Background During the COVID-19 pandemic people affected by rare diseases (RD) or caregiver of affected children have faced additional challenges. The pandemic has affected physical and mental health, social life and has led to financial consequences. Our objectives were to identify the impact of COVID-19 (1) on health care and (2) on daily life and participation of patients with RDs or caregivers from the perspective of representatives of patient organizations. Moreover, we explored their perspective on experiences of pandemic stress and resources during the pandemic. Results We conducted 18 semi-structured interviews with representatives of patient organizations (e.g. chairperson, members of the steering committee), who were asked about the experiences of their members. The interviews were transcribed verbatim and analyzed using the framework approach. We contextualized our findings on the basis of the International Classification of Functioning, Disability and Health (ICF) model and adapted it according to identified subthemes. Patients and caregivers were confronted with aspects of pandemic stress such as lack of information, access and information regarding vaccination and being a risk group for COVID-19 infection. Physical and mental functioning was reported to be negatively impacted. Lock downs and contact restrictions led, e.g., to increasing lack of nursing services or lack of necessary informal support. Participation e.g. in social life and work was reduced. Health care services including medical care and supportive care as well as additional therapies were disrupted and greater effort was necessary to organize care. According to participants, central resources were informal support networks, digitalization, patient organizations and individual characteristics. Conclusions Our study highlights the consequences of the COVID-19 pandemic on the situation of people affected by RDs and caregivers. Contextualization of the results into the biopsychosocial model reinforces the impact of the pandemic on health care as well as daily life and participation. Major challenges and difficulties were experienced during lockdowns and contact restrictions. Depending on the risk of an infection with COVID-19, certain patient groups were still isolated and reduced social contacts or still followed strict hygienic measures (e.g., wearing medical masks). Future pandemic control measures, e.g. on lockdowns and closing facilities, should consider the challenges of people with RDs and caregivers of affected children.
IntroductionPatients 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 analysisWe 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 disseminationThis 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 numberGerman Clinical Trials Registry (DRKS00020488).
Zusammenfassung Hintergrund Seltene Erkrankungen (SE) sind häufig durch komplexe Beschwerdebilder charakterisiert und erfordern in der Regel im Diagnose- und Versorgungsverlauf die Zusammenarbeit von spezialisierten Zentren und Primärversorgenden. Reibungslose Schnittstellen mit geringem Informationsverlust und Kooperation stellen daher eine essenzielle Grundlage in der Versorgung dar. Das Projekt „Evaluation von Schnittstellenmanagementkonzepten bei Seltenen Erkrankungen“ (ESE-Best) verfolgte mittels verschiedener Erhebungsinstrumente das Ziel, Empfehlungen für die Gestaltung und Umsetzung von Schnittstellen in der Versorgung von Menschen mit Seltenen Erkrankungen zu entwickeln. Methoden Es wurden mittels quantitativer und qualitativer Befragungen die Perspektiven der Zentren für Seltene Erkrankungen (ZSE), der Primärversorgung und der Betroffenen (Patient:innen, Eltern) erfragt sowie 2 Expert:innen-Workshops durchgeführt. Ergebnisse Es wurden insgesamt 28 Empfehlungen in den folgenden 5 Bereichen formuliert: 1) Vernetzung zwischen Primärversorgung und Zentren für Seltene Erkrankungen (ZSE), 2) Schnittstellen innerhalb der ZSE, 3) Bekanntheit von Seltenen Erkrankungen, ZSE-Strukturen und Zuständigkeiten, 4) Schnittstellen zwischen ZSE und Patient:innen sowie 5) weiterführende Empfehlungen. Diskussion Die Empfehlungen sollen zukünftig zu einem funktionierenden Schnittstellenmanagement bei der Versorgung von Menschen mit Seltenen Erkrankungen beitragen. Da die Erfahrungswerte von Primärversorgenden, ZSE und Betroffenen in die Entwicklung der Empfehlungen eingeflossen sind, können die externe Validität und damit die Umsetzbarkeit im Alltag angenommen werden. Es ist zu bedenken, dass zeitliche und personelle Ressourcen sowie organisationale Strukturen die Schnittstellenarbeit im Einzelfall beeinflussen können. Die Empfehlungen können an örtliche Gegebenheiten adaptiert werden.
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