Developing a rare disease chronic care model: Management of systemic sclerosis (MANOSS) study protocol

• Published in: Journal of advanced nursing Vol. 75; no. 12; pp. 3774 - 3791
• Main Authors: Kocher, Agnes, Simon, Michael, Dwyer, Andrew A., Villiger, Peter M., Künzler‐Heule, Patrizia, Geest, Sabina, Berben, Lut, Nicca, Dunja
• Format: Journal Article
• Language: English
• Published: England Wiley Subscription Services, Inc 01-12-2019
• Subjects: access to health care; chronic care model; chronic illness management; Chronic illnesses; Contextual analysis; eHealth; Ethics; Experts; Feasibility; Health care access; Health disparities; implementation science; Medical personnel; mixed methods; Mixed methods research; Nursing; Patient communication; Polls & surveys; rare disease; Rare diseases; Relatives; Research ethics; Scleroderma; self‐management; Stakeholders; Systemic sclerosis; Technology; access to health care; chronic care model; rare disease; systemic sclerosis; chronic illness management; mixed methods; nursing; self-management; eHealth; implementation science
• ISSN: 0309-2402; 1365-2648
• DOI: 10.1111/jan.14185

Aim
The aim of the management of systemic sclerosis (MANOSS) study described in this protocol is to develop a chronic care model, based on a contextual analysis and stakeholder involvement, for patients living with the rare disease systemic sclerosis (SSc) in Switzerland.
Design
Applying an implementation science approach, this study starts with an explanatory sequential mixed method study for contextual analysis, followed by broad stakeholder involvement for model development and a Delphi study to reach consensus.
Methods
First, a quantitative cross‐sectional survey with patients and healthcare professionals (HPs) will be conducted to identify current practice patterns of chronic illness management and technology readiness. Second, qualitative interviews with patients, family members and HPs will be performed to gain a deeper understanding of care needs identified in the quantitative survey. Third, a model of care will be co‐created with input from patients, HPs and other experts. The eHealth enhanced Chronic Care Model will serve as a guiding framework. The new model and corresponding outcome parameters will be refined using a Delphi‐study approach to reach consensus on a testable model of care for persons living with SSc. The protocol has received research ethics committee approval in September 2018 by the Swiss Ethics Committee.
Discussion
The MANOSS study's participatory approach is essential for contextual fit of the model for patients with SSc in this setting. Subsequent feasibility testing and implementation are planned to evaluate the model's value in relation to health disparities faced by this patient population.
Impact
Patients living with this rare disease lack access to coordinated, specialized care and self‐management support from qualified HPs. Reengineering of current care, with consideration for technological opportunities, is warranted to meet patients’ and families’ needs.
目的
本方案所述的系统性硬化症管理研究旨在基于语境分析和涉众,为瑞士患有罕见疾病系统性硬化症的患者建立一种慢性护理模式。
设计
本研究采用实践科学的方法,首先用解释性顺序混合法进行语境分析研究,然后再根据广泛的涉众建立模型,并进行德尔菲研究以达成共识。
方法
首先,将对患者和医疗保健专业人员进行定量横断面调查,以确定目前采用的慢性病管理和技术准备的实践模式。其次,将与患者及其家属和专业医疗人员进行定性访谈,以便更深入地了解定量调查中确定的护理需求。最后,将采取患者、专业医疗人员和其他专家的意见共同创建护理模式。电子健康增强型慢性病护理模式将作为指导框架。将使用德尔菲研究方法对新模式及其相应的结果参数进行完善,以就系统性硬化症患者的可测试护理模式达成共识。该方案已于2018年9月获得“瑞士伦理委员会”研究伦理委员会批准。
讨论
在本背景下,本研究采用的参与式方法对于系统性硬化症患者模型的语境匹配至关重要。随后,计划进行可行性测试及实施,以评估该模式对于该患者群体面临的健康差异的价值。
影响
患有这种罕见疾病的患者无法从合格的专业医疗人员处获得一致、专业的护理和自我管理支持。考虑到技术机会,有必要重新设计当前的护理,以满足患者及其家属的需求。