Facilitating [corrected] More Efficient Negotiations for Innovative Therapies: A Value-Based Negotiation Framework.

OBJECTIVES: An increasing number of innovative therapies (e.g., gene- and cell-based treatments) have been developed in the past 20 years. Despite the significant clinical potential of these therapies, access delays may arise because of differing perspectives of manufacturers and payers regarding issues such as the value of the product, clinical and financial uncertainties, and sustainability.Managed entry agreements (MEAs) can enable access to treatments that would not be reimbursed by conventional methods because of such concerns. However, although MEA typologies exist, there is currently no structured process to come to agreements on MEAs, which can be difficult to decide upon and implement.To facilitate more structured MEA negotiations, we propose a conceptual "value-based negotiation framework" with corresponding application tools. METHODS: The framework was developed based on an iterative process of scientific literature review and expert input. RESULTS: The framework aims to (i) systematically identify and prioritize manufacturer and payer concerns about a new treatment, and (ii) select a mutually acceptable combination of MEA terms that can best address priority concerns, with the lowest possible implementation burden. CONCLUSIONS: The proposed framework will be tested in practice, and is a step toward supporting payers and manufacturers to engage in more structured, transparent negotiations to balance the needs of both sides, and enabling quicker, more transparent MEA negotiations and patient access to innovative products.

Identifying relevant factors for successful implementation into routine practice: expert interviews to inform a heart failure self-care intervention (ACHIEVE study).

BACKGROUND: Adherence to heart failure (HF) self-care behaviours has been found to be effective for alleviating illness symptoms, increasing quality of life and reducing hospital re-admissions and mortality. However, many patients fail to implement on-going self-care into their daily lives. It is therefore crucial to improve the behaviour of HF patients to increase self-care adherence. The aim of this study is to identify relevant factors to successfully implement a complex, theory-based HF self-care intervention into routine practice. METHODS: We conducted semi-structured interviews to obtain key stakeholders' opinions on previously developed behaviour change techniques for enhancing HF patients' self-care behaviours, in order to optimise implementation of these techniques in an intervention. The interview topic guide was developed based on the Normalisation Process Theory (NPT), a tool that takes into account the feasibility of implementation and the acceptability to stakeholders. Interviews were analysed using thematic analysis and supported by MAXQDA 2020, a software for qualitative research. RESULTS: Interview participants included 18 key stakeholders consisting of three crucial groups: clinical experts (n = 7), patients (n = 3) and high calibre policy makers/potential funders (n = 8). The interviews revealed numerous factors to consider for successful implementation of an intervention into routine practice. The findings are presented according to two major categories: (1) themes within the NPT framework and (2) themes beyond the NPT framework. Themes within the NPT component 'Coherence' include three sub-themes: 'understandability', 'value beyond existing interventions' and 'perceived benefits'. The NPT component 'Cognitive participation' revealed two sub-themes: 'time resources' and 'financial sustainability'. Finally, the NPT component 'Collective action' uncovered three sub-themes: 'need for training', 'compatibility with existing practice' and 'influence on roles'. A further two themes were identified beyond the NPT framework, namely: 'structural challenges' and (2) 'role of carers'. CONCLUSIONS: Factors identified previously by NPT were validated, but stakeholders further identified relevant aspects beyond NPT. Based on these findings, we suggest the existing NPT framework could be expanded to include a fifth component: questions considering specific environmental factors (contextual considerations). Sensitising researchers to these issues at an early stage when designing an intervention can facilitate its later success.

Examining the impact of different country processes for appraising rare disease treatments: a case study analysis.

BACKGROUND: Conventional appraisal and reimbursement processes are being challenged by the increasing number of rare disease treatments (RDTs) with a small evidence base and often a high price. Processes to appraise RDTs vary across countries; some use standard processes, others have separate processes or adapted processes that explicitly deal with rare disease specificities. The objective of this study was to examine the impacts of different appraisal processes for two RDTs. METHODS: A case study analysis was conducted using countries with different forms of appraisal processes for RDTs for which public health technology assessment (HTA) reports were available. Two contrasting RDTs were chosen according to the criteria: rare versus ultra-rare treatment, affecting child versus adult, life-threatening versus disabling. Information from public HTA reports for each country's RDT appraisal was extracted into templates, allowing a systematic comparison of the appraisals across countries and identification of the impact of the different processes in practice. RESULTS: Reports from Belgium, England, France, Germany, Italy, Netherlands, Norway, Scotland, Sweden, and the USA were selected for nusinersen (for spinal muscular atrophy) and voretigene neparvovec (for inherited retinal disorders). Countries with separate or adapted processes had more consistent approaches for managing RDT-related issues during appraisal, such as stakeholder involvement and criteria to address the specificities of RDTs, creating more transparency in decision-making. CONCLUSIONS: Findings suggest that separate or adapted approaches for RDT appraisal may facilitate more structured, consistent decision-making and better management of RDT specificities.

The Mediator Roles of Problematic Internet Use and Perceived Stress Between Health Behaviors and Work-Life Balance Among Internet Users in Germany and China: Web-Based Cross-Sectional Study.

BACKGROUND: Work-life balance is associated with health behaviors. In the face of digitalization, understanding this link requires a theory-based investigation of problematic internet use and perceived stress, which are so far unknown. OBJECTIVE: On the basis of the compensatory carry-over action model, this study aimed to determine whether problematic internet use and perceived stress mediate the relationship between health behaviors and work-life balance in two groups of internet users from different environments (residents in Germany and China). We also investigated whether the place of residence was a moderator. METHODS: An online questionnaire (N=877) was administered to residents from Germany (n=374) and China (n=503) in 3 languages (German, English, and Chinese). Moderated mediation analyses were run with health behaviors as the independent variable, work-life balance as the dependent variable, problematic internet use and perceived stress as the mediator variables, and place of residence as a potential moderator. RESULTS: On a mean level, individuals in Germany reported less problematic internet use and more health behaviors than individuals in China; however, they also had lower work-life balance and higher perceived stress. Results showed that health behaviors seem to be directly related to work-life balance in both groups. Among the residents of Germany, a partial mediation was revealed (ß=.13; P=.01), whereas among the residents of China, a full mediation was found (ß=.02; P=.61). The mediator role of perceived stress was compared with problematic internet use in all the serial models and the parallel model. Residence moderated the relationship between health behaviors and work-life balance: The interrelation between health behaviors and work-life balance was stronger in Germany (ß=.19; P<.001) than in China (ß=.11; P=.01) when controlling for other variables. CONCLUSIONS: The findings of this study are in line with the compensatory carry-over action model. To promote work-life balance, individuals should perform health behaviors to help overcome problematic internet use and perceived stress. Both problematic internet use and perceived stress mediated health behaviors and work-life balance partially in German study participants and fully in Chinese study participants.

Investigating acculturation orientations of patients with an immigration background and doctors in Canada: implications for medical advice adherence.

BACKGROUND: Increased immigration requires successful interaction of different cultures in various life domains, such as health. This study investigates acculturation orientation (AO) of immigrant patients and doctors native to the country, as a potential factor related to perceived medical advice adherence. DATA AND METHODS: N = 171 immigrant patients (M = 54.38 years, SD = 17.94, range = 23-96, 74.3 % female) and their N = 12 doctors (M = 38.88 years, SD = 13.42, range = 27-66, 83 % female) from a hospital in Montreal, Canada, participated in a paper-based survey to assess AOs, patients' perceived expectations of their doctor (regarding adopting the new culture or keeping their previous culture), doctors' actual expectations, perceived quality of care and perceived adherence. RESULTS: AO of patients significantly related to perceived adherence, via a path model involving perceptions of doctors' expectations and perceived quality of care. Integration was positively related to perceived adherence, while Marginalization was negatively related to it. Doctors' AOs were not significant. CONCLUSION: Patient perceptions seem to be a significant factor to be considered when striving to improve immigrant medical advice adherence.

Investigating patients with an immigration background in Canada: relationships between individual immigrant attitudes, the doctor-patient relationship, and health outcomes.

BACKGROUND: Increasing immigration in the world today leads to more intercultural interactions. This is a particularly crucial fact in doctor-patient relationships, which often become more complex and suboptimal within an intercultural context. Since acculturation is a particularly important factor in this process, and the doctor-patient relationship is a key component in patient health outcomes, this study investigates the interrelation of individual immigrant acculturation orientations with the quality of the doctor-immigrant patient relationship, the patients' perceived quality of care, and how this relates to immigrant health behaviours and quality of life of the patients. METHODS: 171 immigrant patients of various backgrounds participated in a paper and pencil questionnaire to assess the role of acculturation orientations (AO) on patients' perceived expectations of their doctor, perceived quality of care (PQOC), health behaviours and quality of life. Data were analyzed using ANOVA, regression and correlation procedures with SPSS statistical software. RESULTS: Significant correlations were found between all AOs and measures of the participant feeling connected to the host or home culture, thereby verifying the measure of AO. All four AOs were significantly interrelated directly with the patient's perception of what the doctor expects of him/her, and the patients' quality of life. Patients' perceived expectations of their doctors were significantly related to the patients' PQOC, and PQOC was associated with improved health behaviours (adherence to doctor recommendations, physical activity maintenance self-efficacy). CONCLUSIONS: AO may be an important factor in the doctor-immigrant patient relationship, via a complex process involving the patients' perceptions of doctors' expectations and perceived quality of care. This has important implications, since such an understanding can be used to create interventions for both doctors and immigrant patients to learn about their own AO, how it can relate to the quality of their relationship, and ultimately, the quality of care, health and quality of life of the patient.

Effects of individual immigrant attitudes and host culture attitudes on doctor-immigrant patient relationships and communication in Canada.

INTRODUCTION: In many countries doctors are seeing an increasing amount of immigrant patients. The communication and relationship between such groups often needs to be improved, with the crucial factor potentially being the basic attitudes (acculturation orientations) of the doctors and patients. This study therefore explores how acculturation orientations of Canadian doctors and immigrant patients impact the doctor-patient relationship. METHODS: N = 10 participants (five doctors, five patients) participated in acculturation orientation surveys, video recordings of a regular clinic visit, and semi structured interviews with each person. Acculturation orientations were calculated using the Euclidean distance method, video recordings were analyzed according to the Verona Coding System, and thematic analysis was used to analyze the interviews. Interviews were used to explain and interpret the behaviours observed in the video recordings. RESULTS: The combined acculturation orientations of each the doctor and immigrant patient played a role in the doctor-patient relationship, although different combinations than expected produced working relationships. Video recordings and interviews revealed that these particular immigrant patients were open to adapting to their new society, and that the doctors were generally accepting of the immigrants' previous culture. This produced a common level of understanding from which the relationship could work effectively. CONCLUSION: A good relationship and level of communication between doctors and immigrant patients may have its foundation in acculturation orientations, which may affect the quality of care, health behaviours and quality of life of the immigrant. The implications of these findings are more significant when considering effective interventions to improve the quality of doctor-patient relationships, which should have a solid foundational framework. Our research suggests that interventions based on understanding the influence of acculturation orientations could help create a basic level of understanding, and therefore improved interaction between doctors and immigrant patients.

Improving Interpretation of Evidence Relating to Quality of Life in Health Technology Assessments of Rare Disease Treatments.

Rare diseases are often severe, debilitating, life-limiting conditions, many of which occur in childhood. These complex conditions have a wide range of clinical manifestations that have a substantial impact on the lives of patients, carers and families and often produce heterogeneous clinical outcomes. Therefore, the evaluation of quality-of-life (QoL) impacts is important. In health technology assessment (HTA), patient-reported outcome measures (PROMs) and/or health state utility values (HSUVs) are used to determine QoL impacts of new treatments, but their use in rare diseases is challenging due to small and heterogeneous populations and limited disease knowledge. This paper describes challenges associated with the use of patient-reported outcomes (PROs)/HSUVs to evaluate QoL in HTA of rare disease treatments (RDTs) and identifies five recommendations to ensure appropriate interpretation of QoL impacts. These were derived from mixed methods research (literature reviews, appraisal document analyses, appraisal committee observations and interviews) examining the use of PROs/HSUVs in HTA of RDTs. They highlight that HTAs of RDTs must (1) understand the QoL impacts of the disease and of treatments; (2) critically assess PRO data, recognising the nuances in development and administration of PROMs/HSUVs, considering what is feasible and what matters most to the patient population; (3) recognise that lack of significant effect on a PRO does not imply no QoL benefit; (4) use different forms of evidence to understand QoL impacts, such as patient input; and (5) provide methodological guidance to capture QoL impacts on patients/carers.

Consideration of quality of life in the health technology assessments of rare disease treatments.

OBJECTIVES: Challenges with patient-reported outcome (PRO) evidence and health state utility values (HSUVs) in rare diseases exist due to small, heterogeneous populations, lack of disease knowledge and early onset. To better incorporate quality of life (QoL) into Health Technology Assessment, a clearer understanding of these challenges is needed. METHODS: NICE appraisals of non-oncology treatments with an EMA orphan designation (n = 24), and corresponding appraisals in the Netherlands, France, and Germany were included. Document analysis of appraisal reports investigated how PROs/HSUVs influenced decision-making and was representative of QoL impact of condition and treatment. RESULTS: PRO evidence was not included in 6/24 NICE appraisals. When included, it either failed to demonstrate change, capture domains important for patients, or was uncertain. In the other countries, little information was reported and evidence largely did not demonstrate change. In NICE appraisals, HSUVs were derived through the collection of EQ-5D data (7/24 cases), mapping (6/24), vignettes (5/24), and published literature or other techniques (6/24). The majority did not use data collected alongside clinical trials. Few measures demonstrated significant change due to lack of sensitivity or face validity, short-term data, or implausible health states. In 8/24 NICE appraisals, patient surveys or input during appraisal committee meetings supported the interpretation of uncertainty or provided evidence about QoL. CONCLUSIONS: This study sheds light on the nature of PRO evidence in rare diseases and associated challenges. Results emphasise the need for improved development and use of PRO/HSUVs. Other forms of evidence and expert input are crucial to support better appraisal of uncertain or missing evidence.

The Use of Patient-Reported Outcome Measures in Rare Diseases and Implications for Health Technology Assessment.

BACKGROUND: Patient-reported outcome measures (PROMs) are used in health technology assessment (HTA) to measure patient experiences with disease and treatment, allowing a deeper understanding of treatment impact beyond clinical endpoints. Developing and administering PROMs for rare diseases poses unique challenges because of small patient populations, disease heterogeneity, lack of natural history knowledge, and short-term studies. OBJECTIVE: This research aims to identify key factors to consider when using different types of PROMs in HTA for rare disease treatments (RDTs). METHODS: A scoping review of scientific and grey literature was conducted, with no date or publication type restrictions. Information on the advantages of and the challenges and potential solutions when using different types of PROMs for RDTs, including psychometric properties, was extracted and synthesized. RESULTS: Of 79 records from PubMed, 32 were included, plus 12 records from the grey literature. PROMs for rare diseases face potential data collection and psychometric challenges resulting from small patient populations and disease heterogeneity. Generic PROMs are comparable across diseases but not sensitive to disease specificities. Disease-specific instruments are sensitive but do not exist for many rare diseases and rarely provide the utility values required by some HTA bodies. Creating new PROMs is time and resource intensive. Potential solutions include pooling data (multi-site/international data collection), using computer-assisted technology, or using generic and disease-specific PROMs in a complementary way. CONCLUSIONS: PROMs are relevant in HTA for RDTs but pose a number of difficulties. A deeper understanding of the potential advantages of and the challenges and potential solutions for each can help manage these difficulties.

Cardiopulmonary capacity and psychological factors are related to return to work in orthopedic rehabilitation patients.

Health psychology sheds light on the process of returning to work after sick leave and subsequent medical rehabilitation. A 15-month longitudinal study with N = 201 orthopedic rehabilitation patients is reported. It examined whether and how both physiological health (e.g. body mass index and oxygen reabsorption) and psychological/social-cognitive factors (e.g. self-efficacy and social support) are related to returning to work. It was found that social-cognitive and physiological variables, such as oxygen consumption at anaerobic threshold, are important for returning to work. While physical activity was significantly correlated with social-cognitive variables, it was not significantly correlated directly with returning to work. Results suggest that self-efficacy and oxygen consumption at anaerobic threshold should be improved during medical rehabilitation.

What the guide does not tell you: reflections on and lessons learned from applying the COM-B behavior model for designing real life interventions.

Substantial inconsistency exists in the effectiveness of existing interventions to improve heart failure (HF) self-care, which can be partially explained by the fact that self-management interventions often lack theoretical models that underpin intervention development. The COM-B behavior model is a comprehensive theoretical framework that can be used to develop effective, theory-based interventions. The aim of this article is to highlight the challenges and practical solutions when applying the COM-B model to HF self-care, in order to provide useful support for researchers intending to use the model for designing behavior change interventions. "The Behaviour Change Wheel" handbook provides a step-by-step guide to understand and change behavior. When following the guide, some practical and methodological challenges were encountered. Lessons learnt to overcome these challenges are reported. Although the handbook is a comprehensive guide for designing behavior change interventions, a number of challenges arose. For example, the descriptions provided in the guide were not always sufficient to make solid judgments on how to categorize determinants; narrowing down intervention possibilities to a manageable number and prioritizing potential behavior change techniques over others involved a certain amount of subjectivity in an otherwise highly systematic and structured approach. For the encountered challenges, solutions are provided to illustrate how the model was applied practically to design theory-based behavior change interventions. This article provides a useful reference for researchers' use of the COM-B behavior model, as it outlines challenges that may occur and potential solutions to overcome them.

Determining Contextual Factors for a Heart Failure Self-Care Intervention: A Consensus Delphi Study (ACHIEVE).

There is a rising recognition of the crucial role self-care plays in managing heart failure (HF). Yet patients often have difficulties implementing ongoing self-care recommendations into their daily lives. There is also recognition of the importance of theory for successful intervention design, and understanding of key factors for implementation so interventions fit a given context. Local key stakeholders can provide valuable insights to help understand relevant context-specific factors for intervention implementation. This study sought to engage stakeholders to explore and determine relevant contextual factors needed to design and facilitate successful implementation of an HF self-care intervention in the German health care system. A ranking-type Delphi approach was used to establish consensus from stakeholders (i.e., clinicians, patients, policymakers/potential funders) regarding eight factors (content, interventionist, target group, location, mode of delivery, intensity, duration, and format) to adequately define the components and implementation strategy of the intervention. Seventeen participants were invited to participate in the first Delphi round. A response rate of 94% (16/17) was achieved and maintained for all three Delphi rounds. Stakeholder consensus determined that nurses specializing in HF are the most appropriate interventionists, target groups should include patients and carers, and the intervention should occur in an outpatient HF clinic, be a mixture of group and individual training sessions, and last for 30 minutes. Sessions should take place more frequently in the beginning and less often over time. Local stakeholders can help determine contextual factors that must be taken into account for successful delivery of an intervention. This enables the intervention to be developed and applied based on these factors, to make it suitable for the target context and to enhance participation to achieve the desired outcomes.

Applying the COM-B behaviour model to overcome barriers to heart failure self-care: A practical application of a conceptual framework for the development of complex interventions (ACHIEVE study).

BACKGROUND: Effective interventions to enhance adherence to self-care recommendations in patients with heart failure have immense potential to improve health and wellbeing. However, there is substantial inconsistency in the effectiveness of existing self-management interventions, partly because they lack theoretical models underpinning intervention development. AIM: To outline how the capability, opportunity and motivation behaviour model has been applied to guide the development of a theory-based intervention aiming to improve adherence to heart failure self-care recommendations. METHODS: The application of the capability, opportunity and motivation behaviour model involved three steps: (a) identification of barriers and facilitators to heart failure self-care from two comprehensive meta-studies; (b) identification of appropriate behaviour change techniques to improve heart failure self-care; and (c) involvement of experts to reduce and refine potential behaviour change techniques further. RESULTS: A total of 119 barriers and facilitators were identified. Fifty-six behaviour change techniques remained after applying three steps of the behaviour model for designing interventions. Expert involvement (n=39, of which 31 were patients (67% men; 45% New York Heart Association II)) further reduced and refined potential behaviour change techniques. Experts disliked some behaviour change techniques such as 'anticipated regret' and 'salience of consequences'. This process resulted in a final comprehensive list consisting of 28 barriers and 49 appropriate behaviour change techniques potentially enhancing self-care that was put forward for further use. CONCLUSION: The application of the capability, opportunity and motivation behaviour model facilitated identifying important factors influencing adherence to heart failure self-care recommendations. The model served as a comprehensive guide for the selection and design of interventions for improving heart failure self-care adherence. The capability, opportunity and motivation behaviour model enabled the connection of heart failure self-care barriers to particular behaviour change techniques to be used in practice.

Are supplemental appraisal/reimbursement processes needed for rare disease treatments? An international comparison of country approaches.

BACKGROUND: There is increasing recognition that conventional appraisal approaches may be unsuitable for assessing the value rare disease treatments (RDTs). This research examines what supplemental appraisal/reimbursement processes for RDTs are used internationally and how they can be characterised. A qualitative research design was used that included (1) documentation of country appraisal/reimbursement processes for RDTs via questionnaires, desk research and iterative interactions with country experts to produce country vignettes, and (2) a cross-country analysis of these processes to identify and characterise features in supplemental processes for RDTs, and compare them to countries without supplemental processes. RESULTS: Thirty-two of the 37 invited countries participated in this research. Forty-one percent (13/32) use supplemental processes for RDTs. Their level of integration within standard processes ranged from low to high, characterised by whether they are separate or partially separate from the standard process, adapted or accelerated standard processes, or standard processes that may be applied to RDTs. They are characterised by features implemented throughout the appraisal process. These features are mechanisms that allow application of different standards to assess the value of the medicine, support to the appraisal/decision-making process, overcome the issues of lack of cost-effectiveness, or exempt from part of/the full appraisal/reimbursement process. They increase the likelihood of reimbursement by adjusting and/or foregoing part of the assessment process, or accepting to pay more for the same added benefit as for common conditions. A large proportion of countries with standard processes include one or more of these features (formally or informally) or are discussing potential changes in their systems. CONCLUSIONS: Results suggest revealed preferences to treat RDTs differently than conventional medicines. Some of the challenges around uncertainty and high price remain, but supplemental process features can support decision-making that is more flexible and consistent. Many of these processes are new and countries continue to adjust as they gain experience.

Problematic Internet Use and Perceived Quality of Life: Findings from a Cross-Sectional Study Investigating Work-Time and Leisure-Time Internet Use.

Avoiding the potential negative impact brought by problematic internet use is becoming more important. To better understand public health and addiction, this study investigated to what extent work-time and leisure-time internet use relate to problematic internet use and perceived quality of life among college students and highly educated adults. An online cross-sectional survey with 446 individuals was assessed in Germany. Linear regression analyses were used to predict problematic internet use. Ordinal regression analyses were applied to predict perceived quality of life. Results showed that leisure-time internet use, but not work-time internet use, was positively associated with problematic internet use. Participants whose work-time internet use could be considered balanced (5-28 h/week in this study) indicated a higher perceived quality of life compared to individuals with little or large amount of internet use for work. The findings still emerged when taking negative feelings, perceived stress, smoking status and alcohol consumption into account. As both work-time and leisure-time internet use can be risk factors for mental health in terms of problematic internet use and perceived quality of life, well-controlled internet use rather than excessive use is recommended. This should be kept in mind when dealing with the Coronavirus pandemic and its aftermath.

'Mapping' Health State Utility Values from Non-preference-Based Measures: A Systematic Literature Review in Rare Diseases.

BACKGROUND: The use of patient-reported outcome measures (PROMs) to monitor the effects of disease and treatment on patient symptomatology and daily life is increasing in rare diseases (RDs) (i.e. those affecting less than one in 2000 people); however, these instruments seldom yield health state utility values (HSUVs) for cost-utility analyses. In such a context, 'mapping' allows HSUVs to be obtained by establishing a statistical relationship between a 'source' (e.g. a disease-specific PROM) and a 'target' preference-based measure [e.g. the EuroQol-5 Dimension (EQ-5D) tool]. OBJECTIVE: This study aimed to systematically review all published studies using 'mapping' to derive HSUVs from non-preference-based measures in RDs, and identify any critical issues related to the main features of RDs, which are characterised by small, heterogeneous, and geographically dispersed patient populations. METHODS: The following databases were searched during the first half of 2019 without time, study design, or language restrictions: MEDLINE (via PubMed), the School of Health and Related Research Health Utility Database (ScHARRHUD), and the Health Economics Research Centre (HERC) database of mapping studies (version 7.0). The keywords combined terms related to 'mapping' with Orphanet's list of RD indications (e.g. 'acromegaly') in addition to 'rare' and 'orphan'. 'Very rare' diseases (i.e. those with fewer than 1000 cases or families documented in the medical literature) were excluded from the searches. A predefined, pilot-tested extraction template (in Excel®) was used to collect structured information from the studies. RESULTS: Two groups of studies were identified in the review. The first group (n = 19) developed novel mapping algorithms in 13 different RDs. As a target measure, the majority used EQ-5D, and the others used the Short-Form Six-Dimension (SF-6D) and 15D; most studies adopted ordinary least squares (OLS) regression. The second group of studies (n = 9) applied previously published algorithms in non-RDs to comparable RDs, mainly in the field of cancer. The critical issues relating to 'mapping' in RDs included the availability of very few studies, the relatively high number of cancer studies, and the absence of research in paediatric RDs. Moreover, the reviewed studies recruited small samples, showed a limited overlap between RD-specific and generic PROMs, and highlighted the presence of cultural and linguistic factors influencing results in multi-country studies. Lastly, the application of existing algorithms developed in non-RDs tended to produce inaccuracies at the bottom of the EQ-5D scale, due to the greater severity of RDs. CONCLUSIONS: More research is encouraged to develop algorithms for a broader spectrum of RDs (including those affecting young children), improve mapping study quality, test the generalisability of algorithms developed in non-RDs (e.g. HIV) to rare variants or evolutions of the same condition (e.g. AIDS wasting syndrome), and verify the robustness of results when mapped HSUVs are used in cost-utility models.

Patient Perspectives to Inform a Health Literacy Educational Program: A Systematic Review and Thematic Synthesis of Qualitative Studies.

Patient-centred care is tailored to the needs of patients and is necessary for better health outcomes, especially for individuals with limited health literacy (LHL). However, its implementation remains challenging. The key to effectively address patient-centred care is to include perspectives of patients with LHL within the curricula of (future) healthcare providers (HCP). This systematic review aimed to explore and synthesize evidence on the needs, experiences and preferences of patients with LHL and to inform an existing educational framework. We searched three databases: PsychInfo, Medline and Cinahl, and extracted 798 articles. One-hundred and three articles met the inclusion criteria. After data extraction and thematic synthesis, key themes were identified. Patients with LHL and chronic diseases encounter multiple problems in the care process, which are often related to a lack of person-centeredness. Patient perspectives were categorized into four key themes: (1) Support system; (2) Patient self-management; (3) Capacities of HCPs; (4) Barriers in healthcare systems. "Cultural sensitivity" and "eHealth" were identified as recurring themes. A set of learning outcomes for (future) HCPs was developed based on our findings. The perspectives of patients with LHL provided valuable input for a comprehensive and person-centred educational framework that can enhance the relevance and quality of education for (future) HCPs, and contribute to better person-centred care for patients with LHL.

Testing a Photo Story Intervention in Paper Versus Electronic Tablet Format Compared to a Traditional Brochure Among Older Adults in Germany: Randomized Controlled Trial.

BACKGROUND: To increase effective communication in primary care consultations among older adults in Germany, the photo story is considered to be a useful tool based on Bandura's social cognitive theory. With information technology helping to increase effective communication, the use of tablets is gaining attention in health care settings, especially with older adults. However, the effectiveness of tablet technology and photo stories has rarely been tested. OBJECTIVE: The aim is to compare the effectiveness of a photo story intervention to a traditional brochure. Both were delivered either in paper or tablet format. METHODS: A trial was conducted with 126 older adults, aged 50 years and older, who were approached and recruited by researchers and administrative staff from senior day care, doctors in rehabilitation centers, and trainers in sports clubs in Germany. Open and face-to-face assessment methodologies were used. Participants were randomly assigned to one of four intervention conditions: traditional brochure in paper format (condition 1) and tablet format (condition 2), and photo story in paper format (condition 3) and tablet format (condition 4). Each participant received a questionnaire and either the traditional brochure or photo story in a paper or tablet version. To evaluate the effectiveness of each intervention, participants completed evaluation questionnaires before and after each intervention. The second part of the questionnaire measured different indicators of health literacy, communication skills, health measurements, and possible underlying mechanisms. RESULTS: Compared to the traditional brochure, participants considered the photo story easier to understand (t124=2.62, P=.01) and more informative (t124=-2.17, P=.03). Participants preferred the paper format because they found it less monotonous (t124=-3.05, P=.003), less boring (t124=-2.65, P=.009), and not too long (t124=-2.26, P=.03) compared to the tablet format. Among all conditions, the traditional brochure with a tablet (condition 2) was also perceived as more monotonous (mean 3.07, SD 1.08), boring (mean 2.77, SD 1.19), and too long to read (mean 2.50, SD 1.33) in comparison to the traditional brochure in paper format (condition 1). Moreover, the participants scored significantly higher on self-referencing on the traditional brochure in paper format (condition 1) than tablet format for both types of the brochure (conditions 2 and 4). CONCLUSIONS: Traditional brochures on a tablet seem to be the least effective communication option in primary care consultations among all conditions for older adults. The findings might be specific for the current generation of older adults in Germany and need to be replicated in other countries with larger sample sizes. Although information technology brings advantages, such as effective interventions in different fields and settings, it may also come with several disadvantages, such as technical requirements of the users and devices. These should be considered when integrating information technology into wider situations and populations. TRIAL REGISTRATION: ClinicalTrials.gov NCT02502292; https://clinicaltrials.gov/ct2/show/NCT02502292 (Archived by Webcite at http://www.webcitation.org/747jdJ8pU).