Living with unpredictability in Multiple Sclerosis Hannah R Wilkinson BSc (Hons), MSc 2014 A thesis submitted in partial fulfilment of the requirements of the University of Lincoln for the degree of Doctorate in Clinical Psychology Page 1 of 195 Portfolio Abstract Background: Unpredictability is identified as a major factor in Multiple Sclerosis (MS) and reported to be a “constant companion” for individuals living with MS. It is reported to be a factor of uncertainty, which is indicated to have a biopsychosocial impact on these individuals. However the impact of uncertainty is argued to diminish over time. Whereas, the lived experience of unpredictability is reported to be continual. Furthermore, unpredictability is argued to have a continual psychological effect on individuals with MS. However, unpredictability is an aspect that remains relatively unaddressed in the research. Qualitative research has been highlighted to be fundamental in exploring the individual experience of illness, providing rich detailed descriptions. In addition it has been instrumental in the development of services to meet the needs of these individuals. The significance of unpredictability in MS highlights the need for further exploration of lived experience of this aspect of the illness to support healthcare professionals and the development of services to meet the needs of these individuals. Aim: This study aimed to address the limited research examining unpredictability in MS, to develop a comprehensive understanding of how this aspect affects the lives of individuals with MS. Method: This study employed a qualitative design. Twelve participants were recruited through the MS Society, local advertisements and by a ‘snowballing’ sampling technique. All participants lived in England, had a diagnosis of MS, and were able to provide consent to participate. Individual interviews were conducted (7 face-to-face and 5 telephone interviews) using a semi-structured interview. Interviews were transcribed verbatim and analysed by the researcher using a thematic analysis (TA) approach. Results: Three main themes were derived from the analysis: 1) Challenges to meaning-making; 2) A wide picture of unpredictability; and 3) Surviving unpredictability. Each of the themes generated between two or three sub-themes. The themes told the participant’s story for living with unpredictability. Page 2 of 195 Conclusion and recommendations: Unpredictability was reported to be a key factor of living with MS, yet it was challenging and difficult to understand. Participants described the wide reaching impact of unpredictability influencing them personally and socially. Pragmatic and psychological approaches were employed by participants to deal with unpredictability. The findings offer insight into the individual’s experience of living with unpredictability. It is argued the findings represent individuals who have reached a stage of acceptance with their illness and therefore may offer health professionals guidance around how to support individuals who have not been able to come to terms with the unpredictability of their illness. Furthermore, the findings highlight the wide reaching impact of unpredictability emphasising the importance of the family team. These findings may offer health professionals guidance around providing family education and support to help develop a cohesive network of support for the individual and the family experiencing the effects of unpredictability in MS. Page 3 of 195 Acknowledgements First and foremost, I would like to take this opportunity to thank my research supervisor Dr Roshan das Nair. His support, guidance and constructive feedback have been invaluable in devising, implementing and writing up this research project. Secondly, I would like to thank all the individuals who took time to participate in the study and shared their views of living with an unpredictable illness. Without their help and willingness to give their time and support in further recruitment, this research would not have been possible. Furthermore I would like to say an extra special thank you to the MS Society Research Network for their support during the early stages of the project, advertising the research and supporting me during the recruitment phase. Thirdly, I am extremely grateful for the endless encouragement from my family and friends, especially my Mum and Dad, for supporting me and offering their invaluable support and confidence in my ability to reach my goal. Finally, a heartfelt thank you goes to my husband, Jon, who has endured this journey with me, taking on many of the household roles, providing a sound board for my ideas and trying to make me smile even in the hardest of times. Your support and encouragement have helped me enormously. Thank you Page 4 of 195 Statement of Contribution Hannah Wilkinson was responsible for the design of this project, applying for ethical approval, reviewing the relevant literature, recruitment of participants, data collection, transcription of five of the interviews, analysis and writing up the research. Dr Roshan das Nair provided supervision and guidance throughout the research process. He was also responsible for independently coding the transcripts, reviewing themes, and reviewing the thematic map that was derived from the analysis. The MS Society provided the means to advertise the study, on their website and through local MS Society Newsletters. In addition local therapy centres and participants helped to advertise for recruitment to the study. A transcription service transcribed seven interviews. Page 5 of 195 Contents SYSTEMATIC LITERATURE REVIEW ................................................................ 9 ABSTRACT ................................................................................................................................ 11 INTRODUCTION .......................................................................................................................... 12 METHODS ................................................................................................................................. 13 RESULTS................................................................................................................................... 16 DISCUSSION .............................................................................................................................. 32 APPENDIX ................................................................................................................................. 36 REFERENCES ............................................................................................................................ 37 JOURNAL PAPER ............................................................................................. 42 ABSTRACT ................................................................................................................................ 45 METHOD ................................................................................................................................... 49 FINDINGS .................................................................................................................................. 54 DISCUSSION .............................................................................................................................. 70 CONCLUSION ............................................................................................................................. 75 JOURNAL REFERENCES ................................................................................. 76 EXTENDED PAPER ........................................................................................... 86 EXTENDED INTRODUCTION ............................................................................ 87 MULTIPLE SCLEROSIS. ............................................................................................................... 87 Aetiology and prevalence. ...................................................................................................... 87 Diagnosis and disease course. .............................................................................................. 87 Symptoms and consequences of MS. ................................................................................... 88 UNCERTAINTY AND UNPREDICTABILITY ........................................................................................ 89 Chronic illness literature. ........................................................................................................ 89 MS literature. .......................................................................................................................... 90 Effects of uncertainty and unpredictability. ............................................................................ 91 Psychological effects of unpredictability. ............................................................................... 92 THEORIES OF COPING ................................................................................................................ 93 IMPORTANCE OF LIVED EXPERIENCE ........................................................................................... 95 RATIONALE FOR PRESENT STUDY ............................................................................................... 96 RESEARCH AIM ......................................................................................................................... 96 EXTENDED METHODOLOGY ........................................................................... 97 RESEARCH DESIGN ................................................................................................................... 97 Ontology and epistemology. ................................................................................................... 97 Researcher’s epistemological position. .................................................................................. 98 Rational for qualitative methodology. ..................................................................................... 99 Methodology considerations. ............................................................................................... 100 Rational for individual interviews. ......................................................................................... 100 Limitations of individual interviews. ...................................................................................... 101 Rational for focus groups. .................................................................................................... 102 Page 6 of 195 Limitation of focus groups. ................................................................................................... 102 Photo methods. .................................................................................................................... 103 Service user input. ............................................................................................................... 103 Rational for integrated methods. .......................................................................................... 104 Methodology used in the study. ........................................................................................... 104 PROCEDURE ........................................................................................................................... 104 Rational for number of participants. ..................................................................................... 104 Sampling and recruitment. ................................................................................................... 105 Preparation for data collection. ............................................................................................ 106 Demographic interview. ........................................................................................................ 106 Semi-structured interview schedule. .................................................................................... 106 Interviews. ............................................................................................................................ 107 Transcription......................................................................................................................... 107 Ethical considerations and approval. ................................................................................... 107 DATA ANALYSIS ....................................................................................................................... 109 Qualitative approaches. ....................................................................................................... 109 Rational for TA. .................................................................................................................... 111 A priori decisions. ................................................................................................................. 112 TA procedure........................................................................................................................ 113 Establishing quality. ............................................................................................................. 114 Researcher’s statement of perspective. ............................................................................... 117 EXTENDED RESULTS ..................................................................................... 118 THEMES AND SUB-THEMES ....................................................................................................... 118 Challenges to meaning-making............................................................................................ 119 Difficulties of understanding and articulating unpredictability.......................................................... 119 How people understand and articulate unpredictability. ................................................................. 120 A wide picture of unpredictability.......................................................................................... 124 Self. ................................................................................................................................................ 124 Others. ............................................................................................................................................ 131 Surviving unpredictability. .................................................................................................... 134 Psychological. ................................................................................................................................. 134 Pragmatic. ...................................................................................................................................... 136 EXTENDED DISCUSSION ............................................................................... 138 SUMMARY OF FINDINGS ............................................................................................................ 138 FINDINGS IN RELATION TO PREVIOUS RESEARCH ........................................................................ 140 STRENGTHS AND LIMITATIONS .................................................................................................. 143 CLINICAL IMPLICATIONS ............................................................................................................ 145 FUTURE RESEARCH ................................................................................................................. 146 CRITICAL REFLECTION ................................................................................. 147 INITIAL STAGES OF THE RESEARCH ........................................................................................... 147 Personal orientation to research. ......................................................................................... 147 PLANNING AND CONDUCTING THE RESEARCH ............................................................................ 148 A research idea to a research proposal. .............................................................................. 148 Epistemological position. ...................................................................................................... 149 Decisions that had to be made............................................................................................. 149 Planning the interviews. ....................................................................................................... 150 REFLECTIONS DURING RESEARCH ............................................................................................ 150 Page 7 of 195 Interviews. ............................................................................................................................ 150 Transcription......................................................................................................................... 152 Analysis. ............................................................................................................................... 153 Write-up. ............................................................................................................................... 153 DEVELOPMENT AS RESEARCHER .............................................................................................. 155 PORTFLIO REFERENCES .............................................................................. 156 APPENDICES ................................................................................................... 170 APPENDIX A: JOURNAL MANUSCRIPT GUIDELINES ..................................................................... 170 APPENDIX B: INTERVIEW SCHEDULE ......................................................................................... 171 APPENDIX C: MS EXPERIENCE AND DEMOGRAPHICS INTERVIEW ............................................... 172 APPENDIX D: PARTICIPANT INFORMATION SHEET ...................................................................... 174 APPENDIX E: PARTICIPANT CONSENT FORM .............................................................................. 177 APPENDIX F: ADVERT FOR RECRUITMENT ................................................................................. 179 , APPENDIX G: PARTICIPANT DEMOGRAPHICS ............................................................................. 180 APPENDIX H: EXAMPLE OF INITIAL CODING AND INITIAL THEMES ................................................. 181 APPENDIX I: EXCERPT OF THEMES AND SUB-THEMES WITH ASSOCIATED CODES .......................... 182 APPENDIX J: EARLY THEMATIC MAP ......................................................................................... 186 APPENDIX K: THEMATIC MAP – FINAL VERSION ......................................................................... 187 APPENDIX L: EXCERPTS FROM REFLECTIVE DIARY .................................................................... 188 APPENDIX M: ETHICS APPROVAL LETTER ................................................................................. 189 APPENDIX N: ETHICS APPROVAL – E-MAIL TRAILS FOR AMENDMENTS .......................................... 190 APPENDIX O: SIGNED CONFIDENTIALITY FORM – TRANSCRIPTION SERVICE ................................ 193 APPENDIX P: DESIGNING THE STUDY - RESEARCH NETWORK QUESTIONNAIRE ........................... 194 APPENDIX Q: PROCEDURE FLOW DIAGRAM .............................................................................. 195 Tables and Figures Table 1: Summary of studies examined for relevance ....................................................18 Table 2: General Characteristics of review studies ........................................................21 Table 3: Summary of the themes of psychological effects of unpredictability .................27 Table 4: Model of the psychological impact of unpredictability in MS .............................32 Table 5: Summary demographics .................................................................................52 Table 6: Themes and Sub-themes .................................................................................55 Figure 1: Flow Chart – Identifying relevant studies .........................................................17 Figure 2: Shifting perspectives model ............................................................................94 Figure 3: Thematic Map of Themes and sub-themes .................................................. 118 Page 8 of 195 SYSTEMATIC LITERATURE REVIEW Page 9 of 195 The Psychological impact of living with an unpredictable illness: A meta- 1 synthesis of qualitative research literature in Multiple Sclerosis 2 3 Hannah Rose Wilkinson (BSc Hons, MSc) and Roshan das Nair (BA, MSc, MPhil, PhD, CPsychol, AFBPsS) For all correspondence contact: Hannah Wilkinson Trainee Clinical Psychologist DClinPsy Course Faculty of HLSS University of Lincoln 1st Floor, Bridge House Brayford Pool Lincoln LN6 7TS