Learning from a 7 year long Public Patient Involvement (PPI) journey with the rare disease patient organisation 22q Ireland

Introduction: This research reflects on multi-stakeholder perspectives on the outcomes, enablers, barriers and lessons learnt from a 7 year Public Patient Involvement (PPI) partnership with a rare disease patient charity. Context: In 2014, families of children with the rare disorder, 22q11.2 deletion syndrome (22q) reported frustration and exhaustion as they sought care from a range of different healthcare providers who were unaware of their children's condition or needs (Alugo,T et al, 2017). Additionally, there was a complete lack of research literature on the lived experience of people living with 22q (Kerin, L. et al, 2017). The patient charity 22q Ireland contracted the author, a participatory health researcher, who co-designed successive research projects to hear the lived experiences and needs of young people living with 22q and their parents (2). The findings subsequently informed PPI partnerships with the charity, clinicians and healthcare service managers to develop clinical services in response to the identified needs. This research reflects on the enablers, barriers, outcomes and lessons learnt from the 7 year PPI partnership, from the perspective of the different stakeholder groups including young adults living with 22q, their parents, hospital clinicians, healthcare service managers and researchers. Public Patient Involvement approach: This research was co-designed with a PPI panel, involving members of the patient charity board as well as young adults living with 22q at different stages of the research cycle, including the funding application, research design, research ethics materials,, research write up and research dissemination. What did we do? A rapid literature review on Public Patient Involvement (PPI) in rare disease research informed initial qualitative research design, subsequently refined with the PPI panel. Interviews with 23 multi-stakeholder respondents explored their experience of the PPI in research journey across the time frame of 7 years (2014 -2021). Data was coded using a theoretical analysis approach (Braun & Clarke 2021) and analysed using Maxqda software. Findings were shared with the PPI panel and collaborative dissemination opportunities were agreed. Impact: The impact of the PPI research projects included a range of personal, professional, clinical and population level benefits. Of particular note was the co-production of young expert by experience panel (22q YEEP), a clinical and research advocacy network (22q CARA), a specialist paediatric service inclusive of transition support (CHI at Crumlin), statutory funding for care coordination and ultimately national status as a pilot for complex care service provision in the national healthcare service in the Republic of Ireland. What is the learning for the international audience? Thematic analysis identified key enablers, critical barriers and recommendations to develop successful Public Patient Involvement in research and health service development. These findings are transferable to international contexts, as are the innovative clinical service developments. What are the next steps? Based on this innovative PPI initiative, work is underway with the Health Service Executive (HSE) to progress further service development, and to transfer learning to inform integrated care for populations living with complex care needs across the island of Ireland.