Measuring Health-Related Quality of Life in Amyotrophic Lateral Sclerosis: A Systematic Review and Conceptual Framework.

BACKGROUND AND OBJECTIVES: The assessment of health-related quality of life (HRQoL) in patients with amyotrophic lateral sclerosis (ALS) is heterogeneous and inconsistent. The objectives of this study were (1) to develop a comprehensive conceptual framework of HRQoL in ALS and (2) map the content of existing patient-reported outcome measures (PROMs) used in ALS to this novel framework. METHODS: Our model of HRQoL in ALS (Health-related Quality of life in Amyotrophic Lateral Sclerosis, QuALS) was developed from a systematic literature review and consultative input from key stakeholders (patients, carers, and health care professionals). Five electronic databases were searched in April 2022. Primary studies of any design that assessed HRQoL in ALS by using a multi-item PROM and/or qualitative methods were identified. Using an a priori framework, HRQoL themes were extracted and iteratively modified from the content of each PROM and qualitative study quotations identified in the literature. The conceptual framework was ratified by stakeholders with lived experience and clinical experts. The QuALS framework was used to map the content of identified PROMs and qualitative studies based on thematic coverage. RESULTS: QuALS covers 3 high-level domains of HRQoL (physical, psychological, and social functioning) and consists of 7 themes (Activities; Physical Health; Autonomy; Cognition; Feelings and Emotions; Self-identity; Relationships), characterized by 42 subthemes. Of 8,220 studies identified, 274 were included in the review that informed QuALS. In these studies, 111 PROMs were used to assess at least 1 aspect of HRQoL, and 11 studies used qualitative methods. Of the 3 high-level domains, physical functioning was the most commonly assessed, particularly within ALS-specific PROMs where almost one-quarter of PROMs exclusively assessed physical functioning. None of the PROMs or qualitative studies identified assessed all aspects of HRQoL in the QuALS framework. DISCUSSION: This study presents a new comprehensive conceptual framework of HRQoL in ALS (QuALS), informed by a robust systematic review of existing literature and stakeholder input, incorporating lived experience. QuALS provides a valuable resource for researchers and clinicians interested in taking a holistic approach to assessing and understanding the full impact of ALS on HRQoL and how this may be affected by treatments.

White Paper: Open Digital Health - accelerating transparent and scalable health promotion and treatment.

In this White Paper, we outline recommendations from the perspective of health psychology and behavioural science, addressing three research gaps: (1) What methods in the health psychology research toolkit can be best used for developing and evaluating digital health tools? (2) What are the most feasible strategies to reuse digital health tools across populations and settings? (3) What are the main advantages and challenges of sharing (openly publishing) data, code, intervention content and design features of digital health tools? We provide actionable suggestions for researchers joining the continuously growing Open Digital Health movement, poised to revolutionise health psychology research and practice in the coming years. This White Paper is positioned in the current context of the COVID-19 pandemic, exploring how digital health tools have rapidly gained popularity in 2020-2022, when world-wide health promotion and treatment efforts rapidly shifted from face-to-face to remote delivery. This statement is written by the Directors of the not-for-profit Open Digital Health initiative (n = 6), Experts attending the European Health Psychology Society Synergy Expert Meeting (n = 17), and the initiative consultant, following a two-day meeting (19-20th August 2021).

Telehealth in long-term neurological conditions: the potential, the challenges and the key recommendations.

Long-term neurological conditions (LTNCs) cause physical and psychological symptoms that have a significant impact on activities of daily living and quality of life. Multidisciplinary teams are effective at providing treatment for people with LTNCs; however, access to such services by people with disabilities can be difficult and as a result, good quality care is not universal. One potential solution is telehealth. This review describes the potential of telehealth to support people with LTNCs, the challenges of designing and implementing these systems, and the key recommendations for those involved in telehealth to facilitate connected services that can benefit patients, carers and healthcare professionals. These recommendations include understanding the problems posed by LTNCs and the needs of the end-user through a person-centred approach. We discuss how to work collaboratively and use shared learning, and consider how to effectively evaluate the intervention at every stage of the development process.

A Road Map for Remote Digital Health Technology for Motor Neuron Disease.

Despite recent and potent technological advances, the real-world implementation of remote digital health technology in the care and monitoring of patients with motor neuron disease has not yet been realized. Digital health technology may increase the accessibility to and personalization of care, whereas remote biosensors could optimize the collection of vital clinical parameters, irrespective of patients' ability to visit the clinic. To facilitate the wide-scale adoption of digital health care technology and to align current initiatives, we outline a road map that will identify clinically relevant digital parameters; mediate the development of benefit-to-burden criteria for innovative technology; and direct the validation, harmonization, and adoption of digital health care technology in real-world settings. We define two key end products of the road map: (1) a set of reliable digital parameters to capture data collected under free-living conditions that reflect patient-centric measures and facilitate clinical decision making and (2) an integrated, open-source system that provides personalized feedback to patients, health care providers, clinical researchers, and caregivers and is linked to a flexible and adaptable platform that integrates patient data in real time. Given the ever-changing care needs of patients and the relentless progression rate of motor neuron disease, the adoption of digital health care technology will significantly benefit the delivery of care and accelerate the development of effective treatments.

Reflexive thematic analysis exploring stakeholder experiences of virtual pulmonary rehabilitation (VIPAR).

INTRODUCTION: To enable greater availability of pulmonary rehabilitation (PR), video-conferencing technology was employed to link a community site to a standard outpatient PR service to deliver the programme closer to those with chronic respiratory disease. The service was called virtual pulmonary rehabilitation (VIPAR). To understand the experiences of those involved in this service, this study aimed to answer the question: How do the different stakeholders that interact with VIPAR experience the programme? METHODS: Focus groups were conducted with people with chronic lung disease attending either the community or outpatient PR site, in addition to the staff involved in the running of the PR groups. A total of five focus groups were conducted. Reflexive thematic analysis was used to analyse the transcripts. RESULTS: Participants who received VIPAR were positive regarding the programme and described the benefits of the service. Additionally, participants discussed the trade-off between convenience and relatedness. Staff described barriers to the service and solutions that could be employed to mitigate these, including greater training, organisation and understanding of risk. DISCUSSION: Using technology to increase the availability of PR is acceptable to the stakeholders involved, providing problems and organisational issues are resolved with training.

Using self-determination theory to predict self-management and HRQoL in moderate-to-severe COPD.

Objective: Chronic Obstructive Pulmonary Disease (COPD) is a long-term condition that detrimentally affects health-related quality of life (HRQoL), with self-management proposed as an effective treatment. Using self-determination theory (SDT), this research explored psychological need satisfaction, frustration, and behavioural regulation to explain indicators of self-management. Design and Main Outcome Measures: Cross-sectional, questionnaire-based methods in people on a pulmonary rehabilitation waiting-list. 72 participants completed SDT, HRQoL, and self-management knowledge questionnaires. Path analyses investigated the ability of SDT concepts to predict self-management knowledge and HRQoL. Results: Chi-square tests found no significant differences (χ2(13, N=72) = 16.7, p > 0.05) between the just - and over-identified models, and multiple measures suggested an acceptable fit to the data. Relatedness frustration positively predicted controlled regulation and autonomy and relatedness satisfaction positively predicted autonomous regulation. The associations between the other needs and the different regulation types were not statistically significant. Both regulation types strongly predicted HRQoL (35% variance explained) and self-management knowledge (22% variance explained). Conclusion: SDT concepts can predict more self-determined self-management regulation, self-management knowledge, and HRQoL and provide a framework for researchers and healthcare professionals to develop future health interventions for people with COPD. Greater research is needed to understand basic psychological need frustration in health contexts.

Using the Technology Acceptance Model to conceptualise experiences of the usability and acceptability of a self-management app (COPD.Pal®) for Chronic Obstructive Pulmonary Disease.

Chronic Obstructive Pulmonary Disease (COPD) is a long-term progressive inflammatory lung disease causing chronic breathlessness and many hospital admissions. It affects up to 1.2 million people in the UK. To help people with COPD self-manage their condition we developed, in partnership with healthcare users, a digital mobile phone app called COPD.Pal®. We report the first user feedback of COPD.Pal®, applying the Technology Acceptance Model (TAM) theoretical framework. 11 participants engaged with a click dummy version of COPD.Pal® before being asked questions relating to their experiences. A deductive, semantic, reflexive thematic analysis was conducted to analyse their individual and collective experiences. The study was registered at Clinical Trials.gov (NCT04142957). Two overarching themes resulted: Ease of Use and Perceived Usefulness. Within the former, participants discussed how they wanted flexibility and choice in how they engaged with the app; including how often they used it. Additionally, they discussed how the app layout should make it straightforward to use, whilst unanimously agreeing that COPD.Pal® provided this. Within Perceived Usefulness, participants discussed how they wanted the information they entered into the app to be useful, in addition to the app providing resources regarding COPD. Lastly, there was disagreement regarding preferences for further app development. We found that COPD.Pal® was usable and acceptable by people with COPD and TAM provided a useful theoretical framework for both structuring discussions with users and analysing their comments.

Safety, feasibility, and effectiveness of virtual pulmonary rehabilitation in the real world.

Purpose: To assess the feasibility, safety, and effectiveness of a VIrtual PulmonAry Rehabilitation (VIPAR) program in a real-world setting. Patients and methods: Twenty-one patients with stable chronic lung disease at a spoke site received (VIPAR) through live video conferencing with a hub where 24 patients were receiving 14 sessions of standard, outpatient, multi-disciplinary pulmonary rehabilitation (PR) in a hospital. We studied three such consecutive PR programs with 6-10 patients at each site. The hub had a senior physiotherapist, occupational therapist, exercise assistant, and guest lecturer, and the spoke usually had only an exercise instructor and nurse present. Uptake, adverse events (AEs), and early clinical changes were compared within and between groups. Travel distances were estimated using zip codes. Results: Mean attendance was 11.0 sessions in the hub and 10.5 sessions in the spoke (P=0.65). There was a single (mild) AE (hypoglycemia) in all three hub programs and no AEs in the three spoke programs. Mean COPD Assessment Test scores improved from 25.3 to 21.5 in the hub (P<0.001, 95% CI 2.43-5.17) and from 23.4 to 18.8 (P<0.001, 2.23-7.02) in the spoke group, with no difference between the groups (P=0.51, -3.35-1.70). Mean incremental shuttle walk test scores improved from 142 to 208 m (P<0.001, 75-199) in the hub and from 179 to 316 minutes in the spoke (P<0.001, 39.3-92.4), with a greater improvement in the spoke (P=0.025, 9.31-133). Twenty-one patients saved a total of 8,609.8 miles over the three programs by having the PR in their local spoke, rather than traveling to the usual nearest (hospital) hub. Conclusion: Video-conferencing, which links a local site to a standard PR program is feasible, safe, and demonstrates at least equivalent short-term clinical gains. Throughput can be increased, with less staffing ratios and significantly less traveling.

Quality of life in patients receiving telemedicine enhanced chronic heart failure disease management: A meta-analysis.

Background Previous reviews have investigated the effectiveness of telemedicine in the treatment of heart failure (HF). Dependent variables have included hospitalisations, mortality rates, disease knowledge and health costs. Few reviews, however, have examined the variable of health-related quality of life (QoL). Methods Randomised controlled trials comparing the delivery methods of any form of telemedicine with usual care for the provision of HF disease-management were identified via searches of all relevant databases and reference lists. Studies had to report a quantitative measure for mental, physical or overall QoL in order to be included. Results A total of 33 studies were identified. However, poor reporting of data resulted in the exclusion of seven, leaving 26 studies with 7066 participants. Three separate, random effects meta-analyses were conducted for mental, physical and overall QoL. Telemedicine was not significantly more effective than usual care on mental and physical QoL (standardised mean difference (SMD) 0.03, (95% confidence interval (CI) -0.05-0.12), p = 0.45 and SMD 0.24, (95% CI -0.08-0.56), p = 0.14, respectively). However, when compared to usual care, telemedicine was associated with a small significant increase in overall QoL (SMD 0.23, (95% CI 0.09-0.37), p = 0.001). Moderator analyses indicated that telemedicine delivered over a long-duration (≥52 weeks) and via telemonitoring was most beneficial. Conclusion Compared to usual care, telemedicine significantly increases overall QoL in patients receiving HF disease management. Statistically non-significant but nonetheless positive trends were also observed for physical QoL. These findings provide preliminary support for the use of telemedicine in the management of heart failure without jeopardising patient well-being.