Patient engagement in research: lessons learned from CAPTURE ALS, a longitudinal observational ALS study.

OBJECTIVE: There are compelling ethical and practical reasons for patient engagement in research (PEIR), however, evidence for best practices remains limited. We investigated PEIR as implemented in CAPTURE ALS, a longitudinal observational study, from study inception through the first 2.5 years of operations. METHODS: Data were drawn from three engagement initiatives: a community-led letter-writing campaign; consultation with patient and caregiver focus groups; and a study-embedded 'participant partner advisory council' (PPAC). Data were derived retrospectively from study documentation. We used the International Association of Public Participation (IAP2) participation spectrum as a framework for investigation. RESULTS: 2401 letters from community members to the Canadian government affirmed study objectives and advocated for funding. Feedback from focus group consultation influenced study design and supported the study's data-sharing plan. PPAC collaboration shaped all aspects of the study. Contributions included: co-creation of governance documents, input on study protocols and public-facing communication, and development of engagement webinars for study participants and feedback surveys. Effective communication practices fostered collaboration and helped avoid tokenistic engagement. CAPTURE ALS encompassed all IAP2 participation levels. CONCLUSIONS: CAPTURE ALS was shaped by meaningful engagement initiatives over the course of the study. Lessons learned included: begin early and embed PEIR within research; build relationships and foster mutual learning; be flexible, open to adaptation, and seek diversity. Primary challenges included funding for early implementation, time needed to maintain relationships, and attrition due to disease progression. All IAP2 participation levels contributed to meaningful PEIR. 'Empowerment' was demonstrated through advocacy.

Asynchronous online focus groups for research with people living with amyotrophic lateral sclerosis and family caregivers: usefulness, acceptability and lessons learned.

BACKGROUND: People with amyotrophic lateral sclerosis (ALS) face disability- and travel-related barriers to research participation. We investigate the usefulness and acceptability of asynchronous, online focus groups (AOFGs) for research involving people affected by ALS (patients and family caregivers) and outline lessons learned. METHODS: The ALS Talk Project, consisting of seven AOFGs and 100 participants affected by ALS, provided context for this investigation. Hosted on the secure itracks Board™ platform, participants interacted in a threaded web forum structure. Moderators posted weekly discussion questions and facilitated discussion. Data pertaining to methodology, participant interaction and experience, and moderator technique were analyzed using itracks and NVivo 12 analytics (quantitative) and conventional content analysis and the constant-comparative approach (qualitative). RESULTS: There was active engagement within groups, with post lengths averaging 111.48 words and a complex network of branching interactions between participants. One third of participant responses included individual reflections without further interaction. Participants affirmed their co-group members, offered practical advice, and discussed shared and differing perspectives. Moderators responded to all posts, indicating presence and probing answers. AOFGs facilitated qualitative and quantitative data-gathering and flexible response to unanticipated events. Although total participation fell below 50% after 10-12 weeks, participants valued interacting with peers in an inclusive, confidential forum. Participants used a variety of personal devices, browsers, and operating systems when interacting on the online platform. CONCLUSIONS: This methodological examination of AOFGs for patient-centred investigations involving people affected by ALS demonstrates their usefulness and acceptability, and advances knowledge of online research methodologies. Lessons learned include: early identification of research goals and participant needs is critical to selecting an AOFG platform; although duration longer than 10-12 weeks may be burdensome in this population, participants were positive about AOFGs; AOFGs offer real world flexibility enabling response to research challenges and opportunities; and, AOGFs can effectively foster safe spaces for sharing personal perspectives and discussing sensitive topics. With moderators playing an important role in fostering engagement, AOFGs facilitated rich data gathering and promoted reciprocity by fostering the exchange of ideas and interaction between peers. Findings may have implications for research involving other neurologically impaired and/or medically vulnerable populations.

What do people affected by amyotrophic lateral sclerosis want from health communications? Evidence from the ALS Talk Project.

INTRODUCTION/AIMS: Health communication is central to effective, supportive amyotrophic lateral sclerosis (ALS) clinical care. Guidance for ALS communication is limited, focuses on diagnosis disclosure, and frequently relies on expert consensus and/or reviews. Patient-based evidence is needed to guide ALS health communication. We investigated how the experiences of ALS patients and family caregivers can inform effective communication practices from diagnosis to end-of-life. METHODS: Data were drawn from the ALS Talk Project, an asynchronous, online focus group study. Seven focus groups and five interviews (105 participants) were conducted. Data were qualitatively analyzed using directed content analysis and the constant-comparative approach. RESULTS: We found four primary themes: communication content, communication circumstances, information sufficiency, and communication manner. Data indicate participants relied on clinicians for medical information but also wanted practical information; health communication should attend to the circumstances within which conversations occur; information must be sufficient for individual needs, without overwhelming; and an empathetic, direct, and honest manner facilitated trust. Participants identified communication challenges and strategies to improve communication across major themes, including stepwise approaches and conversations tailored to individuals and their heterogeneous disease experiences. DISCUSSION: Healthcare professionals should discuss patient/caregiver communication preferences early in the therapeutic relationship, co-develop a communication agreement, and update the agreement in response to changing needs and disease progression. This will foster regular discussion of information needs and promote timely discussions of challenging topics, including advance care, while giving patients and families a sense of control. Findings may have implications for other neuromuscular disease and/or seriously ill populations.

Amyotrophic lateral sclerosis (ALS) health charities are central to ALS care: perspectives of Canadians affected by ALS.

OBJECTIVE: Expert consensus guidelines recommend referral of people with amyotrophic lateral sclerosis (ALS) to ALS health charities for support. Limited research indicates that patients and families value interaction with these volunteer sector organizations. We investigated how patient support from Canadian ALS health charities (ALS Societies) is experienced by those affected by ALS, and whether patient-centered outcomes validate recommendations for referral. METHODS: Data were drawn from the ALS Talk Project, an asynchronous online focus group study. Patients and family caregivers were recruited from regions across Canada. Seven groups met online for 14 weeks between January and July 2020. Seventy-eight participants made statements about ALS Societies. Data were qualitatively analyzed using directed content analysis and the constant-comparative approach. RESULTS: Participants viewed ALS Societies as integral to the healthcare system. The Societies acted as patient navigators and filled perceived care gaps, including psychological support. They provided critical practical assistance, particularly equipment loans and peer support groups; comprehensive disease-related and real-life information; and personal connections. They facilitated knowledge of research, emerging therapies, and research opportunities. Delayed referral to ALS Society supports and information resources was a concern for some participants. CONCLUSIONS: ALS Societies provide patients with critical practical, informational, and emotional support and play an overarching role as patient/research navigators. Patient-centred outcomes support patient referral to ALS Societies. Communication about the services provided should be a standard component of clinical care, with choice of access left to individuals. Clinical conversations should be supplemented with information resources developed by these voluntary sector organizations.

Covid-19 threat and coping: application of protection motivation theory to the pandemic experiences of people affected by amyotrophic lateral sclerosis.

BACKGROUND: People with amyotrophic lateral sclerosis (ALS) are at high risk for severe outcomes from Covid-19 infection. Researchers exploring ALS and Covid-19 have focused primarily on system response and adaptation. Using Protection Motivation Theory, we investigated how people with ALS and family caregivers appraised and responded to Covid-19 threat, the 'costs' associated with pandemic response, and how health professionals and systems can better support people affected by ALS who are facing public health emergencies. METHODS: Data were drawn from the 'ALS Talk Project,' an asynchronous, moderated focus group study. Participants were recruited from regions across Canada. Seven groups met online over 14 weeks between January and July 2020. Fifty-three participants contributed to Covid-19 discussions. Data were qualitatively analyzed using directed content analysis and the constant-comparative approach. RESULTS: Participants learned about the Covid-19 pandemic from the media. They rapidly assessed their vulnerability and responded to Covid-19 threat by following recommendations from health authorities, information monitoring, and preparing for worst-case scenarios. Adopting protective behaviors had substantial response costs, including adaptations for medical care and home support workers, threatened access to advance care, and increased caregiver burden. Participants expressed need for ALS-specific, pandemic information from trusted health professionals and/or ALS health charities. Telemedicine introduced both conveniences and costs. Prior experience with ALS provided tools for coping with Covid-19. Threat and coping appraisal was a dynamic process involving ongoing vigilance and adaptation. Findings draw attention to the lack of emergency preparedness among participants and within health systems. CONCLUSIONS: Clinicians should engage ALS patients and families in ongoing discussions about pandemic coping, strategies to mitigate response costs, care pathways in the event of Covid-19 infection, and changing information about Covid-19 variants and vaccines. Healthcare systems should incorporate flexible approaches for medical care, leveraging the benefits of telemedicine and facilitating in-person interaction as needed and where possible. Research is needed to identify strategies to mitigate response costs and to further explore the interaction between prior experience and coping. Further study is also needed to determine how communication about emergency preparedness might be effectively incorporated into clinical care for those with ALS and other medically vulnerable populations.

Lung volume recruitment improves volitional airway clearance in amyotrophic lateral sclerosis.

INTRODUCTION/AIMS: In this study we evaluated the effects of lung volume recruitment treatment (LVR), a low-tech, low-cost, manual "breath-stacking" technique used to help people cough with enough force to clear their airways, thereby reducing the risk of aspiration and choking, on five volitional airway clearance and protection behaviors used by people living with amyotrophic lateral sclerosis (PwALS). METHODS: Using a repeated-measures cross-over design, 29 PwALS performed five volitional airway clearance and protection behaviors in LVR treatment and in no-treatment, control conditions. Peak cough flow (PCF) was used to measure maximum expiratory rate during forced expiration, throat clearing, hawking, post-swallow coughing, and the supraglottic swallowing maneuver. Comparisons were made as a function of condition (treatment or control) and three time-points (pretreatment, and 15 and 30 minutes posttreatment). RESULTS: LVR treatment had a significant positive effect on maximum expiratory rates during all tested airway clearance and protection behaviors. Increased PCF values lasted for up to 30 minutes post-LVR for all tested behaviors in the treatment condition. DISCUSSION: We found that LVR treatment could increase control over airway clearance in PwALS, as well as provide improved airway protection for up to 30 minutes, the duration of a typical meal. This study has implications for patient care. These include offering patients control over some of the most feared symptoms of ALS, particularly choking during activities of daily living, and enhanced ALS respiratory care in low-resource settings. Findings may have implications for other neurodegenerative disorders in which dysphagia occurs with retained sensory function.

Communication About End of Life for Patients Living With Amyotrophic Lateral Sclerosis: A Scoping Review of the Empirical Evidence.

Background: Communication about end of life, including advance care planning, life-sustaining therapies, palliative care, and end-of-life options, is critical for the clinical management of amyotrophic lateral sclerosis patients. The empirical evidence base for this communication has not been systematically examined. Objective: To support evidence-based communication guidance by (1) analyzing the scope and nature of research on health communication about end of life for amyotrophic lateral sclerosis; and (2) summarizing resultant recommendations. Methods: A scoping review of empirical literature was conducted following recommended practices. Fifteen health-related and three legal databases were searched; 296 articles were screened for inclusion/exclusion criteria; and quantitative data extraction and analysis was conducted on 211 articles with qualitative analysis on a subset of 110 articles that focused primarily on health communication. Analyses summarized article characteristics, themes, and recommendations. Results: Analysis indicated a multidisciplinary but limited evidence base. Most reviewed articles addressed end-of-life communication as a peripheral focus of investigation. Generic communication skills are important; however, substantive and sufficient disease-related information, including symptom management and assistive devices, is critical to discussions about end of life. Few articles discussed communication about specific end-of-life options. Communication recommendations in analyzed articles draw attention to communication processes, style and content but lack the systematized guidance needed for clinical practice. Conclusions: This review of primary research articles highlights the limited evidence-base and consequent need for systematic, empirical investigation to inform effective communication about end of life for those with amyotrophic lateral sclerosis. This will provide a foundation for actionable, evidence-based communication guidelines about end of life. Implications for research, policy, and practice are discussed.

Transitions in Amyotrophic Lateral Sclerosis: Patient and Caregiver Experiences.

BACKGROUND: Amyotrophic lateral sclerosis (ALS) presents many transitions for persons/people with ALS (PwALS) and their caregivers. Transitions are passages from one life phase, condition, or status to another. We used qualitative methods to understand how PwALS and caregivers experience transitions throughout their ALS journey. METHODS: PwALS and their caregivers were recruited from a multidisciplinary ALS clinic in Edmonton, Canada. We recruited patients at the stage of ALS that home mechanical ventilation, a feeding tube, and/or assistive communication technology had been offered. Semi-structured interviews were audio-recorded, transcribed, and analyzed using qualitative thematic analysis. RESULTS: We interviewed 14 PwALS and 15 caregivers and identified 5 recurring themes. The importance of community was identified by many PwALS and caregivers who expressed feelings of loneliness and isolation. Most caregivers were spouses and couples navigated a change in their relationship roles as one spouse transitioned to becoming a caregiver while the other transitioned to dependency. The caregiver spouses reported a sense of "total responsibility" that encompassed continual vigilance for the PwALS's well-being, managing their household and finances. PwALS and caregivers reported transitioning to reliance on life-sustaining medical devices; early adoption and information on these devices increased their quality of life. Participants also wanted more and earlier information on advanced care planning. PwALS and caregivers identified adapting to new forms of communication as a necessity. CONCLUSION: ALS presents many transitions for PwALS and caregivers. Understanding these transitions is important for ALS healthcare professionals who seek to implement best care practices.

Medical Information Commons to Support Learning Healthcare Systems: Examples From Canada.

We explore how principles predicting the success of a medical information commons (MIC) advantaged or disadvantaged three MIC initiatives in three Canadian provinces. Our MIC case examples demonstrate that practices and policies to promote access to and use of health information can help improve individual healthcare and inform a learning health system. MICs were constrained by heterogenous health information protection laws across jurisdictions and risk-averse institutional cultures. A networked approach to MICs would unlock even more potential for national and international data collaborations to improve health and healthcare.

Human Excretion of Polybrominated Diphenyl Ether Flame Retardants: Blood, Urine, and Sweat Study.

Commonly used as flame retardants, polybrominated diphenyl ethers (PBDEs) are routinely detected in the environment, animals, and humans. Although these persistent organic pollutants are increasingly recognized as having serious health implications, particularly for children, this is the first study, to our knowledge, to investigate an intervention for human elimination of bioaccumulated PBDEs. Objectives. To determine the efficacy of blood, urine, and perspiration as PBDE biomonitoring mediums; assess excretion of five common PBDE congeners (28, 47, 99, 100, and 153) in urine and perspiration; and explore the potential of induced sweating for decreasing bioaccumulated PBDEs. Results. PBDE congeners were not found in urine samples; findings focus on blood and perspiration. 80% of participants tested positive in one or more body fluids for PBDE 28, 100% for PBDE 47, 95% for PBDE 99, and 90% for PBDE 100 and PBDE 153. Induced perspiration facilitated excretion of the five congeners, with different rates of excretion for different congeners. Conclusion. Blood testing provides only a partial understanding of human PBDE bioaccumulation; testing of both blood and perspiration provides a better understanding. This study provides important baseline evidence for regular induced perspiration as a potential means for therapeutic PBDE elimination. Fetotoxic and reproductive effects of PBDE exposure highlight the importance of further detoxification research.

Partnering with Indigenous student co-researchers: improving research processes and outcomes.

OBJECTIVE: To examine the contribution of student co-researchers to a community-based participatory Photovoice investigation of Indigenous children's food-related lived experience. We examine co-researchers' contributions to the research process, their role in knowledge co-generation and dissemination, and factors that fostered research partnership with the teenage co-researchers. METHODS: High school students attending a First Nation community school in Canada were trained as research partners. They contributed to aspects of research design, conducted interviews with grades 3 and 4 Photovoice participants, and participated in data analysis and the development of a culturally relevant photobook. The study was initiated by the community's research committee. It is informed by critical consciousness theory and the positive youth development framework. RESULTS: Student co-researchers incorporated culturally appropriate strategies as they interviewed participants. Co-researchers adopted conversational approaches, built rapport by articulating personal and cultural connections, and engaged in mentoring and health promotion as they interviewed participants. They made critical contributions to dissemination by developing photobook content that promoted the importance of traditional foods and the vital role of family and community in healthy eating practices. Relationships and "dialogic" space were important to building partnership with and promoting capacity development among youth co-researchers. CONCLUSIONS: Partnership between university researchers and Indigenous student co-researchers holds great promise for health promotion in communities. Co-researchers developed research and leadership skills, gained understanding of health challenges facing their community, and initiated health and cultural promotion through the project's Photobook. This investigation supports the powerful potential of student co-researchers to meaningfully contribute to research processes and to build knowledge that is relevant and credible both within and outside of their communities. Findings have implications for youth, communities and researchers.

Social Positioning Theory as a lens for exploring health information seeking and decision making.

In this article I use Social Positioning Theory to explore the experiences of women as they interact with and make sense of evolving health information mediated by formal and informal sources. I investigate how women position themselves within their accounts of information seeking, and the influence of positioning on interactions with health professionals (HPs). Interviewed women gathered and valued information from a range of sources, and were likely to position themselves as autonomous, rather than collaborative or dependent. Faced with evolving health information, women felt responsible not only for information seeking, but also for making sense of gathered and encountered information. Participants did, however, value information provided by HPs and were likely to view decision making as collaborative when HPs fostered information exchange, appeared to appreciate different types of knowledge and cognitive authority, and supported women in their quests for information. Implications for shared decision making are discussed.

Exploring the continuum: medical information to effective clinical practice. Paper I: the translation of knowledge into clinical practice.

This paper investigates the translation of medical information into clinical practice and the role of thoughtful dissent by exploring the influence of sociological factors on change, the impact of evidence-based medicine (EBM), and the role of industry. Changing practice related to hormone therapy for menopausal and post-menopausal women provides context for this discussion. Medical change involves diffusion of ideas to potential users and ongoing reconciliation of new information with old ideas; this process is influenced by sociological factors including values and experiences, interpersonal relationships and local context. While EBM has alerted doctors to the importance of high quality research and theoretically provides a tool for translating research into practice, there are important problems with its application: (1) it has resulted in a reductionist approach to research and illness; (2) there is a considerable gap between research findings and the complex environment of clinical practice; and (3) EBM has been appropriated by experts, thus corporately developed 'standard-of-care' documents have become instruments of external regulation, and EBM has ceased to be a tool in the hand of individual clinicians. In addition, industry impacts the translation of knowledge by significantly influencing academia, researchers, medical publications, consensus conferences, and practising doctors. While questioning doctrinaire practices or directives is a daunting prospect for individual clinicians, the translation of knowledge into practice and evolution of medical thought is dependent on the progressive role of thoughtful dissent.

Exploring the continuum: medical information to effective clinical practice. Paper II. Towards aetiology-centred clinical practice.

Contemporary clinical practice increasingly functions within a disease management paradigm aimed at finding and implementing therapeutic interventions that demonstrate efficacy in clinical trials. Disease prevention, elucidation of illness aetiology and proactive health promotion have taken a back seat. Current clinical care often includes a 'fast-food' type of medical encounter, which frequently neglects disease causality. The medical community is presently challenged by unique administrative and professional adversities as well as undue commercial influence; these factors contribute to a lethargic response to escalating rates of chronic illness and to mainstream medicine's relative inattention to emerging research about disease aetiology. Individual medical practitioners and the medical establishment must strategically seek to advance patient health and maintain professional relevancy by a renewed emphasis on the following principles: patient-based clinical care, aetiology-centred medicine, and proactive health promotion.

Implications of cyberspace communication: a role for physicians.

Through the presentation of three clinical case reports and subsequent discussion, it is demonstrated that physicians must begin to familiarize themselves with the health-related implications of online communication, and must proactively address Internet use as it relates to health and well-being. Included case presentations highlight the following: the established association between those seeking sexual partners through the Internet and an increased risk for sexually transmitted disease; the implications of cyber-communication for young people and concerns related to unsafe online behaviors including sharing identifying information with strangers; the potential use of strategically constructed virtual identities to facilitate sexual exploitation; the impact of accelerated intimacy and disinhibition evident in online communication; and the invasive nature of Internet sexual harassment or bullying. Although it is recognized that most online activities do not negatively affect health, doctors must be prepared to ask patients about Internet use and become involved in educating children, teenagers, and parents about safe online relationships to promote optimal physical, mental, and social health.

Managing the sexually transmitted disease pandemic: a time for reevaluation.

The serious implications of the sexually transmitted disease (STD) pandemic that currently challenges educators, medical practitioners and governments suggest that prevention strategies, which primarily focus on barrier protection and the management of infection, must be reevaluated and that initiatives focusing on primary prevention of behaviors predisposing individuals to STD risk must be adopted. Human immunodeficiency virus/acquired immunodeficiency syndrome, human papillomavirus, genital herpes, and Chlamydia are used to illustrate the pervasive presence of STDs and their serious consequences for individuals and national infrastructures. Long-term sequelae are discussed, including the emerging link between various sexually transmitted infections and cancer, and the psychosexual and psychosocial factors which impact infected individuals. Although risk reduction and treatment of existing infection is critical, the promotion of optimal life-long health can be achieved most effectively through delayed sexual debut, partner reduction, and the avoidance of risky sexual behaviors.

Human exposure assessment and relief from neuropsychiatric symptoms: case study of a hairdresser.

Human exposure assessment and the results of implementing 'precautionary avoidance' suggested a relationship between a hairdresser's neuropsychiatric symptoms and occupational exposure to potentially hazardous chemicals. A variety of investigations in response to patient complaints of depression, emotional instability and various physical symptoms revealed no objective abnormality; the CH2OPD2 mnemonic (community, home, hobbies, occupation, personal habits, diet and drugs) recommended by the Ontario College of Family Physicians was used as a first-line screening tool to assess potential environmental exposure to toxins. After occupational leave of absence, the patient reported cessation of symptoms. Environmental causes for familiar medical problems are frequently undiagnosed; it is recommended that, where appropriate, a screening tool for evaluation of environmental exposure to toxics be incorporated into primary care assessment and management of patients.