A mixed methods inquiry into the disclosure behaviours and experiences of children living wi

503 Pages·2016·11.34 MB·English

by  Ailbhe Benson

Breaking the Cycle of Invisibility:
A mixed methods inquiry into the
disclosure behaviours and experiences
of children living with epilepsy and their
parents

A thesis presented to Dublin City University for the Degree of Doctor in
Philosophy
By
Ailbhe M. Benson
MPsychSc (Hons), BA (Hons)

Supervisors:
Dr. Veronica Lambert, Dublin City University
Prof. Pamela Gallagher, Dublin City University
Dr. Amre Shahwan, Temple Street Children’s University Hospital

School of Nursing and Human Sciences, Dublin City University
September 2016
Declaration
I hereby certify that this material, which I now submit for assessment on the programme of
study leading to the award of Doctor in Philosophy is entirely my own work, that I have
exercised reasonable care to ensure that the work is original, and does not to the best of my
knowledge breach any law of copyright, and had not been taken from the work of others
save and to the extent that such work has been cited and acknowledged within the text of
my work.

Signed: ____________________ (Candidate) ID No.: 12212313 Date:________________

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Acknowledgements
Undertaking this PhD, although challenging at times, has been extremely interesting and
rewarding. There are a number of people that I am eternally grateful to who have contributed
enormously to ensuring that this project came to its realisation. First, I feel hugely privileged to
have worked with Dr. Veronica Lambert and Prof. Pamela Gallagher as my supervisors. Their
insightful appraisal of my work, rigorous exactitude of standard, patient instruction, ability to
challenge me, and thus bring out the best in me, and personal encouragement constantly served
to inspire me. Their endless support was crucial in this project moving beyond a mere concept
and coming to its fruition. I am quite sure that the experience and insights I have gained in
working with them will stand to me for the rest of my professional life. To them I wish to
extend my sincerest gratitude.
I would also like to offer particular thanks to Dr. Amre Shahwan. It is rare to meet a clinician so
invested in his patients; if only all children and parents were lucky enough to get to engage with
such a wonderful neurologist. His keenness to constantly ensure that everything ran as smoothly
as possible was critical in ensuring the success of this study. He constantly went above and
beyond to encourage and support not only the research, but also my personal and professional
development. I feel extremely thankful to have had the opportunity to work with such a brilliant
mind.
To Prof. Joan Austin, I must also express my appreciation. Her remarkable statistically-minded
brain, extensive and encyclopaedia-like knowledge about childhood epilepsy, personal
experiences with epilepsy, and kind nature were wonderful assets to this project.
Thanks are also due to the HRB who funded this research project; and to the neurology team in
TSCUH (in particular, Therese Nestor), Epilepsy Ireland (specifically Peter Murphy), Dr. Colin
Doherty and the staff in St. James’s hospital, and Dr. Siobhán Gormally and colleagues in Our
Lady of Lourdes Hospital, all of whom assisted with recruitment.
My mother, Máirín, and my father, Eddie, deserve particular mention. When I received a
diagnosis of epilepsy at 14 years of age, they instilled in me the belief that nothing was ever
beyond my reach. I couldn’t have been luckier to have parents who never saw my epilepsy as a
limitation and ensured that I didn’t see it as such either. I’m not sure either of them will ever
know quite how important this was in shaping me into the individual I am today. Mum, you
taught me everything I know and ignited in me a deep passion for education and learning -
although I’m not sure your original intention was for me to still be in education at 26 years of
age, so as usual you overachieved! Dad, I may tease you that you are fishing for compliments
when you claim to others that you played a pivotal role in my success in applying for this
position – I will finally admit it...your claims may have some merit! I’m not sure I ever would
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have come so far without your continuous belief in me (even when I didn’t believe in myself).
My brother Liam was also hugely supportive throughout this process. To him I must apologise
for those times when I just needed to vent; although a man of few words of advice, his advice is
always on the money. His capacity to always make me see the humorous side of stressful
situations is second to none. Additionally, my (huge) extended family warrant mention as they
have made this whole process easier – I love you all!
Thanks are also due to my friends for their enthusiasm and interest. The Muckross girls were
unwavering in their support – I would have been lost (and bored!) without them by my side
throughout this journey. Furthermore, I would like to acknowledge the support and friendship
of my fellow postgraduates in H101 over the last 4 years. Steph needs particular mention – I am
fortunate to say that whilst we started off as colleagues who initially acted so formally in our
correspondence with each other, all such formality has now been abandoned and I can now
count you amongst my close friends. Thank you for being my second set of ears and eyes, for
accompanying me on our many wild pursuits (e.g. our twelve runs for TSCUH/EI) and for
always joining me when I fall around the place laughing at something that only we could see the
humorous side of. I also want to express my particular gratitude to Alan. You never failed to
put a smile on my face throughout this process, no matter what stage I was at or how stressful
things seemed. Thank you for always being available to come for long walks with me at short
notice (when I needed to destress) and for always listening to me in my moments of madness.

Finally, I owe a particular debt of gratitude to the children and parents that participated in this
study. I am extremely thankful that you granted me the privilege of learning more about your
experiences of living with epilepsy. This study is dedicated to you.

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Table of Contents
Declaration.................................................................................................................................... i
Acknowledgements ..................................................................................................................... ii
List of Tables ............................................................................................................................ xiii
List of Figures ........................................................................................................................... xvi
List of Appendices ................................................................................................................... xvii
Operational Definitions ........................................................................................................... xix
Abbreviation List ..................................................................................................................... xxi
Abstract.....................................................................................................................................xxii
_____________________________________________________________________________
Chapter 1: Introduction ......................................................................................................... 1
1.0 Introduction ............................................................................................................................. 1
1.1 Epilepsy: Definitions, Epidemiology, Aetiology and Consequences ..................................... 1
1.2 Epilepsy-related Stigma .......................................................................................................... 3
1.3 Disclosing Epilepsy: A Concealable Stigmatised Identity ...................................................... 5
1.4 Thesis Conspectus ................................................................................................................... 7
_____________________________________________________________________________
Chapter 2: Literature Review ............................................................................................... 8
2.0 Introduction ............................................................................................................................. 8
2.1 Conducting a Systematic Review ........................................................................................... 8
2.2 Methods of the Systematic Review ......................................................................................... 9
2.2.1 Search Strategy ................................................................................................................ 9
2.2.2 Study Selection Criteria ................................................................................................... 9
2.2.3 Methods of the Review .................................................................................................. 10
2.2.4 Data Extraction and Data Synthesis ............................................................................... 11
2.2.5 Quality Assessment ........................................................................................................ 11
2.3 Results of the Systematic Review ......................................................................................... 12
2.3.1 Description of the Studies .............................................................................................. 14
2.3.2 Quality Appraisal of the Included Studies ..................................................................... 15
2.3.3 Quantitative Measures of Epilepsy Disclosure Behaviours ........................................... 18
2.3.4 Disclosure-Specific Findings across the Identified Studies ........................................... 19
2.3.5 Parent Disclosure Behaviours ........................................................................................ 19
2.3.6 Barriers/Enablers for Parent Disclosure ......................................................................... 21
2.3.7 Consequences of the Disclosure Management Strategies Adopted by Parents of CWE 22
2.3.8 Relationship between Parental Disclosure Management and Demographic, Clinical and
Psychosocial Factors ............................................................................................................... 23
2.3.9 CWE Disclosure Behaviours ......................................................................................... 23
2.3.10 Barriers/Enablers for CWE Disclosure ........................................................................ 24
2.3.11 Consequences of the Disclosure Management Strategies Adopted by CWE .............. 25
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2.3.12 Relationship between CWE’s Disclosure Management and Demographic, Clinical and
Psychosocial Factors ............................................................................................................... 25
2.4 Discussion of the Systematic Review Findings .................................................................... 26
2.4.1 Disclosure Behaviours and their Situational Context .................................................... 26
2.4.2 The Consequences of Specific Disclosure Management Strategies ............................... 28
2.4.3 Quantitative Measures of Disclosure ............................................................................. 28
2.4.4 Contextualising the Evidence......................................................................................... 28
2.4.5 Strengths and Limitations of the Review ....................................................................... 29
2.5 Conclusions of the Systematic Review ................................................................................. 30
_____________________________________________________________________________
Chapter 3: Methodology ....................................................................................................... 32
3.0 Introduction ........................................................................................................................... 32
3.1 Study Aims............................................................................................................................ 32
3.2 Research Questions ............................................................................................................... 32
3.3 Mixed Methods Research ..................................................................................................... 33
3.3.1 Pragmatism .................................................................................................................... 34
3.3.2 The Advantages and Disadvantages of Mixed Methods Research ................................ 35
3.4 Present Study Design ............................................................................................................ 36
3.4.1 Rationale for Using a Sequential Exploratory Design ................................................... 36
3.4.1.1 Type of Design ........................................................................................................ 36
3.4.1.2 Timing of Phases ..................................................................................................... 37
3.4.1.3 Weighting of Phases ............................................................................................... 38
3.4.1.4 Data Integration Approach ...................................................................................... 38
3.4.1.5 The Advantages and Disadvantages of a Sequential Exploratory Design .............. 38
3.4.1.6 Summary ................................................................................................................. 39
3.5 Conclusions ........................................................................................................................... 39
_____________________________________________________________________________
Chapter 4: Phase One: Qualitative Method ..................................................................... 41
4.0 Introduction ........................................................................................................................... 41
4.1 Design ................................................................................................................................... 41
4.2 Aims and Objectives for Phase One ..................................................................................... 41
4.2.1 Aim of Phase One .......................................................................................................... 41
4.2.2 Objectives for Phase One ............................................................................................... 41
4.3 Participants ............................................................................................................................ 42
4.3.1 Inclusion and Exclusion Criteria .................................................................................... 42
4.4 Procedure .............................................................................................................................. 43
4.4.1 Ethical Approval ............................................................................................................ 43
4.4.2 Interview Schedule Development .................................................................................. 43
4.4.3 Recruitment .................................................................................................................... 44
4.4.4 Conducting the Interviews ............................................................................................. 45
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4.5 Ethical Considerations .......................................................................................................... 46
4.5.1 Beneficence .................................................................................................................... 46
4.5.2 Non-maleficence ............................................................................................................ 47
4.5.3 Autonomy/Self-determination ....................................................................................... 47
4.5.4 Anonymity ..................................................................................................................... 48
4.6 Data Analysis ........................................................................................................................ 48
_____________________________________________________________________________
Chapter 5: Phase One: Qualitative Findings ................................................................... 49
5.0 Introduction ........................................................................................................................... 49
5.1 Overview of Participants, Recruitment & Interview Setting ................................................ 49
5.1.1 Sample Description: Demographics and Clinical Characteristics.................................. 49
5.1.2 Recruitment Source ........................................................................................................ 51
5.1.3 Interview Setting ............................................................................................................ 52
5.2 Child Findings ....................................................................................................................... 53
5.2.1 Children’s Disclosure Management Strategies .............................................................. 55
5.2.1.1 Concealment ........................................................................................................... 55
5.2.1.2 Open and Voluntary Disclosure .............................................................................. 55
5.2.1.3 Preventive Disclosure ............................................................................................. 55
5.2.1.4 Selective Disclosure ................................................................................................ 56
5.2.1.5 Unplanned Revelations ........................................................................................... 56
5.2.1.6 Indirect Telling (via others) .................................................................................... 56
5.2.2 Disclosure Targets for Children ..................................................................................... 57
5.2.2.1 Extended Family ..................................................................................................... 57
5.2.2.2 Peers ........................................................................................................................ 58
5.2.2.3 School Personnel ..................................................................................................... 61
5.2.2.4 Healthcare Professionals ......................................................................................... 62
5.2.2.5 Sports Team Coaches and/or Instructors of Extra-Curricular Activities ................ 62
5.2.2.6 Adults vs. Children ................................................................................................. 63
5.2.3 Content of Child Disclosure Exchanges ........................................................................ 63
5.2.3.1 Descriptions of Epilepsy and Seizures .................................................................... 63
5.2.3.2 Impact of Epilepsy on the Child ............................................................................. 65
5.2.3.3 Hospital Appointments ........................................................................................... 66
5.2.3.4 Medication and/or Other Epilepsy Treatments ....................................................... 66
5.2.4 Situational Context of Disclosure for the Child ............................................................. 66
5.2.4.1 In Secure Relationships with Disclosure Targets.................................................... 67
5.2.4.2 Cues Make the Invisible Condition Visible ............................................................ 67
5.2.4.3 Others are Curious about Epilepsy .......................................................................... 68
5.2.4.4 In an Environment where the Topic of Disability and/or Epilepsy is Salient ......... 68
5.2.4.5 The Mood of the Disclosure Target is Deemed Appropriate .................................. 69

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5.2.5 CWE’s Perceived Barriers to Disclosure ....................................................................... 69
5.2.5.1 CWE’s Desire for Normalcy ................................................................................... 71
5.2.5.2 Out of Sight but in the Mind ................................................................................... 72
5.2.5.3 Contending with Negative Responses to Disclosure .............................................. 74
5.2.5.4 The Complexity of Epilepsy ................................................................................... 75
5.2.5.5 Self and Others’ Perceptions of Epilepsy ............................................................... 76
5.2.6 Children’s Perceived Enablers of Disclosure ................................................................. 78
5.2.6.1 CWE’s Positive Perceptions of and Attitudes toward Epilepsy .............................. 78
5.2.6.2 Open and Positive Family Communication about Epilepsy ................................... 78
5.2.6.3 Others Reacting Positively to Disclosure ................................................................ 79
5.2.6.4 The Child’s Seizure Characteristics ........................................................................ 79
5.2.6.5 Getting Help with Disclosure .................................................................................. 80
5.3 Parent Findings ..................................................................................................................... 80
5.3.1 Parental Disclosure Management Strategies .................................................................. 82
5.3.1.1 Concealment ........................................................................................................... 82
5.3.1.2 Open and Voluntary Disclosure .............................................................................. 82
5.3.1.3 Preventive Disclosure ............................................................................................. 82
5.3.1.4 Selective Disclosure ................................................................................................ 83
5.3.1.5 Unplanned Revelations ........................................................................................... 83
5.3.2 Disclosure Targets for Parents ....................................................................................... 84
5.3.2.1 Extended Family ..................................................................................................... 84
5.3.2.2 Peers ........................................................................................................................ 85
5.3.2.3 School Personnel ..................................................................................................... 85
5.3.2.4 Healthcare Professionals ......................................................................................... 86
5.3.2.5 Caregivers ............................................................................................................... 87
5.3.2.6 Other Families ......................................................................................................... 87
5.3.2.7 Sports Coaches and/or Instructors of Extra-Curricular Activities .......................... 88
5.3.2.8 Parents of Children with Medical Conditions ......................................................... 88
5.3.2.9 Employers and Work Colleagues ............................................................................ 88
5.3.2.10 Sources of Help and/or Support ............................................................................ 89
5.3.3 Content of Parental Disclosure Exchanges .................................................................... 89
5.3.3.1 The Child’s Specific Seizure Symptomatology ...................................................... 89
5.3.3.2 Seizure First Aid Protocols ..................................................................................... 90
5.3.3.3. Impact of Epilepsy (Beyond Seizures) ................................................................... 91
5.3.3.4 Emotional Impact of the Diagnosis ......................................................................... 93
5.3.4 Situational Context of Parental Disclosure .................................................................... 94
5.3.4.1 Child Entering a New Environment ........................................................................ 94
5.3.4.2 Parents Perceive there are Risks for the Child ........................................................ 95
5.3.4.3 Others Raise the Topic of and/or Pose Questions about Epilepsy .......................... 95
5.3.4.4 Children’s Difficulties Arise during Conversation with Others ............................. 96
5.3.4.5 Seizure Occurrences ................................................................................................ 97
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5.3.4.6 Hospital Appointments ........................................................................................... 97
5.3.4.7 In the Presence of Others with Experience of Epilepsy .......................................... 97
5.3.4.8 Periods of Emotional Struggle due to the Child’s Diagnosis .................................. 98
5.3.5 Parents’ Perceived Barriers to Disclosure ...................................................................... 98
5.3.5.1 Seeking Normalcy for the Child ........................................................................... 100
5.3.5.2 The Invisibility of Epilepsy................................................................................... 101
5.3.5.3 Negative Reactions to Disclosure ......................................................................... 102
5.3.5.4 Contending with Poor Public Perceptions of Epilepsy ......................................... 104
5.3.5.5 Coming to Terms with the Diagnosis ................................................................... 105
5.3.6 Parents’ Perceived Enablers of Disclosure .................................................................. 108
5.3.6.1 Parental Perceptions of Epilepsy ........................................................................... 108
5.3.6.2 Disclosure Perceived as Enhancing Safety and Others’ Understanding of the Child
.......................................................................................................................................... 110
5.3.6.3 Positive Reactions to Disclosure ........................................................................... 110
5.3.6.4 The Child’s Seizure Characteristics ...................................................................... 111
5.3.6.5 Disclosure Perceived as an Educational Tool and a Method of Fighting Against
Epilepsy-related Stigma .................................................................................................... 111
5.3.6.6 Getting Used to It .................................................................................................. 112
5.3.6.7 Public Awareness and Media Coverage of Epilepsy ............................................ 112
5.4 Summary of Child and Parent Perspectives ........................................................................ 113
_____________________________________________________________________________
Chapter 6: Phase One: Discussion of the Qualitative Findings ................................. 115
6.0 Introduction ......................................................................................................................... 115
6.1 Disclosure Management Strategies ..................................................................................... 115
6.1.1 Child and Parent Perspectives: Disclosure Management Strategies ............................ 115
6.1.2 Child and Parent Perspectives on Disclosure Management Strategies: Similarities and
Differences ............................................................................................................................ 118
6.2 Disclosure Targets .............................................................................................................. 118
6.2.1 Child Perspectives: Disclosure Targets ........................................................................ 119
6.2.2 Parent Perspectives: Disclosure Targets ...................................................................... 121
6.2.3 Child and Parent Perspectives on Disclosure Targets: Similarities and Differences ... 123
6.3 The Content of Disclosure Exchanges ................................................................................ 124
6.3.1 Child Perspectives: The Content of Disclosure Exchanges ......................................... 124
6.3.2 Parent Perspectives: The Content of Disclosure Exchanges ........................................ 125
6.3.3 Child and Parent Perspectives on the Content of Disclosure Exchanges: Similarities and
Differences ............................................................................................................................ 126
6.4 The Situational Context of Disclosure Exchanges .............................................................. 127
6.4.1 Child Perspectives: The Situational Context of Disclosure Exchanges ....................... 127
6.4.2 Parent Perspectives: The Situational Context of Disclosure Exchanges ...................... 128
6.4.3 Child and Parent Perspectives on the Situational Context of Disclosure Exchanges:
Similarities and Differences .................................................................................................. 128

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6.5 Barriers to Disclosure ......................................................................................................... 130
6.5.1 Child Perspectives: Barriers to Disclosure ................................................................... 130
6.5.2 Parent Perspectives: Barriers to Disclosure ................................................................. 133
6.5.3 Child and Parent Perspectives on Barriers to Disclosure: Similarities and Differences
.............................................................................................................................................. 135
6.6 Enablers of Disclosure ........................................................................................................ 136
6.6.1 Child Perspectives: Factors that Enable Disclosure ..................................................... 136
6.6.2 Parent Perspectives: Factors that Enable Disclosure ................................................... 136
6.6.3 Child and Parent Perspectives on Enablers of Disclosure: Similarities and Differences
.............................................................................................................................................. 137
6.7 Strengths and Limitations of Phase One ............................................................................. 139
6.8 Implications for Phase Two ................................................................................................ 140
6.9 Conclusions ......................................................................................................................... 141
_____________________________________________________________________________
Chapter 7: Phase Two: Quantitative Method ............................................................... 143
7.0 Introduction ......................................................................................................................... 143
7.1 Design ................................................................................................................................. 143
7.2 Aims, Objectives and Hypotheses for Phase Two .............................................................. 143
7.2.1 Aim of Phase Two ....................................................................................................... 143
7.2.2 Objectives for Phase Two ............................................................................................ 143
7.2.3 Hypotheses for Phase Two ........................................................................................... 144
7.3 Participants .......................................................................................................................... 147
7.3.1 Inclusion and Exclusion Criteria .................................................................................. 147
7.4 Procedure ............................................................................................................................ 148
7.4.1 Ethical Approval .......................................................................................................... 148
7.4.2 Recruitment .................................................................................................................. 148
7.5 Designing the Surveys ........................................................................................................ 149
7.5.1 Pre-validated Child Instruments ................................................................................... 150
7.5.2 Pre-validated Parent Instruments ................................................................................. 152
7.5.3 Child and Parent Epilepsy Disclosure .......................................................................... 153
7.6 Piloting the Surveys ............................................................................................................ 155
7.7 Data Analysis ...................................................................................................................... 156
7.8 Ethical Considerations ........................................................................................................ 157
_____________________________________________________________________________
Chapter 8: Phase Two: Quantitative Results ................................................................ 158
8.0 Introduction ......................................................................................................................... 158
8.1 Sample Characteristics, Survey Distribution Details and Response Rates ......................... 158
8.1.1 CWE: Demographic Profile and Seizure Characteristics ............................................. 158
8.1.2 Parents: Demographic Profile and Child Seizure Characteristics ................................ 160
8.1.3 Participating Dyadic Families ...................................................................................... 164
8.1.4 Survey Distribution Details and Response Rates ......................................................... 164
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