People with disabilities face inequalities in mental wellbeing, for which social exclusion is a contributing factor. Musical activities offer a promising but complex intervention, making impacts on a population with highly varied characteristics and needs challenging to capture. This paper reports on a mixed methods, person-centered study investigating a community music intervention for such a population. Three groups of adult service users with varied disabilities , took part in weekly music workshops in different locations. Music staff, housing and resource center staff, as well as participants and members of their families, took part in semi-structured interviews. A quantitative measure administered by service staff was used to rate service users’ social development. Two lay researchers, both individuals with a disability contributed to all aspects of the study. Interviews were analyzed through thematic analysis. Improvements in individuals’ self-expression, confidence, mood, and social skills were consistent with previous findings. Differences in effect between centers included: Group 1, some of whom had previous experience of workshops, showed an improvement in musical skills; Group 2 showed a mixed response, some participated with enthusiasm but others chose art activities over music workshops; Group 3 had lasting positive impact, this group had very limited opportunities for music due to their rural location. Quantitative analysis showed significant increase over all groups in communication, interaction with others, and joint attention. The intervention was beneficial for participants in separate locations in similar ways, but also highlighted that context and prior experience mediated effects in distinct ways. The lay researchers enhanced the qualitative analysis by emphasizing (1) the importance of recognizing participants’ self-expression in non-verbal modes of communication and (2) the importance of having music staff with a disability to provide a positive role model. This paper proposes that mixed methods person-centered research is the most suitable approach to capture and understand the multiple and varied effects of this complex intervention for a diverse group of participants. Tackling social isolation is a priority in addressing the widespread inequalities experienced by people with disabilities. In Scotland, for example, mental wellbeing is lower among adults living with a limiting long-term health condition than in those living with no long-term health condition . IsolatiParticipation in music in particular can address these priorities, through music therapy or community music. There are many approaches to and definitions of music therapy but key to all is the therapeutic relationship between a qualified music therapist and client, where music is used to achieve beneficial effects . These cconnectivity recognizes that researchers should accommodate other viewpoints; attentiveness and dialog require mindfulness of context and the implications of researcher’s co-presence; empowerment and participation afford participants agency in their choices; and critical reflexivity necessitates examining how power relationships are cultivated and sustained and who benefits from them.It is also vital that any research on the experiences of marginalized groups attends to their unique experiences and perspectives. Four research principles were proposed to recognize and accommodate the needs of the individual in a priori categories based on Landry et al., rather than seeking to understand subjective experiences of impact on participants’ wellbeing Do the effects of community music workshops vary for different groups of adults with disabilities, and if so, how?(2)How can qualitative, quantitative and participatory methods be combined to evaluate the range of impacts of community music programs?(3)What potential do community music programs have to benefit a broad range of people with disabilities?concurrent  participants were recruited via three separate resource centers from an accessible rural location in Central Scotland. Ages ranged from 18 – 53. Group 2: Sixteen  participants were recruited via two separate resource centers in an urban location in Central Scotland. Ages ranged from 30 to 73. Group 3: Twelve  participants were recruited via one resource center in a rural location in Southern Scotland. Ages ranged from 22 to 67.Within all groups, participants’ disabilities ranged from mild to profound and complex, and their levels of independence ranged from requiring constant one-to-one care to living alone in sheltered accommodation. More specific information about the nature of participants’ disabilities was not officially disclosed by the resource centers. This policy is guided by the recognition that the medical and personal information of a person with a disability is private to that individual . EthniciWorkshops delivered followed the format, content and objectives of these detailed in Activities were tailored for each of the groups above based on preferences expressed through the participatory process of the workshops. Two examples of these are: (1) inclusive activities which were differentiated. For example, participants could drum, sing, play adapted guitars, or keyboards according to their preference and (2) the flexible structure, whereby space was given to hear service users’ thoughts both during and between activities. Choosing the next song was often based on views from the group.Many of Group 1 had participated in Stage 1 see  so had eGroup 2 had not participated in Stage 1, however, five participants had previously attended workshops run by other disability music organizations in the city. They contributed song lyrics and suggested narrative directions of songs. Two participants requested songs they wanted the music workers to play, so they could perform them for the rest of the group.Group 3 had not participated in Stage 1 and received 10 workshops from August-October, rather than the initially planned 20. This was due to the rural center that was first identified being unable to recruit sufficient attendees. Group 3 had very limited previous access to music apart from singing or playing percussion along with recorded music in sessions led by non-specialists. Outwith the music sessions, participants asked staff if they could sing workshop songs. Toward the end of the project, the center purchased a set of BoomWackers so they could continue making music in ways introduced to them during the workshops.In-depth interview topic guides were devised to gather information about particular difficulties faced by participants, their levels of confidence and sociability, aims for their personal growth in both center and home contexts, views on the workshops and any impacts or issues they perceived; and at the final interview, views on whether these impacts were still appreciated some weeks after the program. Interviews with music staff delivering the workshops gathered their perceptions of any changes in the participants’ musicianship, sociability and any other perceived impacts.t-tests were conducted to compare scores in five key areas of social development before and after the workshop program. These were: joint attention, ; communication (I am making my needs known and responding to the communication of others); interaction with others ; social awareness  and self-expression (I am able to let others know what I am thinking/feeling).Quantitative data comprised a survey developed for service staff to assess participants’ social development. Participants were scored in five key areas of social development on a 5-point scale from very limited to very good. Paired-samples Two lay researchers, both individuals with a disability, were recruited to take detailed observational notes on how they perceived participants engaged musically and socially in workshops for Groups 1 and 2 and to contribute to analysis.The programs of music workshops were delivered by a registered charity highly experienced in this work (Limelight Music). Groups 1 and 2 received 10 workshops from April to July 2018; all three centers then received 10 workshops between August and October 2018. Workshops were weekly, with occasional breaks due to day center closures for holidays and a summer holiday of 5 weeks. Sessions were for 2 h with a 15-min break. Individuals who attended all sessions from Groups 1 and 2 received 35 h over 28 weeks, and those from Group 3 experienced 17.5 h of workshops over 12 weeks. All workshops took place in accessible rehearsal spaces, and encouraged continued social interaction between participants at breaktimes. In Group 2, six participants dropped out of the workshop program; all were given the choice to attend at the beginning of every workshop and chose to take part in art and crafts activities instead.Interviews were gathered at three timepoints: baseline; post-program; and at five-week follow-up. Interviews were sought with service staff for each participant (center life interview) and either the participant, a family member, or key housing staff for participants (home life interview). Interviews were held in centers or in homes as convenient for participants. At baseline, 44 center life, 31 home life, and three music staff interviews were gathered. Post-program, 38 center life; 17 home life, and four music staff interviews were gathered. At five-week follow-up, 38 center life and 18 home life interviews were gathered.The survey was administered by service staff at the program midpoint in early August; post-program; and at five-week follow-up. 1. Two overarching categories structure the results reported here: (i) common impacts which were described in all three groups and (ii) contextual impacts, which were themes distinct between groups or unique to one.Interviews were analyzed using thematic analysis . Transcrt-tests, the analysis focused on identifying statistically significant change in reported social development aspects, looking at scores before and after the workshop program.The quantitative data collected was analyzed using IBM SPSS V25 statistical software . Throughconfirms or contradicts one another; is mixed, with both confirmatory and contradictory elements, or enhances, where extra understanding and depth is added to the themes.In complementarity mixed methods research, data are initially analyzed separately following the chosen methodological approaches (as described above) see . FollowiQuantitative results were only topically relevant to the first category, common impacts and not to the second, contextual impacts.Informed consent for research participation was gathered in advance from participants, or from an identified person with responsibility if they lacked capacity for this. To inform potential interviewees, accessible printed or recorded information sheets were prepared detailing the voluntary, confidential, and anonymous nature of participation and how data would be used.This research received ethical approval from the Edinburgh College of Art Research Ethics Sub-Committee, University of Edinburgh, which includes consideration of any risk of disadvantaging individuals through the research.Two overarching categories were identified, common impacts and contextual impacts. Common impacts were consistently noted in all types of interviewees’ accounts and in all of the groups; contextual impacts were mutually exclusive to each location. The category of common impacts was supported by relevant quantitative results and mixed methods analysis. The sections that comprise results are: common impacts, contextual impacts, quantitative results, and mixed methods analysis.Improvements in individuals’ self-expression, confidence, mood, and social skills were attributed to workshop participation at all three settings.Participants’ emotions were recognized and accommodated through music staff altering existing songs or creating new ones. An example of a new song was “I feel alone” which explored feelings of, and coping with loneliness. The act of playing or singing was also seen to be a separate channel of expression. For example:2.He’s actually playing the instruments, joining in and listening to music, songs being sung, and expressing himself through singing, which I have to say wasn’t something he did that often until he was actually working with the group (Service staff/G1/7)An increase in verbal expression from some service users during the workshops was also noted: “he can speak freely (in workshops) as well” (Service staff/G2/4). Lay researcher KB commented that through the workshops participants were able to express their own “voice.” KB defined “voice” as a form of self-expression that could be “non-traditional”  in how it was communicated. For example, participants could sing, speak, play instruments, but also express their “voice” through increased eye contact with others, bodily movement or gesture.Staff and parents described participants having increased belief in themselves and their choices, pointing to them “speaking up” (Service staff/G1/2) in workshops or being more likely to make decisions for themselves outwith music sessions. For instance, one participant chose to sit next to different people in the workshops after a period of always choosing to sit between the same two service users.Participants themselves talked about developing confidence, for example in their feelings about going into the sessions:Well, the first day I was a bit nervous because I wasn’t sure what I was going into. I was like “what is this?” but as the weeks went on I’m confident enough to go to the workshops now (Participant/G1/1).Developments in self-efficacy were also described in the everyday lives of service users:Certainly in the last couple, two or three (months), I would say there’s been even more of a positive change in Cassie’s life. She’s even involved in making more decisions, she’s been very vocal about things that she wants to do and places she wants to go .3 :For Cassie, her ability to be more “vocal” gave her greater agency to improve her experiences in a way which was meaningful to her. The staff saw her growth of confidence at the workshops as transferable. This was also demonstrated by three individuals (from Group 1) who took part in a government-led focus group to gather young adults’ opinions about key issues for themI’m not sure how a year ago the girls would have got on going into an environment with young folks… who very much know each other. I’m not sure how much they would have spoken and contributed. Whereas the progression, I think they’ve went into it with more confidence than they would have done before. I think that’s four meetings that they’ve been part of, they’re absolutely in the mix now. the girls are doing a great job. They’re raised loads of points about different things about inclusivity (Service staff/G1/9).By making an effective contribution to this focus group, the three participants exercised newfound skills of listening, synthesizing information and being able to put views across on behalf of young adults with a disability.Staff in all groups noted increases in happiness and relaxation among participants both during and after the workshops. Enjoyment was seen to be crucial in this:I just enjoy the workshops, it’s good fun, a good laugh. It’s a good social activity (Participant/G1/3).Interviewer: How do you feel after a workshop? Participant: Happy. That’s the overwhelming feeling is happy (Participant/G1/2).It seems to be making him feel relaxed enough to be more open, rather than just being an observer, he’s obviously joining in (Service Staff/G3/3).Shortly before Stage 2, staff in one of the Group 1 centers were researching strategies to target absenteeism and low mood through creating rewarding, meaningful activities. However, after the workshops started, those taking part in the workshops were seen to have improved past the need for extra intervention at their center. Staff also saw the music sessions as having “a calming influence” on anxious participants. Some non-verbal participants were observed displaying fewer compulsive behaviors associated with their anxiety, such as rubbing their head or chewing clothes. One staff member thought music helped a service user prone to frustration and anger because:it kind of gets his mind off things. It looks… like he partially loses himself in the music; it’s a bit of escapism (Service staff/G2/1).The workshops became an important part of the week, parents and staff reporting the participants’ positive anticipation the night before, or morning of the workshops.Participants were described by others as becoming more inclined to meet with new people or people they did not know so well, and as more likely to enter situations where they did not know people. In the following extract a staff member describes how he understood a service user becoming more socially engaged through workshop participation:I felt she (Violet) was just getting used to looking around the room … in the session and enjoying it. She became a lot more expressive, maybe shaking maracas and joining in… But she’d always arrive in a shell, and then blossom, if you like, by the end of it (Service staff/G1/5).For this staff member, the first observable changes were in Violet’s gaze interactions. The workshops provided a space for Violet to interact with others at a pace that was suitable for her. She was able show this interaction though playing music, “joining in” when she chose. Another participant explained herself how her gaze shifted toward others as she felt more sociable:When I first started I wasn’t looking at anybody, and I was just looking at the ceiling, but now I’ve stopped doing that, and talking to other people instead of Jackie (Participant/G1/1).The expansion of her social circle was an important change for this individual. For others, this was as a motivating factor to attend more workshops, for example:I think it would change my life a bit to come and do another music thing. Like I’m doing the dance group as well on a Wednesday and I feel like the Monday’s a good day for me to come and meet new people (Participant/G1/2).This participant appreciates a potential gain from getting to know people at workshops so far, but also sees further possible benefits: “I think it would change my life.”Self-expression, confidence and mood were described through change in either individuals’ behavior, self-perception, or wellbeing. They can be seen as first affecting individuals on a personal level, whereas social skills developed through change in individuals’ communications and interactions with others. These effects are linked, as increased capacity to speak up (confidence) can result in enhanced self-expression. Another interpretation is that successful self-expression can give individuals a growing belief in themselves as competent communicators, which in itself leads to greater confidence. A combination of both of these qualities contributes to participants’ sociability, which can be understood as the ways their confidence and self-expression are enacted with others.The next section reports on impacts for particular contexts.Not all impacts from the workshops were observed in all three groups. This section presents distinct impacts found in each group.An improvement in both, or either, technical and musical skills was noted by service users and key stakeholders for Group 1 only. Music staff described progress in the following ways:So, you can see the development there. And with everybody, everybody’s exerting more pressure, so things are becoming more physical as well as more musical and more social (Music staff/1).An important stage in learning string instruments is when a student can effectively push the strings down to create a good, clear tone without buzzing. Music staff can appreciate the change in sound that the participants make over time. Being “more musical” may refer to the point in a person’s musical development where they can manipulate more parameters, for example, tone, dynamics and timing to find their own way of playing a phrase. This is a complex process which needs skill, time and experience to realize.Service users also perceived their own musical skills improving:When I first started I didn’t know how to play a guitar, but now I’m really good at it. [music staff] actually found me improving in how to play it, even though it’s a left handed one (Participant/G1/1).This person appreciated a big change in what they were able to play, coming from being a complete beginner to being “good.” They also overcame an additional barrier in negotiating a left-handed guitar, which can be confusing when everyone else in the room is playing the standard right-handed instrument.Lay researcher KB identified sophisticated musical skills in a service user who liked drumming:he’s got such a good musical ear that he picks up the drumming, back to the drumming section again. He picks up almost immediately what (music staff) is doing (KB).Different aspects of musicianship are expressed in this extract. Having a “musical ear” in this context demonstrates knowing the structural features of a song, i.e., what drumming pattern you are meant to play where. “Picking” up on the intentions of the music staff correctly requires good contextual knowledge of the song and happens quickly, often in a few words and gestures rather than step-by-step explanation or reading music.Lay researcher MM identified the benefits of having music staff who also had a disability as making a positive contribution to an inclusive musical environment. He attributed this to Limelight Music creating an atmosphere where “it’s not them and us,” exemplified by having a role model, a musician with a disability. Another factor which may have influenced the strength of this effect for Group 1 is the lack of opportunity in music college courses or modules available in the area. One parent explained “college courses are all about life skills, about surviving, how to do what you need to do just now” (Parent/G1/9). The workshops provided her son with a chance to refine his music skills with experts. A final contributing factor is that some of Group 1 had been part of Stage 1 and so had a strong basis from which to extend existing musical skills.mixed response to the workshop content. They all enjoyed the music staff’s use of humor: “sometimes it’s funny, because usually you get a bit of banter” (Participant/G2/2). They all enjoyed the social aspects of the workshops, but the musical activities were viewed in sharply different ways.Staff, family and participants of Group 2 reported a One participant expressed a wish to hear and play different music:Participant: Aye. It’s the same songs every time.Interviewer: What other songs would you like to do?Participant: Pop music…. But fast music, rock and roll, Rod Stewart (Participant/G2/4).One staff member offered an explanation for the service users’ feeling about the content: “If things get too repetitive they’re like, we’ve done it!” It is possible that the purpose of the workshops was not understood fully; the nature of development in music workshops to where change is comfortable for all may have felt too slow. If participants missed workshops then music staff would have to repeat songs so that the group had a cohesive understanding before moving to new material. In this way non-attendance can create a hiatus in the process of improvement in the music group and consequently repetition of known material could affect motivation to come back.On the other hand, other participants in Group 2 were identified as benefiting from a known structure:He likes that he knows when he goes in …what they’re going to be doing and he knows what’s coming next, he knows the words to the songs, he knows the instruments that they use and stuff like that. Obviously (music staff’s name) is there every week so he knows that that is who is going to be taking the class. So that’s important to him, so I think that is what works for him (Service staff/G2/5).Balancing different needs and preferences presents a challenge for music staff. Groups 1 and 3 had very good attendance and all were able to contribute to and enjoyed playing in the group. This suggests other external factors outside of workshop content can impact on cohesiveness of a group and motivation to attend. Group 2 were presented with an alternative activity, arts and crafts, during the scheduled workshop time which six participants chose to pursue instead of the music sessions.n = 8).In Group 3, both center and residential staff, as well as families of the participants described a lasting positive change in mood as a key impact in both post-workshop and follow up interviews, 2 months after the workshop program was complete. This was noted strongly in over half of the center users  and started joining in with enthusiasm. Jack began making jokes during the workshop and at home, using the songs he learned in the workshops to play with expectations. For example, in the breakfast song, he claimed that he had very different and extravagant food than normal. His mum saw this as him “regaining sense of humor, even in a small way.” She said that staff reported that during the workshops:it’s like seeing the old Jack. You know, wee windows. I don’t think for the whole time, because his concentration’s not … you’re never going to get that length of time. But they said they’ve just seen bits of him that’s like they used to see years ago, coming back. So they thought it was great. Because sometimes I don’t think they can get him to participate in very much (Parent/G3/1).During the workshops, Jack would come home and put music on of his own volition, continuing this after the workshop program ended.When we come home, as soon as he came in he puts music on in his room and he was singing away….he’d be quite enthused… it was all himself, self-motivated sort of thing, which is good (Parent/G3/1).Housing staff for Group 3 described an ongoing positive mood for eight service users: “Laura herself has continued to have a very positive outlook” , attributing this to their experience in the workshops that contributed to a growing resilience. For example, one participant who was in a married couple living in sheltered housing had dealt with her husband’s sickness better than expected: “some of that could be contributed to the fact she’s feeling quite positive about life generally at the moment, even though she is facing difficulty” . Other participants said to be “really positive with the way things are in their lives”  and this had an impact on previously “fractious” relationships with others in sheltered housing.Staff identified the music workshops as providing a high quality and inclusive experience for their service users: “I think it’s something she can genuinely interact with and get something back from” (Service staff/G3/4). They compared the workshops favorably to other available activities, as they found that everyone in the group could join in some way:Whereas music, she can genuinely contribute to that. Something can be put in her hand that she can shake if she wants to. If there was a vibration, if her hand was put on a speaker, she can feel all of that stuff. She can shout. I think it’s stress-relieving. All of that stuff. So, I think it’s something she can genuinely be involved in and it’s not just tokenism .Inclusiveness was enacted by music staff creating bespoke arrangements of songs for the group so individuals could contribute at a level that was appropriate for them.As well as impacts reported across all three groups, interviewees from each center described distinct outcomes from the workshops. Group 1 described how the musicianship of several members improved over the workshop program, as well as the group itself improving. Family members attributed this to the workshops addressing a gap in available education. As well as this, one of the music staff with disability provided a positive role model for the group. Potentially due to lower social cohesion, Group 2 reported mixed responses toward the music workshops, with some participants finding their structure too repetitive, while others enjoyed the predictability and familiarity of the activities. This led to some participants choosing to attend arts and crafts activities instead of the workshops, as they took place at the same time. Family and housing staff from Group 3 described them improving in mood and that this resulted in increases in self-initiated music listening and greater resilience. Participants found the workshop activities to be fun and engaging and they were able to contribute musically on different levels as suited their skills.The next section reports the quantitative results on dimensions of change in social skills and the section following that reports the results from mixed methods analysis.There was a statistically significant increase in the following areas of the survey, comparing scores before and after the workshop program:•t(14) = −3.389, p = 0.004, M = 3.60, and SD = 1.242 before the workshop program and M = 4.47, SD = 0.640 after the workshop program.Joint attention: •t(14) = −3.523, p = 0.003, M = 3.33, and SD = 1.397 before the workshop program and M = 4.47, SD = 0.834 after the workshop program.Communication: •t(14) = −3.108, p = 0.008, M = 3.53, and SD = 1.302 before the workshop program and M = 4.47, SD = 0.915 after the workshop program.Interaction with others: There was no statistically significant increase found in self-expression or social awareness, as measured in the survey used.By combining qualitative and quantitative results, a deeper understanding of the identified themes from the interviews is possible, as well as where they overlap. As the survey was concerned with social development, the theme of mood was not topically relevant and there was no convergence of data for this theme. The following section expands on confirms the qualitative data, as it was a measure of participants’ ability to form and articulate a request for themselves.This theme explored the capacity of the participants in the following ways: ability to tell “my story” through creating or contributing to song lyrics; convey feelings through playing instruments or singing and communicate musical intent through non-verbal modes of communication . The survey element of communication confirm this theme.The confidence theme was articulated as participants believing in their personal choices and being able to communicate these; coping better with uncertainty and change; and initiating communication with others. Survey elements of communication and interaction Improvements in social skills were described as participants having increased number of interactions with others and in more ways  as well as their social circles expanding. Survey elements of joint attention, communication and interaction with others confirm this theme. Joint attention enhances this theme as interviews did not describe social skills in this way. The music workshops were a clear example of a shared focus which was perhaps taken for granted.In summary, aspects of the qualitative results were confirmed and enriched by quantitative results. Combining results also demonstrated the overlapping relationships between themes, for example self-expression and confidence were confirmed by the quantitative element of communication. Social skills overlapped with both self-expression and confidence, with quantitative elements of communication and interaction with others.Results confirmed that a community music program can benefit adults with disabilities, but evidenced through the use of innovative mixed methods, these impacts can vary for groups with different characteristics and needs. Common effects were positive impacts in self-expression, confidence, mood and social skills. These are related processes, for example, self-expression could be appreciated in different modes of communication in this inclusive workshop program, as participants had space to express themselves in their own way. The development of the personal qualities  and improvements in wellbeing (mood) could be viewed in a range of social interactions . As wellMixed methods were the most suitable way to appreciate the multiple and varied effects of this complex intervention for the diverse people taking part in the same workshop. The person-centered research design was qualitatively led. This had the benefit of gaining a greater understanding of the participants’ environments, such as challenges due to location, lack of opportunity or clash in activities, a crucial feature in research with those who have ASC . CombiniCommunity music programs have potential to benefit the mental health and wellbeing of a broad range of people with disabilities, but importantly this research highlights that they can do so in different ways for particular groups and settings. Positive effects on different dimensions of mental health can be appreciated in addition to improvements in mood. For Group 1, improvements in musical skills created a growing feeling of competency and for some, a powerful feeling that music participation could change their life. The flexibility with which self and other are brought into being is seen as being at risk in individuals with ASC in particular but amenable to creative interventions , and thiParticipants in rural areas have more barriers, such as loneliness, lack of agency, and boredom due to fewer opportunities. There is a similar structure and activities in school and for school leavers in day centers. Reasons for this intervention being particularly beneficial for mood in Group 3, could be that it offered enough stability to create a secure environment and sufficient change to be engaging. Taking part in music groups has been proposed as providing a positive coping strategy for those with a disability , an asseResearch Principles in Person-Centered Research  InformedThis study had many stakeholders who were able to contribute their views through different channels. These were an advisory group with monthly meetings, and open public discussion during the showcase event at the end of the project. Two lay researchers who have a disability, KB and MM, contributed their thoughts on a draft of this paper, resulting in further conceptual development of themes, in particular, KB’s description of a participant’s “voice” being expressed in non-verbal modes of communication.The research team made several visits to all centers to both observe and participate in the workshops. An important part of these visits was for service users to meet the researchers as “a relaxed, informal interaction …with trust and rapport” are crucial when interviewing individuals with a disability , p109.Participants had agency within the music workshops, they were able to choose whether to attend, how to join in (or not), what instruments they wanted to play, and their ideas became part of songs which formed the group’s repertoire. Before every workshop, verbal assent was sought and six participants from Group 2 chose to attend art instead. One participant was very gifted in this discipline and was perhaps more motivated to participate in an activity in which he had more mastery. Interestingly, the participants who dropped out were from one of the two subgroups comprising Group 2 and so may have demonstrated a social cohesiveness of their own choosing, by taking part in art classes together.In taking a person-centered approach, the rights and views of the participants were prioritized throughout this study. This is in line with demands for a commitment and focus among non-disabled researchers toward genuine engagement, rethinking research relationships, inclusive design, and an empowered voice for people with intellectual disabilities . It has Developing holistic research projects that are ecologically valid, including qualitative and quantitive aspects and involving service users and stakeholders, is of paramount importance to increase the impact and relevance of academic research. This study apprehended and measured the complex impacts of a community music intervention through a mixed-methods, qualitatively led person-centered approach. This approach adds to the small but growing body of mixed methods research in community music e.g., , particuThis research has identified positive effects of music participation on aspects of mental health in terms of self-expression, confidence, mood, social skills, communication skills, joint attention and interaction with others, which align with previous work . This haThe datasets for this study will not be made publicly available because the terms of consent for research participation did not specify that data would be accessible other than by the research team.This research received ethical approval from the Edinburgh College of Art Research Ethics Sub-Committee, University of Edinburgh, which includes consideration of any risk of disadvantaging individuals through the research. Informed consent for research participation was gathered in advance from participants, or from an identified person with responsibility if they lacked capacity for this. To inform potential interviewees, accessible printed or recorded information sheets were prepared detailing the voluntary, confidential and anonymous nature of participation and how data would be used.All authors listed have made a substantial, direct and intellectual contribution to the work, and approved it for publication.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 rapidly decreasing cost of gene sequencing has resulted in a deluge of genomic data from across the tree of life; however, outside a few model organism databases, genomic data are limited in their scientific impact because they are not accompanied by computable phenomic data. The majority of phenomic data are contained in countless small, heterogeneous phenotypic data sets that are very difficult or impossible to integrate at scale because of variable formats, lack of digitization, and linguistic problems. One powerful solution is to represent phenotypic data using data models with precise, computable semantics, but adoption of semantic standards for representing phenotypic data has been slow, especially in biodiversity and ecology. Some phenotypic and trait data are available in a semantic language from knowledge bases, but these are often not interoperable. In this review, we will compare and contrast existing ontology and data models, focusing on nonhuman phenotypes and traits. We discuss barriers to integration of phenotypic data and make recommendations for developing an operationally useful, semantically interoperable phenotypic data ecosystem. Organism traits determine the role of species in economies and ecosystems, and the expression of those traits relies on interactions between an organism’s genes and environment. The key to predicting trait expression is having a large pool of data to derive models, but most organism trait observations are recorded in ways that are not computational. In this paper, intended for an interdisciplinary audience, we discuss data models for representing organism traits in a computable format. Increasing acceptance of a data model for traits will greatly increase the pool of available data for studying the dynamic processes that determine trait expression. We hope that explaining these data models in a straightforward way and articulating their potential for accelerating discovery will increase adoption of this promising data standard. An organism’s phenotype is the product of interactions between its genetic endowment and environmental conditions over its lifetime, but the ability to predict phenotypes from genotype and environmental data is limited. The models we currently have that predict organism phenotype from genotype rarely include environments, have lower performance on multigene phenotypes, and often only apply to a single taxon –18. The Worldwide recognition of climate change has created urgency around addressing this problem for agricultural sustainability and conservation of essential ecosystem functions . Models Information about organismal phenotypes has been collected by thousands of observers for a myriad of purposes over centuries. Much of this information is contained in countless small, heterogeneous data sets , which aA “phenome” is the set of all phenotypes expressed by an organism at all life stages . It is analogous to the genome or the proteome, which are the sets of all genes and proteins of an organism, respectively. Thus, phenomics is the study of the phenome and how it is determined, especially in relation to genes and environmental influences. Integrated computational analysis of genotype and phenotype is at the heart of precision medicine , evolutiMany different research disciplines, such as biodiversity science, environmental science, agronomy, biomedicine, and phylogenetics, document characteristics of organisms and taxa. These characteristics have been referred to as traits, phenotypes, characters, and qualities, sometimes interchangeably or inconsistently within and between disciplines. Characteristics of organisms have been represented using several different data models, terminologies, and perspectives, and we will use terms according to the definitions in Character or trait: Any descriptor of an organism that can have multiple states/phenotypes . In the phylogenetics community, characters are a special subset of traits that are important for inferring the process of evolution.Character state or phenotype: The specific state or manifestation of a character or trait in an organism .Quality: Any descriptor of an organism and its multiple states. In an ontology, the states are subclasses of the descriptor .Value: Numerical measurement of a phenotype Specimen: A physical object collected for research purposes. In this context, an organism, part of an organism, or collection of organisms. Specimens are often accompanied by metadata such as time and place of collection.Taxon concept: A hypothesis about how to group individual organisms into species or higher-level taxa.Ontology: An ontology is a classification of concepts in a field of knowledge, or a domain, such as organisms or anatomical entities. Concepts are hierarchically arranged and formally defined in a human-readable format (using text definitions) and computer/machine-readable format (encoded with a knowledge representation language like Resource Description Framework Schema (RDFS), Web Ontology Language (OWL), and Open Biomedical Ontologies format (OBO). In addition, the relationships between concepts are defined, which allows disparate data types to be connected in a formal way.7 barriers that stand in the way of integrating phenotypic data:There are multiple challenges to making phenotypic data available and interoperable, including variable formats, lack of digitization, and linguistic problems such as ambiguity and poor language translation ,37,41. WMany names for 1 thing, 1 name for many things . By logically defining the phenotype concepts, providing text definitions and synonyms, ontologies solve the problems of homonymy, synonymy, polysemy, and most importantly, ambiguity (barrier 1 and 2). For example, the biomedical Natural Language Processing (NLP) community has developed several tools that use reference ontologies (in addition to other resources) for addressing these “word sense disambiguation” problems, enabling a machine to extract meaning from human-readable text –49. The Although model organism phenotypic data are rarely described as corresponding to a specimen (and such specimens are rarely preserved), these data are always associated with a genotype. While such data are not foolproof against future changes in taxonomy, the relationship to a known genotype does provide higher precision than simply a species name and facilitates combining model organism phenotypic data with data from nonmodel species . The usThere are several databases containing information about organism characteristics . All of An important concept in understanding the diversity of phenotypic data is the recognition that some assertions are made at the “class” level  and others at the “instance” level . TBox reasoning is defined as logical entailments regarding axioms about classes and properties, whereas ABox reasoning utilizes axioms about instances . The terIn biological domains, the TBox is often implemented as an ontology with “classes” that describe kinds of things, like “femur,” “leg,” and “bone,” and “properties” that describe the relationships among the classes or relationships among instances of those classes. The properties have machine-readable rules to describe how the kinds of things relate to each other . So, in the example above, that “femur” “is a” type of “bone” and “part of” a “leg;” “femur,” “leg,” and “bone” are classes, while “part of” and “is a” are the properties. The ABox uses the classes and properties described in the ontology to model instance data. The bridge between the TBox and ABox is the use of common classes, in this case, “femur.” In the ABox, the data represent instances of the class “femur,” which is described in the TBox.Entities in ontologies may be defined by or composed of multiple other classes. For phenotypes or traits, this is done using the Entity–Quality (EQ) Formalism . This moWhen such EQ-based traits are made into named classes, they are said to be “pre-composed” , which is axiomatized as [“color” and (“inheres in” some “flower”)] where “color” comes from Phenotype and Trait Ontology (PATO), “inheres in” comes from RO, and “flower” comes from PO). Alternatively, one could assert that an instance of the “length” class is a quality of an instance of the “femur” class without making a new, named class. In this latter case, femur length is “post-composed.” These 2 approaches are logically equivalent and can be reasoned over  . The choCategorical phenotypes, for example, “shortened femur” or “delayed germination,” are often described as relative to some wild type  and can readily be represented using EQ Formalism. As described above, the EQ Formalism can precompose phenotype classes using an anatomy or process ontology and the PATO . QualitaMore recently, several phenotype ontologies implemented the Dead Simple Ontology Design Pattern (DOS-DP) ontology building process to consistently precompose classes and represent more granular phenotypes ,115. ThiThe Character/Character State semantic model reflects the long history of research strategies and data structures in systematics, taxonomy, and phylogenetics, which is dominated by tabular data exchange standards. Phylogenetic data  are represented in a matrix for tree-building algorithms . TaxonoThis model is quite flexible and capable of accommodating historical data. The biggest advantage to this model is its compatibility with the existing infrastructure of biodiversity databases that are using the Darwin Core standard. The “spreadsheet” data format is familiar to many researchers, citizen scientists, and local experts, unlike OWL. The use of ontology classes to define the characters and character states aids in translating these data from tables to graphs; however, there are limitations in translating from class-based to instance-based because of the difficulty in retaining specific collection metadata in the class-based approach . The ChaThis model is an extension of the EQ Formalism that accommodates measurements from individual specimens and is frequently utilized in tools for users to record observational data . DetailsThe largest volume of Measurement-Based assertions about organism phenotypes comes from the clinical domain. A single person may have thousands of assertions about them and be in a collection of hundreds of thousands of people. A comprehensive list of these repositories and their data models is outside the scope of this manuscript.Representing organismal characters in a machine-readable form brings modern data science and computational power to the study of organismal diversity. The most common structured representations of phenotypic data are a phylogenetic matrix, data table, or semistructured text, but most phenotypic data are semistructured in hard-to-find supplementary files or unstructured in narrative text or images . Much ofThe community has not yet reached a consensus around how phenotypic data should be modeled in semantic knowledge bases using ontologies; however, some discipline-specific best practices have been developed. The Biolink model has been an effective meta-model for integrating phenotypic data across biomedical knowledge graphs that could potentially be used in other disciplines . The MinIt is unlikely that existing knowledge bases will be able to quickly redesign their systems to adopt a new, unified model; thus, it becomes important to map across the different models. Translation from 1 model to another can be straightforward, especially if shared ontologies or a meta-model, such as Biolink, are used. Difficulty arises when trying to combine or transform phenotypes reported at the individual organism level with phenotypes reported at the group level. When possible, phenotypes should be reported at the individual level because these can be aggregated to calculate a group-level phenotype. Breaking up group-level phenotypes into the more granular individual-level phenotypes is not possible. One possible exception is the reporting of traits for strains, cultures, or cultivars, wherein all individuals are supposed to be genetically identical . In addition, reporting phenotypes for individuals then allows integration of the phenotypes with any other metadata collected about that individual, such as its environment, biotic interactions, or genotype. While recording data at the individual level is preferable to the group level, this is not always possible for existing knowledge bases with established data models and a method for mapping across models is needed.Ontologies and semantic knowledge bases provide a way to overcome barriers to integration at scale but are limited by the lack of supporting infrastructure to make them easy to use in practice, which requires a balance between human usability and computational capabilities. Essential usability components include provenance tracking and documentation of design decisions and the collaborative decision-making process ,147. ResLastly, the complexity of ontologies requires documentation in addition to standard approaches –159, sucThis paper discusses 3 different phenotypic data models, EQ Formalism, Character/Character State, and Measurement-Based. The EQ Formalism and Measurement-Based models are closely related in that they both have significant logical semantics. The Measurement-Based approach links specific values to specimens, rather than linking averages to taxon concepts, and thus is more easily adaptable to taxonomic changes that can rearrange the assortment of specimens (and their phenotypes) within taxa. Conversely, the Character/Character State model is much more straightforward for individual researchers, is more closely conformant to existing biodiversity standards, and can represent qualitative and quantitative data for a class or an instance in a similarly straightforward manner. As a result, numerous data sets are developed for aggregators using the Character/Character State model that may not be made conformant to any other standard. Thus, we recommend development of a workflow for passing data from individual researchers to content aggregators using the Character/Character State model that can be translated to OWL semantics using a Measurement-Based or EQ approach. Such a workflow would include transforming small data sets to conform to an aggregator standard (similar to the process EOL TraitBank currently uses) and then transformation of these data into OWL semantics. Significant work is required to develop the infrastructure to support this workflow including expanding the coverage of ontologies and semantic data models, developing an interface for data access, and creating a governance model for long-term sustainability and maintenance of the resource. This workflow is dependent on source data sets being properly licensed for sharing and reuse , which m


Hyperfocus is most often mentioned in the context of autism, schizophrenia, and attention deficit hyperactivity disorder, but research into its effect on cognitive and neural functioning is limited. We propose that hyperfocus is a critically important aspect of cognition, particularly with regard to clinical populations, and that it warrants significant investigation. Hyperfocus, though ostensibly self-explanatory, is poorly defined within the literature. In many cases, hyperfocus goes undefined, relying on the assumption that the reader inherently knows what it entails. Thus, there is no single consensus to what constitutes hyperfocus. Moreover, some studies do not refer to hyperfocus by name, but describe processes that may be related. In this paper, we review how hyperfocus (as well as possibly related phenomena) has been defined and measured, the challenges associated with hyperfocus research, and assess how hyperfocus affects both neurotypical and clinical populations. Using this foundation, we provide constructive criticism about previously used methods and analyses. We also propose an operational definition of hyperfocus for researchers to use moving forward. A PubMed search for the same terms in the title or abstract returned 19 results. Of these, 7 are empirical studies explicitly focused on assessing cognitive and neural states associated with hyperfocus  for hyperfocus and variations of the term, namely “hyper-focus”, “hyper focus”, “hyperfocusing”, “hyper-focusing”, and “hyper focusing” in the In this review, we will attempt to explain why hyperfocus research has been so limited and we will assess if other phenomena in the literature may essentially be hyperfocus going by another name. Based on this, we will propose a clear and testable operational definition of hyperfocus. Finally, we will assess if the definitions of hyperfocus used in the psychiatric literature match our proposed operational definition.There are three major issues to consider. First, there is no clear general or operational definition of hyperfocus in the literature, which usually assumes that the reader inherently knows what it is. As there is limited explicit literature on the topic, references to and descriptions of hyperfocus are typically anecdotal and can differ from paper to paper. Table Hyperfocus is characterized by an intense state of concentration/focus.When engaged in hyperfocus, unrelated external stimuli do not appear to be consciously perceived; sometimes reported as a diminished perception of the environment.To engage in hyperfocus, the task has to be fun or interesting.During a hyperfocus state, task performance improves.Despite these issues, there appear to be four general features or criteria of hyperfocus that are consistently reported Table :HyperfocSecond, it is very difficult to experimentally manipulate a subject into a hyperfocus state Sklar, . The natThird, some studies do not refer to hyperfocus by name, but describe processes that appear to be related, such as “in the zone” and “flow”. For example, several researchers ;a sense that one can control one’s actions; that is, a sense that one can in principle deal with the situation because one knows how to respond to whatever happens next;distortion of temporal experience ;experience of the activity as intrinsically rewarding, such that often the end goal is just an excuse for the process.Under these conditions, experience seamlessly unfolds from moment to moment and one enters a subjective state with the following characteristics:The conditions for entering flow include:The conditions for entering flow and the subjective experiences associated with it appear to map onto the most commonly reported features of hyperfocus Table . TherefoLinnell and Caparos  have sugIn terms of cognitive psychology, we could phrase “intense concentration” as the intense engagement of sustained and selective attention mechanisms. Unfortunately, there are limited cognitive studies of attentional processes in the flow literature. As with the hyperfocus literature, this is largely due to the reasons discussed at the beginning of this review. Although mostly speculative, this section highlights attentional processes that may contribute to flow and hyperfocus. As attention is a limited resource, if a significant amount of those resources are focused on a particular task, peripheral and task irrelevant information may be lost  and found that subjects in the adaptive condition (where subjects purportedly were in a flow state) had higher top scores than subjects in the other conditions. This was interpreted as evidence that flow experiences result in improved performance. Additionally, Schüler  hyperfocus is induced by task engagement; (2) hyperfocus is characterized by an intense state of sustained or selective attention; (3) During a hyperfocus state, there is a diminished perception of non-task relevant stimuli; and (4) During a hyperfocus state, task performance improves.Flow states do show evidence that they are induced by at least interesting, if not fun tasks. In particular, they are induced by engaging tasks, irrespective of the source of motivation. Although the specific language is different from what has been typically used to describe hyperfocus (engagement vs fun/interesting), it is arguable that in context they mean the same thing. There is no explicit evidence that flow induces intense states of sustained and selective attention. However, based the reported effects of flow, it is reasonable to hypothesize that sustained and selective attention likely play a significant role. Moreover, applying a cognitive framework can provide a reasonable account of how intense states of attention could lead to the reported effects of flow. There is ample evidence that flow states induce a “diminished perception of non-task related stimuli”, though this has been only reported in subjective questionnaires, after the flow state had ended. To date, there have been no explicit cognitive or psychometric measurements taken during a flow state. Regardless, the subjective reports match the anecdotal reports of hyperfocus closely. Finally, there is some limited evidence that flow states improve task performance, but this is all correlational and indirect. Overall, the evidence suggests that flow states and hyperfocus appear to be the same phenomena, just with different names and initially reported in different fields of psychology. That being said, more research will be necessary to confirm and strengthen this claim Table .Table 3E“In the zone” states show no evidence that they are induced by fun or interesting tasks. They do show some evidence that they are characterized by intense states of sustained attention, but it is unclear how “intense” these states really are. “In the zone” states seem to reflect the upper range of normal fluctuations in attentional performance, rather than a distinct state of attention. “In the zone” states also show clear evidence of increased distractor suppression, resulting in a diminished perception of non-task relevant stimuli. Finally, there is no evidence that “in the zone” states result in improved task performance. Overall, the evidence leads us to conjecture that while “in the zone” states may still be mediated by similar mechanisms to hyperfocus, they are nevertheless distinct phenomena. More research will be needed to investigate this possibility Table .References to hyperfocus most frequently arise in research on ADHD, schizophrenia, and autism. Each disorder is reported to increase the frequency and/or magnitude of hyperfocus states, sometimes in different contexts. This phenotypic overlap may not be surprising given evidence for a genetic overlap across the three conditions  is characterized by clinically significant  hyperactivity, impulsivity, and inattention. It should be noted that, despite its seemingly self-descriptive name, ADHD is not solely a disorder of attention, but also executive functions . It was speculated that this might reflect the “experience of the activity as intrinsically rewarding” element of hyperfocus. Third, in the parietal lobe, the mean absolute power was higher in the ADHD patients than the controls. This was notable because typically ADHD patients show lower parietal activation than controls, which is thought to reflect impaired attentional process in ADHD. Sklar  experienced hyperfocus while playing the games. Sklar , like WeSecond, Sklar  did not ADHD is synonymous with a high degree of distractibility and having a short attention span. However, the oft reported hyperfocusing states in this condition suggest that individuals with ADHD can, paradoxically, sustain attention excessively. In fact, attentional control may not be as impaired in patients with ADHD as once thought. For example, Roberts, Ashinoff, Castellanos, and Carrasco  have shoAutism spectrum disorders (ASD) are neurodevelopmental disorders associated with impairments in social development, language, and repetitive, circumscribed behaviors/interests. Two defining symptoms of ASD are “B1) Stereotyped or repetitive motor movements, use of objects, or speech ” and “(B3) Highly restricted, fixated interests that are abnormal in intensity or focus ” APA, . Of part StereotyTo our knowledge, there are no studies that specifically attempt to measure behavior or cognitive performance during hyperfocus or flow states in ASD. In a review paper, Geurts et al.  defined Geurts, Corbett, and Solomon  also proOverall, it appears as if hyperfocus is a real phenomenon that occurs in ASD, but care must be taken to distinguish it from other symptoms such as stereotypic behaviors. Research into hyperfocus in ASD is important because, as of now, it is unclear if hyperfocus is a primary symptom or a secondary symptom that is merely induced by other ASD-related behaviors. It is possible that mechanisms underlying hyperfocus behaviors are not autism-specific, but rather that ASD behaviors happen to trigger the same kind of hyperfocus seen in the general population more frequently or strongly.Schizophrenia is a disorder characterized by abnormal social behavior, deficits in emotional processing, and psychosis. Symptoms of schizophrenia are divided into three categories: positive symptoms, negative symptoms, and cognitive dysfunction APA, . PositivLuck et al.  showed tMore recent studies have also supported the hyperfocusing theory of schizophrenia. Sawaki et al.  found thKreither et al.  addresseConsidering the symptom variability associated with schizophrenia, a natural question is whether hyperfocus is associated with negative symptoms, positive symptoms, or cognitive dysfunction. Luck et al.  tested fMore generally, although we do not doubt the results or value of the “hyperfocusing theory of schizophrenia”, it is debateable if its operational definition of hyperfocusing reflects the same process that is typically described in anecdotal reports of hyperfocusing. The hyperfocusing theory does appear to reflect intense concentration and improved task performance. And, although it is generally consistent with reports that people who hyperfocus “tune everything else out”, it is not clear if, in anecdotal reports, people were always focused on stimuli at the center of vision. A future study will need to assess if hyperfocus can only occur at the center of vision or the loci of hyperfocus can move, such as with the spotlight of attention. Moreover, there was nothing particularly fun or interesting about the tasks. A further consideration was that the effect of “hyperfocus” was relatively consistent over time in these studies, which is inconsistent with the notion that hyperfocus reflects an irregular, inconsistent state of attention that is difficult to induce. One possibility is that the “hyperfocus theory” studies were tapping into a visual attention counterpart of hypersalience (rather than hyperfocus) that has been previously reported in individuals with schizophrenia in decision-making tasks. Speechley, Whitman, and Woodward  showed tDoes hyperfocus, reported in the ADHD, autism, and schizophrenia literature, refer to the same phenomenon in all three conditions? Before we can address this, we must first consider more generally if these disorders present with similar symptoms.ADHD and autism Panagiotidi, Overton, and Stafford  to engage in hyperfocus, the task has to be engaging . (2) Hyperfocus is characterized by an intense state of sustained or selective attention. (3) When engaged in hyperfocus, there is a diminished perception of non-task relevant stimuli. (4) During a hyperfocus state, task performance improves. Although this definition may change in light of future research, it provides a reasonable starting point for the investigation of this phenomenon. Moreover, we proposed and provided supporting evidence for the notion that the phenomenon of flow is synonymous with hyperfocus. Last, we reviewed the references to hyperfocus in the psychiatric literature to determine if they fit with our newly established definition. We propose that the hyperfocus referenced in the ADHD and autism literature does fit with our definition, but the hyperfocus referred to in the schizophrenia literature does not.In all, the purpose of this review was to bring attention to an important, but generally forgotten or ignored phenomenon that may be a critical element in several psychiatric disorders. We hope that our operational definition of hyperfocus will facilitate the development of new research paradigms that are specifically tailored to assess the hyperfocus state and underlying mechanisms in both healthy and psychiatric populations. One possibly fruitful avenue for future research is to explore the potential of gamification of cognitive psychology paradigms combined with assessments of performance over the course of the task (like in Esterman et al.,


Clinical Ethics Committees (CECs), as distinct from Research Ethics Committees, were originally established with the aim of supporting healthcare professionals in managing controversial clinical ethical issues. However, it is still unclear whether they manage to accomplish this task and what is their impact on clinical practice. This systematic review aims to collect available assessments of CECs’ performance as reported in literature, in order to evaluate CECs’ effectiveness. We retrieved all literature published up to November 2019 in six databases , following PRISMA guidelines. We included only articles specifically addressing CECs and providing any form of CECs performance assessment. Twenty-nine articles were included. Ethics consultation was the most evaluated of CECs’ functions. We did not find standardized tools for measuring CECs’ efficacy, but 33% of studies considered “user satisfaction” as an indicator, with 94% of them reporting an average positive perception of CECs’ impact. Changes in patient treatment and a decrease of moral distress in health personnel were reported as additional outcomes of ethics consultation. The highly diverse ways by which CECs carry out their activities make CECs’ evaluation difficult. The adoption of shared criteria would be desirable to provide a reliable answer to the question about their effectiveness. Nonetheless, in general both users and providers consider CECs as helpful, relevant to their work, able to improve the quality of care. Their main function is ethics consultation, while less attention seems to be devoted to bioethics education and policy formation. Clinical Ethics Committees (CECs) or Hospital Ethics Committees are bodies originally established with the aim of supporting healthcare professionals in managing controversial ethical issues affecting clinical practice .Drawing from these premises, the overarching aim of this systematic review is to gain a comprehensive overview on the By collecting and clarifying evaluation tools used to assess the effectiveness of CECs in healthcare, we also try to answer the question whether CECs are useful resources.clinical ethics committees, hospital ethics committees. Therefore, as terms and/or keywords  all the expressions referring to—or containing under their trees—the aforementioned terms were included.A large number of studies refer to ethics committees as broadly conceived, thus including both CECs and RECs. Therefore, the search string had to be narrowed down in order to include only ethics committees devoted to clinical practice. The search focus was represented by CECs. Furthermore, all the possible definitions of CECs had to be taken into account: On these premises, the string was built in relation to two semantic groups: group A included all possible definitions and mesh terms related to CECs; group B contained all terms pertaining to assessment, impact, and/or evaluation. In particular, group A contained the following terms: Clinical ethics committee*, hospital ethics committee*; while group B contained: impact, effectiveness, evaluation*, assessment*.The two groups were then gathered according to the properties and Boolean operators of each database (see Table The screening process was then carried out by the first (CC) and the second author (VS) according to the PRISMA guidelines  were excluded. The first author (CC) screened the remaining titles, according to preset inclusion and exclusion criteria (see below). The abstract screening (115) was then performed by the first author (CC) and the second author (VS) independently, to ensure scientific and methodological rigorousness of the abstract selection. In 91.5% of the abstracts there was agreement between the two authors, but consensus was reached after discussing doubtful candidate abstracts. A screening of the full text of the remaining records (71) was then performed by first author (CC) and the second author (VS) independently. After this step, a total of 27 articles was included in the review process.Bibliographies of relevant articles were examined and two additional articles that met the inclusion criteria were retrieved through reference manual searching and included..Finally, a total of 29 studies was included in the review. All the articles included were considered of a sufficient quality, based on the peer review process and on the academic reputation of the journalsThe full process of selection is reported in the flow chart in Fig. theoretically—such as the description of an assessment model—or empirically, through quantitative and/or qualitative measures.Publications were included on the basis of both the following conditions: (a) address CECs as a specific topic and (b) provide an evaluation, assessment, impact of one or more aspects of CECs’ performance, whether The following publications were excluded from the review: (a) studies addressing topics other than CECs as their main focus; (b) studies concerning CECs but not providing any form of evaluation, assessment, impact ; articles (a) with no full text available (d) and not published in English, (e) editorials, books, and book chapters.Twenty-seven articles from the research queries and two more papers identified through the snowball method met our inclusion criteria and became part of this systematic review see Fig.  Table 33.Table 3Publication dates range from 1982 to 2019, with five articles published in the last 5 years . Of the twenty-nine articles included in the review, 23 made an evaluation based on data collected through empirical research and/or on the documents drafted by CECs’ members, such as the reports of meetings and discussions . The remaining six articles describe theoretical models for CECs’ evaluation . Amongst the latter, two articles also provide empirical data in support of (2) and/or to test (19) such model. It should be noted that two articles included in the review refer to the same study . However, since they report different aspects of the same study, respectively the theoretical (7) and empirical (23) part of an assessment model for CECs’ effectiveness, we decided to include both publications.The tools used for CECs’ evaluation were the followings: surveys only , interviews only , survey plus interviews , survey plus anecdotal evidence (22). In addition, the authors of three studies made qualitative analyses on reports from case consultation , or used medical charts to compare data from surveys and interviews . The assessment tools are outlined in detail in Table The enrolled participants are physicians in twelve studies (41.4%) , CECs’ members in eight studies (26.6%) , and the category of those who requested CEC’s intervention or were somewhat involved in the CEC’ processes, mainly as part of the attending healthcare team, in nine studies (30%) . Patients and their families who took part in ethics consultation were invited to participate only in 10% of studies, in which they were asked to provide comments about the ethics services offered by the CEC . In two studies, the composition of the sample is not clear, as the identity of respondents is not specified . There was no sample in the three studies analyzing reports from case consultation  and in the four theoretical studies .ethics/clinical ethics consultation” . The same can also be labeled as “case consultation” , prospective and retrospective “case review” , “discussion forum” (19), and “case discussion” (22).Of the three functions that are typically attributed to CECs—ethics consultation, bioethics training, and revision and/or development of ethics policies—the mostly evaluated is ethics consultation, being the only subject assessed in sixteen studies (55.2%) . This function may be performed in different ways, often in relation to the context in which the CEC is located  and one outlines a model to perform assessments (16).Two studies propose a framework for measuring (13) and/or reaching (18) CEC’s success in all the three above-mentioned functions. Among the theoretical papers, one deals with the function of preparation and/or revision of ethics policies and provides a model for their successful development (24). The function of policy preparation and/or revision is also assessed, together with ethics consultation, in study 22.The selection process did not identify studies focusing only on education and training in bioethics, though this is considered a core function of CECs , with a positive impact for the healthcare staff (17).Finally, study 19 investigates whether CECs carry out some kind of self-evaluation.No selected article provides a comprehensive evaluation of CECs, looking at CECs’ functions separately.expressions used to indicate CECs’ performance, but also the meaning of these expressions, as well as the outcomes considered as index of effectiveness, differ. In general, all the above mentioned terms may refer either to more objective outcomes, namely the tangible consequence of CECs’ activities on clinical practice , or to a more subjective outcome, namely, the experiences of the stakeholders—healthcare professionals, patients, and their families—who benefited from CECs’ services . In this second meaning, CEC’s impact was measured mainly through questionnaires and/or semi-structured interviews.The aim of the present work is to review the results of CECs’ assessments in order to clarify their effectiveness. To reach this aim, we systematically looked into the included articles in order to identify the exact expressions that refer to CECs’ evaluation. We found a variety of terms referring to CEC’s evaluation: effectiveness , which is the most recurrent expression, efficacy , impact , success , performance , usefulness , helpfulness  , neither there is a unique way for measuring it, nor it may be found a validated instrument justifying its use.Most findings concern users’ perceptions. In particular, most studies investigate whether users and providers consider CEC’s activities, especially ethics consultation, to be helpful . Users are represented by physicians, staff members, residents or trainees , nurses , members of the healthcare team in general , or professionals working within the hospital, such as social workers and pastoral care staff (3). Patients and their families are also included as users of the consultancy service, but only in a minority of studies .Despite potentially raising conflicts of interests, in some cases the evaluation of CEC’s performance is provided by hospital administrators  and CEC’s own members, who are asked to assess how they perceive the impact of their own consultation activities .Satisfaction and positive overall judgment towards ethics consultation prevail over dissatisfaction not only in all the studies involving CEC’s members, as expected , but also when they involve users, with only one study reporting a higher percentage  of physicians’ negative impression (10). Although data reported by the studies and tools used to collect them are too diverse to enable real comparisons, there seems to be a difference in users’ reported satisfaction levels. For instance, patients’ families or surrogates  express appreciably lower average scores in satisfaction than the other groups of respondents. In fact, they rate ethics consultation helpful in 57% of the cases, according to study 29, and two out of six participants (33.3%) of study 5 claim they were very dissatisfied with the consultancy. On the other hand, according to the studies reporting percentages, perceived helpfulness ranges from 65% (4) to 96% (3) for healthcare professionals and from 81% (1) to 88% (4) for CEC’s members.Among healthcare professionals, physicians seem the least satisfied category. In general, physicians are usually more critical towards different aspects of consultation services, even when they declare an overall satisfaction. They complain about the long response timelines to receive recommendations about cases submitted , the lack of any systematic structure, improper analyses (9), and biases in case discussions (21). Physicians also express concerns about the composition of CECs, by which the presence of specialized professionals, or key figures whose presence during consultation sessions is essential for the completeness of the case discussion, should be increased . In this view, including an acceptable number of clinicians would also prevent CEC discussions from being too theoretical and far from the daily routine of clinical practice (10). Other physicians raise doubts over CEC members’ expertise on matters discussed  as well as on consultations’ real usefulness, questioning their need (12) and their effectiveness (10).Differently, in all the studies in which they were enrolled, nurses appear as more satisfied than physicians, especially in relation to ethics consultation: although they seem to have less awareness and access to the ethics consultation services, 83% of nurses rate it as effective, in comparison to 65% of physicians .Although a unique and standardized tool for measuring CECs’ effectiveness was not found, articles selected provide relevant data on the impact of CECs’ activities, which may help in shedding some light on this topic. In more than one article, ethics consultation is considered to strengthen the decisions regarding patient management and to support physicians in their treatment intentions . Many physicians also report they learnt how to fruitfully discuss ethically sensitive issues from case consultations . Other studies find the process of ethics consultation useful to improve quality of care (3) and to promote care values, even, in some cases, by helping hospitals to preserve their (religious) identity (12).Other authors report a positive correlation between the degree of clinicians  and/or patients’ families (29) satisfaction with respect to ethics service and the change in patient’s treatment, perceived as a positive result of the ethics consultation process. Remarkably, some changes in treatment plan occurred in thirty-one out of fifty-nine patients in study 20 and in 33% of patients in study 29.Meetings devoted to ethics consultation are also considered as helpful opportunities to discuss ethically relevant issues , insofar as they are also able to provide healthcare professionals and patients’ families with emotional and social support (4). This evidence is also supported by studies showing a correlation between ethics consultation and a decrease in the level of distress among hospital staff members (14), and among patients and their surrogates (29). In paper 14, twenty-eight out of the thirty-five healthcare professionals involved in the study reported a decrease in moral distress due to consulting ethics services, while in study 29, patients and their surrogates declare that ethics consultation was “reassuring”, “supportive”, and “took the weight off” their shoulders . In general, ethics consultation may give a voice to all the individuals facing—albeit differently—ethical issues in clinical practice, thus making physicians, patients and their families feel that their concerns and perspectives matter .More objective evaluation measures include qualitative analyses of ethics consultation reports, with the aim of evaluating how CECs work during case deliberation and/or how case discussion is conducted . These studies also report, when available, the number of cases in which CECs’ suggestions have been then actually followed by relevant players (18), as well as the following information: the reason for requesting the consultation (18), whether ethical issues have been correctly identified and analyzed, by what method , if the discussion takes place following a specific structure or set of steps (15), how much time is dedicated to the meeting , and how much time is needed to provide requesters with a response .meaningful consensus as a criterion for successfully delivering ethics consultations (13). With respect to the latter, the idea was raised that consensus among CEC’s members in case discussion is not necessarily a value per se, as it could be due to a lack of divergent views or the dominance of a single committee member.Considerations resulting from the theoretical articles are in line with the aforementioned empirical data. More than one article underlines the importance of multidisciplinarity, encouraging CEC to be composed in such a way so as to incorporate all relevant expertise and disciplines . They also highlight the importance of having systematic discussions during CEC’s meetings . Another point is the concept of This review shows that CECs seem to exert a positive impact both on the healthcare personnel and the institutions in which they work, but many aspects of their functioning are still left to dissect. It is apparent that there is a great diversity in the procedures they adopt, mostly in relation to their cultural and geographical contexts. This also makes it difficult to get to shared criteria for their evaluation.Heterogeneity in assessments raises methodological difficulties to make straightforward comparisons and to identify the key factors for a positive impact. Criteria by which CECs’ activity is considered successful, and the definition of success itself, varies considerably from study to study, and from context to context. This makes it difficult to evaluate CECs’ performance. Therefore, the adoption of clear  standards would be useful. However, cultural diversities should be also respected. CECs are meant to be so close to clinical practice that a globally harmonized metric of their success may be unconceivable and possibly not desirable. Nonetheless, as a matter of fact, CECs—particularly in regard to their function of ethics consultation—were largely reported as beneficial by both users and providers in many studies.Clearly, ethical consultation is perceived as the main core business of CECs. Unfortunately, assessing its efficacy is problematic Hoffmann . There iAlbeit few studies have investigated this aspect and more research is needed, this finding could indicate how a broadening of perspectives as allowed by the ethical multidisciplinary review can affect the decision-making process and impact on clinical decisions, thus improving the quality of patient care , we proposed a quality assessment (from low to high) for the ones reporting empirical data. Data considered for quality assessment were the followings: the type of evaluation tool employed, whether the complete dataset was reported, the number and description of enrolled subjects or the number of documents analyzed, and the response rate. We excluded potentially interesting papers (i.e. papers that could have met our inclusion criteria) if they showed a low quality, according to our assessment criteria .With respect to the When training also aims to transfer operational skills (as stated by the American Society for Bioethics and Humanities), any assessment on the application of such skills should take into account that this is an ongoing and iterative process. The evaluation methods should also be modeled according to the specific skill/s conveyed, and on the audience it is addressed—namely, the hospital staff or the internal members of the CEC’s itself. As an example, in the first case, if the skills conveyed regard performing ethics consultation, the training sessions should teach the healthcare professionals first to recognize whether a case is ethically sensitive and then the key elements of ethics consultation ; in this case, the assessment should require the trainee to apply the acquired skills, for instance by asking trainees to discuss an ad hoc clinical ethics case, recognizing the moral dilemma and analyzing the underlying ethical problem. Depending on the resources available, such an assessment can be made either through an oral test, or a focus group, or through a written examination.Concerning the in-house training for CEC's members, as this is on-going training, the assessment should also be on-going. In this case, the members' skills to provide ethics consultation can be tested either through a test at the end of each course ; or through a training day in which this skill is updated and reinforced, for example by collecting particularly relevant cases and using them to practice moral case analysis. Again, the evaluation can be either oral or written.policy preparation and/or policy revision—a key element to evaluate CECs’ performance is to verify if policies have been approved and enforced. Moreover, as it is always fundamental that healthcare professionals of a given institution develop an “ownership feeling”  were written and published before the year 2000. Data reported by those studies would need an update. Only in one case, we noticed an update of data concerning the same CEC through the use of the same questionnaire (Gaudine et al. The aim of this systematic review was to provide an answer to the question whether CECs may be useful, by collecting all evaluation tools used by researchers to assess their impact in clinical practice. Although a definitive answer to this question cannot be provided, our work systematically collected available information. By doing this, our study provides a comprehensive overview of CECs’ impact, highlighting some key elements of their performance. Amongst the three most typical functions of CECs—namely, ethics consultation, policy formation and/or revision, and bioethics education—ethics consultation is largely overwhelming.Despite the lack of standardized assessment tools, CECs appear to be beneficial at the very least in terms of healthcare professionals’ satisfaction. Indeed, the presence of CECs correlates with a lower moral distress among staff members.However, this systematic review stresses the importance of developing standardized tools for evaluating ethics consultation. More work is needed to collect more data with respect to patients and/or their surrogates’ perspectives on this issue. Definitely, in view of an increasingly demand for personalized medicine, the patient’s perspective cannot be left aside.