Dataset Information

Interface management concepts in healthcare for rare diseases in Germany: a study protocol for a mixed-methods study to develop best practice recommendations.

ABSTRACT: Introduction: Patients and families affected by a rare disease are burdened in multiple ways. Functional interface management can unburden patients or relatives from the need to be solely accountable for the navigation through the healthcare system. This study aims at (1) providing an assessment of approaches and interface management concepts in the care of rare diseases, (2) evaluating selected existing approaches and concepts and (3) developing best practice recommendations for interface management.

Methods and analysis: We will conduct a mixed-methods study with three phases. In phase 1, we will develop a tool to assess existing concepts of interface management for rare diseases based on a literature search and an expert workshop. The tool will be applied in a telephone survey with representatives of centres or clinics of expertise for rare diseases (target: n=100) and cooperating practitioners (target: n=60). Based on the results of phase 1, we will select four to six centres of expertise with interface management concepts, which will be evaluated extensively in phase 2. For the evaluation, we will conduct semistructured interviews with practitioners cooperating with centres or clinics for rare diseases (target: n=50), a paper-based survey including patients or parents/legal guardians (target: n=300) from the selected centres or clinics, and semistructured interviews with patients or parents/legal guardians (target: n=50). The final phase of the study will be an integration of results from phases 1 and 2 to develop best practice recommendations for interface management in healthcare of rare diseases. In a concluding expert workshop, recommendations will be presented and finalised.

Ethics and dissemination: This study was approved by the Local Psychological Ethics Committee of the Center for Psychosocial Medicine of the University Medical Center Hamburg-Eppendorf (LPEK-0062). The findings of our study will be presented on national and international conferences and published in scientific, peer-reviewed journals. To assure that centres for rare diseases get access to the study results, centres are invited to send a representative to a final expert workshop in phase 3. Moreover, an executive summary will be provided and sent to relevant stakeholders.

Trial registration number: German Clinical Trials Registry (DRKS00020488).

SUBMITTER: Inhestern L

PROVIDER: S-EPMC7705519 | biostudies-literature | 2020 Nov

REPOSITORIES: biostudies-literature

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Interface management concepts in healthcare for rare diseases in Germany: a study protocol for a mixed-methods study to develop best practice recommendations.

Inhestern Laura L Zybarth David D Otto Ramona R Brandt Maja M Härter Martin M Bergelt Corinna C

BMJ open 20201130 11

<h4>Introduction</h4>Patients and families affected by a rare disease are burdened in multiple ways. Functional interface management can unburden patients or relatives from the need to be solely accountable for the navigation through the healthcare system. This study aims at (1) providing an assessment of approaches and interface management concepts in the care of rare diseases, (2) evaluating selected existing approaches and concepts and (3) developing best practice recommendations for interfac ...[more]

PMID: 33257484

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Project description:BackgroundIndividuals with Friedreich ataxia (FRDA) can find it difficult to access specialized clinical care. To facilitate best practice in delivering healthcare for FRDA, clinical management guidelines (CMGs) were developed in 2014. However, the lack of high-certainty evidence and the inadequacy of accepted metrics to measure health status continues to present challenges in FRDA and other rare diseases. To overcome these challenges, the Grading of Recommendations Assessment and Evaluation (GRADE) framework for rare diseases developed by the RARE-Bestpractices Working Group was adopted to update the clinical guidelines for FRDA. This approach incorporates additional strategies to the GRADE framework to support the strength of recommendations, such as review of literature in similar conditions, the systematic collection of expert opinion and patient perceptions, and use of natural history data.MethodsA panel representing international clinical experts, stakeholders and consumer groups provided oversight to guideline development within the GRADE framework. Invited expert authors generated the Patient, Intervention, Comparison, Outcome (PICO) questions to guide the literature search (2014 to June 2020). Evidence profiles in tandem with feedback from individuals living with FRDA, natural history registry data and expert clinical observations contributed to the final recommendations. Authors also developed best practice statements for clinical care points that were considered self-evident or were not amenable to the GRADE process.ResultsSeventy clinical experts contributed to fifteen topic-specific chapters with clinical recommendations and/or best practice statements. New topics since 2014 include emergency medicine, digital and assistive technologies and a stand-alone section on mental health. Evidence was evaluated according to GRADE criteria and 130 new recommendations and 95 best practice statements were generated.Discussion and conclusionEvidence-based CMGs are required to ensure the best clinical care for people with FRDA. Adopting the GRADE rare-disease framework enabled the development of higher quality CMGs for FRDA and allows individual topics to be updated as new evidence emerges. While the primary goal of these guidelines is better outcomes for people living with FRDA, the process of developing the guidelines may also help inform the development of clinical guidelines in other rare diseases.

Project description:OBJECTIVES:To investigate the practices of healthcare professionals in relation to best practice recommendations for the multidisciplinary management of people with diabetes and periodontitis, focusing on two clinical behaviours: informing patients about the links between diabetes and periodontitis, and suggesting patients with poorly controlled diabetes go for a dental check-up. DESIGN:Cross-sectional design utilising online questionnaires to assess self-reported performance and constructs from Social Cognitive Theory (SCT) and Normalisation Process Theory. SETTING:Primary care medical practices (n=37) in North East, North Cumbria and South West of England Clinical Research Networks. PARTICIPANTS:96 general practitioners (GPs), 48 nurses and 21 healthcare assistants (HCAs). RESULTS:Participants reported little to no informing patients about the links between diabetes and periodontitis or suggesting that they go for a dental check-up. Regarding future intent, both GPs (7.60±3.38) and nurses (7.94±3.69) scored significantly higher than HCAs (4.29±5.07) for SCT proximal goals (intention) in relation to informing patients about the links (p<0.01); and nurses (8.56±3.12) scored significantly higher than HCAs (5.14±5.04) for suggesting patients go for a dental check-up (p<0.001). All professional groups agreed on the potential value of both behaviours, and nurses scored significantly higher than GPs for legitimation (conforms to perception of job role) in relation to informing (nurses 4.16±0.71; GPs 3.77±0.76) and suggesting (nurses 4.13±0.66; GPs 3.75±0.83) (both p<0.01). The covariate background information (OR=2.81; p=0.03) was statistically significant for informing patients about the links. CONCLUSIONS:Despite evidence-informed best practice recommendations, healthcare professionals currently report low levels of informing patients with diabetes about the links between diabetes and periodontitis and suggesting patients go for a dental check-up. However, healthcare professionals, particularly nurses, value these behaviours and consider them appropriate to their role. While knowledge of the evidence is important, future guidelines should consider different strategies to enable implementation of the delivery of healthcare interventions.

Dataset Information

Interface management concepts in healthcare for rare diseases in Germany: a study protocol for a mixed-methods study to develop best practice recommendations.

Publications

Interface management concepts in healthcare for rare diseases in Germany: a study protocol for a mixed-methods study to develop best practice recommendations.

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