Hypermobile Online Pain managemEnt (HOPE) program for people with hypermobile Ehlers-Danlos syndrome or hypermobility spectrum disorder

<p dir="ltr">There are six chapters presented in a sequential flow that details the work taken to develop and test the final product of this thesis: the Hypermobile Online Pain managemEnt (HOPE) program for people with hypermobile Ehlers-Danlos Syndrome or hypermobility spectrum disorder. This is a thesis by publication and Macquarie University permits published manuscripts to be included. I started my MPhil part-time in the University of Sydney in 2021 and transferred to a full-time PhD at Macquarie University in 2022. This thesis was completed at Macquarie University. </p><p dir="ltr"><b>Chapter 1</b> starts off with an overview of symptomatic joint hypermobility, followed by deeper discussion of a group of syndromic joint hypermobility conditions known as Ehlers-Danlos Syndrome (EDS). The chapter narrows down the scope of this thesis to two subgroups of EDS, known as hypermobile Ehlers-Danlos Syndrome (hEDS) and Hypermobility Spectrum Disorder (HSD). Since pain is one of the most commonly reported symptom in hEDS and HSD, this chapter discusses the mechanisms of pain as well as how it interplays with other classical signs and symptoms common in hEDS and HSD. The chapter goes on to explore why pain self-management options are important and what is available for this population. Online interventions are increasingly being used, so the chapter ends of by presenting what online pain management interventions been investigated in hEDS and HSD and closes with the aims of this thesis. </p><p dir="ltr"><b>Chapter 2 </b>consists of a systematic review and meta-analysis of online pain management programs in chronic, widespread musculoskeletal conditions. This manuscript was prepared while I was at the University of Sydney and was submitted after I transferred to Macquarie University. In chapter two, it is presented as published in Pain Practice. </p><p dir="ltr"><b>Chapter 3</b> is a published development paper of the Hypermobile Online Pain managemEnt (HOPE) program that was created as part of this thesis. An international, modified Delphi was conducted in the development stage to gain input from stakeholders, and the HOPE program was created using their input. This manuscript is presented as published in Disability and Rehabilitation. </p><p dir="ltr"><b>Chapter 4</b> is a manuscript of the pilot randomised controlled trial of the HOPE program. This is a mixed method study and there are two manuscripts from this pilot trial. Chapter 4 reports the quantitative outcomes, with primary outcomes being feasibility, acceptability and appropriateness, and secondary outcomes being e`ectiveness of the intervention on clinical outcome measures. This manuscript is presented in the format submitted to the European Journal of Pain on 2 January 2025. </p><p dir="ltr"><b>Chapter 5</b> reports the qualitative feedback from a sample of participants that took part in the HOPE intervention. Semi-structured, 1:1 interviews were conducted to investigate the feasibility, acceptability and appropriateness of the HOPE program. This manuscript is presented as published in Health Expectations. </p><p dir="ltr"><b>Chapter 6</b> examines and discusses the key findings of the results produced from the studies included in this thesis. It will present a synthesis of the various findings and compare and discuss these results to other relevant studies, as well as discuss the clinical implications of this thesis. This chapter concludes with future direction and recommendations for the HOPE program and future research. </p><p dir="ltr">Each chapter in this thesis contains its own reference list and appendices. Additional appendices unrelated to individual chapters are included at the end of the thesis. PROSPERO registration of the systematic review in Chapter 2, ethical and scientific approval was obtained from the Macquarie University Human Research Ethics Committee for the pilot randomised controlled trial in Chapter 4 and Chapter 5 are provided in the appendices.</p>