Recovery Education Centres (REC) in mental health offer a new model of providing recovery supports through emancipatory adult education and recovery-oriented service principles. Despite the widespread adoption of RECs, there is limited evidence regarding factors enabling engagement and participation, particularly for unique subpopulations or service delivery contexts. The Supporting Transitions and Recovery Learning Centre (STAR) in Toronto, Ontario is the first REC in Canada and one of few worldwide supporting adults transitioning out of homelessness. This research aimed to investigate individual and program level enablers of engagement and participation in a REC for this population.Qualitative methods were used to explore the experiences of 20 service user participants through semi-structured interviews exploring their experiences of REC participation and perceived key program features. Interviews were conducted between July 2017 and June 2018, six to 14 months following REC enrollment, and analyzed using inductive thematic analysis.In contrast to past experiences with health and social services, participants described a welcoming and respectful physical and interpersonal environment with low-barrier seamless access facilitating their engagement and participation. Although the realities of homelessness presented barriers for some, participants described that the involvement of peers, as role models, and the self-directed, strengths, and skills-based curriculum, co-produced and co-delivered by peers and professionals, were instrumental in activating the process of recovery through education.Findings are consistent with the growing evidence base of the defining features of RECs and suggest this model can be successfully extended to support recovery among adults transitioning out of homelessness. This unique examination of Canada’s first REC for adults exiting homelessness can help guide program and policy development to better support this disadvantaged population. Homelessness and housing insecurity are increasing challenges in many jurisdictions . The citAmong health conditions, the prevalence of mental health and addictions problems and illnesses in homeless populations is alarmingly high . SeveralIn recent years, recovery has become a new paradigm and target for intervention within mental health services, and in the community. Mental health recovery is defined as a process of personal change leading to a satisfying, hopeful, contributing life, even within the limitations of mental illness , 16. RecAmong recovery-focused interventions, Recovery Education Centres (RECs) offer recovery supports through education rather than traditional health and social services . First eDespite the rising popularity of RECs, and progress in defining key ingredients and fidelity measures , there rThe Supporting Transitions and Recovery Learning Centre (STAR) was the first REC in Canada and the only REC in North America with the mandate of supporting individuals transitioning out of homelessness , 27.At STAR, members participated in classes of their choice led by peers with lived experience of homelessness and mental health challenges, as well as social and health service providers, within a curriculum co-developed with service users . IndividSTAR employees, overseen by a program manager, included a team leader and an administrative assistant, as well as five peer specialists, employed in a part-time capacity, from 0.2 to 0.6 of a 40-h full-time equivalent work week. STAR’s caseload was between 60 and 80 unique service users at any one time, with the majority of individuals taking one to two courses per semester.The center operated as a “hub-and-spoke” model where the hub is a community center with a central classroom and staff offices  in the RThis study is part of a rigorous mixed methods quasi-experimental evaluation of the STAR Learning Centre, located at St. Michael’s Hospital in Toronto, Canada. Qualitative methods were used to explore the experiences of participation of homeless and precariously housed participants . A realiSTAR participants were 16 years of age or older, and had experienced challenges maintaining housing within the past 2 years. Furthermore, study eligibility criteria included: i) capacity to provide informed consent for research participation; ii) being a new member of STAR during the study recruitment period; iii) completion of at least 10 h of STAR classes .Of the 92 participants in the intervention arm of the larger study, 23 were recruited through purposive sampling to participate in an in-depth, in-person, semi-structured interview. Participants were selected based on willingness to participate, and ability reflect on and provide insights into their experiences with STAR as well as other services. Recruitment took place between July 2017 and June 2018, 6–14 months following program enrollment, and continued until data saturation was reached and no new themes emerged . Of the A semi-structured participant interview guide was created during the research protocol development phase . Key topAll participants provided written informed consent to participate. Participants received a $30 honorarium and two transit tokens for each interview. All interviews were recorded with participant permission and transcribed verbatim.Qualitative interview and focus group data were analyzed through an inductive thematic analysis approach , using QSome codes, related to potential key ingredients and other key program characteristics were identified based on literature reviews, using a deductive approach, while the majority of codes, themes, and subthemes emerged inductively during analysis of the transcripts. During analysis, similar codes were grouped into a set of high-level themes supported with direct examples and quotations from the sources. The research team discussed the categories, collectively reduced them to a smaller set of higher-level themes and refined them through an iterative process of review and feedback  to enhanParticipant interviews lasted from 35 min to 100 min. The mean hours of STAR participation among study participants was approximately 80 h. At the time of their interview, the mean age of participants was 44.6, the majority identified as female (N=13 or 65%) and Caucasian . Further demographic characteristics of participants are shown in Participants described largely negative past experiences with local health and social services, highlighting challenges in accessing needed services and revealing personal contextual factors that made engagement in recovery challenging for some. Participants’ perspectives and experiences with STAR were overwhelmingly positive, with no negative experiences emerging as a theme. Participants further highlighted factors that facilitated engagement and participation in STAR, such as the low barrier access and the welcoming and respectful physical and interpersonal environment. Finally, participants described defining program characteristics supporting their engagement in the process of recovery, including the involvement of peers, as role models, and the self-directed, strengths, and skills-based curriculum, co-produced and co-delivered by peers and professionals. These themes and subthemes are further detailed below.Most participants had extensive prior service use, including emergency shelters, housing services, hospitals, mental health assistance and drop-in programs. This allowed for rich descriptions of experiences with both STAR and other services, as described below:The majority of participants (n=15) reported few past positive experiences with services, highlighting a lack of availability of needed services, lack of awareness of existing services, extended wait-times, time-limited availability, and services that were not geared to their health needs. As one participant described:I went in there when I was homeless and when I got there they said, “We won’t be able to give you an appointment for another month.” So, like, so what do you do here? … Who knows if it was out in the winter you’d be out in the cold for a month, and they have no alternative. (P62)[I] put myself back into emergency, again was observed for seven hours and released again, with the promise that I would get a follow-up within 2 weeks for counseling. The call never came. I called them three weeks later, [they said] “Well we don’t do that anymore”. So, I was basically [abandoned]. (P78)Participants also highlighted the lack or responsiveness of services, as this participant described:[l]ook a certain way, or … look more challenged … if you don’t reek of piss, they kinda don’t talk to you. Some services I find are like that and how clean or dirty do I have to be to get my needs met. (P114)Other participants described that available services were not suited to their needs, or were experienced as stigmatizing:Many participants described how their personal circumstances hindered their full engagement with STAR and other services. The majority of participants endorsed barriers such as active physical health, mental health, and/or addiction challenges (n=13), while several highlighted the lack of social capital such as family supports or education (n=8). Others described the impact of precarious housing on their ability to focus on participation and recovery (n=9). One participant discussed turning to alcohol to manage their stress: “two weeks into being homeless I started drinking, daily. I’m a daily drunk when I drink … life is just easier so to speak” (P1). Others described wishing they had the support of others: “I had some family members, but again, being judged from “It’s all your fault, you should’ve been more conscious about stuff” (P108) while another participant considered if their outcome would have been different had neighbors been supportive: “maybe had they been friends I would have gotten a weekend on a couch or something” (P1).[S]he gave me the right contact [information], there’s no wait list to get in, …. There wasn’t all this form you had to fill in, there’s no intimidation, it’s all straightforward. [I was] totally supported (P160).Notably, participants described overwhelmingly positive experiences with STAR, and described program characteristics facilitating engagement and participation. They highlighted the importance of the attractive and dignifying physical space, the low barrier access, the seamless experiences with registration and enrollment, and the welcoming interpersonal environment. As this participant commented: “I remember being like I don’t know what I’m doing, I’m nervous but this community center is gorgeous, and I’ve always wondered about it” (P109). Another participant shared:“you have to actually physically enroll, and that’s what makes it different compared to other programs that I’ve been at” (P63).Participants alluded to ownership being established in the enrollment process: ) and another described being listened to and treated with kindness, justifying their continued engagement: “I feel really listened to, really cared about” (P166).The majority commented on feelings of support and belonging fostered by the welcoming and hospitable nature of staff and co-participants. One participant stated, “I belonged somewhere”  strongly valued lived experience being part of every level of the program. Participants explained that “[b]ook smarts is great but book smarts plus an experience is a little bit stronger” (P133) because “I know that these people have the experience, so they know what they’re talking about” (P136). Specifically, participants believed the inclusion of staff and volunteers with lived experience made the program feel more relevant, practical, approachable, and effective. As one participant described,[i]t makes the program a lot more honest, a lot more relevant, a lot more down to earth, when you’ve got someone like the program director willing to share his struggles when he was younger with addiction … and homelessness … just adds a huge amount of legitimacy to the whole program … Makes it, in my mind, ambitious, inclusive. (P115).[T]he teachers, the peers support workers also coming from lived experience … it is not a clinical approach or a therapeutic approach. It’s one of facilitation and support that I would find very hard to achieve in a therapeutic or clinical setting. The power and value of the lived experience … is invaluable … I believe it’s mandatory in how effective STAR is. (P78)That is, the level of participant engagement, support and connection was bolstered by a culture at STAR where experience with mental health challenges was both valued and shared. For some participants, this program feature was instrumental to engaging them in the model:(P88).As another described: “[i]f you don’t abide by their curriculum then you’re perhaps not accepted to certain programs, whereas with STAR, they’re open to those things and help people with that in this very relatable way” via facilitation.Nearly two thirds of participants (n=13) spoke about the participatory approaches to teaching and learning at the center. Participants valued that programming was developed and delivered in an inclusive way, such a way that “there’s no hierarchy” (P160) and “they [program staff] never feel like they are superior” (P1). Participants described a “very nimble, very open environment for making suggestions” (115) and valued opportunities to become involved in content design and delivery Feeling their voices were heard, and opinions were respected within the program was important to participants as they explained how regular town hall meetings were held during which they were encouraged to contribute to conversations about proposed programming changes. As one of the participants further described,[e]verybody had an opportunity to give feedback or input … they ask for feedback on how they can improve the class and what kinds of things you think you want to learn … they’re always concerned about your feedback too to make it better. (P63).Others described overcoming a fear of failure by participating as a facilitator through STAR: “it’s [okay] to fail … I felt really welcomed … I’ve been given the chance to facilitate classes which gives me that, you know, my self-esteem is right back up there” (P108).Over half of participants (n=11) described their ability to self-direct their program participation and overall recovery as a defining feature. Participants described Individual Learning Plans as a key aspect that fostered their “self-direct[ion]” (P62) and “self-leadership” (P66). These plans created recurring opportunities to “make the choices that I want to make and when I say, ‘Hey, I want to follow this path,’ or ‘I want to get to this point’…they’re able to show me which courses I can do and I can choose whether I want to do them or not” (P109); and because “you have a say in it … you feel in charge” (P88). Participants used their learning plans as tools to develop their self-determination, self-management and keep their goals on track; for example, “always start[ing classes] off with ‘Why do you think you’re here’ or who I want to be today, not tomorrow” (P50).Similarly, participants spoke about the “well-rounded curriculum based on the variable needs of the members” (P78), and the “different types of [courses] … different things to do…” (P89). As another participant explained,[I]n our goal plan … we say what we need and … they find courses that fit that for us. Say you want to learn more about feelings and emotions and triggers, they will add that in. They will add in an art group. They will try to bring someone in for writing. (P50)Others described how the curriculum enabled specific career goals, for example: “I’ve chosen the peer support route and they have those courses to achieve the peer support that I want to achieve” (P114). This STAR feature underscored contextual fit: “[i]t’s just a better fit than a lot of other things that I’ve tried…… it’s about learning” (P113) and [the program] was “very appropriate and supportive in what my goals are” (P62).Despite the widespread adoption of REC worldwide, there has been little research examining key features of RECs in diverse service delivery contexts, or diverse subpopulations of people experiencing mental health challenges. This is the first study to examine service user perspectives and experiences with a REC for people experiencing homelessness and mental health or addictions challenges.Our findings expose past experiences with services for this population in the local context and highlight key enablers of engagement and participation in recovery education. Findings also highlight valued key features perceived as instrumental in facilitating the process of recovery.With multiple past negative experiences with services, and in the context of challenging personal circumstances, engaging in recovery education was not smooth for some participants, who struggled with the realities of homelessness, few social supports, and other personal barriers. These findings are in line with previous literature with participants attesting to physical and mental health challenges, homelessness, and precarious housing in addition to poor connections to the healthcare system as being experiences in their journey to recovery , 33. TheParticipant narratives also described valued program features facilitating engagement and participation, including the open and inviting programming space and the streamlined enrollment and registration process. These findings are consistent with previous literature, highlighting that participants were not engaging with recovery programming that occurred in an unwelcoming environment –41. PartSimilar to previous work with RECs in the UK, participants highlighted several key ingredients facilitating the process of recovery, including the involvement of individuals with lived experience of homelessness in the design and delivery of services; a participatory approach to teaching and learning; and an individualized, skills-focused curriculum, which emphasized self-determination . The keyThe engagement of individuals experiencing homelessness and mental health and addiction challenges in the process of recovery could be mapped onto the Transtheoretical Model of Change  with indFinally, this study exposed, similar to prior research with this population, critical areas where current programming in the local context is failing. Participants reflected on limited service availability and accessibility, long wait times, and time-limited service provision or inappropriate services. Given the complex health needs and poor health and social outcomes of this population, efforts to promote housing stability as well as recovery are urgently needed. Additional research is needed to guide policy and practices that consider and address the structural barriers and the broader social context in which Recovery Education interventions operate, particularly for socially disadvantaged populations.The individual REC described in the current study has fidelity to the overarching REC model of care and consequently, findings of factors facilitating engagement and participation and key program features supporting the process of recovery may be generalizable to other recovery colleges and settings , 44. FurStudy limitations include the point-in-time observations and as a result, do not provide a long-term perspective of how program features impact longitudinal experiences of recovery education. Furthermore, the key ingredients discussed were elucidated from a single study site; multiple settings would be ideal to deduce and concretize key program ingredients for this population. Other limiting factors include the characteristics of our sample, including participants who were predominantly women, Caucasian, and had some post-secondary education. These characteristics may not be representative of the larger population of individuals experiencing homelessness in different settings, or of STAR program participants. STAR service users were ethnically diverse with slightly more male (56%) than female service users; the STAR hub was also located in an ethnically diverse neighborhood. It is possible that participants from diverse backgrounds were reluctant to participate in this component of the research study or faced additional barriers to participation. Finally, although people with lived experience of homelessness contributed to the study protocol and interview guides, and gave early feedback on emerging themes, they did not participate in drafting or revising this manuscript.The study exposes key contextual factors affecting engagement with recovery education, and perceived key ingredients of a Recovery Education Centre for people experiencing homelessness and mental health challenges. Findings can inform efforts to enhance health and housing interventions and improve recovery outcomes for this disadvantaged population.The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.The studies involving human participants were reviewed and approved by St. Michael’s Hospital and the Centre for Addiction and Mental Health, Toronto, Ontario, Canada. The patients/participants provided their written informed consent to participate in this study.BK led the data analysis and manuscript preparation. NR participated in data analysis and drafting of the manuscript. RB participated in data collection, data analysis and drafting of the manuscript. NK participated in drafting of the manuscript. VS secured funding for the study, developed the research protocol, participated in data analysis, and co-led drafting of the manuscript. All authors contributed to the article and approved the submitted version.This study was funded by the Canadian Institutes of Health Research (grant number: 201409FDN). The funders had no role in study design, data collection and analysis, interpretation of data, or in writing of this manuscript.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 ongoing generalization of Internet of Things and its presence and application in multiple fields is generating a large amount of data that can be used to extract knowledge, among other purposes. In this context, algorithmic techniques and efficient computer systems provide an opportunity to successfully address efficient data processing and intelligent data analysis. As a result, multiple services can be improved, resources can be optimized and real-world problems of interest can be solved. This Special Issue on Algorithm and Distributed Computing for the Internet of Things gives the opportunity to know recent advances in the application of modern technologies hardware and software to the Internet of Things. There is a growing interest in applying technologies, such as the Internet of Things (IoT), derived from the intense deployment of Wireless Sensor Networks (WSNs) over recent decades, in relevant fields such as Industry 4.0, smart cities, and connected infrastructures. The use of IoT in these areas can lead to significant benefits, but it also generates a huge amount of data, which should be processed by a system to, in many cases, detect patterns or optimize certain processes of interest. To this end, big data, machine learning, and deep learning approaches are usually considered, leading to more complex and powerful systems than those of previous WSN applications, which also means new challenges to address. Moreover, for critical areas such as connected infrastructures, dependability, sustainability, and security issues should be addressed to ensure specific application requirements.In this Special Issue, we have collected original high-quality articles, clearly focused on theoretical and implementation solutions for IoT, including intelligent approaches , network levels , embedded systems, sensing devices, nonfunctional requirements , deployment strategies, and management platforms. The papers submitted to this Special Issue fitted some of the following topics, which were listed in the call for papers: Smart manufacturing and Industry 4.0; connected vehicles; smart cities; cognitive computing and deep learning; big data processing; edge computing and network intelligence, bringing the computation closer to the data; dependability ; sustainability ; security and privacy; distributed and embedded computing for networked systems; applications, deployment, and management in smart environments; human behavior and context-aware aspects; prediction models; and optimization and metaheuristics.Although each paper published in this Special Issue covers different topics, we can identify four groups where the papers can be classified according to their main focus: Privacy and security; efficiency and performance; algorithms and intelligence; and protocols, networks and infrastructures. However, some of these papers could be classified into more than one of these groups.With regard to privacy and security, we find two contributions.The first article in this group is entitled “Location Privacy Protection in Distributed IoT Environments Based on Dynamic Sensor Node Clustering” , by KonsThe second article in this group is entitled “Pseudo-Random Encryption for Security Data Transmission in Wireless Sensor Networks” , by LianWith regard to efficiency and performance, we find other two contributions.The first article in this group is entitled “Mapping Neural Networks to FPGA-Based IoT Devices for Ultra-Low Latency Processing” , by MaciThe second article in this group is entitled “Distributed Egocentric Betweenness Measure as a Vehicle Selection Mechanism in VANETs: A Performance Evaluation Study” , by AdemWith regard to algorithms and intelligence, we find a wide area of interest where this Special Issue provides seven contributions.The first article in this group is entitled “DNN-MVL: DNN-Multi-View-Learning-Based Recover Block Missing Data in a Dam Safety Monitoring System” , by YingThe second article in this group is entitled “Coverage-Balancing User Selection in Mobile Crowd Sensing with Budget Constraint” , by YanaThe third article in this group is entitled “Optimizing Movement for Maximizing Lifetime of Mobile Sensors for Covering Targets on a Line” , by PeihThe fourth article in this group is entitled “Wireless Charging Deployment in Sensor Networks” , by Wei-The fifth article in this group is entitled “A oneM2M-Based Query Engine for Internet of Things (IoT) Data Streams” , by PutuThe sixth article in this group is entitled “Natural Computing Applied to the Underground System: A Synergistic Approach for Smart Cities” , by ClemThe last article in this group is entitled “Connected Vehicle as a Mobile Sensor for Real Time Queue Length at Signalized Intersections” , by Kai The last group focuses mainly on protocols, networks and infrastructures. We find two contributions in this group.The first article in this group is entitled “An Optimized Probabilistic Delay Tolerant Network (DTN) Routing Protocol Based on Scheduling Mechanism for Internet of Things (IoT)” , by YuxiThe second article in this group is entitled “Design and Analysis of a General Relay-Node Selection Mechanism on Intersection in Vehicular Networks” , by Dun Finally, we thank all authors who have submitted their manuscripts to this Special Issue for considering Sensors and the reviewers for their hard work with the reviewing process.


Several efforts have been made to change management of neuropsychiatric symptoms (NPS) in nursing homes, however only few were successful. Numerous barriers to change in healthcare were identified, yet only one conceptual model is known to study their interrelationships. Unfortunately, this model does not discuss specific barriers encountered in nursing home practice. The aim of this study is to explore perceived barriers to change in the management of NPS in nursing homes and to construct a conceptual framework providing insight into the relative importance and interrelationships of these barriers when improving quality of care.Four focus groups were conducted in different dementia special care units of one Dutch nursing home. Participants were either nursing staff, treatment staff or relatives of residents. Qualitative thematic analysis was conducted according to the five phases defined by Braun & Clarke. Finally, a conceptual framework showing the interrelations of barrier-themes was constructed using text fragments of the focus groups.We constructed a conceptual framework consisting of eight themes of barriers explaining the extent to which change in NPS-management can be achieved: ‘organizational barriers’, ‘personal barriers’, ‘deficiency of staff knowledge’, ‘suboptimal communication’, ‘inadequate (multidisciplinary) collaboration’, ‘disorganization of processes’, ‘reactive coping’ and ‘differences in perception’. Addressing ‘organizational barriers’ and ‘deficiency of staff knowledge’ is a precondition for change. ‘Suboptimal communication’ and ‘inadequate (multidisciplinary) collaboration’ play a key role in the extent of change achieved via the themes ‘differences in perception’ and ‘disorganization of processes’. Furthermore, ‘personal barriers’ influence all themes - except ‘organizational barriers’ - and may cause ‘reactive coping’, which in turn may lead to ‘difficulties to structure processes’.A conceptual framework was created explaining the relationships between barriers towards achieving change focused on improving management of NPS in nursing homes. After this framework has been confirmed and refined in additional research, it can be used to study the interrelatedness of barriers to change, and to determine the importance of addressing them for achieving change in the provided care. In the Netherlands 80.000 people with dementia reside in care facilities such as nursing homes . Of thesThe small effects of interventions for psychotropic drug use may be a result of difficulties to implement interventions and induce change in nursing homes . In thatIn the past, research has been conducted to identify barriers and to classify these into categories i.e., themes. For example, Mentes & Tripp-Reimer  provide In relation, Van Bokhoven et al.  mention A pilot study was conducted in preparation of a larger trial ‘Reduction of Inappropriate psychotropic Drug use in nursing home patients with dementia’ (RID) . In thisTo illustrate the Dutch nursing home setting; in the Netherlands, a psychogeriatric unit of a nursing home has been built in either two ways to adapt to the needs of their residents with dementia: (1) a  larger scale care unit with multiple living rooms, interconnecting corridors and closed-door systems or (2) a small-scale living facility, wherein the unit resembles a general home, which is equipped with a kitchen, living room and individual bedrooms . Life inFour (monodisciplinary) focus group discussions were organized in two care units of one nursing home in the Northern part of the Netherlands. To increase diversity of the sample, one traditionally built large scale care unit and a small-scale living facility were included in this research. Two focus groups included nursing staff and their manager group 1 & 4), one included only treatment staff (group 2) and one relatives (group 3), see Fig.  & 4, oneParticipants of the focus groups were stimulated to express their views and exchange opinions on difficulties in the care process of their care unit for residents with dementia, with a specific focus on NPS and psychotropic drug use. Furthermore, participants were stimulated to discuss general barriers concerning possible implementation of interventions to address and improve the treatment of NPS and reduce psychotropic drug use. A guide to direct the discussion was developed, based upon literature and consultation of clinical experts, following guidelines for conducting focus groups . The focAll interviews were transcribed ad verbatim, and transcriptions were cross-checked with the recordings afterwards. Qualitative thematic analysis was used by continued open coding, wherein barrier-themes identified in previous research were used as background information. Furthermore, the framework was refined until no new information could be added from the existing data gathered in the four focus groups, and the stage of conceptual saturation was reached . The ultData analysis was an iterative process according to the five phases described by Braun & Clarke (2006) and was conducted by two researchers (C.T. and K.V.). C.T. has a background in medicine, while K.V. has a background in psychology. The researchers started the analysis by reading and familiarizing with the data (phase one: familiarizing yourself with your data). Hereafter, relevant quotations for answering the research question were independently marked as free quotations using Atlas.ti software v 7.5.10, . Next, the researchers individually labelled these quotations with codes, staying as close to the text as possible. In addition, memos were attached to contradictions and deviating opinions that were observed by the researchers between or within the transcripts of the focus groups. Then, the researchers discussed all codes until consensus was reached . Subsequently, both researchers independently categorized all codes into barrier-subthemes (using ‘clustered codes’ and ‘subthemes’ in Atlas.ti) and discussed these until consensus was reached to ensure reliability. Afterwards, the researchers (C.T. and K.V.) had multiple meetings to analyze and discuss the relation between different barrier-subthemes. Barrier-subthemes that were related, were brought together in themes of barriers (themes) by D.G. and C.T. . In addition, all memos were crosschecked with the identified themes to check for new insights and content that was not in line with comments from individual participants. Memos with the same content were categorized together and included in the analysis. After grouping all barrier-subthemes into themes, themes were named according to their content (phase five: defining and naming themes). The interrelations between themes of barriers were defined by using text fragments of the focus groups and hereafter visualized in a conceptual framework. Four researchers  had multiple discussions to construct this framework.The study was undertaken in accordance with the declaration of Helsinki , the appN = 2; EQF level 3), RLPN , and the unit manager  of the care unit. Focus group 4 consisted of the following participants: LPN , RLPN , NA  and UM . The focus group of treatment staff consisted of: registered nurses who are responsible for behavioral treatment decisions outside office hours , psychologists , a nurse practitioner who functions at the level of a physician  and a behavioral coach who is responsible for behavioral treatment decisions within office hours , one of whom was male. The last focus group consisted of four partners and two adult children of residents. Half of the relatives was female, half was male. The focus groups took between 84 and 115 min. In the results presented below, the word ‘participants’ is used when participants of all four focus groups reported these findings, in any other case the participant’s function is mentioned.Four focus groups were conducted, see Fig. The analysis resulted in the identification of eight themes of barriers: ‘Organizational barriers’, ‘Personal barriers’, ‘Deficiency of staff knowledge’, ‘Inadequate (multidisciplinary) collaboration’, ‘Suboptimal communication’, ‘Disorganization of processes’, ‘Reactive coping & resilience of organization’ and ‘Differences in perception’. These interacting themes of barriers were brought together in a conceptual framework explaining the extent to which change regarding management of NPS is impaired in a nursing home, given the existing barriers. Some of these barriers are explicitly linked to prohibiting change, as shown in corresponding quotations, others regard impediments to good care, indirectly impairing change. Firstly, we will describe the barrier-subthemes and themes: the building blocks of which the framework is composed. Thereafter, the conceptual framework, which shows the relationships between the themes, will be described.Additional quotations to the ones mentioned in the results below, are included in Table 1 . Each quA11, “We have actually had many different physicians here the past year, now another new one. And every physician also has their own method. And own mindset. And has their own vision on this [psychotropic drug prescription]. And we have to change.”RLPN (pa22)Moreover, a lack of time influenced the transfer and consistency of information between nursing staff (A16) and has been mentioned by the psychologist to impair the information extraction about residents (A17). Lastly, participants indicated that continuous (cyclic) training for nursing home staff was important to get inspired, acquire new insights, and to incorporate these insights into daily practice (A18). The absence of continuous (cyclic) training is a barrier to change.The first theme consists of barriers that were related to the organization and organizational decisions. This theme is composed of the following subthemes: ‘Use of temporary staff’, ‘Insufficient staff on the unit’, ‘discontinuity by frequent staff turnover’, ‘Lack of time’ and ‘Lack of continuous education’. The ‘use of temporary staff’ and a ‘lack of sufficient staff’ on the unit (A4) inhibited the implementation of interventions as well as the continuity of care (A1). In addition, a difficulty in maintaining the continuity of care was caused by ‘turnover’ within the ranks of the physicians (A13) and a ‘turnover’ within the nursing staff . Furthermore, these barriers impeded the extent of change reached.B1: “It’s also up to the person, I think. One is interested more quickly, as you said yourself, to search themselves, what fits with this disease, what should I think of? Is there another approach necessary? Someone else might think: Do I care? I work here and that’s it. { … } I think there are a lot of differences between colleagues.RLPN (pa4)One will deepen their knowledge more than others.”RLPN (pa4)Furthermore, another important barrier-subtheme within the theme personal barriers was ‘negative staff emotions’. It primarily entailed hopelessness of nursing staff regarding the interaction with residents or treatment staff and the proposed treatment of behavior (B4-B6).The second theme consists of barriers that are related to personal factors of staff members and relatives. This theme is composed of the following subthemes: ‘Reduced staff motivation and effort’, ‘Negative staff emotions’ and ‘Dissatisfaction of relatives’. Participants stressed differences in ‘reduced staff motivation and effort’ among staff members. It was considered important to show motivation by showing effort to gain more knowledge, for example on diseases, but participants mentioned that others did not.Lastly, the ‘dissatisfaction of relatives’ might negatively influence the amount of change possible, through repeatedly expressing their disappointment in the matters at hand. In particular, dissatisfaction of relatives was apparent when problems arose on the unit with their relative. Relatives sometimes felt disappointed about turnover of staff and temporary workers (B7).C3, “And if someone totally panics because he sees big spiders walking on the wall, then you know … . Oh … that fits the picture of the disease. So, he sees things that are not there. You can panic about that and so yes … as long … if you don’t have that knowledge … then you would think … that man is not well at all. I have to call the physician quickly as he has to go to the hospital.”LPN (pa3)The third theme consists of barriers that are related to knowledge and has no subthemes. The treatment of NPS and therefore also prescription of psychotropic drugs was strongly related to knowledge of staff, or a deficiency thereof (C1).D1, “In past several years, if someone was given a physical restraint, then that usually remained that way. And before it comes up for discussion again or before it gets discussed like ‘is it actually still necessary that someone is restrained’, that woman is not going to get up anymore. That you … If no one makes a remark about it, that sometimes persists longer than necessary.”BC (pa10)Additionally, not consulting other key staff members, such as RN, LPN and RLPN, impaired a healthy (multidisciplinary) collaboration, even though the exclusion of these members was not done consciously (D3). Lastly, the lack of frequent meetings with this staff was considered odd and might have impaired the establishment of new and effective treatments for residents (D5).The fourth theme consists of barriers that are related to inadequate (multidisciplinary) collaboration. This theme is composed of the following subthemes: ‘Lack of evaluation’, ‘Lack of (multidisciplinary) consultation of key disciplines’ and ‘Lack of multidisciplinary consultations / meetings’. The participants indicated that lack of evaluations of initiated processes of change and of treatments started was a key barrier in inadequate (multidisciplinary) collaboration.The fifth theme consists of barriers that are related to communication. This theme is composed of the following subthemes: ‘Flawed internal reporting and communication’, ‘Lack of sharing experiences’, ‘Unclear communication of changes with family’ and ‘Communication with relatives is considered time consuming’. The theme ‘suboptimal communication’ is a very broad theme, entailing different kinds of aspects such as: 1) communication between staff as seen by relatives, 2) communication between staff as seen by the staff and 3) communication between relatives and staff as seen by staff and relatives.E5, “Especially the old school [LPN], they really have a … really a … a culture of wanting to control, they want to have the right touch. And if they need to ask for help, sometimes that is a … that is too much to ask. Or a … Or … One is not so easily inclined to share a problem. They keep it to themselves. And I find that very unfortunate.”BC (pa10)In addition, there was confusion about the communication of alternations (for example in medication) with family. The physician expected nursing staff to discuss certain alternations in medication with relatives, while the nursing staff experienced difficulties explaining these to the relatives due to flawed reporting by the physician in the patient file (E7). Furthermore, an LPN remarked she thought the communication about the resident with relatives was time consuming. Therefore, often only the bare essentials about the resident were discussed. This resulted in incomplete information in the patient file (E9).One of the relatives of a resident described the communication between nursing staff members as flawed, impairing the process of care (E1). In addition, one of the psychologists mentioned he does not always communicate with his colleagues on who will communicate with the relative (E3). Participants stated ‘lack of sharing experiences’, such as asking for help and sharing success stories, was important to inspire each other into improving care, whereas lack thereof was seen as a barrier.F6, “I think it is important, that they [physician and psychologist] are in a position … in which they can collaborate. So that it is clear, who does which task? Eh … Who is the coordinator? Is the physician the main point of contact in case of NPS or is it the nurse practitioner? Or is it the psychologist? I sometimes find that difficult, I sometimes think who is the captain on that ship?”P (pa6)Lastly, within this theme, the ‘unfulfilled expectations of management’ and their support of staff are important barriers. Staff expected the unit manager to coach and inspire the nursing staff, while in practice the unit managers were predominantly busy with planning tasks (D7).The sixth theme consists of barriers that are related to disorganization of processes. This theme is composed of the following subthemes: ‘Unstructured processes’, ‘Ambiguity of the division of responsibilities and tasks’ and ‘Decision-making culture’. This theme entailed information related to the obstacles, either culture-based or related to a key person, in organizing (care) processes. The necessity of structuring evaluation and consultation about NPS and its treatment was primarily mentioned by the nurse practitioner and psychologists . Furthermore, obstacles in structuring processes were mentioned, such as ideas that do not converge (F4). Moreover, participants expressed confusion concerning the division of responsibilities and tasks. Especially ambiguity about the person who manages the process of care was mentioned .The last item mentioned in this theme was the culture of trying to reach consensus when making a decision. This culture was seen as frustrating by participants, which elongated the time necessary to structure processes (F12).G1, “ … things that are going like this for years, yes that is very hard to break through, to change. That is in everything on this care unit.”UM (pa1)Furthermore, the organization did not proactively involve the relatives in the decision process. Relatives voiced their concerns about the way their input about the care of their relative was not used in the nursing home. They said they did not have any influence on the care process (G4) and that although the relatives were sometimes consulted by the nursing staff, this consultation took place after the final decision already had been made (G5).The seventh theme consists of barriers that are related to resilience of the organization or reactive coping of the persons within that organization. Reactive coping is a coping style in which one awaits circumstances to unfold before responding, which may complicate initiation or maintenance of change. This theme is composed of the following subthemes: ‘Difficulty breaking patterns’, ‘Concerns relatives on changing practice’, ‘Responding late to behavior’ and ‘Not signaling changes in behavior’. Participants mentioned how difficult it was to change existing practice and that sometimes they encountered resistance . The manager of one of the care units explained that it is difficult to break existing patterns, to change.In addition, an LPN mentioned a tardiness in responding to behavior of residents by involving other disciplines afterwards, when the damage was already done (G10). Although interventions have been used to improve the timing, nursing staff maintained their behavior of delayed responding. ‘Responding late to behavior’ and ‘Not signaling changes in behavior’ by staff impaired the care process (G11).The eighth theme consists of barriers that are related to differences in perception. This theme is composed of the following subthemes: ‘Expressed differences in perception between colleagues’ and ‘Observed differences in perception between focus group participants’. The first subtheme was mentioned by participants in the focus groups, while the second was observed in the transcripts between and within the different focus groups by the researchers. These two subthemes are a broad collection of all differences and controversial views expressed and observed in the focus groups., as was illustrated by the psychologist and nurse practitioner.H4, “I think those [restrictions of freedom of the resident] are being evaluated by the physician in the rounds, monthly. That’s not something that’s discussed multidisciplinary … ”P (pa6)If I’m honest, I have never experienced that [evaluation of restrictions of freedom of the resident] before.”NP (pa7)These quotes show that the different disciplines were not aware of the activities, work and tasks of the other. In addition, several differences in perception between focus group participants were observed by the researchers, while transcribing and analyzing the data, using memos. The psychologist mentioned he did not see any need in the presence of registered nurses in the multidisciplinary meetings about behavior of residents, while later on in the same focus group, the nurses emphasized it would have been useful for them to be present in such meetings.H5, “People are broadly discussed in the multidisciplinary meetings. There we address what they need … { … } What would be good interventions, fitting for that person. So, then we have a much broader context than … where we talk about someone. Of course, not everyone is present. For example, you [registered nurses] do not have anything to do with that.”P (pa5)H6, We are actually never present at such meetings [multidisciplinary consultation]. { … } It would be relevant if we’d be present there. Because we work in the evenings, we work at night, the weekends. We are here such a big part of the time. We are always the ones that get called .”RN (pa8)Furthermore, the nurse practitioner thought nursing staff informed relatives about changes in medication. However, nursing staff were under the impression that the nurse practitioner or physician would inform the relatives . Another contradiction was observed about the assumptions on necessity to structure meetings between a unit manager and behavioral coach/nurse practitioner. The unit manager did not want to structure the frequency of evaluation meetings; according to her, this was not necessary in a small setting. The other group, however, emphasized that structuring the frequency and time of these meetings would improve the continuity of care., because the meetings often didn’t take place .There were two ways by which the ‘expressed differences in perception between colleagues’ became clear. First, the participants mentioned differences in the experience of norms and values (H1), vision and work approach and attitude between colleagues . Secondly, there was a difference in view on the course of affairs on for example evaluations by physicians/psychologists and care staffMoreover, the staff remarked that relatives had little complaints, while relatives mentioned many complaints in their focus group, for example on staff turnover .Next, based on the accounts of the participants and our observations in the transcripts of the focus groups, we will explain the relations and hierarchy between the different themes by means of a conceptual framework see Fig. .Fig. 2F“They are appointed by the employment agency, well … nine out of ten times, they do not know chalk from cheese.”FM (pa14).Figure “I think it is important, that they [physician and psychologist] are in a position … in which they can collaborate. So that it is clear, who does which task? Eh … Who is the coordinator? Is the physician the main point of contact in case of NPS or is it the nurse practitioner? Or is it the psychologist?”P (pa6).The third layer consists of an interaction between the themes ‘Suboptimal communication’ and ‘Inadequate (multidisciplinary) collaboration’, ‘Differences in perception’ and ‘Disorganization of processes’. ‘Suboptimal communication and ‘Inadequate (multidisciplinary) collaboration’ were so strongly related that they were put in the same box, there was no way to say which of these themes influenced the other. A poor quality of communication impeded good collaboration and sharing of information, which disrupted structuring of processes. The following was said about this relation: ‘Suboptimal communication’ and ‘Inadequate (multidisciplinary) collaboration’ were causes for observed discrepancies in perception and assumptions. These observed discrepancies in perceptions and assumptions led to unstructured processes, according to the participants . There was no structured approach and there were many ambiguities about agreements made (G7 – G10). Moreover, the unstructured approach and ambiguous agreements resulted in impediments for a structured collaboration and structured deliberations on NPS.“But, again, today I encountered that the behavioral coach wasn’t contacted. So, I think that’s very frustrating.”NP (pa7)It was difficult for care professionals to break already existing behavioral patterns and try a new approach, which impeded collaboration to structure processes . ‘Personal barriers’ were related to all themes except organizational barriers. They were strongly related to the theme ‘deficiency of staff knowledge’, since a ‘reduced staff motivation and effort’ lead to a decrease in knowledge of staff (B2). Furthermore, regarding the identified barriers ‘reduced staff motivation and effort’, ‘suboptimal communication’ and ‘inadequate (multidisciplinary) collaboration, some participants explained that communication and collaboration were a result of motivation and effort of staff. ’.“I’m always a little bit earlier, you [other LPN] always come a little earlier too, so you’ll sit down or leave later. That facilitates information exchange.RLPN (pa23)Because I just joined the team, I think it’s very important for me to receive more information. Obviously, you read, but it is more pleasant to consult like this [face-to-face]. So sometimes I stay a little bit longer.”LPN (pa19)Finally, the result of all previously mentioned themes of barriers, seemed to influence the extent to which change of care processes was impaired in the nursing home.Next, there were two relations: first, ‘Personal barriers’ separately enhanced the negative influence of ‘Reactive coping & resilience of organization’, which was strongly related to ‘Disorganization of processes’ and, through that theme, to the extent of change. Second, an interaction was present between ‘personal barriers’ and ‘disorganization of processes’, via ‘reactive coping’. When staff motivation and effort was reduced, there was usually a reactive coping style, inhibiting the start of structuring processes. In their turn, the subsequent difficulties which can be encountered, caused a reactive coping style and frustration (negative emotions) in staff.In this study we focused on the identification of perceived barriers to change in nursing homes and we aimed to construct a conceptual framework explaining the relation between these different barriers. We extracted eight themes of barriers that impede the extent to which change is likely. Some are direct barriers and some are indirect barriers. For example, ‘suboptimal communication’, ‘inadequate (multidisciplinary) collaboration’ and ‘reactive coping & resilience of organization’ are indirect barriers. These themes do not necessarily influence the extent of change directly, but do so via another theme or route. All identified themes are hierarchically related, wherein ‘organizational barriers and ‘deficiency of staff knowledge’ were the foundation of all other themes. Hereafter, ‘suboptimal communication’ and ‘inadequate (multidisciplinary) collaboration’ may cause ‘differences in perception’, which in turn can lead to disorganization. In addition, ‘personal barriers’ may influence ‘reactive coping & resilience of organization’ and via that route influence ‘disorganization of processes’. Moreover, ‘personal barriers’ influence these interacting layers. Especially ‘reduced staff motivation and effort’ and ‘negative staff emotions’ play an important role herein. The extent to which possible change is impaired can be determined by identifying existing barriers and categorizing them according to the conceptual framework. Subsequently addressing these barriers could enhance the possibility to change.Various barriers, found in this research, are known from previous research. For example, ‘organizational barriers’, ‘personal barriers’ (such as a reduced motivation and effort) and ‘deficiency of staff knowledge’ are well-known categories of barriers to change , 14–22. Although many studies have identified themes of barriers, only one elaborated on the relations between the different themes . WhereasSome of the major studies included nursing staff and treatment staff in their focus groups , 21, ackAlthough our study resulted in a novel framework explaining the relationships between barriers to change in nursing homes, it had some possible drawbacks. First, the study was carried out in preparation of selecting and implementing an intervention for reducing inappropriate psychotropic drug use. The focus of the focus group questions was therefore on management and treatment of NPS in combination with the prescription of psychotropic drugs. We asked concrete questions about suboptimal care and did not use the more abstract terminology of barriers to change. Due to this strategy we hope to have facilitated the conversation and to have elicited specific information about everyday practice. However, there is a possibility that we missed some of the barriers encountered. Secondly, the fact that the manager invited focus group participants and was present in the focus groups of the nursing staff and that the moderator sometimes asked provoking questions, could have negatively influenced participants to speak frankly. Next, there was no physician present in the focus groups since the attending physician was newly employed in this nursing home at the time of the research and was therefore unable to reflect on processes and change in this nursing home. Yet, a nurse practitioner functioning at the level of a physician was present, as was described in our methods section. Nevertheless, although many barriers mentioned concern actions of the physician, it is uncertain if the physician or nurse practitioner was meant. This might lead to a skewed interpretation of barriers. Finally, some barriers found in other research did not emerge in the focus groups in this study, such as culture on the care unit and complexity of the change or intervention trying to be achieved –16. ThisWe can conclude that we have provided a basic, conceptual framework explaining the relationships between different overarching themes of barriers towards achieving change in management of NPS in nursing homes. The framework may be used as a fundament to assess and to classify barriers to change after it has been confirmed and refined in additional research. It can assist in future research in the determination of steps to be taken when wanting to either improve the extent of change possible, or to establish the current extent to which change may be hindered. Future research could focus on the classification of local barriers and try to resolve and address these barriers. Specifically, the ranks of suboptimal communication, inadequate (multidisciplinary) collaboration and personal barriers call for action into resolving the barriers before attempting implementation of an intervention, to provide optimal implementation.Additional file 1: Supplement A. Detailed applied methodology following the consolidated criteria for reporting qualitative studies (COREQ) 32-item checklist


A minority of patients have a late-onset form of disease that presents from late-childhood to adulthood and has a slowly progressive course with prolonged survival.The GM2 gangliosidoses (GM2), Tay-Sachs and Sandhoff diseases, are rare, autosomal recessive genetic disorders caused by mutations in the lysosomal enzyme β-hexosaminidase A Little research has been published documenting patient experiences with late-onset Tay-Sachs and Sandhoff diseases and how the disease impacts their daily lives and functioning. This study explored the most frequent symptoms and functional impacts experienced by patients with late-onset GM2 gangliosidosis through interviews with patients and caregivers.A qualitative research study design was employed, using three focus groups and 18 one-on-one interviews with patients who were recruited at the National Tay-Sachs and Allied Diseases Annual Family Conference. Transcripts were generated from the discussions, and patient quotes were analyzed using a content analysis approach. Concepts were aggregated into symptom and functional impacts, and the frequency of mention in the focus groups and individual interviews was calculated.n = 19, 95%), “clumsy” gait , fatigue ] and impacts  disclosed by patients and caregivers were similar to those previously reported in the literature. However, less frequently described symptoms such as gastrointestinal issues  and coughing fits  have been expanded upon. This study evaluated the immediate impact of these symptoms on the patients’ lives to highlight the burden of these symptoms and the functional limitations on daily living activities, independence, and emotional well-being. The findings were used to develop a conceptual disease model that could serve as a foundation for patient-centered outcomes in clinical trials and provide insights to the medical community that may benefit patient care.Many of the frequently described symptoms [muscle weakness (This study contributes to the current understanding of symptoms associated with late-onset GM2 gangliosidosis, and further identifies the many consequences and impacts of the disease. These symptoms and impacts could be measured in clinical trials to examine the effects of novel treatments from the patient perspective. HEXA) or β subunit (HEXB), respectively, of the two β-hexosaminidase isoenzymes, hexosaminidase A (αβ) and hexosaminidase B (ββ). Tay-Sachs disease is caused by a deficiency of β-hexosaminidase A activity, whereas Sandhoff disease results from a combined deficiency of β-hexosaminidase A and B activities. The reduced ability of patients’ cells to catabolize GM2 ganglioside leads to progressive lysosomal accumulation of GM2 and related glycolipids, hence their classification as GM2 gangliosidoses  and just kind of like give up. Some people just say ‘Huh? Huh?’ … He’ll order a lager and they give him a water and he’ll get annoyed” .Embarrassment was centered on symptoms, such as muscle weakness, gastrointestinal issues, and coughing, as well as impacts such as falling and going out. One patient was embarrassed by how muscle weakness prevented him/her from being able to stand up to use the bathroom, stating “that’s really bad for my psyche and ego … I wear “Depends” and I have my help clean it up at night” (Interview #4). One patient mentioned that the frequent coughing was embarrassing as it caused a lot of unwanted concern from others, particularly when “going out to eat and then you eat and you’re either…you start that cough and people think you’re constantly choking” (Interview #13).A number of patients were worried about disease progression and worsening symptoms. After a bad fall that resulted in a broken patella, one patient was worried, stating “I was scared I would never walk again” (Interview #14). Though some patients were worried about falling, another patient specified that his/her fear was not of falling, but of not being able to get up again. Lastly, the fear of the future and how quickly the disease might progress was a tangible concern, and a patient stated that “I’ve noticed over the last probably six months that I’m definitely starting to progress…not quickly, but much quicker than I have in the previous four to five years” (Interview #16). What was even more concerning for this patient was that it would be many years before he/she would be able to collect a disability check, and “I don’t know if I’m going to be able to make it that far” (Interview #16).Patients were acutely aware of the effect that late-onset GM2 gangliosidosis has on other people. They felt that their disease is burdensome to others and that these feelings made it difficult for them to proactively engage with other people. Difficulty communicating was reported in 11 interviews (55%) and affected communication with family, friends, and strangers. Despite patients’ best efforts to communicate, “some people won’t take the time to try and listen” (Interview #6).Due to the actual and perceived social hurdles, patients and caregivers felt that their disease negatively impacted relationships as “friends, family, everything, his whole life was affected by this” (Interview #6). Engaging in romantic relationships was difficult, particularly for one patient who “used to be engaged and it never worked out because she couldn’t handle my condition” (Interview #4). Another patient mentioned that having the disease was very challenging for his/her self-esteem to handle having “such a difficult diagnosis, looking for a partner. Most women, they’ll run for the hills when they see that” (Interview #11). One patient felt socially isolated due to limited mobility, stating “I can’t dance at weddings. When people go to weddings and they are getting up to do the Hora or whatever, I am not and I am not too happy about that. My girl got married two years ago and I was sitting down. My husband danced, but I could not dance” (Patient Focus Group #1).Most patients were unable to work in the career of their choice due to the physical limitations of the job, communication obstacles, or financial constraints. Six patients (30%) reported that the disease affected their productivity, as muscle weakness affected jobs that ranged from physical labor –“I just could not handle the climbing up and down in the truck all day long” (Interview #15) to fine motor skills – “He had problems with the weakness and typing in the computer” (Interview #14). A caregiver stated that due to the patient’s worsening speech difficulties, “she ultimately lost her job. … So rather than having a career in marketing, she’s a cashier at a supermarket. …. Her career is no longer an option for her” (Interview #8). Lastly, patients had few opportunities available as the job needed to meet certain requirements, such as “I have to get a job that I could do in my wheelchair. And I have to get a job that’s close enough to home for me … That dictates where I can work” (Interview #1).n = 10, 50%). Being unable to work caused financial difficulties and many patients needed to live with their parents for symptom and financial support. As one patient was unable to work a full-time salaried job and was on disability, her caregiver stated that “The disability only allows her to make a certain amount of money a month, which limits whatever kind of job she can get. … She could no longer afford to support herself” (Interview #8). Due to their disease symptoms, patients often had to purchase specialty items, such as an electric chair to navigate stairs, and others made changes to their homes such as “put[ting] a foam floor on the top of a packed … floor, so when she falls, it’s not as bad” (Caregiver Focus Group). When a patient mentioned in a focus group that “I will have to sell my house and move to a one level eventually” (Patient Focus Group #1), others also echoed the need to move to a more accommodating residence. Since some patients required non-medical services, such as a trainer or nursing service, these were not covered by health insurance and required out-of-pocket payment. Patients were acutely aware of the amount of money they earned in order to remain eligible for Medicaid, which was necessary as “prescriptions are over $1,000 a month, so I can’t be without Medicaid” (Interview #1).Patients and caregivers were significantly impacted financially due to disease management or securing their future financial stability. Patients expressed an overwhelming concern about being eligible for or staying on disability (n = 3) and speech and walking (n = 2 each). For treatment, four patients each felt that preventing disease progression and being able to walk would be the most important treatment benefits. A meaningful improvement in symptoms would be reducing muscle weakness (n = 4), improving the ability to communicate, improving clarity of speech, and not needing to use a wheelchair (n = 2 each). Overall perspectives are presented in Tables Patients were asked to consider what the most bothersome symptoms of late-onset GM2 gangliosidosis were and which were the most important to treat. Due to time constraints, not all patients were asked these questions. Five patients felt that the most bothersome symptom was muscle weakness, followed by lack of independence . Caregivers noted that many patients experienced frustration in their inability to self-care, help their caregivers around the household, and move independently. One caregiver stated that, “I think the biggest problem [he] had was stopping driving. That was his last vestige of independence. It didn’t matter that he was in a wheelchair” (Caregiver Focus Group). With the progression of disease over time, caregivers expressed that patients felt increasingly isolated as a result of their reduced ability to express themselves or actively engage with others in social situations.The aspects of late-onset GM2 gangliosidosis perceived by caregivers to be most burdensome for patients were the lack of independence, impact on self-esteem, and muscle weakness, confirming patient reports that muscle weakness was among their most bothersome symptoms. Caregivers noted that the lack of independence and impact on self-esteem were often linked. For one caregiver, the patient who he/she cared for “feels he is a burden. It doesn’t matter what I say or do. I can’t seem to help him overcome that lacking that he has” (Caregiver Focus Group).A conceptual model Fig.  was deveThis study used a semi-structured interview method that consisted of focus groups and one-on-one interviews to elicit patient perspectives (complemented by caregivers’ views) on the symptoms and functional limitations experienced by patients with late-onset GM2 gangliosidosis. A conceptual model was developed to capture the relationship between symptoms, functional limitations, and other impacts of the disease. As no instruments exist that specifically measure the impact of late-onset GM2 gangliosidosis on the patient experience, this conceptual model may be used to develop new disease-specific clinical outcome assessments (COAs) and inform clinical trial and registry designs.7,8 In both late-onset Tay-Sachs and Sandhoff diseases, patients described their balance and frequent falling as being “clumsy” (Patient Focus Group #2). Development of a broad-based ataxic gait that made walking difficult is well-documented, and this study captured various adaptations by patients, including assistive devices, specialty adaptations to the home, and changes to gait to assist with mobility.Patients mentioned experiencing symptoms of muscle weakness and muscle tremors, as well as altered speech , all of which are cited as being more apparent in the first decade of disease manifestation, indicating underlying neurologic problems.Some of the symptoms of late-onset GM2 gangliosidosis disclosed by patients and caregivers in this study are similar to those reported elsewhere, particularly problems with balance and walking, muscle weakness, and cognitive function , 10. HowThe main strength of this study is the patient-centered approach, which focused on generating a representative view from patients and caregivers. Considering the rarity of the disease, a relatively large sample size of 20 patients was attained. The qualitative study design involved both focus group and individual interviews, which minimized any potential limitations that may exist with either method of data collection alone. Data saturation was achieved with the patient interviews, and the caregiver group corroborated the most salient concepts. The consistent input from participants supports the likelihood that the perspectives documented here are representative of patients with late-onset GM2 gangliosidosis. However, it is possible that since the population of this study was drawn from people who attend a disease-specific patient advocacy conference, they may differ from other patients with late-onset GM2 gangliosidosis. Nevertheless, the opportunity to use national patient/family meetings to collect qualitative data from a broad spectrum of patients and caregivers is an important one and represents a patient-centered approach that recognizes patients as the experts of their conditions.The limitations of this study are those that are common to similar studies in a population of patients with a rare disease, especially one that has limited peer-reviewed literature. The retrospective nature of the research, the self-reporting by parents and other family members, and the presence of disease-related features makes it difficult to exclude recall bias and ascertainment bias. As this study was not designed to evaluate subgroups such as gender or age, there may be different experiences of the disease across populations. Additionally, there were only four patients with late-onset Sandhoff disease who participated in the study, and thus the findings may not fully reflect the experiences of all patients with late-onset Sandhoff disease. However, the four patients with late-onset Sandhoff disease who participated in this study largely reported similar experiences to what was reported by patients with late-onset Tay-Sachs disease. Clinically, late-onset Tay-Sachs and Sandhoff diseases are considered as one entity.This research has demonstrated that patients experience many consequences of the symptoms of their disease, some of which have a direct impact , while other, more indirect impacts, extend over a longer term . The symptoms with the most impact present viable opportunities for evaluation as possible primary or secondary endpoints in late-onset GM2 gangliosidosis clinical trials. Patients were most frustrated by their muscle weakness, lack of independence, altered speech, and lack of/limited mobility, and many expressed a desire to improve these symptoms. In more advanced states of the disease, speech becomes more burdensome for patients as communication becomes more difficult. The conceptual model will be validated in a future study and can provide insight to the medical community. In addition, a repeated measures approach could be useful in order to better understand the trajectory of the condition and how patients are impacted by and cope with it over time.There is a clear need for additional support and solutions for patients that would enable more independence and management of their own lives. Research into ways to enable patients to move more independently, especially in moderate to advanced disease stages, as well as being able to communicate more effectively, would strongly improve patients’ quality of life by enabling them to be mobile, better understood, and independent.This study revealed that patients with late-onset GM2 gangliosidosis experience difficulties with walking, balance, and communication that limit many aspects of their lives, from relationships to career choices. The findings and development of this preliminary conceptual model may prove useful to the recognition or development of instruments that will optimally measure relevant clinical outcomes in clinical trials, registries, or other studies. In the long term, these findings are an important contribution to the current understanding of patients’ and caregivers’ experiences with late-onset GM2 gangliosidosis and may lead to the introduction of interventions designed to better support patients.Sanofi Genzyme collaborated with the National Tay-Sachs and Allied Diseases (NTSAD) patient association in the United States (US) to identify potentially eligible patients and to conduct the interviews. Adult patients with late-onset GM2 gangliosidosis and their caregivers were recruited through their attendance at the NTSAD Annual Family Conference in 2015 and/or 2018. No other screening was conducted to determine eligibility for participation.The protocol, informed consent form, and relevant supporting information were submitted for review and approved by Schulman IRB and Advarra IRB. The study was conducted in accordance with the ethical principles outlined in the Declaration of Helsinki and consistent with Good Clinical Practice and applicable regulatory requirements.Informed consent was obtained in writing from interested patients and caregivers prior to participation in the focus groups and individual interviews. To preserve patient confidentiality, all transcripts and audio recordings were de-identified and no personal identifying information was collected.A qualitative research design using semi-structured focus group and individual interviews with patients and caregivers was adopted to elicit multiple perspectives on each topic . In 2015Each focus group comprised between five and seven participants. To ensure homogeneity between focus group participants, the GM2 gangliosidosis patient groups differed by age distribution, time since diagnosis, and living situation: one group with older and more advanced patients (Patient Focus Group #1), and the other one (Patient Focus Group #2) with primarily younger patients diagnosed within the previous 5 years and living alone or with parents. The sessions were audio recorded with participant permission to capture the discussion verbatim in the transcripts in order to be analyzed a posteriori. The patient focus groups were designed to improve the understanding of the signs, symptoms, and burden of disease by obtaining the views directly from the perspective of patients with late-onset GM2 gangliosidosis. To further confirm the symptoms and impacts that were experienced by patients, a single focus group interview was held with seven caregivers of some of these patients. This focus group was designed to expand upon the concepts spontaneously elicited by patients and obtain information on concepts that were not fully discussed with patients.In 2018, 18 interviews were conducted with patients with late-onset GM2 gangliosidosis, nine of whom had also participated in the 2015 focus group interviews. All of the interviews were conducted one-on-one, with the exception of five patients who were interviewed with their caregivers assisting in communicating the full breadth of their experience. The interviews lasted approximately 75 min and the goal was to confirm the most salient symptoms and impacts for patients with GM2 gangliosidosis.Data were pooled across the 2015 and 2018 interview cohorts to describe the symptoms and functional limitations of GM2 gangliosidosis. While this included data from the nine patients who participated in both the focus group and individual interviews, their 2018 data was retained in the analysis because the one-on-one interviews provided a more in-depth assessment of their experiences. There also was the possibility that patient experiences may have changed in the three-year period between the interviews. The initial analytic step involved repeatedly listening to recordings and reading transcripts while taking notes, and comparing the recordings and transcripts with the minutes made during the focus group sessions. Meaningful information was organized into themes that described and categorized the possible observations. De-identified interview transcripts were reviewed and analyzed in ATLAS.ti 8 software using content analysis techniques to extract meaningful themes and concepts. The frequency with which themes and concepts were mentioned was the primary method for establishing the salience of their relevance to GM2 patients. The transcript from each focus group was treated as a single patient in analysis, as it was not possible to distinguish the identities of individual speakers from the focus group transcripts.Concept saturation was assessed to ensure that the number of interviews was sufficient to confirm that all of the most relevant concepts related to the late-onset GM2 gangliosidosis patient experience were identified.


During the Covid-19 pandemic, risk communication has often been ineffective, and from this perspective “fake news” has found fertile ground, both as a cause and a consequence of it. The aim of this study is to measure how much “fake news” and corresponding verified news have circulated in Italy in the period between 31 December 2019 and 30 April 2020, and to estimate the quality of informal and formal communication. We used the BuzzSumo application to gather the most shared links on the Internet related to the pandemic in Italy, using keywords chosen according to the most frequent “fake news” during that period. For each research we noted the numbers of “fake news” articles and science-based news articles, as well as the number of engagements. We reviewed 2102 articles. Links that contained fake news were shared 2,352,585 times, accounting for 23.1% of the total shares of all the articles reviewed. Our study throws light on the “fake news” phenomenon in the SARS-CoV-2 pandemic. A quantitative assessment is fundamental in order to understand the impact of false information and to define political and technical interventions in health communication. Starting from this evaluation, health literacy should be improved by means of specific interventions in order to improve informal and formal communication. Severe acute respiratory syndrome coronavirus 2), due to the similarities with the SARS virus (SARS-CoV) [On 11 March 2020, only ten years after the H1N1 swine influenza pandemic . This isARS-CoV) .The first cases in Italy were reported on 30 January 2020, when a couple of tourists tested positive in Rome . Since tBy mid-July 2020, the pandemic accounted for 13,486,823 cases, with 581,965 deaths and 7,876,115 recovered patients worldwide . This faIn such times, people can be prone to scapegoating; religion, ethnics, wealth, and gender can all become social dividers that can heighten the social unrest and uneasiness . The uneConspiracy theories and alternative facts find fertile ground on the Internet, feeding off the restlessness and helplessness of people, frustrated firstly because they could not foresee the disaster and secondly because they are unable to indicate the culprit of such tragedy. The void left by the absence of certainty can make any kind of explanation plausible and reasonable to the eyes of the general population. United Nations Educational, Scientific and Cultural Organisation) recently publicized a handbook about the issues affecting journalism and information. One of the purposes is warning that the term “fake news” does not have a straightforward or commonly understood meaning. In fact, “news” means verifiable information in the public interest; information that does not meet these standards does not deserve the label of “news”. Therefore, “fake news” is an oxymoron that lends itself to undermining the credibility of the “real” news. Thus, it would be better to use other terms to describe a phenomenon where there is a spreading of untruths via social media and the Internet, involving disinformation , misinformation , and mal-information  [Although there is still not a clear definition of this false information, or so-called “fake news”, we can affirm that such articles are born from a piece of news that is deliberately built from false and mendacious information but looks plausible and factual to the eyes of the reader. The aim is to manipulate and mold public opinion. While a factual news story is created by a journalist, fake news is a product of both the writer and the audience, inasmuch as it only exists if it is perceived to be real . The Intcountry) .A recent study claims that 82% of Italians are not able to recognize fake information . The Intth Generation) technology being implemented in many countries [Digital and health illiteracy can account for most of the inaccurate news stories depicting the cause of the pandemic, whether suggesting it is a virus engineered purposely in a Wuhan laboratory , a bioloountries . Some peountries . We can ountries ,26. Such fake stories are intentionally built to rapidly and tumultuously spread over the Internet and social media. Their popularity is rapidly increasing, such that retractions are always published too late. As many philosophers used to say, “a lie can run around the world before the truth can get its boots on” .The seriousness of the situation was duly described in a recent issue of The Lancet, which published a statement in support of scientists, public health, and medical professionals, condemning all theories validating a non-natural origin of the SARS-CoV-2 virus and citing a list of articles proving the enormous amount of studying that was going into the sequencing of the genome of this novel pathogen ,30,31,32The Lancet again published an article in 2015 denouncing the still underestimated inadequateness of health literacy in Europe , and mor“Fake news” has inevitably and indubitably influenced health communication in the Covid-19 emergency and it is clear that it might continue to do so for the foreseeable future, contributing to social unrest and uneasiness. The aim of this study is to measure—using nine specific key words—how much false and true information have circulated in Italy in the period between 31 December 2019 and 30 April 2020, as part of the phenomenon called infodemia . More geWe used the BuzzSumo pplication  in orderBuzzSumo gathers data across the social media platforms Facebook, Pinterest, Reddit, and Twitter to generate a list of article links with the highest online engagement. Engagement is defined as the total number of interactions that users have with a particular article link, including actions such as “liking”, “commenting”, and “sharing” on social media ,40. SincWe restricted our search to three specific date ranges: (1) 31 December 2019–19 February 2020; (2) 20 February 2020–10 March 2020; (3) 11 March 2020–30 April 2020. The data ranges were selected according to self-evident pivotal moments in the events timeline of the Covid-19 pandemic in Italy , “origin” (“origine”), “laboratory” (“laboratorio”), “plot” (“complotto”), “HIV” (“HIV”), “vitamin C” (“vitamina C”), “vitamin D” (“vitamina D”), “garlic” (“aglio”), “5G” (“5G”). The keywords were chosen according to the most frequent topics of the “fake news” that spread during the Covid-19 pandemic. Such a specific set of keywords was purposefully chosen because it reflected our intent to only look for health-related news stories about Covid-19. The 9 keywords were chosen in a consensus meeting of the research group, since they were the most likely to uncover health-related false information using the BuzzSumo search engine, and specifically fake news that would not meet our exclusion criteria.Searches were filtered by time and language , and for each one of the 9 terms a search was conducted on the three specific date ranges we exposed previously. Therefore, we conducted 27 separated searches.We noted the total findings that the BuzzSumo application was able to provide. We manually revised each one of the top shared results , looking for false information  and incomplete or false coverage of Covid-19-related content, as well as science-based news and articles about those topics. We applied strict exclusion criteria when conducting our review. We excluded content that was merely a report of events, focusing on medical and scientific subjects. An article was immediately excluded when the content did not deal specifically with health or science, i.e., the focus may have been on the socioeconomic consequences of the pandemic, which was a topic we excluded from our fake news review. Moreover, we decided to include articles that were reporting fake news about SARS-CoV-2 to give a sense of reasonable doubt, i.e., claiming that there were certain allegations about a certain topic but pointing out that such allegations had no evidence whatsoever supporting them. Articles were classified as fake news if the content was not supported by scientific literature or when the data reported were used to make inappropriate conclusions about SARS-CoV-2. The contents of the most frequent fakes found for each topic are reported in Overall, we reviewed 2102 articles that were generated by our keywords search on BuzzSumo. The analyzed topics attracted public attention with unequal distribution:—laboratory, vaccine, and 5G articles accumulated more shares than other topics.Altogether throughout the three periods, links that contained untrue information were shared 2,352,585 times, accounting for about 23.1% of the total shares of all the articles reviewed. The topics most contaminated with fakes were vitamin D (89.4%), HIV (77.8%), and garlic (71.2%). A synoptic comprehensive analysis of the data we gathered from the keyword search is shown in D. Comprehensive and synoptic data analysis of all content analyzed are reported in The increasing percentage of “fake news” for numerous topics of research seems directly linked to specific events, for example when popular or well-established sources start supporting theories about SARS-CoV-2 without presenting evidence for their claims.As a significant example, we can analyze the fluctuations of this false information when searching “coronavirus HIV”. In the first period, there was 1 fake article with 767 total shares, in the second period there was 1 fake article with 3 total shares, and finally in the third period there were 15 fake articles with 72,715 total shares. Such a rapid increase can be traced back to some statements about the origin of the virus by the 2008 Nobel Prize for Medicine winner Dr. Montagnier, which were not supported by any evidence. Something similar seemed to happen when searching for “coronavirus laboratory” (“coronavirus laboratorio”). In the first period, this term accounted for 55 fake articles and 1,039,224 total shares, while in the second period only 23 fake articles and 12,972 total shares were counted. The first period coincided with claims by a news reporter from a popular newscast about the alleged laboratory origin of the virus in Wuhan. The trend even more evident when searching “coronavirus garlic” (“coronavirus aglio”), where in the first and second periods 0 fake articles and 0 shares emerged, while in the third period 7 fake articles received 5284 total shares, whereby garlic was described as a miraculous treatment for Covid-19 without presenting any evidence whatsoever . When searching for “coronavirus vaccine” (“coronavirus vaccine”), the total numbers of shares between the first and third period increased from 14,106 to 206,900, an 18.5-fold increase, following the more extensive media coverage about vaccine research for SARS-CoV-2. Similar patterns of fluctuations of false information were found for the other keywords for topics dealing with prevention or treatment of Covid-19. In conclusion, it was concluded with no exception for any given keyword that fake news in the Covid-19 pandemic succeeded multiple times in overshadowing formal verified news. This phenomenon was in many cases not only limited to the gross number of new articles produced, but also the number of shares on social media. Fake news indubitably and perceivably affected health communication during the Covid-19 pandemic. What this study tried to achieve was to quantitively gauge the amount of fake news and to decipher the common patterns and mechanisms that underlie the tumultuous spread of fake news on social media. The data gathered was also able to show how apparently few fake news story  can account for a vast majority of shared news stories on social media . This is very self-evident when looking at the data for the keyword “garlic” (“aglio”), where 18% of the news stories that were classified as fake accounted for almost 70% of the shares on social media. The spread of false information, or so-called “fake news”, in non-official communication can ultimately be considered a very disruptive and dangerous phenomenon that can profoundly undermine health and risk communication, particularly in an emergency, such as the one we are living through.The number of news stories regarding the Covid-19 pandemic continued to increase through the three periods we considered, which is easily understandable, as the virus started to spread dramatically throughout the globe and newscasts increasingly focused on that subject. As the number of news stories increased, so did the number and percentage of untrue information about the Covid-19 pandemic. Our observations regarding the percentage distribution between shares and news were particularly interesting . In 6 ouThe tendency to produce false content is born and spread for a variety of reasons. One of the main reasons is when such news stories are conveyed by authoritative figures, as in the case of the lab incident in Wuhan while testing an HIV vaccine . When thOther reasons can be attributed to the fact that Covid-19 is not only a health issue, but also a large-scale socioeconomic disaster, which is so deeply impactful that it has monopolized international newscasts. The pandemic contributed further complicated the political relations between the US and China, the two biggest economies in the world, to the point that some are calling it a new cold war . In thisThe last and perhaps more important reason can be found in the scapegoating phenomenon. One dramatic aspect of the response to the pandemic was the desire to assign responsibility by identifying the culprit, which is a recurrent situation in history. For instance, during the Black Death in 1347, this role was attributed to the Jewish people—they were accused of poisoning the water wells with the intent of killing all Christians. The same happened in the 1630 plague—in “The Betrothed”, Manzoni described how the blame was casted upon disease spreaders, astral influences, or poisonous exhalations, the so-called miasmas .The scapegoating phenomenon exploits existing social divisions of religion, race, ethnicity, class, or gender identity, fueling social conflict and friction between governments and the general population . To understand the reasons for the popularity of “fake news”, it will be necessary to deeply analyze and better understand the psychological mechanisms underlying its success.The universe of “fake news” is heterogeneous, but recurring elements are identifiable in terms of the context, methods of diffusion, and the target population that the fake news is addressed to. As for the context, social networks have proven to be the most fertile ground for the spread of false facts. They offer a great space for profiles and pages that are created with the precise intent of generating and spreading fake news ,51. False facts are usually presented with some distinguishing features—the content is mostly represented in the title, while the page that shows the entire text (once the title is clicked) often shows only a bare repetition of the key concept .Titles are usually short with “clickbait” features, written in larger fonts, with a sly use of colors and photos. The aim is to appeal and attract an audience that is used to ratiocinating intuitively, through “heuristics”  that lead to “biases” (cognitive distortions that lead to misinterpretation of the text) .The intrinsic features of the target population also play a role in the spread of “fake news”. Recent studies show that subjects prone to depression, disappointment, suspicion, and religious fundamentalism are more susceptible to this type of content . Age is Although the key psychological mechanisms still require thorough research, many experiments point to a correlation with the scores obtained in tests that assess analytical skills, such as the cognitive reflection test (CRT) . CRT is Social media also allows for massive repetition of “fake news” content, creating some sort of “echo chamber” phenomenon. The constant repetition of the same false information can trigger a feeling of familiarity in the reader, determining a further drop in attention and fact-checking. This all finally results in a greater acceptance of the content propagated by the “fake news” story—the more false content is shared, the more a user is led to believe it ,60,61,62In this scenario, several research studies are aimed at finding strategies to reduce the impact of “fake news” and inaccurate information. This goal could be achieved by limiting the repetition and massive sharing of inaccurate content. However, it is a difficult approach, since repetition and echo-chamber effects are intrinsic characteristics of social media. Another possible solution would be to immediately deny and discredit “fake news”. This strategy could work provided that the retraction is rapid and effective. It has been proven that even after pointing out incorrect information, the initial influence of false information cannot be undone. According to a study that investigated the persistence of feelings, some individuals, especially those with lower cognitive abilities, may continue to foster negative feelings or express discomfort towards a certain topic even after disconfirmation of the false information .A strategy that has been more successful in the fight against “fake news” promotes the use of nudging; it would be enough, in fact, to remind the subjects to pay attention in assessing the veracity of some statements before reading them in order to improve their ability to understand which news is false and which is not . FurtherOur study illustrates the “fake news” phenomenon in the Covid-19 pandemic. The spread of false content related to health communication was a very prominent feature throughout the three periods we considered, which had a deep and meaningful impact upon the general population. It was an element that undoubtedly undermined informal communication during the health emergency. Health-related fake articles have been thoroughly studied and analyzed, especially in the last few years, when they became a disruptive element in the conversation about vaccines. It was not a surprise to encounter false facts during the Covid-19 pandemic.Such mendacious and false new articles are easily traced back to their source and probably do not require more top-down, specific qualitative studies about their origin and nature. However, we believe that a quantitative and qualitative evaluation is fundamental in order to assess the real impact of fake news and to start defining political and technical interventions in health communication.In conclusion, we believe that there are two paths to minimize the impact of “fake news”. On the one hand, we must improve health and digital literacy—it has been demonstrated that a low level of health literacy brings people who have suspected symptoms related to Covid-19 to feel more stressed and depressed than people who have higher levels of health literacy , which a