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Project Topics Pdf 121453 | S13023 022 02514 3

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                          et al. Orphanet Journal of Rare Diseases          (2022) 17:357  
                    Neff 
                    https://doi.org/10.1186/s13023-022-02514-3
                      RESEARCH                                                                                                                                      Open Access
                    Identifying project topics and requirements 
                    in a citizen science project in rare diseases: 
                    a participative study
                                          1*                        1                        1                     2                     2                          2
                    Michaela Neff    , Holger Storf , Jessica Vasseur , Jörg Scheidt , Thomas Zerr , Andreas Khouri  and 
                    Jannik Schaaf1 
                       Abstract 
                       Background:  Due to their low prevalence (< 5 in 10,000), rare diseases are an important area of research, with 
                       the active participation of those affected being a key factor. In the Citizen Science project “SelEe” (Researching rare 
                       diseases in a citizen science approach), citizens collaborate with researchers using a digital application, developed as 
                       part of the project together with those affected, to answer research questions on rare diseases. The aim of this study 
                       was to define the rare diseases to be considered, the project topics and the initial requirements for the implementa-
                       tion in a digital application.
                       Methods:  To address our research questions, we took several steps to engage citizens, especially those affected by 
                       rare diseases. This approach included the following methods: pre- and post-survey (questionnaire), two workshops 
                       with focus group discussion and a requirements analysis workshop (with user stories).
                       Results:  In the pre-survey, citizens suggested 45 different rare diseases and many different disease groups to be 
                       considered in the project. Two main project topics (A) “Patient-guided documentation and data collection” (20 votes) 
                       and (B) “Exchange of experience and networking” (13 votes) were identified as priorities in the workshops and ranked 
                       in the post-survey. The requirements workshop resulted in ten user stories and six initial requirements to be imple-
                       mented in the digital application.
                       Conclusion:  Qualitative, citizen science research can be used to collectively identify stakeholder needs, project top-
                       ics and requirements for a digital application in specific areas, such as rare diseases.
                       Keywords:  Rare diseases, Citizen science, Patient science, Qualitative research
                    Background                                                                              expertise, and lack of available health services require 
                    In the European Union, the prevalence of a rare diseases                                special efforts to obtain a specific and correct diagnosis 
                    (RD) is defined as affecting no more than 5 in 10,000 per                               and appropriate treatment [3–5].
                                                                                                      -
                    sons. There are more than 6000 known different RDs, of                                     As they face all these difficulties, patients with RDs and 
                                                                                                            their relatives become experts on their disease. There
                    which almost 72% caused by genetic abnormalities [1,                                                                                                                     -
                    2]. Low prevalence, complex symptomatology, limited  fore, even more than for common diseases, it is necessary 
                                                                                                            to recognise them as informed and active project partici
                                                                                                                                                                                             -
                                                                                                            pants [6, 7]. People affected by a RD should therefore be 
                     *Correspondence:  michaelachristina.neff@kgu.de                                        directly involved in research projects, e.g. in Citizen Sci
                                                                                                                                                                                             -
                     1 Institute of Medical Informatics, Goethe University Frankfurt, University            ence (CS) projects, which engage people in the scientific 
                     Hospital, Frankfurt am Main, Germany                                                   process who do not work professionally in this field of 
                     Full list of author information is available at the end of the article
                                                                © The Author(s) 2022. Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which 
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                    Neff et al. Orphanet Journal of Rare Diseases          (2022) 17:357                                                                                    Page 2 of 12
                    research. Muki Haklay describes "participatory science",                             the local area). Those interested in joining the study were 
                    which includes CS, as the involvement of the population                              asked to participate in a pre-survey by completing a ques-
                    already in the formulation of the research question as                               tionnaire in PDF format available on the project website 
                    well as in data collection [8].                                                      (www. selee. de) and returning it by e-mail or letter.
                       The Project ’Seltene Erkrankungen bürgerwissen                                       The questionnaire contained six semi-open and open-
                                                                                                   -
                    schafltich erforschen! (SelEe)’ (engl. ‘Researching rare                             ended questions in German language (Additional file 2), 
                    diseases in a citizen science approach’) is a joint CS pro-                          covering two categories of questions:
                    ject on RDs by the Institute for Information Systems at 
                    Hof University of Applied Sciences (iisys) and the Insti
                                                                                                   -        1.  Questions 1–3 (Q1–3): Background of the interested 
                    tute of Medical Informatics (IMI) at Goethe University                                      person,
                    Frankfurt. The project is funded by the Federal Ministry                                2.  Questions 4–6 (Q4–6): Proposal of diseases and dis-
                    of Education and Research in Germany (BMBF) and sup-                                        ease groups with optional justification and first topic 
                    ported by the Alliance of Chronic Diseases (ACHSE e.V.)                                     suggestions regarding the SelEe project.
                    [9, 10]. In SelEe, scientists and citizens aim to investigate 
                    RDs together by collecting data using a digital applica-                                The survey was conducted over four weeks in August 
                    tion. Citizens can contribute their knowledge and ideas                              2021. Data analysis of the survey was conducted using 
                    directly to the project, formulate requirements, and  Microsoft Excel. To assign participants to expertise in 
                    improve collaboration between all stakeholders—starting                              specific RD groupings (RD, not a RD, unclear), the named 
                    with the initial phase of the project. During this phase,                            disease in Q3 was checked using orphanet nomenclature 
                    the challenges and problems in the daily lives of people                             [12].
                    affected by RDs—patients as well as their relatives (fur
                                                                                                   -
                    ther referred to as ‘RD-affected persons’)—will be iden-                             Focus groups topic definition
                    tified and addressed. In the context of the project, the                             After the pre-survey, two focus groups were conducted. 
                    term citizen also includes any interested non-scientists                             These moderated group discussions were used to engage 
                    with no connection to RDs (further referred to as "inter
                                                                                                   -     citizens in the decision-making process and to collect 
                    ested persons"). The project will initially be carried out in                        and discuss different facets of challenges and topics of 
                    Germany, with the possibility of a gradual international                             RD-affected persons [13–15].
                    expansion.
                       To create a long-term benefit for all RD-affected per
                                                                                                   -     Setting and sampling
                    sons, several steps were taken at the beginning of the                               The participants of the focus groups were selected from 
                    project to identify topics that should be explored and                               those who had completed the pre-survey, based on 
                    implemented. The objectives of this study were to answer                             one of the following inclusion criteria: affected by an 
                    the following questions: (1) which RDs and groups of                                 RD according to the EU-wide definition of RD, has an 
                    RDs should be considered, (2) which topics should be                                 unclear diagnosis, or relative of an affected person. After 
                    investigated for joint research on RDs using a digital                               pre-selection according to the inclusion criteria, partici
                    application, and (3) which requirements for the digital                                                                                                              -
                    application are considered most useful.                                              pants were randomly selected and distributed to the two 
                                                                                                         focus groups until a maximum number of participants of 
                    Methods                                                                              12 persons per focus group was reached [13, 15]. Finally, 
                                                                                                         the participants were invited by e-mail.
                    A multi-step approach was used to answer the research 
                    questions, including the following methods: question
                                                                                                   -     Data collection
                    naires, focus groups and a requirements analysis work-                               Prior to conducting the focus groups, all participants 
                    shop. Figure 1 illustrates the steps of this study, which                            received and signed a consent form and were provided 
                    will be described in more detail in the following sec
                                                                                                   -     with information about the study (including information 
                    tions. The Standards for Reporting Qualitative Research                              about the researchers). The focus groups were performed 
                    (SRQR) guideline was considered for reporting the focus                              online via a video-conference application in October and 
                    groups [11]. A checklist is available in Additional file 1.                          November 2021. Each focus group lasted approximately 
                                                                                                         120 min and was held in German language.
                    Pre‑survey                                                                              A semi-structured interview guide (Additional file 3) 
                    The invitation to the project was disseminated via various                           was developed in preparation for the focus groups. 
                    media by ACHSE e.V. (e-mail distribution list) and the                               In addition, an interactive word cloud online appli
                                                                                                                                                                                         -
                    science communication department of Hof University  cation [16] was used as a stimulus during the discus-
                    of Applied Sciences (websites, newspapers and radio in                               sion (Fig.  2). First proposals for project topics were 
        Neff et al. Orphanet Journal of Rare Diseases          (2022) 17:357 
                                                               Page 3 of 12
        Fig. 1  Steps of the study’s multi-step approach
                    Neff et al. Orphanet Journal of Rare Diseases          (2022) 17:357                                                                                    Page 4 of 12
                                                                                                         Post‑survey
                                                                                                         To identify a final project topic, a post-survey was con-
                                                                                                         ducted. In terms of CS, this survey was conducted as 
                                                                                                         a follow-up questionnaire to the focus group with an 
                                                                                                         expanded group of participants and was thus sent to all 
                                                                                                         citizen (RD-affected persons and interested persons) in 
                                                                                                         the project who had completed the pre-survey (Sect. 2.1), 
                                                                                                         excluding those who had by then revoked their participa
                                                                                                                                                                                         -
                                                                                                         tion in the project.
                                                                                                            The survey was conducted in anonymized form using 
                                                                                                         the online tool LimeSurvey [20]. Repeated participations 
                                                                                                         were ruled out using a dedicated feature of LimeSur
                                                                                                                                                                                         -
                      Fig. 2  Interactive word cloud—which project topics would you like                 vey. The survey was distributed via e-mail in November 
                      to propose?                                                                        2021 and was conducted over two weeks in November/
                                                                                                         December 2021. In the questionnaire, each participant 
                    collected, initially showing suggested topics from Q6 of                             had the opportunity to vote for exactly one project topic 
                    the pre-survey. The word cloud was then interactively                                (Additional file 4). Data analysis for the survey was con-
                    updated and discussed by all participants.                                           ducted using Microsoft Excel.
                       Following a short round of introductions, the dis-                                Requirement analysis workshop
                    cussions during the focus groups were recorded via  After establishing the project topic, a workshop was 
                    audio recording and moderated by two experienced  performed together with RD-affected persons to define 
                    female moderators from ACHSE e.V.. Two research-                                     specific requirements of the digital application for the 
                    ers from the SelEe project team created protocols of                                 implementation of the project topic. In this study, a 
                    the project topic discussion to capture chat notes of                                requirement was defined as a software function that 
                    the participants, visualize the topics in table form  could be used by a user in a software system. The par
                    and prepare them for voting. Subsequently, all par                                                                                                                   -
                                                                                                   -     ticipants of both previous focus groups (Sect. 2.2) were 
                    ticipants were asked to vote on the topics on a scale                                invited as the designated primary user group of the digi
                    of 1 to 3 (1 = "most important", 2 = "very  important",                                                                                                              -
                    3 = "important").                                                                    tal application. The invitation was sent in January 2022 
                                                                                                         via e-mail. All participants again received and signed a 
                                                                                                         consent form and further information before workshop 
                    Data analysis and processing                                                         participation.
                    The audio recordings were transcribed and reviewed                                      The workshop lasted 120 min and started with a short 
                    independently by two researchers using the transcrip                                 presentation on the topic. Afterwards, user stories, visu-
                                                                                                   -     alised with story cards, were collected interactively and 
                    tion system of Kuckartz et al. [17, 18]. The affiliation of                          common requirements were discussed. A user story is an 
                    the statements (participants/moderations team) were                                  informal, general explanation of a software feature writ
                    marked and the statements of the participants were                                                                                                                   -
                    anonymized. The transcripts were not distributed to                                  ten from the end user’s perspective [21]. Participants 
                    the participants for correction or comments. However,                                were advised to share their suggestions using the follow-
                    participants received an anonymized summary of the                                   ing user story template of Mike Cohn [22–24]: As  I 
                    results in German language. A translation of the quota                               want  so that .
                                                                                                   -        The user stories were visualised and documented in 
                    tions was made for the purpose of this publication.                                  Microsoft PowerPoint by two researchers and were vis-
                       Based on the transcript materials, the focus group  ible to all participants. The common requirements were 
                    protocols, and the results of the project topic ranking,                             also noted visibly for the participants and documented 
                    central topics were identified. For this purpose, a content-                         in the researchers’ notes. They were transferred from the 
                    structuring qualitative content analysis [19] was applied                            researchers’ notes into Microsoft Word.
                    to combine the proposed topics from both focus groups 
                    and form categories to represent project topics. The 
                    main categories, including their sub-categories, which                               Results
                    achieved the highest prioritization in the combination of                            Pre‑survey
                    both focus groups (taking into account the average of the                            The pre-survey conducted during participant recruit-
                    voting of topics) were prepared for the post-survey.                                 ment was answered by 69 candidates, with affected 
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...Et al orphanet journal of rare diseases neff https doi org s research open access identifying project topics and requirements in a citizen science participative study michaela holger storf jessica vasseur jorg scheidt thomas zerr andreas khouri jannik schaaf abstract background due to their low prevalence are an important area with the active participation those affected being key factor selee researching approach citizens collaborate researchers using digital application developed as part together answer questions on aim this was define be considered initial for implementa tion methods address our we took several steps engage especially by included following pre post survey questionnaire two workshops focus group discussion analysis workshop user stories results suggested different many disease groups main patient guided documentation data collection votes b exchange experience networking were identified priorities ranked resulted ten six imple mented conclusion qualitative can used c...

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