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Information and communication technology (ICT) has made remarkable progress in recent years and is being increasingly applied to medical research. This technology has the potential to facilitate the active involvement of research participants. Digital platforms that enable participants to be involved in the research process are called participant-centric initiatives (PCIs). Several PCIs have been reported in the literature, but no scoping reviews have been carried out. Moreover, detailed methods and features to aid in developing a clear definition of PCIs have not been sufficiently elucidated to date.
The objective of this scoping review is to describe the recent trends in, and features of, PCIs across the United States, the United Kingdom, and Japan.
We applied a methodology suggested by Levac et al to conduct this scoping review. We searched electronic databases—MEDLINE (Medical Literature Analysis and Retrieval System Online), Embase (Excerpta Medica Database), CINAHL (Cumulative Index of Nursing and Allied Health Literature), PsycINFO, and Ichushi-Web—and sources of grey literature, as well as internet search engines—Google and Bing. We hand-searched through key journals and reference lists of the relevant articles. Medical research using ICT was eligible for inclusion if there was a description of the active involvement of the participants.
Ultimately, 21 PCIs were identified that have implemented practical methods and modes of various communication activities, such as patient forums and use of social media, in the field of medical research. Various methods of decision making that enable participants to become involved in setting the agenda were also evident.
This scoping review is the first study to analyze the detailed features of PCIs and how they are being implemented. By clarifying the modes and methods of various forms of communication and decision making with patients, this review contributes to a better understanding of patient-centric involvement, which can be facilitated by PCIs.
RR2-10.2196/resprot.7407
The use of information and communication technology (ICT) is increasing in all aspects of health care delivery and medical research, enabling vast amounts of data to be accumulated and analyzed at an unprecedented rate. Furthermore, a growing number of people are participating in research using smart devices, such as smartphones, tablets, and wearable devices [
In parallel, attitudes toward medical research are also shifting to more active involvement of research participants. This emerging model of research, in which researchers and participants collaborate through the research process, is gaining momentum internationally. Several research funders, such as INVOLVE in the United Kingdom, which was established by the National Institute for Health Research (NIHR) to support public involvement in the National Health Service (NHS), and the Patient-Centered Outcomes Research Institute (PCORI) in the United States, are known as leading organizations promoting the involvement of research participants [
The definition of patient and public involvement and engagement has not yet been established. There is no designated terminology to describe the active involvement of participants in research. According to INVOLVE,
In this review, we use the definition from INVOLVE [
Participant-centric initiatives (PCIs) are new initiatives that employ ICT for facilitating active involvement in research and are defined as “digital tools, platforms, or projects that have been developed to help participants become more actively involved in the research process” [
While examples and features of PCIs have been reported by Anderson et al [
By applying a scoping review methodology, we examined PCIs that have been implemented in the United States, the United Kingdom, and Japan, and we systematically analyze their detailed functions and features. The reason for selecting these countries for the study was that the United States and the United Kingdom have been actively advancing patient-centric approaches in medical research, and the majority of reported PCIs in previous research was located in these two countries [
Therefore, the aims of this study were (1) to identify existing PCIs used for medical research in the United States, the United Kingdom, and Japan, (2) to describe recent trends and features of PCIs, and (3) to highlight the methods of participant involvement facilitated by PCIs.
The methodology followed here is based on the previously published study protocol [
According to the features of PCIs described by Anderson et al [
In this study,
We first conducted a literature search in June 2017 in the following databases: MEDLINE (Medical Literature Analysis and Retrieval System Online), Embase (Excerpta Medica Database), CINAHL (Cumulative Index of Nursing and Allied Health Literature), PsycINFO, and Ichushi-Web. We conducted our search by using subject headings and keywords. Search terms included a combination of keywords and subject headings such as Medical Subject Headings (MeSH). Keywords were comprised of the following terms: “participant” AND “centric” OR “centered” OR “centred” OR “engage” OR “involve” OR “collaborat” OR “partner” OR “led” OR “driven” OR “initiat” OR “oriented.” Subject headings for “participation,” “technology,” and “research” were searched and adjusted to best meet the requirements of each database. The detailed search strategies and history are shown in
A grey literature search was also conducted using Open Grey in December 2017, and a website search was conducted using Google and Bing from April to June 2018. The grey literature and website searches were conducted by using the same search keywords as in the literature database search. Some searches showed a large number of items, for example, more than 100,000 hits. However, as it was practically challenging to identify relevant websites by screening all items, we took a pragmatic approach and screened websites that had appeared within the first 50 results for each keyword.
The relevant articles and websites were screened based on the inclusion criteria described in
Inclusion criteria:
Research enables participants to become actively involved in the research design
Complies with participant-centric initiative (PCI) features described by Anderson et al [
Articles, documents, or websites published in English or Japanese
Adult population (ie, over 18 years of age)
Focuses on medical research purposes
Available to participants in the United States, the United Kingdom, or Japan
Exclusion criteria:
Platforms that enable patients to connect and communicate with other patients only
Platforms that use data for research, but there is no interaction between participants and researchers
Research activity directed and conducted by citizens without the support of scientists
Research intended to improve the efficiency of clinical practices or to develop tools for health care services
Medical research that aims to engage with participants without using a digital platform
The features of PCIs were extracted and mapped from the relevant articles and information on the websites of each PCI. The data extraction was completed by NH, and the preliminary results were reviewed by research study members to ensure validity. Characteristics to extract included the following: characteristics of PCI websites, type of medical research, and method of involvement. The list of data elements that were extracted are shown in
The PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) flowchart template [
A consultation is recommended as an optional stage in conducting a scoping review [
The search results and screening process are shown in
In the database search, 10 relevant articles [
The grey literature and website searches were conducted in an iterative process reflecting the results of the database search that had been performed earlier. In total, 3622 documents and websites were screened, but none described the implementation of PCIs for medical research. However, 16 additional PCIs were identified by a supplementary search of 2 excluded articles [
Flowchart for study selection. MEDLINE: Medical Literature Analysis and Retrieval System Online; Embase: Excerpta Medica Database; CINAHL: Cumulative Index of Nursing and Allied Health Literature; PCI: participant-centric initiative.
Participant-centric initiatives (PCIs) identified by our scoping review.
No. | Name of PCI | Location | Type of organization | Areas of focus | Launch year | Number of usersa |
1 | 23andMe [ |
United States | Industry | Diverse (more than 230) | 2006 | >1,200,000 |
2 | PatientsLikeMe [ |
United States | Industry | Diverse (more than 2800) | 2006 | >600,000 |
3 | PEERb [ |
United States | NPOc | Diverse (about 50) | 2014 | >15,000 |
4 | GenomeConnect [ |
United States | Research institute | Diverse (genetic disorders) | 2014 | 1400 |
5 | RUDYd [ |
United Kingdom | University | Fibrous dysplasia, vasculitis, osteogenesis imperfect, etc | 2014 | 993 |
6 | MoodNetwork [ |
United States | Research hospital | Mood disorders | 2015 | Unknown |
7 | mPower [ |
United States | NPO | Parkinson disease | 2015 | 15,000 |
8 | J-RARE [ |
Japan | NPO | Distal myopathy, relapsing polychondritis, Marfan syndrome, etc | 2013 | ≥47 |
9 | ABOUT Network [ |
United States | NPO and university | Hereditary breast cancer | 2016 | 10,500 |
10 | Arthritis Power [ |
United States | NPO | Rheumatoid arthritis, fibromyalgia, inflammatory bowel disease (IBD), etc | 2014 | 15,365 |
11 | IBDe Partners [ |
United States | NPO and university | Crohn disease | 2011 | 15,680 |
12 | Rare Epilepsy Network (REN) [ |
United States | NPO | Rare epilepsy | 2014 | 1392 |
13 | COPD PPRNf [ |
United States | NPO | Chronic obstructive pulmonary disease and asthma | 2014 | 75,000 |
14 | Health eHeart [ |
United States | University | Cardiovascular diseases | 2013 | 75,000 |
15 | IANg [ |
United States | NPO | Developmental disorder | 2006 | >20,000 |
16 | iConquerMS (multiple sclerosis) [ |
United States | NPO | Multiple sclerosis | 2015 | ≥3100 |
17 | AD-PCPRNh [ |
United States | Research hospital | Alzheimer disease and dementia | 2014 | 57,000 |
18 | NephCure Kidney Network Patient Registry [ |
United States | NPO | Primary nephrotic syndrome | 2014 | 666 |
19 | PIi CONNECT [ |
United States | NPO | Primary immunodeficiency | Unknown | 5040 |
20 | V-PPRNj [ |
United States | University | Behçet disease, vasculitis, polyarteritis nodosa, etc | Unknown | Unknown |
21 | MyApnea [ |
United States | Research hospital | Sleep apnea | 2013 | 12,677 |
aThe number of registrants (ie, users) is based on information publicly available in July 2018.
bPEER: Promise for Engaging Everyone Responsibly.
cNPO: nonprofit organization; includes patient organizations and research organizations.
dRUDY: Rare and Undiagnosed Diseases Study.
eIBD: Inflammatory Bowel Disease.
fCOPD PPRN: Chronic Obstructive Pulmonary Disease Patient-Powered Research Network.
gIAN: Interactive Autism Network.
hAD-PCPRN: Alzheimer's Disease Patient- and Caregiver-Powered Network.
iPI: Primary Immunodeficiency.
jV-PPRN: Vasculitis Patient-Powered Research Network.
During the database search, it became clear that there were no designated subject headings to describe features of the PCIs within this emerging field; for instance, the existing MeSH do not have terms to include “participant-centric,” “engagement,” or “involvement”; alternatively, “patient participation” was suggested as the only MeSH option. The subject heading is typically used as an effective search tool in a database search. In this case, however, we had to combine keywords to search for articles relevant to PCIs. The lack of effective search terms may reflect the novelty of the field. For the same reason, the results of grey literature and website searches using Google and Bing were also limited.
Many of the documents that were excluded in the screening process were related to patients’ decision making or participation in clinical practice or medical interventions, including test screening, rather than medical research. Another major category of excluded items concerned the digitization of research and health care with the aim of developing tools for services, with no description of the involvement of participants.
After the consultation, all members of the expert panel commented on the obtained results. Overall, they considered the results “important and well worthy of publication” and the search strategy as “sensible.” In addition, the experts gave feedback on the analysis and discussion sections of the paper. The feedback was incorporated into our analysis of results and the discussion.
There were also suggestions on potential PCIs we had not identified through the literature search. A total of 5 potential platforms were suggested by two members of the panel—one from Japan and one from the United Kingdom. Moreover, one member suggested that disease registries were underrepresented in the obtained results. The main reasons that we did not pick up these registries with our literature search is that any scientific papers generated by them appeared after our cutoff point or they did not indicate active involvement of patients in existing papers.
In response, we screened the registries listed on the National Institutes of Health (NIH) website. A total of 62 registries were screened by NH and AK. As a result, we identified 5 PCIs in the United States and Japan, which had not come out in our original literature search. Moreover, an anonymous reviewer suggested an additional PCI during the peer-review process. As we had decided that the results of the literature searches would be our endpoint for this study, these additional PCIs are offered here as supplementary data (see
PCIs have been implemented for research focusing on various diseases, including rare diseases, mood disorders, heart diseases, and dementia. Of the 21 kinds of PCIs identified in this scoping review, 4 (19%)—23andMe, PatientsLikeMe, PEER (Promise for Engaging Everyone Responsibly), and GenomeConnect—had features that included a variety of disease areas and had cross-cutting registry functions ranging from dozens to thousands of diseases. One of them was a direct-to-consumer genetic testing company (ie, 23andMe). In these PCIs, multiple kinds of medical research were being conducted. Furthermore, these 4 PCIs aimed to match the genomic information of research participants with their disease phenotype. On the contrary, the main registration objectives of the other 17 PCIs for medical research concerned specific disease areas; some of them had gradually expanded their disease areas of focus, such as RUDY (Rare and Undiagnosed Diseases Study) and V-PPRN (Vasculitis Patient-Powered Research Network).
Examining the year in which each PCI was established, our results show a dramatic upward trend from 2013 onwards (see
The trend in the number of participant-centric initiatives (PCIs) identified in the scoping review, cumulative total by year. NB: For 2 PCIs, the launch year was unknown.
The medical research conducted using digital tools, such as personal computers and smartphones, had one or both of the following purposes:
Understanding the symptoms: changes in the presentation of symptoms over time, genetic information and disease phenotype matching, daily changes of conditions, and health conditions associated with everyday life, including exercise and diet.
Comparison of treatment effectiveness: a case-control study comparing the effects of interventions, such as comparing medication records with symptoms or the impact of an online exercise class.
We found that at least 277 articles had already been published by several PCI research groups by January 2018, including some in major scientific journals [
Types of data collected by participant-centric initiatives (PCIs).
Types of data | Number of PCIs (N=21), n (%) |
DNA test result | 7 (33) |
Closed-ended questionnaire | 17 (81) |
Open-ended questionnaire | 5 (24) |
Treatment and medication | 16 (76) |
Motion and voice | 3 (14) |
Self-reported measurement | 4 (19) |
Electronic health record | 5 (24)a |
aIn addition to the 5 PCIs that collected electronic health record data, 2 were in preparation.
In total, 3 PCIs out of 21 (14%)—PEER, RUDY, and J-RARE—implemented a
Model of consent implemented in participant-centric initiatives (PCIs).
Types of data | Number of PCIs (N=21), n (%) |
Specific consent | 12 (57) |
Broad consent | 1 (5) |
Dynamic consent | 3 (14) |
Treatment and medication | 16 (76) |
Unknown | 3 (14) |
By examining the activities undertaken by the 21 PCIs, it became clear that various modes of communication had been used, including patient forums, webinars, and dialogue (ie, patients and researchers exchanging messages online) (see
Modes and methods of communication for the participant-centric initiatives (PCIs).
No. | Name of PCI | Patient foruma | Webinarb | Dialoguec | Use of social media for communication | Other modes of communication | |||
YouTube | Other | ||||||||
1 | 23andMe |
|
✓ |
|
✓ | ✓ | ✓ | ✓ | News |
2 | PatientsLikeMe | ✓ |
|
✓ | ✓ | ✓ | ✓ | ✓ | News |
3 | PEERd | ✓ |
|
|
✓ | ✓ | ✓ |
|
MOSAICe |
4 | GenomeConnect |
|
|
Unclear | ✓ | ✓ | ✓ | ✓ | Newsletter and mailing list |
5 | RUDYf | ✓ |
|
|
✓ | ✓ |
|
|
N/Ag |
6 | MoodNetwork | ✓ |
|
✓ | ✓ |
|
✓ |
|
N/A |
7 | mPower |
|
✓ |
|
|
|
✓ | ✓ | Patient satisfaction questionnaire |
8 | J-RARE |
|
|
|
|
|
|
|
Questionnaire |
9 | ABOUT Network |
|
✓ |
|
✓ | ✓ |
|
|
GAP360h |
10 | Arthritis Power |
|
|
✓ | ✓ | ✓ | ✓ |
|
N/A |
11 | IBDi Partners |
|
✓ | ✓ | ✓ | ✓ | ✓ | ✓ | Blog and dashboard for research ideas |
12 | Rare Epilepsy Network (REN) | ✓ | ✓ |
|
✓ | ✓ |
|
|
Dashboard |
13 | COPD PPRNj |
|
|
✓ |
|
|
|
|
COPD360° |
14 | Health eHeart |
|
|
✓ | ✓ | ✓ |
|
✓ | Health eHeart community |
15 | IANk |
|
✓ |
|
✓ | ✓ | ✓ |
|
N/A |
16 | iConquerMS (multiple sclerosis) | ✓ |
|
✓ | ✓ | ✓ | ✓ | ✓ | Newsletter and iConquerMS community |
17 | AD-PCPRNl |
|
|
|
✓ | ✓ | ✓ |
|
|
18 | NephCure Kidney Network Patient Registry |
|
✓ |
|
✓ |
|
|
|
Patient story and regional volunteer community |
19 | PIm CONNECT | ✓ | ✓ |
|
✓ | ✓ | ✓ |
|
N/A |
20 | V-PPRNn |
|
✓ |
|
✓ | ✓ |
|
✓ | N/A |
21 | MyApnea | ✓ |
|
✓ | ✓ | ✓ |
|
|
Online bulletin board, blog, and personalized report |
aIncluded community day, leadership summit, and research forum.
bIncluded content for general use.
cIncluded the sharing of experiences, thoughts, and information with researchers and other patients; networking.
dPEER: Promise for Engaging Everyone Responsibly.
eMOSAIC: Model of Observational Screening for the Analysis of Interaction and Communication.
fRUDY: Rare and Undiagnosed Diseases Study.
gN/A: not applicable.
hGAP: Generate, Assess, Prioritize, Plan, Perform, and Publish.
iIBD: Inflammatory Bowel Disease.
jCOPD PPRN: Chronic Obstructive Pulmonary Disease Patient-Powered Research Network.
kIAN: Interactive Autism Network.
lAD-PCPRN: Alzheimer's Disease Patient- and Caregiver-Powered Network.
mPI: Primary Immunodeficiency.
nV-PPRN: Vasculitis Patient-Powered Research Network.
It became clear that there were various phases in decision making by research participants. First, the 3 PCIs that implemented the dynamic consent model allowed each participant to control the range of data sharing. Most PCIs used an interface that enabled inputs of participant feedback on research and operations or agenda setting by allowing participants to propose new research questions. Furthermore, more than half the PCIs (14/21, 67%) had a governance structure that included participant representatives in the decision-making process of the research design and conduct of the research (see
Decision-making process implemented in participant-centric initiatives (PCIs).
No. | Name of PCI | Data-sharing control | Individual feedback and suggesting research questionsa | Research design and governanceb |
1 | 23andMe |
|
✓ |
|
2 | PatientsLikeMe |
|
✓ | ✓ |
3 | PEERc | ✓ | ✓ | ✓ |
4 | GenomeConnect |
|
✓ | ✓ |
5 | RUDYd | ✓ | ✓ | ✓ |
6 | MoodNetwork |
|
✓ | ✓ |
7 | mPower |
|
✓ |
|
8 | J-RARE | ✓ |
|
✓ |
9 | ABOUT Network |
|
✓ | ✓ |
10 | Arthritis Power |
|
✓ | ✓ |
11 | IBDe Partners |
|
✓ |
|
12 | Rare Epilepsy Network (REN) |
|
✓ |
|
13 | COPD PPRNf |
|
|
✓ |
14 | Health eHeart |
|
✓ | ✓ |
15 | IANg |
|
✓ |
|
16 | iConquerMS (multiple sclerosis) |
|
✓ | ✓ |
17 | AD-PCPRNh |
|
|
✓ |
18 | NephCure Kidney Network Patient Registry |
|
✓ | ✓ |
19 | PIi CONNECT |
|
✓ |
|
20 | V-PPRNj |
|
✓ |
|
21 | MyApnea |
|
✓ | ✓ |
aIndividual comments, suggestions of research questions, decisions of priority, etc.
bThe main purpose is to determine the overall policy as representative of research participants, such as an advisory board, a steering committee, and a governor group.
cPEER: Promise for Engaging Everyone Responsibly.
dRUDY: Rare and Undiagnosed Diseases Study.
eIBD: Inflammatory Bowel Disease.
fCOPD PPRN: Chronic Obstructive Pulmonary Disease Patient-Powered Research Network.
gIAN: Interactive Autism Network.
hAD-PCPRN: Alzheimer's Disease Patient- and Caregiver-Powered Network.
iPI: Primary Immunodeficiency.
jV-PPRN: Vasculitis Patient-Powered Research Network.
In this study, we conducted a scoping review to capture the recent trend and features of PCIs for medical research, in particular, by focusing on active involvement. A total of 21 PCIs were identified by the scoping review. After analyzing the detailed functions and characteristics of each, the landscape of PCIs became clearer. To our knowledge, this is the first scoping review conducted in this emerging area to map the extent and range of PCIs currently available across the United States, the United Kingdom, and Japan.
The PCIs identified in this study were utilized in medical research in various fields. The number of participants registered for each PCI ranged from approximately 50 to 100,000 (see
Another noteworthy trend was the sharp increase in the number of PCIs after 2013. The reasons for the increase may be improvements in technology and greater understanding of the benefits of using digital technologies in health care, as well as an increasing awareness of the importance of engaging with patients. As 12 of the 21 PCIs (57%) were funded by PCORI in the United States, the trend may also have been influenced by the year of its establishment. PCORI was established in 2010 with the aim of funding comparative clinical effectiveness research for patients and those who care for them to make better-informed health decisions [
There are at least 270 scientific papers published that used data collected by PCIs. Out of 21 PCIs, 2 (10%)—23andMe and PatientsLikeMe—are notable because they have published nearly 100 papers in the area between them; both are organized by for-profit organizations. It is necessary to analyze them further to understand the kinds of research papers that are published by these PCIs and why these for-profit organizations seem to have a greater reach than others.
The features of PCIs have been described in a prior paper [
Dynamic consent was implemented in 3 out of 21 PCIs (14%)—PEER, RUDY, and J-RARE—while 12 PCIs (57%) implemented the specific consent model. This result indicates that not all PCIs embedded individual control into the design of the interface.
The use of social media technology, such as information dissemination, was observed in most of the 21 PCIs. This allows participants to see the progress in research and the results. PCIs also generally provide user-friendly platforms that facilitate two-way interaction between participants and researchers and, in some PCIs, between the participants. In more than half of the 21 PCIs, participants were involved in suggesting research questions. These results indicate that PCIs enable participants to play an active role, not only in terms of controlling their own data-sharing settings but also by way of contributing to decision making in research design. They encourage participants to become involved in the agenda setting of the entire research community. A follow-up study is needed to examine the method and evaluation of these involvement activities.
The definition of PCIs was suggested in prior studies in 2012 [
PCIs cultivate an environment to establish collaborative partnerships between participants and researchers. As this
Of the 21 PCIs identified, 19 (90%) were initiated in the United States. There may be a number of possible reasons for this, some of which suggest avenues for future investigation. Firstly, it is possible that the culture of involving patients and members of the public in research is more established in the United States.
Our results indicated that many of the PCIs were funded by PCORI. The Patient-Powered Research Network is one of the research networks that is operated and governed by patient groups, and it has launched a number of projects using online platforms to collect self-reported data [
Furthermore, the differences in the way that health care is organized may be an additional explanatory factor, but this would need further exploration. The US system is rather unique in the way it organizes health care and this may be tied into the more populous PCI landscape in the country. We are aware that patients in the United States are often engaged around social entrepreneurship in health care and play key roles in initiating new research projects [
Future research is needed to disentangle the factors impacting the establishment and sustainability of PCIs in order to better understand the underlying reasons for the differences between these countries.
In this paper, we investigated PCIs, which enable the active involvement of patients, by conducting a scoping review to understand this new field that has not yet been conceptually established. Therefore, there are a number of limitations to this work.
First, due to limitations in the search method, some PCIs might have been overlooked in our search. For example, any PCI-related documents that do not contain our keywords indicating active involvement of patients in the titles or abstracts would have been extremely difficult to detect. This would also hold for the website search results. We suggested that consistent definition and terminology of participant involvement should be established to overcome these methodological limitations. Second, any PCIs that do not publish their research in scientific articles, or those whose websites do not appear in any search engine results, would not have been found. Further, research articles and websites that were published after our search period would also have not been included in our findings.
For future research, PCIs implemented in countries other than those focused on in this study also deserve attention. For instance, other English-speaking countries, such as Canada, Australia, and New Zealand, where there is a push toward patient and public involvement in medical research could be prioritized in future research. Furthermore, it may be desirable to seek a method to evaluate PCIs by a variety of means, such as careful observation, surveys of participant opinions, and exploration of theoretical considerations. Such results will offer insights to further improve ongoing PCIs and aid the establishment of new ones in the future.
We can also propose possible ideas for moving forward the area of PCIs. In this review, we have documented the important activities of PCIs; at the same time, we have noted that there are still only a small number of projects that meet the criteria we have set for patient involvement. Therefore, the future challenge in the field is how to expand the number of PCIs and also the level of patient involvement that these platforms enable. Ideas for improvement can include the following: (1) wider dissemination of information about the value of PCIs for patients and patient organizations, (2) encouragement of networking between PCIs to facilitate the adoption of good practices, and (3) increase of support by government bodies for PCIs.
A scoping review was sufficient to capture recent trends in PCIs designed to facilitate medical research. We identified 21 PCIs currently operating in the United States, the United Kingdom, and Japan. This review contributes to a better understanding of the concept of
The search history of the literature database search.
The list of data elements extracted from relevant articles and information on websites of each participant-centric initiative (PCI).
Detailed characteristics of identified participant-centric initiatives (PCIs).
The list of participant-centric initiatives (PCIs) that had not come out of our original literature search.
Cumulative Index of Nursing and Allied Health Literature
Excerpta Medica Database
Hereditary Angioedema International
internet and communication technology
Interdisciplinary Program for Biomedical Sciences
Japan Society for the Promotion of Science
Grants-in-Aid for Scientific Research
Medical Literature Analysis and Retrieval System Online
Medical Subject Headings
Medical Research Council
National Health Service
National Institutes of Health
National Institute for Health Research
participant-centric initiative
Patient-Centered Outcomes Research Institute
Promise for Engaging Everyone Responsibly
Preferred Reporting Items for Systematic Reviews and Meta-Analyses
Rare and Undiagnosed Diseases Study
US Hereditary Angioedema Association
Vasculitis Patient-Powered Research Network
We thank Mr Toshiyuki Suwa for helping develop our search strategy. We are also grateful to Mr Anthony Castaldo from the US Hereditary Angioedema Association (US HAEA) and Hereditary Angioedema International (HAEi), Mr Simon Denegri from the NIHR, Dr Tiffany Boughtwood from the Murdoch Children’s Research Institute, Dr Mizuki Morita from Okayama University, and Dr Soichi Ogishima from Tohoku University for consultations on the obtained results as the expert panel members. We thank Dr Jessica Bell and Dr Teresa Finlay from the University of Oxford for contributing to the initial study design. We thank Dr Saad Anis from Edanz Group for editing a draft of this manuscript. This work is supported by JSPS (Japan Society for the Promotion of Science) Grants-in-Aid for Scientific Research (KAKENHI) (grants No. JP15K15167 and No. JP17K19812), Osaka University International Joint Research Promotion Program, and the Interdisciplinary Program for Biomedical Sciences (IPBS).
None declared.