Public involvement is increasingly considered a prerequisite for high-quality research. However, involvement in palliative care is impeded by limited evidence on the best approaches for populations affected by life-limiting illness.To evaluate a strategy for public involvement in palliative care and rehabilitation research, to identify successful approaches and areas for improvement.Co-produced qualitative evaluation using focus groups and interviews. Thematic analysis undertaken by research team comprising public contributors and researchers.Researchers and public members from a palliative care and rehabilitation research institute, UK.Seven public members and 19 researchers participated. Building and maintaining relationships, taking a flexible approach and finding the ‘right’ people were important for successful public involvement. Relationship building created a safe environment for discussing sensitive topics, although public members felt greater consideration of emotional support was needed. Flexibility supported involvement alongside unpredictable circumstances of chronic and life-limiting illness, and was facilitated by responsive communication, and opportunities for in-person and virtual involvement at a project- and institution-level. However, more opportunities for two-way feedback throughout projects was suggested. Finding the ‘right’ people was crucial given the diverse population served by palliative care, and participants suggested more care needed to be taken to identify public members with experience relevant to specific projects.Within palliative care research, it is important for involvement to focus on building and maintaining relationships, working flexibly, and identifying those with relevant experience. Taking a strategic approach and developing adequate infrastructure and networks can facilitate public involvement within this field. What is already known about the topic?● Public involvement in palliative care is impeded by limited evidence on the best approaches to use in populations affected by life-limiting illness.What this paper adds?● We provide a qualitative evaluation of an institutional-level strategy supporting patient and public involvement in palliative care and rehabilitation research.● Public involvement in palliative care research requires a focus on building and maintaining relationships with careful consideration of emotional support when broaching sensitive topics; the ability to work flexibly with people living in complex and unpredictable circumstances; and an emphasis on involving people across the diversity of our field who have experience relevant to the specific research project.Implications for practice, theory or policy● Taking a strategic approach to public involvement within a research institute based on the above principles can facilitate involvement for people with life-limiting illness in research on palliative care and rehabilitation.● Ongoing evaluation of approaches to public involvement is crucial to understand what works well and to minimise unintended consequences.● A collaborative approach to public involvement in palliative care and rehabilitation research across organisations may help progress the quality, diversity and extent of public involvement within this field.1 This can include involving the public to identify research priorities, plan study designs, collect, analyse and interpret data, disseminate and/or implement research findings.1 Public involvement is increasingly considered a prerequisite for high-quality research, with the potential to improve research relevance, quality and impact.2 It is required by most research funders in the UK,4 and is becoming more common internationally.6Public involvement is the process by which research is conducted with or by patients, carers or members of the public, to ensure research remains relevant to the populations in which it is conducted.8 aid recruitment to studies,8 and support dissemination of findings to a wider community.79 Yet, there is a lack of evidence on the best approaches to public involvement in palliative care. Accessing the views of those who may have advanced illness and considerable disability can be challenging, and involvement can be time consuming and resource-intensive for both researchers and public contributors.10 A recent review identified eight broad themes which facilitated and hindered successful involvement within this field.11 These included definitions and roles, values and principles, organisations and culture, training and support, networking and groups, perspectives and diversity, relationships and communication, and emotions and impact.11 However, most involvement in palliative care research to date has been consultative, rather than co-productive, and many of the approaches which have been evaluated have focused only on cancer-specific populations.1012Public involvement within palliative care research can help to develop patient-focused research questions,13 Stakeholders emphasised the following principles: early involvement; flexible involvement; and recognising and promoting the contribution of users.13 These principles formed the basis of a novel three-year (2017–2020) public involvement strategy at our palliative care and rehabilitation research institute. The strategy details a collaborative approach to how public involvement is implemented and evaluated to enhance research productivity, quality and clinical relevance. Alongside the core principles, the strategy commits to key infrastructure to support involvement, including a public involvement coordinator, public involvement training, and an online forum14 , from both the researcher and public perspective, to support continued development of public involvement in palliative care.Our aim was to evaluate this strategy for public involvement in palliative care and rehabilitation research, to identify successful approaches and areas for improvement. Importantly, this was not intended as an evaluation of ‘outcomes’ of public involvement as if it were a complex intervention.16 By co-production, we mean that there was shared input and decision making in developing and undertaking the evaluation, as well as having shared power in the generation of knowledge which came from the evaluation.16 Specifically, public contributors were involved in the formation of project aims and objectives, protocol development, ethics application, development of topic guide, and were heavily involved in data analysis, interpretation and dissemination .We took a co-productive approach to this project, collaborating with three public contributors  at all stages.A palliative care and rehabilitation research institute, UK, comprising approximately 50 researchers and 30 public members.Institute public members were invited via email to participate in a focus group during a routine workshop in June 2018 , and researchers were invited via email to participate in a focus group during a routine meeting in July 2018 . Convenience sampling was used to maximise parti-cipation.16 The topic guide explored participants’ experiences of involvement in relation to the strategy . Written informed consent was obtained prior to audio-recording of the focus groups and interviews.Focus groups for both researchers and public members were facilitated by members of the research team with experience of qualitative research . Field notes were taken during, and following, the focus groups. Two interviews were also conducted with public members who were unable to attend the focus group due to illness and caring commitments. Focus groups and interviews were scheduled for one hour and followed a semi-structured topic guide, which was co-produced by researchers and public contributors.16 Focus group and interview data were transcribed verbatim and pseudonymised. We used NVivo 11 for data management.18 Data were analysed thematically,19 using the key principles of the public involvement strategy as an overarching framework, but coding inductively within these domains, and paying attention to novel themes not considered in the initial framework. To ensure rigour and trustworthiness, researchers  independently examined transcripts to identify initial codes and then held a collaborative meeting with public contributors to refine the coding framework . This framework was applied to the data by researcher (HJ), and double coding of a sub-section of the transcripts was completed by public contributor (MO), to check rigour of application and to ensure interpretation was not dominated by a researcher perspective. Where coding differed between researcher (HJ) and public contributor (MO), discrepancies were resolved through discussion. From coded transcripts HJ and MO identified overarching themes, incorporating public and researcher perspectives. At this stage we considered areas which converged, complemented, and were discrepant as well as instances of silence – where a theme or finding was identified in one group but not the other. Additionally, HJ and MO considered where involvement had worked well and where there were opportunities to enhance involvement. During interpretation we considered how the findings related to the recently developed National Standards for involvement20 as well as relevant theoretical models of involvement.21Analysis was conducted jointly by researchers and public contributors within the research team.Ethical approval was obtained from the King’s College London’s Research Ethics Committee (LRS-17/18-6473). Due to the research team’s relationship with potential participants, and to minimise any perceived pressure to participate, information sheets clearly communicated that participation was entirely voluntary. Although identifiable information on individual participants have been removed, participants were made aware that the institute would be identified when reporting the findings.n = 19) and one focus group (n = 5), one face-to-face interview (n = 1), and one telephone interview (n = 1) with public members. Participant characteristics are provided in We conducted three focus groups with researchers  building and maintaining relationships; (ii) flexible approaches to involvement; and (iii) finding the ‘right’ people see .These themes demonstrated what was working well within the current strategy and associated infrastructure, alongside areas for refinement and improvement, including several practical examples .The public involvement strategy emphasised early involvement and promoting the contribution of public members, but these had become part of a wider theme of building and maintaining relationships. High-quality relationships were characterised by flexible methods of communication, early involvement, infrastructure to support relationships, continuity and emotional support.Overall, public members felt relationships were managed well. Frequent and open communication channels and researchers extending opportunities to be involved made public members feel included, supported and valued, and created a sense of continuity.. . .you very much involve people, you let them know what opportunities there are, those are circulated and communicated, ah-and sometimes you reach out to individuals knowing their skill set, and say you know ‘would you be interested in this’ opportunity, and so I feel very valued (Public member 7)Offering multiple formats of involvement was key to initiating and sustaining relationships, enabling individuals to tailor involvement to their needs. Public members living with chronic illness and/or managing caring responsibilities valued the availability of virtual methods of communication and information transfer. This allowed them to be involved in key project meetings if unable to attend face-to-face and receive key documents without delay, minimising the burden of involvement and increasing accessibility.. . .without the skype and teleconferencing, this might have been a non-starter for me to be honest because I’ve got an illness. . .that’s got its limitations, I’ve got caring responsibilities . . .and, I am really busy that if I was having to come to London all the time, I think it would be too much. (Public member 6)However, some public members reiterated the importance of face-to-face meetings, which were perceived to enable higher quality communication, and the availability of hard copy documents to support readability.The authenticity of the information you get. . .face to face I think is really important. . . I think you learn more and you’re able to modify your views more if you want to when you hear other people sitting around a table. . . I think the quality of that information that the researchers pick up is probably of a higher quality than what you might get from emails and online. (Public member 4)Well my reading email is on my phone, so it’s a nightmare because, I’ve got a laptop but, I’m only half way getting it set up, cause I am not e-minded, so, to me, a piece of paper to read it is, how I like it. (Public member 2)Early involvement in a research study remained key and was perceived by public members to foster more collaborative relationships. This included involvement activities pre-funding, such as developing research questions, plus early post-funding activities including reviewing study protocols and attending ethics committee meetings. At this stage, public members felt they could provide critical input and shape research in terms of its acceptability and relevance. Participants commented that this had improved over the period in which the strategy had been introduced.You’ve got your researchers coming in now, talking about their project pre-funding. . . so at very early stages. . . it’s a bit more like co-design, and there’s more personal involvement . . .so, it is collaborative from the beginning . . . and that is just unbelievably important, because it shapes where you go (Public member 4)There were also challenges of involving people early. Researchers found it difficult to manage the disappointment and expectations of public members if grant applications were unsuccessful. Early involvement was also limited by researchers’ uncertainty around how to fund public members’ time at this stage and limited awareness of funding available from local organisations to support such activities. Public members also felt that more transparent discussions about their role(s) and time/resource required for the project were needed during grant development. This would allow public members to consider their involvement in line with potential disease trajectory and/or caring responsibilities.It would be really good to get the, er, a magnitude er of, of the project, is it a six-month’s program, is it a six-year’s program, it just helps to understand how much involvement is there going to be, and, and, you know, am I going to be alive in that period, er, because that’s important. . . (Public member 3)Having suitable infrastructure in place to support researchers and public members, alongside increasing awareness of external funding and support, was perceived as key to overcoming some of these challenges. At a department level, the core institute public involvement group, public involvement workshops and the public involvement online forum were key to facilitating early involvement.. . . at a structural level. . . I can use the Dragon’s Den, or I can use the award-winning web-based platforms, or I can just ask a colleague and they have a database of people. . .-that sort of infrastructure, as a researcher, is amazing and I’m really grateful cos I think it really helps (Researcher 17)Challenges relating to maintaining relationships were also seen towards the end of projects. Some public members felt ‘abandoned’ when projects ended, and researchers struggled with meeting public members’ expectations about continuing project activities when funding had ceased, and often, employment contracts linked to the project were ending.. . . I’ve immersed myself in a project and then when it comes to end I can feel a little bit abandoned, and that’s something about drawing things to a close. . . especially in a project where you’ve really enjoyed it. . .and you, erm, sort of, got on well with the other PPI [patient and public involvement] reps and the researchers. You don’t really wanna see it disband. . . (Public member 6). . . I feel like-r-we’re coming to the end of our project now, and, we have finite resources . . . funders saying ‘the money’s gonna stop at this point,’ and our PPI [patient and public involvement] are really, really great at ideas . . .and they really want to kind of think about the bigger picture which drives us forward . . . but I find myself caught in a tension between, what’s realistic, and what they want to do.. . .I can’t really commit to anything beyond this project. (Researcher 3)Further integration of public members into the research institute, and core public involvement activities, rather than just project-specific involvement, was suggested to enable continuity when projects ended.My first experience here was just ah-straight in to um the meeting room. . .you were just straight into that project and it was very much that little silo that you worked in, and it was a long long time down the line, coming to other events, where I learnt about who was here, and get a whole feeling and sense of how the institute actually runs in an entity, rather than just coming in on that tiny little bit. (Public member 7). . . putting them into the institute level, full of PPI [patient and public involvement] members. . .so their activities shouldn’t kind of end with a project, or their involvement shouldn’t end with a project, it should continue. I think that’s the way they’re gonna feel sort of valued. (Researcher 19)Finally, public members suggested that further emotional support should be considered to maintain relationships, with the sensitive nature of palliative care research often meaning that involvement could require discussing emotional or distressing experiences. This could include offering a debrief before or after project meetings to ensure any concerns are communicated and discussed.. . .it can bring up a lot of memories from the past, and ah emotions, and that can be quite difficult to manage personally, because you might cope with it in that meeting or that setting, but then it sets off a whole train of thought and sort of sad reflections when you leave that meeting, and um, the impact can stay with you, especially reading transcripts, you need to be very mindful of that, and you know, protect yourself. (Public member 7)Consistent with the core principles of the public involvement strategy, a flexible approach to involvement remained important. Researchers used a variety of communication methods, and utilised infrastructure such as public involvement workshops and the online forum to support flexible involvement.We have adopted a variety of ways to involve them [Patient and Public Involvement members], for example skype, and telephone call. And even, for example, they haven’t, been able to make the meeting, we always send them the newsletters or, our meeting minutes, for them to ev-still like involve in a later subsequent discussion. (Researcher 9)However, discussions also highlighted how researchers needed to think in more flexible and meaningful ways about involvement for their individual projects. Researchers often took a standardised approach to involvement; whereby public members were being included at every stage of the research. However, doing so, without considering the aim or purpose of involvement at each stage was problematic.Potentially, we do a bit of a disservice sometimes when we just include them, without thinking about why, and I’ve definitely been in a room before, including on my own project, when there have been PPI [patient and public involvement] people in the room but I’m not really sure what the purpose is . . .it really hasn’t been beneficial. (Researcher 16)Researchers agreed that there was a need to tailor approaches to involvement to align with the study design and the skills, expertise and development needs of public members involved. For example, a primary data collection study may find it beneficial to have input from public members regarding the recruitment strategy and participant information materials, whereas routine data projects would not require this type of input and involvement may be focused more on, for instance, dissemination of project findings.. . . it makes sense to probably have a slightly different sort of role and format for PPI [patient and public involvement] within secondary analysis because, you already have the data and the data is there, a lot of the input is already, you’ve cut that off, and if you’re collecting primary data, it’s a lot more that, they, you know, that’s really when you need to engage them and so on and so forth. Erm, so it seems to me like, now we’re thinking of it and coming up with ideas and saying, actually, the system should be s-tailored to, I suppose, the study type. (Researcher 14)Having opportunities in meetings or workshops to share good practice and resources, including examples of public involvement across projects, was suggested by researchers as a useful way to build knowledge in different approaches to involvement, fostering greater flexibility.I know that there’s a wealth of ways that people have done that [patient and public involvement] here erm, but as someone who’s new, it’s like ‘ok, ah, exactly how am I gonna do that?’ . . .and what’s the right way to do it? . . . and it might be different for quant and qual. (Researcher 6)So sharing that, I guess sharing what’s worked, what we’ve done and . . . (Facilitator 1)Yeah, what’s already known. And then it could be added to as we move forward and pick up new ways to do things. (Researcher 6)Developing clear aims and plans for involvement in collaboration with public members at an early stage was also key for ensuring involvement was tailored and meaningful. Further, reflecting on and evaluating whether the initial aims of involvement were being met throughout the project, and adapting approaches flexibly was important.. . .have all the objective, including the PPI [patient and public involvement] objective been met? . . . when you’re writing the report, concluding . . .you need to address, ‘this was raised at the, at t-as-one of the aims or, an objective, and this is what was concluded,’ . . . (Public member 3). . .so making a bit more explicit I guess almost the aim of your patient and public involvement and, whether that worked out . . . (Facilitator 1). . . absolutely, yes, and it, in some cases, you may have been able to address, and in some cases, you may not have been, but at least an acknowledgement of that, and, explaining the reason why you could not meet the objectives of PPI [patient and public involvement], ahh, will at least emphasise that, some more work needs to be done. . . (Public member 3)Participants agreed that feedback  played an important role in this. However, public members highlighted that current provision of feedback across projects varied. This inconsistency may be explained by researchers’ uncertainty on how to capture the difference and impact of involvement. Participants noted the more intangible benefits of involvement, which is often challenging to record and quantify. One public member also highlighted how the impact of involvement on research, while sometimes immediate, often produces a domino effect.I would say that, sometimes the PPI [patient and public involvement] member just helps reframe a conversation in a slightly different way, and you, it’s a little bit intangible, the benefit (Researcher 17). . .because that’s the thing with PPI [patient and public involvement], it sometimes doesn’t have an immediate effect on the project that you’re dealing with now, but because other projects follow on, sort of like a domino effect, you never know what you say at one meeting, is going to have an effect several projects down the line. . .(Public member 6)Researchers suggested a need to standardise and embed feedback processes throughout the research cycle, including opportunities to reflect on the contribution of public members, whether the initial aims of involvement have been achieved, and if any improvements were required to facilitate involvement.it’s that process of feeding back . . .and I think, if I was to do that more regularly, that would be a good way of being more mindful about how they [public members] have changed, projects over time (Researcher 18)Participants emphasised the importance of engaging the ‘right’ people for particular research projects, referring to those who had lived experience relevant to the study population, including experience of specific conditions or health services being investigated. While this was not considered within the current strategy, participants perceived it as key to ensuring meaningful involvement and enhancing the acceptability and relevance of research. Despite offering flexible methods of involvement, researchers were regularly not collaborating with public members who had experience sufficiently relevant to their project, often relying on the core public involvement group instead. This also led to the same public members often being involved across projects, limiting the inclusion of new perspectives within the research.I think it also comes down to actually making the effort to, to ensure you’ve got the ‘right’ people . . . I think it’s about not being complacent that, you put an advert out and, four people reply so it’s like. . . great you’ve got four people. . . Have you got the ‘right’ people? (Researcher 7)Participants suggested that more should be done to diversify and expand the pool of public members to ensure it reflected the diversity of people which palliative care serves. This included actively engaging with individuals with different conditions, different experiences and those currently not involved in research. Establishing links with wider groups and networks was perceived as one way to enhance diversity, alongside developing resources to support researchers when identifying people to involve.. . . it’s principally the, the people who are involved i-it’s trying to get all levels of society involved in PPI [patient and public involvement], and I know that’s difficult, and I know it can be unbelievably frustrating - I’ve watched it, but I think if you want to do, a k- a kind of – if you want to be inclusive. . . you need to do-be doing that. (Public member 4)So, diversifying and growing the group so it’s flexible in lots of different directions at different points, that reflect the needs of the institute would be useful sometimes, or at least having the resource to know where to go. . .. (Researcher 12)Due to the nature of palliative care and rehabilitation research, public involvement requires particular consideration of how we build and maintain relationships, work flexibly, and find the ‘right’ people to work alongside. Building and maintaining relationships was essential for providing a safe environment to discuss the emotionally sensitive research topics often prevalent in palliative care and rehabilitation research. Flexible approaches are also needed when involving those living in the complex and unpredictable circumstances of chronic and life-threatening illness. Opportunities for institution-level involvement (e.g. the workshops) alongside project-specific collaborations and infrastructure for in-person and online opportunities, plus open and responsive communication, facilitates this. However, participants noted a need for more careful focus on emotional support and flexible approaches that stem from iterative feedback . Purposeful consideration of how involvement may be tailored to a particular project, and the need to seek out the ‘right’ people to work collaboratively with were considered important. The ‘right’ people were considered as individuals with relevant lived experience of the health condition or service being researched.11 we did not find the principles of public involvement in palliative care research to be entirely different from other research fields. In fact, our data often reflected many elements of the eight themes identified in Chambers et al.’s review,11 and those listed in the broader UK Standards for Public Involvement in Research.20 Rather, our findings identified areas of particular focus when considering the specific context of palliative care and rehabilitation research, and how core strategy and infrastructure can influence these. For example, our findings particularly highlight the need to establish support systems for public members involved in research which may be emotionally demanding. Opportunities for emotional support  should be considered and discussed with public members at the early stages of involvement, as a core part of building and maintaining relationships.In line with Chambers et al.’ s recent review,2426 In palliative care, providing flexible methods of involvement  was seen to be essential to successfully collaborate with individuals living in complex and unpredictable circumstances.12 Our findings, however, also highlighted the need for more flexible approaches within specific projects to help facilitate meaningful involvement. Ensuring embedded feedback processes throughout the project would improve flexibility, by ensuring that involvement could be tailored and adapted over time in response to feedback by public members to researchers (or vice versa), and in response to the needs of the specific project. Similar to previous research, the provision of feedback within projects in this evaluation was variable.27Similar to other studies, our findings showed the importance of the broad principle of flexibility to facilitate involvement.28 and was raised as an important consideration in this evaluation. In palliative care and rehabilitation research, particular care is needed to ensure that those involved reflect the wide diversity of demographics and conditions which palliative care serves. Within research projects, greater attention is needed to find the ‘right’ people; those who have experience relevant to the study population. When done well, this can help improve the relevance and impact of research, but when done poorly this can result in missed opportunities. As such, allocating increased time and resources to this early stage of involvement may help researchers to reach out to a wider diversity of public experience, and involve those who have the most to offer within each project.Greater attention to diversity in involvement in research has been emphasised widely29 and working together across research groups to share expertise and increase reach.30 Having identified how well-intentioned principles (e.g. a focus on early flexible involvement) can lead to unintended consequences (e.g. blanket approaches to involvement without careful consideration of its purpose), ongoing critical reflection will be required. Ultimately, this may improve the quality and relevance of palliative care research for those it is intended to benefit.More widely in palliative care research, participants acknowledged that while areas of good practice exist locally, there is scope for wider sharing of experiences and learning both nationally and internationally. This would not only help reduce duplication, but also provide inspiration of novel approaches to involvement across different project types and institutions. A more collaborative approach to public involvement in palliative care and rehabilitation research across organisations may help progress the quality and extent of involvement, internationally. This might include sharing examples of consumer-led work, which is currently limited in palliative care research and practice,31 We used a mixture of focus groups and interviews (including a telephone interview) to facilitate flexible involvement of public members who were unable to attend a certain time or venue. Moreover, we strengthened our analysis and interpretation by incorporating recent models of public involvement within and outside of palliative care.21We purposefully took a co-productive approach to this study, working in a team of researchers and public contributors. Involvement from the conception of the project allowed for joint input on the study design, ethical considerations and data collection tools. Public contributors’ understanding and experience of the topic being explored, public involvement, aided their involvement in the analysis and interpretation stage, and ensured the work was not limited to a researcher perspective.Due to convenience sampling, using existing meetings, it was not possible to determine the response rate to the study. Although no-one who attended the meetings declined to participate in the study, individuals who did not attend the meetings may have experiences that differ to those who did contribute. Further, as the evaluation was undertaken by researchers familiar to participants, participants may have censored some of their responses. We note, however, that we received both positive and negative responses during focus groups and interviews. Finally, our study took place at a single research institute in the UK where the strategy was originally developed and implemented. Although we anticipate the core principles of successful involvement are likely to be similar, further work is needed to understand which elements of the processes and infrastructure of our public involvement strategy might need adaptations to different contexts.Palliative care research often involves emotionally sensitive topics and diverse populations of people living in complex and unpredictable circumstances. Public involvement in palliative care research therefore needs to prioritise building and maintaining relationships, flexible approaches to involvement, and finding those with relevant lived experience for specific projects. Taking a strategic approach to public involvement within a research institute can facilitate this, but ongoing critical reflection on such initiatives is essential to build on and sustain successes and minimise unintended consequences. Future work is needed to understand the strategies and resources required to support this approach to involvement in different palliative care and rehabilitation research contexts and support wider national and international development of public involvement in palliative care and rehabilitation research.Click here for additional data file.Supplemental material, Supplementary_File_1_-_Focus_Group_topic_guides_1 for Patient and public involvement in palliative care research: What works, and why? A qualitative evaluation by Halle Johnson, Margaret Ogden, Lisa Jane Brighton, Simon Noah Etkind, Adejoke O Oluyase, Emeka Chukwusa, Peihan Yu, Susanne de Wolf-Linder, Pam Smith, Sylvia Bailey, Jonathan Koffman and Catherine J Evans in Palliative MedicineClick here for additional data file.Supplemental material, Supplementary_File_2-_Coding_framework_2 for Patient and public involvement in palliative care research: What works, and why? A qualitative evaluation by Halle Johnson, Margaret Ogden, Lisa Jane Brighton, Simon Noah Etkind, Adejoke O Oluyase, Emeka Chukwusa, Peihan Yu, Susanne de Wolf-Linder, Pam Smith, Sylvia Bailey, Jonathan Koffman and Catherine J Evans in Palliative MedicineClick here for additional data file.Supplemental material, Supplementary_File_3_-GRIPP2_Short_Form_Resubmission for Patient and public involvement in palliative care research: What works, and why? A qualitative evaluation by Halle Johnson, Margaret Ogden, Lisa Jane Brighton, Simon Noah Etkind, Adejoke O Oluyase, Emeka Chukwusa, Peihan Yu, Susanne de Wolf-Linder, Pam Smith, Sylvia Bailey, Jonathan Koffman and Catherine J Evans in Palliative Medicine


For those of us who believe deeply in a collaborative relationship between patients and doctors, the chaos created by the COVID-19 pandemic has brought an uncomfortable question to the fore: Is participatory medicine still relevant during a pandemic? Drawing liberally upon the Jewish tradition of Talmudic reasoning, I would like to offer 3 considered replies: “Yes,” “no,” and “it depends.” Sometimes, patients may have no choice but to cede control to medical professionals, even though patients are still the experts on their own lives. Other times, the shared control of participatory medicine is both an ethical and clinical imperative. However, as the worldwide toll exacted by COVID-19 has made us grimly aware, no one is really in control. That is why, in these uncertain times, the path forward requires maintaining mutual trust between health care providers and patients, whatever the circumstances. After all, it is our bodies and our selves at stake. Consider the following scenario: you are at home with your significant other, and that low-grade fever you have been running is rising. You have also developed a persistent cough, and you are losing your sense of smell. In a panic, you phone your doctor, who tells you to go immediately to a special area set aside for patients with COVID-19 by your local hospital. He will call ahead.“Thank you so much, doctor,” you blurt out. Yet even in your dazed state, you know there are a few more questions you just have to ask.“As an informed consumer,” you begin, “I’m wondering if the hospital’s online price list includes COVID-19 treatment costs.” You add, “I also want to make sure the emergency room physicians understand that I expect to be a partner in the coproduction of my care , but I’mYour significant other violently grabs the phone and starts apologizing profusely. “I’m really sorry,” they tell the doctor. “That must be the fever talking. We’re on our way to the hospital right now.”For those of us who believe deeply in a collaborative relationship between patients and doctors, the chaos created by the COVID-19 pandemic has brought an uncomfortable question to the fore: Is participatory medicine still relevant during a pandemic? Drawing liberally upon the Jewish tradition of Talmudic reasoning, I would like to offer 3 considered replies: “Yes,” “no,” and “it depends.”Let us start by expanding on the “no” answer. The term “participatory medicine”  containsNeither of these assumptions is invariably true. Someone rushed into surgery after a serious accident, for example, is in no position to speak up with a perspective on how the surgeon should close their wounds, nor would it be helpful to engage in shared decision making about staples versus sutures. A drug allergy or similar critical information that a family member or caregiver might provide is, of course, a different matter. Unfortunately, caring for a SARS-CoV-2 infection still too often feels like an emergency. There is extraordinary stress not just on patients and family, but also on the medical staff, as they, too, worry about getting sick or infecting others . PerhapsTo be clear, the term “participatory” in this context does not mean the kind of public health actions that everyone, infected or not, should be participating in . Rather, it refers to the deliberate incorporation of the patient voice into the national COVID-19 response. For instance, in June 2020, the National Institutes of Health launched a data analytics platform, and its usefulness will ultimately depend upon Americans with COVID-19 sharing their electronic health record information. This platform was opened to researchers in September 2020, and it incorporates analytic techniques that are designed to examine everything from potential COVID-19 risk factors to the effectiveness of different therapies .The pandemic has prompted clinicians, employers, and public health officials to ask individuals to self-monitor at different times, such as when they are healthy, if and when they become symptomatic, and during recuperation. Digital tools for web-based symptom checks and consultations are key components of combating the COVID-19 pandemic . As a reTo give an example of the central role being played by patient-generated health data, Fitbit Inc has developed a COVID-19 symptom tracking service called Ready for Work. It lets employees self-report their symptoms on a Fitbit device and lets employers see worker information on a central dashboard. This helps employees and employers determine when it is safe to return to the workplace. Preliminary data from a nonpeer-reviewed Fitbit study written in mid-August 2020 has suggested that “hospitalization risk can be calculated from self-reported symptoms, and relevant and predictive physiological signs related to COVID-19 may be detected by consumer-wearable devices” .Similar initial results were reported at the end of October 2020 by the Scripps Research Translational Institute in its DETECT  study, which involved large-scale epidemiological research on consumers who use a wide range of smart wearables . AdditioClinical uncertainty about COVID-19 has also given birth to online patient support groups, such as Survivor Corps, COVID-19 Recovered - Survivors, and the Body Politic COVID-19 support group. Participation in these support groups now includes tens of thousands of individuals around the globe. As Fiona Lowenstein, founder of one such group, wrote in Vox, “As we wait for institutions to catch up with a new and fast-moving virus, parallel forms of information-sharing via communities, personal stories, and support groups…have become crucial” .Now that we have seen the “yes” and “no” arguments for the relevance of pandemic-era participatory medicine, here is the last answer: “It depends.”Listen for a moment to a woman named Byllye Avery recounting what she learned from fellow patients after a traumatic encounter with the health care system. “If you don’t know how to take care of yourself, you are basically ignorant,” she wrote. “And health information,” she added, “had to be shared within the context of one’s life. [There was a] right to have medical information and…patient participation.”"Our Bodies, Ourselves". Avery, who later founded the Black Women’s Health Imperative, shared her memories in a preface to that groundbreaking book’s 25th anniversary edition [That advice was given to Avery nearly a half century ago at a 1971 women's health meeting whose attendees were part of a group that authored the book,  edition . As note edition .Although it has taken decades, the health care system has finally accepted the principles of participatory medicine as valid. However, putting all these principles into practice may not always be possible, especially at a time when patients, their friends and loved ones, and health care providers are all extraordinarily stressed by the mortal threat of a dangerous and incompletely understood pandemic. Health care, like much of life, is complicated and messy. Evolving circumstances dictate the appropriate response. Nonetheless, whether during a period of pandemic or relative placidity, certain bedrock principles must remain part of the relationship between professionals and patients. These principles include honesty, mutual respect, and the mutual sharing of information. Such sharing needs to continue, even when it means admitting to uncertainty, fear, guilt, or other uncomfortable emotions.Sometimes, patients may have no choice but to cede control to medical professionals. This decision, though, does not relieve medical professionals of the obligation to listen to patient and family concerns; patients are still the experts on their own lives. Other times, the shared control of participatory medicine is both an ethical and clinical imperative . However


High-volume ventricular tachycardia (VT) ablationists are often asked what their chosen “technique” is for VT ablation. Should VT be induced and mapped during tachycardia, or should a substrate-based approach be undertaken instead?The inherent problem in this question is that every patient, every VT, and every scar are different. As physicians, we like to have clear “black or white” answers to clinical questions and to be able to state that the “right way” to do a procedure is “X”—in effect, do A, B, and C, and you will obtain the same result every time. However, the reality is that the difference between success and failure in a highly complex VT ablation may come down to experience and the willingness to think about all of the possible critical regions for a given VT or VTs, in a given scar, in a given substrate, in a given patient.The Journal of Innovations in Cardiac Rhythm Management, we electrophysiologists generally like to label VT origin by the 12-lead electocardiogram morphology. However, QRS morphology only informs us of the exit site, and ablation that is performed near an exit may not in fact yield lesions located anywhere near the critical isthmus in a large scar.VT doesn’t always behave rationally. A VT that exits on the epicardial lateral left ventricle (LV) may have perfect pacemaps on the endocardial septal LV but have a critical isthmus in the midmyocardial anterior LV, for example.Regardless of whether mapping in VT or employing a purely substrate-based approach, without the performance of comprehensive mapping, critical regions may be missed. Sohinki et al.Additionally, new techniques can often elucidate substrates not seen with standard voltage mapping. Isochronal late-activation mappingIn the end, there is no “right way” to ablate VT. Often times, a combination of substrate and VT mapping is required. Comprehensive mapping, operator experience, and a thoughtful assessment of the data available can lead to improved outcomes.


There is a stark disparity between the number of patients awaiting deceased-donor organ transplants and the rate at which organs become available. Though organs for transplantation are assumed to be a community resource, and the organ supply depends on public willingness to donate, current allocation schemes do not explicitly incorporate public priorities and preferences. This paper seeks to provide insights regarding the Israeli public’s preferences regarding criteria for organ  allocation, and to determine whether these preferences are in line with current allocation policies.A market research company administered a telephone survey to 604 adult participants representing the Jewish-Israeli public . The questionnaire comprised 39 questions addressing participants’ knowledge, attitudes, and preferences regarding organ donation and criteria for organ allocation, including willingness to donate.The criteria that respondents marked as most important in prioritizing waitlist candidates were maximum medical benefit (51.3% of respondents) and waiting time (21%). Donor status  was ranked by 43% as the least significant criterion. Most participants expressed willingness to donate the organs of a deceased relative; notably, they indicated that they would be significantly more willing to donate if organ allocation policies took their preferences regarding allocation criteria into account. Unlike individuals in other countries  who responded to similar surveys, Israeli survey respondents did not assign high importance to the candidate’s age (24% ranked it as the least important factor). Interestingly, in some cases, participants’ declared preferences regarding the importance of various allocation criteria diverged from their actual choices in hypothetical organ allocation scenarios.The findings of this survey indicate that Israel’s citizens are willing to take part in decisions about organ allocation. Respondents did not seem to have a strict definition or concept of what they deem to be just; yet, in general, their preferences are compatible with current policy. Importantly, participants noted that they would be more willing to donate organs if their preferences were integrated into the allocation policy. Accordingly, we propose that allocation systems must strive to respect community values and perceptions while maintaining continued clinical effectiveness. Organs for transplantation are a scarce resource. In many countries, there is a stark disparity between the number of patients awaiting deceased-donor organ transplants and the rate at which organs become available  , 10, polThe current study focuses on an element that remains noticeably absent from this debate and from the allocation decisions made in practice: namely, public perceptions and opinions regarding organ allocation criteria. Owing to the fact that the organ supply depends on public willingness to donate, and given that the organ shortage can be defined as a public health problem or at least a critical challenge, it can be assumed that deceased-donor organs for transplantation are a community resource or public goods. In other words, people have a shared interest in ensuring that donor organs are available to all, and thus would be better off cooperating with regard to the provision and allocation of such organs. Therefore, it seems that allocation policies should, to some extent, consider community preferences regarding which factors should be prioritized in allocation decisions . Though It is worth mentioning that, in the US and in the UK, the public does have a formal role in health-related policy-making. In particular, in the US, the public can comment and provide feedback on policy proposals put forward by the US Health Resources and Services Administration. Moreover, the public’s comments and remarks form a vital part of the policy development process. Significantly, as a means of learning about public concerns, the Department of Health and Human Services hosts forums, public hearings, and summits that allow community members to share ideas, with the aim of assisting the department in identifying areas for improvement , 18. In To begin to address the lack of knowledge on public opinions regarding criteria for organ allocation, we carried out a survey to systematically measure such opinions among members of the Jewish-Israeli public. Specifically, we focused on allocation of kidneys, the organ type with the highest proportion of patients on waiting lists . In IsraIn our survey, we sought to achieve the following goals: (1) to evaluate the general public’s knowledge and understanding regarding organ transplantation in general, and kidney transplantation in particular; (2) to examine which criteria the community prioritizes in the organ allocation process—both in terms of declared preferences, and in terms of hypothetical allocation decisions ; (3) to assess whether taking public opinion into account in a kidney allocation policy has the potential to increase willingness to donate organs. In evaluating the results of the survey, we devoted particular attention to the question of whether current organ allocation policies are consistent with societal values and public preferences. We note that an abundance of surveys conducted in countries such as the US, the UK, and Australia have suggested that the vast majority of the public is willing to sacrifice some degree of medical efficiency of transplantation programs in exchange for an increase in equity or fairness in the allocation of donor organs , 26–28. We note that several studies have previously been conducted in Israel to shed light on various aspects of the public’s perception of organ donation. These studies investigated the attitudes of a specific group towards organ donation , immigration status , level of religiosity (secular and orthodox) and socio-economic level. Sampling was done so that the probability of each statistical area to be included in the sample was proportional to the size of the population in the area. Such sampling ensures representation of various population groups, particularly those with a relatively small proportion.Mage = 49.31, SD = 16.76); half the participants were male.The market research company approached 1438 households. Among these selected households, 300 people were unavailable when the survey was executed ; 526 people refused to take part in the survey; and 8 participants only partially answered the survey questions, so they were excluded from the research. Therefore, the survey pool included 604 Israeli participants (42% response rate), ranging in age from 18 to 95 years  data.n = 226) declared they are organ donor-card holders. Other features that characterize the survey pool are as follows: 79% of the participants are parents to children, 34% have an above-average income, and 29% have below-average income.Table Eighty percent of the participants specified that they were aware of most or all of the information presented at the beginning of the survey regarding the repercussions of renal failure and comprehended the customary treatment methods. Fifteen percent noted that they were aware of some parts of the information, while only 5% stated that they were not at all aware of the information.Figure When asked to identify the criterion that should be attributed the least significance in allocation decisions, 43.9% of respondents chose the recipient’s donor status . Contrary to expectations, respondents did not indicate that they perceived recipient age as a criterion that should be attributed high importance in organ allocation. In fact, 24% of the interviewees ranked it as the least important factor in determining transplantation priorities. Fig. Notably, the declared preferences of respondents who were registered donors were similar to the preferences of respondents who had not signed a donor card. Specifically, among registered donors, 43.2% chose the recipient’s donor status as the least important attribute to be considered. This finding is surprising in light of the fact that, as noted above, Israel’s allocation policy prioritizes registered donors, as an incentive to increase willingness to donate.χ2 = 45.62, p < 0.001). Moreover, participants significantly prioritized an older patient who had signed an organ donor commitment (36%) over a younger candidate who had not committed to organ donations  significantly higher than they ranked waiting time , (t (603) = 4.72, p < .002). Yet, when asked to allocate a kidney to one of two patients—patient A, who has a 70% chance of a successful transplant and has been waiting 4 years, and patient B, who has a 90% chance of a successful transplant and has been waiting only 1 year . This decision seems to violate their own declared preference.Nevertheless, we did observe several discrepancies between declared preferences and the hypothetical allocation decisions. For example, when assigning scores to the various criteria, participants generally ranked maximum benefit  = 8.4, p < 0.001). Yet, when asked to choose between a patient who was a registered donor with a 70% chance of receiving another kidney in the coming year versus a non-registered donor with only a 30% chance .Moreover, as noted above, participants seemed to assign low priority to the candidate’s status as a registered donor, but their decisions in the trade-off scenarios revealed a more nuanced picture. For example, when rating the various criteria on Likert scales, participants assigned a significantly lower rating to donor status than to the likelihood of finding another kidney in the coming year , with registered donors expressing significantly greater willingness to donate  compared with participants who had not signed a donation commitment  = 9.10, p < .001).Participants’ willingness to donate a deceased relative’s organs was relatively high, 64% indicated that they would definitely agree, and 26% said that they would probably agree (but note that 16% did not want to answer this question)  = 9.53, p = 0.002). For example, among 48 respondents who were reluctant to donate their loved one’s organs , 18% expressed higher willingness to donate if their preferences were incorporated into the organ allocation policy.Interestingly, participants indicated that they would be significantly more willing to donate a relative’s organs if the criteria they had specified as most important were integrated into the allocation policy . However, when asked to rate their willingness to sign an organ donor commitment given that their preferences will be taken into account in the allocation policy, participants’ willingness to sign the card was significantly higher  = 4.80, p < .001).As noted above, 61.1% of the participants were not registered organ donors. When asked about their willingness to sign an organ donation commitment in the near future, participants expressed relatively low intention to sign a commitment . In scenario 6, which traded off organ donation commitment against waiting time, the public assigned lower weight to organ donation commitment than the point system did.This comparison suggests that, in most cases, the views of the public are in line with those of the National Transplant Center. In particular, both the public and decision-makers assign high importance to maximum  benefit and acknowledge the importance of waiting time. Nevertheless, we do observe some divergence. For example, the public attributes lower importance to recipient age than the point system does (scenario 1). The public’s decisions also illustrate their somewhat “fickle” perceptions of the significance of organ donation commitment: For example, in scenario 4, which traded off organ donation commitment against the likelihood of receiving another kidney, the public assigned Participation in the survey was voluntary. Interviewees were not incentivized to take part in the research and were contacted during their free time. They were asked to confront hard questions, judge difficult scenarios, and share deep insights. We suggest that the fact that 604 people (42% response rate) from all walks of life decided to dedicate their time to tackle questions that are not mundane for the layman shows that the general public is interested in and ready to answer profound and complex questions regarding organ donation issues. This proposition is in line with findings of surveys of other populations e.g., , 28). Mo. Mo28]).In interpreting the survey’s results, it is important to acknowledge potential differences between Israel’s population distribution and the characteristics of the survey population. The extent of divergence varies across different parameters. Specifically, participants’ countries of origin are in line with those of Israel’s general population, with 75% of survey participants stating they were born in Israel . Likewisn = 226), whereas only 14% of adults in the general Israeli population  have signed an organ donor card )At first glance, our survey results might seem to suggest that people were more willing to donate the organs of a deceased relative than to sign an organ donation commitment card. However, a closer look reveals that in comparison to participants who had not signed a donation commitment, registered donors expressed considerably greater willingness to donate the organs of a deceased relative. Hence, the differences between the results are not substantial.In spite of the study’s contribution to the understanding of the Israeli public’s preferences and beliefs regarding organ allocation, it has several limitations. First, the number of scenarios included in our survey was chosen according to the recommended length for a telephone survey. Obviously, these examples are limited and can only provide initial ideas regarding the directions of potential discrepancies between declared policy and specific allocation decisions. Second, we note that in the trade-off scenarios, participants were not forced to make a binary choice between the two candidates described; that is, they could select a “no preference” option and thereby avoid making a decision. Yet, in real life, due to organ scarcity, medical professionals do not have the privilege to avoid deciding between two equally deserving transplantation candidates. It would be interesting for future surveys to omit the “no preference” option and to observe participants’ responses when forced to make a choice, as in the real world. Third, the study’s population was composed of the general adult Jewish population in Israel. We decided to focus on the Jewish population since we assumed that cultural and religious differences might impact preferences and standpoints. The Arab citizens of Israel constitute about 20% of the population and are an integral part of the country, and therefore their stances and attitudes regarding organ allocation are of immense importance. Future studies ought to examine and evaluate Arab citizens’ values and beliefs. Fourth, since Israel is one of the first countries to implement non-medical criteria in organ allocation, the survey particularly focused on the population’s stance regarding the candidate’s status as a registered donor. In future studies, more balanced trade-off scenarios should be displayed. Fifth, when composing the trade-off scenarios, we did not notice that we had designed several scenarios in which, according to the point system decision, the score for both patients was identical  when they feel that their own voices are heard. Accordingly, we propose that the public should be allowed to seek clarification, engage in discussions, express their views, and listen to other opinions and that in turn, decision-making entities should take community preferences into account when formulating their guidelines. Though we are aware that it is infeasible for a complex system to perfectly reflect the priorities of all stakeholders, ultimately, we propose that an optimal allocation system must strive to find a balance that respects community values and perceptions while maintaining continued clinical effectiveness , 44. Mor


The results showed a clear difference between ASD children and TD children in the how, but not in the what, condition. Indeed, while TD children distinguished the vitality forms to be expressed by mostly varying a specific spatiotemporal parameter (i.e. movement time), no significant variation in this parameter was found in ASD children. As they are not prone to express vitality forms as neurotypical individuals do, individuals with ASD’s interactions with neurotypical peers could therefore be difficult to achieve successfully, with cascading effects on their propensity to be tuned to their surrounding social world, or so we conjecture. If this conjecture would turn out to be correct, our findings could have promising implication for theoretical and clinical research in the context of ASD.The notion of “vitality form” has been coined by Daniel Stern to describe the basic features of action, which may reflect the mood or affective state of an agent. There is general consensus that vitality forms substantiate social interactions in children as well in adults. Previous studies have explored children with Autism Spectrum Disorder (ASD)’s ability in copying and recognizing the vitality forms of actions performed by others. In this paper we investigated, for the first time, how children with ASD express different vitality forms when acting themselves. We recorded the kinematics of ASD and typically developing (TD) children while performing three different types of action with two different vitality forms. There were two conditions. In the  Daniel Stern coined the term of “vitality forms” claiming that their expression and recognition substantiate social interactions in children as well in adults, providing them with a primordial way of relating to each other5. Vitality forms could therefore offer an almost unique opportunity to explore core social skills, even when their development is not typical6.Our actions may take distinct forms according to our mood or affective state. For instance, our grip can be vigorous or delicate, our caress can be gentle or rushed. These action forms have been variously conceived by different researchers8. Previous studies found that children with ASD show selective difficulties in imitating and recognizing vitality forms expressed by others12. For instance, children with ASD were observed to have more difficulty than typically developing (TD) children in performing an imitation task which requires copying the vitality form of an observed action (e.g. its being gentle or forceful), although they did not differ from TD children when performing an imitation task which requires copying the goal of an observed action10. A similar dissociation between vitality form and action goal was found in a recognition task. ASD and TD children had to judge two observed actions as the same or different with respect to their goals or their vitality forms. The results showed that both ASD and TD children were able to identify the goals of the observed actions. However, unlike TD children, ASD children mostly failed to recognize which vitality form characterized these actions11.Autism spectrum disorder (ASD) is a neurodevelopmental condition with heterogeneous clinical manifestations. These include difficulties in the social domain, verbal and nonverbal communication, and patterns of restricted and repetitive behavioursAlthough these findings suggest that vitality form may contribute to a better understanding of social interaction difficulties in ASD, there are no studies investigating the way in which children with ASD express their own vitality forms when acting themselves, rather than observing someone else acting. The main aim of this paper is to fill this gap.how actions unfold in space and time, with each vitality form being characterized by a specific kinematic “contour”5. In order to assess whether and how such contours individuate action forms in autism, we compared upper limb kinematics of a group of 14 ASD children  to a control group of 14 TD children  (Table gentle and rude) in two separate experimental sessions. Indeed, in a first session ASD and TD children had to perform the three different actions gently, while in a second session the same actions were performed rudely, or vice-versa.According to Stern’s notion, vitality form primarily refers to what and how, respectively. In the what condition we contrasted the three different types of action  performed with a same vitality form (gentle or rude), while in the how condition we contrasted the same action  performed with two different vitality forms (gentle and rude). To focus on potential differences in expressing vitality forms in ASD, one should expect to find any dissociation between groups in the how condition, and substantial similarities in the what condition. And this was what we actually found. The implication of this finding for better understanding social cognition in ASD will be discussed.ASD and TD children’s kinematics was analysed in two different conditions: X, MaxDY, MaxDZ) . Concerning the Cartesian axes, X corresponds to the dorso-ventral axis of the body, Y to the vertical axis of the body, and Z to the lateral axis of the body , whereas the second measured the amount of variation of each parameter when the same type of action was performed with the two different vitality forms . Intra-group and inter-group analyses were carried out.Upper limb kinematics of ASD and TD children were analysed by means of the following kinematic and spatiotemporal parameters: movement time (MT), peak velocity (pV), peak acceleration (pA), peak deceleration (pD), time to peak velocity (TpV), percentage of time spent in acceleration (T%Acc), percentage of time spent in deceleration (T%Dec), maximum displacements along the three Cartesian axes  was higher than in the what condition (|what| index), suggesting that this parameter was used by the control group mainly to modulate different vitality forms rather than different types of action (p = 0.005). The pV, pA and pD were also mainly used to modulate different vitality forms; indeed the |how| indexes were significantly higher than the |what| indexes for all three of these parameters . No differences between the two conditions emerged in the amount of variation of temporal parameters, such as TpV (p = 0.357), T%Acc (p = 0.816) and T%Dec (p = 0.545). This suggested that these parameters varied across different vitality forms to the same extent as they vary across different types of action. Amount of variation of MaxDX was lower in the how (|how| index) than in the what condition (|what| index) (p = 0.007). Therefore, the amplitude of MaxDX was mainly varied across different types of action performed with the same vitality form (what condition). No differences emerge between the two conditions for MaxDY (p = 0.592) and MaxDZ (p = 0.567) , pA , pD  and MT  were the only parameters explained with a moderate or reasonably high goodness of fit. On the contrary, when using “type of action” as predictor, no parameter was explained with R2 > 0.25. Extended results of linear regression analyses are reported in Supplementary Table Using “vitality form” as predictor, linear regression analysis showed that pV (Rhow| index Vs. |what| index) of most parameters were similar to the control group. More specifically, in the ASD group, the parameters pV, pA, and pD  vary between different vitality forms (how condition) more than they do between different types of action (what condition), as found in the TD group. Furthermore, as in the TD group, variations of temporal parameters such as TpV (p = 0.214), T%Acc (p = 0.080) and T%Dec (p = 0.091), were not significantly different between the two conditions. The same was found for MaxDY (p = 0.899) and MaxDZ (p = 0.421), which did not vary differently between the two conditions. Strikingly, things were different for two critical parameters, that is, MT and MaxDX. Contrarily to the TD group, in the ASD group the amounts of variation of both MT (p = 0.840) and MaxDX (p = 0.901) were not significantly different between the how and the what conditions , pA , pD  were the only parameters explained with a moderately high goodness of fit. Contrarily to what emerged for the TD group, “vitality form” does not explain the model of MT (p = 0.107). This means that “vitality form” explained pV, pA, and pD data in both groups in a comparable way, but MT was predicted only in the TD group. As in the TD group, when using “type of action” as predictor, no parameter was explained with an adequate goodness of fit . Extended results of the linear regression analyses are reported in Supplementary Table Using “vitality form” as predictor, linear regression analysis showed that pV .For each parameter, the |what| index, no significant differences were found between the two groups . This indicates that there were no differences in the kinematic and spatiotemporal parameters between ASD group and TD controls that can be attributed to the execution of different types of action (what condition) (see Table how condition. A higher variation of the pD parameter (p = 0.014) was reported for the TD group compared to the ASD group. The result of the pD parameter simply suggested a “quantitative” inter-group difference . This was also corroborated by regression analysis, in which “vitality form” is a regressor factor of pD parameter both for TD  and ASD  participants . This was consistent with what we found in the intra-group analysis: in the TD group, a significant difference was reported between the how condition and what condition for this parameter, while in the ASD group no significant difference emerged (Table Y (p = 0.918), MaxDZ (p = 0.229) and temporal parameters such as TpV, T%Acc and T%Dec .When considering the |how| index one could have expected an inter-group difference also in the modulation of MT, but this was not the case. Indeed, the intra-group analysis showed a significant difference between the how and what conditions in the TD group (p = 0.005), but this difference did not emerge in the ASD group (p = 0.840)  taking into account the “direction” of the modulation in the how condition. Graphically, this modulation of the MT parameter was clearly shown by the individual distributions (“rain”) in the raincloud plots , with higher values in the TD group. As expected, results on MaxDX parameters were consistent between the |how| index and the howSign index   Table . HoweverA warning should be reported. Caution is needed when dealing with data that should be replicated in further studies. This is generally true with any experimental approach, but it seems particularly urgent for kinematic and motion capture studies considering potential intra- and inter-subject(s) variability. Our results should therefore not be overestimated or oversimplified.how  than in the what condition . A graphical representation of this tendency is reported in Fig. Statistical Analysis Section). Regression analysis corroborates this finding, by showing that in both groups the explained variation in the data  was globally higher when “vitality form” was selected as predictor  with two different vitality forms (gentle and rude). Participants’ kinematics were generally found in both groups to be modulated more in the ta i.e. R values wX were differently modulated in ASD and TD children . Indeed, in TD children, MaxDX significantly varied more in the what condition compared to the how condition, while MT significantly varied more in the how condition, reflecting the difference in expressing vitality forms. Strikingly, children with ASD did not present any significant variation in either parameters (MaxDX and MT) between the conditions . It is also true that recent literature exploring motor functioning in infancy and childhood has not found a general consensus in ascertaining motor dysfunctions in ASD, but instead describes a complex picture of similarities and differences as compared to neurotypical controls24.It is true that there is a certain clinical agreement regarding a somehow generic reference to atypical aspects of motor functioning in ASD. This is certified by classical studies in the literaturewhat condition. But this was not the case . This was not surprising, giving the type of actions all the participants had to perform, that is, moving a bottle to a fixed point, throwing a ball, and giving a packet of crackers . In contrast with TD children, participants with ASD did not capitalize on a specific parameter in expressing their vitality forms. Indeed, they modulated both parameters MaxDX and MT in a similar way in the what and how conditions. Although ASD participants clearly understood our instructions and tried to implement different vitality forms , ASD children’s kinematic strategies were similar to those identified in the what condition .Taken together, our findings indicate that TD children took advantage of two different parameters in the two different conditions. In the ers Fig. . In the what and how to unfold an action to be performed, thus reflecting the action goal and one’s own internal mood, respectively. In other words, ASD participants would not represent the how independently of the what, that is, they would not represent vitality form as an action component different from an action goal. This hypothesis seems to be in line with previous evidence suggesting that children with ASD show selective difficulties in imitating actions with different vitality forms.How can this difference between TD and ASD children in vitality form expression be explained? A likely hypothesis is that, in contrast with TD, motor parameters in ASD would not be settled in such a way as to flexibly represent 9 explicitly contrasted the what and the how by asking a group of children and youths with ASD and TD controls to imitate, after a delay, some observed actions performed with different vitality forms. For instance, participants had to imitate the experimenter holding a pipe rack against the upper part of his left shoulder and running a wooden stick across it with his right hand, as if he were playing a violin. The experimenter strummed the stick over the pipe rack in either a rude or a gentle way. The results showed that while the TD controls imitated both the what and the how of the observed actions, participants with ASD were able to imitate the goal of the action only. Indeed, they had no difficulties in applying the stick to the pipe rack, but mostly failed in reproducing the corresponding vitality form. Similar results have been obtained by a subsequent study in which ASD and TD participants were asked to imitate actions performed with different vitality forms10. Groups did not differ in their ability to copy the action goals, even when accomplishing the goals required relatively complex movements. On the contrary, their imitative performance was significantly different when the vitality forms had been copied, with participants with ASD showing difficulties in imitating the vitality forms of the observed actions, especially when they were incidental to accomplishing their goals.In a first study Hobson and Leeprimarily to their inability to represent the how independently from the what when they have to perform an action themselves. This could also shed new light on vitality form recognition difficulties in ASD. Rochat and colleagues11 asked ASD and TD children to judge whether two observed actions were similar/different relative to either their vitality forms (how task) or their goals (what task). The results indicated a dissociation between the two tasks, with children with ASD exhibiting difficulties in the how, but not in the what, task.If our hypothesis turned out to be right, the difficulties of participants with ASD in vitality form imitation would be likely to be due 28. In particular, it has been shown that acting, imaging acting and observing someone else acting with a given vitality form share the recruitment of a dorso-central portion of the insula27, which is known to receive information on the affective state of an agent30 and to modulate the parieto-frontal circuits directly involved in representing and executing actions31.It is tempting to link these reported difficulties concerning individuals with ASD in vitality form recognition to their differences in representing and expressing vitality forms. Indeed, there is evidence that vitality form recognition may elicit, in the observer’s brain, processes and representations similar to those involved in vitality form expression32. However, our data indicate that individuals with ASD do not represent and express their vitality forms as neurotypical individuals do. So, when observing someone else acting, they may be less inclined to rely on their own processes and representations of the corresponding vitality forms. This could explain why they had difficulties in recognizing them when expressed by others.Accordingly, observing someone else acting may involve a transformation of the sensory information concerning the observed vitality form into processes and representations which would occur if the observer were expressing that vitality forms herself, and this would allow her to recognize it, by tracking another’s corresponding mood33). For instance, Cattaneo and colleagues34 found that children with ASD did not represent motorically a sequence of basic actions (e.g. reaching-grasping-putting into the mouth) as chained to each other in virtue of their means/end relations (reaching for grasping for putting into the mouth) both when they were performing those actions themselves and also when they were observing someone else performing them . Strikingly, Boria and colleagues37 showed that children with ASD had no difficulties in identifying single action goals such as taking a glass , but they did show difficulties in understanding why those actions had been performed (e.g. taking a glass for drinking or moving away). These difficulties were especially pronounced when understanding required taking into consideration specific motor cues (i.e. object affordances).It is worth noting that motor cognition  have been shown to be anomalous in ASD by previous studies , and 14 children  for TD group. Left-handed individuals, as indicated by a questionnaire adapted from the Edinburgh Handedness Inventory40); (2) absence of pharmacological therapy; (3) normal or corrected-to-normal vision and hearing; (4) absence of co-diagnosis of Developmental Coordination Disorder (CDC) or other major behavioural disorders ; (5) absence of any other neurological and psychiatric comorbidity. ASD participants were recruited at the Child Psychopathology Unit of the Scientific Institute IRCCS MEDEA . Clinical diagnosis of ASD was made by licensed clinicians with specific experience in the assessment of ASD in terms of DSM-IV diagnostic criteria and the Autism Diagnostic Observation Scale (ADOS). For the control group, parents reported no prior history of psychiatric or neurological disorder. TD participants’ cognitive level was screened with 2 subtests of the WISC-III that test verbal and visuo-perceptual abilities . No statistically significant difference between groups is reported neither for block design (t(26) = 0.37, p = 0.71) nor for vocabulary (t(26) = 1.94, p = 0.06) subtest. Comprehension receptive skills were also tested with the Peabody Picture Vocabulary Test (PPVT-R), and performance do not differ between ASD group and controls (t(26) = 0.96, p = 0.35) (see Table Inclusion criteria for the ASD group were: 1) full scale IQ ≥ 70 as measured by the Italian version of Wechsler Intelligence Scale for Children-Revised  and was conducted according to the Declaration of Helsinki. Participants’ parents or legal guardian(s) signed the informed consent.(I) placing a bottle initially located in a specific point  on another point on the table along the forward axis ;(II) throwing a ball initially located in a specific point  on the table along the forward axis ;(III) giving a packet of crackers initially located in a specific point  along the forward axis (without placing it on the table).The experiment was conducted in a quiet room, where participants were seated at a table facing the experimenter  with their right hand resting on a fixed point of the table. They were asked to perform three different types of action with their right hand  the first one, from the “hand starting position” to the “object initial position”, correspondent to the reaching phase;(b) the second one, from the “object initial position” to its end position, correspondent to the final phase of the action;Each action was split into two motor acts:27), and accordingly to a pilot study, which confirmed that those ones were the most suitable in our experimental setting . All objects were initially located  at a distance of 12 cm from the starting position (= “hand starting position”). All these three types of action were executed, alternatively, using two vitality forms: rude and gentle .Actions belonged to the participants’ motor repertoire. Moreover, they have been selected among a group of daily life ones from previous studies ; from the other it made very unlikely the possibility that one participant may express both rude and gentle vitality form in the same trial or she/he could be confused about the experimental request.Each action was performed six times for each vitality form, for a total of thirty-six trials . To improve children’s compliance and data reliability, the experimental procedure was split into two sessions , one for each vitality form, carried out on different days. Critically, from the one side this also minimized the risk of kinematic “dragging” effects . In the “types-of-action-familiarization” phase, he pushed participants to do actions by using verbal hints . Noteworthy, in this familiarization phase it did not matter the vitality form, whereas we exclusively focused on the type of action executed by participants. In the “vitality-form-familiarization” phase, the experimenter familiarized with participants to verbally evoke their (kinematic) expression of rude/gentle vitality form. Distinct objects  and distinct requests  were provided in order to avoid any bias during the test session. Noteworthy, during this vitality-form-familiarization phase it did not matter the type of action, whereas we exclusively focused on the how of the action expressed by participants.Order of presentation of both the types of action and vitality forms was randomized within and between participants. At the beginning of each session, all (both TD and ASD) participants were familiarized with the experimental procedure by the same experimenter (LC) that—in order to avoid any imitative effects—never performed any actions and never stated phrase such as “this is gentle” or “this is rude”. In addition, he never used any terms that may be a bias for our analysis . Generally speaking, the experimenter avoided any what condition, all but three parameters in the how condition) and by regression analyses, and performed the task generally well  or very well (no errors). Furthermore, 2D video recordings of the experimental sessions were also checked to verify compliance.During the test session, after each trial participants returned with their right hand at the “hand starting position”, and the experimenter placed the right object expected for the next trial on the “object initial position”. All participants clearly comprehended the experimental requests, as indirectly corroborated by kinematic similarities for many parameters between groups  able to detect the 3D position and displacement of passive markers composed of adhesive plastic hemispheres, 10 mm in diameter, covered with reflective paper. This system consists of eight infra-red (IR) cameras, working at a sampling rate of 60 Hz, able to detect light reflected by the markers placed on specific body landmarks. Spatial resolution of the system is 0.3 mm. To maximize flexibility of analysis and minimizing potential data loss, we defined a marker-set of 6 markers placed, respectively, on the right arm acromion, elbow , wrist , third metacarpal head, fingernails of the index and of the thumb . In fact, it is more reliable given that it excludes potential confounds related to the grasping phases of different objects. Preliminary analysis indicated that the most reliable marker for our aim was the one on the wrist , as it was the more stable and visible in the second act recording. Thus, results reported in this paper focus only on the second act kinematics of the WRI-marker.Raw data were filtered by means of a triangular moving window filter (window duration of 66.7 ms)43). In our experimental setting, X axis corresponds to the dorso-ventral axis of the body, Y to the vertical axis of the body, and Z to the lateral axis of the body  exceeds the value of spatial resolution of the system (0.3 mm). To determine the end of the reaching phase, we calculated the first frame following the onset in which the X, Y and Z displacements of the WRI-marker were all less than 0.3 mm , peak velocity (pV), peak acceleration (pA), peak deceleration (pD), time to peak velocity (TpV). The acceleration phase was defined as the portion of movement from the movement onset up to pV. The deceleration phase is defined as the portion of movement from pV to the movement offset. Subsequently, we computed the percentage of acceleration time (T%Acc), defined as the duration of the acceleration with respect to MT, and the percentage of deceleration time (T%Dec) defined as the duration of the deceleration with respect to MT. Maximal displacements of the WRI-marker along the three Cartesian axis were also calculated. Maximal displacements of the WRI-marker, MaxDR), “ball-gentle” , “crackers-rude” (Crackers R), “crackers-gentle” (Crackers G), “bottle-rude” (Bottle R), “bottle-gentle” (Bottle G). The average values of each parameter were then used for further analysis. In particular, we considered the what condition  and the how condition . To analyse the amount of variation of each parameter in the two different conditions (what and how), we built distinct indexes. The index of absolute differences for the what condition (|what| index) measured the amount of variation of the parameter of interest among the different types of action, when performed with the same vitality form (rude or gentle). For each participant and each parameter, the absolute differences between different types of action performed with the same vitality form were computed. To recap, the following combinations were computed for each parameter :what| index for each parameter was obtained computing the median value  over all the combinations and participants .For each participant and for each parameter, we computed the average values  over the 6 trials performed for each type of action/vitality form: “ball-rude”  measured the amount of variation of the parameter of interest on varying the two vitality forms, when the same type of action was performed. For each participant and each parameter, the absolute differences between the rude (R) and gentle (G) vitality form for each of the three types of action were computed. To recap, the following combinations were computed for each parameter (P):how| index for each parameter was obtained computing the median value (IQR) over all the combinations and participants . The |how| index measured the amount of variation of each parameter on varying the vitality forms without taking into account the “direction” of such variation . Thus, we also computed an index that included differences with sign  to assess how each parameter was modulated on varying the vitality forms and to clarify the direction of the modulation.The index of absolute differences for the how condition and what condition, the Mann–Whitney U test was performed within each group, comparing the |how| and the |what| indexes for each parameter. This analysis led to identify, in each group, which were the spatiotemporal parameter(s) more relevant to modulate the kinematics of different vitality forms (how condition) or different types of action (what condition). Thus, when “|how| index >|what| index” for a specific parameter, it means that such a parameter encoded more the modulation of different vitality forms than the modulation of different types of action . A significance level of 0.05 was used.To test differences between p < 0.05) with at least moderate goodness of fit  were discussed  explain variation in spatiotemporal and kinematic data. Significant models (how condition) and different types of action (what condition), the Mann–Whitney U test between groups was used. For each parameter, the |how| index and the |what| index were compared between the two groups in order to explore differences in terms of amount of variation (absolute difference) . Similarly, for the Sign indexes, the two datasets were merged to obtain z-scores (howSign_TD and howSign_ASD). Once the z-scores were obtained, values referring to conditions  and groups  were plotted in different maps, with rows representing participants and columns representing parameters. We sorted the z-scores within the columns in increasing order from top to bottom; this facilitates the identification of patterns within each group but precludes the possibility to perform intra-subject comparison between different conditions, being the values relative to a single participant not on the same row (Supplementary Figure Heat maps showing amount of modulation (|Supplementary file1Supplementary file2Supplementary file3Supplementary file4