C a r d i f f UNIVERSITY PR1FYSGOL Ca e RDy£> The Amniocentesis Dilemma Needs Assessment, Development and Field-Testing of a Theory-Based Decision Support Intervention Marie-Anne Durand Thesis submitted for the degree of Doctor of Philosophy at Cardiff University July 2009 UMI Number: U584B94 All rights reserved INFORMATION TO ALL USERS The quality of this reproduction is dependent upon the quality of the copy submitted. In the unlikely event that the author did not send a complete manuscript and there are missing pages, these will be noted. Also, if material had to be removed, a note will indicate the deletion. Dissertation Publishing UMI U584394 Published by ProQuest LLC 2013. Copyright in the Dissertation held by the Author. Microform Edition © ProQuest LLC. All rights reserved. This work is protected against unauthorized copying under Title 17, United States Code. ProQuest LLC 789 East Eisenhower Parkway P.O. Box 1346 Ann Arbor, Ml 48106-1346 The Amniocentesis Dilemma Needs Assessment, Development and Field-Testing of a Theory-Based Decision Support Intervention Marie-Anne Durand Thesis submitted for the degree of Doctor of Philosophy at Cardiff University July 2009 Supervisors: Professor Glyn Elwyn and Doctor Jacky Boivin 1 Abstract The amniocentesis dilemma: needs assessment, development and field-testing of a theory-based decision support intervention Background: Amniocentesis is the most common prenatal diagnostic procedure undertaken in the United Kingdom, usually performed after 15 completed weeks of pregnancy. The procedure is reported to have a 1 % risk of miscarriage and the results of the chromosome tests may require further decision making about whether to continue with the pregnancy. Deciding about amniocentesis is a complex and emotionally charged decision, often undertaken in a short period of time and, under current practice, with little systematic decision support. Decision Support Interventions, also known as Patient Decision Aids, have been developed to help individuals learn about the features and implications of their treatment or screening options while improving communication with their health professionals. Those interventions are specifically targeted at preference-sensitive decisions with significant harms, benefits and uncertainty, where no screening or treatment option is objectively better than the other. This thesis proposed to assess information and decision support needs of pregnant women undertaking amniocentesis testing and to design and field-test, in collaboration with pregnant women and health professionals, a theory-based Decision Support Intervention for amniocentesis testing (amnioDex). Methods: A multi-method approach was adopted that included a systematic review, theoretical review, and qualitative analysis to develop and pilot a theory-based intervention intended for pregnant women facing a decision to undertake amniocentesis testing. The content areas and themes to be covered in the intervention were determined by a literature review and needs assessment conducted with pregnant women and health professionals. The prototype development of amnioDex (amniocentesis decision explorer) was guided by theory and included heuristic-based deliberation tools. Incremental prototypes of amnioDex and embedded deliberation tools were field-tested with lay users, health professionals and pregnant women facing a decision to undertake amniocentesis, using the “think-aloud” technique. Results: The amnioDex intervention was developed over a period of two years and field-tested for eight months. Conclusion: Findings from this thesis showed that it was feasible to use theory to generate a Decision Support Intervention acceptable to women facing amniocentesis testing and to health professionals counselling them. Future research needs to evaluate the effectiveness of amnioDex in a randomised controlled trial and to examine methods for effectively transferring theory into practice. 1 APPENDIX 1: Specimen Layout for Thesis Summary and Declaration/Statements page to be included in a Thesis DECLARATION This work has not previously been accepted in substance for any degree and is not r any degree. (candidate) Date S\% e^ ^ a a A J ^ ' 2 U-1'0 n partial fulfillment of the requirements for the degree of i, MD, MPhil, PhD etc, as appropriate) (candidate) Date STATEMENT 2 This thesis is the result of my own independent work/investigation, except where otherwise stated. d by explicit references. (candidate) Date STATEMENT 3 I hereby give consent for my thesis, if accepted, to be available for photocopying and for inter- nd summary to be made available to outside organisations. (candidate) Date - BAR ON ACCESS APPROVED I hereby give consent for my thesis, if accepted, to be available for photocopying and for inter- library loans after expiry of a bar on access approved bv the Graduate Development Committee. Signed (candidate) Date Acknowledgements I would like to thank all the patients and health professionals who kindly took part in the needs assessment and development of amnioDex. This research would not have been possible without the exemplary support of the midwives at the antenatal clinic of the University Hospital of Wales. I am very grateful to the members of the amnioDex steering group for their support, advice and expertise: Lynne Taylor, Doctor Annie Procter, Doctor Andrew Dawson and Jane Fisher to name a few. 1 am extremely grateful to my supervisors, Professor Glyn Elwyn and Doctor Jacky Boivin for their continued support, advice and ever so inspiring research interests and ideas. They were courageous enough (some might even say adventurous) to let me undertake a PhD as an international student and I am forever indebted to them for their unwavering patience and sense of humour, on those many occasions where I got lost in translation. Their ideas and opinions have shaped my career aspirations, vision and understanding of research in healthcare. Three years on, I maintain that I could not have chosen better supervisors. My PhD studentship was funded by the Sir Hailey Stewart Trust, and I would like to sincerely thank them for giving me the opportunity to embark on the most rewarding as well as challenging journey that has been my PhD. I would like to thank Mareike Stiel, for her continuous support and help reviewing articles and coding interview transcripts in the first and second year of my PhD. 2 I am grateful to my colleagues in the Department of Primary Care and Public Health and in the School of Psychology, for their insightful comments and continued support (Rhodri Evans, Nathalie Joseph-Williams, Paulina Bravo and Stephanie Sivell to only cite a few). I would like to thank Joanna and all my friends on the other side of the Channel for their support, optimism and genuine or feigned interest in my PhD. Finally, I am forever grateful to Will, and to my family, for their enduring patience, support and love and for tolerating the presence of my laptop at all times, including on the beach. This thesis is dedicated to my parents, Solange and Jacques, and to my sister, Natacha. 3 Contents Abstract 1 Acknowledgements 2 Contents 4 List of Tables 9 List of Figures 10 Chapter 1 11 1 Introduction and Thesis Overview 11 1.1 Involving Patients in Medical Decision Making: the Patient Centred Approach II 12 The Amniocentesis Dilemma or How to Cope With Clinical Equipoise 13 1.3 Decision Support Interv entions 16 1.4 Thesis 0 \er\iew 20 1.4.1 Identifying Evidence and Theoretical Basis (Chapters 2, 3 and 4) 20 1.4.2 Needs Assessment and Prototype Development (Chapters 5, 6 and 7) 21 1.4.3 Qualitative Field-Testing of AmnioDex and Deliberation Components (Chapters 8 and 9) 22 Chapter 2 24 2 Theoretical Review 24 2 1 Introduction 24 2.2 Methods 25 2 3 Results 26 2.3.1 Prevalence of “Atheoretical” Interventions 27 2.3.2 Normative Theories of Decision Making 27 2.3.3 The Ottawa Decision Support Framework 31 2.3.4 Combination of Decision Making Theories 33 2.3.5 Cognitive and Social Theories of Decision Making 34 2.3.6 Behavioural Theories 36 2 4 Discussion 3 7 2 5 Conclusion ^9 Chapter 3 41 3 Amniocentesis Testing 4* 3 I Introduction 3.2 Screening for Down’s Syndrome *2 4 3.3 The Amniocentesis Procedure and Chromosome Tests 46 3.4 Existing Information and Decision Support Arrangements 49 3.5 Summary• 51 Chapter 4 52 4 Review of Decision Support Interv entions for Amniocentesis 52 4.1 Introduction 52 4.2 Methods 53 4.2.1 Definitions 53 4.2.2 Literature Search Strategy 53 4.2.3 Study Inclusion and Exclusion Criteria 54 4.2.4 DES1 Assessment 54 4.3 Results 57 4.3.1 Selection of DESIs 57 4.3.2 How Many DESIs for Amniocentesis Exist and What Are Their Aims? 58 4.3.3 Do DESIs for Amniocentesis Meet Published Quality Standards? 62 4.3.4 What is the Effectiveness of Existing DESIs? 66 4.4 Discussion 68 4.5 Conclusion 7/ Chapter 5 ^ 5 User Perspective 73 5.1 Introduction 7J 5.2 Methods ^4 5.2.1 Participants 74 5.2.2 Data Collection 75 5.2.3 Data Analysis 75 5.3 Results ^6 5.3.1 Information Needs 77 5.3.2 Emotions and Decision Support 5.3.3 Reasons for Accepting/Declining an Amniocentesis 81 5.3.4 Making a Decision **2 5.3.5 Synthesis ^ 5.4 Discussion 5.5 Conclusion ^ Chapter 6 ^ 5 6 Stakeholder Analysis 9 0 6.1 Introduction gg 6.2 Methods gj 6.2.1 Participants 91 6.2.2 Data Collection 91 6.2.3 Data Analysis 92 6.3 Results g2 6.3.1 Information Needs 92 6.3.2 Emotions and Decision Support 96 6.3.3 Reasons for Accepting/Declining an Amniocentesis 97 6.3.4 Professionals’ Perceptions of the Amniocentesis Decision 99 6.3.5 Counselling