Adaptation to Inflammatory Bowel Disease

by

Lawrence Matini

Thesis submitted for the award of Doctor of Philosophy January 2017

School of Psychology

Faculty of Health and Medical Sciences

University of Surrey

Declaration of originality

This thesis and the work to which it refers are the results of my own efforts. Any ideas, data, images or text resulting from the work of others (whether published or unpublished) are fully identified as such within the work and attributed to their originator in the text. This thesis has not been submitted in whole or in part for any other academic degree or professional qualification. I agree that the University has the right to submit my work to the plagiarism detection service TurnitinUK for originality checks. Whether or not drafts have been so-assessed, the University reserves the right to acquire an electronic version of the final document (as submitted) for assessment as above.

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Abstract

Inflammatory bowel disease (IBD) is a term used to describe two chronic diseases of the gastrointestinal tract: Ulcerative Colitis (UC) and Crohn's Disease (CD). Although the efficacy of treatment is continuously improving, Quality of Life (QoL) in this illness population remains low with many patients suffering from psychological and psychiatric comorbidities. Psychological interventions aimed at improving outcomes in these patients have largely demonstrated little improvement. This thesis argues that this may be the result of poor understanding of the experience of living with this condition with too little focus on the adaptation of the patient to their illness. This thesis aimed to address this gap in the literature through four empirical studies. Firstly, Study 1 used a qualitative design to (n = 22) to explore the lived experiences of patients with IBD and to conceptualise adaptation to IBD. The results highlighted the importance of making sense of the illness and the impact and feelings associated with the illness. This was transcended by a notion of uncertainty which was resolved by employing coping mechanisms to restore equilibrium between their identity before and after diagnosis, resulting in a 'new normal'. Study 2 then employed a cross-sectional design (n = 307) to develop a new measure of adaptation to IBD (the A-IBD) which after psychometric analysis revealed four subscales including person identity, patient identity, acceptance and expectations. This study also explored the degree of association of the A-IBD with existing measures of sense making (BIPQ) and QoL (IBDQ), to assess the ability of the A-IBD in predicting QoL and ascertain whether it could predict QoL over and above sense making. The results suggested the A-IBD was not synonymous with these constructs and had utility as a predictor of QoL even when accounting for the predictive ability of the BIPQ. Finally, Study 3 used a combination of qualitative and quantitative design (n = 16). Patients scoring in the top and bottom 25% of the A-IBD from Study 2 completed the measure again to assess the dynamic nature of adaptation and were interviewed about the factors that either encouraged or inhibited their degree of adaptation. This analysis revealed that adaptation is indeed dynamic, and that antecedents of adaptation include 'engagement', 'resilience' and certain 'contingencies' including disease and social factors. Overall, the findings from this thesis indicate that the treatment of IBD must be approached in a biopsychosocial manner, that adaptation can be measured effectively with the new tool and that adaptation, with an emphasis on the notion of person, not patient, predicts quality of life.

Acknowledgements

Firstly, I would like to extend my utmost gratitude to my supervisor, Professor Jane Ogden, who has supported and encouraged me every step of the way over the past three years. Your understanding throughout the toughest point in my life health-wise gave me the time and space I needed to re-focus and see this PhD through to the end. I couldn't have wished for a better person to be my supervisor, so thank you.

I would also like to thank Dr. Deborah Cooke for her support and guidance as well as the numerous other staff and students of the University of Surrey School of Psychology that have assisted me along this journey. I must also thank the School of Psychology for selecting me to be a recipient of the Russell Wicks memorial fund which was of great help in seeing me through the final year of this PhD.

I of course also owe a huge thanks to all the individuals who took part in my research. The willingness of those who gave up their time and shared their personal stories with me was staggering and I am forever grateful. I hope this PhD contributes to an increased awareness and better management of IBD and thus goes some way towards paying back all that were involved.

Finally, I would like to thank my family and closest friends. My Mum, Dad and Brother have been unwavering in their support ever since I expressed my desire to one day complete a PhD a number of years ago. Through the toughest of times they continued to stand by me and help me realise this dream. I'm particularly glad my Dad can now tell everyone about how his son is a doctor without me having to stop him.

Table of Contents

Chapter One
Literature Review / 1
1.1 Definition of IBD / 1
1.2 Prevalence of IBD / 5
1.3 Causes of IBD / 10
1.4 Consequences of IBD / 15
1.4.1 Physiological consequences / 15
1.4.2 Psychological consequences / 21
1.5 IBD as a chronic condition / 25
1.6 The process of having a chronic condition / 26
1.6.1 Coping / 26
1.6.2 Criticisms of the coping literature / 30
1.6.3 Sense making / 32
1.6.4 Criticism of the SRM and CAT / 42
1.7 Outcomes of a chronic condition / 43
1.8 The working model of adjustment to chronic illness – integrating both processes and outcomes / 46
1.9 The central role of adaptation / 49
1.10 Process, outcomes and adaptation in IBD / 50
1.11 Overview of aims for thesis / 54
Chapter Two
Study 1: Patients’ experiences of living with IBD: a qualitative study / 57
2.1 Overview / 57
2.2 Introduction / 57
2.3 Method / 60
2.3.1 Design / 60
2.3.2. Sample / 60
2.3.3. Procedure / 63
2.3.4 Interview schedule / 63
2.3.5 Data analysis
2.4 Results / 64
2.5 Discussion / 76
2.5.1 Summary of findings / 76
2.5.2 Links to literature / 77
2.5.3 In summary / 78
2.5.4 Methodological limitations / 78
2.5.5 Conclusion / 80
Chapter Three
Study 2: The development of a new measure to assess adaptation in patients with IBD and its utility in predicting QoL: a cross-sectional study / 81
3.1 Overview / 81
3.2 Introduction / 3.2.1 Literature review
3.2.2. Steps involved in development and validation of the adaptation to IBD questionnaire / 82
82
85
3.3 Method / 87
3.3.1 Design / 87
3.3.2 Sample / 87
3.3.3 Procedure / 87
3.3.4 Measure / 88
3.4 Results / 89
3.4.1 Conceptualisation / 89
3.4.2 Operationalisation / 90
3.4.3 Assessment of the scale’s psychometric properties / 91
3.4.4 Reliability assessment / 97
3.4.5 Final scale with response options and instructions / 97
3.4.6 Association of the A-IBD with existing measures of sense making and QoL / 99
3.4.7 Using the A-IBD subscales to predict QoL / 102
3.4.8 Adaptation formula / 111
3.4.9 Does the A-IBD predict QoL over and above the BIPQ? / 111
3.5 Discussion / 113
3.5.1 Summary of findings / 113
3.5.2 Links to literature / 115
3.5.3 In summary / 117
3.5.4 Methodological limitations / 119
3.5.5 Conclusion / 122
Chapter Four
Study 3: Patients’ explanation for their degree of adaptation: a qualitative study with a quantitative component / 123
4.1 Overview / 123
4.2 Introduction / 124
4.3 Method / 126
4.3.1 Design / 126
4.3.2 Sample / 126
4.3.3 Classifying degree of adaptation / 127
4.3.4 Change in adaptation / 127
4.3.5 Procedure / 129
4.3.6 Interview schedule / 129
4.3.7 Data analysis / 130
4.4 Results / 130
4.5 Discussion / 4.5.1 Summary of findings- thematic analysis / 148
148
4.5.2 Links to literature - thematic analysis / 148
4.5.3 Summary of findings – adaptation - state or trait? / 150
4.5.4 Links to literature - adaptation – state or trait? / 150
4.5.5 In summary / 151
4.5.6 Methodological limitations / 152
4.5.7 Conclusion / 153
Chapter Five
General Discussion / 154
5.1 Overview / 154
5.2 Summary of findings / 155
5.3 Story of the thesis – What is adaptation? / 157
5.4 Methodological limitations / 160
5.5 Implications for literature / 163
5.6 Implications for research / 165
5.7 Implications for practice / 168
5.8 Conclusion / 169
References / 171
Appendices / 195
Appendix A - Study One Information Sheet / 195
Appendix B - Study One Consent Form / 199
Appendix C - Study One Interview Schedule / 200
Appendix D - Study One Debrief Sheet / 201
Appendix E - Study One University Ethics Committee Approval / 203
Appendix F - Information Sheet (Studies Two, Three and Four) / 204
Appendix G - Consent Form (Studies Two, Three and Four) / 208
Appendix H - Debrief Sheet (Studies Two, Three and Four) / 209
Appendix I - Adaptation to IBD Questionnaire (Study Two) / 211
Appendix J - Brief Illness Perceptions Questionnaire (Study Three) / 213
Appendix K - Inflammatory Bowel Disease Questionnaire (Study Three) / 214
Appendix L - Inflammatory Bowel Disease Questionnaire Stoma (Study Three) / 221
Appendix M - Study Four Interview Schedule / 232
Appendix N - University Ethics Committee Approval (Studies Two, Three and Four) / 234

Index of tables

Table 1. Participant demographic information 62

Table 2. Scale development and validation steps included and not included 86

Table 3. Factor loadings for 18 item adaptation questionnaire 95

Table 4. Means and standard deviations for 18 item adaptation questionnaire 96

Table 5. Correlations between A-IBD, Brief IPQ and IBDQ/IBDQ-S 100

Table 6. A-IBD and BIPQ predicting IBDQ scores 112

Table 7. A-IBD and BIPQ predicting IBDQ-S scores 113

Table 8. Participant demographic information 128

Table 9. Themes and subthemes of thematic analysis 131

100

Chapter One

Literature Review

1.1 Definition of IBD

Inflammatory Bowel Disease (IBD) is an umbrella term that encapsulates two very similar yet distinct idiopathic, chronic diseases of the gastrointestinal system: Ulcerative Colitis and Crohn's Disease. The validity of this grouping term is justified by the fact that both diseases are characterised by inflammation and ulceration of the gastrointestinal tract, frequent recurrences, and many gastrointestinal and systemic complications (Kirsner, 1995). The two diseases do however possess individual nuances. Ulcerative Colitis is a diffuse disorder of the colon which always affects the rectum and extends proximally to include variable degrees of the rest of the bowel. If only the rectum is diseased then this is often referred to as 'proctitis', which may be followed by 'distal colitis' involving the left or 'distal' side of the large intestine and finally 'total colitis' if the disease has spread throughout the large intestine. The disease never extends beyond the caecum (the intersection between the large and small intestine) and thus does not involve the small intestine. With Ulcerative Colitis only the innermost lining of the bowel wall becomes inflamed. In contrast, Crohn's disease can affect any part of the gastrointestinal tract. Although most commonly found in the ileocaecal region involving the terminal ileum (the most distal part of the small intestine) and the caecum, it is possible but rare for the disease to manifest itself solely in the anus, where it is referred to as 'perianal Crohn's', the mouth,known as 'oral Crohn's', or in the upper gut region - the oesophagus, stomach or duodenum, in which case it is referred to as 'gastroduodenal Crohn's'. Crohn's disease may involve several segments of intestinal inflammation simultaneously (known as 'skip legions') with regions of histologically healthy bowel in between (Jewell et al., 1992) and unlike Ulcerative Colitis, inflammation in Crohn's disease is transmural, i.e. all layers of the bowel wall may be inflamed. Patients with Crohn's disease also frequently suffer from strictures of the bowel (also known as 'stenosis', a narrowing of the intestinal wall) or fistula formation (ulcers that extend completely through the intestinal wall creating abnormal connections between different body parts) into adjacent loops of the intestine (enteroenteric or enterocolic fistulas), the bladder (enterovesical or colovesical fistulas), the vagina (enterovaginal fistulas), the skin (enterocutaneous fistulas), often occurring following surgery along the incision line on the abdomen, or most commonly the anal area (perianal fistulas) (Crohn's and Colitis UK, 2013).

While the areas of the gastrointestinal tract that the two diseases may inhabit differ, their symptoms share more similarities. The symptoms that IBD patients suffer from are commonly characterised by phases of exacerbation and intermittent remission (Jewell et al., 1992). During periods of disease relapse, symptoms that are commonly experienced by patients are phases of acute abdominal pain, spasms, nausea, fever, fatigue, diarrhoea and rectal bleeding (Cooper et al., 2010; Daniel, 2001). Even in times of remission, which are usually scarce, fatigue still largely affects patients' daily functioning (Czuber-Dochan et al., 2013).In conjunction with this, some may suffer from extraintestinal manifestations (EIMs) of the disease that may include maladies of the skin (most commonly 'erythema nodosum' - a condition where the layer of fat underneath the skin becomes inflamed causing red rounded, tender nodules to form, and 'pyoderma gangrenosum' - where pustules form on the skin and enlarge, coalesce and ulcerate) joints (arthritis and spondylitis) and eyes ('episcleritis' - mild pain and redness of the thin layer of tissue between the conjunctiva and connective tissue that forms the sclera of the eye, and 'uveitis' - inflammation of the middle layer of the eye called the uveal tract) (Vermeire & Rutgeerts, 2005). As well as these dermatologic, musculoskeletal and ocular EIMs, manifestations of the hepatopan-creatobiliary system such as 'primary sclerosing cholangitis - inflammation of the bile ducts inside and outside the liver causing scarring (Navaneethan & Shen, 2010)as well as renal and pulmonary manifestations such as 'nephrolithiasis' - the formation of kidney stones (Levine & Burakoff, 2011). These EIMs can be classified into two major groups: the first including reactive manifestations often associated with intestinal inflammatory activity and thus mimicking a pathogenic mechanism common with intestinal disease (of which arthritis, pyoderma gangrenosum and uveitis are examples), while the second includes many autoimmune diseases that are not considered specific to IBD but are associated with a major susceptibility to autoimmune diseases as IBD is thought to be (including spondylitis and alopecia) (Danese et al., 2005).Some form of these EIMs are observed in as many as 25-40% of patients with IBD (Bernstein et al., 2001).