Modelling logical intervention pathways through a stakeholder authored concept map to design complex interventions aimed at improving participation for people with Sjögren’s syndrome

Background: Participation in daily activities is problematic for many people with primary Sjögren’s syndrome (pSS). A group concept map, co-developed with 232 patients, family and healthcare staff stakeholders (patients with pSS, family members and health professionals) contained 7 themed clusters of prioritized ideas. To utilize the map to develop future interventions, our aims were: 1) model logical clear pathways through the map; 2) identify specific facilitators and outcomes from the ideas within the map which relate to agreed intervention targets within the priority logical pathway. Methods: To utilize these maps to develop future interventions, we explored their content further during three steering group interpretation sessions and through a validation process with clinicians. Steering group members (n = 6) individually considered the map and agreement was reached on the logical pathways following three interpretation meetings. Specific priority intervention targets within one pathway were selected from within the concept map through discussion and reaching consensus. The priority intervention targets and all individual highly rated ideas from within the concept map which explicitly related to these targets were presented to experienced psychologists, physiotherapists and occupational therapists (n = 10) for validation. The clinicians were asked to provide specific examples of how these related ideas could be applied and evaluated within a clinical setting. Results: The themed clusters in order of priority included; patient empowerment, symptoms, wellbeing, access & coordination of healthcare, knowledge & support, friends/family and public awareness. Four logical pathways were drawn through the themed clusters within map and are listed in order of priority: 1) To improve patient empowerment the priority symptoms of fatigue, dryness, pain and sleep need to be addressed in order to improve wellbeing. 2) Priority symptoms can be addressed through appropriate access & coordination of healthcare which could positively impact on patient empowerment. 3) Knowledge and support could be delivered through an appropriate healthcare system and may impact on public awareness and support. 4) Friends and family are more likely to be supportive if there was better public awareness of pSS. Their support could facilitate patient empowerment. Pathway 1 included the highest rated themed clusters. The steering group agreed to focus on the intervention targets of fatigue, pain and sleep as these were often not being adequately addressed in current clinics. The clinician validation process provided specific intervention examples for each priority idea relating to the targets of fatigue, pain and sleep and suggested outcome measure tools. Conclusion: A priority logical intervention pathway to improve participation in Sjögren’s syndrome has been developed through a rigorous process of stakeholder engagement and a consensus activity. A clinician validation process has produced specific ingredients and outcomes to form components of a future complex intervention to improve participation in pSS patients