Patient-driven innovations reported in peer-reviewed journals: a scoping review.

BACKGROUND: Awareness of patients' innovative capabilities is increasing, but there is limited knowledge regarding the extent and nature of patient-driven innovations in the peer-reviewed literature.
OBJECTIVES: The objective of the review was to answer the question: what is the nature and extent of patient-driven innovations published in peer-reviewed scientific journals? ELIGIBILITY CRITERIA: We used a broad definition of innovation to allow for a comprehensive review of different types of innovations and a narrow definition of 'patient driven' to focus on the role of patients and/or family caregivers. The search was limited to years 2008-2020. SOURCES OF EVIDENCE: Four electronic databases (Medline (Ovid), Web of Science Core Collection, PsycINFO (Ovid) and Cinahl (Ebsco)) were searched in December 2020 for publications describing patient-driven innovations and complemented with snowball strategies. CHARTING
METHODS: Data from the included articles were extracted and categorised inductively.
RESULTS: A total of 96 articles on 20 patient-driven innovations were included. The number of publications increased over time, with 69% of the articles published between 2016 and 2020. Author affiliations were exclusively in high income countries with 56% of first authors in North America and 36% in European countries. Among the 20 innovations reported, 'Do-It-Yourself Artificial Pancreas System' and the online health network 'PatientsLikeMe', were the subject of half of the articles.
CONCLUSIONS: Peer-reviewed publications on patient-driven innovations are increasing and we see an important opportunity for researchers and clinicians to support patient innovators' research while being mindful of taking over the work of the innovators themselves. © Author(s) (or their employer(s)) 2022. Re-use permitted under CC BY-NC. No commercial re-use. See rights and permissions. Published by BMJ.

To our knowledge, this study is the first attempt to perform a comprehensive review of what has been published in peer-reviewed journals about patient-driven innovations.

The review had a systematic approach in searching four large databases, complemented with snowball sampling.

Patient-driven innovations are not always labelled in the research as patient-driven innovations and as such, despite the methods used including snowball sampling, the result is likely an under-representation of research of patient driven innovations.

The study contributes to concepts and operational definitions related to patient innovations, pointing out the result of using an inclusive and broad definition of innovation and a narrow definition of what is meant by patient driven.

Background

Traditionally, patients have been considered as passive recipients of medical care, merely ‘buying’ and consuming the services and products that experts (eg, researchers, healthcare professionals, ‘medical producers’) have created.1 However, healthcare providers are increasingly regarding patients as experts in their own conditions, involving them and their family caregivers as active participants in care.2–4 Although most policies promote a more active patient role in care, research has found that in reality, patients’ role remains limited.5 6 Patients repeatedly report having too little influence over their care while their needs remain unmet in the current healthcare systems.7 8

Many patients want to play a greater role in decisions about their care, to perform more effective self-care, and to engage in improving healthcare systems.3 4 For example, patient innovators take part in the development and spread of patient-driven innovations.9–11 The Patient Innovation website (www.patient-innovation.com), which was created to collect innovations by patients and/or family caregivers, lists over 1200 innovations. This gives an indication of significant activity by patients and their family caregivers driving health innovations, often independently of the health system. Patient innovators have been defined as ‘patients or their nonprofessional caregivers (eg, parents and family members), who modify or develop a treatment, a technical aid product, or a medical device to cope with a health condition’.7 Although the awareness of patients’ innovation capacity is increasing, there is still limited knowledge regarding the extent and nature of patient-driven innovations in the peer-reviewed literature.7 12 In the discussion section of this paper, we consider possible explanations for this. The objective of the review was to answer the following research question: What is the nature and extent of patient-driven innovations published in peer-reviewed scientific journals?

Method

Design

A scoping review method was chosen as the most appropriate for the objective of the study because our initial investigations revealed a diverse range of types of studies and publications, and the method is recommended as useful when examining emerging areas of research.13 It was performed according to the five-stage framework proposed by Arksey and O’Malley13 and is reported according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses Extension for Scoping Reviews (PRISMA) Checklist14 (online supplemental appendix 1). Critical appraisals of articles were not performed since the aim was to explore the extent and nature of patient-driven innovations, not assess the quality of these. The review does not follow a preregistered protocol.

Definition of key concepts

To find a shared understanding of the concept ‘patient-driven innovation’, the research team performed a concept analysis inspired by Walker and Avant.15 The concept analysis was performed in a collaborative workshop with patient innovators and researchers where we used key articles to identify model cases of patient-driven innovation, and borderline cases, related cases and contrary cases. Using the concept analysis method, the team agreed to define ‘innovation’ based on the WHO definition of health innovation as ‘an innovation that identifies new or improved health policies, systems, products and technologies, or services and delivery methods that improve people’s health and well-being. The innovation aims to add value in the form of improved efficiency, effectiveness, quality, sustainability, safety and/or affordability. The innovation can be preventive, promotive, curative, rehabilitative, assistive and/or palliative care.’ The other part of the concept was ‘patient driven’ that we agreed consisted of two parts and was defined by: (1) The innovation is user driven, meaning that it is both initiated and driven (in development, implementation, etc) by patients and/or family caregivers and (2) The innovation responds to one or more unmet needs of the innovator. Unmet needs are defined by the innovator. This provided a definition broad enough to allow for a comprehensive review of the nature of patient-driven innovations but limited ‘patient driven’ to focus on the role of patients and/or family caregivers (see table 1).

Table 1

Eligibility criteria for inclusion

Inclusion criteria	Exclusion criteria
1. English language	Other language than English
2. Published between January 2008 and December 2020	Published earlier than 2008 or later than 2020
3. Published in a peer-reviewed journal	Not published in peer-review journal
4. Reporting on patient-driven innovation(s) as defined by three criteria:4.1. Based on WHO’s definition of health innovations the innovation identifies new or improved health policies, systems, products and technologies, or services and delivery methods that improve people’s health and well-being. The innovation aims to add value in the form of improved efficiency, effectiveness, quality, sustainability, safety and/or affordability. The innovation can be preventive, promotive, curative, rehabilitative, assistive and/or palliative care.4.2. The innovation is user driven, meaning that it is both initiated and driven (in development, implementation, etc) by patients and/or family caregivers.4.3. The innovation responds to one or more unmet needs of the innovator. Unmet needs are defined by the innovator.	Article is out of scope (context other than healthcare)No innovation describedDescribed innovation is not patient-driven
5. The innovation is the focus of the article	The innovation is used for data collection but not described in the article

Eligibility criteria

Eligibility criteria are presented in table 1. We included studies published in peer-reviewed journals (publication years 2008–2020) that covered health innovations initiated and driven by patients and/or family caregivers (hereafter referred to as patient-driven innovations, as defined in table 1, point 4.1–4.3.). Review articles were used to identify original articles, and review articles that presented original data not presented elsewhere were included. We limited the review to reports and publications made from the year 2008 and onwards because our initial searches found few reports or publications about patient innovations before 2008. Articles where the innovation (primarily the online platform PatientsLikeMe) was used solely for data collection were excluded.

Information sources

Four electronic databases were searched in October 2019 and the search was updated in December 2020: Medline (Ovid), Web of Science Core Collection, PsycINFO (Ovid) and Cinahl (Ebsco). We also employed snowball sampling: (1) The webpage www.patient-innovation.com was screened for names of innovators and innovations and those names were used to search records in PubMed (January 2020); (2) Reference lists of included articles were screened (August 2020) and (3) Names of identified patient innovators were used for author search in Web of Knowledge (August 2020).

Search

Key articles on patient-driven innovations were used by MR to form a search strategy in consultation with librarians at the Karolinska Institutet University Library. The search strategy was tested and refined three times to ensure that all key articles were identified. A complete search strategy for Web of Science is presented in table 2 and for all databases (Medline (Ovid), Web of Science Core Collection, PsycINFO (Ovid) and Cinahl (Ebsco) in online supplemental appendix 2.

Table 2

Search strategy used in MEDLIN

Interface: Ovid MEDLINE(R) and Epub Ahead of Print, In-Process & Other Non-Indexed Citations and Daily Date of Search: 15 October 2019 Number of hits: 3443 Comment: In Ovid, two or more words are automatically searched as phrases; that is, no quotation marks are needed		Field labels · exp/=exploded MeSH term /=non exploded MeSH term ti, ab, kf.=title, abstract and author keywords adjx=within x words, regardless of order *=truncation of word for alternate endings
#	Searches	Results
1	((adult child* or patient* or caregiver* or caregiver* or carer* or family or husband* or “next of kin*” or partner* or spouse* or user or wife or wives) adj1 (directed or driven or driving or initiated)).ti, ab, kf.	4150
2	(co creat* or co design* or collaborative creation* or collaborativ* created or “do it yourself” or “doing it for themselves” or diy or e patient* or lead patient* or participatory design* or public driven or superuser*).ti, ab, kf.	3444
3	Patient participation/	24 568
4	or/1–3	31 910
5	Equipment design/	144 620
6	Inventions/	1720
7	Organisational innovation/	23 978
8	Diffusion of Innovation/	17 239
9	(innovat* or invention* or invented).ti, ab, kf.	128 952
10	Self-Management/	1492
11	exp Self-Help Devices/	11 126
12	exp Self care/	52 825
13	(assistive technolog* or co care or self care or self help device* or self management* or self monitor* or self track*).ti, ab, kf.	42 096
14	or/5–13	388 764
15	4 and 14	3235
16	(co innovat* or patient* innovat* or patient design*).ti, ab, kf.	219
17	15 or 16	3443

Selection of sources of evidence

Records were screened by six authors (MR, AB, HJ, SR, HH and CW) and two research assistants (see the Acknowledgements section) in the open-source software Rayyan,16 according to eligibility criteria. To screen the large number of records identified at this stage, we first collected and applied the selection criteria to titles and abstracts of the papers discovered in the search. When abstracts were not available, we retrieved the full paper to decide if the selection criteria were met or not met to carry forward to the next stage of the review.

Screening was blinded and a minimum of two researchers conducted the screening for each article. Inclusion/exclusion decisions were compared. In 7% of the cases, researchers disagreed about inclusion/exclusion, and these conflicts were resolved by consensus through discussion among authors based on a full-text screening.

Data charting process and data items

A template for data charting in Microsoft Excel 2013 was developed iteratively by all authors, who worked in pairs to extract data. Extractions were compared within the pairs by those who extracted the data and merged by MR.

The final extraction form included items on the characteristics of the studies (journal, publication year, first author country of affiliation, publication type, study aim, study design, data and sample size), and on the innovations (name of innovation, name of innovator(s), description of innovation, unmet needs that the innovations aimed to fulfil, and medical condition). Extracted data is published in online supplemental appendix 3.1–3.3.

Analysis of review findings

MR performed an initial overview of the extracted data and proposed preliminary categories for each data item. PM, CW, CS, HH, AB and HJ worked in pairs with sorting the data according to suggested categories and refined the categories and suggested additional categories when needed. Detailed description of the categorisation of data ispresented in online supplemental appendix 4.

Patient and public involvement

This study was performed within the auspices of the cocreated research programme ‘Patients in the driver’s seat! A multimethod partnership programme on patient-driven innovations’, where patient innovators are engaged as equal partners in research. The research programme members were engaged in the research meetings and contributed to the research questions, definition of patient-driven innovations and selection of sources of evidence (see the Acknowledgements section). Furthermore, SR, a patient researcher17 living with Parkinson’s disease, coauthored the current paper and was involved in all stages of the process.

Findings

The systematic search generated 7220 records after duplicates were removed; the snowball sampling generated 559 additional records. In total, 7629 records were screened by title and abstract and 414 records were screened in full text for eligibility. Of these, 96 articles on 20 patient-driven innovations were included. The study selection process is reported in a PRISMA flow diagram (figure 1).

Figure 1

PRISMA 2009 flow diagram. PRISMA, Preferred Reporting Items for Systematic Reviews and Meta-Analyses.

The nature of patient-driven innovations

The 20 identified innovations addressed the unmet needs of patients and family caregivers with diabetes (7 innovations, 46 publications18–63); cancer (1 innovation, 1 publication64); rare diseases (3 innovations, 5 publications65–69); gastrointestinal diseases (2 innovations, 4 publications70–73); disabilities (2 innovations, 3 publications74–76); Parkinson’s disease (2 innovations, 3 publications77–79) and mental illness (1 innovation, 2 publications80 81). There were also innovations targeting unmet needs of multiple conditions (2 innovations, 32 publications82–113). Data by innovation are presented in table 3.

Table 3

Innovation-specific information

Medical condition	Innovation	N articles	Innovator(s) mentioned in the article(s)	Type of innovation	Innovator(s) listed as author
Cancer (breast cancer)	Jacki Jacket	1	Cathy McGrath	Patient clothing	No
Diabetes	Autosens	1	Dana Lewis, Scott Leibrand	Technical innovation for diabetes care	Yes
	Autotune	1	Dana Lewis, Scott Leibrand	Technical innovation for diabetes care	Yes
	DIYAPS, Do-It-Yourself Artificial Pancreas System	21	#wearenotwaiting-community, Dana Lewis, Adrien Tappe, Bastian Hauck, Tebbe Ubbe, Saskia Wolf, Timothy Omer	Technical innovation for diabetes care	Yes
	MySugr	2	Fredrick DeBong	Mobile app	Yes
	Nightscout (CGM in the cloud)	3	#Nightscout movement, specific innovators not reported	Technical innovation for diabetes care	Yes
	Omnipod	2	John Brooks III	Technical innovation for diabetes care	Yes
	T1resources.uk	1	Mike Kendall	Online network	Yes
	Webdia	1	Jean-Luc Mando	Mobile app	Yes
Disabilities	Upsee	2	Debby Elnatan	Wearable devise	No
	(No name)Auditory stimulation	1	Debby Elnatan	Auditory stimulation	Yes, last author
Gastrointestinal diseases	Ostom-i-Alert	3	Michael Seres	Technical	Single author or not author
	(No name) Helminth therapy	1	Sean Ahrens	Alternative treatment	Single author
Mental illness	No name (self-tracking number of hallucinations)	2	Spencer Roux	Technical innovation for self-tracking	Yes
Multiple	PatientsLikeMe	29	Jamie Heywood, Benjamin Heywood, Jeff Cole	Online network	Yes in some
	Medistory	1	Olive O’Connor	pen and paper medical journal	Yes
Parkinson’s disease	No name (A painted staircase)	2	Mileha Soneji	Paint on floor, optical illusion	Yes
	No name (a smartphone app for collecting data on drug intake and well-being)	1	Sara Riggare	Mobile app	Yes
Rare diseases (22q11 deletion syndrome)	(No name) patient driven collaborative initiative	1	Anne Lawlor	Collaborative network	Yes
Rare diseases (cystic fibrosis (CF))	Upstream dream, Genia	2	Andreas Hager	Mobile app	Yes
	Sweden CF Coalition	2	Andreas Hager	Collaborative network	Yes

DIYAPS, Do-It-Yourself Artificial Pancreas System.

Many of the innovations involved digital technologies, with four mobile apps, four collaborative networks, five technical innovations for diabetes care and one sensor that measures output volume from one’s ostomy. Other innovations included one jacket with pockets for postoperative drain tubes, one pen-and-paper form for personalised medical records, a painted staircase (optical illusion) to prevent gait freezing, and ingestion of pig-worms to improve symptoms from Crohn’s disease. Among the 20 innovations reported, the Do-It-Yourself Artificial Pancreas System (DIYAPS) and PatientsLikeMe.com, an online health-related social network, accounted for half of the articles.

Extent of publications

Author affiliations were exclusively in high income countries with 56% of first authors with affiliations in North America, followed by 36% from European countries (see table 4). One first author had their affiliation in Asia and six in Oceania. The number of publications increased in later years, with 69% of articles published 2016–2020 (figure 2). A majority (65%) of the studies were published in journals with a scope of general or specialised medicine, with some in journals focused on research or healthcare processes (12%), or journals focused on digital health (10%). One article was published in a journal focused on patients’ health. About half of the studies were classified as peer-reviewed research with 38% original articles, 2% short reports, 2% protocols and 6% reviews that published original results. Remaining articles were published in a peer-reviewed journal but in formats that commonly are not peer-reviewed: 24% Letters to the editor/commentaries, 18% published conference abstracts, 6% editorials and 3% in special sections dedicated to patients. Patient innovators mentioned in articles were listed as authors in most, but not all, publications. The articles seldom described the patient innovator’s role in the research process.

Table 4

General characteristics of studies

	N studies (%)
Continent (first author affiliation)
Europe	35 (36)
North America	54 (56)
South America	0
Asia	1 (1)
Oceania	4 (6)
Africa	0
Publication year
2008	3 (3)
2009	2 (2)
2010	3 (3)
2011	4 (4)
2012	2 (2)
2013	2 (2)
2014	5 (5)
2015	6 (6)
2016	10 (10)
2017	10 (10)
2018	15 (16)
2019	19 (20)
2020	15 (16)
Type of journal
General medicine	20 (21)
Specialised medicine	51 (53)
Process related	14 (14)
Digital health	10 (10)
Patient oriented health	1 (4)
Publication type
Original research	34 (35)
Short report	2 (2)
Protocol	2 (2)
Review (presenting original results)	6 (6)
Letter to editor/commentary	25 (26)
Conference abstract	17 (18)
Editorial	7 (7)
Special section dedicated to patients	3 (3)
Study aim
Describe the innovation and/or development of innovation	23 (24)
Describe users and/or how users perceive the innovation	21 (22)
Test effect/impact of innovations	23 (24)
Describe/discuss ethical issues and/or policy change	8 (8)
Test feasibility of innovation	2 (2)
Aim not presented/not relevant	19 (18)
Study design
Descriptive	42 (44)
Observational	11 (11)
Experimental	8 (8)
Design not presented/not relevant	35 (36)
Sample size
1	8 (8)
2–100	21 (21)
101–500	9 (9)
501–1000	2 (2)
>1000	11 (11)
Sample size not presented/not relevant	45 (47)

Figure 2

Number of publications per year.

Aims and design of included articles

Almost half of the studies (47%) used a descriptive design, while smaller proportions used an observational design (15%) or experimental design (9%) and 29% of the articles were categorised as not having a study design, for example, editorials. Sample sizes ranged from one participant (8% of studies) to over 1000 participants (14% of studies). Of the 96 articles included, 77% presented a study aim, and approximately one-third of these had a study aim that focused on describing or testing the innovation: 24% aimed to describe the innovation and/or the development of the innovation, 24% to test the effect and/or impact of the innovation and 2% to test the feasibility of the innovation. Other articles aimed to describe characteristics of users of the innovation and/or describe how users perceived the innovation (22%). A small proportion of the articles described and discussed ethical issues and/or or policy changes relating to patient-driven innovations (8%).

Discussion

This article reports a scoping review of publications about patient-driven innovations in peer-reviewed journals. The review identified 96 articles published from year 2008 to 2020, reporting 20 different patient-driven innovations and the number of publications increased in the later years. Among the 20 innovations reported, ‘DIYAPS’ and the online health network ‘PatientsLikeMe’ accounted for half of the articles. Considering that over 1200 patient-driven innovations are listed on www.patient-innovation.com, the number of 20 patient-driven innovations published in peer-reviewed journals is remarkably small.

Canhao et al9 point out that the lack of patients scaling up and spreading their innovations to others may be an example of market failure. Based on the potential benefits of patient-driven innovations, actors such as medical product and service producers and government regulators could support patient innovators in the development and diffusion of their innovations.114 We suggest that the lack of patient-driven innovations reported in peer-reviewed journals may also be seen as an academic failure as scientific peer-reviewed journals are important arenas for disseminating, evaluating, improving and discussing ideas in healthcare. The research community has an important part to play in complementing other ways of support for the creative contributions of the patients by using the systematic methods of research to evaluate, develop, and integrate these solutions into patients’ daily lives and healthcare systems. According to Canhao et al,9 there are several barriers for patient innovators that prevent them from sharing their innovations, including lack of resources, skills or access to the process of approval and commercialisation. In this review, only a minor portion of the studies had an observational or experimental design, and it is possible that similar barriers that prevent patient innovators from sharing their innovations apply to research and scientific writing.

Strengths and limitations

The strengths of the study include the broad scope of the review. We followed the process outlined in Arksey and O’Malley13 and the review was guided by a predetermined strategy for data collection and analysis. Methodological strengths lie in this systematic approach to searching the four large databases, complemented by snowball sampling. Earlier research has reviewed specific patient-driven innovations, for example, a review of ‘DIYAPS’ by Kesavadev et al,115 or investigated characteristics of patient innovators.7 This is the first review undertaken of patient-driven innovations, according to a broad and comprehensive definition, and one of the very few undertaken of innovations in which patients have played a significant role in development of the innovation. This builds on Oliveira et al’s definition of patient innovations where innovation is limited to ‘a treatment, a technical aid product, or a medical device’.7 We cocreated a broader definition together with patient innovators in order to include social innovations such as collaborative or social networks. Thus, this broader scope and definition of patient-driven innovations was able to capture more innovations in which patients have played a significant role in the development.

The limitations of the study include the choice to only select peer-reviewed articles, but this was motivated by our aim to explore the proliferation of patient-driven innovations within the scientific literature. If the purpose had been to create an inventory of patient-driven innovations, the inclusion of grey literature would have yielded more results. Furthermore, the source of information was restricted to the included articles and in some cases the webpage wwwpatient-innovationcom. Patient-driven innovations are not always labelled as such in the publications. Therefore, despite the broader definition and the use of snowball sampling and online searches to identify the drivers of innovations, our results are likely an under-representation of research on patient-driven innovations. Also, as patient-driven innovations may initially go through commercialisation processes with a shift of ‘drivers’, it is possible that we would relabel some innovations as not being patient driven if we had access to more information.

Unanswered questions and future research

As this field of research is relatively new, there are several unanswered questions for future research. Considering the potential benefits that patient-driven innovations can have if they become widely used, it will be important to understand factors that may facilitate or hinder implementation, spread and scale-up of patient-driven innovations; none of the included articles in this review systematically examined these questions. It may also be important to gain deeper understanding of patient-driven innovations in general, what unmet user need they address, how they are used and by whom and what outcomes they have for patients and healthcare systems. A further unanswered question is what determines whether patient innovators decide to publish their results and if so, in which journal(s). Patient innovators were often listed as coauthors in publications related to their innovations, there was a broad variation in type of publication, and it was common for patient innovators to coauthor articles together with established researchers and/or clinicians. Some patient innovators presented their innovations in single authored papers and others were not listed as authors. A suggestion for future studies is to interview or survey patient innovators who publish in peer-reviewed journals and explore how their reasoning around research collaboration and publishing.

This review was restricted to innovations, and we acknowledge that we, in the study selection, have excluded studies published by patient researchers if the studies were not related to patient-driven innovations. For further research we suggest a review of literature published by patient researchers and citizen scientists in general.

Conclusions

Peer-reviewed publications on patient-driven innovations are increasing and peer-reviewed journals constitute an arena where patient-driven innovations can be evaluated, discussed and developed further. We see an important opportunity for researchers and clinicians to support patient innovators’ research and publication while being mindful about not taking over the work of the innovators themselves.