendations
•Outcomes
•Reporting at least 1 of the following elements: What information to communicate to patients or members of the public; how to communicate
the information (eg, format and support).
•Context
•Secondary use of data
•Uses in research
•Data type
•Health data such as hospital data, electronic record data, administrative data, or medical data
Exclusion criteria
•Language and article availability
•Other language
•Abstract only
•Article type
•Review articles (Review articles that met the other inclusion criteria were screened to include potential studies of interest.)
•Study protocols
•Context
•Primary use of data (eg, for health follow-up)
•Other secondary uses of data (eg, quality improvement, safety monitoring, clinical decision-making, or policy making)
The 2 reviewers first performed a joint screening on a 10%
sample of the articles to familiarize themselves with the selection
criteria and to standardize their approach. Every discrepancy
between the 2 reviewers was discussed to obtain a consensus.
If a consensus could not be reached, a third reviewer (AC or
J-FM) was consulted. The team met weekly to discuss the
progress of the literature review. The search and screening
process was initially completed in July 2021 but was updated
up to November 2022 following the same methodology to
capture and include recent publications.
Data Extraction and Analysis
The extraction, organization, and analysis of information from
the articles included in this scoping review was performed using
NVivo 11 Pro software (QSR International), a tool to conduct
extensive literature reviews [29]. On the basis of the
aforementioned research questions and objectives, the research
team developed a codebook with an initial tree of nodes to chart
the data into NVivo. This codebook also includes the coding
procedure and definitions of the nodes for the reviewers
responsible for the data extraction. All selected articles were
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imported into NVivo software. Four reviewers (RD, FL, AC,
and 1 research assistant) coded all the articles, a process that
was subsequently validated by 2 reviewers (AC and J-FM). All
reviewers received detailed training in the software, project,
and codebook. They all simultaneously coded a sample of the
articles to familiarize themselves with the tree of nodes and
coding method. During training, the reviewers’ coding was
compared with determine whether their approach to data
extraction was consistent, and discrepancies were discussed in
team meetings. This process was repeated until all reviewers
were comfortable with the coding. The coding was performed
in an iterative manner to allow the emergence of new concepts
and themes in the tree of nodes. Weekly team meetings were
held between reviewers to discuss and validate coding decisions
as well as emerging themes. The information was subsequently
analyzed to examine what information, time frame, and means
of communication were proposed, recommended, or desired for
various research activities regarding the access and secondary
use of health data for research purposes. We also aimed to
identify topics that were not addressed in the literature for future
work. The following 3 themes emerged from a text analysis:
content to communicate, how to communicate, and situations
in which communication becomes relevant. A fourth theme also
emerged, namely, the impacts of communication, which were
further divided into potentially negative, neutral, or positive
impacts. This last theme was not directly linked to our research
question and was the focus of a separate article. Note that the
literature update from July 2021 to November 2022 did not
bring any new concepts to light but rather confirmed that we
had reached theoretical data saturation.
Results
Search Outcome and Bibliographic Overview
The detailed selection process is illustrated in Figure 1. After
removing duplicates, 2964 articles were identified through the
database search. The screening of titles and abstracts led to the
assessment of 518 articles in a full-text review. In total, 152
original articles and 13 review articles met our inclusion criteria.
The manual assessment of the references from the review articles
added 26 original articles. Only original articles of those reviews
that met our inclusion criteria were included in the final selection
of this scoping review. Finally, 178 articles were included in
this scoping review.
Figure 1. Article selection process. *Restricted to publications in the last 2 years for more recent indexation.
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The main characteristics of the articles included in this review
are presented in Table 1, and 178 complete references are presented in Multimedia Appendix 3 [6,8,11,18,19,30-202].
Table 1. Characteristics of articles included in the scoping review (n=178).
Articles, n (%)Characteristics
Year of publication
9 (5.1)2002-2006
71 (39.9)2007-2017
10 (5.6)2018
27 (15.2)2019
22 (12.4)2020
24 (13.5)2021
15 (8.4)2022
Continent where study was conducteda
86 (48.3)North America
69 (38.8)Europe
6 (3.4)Asia
5 (2.8)Oceania
4 (2.2)Africa
8 (4.5)Worldwide
Study designb
91 (51.1)Qualitative
52 (29.2)Quantitative
9 (5.1)Mixed approach
9 (5.1)Commentary
16 (8.9)Law and politics
1 (0.6)Descriptive
Perspectives reported in the studyc
68 (38.2)Public
59 (33.1)Patients
26 (14.6)Researchers
22 (12.4)Law and politics
15 (8.4)Ethics
11 (6.2)Health care
8 (4.5)eHealth or informatics
aNorth America: Canada, United States; Europe: United Kingdom, Iceland, Ireland, Sweden, Italy, Germany, Denmark, Switzerland, Norway, Portugal,
the Netherlands, Belgium, France, and Finland. Asia: Saudi Arabia, Taiwan, China, India, and Japan. Oceania: New Zealand and Australia. Africa:
Ghana, Uganda, Zambia, Kenya, and Singapore. Worldwide: studies with multiple countries involved.
bQualitative: workshops, interviews, focus groups, councils, and panels; Quantitative: survey and Delphi; Mixed approach: combination of quantitative
and qualitative designs; Commentary; Law and politics: ethical, legal, or governance frameworks, guidelines, requirements, and other policies; and
descriptive study.
cNot mutually exclusive. Public: Indigenous communities, minority community members, older adults, blind and low-vision communities, and early
adopters of emerging technologies. Patients: susceptible, rare diseases, cancer, cardiac, Parkinson disease, mental health, pediatric (parents and families),
and representatives of patients’ organizations. Researchers: research participants, students, researchers, recruiters, sponsors, investigators, scientific
staff, data infrastructure employers and managers, and research governance experts. Law and politics: policy makers, legal professionals, and regulatory
staff. Ethics: ethicists and research ethics committee members. Health care: health care professionals, health managers, and health systems leaders.
eHealth or informatics: eHealth experts, device or app developers, and data-sharing experts.
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What to Communicate?
There were 2 distinct types of content that emerged from this
review. We refer to them as “generic content” (communication
on broader aspects than the secondary use of their own personal
health data) and “specific content” (communication on the
secondary use of their own personal health data).
Generic Content
Overview
Table 2 lists an overview of the types of generic content that
various stakeholders wish to see communicated with exemplary
citations extracted from the studies reviewed.
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Table 2. Types of generic content: description and examples of stakeholders’perspectives.
Exemplary citations of different stakeholders extracted from reviewed studiesDescriptionType of generic
content
General infor-
mation on sec- •“[...] I think that education in general is a really good tool for the layperson to under-
stand what’s happening and I just think that that’s probably the key, educating away
•Education around data types and
secondary uses [49,54-56,59-61]
ondary use of the fears but also disclosing—what are we responsible for? What could happen? It’s
•Education on Research Ethics
Board role [11,46,50-53]health data
[11,19,30-61]going to help people trust what you’re doing a little more too” (biobank participant
[39]).
•“Participants clearly identified a number of areas where there was a need for more
knowledge and work around data sharing. [...] There were four main areas where
greater knowledge was required: (1) clarity regarding the legal and clinical implica-
tions of shared data for patients and providers, (2) an understanding of legislation
across Canada, (3) decision-making about what data are needed and (4) being aware
of the context of electronic medical records [EMR] data” (Terry et al [35] on the
views of a variety of stakeholders).
Information on
data governance •“Respondents identified biobank objectives, governance structure and accountability
as the most important information to provide participants. Respondents’concerns
•Who: identity of data overseers
[11,31,41,49,53,76,83,85,98,
100-102,115] and data users[11,31,33,34,36,about biobanking generally centered around the control and ownership of biological
[11,31,52,73-77,79-81,86,91,113,114,40-42,49,52-54,57,
59,60,62-118]samples and data, especially with respect to potential misuse by insurers, the govern-
ment and other third parties” (Joly et al [64] on the views of members of the public).117,118]
•“I’m just trying to say there is this framework, you know we say that there is a
governance system in place which will protect the patient and we can look at them
•What and what for: type of data
used and type of secondary use like we do the financial institutions and we’re quite happy with how they exist, well[53,54,59,60,62,75,86,94,108-110,they’re quite well developed. There’s a framework around this and we want some117,118]assurance” (patient with a rare disease [67]).
•With whom: data-sharing practices
[59,60,65,90,97,105,106,111,118]•“Well, I did not know how freely they could share the information, that they are
actually sharing them with payers. So, something needs to be done with that because
•How: sources of funding [95,96],
safeguards [34,53,73,103,104,112] we have a right to know where our information is going [...]” (oncology patient
[114]).
•“I guess, for me I think it’s not so much the party accessing the data, but rather how
is the data being used for and for what purpose. So knowing that, then I’m able to
make a better decision as in whether I want to participate. ... if it’s from a big phar-
macy company, then I think it may be for a commercial gain, but again it still help
people. So I guess it’s still the purpose, how the data being used, the purpose what
is it used for” (member of the public [94]).
•“Similar to other findings [...], our discussants emphasized, however, that disclosure
of data-sharing practices was important in order to make a truly informed decision
and fulfill the fundamental ethical principles of participant autonomy and respect”
(Haga and O’Daniel [63] on the views of members of the public).
•“Overall, workshop participants felt that if they knew more about the processes and
safeguards in place, they might feel more empowered, and hence more open and
trusting in the decision-making process around data collection and sharing (and may
not, therefore, need to opt-in)” (Ipsos [112] on the views of members of the public).
Information on
ethical and legal •“It was also suggested that there would be some benefit in raising public awareness
of the complex legal environment surrounding data sharing and that this might
•Legal framework
[35,81,124,126,127]
aspects [31,33, demonstrate the legitimacy of researchers’access to data” (Mamo et al [40] on the
•Confidentiality and privacy mea-
sures [33,60,63,71,35,39,40,52,53, views of members of the public).
60,63,64,71,73,75,75,80,86,117,118,123,129,130]•“How it is being used, how I am protected from corrupt or evil activities, and what
precautions are taken to protect it” (member of the public [64]).77,80,81,85,86,
103,111,118-131]•Security breach [31,52,60,103]
•“To protect our privacy in a world where we no longer control our data, we must
obfuscate health data, penalize the misuse of health data, and improve transparency
•Penalties for misuse
[31,39,60,73,77,85,103,111,124,128]around who shares our data and for what purposes” (expert opinion [111]).
•Participant rights [73,120-122,131]
•Intellectual property rights [52]
Information on
technical as- •“I would want to know what kind of security the central network is using. Are they
using any type of encryption at all, who has access to the system? How do they
•Data linkage [53,83,96,103,132]
•Types of data sets [34,62]
pects [33,34,38,maintain that type of access, you know, just general [questions]” (patient [40]).
•Whether medical records are ac-
cessed [78]
40,52,53,62,63,73,
78-80,82,83,86,•“Patients also brought up the importance of restricting data access, oversight of such
restrictions and voiced specific questions about data security, for instance, wanting
•How data are shared
[38,80,91,92,106,130]
91-93,95-97,103,106,
118,120,130,132,133]details on how the data would be transferred. Some patients expressed uncertainty
about current practices; as one patient said, ‘I don’t know who has access to my in-
formation’” (Mazor et al [106] on the views of patients).
•Data security
[52,73,79,97,103,117,120,133]
•“Kept in a very safe location. I hate qualifiers like that. It doesn’t make me feel very
safe. I: What would make you feel safe? P: When I see ‘will be kept in a very safe
•Duration of data storage and how
they are stored [78,93,95,97,118]location.’I would want specifics” (patient [73]).
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Exemplary citations of different stakeholders extracted from reviewed studiesDescriptionType of generic
content
•“Around half of the respondents want to receive an easily understandable summary
of project (51%) and information about the management rules (49%)” (Courbier et
al [76] on the views of patients with rare diseases).
•“It would be very helpful to the reader and potential study subject to have some, at
least, some examples of the type of research the researchers intend to do” (patient
[73]).
•“[My mobile data] shows a terrible step count, but that’s because I don’t hold my
phone while playing netball, long walks etc” (member of the public [75]).
•Nature of the research and objec-
tives [11,31,64,68,76,77,79,95,
97,115,132,136]
•Research methodology [79,93]
•Risks and benefits [46,53,65,73,
75,77,79,85,89,92,95,96,99,134]
•Data validity and possible biases
[75,79,96]
Information on
scientific as-
pects [11,31,46,
53,64,65,68,73,
75-77,79,85,89,
92,93,95-97,99,
115,132,134-136]
General Information on Secondary Use of Health Data
The need for general knowledge about information systems and
data-driven research was a transversal theme in our review
(Table 2, row 1), whether in the context of biobanking
[19,30-45,71,83,84], precision medicine [92], mining of health
data [34,44,49,72,115], or data sharing with the industry
[71,73,75,134]. Some stated that the need for education extended
to all stakeholders, including patients, research participants, and
researchers. Clinicians were also targeted for education on how
to integrate genomics into clinical care [40,55,137] and training
in information technologies [120]. Members of the public and
research participants highlighted the value of [31,83]
communicating positive messages about how data
are used: ‘promoting the success stories’, And I can
see an advantage in updates because I think it creates
a positive view of things, a positive view while there’s
so much bad information. You know that here’s a
group of people working for the human good and
you’ve participated in it, you know. It’s uplifting
really [Biobank participant]
Information on Data Governance
The second set of generic content is related to data governance
(Table 2, row 2). Comprehensive and transparent governance
emerged as a key point in the literature from biobanking
[64,69,78,102] and appeared to apply to more novel uses of
data; for example, secondary use of anonymized mobile phone
data [33,65,66,80], unregulated mobile apps [81], novel data
linkages [75,132], sharing genomic data [57,85,91], distributed
data networks [40,109,138], or artificial intelligence
[79,108,139]. The most common content elements pertain to
the structure of governance, nature of secondary use [42,76],
and the identity of data users, in particular if sharing with the
industry [38,40,61,75,93,97,104]. In a recent survey, knowing
the names of the individuals in charge of data governance was
particularly important if identifiable health data were being
accessed in the context of data mining [115]. In the same line
of thought, an analysis on 836 respondents by Kisekka et al [19]
concluded that in the context of e-portals and health technology
use, “[...] individuals worry more about who possesses the right
to access their health data (i.e., who, what, when, and why) than
the mechanisms used to safeguard data from unauthorized
access.” Of interest, the composition of the governance body;
distribution of power; presence of a data overseer (at times
referred to as an ombudsman); and the integration of patients,
citizens, or the community in governance were suggested as
important information to foster trust.
Information on Ethical and Legal Aspects
Stakeholders wished to obtain some information on the legal
frameworks protecting the data, confidentiality and privacy
measures, penalties for misuses, participant rights, and
intellectual property rights (Table 2, row 3). The participant
rights most mentioned were related to a need for control over
future use, with a focus on individual consent. In a survey of
public perspectives on genomic data sharing with responses
from 36,268 individuals across 22 low-, middle-, and
high-income countries, the right to withdraw one’s data was
ranked highly as a measure to increase trust (second to
information on who would benefit from the data sharing) [85].
Information on Technical Aspects
Many raised the issue of technical information, such as measures
taken to protect the data (Table 2, row 4). Fewer stakeholders
wished for information about data linkage, how data are shared,
and information security.
Information on Scientific Aspects
Some members of the public and patients suggested that
information regarding the nature of the research would be
welcome with a focus on general objectives [64] and benefits
for the self or society [33,37,85,99,134] (Table 2, row 5).
Stakeholders occasionally raised the issue of communicating
measures taken to ensure data accuracy or validity [75,79,96].
Specific Content
Overview
Table 3 presents the major types of specific content that emerged
from our review. Of note, when the comments concerned
specific content, they emphasized the motivation for receiving
such content. This was different from the comments on generic
content that focused more on details around the type of content;
hence, the difference in headers for the second column of Tables
2and 3.
By far, the most frequent elements cited in the literature for
specific information were related to the return of results from
individual tests (Table 3, row 2) and the sharing of one’s data
(Table 3, row 4).
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Table 3. Types of specific content: motivations and examples of stakeholders’ perspectives.
Exemplary citations of different stakeholders extracted from reviewed studiesMotivation for receiving this contentType of spe-
cific content
Information
updates on •“I’d probably just want to be told that the study had expanded a little bit... that it was
something different. Yeah, to keep everything above board. I would still say go ahead
•To remain informed
[68,110,131,138,142]
the use of and use it, but... provided that the patient is aware” (patient [68]).
•To remain engaged
[68,78,87,129,140,144]one’s data •“Concerning genetic data, all interviewees thought that next-of-kin should be informed
about the fact that post-mortem genetic data analysis is taking place and be given the[48,51,68,83,95,•As a form of reciprocity
[83,117,145]105,110,131,138,
140-145]choice to be contacted about findings with potential relevance for their own health, if
no prior preferences had been reported by the deceased” (Bak et al [54] on the views
of patients and family members).
•“Well, it is all about giving and taking. You are giving information about yourself,
about your state of health, in the end intimate details. And in return I want something
back [...]” (member of the public [83]).
Information
on results •“I never heard any results. Our specimens [are] just being kept, being used however
they might. What I would like to see is if specific tests are run. I would like to know
•For follow-up on one’s health
[51,54,70,98,127,144,146,147]
from individ- the results” (research participant from an Indigenous community [144]).
•As a form of reciprocity
[41,78,83,118,146]ual tests [41,•“I thought it would be great if I could delve into the relationship between my... geneal-
ogy and my cancer” (patient [146]).51,54,64,69,70,•As a condition for participation in
research [80,83,148]78,80,83,98,•“To participate in a study where you get specific results would be very, for me, very
positive. It would make me feel that I am contributing more instead of being lumped118,119,127,140,
144,146-154]•To mitigate concerns about privacy
[119] into this mass of people” (patient [146]).
Information
on global re- •“[...] I think if I was to take part in anything like this I’d like to be able to see how the
research was actually being used and its effects within society and how it’s helping
•For transparency and to increase
trust [36,49,53,77,78,89,97,
105,118,128,143,144]search re- people; that would be quite important for me to get something back” (biobank participant
sults of [83]).
•To help individuals or communities
make informed choices about theirprojects that •“When asked directly if they would like to be informed about the outcome of a data-
sharing project in which they are participating, almost 100% of the respondents (99.7%)used one’s health [47,50,78,92,127,129]
data [41,47, answer positively” (Courbier et al [76] on the views of patients with a rare disease).
•As a gesture in return for participa-
tion [51,53,54,76,81,118,143,157]49,50,53,54,69,•“Yes, okay, you’re going to share your data, but now we want you to share the results,
positive or negative... One of the conditions for say getting our data is you have to share76-78,80,81,92,•To share successes and increase re-
search efficiency97,105,118,127,it and that shareable thing can be shareable with the public as well” (Parent of participant
128,135,138,in a pediatric repository [138]).[41,77,83,105,138,143]
143,144,146,
155-160]•• “It makes you feel like... what you’ve done is helpful and meaningful” (biobank partic-
ipant [157]).
If individual results are not avail-
able [146]
•“I’d like to know the results and whatever the issue is, how we can help communities,
how we can help one another. Just what the next steps are. Where do we go from here?”
(research participant from an Indigenous community [144]).
•“I mean, I would prefer linked because obviously there’s personal interest there, but if
it’s done without that then I’d still be interested in the overall results” (patient with
cancer [146]).
Information
on the shar- •“I don’t need to manage it but do want to know who and when they check my file. That
way I can decide whether grant access or not” (patient [45]).
•To retain control over future use
[45,67,73,75,81,83,88,99,100,113,118,
119,121,143,155,159,165]ing of one’s •“Knowing what they’re doing or what they’re planning to do. To know exactly what
everything [is that] they’re doing… and when and how it’s been used. Because, like Idata [11,45,•To know who can access or has ac-
cessed the data [63,74,80,161]48,51,60,62,63,said, because it’s her genes, her stuff—you know” (parent [155]).
67,73-75,78,•“One participant said they would need to know, ‘exactly who, where, [and] how my
information will be used [...]’” (Franklin et al [80] on the views of a patient with cancer).
•To enact ongoing consent
[11,48,60,95,105,140,141,143]80,81,83,86,
88,95,99,100,•“I’d like to be notified anytime anybody accesses my medical records. Even if it’s my
primary care physician... I’d either be notified through email or whenever you log on...105,113,115,
118,119,121,When you log on, you should be able to see a list of everybody who’s accessed your
135,140,141,file. If it’s electronic, you’d be notified if they’re trying to access something that’s more
143,155,158,
159,161-166]confidential” (patient [74]).
•“I think however they plan to [share the data]—they should inform so that you know
what they are doing, and [where] it’s going to go—any method that they use” (member
of the public [63]).
•“Yeah well, I feel if it’s confidential it’s confidential... and it’s anonymous, so… I
suppose maybe I’d prefer to know personally... but then if you never know it is going
to be released then it’s not going to bother you. But personally, I would prefer to know”
(patient [161]).
•“I would like to be asked because if I think it’s important and it can help some sick
people to be healed, yes, there’s no problem. But if I see that it’s not relevant and that
it could be a bit of anything, then I might refuse” (member of the public [11]).
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Exemplary citations of different stakeholders extracted from reviewed studiesMotivation for receiving this contentType of spe-
cific content
•“If they collect the data, you should have some sort of report. You could say when
something is missing” (member of the public [79]).
•To be able to analyze one’s data
[74,111,129,167]
•To be able to verify one’s data
[44,53,60,79,85,109]
•To be able to withdraw data
[85,168]
Information
on how to
access one’s
data [44,53,
60,74,79,85,
99,100,109,
111,129,167,
168]
Information Updates on the Use of One’s Data
Patients and members of the public proposed that information
updates be sent at the time of secondary use of their data (Table
3, row 1). Some referred to a need for control over future use,
whereas others saw the ongoing relationship as a path to
engagement or as a form of reciprocity. Of note, the need for
information and control appeared prominently in certain groups,
including racial and ethnic minorities [18,38], people who have
lived through experiences of exclusion [169], and members of
Indigenous communities [78,92,144].
Information on Results From Individual Tests
Across the literature, patients and members of the public alike
expressed a strong expectation for being informed of results
from tests conducted in research contexts (Table 3, row 2), even
when it only concerned a marker for disease susceptibility [56],
an indication of increased or decreased risk [153], exploratory
(for example microbiome) research [127], and even when not
performed in a certified laboratory [148]. This expectation was
often merged with the request of the right to access one’s clinical
data, which is outside the scope of the secondary use of health
data for research. Clinicians and researchers, perhaps more
cognizant of the distinctions between results that are part of a
diagnostic process and nonvalidated research results, were less
unanimously in support of the return of results [127,156,170].
In a cross-sectional study of the general Dutch population,
participants wanted to receive both individual and aggregated
results, with a preference for the former [69]. Notwithstanding
this important preference for the return of individual results,
many underlined the associated challenges, including the
interpretation of results by the individual participants
[127,146,152], and to a lesser extent, the risk of breach of
confidentiality [150]. In addition, other factors seemed to
influence preferences regarding the return of individual results,
such as whether the medical condition was rare [76]; whether
a health professional would be available to explain the results
[69]; whether it had been agreed upon at the time of initial
consent [81,91,163] (eg, at time of broad consent in the context
of a biobank); whether there would be sharing of data with third
parties [97], in particular those belonging to nonregulated,
nonresearch settings [81]; and whether there might be logistical
hurdles or excessive use of resources [146,152,170]. Indeed,
many biobank participants may be primarily driven by altruistic
motives and do not wish to allocate excessive resources to the
return of results [83]. A participant in the study by Richards et
al [157] stated the following:
Because the most important thing is to find, um, is
the research itself. That’s the most important thing.
So, to me, getting updates on what’s going on is a
nice to have, but it’s not a must have. [Biobank
participant]
On the other hand, the return of results seemed to mitigate
privacy concerns for other biobank participants [119].
Information on Global Research Results
The return of global research results of projects that used one’s
health data (Table 3, row 3) was another important theme for
several stakeholders, including members of Indigenous
communities. Return of global results, whether positive or
negative [138], feeds interest, fosters trust, and can permit
individuals and communities to take more informed decisions.
Participants from Indigenous communities underlined how
providing information would be the way to gain and maintain
their communities’ trust in research [78,89,144]. It was also
seen as a gesture of gratitude in return for participation.
Interestingly, in a survey of 80 Dutch researchers involved in
biobank research, 23% disagreed in part or completely with the
statement: “Participants have to be informed about aggregate
research results” [156].
This opinion was echoed by 81.6% of patients with
cardiovascular disease who responded to a survey distributed
by the European Heart Network [135].
Information on How to Access One’s Data
Information on how to access one’s data (Table 3, row 5) was
not frequently mentioned in the context of governance but
seemed to be increasingly mentioned in recent years by members
of the public in relation to a need to take control of one’s health,
ensure data accuracy, and exert some control in the context of
novel health data uses [44,79,85] or sharing of genomic data
[155]. In the context of data mining, Watson and Payne [44]
stated the following:
Structures to allow individual access are required to
address inaccuracies in the data and to provide a
sense of fairness and comfort in knowing that there
is some recourse to address problems.
How to Communicate?
Overview
Stakeholders proposed several characteristics of communications
to ensure that they were effective (Table 4).
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Table 4. Characteristics of communications: description and examples of stakeholders’ perspectives.
Exemplary citations of different stakeholders extracted from reviewed studiesDescriptionCharacteristic of
communication
Frequency [11,
18,33,36,38,49,•“The most appropriate approach would be to design consents and notices that are
like that as well—real-time, updated, frequently communicating with you and letting
•Periodically, on an ongoing basis
[33,36,38,74,76,78,79,81,140,156,
171,172]59,74,76,78,79, you know not only how your data is going to be used and how it will be protected
81,100,105,125,
140,156,171-173]privacy and security wise... I think a consent information type notice should happen
regularly [and] keep you engaged in understanding the continued use of this data”
(regulatory expert opinion regarding unregulated mobile app research [81]).
•Linked to health care encounters
[18,49,59,100,125]
•Upon request [78,105]
•“At least once a year. If nothing else, you know what is going on” (patient from a
US Veterans Affairs facility [77]).
•“Maybe every half a year, or maybe even once a month [...]. It would be good every
six months to get follow-up information” (2 members of the public [79]).
•“I’d like to be notified anytime anybody accesses my medical records. Even if it’s
my primary care physician... I’d either be notified through email or whenever you
log on... When you log on, you should be able to see a list of everybody who’s ac-
cessed your file” (patient [74]).
•“What does the quantity look like? I mean, if we are getting 10 emails a day, we
might get annoyed” (member of the public [11]).
Associated sup-
port [11,19,32,•“They [care providers] all use very strange words and it’s in one ear and out the
other, and then when you get home, you have forgotten. But now, you can check
•Support that is electronic
[73,74,76,78,81,88,143,180]
36,40,41,43, again and you can look it up on the internet” (patient in conditions of vulnerability
•Support that is delivered by mail
[32,59,78,158]45-48,50-53,56, [175]).
59,68,69,73,74,•“I like how it’s (the electronic version) broken up so it’s easier to read. It’s less in-
timidating upon first glance than a packet of paper” (patient [73]).
•Support that is face-to-face
[43,59,79,83,139,160,175,177]76,78,79,81-83,
85,86,88,90,92,•“If someone is telling face-to-face, it’s easier to motivate or convince the person.
But if it’s some odd papers, sometimes you just skip the part that you didn’t need”
•Support that is delivered through
technology [48,52,152]95,100,103,105,
108,114,123,127,(member of the public [79]).
•Support that is delivered tradition-
ally [41,43,47,59,105]129,132,133,138,•“Having a website is cost-effective because if people are interested they can go on
it and have a look; if they're not then they don’t have to. Leaflets and things like139,141,143,150-153,•Support that is delivered through
social media [88,108,132,172]155,158,160,165,
171,172,174-183]that, I think, are expensive and unnecessary because 95 per cent of them will just
end up at the bottom of a bird cage” (member of the public [105]).
•Support that is delivered through
emails [138,158,171]•“Well then you’d feel that you were doing something that was very worthwhile
wouldn’t you, you’d think you were part of it, instead of just wondering what’s going
•Support that is delivered through
newsletters [69,83,171] to happen, even a website we could come on and just see what’s happening, to keep
us updated” (biobank participant [83]).
•Support that is delivered through
academic and health institutions •“[I]f there’s sort of a portal to a web-based feedback that’s easy for physicians to
use in the little time they have during the day, that would be good” (oncologist fa-[40,85,108,132,141]miliar with rapid learning systems [123]).
•Support that is delivered through
health [11,19,32,40,45,50,56,59,•“For me, it [consent portal]’s a must because it’s kind of a control thing. I would be
able to see who’s using it and why” (member of the public [11]).68,69,76,82,83,100,123,143,150-153,
155,165,174,176-178,183] or re- •“And if it works for me, I can point [to] my other nephews and cousins and everybody
else and say: ‘Go try this out. Go see these people.’ Because I want to be a spokesmansearch [123,177] professionals
and peers and I will say, you know, ‘This is what works. This is how I combat this or that.’
[51,76,92,129,132,143,153] And I’d have an avenue to say, ‘Hey, go try that program out’” (patient from an In-
digenous community [92]).
Examples of technological supports
include the following: •“[...] To perceive leaflets as light reading while awaiting their appointments: ‘People
pick them up and read them while they are waiting and them put them back’” (patient
•Patient portals
[114,123,158,172,175][59]).
•“The problem I have is that not everybody has a cell phone. Not everybody has access
to electronics, and probably the people who are most underserved are those people.
•Public databases [53,158]
•Websites
[41,53,69,83,88,90,127,138,141,181]Probably the socioeconomic group odds are they don’t have money to buy these fun
things, or they don’t have the education to be able to use them. So they’re left in the
•Web-based FAQasection [88,103]dark, and they’re probably the ones that are most easily taken advantage of” (oncol-
ogy patient [114]).
•Short videos
[11,36,103,132,174,179]
•Mobile apps [73,86]
Examples of physical support included
the following:
•Posters [36,95,123,176,181]
•Flyers [46,53,59,132,133,176]
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Exemplary citations of different stakeholders extracted from reviewed studiesDescriptionCharacteristic of
communication
•“If someone can answer, ‘Here’s where it’s stored, here’s how we use it’in simple
ways, not this 30-page agreement. Very simply [...]” (early adopter of eHealth
technology [167]).
•“I sort of wish that I could provide some second bulleted point of one page that was
like, ‘In plain language this is what you've just agreed to’” (researcher [127]).
•“The average person might not understand the meaning of all of [the results] so
[results] would have to be returned in a format they can relate to” (researcher [127]).
•Brief [127,160,167,174,176]
•Interpreted
[55,127,175,176,184,185]
•Layered to permit little or more
information [33,56,73,91,103,127]
Format [33,55,
56,65,72,73,91,
103,116,127,160,
167,174-176,184,185]
•“Researchers should state exactly what is being done with data and make it simple
for people to understand” (citizen council member [93]).
•“This [Health Care Information Directive] is too busy, it’s too much. If I’m sick, I
friggin’don’t want to be bothered with it... Look at this. English is my first language.
How would somebody whose mother tongue is something other than English? [sic]
It’s too complicated” (senior citizen [100]).
•“In terms of getting the information out to Alaskan Native people, just providing
this in a very clear manner about what it is, what it means, what it can do for our
system, what it can do for them individually. So, I think that, again, transparency is
really huge” (health care provider [92]).
•In accessible, plain language
[44,46,56,65,72,73,76,79,81,
87,92,93,99,100,103,105,111,
114,127,130,132,134,139,160,167,
173,175,176,188]
•Attention to levels of literacy
[11,32,56,131,172,176,187]
•Use of mother tongue
[51,70,92,188]
•Attention to special needs
[101,143,176]
•Attention to cultural competency
[51,92,131,186]
•Attention to tone [54,103]
•With explicit language
[39,65,81,103,187], use of visual
supports [103,176] and examples
[103,176]
•Impartial and uncensored [31]
Wording [11,31,
32,39,44,46,51,
54,56,63,65,70,
72,73,76,79,81,
87,92,93,99-101,
103,105,111,114,
127,130-132,134,
139,143,160,167,
172,173,175,176,
186-188]
aFAQ: frequently answered questions.
Communication Approaches That Are Ongoing and
Varied
To raise awareness and education, continuous communication
methods (Table 4, row 1) as well as citizen forums were deemed
appropriate strategies by all stakeholders, including patients
and members of the public, as the best ways to reach all citizens
“[...] before they actually become patients” [42]. For generic
content for the purpose of informing with regard to a particular
data set or data use or for specific content, mixed strategies with
recourse to traditional media, social media, websites, patient
portals, phone lines, posters, and flyers were proposed, again
with the intention of reaching as many individuals as possible
(Table 4, row 2). There was a concern that certain groups of
citizens may be excluded if web-based communication is the
only approach used [73,105,114]. Others favored information
on demand as a cost-effective way for individuals to obtain the
information needed [105]. Some underlined the virtue of
centralizing the information to avoid being overwhelmed by
the amount of information on the web [33,105,129]. Bernaerdt
et al [175] outlined obstacles to the use of e-portals by
“susceptible patients” and proposed recommendations for their
design, an initial face-to-face encounter, and ongoing education.
Frequency
There was no consensus on the appropriate frequency, but the
concept of continuous or periodic communication was frequently
raised, with some practical suggestions to link to other
communications and health encounters (Table 4, row 1). Overby
et al [41] found that 51% of respondents stated that regular
updates increased their likelihood of participating in a biobank
[41].
Associated Support
Many stakeholders emphasized providing support around the
communications (Table 4, row 2). Examples of institutional and
technological support were proposed but the need to be able to
converse directly with a person, whether a peer, a health
professional, or a research coordinator, was deemed important.
Overall, stakeholders, including the European Commission
[203], encouraged person-centered approaches to
communications [51,74]. Rake et al [179] presented a model of
personalized consent flow as a starting point to meet all
requirements for sharing personally collected and controlled
health data for research [179]. In the context of biobanking,
Dirks et al [78] emphasized the importance of not making
assumptions regarding preferences about communication but
to “[…] instead consider using communication strategies that
use iterative inquiry to learn about and engage communities in
which they [researchers] wish to conduct research.”
Format
Ideal formats of communications were deemed to be brief,
accessible to all, notwithstanding impairments such as visual
handicaps, and included an interpretation of results (when
applicable) (Table 4, row 3). Of note, 43% of blind or low-vision
respondents to a national survey exploring the views of persons
with disabilities about participation and barriers to participation
in precision medicine research agreed with the statement,
“information about medical research is not accessible to me”
[101].
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In the context of eHealth, it was suggested that we need to move
away from traditional terms and conditions that are infrequently
read by users. The latter were reported to not be written in a
way to engage people as it often does not use basic language or
sufficient concise content, but at the same time, not explicit
enough on certain aspects considered essential by the
participants [65,167]. An interesting example involving layered
communication was proposed in the context of communicating
with patients about software to enhance privacy in secondary
database research involving record linkages [103]. This strategy,
which uses expandable text and on-demand definitions, allows
to provide information to those who want it while reducing
on-screen text for those who feel overwhelmed [103].
Wording
Stakeholders suggested considerable advice on the question of
wording (Table 4, row 4). Nelson et al [176] proposed a whole
framework to communicate health data that emphasizes the
importance of plain language devoid of medical jargon and
provide contextual information to assist interpretation.
Communications should be explicit and consider special needs,
native language, cultural competencies, tone, and level of
literacy. Some have suggested dedicated resources to a phone
line for direct access to information and explanations [53].
Patients [103] and other stakeholders [176] proposed the use of
visual aids and inclusion of examples. Of interest, we came
across a single stakeholder (member of a citizen jury) who raised
concerns about the choice of certain terms and their legal
implications [97].
When to Communicate: Situations Where
Communication Becomes Necessary
Overview
When discussing the secondary use of health data, stakeholders
often referred to situations in which they considered
communication necessary (Table 5). The 4 categories of
situations that could or should trigger a communication, reaffirm
themes that emerged around the types of information desired
(Tables 2-3) and foreshadow, to a certain extent, the ideal format
(Table 4).
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Table 5. Situations where communication becomes necessary: description and examples of stakeholders’ perspectives.
Exemplary citations of different stakeholders extracted from reviewed studiesDescriptionSituations
Before the reuse of
data [40,53,59,
67,74,80,81,86,
91,97,108,138,
144,145,152,158,
166,167,189-192]
•• “The most appropriate way is to inform the patient every time their data moves to the
researcher or moves for a purpose and give them a chance to opt out or opt in each
time. It may not be the most ideal for the company, but it’s much more ideal for the
patient” (expert opinion regarding unregulated mobile app research [81]).
To exert control on the sec-
ondary use of one’s data
[40,67,81,91,152,189,190,192]
•When the reuse involves the
private sector
[53,81,97,158,191]•“I don’t like it [one-time broad consent]. That’s just me because I mean it’s just like
you sign the form once and you never see it again and then later on in life it ends up
biting you in the ass cause well you signed the form once and you never saw it again.
But someone goes out and dusts off your records and says, ‘Hey look here.’ I’m like,
‘Well Goddam I guess I did sign it.’ And you can’t do anything about it. There’s no
option” (patient [40]).
•When a minor participant
reaches the age of majority
[67,189]
•When the data are sensitive
[53,59,74]•“I’m down with that... People can do whatever they want with our data... But what
you’re trying to tell me is you’re now doing research that will put my name back on
the data I gave you. In a way, you’re not just doing research on my data. You’re doing
research on my data that will add data to my data that I didn’t give you for a reason”
(early adopter of health technology [167]).
•In the context of public health
emergencies when consent is
not required [108]
•“In an ideal world I would include that a company, when they share and sell the data,
would need to have a site that users could access to see with whom their data has been
shared” (patient advocate regarding unregulated mobile app research [81]).
When individual
results become
available [51,54,
56,69,81,83,91,
98,144,193]
•• “Yes, definitely. The reason that this [cardiac arrest] doesn’t bother me anymore in
daily life is that the blood clot was taken out and they explained to me what had hap-
pened. What had gone wrong in my body. I could see it clearly on the monitor during
the catheterisation. So you finally know what it was that made you feel unwell. That
was really nice. So in ninety percent of the cases I’d say, ‘tell me everything you can
find about me, please’” (patient survivor of cardiac arrest [54]).
Results that are actionable
[81,83,91,144] or not [56]
•Incidental findings with clini-
cal relevance [54,193] or not
[193]
•“Well, I was pleased with it and I’m a bit like, that’s one of the incentives for me to go
in for I was interested to know how well I was and I was also interested to know about
my cholesterol as well because my father had really, really high cholesterol and I’ve
never had mine done, so I thought, ‘well, that’s a way to find out what mine is’” (biobank
participant [83]).
•“I would have liked to have had a more detailed summary than we actually got. I think
there were other things that they could have given and, for example, had there been
any major medical problems I think it would have been good if they’d have pointed
those out at some stage or other” (biobank participant [83]).
•“It may not be a guarantee that this will happen, but one of the key issues in a disease
such as this... is early identification and spurring people to action. Melanoma is probably
one of the cancers that kills a lot of people, I would imagine because they aren’t aware
of it and don’t act early enough... So, if he has an algorithm that’s more than 50% accu-
rate, it’s imperative that he let the individuals be aware” (patient advocate regarding
unregulated mobile app research [81]).
•“If that gentleman thinks he’s the carrier for something and he’s not, he needs to know
that” (genetics specialist on the subject of false paternity [193]).
When new global
research results be-
come available
[76,78,98,129,
144,156,158,186]
•• “[...] A lot of the researchers were always promising verbally that they were going to
share the information with you and... more than half the time they never see that the
results of the data after they leave your community. That’s part of the reason why a lot
of the Natives in small communities don’t trust the researchers” (member of an Indige-
nous community [144]).
To maintain trust and commu-
nity engagement [144,186]
•Research conclusions may
influence a person [98,129]
or a community’s actions
[76,186]
N/Aa
In the occurrence
of a breach in con-
fidentiality
[52,73,97,164]
•“If my health information is compromised, how will I be informed?” (patient [73]).
•“If you do the wrong thing, you should face the consequences... then maybe they won’t
do it again” (citizen jury [97]).
•“Certainly, there is reportability back to the Institutional Review Boards [IRB]. There
is probably reportability back to our audit committee of our board, which oversees
compliance in Health Insurance Portability and Accountability Act [HIPAA] at the
very minimum, depending if it’s identified... I mean, it could go all the way out to no-
tification...Certainly notification to the patient if it's identified, but also perhaps the
government agencies” (institution compliance officer [52]).
aN/A: not applicable.
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Before the Reuse of Data
In the context of data sharing, many patients (67%) believed
that they should be informed before their data were reused
[86,192], and this feeling was enhanced when the data were
viewed as sensitive [53,74] (Table 5, row 1). Sharing data with
the private sector raised concerns [119,194]. In the context of
Canadian citizens participating in a week-long dialogue, “[...]
56% of participants indicated that their tissue samples should
never be used or that they must always be asked if profit is
involved” [53]. Another interesting situation is that of minor
participants who may want to be informed of past, current, and
future data uses at the time of their majority [67,189].
When Individual Results Become Available
The return of individual results and the management of clinically
important incidental findings have been largely discussed in the
context of biobanking, but appear important in other contexts,
including administrative databases [54,97], digital data [153],
and mobile apps [81]. Some stakeholders highlighted how these
communications—whether generic when global research results
become available or specific when new individual research
results become available— were deemed important to them as
they would permit them to act on their own health or the health
of their communities (Table 5, row 2). Finally, a few
stakeholders deemed it necessary to communicate in the case
of a breach of confidentiality.
Discussion
Principal Findings
This scoping review presents an extended analysis of the
literature on patients, public, and other key stakeholders’
perspectives on transparency and secondary use of health data.
It reports findings from 178 studies with various designs
published over the past 2 decades. This extensive work is the
first to map the views of a broad range of stakeholders on what
information should be communicated to data contributors and
how and in what situations to ensure transparency with the
secondary use of health data. Figure 2summarizes the findings
of this study. There was a major overlap in the perspectives of
different stakeholders. Some groups expressed opinions that
reflected specific needs, such as data sovereignty in the context
of Indigenous community data or access to results for patients
with rare disorders. Overall, communication was deemed crucial
for many purposes, including educating patients and members
of the public on potential benefits, giving some control over
data use as a form of reciprocity and as a condition to build and
maintain trust. Elements that should be communicated include
generic content, such as governance and regulatory frameworks,
scientific aims, potential future uses of the data, and specific
content that is relevant to each person with regard to the use of
their data. Regarding methods of communication, broad
approaches were generally favored, such as nationwide publicity
campaigns, mainstream and social media for generic content,
and mixed approaches for specific content, including websites,
patient portals, and face-to-face encounters. Patients and
members of the public rarely specified on whom the onus for
these communications would fall, but we can imagine a shared
responsibility according to intent and means between
governmental, national, health care organizations, other large
data custodians, and, to a lesser extent, individual researchers.
Content should be customized to the individual as much as
possible with regard to the length, avoidance of technical terms,
cultural competence, and level of detail.
Figure 2. Summary of the scoping review’s findings.
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Interpretation of Findings
At this moment, in Western societies, sharing both generic and
specific content seems necessary to achieve the goals of raising
awareness, creating and maintaining trust by respecting the
moral concerns of donors [110], and addressing the specific
information needs of populations that have experienced racism
and research violations [166,186,190]. Raised awareness and
education are fundamental for members of the public to
understand the legitimacy of various secondary uses of health
data [58,79,134,163,194,195]. However, the data suggest that
the current knowledge deficit is important [196,197]. In the
international survey by Milne et al [85], providing transparent
information about who will benefit from access to genomic data
was the most important measure for increasing trust. There is
an ongoing perceived need for control over the secondary use
of health data and an aspiration for greater equity in the
science-public relationship [31]. These findings confirm the
conceptual framework by Brall et al [147] of individual
willingness to participate in personalized health research and
emphasize certain elements. The framework identified 20
elements pertaining to attitude, motivation, utility of results,
data sharing and use, data management, and data governance
that can have an impact on individual willingness. All these
elements are presented in our review. Their relative importance
could evolve with society’s greater understanding of data,
measures taken to make their reuse secure, and confirmation of
the benefits of secondary use. Achieving the objective of
transparency needs to address its various facets whether it is
“informational transparency” defined by Aitken et al [31] as
“requiring disclosure of information on which decisions are
based,” “participatory transparency” defined as “enabling public
participation in decision-making processes,” and “accountability
transparency” defined as “decision-makers are held
accountable.” Any step toward greater communication and
transparency should be carefully considered to ensure relevance
and quality and decrease the risk of negative backlash [204].
A narrative review of the literature highlights the fact that social
licenses for data-intensive health research cannot be assumed
[205]. Trust and its absence had a considerable impact on the
acceptance of the secondary use of health data without specific
consent [136,191,198]. Muller et al [206] emphasized the merits
of using the concept of social license as a reference for ethical
governance and highlighted the strategic importance of dialogue
with patients and members of the public. Research is ongoing
to define what secondary uses of health data are within a social
license and how perspectives could change with greater
transparency [207]. On the basis of 8 focus groups with
Canadian members of the public, Paprica et al [194] concluded
that “[r]esearchers and organizations that hold health data should
engage with members of the public to ensure that research is
aligned with social license, particularly where there is
private-sector involvement.” These findings are also reflected
in a similar study of deliberative focus groups conducted with
United Kingdom citizens [208]. Furthermore, we would do well
to not assume and rather gather more evidence to support the
idea that transparency and trust will necessarily go hand in hand
[20].
Reciprocity was also a transversal theme in this review and is
likely a foundation on which to build this common
understanding with different communities and populations.
Indeed, Sexton et al [34] linked trust and reciprocity and raised
the concern that “[f]aceless processes of governance are
increasingly foregrounded over traditional relational bonds.”
Hobbs et al [83] explored the privacy-reciprocity connection,
whereby participants attached certain conditions to their research
donations. Patient engagement is another form of reciprocity
that could increase trust [31]. Finally, reciprocity from the
private sector appeared to be an important condition for sharing
health data [136,191].
The return of individual results, particularly if perceived as
potentially actionable, was a commonly cited example of a
specific communication that patients and members of the public
wanted, and this was substantiated by other research [209,210].
For some but not all, it was a motivation to donate health data,
including genomic data [199]. What is less clear is whether, in
the absence of the return of individual results, the need for
information could be satisfied by communication on aggregated
or global research results.
We also tried to distinguish whether individuals wished to
receive specific communication about the secondary use of
information by a single research project or by a group of projects
with similar features. However, that distinction was not clearly
present in our analysis. It could be interesting to confirm that
an intermediate level of information is linked to the category
of projects (eg, academic research, research with genomic data,
and research with a private partner) that may or is using the data
could meet individuals’need for information without excessive
use of resources [192]. This would also align well with a
metaconsent model, a promising dynamic approach to
information and consent in the context of LHS [11].
There will be no one size fits all—some individuals wanted
regular communications by varied means and at specific times,
for example, each time their data were used, whereas others
wished for basic information to be available on demand so that
resources can be allocated elsewhere. Individual factors,
including age, ethnicity, gender, education, general and scientific
culture, curiosity, and motivations to participate in research,
were key to explaining the variety of views on the subject
[56,59,83,151,158,166]. Biobank participants, for example, may
have a more altruistic outlook and a reduced need for the return
of information than members of the public at large who are less
aware of the risks and benefits of research. It will also be
important to pay attention to the “unheard voices” of people
who are marginalized in society [169]. Public deliberation is
an interesting approach to better understand specific population
needs and preferences for notifications about data sharing [114].
Any communication strategy will need to be patient-centered
and modifiable so that low information seekers are not
overwhelmed by unwanted communication, and high
information seekers can equally meet their needs [11,69].
Communications should also be adapted to the needs of people
with disabilities, especially considering that technology is
creating an information accessibility gap as it tends to replace
face-to-face communication [101]. Interesting models of
communication include the frequently asked questions section
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that provides layered information so that an individual could
obtain as much or as little information as desired [103] and the
OPT-IN framework by Nelson et al [176] “[...] to help guide
communicators with selecting and presenting data to lay
audiences, taking into account the broader communication
perspective.” Guidelines on how to discuss the protection and
use of personal health information would also be welcome [59].
Findings in Relation to Broader Context
There are other noteworthy initiatives to help us pave the way
forward and find a balance between information needs, trust,
and societal benefits from the secondary use of health data [200].
We would like to highlight 4 key features: patient or public
engagement, harmonization of policies, the importance of
user-centered informational tools, and innovations in data
security.
First, transparent governance and patient engagement are
increasingly being proposed as important avenues toward social
license. Paprica et al [87] proposed the minimal specifications
for data trust. Participatory governance is a key concept, as
exemplified in the Academic Research Network of Diabetes
Action Canada [72], the Research Data Alliance COVID-19
Working Group guidelines [201], and in 1 of the 7
recommendations from the work by Courbier et al [76] with
patients with rare diseases. Participatory governance is also
necessary to respect the Principles for Indigenous Data
governance [201].
Second, the adoption of coherent policies and practices can help
harmonize different initiatives and reduce risks, such as privacy
breaches [44]. National or institutional standards such as
Quebec’s Fonds de Recherche Standards for Data Access
Centers [211], New Zealand’s integrated data infrastructure
[166], and the United Kingdom’s data registry standard [212]
are a few examples of initiatives that can serve as models.
Third, educational tools will be required to raise awareness
among all stakeholders, with a focus on members of the public
and tailored to specific populations. Such large-scale initiatives
could address the frequently expressed need for information to
help individuals control their use of data. Indeed, studies have
demonstrated that once informed of regulatory safeguards,
presented with conditions around use [6], informed of impacts
[213], or sensitized to logistic hurdles, opinions regarding
individual consent and control can shift.
Fourth, we should build on existing communication strategies.
The Health Care Information Directive is an example of a patient
decision aid that aims to delineate the level of health information
that an individual is willing to share [100,214]. Caine et al [74]
derived 6 implications for the design of a patient-centered tool
to allow individual choices in the disclosure of health data from
patient interviews. In their systematic review of the design of
patient aids, Vaisson et al [215] proposed a user-centered design
framework that could help design future aids to track the
secondary use of one’s health data. The proposed concept
interface enables contextual control by allowing patients to set
their privacy levels in the context of viewing events within an
electronic health record. Fagerlin et al [216] proposed 10
pragmatic recommendations to help patients make decisions
when faced with complex information on risks and benefits that
could be applied to making decisions about data sharing. On a
more theoretical level, Zikmund-Fisher [217] proposed a
standard taxonomy of risk concepts that could inform or evaluate
future communications. Riso et al [70] outlined a framework
with 6 core values to improve the ethical standards of
data-sharing platforms. For example, the GA Registry [218] is
patient-led and permits large-scale data sharing while letting
users decide which data sets to share and with whom. Exemplary
projects and patient portals include the tailored, ongoing
communication of the CHRIS longitudinal study [202],
NIH-funded All of Us Research Program [219], Connect Care
(Canada) [220] that allows bidirectional interaction with the
patient, Sundhed (Denmark) [221], and Health data hub(France)
[222].
Finally, ongoing innovations in data security will be important
to enhance privacy and trust in all these initiatives. Examples
include CrowdMed, a blockchain approach proposed by Shah
et al [180] and MiNDFIRL, a software to enhance privacy in
secondary database studies [103].
Limitations
Our search yielded most articles originating from North America
and Europe and documenting Western contexts and attitudes;
therefore, our review may not entirely represent other societies.
In addition, although we cited research conducted with
stakeholders from Indigenous communities, we cannot conclude
that the views collected are representative of all Indigenous
communities. Furthermore, many earlier articles focused on
research conducted in the context of biobanking. We restricted
our analysis to articles that explored stakeholder perspectives
with a focus on the future use of samples or data. During
thematic analysis, we were able to conclude that many of the
perspectives overlapped with research conducted in other
contexts, such as administrative databases, data mining, and
mobile app. This suggests that for the person concerned by the
secondary use of health data, there are similarities between the
research contexts of biobanking and newer contexts.
Conclusions
This scoping review highlights the diversity and extent of patient
and public expectations regarding information on the secondary
use of health data. It also attests how and in which situations
the communication of different types of information could
contribute to the objective of greater transparency. These
findings suggest that governing bodies should actively invest
in widespread and targeted activities to increase public
awareness and understanding about the secondary use of health
data. Modern societies would do well to foster a culture of
information and health literacy, which in turn is necessary for
individual empowerment in the context of health care. Indeed,
our results highlight the importance of patient, citizen, and
community engagement in different secondary uses of health
data [104]. This is particularly true for some populations such
as Indigenous communities that have developed their own
models of data governance [223].
Future studies should consider building on existing models to
develop thoughtful strategies for communicating with the
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secondary use of health data. This review did not identify
resources specifically considering the LHS context; therefore,
the impact of such a framework on the desires of patients and
members of the public remains to be explored. To support the
implementation of LHSs in Quebec (through a transparency
portal), the next steps would be to validate some of the results
with patients and members of the public, including the
following: (1) the need for both types of content (generic and
specific); (2) the level of generic content to meet information
needs and foster and sustain trust; and (3) the resulting most
appropriate format considering resource use and development
of web portals to support LHSs [224]. Other research could also
validate how we can match actual citizen and patient needs with
congruent types of knowledge and informational tools (Table
2 in the study by Zikmund-Fisher [217]), and how the
personalization of communications meets the demands of
different regulatory bodies, for example, the recommendations
of the European Commission [203]. Another important question
is whether transparency leads to unexpected outcomes, such as
desensitization and “systematic exploitation” [111]. Finally,
these strategies should be evaluated to ensure that they reach
their objectives, notably with regard to transparency and even
improving health outcomes at the individual and system levels.
Beyond just informing members of the public on the secondary
use of their data, we should aim to foster a culture in which all
citizens are given tools to participate in their health care and
determine research priorities [139].
Acknowledgments
The authors would like to express their sincere thanks to Professor Daniel Caron and Emmanuel Bilodeau for their critical review
of the manuscript draft. They also want to thank Dominique Wolfshagen for creating Figure 2, Timothey Bédard for his important
work in the coding of the articles, and Denis Boutin and Karina Prévost for their thoughtful and wise recommendations as patient
partners in the research program. This work was supported by J-FE personal fundings.
Data Availability
The data generated and analyzed during this study are available from the corresponding author upon reasonable request.
Authors' Contributions
AC, J-FM, AB, RD, and J-FE participated in the identification of the research questions, conception and design of this scoping
review, and data analysis and interpretation. RD, FL, AC, and J-FM participated in the study selection, data extraction, and
interpretation. AC drafted the paper, but all authors critically revised it for important intellectual content. All authors granted
final approval for the version to be published and take accountability for all aspects of the work.
Conflicts of Interest
None declared.
Multimedia Appendix 1
PRISMA-ScR Checklist.
[DOCX File , 79 KB-Multimedia Appendix 1]
Multimedia Appendix 2
Research strategies.
[DOCX File , 24 KB-Multimedia Appendix 2]
Multimedia Appendix 3
Characteristics of the articles included in the scoping review.
[DOCX File , 62 KB-Multimedia Appendix 3]
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Abbreviations
LHS: learning health system
PRISMA-ScR: Preferred Reporting Items for Systematic Reviews and Meta-Analyses extension for Scoping
Reviews
Edited by A Mavragani; submitted 12.12.22; peer-reviewed by D Willison, J van Delden; comments to author 25.01.23; revised version
received 09.02.23; accepted 03.03.23; published 13.04.23
Please cite as:
Cumyn A, Ménard JF, Barton A, Dault R, Lévesque F, Ethier JF
Patients’and Members of the Public’s Wishes Regarding Transparency in the Context of Secondary Use of Health Data: Scoping
Review
J Med Internet Res 2023;25:e45002
URL: https://www.jmir.org/2023/1/e45002
doi: 10.2196/45002
PMID: 37052967
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©Annabelle Cumyn, Jean-Frédéric Ménard, Adrien Barton, Roxanne Dault, Frédérique Lévesque, Jean-François Ethier. Originally
published in the Journal of Medical Internet Research (https://www.jmir.org), 13.04.2023. This is an open-access article distributed
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Medical Internet Research, is properly cited. The complete bibliographic information, a link to the original publication on
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