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

Abstract

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.

Room for improvement in non-pharmacological systemic sclerosis care? — a cross-sectional online survey of 650 patients

“…However, poor communication and coordination, can create additional barriers to care access. SSc patients and their families are feel exposed to great barriers in access to and the quality of specialized and coordinated healthcare [ 16 , 33 , 34 ]. They describe themselves as being “passed around”, have difficulties to find reliable information about their illness and treatment, and experience follow-up appointments logistically, physically and emotionally demanding.…”

A rare disease patient-reported outcome measure: revision and validation of the German version of the Systemic Sclerosis Quality of Life Questionnaire (SScQoL) using the Rasch model

“…The study has several limitations. First, the validation was only planned when the MANOSS project was already established and did not allow for confirmation of the self-reported diagnosis, multiple measurement points and multinational validation [ 18 ]. For the cognitive interviews, only six Swiss and German patients were included.…”

“…In Phase 1, the SScQoL was refined in accordance with the International Society for Pharmacoeconomics and Outcomes Research (ISPOR) guideline [ 16 , 17 ]. Phase 2, drew on data from the MANagement Of Systemic Sclerosis (MANOSS) cross-sectional study carried out in Switzerland [ 18 , 19 ]. The MANOSS project aims to fill existing gaps in SSc care by developing an eHealth-enhanced rare disease chronic care model for SSc patients in Switzerland.…”

“…In phase 2, the validation study, German-speaking SSc patients of the MANOSS cross-sectional survey (March–August 2019) completed the revised (polytomous) SScQoL [ 18 ]. Participants completed either a paper format version and returned it by mail or completed the revised SScQoL in a web-based format.…”

“…They were asked to assess the face validity of the revised SScQoL. For phase 2, patients were recruited according to the MANOSS protocol [ 18 ]. Patients were recruited from four Swiss University hospitals, one regional (cantonal) hospital, rheumatology outpatient clinics, and the Swiss SSc patient association.…”