A Realist-informed Investigation of the Organisation and Delivery of Health and Social Care Services for People With Fibromyalgia Living in the UK
Conditions: Fibromyalgia, Fibromyalgia, Primary, Fibromyalgia, Secondary
Sponsor: University of Aberdeen

 
 
 
i 
 
 
FULL/LONG TITLE OF THE STUDY 
 
A realist-informed investigation of the organisation and delivery of health and social care services for 
people with fibromyalgia living in the UK. 
 
SHORT STUDY TITLE / ACRONYM 
Understanding health services delivery for fibromyalgia 
 
PROTOCOL VERSION NUMBER AND DATE 
V2  [15.11.2020] 
 
RESEARCH REFERENCE NUMBERS 
 
IRAS Number: 
275725 
 
SPONSORS Number: 
2-068-20 
 
FUNDERS Number: 
21958 
 
 
 

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SIGNATURE PAGE 
The undersigned confirm that the following protocol has been agreed and accepted and that the Chief 
Investigator agrees to conduct the study in compliance with the approved protocol and will adhere to 
the principles outlined in the Declaration of Helsinki, the Sponsor’s SOPs, and other regulatory 
requirement. 
I agree to ensure that the confidential information contained in this document will not be used for any 
other purpose other than the evaluation or conduct of the investigation without the prior written 
consent of the Sponsor 
I also confirm that I will make the findings of the study publicly available through publication or other 
dissemination tools without any unnecessary delay and that an honest accurate and transparent 
account of the study will be given; and that any discrepancies from the study as planned in this 
protocol will be explained. 
 
 
Chief Investigator: 
Signature:  
...................
...................................... 
 
Date: 
15/11/2020 
Name: (please print): 
PROFESSOR GARY MACFARLANE 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 

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LIST of CONTENTS 
GENERAL INFORMATION 
Page No. 
TITLE PAGE  
i 
RESEARCH REFERENCE NUMBERS 
 
i 
SIGNATURE PAGE 
ii 
LIST OF CONTENTS  
iii 
KEY STUDY CONTACTS 
iv 
STUDY SUMMARY 
vi 
FUNDING 
 
vii 
ROLE OF SPONSOR AND FUNDER 
vii 
ROLES & RESPONSIBILITIES OF STUDY STEERING GROUPS AND INDIVIDUALS   
vii 
STUDY FLOW CHART 
 
x 
SECTION 
1. BACKGROUND 
 
1 
2. RATIONALE 
 
2 
3. THEORETICAL FRAMEWORK 
2 
4. RESEARCH QUESTION/AIM(S)  
3 
5. STUDY DESIGN/METHODS 
4 
6. STUDY SETTING 
7 
7. SAMPLE AND RECRUITMENT 
7 
8. ETHICAL AND REGULATORY COMPLIANCE 
11 
9. STUDY CONDUCT RESPONSIBILITIES 
14 
10. DISSEMINATION POLICY 
14 
10. REFERENCES 
15 
11. APPENDICES 
18 
 
 

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KEY STUDY CONTACTS 
Chief Investigator 
Professor Gary Macfarlane 
Email: g.j.macfarlane@abdn.ac.uk 
Tel: 01224 437143 
Study Co-ordinator 
Marcus Beasley 
Email: m.beasley@abdn.ac.uk 
Tel: 01224 43 7067 
Sponsor 
Louise King 
Research Governance Manager 
University of Aberdeen and NHS Grampian 
Polwarth Rm: 1.126 
Forsterhill 
Aberdeen AB25 2ZD 
Email: researchgovernance@abdn.ac.uk 
Tel: 01224 437220 
Joint-sponsor(s)/co-sponsor(s)  
N/A 
Funder(s) 
Versus Arthritis 
Copeman House, St Mary’s Court 
St Mary’s Gate 
Chesterfield S41 7TD 
Key Protocol Contributors 
Debra Dulake (DD) 
Patient research partner 
 
Rosemary Hollick 
The University of Aberdeen 
rhollick@abdn.ac.uk  
01224 437275 
 
Louise Locock 
The University of Aberdeen 
Louise.locock@abdn.ac.uk 
01244 438143 
 
Gary Macfarlane 
The University of Aberdeen  
g.j.macfarlane@abdn.ac.uk  

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01224 437143 
 
Catherine Pope 
University of Oxford 
Catherine.pope@phc.ox.ac.uk  
01865 289335 
 
Simon Stones (SS) 
Patient research partner 
 
Nicky Wilson 
King’s College Hospital NHS Foundation Trust 
Nicky.wilson1@nhs.net  
020 3299 5809 
Committees 
N/A 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 

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STUDY SUMMARY 
Study Title 
A realist-informed investigation of the organisation and 
delivery of health and social care services for people with 
fibromyalgia living in the UK. 
Internal ref. no. (or short title) 
Understanding health services delivery for fibromyalgia. 
Study Design 
Qualitative case study design  
Study Participants 
Up to ten UK-based case studies will be conducted in total: at 
least one in England, Scotland and Wales. 
Participants will include healthcare professionals (for 
example, doctors, nurses, allied health professionals), social 
care practitioners (for example, social prescribers), service 
delivery managers, commissioners and other individuals 
involved in the organisation and delivery of health and social 
care services for people with fibromyalgia living in the UK.  
Planned Size of Sample (if applicable) Interviews - approximately 10 participants per case study 
(total 100 participants). 
Qualitative observations (in-person or on-line) involving 
similar numbers of people (total 100 participants). 
Online focus groups with approximately 6 to 8 participants per 
group (up to ten groups - total 60-80 participants)  
Follow up duration (if applicable) 
N/A 
Planned Study Period 
January 2021 – January 2022 
Research Question/Aim(s) 
 
This research study aims to: 
i) 
Explore and understand how health and social 
care for people with fibromyalgia living in the UK is 
organised and delivered.  
ii) 
Identify models of practice to inform codesign of 
new care pathways for people with fibromyalgia 
living in the UK. 
 
 
 
 
 
FUNDING AND SUPPORT IN KIND 

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FUNDER(S) 
(Names and contact details of ALL 
organisations providing funding and/or support 
in kind for this study) 
FINANCIAL AND NON FINANCIAL SUPPORT 
GIVEN 
Versus Arthritis (funding) 
£1,185,670.91 (PAtient-centred Care for 
Fibromyalgia: New pathway Design [PACFiND] 
research programme funding – 60 months) 
 
 
 
ROLE OF STUDY SPONSOR AND FUNDER 
 
i) 
The University of Aberdeen, as the study sponsor will be responsible for initiation, 
management and reporting of the study and will ensure appropriate indemnity before the study 
starts. 
 
ii) 
Versus Arthritis as the funder, will provide staff, travel and subsistence, and transcription costs 
associated with the conduct of this study. 
 
iii) 
The design, management, analysis and reporting of the study will be entirely independent of 
the funder. 
 
 
ROLES AND RESPONSIBILITIES OF STUDY MANAGEMENT COMMITTEES/GROUPS & 
INDIVIDUALS 
 
A study management team, including the Chief Investigator, deputy Chief Investigator, study manager, 
academic and clinical staff, will be responsible for the delivery of this study. The study management 
team will provide guidance and expert advice to the researchers on all aspects of this study through 
monthly teleconference meetings and email/telephone correspondence as necessary. The study 
management team will, in turn, be supported by a wider all-investigator team associated with the 
PACFiND programme, who will provide advice and guidance about issues arising. 
 
Patient & Public Involvement Group 
In addition to our two patient research partners (DD and SS) who are members of the study 
management team, a local patient and public reference group of 8 to 10 members has been convened 
in South London to advise on the development and co-ordination of this research. The group will 

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ensure that the research undertaken is accountable, transparent and relevant to patients and the 
public. Members of the group will provide a patient and public perspective on the research, read and 
review research project documents, highlight any issues of concern relating to ethics and governance 
of the study, and advise on and be involved in the dissemination of the study findings. Two further 
patient and public reference groups (one each in Scotland and Wales) will also be established to 
advise on and support the study.  
 
 
PROTOCOL CONTRIBUTORS 
 
Principal Investigators:  
 
Dr Nicky Wilson, King’s College Hospital NHS Foundation Trust & Professor Catherine Pope, Nuffield 
Department of Primary Care Health Sciences, University of Oxford. 
 
Co-investigators (PACFiND programme) 
Dr Rosemary 
Hollick 
Senior Clinical 
Lecturer/Honorary Consultant 
Rheumatologist 
University of 
Aberdeen 
rhollick@abdn.ac.uk  
Prof Louise 
Locock 
Associate Director: Research 
University of 
Aberdeen 
Louise.locock@abdn.ac.uk 
Prof Corrinda 
Black 
Consultant in Public Health 
University of 
Aberdeen 
Corri.black@abdn.ac.uk 
Prof Catherine 
Pope 
Professor of Medical Sociology 
University of 
Oxford 
Catherine.pope@phc.ox.ac.uk 
Dr Nicky 
Wilson 
Consultant Physiotherapist 
King’s College 
Hospital 
Nicky.wilson1@nhs.net 
Prof Paul 
McNamee 
Professor of Health Economics 
University of 
Aberdeen 
p.mcnamee@abdn.ac.uk 
Simon Stones 
Patient research partner 
 
   
Debra Dulake 
Patient research partner 
 
 
Dr Neil Basu 
Clinical senior lecturer 
/Honorary Consultant 
Rheumatologist 
University of 
Glasgow 
 Neil.Basu@glasgow.ac.uk 
Dr Kathyrn 
Martin 
Lecturer in epidemiology 
University of 
Aberdeen 
Kathyrn.martin@abdn.ac.uk 
Dr Sarah 
Maclennan 
Senior health psychologist 
University of 
Aberdeen 
s.maclennan@abdn.ac.uk 
Prof Ernest 
Choy 
Professor and Head of 
Rheumatology 
Cardiff 
University 
choyeh@cardiff.ac.uk  

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Dr Gareth 
Jones 
Reader in Epidemiology 
University of 
Aberdeen 
gareth.jones@abdn.ac.uk 
Prof Peter 
Murchie 
Professor of primary care 
University of 
Aberdeen 
p.murchie@abdn.ac.uk 
Prof 
Christopher 
Eccleston 
Professor of Psychology 
University of 
Bath 
C.Eccleston@bath.ac.uk 
Prof David 
Williams 
Professor of Anesthesiology, 
Medicine, Psychiatry, and 
Psychology 
University of 
Michigan 
daveawms@med.umich.edu  
Prof Karen 
Walker-Bone 
Professor in Occupational 
Rheumatology and Honorary 
Consultant in Rheumatology 
University of 
Southampton 
kwb@mrc.soton.ac.uk 
Prof Sue 
Ziebland 
Professor of Medical Sociology, 
Director Health Experiences 
Research Group 
University of 
Oxford 
sue.ziebland@phc.ox.ac.uk  
 
 
 
 
KEY WORDS:  
 
Fibromyalgia 
Case study 
Realist research 
Health service delivery  
 
 
 
 
 
 
 
 
 
 
 
 
 
 

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STUDY FLOW CHART 
 

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STUDY PROTOCOL 
A realist-informed investigation of the organisation and delivery of health and social care services for 
people with fibromyalgia living in the UK. 
 
1 
BACKGROUND 
Fibromyalgia is a multi-symptom long-term condition that affects approximately 2% of the general 
population (Heidari et al. 2017). It is characterised by persistent generalised pain, fatigue, cognitive 
symptoms, unrefreshing sleep and psychological distress (Wolfe et al. 2016). While recovery from 
fibromyalgia is possible (Grape et al. 2015), many individuals report ongoing pain and disability 
decades after diagnosis, and the personal, social and economic impact of fibromyalgia is substantial 
(Sim & Madden 2008; Mengshoel & Grape 2017; Arnold et al. 2019). 
 
Furthermore, diagnosis of fibromyalgia can be challenging. Patients report that the lead up to 
diagnosis is often protracted and punctuated by consultations with different healthcare providers 
during which hopes are raised and dashed (Mengshoel et al. 2018). For clinicians, the lack of 
conclusive diagnostic laboratory and imaging tests, and the variable nature of the condition is 
problematic, often resulting in healthcare provider and patient frustration and dissatisfaction (Briones-
Vozmediano et al. 2013; Arnold et al. 2019). 
 
Dissatisfaction with healthcare services often persists after diagnosis (Mengshoel & Grape 2017) 
despite the existence of evidence-based non-pharmacological and pharmacological interventions to 
treat and manage the signs and symptoms of fibromyalgia (Macfarlane et al. 2017). Patient 
experiences of UK based healthcare services reflect unmet need (Lempp et al. 2008; Patient and 
Client Council 2016) and there is a lack of a universally accepted model or package of health and/or 
social care for all patients with the condition (Able et al. 2016; Doebl et al. 2020). This negatively 
impacts quality improvement activities, patient empowerment, and ultimately, outcomes for people 
with fibromyalgia.  
 
To address this issue, the University of Aberdeen, in collaboration with a number of other universities 
in the UK and the University of Michigan, has received funding from the charity Versus Arthritis to 
develop new pathways of care with and for people with fibromyalgia. PAtient-centred Care for 
Fibromyalgia: New pathway Design (PACFiND) is a five-year funded programme of work that seeks to 
develop better services for people with fibromyalgia 
(https://www.abdn.ac.uk/iahs/research/epidemiology/pacfind-1483.php). This study is one component 
of the PACFiND research programme and seeks to understand how health and social care is currently 

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organised and delivered to people with fibromyalgia living in the UK to help identify what works, for 
whom and in what circumstances. 
 
2 
RATIONALE  
Health and social care interventions (e.g. drug therapy) or packages of inter-related interventions (e.g. 
drug therapy plus counselling) work differently for different groups of people under different conditions 
in different settings (Pawson & Tilley 1997). Developing and refining a transferable theory (or theories) 
about how interventions or packages of inter-related interventions (from here on referred to as 
‘programmes’) deliver particular outcomes for people with fibromyalgia, will potentially open up 
opportunities to improve health-related outcomes and patient satisfaction for people with fibromyalgia 
living in the UK. 
 
Building a programme theory (or theories) to underpin the organisation and delivery of health and 
social care for patients with fibromyalgia, from which new pathways or models can be advanced, is 
likely to be of benefit to people with fibromyalgia, their families and carers, health and social care 
practitioners and policy makers concerned with the organisation of health and or social care.  
 
3 
THEORETICAL FRAMEWORK 
Realist-informed qualitative case studies will be undertaken to explore and understand how health and 
social care programmes delivered to people with fibromyalgia living in the UK work and bring about 
their effects (Pawson & Tilley 1997; Exworthy et al. 2012; Russell et al. 2015). Realist research 
approaches aim to tease out for a particular programme what works, for whom, and in what 
circumstances, in order to develop and/or refine a transferable theory or set of theories that can be 
used to guide future programme design, planning and implementation in different settings (Wong et al. 
2016).  
 
Realist investigations focus on the causal mechanisms by which programmes bring about specified 
outcomes in different settings. In realist terminology, context (C), mechanism (M) and outcomes (O) 
form configurations (context-mechanism-outcome configurations (CMOCs)) that are central to enquiry 
(Pawson & Tilley 1997).  
 
The detection of CMOCs commonly starts with the identification of specified outcomes, with 
explanations about the causal mechanisms of these outcomes informed by an initial rough programme 
theory (Shearn et al. 2017). In this study, key outcomes of interest will focus on what matters to people 
with fibromyalgia and will be developed through patient and public involvement workshops, informed 

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by patient experience literature and interviews conducted with people with fibromyalgia gathered from 
other research studies associated with the PACFiND programme.  
 
 
4 RESEARCH AIM(S) 
 
1. Explore and understand how health and social care for people with fibromyalgia living in the 
UK is organised and delivered.  
 
2. Identify models of practice to inform codesign of new care pathways for people with 
fibromyalgia living in the UK. 
 
 
4.1 
Objectives 
 
 
1. To develop an initial rough programme theory (or theories) to guide and structure data 
gathering, analysis and synthesis within and across the case studies. 
 
2. To describe existing care practices and processes associated with identifying and supporting 
people with fibromyalgia in relation to outcomes that matter to people with fibromyalgia living in 
the UK. 
 
3. To identify, map and explain the causal mechanisms associated with change / outcomes that 
matter to people with fibromyalgia living in the UK. 
 
4. To specify key CMOCs within and across case studies.  
 
5. To develop a realist programme theory (or theories) underpinning positive outcomes in relation 
to identifying and supporting people with fibromyalgia living in the UK. 
 
 
 
 
4.2 Outcome 
 

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A primary focus of data collection during the case studies will be on outcomes that matter to people 
with fibromyalgia. A preliminary set of outcomes has been developed, informed by peer-reviewed 
literature about patient experiences of living with and managing fibromyalgia (for example, Mengshoel 
and Grape 2017; Mengshoel et al. 2018; Muraleethran et al. 2018) and data taken from new 
interviews conducted with people with fibromyalgia as part of the wider PACFiND programme. These 
are: 
 
i) 
Validation of patient experiences of signs and symptoms of fibromyalgia and prompt diagnosis.  
ii) 
Interactions between people with fibromyalgia and health and/or social care providers that 
generate hope, are individualised and patient-centred, and focussed on ‘what matters to me’ 
through processes of shared decision making. 
iii) 
Recognition of fibromyalgia as a disability (Barber et al. 2019).  
iv) 
Sustained and supportive relationships with skilled and knowledgeable health and/or social 
care providers over time to help build self-management and facilitate re-integration into life and 
society.  
v) 
Prompt access to interventions/programmes of health and/or social care that are coordinated 
across providers. 
vi) 
Increased knowledge, skills and confidence to manage independently.      
These outcomes will be further developed and refined through discussions with members of our 
affiliated patient and public reference groups. 
 
 
5 
STUDY DESIGN and METHODS of DATA COLLECTION AND DATA ANALYSIS 
 
We will purposively sample up to ten qualitative case studies in the UK to explore current health and 
social care pathways for identifying and supporting people with fibromyalgia living in the UK. Case 
studies may include one service/provider or multiple services/providers (NHS or non-NHS) within a 
locality, reflecting configurations and delivery of health and social care services for people with 
fibromyalgia. 
 
Data collection 
 

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We will: 
 
  
a) Gather documentary evidence, such as: policy and strategy documents; local care pathways 
and services processes relating to the provision of health and social care for people with 
fibromyalgia or signs and symptoms suggestive of fibromyalgia; staff training and development 
documents and service and programme evaluations. 
 
b) Undertake focussed observations (in-person or on-line depending on the coronavirus 
pandemic situation) of the work done by staff relating to i) the practices and processes 
associated with identifying people with fibromyalgia and ii) the organisation and delivery of care 
to support people with fibromyalgia. Observations, (which may include informal conversation 
with staff during quiet periods) will focus on outcomes that matter to people with fibromyalgia 
and the circumstances within which these outcomes emerge or not. As part of our 
ethnographic work, we will capture information about: (i) providers, such as the type, skill mix 
and capacities of health and social care worker(s) associated with the delivery of a 
programme; (ii) programmes of care, including the location, duration and nature of 
interventions (for example face-to face or remote interventions); (iii) the circumstances in which 
programmes are delivered (for example, organisational culture and constraints); and (iv) inter-
personal interactions (for example, between staff and patients, staff and others) in order to 
understand the infrastructure underpinning a programme (Pawson 2006). Data will be collected 
via field notes using an observation record template and later word processed. 
 
c) Undertake face to face, telephone or on-line semi-structured interviews with staff delivering 
/organising health and social care for people with fibromyalgia. The aim of the interviews will 
be to explore participant’s views and experiences of circumstances influencing outcomes that 
matter to people with fibromyalgia in relation to identification and support of people with the 
condition, in order to build explanations about what works, how it works and the circumstances 
in which it works. A topic guide (see Interview schedule) will inform the interviews. Face-to-face 
or telephone interviews will be digitally audio-recorded, encrypted and subsequently 
transcribed verbatim. On-line interviews will be audio-recorded using Microsoft Teams. Data 
will be de-identified at the point of transcription. In line with the development of a realist 
programme theory (or theories), up to two shorter follow up interviews may be requested with 
participants to clarify any supplementary related points (Manzano 2016). These can be 
conducted by telephone, email or on-line. 
 

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d) Undertake on-line focus groups within and or across case studies with key stakeholders 
involved in the organisation and delivery of healthcare services for people with fibromyalgia.  
The aim of the focus groups will be to further develop a realist programme theory (or theories), 
through integration of stakeholder theory with middle range theories, about how programmes 
of care for people with fibromyalgia work. Focus groups enable participants to build on each 
other’s views, and explanations or theories underpinning positive outcomes relating to 
identification and support of people with fibromyalgia will be presented to participants for 
confirmation and refinement (Astbury 2018). A deliberative discussion focus group approach 
will be taken (Rothwell et al. 2016), using both live chat and discussion boards over a period of 
one week to allow reflection and development of the discussion. The online groups and 
discussion boards will be facilitated and moderated by members of the research team. A topic 
guide will be further developed (see outline focus group sample guide), drawing on data 
gathered during observations and interviews to formulate theory or theories, to guide the focus 
groups discussion. Live online discussion sessions will take place on Microsoft Teams and will 
be audio-recorded directly from Microsoft Teams. The recording will be encrypted and 
subsequently transcribed. Microsoft Teams channels with access restricted to specified users 
will be used for discussion boards. The discussions in the Microsoft Teams chat feature will be 
copied and pasted into word documents. All data will be de-identified at the point of 
transcription/extraction.  
 
Data will be collated using Microsoft Word and Excel packages available to the researchers. 
 
Data analysis 
 
Data management and analysis will be aided by qualitative data software (NVivo) and informed by a 
realist approach. Following familiarisation with the data, initial coding of texts from observations and 
interviews will focus on descriptions of the actual practices and processes underpinning an 
intervention/programme, observed outcomes, the contextual conditions and the mechanisms, in order 
to formulate of a range of CMOCs for each case. Analysis of data will be iterative. Subsequent 
development of codes will focus on theory-driven interpretations of the data informed by an initial 
rough programme theory (or theories). Within each case, CMOCs will be compiled into narrative 
summaries and represented in tabular and or diagrammatic form to facilitate comparison within and 
between case study sites. This will enable the development and refining of the initial rough programme 
theory (or theories) into one or more realist programme theories underpinning the organisation and 
delivery of health and social care for people with fibromyalgia. Focus group discussions will be 
analysed thematically (Braun and Clarke, 2006).  

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Whilst a realist methodology will guide data analysis, we recognise that the distinction between 
mechanisms and contexts may not be clear-cut (Shaw et al. 2018). Therefore, we will also adopt a 
wider comparative case study analytic lens (Dopson and Fitzgerald, 2009) and take an interpretive 
holistic approach to understanding our cases and how they compare with others. 
 
The final set of CMOCs and realist programme theory or theories resulting from the case studies will 
be used as a framework for a subsequent co-design study within the PACFiND research programme. 
 
6 
STUDY SETTING 
 
Up to ten case studies will be selected. There will be a minimum of one case study in England, 
Scotland and Wales. Case studies may include one service/provider or multiple services/providers 
within a locality, reflecting configurations and delivery of health and social care services for people 
with fibromyalgia. 
 
Health and social care delivered to people with fibromyalgia will be the unit of interest of our case 
studies.  
 
Our choice of case studies will be informed by the intersection of four dimensions - (i) providers and 
personnel (for example NHS primary/secondary care and or non-NHS services); (ii) the nature of 
programme(s) delivered to people with fibromyalgia; (iii) inter-relations/local pathways connecting 
providers, services and organisations and (iv) local accessibility arrangements and permissions. 
 
 
7 
SAMPLE AND RECRUITMENT 
 
7.1  
Eligibility Criteria 
 
7.1.1 Inclusion criteria  
 
Case studies 
1. NHS and/or non-NHS organisations in the UK providing health and social care to people with 
fibromyalgia. 
 
Interviews, observations and focus groups 

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1. Health and social care professionals identifying and or providing support for people with 
fibromyalgia in one of the ten case studies. 
2. NHS/non-NHS staff influential in the organisation and/or delivery of health and social care to 
people with fibromyalgia in one of the ten case studies, for example, service managers, 
professional service leads, commissioners. 
 
7.1.2 Exclusion criteria  
 
Case studies 
1. NHS/non-NHS organisations in the UK not providing health or social care to people with 
fibromyalgia. 
 
 
Interviews, observations and focus groups 
1. NHS/non-NHS staff who are not able to communicate sufficiently well in English to participate 
effectively. 
 
 
7.2  
Sampling 
 
7.2.1  Size of sample 
 
Two professors (Pope and Locock) with combined experience of conducting case study research in 
healthcare totalling nearly 60 years designed this study. The numbers of cases are in line with sample 
sizes and case selection for numerous funded and successfully completed NIHR and charity funded 
projects using case study approaches. Please see for example:  
 
Pope C, Halford S, Turnbull J, Prichard J, Calestani M, May C. Using computer decision support 
systems in NHS emergency and urgent care: ethnographic study using normalisation process theory. 
BMC Health Serv Res. 2013 Mar 23;13:111. doi: 10.1186/1472-6963-13-111. ; 
 
Turnbull J, Pope C, Rowsell A, et al. The work, workforce, technology and organisational implications 
of the ‘111’ single point of access telephone number for urgent (non-emergency) care: a mixed-
methods case study. Southampton (UK): NIHR Journals Library; 2014 Feb. (Health Services and 
Delivery Research, No. 2.3 
Locock, L. Montgomery, C., Parkin, S. et al. How do frontline staff use patient experience data for 
service improvement? Findings from an ethnographic case study evaluation. 
J Health Serv Res Policy, 2020, 25:151-161. 
   
Case study sites 

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We will select up to ten case studies in the UK (at least one will be from England, Scotland and Wales) 
in order to capture a variety of contemporary NHS and non-NHS health and social care services 
delivered to people with fibromyalgia. 
 
Observations 
 
The exact number of observations undertaken in each case study cannot be determined in advance 
and will be shaped by the post-COVID health service landscape. However, we anticipate an 
observational period (in person or online) of up to two weeks for each case study to ensure in-depth 
data collection. 
 
Interviews 
 
The nature of ethnographic case study work informed by realist principles and the post-COVID health 
service landscape means that the participant sample has to remain flexible. The exact number of 
interviews undertaken in each case study will be dependent on the mix and configuration of staff 
involved in the organisation and delivery of health and social care for people with fibromyalgia and 
their availability. We estimate that the number of participants interviewed in each case study will be 
approximately ten (100 participants in total). 
 
Online focus groups 
Up to ten online focus groups with between six and eight participants per group will be conducted 
within and across case studies (60-80 participants in total). 
 
7.2.2  Sampling technique 
 
Case studies 
To facilitate variation in our case studies we will construct a sampling frame incorporating a range of 
factors, including geographical region, primary/secondary/third sector care, co-location and or inter-
related services providing health and social care for people with fibromyalgia living in the UK. We will 
review the findings of two national surveys recently undertaken as part of the PACFiND programme and 
use these in combination with intelligence about services, gathered through online searches, local 
contacts and insights from members of the research study team.  
 
Interviews, observations and focus groups 

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Purposive sample, including staff delivering health and social care to people with fibromyalgia, service 
managers and professional leads involved in the organisation of health and social care for people with 
fibromyalgia, commissioners and policy makers.   
 
 
7.3 
 Recruitment 
 
7.3.1 Sample identification 
 
Once an organisation/provider has agreed to participate in the study, a local collaborator will facilitate 
access to the site and potential participants. The researchers will work with local collaborators to identify 
relevant stakeholders to invite to participate in the study following the granting of local permissions.  
 
7.3.2 Consent 
 
Health and social care staff wishing to participate in the study will be required to provide written 
informed consent prior to taking part. Consent will be taken by the researchers responsible for 
conducting the study (Nicky Wilson, Catherine Pope and a Research Associate appointed to work on 
the study). Separate consent forms will be used for observations, interviews and focus groups. When 
data collection of an observation, interview or focus group is fully online, one of the researchers will 
read through the consent form individually with each participant and ask the participant to verbally 
agree to each point. This will be recorded on the consent form. In addition, verbal consent to observe 
interactions between participating staff and others (for example other staff during healthcare provider 
group meetings) will be obtained from all those involved and recorded in the observation field notes. 
Patients attending services during periods of observation, and non-participating staff, will be informed 
about the study via an information leaflet given out by one of the researchers and/or a member of the 
patient’s direct care team. Information leaflets will also be displayed within the department. For online 
observations, any person who may be part of the observation will be sent an information leaflet prior to 
an observation taking place.  
Verbal permission to observe a consultation/ interaction and make field notes will be sought from each 
person present during an observation. Consent will be recorded in field notes. It will be made clear to 
all patients attending services during periods of observations that the purpose of the research is to 
understand how services for people with fibromyalgia are organised and delivered and that all data 
gathered will relate to this and not to individual patients. 
7.3.3 Withdrawal from the study  

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Participants can choose to withdraw from the study at any time. Withdrawals will be recorded, and 
permission will be sought from the participant to retain their study data up to the point of withdrawal. If 
a participant withdraws from the study early in the data collection phase, another participant may be 
approached and invited to take part in the study.  
 
8 
ETHICAL AND REGULATORY CONSIDERATIONS 
 
8.1 
Assessment and management of risk 
 
Risk of harm to participants  
Minimal risk of harm to participants is anticipated from involvement in this study. Participants may 
perceive intrusion or inconvenience as a result of having a researcher observe their clinical practice or 
service-related work. The researcher will endeavour to not create additional work for participants. In 
addition, the researcher will ensure that interviews are conducted at a convenient time for participants. 
 
Risk of harm to patients with fibromyalgia attending NHS or non-NHS services 
Patients attending services during periods of observational data collection may become distressed 
during consultations with healthcare providers. Should this occur, the researcher will offer to leave the 
room/area/virtual platform. 
 
Risks to researchers 
This study poses minimal risk of physical or emotional harm to the researchers. A risk assessment, 
sensitive to the local research environment, will be undertaken prior to data collection at each case 
study site. The researchers will follow relevant policies relating to the coronavirus pandemic, lone 
working and safeguarding and will seek advice if necessary.  
 
 
8.2  
 Research Ethics Committee (REC) and other Regulatory review & reports 
 
A favourable opinion will be sought from an NHS Research Ethics Committee and local research and 
development offices for participating organisations prior to commencement of the study. The study will be 
conducted in accordance with Good Research Practice and Good Clinical Practice guidance. Any 
amendments requiring ethical review will not be implemented until a favourable opinion is granted and 
mechanisms to implement change at a specific site are in place.  
 
8.3  
Peer review 

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The PACFiND programme (incorporating this study) was subject to external peer review as part of a 
competitive funding application.  
 
8.4  
Patient & Public Involvement 
 
Nine patient partners, identified through the charity Fibromyalgia Action UK contributed to the 
development of the proposal for the PACFiND research project and two patient research partners are 
members of the study management team that meets monthly to discuss aspects of project design, project 
development and implementation. In addition, PPI groups in Scotland have contributed to the 
development and implementation of two studies embedded within an earlier workpackage of the 
PACFiND programme, the findings of which have informed this study. 
 
In relation to the research described in this proposal, a patient and public representative group in 
South London of 8 to 10 people with fibromyalgia has been convened from support groups and NHS 
services provided by King’s College Hospital NHS Foundation Trust. The group met face-to-face in 
January 2020 and on-line in April 2020 and will meet again on another two occasions over the 
duration of the study. Group members will be asked to: (i) contribute to the development of outcomes 
that are important to people with fibromyalgia, in relation to the organisation and delivery of health and 
social care services; (ii) reflect on and discuss early findings of the study and (iii) contribute to the 
development of narratives about the most relevant CMOCs. In addition, group members will be asked 
to review patient information or dissemination material. This will be done via mail or email, with 
information sent out at least one week ahead of meetings. Two further PPI groups (one each in 
Scotland and Wales) will also be established to advise on and support the study. 
 
PPI group members will be reimbursed for attendance at meetings and for time revising study 
materials. Rates will be informed by INVOLVE guidance and will be context specific.  
 
 
 
8.5 
Data protection and patient confidentiality  
 
The sponsor will act as the research data controller for this study. All members of the research study 
team will comply with the requirements of GDPR and the Data Protection Act 2018. Audio-recorded 
interview and focus group data will be encrypted and sent securely to a University of Aberdeen 
approved third party transcribing service provider. Notes taken during observations will be typed up on 
hospital or university password-protected computers. Any identifiable information will be removed from 

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the data at the point when it is typed up. Audio-recordings, field notes and texts from the discussion 
boards and chat during online focus groups, will subsequently be destroyed. De-identified data and 
non-paper consent forms will be uploaded by members of the research team to a password-protected 
drive on the University of Aberdeen’s secure servers. Paper consent forms will be stored in locked 
filing cabinets in secure offices at the researchers place of work (Wilson, King’s College Hospital; 
Pope & Research Associate, Nuffield Department of Primary Care Health Sciences).  These data will 
be stored under a unique participant identification for ten years and will be accessible to the three 
members of the research study team only.  
 
Participant personal data (e.g. names and contact details) will be kept electronically and stored 
separately within a restricted folder on a password-protected drive on the University of Aberdeen's 
secure servers. Only the researchers collecting data will have access to this restricted folder. 
Anonymised data may be shared by ZendTo with members of the research team who do not have 
access to this folder. Participant personal data will be deleted three months after the end of the study.  
 
Data collected during this study will be kept confidential. However, participants, patients and non-
participant staff will be informed (via Participant Information Sheets, Consent forms and the 
information leaflet) of the limits of confidentiality if potential safeguarding issues, poor standards of 
care, or malpractice are identified during the period of data collection.  
 
Reports or publications related to the study findings will not contain information that could identify 
individuals or organisations.  
 
8.6 
Inspection of records 
 
Direct access to all study records and source documentation will be granted by the Chief Investigator, 
Principal Investigators, and all institutions involved in the study, to authorised representatives from the 
Sponsor or host institution for study-related monitoring and/or audit of the study to ensure compliance 
with regulations. 
 
8.7 
Indemnity 
 
The University of Aberdeen is the Sponsor of the study and holds a policy of public liability insurance for 
legal liabilities arising from the conduct of the study.  
 

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The researchers conducting the study will hold honorary contracts with the University of Aberdeen and 
will have cover under the University of Aberdeen’s insurance policy. 
 
The Sponsor does not provide study participants with indemnity in relation to participation in the study. 
 
8.8 
Access to the final study dataset 
 
Only the researchers responsible for conducting the study and gathering data (Wilson, Pope and a 
Research Associate appointed to work on the study) will have access to the full data set including 
participant names and ID numbers. The Chief Investigator, Deputy Chief Investigator and other 
members of the study management team will have access to the full but de-identified data set. De-
identified transcripts or extracts of transcripts may be shared with other members of the research team 
for analysis and discussion using ZendTo. 
 
9 
STUDY CONDUCT RESPONSIBILITIES 
 
9.1 
Protocol amendments, deviation and breaches  
 
If a protocol or study document amendment is necessary, the Chief Investigator will seek approval 
from the Sponsor, Research Ethics Committee and NHS R&D Offices. An amendment will not be 
implemented without approval.  Any deviation from the protocol, (including the nature and reason for 
deviation) will be documented and reported to the Chief Investigator and the study Sponsor. Any 
subsequent amendment will be submitted to the Sponsor for approval and then to the appropriate 
Research Ethics Committee and NHS R&D Offices for review and approval. If a serious breach of 
GCP is suspected, it will be reported to the Sponsor immediately using the ‘Breach Report Form’.   
 
9.1  
Study record retention 
 
Study records will be archived and stored on the University of Aberdeen’s secure server (in the 
PACFIND restricted access folder) for a period of ten years. 
 
9.2  
End of study 
 
The end of the study, defined as the last live online discussion of the last focus group, will be reported 
to the Sponsor and the Research Ethics Committee within 90 days of final data collection or within 15 

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days if the study ends prematurely. A summary report of the study will be provided to the Sponsor and 
Research Ethics Committee within one year of the end of the study. 
 
 
10 
DISSEMINATION POLICY 
 
10.1  
Dissemination policy 
 
On completion, the findings of this study will be written up as a full report and made available on the 
PACFiND website and disseminated to key stakeholders, including public involvement groups working 
alongside the PACFiND study team and national charities such as Versus Arthritis and Fibromyalgia 
Action UK. We will offer all participants the opportunity to be sent a summary of the findings at the end 
of the study. In addition, the study findings will be presented at national and international conferences 
and disseminated through peer reviewed professional journals.  
 
10.2  Authorship eligibility guidelines and any intended use of professional writers 
 
Authorship of the final study report and subsequent papers for publication will follow guidance set out 
by The International Committee of Medical Journal Editors. Manuscripts will include acknowledgement 
of contributors and disclaimer statements as required by the funder. 
 
 
 
11 
REFERENCES 
 
Able, SL., Robinson, RL., Kroenke, K., Mease, P., et al. (2016) Variations in the management of 
fibromyagia by physician speciality: rheumatology versus primary care. Pragmatic and Observational 
Research 7;11-20. 
 
Arnold, LM., Bennett, RM., Crofford, LJ., Dean, LE., et al. (2019) AAPT Diagnostic criteria for 
Fibromyalgia. The Journal of Pain 20(6): 611-28. 
 
Astbury, B (2018) Making claims using realist methods, In: Emmel, N., Greenhalgh, J., Manzano, A., 
Monaghan, M, and Dalkin, S. (eds) Doing realist research. London, Sage, 59-77. 
 
Barber, S., Kennedy, S., McInnes, R., Pyper, D. and Sutherland, N. (2019) Recogniton of fibromyalgia 
as a disability. Debate pack 2019/0003. Available from: 
https://researchbriefings.parliament.uk/ResearchBriefing/Summary/CDP-2019-0003#fullreport [Last 
accessed 03 January 2020]. 

 Understanding health services delivery for fibromyalgia 
V2 [15.11.2020] 
16 
 
 
Braun, V. and Clarke, V. (2006) Using thematic analysis in psychology. Qualitative Research in 
Psychology 3(2): 77-101, DOI: 10.1191/1478088706qp063oa 
 
Briones-Vozmediano, E., Vives-Cases, C., Ronda-Perez, E. and Gil-Gonzalez, D. (2013) Patients’ and 
professionals’ views on managing fibromyalgia. Pain Research & Management 18(1):19-24. 
 
Doebl, S., Macfarlane, GJ. and Hollick, RJ. (2020) ‘No one wants to look after the fibro patient’. 
Understanding models, and patient perspectives, of care for fibromyalgia: reviews of current evidence. 
PAIN doi: 10.1097/j.pain.0000000000001870. [Epub ahead of print] 
 
Dopson S, Fitzgerald L. Comparative case study designs: their utility and development in 
organizational research. In D. Buchanan and A. Bryman (eds). The Sage Handbook of Organizational 
Research Methods. London: Sage, 2009 
 
Grape, HE., Solbraekke, KN., Kirkevold, M. and Mengshoel, AM. (2015) Staying healthy from 
fibromyalgia is ongoing hard work. Qualitative Health Research 25(5): 679-88. 
 
Heidari, F., Afshari, M. and Moosazadeh, M. (2017) Prevalence of fibromyalgia in general population 
and patients, a systematic review and meta-analysis. Rheumatology International 37: 1527-39. 
 
Hsieh, HF. and Shannon, SE. (2005) Three approaches to qualitative content analysis. Qualitative 
Health Research 15(9): 1277-1288.  
 
Macfarlane, GJ., Kronisch, C., Dean, LE., Atzeni, F., et al. (2017) EULAR revised recommendations 
for the management of fibromyalgia. Annals of Rheumatic Diseases 76(2):318-328. 
 
Manzano, A. (2016) The craft of interviewing in realist evaluation. Evaluation 22(3):342-360. 
 
Mengshoel, AM. And Grape, HE. (2017) Retinking physiotherapy for patients with fibromyalgia – 
lessons learnt from qualitative studies. Physical Therapy Reviews 22(5-6): 254-9. 
 
Mengshoel, AM., Sim, J., Ahlsen, B. and Madden, S. (2018) Diagnostic experience of patients with 
fibromyalgia- A meta-ethnography. Chronic Illness 14(3): 194-211. 
 
Patient and Client Council (2016) A hidden condition. Ten people living with fibromyalgia tell their 
story. Available from: https://www.myni.life/wp-
content/uploads/2019/05/Patient_stories_A_Hidden_Condition.pdf [Last accessed 31 December 2019] 
 
Pawson R & Tilley, N. (1997) Realistic evaluation. London: Sage.  
 
Pawson, R. (2006) Evidence-based policy: A realist perspective. Sage Publications. 
 

 Understanding health services delivery for fibromyalgia 
V2 [15.11.2020] 
17 
 
Rothwell, E., Anderson, R. and Botkin, JR. (2016) Deliberative discussion focus groups. Qualitative 
Health Research 26(6):734-740. 
 
Russell, J. Greenhalgh, T. and Kushner, S. (2015) Case study evaluation: past, present and future 
challenges. Bingley, Emerald Group Publishing Limited. 
 
Segar, J. Checkland, K., Coleman, A. and McDermott, I. (2015) Thinking about case studies in 3-D: 
Researching the NHS clinical commissioning landscape in England. In: Russell, J., Greenhalgh, T. 
and Kushner, S. (eds) Case study evaluation: past, present and future challenges. Advances in 
Program Evaluation. Bingley, Emerald Group Publishing Limited, 15:85-105. 
 
Shaw, J., Steele Gray, C., Baker, GR., Denis JL., et al. (2018) Mechanisms, contexts and points of 
contention: operationalizing realist-informed research for complex interventions. BMC Medical 
Research Methodology 18: 178. 
 
Shearn K., Allmark, P., Piercy, H. and Hirst, J. (2017) Building realist program theory for large complex 
and messy interventions. International Journal of Qualitative Methods 16: 1-11. 
 
Sim, J. and Madden, S. (2008) Illness experience in fibromyalgia syndrome: A meta-synthesis of 
qualitative studies. Social Science & Medicine 67: 57-67. 
 
Wolfe, F., Clauw, DJ., Fitzcharles, MA., Goldenberg, DL. et al. (2016) 2016 Revisions to the 
2010/2011 fibromyalgia diagnostic criteria. Seminars in Arthritis and Rheumatism 46: 319-29. 
 
Wong G (2015) Special Invited Editorial: Getting started with realist research. International Journal of 
Qualitative Methods 1-2. 
 

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12.  
APPENDICIES 
 
12.1 
Required documentation  
Letter of invitation to participants (v1 26.07.2020) 
Participant information sheet observation &/or interview (v2 15th November 2020) 
Participant information sheet online focus groups (v2 15th November 2020) 
Information leaflet about observations (v2 15th November 2020) 
Informed consent form observations (v2 15th November 2020) 
Informed consent form interviews (v2 15th November 2020) 
Informed consent form focus groups (v2 15th November 2020) 
Observation record template (v1 5th October 2020) 
Interview schedule (v1 5th October 2020)] 
Outline focus group sample guide (v1 5th October 2020) 
 
 
13. 
Appendix 3 – Amendment History 
 
Amendment 
No. 
Protocol 
version no. 
Date issued 
Author(s) of 
changes 
Details of changes made 
1 
2 
15.11.2020 
Nicky Wilson 
Information about the limits of 
confidentiality in the event of the 
identification of safeguarding issues, 
poor standards of care or malpractice 
has been added to Section 8.5.