The dissemination of evidence-based interventions  is a core function of US state health departments (SHDs). However, interventions are originally designed and tested with a specific population and context. Hence, adapting the intervention to meet the real-world circumstances and population's needs can increase the likelihood of achieving the expected health outcomes for the target population from the implemented intervention. This study identified how SHD employees decide to adapt public health programs and what influences decisions on how to adapt them.n = 45) were interviewed using a qualitative semi-structured interview guide. Telephone interviews were audio-recorded and transcribed verbatim. The transcripts were consensus-coded and themes were identified using thematic analysis. Several themes aligned with the Model for Adaptation Design and Impact.SHD employees , and reasons to adapt a program included organizational and sociopolitical contextual factors. SHD middle-level managers, program managers and staff, and local agencies were involved in the decisions to adapt the programs. Finally, the goals for adapting a program included enhancing effectiveness/outcomes, reach and satisfaction with the program; funding; and partner engagement. After SHD employees decided to adapt a program, data and evidence guided the changes. Program staff and evaluators were engaged in the adaptation process. Program managers consulted partners to gather ideas on how best to adapt a program based on partners' experiences implementing the program and obtaining community input. Lastly, program managers also received input on adapting content and context from coalition meetings and periodic technical assistance calls.The findings related to decisions to adapt public health programs provide practitioners with considerations for adapting them. Findings reaffirm the importance of promoting public health competencies in program evaluation and adaptation, as well as systematically documenting and evaluating the adaptation processes. In addition, the themes could be studied in future research as mechanisms, mediators, and moderators to implementation outcomes. In the U.S., state and local health departments deliver essential public health services, including preventing and controlling diseases with population-level approaches . The delEvidence-based interventions are typically designed and tested with a specific population within a specific context and research setting  and are Program adaptation frameworks, summarized in a scoping review by Escoffery et al. , provideAdaptation frameworks and models are tools designed for practitioners and researchers to aid planning, monitoring, evaluating, reporting, and studying the adaptation of interventions. These are comprehensive instruments and encompass different components in the adaptation process. Subsequently, the systematic use of these instruments strengthens the type 3 evidence generated in research and facilitates the implementation of interventions in the real world. Type 3 evidence provides information on the design and implementation of an intervention, the contextual circumstances in which the intervention was implemented, and how the intervention was received . In addiYet, the process of adapting a program has inherent implementation challenges. There is no guarantee that an adapted program will generate the expected outcomes even when considering contextual factors and employing frameworks. Balancing adaptation with fidelity is a frequent struggle when implementing evidence-based public health interventions . AdditioSeveral research teams have created models that can inform adaptation decision-making. Miller et al.  mentionehow adaptations should be made, referred to as type 3 evidence  employees' decision-making processes around program adaptation to improve the effectiveness of public health programs. Using a qualitative description approach , we inveevidence , 21. In This study involved qualitative interviews with public health professionals working at state health departments. A qualitative description approach  was usedThe interview guide questions aimed at understanding decision-making processes and factors related to mis-implementation of public health programs, i.e., ineffective programs that continue when they should have ended or effective programs that ended prematurely . The quevia email to inform them that the research team would be inviting their staff to participate in interviews. During this contact, chronic disease program directors were also invited to participate and asked for recommendations for other interview participants. In one case, the program director requested that their staff not be contacted, resulting in the research team replacing this state with an alternative state.Eight states were selected to recruit participants for this study. States were chosen to be representative of various population sizes and geographical locations, as well as high and low perceived levels of mis-implementation based on the results of a previous national survey . Each stPotential participants were then invited from each chosen state's chronic disease program. They were identified as having responded to the research team's previous national survey and/or based on recommendations from chronic disease directors or other staff. Participants were eligible if they served in a programmatic role in the chronic disease program . Their recruitment details are found elsewhere , 25. In via phone between February and June 2019. The interview guide was provided to participants prior to the interview. The interviews were conducted by an all-female trained research team, including three graduate research assistants, the project manager, and a faculty member . Participants were offered a $40 gift card. Alternatively, for completing the interview, the interviewees were offered a $40 donation to a public health charity of their choice made from the project's budget on their behalf. All interviews were audio-recorded and professionally transcribed (Rev.com) for analysis in NVivo 12  deciding to adapt the program (pre-adaptation) and (2) adapting the program (during adaptation) are presented below and found in The themes related to the decision-making processes around adaptation for the topics Participants reported different elements depicted by the themes when deciding to adapt a program. The themes included the factors influencing the decision to adapt a program, who was involved in this process, and the goals for those adaptations.Participants pointed out how much they relied on program evaluations or other data such as behavioral surveillance survey trends or return on investment calculations to guide their decision to adapt a program. Program evaluation data were the data on which participants relied for adaptation decision-making. Program evaluation data discussed by participants included quantitative participant surveys before and after implementation to detect changes in intervention clients' behavior or health status, as well as implementation process information from qualitative interviews.So we do have an evaluator. We have an epidemiologist and then the program managers. And we all kind of serve as an umbrella hub to really ask those hard questions, ask the critical questions. We utilize our evaluation plans. We utilize our performance management plans. We really have those hard conversations with staff about, ‘The needle is not moving. Help us understand why,' or ‘Do we really need to go back and let go of this approach and try a new thing altogether?”[Participant 1]“I think it really takes diligent and observant program directors or even the leaders of the DPPs [Diabetes Prevention Program], if they think about how... and they're evaluating their program itself, and who they're targeting and who they're reaching. I think it would take those types of people to go to the decision-maker and say, I think that this program isn't as effective as it could be, or that we could try to reach a different population in a different way. I also think that data speaks volumes, too, so if enough data can be shown that something needs to change, or what we're doing isn't working.” [Participant 2]“And I think that's where evaluation comes in and we have a very strong evaluation team, and so we're able to look at from beginning to end, how does this work and is it effective and do we want to continue or do we need to adjust it to adapt it to the different needs in our communities.” [Participant 20]“So I think any time that we are making those hard decisions about changes in the programing or letting something go or adding something new in, our conversations really now are, ‘At the end of a certain grant cycle, will we be able to show the outcomes that are needed to secure funds for [our state] or put us in the best place to receive those funds?” [Participant 1]“Other organizational and sociopolitical factors also affected the decision to keep a program and adapt it. Participants discussed the need to adapt programs to align with changing federal landscapes. The main example several participants described was the need to adapt recruitment processes and target recruitment populations for breast and cervical cancer screening or colorectal cancer screening after the Affordable Care Act expanded access to cancer screening and after many states expanded Medicaid eligibility.Evaluation, outcomes on that was a priority, our return on investment was a priority, and then just overall community engagement, community support.” [Participant 3]“When the new hypertension guidelines came out, we adopted them. I think it's based on funding and it's based on whether or not national funding partners decide, ‘We need to change that.” [Participant 4]“So for example, prior to the Affordable Care Act, the women's cancer screening program, our primary objective was to help uninsured women. The amount of uninsured women decreased significantly post-ACA so we had to change the way we implement our program a little bit and that involved expanding our services to include under-insured women. So that was women who have insurance but who might have some difficulty paying for... like copays and deductibles associated with screening services. Particularly like follow-up diagnostic services.” [Participant 12]“The decision to adapt a program was often made by SHD managers after discussion with team members, by program managers and their staff, or by SHD program teams in consultation with local agencies. Participants said most decision-making about adaptation happens at the program staff and program manager levels. Moreover, they mentioned that only programs with media attention or political considerations needed to go further up the SHD chain of command for decisions about modifying program content, target population, or recruitment or implementation strategies. Other participants said program staff and managers are involved in the adaptation decision-making in consultation with external partners, especially partners the SHD is contracting with to implement the program.It would be myself; I pretty much oversee this program, and then also my program manager.” [Participant 5]“Our chronic disease team meets. They're part of the same team, and so they have regular, ongoing staff meeting, communication meeting, all of that. They do their planning and they work together. And they're also connected with the larger provider community that administers such programs. And so I think that they're the ones that would be more in conversation and discussion about how programs are implemented and which ones are effective and what they hear from their colleagues and others.” [Participant 6]“Participants often noted what they aimed to achieve when making a change. Some goals seemed to be directly related to addressing the lack of outcomes or due to sociopolitical and organizational reasons. Other goals seemed to be indirectly related and included the goals to enhance funding or partners' engagement and satisfaction by refreshing the program. Here are examples of the adaptation goal of increasing the reach of the program:There was the building awareness and the outreach for recruitment of participants for the different counties. They were also very creative in identifying how one method of maybe recruitment or advertising of the program wasn't quite working, so they then tried a different way.” [Participant 7]“And now, since we've refreshed the materials, they look new, they look different, they look exciting. So that has helped a little bit.” [Participant 8]“After deciding to adapt a program, four approaches were reported by the participants as part of the adaptation process. The following themes summarize the approaches used for selecting how to adapt a program and who is involved in this process.Participants mentioned using evaluation data and other evidence to inform their decisions when adapting their program. For this process, they relied on accessing available evidence from research, including evidence-based approaches and pilot projects, learning what was working in other states; and obtaining data from their program evaluations, including data from participant surveys, partner engagement surveys, and interviews with participants and partners.An ineffective program I think is one that really isn't based in our evidence. I fully understand the importance of emerging evidence and research and piloting projects. …I rely a lot on evaluation and outcomes from evaluation initiatives that we do within our various programs. So, you know, just taking a look at the evidence, seeing what is working within our programming efforts, what may not be working, but adapting to that feedback too. So not just staying stagnant in our activities and initiatives, but learning from evaluation reports, learning from data collection and looking at data trends and making meaningful change that way.” [Participant 9]“…So we're in the process of transitioning and that was all based on the evaluation results and there is... school-based programs are an evidence-based approach. ….We did a lot of research into this, the best ways to implement it. So we're in the process now of transitioning. It's a lot of work. We're just trying to implement it to the best of our knowledge, following all the evidence that's out there.” [Participant 10]“The coalition member that was the lead on it, she found I believe it's a study done on library staff in Washington State. So she did some background research, learning what works, but again tweaking it so instead of library staff doing the screening and education it was hairstylists.” [Participant 21]“Program staff met regularly, e.g., weekly, monthly, or every 6 months, to discuss and plan the changes they wanted to make in the programs. Program managers and staff often brought in program evaluators, who were an important part of the process of deciding how to adapt a program. In some instances, leadership needed to approve the changes. In this process, they used data from evaluations to inform their changes.The evaluator, our internal... our director of evaluation will usually do at least... as he's establishing the evaluation plans, he will meet frequently with the staff to go through that process of coming up with a good plan. He will meet often with stakeholders a number of times throughout the year and with program staff at least once a month as well. So, you know it's pretty frequent. And then, once an evaluation is underway then there's at least an annual review of what have we found? Where are we going? What do we... is there anything that we need to tweak? It's probably done more every six months actually.” [Participant 11]“Well, honestly I think that's something in my unit that we're always thinking about. We have weekly meetings. Part of our meetings is how we can best approach programming and evidence base, and how we can meet our PM [performance management objectives]. So yeah, it's a continuous thought and conversation.” [Participant 22]“Well, certainly during the planning process it's a lot. It's in a really short compressed timeframe. And then, once the funding is received and we start to really solidify the programs, then it's the program staff who will get together at least monthly, usually more frequently in the beginning but move to a monthly meeting to where they talk through what's happening. How are things going? What are we seeing? What aren't we seeing? Are there things that need to be tweaked?” [Participant 11].“In the process of making changes, program managers sometimes contacted their partners in other states. They paid attention to what was working in these states to rethink how they should do things in their SHD. Another approach used to decide on the changes was getting feedback from community stakeholders.I do make, at the end of the day, decisions about what happens with the programs and the contracts that we make and the direction that we're moving in. But I would like to think that I sort of consult my peers as much as possible.” [Participant 12]“The community came back and said no we want to do Zumba but the … program coordinator wanted to implant a yoga program and so the community said it is great that you are offering that it is better than nothing but if you really want to get this community up and moving Zumba is what you are going to need to offer, because people love to dance. So they [program coordinator] acted fast and started a Zumba program and that's what I saw there that I haven't seen in a lot of other programs.” [Participant 3]“..we would often during our TA [technical assistance] calls at CDC, or in attending meetings or conferences across the state, across the country, we would find best practices of what other states were doing with similar funding, if not the same funding. We would also get feedback from the staff at the districts and also our partners. So we did partner engagement surveys to determine what was working, what wasn't working. We had calls often with our district-level folks, again to determine what was working, what wasn't working, and how we could kind of re-steer the ship to ensure that we were still meeting our goals and deliverables.” [Participant 13]“I think here our leadership would generally rely on the programmatic folks and the division directors to sort of research and understand what other alternatives would be and to come up with a recommendation. I think if they felt like that wasn't working or wasn't the right way to go, they would reach out to other states to find out what they're doing.” [Participant 14]“And obviously states are really willing to share, and that is really helpful because if somebody does find something that works in a new space, it really is helpful to kind of take their lessons learned, maybe avoid some of their pitfalls and their challenges that they had, but we always have to tweak it to the [state] landscape…we can't just pick up something that they've done and run with it because the partnerships are different, the infrastructures are different, the relationships are different.” [Participant 1].“Participants mentioned SHD program team meetings, regular technical assistance calls, committees, and coalitions as opportunities to discuss, receive and provide feedback on the adaptation process. Some coalitions and committees were statewide, but participants also described instances where a local county or city coalition or committee provided input on which aspects of the program to adapt and how.I have a statewide coalition and each one of my programs, our strategies have committee around it and we review the progress together and decide what's working out and what's not and how to either tweak it [program] or move it [the program into] something slightly different, I don't think it [program] was ever really discontinued unless there was no funding.” [Participant 15]“We have monthly TA [technical assistance] calls. Our nurses do monthly TA calls with their assigned specially qualified health center, and the local health departments have a community health educator on our staff that is one of their TAs. A lot of times, they'll talk to their TA about an issue. If it's something that the TA can't address, then they'll bump it up to management. Then we'll discuss in grand rounds or sit in on a monthly call to see if we can address their concerns or figure out what needs to be done differently.” [Participant 16].“This study provides descriptions of state health department employees' decision-making processes around program adaptation of public health programs that others can apply when deciding whether or how to adapt a public health program. Program managers and section directors shared their decision-making approach to adapt a program. The decisions were (a) directed by data, outcomes, and evaluation, (b) influenced by reasons that included sociopolitical and organizational contextual factors, (c) involved SHD middle-level managers, program managers and staff, and the local agencies, and (d) aimed at increasing program effectiveness/outcomes, reach, satisfaction with the program, funding, and partner engagement. The program adaptation processes encompassed (a) using data and evidence to guide changes, (b) engaging with program staff and program evaluator, (c) consulting with partners and stakeholders, and (d) using systems and groups already in place to get input on how to adapt content and contexts. The findings provide practitioners and researchers with insights for decision-making around adapting public health programs.For both topics, “deciding to adapt a program” and “adapting the program,” a link to evidence-based public health (EBPH) was identified as important. EBPH is defined as  “the proWhen deciding to adapt a program, interview participants mentioned EBPH approaches like using data, evaluating outcomes, and engaging the community in decision-making. Participants also involved programming staff in the decision to adapt the program, which is important since participatory decision-making when adapting a program may predict the impact of the changes . AdditioWhen using data and the best available evidence to adapt a program, participants were applying EBPH skills , indicatOur findings are aligned with the interview guide framework  used forThe findings reinforce the importance of promoting public health capacities in program evaluation and adaptation. Participants often mentioned that adaptation needs were identified once someone with evaluation skills was involved in the program, highlighting program evaluation's critical contribution to deciding whether to adapt and successfully adapt a program . TraininDeliberate efforts are needed to promote program adaptations as a systematic and evidence-informed practice. Having established procedures for documenting translation and adaptation can help recognize whether the adaptation is effective . To implThe study has a few limitations. Our data are from eight states; although we aimed for representativeness in the selection strategy of the participating states, it is still a limitation. Differences in how programs are governed in different states and territories and other organizational and environmental characteristics might have led to variations in the approaches used to adapt programs. Another limitation was that program effectiveness was based on the participants' perceptions and it was not investigated if the changes resulted in the expected outcomes for the programs. Adaptation decision-making was only a portion of each interview, so our findings are not a comprehensive view of adaptation but inform this area of research. Additionally, we were not able to analyze adaptation decision-making power by equity-relevant subgroups  since we did not know which partner groups were from marginalized communities.The findings have implications for public health practice, policy and research. First, the results depict the decision-making process for adapting programs in state health departments, providing practitioners with potential directions for adapting public health programs. Second, the findings reinforce the importance of promoting public health capacities in program evaluation and adaptation, indicating that agencies' policies could support more investments and plans to enhance skills in these areas. Third, documenting the adaptation process is important  and shouUsing a qualitative analytical method , we invel.farah@wustl.edu.The datasets presented in this article are not readily available because of participants confidentiality concerns regarding the raw data, but data summaries are available upon reasonable request. Requests to access the datasets should be directed to LF: The studies involving human participants were reviewed and approved by Washington University in St. Louis Institutional Review Board. Written informed consent for participation was not required for this study in accordance with the national legislation and the institutional requirements.RB conceptualized, designed the study, and outlined the introduction section. SM, SM-R, ER, PA, MP, and RB developed the interview guide. SM-R, ER, and MP conducted the interviews. SM, SM-R, LF, ER, PA, and MP coded the interviews' transcripts. ER and LF synthesized the themes and reviewed the first draft. ER drafted the methods section. LF drafted the other sections. PA and SM contributed to the discussion of the findings. All authors read, contributed to manuscript revision, and approved the submitted version.This project is funded by the National Cancer Institute of the National Institutes of Health (R01CA214530). Additional support came from National Cancer Institute , the National Institute of Diabetes and Digestive and Kidney Diseases , the Centers for Disease Control and Prevention (U48DP006395), and the Foundation for Barnes-Jewish Hospital. The findings and conclusions in this manuscript are those of the authors and do not necessarily represent the official positions of the National Institutes of Health or the Centers for Disease Control and Prevention.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. The handling editor MM declared a past collaboration with the author RB.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


BackgroundResidency program websites are an important resource widely used by prospective applicants when applying to programs. The objectives of our study were to evaluate the program content available on Canadian anesthesiology residency program websites using established criteria and identify any areas for improvement. MethodsIn this cross-sectional study, we evaluated the content available on accredited anesthesiology residency training program websites, between July and August 2021, using 54 criteria provided in the following domains: recruitment; faculty; residents; education and research; clinical work; incentives; wellness; and environment. Website scores were analyzed using descriptive statistics and presented as median (interquartile range), percentage (%), and range.ResultsWe identified 17 programs with publicly available functional websites. Overall, residency programs met a median of 28 (interquartile range: 18-36) website criteria out of 54 (51.9%). Education and research was the highest-scoring domain among residency programs (median 77.8% of criteria met), while resident information and incentives were the lowest (14.3%).ConclusionCanadian anesthesiology residency program websites include information on many domains relevant to prospective applicants, including education and research. However, most websites require improvement and content updates for faculty information, resident information, incentives, wellness, and environment. The process of choosing and applying for residency programs is challenging for medical students -3. OnlinTo better facilitate applicant decision-making, it is pertinent to maintain a resource of comprehensive information for prospective applicants. This will help both medical trainees and program recruitment committees to find the most suitable match for their respective goals, aims, and needs. Accordingly, the objectives of our study were to: (1) assess the content available to prospective applicants on Canadian anesthesiology residency websites using established criteria ,7,11-14;Study designWe conducted a cross-sectional study to evaluate program information available to applicants on Canadian anesthesiology residency training websites between July and August 2021. The study did not require research ethics board approval as publicly available data was evaluated (Tri-Council Policy Statement 2 Article 2.2). A 54-criteria system used to evaluate program websites was created using: (1) established criteria from previously published postgraduate medical program website evaluations ,11-14; (Data collectionhttps://www.carms.ca/match/r-1-main-residency-match/program-descriptions/). Anesthesiology Clinician Investigator Programs (CIP) were excluded from this study because RCPSC training requirements and goals for CIP differ from regular stream anesthesiology residency programs; it would not be appropriate to evaluate Anesthesiology CIP website content using our 54-criteria system (Table A list of RCPSC accredited anesthesiology residency programs was obtained from the Canadian Residency Matching System (CaRMS) website (em Table  18]..https://https://www.random.org/lists/). The reviewers were not blinded because they were viewing the actual website [Two website reviewers (AST and DJW) were trained to use the 54-criteria system by examining three randomly selected program websites. Discrepancies in scores were discussed, and coding was determined as a team to establish interrater reliability. Information was considered present on a program website if it was available on: (1) the program website or subpages; (2) linked documents ; or (3) websites hyperlinked on the official page . A score of one was recorded for each criterion met by a program website, and zero for every criterion absent. The trained reviewers independently analyzed each anesthesiology program website; the order of program websites for each reviewer was randomly selected using an online generator . Website scores were analyzed using descriptive statistics and presented as median (interquartile range), percentage (%), and range. Statistical analyses were performed using SPSS Statistics .There are a total of 17 RCPSC accredited anesthesiology residency programs; all programs had a functional program website. Overall, the 17 residency program websites met a median of 28 (interquartile range: 18-36) website criteria out of 54 total criteria (51.9%) with scores ranging from 14 to 44 (Table Within the highest-scoring domain (education and research), research opportunities were the most prevalent criterion present on program websites  followed by curriculum/didactic learning  and assessments/evaluations . Resident information and incentives were the lowest-scoring domains among residency websites Table . SpecifiThe purpose of our study was to assess program content available to prospective applicants on Canadian anesthesiology residency websites using a 54-point criteria system. Education and research were the highest-scoring domain among residency programs, while resident information and incentives were the lowest.Residency websites effectively provided program information regarding research opportunities , curriculum/didactic learning , and assessments/evaluations  within the education and research domain. Similarly, a study of obstetrics and gynecology program websites in Canada and general surgery residency websites in the United States (US) and Puerto Rico found that research opportunities and didactic learning were prevalent criteria on residency program websites ,20. AddiInformation about faculty, residents, incentives, wellness, and environment were often omitted from anesthesia residency websites. Faculty and resident information  are considered important content areas on program websites among applicants . Facultyhttps://www.royalcollege.ca/rcsite/documents/about/covid-19-wellness-resources-hp-e).Information regarding wellness  and incentives  were also among the least commonly met criteria on anesthesia residency websites. This is despite the increasing emphasis of the anesthesiology specialty on work-life balance and burnout avoidance ,23,24. Whttps://www.carms.ca/match/r-1-main-residency-match/salary/#1511459027032-06ec5e41-5301). Opportunities such as moonlighting, a practice permitted by the RCPSC, support trainees dealing with financial hardship, reduce emotional exhaustion, and improve quality of life [Similarly, information about incentives, including salary, benefits, and moonlighting may help applicants with financial planning. Medical school graduates often have significant educational debt, a factor commonly associated with depression, burnout, low quality of life, and decreased job satisfaction . Salary  of life ,29. It iA multitude of reasons may explain the absence of information on residency websites. There is the possibility that programs may not recognize the changing trends for obtaining residency information ; programLimitationsData collection took place between July and August 2021 and did not account for website updates that may have taken place after this study period. The 54 criteria used to assess each program website were adapted from previous studies and although they provide a standardized framework for assessing program websites, there is no one set of criteria that can reflect the values of all prospective applicants. Future studies investigating which criteria are important to postgraduate Canadian anesthesiology trainees may be helpful to create a more representative criteria list. Data collection by different individuals may lead to varying results due to the subjectivity involved in identifying the presence of information on program websites. Nevertheless, two trained investigators independently evaluated each program website to mitigate the potential for variability.In this cross-sectional study, we found that Canadian anesthesiology residency training program websites met most criteria in the education and research domain. However, many program websites lacked content pertinent to prospective applicants. Domains including faculty information, resident information, incentives, wellness, and environment are areas that require improvement and content updates. Addressing these insufficiencies by updating websites may help anesthesiology programs attract prospective applicants as part of their recruitment efforts and may help applicants make informed decisions surrounding their postgraduate training.


Rates of young people who are neither in employment, education, or training (NEET) are fairly high in the European Union. Correspondingly, there has been a growing tendency to regard sport as a suitable tool to develop soft skills and raise NEETs' level of employability. However, if and how such sport for employability (SfE) programs are able to realize these major claims has been called into question. The purpose of the present study was, therefore, to explore how an actual SfE initiative constructs and delivers its program. In addition, the article assessed whether the investigated program operates in line with researchers' recent calls for theory-based approaches. Guided by a case study approach set up within an initiative located in Flanders, data were gathered through 12 semi-structured interviews with 8 program providers. Results, analyzed using thematic analysis, indicated that the program was characterized by an absence of well-defined desired outcomes, imprecision as to how the program should contribute to these outcomes, and consequently minimal attention to the follow-up of participants' progress. As such, these findings and the accompanying challenges point to the absence of a theory-based approach. Several possible sources for the lack of a systematic approach are discussed. According to Eurostat , over 13Different strategies have been proposed to face this societal challenge. One strategy consists of increasing employers' likelihood of hiring young people by providing, for example, wage and training subsidies or tax and national insurance credits . AnotherDespite the lack of consensus about its conceptualization, some recurring components can be found in most employability definitions. For example, both academic literature  and polSport is proposed as a suitable tool to develop the aforementioned soft skills. It is argued that sport can contribute to the development of such skills as communication, teamwork, self-motivation, perceived self-efficacy, self-discipline, problem-solving and perseverance –32. AccoSuch initiatives are captured under the rubric of 'sport for employability (SfE) practices' and can be regarded as a specific subset of sport for development (SfD). However, given the limited number of studies concerned with increasing employability through sport , 36, we Sport is claimed to contribute to numerous social, educational and health goals . It is bSport, Plus Sport or Sport Plus. Sport can be organized just for the sake of sport. But when sport functions as a ‘fly paper’ to engage targeted youth within programs of education and training, it is labeled as Plus Sport. A fairly different approach is reflected in Sport Plus practices whereby sport is not only deployed to attract targeted youth, but sporting activities are tailored and supplemented by non-sporting activities  to achieve certain developmental goals. Both Plus Sport and Sport Plus approaches explicitly use the social values of sport.Based on the prominence of the social role of sport, Coalter , 49 distDespite sports' social or developmental potential depending on its intentionally created development opportunities , SfD prTherefore, several researchers pointed to the importance of providing insight into the process of SfD programs by using a “program theory” or a “theory of change” e.g., , 51]. Ac. Ac51)].Such a theory-based approach “seeks to identify the components, mechanisms, relationships and sequences of cause and effect that are presumed to lead to desired impacts and outcomes”  (p. 53).A theory of change ideally starts with the aimed impact of the program and contains short-term outcomes and outputs, the various activities of the program, and the resources or organizational inputs required to implement these activities . CharactSeveral benefits are linked to this theory-based approach. First, it offers the possibility to intentionally and commonly stimulate these outcomes or impacts by focusing on the conditions and mechanisms that are responsible for triggering them . Second,According to Coalter et al. , sport cAlthough the current literature provides valuable information based on an analysis across different programs, in-depth insight into the workings of a particular existing SfE program remains absent. Thus, the purpose of the present study is to take a closer look at a SfE program and explore how it structures and organizes its program in an applied setting. Such a detailed case study analysis  might prThis study adopted a case study approach to address the research questions. Data were collected within a municipal sports-based employability program in Flanders, the Northern Dutch-speaking part of Belgium. Data of this municipality (from 2019) show that only 65.4% of the local population between 18 and 64 years old is employed . More spIn 2017, the municipal employment service and a social sport initiative (which can be considered as the main program organizers) joined forces with several local partners and initiated two SfE programs targeting NEETs . While the project description of the male program specifically aimed at young adult males (between 18 and 30 years old) unknown by the Flemish employment service and therefore missing opportunities for support to find work, the female program targeted women (between 18 and 50 years old) lacking the skills and resilience conducive to their integration into the labor market. This also included their low Dutch language level. The program was organized three times a week and lasted 10  or 20  weeks. The programs had different lengths as the specific organizers of the women's program thought 20 weeks was too long to keep participants motivated. The programs included various workshops and activities related to (non-sports-based) experiential learning (cooperation and outdoor activities complemented by reflection), sport, job coaching and social orientation and additional job coaching support if needed. The sessions experiential learning accounted for half a day per week within both programs and consisted of, among others, climbing, cooperation and problem-solving games . Sport sessions were more prominent in the male program as it covered a full day per week compared to half a day within the female program. These sessions included both team and individual sports . The final combined component of job coaching and social orientation was in the female program organized during one and a half day per week and one day within the male program. In general, the content of this final component covered informal training on social skills, work attitudes, labor market expectations, personal strengths and gaining insight into labor-related organizations. Example activities include a game about one's personal strengths, a quiz on labor market expectations, interview training, and a visit to the Flemish employment agency. The activities were distributed equally over the duration of the program. Evidently, the specific content of the activities became more labor market oriented as the program progressed . The aim of both programs was to enter the labor market in a sustainable manner. Twelve female participants participated in the program and were always present over the course of the program. Within the male program, an average of 15 people attended the program. During the course of the program, the number of male participants fluctuated as some participants dropped out, while others entered the program at a later stage.This study used a case study approach as it facilitates an in-depth exploration of phenomena  and can In total, 12 individual semi-structured interviews were conducted with 8 staff members . Some were interviewed twice as they were related to both the male and female program or to expand on information. Respondents' average age was 36 years (ranging from 26 to 53) and there was some variation with regard to years of experience in the field. The selection covered a variety of staff roles and types of organizations involved in both programs see .The selected data collection method enabled interviewees to report on their own thoughts and feelings and made it possible to obtain a deeper knowledge on their experiences . The intAll interviews were tape-recorded and transcribed verbatim afterwards. Thematic analysis was used as an accessible method to identify, analyze and report patterns or themes within the data . Followivia a critical friend (second author) who offered a theoretical soundingboard by eliciting critical dialogue and encouraging reflexivity on the lead researchers' interpretations  were in congruence with the research questions. Furthermore, a fourth topic, relating to the integration of stakeholders, was added during data analysis as it provided important information to understand how the program operates. The relationship between the different topics, higher order themes, core themes, and raw data themes is illustrated in The organizers listed 18 intended program outcomes. The list covered psychosocial outcomes including, among other things, perseverance, oral communication, motivation, stress management, attendance, and initiative. However, a follow-up conversation with one of the providers revealed that the list was not based on an actual consultation with the other program providers and was not consistently put into practice . It was developed intuitively and primarily designed for the project to be submitted for funding.Furthermore, providers reported that they experienced political pressure to modify the program results in a positive way. The coordinator of one of the partners argued policy wanted to present simple evidence, including figures of those who access the labor market, without taking into account participants who may not fit into the project: “that's a bit of a trap with this kind of interventions, when you look at policy, they obviously want quantitative data” (P7).The program organizers had a general idea with regard to the intended target group, but recruitment and selection was not a conscious process and the selection procedure was not clearly defined. Extensive recruitment was done by drawing on its own network of potential participants and through contacting and promoting the program among specific local organizations who are likely to target NEETs and vulnerable groups . A number of general selection criteria were used for the inclusion of participants in the program. Reference was made to “coming from the municipality”, “between 18 and 30 years old” and “having a distance from the labor market”.However, interviewees stated that selecting participants was not a conscious process: “That is not a deliberate decision. (…) I find it very difficult to say who I do and do not select and why” (P5). Accordingly, the selection procedure was not clearly defined. Although a selection day (including intake interviews and a cooperation assignment) was organized, participants could enter the program at any time during the whole program and without being formally selected on the selection day: “In the beginning, it was a mess: who will come and who will not and based on what do you participate in the program?” (P1).The fact that clear selection guidelines were lacking also resulted in some cases in failing to attract people with a distance from the labor market: “Our local authority says 6 out of 8 former participants are currently working in a sustainable manner. That's basically correct, but they shouldn't have been in this project as they would have found employment anyway” (P7).Both programs consisted of three major activities: (non-sports-based) experiential learning, sport, and job coaching and social orientation. Respondents indicated the aim of the experiential learning sessions was to raise participant's level of self-awareness. Experiences such as cooperation and outdoor activities were complemented by a reflection component where participants were encouraged to reflect on and discuss their own attitudes and behavior. According to the experiential learning coach, an underlying process of recognizing, acknowledging, and exploring was intended to approach things differently in the future:For instance, x is a participant who encounters many difficulties at work and is only able to complete short contracts. Here we see someone that complains a lot and is always making remarks. That is mentioned and discussed. During the one-week training camp, the other participants also mentioned: “grumpy bear is at it again”. If a person acknowledges something, he or she can see what the effects are and then you have the choice to do something with it. (P1)The coaches explained that the experiential learning sessions were characterized by a specific structure focusing first on “creating a safe learning environment and getting to know each other” (P1), while gradually shifting the emphasis to incorporating reflection and developing soft skills. However, a major barrier within this process consisted of irregular attendance of some participants. As one of the coaches stated: “This made it difficult to use the group as a tool and created a constantly fluctuating group dynamic” (P1).Another main component of the program related to the sport sessions, which in the male program intended to be used as, among other things, an attraction pole or “flypaper”. Furthermore, respondents highlighted that involvement in these sessions was expected to trigger several positive outcomes, including feeling better, enhanced social skills , enhanced perseverance and improved group cohesion. While referring to broadly the same suggested positive outcomes, the interviews revealed that sport was used differently within both programs. The male sport sessions included, among others, football, running, boxing, climbing, swimming and fitness. The program providers' accounts reflected that sport was intended to be used as a learning tool but did not reach its full potential as many learning opportunities were left unused. It was believed that a lack of expertise was primarily responsible for this: “Here you can work more deeply and on an individual base compared to a regular sport class. But that's still a learning process for me. I feel I need more background and frameworks to apply this even more” (P1). Where reflections were included, they rather took the form of “how did it go?”. The only notable exception related to a session whereby participants had to teach a boxing lesson to an external group. In this case, participants were instructed to specifically focus on job interview skills such as “speaking loud and clearly” and “a proper appearance”. Afterwards, they concluded with a reflection including feedback from the people who received the boxing lesson, the other participants of the program and themselves.Female sport sessions included, among other things, body shaping, fitball, boxing, badminton, and netball. Coaches believed that participants would be more interested in feminine sports and therefore “consciously opted for more feminine sports such as Zumba and fitball” (P7). Although reference was made to a number of expected positive outcomes, a number of interviewees explicitly stated that the role of sport was rather ill-considered:ad hoc basis. It wasn't originally the idea to use sport to enhance employability. I think we had too little thought about it beforehand. (P7)We did insufficiently deploy sport as a means to develop skills. Things happened and were discussed afterwards but this was all on an Furthermore, it is interesting to note that there was a lack of common understanding regarding the role of sport within the program. When asked why sport was part of the program, the sport coach replied: “You have to ask them [the municipal employment service and the social sport initiative] why sport is part of the program, the main program organizers didn't tell me” (P6). Program providers, for example, stated that reflection or a link with a work situation was not part of their sport activities: “There is little explanation regarding sport. The morning already requires reflection during the sessions experiential learning, so we'll just let the afternoon be relaxing” (P5).The final major component of the program involved job coaching and social orientation and consisted of both group sessions and individual guidance by a job coach. Collective sessions included three parts: (a) understanding participants' interests and aspirations, (b) support with CV writing, interview training and job searching, and (c) an introduction to various sectors and jobs including visits to potential employers and labor-related organizations . Subsequently, intensively working toward work, education or any other suitable goal tailored to the participant  was the key aim of the individual support provided by the job coach. However, this individual phase was not quite specified in advance and therefore “not sufficiently integrated within the intervention” (P2). This ambiguity was also reflected in both the format, frequency, and duration of the individual job coaching support. To illustrate, the job coach of the female program described this phase as: “There is no fixed period on how long I guide them” or “For some participants I go on home visits and for others I don't … some need more guidance than others” (P5).ad hoc basis. We did a lot of shuffling within the planning” (P7). Instead, providers stated that the content of the program was mainly driven by emerging individual and group related needs or topics, rather than deliberately integrating explicit learning opportunities based on predetermined soft skills. The following quote illustrates the focus to adapt to the group needs:Despite the existence of three major activities, there was no adherence to a predetermined schedule in terms of the particular activity and its content. This was illustrated by some of the coaches: “There was written ‘sport’ within the planning, but when each part was delivered was often determined on an It was not fixed within the program when we would address certain issues. Rather, we tried to sense what the group needs and what the group is open to at a certain moment. In the beginning, there was a negative person who made fun of everything and our group was not strong enough to handle that. That could have resulted in a different group dynamic. In order to have that positive group dynamic, it's essential to have a group where you can build upon at that time. (P7)Respondents also pointed out that there was only minimal integration of the different program activities. Coaches felt that the main activities “remain too much three separate parts that do not form a whole” (P2). This lack of integration, exemplified for instance by some partners' absence from general meetings, resulted in a lack of alignment:The sessions at the center for adult education [one of the partners] went quicker than we [one of the partners] did. For example, they already put their CV's online and received calls from employers while job search was only on our agenda after 10 weeks. (P7)Furthermore, there was a difference in approaches between the different program providers of the male program. This difference can be marked by a strong reliance on the method of experiential learning including offering experiences, responsibilities, and reflection on the one hand versus an authority that gives direct instructions without any kind of reflection on the other hand. This difference in approaches became more pronounced over the course of the program and had a hampering effect on the overall coherence of the program. Respondents argued that as not everyone was convinced of the contribution of each component of the program, coaches started to only deliver their own component properly:Normally, I was supposed to attend more often, also on Mondays. But that was practically unfeasible. Or also because I didn't support those sessions. As for those Mondays, I've taken a step back. The coach of the youth work organization [one of the partners] is doing his thing there and I'll do my thing on Friday. Sitting squatted against a tree, starting to hyperventilate, … I don't see the point. (P1)Coaches referred to the importance of creating a safe social climate and establishing trusting relationships with participants. In this sense, social relationships were considered to be “the success of the program” (P4). Already since recruitment, there was a strong focus on “feeling comfortable and at home”, “feeling appreciated with both your strengths and weaknesses”, “value one another”, “a caring relationship”, “a sense of belonging” and “always be there for them”. Interested and caring adults and a sense of safety, acceptance and belonging are also part of the “protective factors” as identified by Witt and Crompton . These fIn relation to these mentoring styles, a notable divergence became apparent throughout the male program. The interviews indicated that one of the coaches provided unconditional support: “coach x was always there, he did everything for those guys” (P1). As a result, this relationship was marked as “coddling” as “he moved away from the rules” (P7). Most of the coaches, on the other hand, rather underpinned a relationship based on critical support to empower participants. As such, participants should “come to important insights themselves” by “offering experiences” and “promoting responsibility” (P1). According to one of the providers, this imbalance of support between the coaches led to “good cop, bad cop, which causes the program to fail in terms of quality” and “the loss of learning opportunities for the group” (P7).Another aspect that came to the surface during the interviews was the distinctive relationship that participants developed with coaches that are seen as “equal to them”. According to Bandura , perceivI think the boys look different to me than to coach x or coach y. I think they see them more as equals. You feel the difference. Not that I have a bad relationship but if I wanted to make someone aware “this job is something for you”, if x said that, it had more impact. (P2)Conversely, coaches who do not seem to meet these requirements were sometimes being told: “It's easy for you, you are not raised in my neighborhood, you don't know what that means” (P7).Although the initiative was characterized by several promising elements with regard to the monitoring and evaluation (M&E) of participants' progress, it was evident that a systematic approach was missing. This will be explained by successively discussing the different elements  that relate to the stages that participants went through during the program.Starting with the intake procedure, the male program had no formal diagnosis of participants' personal situation at the start of the program: “It's not like we're evaluating their needs during the selection process” (P7). In contrast, in the female program participants' needs were assessed as part of the intake interview with the job coach:I have a conversation of an hour to an hour and a half to find out who that person is and what that person needs. “What has caused that you are in the current situation and that you are not getting to where you want to be”. (P5)The job coach also regularly attended various workshops to get to know the participants, identify their strengths and weaknesses and points that she could work on. Additionally, the program sought to evaluate participants' competences and subsequent development by means of a low-threshold tool. In the absence of a personalized instrument, they looked at existing instruments and were given permission to test a competence measurement tool commissioned by the sport administration of the Flemish government. The instrument was mainly targeted at volunteering in sport but covered the same general target group of people with a certain distance from the labor market. Upon admission, female participants were asked to complete the competence measurement tool representing seven roles or personalities measured by several items encompassing skills and attitudes . Although results were displayed immediately after finishing the test, results were not discussed in detail with the participants, nor did they provide a basis for setting objectives within the program: “Participants got their results, but we didn't discuss it with them afterwards” (P4).Regarding goal setting, one of the members of the steering committee expected the female program to work with a personal development plan including goals for each of the participants. For the sole purpose of meeting this requirement, the program providers completed all personal development plans, but this tool was not used within the context of the program as one interviewee indicated “… we didn't do anything with it. […] we never discussed them with the participants” (P4). In contrast, the male program did not have a formally drawn up plan, but objectives were established and subsequently discussed during the individual progress meetings: “Our evaluation, including qualities and areas in need of development, were discussed with the participants. The second time, we included possible changes compared to the previous time as part of the conversation” (P1).Considering the final element, that is monitoring and evaluating participants' progress, neither program made use of a systematic monitoring system. The initial completion of the competence measurement tool relating to skills and attitudes was not followed by an evaluation on a later occasion within the program: “We didn't do much with the competence measurement tool” (P4). As opposed to the female program, the male intervention did organize some individual progress meetings to discuss participants' development. However, providers of the male program reported that participants still faced difficulties to identify their own strengths: “Naming one's own qualities remained difficult at the end [of the program]” (P1). Coaches stated that they pointed out participants' qualities and development needs, but that they did not subsequently monitor them on a regular basis: “We explicitly mentioned their qualities, but it's not that we looked back at it the following week. Maybe that could have been more concrete. With specific development needs which we might then consider on a weekly basis” (P1).In addition, the interviews revealed that the decision whether someone is ready to enter the job market is associated with some ambiguity. Reference was made to things such as “participants must first have a strong foundation” and “motivation and attitude are the most important criteria to consider in order to declare participants ready for the job market” (P2). However, the job coach explained that although she did experience a number of differences with the participants concerned, it was hard to substantiate these exact differences. She indicated that it was rather a perception that relied heavily on intuition:That's pretty much a guess I'm afraid. […] Gradually you feel, but that is some kind of gut feeling, I can send that person to an employer while I did not support that idea in the beginning of the program. Adhere to agreements, punctuality, be realistic, motivation, … […] Those participants that are currently in employment did undergo a few changes. (P2)Moreover, program providers stated “ambiguity” as to whether participants are expected to reach this goal already before the end of the group program and therefore called for more clarity. Reference was made to former participants who repeatedly dropped out of employment due to recurring issues such as “showing up late”, “an argument with the boss” or “not able to work in a team”. According to one of the providers, the program was partly responsible for this “… as they allow participants to move on when they are not yet ready” (P7).The program was characterized by only minimal integration with stakeholders . With regard to referral agencies which had the possibility to terminate participants' benefits, several respondents reported that the intervention would benefit from a specific cooperation which respects the rationale of the program. This means, providing optimal chances for participants but also supervising certain obligations since the intervention should prepare participants for the labor market.Similarly, the program did not engage in any specific partnerships with employers. Rather, support provided by the job coach focused solely on the participants and on bringing them into contact with potential employers who originate from the job coach's network. Such a focus on participants' own ability, including building on strengths and removing personal barriers, seemed also reflected within the program: “our program focuses on strengthening the job search: overcoming obstacles and enhancing qualities to make the step toward work easier” (P5). Due to this lack of involvement of  employers within the program, one of the coaches expressed concern as to whether participants were able to transfer any achieved outcomes to the labor market: “it's not clear whether the development of some women has continued within the labor market afterwards, because by then, of course, that safe environment will be gone” (P7).The aim of the present case study was to gain more insight into how a SfE program defined and worked toward its desired outcomes and how it monitored progress. As a secondary research question, we intended to assess whether the investigated program operated in line with  theory-based approaches. Our findings revealed that the program providers were driven by high level ambitions such as closing participants' gap to the labor market or education. However, the results mainly illustrated the potential pitfalls or challenges that SfE programs may face. Correspondingly, our findings suggest that the investigated program was designed based on intuition and experience without reflecting on the underlying principles that undergird program activities . As suchThe first observation and challenge related to the absence of well-defined definitions and operationalizations of desired outcomes. Findings suggest that there was limited (shared) reflection on what the program should achieve to sustainably strengthen participants' position in the labor market. As such, the relative lack of focus hindered intentional work toward these outcomes and prevented program providers from evaluating their initiative on the basis of related performance indicators. This finding is in line with research conducted within the broader field of SfD. A number of researchers already concluded that sports' added value is often described in ambiguous, ill-defined, over-inflated and imprecise terms e.g., , 48]. Ac. Ac48)].Second, and in line with the first observation, it was found that the case under study did not use a systematic approach to its program strategy. The content of the program was not well considered with regard to the nature, extent and duration of participants' involvement and contained numerous vague elements. Correspondingly, it was unclear how the program would contribute to the list of intended outcomes. This was exemplified by, for example, unclear selection guidelines to enter the program, a lack of integration of the various program elements and providers, and the unclear role of sport. According to Coalter , 40, manThird, and not entirely unexpected, the imprecise formulation of desired outcomes had implications for the M&E of participants' progress. Despite several promising elements regarding tracking participants' progress, there was no coherent strategy to address the different stages  that participants go through during the program. This resonates with findings from a European study on the contribution of sport to the employability of NEETs , which cA number of possible sources for the lack of a systematic approach can be suggested. First, program providers may underestimate the importance of defining precisely both specific outcomes and accompanying strategies to intentionally facilitate those outcomes. Respondents organizing the female program in our study reported that the role of sport was not a subject of reflection when designing the program. One of the partners, responsible for teaching the sport sessions, was unaware of why sport was part of the program. It suggests that the main program organizers had only minimal reflection on what they are doing with sport, how they are doing it and why. In addition, they did not inform the partner responsible for teaching these sessions. This lack of consideration might be related to a general belief in the power of sport in which sport will unconditionally benefit the employability level of the participants. Sport is often considered as “a unitary experience”  (p. 136)Second, even if program developers are aware of the relevance of clearly defining outcomes and program processes, they might lack specific knowledge and frameworks to successfully complete this task of outlining and implementing. According to Weiss , construThird, although the program in our study consisted of a cooperation between several partners, the adopted program strategy was not discussed and aligned among these partners. The failure to do so prevented working from shared program aims, supported by all partners. Several examples in the present study show evidence of a lack of integration of the various program activities. A notable illustration relates to the different guidance styles used by different program providers. The male program was characterized by different approaches regarding the method and content of the program . These different approaches had a hampering effect on the general cohesion of the project as coaches withdrew themselves from activities they did not support. In parallel, the interviewees reported that the imbalance in critical support between the different coaches  caused the program to fall short in quality and is therefore a proper example of what Weiss  would deA final determining factor for the lack of a systematic approach concerns the pressure to comply with the requirements of funders. As providers reported pressure from local authorities to provide simple and quantitative evidence and to put a focus on positive results, the present study confirms the pressure of sports-based employability programs to deliver funder-favored numerical outcomes . MoreoveBased on this study, several applied and research implications can be derived. First, as stated, it is advantageous to adopt a theory-based approach when implementing or conducting research into SfE programs. Practically, and in line with a theory of change approach as outlined by Weiss , this imSecond, future work should advance our understanding of generative mechanisms that operate within SfE programs. Generative mechanisms entail the mechanisms that underpin the relationship between complex social interventions and their outcomes . Within Third, when adopting a theory-based approach, it becomes possible for organizations and researchers to test the specified assumptions and measure desired outcomes directly related to the program , 52. To Fourth, in line with the need for a better understanding of how SfE programs work, future work should acquire more specific knowledge and expertise on how SfE programs can organize ‘sport plus’ sessions. As indicated, using sport as a tool to teach soft skills requires programs to go beyond traditional sport sessions and draw on the approach of experiential learning . HoweverThis study has a number of limitations that should be acknowledged. First, the presented findings are based on program providers' perspectives only. Although our aim was to investigate how organizers construct and deliver their program, obtaining information from other stakeholders such as participants and funders might have increased our understanding of how the program was constructed and delivered. Second, as this study was exploratory in nature, we opted for a rich description of a phenomenon . This alTo summarize, the present study was an attempt to gain insight into a particular and already existing SfE program. We specifically explored how it constructed and delivered its program. This question was prompted by the limited amount of research within the area, despite the large and growing interest in using sport to increase employability among NEETs , 88. Ourtessa.commers@vub.be.The datasets presented in this article are not readily available because it concerns confidential data. We consciously opted to keep the selected case and its participants anonymous within this study. Requests to access the datasets should be directed to TC, The studies involving human participants were reviewed and approved by Ethical Committee for Human Sciences - Vrije Universiteit Brussel. The patients/participants provided their written informed consent to participate in this study.TC, MT, and FC jointly conceptualized the study. The data collection and data analysis was conducted primarily by TC, with MT acting as a critical friend and supervisor who provided regular feedback. TC wrote the first draft of the manuscript, with MT and FC providing extensive feedback. All authors contributed to the article and approved the submitted version.Funding was provided through external financing by a Flemish municipality.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


Backgrounds Hereditary transthyretin amyloidosis (ATTRv) presymptomatic subjects undergo multidisciplinary evaluation to detect, as early as possible, a subclinical involvement of multisystem disease. Quantitative sensory testing (QST) that investigates and discriminates the function of C, Aδ and Aβ fibers is included as an instrumental test to monitor nerve fiber function. The purpose of this study was to evaluate the role of QST in the context of the multidisciplinary evaluation in late onset carriers. Methods Four-teen presymptomatic (namely carriers) were enrolled. Subjects underwent thermal  and tactile QST in four body sites: foot dorsum, distal lateral leg, distal thigh, hand dorsum. Results Overall, presymptomatic subject showed a significant difference in all thermal QST findings compared to the control group. All subjects had at least one altered thermal QST finding; the sites more frequently altered were foot and leg, whilst the thermal modalities which were more frequently abnormal were CDT, WDT and CP. Conclusions Our study highlights the importance of performing thermal QST in subjects carrying TTR mutation, given the high frequency of abnormal findings. Notably, performing both innocuous and painful stimulation in foot and/or leg increases the chance of detecting nerve fiber dysfunction. Moreover, the investigation of the hand may provide useful information in monitoring disease progression before the Predicted Age of Disease Onset (PADO). Moreover, we collected demographic data , estimated the Predicted Age of Disease Onset (PADO)  and calcn = 6; −3.5 ± 6.3; −15–4) and >10 years to PADO group . p values lower than 0.05 were deemed as statistically significant. Analyses and graphics were performed with statistical software IBM SPSS Statistic version 25 .Descriptive statistics were based on mean ± standard deviation in the case of numerical variables and on percentage in the case of categorical data. Since the variables had Gaussian distribution (verified through the Kolmogorov-Smirnov test), a Student’s T test was used to compare QST variables between carrier and control groups, between gender and between type of mutation, while a Chi-squared test was used to compare categorial variable (gender) between the two groups. To test if subjects closer to PADO had higher QST thresholds, ANOVA one-way analysis was performed between three groups: controls, ≤10 years to PADO group (We recruited 14 presymptomatic subjects (belonging to 11 families) with no ATTRv symptoms or signs and with normal nerve conduction study. Demographic data and QST findings of ATTRv carrier and healthy controls were summarized in Analyzing the frequency of abnormal QST results (≥+2 z-score) in carrier cohort , all subThe sites more frequently altered were foot (62.3%) and leg (62.3%), while less frequently involved were thigh (47.8%) and hand (44.9%). The sensory modality more frequently altered were CDT (74.1%), WDT (70.4%) and CP (68.6%), whilst less frequently involved was HP (48.1%) .p < 0.05). On the other hand, no statistical differences were found in tactile QST data between groups (p > 0.05). The results of post-hoc Tukey’s HSD Test for multiple comparisons are graphically summarized in ANOVA one-way analysis, to explore if QST results worsened with the PADO approaching, revealed that there was a statistically significant difference in all thermal QST findings between at least two groups (p < 0.05) of CDT and CP at foot, CP at leg and thigh, and WDT, CP and HP at hand site.Briefly, the post-hoc comparisons found that the mean value of all thermal QST findings were significantly different between the control group and the two carrier groups (> and ≤10 years to PADO). Conversely, the post-hoc analysis between the two carrier groups (> and ≤10 years to PADO) showed a significant mean difference  ,14,16,17In this study, we used the QST to further describe the involvement of nerve fibers in a late-onset ATTRv carrier cohort. Our results support the notion that the smallest fiber dysfunction was evident, since all thermal thresholds were higher compared to the control group. These findings support that C and Aδ fibers are precociously involved compared to the larger Aβ fibers (explored through tactile stimuli). Our results corroborate the morphological findings of sural nerve  and skinIn our cohort, all patients showed at least one abnormal threshold in one site. In the context of carrier’s multidisciplinary evaluation, QST allows us to always detect at least one abnormal test among those suggested by European consensus  and to cThe high frequency of abnormal QST findings, especially in the distal sites (foot and leg), appears to be in contrast with previous reports ,14 that Moreover, this study surprisingly demonstrated that Aδ fibers were more frequently abnormal for both innocuous (74.1%) and painful (68.6%) cold stimulations compared to the C fibers that were altered more for innocuous (70.3%) respect to the painful (48.1%) warm stimulations. These results might support that in late-onset carriers the smallest myelinated fibers (Aδ) were more frequently involved compared to the unmyelinated C fibers. To support this, CP was the threshold more sensitive to detecting progression when subjects approached PADO, since in all explored sites CP were higher in < 10 years to PADO group . InteresLastly, the site more sensitive to detecting the progression due to approaching the PADO was the hand since three different sensory modalities  out of four were different between the two groups .In conclusion, this research, although presenting several limitations as a small cohort and the retrospective study design, is the first study focused on the role of QST in a group of carriers of late-onset ATTRv phenotype, including four sites and both innocuous and painful thermal stimuli. Our work suggests that thermal QST, despite the limits of the methodology due to its subjective nature, can be useful in multidisciplinary evaluation of carriers given the high frequency of abnormal findings. Notably, performing both innocuous and painful stimulation in foot and/or leg increases the chance of detecting nerve fiber dysfunction. Moreover, the investigation of the hand may provide useful information in monitoring disease progression before the PADO.