Enhancing human aspects of care with young people with muscular dystrophy: An evaluation of a participatory qualitative study with clinicians.

PURPOSE: This paper evaluates a study which aimed to enhance clinical care of young people with Duchenne or Becker muscular dystrophy (MD) and their families in two Canadian neuromuscular clinics. We report on how/why the study changed clinical practices in relation to the 'human' (e.g., emotional, social, existential, cultural) dimensions of living with MD. MATERIALS AND METHODS: The intervention involved regular dialogical exchanges with clinicians across the two sites, during which direct observations of the clinics' care practices were discussed and changes were planned. We drew from realist evaluation approaches to assess changes in clinical care associated with the intervention. Data sources included dialogical exchanges; clinic observations; interviews with clients, families and clinicians; and team analysis sessions. RESULTS: Our evaluation suggests the clinical teams shifted their thinking and practices towards greater consideration of human aspects of living with MD including: more routinely attending to emotional, social and experiential dimensions of living with MD; reconceptualisation of risk; and considerations of affective aspects of clinical care. Not all clinicians changed their thinking and practices in the same ways, or to the same extent, and there were differences between the sites. These differences were likely due to numerous factors, including varying levels of clinician comfort with examining and shifting their own practices, and differing formal and informal clinic routines at each site. CONCLUSIONS: Overall, this intervention was able to shift clinic practices, and could feasibly be adapted across rehabilitation settings.

A qualitative exploration of the priorities and experiences of children with Duchenne muscular dystrophy, their parents, and healthcare professionals around weight management.

PURPOSE: The quality and length of life for boys with Duchenne muscular dystrophy (DMD) has improved due to medical advancements, but obesity has emerged as and may pose a risk to their physical health. Clinical guidelines recommend attention to weight management, but healthcare professionals (HCPs) find implementing them in clinical care challenging. Little information is available about the perspectives of children with DMD and their families around weight management. This study explored the key priorities of children with DMD, their parents, and HCPs who treat them, around weight management. METHODS: Qualitative, individual, semi-structured interviews were conducted and analyzed using a qualitative descriptive approach. RESULTS: Participants included parents of children with DMD (n = 13), children with DMD (n = 10), and HCPs (n = 14). Theme one: "Competing priorities between healthcare providers, parents, and boys" contained two sub-themes: (i) Body mechanics and function; and (ii) Psychosocial well-being. Theme two: "The realities of living with Duchenne's Muscular Dystrophy," with subthemes: (i) Striving for normality; (ii) The trajectory of DMD; (iii) The labour associated with DMD. CONCLUSION: HCPs, parents, and boys have diverging worldviews around weight management, highlighting the importance of integrating the priorities of families into care, even when not aligned with guideline recommendations.IMPLICATIONS FOR REHABILITATIONHCPs must understand the competing priorities in the lives of children with DMD and their families when discussing weight, weight management, and lifestyle changes.Quality of life and living a "normal" life are prioritized by children and families over the surveillance and time demands of lifestyle routines recommended by clinicians for weight management.Weight management recommendations should be based upon the individual needs and priorities of the family.

Enhancing the human dimensions of children's neuromuscular care: piloting a methodology for fostering team reflexivity.

For those with chronic, progressive conditions, high quality clinical care requires attention to the human dimensions of illness-emotional, social, and moral aspects-which co-exist with biophysical dimensions of disease. Reflexivity brings historical, institutional, and socio-cultural influences on clinical activities to the fore, enabling consideration of new possibilities. Continuing education methodologies that encourage reflexivity may improve clinical practice and trainee learning, but are rare. We piloted a dialogical methodology with a children's rehabilitation team to foster reflexivity (patient population: young people with Duchenne's or Becker's muscular dystrophy). The methodology involved three facilitated, interactive dialogues with the clinical team. Each dialogue involved clinicians learning to apply a social theory (Mol's The Logic of Care) to ethnographic fieldnotes of clinical appointments, to make routine practice less familiar and thus open to examination. Discourse analyses that preserve group dynamics were completed to evaluate the extent to which the dialogues spurred reflexive dialogue within the team. Overall, imagining impacts of clinical care on people's lives-emphasized in the social theory applied to fieldnotes-showed promise, shifting how clinicians interpreted routine practices and spurring many plans for change. However, this reflexive orientation was not sustained throughout, particularly when examining entrenched assumptions regarding 'best practices'. Clinicians defended institutional practices by co-constructing the metaphor of balancing logics in care delivery. When invoked, the balance metaphor deflected attention from emotional, social, and moral impacts of clinical care on patients and their families. Emergent findings highlight the value of analysing reflexivity-oriented dialogues using discourse analysis methods.

Evaluation of the Reliability of the Challenge when used to Measure Advanced Motor Skills of Children with Cerebral Palsy.

AIMS: The Challenge was designed as an extension to the GMFM-66 to assess advanced motor skills of children with cerebral palsy (CP) who walk/run independently. This study evaluated the Challenge's inter-rater and test-retest reliability. METHODS: Thirty children with CP (GMFCS level I [n = 24] and II [n = 6]) completed the Challenge, with re-testing one to two weeks later. Seven physiotherapist assessors passed the Challenge criterion test pre-administration. A single assessor administered and scored test and retest sessions (test-retest reliability). A second assessor independently scored one of these sessions (inter-rater reliability). RESULTS: Inter-rater reliability was excellent (ICC = 0.97, 95%CI 0.94-0.99, CoV < 10%), with no bias (Bland-Altman plot). Test-retest ICC was excellent (ICC = 0.94, 95% CI 0.88-0.97. CoV < 10%, and Minimum Detectable Change (MDC90) was 4.47 points. Many participants indicated practising at home pre-retest session. CONCLUSIONS: There was strong rating consistency between assessors. While test-retest ICC estimates were also high, Challenge scores were higher at retest. The MDC90 was still in a range (>4.5 points) that seems clinically viable for change detection. Test-retest reliability could be reassessed with children instructed not to practice between assessments to determine the extent to which between-session practice influenced scores.

Development of Child and Family-Centered Engagement Guidelines for Clinical Administration of the Challenge to Measure Advanced Gross Motor Skills: A Qualitative Study.

AIMS: This article describes a qualitative study aimed at producing child-centered guidelines for the administration of a measure of children's advanced gross motor skills, the Challenge. The purpose of the guidelines is to promote collaborative interpretation and application of results. METHODS: The study was conducted in three Canadian cities and included 31 children with cerebral palsy (GMFCS Level I or II) ages 8 to 18 and one parent/caregiver per child (N = 62 participants). Following Challenge administration, each child and one of their caregivers took part in separate qualitative interviews. Analyses were oriented to exploring understandings of the purposes of testing, impressions of the child's performance, and perceptions of how results might inform activity choices and interventions. RESULTS: Three themes were generated: investments in doing well; I know my child/myself; and caregivers' interpretations of child's performance. Themes were then integrated with principles of child and family-centered care to develop The Challenge Engagement Guidelines directed at reducing test anxiety and enhancing shared decision making. CONCLUSIONS: The Guidelines are the first of their kind to integrate child and family-centered principles into the administration protocol of a motor measure. Although developed for the Challenge, the principles have applicability to other rehabilitation measures.

Assembling activity/setting participation with disabled young people.

Rehabilitation research investigating activity participation has been largely conducted in a realist tradition that under-theorises the relationship between persons, technologies, and socio-material places. In this Canadian study we used a post-critical approach to explore activity/setting participation with 19 young people aged 14 to 23 years with complex communication and/or mobility impairments. Methods included integrated photo-elicitation, interviews, and participant observations of community-based activities. We present our results using the conceptual lens of assemblages to surface how different combinations of bodies, social meanings, and technologies enabled or constrained particular activities. Assemblages were analysed in terms of how they organised what was possible and practical for participants and their families in different contexts. The results illuminate how young people negotiated activity needs and desires in particular 'spacings' each with its own material, temporal, and social constraints and affordances. The focus on assemblages provides a dynamic analysis of how dis/abilities are enacted in and across geotemporal spaces, and avoids a reductive focus on evaluating the accessibility of static environmental features. In doing so the study reveals possible 'lines of flight' for healthcare, rehabilitation, and social care practices.

Transitioning to adulthood with a progressive condition: best practice assumptions and individual experiences of young men with Duchenne muscular dystrophy.

PURPOSE: Youth with progressive conditions are living longer, and there is increased health care focus on assisting them with "transitioning" to adult services and adult life. The purpose of this investigation was to examine key discourses and normative assumptions underpinning transitions best practices and how they are reflected in the experiences of young men with Duchenne muscular dystrophy (DMD). METHODS: Using a critical perspective, we qualitatively analyzed influential transitions best practice documents to identify their underpinning discursive assumptions. We compared these to the analysis of qualitative interviews and diary data from a study of 11 young men with DMD. RESULTS: Transitions best practices are underpinned by discourses of developmental progression. They reproduce notions that associate successful transitions with becoming as independent as possible, approximating normal life trajectories, and planning for future adulthood. The accounts of youth with DMD both reflected and resisted these future-oriented discourses in creative ways that maintained positive personal identities. CONCLUSIONS: Normal developmental progression towards typical adult roles constitutes the generally accepted aims of transitions practices. Such aims may not be appropriate for all youth with disabilities. We suggest that alternative understandings of the life course and approaches to care need to be considered alongside dominant practices. Implications for Rehabilitation Children and youth with progressive conditions, such as DMD, are living longer and there is increased interest in designing programs that will assist them with "transitioning" to adulthood. Transitions best practices reflect dominant social values and assumptions about what constitutes a successful adulthood, embedded in goals such as independent living, self-management and obtaining work. Rehabilitation professionals should be aware of both positive (e.g. feelings of achievement) and negative (e.g. anxiety about the future) consequences of transitions practices that emphasize normal social developmental trajectories and milestones. Discussions with youth should offer multiple possibilities for living a good life in the present and provide support to address negative feelings and the progressive effects of DMD.

An integrated methods study of the experiences of youth with severe disabilities in leisure activity settings: the importance of belonging, fun, and control and choice.

PURPOSE: The aim was to examine the leisure activity setting experiences of two groups of youth with severe disabilities - those with complex continuing care (CCC) needs and those who have little functional speech and communicate using augmentative and alternative communication (AAC). METHOD: Twelve youth took part in a mixed methods study, in which their experiences were ascertained using qualitative methods (observations, photo elicitation and interviews) and the measure of Self-Reported Experiences of Activity Settings (SEAS). Data integration occurred using a "following a thread" technique and case-by-case analysis. RESULTS: The analysis revealed several highly valued aspects of leisure activity setting experiences for youth, including engagement with others, enjoying the moment, and control and choice in selection and participation in activity settings. CONCLUSIONS: The findings provide preliminary insights into the nature of optimal activity settings for youth with severe disabilities, and the mediators of these experiences. Compared to other youth, the data illustrate both the commonalities of experiences and differences in the ways in which these experiences are attained. Implications for research concern the utility of mixed methods approaches in understanding the complex nature of participation experiences. Implications for clinical practice concern the importance of not assuming the nature of youths' experiences.

Becoming men: Gender, disability, and transitioning to adulthood.

Children and youth with progressive conditions are living longer, and there is increased interest in designing programs that will assist them with "transitioning" to adulthood. Almost none of the transitions research to date, however, has attended to the experiences of disabled boys in "becoming men," nor has there been critical conceptual work problematizing notions of "normal" adulthood or theorizing the complex, diverse, and gendered experiences of transitioning. In this Canadian study, we investigated the intersectionality of gender, disability, and emerging adulthood with 15 young men with Duchenne muscular dystrophy. Participants created audio diaries and photographs that were explored in in-depth interviews. Using a Bourdieusian lens and Arthur Frank's notion of the narrative habitus, we examined how participants re/negotiated identities in everyday practices. Our analysis suggested that disability, masculinities, and generational (life stage) identities intersected through "narratives of nondifference," wherein participants worked to establish identities as typical "guys." Within limited fields of school and work, participants distanced themselves from the label of "disabled" and discussed their successes and challenges in terms of normative developmental trajectories. We suggest that the pursuit of "normal" is reproduced and reinforced in health and social programs and closes off other narratives and possibilities.

A multi-method approach to studying activity setting participation: integrating standardized questionnaires, qualitative methods and physiological measures.

PURPOSE: This study investigated the feasibility of integrating three diverse methods for studying disabled youth's experiences of activity settings: standardized questionnaires, qualitative methods and physiological measures. The article describes a case study of data collection in a real-life setting to outline the opportunities, challenges and lessons learned for future research. METHODS: The methods included (a) two newly developed quantitative measures of qualities of home and community activity settings (MEQAS) and youth experiences (SEAS); (b) youth-friendly qualitative methods, including photo-elicitation, observations, electronic interviews and face-to-face interviews; and (c) an innovative system to collect physiological data (the HEART system) that provided insight into the experiences of youth beyond other methods. RESULTS: The study demonstrated that these diverse methods measures can be practically combined to study activity setting participation experiences. The measures provided different types of data that informed one other and allowed a rich interpretation and deep understanding of the participant's experiences. We discuss practical lessons concerning the joint use of the three methods, lessons specific to each method, and lessons concerning next steps for integrating the data. CONCLUSIONS: Combined methodologies hold great promise for investigations of the participation experiences of disabled youth in future research.

Disability and dignity-enabling home environments.

In Canada where long-term care is primarily oriented to elderly persons and affordable accessible housing is limited, younger disabled adults may be living in circumstances that do not meet their health needs and contribute to their social exclusion. The purpose of this study was to undertake an ethical analysis of what constitute an 'adequate' home environment for adults with significant mobility disabilities. An integrated design was used that combined qualitative interviews with normative ethical analysis in an iterative process. Twenty interviews with 19 participants were conducted in Ontario, Canada with two groups: younger adults (ages 18-55) with mobility disabilities and 'decision-makers' who consisted of policy makers, program administrators and discharge planners. Data were analyzed using a critical disability ethics approach and processes of reflective equilibrium. Drawing on Nora Jacobson's (Jacobson, 2009) taxonomy of dignity and pluralistic approaches to social justice, the concept of 'social dignity' provides a lens for exploring the adequacy of home environments for disabled people. Analyses suggested seven threshold conditions necessary for a dignity-enabling home: the ability to form and sustain meaningful relationships; access to community and civic life; access to control and flexibility of daily activities; access to opportunities for self-expression and identity affirmation; access to respectful relationships with attendants; access to opportunities to participate in school, work or leisure; access to physical, psychological and ontological security. The results have implications for housing, health and social care policies, and political reform. Social dignity provides a normative ethical grounding for assessing the adequacy of home environments. The threshold elements outline specific dignity-enabling conditions that are open to further specification or elaboration in different contexts.

Using drama to improve person-centred dementia care.

AIMS AND OBJECTIVES: We implemented a 12-week drama-based educational intervention to introduce to dementia practitioners person-centred care that emphasizes the notion of embodied selfhood (defined as non-verbal self-expression). BACKGROUND: Person-centred dementia care guidelines emphasize the assessment of individual needs, and where appropriate, the use of non-pharmacological interventions before resorting to pharmacological management. However, dementia care is not consistent with these guidelines suggesting conceptual limitations and reliance on passive knowledge translation strategies. DESIGN AND METHODS: Focus groups and semi-structured interviews with practitioners (n = 24) in two nursing homes in central Canada were undertaken to assess the effectiveness of the drama-based components of the intervention. RESULTS: Our findings suggest that drama was effective as an educational modality, and helped implement the person-centred approach into practice. Significant practice outcomes included: new awareness that residents' body movements and dispositions can convey meaning; seeking biographical information from families; increased time efficiency; and supporting residents' independence. CONCLUSIONS: Our findings make an important contribution to person-centred dementia care by broadening the notion of personhood, and by facilitating implementation using drama. IMPLICATIONS FOR PRACTICE: As an enhancement of person-centred care, the support of embodied selfhood may significantly improve residents' quality of life, quality of care, and practitioners' care-giving experience.