Cancer Related, Lower Limb Lymphoedema During the Intensive and Maintenance Phases of Complex Decongestive Therapy and Its’ Impact on Their Lives

Exploring the experiences of patients with primary and secondary, non- cancer related, lower limb lymphoedema during the intensive and maintenance phases of Complex Decongestive Therapy and its’ impact on their lives.

Mary Costello, BScN, MScN.

School of Nursing and Midwifery

RCSI

A thesis submitted to the School of Postgraduate Studies, Faculty of Medicine and Health Sciences, Royal College of Surgeons in Ireland,

In fulfilment of the Degree of Doctor of Philosophy

Supervisors: Dr Declan Patton

Professor Zena Moore

March 2019

Declaration

I declare that this thesis, which I submit to RCSI for examination in consideration of the award of Doctor of Philosophy is my own personal effort. Where any of the content presented is the result of input or data from a related collaborative research programme this is duly acknowledged in the text such that it is possible to ascertain how much of the work is my own. I have not already obtained a degree in RCSI or elsewhere on the basis of this work. Furthermore, I took reasonable care to ensure that the work is original, and, to the best of my knowledge, does not breach copyright law, and has not been taken from other sources except where such work has been cited and acknowledged within the text.

Signed ______

Student No. 10100768

Date: 02/04/2019

Table of Contents

Declaration ...... 2 Table of Contents ...... 3 List of Abbreviations ...... 9 List of Figures ...... 10 List of Tables...... 11 Presentations arising from the dissertation ...... 12 Summary ...... 14 Acknowledgements ...... 17 Chapter 1 Introduction ...... 18 1.1 Background ...... 18 1.2 Research Questions ...... 21 1.3 Purpose of the study ...... 21 1.4 Research Aims and Objectives ...... 22 1.5 Presentation of the thesis ...... 22 1.6 Summary ...... 23 Chapter 2 Literature Review ...... 25 2.1 Introduction ...... 25 2.2 Search Strategy ...... 25 2.3 Aetiology of Lymphoedema ...... 26 2.3.1 Introduction ...... 26 2.3.2 Anatomy of the Lymphatic System ...... 27 2.3.3 Scientific Advances in Lymphology ...... 28 2.4 Classification and staging of Lymphoedemas ...... 29 2.4.1 Introduction ...... 29 2.4.2 Primary and Secondary Lymphoedema ...... 30 2.4.3 Summary ...... 32 2.5 Lymphoedema – Epidemiology ...... 32 2.5.1 Introduction ...... 32 2.5.2 Definitions ...... 33 2.5.3 Prevalence of lymphoedema ...... 34 2.5.4 Primary Lymphoedema ...... 36 2.5.5 Non-Cancer Related secondary lymphoedemas ...... 36

2.5.6. Summary ...... 38 2.6 Complex Decongestive Therapy ...... 38 2.6.1 Introduction ...... 38 2.6.2 Manual lymphatic drainage ...... 39 2.6.3 Compression Bandaging and Hosiery ...... 41 2.6.4 Skin care and Exercise ...... 44 2.6.5 Complex Decongestive Therapy ...... 46 2.6.6 Summary ...... 48 2.7 Impact of lymphoedema on health and social gain ...... 48 2.7.1 Introduction ...... 48 2.7.2 Economic impact of lymphoedema on service provision ...... 49 2.7.3 The economic impact of lymphoedema on the patient ...... 50 2.8 Lymphoedema and Quality of Life ...... 52 2.8.1 Summary ...... 55 2.9 Self-management of Lymphoedema ...... 55 2.9.1 Summary ...... 59 Chapter 3 Methodology and Methods ...... 60 3.1 Introduction ...... 60 3.2 Research Aims and Objectives ...... 60 3.3 Justification for choice of methodology ...... 61 3.4 Mixed Methods Research ...... 63 3.5 Definitions of Mixed Methods Research ...... 64 3.6 Classification of Mixed Method Designs ...... 65 3.7 Explanatory Sequential Design ...... 65 3.8 Philosophy underpinning Research Methodologies ...... 67 3.9 Pragmatism and Mixed Methods Research ...... 68 3.10 Methodology ...... 69 3.11 Sample ...... 73 3.12 Study subjects and the setting ...... 76 3.13 Data Collection – Part 1. Quantitative Phase ...... 79 3.14 Data Collection – Part 2 Qualitative Phase ...... 83 3.15 Data Analysis – Part1 Quantitative ...... 87 3.16 Data Analysis – Part 2 Qualitative ...... 88 3.17 Pilot Study ...... 90

3.17.1 Data Collection Part 1 Quantitative...... 90 3.17.2 Data Collection Part 2 Qualitative ...... 91 3.17.3 Data Analysis Part 1 Quantitative ...... 92 3.17.4 Data Analysis Part 2 Qualitative ...... 92 3.18 Reliability and Validity ...... 93 3.18.1 Reliability and validity in Mixed Methods Research ...... 93 3.18.2 Veracity ...... 93 3.18.3 Consistency ...... 94 3.18.4 Applicability ...... 95 3.18.5 Neutrality ...... 96 3.19 Ethical Considerations ...... 96 3.19.1 Risks ...... 98 3.19.2 Data Protection ...... 98 3.20 Summary ...... 99 Chapter 4 Results - Quantitative Findings ...... 100 4.1 Demographic Findings ...... 100 4.2 Quality of Life ...... 105 4.3 Quality of Life scoring in the domain of function ...... 109 4.4 Quality of Life Scoring in the domain of appearance ...... 114 4.5 Quality of Life Scoring in the domain of Symptoms ...... 116 4.5.1 Quality of Life scoring in the domain of Mood ...... 119 4.5.2 Summary of QoL changes in the 4 domains from baseline to week 24 ...... 122 4.6 Limb Circumferential and Volume changes ...... 124 4.7 Self Efficacy ...... 128 4.7.1 Short Form for Self-Efficacy for Managing Daily Activities ...... 129 4.7.2 Short Form for Self-Efficacy for Managing Emotions ...... 131 4.7.3 Short Form for Self-Efficacy for Managing Social Interactions ...... 133 4.7.4 Short Form for Self-Efficacy for Managing Symptoms ...... 134 4.7.5 Summary of Self-efficacy scoring ...... 136 Chapter 5 Results - Qualitative Findings of Interviews ...... 137 5.1 Thematic Analysis ...... 137 5.2 Familiarisation with data ...... 138 5.3 Generating Initial Codes ...... 138 5.4 Searching for themes ...... 140 5

5.5 Reviewing Themes ...... 141 5.6 Defining and naming themes ...... 142 5.7 Theme One: Health professionals lack of knowledge in the field of lymphoedema ...... 143 5.7.1 Delayed diagnosis ...... 143 5.7.2 Concept of low importance of lower limb oedema ...... 144 5.7.3 Absence of referral pathways ...... 145 5.7.4 Absence of patient information ...... 146 5.8 Theme 2: The impact of lymphoedema: psychosocial, physical and financial ...... 147 5.8.1 The psychosocial impact of lymphoedema ...... 147 5.9 Physical impact of lymphoedema ...... 151 5.9.1 Reduced mobility and symptoms ...... 152 5.10 Financial impact of Lymphoedema ...... 152 5.10.1 Cost of treatment and garments ...... 153 5.10.2 Inequality of services ...... 154 5.10.3 Loss of earnings ...... 154 5.11 Therapeutic Benefits of CDT ...... 155 5.11.1 Compression: A means to an end ...... 155 5.11.2 Positive physical outcomes ...... 157 5.11.3 Positive Emotions ...... 158 5.12 Summary of Qualitative Analysis of interview data ...... 158 Chapter 6 Results - Qualitative Findings of Diary ...... 160 6.1 Maintenance Diary ...... 160 6.2 Compression Hosiery ...... 161 6.3 Skin care ...... 163 6.4 Exercise ...... 163 6.5 Simple Lymphatic Drainage ...... 164 6.6 Explanation of QoL and limb volume changes using diary data ...... 165 6.6.1 Participant ID 9 Diary Analysis ...... 165 6.6.2 Participant ID 10 Diary Analysis ...... 166 6.6.3 Participant ID 5 Diary Analysis ...... 167 6.7 Self-efficacy and diary analysis ...... 167 6.8 Summary of qualitative findings of the participant diary ...... 169 Chapter 7 Discussion ...... 171

7.1 Introduction ...... 171 7.2 Demographic Profile ...... 171 7.2.1 Age ...... 171 7.2.2. Aetiology and stage of lymphoedema...... 172 7.2.3 Body Mass Index ...... 173 7.2.4 Treatment history of participants before commencing the study ..... 174 7.3 Key Findings of the research study...... 175 7.3.1 Research Question 1 and key findings ...... 176 7.3 2 Complex Decongestive Therapy does improve quality of life and reduce limb volume: ...... 176 7.3.3 Summary ...... 182 7.4 Research Question No. 2 and No.3 and Key Findings ...... 183 7.4.1 The patient perspective in 2018 in Ireland remains unchanged since it was last explored in 2010 ...... 183 7.4.2 Compression bandaging, a means to an end...... 185 7.4.3 Heat can be a factor that causes patients with lower limb lymphoedema to be non-concordant with wearing compression hosiery . 187 7.4.4 Elevated BMI has a negative influence on self-efficacy in lymphoedema self-care ...... 190 7.5 Summary ...... 194 Chapter 8 Conclusion ...... 197 8.1 Introduction ...... 197 8.2 Strengths of the study ...... 197 8.3 Limitations of the study ...... 198 8.4 Plans for dissemination ...... 199 8.5 Recommendations for Nursing Practice ...... 200 8.6 Recommendations ...... 202 8.6.1 Recommendations for Education ...... 202 8.6.2 Recommendations for future research ...... 203 8.6.3 Recommendations for Policy ...... 204 8.7 Reflection ...... 204 8.8 Conclusion to the Thesis ...... 206 References...... 207 Appendices ...... 222 Appendix 1: Ethical Approval Letter ...... 223

Appendix 2: Ethical Approval RCSI January 2016 ...... 224 Appendix 3: Ethical Approval RCSI April 2017 ...... 225 Appendix 4: Patient Information Leaflet ...... 226 Appendix 5: Patient Consent Form ...... 231 Appendix 6: Letter to Director of Public Health Nursing ...... 233 Appendix 7: Response to query from Tullamore Ethics Committee ...... 234 Appendix 8: Research Ethics Approval...... 235 Appendix 9: Lymphoedema Handout...... 237 Appendix 10: Simple Lymphatic drainage handout ...... 240 Appendix 11: Lymphoedema Measurements – Week 1 ...... 243 Appendix 12: LYMQOL LEG ...... 245 Appendix 13: LYMQOL LEG scores ...... 248 Appendix 14: PROMIS Item Bank v1.0 Short Form ...... 251 Appendix 15: Grant Approval Tullamore ...... 253 Appendix 16: Grant Amendment ...... 255 Appendix 17: Interview Sample ...... 257 Appendix 18: Searching for Themes...... 268 Appendix 19: Diary ...... 269

List of Abbreviations

BCRL Breast Cancer Related Lymphoedema

CDT Complex Decongestive Therapy

CREST Clinical Resource Efficiency Support Team

DPHN Director of Public Health Nursing

EWMA European Wound Management Association

GP General Practitioner

HSE Health Service Executive

ICHN Institute of Community Health Nursing

ISL International Society of Lymphology

MLD Manual Lymphatic Drainage

NLLUC Nurse Led Leg Ulcer Clinic

NMPDU Nursing and Midwifery Planning and Development Unit

QoL Quality of Life

SLD Simple Lymphatic Drainage

WMAOI Wound Management Association of Ireland

List of Figures

Figure 3.1 Explanatory Sequential Design of This Study ...... 67 Figure 5.1 Braun and Clarke (2006) Six Phase Thematic Framework ...... 137 Figure 5.2 Thematic Map ...... 141

List of Tables

Table 3.1 Interview Schedule ...... 71 Table 3.2 Maintenance Diary Daily Record ...... 72 Table 4.1 Baseline Characteristics (n=20) ...... 101 Table 4.2 History of oedema duration/ initial diagnosis and treatment ...... 103 Table 4.3 Treatment Modalities of participants (n=11) ...... 104 Table 4.4 Overall QoL changes week 1 to week 8, week 8 to week 24 and week 1 to week 24 ...... 106 Table 4.5 Baseline QoL scoring in the domain of function ...... 109 Table 4.6 Characteristics of 6 participants who scored 1-2 on the Function Domain at baseline...... 110 Table 4.7 Characteristics of participants scoring 3.1 to 4 in the Function Domain at baseline ...... 111 Table 4.8 Percentage change in QoL in the domain of function baseline to week 24 ...... 112 Table 4.9 Baseline QoL scoring in the domain of appearance ...... 114 Table 4.10 Percentage change in QoL scoring in the appearance domain from baseline to week 8...... 115 Table 4.11 Baseline QoL scoring in the domain of appearance ...... 117 Table 4.12 Percentage change in QoL scoring in the symptom domain from baseline to week 24...... 118 Table 4.13 Baseline QoL scoring in the domain of mood ...... 120 Table 4.14 The percentage of change in QoL in the domain of mood from baseline to week 2...... 121 Table 4.15 Percentage change in limb volume week 1/ week 8/ week 24 ...... 125 Table 4.16 Cumulative Scores – Self-efficacy for Managing Daily Activities ... 130 Table 4.17 Cumulative Scores - Self-efficacy for managing emotions ...... 132 Table 4.18 Cumulative Scores – Self-efficacy for Managing Social Interactions ...... 133 Table 4.19 Cumulative Scores – Self-efficacy for Managing Symptoms ...... 135 Table 5.1 Example of Initial Coding Framework...... 139 Table 5.2 Final Themes and Subthemes ...... 142 Table 7.1 Self-efficacy and BMI of 5 participants ...... 192

Presentations arising from the dissertation

Oral Presentations:

Exploring the experiences of patients with primary and secondary, non-cancer related lower limb lymphoedema during the intensive and maintenance phases of Complex Decongestive Therapy and its’ impact on their lives.

Wounds UK, Harrogate, Nov 2018

Nursing and Midwifery Planning and Development Unit, Research Conference, Tullamore, Co-Laois. March 9th, 2018.

Poster Presentations:

European Wound Management Association (EWMA) Conference, Krakow, Poland. May 9th -11th, 2018.

Nursing & Midwifery Planning & Development Unit Conference. March 9th, 2018

Community Nursing: Advancing the Quality Agenda The Institute of Community Nursing Annual Conference. May 18th, 2016

Wounds UK, Harrogate, 2017

Royal College of Surgeons in Ireland Research Day, Feb 25th, 2016

Summary Introduction Lymphoedema is an incurable, progressive condition, which results in the swelling of a limb or limbs and impairs mobility (Lymphatic Framework, 2006). The psychosocial impact of lower limb lymphoedema is also devastating as body image and quality of life are greatly affected (Lasinski et al, 2012). Although lymphoedema is a chronic condition, it is manageable through treatment modalities which include manual lymphatic drainage, compression bandaging, compression hosiery, skin care and exercise, otherwise known as Complex Decongestive Therapy (CDT) (Todd, 2012). The area of lower limb, non-cancer related, lymphoedema is poorly resourced and poorly researched, therefore this thesis proposes to explore treatment outcomes for patients with non-cancer related lower limb lymphoedema during the intensive and maintenance phases of CDT,examine their experiences of living with lymphoedema and the challenges of lifelong self-care maintenance.

Research Questions The research questions for the study were as follows; 1. What is the impact of Complex Decongestive Therapy (CDT) as a treatment for primary and secondary lower limb lymphoedema during the intensive and maintenance phases of CDT, in relation to; a. Quality of life? b. Limb circumferential and volumatic changes? c. Self-efficacy in managing lymphoedema self care? 2. What are the patients’ experiences of living with lymphoedema? 3. What are the patients’ experiences of the four elements of CDT used during the intensive and maintenance phases of treatment?

Aims & Objectives The aims of this study were twofold; 1. To examine the impact of Complex Decongestive Therapy (CDT) as a treatment for primary and secondary, non-cancer related, lower limb lymphoedema during the intensive and maintenance phases of CDT;

2. To explore the experiences of patients with primary and secondary, non- cancer related lower limb lymphoedema during the intensive and maintenance phases of CDT.

The objectives of the study are as follows: 1. To determine the impact of CDT on limb circumference and quality of life. 2. To explore the patients’ experience of living with lymphoedema. 3. To investigate the patients’ reported experience of the four elements of CDT. 4. To examine the patients’ reported maintenance regimen of self-care and its resulting effect on limb volume and quality of life.

Methods A mixed methodology, using both quantitative and qualitative data in a sequential manner was used. This study was divided into 3 parts, Part 1 a quantitative approach was used to measure; a) Limb volume changes. b) Quality of Life changes c) Self-efficacy Part 2 –A qualitative approach was used to analyse data from face to face, semi-structured interviews with 18 participants exploring the experiences of patients living with lymphoedema and their experiences of Complex Decongestive Therapy. Part 3 – Descriptive analysis of 18 participant diaries was used during maintenance self-care, over a 4-month period, to examine the participants’ experiences of lymphoedema self care.

Results A purposive sample of 20 participants were recruited to this study All participants were diagnosesd with lower limb lymphoedema, 10 participants had primary lymphoedema and 10 participants had secondary, non-cancer related lymphoedema. The study setting was a lymphoedema clinic in the Midlands of Ireland, run by the researcher who is also a Tissue Viability Nurse and Manual Lymphatic Drainage Therapist.

The findings included a reduction in limb volume from baseline to week 8 during intensive therapy and then a fluctuation of oedema for 6 of the participants during the 4-month self-maintenance period. Quality of life improved for all participants from baseline to week 24. Thematic analysis of the interviews identified themes of lack of knowledge, delayed diagnosis, physical, psychosocial and financial burden of lymphoedema and positive therapeutic effect of treatment. The diary analysis identified self-care challenges that were influenced by elevated body mass index and carer support.

Conclusion Primary and secondary, non-cancer related lower limb lymphoedema is an area of lymphoedema that is poorly researched. It is also true to say that there is a sparsity of research in the area of lymphoedema self maintenance care. The aims of this research study were twofold, to explore patient outcomes with regard to limb volume, quality of life and self-efficacy during the intensive and maintenance phases of CDT and to examine the experiences of the patients living with lymphoedema, during their treatment and following on into self maintenance. The findings included reduction in limb volume during therapy, an improvement in quality of life and a fluctuation in self-efficacy that was influenced by elevated body mass index. Thematic analysis of interviews concluded that living with lymphoedema poses many challenges resulting from poor knowledge within the health professions and a lack of referral pathways and services. The success of lymphoedema self maintenance is greatly influenced by the patients physical ability and support structures, especially in the case of the patient with an elevated body mass index.

Acknowledgements I would like to acknowledge and thank my supervisors, Dr Declan Patton and Professor Zena Moore, who have encouraged and supported me throughout this project.

I would also like to acknowledge the funding that I received from the Nursing and Midwifery Practice and Development Unit, Tullamore, Co-Offaly.

I would like to thank my Director of Public Health Nursing, Joan Bourke, and Assistant Director of Public Health Nursing, Yvonne Delaney, for their continued support and encouragement.

I would like to thank my family for their continued understanding and encouragement throughout the years.

Chapter 1 Introduction

1.1 Background

Lymphoedema is an incurable, progressive condition, which results in the swelling of a limb, or limbs, due to the accumulation of protein rich fluids in the interstitial spaces of the tissues (Lymphoedema Framework, 2006). A patient with lymphoedema can experience pain, decreased mobility, and psychological distress (Cooper, 2014a). The lymphatic system may fail due to many reasons, such as, congenital malformation of the lymphatic system or a secondary element which may damage the lymphatics (Clinical Resource Efficiency Support Team (CREST), 2008).

Lymphoedema is classified as either primary, or secondary, depending upon the aetiology. Primary lymphoedema is caused by congenital abnormalities to the lymphatic system, which may be evident from infancy to later life. Secondary lymphoedema is caused by a trauma to the lymphatics following cancer treatment, surgery or infections (Twycross et al., 2000). Chronic venous insufficiency may also cause the overloaded lymphatic system to deteriorate in function (Todd, 2012).

Recently it has been identified that 90% of reabsorption of interstitial fluid occurs through the lymphatic system, which shows the importance of the lymphatic system to normal human functioning (Mortimer and Rockson, 2014, Levick and Michel, 2010).

Many authors have stated that there is a lack of reliable prevalence rates of lower limb lymphoedema due to poor recognition of lymphoedema and resulting under diagnosis (Moffatt et al., 2003, Cooper and Bagnall, 2016, Cooper, 2014a, Cooper, 2010).

Historically, the focus of education in nursing and medicine tended to lean heavily towards the emphasis on the cardiovascular system, with little time dedicated to the lymphatic system and the conditions that pertain to that system. A mixed methods research study conducted by Davies et al. (2012) demonstrated knowledge deficits by generalist practitioners, which included 18 nurses and GP’s, in the area of lymphoedema assessment and diagnosis, with only 14% having taken part in some form of lymphoedema education in the previous 5 years. Both groups felt that lymphoedema training needed to be a part of undergraduate health professional training.

The predominance of prevalence studies revolve around the area of cancer- related lymphoedema, with reported prevalence rates of 70% of patients in Canada who suffer with lymphoedema post breast and prostate cancer treatment (Keast et al., 2015). Meanwhile, in the UK, breast cancer related lymphoedema accounts for between 12% and 40% of lymphoedema incidence (Poage et al., 2008). Approximately, 200-300 million people worldwide, since records began, have experienced lymphoedema at some stage of their lives, with a significant incidence in the Far East as a result of parasitic infection (Wozniewski et al., 2001). Indeed, Lymphatic Filariasis, a parasitic infection transmitted by mosquitos, is endemic in India, affecting an estimated 23 million patients (Narahari et al., 2007). According to Gethin et al. (2012), a prevalence of 2.63% of patients attending a wound management and vascular clinic in Ireland had a diagnosis of lower limb lymphoedema. Interestingly, should the prevalence rates reported by Moffatt et al. (2003) of 1.3/1000 population be reflected on the population in Ireland, 5,500 cases of lymphoedema in the Irish population would be expected (Gethin et al., 2012).

Chronic oedema is a term used to describe “a group of conditions characterised by the presence of swelling within tissues of the body, caused by the accumulation of excess fluid within the interstitial space of the affected area. Oedema most commonly affects the lower or upper limbs, but may also affect midline structures such as the head and neck, trunk, breasts or genitalia” (National Lymphoedema Partnership, June 2015). This umbrella term groups together lymphoedema of all aetiologies and chronic oedema. The relevance of this grouping, for future prevalence data, is that prevalence figures could show significant disease burden in the area of chronic oedema, which in turn would provide policymakers with prevalence data that could assist in decisions about service provision.

The World Alliance for Wound and Lymphoedema Care (2010) predict a growing epidemic of non-communicable disease and increased life expectancy. 19

This will potentially lead to an increase in the incidence of wounds and lymphoedema as a complication of the associated risk factors of ageing and co- morbidities. Chronic oedema is a complication of the many co-morbidities that elderly patients display, such as heart failure, chronic venous insufficiency, reduced mobility, and renal disease. A more recent study by Moffatt et al. (2017) demonstrates an increasing prevalence of chronic oedema (3.93 per 1000 population) in a comparable urban population when compared to her 2003 study (Moffatt et al., 2003).

The psychosocial impact of lymphoedema is also devastating to the sufferer, as body image and quality of life are greatly affected. However, it has been found that quality of life outcomes improve with treatment and reduction of limb volume (Lasinski et al., 2012).

Although lymphoedema is a chronic condition, which is not curable at present, it is manageable through the initiation of specific treatment modalities which include manual lymphatic drainage (MLD), compression bandaging, compression hosiery, skin care and exercise, known collectively as Complex Decongestive Therapy (CDT) (Todd, 2012). The International Lymphoedema Framework (2006) has identified best practice in the treatment of lymphoedema, which includes a holistic, multidisciplinary approach. The management program required should be determined by the severity and site of the lymphoedema. During the Intensive Phase of therapy, CDT is performed by a trained practitioner daily for one hour over three to four weeks in order to reduce limb volume and improve joint range of motion. The Maintenance Phase requires the patient to perform simple lymphatic drainage massage techniques that, when combined with skin care, exercise, and the use of compression garments, will maintain the desired level of limb volume (International Society of Lymphology Executive Committee, 2013). The long-term success of the maintenance phase will ultimately depend on the concordance of the patient with the maintenance plan (Clinical Resource Efficiency Support Team (CREST), 2008).

1.2 Research Questions

The research questions for the study were as follows;

1. What is the impact of Complex Decongestive Therapy (CDT) as a treatment for primary and secondary lower limb lymphoedema during the intensive and maintenance phases of CDT, in relation to;

a. Quality of life?

b. Limb circumferential and volumatic changes?

c. Self-efficacy in managing lymphoedema self care?

2. What are the patients’ experiences of living with lymphoedema? 3. What are the patients’ experiences of the four elements of CDT used during the intensive and maintenance phases of treatment?

1.3 Purpose of the study

The majority of research is focused on the upper limb and cancer-related lymphoedema. A direct transfer of information from upper limb to lower limb is not possible due to the differences in size, volume, location, and function of the limb (Devoogdt et al., 2014). Therefore, a research study was completed which explored the experiences of patients with primary and secondary, non-cancer related, lower limb lymphoedema during the intensive and maintenance phases of Complex Decongestive Therapy and its impact on their lives. Currently in Ireland, there has been no research in the area of CDT outcome measures in a specific program of therapy. The measurement of treatment outcomes plays a very important role in the construction of evidence that is required for resource allocation and service development. This study measured limb volume and quality of life as indicators of the response of lower limb lymphoedema patients to a tailored CDT program. Chronic conditions require life-long commitment by the patient to be independent in self-care. Few studies have examined self- maintenance in this cohort of patients. This study will follow the patient’s maintenance self-care program, up to 6 months from initial assessment, and will 21 examine the barriers and challenges faced by the patients on a day to day basis and their impact on limb volume and quality of life.

1.4 Research Aims and Objectives

The aims of this study were twofold;

1. To examine the impact of Complex Decongestive Therapy (CDT) as a treatment for primary and secondary, non-cancer related, lower limb lymphoedema during the intensive and maintenance phases of CDT; 2. To explore the experiences of patients with primary and secondary, non- cancer related lower limb lymphoedema during the intensive and maintenance phases of CDT.

The objectives of the study are as follows:

1. To determine the impact of CDT on limb circumference and quality of life. 2. To explore the patients’ experience of living with lymphoedema. 3. To investigate the patients’ reported experience of the four elements of CDT. 4. To examine the patients’ reported maintenance regimen of self-care and its resulting effect on limb volume and quality of life.

1.5 Presentation of the thesis

Chapter 1 includes the introduction to the thesis and presents the literature review which has been divided into 6 headings, namely, the aetiology and classification of lymphoedema, epidemiology and prevalence, health and social gain, the role of Complex Decongestive Therapy (CDT) in the treatment of lower limb lymphoedema, quality of life and self-management of lymphoedema.

Chapter 2 presents the methodology and methods and is divided into three sections; the first section provides a review of the mixed methods research, the second section discusses the philosophical underpinnings of mixed methods research, followed by a section discussing the research methods adopted.

Chapter 3 presents the results of the study and is divided into three parts. Part 1 presents the quantitative findings of the circumferential limb measurements, quality of life, and self-efficacy. Part 2 presents the qualitative findings of the semi-structured interviews, while part 3 presents the descriptive findings of the maintenance diary.

Chapter 4 is the discussion and is divided into 2 chapters; the first chapter presents the strengths and limitations of the study and discusses the findings in terms of the current body of knowledge, while the second chapter provides a conclusion and the writer’s recommendations arising from the study.

1.6 Summary

Lymphoedema is an incurable, chronic condition, which can be treated with a combination of manual lymphatic drainage, skin care, exercise and compression (Lymphoedema Framework, 2006). Despite the development of many international consensus documents (International Society of Lymphology Executive Committee, 2013, International Lymphoedema Framework, 2012, Clinical Resource Efficiency Support Team (CREST), 2008) recommending CDT as the gold standard of care, there remains great inequalities in the provision of services, especially to the cohort of patients who suffer from non- cancer related lymphoedema or chronic oedema.

Best practice and allocation of resources must be based on high quality evidence, thus there is a need for research to explore the impact of Complex Decongestive Therapy, as experienced by the patient, on limb volume and quality of life in patients with lower limb non-cancer related lymphoedema. Also, as lymphoedema is a chronic condition requiring long-term maintenance, a further key area that requires exploration is the maintenance practices of

23 patients, the barriers and challenges to life-long self-care, and the impact of these practices on limb volume and patient quality of life.

Chapter 2 Literature Review

2.1 Introduction

Lymphoedema is a chronic, lifelong condition, presenting either from birth to puberty, or at a later stage of life. Regardless of the timing of this disease’s presentation, the patient’s life will be affected, both physically and psychosocially, for the longevity of their lives. Lymphoedema is defined as an accumulation of protein-rich fluid in the interstitial spaces of the tissues (Lymphoedema Framework, 2006). Lymphoedema is classified by its significance of volume and aetiology (International Lymphoedema Society, 2009). Any part of the body can be affected by lymphoedema, but predominately the upper and lower limbs are affected.

The focus of this study was on primary and secondary, non-cancer related, lower limb lymphoedema. This chapter will review the literature pertaining to lower extremity, non-cancer related lymphoedema and chronic oedema in order to inform the study and identify gaps in research. This literature review will investigate the aetiology and classification of lymphoedema, the area of epidemiology, health and social gain, the role of Complex Decongestive Therapy (CDT) in the treatment of lower limb lymphoedema, quality of life, and self-management of lymphoedema.

2.2 Search Strategy

A computerised search of the following databases was undertaken, Medline, Ebsco Cinahl, CENTRAL (Cochrane Central Register of Controlled Trials), Web of Knowledge, Cochrane Database of Systematic Reviews and Embase.

The following keywords were utilised in the search, “lymphedema,” .Also the term “complex decongestive therapy” is frequently used to describe a combination of therapie, therefore, the above term and other derivatives were searched including “complete decongestive physiotherapy”, “complex physical therapy” and “complex decongestive physical therapy”. Additionally, the

25 keywords, “compression bandaging”, “manual lymph drainage”, “hosiery” and “exercise” were searched. Finally, the keyword of “elephantiasis” was searched as this disease is widespread in India and the management of the manifestation of lymphoedema is paramount in this condition.

Mesh headings were used for the above keywords in Cinahyl and Medline. The explode box was used to identify literature under those headings

The only limit that was placed on the search was language. The only language searched was the English language due to the difficulty of the need for translation. From the initial search, the bibliographies of identified systematic reviews were searched for applicable references and other studies were obtained through this method of hand search.

2.3 Aetiology of Lymphoedema

2.3.1 Introduction

The lymphatic system is a one-way circulatory system that drains interstitial fluid from the tissues and delivers it back into the cardiovascular system (McLafferty et al., 2012). In order to understand the varying aetiologies of lymphoedema, it is imperative to have a good understanding of how the lymphatic system works in normal functioning and then the intrinsic and extrinsic factors that can have a negative influence on its function. Many researchers have suggested that health professionals receive only minimal foundation education on the lymphatic system, thus supporting the opinion that the lymphatic system is of lesser importance than other systems, such as the cardiovascular system (Piller and Van Zanten, 2016, Cooper, 2014b, Mortimer and Rockson, 2014). However, more recent research has changed the focus on the level of importance of the venous system in the reabsorption of interstitial fluid by concluding that 90% of reabsorption occurs via the lymphatic system (Levick and Michel, 2010). This section will briefly discuss the anatomy and physiology of the lymphatic system, along with scientific evidence in the area of lymphology, functions of the lymphatic system, and classifications of lymphoedema.

2.3.2 Anatomy of the Lymphatic System

As previously stated, the lymphatic system is a one-way system, comprising a superficial and a deep system. The lymphatic system spreads throughout the entire body, starting in the tissue spaces at the capillary bed, with the superficial lymphatics lying just below the skin surface. The initial lymphatics are blind- ended, valveless vessels that are slightly larger than capillaries. They are situated all over the body and are supported by anchoring filaments that allow the vessel walls to open and close. These vessel are fragile, collapse easily, and are barely able to withstand pressures of greater than 20mmHg (Weissleder and Schuchhardt, 2008). Therefore, any manipulation of the skin during treatment must be light in order to stimulate the lymph endings without causing damage.

The initial lymphatics absorb excess water and waste products, especially protein and fat molecules, which are too large to enter the venous end of the capillaries (Todd, 2012). They act as force pumps that react to changes in total tissue pressure caused by movement, muscular contraction, respiration and changes in external pressure caused by massage, gravity or change of position (Twycross et al., 2000). The understanding of the impact of changes in tissue pressure further reinforces the positive influence that mobilisation, exercise, and deep breathing have on lymphatic drainage of the tissues (Cooper, 2016). Once the fluid has entered the initial lymph vessels, it is now known as lymph and is transported first into the pre-collectors and then into the larger collector vessels. Smooth muscle in the vessel walls of the collectors pumps the lymph along and the valves ensure there is no backflow (Cooper, 2014a).

The lymphatic trunks form the main parts of the transporting vessels and finally mix the lymph into the venous blood via lymphovenous anastomoses (Weissleder and Schuchhardt, 2008). Lymph fluid passes through several lymph nodes throughout the body of which there are approximately 600-700. The lymph nodes essentially function as a biological filtering station. Once through the nodes, the filtered lymph returns to the circulation and enters the venous system at the right and left subclavian veins (Twycross et al., 2000).

2.3.3 Scientific Advances in Lymphology

The principle of movement of fluid from the tissues and back into the microcirculation, and vice versa, was reported initially by Ernest Starling in 1896. He suggested that the capillary wall acts as a semi-permeable membrane and hydrostatic and colloid osmotic pressure regulate the filtration of fluids across the capillary wall. Starling also presumed that the tissues were protein poor (Jacob and Chappell, 2013). However, Levick and Michel (2010) proposed the glycocalyx model. The glycocalyx is a protein-based substance that lines the vascular endothelium and has a dominant role in maintaining vascular homeostasis and regulating vascular permeability, serving as a gatekeeper to fluid and protein penetration (Biddle, 2013). The glycocalyx reduces fluid reabsorption from the tissues by the venules by the filtration of absorbed interstitial fluid at the underside of the glycocalyx, which raises plasma protein levels and therefore reduces interstitial pressure, which slowly cancels the absorptive force. Therefore, it can now be concluded that the lymphatic system is predominantly responsible for tissue fluid balance and is of fundamental importance to all co-morbidities that contribute to fluid imbalance such as heart failure or venous hypertension (Mortimer and Rockson, 2014)

Genetic testing and identification of specific gene types that cause primary lymphoedema is in progress. To date, 20 genes have been identified in relation to primary lymphoedema and all patients who have a familial history of lymphoedema should be considered for genetic testing (Wigg and Lee, 2014). As causal genes are identified, the possibility arises for a classification built on patient genetic predisposition rather than merely relying on age of onset alone

(Mortimer and Rockson, 2014). Near infrared fluoroscopy is also a developing technology that is being used increasingly to allow the visualisation of the superficial lymphatics and tissue in order to enable the mapping of existing lymphatic pathways or identification of a pathological situation (Wigg and Cooper, 2017). This technology is of particular interest in relation to the objective assessment of the effectiveness of manual lymphatic drainage and also the prescription of drainage pathways that will ensure optimal effect from therapy.

Wigg and Cooper (2017) discuss the improved therapeutic effects that two patients demonstrated post lymphatic imaging. Case study 1 suffered breast cancer related lymphoedema and had been treated with traditional CDT and wore a class 3 sleeve; however, oedema of the hand and arm persisted with an overall excess volume of 38%. Following mapping of the lymphatic system, drainage routes of the hand indicated reverse flow and subsequently a fluoroscopy guided MLD plan was established and the results included rapid reduction of volume and improvements to the texture of the skin. Case study 2 had lower limb lymphoedema post hysterectomy over 11 years ago. Lymphoedema had developed almost 6 months post hysterectomy. The patient reported multiple sessions of CDT over the years; however, the swelling had exacerbated over the last 2 years, with only minimal response from CDT. Near infrared fluorescence mapping of the lymphatics showed drainage pathways with no congestion to the hip or suprapubic area, so, the fluoroscopy guided MLD could be concentrated to the inguinal area. Once again, the reduction in swelling was significant, with a 10cm circumferential difference in 2 weeks. These initial findings from case studies will guide practitioners towards further research in the area of lymphatic mapping and fluoroscopy guided MLD.

2.4 Classification and staging of Lymphoedemas

2.4.1 Introduction

Lymphoedema can be classified according to aetiology, presentation, and severity. Many classification systems are referred to in the literature, which can be a source of confusion when comparison of studies is required. The lack of consensus on a common classification system is also reported as a rationale for the lower than expected prevalence rates of lymphoedema (Moffatt et al., 2003, Cooper, 2016). Experts in the field of lymphology would forecast that in the future that the classification of lymphoedemas will be more specific to the pathology and physiology of the lymphatic system, as genetic testing and advanced imaging technology will be more diagnostic (Piller and Van Zanten, 2016, Wigg and Lee, 2014). This chapter will discuss the classification of lymphoedemas and use of staging to diagnose the severity of the disorder.

2.4.2 Primary and Secondary Lymphoedema

The broad division of lymphoedemas into primary and secondary, dates back to the 1930’s and is still the most frequently used method of classification. Primary lymphoedema refers to a congenital cause of dysfunction of the lymphatic system. Secondary lymphoedema refers to a secondary cause that either damages the lymphatic vessels or obstructs the flow of lymph, most commonly due to a cancer or the treatment of a cancer (Lymphoedema Framework, 2006). Primary lymphoedema is further divided into two branches of sub-classification, noting either the age of onset or the pathophysiology of the lymphatic vessels(Twycross et al., 2000).

Primary lymphoedema per age of onset;

 Congenital – present at birth or within two years of life.  Praecox – presents at puberty and up to 35 years of age.  Tarda – presents from 35 years onwards.

Primary lymphoedema per pathophysiology;

 Aplasia – an absence of formed lymphatic trunks  Hypoplasia – the lymph vessels may be smaller, narrowed or fewer than normally expected  Hyperplasia – the presence of large torturous incompetent lymphatics.

Browse and Stewart (1985) further classified primary lymphoedema into congenital and acquired. The aspects of this classification focused in on the anatomical location of the impaired lymphatic vessels, either peripheral or distal to the affected limb and whether valves or nodes are affected. Although this classification is specific in diagnosis, it is rarely used in literature, as it requires intensive diagnostic interventions.

Currently, the treatment regimens for lymphoedema prevent the advancement of the disease but do not address the pathophysiology, one could argue why the need to diagnose the pathogenesis of the disease. However, advances in imagery for diagnostic purposes, guided therapy and the advent of surgical

30 techniques to correct malformations provide hope for future curative treatments (Mortimer and Rockson, 2014).

Secondary lymphoedema is also known as acquired lymphoedema and as the title suggests is due to a secondary cause. The causes can be divided into four main groups:

 Cancer-related  Infection  Trauma  Inflammation (Hampton, 2010).

Lymphoedema classification can also be based on presentation of the disease. The International Society of Lymphology (ISL) (2013) Consensus Document identifies a 4 stage classification system, which is widely utilised to describe the physical condition of the extremity and to allow comparison in research studies.

 Stage 0 – Latent stage, swelling is not yet evident but subtle changes are occurring in the skin and the patient describes subjective symptoms.  Stage 1 – An early accumulation of fluid relatively high in protein content which subsides with limb elevation. Pitting may occur.  Stage 2 – Tissue swelling is irreversible by elevation. Pitting is manifest and at a later phase in Stage 2 the tissue may no longer pit as excess fat and fibrosis occurs.  Stage 3 – Lymph static elephantiasis where pitting can be absent and skin changes such as hyperkeratosis and papilomatosis is evident due to further deposition of fat and fibrosis.

The ISL (2013) proposes that with improved imaging technology and genetic testing that lymphoedema diagnoses and classification will be more detailed and will give a greater understanding of the pathogenetic mechanisms of lymphoedema. This staging process is widely utilised in the literature and lends itself to determination of treatment pathways depending on the stage of the disease process. The severity of the lymphoedema can also be classified using volumetric measurements. The following is a classification that is based on the

31 volume of lymphoedema in the affected limb, in comparison to the unaffected limb, and it is frequently used to measure treatment outcomes:

 Mild - <20% excess limb volume  Moderate – 20-40% excess limb volume  Severe - >40% excess limb volume (Williams and Whitaker, 2015).

2.4.3 Summary

The fact that there are many classification systems to define the aetiology, stage, and severity of lymphoedema poses a difficulty for the standardisation of definition and diagnosis. The varying standards of diagnosis also pose a question, with regard to the accuracy of diagnosis. Research requires that classifications are standardised and similar studies use similar tools in order that comparisons and conclusions can be made. In addition, accurate diagnoses of lymphoedema must be made in order that research results and findings are reflective of the condition that they are investigating.

2.5 Lymphoedema – Epidemiology

2.5.1 Introduction

A review of the literature demonstrates a predominance of prevalence and epidemiological studies focussed on cancer-related lymphoedema statistics, especially in the area of upper limb lymphoedema associated with breast cancer. These studies report incidence rates of between 12% and 40% depending upon treatment type and follow-up timeframes (Poage et al., 2008, Loika et al., 2018, Acebedo, 2015). The allocation of financial resources in the area of cancer treatment and research, by many government bodies, would explain the volume of research in the area of lymphoedema.

In Ireland, the National Cancer Strategy, 2017-2026 (Department of Health, 2017) recognises that survivors of cancer may have long-term side-effects of cancer treatment, which includes lymphoedema, and funding has been

32 allocated to develop services in Primary Care and nurse-led clinics to support these patients and empower them to self-care. The National Cancer Control Program also recognises the importance of research and audit in the area of cancer treatment and a National Cancer Research Group was established at the end of 2017. Likewise, in Northern Ireland and the U.K., lymphoedema services were initially set up in conjunction with cancer care and hospice facilities as a recommendation from Government initiatives based on audit and prevalence statistics of lymphoedema in that cohort of patients (Department of Health Social Services and Public Safety, 2004). In contrast, the availability of epidemiological data in the area of non-cancer related, lower limb lymphoedema is greatly under-reported in most countries (Moffatt et al., 2003, Cooper and Bagnall, 2016). Piller and Van Zanten (2016) recognise the absence of good quality incidence and prevalence data by concluding that sample sizes can be small and limb volume measurement techniques and follow-up periods can differ. In addition, the fact that both definition and classification of lymphoedema is not standardised, means that many reported prevalence rates cannot be compared.

2.5.2 Definitions

Within the literature, there is quite often an interchange of the terms lymphoedema and chronic oedema. Many would suggest that if chronic oedema was used as an umbrella term for all types of oedema, including lymphoedema, that the prevalence would be of such a significance to draw the attention of health services and budget holders (Riches and Keeley, 2012, Moffatt et al., 2003). Lymphoedema is defined as an accumulation of protein-rich fluid in the interstitial spaces, caused by genetic predisposition or a secondary element such as cancer or the related cancer treatments, namely lymph node removal or radiotherapy (Clinical Resource Efficiency Support Team (CREST), 2008). Other causes of secondary lymphoedema include trauma, immobility, obesity, or infection. In developing countries, lymphoedema is predominately caused by a mosquito, termed filarisis (British Lymphology Society (BLS), 2001b, Lymphoedema Framework, 2006).

Chronic oedema is defined as an accumulation of fluid in the interstitial spaces, present for greater than three months. The aetiology of chronic oedema is 33 secondary to an existing co-morbidity such as congestive heart failure, hypothyroidism, or venous insufficiency. Dependence oedema, secondary to reduced mobility, is also a causative factor (National Lymphoedema Partnership, June 2015). There are similarities between the definitions of both chronic oedema and lymphoedema, and both conditions are a result of an imbalance between the capillary filtration of fluid into the tissues and the transport of interstitial fluid back into the lymphatic system (National Lymphoedema Partnership, June 2015). The interstitial fluid is a protein-rich fluid and the lymphatic system transports 90% of interstitial fluid back into the circulatory system via the lymphatic vessels (Mortimer and Rockson, 2014). Riches and Keeley (2012) discuss the term ‘pure lymphoedema’ as an oedema that occurs only from a genetic deficit in the lymphatic vessels causing a lymphatic transport capacity that cannot manage the protein rich fluid load in the tissues. However, in every case of chronic oedema, there will be some degree of lymphatic impairment (Hampton, 2010).

2.5.3 Prevalence of lymphoedema

The greater proportion of prevalence literature is reported from the following countries, the United Kingdom, United States, Canada, and India (Keast et al., 2015, Moffatt et al., 2017, Narahari et al., 2007). Of note, the majority of prevalence studies are disease specific in a population where lymphoedema is known to be a complication of that disease or its treatment, such as cancer- related lymphoedema (Acebedo, 2015). Therefore, lymphoedema statistics of other aetiologies are grossly under-reported.

A small cohort of prevalence studies are population-based (Moffatt et al., 2003, Moffatt et al., 2017, Cooper and Bagnall, 2016), which can lead to good comparative data. Moffatt (2003) conducted a survey using a questionnaire and subsequent interviewing of a subset of the sample in the urban area of South West London and found that 1.33/1000 of that population had chronic oedema. Chronic oedema was defined as an oedema that was present for greater than 3 months and diagnosed on clinical assessment. This study also identified that the prevalence of chronic oedema increased with age (5.4/1000 of population over 65 years of age) and also that prevalence is greater in women than in men (2.5/0.47 per 1000 of population, women versus men). 34

Following on from the 2003 study, Moffatt et al. (2017) repeated a methodologically similar study, also in an urban population and found that the prevalence of chronic oedema had almost tripled since the 2003 study. The prevalence was identified as 3.93/1000 of population in the Derby area. In addition, this study recognised that prevalence of chronic oedema increased with age, as 28.75/1000 of population over the age of 85 years had chronic oedema and, as confirmed earlier in the 2003 study, the proportion of chronic oedema was greater in women. It was noted that the increase in prevalence figures could be related to an increased knowledge of and onward referral of chronic oedemas in the Derby area and not necessarily an actual increase in prevalence. However, the proportionate increase in prevalence over the 14-year time period is significant.

One Irish study, based in an acute vascular and wound management clinic setting, reported a prevalence rate of 2.63% of patients with lymphoedema over a 4 week point prevalence (Gethin et al., 2012). Similar to Moffatt et al. (2003 & 2017), the inclusion criteria identified patients with un-resolving oedema for greater than 3 months, with or without ulceration. The setting of a vascular/leg ulcer clinic increased the predisposition of the client to a secondary lymphoedema due to venous insufficiency. Of further interest is finding that the prevalence of lymphoedema was greater in the male population, 67% male versus 33% female. This is in contrast to Moffatt et al. (2003) and Cooper & Bagnall’s (2016) findings, in which the prevalence of chronic oedema was greater in the female population. However, a limitation of the Gethin et al. (2012) study was that the sample size of lymphoedema patients (n=13) was quite small and therefore the results may not represent the general population of lymphoedema patients. Similarly, Wang & Keast (2016) conducted a study in an acute care wound clinic in Canada and established that the proportion of male to female patients diagnosed with lymphoedema attending the wound care clinic over the 8 year period of the study was an almost equal proportion of 51.2% women and 48.8% men. The overall prevalence of lymphoedema in the clinic population was 21.2%, which is a dramatically greater prevalence than was reported in the Gethin et al. (2012) study. A possible explanation for the significant difference in prevalence in a similar setting could be the fact that the doctor running the clinic had an expertise in wound care and lymphoedema.

Prevalence data is often also divided into classifications of lymphoedemas, either primary or secondary and the varying aetiologies of lymphoedema.

2.5.4 Primary Lymphoedema

Dale (1985) reported that 1 in every 6000 within the UK are affected with primary lymphoedema. Todd (2010) states that primary lymphoedema is uncommon, with an annual incidence rate of only 8 new cases in the U.K. These figures are quite small which would lead one to question, whether the impact of poor medical knowledge of lymphoedema and lack of standardisation in diagnosis have led to such low prevalence rates. However, in Northern Ireland, the Department of Health, Social Services and Public Safety (2004) report 20 new cases of primary lymphoedema annually.

2.5.5 Non-Cancer Related secondary lymphoedemas

The non-cancer related lower limb lymphoedemas can be related to many risk factors which include venous insufficiency, obesity, cellulitis, and immobility.

2.5.5.1 Prevalence of Venous Related Secondary Lymphoedema

Venous insufficiency has been noted to be the most common cause of secondary lymphovenous disease with an overall prevalence of almost 72.7% (Keast et al., 2015). Moffatt et al. (2017) examined the prevalence of leg ulceration in a population of patients with lymphoedema and 40% of those patients actually had concurrent leg ulceration.

2.5.5.2 Prevalence of Obesity and immobility related Secondary Lymphoedema

Changing demographics of chronic illness, life expectancy and obesity will all have considerable effect on the incidence of secondary lymphoedemas. Lewis and Mogan (2008) establish a direct link between obesity and secondary lymphoedema, highlighting the potential for reduced mobility, increased strain on the lymphatics and increased potential for sleep apnoea in the obese population, which often causes the person to sleep in a chair, which in turn causes limb dependency and oedema. Countries such as the U.K., who

36 forecast figures such as 60% of men and 40% of women will be obese in 2050, face a sharp rise in the incidence of secondary lymphoedemas and wounding (British Lymphology Society, 2013). Ireland also has significant health concerns, as 2 out of every 3 Irish adults are either overweight or obese and rapidly moving towards being one of the most overweight European countries (Department of Health, 2013). Wilkins et al. (2014) conducted a survey of 21 morbidly obese patients attending a bariatric lymphoedema clinic over an 11 month period and found that, non-attendance of clinic appointments was high, patients had a poor uptake of dietician review, and concurrent co-morbidities such as heart failure and diabetes further complicated or ruled out treatment options. Essentially, the success rate of treatment of lymphoedema in this cohort of patients was poor.

Dependency oedema is frequently experienced by people who have reduced mobility or are wheelchair users. The absence or reduced functioning of the calf and foot muscle pump lead to stasis oedema, which over time becomes chronic, non-pitting and eventually will develop into a secondary lymphoedema (Elwell, 2017a). Garcia & Dicianno (2011) conducted a retrospective chart survey of 240 patients in a specialised Spina Bifida Centre in the USA. The aim of the study was to investigate the prevalence of lymphoedema in this Spina Bifida population. The findings were significant, in that a 9% prevalence of lower limb lymphoedema was present, which is 100 times greater than in the general population.

2.5.5.3 Secondary Lymphoedema related to infection

Cellulitis can be a causal factor or complication of chronic oedema (Riches and Keeley, 2012). Quite often, it can be difficult to ascertain which came first, the oedema or the cellulitis (Keeley, 2008). Cellulitis is defined as an acute spreading inflammation of the skin and subcutaneous tissues characterised by pain, warmth, swelling and erythema (British Lymphology Society and Lymphoedema Support Network, 2016). The risk factors associated with the development of cellulitis include lymphoedema, wounding and obesity (Atkin, 2016). Annually, in the UK, cellulitis is associated with up to 3% of Accident and Emergency admissions and the average hospital length of stay for treatment of cellulitis is 10 days (Atkin, 2016). Moffatt et al. (2003) noted that of the 218 37 patients interviewed in the study, 29% had experienced an acute infection and of those 27% were hospitalised with an average length of stay of 12 days. This demonstrates that people with lymphoedema are at a greater risk of development of cellulitis.

2.5.6. Summary

Many authors would conclude that lower limb, non-cancer related, lymphoedema is underdiagnosed and poorly recognised and prevalence rates are thought to be grossly under-estimated as a result The grouping of all oedemas under the same umbrella (Moffatt et al., 2003, Moffatt et al., 2017, Cooper, 2016, Todd, 2013) would provide startling figures, which in turn would have significant economic burden on the health service and could possibly target financial resources towards services.

2.6 Complex Decongestive Therapy

2.6.1 Introduction

The term Complex Decongestive Therapy refers to a combination of four modalities, which together form the basis of lymphoedema management. They include compression bandaging or hosiery, manual lymphatic drainage, exercise and skin care (International Society of Lymphology Executive Committee, 2013). The frequency of therapy sessions varies depending on the severity of the lymphoedema and the economic and resource challenges that will confront the patient and the therapist. Interestingly, there is no consensus on the optimum frequency of treatment in order to provide the best outcomes and the varying treatment programmes found in the literature do not allow for comparison of outcomes.

This section aims to discuss the four cornerstones of complex decongestive therapy (CDT), each in isolation and then as a therapy group. The mode of action, outcomes, and both patient and professional challenges associated with their application.

2.6.2 Manual lymphatic drainage

Manual lymph drainage (MLD) is a gentle massage technique that encourages fluid away from congested areas by increasing the activity of the normal lymphatics and bypassing ineffective lymph vessels. The initial concept of the use of massage to promote the movement of fluid in tissues was proposed by a German surgeon, Winiwarter, more than 100 years ago (Williams, 2010). This method was further developed by an Emil Vodder, in the 1930’s, and was named the Vodder Technique, which combines a gentle, pumping circular massage using pressures of approximately 30mmHg followed by a resting phase (Willis, 2004). Vodder continued to work with colleagues from many countries to develop a physical therapy approach to lymphoedema management, which included MLD, exercise, bandaging, and skin care. Despite the fact that there are varying methods of MLD such as Vodder, Foldi and the Casley-Smith method, the principle of the MLD technique remains similar between the different methods (Williams, 2010).

MLD promotes the uptake of the protein-rich interstitial fluid by stimulating the lymph vessels and increasing the rate and flow of the lymphatic system. The health of the tissues is improved by the transport of fluid from the tissues, which in turn improves the flow of oxygen and nutrients to the tissue cells (Weissleder and Schuchhardt, 2008). At present, there is a consensus of expert opinion that MLD is beneficial in the treatment of lymphoedema (Clinical Resource Efficiency Support Team (CREST), 2008, International Society of Lymphology Executive Committee, 2013). The Lymphoedema Framework (2006) suggests that MLD on its own is not a sufficient treatment for lymphoedema. MLD should be used in combination with compression bandaging, exercise, compression hosiery, and skin care. However, it is also noted that there is an absence of good quality research studies, with adequate sample size, which would allow generalizations to be made.

During the Intensive Phase of therapy, MLD is performed by a trained practitioner at a reported frequency ranging from twice daily to three times per week over three to four weeks in order to reduce limb volume and improve joint range of motion. The Maintenance Phase requires the patient to perform simple lymphatic drainage massage techniques that, when combined with nightly 39 compression bandaging and daytime use of compression garments, will maintain the desired level of limb volume. The long-term success of the maintenance phase will ultimately depend on the concordance of the patient with the maintenance plan (Cooper, 2017).

A number of studies support the effectiveness of MLD in combination with the other modalities of CDT (Noh et al., 2015, Wozniewski et al., 2001, Kim et al., 2012). However, there are very few studies which look at the sole use of MLD in the reduction of limb volume. Harris and Piller (2003), in three case studies of patients with primary and secondary lower limb lymphoedema found a movement of fluid and a softening of tissues in the swollen limb after a 45 minute treatment with MLD using the Vodder method.

Muller et al. (2018) conducted a systematic review of randomised controlled trials in the area of manual lymphatic drainage and quality of life in patients with lymphoedema and mixed oedema. The primary outcome of the studies was the effect of the intervention on health related quality of life. The secondary outcomes were volume and functional changes. Following a search of the literature and data extraction, a total of 8 studies were included, only 2 of which involved patients with lower limb lymphoedema. Of those 2 studies, only one applied MLD as a stand-alone intervention (Dos Santos Crisostomo et al., 2015)(Dos Santos Crisostomo et al., 2015)(Dos Santos Crisostomo et al., 2015)(Dos Santos Crisostomo et al., 2015)(Dos Santos Crisostomo et al., 2015)(Dos Santos Crisostomo et al., 2015). In this study, a sample size of 50 patients were randomized to two groups. The inclusion criteria were patients diagnosed with chronic venous insufficiency, as confirmed by venous duplex scan. The intervention group received 10 sessions of MLD over a 4-week period, plus one educational session. The control group only received the educational session. Over the period of the 4 weeks, both groups continued on with their normal daily self-care, which included skin care and compression hosiery. Both groups were followed up to 2 months’ post treatment. Data collection tools included the Chronic Venous Insufficiency Quality of Life Questionnaire-20 (CIVIQ-20), the Venous Clinical Severity Score and circumferential limb measurement using a tape at 3 points on the lower leg.

The CIVIQ-20 provides scores in 4 domains, pain, psychological, social and physical. The findings concluded that the intervention group had improved pain scores at the end of the 4 weeks, continuing into the follow-up period; however, there were no changes in the physical, psychological, or social domains. In addition, leg volume was unaffected by MLD, however, these patients were already wearing compression hosiery and presumably the level of oedema was well controlled.

As discussed previously, the ability to visualise lymph movement and clearance in the superficial lymphatics by using fluoroscopy, now includes a new world of evidence base that can be applied to MLD. De Vrieze et al. (2018) have recently published a protocol of a three armed double blinded randomized controlled trial regarding the effectiveness of fluoroscopy-guided manual lymphatic drainage in the treatment of breast cancer related lymphoedema (BCRL) to be run in university hospitals in Belgium. A sample size of 201 patients with chronic BCRL need to be recruited. All participants will receive the traditional treatment of skin care, compression therapy, and exercises. The intervention group will also receive fluoroscopy-guided MLD. One control group additionally will receive traditional MLD and a second control group will receive a placebo MLD. All subjects will receive 3 weeks of daily intensive treatments and 6 months of maintenance treatment and will be followed-up for a period of 6 months. The primary outcome is the reduction in lymphoedema volume. This study will provide invaluable evidence to the future of manual lymphatic drainage as a modality in lymphoedema treatment and maintenance.

2.6.3 Compression Bandaging and Hosiery

While a large percentage of research around compression bandaging has a focus on venous ulceration, the area of lymphoedema bandaging requires increased investigation. Many questions need clarity such as the optimum bandage type, duration of bandaging and frequency of bandaging as various studies have varying treatment plans with regard to bandaging (Franks et al., 2013, Morgan et al., 2011, Whitaker et al., 2015).

The therapeutic effects of compression are as follows:

 Reducing capillary filtration rate  Increasing lymphatic drainage  Breakdown of fibrosclerotic tissue  Moving fluid from areas of congestion to non-congested areas for reabsorption (International Lymphoedema Framework, 2012).

Inelastic multilayer bandaging is the recommended choice of bandage in lymphoedema treatment (Moffatt et al., 2005). Inelastic bandage provides a stiff structure which has a low, well-tolerated resting pressure and a high working pressure, as the calf muscle expands during movement. The ideal pressure gradient required to achieve volume reduction, as reported by Flour et al. (2011), is 50-60mmHg at the ankle for inelastic bandages. In addition, it was found that pressures >60mmHg resting pressure in a bandage actually reduced volume reduction, thus proving that greater pressures are counterproductive. These findings also have relevance for the frequency of bandage change as an inelastic bandage when initially applied may have a resting pressure of up to 60mmHg. The volume reduction occurs quickly, thus reducing the sub-bandage pressure to a tolerable level, yet a level that can still achieve volume reduction, especially when combined with the massaging effect of exercise. Therefore, a reduced frequency of bandage change is acceptable, as the inelastic bandage remains therapeutic even with pressure reduction secondary to volume loss.

Franks et al. (2013) conducted a prospective cohort study to examine the effectiveness of a 2-layer short stretch bandage, Coban 2, in the reduction of limb volume as the primary outcome and then the comfort, application frequency and differences across patient groups as secondary outcomes. A sample size of 24 patients were recruited, 12 from the UK and 12 from Canada. The sample size was quite small, as this study was an initial evaluation of Coban 2 in lymphoedema management. The inclusion criteria were the need for intensive bandaging, as assessed by the clinician. Sixteen (16) patients had lower limb lymphoedema and 8 patients had upper limb lymphoedema. Limb volume was measured using a tape for circumferential measurement, which was then converted to volume using the truncated cone formulae. All clinicians received training in application of the bandage. The ‘Measure your medical outcome profile’ questionnaire was used to measure secondary outcomes of

42 symptoms (Paterson, 1996). The patients were asked their opinion about the comfort of the bandage. Durability and slippage of the bandage was assessed at each visit by the clinicians. All of the patients were treated for a period of 19 days receiving CDT as per the protocol of the centre at which they were attending. The findings indicated considerable volume reduction over the 3- week therapy period. In addition, symptoms of tightness, heaviness, and swelling improved following the bandage treatment. Comfort rating of the bandage scored high, especially in the area of ability to walk which is of extreme importance for exercise and compliance. The evidence reported in this study in relation to the patients evaluation of symptoms and comfort concurs with findings by Morgan et al. (2011), who found that Coban 2 increased patients’ mobility, improved confidence, and provided a sense of control.

Compression hosiery is used in the maintenance phase of CDT. The wearing of compression hosiery is a life-long commitment in order to maintain oedema reduction and prevent further accumulation of lymphatic fluid in the tissues (Lay- Flurrie, 2011). In order to achieve patient concordance with the wearing of compression hosiery, the patient must understand the dynamics of compression hosiery and their role in oedema maintenance. The patient also needs to be able to get the garments on and off, either with the use of an assistive device or with the help of a carer. Compression garments can be made in two ways, either a circular-knit, which has increased elasticity, or a flat-knit, which has a greater stiffness as is associated with inelastic bandaging. For the purpose of lymphoedema maintenance, the fabric of choice is the flat-knit, which will not allow oedema to extend beyond the stretch of the fabric and also will not extend into skin-folds thus causing skin damage (Lee and Wigg, 2013). In addition, in order to promote concordance, these garments can be fitted with Velcro or zippers, as required, to aid application by the patient and maintain patient independence.

Cooper (2015) discusses the male perspective towards compression and the need for careful consideration when selecting compression hosiery for the male patient as this may well be a deciding factor in the level of compliance of the patient. Perhaps, because there is a predominance of lymphoedema in the female population, the compression hosiery market is more directed towards

43 the female perspective. In addition, it seems more acceptable for a woman to be seen wearing hosiery, whereas a man may feel it inappropriate. The prevalence of non-cancer related lymphoedema in the male population in a study conducted in the West Midlands of the UK was identified as 21% of the overall caseload (Cooper and Bagnall, 2016). However, it was noted that there is a need for further research in the area of male lymphoedema, both cancer and non-cancer related.

2.6.4 Skin care and Exercise

The recommendations from international groups regarding exercise are mostly anecdotal, especially in the area of lower limb non-cancer related lymphoedema where the absence of research is notable (Lymphoedema Framework, 2006, International Society of Lymphology Executive Committee, 2013). Exercises thought to be beneficial to oedema reduction and maintenance include yoga, swimming, walking and low-impact exercises, however the importance of tolerance and prevention of injury to the patient is noted (Clinical Resource Efficiency Support Team (CREST), 2008, Framework, 2006, Lymphoedema Framework, 2006, Cooper, 2015).

The use of exercise in the management of patients with breast cancer related lymphoedema is supported by research. Keilani et al. (2015) carried out a systematic review of resistance exercise and secondary lymphoedema in breast cancer patients and found nine original research articles that met the inclusion criteria. All nine studies concluded that strength training did not have a negative effect on lymphoedema. Simonavice et al. (2017) conducted an observational study of 27 women who were 6 months’ post cancer treatment. Three of the participants had arm lymphoedema, the other 24 did not have any symptoms of lymphoedema. Circumferential arm measurements at 3 cm intervals were taken at baseline and then every 2 weeks for the 6 months of the study duration. Each participant attended a supervised resistance training session twice weekly for the 6-month duration. Two participants had to discontinue the program due to medical reasons that were not associated with lymphoedema. Otherwise, all other participants completed the study with 100% attendance of the exercise program throughout the 6-month period. In conclusion, it was found that

44 resistance training did not have any adverse effect on patients either with or at risk of lymphoedema post breast cancer.

Skin care and the prevention of infection is paramount in lymphoedema care due to the significant incidence of cellulitis in this group of patients (Moffatt et al., 2017). Many skin changes may be seen as a lymphoedema progresses, including, deepening skin folds, hyperkeratosis, papilomatosis, and fibrosis of the skin. Fungal infection of the skin folds is common due to heat and maceration. Regular skin inspection and keeping the skin folds clean and dry will assist in prevention. A fungal infection may be diagnosed with clinical assessment and skin scrapings if necessary and should be treated with an anti- fungal cream (Nowicki and Siviour, 2013). Recommendations for skin care include the use of a soap-substitute for cleansing, regular emollient application and compression to reverse papillomatosis and fibrosis (Elwell, 2017b).

Todd et al. (2017) conducted an audit of skin changes within the population of lymphoedema patients attending a lymphoedema clinic in Glasgow. The need for this audit was fuelled by an impression within the clinic that patients with ‘true lymphoedema’ did not seem to have the amount of skin changes that the patients with chronic oedema or secondary lymphoedema displayed. An audit was carried out of all new patients referred to the service within a 4-month period. In the Todd et al. (2017) study data were collected on sex, age, duration of swelling, body mass index (BMI), history of cellulitis, and presence of skin changes. The criteria for diagnosis of swelling and definition of skin changes was agreed among the four specialists prior to starting the audit. Skin changes recorded were: papillomatosis, hyperkeratosis, skin folds, fibrosis, superficial ulceration, and inflammation.

A sample size of 100 patients was included in the audit (Todd et al. 2017). A total of 52% of those who presented with chronic oedema had a higher than average BMI and almost half of the group were assessed as being morbidly obese. The group also had the highest incidence of cellulitis, at 36.5%, and a higher incidence of skin changes, than the patients with primary or cancer related lymphoedemas. None of the primary lymphoedema patients developed skin changes. Chronic oedema patients, as displayed by this audit, tended to be older, have many other co-morbidities and reduced mobility, which in turn could 45 explain the higher incidence of skin changes due to the inflammatory nature of the chronic oedema. This study leads the way for further studies in this population, with regard to skin care.

James (2011) examined the barriers that patients perceive that prevent them from carrying out regular skin care. A qualitative study was conducted with a sample size of 8 patients attending a lymphoedema clinic using semi-structured face to face interviewing. All patients would have attended 2 educational sessions on skin care as part of the treatment protocol in the clinic. The common themes that were extracted from the transcribed interviews included fear of the consequences of not doing skin care, physical limitations in reaching lower legs, financial implications of buying emollients, and a view that the patients did not have a clear understanding of the function of the skin as a barrier to infection and thus the need to care for it. The results of this study could possibly inform educators of, perhaps, gaps in education programs that need to be addressed.

2.6.5 Complex Decongestive Therapy

Complex Decongestive Therapy as a unit of treatment modalities continues to be the gold standard in the treatment of all lymphoedema types (Clinical Resource Efficiency Support Team (CREST), 2008, Framework, 2006, International Society of Lymphology Executive Committee, 2013). The greatest amount of research into the use of CDT to treat lymphoedema is in the area of breast cancer related lymphoedema. There is a sparsity of research in the field of lower limb treatment for secondary, non-cancer related lymphoedema and chronic oedema.

Iuchi et al. (2015) conducted a retrospective observational design study to examine associations between treatments and outcomes of patients with upper and lower limb lymphoedema. One hundred and seventy (170) patients were recruited from 4 lymphoedema clinics throughout Japan. However, of the 170 patients, only 7 patients received all components of CDT, thus, no associations between treatment and outcomes could be made for this small cohort. The study did show that the quality of life assessed using the LYMQOL for the patients that received CDT was lower than those who were not treated with

CDT. It may be fair to assume that because such a small cohort of the sample size received CDT, that this group may have had a greater severity of swelling and therefore a reduced scoring of quality of life.

Lower limb lymphoedema can have a significant impact on the physical ability of a person to carry out activities of daily living and as highlighted in some studies it can impact on a person’s ability to work and therefore has detrimental consequences from a financial perspective (Moffatt et al., 2003, Stanisic´ et al., 2012). Stanisic et al. (2012) conducted a non-randomised controlled trial to examine the treatment outcomes of CDT on oedema reduction and ability to work in a group of patients with primary and secondary lower limb lymphoedema. The patients were recruited from a retrospective analysis of patients attending a lymphoedema centre in Poland who were deemed unable to work by the social government. A total of 72 patients were recruited, 25 patients were in Group 1, who consented to receive CDT over a 3-6 week period and 47 patients were in Group 2, where they continued on with their standard treatments. Following completion of the intensive phase of CDT, a total volume of 15,330ml oedema reduction was achieved and all patients were deemed able to return to work by the social government. No change was found in Group 2 and all of the 47 patients remained unemployed. However, it is noted that patients in Group 2 expressed a fear of losing unemployment contributions and therefore chose not to avail of intensive therapy. This study does show that CDT can be effective in oedema reduction and restore ability to work

Similarly, in a prospective observational study, Noh et al. (2015) proposed to examine the therapeutic effects of CDT on limb volume, quality of life, and satisfaction. They also set about to compare the differences in therapeutic effect between arm lymphoedema, lower limb and primary lymphoedema. A sample size of 84 patients were identified, 35 patients had arm lymphoedema, 49 patients had lower limb lymphoedema, and all received 10 sessions of CDT over 2 weeks. They were then fitted with compression hosiery and followed up 3 months’ post discharge. Limb volume was measured using a perometer at baseline, week 2 and at 3-month follow-up. The Short Form 36 questionnaire was administered also at the three intervals as well as a study specific satisfaction survey. (Ware & Donald Sherbourne,1992).

Overall, findings identified that patients with lower limb lymphoedema had a lower baseline QOL scoring than arm lymphoedema patients. However, post treatment, there was very little differences in scoring, therefore the patients with lower limb lymphoedema experienced greater QOL changes with CDT. In addition, satisfaction ratings were higher in the lower limb group.

2.6.6 Summary

Complex Decongestive Therapy incorporates four cornerstones, compression, manual lymphatic drainage, exercise and skin care. While the influence of each of these modalities is evident, much of the evidence is gained through case studies and studies of small sample size, with a focus on cancer-related upper extremity lymphoedema versus lower extremity, non-cancer related lymphoedemas. CDT is also time-consuming and requires the resource of specialist training and specialist clinic settings, therefore, more robust research studies need to be completed in order to influence allocation of resource. However, even from case studies, the value of this program of therapy is evident, as the changes to body image, functional ability, and quality of life in all aspects is improved.

2.7 Impact of lymphoedema on health and social gain

2.7.1 Introduction

Lymphoedema is a disfiguring condition that results in the patient having to deal with a limb that is increasing in size and changing in appearance and integrity as time progresses (Murray et al., 2010). It is a chronic condition that has physical, psychological, and economic impact on the patients’ life (Cooper, 2014a). The strength of this impact may depend on the speed of diagnosis and the availability of appropriate treatment. This chapter will discuss the evidence pertaining to social and economic impact and the patients’ quality of life.

2.7.2 Economic impact of lymphoedema on service provision

There is a paucity of information with regard to the financial cost of lymphoedema to the patient and the economy, once again highlighting the underestimation of lymphoedema as a chronic condition, which has a significant impact on the patient, and the economy. The Department of Health, U.K. (2012) reports that patients with long term illnesses account for 50% of all GP appointments, 64% of outpatient appointments and 70% of all inpatient bed days in the U.K. Interestingly, in this government report, lymphoedema is not listed in the named long term conditions; however, of the named conditions, several of them are recognised co-morbidities of the secondary lymphoedema population such as heart failure, hypertension, hypothyroidism, depression, and cancer. Cellulitis, which is a complication of lymphoedema and chronic oedema, is estimated to cost the National Health Service (NHS) £96 million per annum (ILF, 2006).

Humphries and Thomas (2017) evaluated the economic impact of a lymphoedema service in Wales by means of a pre-post service evaluation questionnaire to examine service use across primary care, 6 months prior to accessing the lymphoedema service and then 6 months’ post accessing the service. A purposeful sample of 50 patients from 7 lymphoedema services across Wales completed the questionnaire. Costing of all primary care resources was included, except for medications and appliances such as compression garments. This study demonstrated that the resource utilisation of 50 Lymphoedema Network Wales patients 6 months prior to their entry to the lymphoedema service shows a mean cost per patient of £4859 for the 6 months, when compared to the 6 months’ post entry cost to the service of £2217, indicating an overall estimated potential saving of £2642 per patient. The most dramatic cost savings were seen in the frequency of district nurse calls, GP visits, and practice nurse clinics. Limitations of the study are the sensitivity of the economic analysis and the small sample size. However, this study does highlight the need for a more robust economic investigation into potential cost savings by having a standardised lymphoedema service.

Humphreys et al. (2017) explored the economic impact of a lymphoedema education program in one district in Wales. The aim of the study was also to 49 assess the cost of implementing such a program. A pre-post testing methodology was utilised to discover the financial costing of service resources 3 months prior to the implementation of the program and then 3 months post the education program. The education program was designed to assist healthcare professionals and patients in the management of lymphoedema in the community. A sample size of 97 patients completed the Resource Utilisation Questionnaire. The findings from the study demonstrated a significant reduction in the costing of resources, which included medication and dressings, in the 3- month post implementation of the education program. The total cost of managing chronic oedema in the 97 patients recruited was £563,729 at baseline and £445,098 at 3 months’ follow-up. In addition, district nurse visits reduced by 47%, and hospital stay costs reduced by 38%. Once again, a limitation of the study would be the robustness of methodology used to economically analyse the figures; however, this study does provide evidence that will lead to further research in this area.

2.7.3 The economic impact of lymphoedema on the patient

Pillar (2013) discusses lymphoedema and the influence of societal factors on access to treatment, including geographical and financial implications. For example, Australia, a country that is considered to have one of the better international health systems, does not provide patients with access to the basic lymphoedema care through the public system. Boyages et al. (2017), in an Australian study, conducted an online national survey of survivors of breast cancer, both with and without lymphoedema, in order to assess the financial impact that lymphoedema can cause in this cohort of patients. A sample of 316 patients responded to the online questionnaire, of whom 152 had lymphoedema. An exploratory qualitative methodology was used to interview 30 of the 152 respondents, all of whom had varying severity of oedema. The findings established that lymphoedema does place significant financial burden on the patient in Australia. In addition, the degree of financial burden increased relative to the severity of the lymphoedema, as patients required made-to- measure garments rather than ready-made garments. Compression hosiery accounted for 40.1% of total cost of management. Therapist costs for treatment was also a consideration, as the average number of therapy visits was 5.8 per

50 year. The total average annual cost per patient was A$977, varying from A$207 for sub-clinical lymphoedema to A$1400 for moderate to severe lymphoedema. In order to access care, the patient must pay privately, thus, if the patient cannot afford private care, then they have no access to treatment. Distance from specialist care, transport, and time off work are all issues to be considered which have a significant financial impact on the patient.

From an Irish perspective, it is evident to this author that specialist lymphoedema care is also predominately available in the private sector, thus creating inequitable access of care such that only those in the higher socio- economic classes can afford specialist care. The Irish patients’ perspective and that of the service providers was investigated in 2010 in a research study commissioned and funded by the Irish Cancer Society (Murray et al., 2010). A mixed methodology was employed using both survey questionnaire and focus group interview to explore the Irish perspectives. A sample size of 108 practitioners returned the completed questionnaire and 33 patients with lymphoedema formed the sample for the focus groups. Once again, as highlighted in the Boyages et al. (2017) study, the patients emphasise the financial burden that lymphoedema places on the patient through cost of garments and therapy. The cost was not itemised in this study. Sweden, in contrast, provides lymphoedema treatment and care that is targeted towards an at-risk population and education on prevention is identified as important. Lymphoedema is recognised at an early stage and appropriate care provided. However, the target population is the cancer-related lymphoedemas (Nikolaidis and Karlsson, 2013)

Moffatt et al. (2003) discuss the fact that chronic oedema results in reduced employment, with 80% of patients having to take time off work and 2% having to give up work completely. Another consideration is the fact that the treatment of lower limb lymphoedema takes considerable time out of the patient’s daily work life and thus can have implications for the possibility that the patient may not be able to take the required time out of their employment.

2.8 Lymphoedema and Quality of Life

The majority of research has focused on breast cancer-related lymphoedema and quality of life and only a few authors reporting insight into other groups with lymphoedema and chronic oedema (Moffatt et al., 2003, Williams et al., 2004b, Huggenberger et al., 2015, Harding, 2012, Iuchi et al., 2015, Greene and Meskell, 2017). Lymphoedema has a negative effect on every aspect of life, including school, social life, relationships and dress style (Harding, 2012, Moffatt et al., 2003, Williams et al., 2004b). Interestingly, Huggenberger et al. (2015) in a cross-sectional study of patients with primary and secondary lower limb lymphoedema, after treatment in a rehabilitation clinic in Switzerland, reported that the overall health and quality of life of these patients was comparable to normal values from a population study. Descriptive analysis was used to analyse the data obtained from the data collection instruments, namely, the Short Form 36 and the FLQA-LK.

The Short Form 36 (SF-36) is a validated questionnaire that is widely used for self-assessment of health and quality of life (Burholt and Nash, 2011). The FLQA-LK (Augustin et al.,2005) is a disease specific quality of life tool that is in the German language. However, it is important to note that all of these patients had received appropriate treatment, as recommended by international guidelines, which leads to the conclusion that, with the addition of specialised care, overall quality of life can be improved.

Herberger et al. (2017) conducted a cross sectional observational study over a 1-year period, in Hamburg, Germany with the aim of evaluating the health related quality of life of patients with lymphoedema in the community. A sample size of 301 patients with oedema for greater than 3 months were recruited from health care providers in all sectors of lympoedema care, including GPs, hospitals, nursing homes, self-referral and specialised wound care and lymphology clinics. The methodology included structured interview, clinical exam, and quality of life measurement using the EQ-5D tool and the FLQA-LK. Contrary to the findings of Huggenberger et al (2015), this study revealed a marked impairment of quality of life in comparison to the general population and, in particular, in the specialist centres, where a lower quality of life rating and social impairment was demonstrated. There is the possibility that the 52 severity and chronicity of the patients attending the specialist centres could have a negative impact on quality of life, therefore, highlighting the importance of early referral to these specialist centres.

A study by Moffatt et al. (2003) examined a population of 228 patients with lymphoedema and reported a significant difference in role physical, role emotional, social functioning, and physical functioning. The Short Form 36, a questionnaire widely used for self-assessment of health and quality of life was used in this study. Also, 50% of these patients reported pain from their oedema, describing the pain as aching (36%), heaviness (33%), and tenderness (29%). Notably, in contrast to the patients in the Huggenberger study (2015), only 64% of these patients were receiving any treatment and of these 54% were accessing specialist lymphoedema care with 12% receiving treatment from community services alone.

Williams et al. (2004), using a phenomenological approach, explored the lived experiences of 15 patients with lymphoedema, with particular emphasis on primary lymphoedema. An in-depth interview was used to collect information from the patients and several themes were then identified, including feelings of depression and anxiety, altered body image, and feelings of isolation. Similar findings were noted by Hardy (2012), following an investigation of the experiences of young people with primary lymphoedema. The participants reported struggling with feelings of low self-esteem, poor self-confidence and alteration in body image. Both studies reported that the patients felt that health professionals were lacking in knowledge, with regard to lymphoedema, and all describe a prolonged journey from diagnosis to actually accessing a specialist service.

Lymphoedema treatments aim to alleviate oedema and improve patient’s quality of life. Iuchi et al (2015) compared associations between treatment and outcome of 170 patients with upper and lower limb lymphoedema using a cross- sectional observational study in a lymphoedema clinic in Japan. Seven percent (7%) of the patients had primary lymphoedema and 53% had secondary lower limb lymphoedema. Of the 91 patients who had lower limb lymphoedema, 83% of these were cancer-related. The subjective outcomes of quality of life were measured using the EQ-5D tool and the LYMQOL. The LYMQOL is a 53 lymphoedema condition-specific tool (Keeley et al., 2010). The patients received varying combinations of treatment, including bandaging, manual lymph drainage, simple lymphatic drainage, skin care, and exercise and compression hosiery. Interestingly, of the patients who received treatment from a lymphoedema expert, only 46.5% were satisfied with their treatment. Questionably, perhaps different methods of treatment programmes may be required. The patients who were treated with CDT had a lower LYMQOL score, thus a more favourable quality of life. However, of this group, only 7 patients had CDT and it is therefore a limitation of this study.

To date, only 3 Irish studies have reported on the influence of chronic oedema on quality of life (Greene and Meskell, 2017, Murray et al., 2010, Gethin et al., 2012). Greene and Meskell (2017) using a quantitative descriptive design aimed to examine the impact that chronic oedema can have on health related quality of life. A purposeful sample of 122 patients were recruited from vascular clinics, public health and specialised lymphoedema clinics. Demographic and clinical data was collected and QOL was measured using the LYMQOL, disease specific tool. LYMQOL is divided into 4 domains, symptoms, appearance, function, and mood. LYMQOL also has an overall score ranging from 0=poor to 10=excellent. The findings of this study identified that, while lower limb chronic oedema has a significantly negative impact on QOL, the overall QOL score had a mean of 5.57 (SD 2.52), thus the patients rated their overall QOL as fairly good.

Gethin et al (2012), using the SF-36 to assess quality of life, found that physical functioning had the greatest negative impact on quality of life and emotional well-being was the least affected. However, a limitation of this evidence is that only 13 patients completed the questionnaire. Murray et al (2010) using a mixed methodology, examined the provision of lymphoedema services in Ireland from both the providers and the patients’ perspective, and in doing so, provided great insight into the experiences of the Irish patient living with lymphoedema, which included frustration with delay in diagnosis, feelings of hopelessness, and isolation. However, a quality of life measurement tool was not utilised.

2.8.1 Summary

The evidence indicates that lower limb lymphoedema and chronic oedema does affect quality of life in a negative manner. However, the significance of the impact of lymphoedema on quality of life can depend upon the severity, duration and location of the oedema. In addition, the resources available to the patient have tremendous influence on the patients’ quality of life. The use of generic quality of life tools, for example, the SF-36 and EQ-5D, prevents the generation of data specific to lymphoedema and quality of life and the comparison of the various studies. The use of a condition specific tool would improve the reliability of data and the comparison of studies in the future.

2.9 Self-management of Lymphoedema

Lymphoedema, as previously described, is a life-long chronic condition that can be maintained with the ongoing care and commitment of the patient. A recommendation for a programme of self-care must be grounded in a sound evidence base; however, a review of the literature indicates that there is a lack of evidence upon which to base a plan of care.

Nursing theories guide the way for nursing practice. Theories provide meaning to nursing practice in everyday practice. Orems’ self-care model, developed by Dorothea Orem in 1971, is based upon the belief that the person has a natural ability, right and responsibility to care for themselves. She believed that self- care is a behaviour that is learned and the person cares for themselves, when able, which contributes to maintaining health, life and well-being. Three theories arise from Orems’ Model, that of self-care, self-care deficit, and nursing systems (Punjani, 2013). Self-care deficit refers to the persons’ inability to provide the required care for themselves, however, the skills required to self-care can be learned by the person with the support and knowledge of the nurse or educator (Orem et al., 2001). Many studies that have explored the area of chronic disease and self-care utilise the Orem model, therefore, the researcher believes that the Orem Model is most appropriate to the area of self-care deficit in

55 lymphoedema maintenance and self-care (O’Shaughnessy, 2014, Maslakpa et al., 2018, Bernier, 2002).

In order to empower the patient the health professional as an educator requires the skills to successfully teach and demonstrate the program of self-care that is required by the patient with lymphoedema. Often times it is assumed that the health professional has these skills, that is not always the case, as health professionals often feel more comfortable in a paternalistic role (Wounds International, 2016). The World Health Organisation (2012) has discussed the meanings of health education and health promotion and has established that health education is not merely the transference of education and knowledge but also the promotion of motivation, skills, and confidence (self-efficacy) necessary to take action to improve health. This statement is relevant to lymphoedema self-maintenance education, as the ideal goal is that the patient displays self- efficacy in their own maintenance care.

The Lymphoedema Framework (2006) recommends that the four cornerstones of treatment; compression, manual lymphatic drainage, exercise and skin care, are integral to the maintenance of limb volume and integrity. However, they do acknowledge that the quality of evidence is not robust. A systematic review of self-management of lymphoedema from 2004-2011, identified 16 articles that contained published research results or expert opinion that focused on lymphoedema care. Only a small sub-group of one article was relevant to primary lymphoedema, thus emphasising a gap in research. The authors reported that, based on the included literature, that full-body exercise in the form of weight-lifting and stretches, is most likely to be effective in the maintenance of limb volume in patients with arm lymphoedema post breast cancer treatment. This recommendation was based on a randomised controlled trial of 141 breast cancer survivors. Seventy-one (71) of the women participated in twice weekly weight-lifting exercises, while the remaining 70 women did not participate in any form of exercise. The resulting volumatic measurements did not show any significant difference, but there was a significant improvement in lymphoedema symptoms and exacerbations in the intervention group (Ridner et al., 2012).

Deng et al (2015) conducted a cross-sectional survey of 803 participants with primary lower limb lymphoedema, 269 with unilateral lymphoedema, and 534 with bilateral lower limb lymphoedema. The survey was available to persons within the United States and worldwide who either were at risk of or had lymphoedema. The aims of the study were threefold and with regard to self- care, the study aimed:

1. To evaluate self-care in persons with primary lymphoedema 2. To compare the differences between unilateral and bilateral lower limb lymphoedema in the areas of self-care 3. To explore any associations between self-care, symptoms and infection.

Following analysis of the data, using descriptive statistical analysis, this study concluded the following: 68.9% of the participants performed some of the aspects of self-care, with compression garments being worn by 48.1% of this group, skin care was performed by 40.1%, exercise performed by 32.6% and simple lymphatic drainage completed by 20.7%. Only 13.7% of the participants performed all four aspects of self-care and, alarmingly, one third of primary lower limb lymphoedema patients did not perform any aspect of self-care. Of those who took part 50% reported that they spent 30 minutes per day on self- care. Participants with bilateral lymphoedema were more likely to carry out skin care than those with unilateral lymphoedema but less likely to wear compression garments. Exercise had a positively significant influence on the patients reporting of pain in lower limb lymphoedema. The participants did report taking part in some form of exercise (n=491), however, the type or duration of exercise was not reported. This study also identified a strong association between self-care and symptom burden, reporting that only 68.9% of the patients were conducting some aspect of self-care on a daily basis and, as a result, almost half of the patients reported symptom burden, ranging from swelling, heaviness, stiffness, pain, and numbness On the contrary, Ridner et al. (2012) reported that 88% of patients with secondary lymphoedema post breast cancer participated in all aspects of self-management, which highlights the need for greater awareness of self-management in primary lymphoedema and the need for further resources and research in that area.

Many barriers identified to self-management include age, educational status, physical or functional ability. However, it is important not to assume that because a person is from a lower socio-economic background and have a lower level of education that this person will not be able to self-manage. By firstly identifying the problem, a forum of discussion can be opened between the clinician and the patient which will deepen both the patient and the clinician’s knowledge of how the chronic condition affects the patient and thus a therapeutic relationship will develop (Wounds International, 2016).

Many studies and consensus documents identify the benefit of group self- management programmes.(McGowan et al., 2013, Wounds International, 2016, Tidhar et al., 2014). Disease specific group sessions allow patients to hear different viewpoints, provide deeper learning and patients report increased levels of confidence in self-management and thus reduced isolation (McGowan et al., 2013). In Scotland, the Long Term Conditions Alliance (2008) developed a self-management strategy to help support individuals with long term conditions and embed self-management in a systematic way, in partnership with local authorities and the third level sector. McGowan et al (2013) presented the findings from a retrospective narrative evaluation of 10 courses on lymphoedema self-management during 2010-2012. The programme was provided to 59 patients divided into 10 groups. All participants were at risk of or had cancer-related lymphoedema. The aim of the study was to explore the client’s experiences of the programme and to describe the facilitator’s experience of developing and delivering the programme. The findings highlighted the benefit of a group approach in bringing people together, learning from each other’s experiences, and thus reducing feelings of isolation. Remnerud and Haag (2015), in a 10 year retrospective analysis of patient surveys and interviews, investigated the needs of patients to understand and apply self-care. They stated that the patient must be supported by the therapist and teamwork is imperative. The therapist must listen to the patient, use simple language, and assist the patient to find their own individualised plan of care for managing their lymphoedema.

Of great importance is the understanding of what the patient wants from self- care. Thidar and Armer (2018) explored the meaning of success in

58 lymphoedema management to the patient. They employed a phenomenological approach using semi-structured interview and open-ended questions to understand what people with lymphoedema consider to be a success in the intensive and maintenance phase of management. A purposive sample of 10 people with lymphoedema was interviewed. All patients expressed a feeling of empowerment in the maintenance phase, as they knew how to manage their condition and were in control. In addition, they defined success as “stability”, whereby the condition was stable and not getting any worse.

Up to now, the self-efficacy of individuals with lymphoedema in the undertaking of the 4 cornerstones of lymphoedema management has been discussed. However, Cooper (2014) considers the barriers to self-care that are in our environment, which include funding and commissioning of services, knowledge, insufficient specialist trained therapists, and staffing. As highlighted in the section on prevalence, the future development of lymphoedema services is dependent upon further research and data of chronic oedema incidence in order to drive policy makers to provide funding for services and training of health professionals in the area of lymphoedema treatment and management (Moffatt et al., 2017, Gethin et al., 2012, Greene and Meskell, 2017).

2.9.1 Summary

Self-management of a life-long condition requires a patient-centred approach. Historically, the clinician tends to use a paternalistic approach, which in general has proven unsuccessful. Lymphoedema self-management requires that the patient has the ability to apply the information that they receive and relate the four cornerstones of lymphoedema care to the anatomy and functions of the lymphatic system. Group sessions provide the patient with a greater insight into how others deal with this condition and help to increase confidence and reduce feelings of isolation (Mc Gowan et al., 2013). Therapists need to listen to their clients, be supportive, and provide the knowledge that they require in order to self-care. Ongoing research is required in order to provide robust evidence to support the recommendations of lymphoedema self-care.

Chapter 3 Methodology and Methods

3.1 Introduction

In order to answer the research questions, a mixed methodology, using both quantitative and qualitative methods in a sequential manner, was utilised. This chapter has a number of components. First, the research questions, aims, and objectives of the research study will be outlined. The rationale for selecting the research method and design will be discussed and the sample population, study setting, treatment and maintenance protocol, data collection, and analysis throughout each part of the study will be described. Finally, the reliability and validity of the research employed and ethical considerations will be addressed.

The research questions for the study were as follows:

1. What is the impact of Complex Decongestive Therapy (CDT) as a treatment for primary and secondary lower limb lymphoedema during the intensive and maintenance phases of CDT, in relation to;

a) quality of life b) limb circumferential and volume changes

2. What are the patients’ experiences of living with lymphoedema? 3. What are the patients’ experiences of the four elements of CDT during the intensive and maintenance phases of treatment?

3.2 Research Aims and Objectives

Chapter 2 highlighted that there is a scarcity of research in the area of lower limb secondary, non-cancer related and primary lymphoedema. In addition, considering the fact that lymphoedema is a chronic condition that requires the patient to self-maintain, knowing more about the experiences of the patient throughout this phase is vital to coming to an understanding of patients’ quality of life and care outcomes. The researcher’s interest in the area of lower limb lymphoedema stems from the work that she has completed as a Tissue Viability 60

Nurse in an Irish Community Nurse Led Leg Ulcer Clinic (NLLUC). Many patients presenting to the clinics had lymphoedema on clinical assessment and had active ulcerations. However, the majority of these patients did not have a diagnosis of lymphoedema and had never received any treatment.

The aims of this study were twofold;

1. To examine the impact of Complex Decongestive Therapy (CDT) as a treatment for primary and secondary lower limb lymphoedema during the intensive and maintenance phases of CDT. 2. To explore the experiences of patients with primary and secondary, non- cancer related lower limb lymphoedema during the intensive and maintenance phases of CDT.

The objectives of the study were as follows:

1. To determine the impact of CDT on limb circumference and quality of life. 2. To explore the patients experience of living with lymphoedema. 3. To investigate the patients’ reported experience of the four elements of CDT. 4. To examine the patients’ reported maintenance regimen of self-care and its resulting effect on limb volume and quality of life.

3.3 Justification for choice of methodology

A mixed methods methodology was chosen as the overall approach to guide the study through its different parts. Mixed methods have been called the ‘third methodological movement’, embracing both quantitative and qualitative methods to inform the research question (Teddlie and Tashakkori, 2009). The method embraces the integration of qualitative and quantitative data collection and analysis, in order to get a fuller understanding of the phenomenon being examined (Creswell and Plano Clark, 2011). In mixed methods research, the use of qualitative and quantitative research methods happens in sequence. In this study, the use of qualitative methods were used after an initial period of quantitative investigation.

This study set out to explore the challenges facing patients with lower limb lymphoedema during their treatment and as a lived experience for a person dealing with a chronic condition so that there would be a deeper and more complete understanding of the problem. Therefore, it was essential to provide the most informative, multi-dimensional, and useful understanding of the phenomena of interest (Plano Clark, 2010). Quantitative methods helped understand the measured changes in quality of life scoring and changes in oedema during treatment and in maintenance, while qualitative methods were employed to help understand the meanings, goals, challenges, and realities of living with lower limb lymphoedema (Lee, 2012). Aligning both sets of data in the final phase helped strengthen the overall outcome of the study by offering a more comprehensive and in-depth set of results (Creswell and Plano Clark, 2011).

Moon (2004) asserted that in order to fully describe a phenomenon, it is necessary to supplement qualitative description with quantitative data. In that way, a full and meaningful rich description of the phenomenon can be achieved. The justification for adopting such an approach is threefold. First, just as Creswell (2013) asserted, a research problem or an issue that needs to be addressed determines what kind of research method needs to be undertaken, not the other way around. Second, pragmatist researchers state that it is the research question that drives the inquiry and its design, with the research methods following the research questions in a way that offers the best chance to obtain the most useful answers (Polit and Beck, 2010a). In this study the research questions pointed towards the use of a mixed methods approach as opposed to the use of either a quantitative or qualitative approach. Third, using a mixed methods approach allowed the researcher to take a closer look into both the micro and macro elements of the challenges facing patients suffering with lower limb lymphoedema within the Irish context by collecting qualitative data using semi-structured interviews coupled with quantitative disease specific quality of life questionnaire and limb measurement. Therefore, the design employing both qualitative and quantitative approaches sequentially was particularly appropriate and advantageous for this study.

3.4 Mixed Methods Research

Mixed method research is a relatively recent development in research, with the majority of mixed methodology studies having been published in the last 20 years. Bressen et al. (2016) conducted a literature search in CINAHL of all studies with ‘mixed methodology’ in the title and found that over 61.9%, of those found arose within the last 4 years. Historically, nursing research in particular has revolved around two traditionally polar opposite methodologies, namely, quantitative and qualitative research. Nursing is embedded in the many schools of sociology, psychology, and natural sciences and therefore can adopt the various research methodologies that encompass each discipline (Watson et al., 2008). One of the central tenets of mixed methods research is that the oppositional views held by strong proponents of either qualitative and quantitative research would be best served through the development and utilisation of a new, third methodological approach, namely, mixed methods research (Creswell, 2014).

The origins of mixed methods stretches back to 1959 when Campbell and Fisk used varying forms of only quantitative tools to examine psychological traits. Following on from then, the concept of triangulation was developed, whereby quantitative and qualitative methods converge in order to answer the research question (Creswell, 2014). By the mid 1990’s, mixed methods research was well established and since then an increasing amount has been written on when to use mixed methods and how best to integrate the differing methodologies (Tashakkori and Teddlie, 2010).

Researchers have conceptualised research designs as sitting on a continuum, with one end of the continuum containing single method designs where only one method is used, with partially mixed designs in the middle and fully mixed designs at the other end of the continuum (Onwuegbuzie and Leech, 2007). Fully mixed methods involve the mixing of quantitative and qualitative techniques within or across one or more stages of a research study. With partially mixed methods, the quantitative and qualitative phases are not mixed within or across stages; instead, the elements occur either concurrently or sequentially in their entirety before being mixed at the data interpretation stage (Curry and Nunez-Smith, 2015). 63

Mixed methods research incorporates the strengths of both qualitative and quantitative methodologies (Cameron, 2011, Burke-Johnson et al., 2007). Reviewing the literature on mixed methods research confirms its current use in nursing and health-related research, such as in pediatric oncology nursing (Wilkins and Woodgate, 2008), family medicine (Stange et al., 2006). mental health services (Creswell and Zhang, 2009), disabilities (Mertens, 2010), and public health (Fox et al., 2013). While mixed methods research takes more time and resources to employ, its value in adding depth to findings far overweighs any difficulty experienced in applying the approach (Creswell & Plano Clark, 2011).

3.5 Definitions of Mixed Methods Research

The fact that mixed methods research incorporates the philosophies of both qualitative and quantitative methodologies would explain the reason why there are many varying definitions of mixed methods research. Johnson et al. (2007), having examined and analysed 19 previously published definitions of mixed methods research, define mixed methods research as:

“Mixed methods research is the type of research in which the researcher or team of researchers combines elements of qualitative and quantitative research approaches for the broad purposes of breadth and depth of understanding and corroboration” (p.123).

Creswell and Plano Clark (2011) describe mixed methods research as the combination of quantitative and qualitative methods in a single study. The integration of the data from both methods gives strength and greater clarity to the research findings than if the methods had been used in isolation (Zhang and Creswell, 2013). Mixed methods research is different to multi method research. In multi method research several methods from the same methodology may be used to collect data and answer the research question, such as focus groups and interviews. The data, however, are not combined, as in mixed method research (Johnson et al., 2007).

3.6 Classification of Mixed Method Designs

The classification of mixed method designs is complex due to the varying approaches that may be used in order to answer multi-faceted health sciences research questions. However, the novice researcher requires a guide or framework in order to direct the practice of mixed methods research (Guest, 2013). Creswell and Plano Clark (2011) identified three basic types of mixed method research designs: convergent, explanatory sequential, and exploratory. Convergent design refers to the simultaneous collection of qualitative and quantitative data which is then, either merged or embedded into each other in order to build or strengthen the evidence that the data has displayed (Curry and Nunez-Smith, 2015)

An exploratory sequential design collects qualitative data first followed by the collection of quantitative data. The qualitative findings can inform the development of the quantitative tool or provide independent findings that can inform the research question. Regardless of the type of mixed research used, the goal of any research design should be to generate the knowledge to effectively address the research questions of the study (Teddlie and Tashakkori, 2009).

For the purpose of this research study, an explanatory sequential design was chosen and employed to support and explain the quantitative findings of the limb volume measurements and quality of life changes.

3.7 Explanatory Sequential Design

Sequential mixed methods data collection strategies involve collecting data in an iterative process, whereby data collected in one phase contributes to the data collected in the next, allowing the ongoing augmentation of data collected from a chosen sample (Creswell and Plano Clark, 2007).

In this study, an initial quantitative phase was followed by a qualitative phase. When choosing a mixed method design, many elements must be considered including resources and the research question. An explanatory sequential

65 design may be used when the quantitative findings may not be clearly understood without the addition of the qualitative data to further explain the quantitative data (Curry and Nunez-Smith, 2015). This has proven true of the findings of this study, as the quantitative data alone could not have offered insight into the rationale for changes in the quality of life values over time.

Guest (2013) suggests that the weighting, or importance of methods in a mixed method study is not always evident until both methods are analysed and the data is integrated. Curry and Nunez-Smith (2015) concur with the idea that, when the plan of integration is stated, then the sequence of each method will fall into place. However, other experts argue that the priority of the methods should be decided from the initial stages (Morgan, 1998).

It is suggested that in using mixed method research the weakness of one method can be offset by combining them with an alternative method that offers different strengths, with both methods complementing one another. The purpose of combining methods is to achieve a broader, more comprehensive picture. However, Gerrish and Lacey (2010) illustrate that it is impossible to know from the outset of a mixed method study whether the findings will converge or diverge. If the study widens out and diverges away from the initial ideas, the researcher must carefully consider these divergences and reflect on the complexity revealed by the study.

Cutter and Jordan (2012), using an explanatory sequential mixed method design, examined the differences in compliance with standard precautions in operating theatres between surgeons and nurses. A postal survey was given to surgeons and nurses working in six NHS trusts in Wales. The survey asked questions regarding compliance with standard precautions such as eye protection and double gloving. The survey was then followed up with face to face interviews with surgeons and nurses and telephone interviews with infection control nurses. The interviewees were selected based on their answers to the survey and those who had greater numbers of incidents, or who were more extreme in their answers. The survey response rate was 51.47%. The quantitative findings, which identified that surgeons expected inoculation injuries as an occupational hazard, were further clarified or explained by the qualitative findings, which suggested that the surgeon considered themselves to 66 have a tendency to take risks and had egotistical characteristics. Nurses were more likely to adhere to protocols. This study gives an example of how the quantitative findings were further clarified and explained by the qualitative data gained through interview.

The following figure gives a presentation of the sequential format of this study.

Quantitative Data

Leg volume measurements Quality of life

Qualitative Data

Interview Diary

Integration: Connected Self-efficacy Data to add objectivity to Quan /Qual Diary Narrative.

Figure 3.1 Explanatory Sequential Design of This Study

The theoretical and philosophical battle between quantitative and qualitative methodologies has long abated with the conclusion that all have a role to play in research, with no one research methodology any better than any other methodology, with many authors calling for a combination of research methods, to improve the quality of research as our approaches for scholarly inquiry continually evolve (Arnd-Caddigan and Pozzuto, 2006).

3.8 Philosophy underpinning Research Methodologies

A research philosophy is a belief about the way in which data about a phenomenon should be gathered, analysed and used (Halcomb and Hickman, 2015). Philosophy does not inform us about how to undertake a mixed methods 67 study, what dilemmas we might encounter, or how to combine both quantitative and qualitative data in a study (Creswell and Garrett, 2008). The term epistemology (what is known to be true) as opposed to doxology (what is believed to be true) encompasses the various philosophies of the research approach. Mixed methods research is, generally speaking, an approach to knowledge that attempts to consider multiple viewpoints, perspectives, positions, and standpoints, including the standpoints of qualitative and quantitative research. Mixed methods researchers use diverse philosophical positions and many find mixed methods research to be challenging because of the tensions created by their different beliefs (Greene, 2007). A philosophical approach is also described as a ‘world view’ that influences how the researcher looks at the world (Halcomb and Hickman, 2015).

Creswell and Plano Clark (2011) have considered world views in mixed methods research. The world view most commonly associated with a mixed method study can be either pragmatism, transformative approach, or critical realism. A transformative approach requires the researcher to adopt a consciousness of cultural awareness and injustices. The critical realist approach requires that the reality can only be discovered by looking at multiple views and opinions (McEvoy and Richards, 2006). Pragmatism was selected as the overriding philosophy in this study due to its emphasis on developing research questions devoted to real world problems and answering them in a manner where the best methods are used to get the answers needed.

The author’s greatest concern whilst undertaking this study was that the study would be both relevant to the research question and rigorous in its operationalisation. Overall, the author believed that a pragmatic philosophy was best suited for study, in order to understand the challenges facing patients with lower limb lymphoedema in treatment and in maintenance self-care.

3.9 Pragmatism and Mixed Methods Research

The philosophical underpinning of pragmatism is based on the work of Pierce, James, and Dewey in the 19th and early 20th century. This offers an alternative

68 view to that of neo or post positivism, participatory inquiry and constructivism paradigms. It focuses on the problem to be investigated and the consequences of that research (Teddlie and Tashakkori, 2009). It recognises that there are multiple and singular realities available to research enquiry and is focused on solving practical problems (Creswell and Plano Clark, 2011).

Pragmatists argue that research should be useful and give the researcher freedom to use whatever techniques or methods they need to answer the research question. Mixed methods research can provide an opportunity to transform these tensions into new knowledge. Pragmatic perspectives draw on employing “what works,” uses diverse approaches, gives power to the importance of the research problem and question, and values both objective and subjective knowledge (Morgan, 2007).

Pragmatism means that the focus of research is on the research question and different methods can be employed to answer this question. According to Burke Johnson et al (2007) and Morgan (2007), pragmatism provides a philosophy that supports paradigm integration and helps mixed research to coexist with the philosophies of quantitative and qualitative research. Furthermore, Yin (2006) stipulates that the bias inherent in employing particular data sources and methods are overcome and neutralised by using mixed methods.

3.10 Methodology

This study was divided into 3 separate parts.

In part one a quantitative approach was used to measure:

1. Limb circumference and volume changes, to provide information on treatment and self-management outcomes. Tape measurements at 4cm intervals, widely reported in literature, was the chosen method. Measurements were taken:

 during the intensive phase of CDT at baseline and then weekly for 4 weeks;

 during the maintenance phase at baseline, week 8, when compression hosiery was fitted and then at week 16 and week 24.

2. Quality of Life changes:

A disease-specific quality of life questionnaire, developed by Keeley (2012) named LYMQOL LEG was used. LYMQOL LEG is comprised of 22 questions that covers 4 domains, namely; symptoms, body image, function, and mood, as well as an overall quality of life score. The tool is validated for use with people with lower limb lymphoedema. The questionnaire was completed by the patients at the following time-frames:

 at baseline, at week 8 and at week 24.

3. Self-efficacy

The participants level of confidence to be able to independently self care for their lower limb lymphoedema was measured at week 24, following 4 months of self maintenance, using the Patient Reported Outcomes Measurement Information System(PROMIS) Self efficacy for managing a chronic condition. This tool which was validated by Gruber-Baldini et al. (2017) is comprised of five domains, daily activities, emotions, social interactions, symptoms and medications. Four of the five domains were used in this study, the domain for managing medications was not utilised as the long term management of lymphoedema does not usually include medications.

In part 2 a qualitative approach was used to explore the following:

1. The patients’ experiences of living with lymphoedema and its impact on their lives; 2. The patients’ experience of the elements of CDT; 3. The patients’ reported experience of self-maintenance and how the commitment to wearing compression hosiery, skin care, exercise, and simple lymphatic drainage has impacted on their lives.

An interview schedule, consisting of 10 open-ended questions, was used to explore experiences outlined above, and is presented in Table 3.1 below.

Table 3.1 Interview Schedule Date of interview - ______Interview Start Time - ______Interview Finish Time - ______Interviewer - ______Interviewee identifier No. ______Questions; Living with lymphoedema 1. When did you first notice that there was a problem with your leg/legs? 2. Tell me about you experience of seeking diagnosis and then therapy for your lymphoedema? 3. In what ways has lymphoedema affected your everyday life from a physical, psychological and social perspective? Experience of treatment 1. What has been your experience of manual lymphatic drainage, both positive and negative? 2. What has been your experience of compression bandaging, both positive and negative? 3. Tell me about your exercise program and any challenges it might present? 4. What has been your experience of the skin care regimen? 5. Moving forward, do you have fears or concerns about maintaining your limb/limbs independently? 6. Do you feel that you have been given the skills and education required to maintain your leg/legs in the future? 7. What aspects of self-maintenance do you feel pose the greatest challenges?

The interviews took place at week 8 when the intensive phase was complete and the patient was fitted with compression hosiery. Dictaphone was used to record interviews in a private room, where the atmosphere was quiet and comfortable. Only the researcher and the participant were present. The interview was transcribed by a person who had signed a confidentiality form

71 prior to transcription. Thematic analysis was used to identify common themes in the data.

In Part 3 a qualitative approach was used. The patient completed a diary, daily over a four-month period, and the content was used in order to explore the experiences of self-maintenance. The patient was given a diary, with one page assigned to each day of treatment. The requirements of the diary record were discussed with the patient and they were given a list of indicators that they were requested to record in the diary on a daily basis. These are presented in Table 3.2.

Table 3.2 Maintenance Diary Daily Record Maintenance Diary – Daily Report The purpose of this diary is to provide a clear and factual account of the day to day care that you complete in order to keep your leg/legs in a healthy state. Please record your daily care under the following headings; Compression hosiery Number of hours worn/ any difficulties getting hosiery on or off/laundry of hosiery/any problems associated with the hosiery Skin Care Time taken and at what stage of the day/ emollients used/ comments about the health of your skin. Exercise Type and time dedicated to exercise. Challenges associated with exercise Simple Lymphatic Drainage(SLD) How often and for what length of time? Comments about SLD. Any general comments or observations about maintenance care of limbs

To facilitate increased compliance with the diary record, the patient was contacted by telephone on a weekly basis and when they went back to the clinic for measurement review on weeks 16 and 24, they were asked to bring the diary along with them.

This mixed method approach meant that quantitative data was collected initially followed by the collection of qualitative data which helped explain and elaborate on the quantitative findings.

3.11 Sample

Appropriate sampling is a vital part of the research process that needs to be carefully planned (Burns and Grove, 2009). Sampling is a key step in any study because it helps establish the quality of inferences a researcher makes from the findings of a study (Collins, Onwuegbuzie and Jiao, 2007). Polit and Beck (2010) describe a sample as a portion of the population chosen to represent the entire population. The purpose of sampling is to attain data from a smaller representative sample, which in turn increases efficiency by allowing generalisations to be made about the population without having to include every member. The sample must also be representative of the target population in as many ways as possible. Purposive sampling was used in this study to generate rich information from participants who could responsibly reflect on their experiences to inform the research question (Holloway and Wheeler, 2010).

While qualitative researchers have historically not placed the same amount of emphasis on sampling as quantitative researchers (Onwuegbuzie and Leech, 2007), sampling is important in all research, as it is central to establishing credibility (Onwuegbuzie and Collins, 2007; Onwuegbuzie and Leech, 2007; Houser, 2008). The sampling technique will affect the validity of the research, therefore it should be undertaken with the maximum degree of rigor (Procter and Allan, 2007).

The population of interest in this study was patients with lower limb lymphoedema. The establishment of inclusion and exclusion criteria strengthened the evidence produced and increased the precision of the study (LoBiondo-Wood and Haber, 2010).

In mixed methods research, sampling can be complicated and arguably needs greater attention (Collins et al., 2007). The challenge proposed by mixed methods sampling is the appropriate balance between the breadth of data 73 generated from quantitative methods and the depth of data generated from qualitative data (Curry and Nunez-Smith, 2015). The aim of qualitative studies is to discover meaning and to uncover multiple realities; therefore, qualitative researchers usually include the kinds of people who can best enhance the researchers understanding of the phenomenon, have the desired experience, will best contribute to the information needs and are eligible to participate in the study (Polit and Beck, 2010a). Convenience sampling and/or some form of purposeful sampling are only one of many types of qualitative sampling schemes available for researchers.

This study used a purposive sampling approach. This approach is used when researchers purposely choose subjects who, in their opinion are thought to be relevant to the research topic (Bowling, 2014). Purposive sampling involves the researcher intentionally selecting who to include in the study on the basis that those selected can present the requisite data (Parahoo, 2006). The researcher used a purposive convenience sampling method to recruit patients who had non-cancer related lower limb lymphoedema, who were considered typical of the study population and who were capable of answering interview questions and completing the diary.

The researcher’s decision to study the population of patients who suffer from non-cancer related, lower limb lymphoedema stems from the researcher’s clinical experience in the area of lower limb ulceration which highlights the reality of both under-diagnosis and non-diagnosis of lower limb lymphoedema. Also, the majority of published research has a focus on the area of cancer- related lymphoedema, in particular breast cancer related lymphoedema, thus demonstrating a definite paucity of research in the area of non-cancer related lower limb lymphoedema.

Purposive sampling implies that the researcher decides what needs to be known and sets out to find people who can and are willing to provide the information by virtue of knowledge and experience (Polit and Beck, 2010a).The non-random selection of participants infers a bias by the researcher in the selection of the study population. The selection of people with lymphoedema is fraught with difficulties as the progression and stage of the condition may be

74 different within the population. Also, the varying pathways to diagnosis and treatment tend to influence the patients experience of living with lymphoedema.

Sampling decisions in mixed methods research are often influenced by practical considerations such as resources and time. It is not always possible to examine a wide population and, therefore, consideration needs to be given to strategic sampling choices (Curry and Nunez-Smith, 2015). Sampling plans can be either independent, interdependent or combined depending upon the relationship between the probability and purposeful sampling techniques. The decision to use a specific sampling plan will be based upon the research study objective, the rationale for using mixed methods and the proposed relevance of the study such as transferability or applicability (Teddlie and Tasshakori, 2006).

In this study, an interdependent sampling plan was used, whereby one sample was used for both the quantitative and qualitative parts of the study. The choice to use the interdependent sampling plan and a sample size of 20 participants was based on the following:

 The sample population was small due to delayed diagnosis and lack of knowledge by health professionals, with regard to treatment pathways;  The possibility of a multi-site study is limited in Ireland, by virtue of the fact that there is a sparsity of lymphoedema practitioners in the Irish health service, and those who do provide a service tend to be lone practitioners;  The sampling approach will commit more strongly to the qualitative, information-rich side of the study;  The study follows the participant from baseline therapy to 4 months self- maintenance allowing for the collection of an in-depth data set.

The small sample size was suitable because of the potentially detailed data that could be generated from each participant. Sandelowski (2007) suggests that determining adequate sample size in qualitative research is ultimately a matter of judgment and experience in evaluating the quality of the information collected against the uses to which it will be put, the particular research method and purposeful sampling strategy employed. Thus, the depth of a study’s scope and

75 its clarity of focus will influence the number of participants needed to describe the phenomenon.

Furthermore, based upon reported prevalence rates, both national (2.63%; Gethin et al., 2012) and international rates (0.13%; Moffatt et al., 2003), and taking into account the population of clients accessing the Leg ulcer/wound management clinic in the current setting as 624 new referrals over a year period, a sample size of 20 clients could provide a strong reference point. In addition, the richness of using both quantitative and qualitative methods, which includes multiple data collection tools, provides the study with significant amounts of data on the topic of research. An unpublished thesis, which explored the impact of CDT on physical activity, also used a sample size of 21 patients (Kunkel, 2010).

Also, when conducting a research study, the researcher needs to consider the resources available and time constraints of study timelines for completion. In this study, each participant was followed for 6 months, from baseline to the end of the four month self maintenance period. Each participant was in active treatment for eight weeks and the researcher could only treat three to four clients at each 8 week therapy interval. Therefore the maximum amount of participants that could have been treated and followed up within the time constraints of the study was 20 participants.

3.12 Study subjects and the setting

The setting was a lymphoedema clinic, run by this researcher, who is a Tissue Viability Nurse (TVN) in a community primary care setting in the Midlands of Ireland. The clinic runs on a Monday/Wednesday/Friday morning. Each client requires up to 90 minutes per therapy session and therefore only 3 clients can be treated each morning. The intensive phase of treatment usually requires an eight-week therapy session. The TVN runs five Nurse Led Leg ulcer clinics from which clients would be identified and clinically diagnosed with lymphoedema. Referrals were sourced from Primary Care, General Practitioners (GP) and vascular clinics.

The inclusion criteria were as follows:

Patients with:

 Unilateral and bilateral primary and secondary, non-cancer related, lower limb lymphoedema,  Stage 2-3 lymphoedema,  Diagnosis confirmed by a vascular consultant,  Referred via leg ulcer clinics, G.P or vascular consultant,  Male and female,  Aged 18 years and over.

The exclusion criteria were as follows:

Patients with:

 Secondary, cancer-related, lymphoedema,  A medical condition that would preclude the use of MLD, such as acute cellulitis, unstable congestive cardiac failure, acute deep venous thrombosis.

Patients were directly referred to the Lymphoedema Clinic via the General Practitioner (GP), vascular consultant, public health nursing, and some self- referred. Patients were referred for the management of lower limb lymphoedema. Each patient was then clinically assessed by the researcher. In general, patients referred by vascular consultants had a diagnosis of lymphoedema, either primary or secondary.

Treatment and Maintenance Protocol

The intensive phase of treatment included a 60-minute therapy session during which the participant received manual lymphatic drainage for approximately 35 minutes followed by either unilateral or bilateral full leg compression bandaging depending upon whether lymphoedema was present in either one or both limbs. During the intensive phase of treatment the participant received treatment 3 times weekly for 4 weeks. At the end of 4 weeks the participant was measured for flat knit, made to measure, compression hosiery. The participant then

77 continued to receive treatment twice weekly until the compression hosiery was available.

Manual lymphatic drainage involves a light massage that follows a particular sequence of steps and specialised techniques. Manual lymphatic drainage can only be performed by a certified therapist.

The compression bandaging that was employed during the study was a 2-layer system which included a polyester padding layer followed by 2 short stretch cohesive bandages applied in a spiral technique.

Skin care was completed before bandaging was applied and this consisted of a wash with warm water and an emollient, followed by a further application of an emollient.

The participants were given verbal and written instruction with regard to exercises that could be completed while in compression bandaging and when bandaging was discontinued and compression hosiery was provided.

The eight-week therapy phase, which included the 4-week intensive phase followed by the 4-week maintenance therapy phase, also provided a forum for one to one education for the participant in preparation for independant self-care. The participants were given demonstration of simple lymphatic drainage during the therapy sessions and they also received written instruction on simple lymphatic drainage.

All participants (n=20) received flat knit made to measure compression hosiery for the maintenance of limb volume reduction. Four participants received tights, six participants received thigh length hosiery, two participants received a combination of knee length sock worn with a Capri (separate extension to cover the knee and thigh) and the remaining eight participants received knee length hosiery. The level of compression of the hosiery ranged from class 2 (n=18) to a class 3 for the remaining two participants.

3.13 Data Collection – Part 1. Quantitative Phase

Bryman (2012) stated that for it to truly count as a mixed methods approach, the data within a study derived from two methods should be mutually illuminating, resulting in an enhanced end product. According to Burns and Grove (2009), a study with a clear focus and concentrated data collection usually has richer, more credible findings.

For the quantitative phase of the study, the researcher used:

1. Circumferential measurements of the affected limb at 4cm intervals from a fixed point at the ankle.

A specific tape measurement tool was used which provided a consistent and reliable point of measurement along the leg that would not change with each comparison measurement. The tape would be initially stabilised at the crease of the foot to leg and thereafter each 4cm interval would be allocated from the tape measure. Thus, reliable comparison from week to week of limb circumference could be made. The number of limb interval measurements varied from 11 points up along the leg to 20 points, depending upon the elevation of oedema in the leg, either to the knee or groin. The measurements were taken at baseline, prior to treatment and then weekly for 4 weeks, at week 8, week 16, and week 24. The maintenance phase began at week 8, at which stage the patient had been fitted with compression hosiery and had begun their journey through self-maintenance, which included simple lymphatic drainage, skin care, exercise, and wearing compression hosiery every day.

As previously stated, tape measurement is well accepted as a reliable method of providing quantitative data regards limb circumference (Williams and Whitaker, 2015). Limb circumference can also be calculated as the volume of oedema by using a specific mathematical calculation. The measurement of volume is mostly of significance in the area of unilateral primary lymphoedema, whereby the unaffected leg volume is noted as the baseline or desired therapeutic goal for the treatment effect of the affected leg. Limb volume measurement is also

used to establish the extent and stage of lymphoedema (Williams and Whitaker, 2015). For the purpose of this study, the collection of circumferential measurements and volume was used to indicate changes in oedema, reduction or increase during the intensive and maintenance phases of CDT.

2. Quality of life scoring using the LYMQOL Leg Tool

Lymphoedema has a negative effect on every aspect of life including school, social life, relationships, and dress style (Hardy, 2012, Moffat et al., 2003, Williams et al., 2004a). For the purpose of this study, a disease specific quality of life tool developed by Keeley was chosen to collect data on quality of life (Keeley et al., 2010).

LYMQOL LEG is a validated condition-specific quality of life assessment tool, which can be used for lymphoedema of the lower limbs both on clinical assessment and as an outcome measure (Keeley et al., 2010). This tool was developed by a group of experts in the field of lymphoedema care. The reliability and validity of the tool was tested in a lymphoedema clinic setting using a sample size of 209 patients. Face validity was confirmed, as all patients found the questionnaire easy to use. Ninety-two percent of patients felt that no questions could be left out; however, only half of the patients returned the questionnaires. Reliability was tested using the test-retest method on 15 patients. The sample size was small due to the fact that the majority of the patients did not return for a second treatment; however, good correlation between scores were recorded at each point of treatment (Keeley et al., 2010). The LYMQOL tool has been widely used however the initial validation process is still quite limited and further validation is ongoing in different patient populations in other countries.

LYMQOL covers 4 domains, namely; symptoms, body image, function, and mood. It also gives an overall quality of life score. Each domain has a series of questions that pertain to that domain, and answers are rated from: ‘Not a lot, A little, Quite a bit to A lot', with a numerical score given to each answer. The scores in each domain are added and then divided

by the number of questions in that particular domain, thus, each domain has an accumulative score. The lower the score, the better the quality of life for that patient. However, in the overall score, the values range from 0=poor to 10=excellent, therefore, the lower the overall score the poorer the quality of life.

LYMQOL Leg was given to each participant at baseline, week 8 and week 24. The patient completed the tool while at clinic; therefore, there was a 100% return of questionnaires. However, one patient did not complete the week 24 quality of life tool due to an illness not related to her lymphoedema.

LYMQOL LEG is presented in Appendix 13.

3. Self-efficacy tool

The original methodology and design did not consider the use of a self- efficacy tool. However, as the study progressed and patients began to fill out their diary on self-management, the issue of self-efficacy became more important. In this study, the researcher is also the therapist who provides the treatment of Complex Decongestive Therapy over the 8-week period. The patient and therapist develop a therapeutic relationship during this time and it would be quite possible that the patient would want to please the therapist by writing in the diary the information that would satisfy the therapist. An informal meeting with an expert in the field of lymphoedema gave the researcher opportunity to discuss the above issue and the suggestion of using a self-efficacy tool as an objective means of rating self-confidence in the completion of self-maintenance tasks was given to the researcher. Following on from this meeting it was decided to use a self-efficacy tool.

A literature review of self-efficacy did not provide a disease specific tool, however, a self-efficacy tool for the management of chronic conditions was discovered, called Patient Reported Outcomes Measurement Information System (PROMIS) - Self-Efficacy for Managing a Chronic Condition tool, developed and validated by Gruber-Baldini et al. (2017). This tool comprises five domains, calibrated across diverse chronic conditions

and shows good internal consistency and cross-sectional validity (Gruber‑Baldini et al., 2017). The five domains are as follows:

1) Self-efficacy for managing daily activities, 2) Self-efficacy for managing symptoms, 3) Self-efficacy for managing medications and treatments, 4) Self-efficacy for managing emotions, 5) Self-efficacy for managing social interactions.

The responses to questions asked ranged from, ‘I am not at all confident’ to‘ I am very confident’, with 5 answer options available. The author was contacted to request permission to use the above tools and same was granted. Four of the five short form questionnaires were used; however, the short form for medication management was not used, as medication is not a part of lymphoedema maintenance. The 4 Short Form Self-Efficacy questionnaires are presented in Appendix 14.

The PROMIS Self efficacy for Managing a Chronic condition tool provides a set of five short forms that have been psychometrically developed and tested. A total of 1,087 participants from the United States, 837 of whom had chronic neurological conditions, which included multiple sclerosis and Parkinsons, and 250 patients who had general medical conditions, including congestive heart failure, completed the 5 short forms between the timeframe of April 2013 and April 2014.The age range was from 18 years and upwards, mean age 53.8. A total of 57% of the study population were female. The internal consistency of the 5 short forms was high, with Cronbach’s alphas ranging between 0.96 to 0.97.

Further work is needed to assess the sensitivity of the tool to change over time after interventions or other changes known to impact on functioning occur. Validation in other samples of chronic disease is also needed.

To ascertain the appropriateness of the tool for the cohort of lymphoedema patients, cognitive interviewing was employed with two participants. Cognitive interviewing requires that the participant ‘think out loud’ about the questions in the questionnaire in order that their perception of the question being asked can

82 be assessed (Ahmed et al., 2009). For the most part, cognitive interviewing is used in the development of questionnaires to assess reliability; however, it can also be used to assess participants understanding of questions when adapting previously validated questionnaires (Kamp et al., 2018). The two participants were asked the questions ‘out-loud’ to assess their understanding of the questions being asked in relation to self-management of lymphoedema. The term ‘symptoms’ in the questionnaire was understood to mean ‘swelling’ by both patients. The term ‘doctor’ in the social questionnaire was understood to mean therapist. The responses from both participants varied from quite confident to very confident in self-managing each domain.

The self-efficacy tool was given to the participants to complete on week 24. The patients completed the tool at home and then posted them back to the researcher so as to avoid any pressure felt by the patient to please the therapist. All but one of the questionnaires was completed. One participant did not complete the study to week 24 due to an unrelated illness.

3.14 Data Collection – Part 2 Qualitative Phase

Qualitative research is a systematic and rigorous form of inquiry that uses methods of data collection such as in-depth interviews, ethnographic observation, and review of documents (Watson et al., 2008). It yields detailed information reported in the voices of participants and contextualised in the settings in which they provide experiences and the meanings of their experiences (Creswell, 2007). Brewer and Hunter (2007) view qualitative research as a more genuine method than quantitative methods, as it yields more descriptive, richer data, which is based on personal experience. Qualitative methods allow for identification of previously unknown processes, explanations of why and how phenomena occur, including the range of their effects, and provide valuable insights and advances to knowledge (Pasick et al., 2009). Data collection is facilitated by qualitative methods when measures are not available, representing the voices of participants through quotes (Creswell et al., 2011). Qualitative data collection methods, such as semi-structured interview, allow an in-depth understanding of complex phenomenon that may

83 not be explored fully by using numbers (Bowling, 2014). Two methods of collecting qualitative data were employed in this study, these were the semi- structured interview and a diary.

Semi-structured interview

For the purpose of this study, 18 participants completed a one to one, semi- structured interview with the researcher. Of the 2 participants who declined, one stated that she was “too nervous” and the other patient was unwilling for no stated reason. Interview questions were asked as per the interview schedule, which includes 10 open-ended questions. The interview questions were divided into 3 parts:

 experiences of living with lymphoedema,  experiences of CDT,  challenges of maintenance.

Interview times ranged from 20- 45 minutes. Interview tape recordings were transcribed after each interview and the patient was allowed to read the transcript to verify that they were satisfied with the contents of the transcript. Semi-structured interviews using the interview guide were employed, as they allowed the researcher to have a framework in which necessary open-ended questions were posed to encourage the participants to talk freely about their experiences (Polit and Beck, 2010). The interview guide contained a list of open-ended questions, which were derived from the literature review and the aims and objectives of the study. Open-ended questions were used, as the researcher wished the participants to respond in their own words, thus enabling richer and more complex data to be collected (Whittemore and Grey, 2006). Whitehead and Annells (2007) added that open-ended questions must be unbiased, balanced, clear and sensitive. This was achieved by using simple language and by clarity of questioning that was easily understood by the participant.

The researcher reflected on familiarity and experience with the topic. When developing the interview schedule, the researcher would often contemplate responses to the questions and opinions on the topic. Therefore, the researcher

84 ensured that care was employed when undertaking the interviews to ensure that the participants were not lead or asked questions in a certain way to elicit the response that the researcher desired or expected.

Diary

A diary was completed by the participants on a daily basis for four months. The patient was requested to record the following under 4 headings:

1. Compression hosiery

 Number of hours worn on a daily basis,  Any difficulties getting hosiery on or off,  Frequency of laundry of hosiery,  Any problems associated with the hosiery.

Best evidence and practice recommends that compression hosiery suitable for the treatment of lymphoedema is measured and prescribed by a certified practitioner competent in the area of lymphoedema treatment and management. In order to maintain the reduction of oedema, a flat knit garment was recommended. Due to the compression levels required to maintain a stage 2 or stage 3 lymphoedema - for example, from up to 32mmHg - these stockings require strength and dexterity on behalf of the participant. Many of the secondary lymphoedema patients in the study were morbidly obese or elderly, thus, the physical functionality required to apply and remove such stockings can be a barrier to concordance. Compression stockings were CE marked, licenced for 6 months, and should have been washed daily to maintain elasticity. However, daily washing and drying of the stockings can also be a burden due to reduced physical ability of the patient and economic challenges of affording 2 pairs of socks (one to wash and one to wear).

2. Skin Care

 Amount of time taken to complete skin care,  At what stage of the day did skin care occur,  Emollients used,  Comments about the health of your skin. 85

Maintenance of the health of skin is paramount in the prevention of complications such as fungal infections, dry skin, ulceration, and cellulitis. Best practice recommends that skin care should include daily washing with a low PH soap substitute, drying the skin meticulously, especially between the toes and skin folds and the application of an emollient at least once daily. Although this may sound like a straightforward task, in this population of patients it can be quite common that they cannot reach their toes or lower limb due to obesity and reduced mobility.

3. Exercise

 Type of exercise,  Amount of time dedicated to exercise,  Challenges associated with exercise.

In the study population, many obstacles to exercise existed, which included, reduced mobility, body image, pain, clothing and footwear, and psychosocial issues. Low impact exercise is well recommended for the patient with lymphoedema in order to avoid injury to joints or tissues. Swimming and aqua exercising has been widely documented to be beneficial to oedema reduction and maintenance. However, the impact of altered body image can pose a great challenge to the patient with lymphoedema when facing the prospect of wearing a swimsuit and exposing a distorted limb.

4. Simple Lymphatic Drainage(SLD)

 How often was SLD initiated,  For what length of time did the patient continue SLD,  Comments about SLD.

Simple Lymphatic Drainage is a modified technique of Manual Lymphatic Drainage that the therapist, or in the case of this study, the researcher, teaches the patient through written and demonstrated instruction. While SLD is a recommended cornerstone in the maintenance of lymphoedema, little is known about the impact of SLD in the maintenance of oedema reduction.

A diary was implemented as a means of gathering qualitative data, with regard to the participants’ perspective on the application of the 4 cornerstones of lymphoedema maintenance. The participants were requested to bring the diary with them when they would present at clinic at 2 months and 4 months’ post treatment. In addition, the patient received a weekly text as a reminder, with regard to completion of the diary.

A strength of the use of a diary as a means of collection of data would be the opportunity to examine the course of living with a chronic condition over a prolonged period of time. However, a limitation of the diary would be the potential that the participant might lose interest and would not complete the diary over the required timeframe.

3.15 Data Analysis – Part1 Quantitative

The quantitative data elicited by the circumferential measurements were analysed using Microsoft Excel. The circumferential measurements of the affected lower limbs were measured in centimetres at 4 cm intervals along the leg. These measurements were then converted to limb volume measurements using the truncated cone formula, which was available on an Excel package. The volume measurements were recorded at the pre-determined timeframes and changes in volume were calculated by converting the volume changes into a percentage of change from week 1 to week 8 and then from week 8 to week 24 and also from week 1 to week 24. This provided a clear picture of limb volume changes.

Demographic data were coded, and descriptive statistics were then used i.e. mean, percentages, and standard deviation and the combined data was presented in tables.

Quality of life data, taken from the LYMQOL Leg Tool, were entered into SPSS and the quality of life scoring under each domain was analysed using descriptive statistics, as percentage change from week 1 to week 8, week 8 to week 24, and also a comparison from week 1 to week 24.

Self-efficacy questionnaire data were analysed using Excel. Cumulative scores for each participant, in each domain, were recorded, and mean, minimum, maximum, and standard deviations were presented in table format.

3.16 Data Analysis – Part 2 Qualitative

Semi-structured interviews

Thematic analysis is a method of analysis that identifies, describes, analyses, and reports themes and patterns from data. Braun & Clarke (2006) contends that although thematic analysis is widely used, it is poorly documented. Guest et al. (2012) reveal that thematic analysis takes a pragmatic approach to identify implicit and explicit themes and complexities of meaning within the data. It is a rigorous, inductive set of procedures to identify and examine themes from data in a transparent and credible method. It extrapolates the stories and experiences of participants in an accurate and comprehensive manner. For the purpose of this study the six phase thematic framework developed by Braun and Clark (2006) was employed.

Audio recordings were listened to repeatedly in order to capture the tone, emphasis, and meaning of the responses. The researcher transcribed the one to one interviews verbatim. After approval from participants that their interview transcript was accurate, the researcher read transcripts were read over and over again and notes were made in the margins of words, theories or short phrases that summed up what was being said in the text. Codes developed to represent the themes and offer summary statements or words for each element that was discussed in the transcript that related to the research question. Guest et al. (2012) point out that codes may be compared for frequency, co- occurrence, and relationships.

Braun and Clarke (2006) defined a theme as an idea that captures an important aspect, response, or meaning in the data in relation to the research question. Theme identification began early in this study from the first reading of the transcripts and continued throughout the coding process until the final phase to produce the final results. Initial thematic categories were identified, 88 and codes were assigned which depicted the themes in the best way possible. When coding and theme identification was complete, they were written in a memo that showed the connections between each theme on a thematic chart.

The Diary

The contents of the diaries were analysed using a simple descriptive narrative approach. Eighteen (18) diaries were returned at the completion of the 4-month period. One participant did not complete the program due to ill-health and one participant did not return the diary. The diaries were read and numerical data were recorded with regard to:

1. Compression hosiery

 Length of time that compression hosiery was worn  How often hosiery was washed

2. Skin Care

 Length of time devoted to skin care

3. Exercise

 The type of exercise  Length of time given to exercise

4. Simple Lymphatic drainage (SLD)

 Length of time given to SLD

Reflections were noted and grouped together for thematic analysis. The diary data was then integrated and combined with the quantitative data of the limb volume measurements and quality of life changes in order to explain and illuminate the quantitative changes that were noted for some of the participants.

For the purpose of this study, quantitative data were analysed initially using statistical analysis, followed by qualitative data analysis using thematic analysis. Finally, both quantitative data and qualitative data in the form of a diary was

89 integrated to explain finding during the maintenance phase of the study when the participants were self-caring.

3.17 Pilot Study

A pilot study can be described as a smaller version or trial run of the proposed study, conducted to refine the methodology, improve clarity, and eradicate problems (Burns and Grove, 2009). Two participants with bilateral primary lower limb lymphoedema consented to taking part in the pilot study, having received the information leaflet and having signed the consent form. Both patients were referred to the service by a vascular consultant, having had a confirmed diagnosis of primary lymphoedema. A pilot sample of 2 participants was deemed appropriate as the total sample was 20 participants, the amount of time required for the pilot was 10 weeks in order to progress through the intensive treatment and into 2 weeks of the self-maintenance and diary record. Both of these participants were representative of the average age group of the sample population and had a diagnosis of lower limb lymphoedema.

3.17.1 Data Collection Part 1 Quantitative

Tape measurements were completed as per protocol and recorded without difficulty. The tape measure was easy to use and secure to the designated points of measurement on the leg. The circumferential measurements were applied to the Excel program for conversion into volume without difficulty. The comparison of measurements showed a significant reduction in weeks 1 and 2, which then came to a plateau at week 4. Also, post fitting of hosiery, the measurements demonstrated the ability or otherwise of the hosiery to maintain limb volume. One participant, week 1 post fitting of hosiery, showed an increase in limb circumference, despite 100% compliance with the prescribed hosiery.

The LYMQOL LEG QOL tool was completed by the participants. They reported that the questionnaire was easily understood, all areas were completed. Comparison of baseline and week 8 did demonstrate an improvement of overall QOL, from a rating of 3 at baseline to a rating of 8 at week 8.

Both participants proceeded through the treatment without event, tending to get a little “fed up” at week 4-5, but then looked forward to getting the hosiery. The researcher completed onsite training with an Advisor from an international wound care company, with regard to the practice of measuring for made to measure stockings and types of styles based on limb definition. This session was very beneficial to the researcher as a lymphoedema therapist.

A trend that the researcher noticed in the patients was that initially they were resistant to a life of wearing heavy hosiery, they were full of worries about the summer and how they would manage in the hot weather. However, by completion of the 8 weeks, they had an understanding and an acceptance with regard to compression. Their focus changed from cure and the need for dramatic changes in their legs, to prevention of complications and worsening of the condition in the future. Through regular discussion about the condition, provision of written and verbal education based on international guidelines that cover skin care and simple lymphatic drainage, the participants reported that they now felt in control.

3.17.2 Data Collection Part 2 Qualitative

The interview was conducted at week 8, using the interview schedule. This enabled the researcher to determine the approximate length of the interview, to test that the audio-recording equipment was working and picking up all voices, the suitability of the environment and that the interview guide was applicable (Davies, 2007). The researcher was initially nervous as she had not interviewed people before as part of a research project and as a novice interviewer. As a result of this, interview 1 lasted 10 minutes and interview 2 lasted 5 minutes. On reflection, the researcher should have given the patients the list of interview questions for a few days before the interview so that they could have written down pointers for themselves. After the interview was concluded, the participants both felt that they forgot to say a lot of the things that they considered important. However, both patients spoke of similar themes and issues around diagnosis and QOL. On completion of the interview, the transcript was typed and read by the participants to ensure that they were comfortable that the content accurately reflected what they had said in the interview. The typing of the transcript did require a lot of time, which lead the researcher to 91 conclude that she would need assistance with the typing of the transcripts as the study progressed.

The diary was completed for one week by the patients, and all aspects were documented upon by the patient.

3.17.3 Data Analysis Part 1 Quantitative

Data collected from the limb circumferential measurements were entered into an Excel table in centimetres and millilitres. The measurements were inputted at each timeline for measurement without difficulty. The researcher did reflect upon the time commitment required to input data and realised that this would need to be allowed for within the therapy day, thus reducing the number of participants that the researcher could treat and input data into the database on a daily basis. This experience led the researcher to plan the actual research study, with an aim of treating three participants per therapy day for the length of the intensive and maintenance phases. Quality of life data were entered into SPSS, which was a learning experience for the researcher and therefore required training in the use of this database. The data was easily entered and analysis was completed using descriptive statistics, with the advice of a statistician.

3.17.4 Data Analysis Part 2 Qualitative

The interview transcripts were read and re-read in order for the researcher to get a true and totally inclusive view of the data. The researcher decided not to use a database to organize the qualitative data, as she felt that she was more comfortable with organizing the data using the steps of the Braun and Clark’s (2006) thematic analysis guide. Once again, the researcher was provided with a clear realisation that this form of qualitative analysis was also time consuming. The diary was completed by the participants for two weeks only and was written up each day by the participants. The diary was analysed using a descriptive narrative of the daily activities of maintenance care.

In summary, having completed the pilot, the tools were specific to the study and easy to use. The researcher’s inexperience in interviewing required practice and time planning and scheduling of data input needed consideration. 92

3.18 Reliability and Validity

When undertaking research, the aim was to collect information that is reliable and unbiased and assures the quality of the research (Health Information and Quality Authority, 2011). For a research study to be accurate, its findings must be reliable and valid. Validity refers to the fact that the measures used within the study are actually measuring the concept of interest(Lipsey and Hurley, 2009). Reliability means that the findings would be consistently the same if the study were done over again (Kimberlin and Winterstein, 2008).

3.18.1 Reliability and validity in Mixed Methods Research

A study conducted by Sale & Brazil (2004) attempted to identify criteria for the purpose of critical appraisal of studies that employed mixed methodologies and the framework upon which they based the appraisal was initially proposed by Lincoln and Guba (1985), known as the Framework of Trustworthiness and Rigor. Both quantitative and qualitative methods have very distinct and separate standards of quality or rigor assessment. The advent of mixed methodologies has opened the debate as to whether both elements of qualitative and quantitative methodologies should be assessed for rigor separately or as a unit. Curry and Nunez-Smith (2017), having drawn upon sources such as Polit & Beck (2010), and Sale & Brazil (2004), developed ‘Common Standards of Quality and Appraisal Criteria for Qualitative and Quantitative Studies’. These standards include veracity, consistency, applicability, and neutrality.

3.18.2 Veracity

The standard of veracity refers to credibility in the qualitative study and internal validity in the quantitative study. Veracity refers to the “truth value” of the study or the level of accuracy by which the results of the study represent the phenomenon of interest (Lincoln and Guba, 1985).

Credibility of the qualitative data is evidenced through establishing that the methods and approaches chosen by the researcher to answer the research question are appropriate and what would be expected in that particular methodology. Measures that may be taken to ascertain credibility would include familiarity or a reasonable timeframe spent with the participants and member- 93 checking (Connelly, 2016). For the purpose of this study, the researcher spent 8 weeks of therapy with the participant prior to the interview process, therefore, a therapeutic relationship had developed, and the researcher was familiar with the participant. Following on from the interview and its transcription, the participant received the transcribed interview for feedback, thus ensuring that the information that the participant gave was reflected accurately in the transcription. In addition, the transcription occurred directly from the audiotape thus no other interpretation could be used. The diary also allowed the participant to reflect in privacy, without the presence of the researcher, and provide truthful narrative description of maintenance of oedema reduction. The objectivity of this information was further consolidated by the use of a self- efficacy tool.

A further method of testing credibility in qualitative data includes data saturation, whereby no new information is gained through interview (Curry and Nunez- Smith, 2015). This study has interviewed 18 participants using a semi- structured approach until data saturation occurred.

Internal validity of the quantitative data is concerned with the degree of accuracy with which the findings of a study represent the relationship between the population and the variable of interest (Curry and Nunez-Smith, 2015). In order to assess internal validity, the following question can be posed, ‘Did the study measure what it intended to measure?’ For the purpose of this study, three variables were measured,limb volume, quality of life and self- efficacy.

All were measured at stated intervals using validated and reliable tools and measured what they had set out to do so. However, elements such as mortality have posed a threat to internal validity, as one client was unable to complete the measurements due to ill-health.

3.18.3 Consistency

The second standard of consistency refers to dependability in the qualitative study and reliability in the quantitative study. Dependability of a study is assessed by the examination of the study for clarity of reporting of processes such that the study could be replicated with the same population and the same

94 results would be found. An external audit by another researcher would be a method of completing this assessment but this would prove challenging and could violate patient confidentiality (Cope, 2014). In this study, an audit was not employed however the transparency of the methods and methodology utilised to conduct the study would assist a researcher to replicate this study with a similar population and quite possibly demonstrate the same results

Reliability of the quantitative method is established by examining the potential to use the same tools of measurement in the same or in a different population and get similar results (LoBiondo-Woods and Haber, 2010). A questionnaire should have evidence of reliability and validity thus making the researcher confident that the findings reflect what is intended to be measured (Sullivan-Bolyai and Bova, 2010).

For the purpose of this research study, the use of circumferential measurement at 4cm intervals is documented as reliable and has good inter-rater reliability (Williams and Whitaker, 2015). The quality of life questionnaire is disease- specific and has been tested for reliability (Keeley et al., 2010). A pilot study was also carried out to test the user friendliness of the tools and no difficulties were experienced by the participants or the researcher. The self-efficacy tool which was applied at week 24 was also tested for validity and reliability by the authors (Gruber‑Baldini et al., 2017). Cognitive interviewing was employed by the researcher to assess the participants’ understanding of the questions in the self-efficacy tool. Both participants had a good understanding of the questions but both felt that the word “doctor” in the questionnaire referred to ‘therapist’ and the word ‘symptoms’ referred to ‘swelling’.

3.18.4 Applicability

The third standard is applicability, which refers to transferability in the qualitative context and generalisability in a quantitative context. The transferability of a qualitative study is associated with the extent to which the study findings could be applied to a different population. Transferability occurs when a description of the research processes and the research population is given such that other researchers can decide if the method can be successfully applied to their population (Cope, 2014). 95

Generalisability refers to the potential that similar results could be found by applying the same quantitative tools to other subjects in the same population. Techniques such as randomisation, inclusion /exclusion criteria and the use of validated tools can enhance the generalisability of findings (Curry and Nunez- Smith, 2015). This study has clearly defined the inclusion and exclusion criteria for participants. Validated tools were also employed, thus, assisting the generalization of the study. However, random sampling was not employed as the sample was purposeful and the sample size of 20 would not be of significance in a purely quantitative study therefore the generalisability of this studies’ results would not be possible.

3.18.5 Neutrality

The last common standard of neutrality includes confirmability in the qualitative study and objectivity in the quantitative study (Curry and Nunez-Smith, 2015). Confirmability of a study depends on the researcher’s ability to report the participants’ actual views without any influence from the researcher’s prior experiences or expectations. Confirmability is enhanced by quotes from the participants which demonstrate their views. For the purpose of this study, the researcher used a framework developed by Braun and Clarke (2006) to analyse qualitative data. Many quotes are provided to explain the development of themes. However, the researcher is also the therapist for the participants and has an in-depth knowledge of the condition. This could possibly provide bias towards expected outcomes of treatment. However, the objectivity of the quantitative instruments, which include the lower limb circumferential measurements and the quality of life scoring, will guard against any subjectivity in the findings.

3.19 Ethical Considerations

The Helsinki Declaration (1964) states that the consent of a participant in research must be voluntary. A valid consent requires that the participant receives sufficient information about the study and that adequate time is given for them to make a decision. The Royal College of Physicians (2007),

96 developed guidelines to assist researchers to adhere to the legal and ethical requirements of research. These guidelines highlighted the following rights of participants.

Participants should:

 have the opportunity to ask questions and comment,  be aware that they can withdraw from the study at any stage they wish and without prejudice to their continued treatment,  know how information about them will be used and how confidentiality is secured.

This study also followed the Heath Service Executive (HSE) National Consent Policy (National Consent Advisory Group, 2013). Each participant was given an information leaflet, which included information about the study and the treatment in order that the participant could make an informed decision about consenting to treatment and inclusion in the study (Appendix 1). The participant was given adequate time to read the information leaflet and ask questions to the researcher. The information leaflet was given to the participant at the initial assessment meeting having ensured that the participant was suitable for recruitment as per the inclusion/exclusion criteria. The participant then had a week within which time they could make their decision to join the study or not. A consent form was given to the participant for signing when they had consented to join the study (Appendix 2). Ethical approval was applied for and granted by the HSE Midlands Area Research Ethics Committee (Appendix 3) and by the Royal College of Surgeons Ethics Committee in January 2016 (Appendix 4). Clarification was requested by the Midlands Area Research Committee on the risk of bandage slippage during treatment and how this would be addressed. The researcher’s response with regard to frequency of bandage change, monitoring of skin integrity, and patient advice was accepted by the Ethics Committee (Appendix 5). One of the most fundamental ethical principles in research is that of beneficence, whereby the participant should come to no harm from the study (Polit, Beck & Hungler, 2001). In this study, the risk of bandage slippage causing skin damage was limited by the frequency of bandage change, especially in the initial 4 week intensive therapy phase when limb volume reduction was quite notable. The frequency of bandage change 97 then reduced in the maintenance phase, when the participant was waiting for hosiery, as the limb volume would have stabilised at this stage.

3.19.1 Risks

The potential for risk was minimal in this study. The four cornerstones of CDT were used, these were, manual lymphatic drainage, compression therapy, skin care, and exercise. Manual lymphatic drainage was provided using a light skin massage technique. This therapy may only be provided by a certified therapist following specialised training and certification. The researcher has completed this training. Compression therapy is also only provided by a competent professional who has received training in bandaging technique. Compression was only applied following vascular assessment for arterial insufficiency and same out-ruled. The risk of bandage slippage was minimised by the increased frequency of bandage change during volume reduction and the use of padding to protect the skin. The participants received advice on bandage removal should they have had any concerns of trauma to skin integrity be of concern.

3.19.2 Data Protection

The use and transfer of patient data associated with research involves legislation such as is set out in the Data Protection Acts 1984 and 1988 and the European ‘Data Protection Directive 95/46/EC’ (European Parliament and of the Council, 1995), which ensures the protection of individuals in relation to processing of personal data, including sensitive data and the exchange of such data.

The researcher and both supervisors are the only people who had access to the patients’ data. Data was coded using a number and the identity was held by the investigator. A locked filing cabinet was in place for all hard copy data and the investigators’ laptop was password secured and encrypted. All data was also transferred and saved directly onto the V: drive of the RCSI server.

3.20 Summary

This chapter has outlined the research questions, aims, and objectives. This was a mixed methods research study using a sequential explanatory design. The philosophical underpinning for this study was pragmatism, which gave the researcher freedom to use whatever techniques or methods they needed to answer the problem. A purposive sample of 20 participants were recruited based on a strict inclusion/exclusion criterion. Quantitative data was collected using circumferential measurement of limb to calculate limb volume, a questionnaire to measure quality of life at stated intervals and 4 Short Form questionnaires to measure self-efficacy in lymphoedema self-care. Qualitative data was collected using a semi-structured interview and a diary. Both datasets were analysed singularly and later the results were compared and contrasted to further explain key findings. Reliability and validity of this mixed methods study was discussed and strengths and weaknesses explored. Ethical considerations were examined and the processes of consent, confidentiality, and anonymity were addressed.

Chapter 4 Results - Quantitative Findings

The aims of this study were to examine the impact of Complex Decongestive Therapy, during the intensive and maintenance phases of treatment, on patients with primary and secondary, non-cancer related lower limb lymphoedema by measuring limb volume and quality of life. This study also explored the experiences of patients living with lower limb lymphoedema in Ireland, their experiences of CDT as a treatment and their self-efficacy during the maintenance phase of self care. In order to answer the research questions, a mixed methodology was used. The findings will be presented in three chapters. This chapter will present the quantitative findings, used: a. to introduce demographic data; b. to ascertain the impact of Complex Decongestive Therapy (CDT) as a treatment for primary and secondary lower limb lymphoedema during the intensive and maintenance phases of CDT, in relation to Quality of life and Limb circumferential and volume changes; c. to examine the participant’s confidence in being capable of carrying out maintenance therapy by measuring self-efficacy.

Chapter 4 will present the qualitative data that emerged from the semi structured interviews. This data will relate to the participants’ experience of living with lymphoedema, their experiences of Complex Decongestive Therapy, and the perceived challenges that maintenance therapy offered the participants. Chapter 5 will present the descriptive narrative generated from the analysis of what was written in the 4 month diary.

4.1 Demographic Findings Following ethical approval, 20 participants were recruited to the research study. Participants were referred by General Practitioner, vascular consultant or via the Public Health Nursing service to the Lymphoedema Clinic and allocated to the study following clinical assessment of suitability, as per the inclusion criteria and having given verbal and written consent. The sources of referral for all participants(n=20) was as follows;  Vascular consultant referral = 11 participants

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 General Practitioner referral = 4 participants  Public Health Nursing Referrals = 5 participants The demographic findings are displayed in Table 4.1.

Table 4.1 Baseline Characteristics (n=20) Characteristics Number (%) of participants Gender Female 18 (90) Male 2 (10) Age Range 18-40 years 2 (10) 40 – 60 years 8 (40) 60 – 80 years 10 (50) Body Mass 18.5-25 (healthy) 3 (15) Index (kg/m2) 25-30 (overweight) 5 (25) 30-35 (moderately obese) 4 (20) 35-40 (severely obese) 2 (10) 40-45 (very severely obese) 3 (15) 45-50 (morbidly obese) 0 50-60 (super obese) 3 (15) Aetiology Primary Lymphoedema 10 (50) Secondary Lymphoedema 10 (50) Related to CVI 2 (20) Related to obesity 4 (40) Related to CVI & obesity 3 (30) Related to trauma & obesity 1 (10)

Area of Unilateral Leg 3 (15) lymphoedema Bilateral leg 17 (85) Stage of Stage 1 0 Lymphoedema Stage 2 2 (10) Stage 3 18 (90)

The group consisted of 90% female (n=18) and 10% male (n=2) participants. The mean age was 59 years (with a range of 19 – 80 years of age). Body Mass

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Index ratings of the study population demonstrated that only 15% (n=3) had a healthy BMI, 25% (n=5) were overweight, with the remaining 60% (n=12) were in the category of obese. Of those deemed to be obese; 20% (n=4) were moderately obese, 10% (n=2) were severely obese, 15% (n=3) were very severely obese and, of note, 15% (n=3) were super obese, indicating a BMI between 50-60 kg/m2. The mean BMI rating was 36 kg/m2 (median= 35kg/m2, SD = 11). The range of BMI scoring was between 22 and 60kg/m2, with 60kg/m2 being the maximal range of super obese.

Lymphoedema aetiology was divided equally, with 50% (n=10) of the participants diagnosed with primary lymphoedema and the further 50% with secondary causes of lymphoedema. Of those with secondary lymphoedema, 20% (n=2) was related to chronic venous insufficiency, 40% (n=4) was related to obesity, 30% (n=3) was related to chronic venous insufficiency and obesity combined, and a remaining 10% (n=1) was related to trauma and obesity. Therefore, in 80% of the cases of secondary lymphoedema, obesity was a contributing factor. Unilateral lymphoedema accounted for 15% (n=3) of the participants, while 85% (n=17) of the participants had bilateral lymphoedema.

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Table 4.2 History of oedema duration/ initial diagnosis and treatment

No. of years Duration of oedema/No.(% No. of years since initial No. of years since last active participants) diagnosis treatment

No. (% of participants) Never 9 (45) 1-10 4 (20) 10 (50) 7 (35) 11-20 8 (40) 8 (40) 4 (20) 21-30 3 (15) 0 0 31-40 1 (5) 1 (5) 0 41-50 2 (10) 1 (5) 0 51-60 2 (10) 0 0 SD 4.14 4.45 3.84 MEAN 5.8 3.33 2.85

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Of the 20 participants, 40% (n=8) had experienced oedema for between 11-20 years, 20% (n=4) had experienced oedema for between 1- 10 years, 15% (n=3) have had oedema for between 21-30 years, 5% (n=1) have had oedema for 31- 40 years, 10% (n=2) had oedema for between 41-50 years, and finally a further 10% experienced oedema for between 51-60 years. The mean length of oedema duration was 24.5 years (ranging from 6 years to 57 years).

The participants reported varying length of time since initial diagnosis, but 50% (n=10) of the participants received diagnosis in the past 10 years and a further 40% (n=8) were diagnosed with lymphoedema between 11-20 years ago. Of the two remaining participants, one participant was diagnosed between 31-40 years ago and the other between 41-50 years ago.

Of note, of the 20 participants who each had a diagnosis of lower limb lymphoedema, (n=9) had never received any treatment. Of the remaining participants, 35% (n=7) had last received treatment between 1-10 years ago, and the remaining 20% (n=4) had received their last active treatment between 11-20 years ago. The mean length of time since last treatment was 3.6 years (ranging from never to 16 years).

Table 4.3 Treatment Modalities of participants (n=11) Treatment No. (%) of participants who received treatment prior to study Manual lymphatic drainage(MLD) only 1 (5) MLD + Compression bandaging (CB) 1 (5) MLD + CB + Hosiery 4 (20) Hosiery only 5 (25) SD 2.36 MEAN 2.75

Of the 11 participants who received treatment for their lymphoedema, 25% (n=5) were treated with compression hosiery only. Of the other participants, 20% (n=4) received MLD, compression bandaging and hosiery. Only one

104 participant received MLD and another one participant received MLD and compression bandaging.

4.2 Quality of Life The LYMQOL Leg questionnaire measures Quality of Life (QoL) data in the four domains of function, appearance, symptoms, and mood. The scoring with each item in each section ranges from 1 – 4, depending upon the effect that lymphoedema has on a particular activity in life; 1= not at all 2= a little 3= quite a bit 4= a lot

The score in that domain is then divided by the number of questions in that domain. The higher the score the poorer the QoL of the participant. The best possible score is 1, indicating that lymphoedema did not affect the participants’ QoL at all. The worst possible score is 4, indicating that lymphoedema has a large negative effect on the persons’ QoL. Also, an overall QoL score is presented using a range from 0 (poor) to 10 (excellent). In this case, the higher the score, the better the quality of life of the participant. The scoring system of the LYMQOL Leg Tool is presented in Appendix 12.

The QoL scoring was calculated at baseline, week 8 (at the end of the intensive treatment period and entering into independent maintenance) and week 24 (four months post maintenance period). The overall QoL score changes over the 24 weeks duration are presented in Table 4.4.

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Table 4.4 Overall QoL changes week 1 to week 8, week 8 to week 24 and week 1 to week 24

Participant Week Week Week % Change % Change % Change Week 1 to Week 8 to Week 1 to 1 8 24 Week 8 Week 24 Week 24 1 5 7 7 40 0 29 2 0 7 8 n/a 14 100 3 6 10 10 67 0 40 4 6 8 9 33 13 33 5 3 8 8 167 0 63 6 5 9 9 80 0 44 7 4 8 8 100 0 50 8 5 7 7 40 0 29 9 0 9 5 n/a -44 100 10 2 7 6 250 -14 67 11 5 8 8 60 0 38 12 4 8 100 -100 n/a 13 9 10 10 11 0 10 14 6 8 8 33 0 25 15 5 10 10 100 0 50 16 8 8 8 0 0 0 17 0 3 5 n/a 67 100 18 4 9 10 125 11 60 19 4 7 7 75 0 43 20 6 9 9 50 0 33 SD 2.43 1.56 1.56 61 30 28 MEAN 4.35 8.0 8.0 78 -3.0 48

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The overall quality of life percentage changes from week 1 to week 8 demonstrates a mean QoL scoring of 4.32 (ranging from 0 to 9).Three participants (15%) rated their QoL as 0 (poor) and only one participant rated their QoL at 9. All of the participants had an improved QoL over the 8 week period, the variance in improvement ranged from 0% to 250%. The 0% change from week 1 to week 8 is presented by the participant number 16 who rated their QoL as 8 at baseline thus they already had a very good QoL. The greater improvements in QoL were demonstrated in participants who entered the therapy program with a particularly low scoring QoL at baseline. The mean percentage QoL change from week 1 to week 8 was 78% (median 67%, SD 61%).

Overall Quality of life percentage changes from week 8 to week 24 demonstrated, a reduction in QoL scoring for three participants. Participant number 9 had a minus 44% reduction in QoL due to having developed a cellulitis and a resulting increase in limb volume. Participant number 10 had a minus 14% reduction in QoL due to difficulties with consistently wearing the lymphoedema garment and therefore the oedema was not controlled adequately. Both participants will be discussed in depth in a later chapter, with the inclusion of narrative data from their respective diaries. Also, a 100% reduction in QoL is presented by participant number 12 but this was due to the fact that this participant did not complete the week 24 QoL scoring due to ill- health and hospitalisation, which had no relevance to lymphoedema. Otherwise, overall QoL scoring either remained static, or improved, from week 8 to week 24. However, there was a reduced percentage change when compared with week 1 to week 8. The mean percentage change from week 8 to week 24 was a minus 3% with a median of 0% change. The 100% reduction due to the absence of one participant at week 24 has affected the mean percentage change scoring.

Overall Quality of life percentage changes from week 1 to week 24 demonstrated a marked improvement. The mean percentage change from week 1 to week 24 was 48% (ranging from 0% to 100% change, median 43%, SD 28%). Therefore, 90% of the participants had an improved QoL overall, from baseline to week 24. The remaining 10% was accounted for by participant number 16 who started with a high scoring QoL (8) at the commencement that 107 remained unchanged, whilst participant number 12 withdrew from the study before week 24 due to health reasons.

Over the study period from baseline through the intensive phase of treatment and into maintenance treatment the following observations can be noted:

1.An important overall QoL change was evident from baseline to week 8, with a maximum of 250% change and a mean of 78% change.

The marked improvement in QoL could be explained by the fact that 40% (n=8) of the participants had never received any treatment in the past and therefore the reduction in limb volume during intensive therapy was certain to improve the participants well-being. Also, of those who had treatment in the past (n=12/60%), only 33.3% (n=4) had received the three modalities of manual lymphatic drainage, compression bandaging, and hosiery.

2. Although there continued to be an improvement in overall QoL from week 8 to week 24, the percentage of change was reduced when compared to the week 1 to week 8 period, with a maximum change of 67% and a median of 0%.

The reduction in percentage change in QoL during the maintenance phase is likely to be explained by the fact that limb volume changes are no longer noticeable, as the volume has reached plateau and the participant is now self- caring without the input of the therapist.

3. Increase in oedema due to cellulitis or non-concordance with compression hosiery negatively affected QoL.

Participants 9 and 10 had an increase in oedema from week 8 to week 24 due to cellulitis and non-concordance with compression hosiery and therefore had reduced overall QoL scoring as the oedema had likely impacted negatively on their body image and function.

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4.3 Quality of Life scoring in the domain of function

As described previously, LYMQOL LEG yields a QoL score in each of 4 domains; function, appearance, symptoms, and mood. The domain of function has a series of 8 questions.

Each question is scored 1-4, depending on the level of affect that lymphoedema places on that activity. Table 4.5 illustrates QoL scoring in the domain of function at baseline.

Table 4.5 Baseline QoL scoring in the domain of function

Range of Scoring in function No. of Participants (%) domain 1-2 6 (30) 2-3 7 (35) 3-4 7 (35) SD 0.57 MEAN 6.66

A total of 30% of the participants felt that lymphoedema either did not affect their function, or affected them a little. Of those 6 participants, it was noted that their BMI’s ranged from 22kg/m2 to 36kg/m2, indicating that these participants were in the range of ‘healthy weight to moderately obese’. Furthermore, the age range of these participants was 20 to 74 years. In addition, their affected limb volume ranged from a mean of 2.93 litres to 8.97 litres for the participants, with bilateral lymphoedema, and an excess volume of 4.42 litres and 16.41 litres for the participants, with unilateral lymphoedema. Table 4.6 displays the characteristics of the six participants.

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Table 4.6 Characteristics of 6 participants who scored 1-2 on the Function Domain at baseline

Participant ID BMI kg/m2 Age years Mean Limb volume litres 1 23 70 2.93 3 30 74 4.2 11 36 63 8.97 13 23 54 2.75 16 22 20 4.42 (excess ) 20 26 56 16.41(excess) SD 5.42 19.33 5.30 MEAN 26.66 56.16 6.61

(excess limb volume refers to the comparison limb volume between the unaffected limb and the limb affected by lymphoedema in unilateral primary lymphoedema)

The remaining participants (n=14) reported at baseline that lymphoedema affected their function domain either quite a bit, or a lot. Table 4.7 displays the characteristics of those participants (n=7) who reported that lymphoedema affected them a lot.

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Table 4.7 Characteristics of participants scoring 3.1 to 4 in the Function Domain at baseline

Patient ID BMI kg/m2 Age years Mean Limb volume litres 5 40 36 27.44 6 43 81 15.92 9 60 46 9.08 10 54 57 31.3 12 34 78 11.83 15 45 81 7.29 17 60 52 13.75 SD 10.14 18.41 9.20 MEAN 48 61.57 16.65

The participants who were most greatly affected in the function domain all had an elevated BMI, ranging from 40kg/m2 to 60 kg/m2, putting them in the category of very severely obese to hyper obese. In addition, all of these participants had a bilateral lymphoedema and, of the 7 participants, 3 were diagnosed with primary lymphoedema. The mean limb volume of this group ranged from 7.29 litres to 27.44 litres, thus all of the participants had significant amounts of oedema on their lower limbs, which in turn has affected their mobility.

Quality of life scoring was then repeated with the participants (n=20) at week 8 and week 24. Table 4.8 illustrates the percentage of change in QoL over the 24 weeks in the function domain.

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Table 4.8 Percentage change in QoL in the domain of function baseline to week 24

FUNCTION

Participant Week 1 Week 8 Week 24 % Change % Change % Change Week 1 Week 8 to Week 1 to toWeek 8 Week 24 Week 24

1 1.50 1.25 1.00 -17 -20 -33

2 2.90 1.40 1.00 -52 -29 -66

3 2.00 1.00 1.00 -50 0 -50

4 2.20 1.90 1.40 -14 -26 -36

5 3.50 1.60 1.60 -54 0 -54

6 4.00 2.90 2.90 -28 0 -28

7 2.40 1.60 1.40 -33 -13 -42

8 2.70 1.70 1.70 -37 0 -37

9 3.90 1.60 3.10 -59 94 -21

10 3.90 2.50 2.40 -36 -4 -38

11 1.70 1.50 1.50 -12 0 -12

12 3.60 2.50 #NULL! -31 n/a n/a

13 1.10 1.10 1.10 0 0 0

14 2.60 1.60 1.60 -38 0 -38

15 3.10 1.60 1.60 -48 0 -48

16 1.10 1.10 1.10 0 0 0

17 4.00 3.40 3.10 -15 -9 -23

18 2.90 1.20 1.10 -59 -8 -62

19 2.60 1.90 1.90 -27 0 -27 20 2.00 1.40 1.40 -30 0 -30 SD 18 25 18 MEAN -32 -1 -34

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A total of 90% of the participants had an improvement in QoL from baseline to week 8. The 10% (n=2) who did not have a change in score, scored 1.1 at baseline, thus their QoL was mostly likely not affected by lymphoedema. Quality of life percentage changes ranged from -12% to -59% (SD 18%). Both the mean and median percentage change in QoL was -32%, indicating an important improvement in function from baseline to week 8, at which stage the participants completed CDT and were commencing self-maintenance in compression hosiery.

Quality of life percentage changes in the function domain from week 8 to week24 demonstrate that scores at this time showed no change from week 8 to week 24 for 11 of the participants, therefore their QoL remained improved. A further 8 participants had an increase in their reported QoL from week 8. One participant did not complete at week 24 due to medical reasons and long term care needs. Finally, participant number 9 reported a reduced QoL at week 24 in comparison to week 8. This participant had increased oedema due to cellulitis. The overall mean QoL change from week 8 to week 24 was -1% (SD= 25%). The level of change was much less from week 8 to week 24 than from baseline to week 8.

All participants had a reduced score and therefore an improved QoL in the function domain from baseline to week 24. The overall mean percentage change was -36% (min 0%, max -66%, SD: 18%). Despite having received intensive CDT, 2 participants, participant number 9 and number 17, still rated their QoL as 3.1, in the function domain. Thus, lymphoedema still affected these participants functional well-being. Both of these participants were assessed as being hyper obese (BMI 60KG/M2) but were relatively young, aged 46 and 52 years old.

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4.4 Quality of Life Scoring in the domain of appearance

The second domain of the LYMQOL LEG QoL tool is the domain of appearance. This domain consists of 7 questions .

At baseline, the scoring for ‘appearance’ is presented in Table 4.9.

Table 4.9 Baseline QoL scoring in the domain of appearance

Score No. of Participants (%) 1-2 3 (15) 2.1-3.1 6 (30) 3.1-4 11 (55) SD 4.04 MEAN 6.66

A total of 15% (n=3) of the participants reported that lymphoedema did not affect their appearance at all. Of these participants, the level of oedema was not markedly noticeable. Two participants had bilateral lower limb oedema, with a mean limb volume of 4.2 litres and 2.75 litres. The third client had a unilateral primary lymphoedema and had a total limb volume excess of 4.42 litres, which indicated a severe lymphoedema; however, as the oedema was evenly distributed over the total length of the leg, and this client was 6 foot 2 inches in height, the appearance of the limb in clothing was not noticeable. Interestingly, 55% (n=11) of the participants reported that the oedema affected their appearance a lot and the remaining 30% (n=6) felt that lymphoedema affected their appearance quite a bit. Table 4.10 presents the percentage changes in QoL scoring in the appearance domain, over the 24 week period.

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Table 4.10 Percentage change in QoL scoring in the appearance domain from baseline to week 8

Appearance

Participant Week 1 Week 8 Week 24 % Change % Change % Change Week 1 to Week 8 to Week 1 to

Week 8 Week 24 Week 24

1 2.10 1.70 1.30 -19 -24 -38

2 3.60 1.00 1.10 -72 10 -69

3 1.40 1.00 1.00 -29 0 -29

4 2.60 1.30 1.70 -50 31 -35

5 3.10 1.60 1.60 -48 0 -48

6 3.60 2.60 2.60 -28 0 -28

7 2.60 1.40 1.30 -46 -7 -50

8 3.70 2.60 2.60 -30 0 -30

9 4.00 2.30 2.80 -43 22 -30

10 2.60 2.00 2.80 -23 40 8

11 3.00 1.80 1.80 -40 0 -40

12 3.00 2.10 #NULL! -30 n/a n/a

13 1.10 1.00 1.00 -9 0 -9

14 3.60 2.10 2.10 -42 0 -42

15 3.30 1.40 1.30 -58 -7 -61

16 1.70 1.70 1.70 0 0 0

17 4.00 3.30 3.40 -18 3 -15

18 3.30 1.70 1.00 -48 -41 -70

19 3.30 1.80 1.70 -45 -6 -48

20 3.70 2.00 1.80 -46 -10 -51

SD 17 18 21

MEAN -36 1 -36

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All participants scored lower at week 8, thus indicating an improved QoL in the domain of appearance. The mean percentage change was -36% (median -41%; min 0%, max -72%, SD: 17%). The maximal changes occurred for participants who had lost significant limb volume over the 8 week period; for example, participant number 2 who had a -72% change in QoL from baseline had a total volume loss of 1.75 litres of fluid from a limb and now found it easier to get footwear.

From week 8 to week 24, during the period of self-maintenance, 40% (n=8) of participants did not have any change in QoL scores since week 8. A further 30% (n=6) of the participants continued to show improvements in QoL. However, 5 of the participants had an elevated scoring and thus a reduction in QoL. Examination of the participants’ diary highlighted that participant number 9 had a reoccurrence of cellulitis and the other 4 participants ( participant numbers 2,4,10 and 17) had issues with compliance in wearing their compression hosiery during the summer and on holidays, resulting in increased oedema and feeling of disappointment in their appearance. The mean percentage change from week 8 to week 24 was 1% (min 40%, max -41%, SD: 18%).

Over the 24-week period, only one participant, participant number 10. had an elevated QoL score from baseline, thus, the QoL with regard to appearance had reduced over the period of the treatment and lymphoedema affected this participants QoL considerably. Another participant, participant number 16 had no change in scoring over the 24 weeks but essentially had a low score and therefore did not feel that lymphoedema affected their appearance at all. The remaining 90% of participants had improved scoring from baseline to week 24. The mean percentage change in QoL from baseline to week 24 was -36% (median -38%, min 8%, max -70%, SD 21%).

4.5 Quality of Life Scoring in the domain of Symptoms

The third domain in the LYMQOL LEG quality of life tool is that of symptoms. This domain consists of 5 questions, each of which are scored between 1 and

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4, depending upon the affect that lymphoedema has on the participants’ symptoms. Baseline scoring of quality of life in the symptom domain is presented in Table 4.11.

Table 4.11 Baseline QoL scoring in the domain of appearance Range of Scoring No. of Participants (%) 1-2 8 (40) 2.1-3.1 10 (50) 3.1-4 2 (10) SD 4.16 MEAN 6.66

A total of 40% (n=8) of the participants scored between 1 and 2, thus lymphoedema affected them either, not at all, or only a little. A further 50% of the participants reported that lymphoedema affected them between, a little and quite a bit, and 10% scored their symptoms between 3.1 and 4, thus their lymphoedema affected them a lot. The low percentage of participants scoring between 3 and 4 indicates that, despite longstanding and gross oedema, this group of participants had minor difficulty with symptoms. Table 4.12 represents the QoL changes in the symptom domain over the 24 weeks

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Table 4.12 Percentage change in QoL scoring in the symptom domain from baseline to week 24 Symptoms

Participant Week 1 Week 8 Week 24 % Change % Change % Change Week 1 to Week 8 to Week 1 to Week 8 Week 24 Week 24

1 1.40 1.00 1.00 -29 0 -29

2 3.00 1.60 1.20 -47 -25 -60

3 2.20 1.00 1.00 -55 0 -55

4 1.80 1.20 1.20 -33 0 -33

5 2.60 1.60 1.60 -38 0 -38

6 2.80 1.40 1.40 -50 0 -50

7 2.20 1.40 1.00 -36 -29 -55

8 2.40 1.40 1.40 -42 0 -42

9 3.00 1.00 1.80 -67 80 -40

10 3.80 2.40 2.20 -37 -8 -42

11 1.60 1.20 1.20 -25 0 -25

12 3.20 1.60 #NULL! -50 n/a n/a

13 1.00 1.00 1.00 0 0 0

14 1.40 1.20 1.20 -14 0 -14

15 2.00 1.00 1.00 -50 0 -50

16 1.00 1.00 1.00 0 0 0

17 3.00 2.40 2.40 -20 0 -20

18 2.40 1.00 1.00 -58 0 -58

19 2.80 1.60 1.40 -43 -13 -50

20 2.00 1.20 1.00 -40 -17 -50

SD 18 22 18

MEAN -37 -1 -37

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From baseline to week 8, two participants who already had no symptoms, remained static, otherwise 90% (n=18) of the participants had a reduced score at week 8 and therefore an improved QoL in the symptom domain. The mean percentage change was -37% (median -39%, min 0%, max -67%, SD: 18%). Consequently, it may be said that 90% of the participants had a reduction in symptoms subsequent to completion of the CDT program.

At week 24, 65% of the participants remained static from their reported score at week 8 and a further 25% of the participants had reduced scores in the domain of symptoms thus had additional improvement in QoL. One participant, participant number 9, had an increased score, thus indicating an increase in symptoms and a reduced QoL. The mean percentage of change between week 8 and week 24 was a -1% (median 0%, min 80%, max -29%, SD 22%).

A total of 10% (n=2) of the participants who had no symptoms at baseline continued to have no symptoms. One participant, participant number 12, did not complete week 24 due to medical reasons. The remaining 85% of the participants all had a reduction in scores and therefore an improvement in QoL in the domain of symptoms. The mean percentage of change from baseline to week 24 was -37% (min 0%, max -60%, SD: 18%).

4.5.1 Quality of Life scoring in the domain of Mood The fourth and final domain in the LYMQOL LEG quality of life tool is the domain of mood, which asks 6 questions, all of which are scored from 1 to 4, depending upon the level of effect exerted by lymphoedema on that domain.

Baseline scoring of QoL in the mood domain is presented in Table 4.13.

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Table 4.13 Baseline QoL scoring in the domain of mood Scores No. of Participants (%) 1-2 9 (45) 2.1-3.1 9 (45) 3.1-4 2 (10) SD 4.04 MEAN 6.66

A total of 10% (n=2) of the participants felt that lymphoedema affected their QoL in the domain of mood, a lot. Of those 2 participants, participant number 11,was assessed as being hyper obese, had reduced mobility, was quite socially isolated and had a large bilateral limb volume (mean of 13.75 litres). The second participant, participant number 18,had a history of repeated cellulitis and two resulting hospitalisation to treat her cellulitis and had never been assessed in a lymphoedema service prior to the study. A further 45% of the participants did feel that lymphoedema affected their QoL quite a bit and the remaining 45% reported that their QoL and mood were not affected at all. The participants who reported that lymphoedema did not affect them at all tended to have lower limb volume than those who were affected, and also their BMI ranged from healthy to severely overweight.

The percentage of change in QoL in the domain of mood from baseline to week 24 is presented in Table 4.14.

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Table 4.14 The percentage of change in QoL in the domain of mood from baseline to week 2 Mood

Participants Week 1 Week 8 Week 24 % Change % Change % Change Week 1 to Week 8 to Week 1 to Week 8 Week 24 Week 24

1 1.50 1.00 1.00 -33 0 -33

2 2.30 1.00 1.00 -57 0 -57

3 1.30 1.00 1.00 -23 0 -23

4 1.20 1.00 1.00 -17 0 -17

5 2.30 1.00 1.00 -57 0 -57

6 2.30 1.00 1.00 -57 0 -57

7 2.60 1.00 1.00 -62 0 -62

8 2.60 1.30 1.30 -50 0 -50

9 2.80 1.00 2.60 -64 160 -7

10 1.80 1.30 1.50 -28 15 -17

11 2.80 1.00 1.00 -64 0 -64

12 2.30 1.00 #NULL! -57 n/a n/a

13 1.00 1.00 1.00 0 0 0

14 1.00 1.00 1.00 0 0 0

15 1.00 1.00 1.00 0 0 0

16 1.00 1.00 1.00 0 0 0

17 4.00 2.80 2.00 -30 -29 -50

18 3.60 1.30 1.00 -64 -23 -72

19 2.60 1.50 1.60 -42 7 -38

20 1.30 1.00 1.00 -23 0 -23

SD 24 38 25

MEAN -36 7 -33

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As illustrated in Table 4.14, 4 scores for participants who reported that lymphoedema did not affect their mood, remained unchanged from baseline to week 24, those were participants number 13, 14, 15 and 16. Meanwhile, from baseline to week 8, all other participants (n=16) had a reduced scoring and therefore an improved QoL in the domain of mood. The mean percentage change from baseline to week 8 was -36% (mode -57%, min 0%, max -64%, SD: 24%)

The majority of participants (n=14, 70%), from week 8 to week 24, remained unchanged in their scoring of QoL in the mood domain, with a score of 1. Thus, their mood remained unaffected by lymphoedema. Two further participants, participant number 17 and 18, had a further reduction in scoring, thus they were affected ‘a little’ by lymphoedema. Three participants had an increase in scoring and were therefore negatively affected by lymphoedema; participant number 9 had developed a cellulitis, and the other two participants, participant number 10 and 19, were inconsistently wearing their lymphoedema garments and therefore had increased swelling in their limbs. The mean percentage change was 7% (min 160%, max -29%, SD 38%).

Overall, from baseline to week 24, 75% (n=15) of participants had an improved QoL in the domain of mood. One participant did not complete the program to week 24. The subsequent 20% (n=4) of participants remained unchanged and had originally scored 1; therefore they were not affected by lymphoedema in the domain of mood. The mean change in percentage in QoL in the domain of mood was -33% (min 0%, max -72%).

4.5.2 Summary of QoL changes in the 4 domains from baseline to week 24

Function

A total of 30% (n=6) of the participants felt that lymphoedema did not affect their function at baseline or affected their function a little. Of those 6 participants, it was noted that their BMIs ranged from 22kg/m2 to 36kg/m2. The participants who were most greatly affected in the function domain all had an elevated BMI, ranging from 40kg/m2 to 60 kg/m2, thus were classed in the category of very 122 severely obese to hyper obese. Further, these participants also had bilateral lymphoedema. A total of 90% of participants (n=18) had an improvement in QoL from baseline to week 8. However, one participant reported a reduced QoL at week 24 in comparison to week 8, due to an episode of cellulitis. All participants had a reduced score and therefore an improved QoL in the function domain from baseline to week 24.

Appearance

A total of 15% (n=3) of the participants reported that lymphoedema did not affect their appearance at all. All participants had a reduced score at week 8, thus indicating an improved QoL in the domain of appearance. A further 30% (n=6) of the participants continued to show improvements in QoL into week 24. However, 5 of the participants had an elevated score and thus a reduction in QoL, due to exacerbation of the oedema secondary to cellulitis in one participant, while the other 4 participants were non-compliant in wearing with compression hosiery, during the summer and on holidays, resulting in increased oedema.

Symptoms

At baseline, 40% (n=8) of the participants scored between 1 and 2, thus lymphoedema affected them either not at all, or only a little. A further 50% (n=10) of the participants reported that lymphoedema affected them between, a little and quite a bit, and only 10% (n=2) scored their symptoms between 3.1 and 4, thus their lymphoedema affected them a lot. From baseline through to week 24, 85% (n=17) of the participants had a reduction in scores and therefore an improvement in QoL in this domain. One participant did not complete the program and the remaining participants remained unchanged and symptom free.

Mood

A total of 10% (n=2) of participants felt that lymphoedema affected their QoL a lot, in this domain. Of those 2 participants, the first participant was assessed as being hyper obese, had reduced mobility, was quite socially isolated and has a significant mean bilateral limb volume of 13.75 litres. The second participant 123 had an ulceration as a complication of lymphoedema and this participant had never been assessed in a lymphoedema service prior to the study.

Overall, from baseline to week 24, 75% (n=15) of the participants had an improved QoL in this domain. One participant did not complete the program to week 24. The remaining 20% (n=4) of participants remained unchanged but had originally scored 1 and therefore were not affected by lymphoedema in this domain.

On examination of the 4 domain scorings from baseline to week 24, and at 3 time periods (baseline, week 8 and week 24), it would appear that the domains of function and appearance are the areas of QoL that are most greatly impacted by lymphoedema. Although, symptoms and mood are a concern for this cohort of patients, the impact of these two domains is not as notable as the impact on function and appearance. Between 70% (n=14), to 85% (n=17), of the participants in this study reported that function and appearance were affected either, quite a bit, or a lot, by lymphoedema, whereas only 10% (n=2) of the participants reported that symptoms and mood were affected either, quite a bit, or a lot, by lymphoedema.

4.6 Limb Circumferential and Volume changes

Circumferential measurements were recorded weekly for the first 4 weeks and then at weeks 8, 16, and 24. The aim of recording these timeline measurements was to monitor the progress or otherwise of the treatment and the participants’ self-maintenance period. Circumferential measurements were taken using a tape measure at 4cm intervals from a fixed point at the ankle. The tape measure was specifically designed for limb measurements and each 4 cm interval was designated on the tape in order to avoid error. The circumferential measurements in centimetres were then converted to a volume measurement in litres using an excel program based on the truncated cone formula and as the circumferential measurements were fed into the program they were automatically converted into volume. Table 4.15 presents limb volume percentage changes at the specified timeframes.

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Table 4.15 Percentage change in limb volume week 1/ week 8/ week 24

Percentage change(%) in limb volumes at the specified timeframes

Pt Week 1to Week 1 to Week 8 to Week 8 to Week 1 to Week 1 to ID Week 8 Week 8 Week 24 Week 24 Week 24 Week 24 Right Leg Left Leg Right Leg Left Leg Left Leg Right Leg

1 - 2.39 8.37 0 0 -2.39 8.37

2 -6.12 -2.96 -2.36 0.32 -8.33.5 -2.65

3 -12.35 0.28 -0.27 0.00 -12.59 0.28

4 -11.90 -3.90 -2.865 -3.24 -14.42 -7.01

5 -19.25 -21.48 23.93 25.51 0.07 -1.45

6 -11.92 -14.65 0.64 0.95 -11.35 -13.84

7 -18.08 -15.36 0.39 0.00 -17.76 -15.36

8 -6.90 0.99 0.45 -0.98 -6.48 0.00

9 -14.22 -7.41 14.51 9.91 -1.76 1.76

10 -23.75 -7.33 15.46 6.15 -11.96 -1.63

11 -18.40 -16.72 -0.27 0.27 -18.63 -16.50

12 -6.59 -9.86 n/a n/a n/a n/a

13 -12.64 -18.15 -0.43 -0.43 -13.01 -18.51

14 -18.21 -18.72 0.45 0.68 -17.85 -18.17

15 -26.89 -23.46 0.19 -0.18 -26.75 -23.59

16 0.0 -24.27 0.0 0.11 0.00 -24.19

17 -27.48 -27.87 0.11 0.00 -27.40 -27.87

18 -27.79 -29.70 -0.62 4.53 -28.23 -26.52

19 -15.46 -6.94 0.00 0.00 -15.46 -6.94

20 0.00 -40.85 0.00 -1.08 0.00 -41.49 sd -8.66 12.07 7.11 6.35 9.19 12.87

M -14.02 -14.00 2.60 2.24 -12.33 -12.38 n

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The first timeframe was from week 1 (baseline) to week 8. All 20 participants had oedema reduction although in varying volumes depending upon the starting oedema volumes. Three participants showed a small increase in volume in one leg. Participant number 1 had a percentage volume increase of 8.37% in the left leg; however, this participant had acquired an injury to her left leg during work which had resulting bruising and oedema. Participant number 3 had a 0.38% (10ml) increase in oedema in the left leg for no apparent reason, whilst participant ID 8 had a 0.99% (24ml) increase in oedema to the left leg. Co- Morbidities may have caused these alterations as mild changes in body fluid volume can be associated with many co-morbidities, including renal impairment, venous disorders, hypothyroidism, and also the participants’ level of activity and mobility

Three participants, participant numbers 8,16 and 20, with unilateral primary lymphoedema had no change in the unaffected limb, but important changes were noted in the affected limb to a maximum percentage volume change of 40.85% (10.92L) for participant ID 20.

The mean percentage volume change for the 20 participants was -14.97% (SD 8.76%), with a maximum mean percentage change of -29.47% and a minimum change of 2.7%

The measurements taken from week 8 to week 24 displayed many minor increases in percentage limb volume and three important exacerbations of oedema. Week 8 to week 24 is the period during which the participants were self-caring for their limbs and no longer were in compression bandaging but were wearing their compression hosiery. This initial phase of self-maintenance can be quite challenging, despite the fact that they have received education and training in all the aspects of self-care.

The minor percentage of limb volume changes would lead one to consider the many variables that may have affected volume change, such as:

 The participants were wearing compression hosiery, which may not have been providing the same static pressure gradient as the compression bandaging. After week 8, all the participants were wearing flat knit class 126

2 compression stockings, which exert a 23-32mm/Hg pressure at the ankle. Prior to this, and during the intensive phase of CDT, a cohesive short stretch bandaging system (Actico) was used in this study, which would have exerted pressures ranging from 29-65mm/Hg.  The participants were not fitted with night-time garments and may have had some refill of oedema to the limbs during the night when their compression was off. The first 2-4 months of maintenance usually requires regular monitoring in order to identify the patients that may require further interventions such as night bandaging, night garments or increased levels of compression in hosiery.

The three participants who had significant increases in oedema from weeks 8 to 24 were Participants ID 5, 9, and 10. Participant ID 5 had concordance issues with wearing her compression hosiery and required assistance of a family member to apply them. In addition, the summer weather was a challenge, as the participant found the compression hosiery too warm to wear in the heat. Participant ID 9 developed a cellulitis in the right leg and was hospitalised for treatment. During the acute treatment, the compression was removed and had not been reapplied when the cellulitis had settled. Participant ID10 also had difficulties with applying compression hosiery due to reduced dexterity and flexibility caused by obesity. This participant also found that the compression hosiery was uncomfortable to wear after a period of time and therefore compliance with wearing the garment was low.

The mean percentage of limb volume change from weeks 8-24 (maintenance period) was a 2.43% increase in volume (min -3.05%, max 24.71%).

From the commencement of treatment at week 1 through to the completion of a 4-month maintenance period, a total of 6 months, 80% (n=16) of the participants had a reduction in limb volume. The mean percentage change in limb volume from week 1 to week 24 was -13.15% (min 2.70%, max -29.93%, SD 9.72%). Only 4 participants had an overall increase in limb volume: participant ID 1 had an 8.37% increase in volume to her left leg due to injury, participant ID 3 had a 0.28% increase to the left leg for an unknown reason, participant ID 5 had a 0.07% increase in volume to the right leg due to non- concordance with wearing compression hosiery, and participant ID 9 had a 127

1.76% increase in overall volume to the left leg due to cellulitis. Of note, both participants ID 5 and 9 had a significant reduction in bilateral limb volume from week 1 to week 8; however, this reduction in limb volume could not be maintained due to non-concordance and cellulitis.

Circumferential measurements were recorded weekly for the first 4 weeks and then at week 8, 16 and 24. The aim of recording these timeline measurements was to monitor the progress, or otherwise, of the treatment and the participants self-maintenance period during the first timeframe. From week 1 (baseline) to week 8 all 20 participants had oedema reduction in varying volumes depending on the initial starting oedema volumes. Three participants showed a small increase in volume in one leg. The mean percentage volume change for the 20 participants was -14.97% (min 2.7%, max -29.47%, SD 8.76%).

The timeframe from week 8 to week 24 displayed many minor increases in percentage limb volume and three significant exacerbations of oedema due to non-concordance in 2 cases and cellulitis in the other case. The mean percentage of limb volume change from week 8 to 24 (maintenance period) was 2.43% increase in volume. From the commencement of treatment at week 1 through to the completion of a 4-month maintenance period, a total of 6 months, 80% (n=16) of the participants had a reduction in limb volume. The mean percentage change in limb volume from week 1 to week 24 was -13.15% (SD 9.72%).

4.7 Self Efficacy

Self-efficacy refers to the level of confidence with which a person feels that they can complete a task. At week 24 of this study, a self-efficacy tool was used to assess each of the participants’ level of confidence in being able to self-care for their limbs. Each participant had entered into the maintenance phase at week 8, equipped with both verbal and written information (Appendix 6, 7, and 8) on the four cornerstones of self-care in lymphoedema. By week 24, the participants had been self-caring for 16 weeks. The participants had also been completing a diary on a daily basis, which provided narrative data on their daily self-care

128 routine. The possibility that the participant would only write the narrative in the diary that they felt would best please the therapist and researcher was a threat to the veracity of the data. Therefore, the self-efficacy tool provided an objective measure of the participants’ perceived confidence in their ability to care for themselves.

In the absence of a disease specific self-efficacy tool, the researcher chose the Patient Reported Outcomes Measurement Information Systems (PROMIS), a validated and reliable self-efficacy tool for the management of chronic conditions. The PROMIS Self-efficacy for managing chronic conditions consists of 5 domains, daily activities, emotions, social, symptoms, and medications. For the purpose of this study, the domain of medication was omitted, as in general, the self-care of lymphoedema does not include medication management. Each domain consists of a short form questionnaire consisting of 4 questions. Each question is awarded a score between 1 and 5, as follows:

 1 = I am not at all confident  2= I am a little confident  3= I am somewhat confident  4= I am quite confident  5= I am very confident

The higher the score, the greater the level of confidence. Each domain consists of 4 questions and the cumulative scores for each domain can span from 4 to 20.

4.7.1 Short Form for Self-Efficacy for Managing Daily Activities The domain short form Self-efficacy for managing daily activities, consists of 4 statements, as follows:  1 – I can perform my household chores  2 – I can go shopping and run errands  3 – I can walk around inside my house  4 – I can maintain a regular exercise program

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Table 4.16 presents the cumulative scores in the domain of daily activities for each participant.

Table 4.16 Cumulative Scores – Self-efficacy for Managing Daily Activities

Pt ID Score Distribution 1 20 2 20 3 20

4 20

5 16 Mean: 16.5 6 04 7 20 Min: 4 8 16 Max: 20 9 11 SD: 4.9 10 15 11 16 13 20 14 16 15 16 16 20 17 05 18 19 19 19 20 20

The mean score for this domain was 16.5 (min 4, max 20, SD 4.9). A total of 42% (n=8) of participants scored 20. Another 47% (n=9) of the participants scored between 10 and 19 and 10% (n=2) scored below 10 to a minimum of 4. Once again, the majority of the participants were either quite confident or very confident that they could manage their daily activities. Participant ID 17 had a low score due to reduced mobility and obesity, which was further compounded by lower limb lymphoedema, causing the participant to be dependent on their 130 family for all household chores and shopping. Participant ID 6 had the lowest score of 4 and was totally dependent on her family for all activities of daily life due to obesity, reduced mobility, and lower limb lymphoedema. She also was one of the older participants, aged 77 years.

4.7.2 Short Form for Self-Efficacy for Managing Emotions The Short Form for Self-efficacy for managing emotions consists of the following 4 statements that are rated from 1-5, depending on the participants’ level of confidence in dealing with that particular emotion. The 4 statements are listed below:  1 - I can handle negative feelings  2 - I can find ways to manage stress  3 - I can avoid feeling discouraged  4 - I can bounce back from disappointment Table 4.17 presents the cumulative scores in the domain of emotion for each participant (n=19).

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Table 4.17 Cumulative Scores - Self-efficacy for managing emotions Pt ID Score Distribution 1 18 2 15 3 20

4 16

5 12 Mean: 14.6 6 09 7 20 Min: 6 8 10 Max: 20 9 11 SD: 4.59 10 10 11 20 13 20 14 20 15 20 16 13 17 06 18 14 19 10 20 14

The mean score in the domain of emotion was 14.6 (SD 4.59), indicating that the majority of the participants were confident in dealing with their emotions. The maximum score was 20, with 31.5% (n=6) of the participants achieving this score. Thus, these participants felt very confident in dealing with the presented emotions. The minimum score was 6 and this came from one participant, Participant ID 17, indicated that they were only a little confident in coping with the emotions presented. This participant had a diagnosed anxiety disorder and had difficulties with social isolation.

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4.7.3 Short Form for Self-Efficacy for Managing Social Interactions Self-efficacy for managing social interactions short form consisted of four statements, as follows:  1 - I can talk about my health problems with someone  2 - If I need help, I can find someone to take me to the doctors’ office  3 - I can get emotional support when I need it  4 - I can ask for help when I don’t understand something Table 4.18 presents the cumulative scores in the domain of social interactions for each participant.

Table 4.18 Cumulative Scores – Self-efficacy for Managing Social Interactions

Pt ID Score Distribution

1 20 2 20 3 20

4 20

5 16 Mean: 18.9 6 20 7 20 Min: 12 8 20 Max: 20 9 12 SD: 2.33 10 20 11 20 13 20 14 20 15 20 16 16 17 15 18 20 19 20 20 20

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The mean score in the social domain was 18.9 (min 12, max 20, SD 2.3). A total of 78.9% (n=15) of the participants scored a 20. In the domain of social functioning, it appeared that the participants were mostly very confident that they could manage the situations presented. The statements in this social interaction domain mainly refer to asking for help and emotional support. Notably, the participants in this study were all well supported by family members despite otherwise being somewhat socially isolated.

4.7.4 Short Form for Self-Efficacy for Managing Symptoms Self-efficacy for managing symptoms short form consisted of 4 questions, as follows:  1 - I can manage my symptoms during my daily activities  2 - I can keep my symptoms from interfering with relationships with friends and family  3 - I can manage my symptoms in a public place  4 - I can work with my doctor to manage my symptoms

Following the pilot run of this tool for suitability in this study population, it was decided that the term “symptom” would suggest “swelling” and the term “doctor” would suggest “therapist”. Table 4.19, presents the cumulative scores in the domain of symptoms for each participant.

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Table 4.19 Cumulative Scores – Self-efficacy for Managing Symptoms

Pt ID Score Distribution 1 18 2 20 3 20

4 16

5 11 Mean: 16.5 6 12 7 16 Min: 9 8 15 Max: 20 9 12 SD: 3.43 10 14 11 16 13 20 14 18 15 20 16 18 17 09 18 17 19 13 20 20

The mean score in this domain was 16.05 (min 9, max 20, SD 3.43). A total of 26.3% (n=5) of the study population scored 20. A further 68.4% (n=13) of participants scored between 10 and 19 indicating that the participants felt that they were quite confident that they could cope with their symptoms. One participant (participant ID 17) scored below 10. This participant was in the classification of ‘super obese’ and her mobility and daily activities were greatly impacted by her obesity.

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4.7.5 Summary of Self-efficacy scoring

Self-efficacy refers to the level of confidence with which a person feels that they can complete a task. At week 24 a self-efficacy tool was used to assess each of the participants’ level of confidence in being able to self-care for their limbs. In the absence of a disease specific self-efficacy tool the researcher had chosen a validated and reliable self-efficacy tool for the management of chronic conditions. The PROMIS Self-efficacy for managing chronic conditions consists of 5 domains; daily activities, emotions, social, symptoms and medications. For the purpose of this study the domain of medication was omitted. Each question is awarded a score between 1 and 5 The higher the score the greater the level of confidence. Each domain consists of 4 questions and the cumulative scores for each domain can span from 4 to 20.

The mean score in the domain of emotion was 14.6 (SD 4.59) therefore indicating that the majority of the participants were quite confident in dealing with the emotions that were presented in the statements. In the domain of social functioning it appeared that the participants were mostly very confident that they could manage the situations presented. The mean score in the social domain was 18.9 (SD 2.3). The mean score in the domain of symptoms was 16.05 (SD 3.43), 68.4% (n=13) of the study population scored between 10 and 19 and one participant (participant ID 17) scored below 10. The mean score for the domain of daily activities was 16.5 (SD 4.9). The maximum score of 20 was rated by 42% (n=8) of the study population. Another 47% (n=9) of the participants scored between 10 and 19 and 10% (n=2) scored below 10 to a minimum of 4.

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Chapter 5 Results - Qualitative Findings of Interviews

This chapter will present the qualitative findings from the semi-structured interviews, which explored the participants’ experience of living with lymphoedema, their experiences of Complex Decongestive Therapy, and the perceived challenges that maintenance therapy posed for the participants.

5.1 Thematic Analysis In this study, the researcher used inductive thematic analysis to obtain common patterns and themes from the data of the transcribed interview texts from the 18 participants, using Braun and Clarke’s (2006) six phase framework. This framework provides a structure for the researcher, whereby the data are reduced and categorised into themes, which directly relate to the answering of a research question.

Figure 5.1 Braun and Clarke (2006) Six Phase Thematic Framework

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5.2 Familiarisation with data First, after the completion of each interview, the researcher transcribed each interview and then read and reread these, taking notes and identifying common words, codes, and themes. The researcher had to be objective and clear in the understanding of each transcribed response in order not to be influenced by her own perceptions of the meaning of what participants talked about. The transcribed interviews were returned to the participants to ensure that they were content that the transcriptions were accurate and reflected what they said during the interview. Every participant agreed with the accuracy of the interview transcription.

5.3 Generating Initial Codes In each transcript, statements with reference to the data were extracted and noted. Words and statements were grouped together in order to give meaning, these were then coded. Attention was given to repeated patterns or coding when all transcripts were amalgamated and the codes then began to form common themes and subthemes between all the transcripts. An example of an initial coding framework is displayed below;

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Table 5.1 Example of Initial Coding Framework

Interview Interview Transcript Initial coding Framework

P 10 Q. Tell me about your experience of finding diagnosis?

I feel the medical profession has

failed me. As a consultant once said to me, when i asked, her did she Health professionals lack have an opinion as to why there of knowledge on weren’t too many medical staff lymphoedema informed or why there weren’t too many Consultants in Lymphedema and she said well it’s not cool, No recommendations

Q. Tell me about your experience of seeking therapy?

I heard from a private therapist that they did do manual lymphatic

drainage, and I rang them up but it was very expensive

I’ve had a medical card for years and nobody could tell me where there was anybody who could do lymphatic drainage, massage, or Financial burden bandaging or anything about it,

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Interview Interview Transcript Initial coding Framework

through my medical card, nobody knew, not even my own G.P to be

quite honest,

Trust / information

5.4 Searching for themes

The next stage of analysis involved combining phrases and similar codes to form themes (Appendix 18). In order to help visualisation of the formation of themes, the researcher developed a thematic map (Figure 5.2) displaying the blending of common words and codes to ultimately form themes and subthemes that clearly present the dataset of the interviews in relation to the research questions.

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CLUSTER OF BROAD THEMES

Psychosocial

Delayed Concept of low Physical diagnosis importance of LOWER LIMB LYMPHOEDEMA limb swelling IMPACT OF LYMPHOEDEMA Absence of LACK OF KNOWLEDGE BY referral HEALTH pathway Financial PROFESSIONALS Negative body image Absence of Social isolation health Failure to information diagnose Negative Heaviness/ emotions Reduced no. tiredness of certified POSITIVE Delay in practitioners Reduced mobility treatment/ THERAPEUTIC inappropriate tx RESULTS Cost of garments / Where to treatment refer? Anxiety, worry

Enjoyable, Trust and security Inability to relaxing Lost and work frustrated

Figure 5.2 Thematic Map

5.5 Reviewing Themes

In this stage of analysis, the researcher examined the transcripts again, looking for any codes that may have been overlooked or missed in order to ensure that the emerging themes were complete and gave a thorough picture of the thoughts and experiences of the participants of the study. This process then led to the final naming of themes and subthemes that represented the data transcribed from the interviews.

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5.6 Defining and naming themes

Each theme, of which there were three, were named and then divided into subthemes to further explain, in depth, the meaning of each theme. Table 5.2 displays the named themes and subthemes

Table 5.2 Final Themes and Subthemes

NO. THEME SUBTHEME Theme 1 Health professionals lack of  Delayed diagnosis knowledge in the field of and inappropriate lymphoedema treatment.  Concept of low importance of limb swelling  Absence of referral pathways  Absence of patient information

Theme 2 Impact of Lymphoedema  Negative body image  Psychosocial  Social isolation  Physical  Negative emotions  Financial  Symptoms  Reduced mobility  Cost of treatment and garments  Inequality of services  Loss of earnings

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NO. THEME SUBTHEME Theme 3 Therapeutic benefits of CDT  Compression: A means to an end  Positive physical outcomes  Positive emotions

5.7 Theme One: Health professionals lack of knowledge in the field of lymphoedema

This theme explores the participants’ experience of living with lower limb lymphoedema and the lengthy road to diagnosis, the thirst for information and knowledge about their condition, and appropriate treatment.

5.7.1 Delayed diagnosis

Ten of the participants, all of whom had a secondary lymphoedema, described how they had presented their medical condition to various different health professionals along their journey but yet were not given a diagnosis. One participant (P 6) started to develop lower limb oedema in her twenties and, almost by default, following a consultation with her diabetes nurse, was referred to the local leg ulcer clinic where there happened to be a nurse who specialised in lymphoedema. This participant was now 77 years old and had a stage 3 lower limb lymphoedema.

P6 “I suppose it would be 10 years ago anyway. I was with the Diabetic Nurse and I told her about it, that my legs were getting sore and all of that. So it was the Diabetic Nurse that recommended me, for someone to see them, yourself or somebody like that. I was 20 when it all first started, I’m 77 years old now and I’m hearing for the first time that I have lymphoedema.”

Other participants had similar experiences of delayed diagnosis despite bringing their condition to the attention of the medical profession.

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P5 “The GP gave me painkillers and injections that’s all.”

P11 “I’ve been attending the Doctor all the time because I have a lot of health problems and I attend an Endocrinologist for my Diabetes and lymphoedema was never mentioned to me.”

P13 “It was the summer of 1999 I think, I had gone to my Doctor because my ankles were swelling and she put me on diuretics for a while. Then by 2001 or around that time maybe I started noticing the pitting, that if you put your finger into it, that it would stay, more diuretics!”

P15“I just thought it was part of me, I never knew I could do anything about it. Never, and no one ever suggested it. All they did was give me water tablets.”

P7 “The GP started me on the fluid tablets, he thought it was fluid, he said there was a lot of fluid building up and then it kind of got a bit discoloured then, do you know, and it started running then like as if an ulcer was on it”

Based on what participants said, it would appear that many physicians considered diuretic therapy as the first line of treatment for oedema and even when the swelling had not resolved over the short-term a further line of investigation was not considered. The majority of the participants were quite young when they first presented with oedema and would not have had other co- morbidities at that stage. Also, for consideration, was the fact that 50% of these participants were between 60 and 80 years old and would have had confidence in the decisions of their doctors and would not have questioned this or looked for other advice. They would not have been familiar with online information and therefore relied on their doctor to be the expert in all medical conditions

5.7.2 Concept of low importance of lower limb oedema

The participants described the feeling that their lower limb oedema was not considered a priority or indeed a symptom of concern with the medical profession. The participants themselves recalled that in the initial stages of swelling development that they too ignored or didn’t consider it something that required them to seek help or treatment. It was only when the swelling became more noticeable and they developed symptoms that they then started to question why it wasn’t going away or was there something that could be done to treat it. The following quotes reflect this,

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P4 “I was just too busy, to be honest with you, you know what I mean, you have a few small babies you are not looking at those things. I just thought it was just one of those things.”

P7 “I kept working away with it in fairness, but it was hard, you know. One of the workers in the yard said “you may get something done with that”, and I just kept going. Eventually, it was getting too hard and I had to do something about it.

P20 “So at that stage then, I had five young children and it was impossible for me to travel to Dublin every day for three weeks for treatment, so to be honest I didn’t do anything. I hid it under a trousers and nobody seen it only me, and I never imagined it to go the way it went”.

P3 “No. As far as I know the GP didn’t really know what it was and they weren’t particularly concerned about it as far as I can remember.”

Interestingly, as 90% of the participants were female, many of them started to develop symptoms of swelling in association with pregnancy and weight gain. It is also evident from this cohort of participants that they prioritised their role as a mother above their own health concerns at the time and only started to look for help when the oedema started to affect their role as a mother and carer. Participant 20 did have a diagnosis of primary lymphoedema and was offered treatment but as she states above she had five small children and it just wasn’t possible for her to take the time out to travel for treatment on a daily basis because she could not ask anyone to mind her children for that length of time. At a later stage this participant also stated,

P20 “ When I knew that it wasn’t going to kill me I decided to just cover it up and get on with raising the family”

5.7.3 Absence of referral pathways

A common theme with many (n=13) of the participants was the fact that even if the General Practitioner was aware of lymphoedema, they either did not refer onwards or did not know where to refer their patients for treatment due to the absence of established referral pathways. The following participants talked about this issue,

P11“I mentioned to my GP that I wanted to see a specialist about my swelling and he was talking about some treatment, you know, probably 145

something like what you did, but that was it, he never recommended or never did anything about it.”

P10 “I feel the medical profession has failed me. My husband and I, we both asked a consultant if she had an opinion as to why there weren’t too many medical staff informed or why there weren’t too many Consultants around who specialised in lymphoedema and she said “well, it’s not cool.”

P8 “I’ve had a medical card for years and nobody could tell me where there was anybody who could do lymphatic drainage, massage, or bandaging or anything about it, through my medical card, nobody knew, not even my own G.P to be quite honest.”

P2 “Nobody could tell me, I was banging my head against the wall, and nobody knew anything.”

P18 “The second time that I was hospitalised with cellulitis, one of the nurses suggested to me that one of the physiotherapists might be able to do something about my leg.”

The participants (n=11) who had accessed treatment for their lymphoedema would seem to have been fortunate to meet someone in the health professions who might have had some exposure to lymphoedema and who then guided the participant to a professional with expertise in the field. Of those who accessed treatment, 7 participants got treatment which included CDT in the private clinic setting and the remaining 4 participants got compression stockings from a local health centre setting. However, this referral seemed to rely on luck rather than a formal referral pathway to treatment and care.

5.7.4 Absence of patient information

For all of the participants, the search for information about lymphoedema and treatment was a difficult and sometimes frightening experience for the participants. Patient information was not available in their doctor office, as would be the case with other chronic conditions such as diabetes or heart disease. The main source of health service information focused around the lymphoedemas secondary to cancer and the treatment of cancer. The lack of readily available information either meant that the participant remained without knowledge or perhaps acquired information from websites and Facebook pages. These are some of the main points that participants raised in relation to this:

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P5 “I remember looking up a website or a book about it alright, but not from medical people, no.”

P11 “One lady said to me in the Doctor’s office, “don’t research Lymphoedema whatever you do “and of course I did, and I saw what could happen and oh God.”

P7 “If I had of read about that, let’s say, without you telling me about it, I would have assumed that, that was for someone else that was a lot worse off than me because you have no reference points on these websites as to who they are talking to.”

P15 “I’ll be honest. I went and looked it up on the net and I saw some legs that I didn’t want to see and I said “oh my god”, and then I said “why didn’t somebody else say this to me.”

P13 “I read up about it and discovered that I possibly had fewer lymph vessels than other people and I even went on the internet to see if I could try to get treatment abroad.”

Many of the statements above describe fear of what they saw on internet pages when they started to research the condition. As one participant stated, “you have no reference point as to who these webpages are talking to” and this can cause anxiety in a situation where expertise and treatment is not easily accessible.

5.8 Theme 2: The impact of lymphoedema: psychosocial, physical and financial

Lymphoedema is a condition that affects the sufferer in many ways. Participants talked in detail about the many ways in which lymphoedema had affected their quality of life, psychosocially, physically, and financially.

5.8.1 The psychosocial impact of lymphoedema

The psychosocial impact of lymphoedema has been divided into 3 subthemes, these are:

 Negative body image,  Social isolation,  Negative emotions.

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5.8.1.1 Negative Body Image

All of the participants discussed the devastating impact that the changes in their physical appearance has had on their body image. They talked about how they use clothing to hide their limbs and also the way in which lymphoedema has affected their personal relationships in a negative way as they no longer feel attractive. What these participants said reflected this.

P20 “It was getting bigger and I would just get bigger trousers and probably was so busy rearing the children that I never really took that interest in the leg but then I realised then, it was just gone out of control and nothing would fit me, I couldn’t get shoes to fit me. I couldn’t hardly climb the stairs because of the weight of the leg, and then it did start affecting lots of parts of my life.”

P2 “It did make a difference as a youngster because I didn’t wear skirts and things like that. I was ashamed of them, used to hide them. Then, as I got older I sort of put up with it, got used to it and then it got to a point where they really started to swell up and I couldn’t take it anymore and started to seek help.”

P4 “I’d wear a pair of jeans or a pair of leggings or something and get on with it.”

P8 “Yes it affects your feeling of attractiveness, it affects your sex life, not just how you appear to your partner but also how you feel. The intimacy with your partner suffers even if they’re quite willing to deal with you.”

Ten participants spoke about the difficulty that they felt when allowing the therapist to see the limb and work with the skin. Two participants said the following.

P 9 “The first times were awkward because like I haven’t been used to showing my skin.”

P20 “With the size of your leg, when you have Lymphoedema, it’s bad enough to have to take off your trousers but then for someone to have to actually try and work through the skin and try and work through this, I can’t even describe emotionally how it feels, you feel so self-conscious.”

All of the participants spoke about hiding their legs with big trousers and 2 participants shared the feelings of denial of their condition in order to help them get on with their lives and raise a family.

P20 “I hid my legs and I didn’t even want to look at them myself, I avoided mirrors and avoided any opportunity for others to see my legs. 148

P8 “If you don’t see it, it’s not there”

Overall, the psychological impact of allowing a therapist to see the affected limbs and massage them has a tremendous impact on the person with lymphoedema and it requires great sensitivity on behalf of the therapist in order to allow the patient to feel comfortable.

5.8.1.2 Social Isolation

A common theme throughout the transcripts was the social isolation that the participants felt due to their altered body image, which made the participants feel that they could not allow people to see their limbs. In addition, they described the avoidance of social outings and some participants totally withdraw from society and stayed in the security of the home. These are some examples of what participants said:

P18 “I can’t do what I used to do and also what you wear, there only so much you can do with a black pair of trousers to dress it up, so socially I don’t do that much unless it’s a family occasion that’s basically it.”

P20 “I wouldn’t go to the beach with the sight of the leg, I wouldn’t go swimming with the kids, I couldn’t go on amusement rides because I couldn’t actually get into the ride, I couldn’t get the bar to come down over the leg because the leg wouldn’t bend enough to let the bar come down. I wouldn’t go on a plane because of the pressure in the plane.”

P8 “You know you get invited to a wedding or there is an occasion and it just deflates you, because you just think, “what am I going to wear”, it was something, like I had to hide it and now all of a sudden I have to expose it and it’s different. You just think “oh I don’t care who sees it, I don’t care”, you just don’t care and it’s an awful way to feel because you do care, it eats you up inside.”

P5 “As for going out to a pub, socialising, not worrying, just forget it. It’s too hard to be comfortable with that level of pain and just feeling uncomfortable, feeling your leg filling up and squeezing up and then you know, when that happens you also feel more tired so I’d always be the first person to leave a gathering. It became a bit of a joke, “you can invite x but be sure she won’t come”.

Participants discussed the effect that their social isolation had on their family and relationships. The data highlighted the fact that lymphoedema affects the extended family and friends, not just the person themselves.

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P20 “My kids would often say to me, “Mam we never did this together” or “we never got to do that” or whatever, but it would have just been easier for me to say to them, “ah we’ll do it another time” or “we’ll do that another time”. I would just cancel going.”

P9 “If we were invited to a social occasion I just wouldn’t go and then that affected my husband because he’d want to go and he would be sorry for me, so yeah it affected my whole family life”.

P8 “My daughters are all starting to grow up now and I have lymphoedema going on eight years and my oldest girl is twenty-two and my other girl is sixteen and she has never seen me in a dress and she has never seen me in high-shoes.”

Participants discussed feelings of “guilt”, having allowed lymphoedema to prevent them from being involved in normal activities with their families such as foreign holidays due to the fear that flying would cause an increase in swelling. The social isolation caused by altered body image can prevent the person with lymphoedema from going to events and social gatherings with their family. Therefore, the extended family are also affected by lymphoedema, as it restricts their ability to have a normal family life.

5.8.1.3 Negative Emotions

Throughout the period of thematic analysis, the striking tone of all the interviews was that of negative emotions and several adjectives, such as ”terrifying, low, embarrassed and ashamed”, were repeated to describe how the participants were feeling as they discussed their condition and journey through diagnosis and treatment. In addition, 12 of the participants stated that they felt that no one understood how they were feeling because there was so little known about the condition.

P9 “It was like very terrifying and that’s why I didn’t meet the people at the time, I didn’t meet my friends and I just stayed in the house all the time. I am disappointed in my friends too, it was very disappointing, they don’t seem to understand.”

P6 “It makes you feel like you’re a burden to people”

P5 “Mammy and Daddy don’t understand about lymphoedema because they don’t understand the heaviness, they don’t understand that I’m not able to do what everyone else is able to do. It put my mental health low, feeling down the whole time.”

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P15 “Well, you were worried about it the whole time.”

P4 “I was ashamed of my legs, used to hide them. Then, as I got older I sort of put up with it, got used to it.”

The use of terms, such as “burden”, “ashamed”, “disappointed”, “put up with”, “worried”, and “low”, all depict a very traumatic and sad picture of living with lymphoedema.

Participants who were obese also voiced the opinion that they felt that because they were overweight that health professionals ignored their lymphoedema and focused only on the weight issue. Four of the participants who were diagnosed with primary lymphoedema stated that their lymphoedema had caused them to have a reduced mobility, which in turn led to an increase in body weight.

P10 “I found that people were not sensitive towards me with lymphoedema because I am fat. If I say, “oh I have lymphedema,” i found it was .as though they were laughing me off, like what are you talking about, you just need to loose weight.”

In total 40% (n=8) of the participants ranged from severely obese to super obese and all of these participants reported feelings of being judged by their weight and the only medical advice that they received was with regard to weight loss, with no attention given to their lymphoedema.

5.9 Physical impact of lymphoedema

Lower limb lymphoedema can exert considerable physical impact on the person with lymphoedema due to the fact that the weight of the affected limb or limbs can reduce mobility and affect many of the activities of daily living that depend on a person being able to be mobile, including dressing, toileting, and household chores. The level of impact would seem to be proportional to the level of oedema, as the participants with a greater volume of oedema on their limb/limbs are less mobile than those with mild oedema.

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5.9.1 Reduced mobility and symptoms

In this study, 70% of the participants have a stage 3 lower limb lymphoedema; of those, only 10% have a unilateral lymphoedema. The participants with bilateral lower limb lymphoedema have a large volume of oedema and thus have reduced mobility. These participants described symptoms of heaviness and tiredness and report reduced ability to engage in various aspects of daily living.

P5 “The stairs is a killer. I would go up it one at a time you know.”

P7 “It was hard to get them out of the truck with the weight that was in the leg, like from all the fluid built up in the leg. There was shocking weight on that right leg the whole time, which I suppose in turn affected the hip.”

P4“Walking was a chore because they were so heavy, it was like carrying around two cement blocks.”

P12 “Physically, I find that I’m more fatigued, that means that I need to rest more. Simple chores like bending down are quite uncomfortable.”

P8 “I have a grandson and I wouldn’t be 100% confident, even though I’m only still young, I’m forty five, but I wouldn’t be confident that I can mind him all day on my own.”

Every task that people do as part of their daily lives requires that they are mobile and relatively physically fit. The above statements describe the burden of trying to move and lift oedematous limbs and the resulting inability to complete simple household tasks without feelings of physical exhaustion.

5.10 Financial impact of Lymphoedema

Lower limb lymphoedema is a chronic condition and as such requires life-long maintenance treatment in the form of manual lymphatic drainage, compression bandaging, compression hosiery, skin care products, and exercise, as recommended by international guidelines such as the Lymphoedema Framework (2006) and the CREST guidelines (2008). The ongoing maintenance regimen may also require night-time garments and can also be assisted by the maintenance use of intermittent pneumatic compression

152 sleeves. However, maintenance regimens such as those described do not come without a price. In addition, the significant physical impact of lymphoedema can also result in reduced working hours or actual inability to work, causing significant financial burden due to loss of employment.

5.10.1 Cost of treatment and garments

The thematic analysis of the interviews in this study has identified a theme of financial burden associated with the cost of therapy and garments which has resulted in some participants being unable to afford garments and therefore allowing their lymphoedema to progress. Some of what participants said is outlined below:

P8 “I had no medical card, so I had to buy two garments and I remember my mother actually gave me the money and paid for those, that was €500 for two pairs.”

P8 “My husband was made redundant, and for two years I think we had a medical card and I got them for free. That was fantastic because it just meant that I didn’t have to worry about staying in saggy garments.”

P20 “For the last three years I have had to manage myself, you know, with a daughter in College and my medical card was gone, there has been no option but to pay for them. But literally the last garment I had, I had it for eight or nine months and it was well gone when I went to my Doctor and I said “I just can’t cope, my leg is getting bigger and bigger.” It is a huge burden. I remember being so overwhelmed with the actual cost of them that you don’t know when you are ever going to be able to buy them.”

P10 “I was ringing people and ringing people myself and everybody was saying “oh it’s €40 or €50 a session”, and I did pay that for a little while, but I just couldn’t afford it, and actually I had to travel as well and that cost more money.”

In this study 20% of the participants (n=4) last had active treatment between 11- 20 years ago, 35% (n=7) last had active treatment between 1-10 years ago. Even those participants who had a diagnosis, and who were aware of the type of treatment that they should have, found it extremely difficult to find the appropriate care within the Irish health service and also found that they could not financially sustain the cost of private treatment.

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5.10.2 Inequality of services

In Ireland, the availability of public or non-private treatment is dictated by aetiology, whereby, those who have a lymphoedema due to cancer or the treatment of cancer have access to a lymphoedema service and those who have a primary or non-cancer related lymphoedema have only a very limited service. Also, the provision of garments is limited to those who are a part of the General Medical Scheme (GMS), otherwise the patient must pay for garments themselves.

Participants in this study, all of whom have non-cancer related lower limb lymphoedema, described a treatment system whereby the public system does not provide treatment pathways for them by virtue of the fact that they do not have lymphoedema as a result of cancer.

P13 “The insurance companies will not fund my manual lymphatic drainage because I don’t have an Oncologist’s report.”

Therefore, they tend to avail of private treatment when they can afford it, but it is not sustainable because the costs are too great, also, because it is a chronic condition the continuous need for maintenance therapy at intervals places ongoing private treatment out of reach for the majority of people. One study participant describes how she “muddles through” and tries to make her garments last for as long as possible.

P8 “I just muddled along with what I had, like I wore a garment and it lasted a year. Some of the garments said “machine washable” but I never machine washed it, I just felt that they would last longer.”

5.10.3 Loss of earnings

Five participants reported that they had to reduce their hours of work and one participant had to actually give up her job, as she could no longer tolerate the journey to work in the car, as she felt that driving for periods greater than one hour exacerbated her symptoms and oedema.

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P4 “I have had to resign from my job with my employer because they moved the head-office to Tallaght, and I couldn’t travel to Tallaght so I’ve lost my job because of my Lymphoedema”.

P11 “I had to retire early because my legs were so swollen and tired by the end of my shift, all I could do when I went home was throw off the shoes and put my feet up, after a while that didn’t even help and my legs were still swollen in the morning.

Lymphoedema is not recognised as a chronic condition such as diabetes or heart disease and therefore if a person has to give up their job or retire early due to their lymphoedema there is no financial assistance available to assist with the purchase of garments or treatment and as a result their condition will often dis-improve and progress.

5.11 Therapeutic Benefits of CDT

All participants were asked for their feedback on their experience of each aspect of CDT and their perceived challenges in maintenance self-care. In general, the overall reported experience was positive for each participant. The following subthemes were identified:

5.11.1 Compression: A means to an end

Compression bandaging proved to be the most challenging aspect of the treatment. However, the visual reduction of the limb volume on each bandage change gave the participants the resolve to continue with the treatment to completion stage. Four participants said the following.

P9 “Oh the compression bandaging was so hard, to get used to them, and they were so hard, and they were hot and they were itchy and like uncomfortable but then when the leg went smaller and smaller and you get used to it, and you just like feel that it is working, and you are not so stressed”

P11 “It took a lot of time, it took a lot of your time and my time. It was quite difficult to wear shoes when the bandages were on, compared to the tights.”

P14 “It works, okay and that is what makes you persevere through it, I see my ankles”.

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P17 “I look at the bandage and I see a means to an end and a future again”

Many of the participants had initially felt that they would not be able to wear compression hosiery because they felt it was not cosmetically pleasing or they did not feel that they would be able to tolerate it. However, by the maintenance stage, all of the participants understood the necessity of wearing compression in order to maintain oedema reduction and did not want their legs to return to their former shape. Three participants verbalised the following:

P2 “When I look at my legs they are much improved. I wear the stockings and I find the stockings fine, because I can put tights over the stockings if I want to wear a skirt. I’m delighted with myself. I’m delighted that somebody has taken me seriously after so long.”

P11 “It was a great relief, I have to say, when the bandaging stopped and I was given the stockings because the stockings are very comfortable and I even feel good in them.”

P1 “Well certainly my aversion to the stockings has changed and I do realise that I can help myself much more than I could. I have got a way of helping myself now.”

Most participants (n=15) considered that the wearing of compression hosiery in the summer could be the greatest challenge of self-maintenance care. They perceived that the heaviness of the compression garments would be difficult to wear during warm weather and their greatest concern was the increase in their oedema if they left their stockings off. Also, other participants (n=8) felt that being able to budget for the purchase of compression hosiery was going to be their greatest challenge.

P8 “I feel that as long as I have access to garments I would have no real problem managing myself”.

P4 “I am a little worried about the summer, am I going to be able to wear the stockings because they are quite warm”.

P11 “Its part and parcel of my day but it’s going to be hard and there’s no use in saying “that every day is going to be good”, there is going to be days when you say “oh frig this, I have had enough”, but you can’t give in.”

All participants reported a fear of the return of their oedema if they didn’t wear their stockings every day and, for those who relied on family members to apply

156 their compression hosiery (n=7), there was the added worry of constantly being dependant on another person and the worry of what would happen if that person wasn’t there.

These interviews occurred at week 8, when participants had just completed their intensive phase of treatment and had received their compression hosiery. From baseline, until week 8, the therapist had been in control of oedema reduction and maintenance. The participants had not yet entered the self-care phase in which they needed to carry on with and be responsible for the four cornerstones of lymphoedema care, namely, compression, skin care, exercise and simple lymphatic drainage. However, the statements above do highlight the concerns of the participants for their future of self-care.

5.11.2 Positive physical outcomes

All of the participants reported a physical change in their limbs, which they felt had improved their levels of mobility and function. The participants spoke about being able to move their joints more easily and they were physically more capable of doing daily tasks. The reduced volume now meant that the participant was carrying less weight in their legs and therefore had more energy.

P8 “The leg feels lighter so now I feel like I’ve got a bit more mobility back between the calf and the knee so you know, when I put my jeans on I can already see a reduction. My whole family sees the reduction so it’s not me imagining it. And I know that with measurements they show too.”

P9 Now when it’s like nearly over and I am walking in the shops, I can walk all day. I can go to dinner with my husband. We can like watch our grandchildren, so it’s like lots more we can do. I even can like wash the whole kitchen with the mop, and it was like an exercise to me. I was so proud that I could do that.”

All of the participants reported that exercise has also become a bigger part of their daily routine. Of course, the level of exercise or tolerance for exercise within this group was a gradual progression as many (n=12) of the participants were coming from a situation where their mobility was severely restricted and quite minimal.

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P9 “I am trying to walk more, to move more, to walk more up and down the stairs to get exercise, and I have this stepper to exercise the leg”

P20 “I think I’ll go swimming now and I’ll do the things, I never could do. I will I’ll go swimming and go cycling and I’ll start living my life again. I really, really will”

Throughout the intensive therapy phase, the participants had been in compression bandaging and were, therefore, restricted in the type and amount of exercise that they could do. The participants received a handout on specific exercises while in bandaging (Appendix 10). As a follow up to this therapy phase, the participants received a diary in which they are asked to record on a daily basis how much time they spent on exercise and what type of exercise.

5.11.3 Positive Emotions

The psychological support of being in treatment and seeing improvements also gave the participants an improved outlook on their condition and participants reported positive emotions and feelings, as distinct from the initial reports of negative emotions as the participants recalled their journey with lymphoedema. Two participants said,

P18 “It is very beneficial to have a practitioner to do the manual lymphatic drainage for you. Also from a psychological point of view, it lifts your mood. You are kind of taking time for yourself, which you wouldn’t normally do, but also you feel like you are being looked after and cared for.”

P11 “Oh I’m very happy that the swelling is going down”.

Complex Decongestive Therapy has given this cohort of participants reduced oedema, improved physical ability and a more positive outlook on living with lymphoedema. However, the participants were also aware of the challenges that maintenance on a daily basis provides for them and their families.

5.12 Summary of Qualitative Analysis of interview data

The researcher used Braun and Clarkes (2006) six phase framework inductive thematic analysis to obtain common patterns and themes from the transcribed interview texts of 18 participants. Three main themes were identified and named and then divided into subthemes to further explain in-depth the meaning of each

158 theme. Many of the themes identified in this study are similar to a previous Irish study in 2010 which leads the researcher to believe that little has changed in lymphoedema services and patient experiences in the last 8 years. There is a paucity of research exploring the patient’s experiences of CDT and this study provides valuable information which identifies the positive therapeutic benefits of CDT resulting in improved physical ability and a positive patient outlook on treatment and maintenance of oedema reduction. This research also brings new information to light in relation to the patient’s perceptions of compression bandaging. All of the participants in the study reported that while they did not particularly like being in compression bandaging, they were committed to continuing with the therapy because they could see the reduction of limb size when the compression was removed. Therefore visual changes in the limb gave the participants the incentive to be concordant with treatment.

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Chapter 6 Results - Qualitative Findings of Diary

This chapter will examine the data which emanated from the diary that each participants completed every day for 4 months.

6.1 Maintenance Diary All participants were asked to complete a four-month self-maintenance diary commencing at week eight and finishing on week 24. Each participant was given a diary and a list of daily self-care tasks that they were asked to record and comment on daily. These were,  Compression hosiery: Number of hours worn/ any difficulties getting hosiery on or off/ laundry of hosiery/ any problems associated with the hosiery  Skin Care: Time taken and at what stage of the day/ emollients used/ comments about the health of your skin.  Exercise: Type and time dedicated to exercise. Challenges associated with exercise  Simple Lymphatic Drainage (SLD): How often and for what length of time? Comments about SLD.

The participants had a visit to the clinic on week 16 for circumferential limb measurements. Each participant brought their diary with them for an interim viewing by the researcher. This acted as a reminder for the participant to keep the diary active. This also gave the participant the opportunity to discuss any difficulties they were having with maintenance care. At week eight, the participants were wearing their compression hosiery and had received both written and verbal information on exercise, simple lymphatic drainage, and general recommendations for persons with lymphoedema (Appendix 9, 10, 11).

At the completion of the four-month diary, 18 diaries were returned. Of the two that were not completed, one participant had left the study after week eight due to ill-health and hospitalisation. The second participant did not return the diary at the end of the 24 weeks. Of the 18 diaries that were returned, 16 diaries were completed each day and all recordings completed as requested. If there was no change in routine from the preceding day, then “no change” was recorded. The 160 remaining two diaries were returned in letter format, detailing each area of the four cornerstones of care, which included compression, skin care, exercise, and SLD. Each task was reported on in the letter and both of these participants had uneventful maintenance periods. In general, all of the diaries were quite similar in content. The following data have been extracted and analysed using a similar approach to that used in the interviews. Key findings will now be presented, narratively.

6.2 Compression Hosiery

Of the 18 participants who completed their diary, 15 participants wore their compression hosiery between 12 and 14 hours each day, usually applying them at around 08.00 hrs and removing them at approximately 22.30 hrs. Of the three participants who had difficulty with compression during the maintenance period, one participant (P10) found that she could only wear her garment for a maximum of six hours before needing to remove them. The second participant (P9) developed a cellulitis and required hospitalisation at week 12. The third participant (P5) relied on someone to assist with putting the socks on and as a result there were many occasions when the compression socks were left off or delayed.

One participant used an assistive device to apply and remove the socks. This participant had issues with hand dexterity and could not manage without the device and even found it quite difficult with the device. She reported a timeframe of 35 minutes to get the stockings on and 10 minutes to remove them. Five participants required the assistance of a person to get the socks on and off. These participants were in the age group range of 45-60 years old; however, they were all in the morbidly obese category and physically could not reach their toes or manipulate a device. Therefore, the success or failure of their maintenance care depended upon the input of another person.

Despite the manufacturing information leaflet, which stated that the socks should be washed daily in order to maintain integrity and elasticity, all participants reported washing the compression hosiery every second day. They

161 all reported that they hand-washed the hosiery. All participants reported that they found it took a full day to dry the socks.

Nine participants reported that they found that their feet were uncomfortable by the end of the day. They used terms such as “burning”, “hot”, and “felt as if they were swelling”. However, these symptoms all eased after the socks were removed. All of the participants reported that they were relieved to remove the socks by the end of the day. The narrative from the diaries indicated that compression hosiery posed quite a challenge for many of the participants either due to difficulties with getting the hosiery on and off, complaints of discomfort as the day progressed and infrequent laundering of the hosiery which leads to reduced effectiveness of the fabric.

Thematic analysis of the 18 interviews also identified that many of the participants had a concern with regard to wearing compression hosiery in the summer and whether they would be able to tolerate the combination of heat and compression. Analysis of the diaries confirmed that seven participants were non-concordant with wearing their compression while on holidays leading to reports of an increase in swelling within 2-3 hours of removal of the hosiery. These participants compensated by elevating the limbs in bed and also then having a rest day for elevation and later being able to get the compression hosiery back on again. Two participants were able to self-bandage and had reduced the oedema overnight and then got back into their hosiery. All participants stated that they would not be making the mistake of taking their hosiery off in the warm weather, as the resulting oedema was even more uncomfortable than the heat.

With regard to the various styles and types of compression hosiery, the following remarks were noted:

 Participants who had zips in their hosiery (n=2) felt that they required a lot of dexterity to get the zip edges together and zipped up. They also noted that the zip seam did leave a mark on the skin;  Participants who had thigh length stockings (n=6) had some difficulty with slippage of the socks, especially if the participant was particularly active;

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 Participants who had a combination of thigh length stockings and Bermuda shorts (n=4) found greater versatility, in that they could remove the shorts if there were issues with comfort or a day where they were less activity and yet still have the compression on their legs.

6.3 Skin care

All participants were compliant with their skin care. They bathed the legs in the evening and applied emollients. The average amount of time spent on skin care each night was 10 minutes. Only one participant required assistance with skin care. No difficulties were reported in this area of self-maintenance. Perhaps the fact that 17 of the 18 participants who returned the diary are female has an influence on the compliance with skin care, as females are quite conscious of their skin care in general. None of the participants reported any feelings of burden with skin care, and they implied that skin care was a natural and easy part of their maintenance care.

None of the participants reported anything about foot care or monitoring of skin folds for fungal infection, despite the fact that they would have received written and verbal information about this topic. None of the participants reported adverse skin reactions or skin breakdown. This may highlight a deficit in knowledge or awareness of the importance of skin inspection or a deficit in the provision of education in this area.

6.4 Exercise

All participants reported spending between 10 minutes to one hour daily doing exercise and all participants reported a greater awareness of the need to move and exercise, which concurs with the interview data analysis. At baseline, only four of the participants were involved in some form of exercise. One participant was going to the gym daily and the other three participants were having a daily walk. The remaining participants were not involved in any activity.

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The amount of time spent doing exercise was proportional to the participants’ physical ability. Five participants who were quite active and did not have any other co-morbidities, reported spending 40-60 minutes daily doing exercise. However, a further four participants reported spending as little as 10 minutes of slow walking in the house due to reduced physical ability as a result of obesity and joint pain.

The most popular exercise among all participants was walking. One participant reported joining a Pilates class and two participants reported that they joined an aqua aerobics class. Four participants reported that they would like to go to the pool, either to walk or swim in the water, but their issues with body image were stopping them and they did not feel that they could overcome that issue at that time. Both of the participants who had commenced aqua aerobics were going to early morning classes in order to avoid too many people. They also reported feeling self-conscious of their limbs in the pool.

6.5 Simple Lymphatic Drainage

Only six participants reported performing simple lymphatic drainage on a daily basis, spending 10-15 minutes doing the massage and stating that they found it relaxing. A further four participants reported doing the massage 1-2 times per week initially over the first 4 weeks and then stopped. The final eight participants reported that they did not do the massage at all. Of the participants who were either doing the simple lymphatic drainage consistently or even occasionally, the predominant comment in the diaries was;

P11 “I don’t know if the massage is doing any good”

P 4 “I think this needs to be done by a professional”

Many of the participants (n-12) did not perceive that simple lymphatic drainage was an important component of self-care. They also wrote in the diary that they felt that they were doing it wrong, despite having written instruction to refer to. One participant had drawn a diagram and had numbered the positions and the sequences in order to remind her of how to do the massage. Perhaps a more

164 visual, diagrammatic format of instruction would be easier for patients to follow and understand.

The participants who were not doing simple lymphatic drainage were not concerned that they might not be completing self-care. Therefore, one can assume that the participants did not regard simple lymphatic drainage as an important aspect of self-care and oedema maintenance.

6.6 Explanation of QoL and limb volume changes using diary data

Quality of life scoring and limb volume measurements display the greatest percentage changes, for a number of participants, over the week 8 to week 24 period. The diary can provide insights into the reasons for percentage changes in both QoL and limb volume. As stated previously, this time period is the stage at which the participant is independent in their self-maintenance and therefore it can be more liable to change, as the therapist no longer has control.

The examination of QoL and limb volume percentage changes are most clearly explained by analysing the diaries of the participants who have demonstrated the most significant changes. Of note, participant’s ID No. 9 and 10 displayed the greatest negative changes, with increases in limb volume from week 8 to week 24 and reduced QoL in the same timeframe. Participant ID No 5 displayed an increase in limb volume from week 8 to week 24, however, there was no reduction in QoL. All other participants demonstrated consistent limb volume reduction and improved QoL in all domains, with only mild fluctuation at times.

6.6.1 Participant ID 9 Diary Analysis

In the overall QoL scoring from week 8 to week 24, participant No. 9 had a minus 44% reduction in QoL. In the domain of function, there was a 94% increase in score, in the domain of appearance there was a 22% increase in score, in the domain of symptoms there was an 80% increase in score, and finally in the domain of mood there was a 100% increase in score. Therefore, all domains indicated a reduction in QoL. In addition, in association with this reduction in QoL, a 14.51% increase in limb volume on the right leg, and a

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9.91% increase in volume on the left leg, was noted within this same time period. Following analysis of the diary of participant No. 9, the changes in QoL and limb volume can be explained. This participant developed a cellulitis to the right leg and required hospitalisation at week 12, the compression was discontinued while the cellulitis was being actively treated and this participant was discharged from hospital without compression. At week 16 measurement, both limbs had returned to pre-therapy circumference and this participant needed to recommence CDT. Participant No.9 discussed in the diary the “disappointment, sadness and feelings of failure” because the oedema had returned to both limbs. This participant reported a definite reduction in QoL from week 8 to week 24.

6.6.2 Participant ID 10 Diary Analysis

In the overall QoL scoring from week 8 to week 24, a minus 14% reduction in QoL was reported. In the domain of appearance, there was a 40% increase in scoring and also in the domain of mood there was a 15% increase, indicating a reduced QoL in those domains. However, in the domain of function, there was a minus 4% reduction in score, and in the domain of symptoms, there was a minus 8% reduction; therefore, an increase in QoL in those 2 domains. For this participant, there was a 15.46% increase in limb volume on the right leg from week 8 to week 24, and a 6.15% increase in limb volume on the left leg. Overall, from week 1 to week 24, there was a total limb volume reduction of minus 11.96% on the right leg and a minus 1.63% reduction on the left leg; this would explain the fact that the domains of function and symptom continued to improve over the 24 week period.

Participant No. 10 had difficulties with consistently wearing the lymphoedema garment thus the oedema was not controlled adequately. This participant wore tights, class 2, including the body bandage. She stated that she felt restricted in movement and after about 4 hours of wear, it started to become uncomfortable, especially around the body. In addition, taking the tights down for toileting was also a challenge. After 3 weeks of perseverance, this participant decided not to wear the compression hosiery.

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6.6.3 Participant ID 5 Diary Analysis

Participant 5 had a limb volume increase of 23.93% on the right leg and 25.51% on the left leg from week 8 to week 24. From baseline to week 8, there was a 19.25% reduction on the right leg and a 21.48% reduction in limb volume on the left leg. However, the overall limb volume percentage change from week 1 to week 24 was a 0.07% increase on the right leg and a 1.45% reduction on the left leg.

There was no reduction in QoL as a result of the increase in limb volume from week 8 to week 24, which is the opposite to participant 9 and 10. This participant required the assistance of her mother to put on and take off the stockings, but some mornings her mother was not available and the stockings were not applied until later in the day. At week 10, the participant discontinued wearing the compression hosiery, as the weather was quite warm and her carer had a medical problem which required rest and no physical exertion. By week 24, the bilateral limb volume had returned to almost baseline measurements. In her diary, the participant writes that she was content to know that there was a specialist service available to her if she needed assistance or had questions; this reassurance has possibly prevented a reduction in QoL.

6.7 Self-efficacy and diary analysis

Self-efficacy refers to the level of confidence with which a person feels that they can complete a task or deal with a particular situation. For the purpose of further exploring the data presented from self-efficacy scoring, the researcher will examine the diaries of the participants who scored lower in the PROMIS short form tools, specifically scores of 12 and under, which would indicate that the participant had little or no confidence in their ability to complete the tasks required for lymphoedema self-maintenance.

Once again, as was presented in the QoL and limb volume percentage changes, the same participants present with low scoring in the PROMIS short form tool and thus low confidence, namely participant ID’s 5, 9, and 10. As was described earlier, Participant no 5 required the assistance of her mother to help 167 her to get the compression hosiery on and thus was dependant in that category. However, she could complete the skin care and was taking short walks independently. Participant 5 scored an 11 in symptoms and 12 in emotions, therefore, a little more confident in dealing with her oedema yet needing the support and assistance of others.

Participant 9 scored 11 in emotion and daily activities and 12 for social and symptoms. The reoccurrence of cellulitis and resulting rebounding of oedema had reduced this participant’s confidence that she could deal with her swelling and symptoms and because the rebound oedema had reduced her mobility and activity level again, she was only a little confident that she could complete her daily activities. In her diary, she described how much more confident she had become when the oedema had reduced, as she was able “to mop the floor, climb the stairs”, but subsequent to the cellulitis and increased oedema, she was “disappointed”.

Participant 10 only scored low in emotions because her functional ability was still quite good. She had made a decision that she would no longer wear the stockings and therefore didn’t have to battle with getting them on. However, she stated in her diary that she was “disappointed” that she could not continue to manage the compression and she stated that she “worries” about the future.

Two remaining participants scored quite low in the self-efficacy tool, participant ID’s 6 and 17. Participant 6 was a 77 year old lady, had a stage 3 lower limb lymphoedema and other co-morbidities, including diabetes and renal insufficiency. This participant’s level of mobility was quite poor and she required the assistance of her carer for all activities of daily living. This participant did very well, reduced oedema, and improved her QoL. However, her success in maintenance was dependant on her carer and thus she scored a 4 in daily activities and a 9 in emotions. Her social score was a 20 because she knew that she had all the help that she needed. This participant’s symptom score was 12 as she had the knowledge to deal confidently with her oedema but needed the physical assistance of her carer.

Finally, participant 17 was in the super obese category and as a result her mobility was reduced and she required the assistance of a carer to assist with

168 general household activities. This participant had an oedema reduction and had improved her QoL. She reported in her diary that she could apply and remove her compression hosiery and complete her skin care. Her activity level, though low, had also improved and she reported taking walks in her back garden. Participant 5 reflected in her diary about being “worried and fed up” that she cannot do more around the house. She scored a 6 in emotions and a 5 in daily activities, as she is not at all confident that she is able to deal with these situations and feelings. However, she scored a 15 in social, as she has good support systems to ask for help when required, and then a score of 9 in symptoms, as she had little confidence that she could manage her oedema.

6.8 Summary of qualitative findings of the participant diary

All participants were asked to complete a four-month self-maintenance diary commencing at week eight and finishing on week 24. At the completion of the four month diary, 18 diaries were returned. Of the 18 participants who completed their diary, 15 participants wore their compression hosiery between 12 and 14 hours each day, usually applying them at around 08.00 hrs and removing them at approximately 22.30 hrs. Of the participants who had difficulty with wearing compression, (n=3) the main reasons were difficulty with applying and removing the garments with one participant developing a cellulitis.

Thematic analysis of the 18 interviews also identified that many of the participants had a concern with regard to wearing compression hosiery in the summer and whether they would be able to tolerate the combination of heat and compression. All participants were compliant with their skin care. However, none of the participants reported anything about foot care or monitoring of skin folds for fungal infection.

All participants reported spending between 10 minutes to one hour daily doing exercise and all participants reported a greater awareness of the need to move and exercise. The most popular exercise was walking. One participant reported joining a Pilates class and two participants reported that they joined an aqua aerobics class.

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Only six participants reported performing simple lymphatic drainage on a daily basis, spending 10-15 minutes doing the massage and stated that they found it relaxing. Many of the participants (n-12) did not perceive that simple lymphatic drainage was an important component of self-care

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Chapter 7 Discussion

7.1 Introduction

This chapter will bring together the findings of this study and align them with current evidence in the area of primary and secondary, non-cancer related lower limb lymphoedema. The discussion section is divided into two separate chapters. Firstly, Chapter 7 will present a discussion on the demographic profile of the study participants, followed by the key findings of the research study, before discussing these findings in relation to current evidence. Chapter 8 will then present the strengths and limitations of the study, followed by a discussion of the implications of the findings in terms of clinical practice and future research. Finally, a plan for the dissemination of the findings, along with a summary and a conclusion, will be presented

7.2 Demographic Profile

7.2.1 Age

The study population consisted of 20 participants recruited by referral from General Practitioners, Public health nursing services, vascular consultants, and self-referred. All participants had a diagnosis of lymphoedema, as confirmed by a vascular consultant. Following an initial assessment meeting to ensure that the participant fulfilled the inclusion/exclusion criteria, they were given verbal and written information on the study protocol in order to make an informed consent.

The group consisted of 90% female (n=18) and 10% male (n=2) participants. The mean age was 59 years (with a range of 19 – 80 years old) and a standard deviation of 16. Only 2 participants were between 18-40 years of age, 8 participants were between 40-60 years, and the remaining 10 participants were over 60 years of age. The demographic profile of this study is similar to previous studies of lymphoedema of any aetiology (!!! INVALID CITATION !!!).

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7.2.2. Aetiology and stage of lymphoedema

The study population was divided equally into 50% (n=10) primary lymphoedema and 50% (n=10) secondary lymphoedema. The even distribution of primary lymphoedema and secondary lymphoedema is unusual as the majority of studies tend to present a larger proportion of secondary lymphoedemas. Cooper and Bagnall (2016) conducted a survey based study to assess the prevalence of lymphoedema in the South West and West Midlands of the UK and reported a prevalence of 1:5129 of primary lymphoedema in that population. Likewise, Wang and Keast (2016) examined the prevalence of lymphoedema patients attending a wound care clinic in Canada and found that, of 326 patients with lymphoedema, only 4% (n=12) had primary lymphoedema.

All of the participants in this study had been assessed and diagnosed by a vascular consultant; 6 participants were diagnosed on lymphoscintigraphy and 4 participants were diagnosed by clinical assessment. The accuracy of clinical diagnosis could be debated in the absence of additional diagnostic testing. In this study the participants were staged on assessment using the International Society of Lymphology (2013) Consensus Document 4 stage classification system;

 Stage 0 - Latent stage, swelling is not yet evident but subtle changes are occurring in the skin and the patient describes subjective symptoms.  Stage 1 – An early accumulation of fluid relatively high in protein content which subsides with limb elevation. Pitting may occur.  Stage 2 – Tissue swelling is irreversible by elevation. Pitting is manifest and, at a later phase in Stage 2, the tissue may no longer pit as excess fat and fibrosis occurs.  Stage 3 – Lymphostatic elephantiasis where pitting can be absent and skin changes such as hyperkeratosis and papilomatosis are evident due to further deposition of fat and fibrosis.

The progression of severity of lymphoedema was evident, as 90% (n=18) had a stage 3 lymphoedema, with significant oedema and skin changes, and only 10% (n=2) had a stage 2 lymphoedema, with none of the study population presenting at stage 1. These statistics support the qualitative theme of delayed 172 diagnosis, as 80% (n=16) of the participants reported symptoms of oedema for between 11 years and up to 60 years in duration and 50% (n=10) of the participants were diagnosed in the last 10 years. Therefore, the 50% of participants who were diagnosed in the last 10 years had persisting and increasing oedema for up to 60 years.

7.2.3 Body Mass Index

Body Mass Index ratings of the study population demonstrate that, only 15% (n=3) had a healthy BMI, 25% (n=5) were overweight, and the remaining 60% (n=12) were in the category of obese. Of those deemed to be obese, 20% ( n=4) were moderately obese, 10% (n=2) were severely obese, 15% (n=3) were very severely obese and, 15% (n=3) were super obese, indicating a BMI between 50-60 kg/m2. The mean BMI rating was 36 kg/m2 (median= 35kg/m2, SD = 11). The range of BMI scoring was between 22 and 60kg/m2, with 60kg/m2 being the maximal range of super obese.

There are strong associations between obesity and lower limb lymphoedema (Wilkins et al., 2014). They have the potential for serious complications and have significant cost implications for a country’s health-care system. Obesity is thought to predispose to the development of lymphoedema through several complex mechanisms, including increased capillary pressure in congestive heart failure leading to peripheral oedema, increased capillary permeability causing recurrent lower limb infections, accumulation of fatty tissue and loss of mobility(Ching and Kumarasinghe, 2015). In general, obesity has the ability to reduce physical activity, which in turn reduces muscular activity and the massaging effect of the lymphatics by the muscles (Todd, 2018).

Piller et al. (2008) discussed the concept of “functional lymphoedema”, whereby there is an absence of lymphatic damage but the lymphatic vessels are obstructed by the pressure of adipose tissue surrounding them. Worldwide figures acknowledge the difficulties posed by obesity and physical inactivity and Ireland is on course to become one of the most overweight populations in Europe, so interventions are necessary (Department of Health, 2016).

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7.2.4 Treatment history of participants before commencing the study

Nine participants (45%) had not received any treatment despite their diagnosis. Of the 11 participants (55%) who received treatment for their lymphoedema, 45.45% (n=5) were treated with compression hosiery only. Of the other participants, 36.36% (n=4) received MLD, compression bandaging and hosiery. Only one participant received MLD only and another one participant received MLD and compression bandaging. Thematic analysis of the participants’ interviews identified the subtheme of absence of referral pathways. All participants reported the struggle to find therapy and also the lack of knowledge within the health professions. Only 36.36% of the participants received the appropriate treatment, there was no provision for exercise or simple lymphatic drainage in their treatment package. IIuchi et al. (2015) conducted a retrospective observational design study to examine associations between treatments and outcomes of patients with upper and lower limb lymphoedema. One hundred and seventy (170) patients were recruited from 4 lymphoedema clinics throughout Japan. However, of the 170 patients, only 7 patients received all components of CDT, despite the fact that they had received treatment in a specialised lymphoedema centre thus demonstrating the variation in the therapeutic programs provided by specialist centres. Therefore treatment outcomes cannot be easily assessed due to the fact that there may be many varying combinations of therapies modalities used to treat lymphoedema.

Moffatt et al. (2003), using a questionnaire to determine the incidence of chronic oedema in a community site, identified that in a total population of 823 patients diagnosed with chronic oedema, only 529 (64%) were receiving treatment for their lymphoedema. Of that total 445/823 (54%) patients were seen by specialist lymphoedema practitioners. In a later study, Moffatt et al. (2017) in a similar study design to her 2003 study, identified a total of 971 patients with lymphoedema in a similar community setting and, of those, 667 patients (69%) were managed in a specialist centre. While the percentage of patients receiving treatment are comparable between this author’s study and both of the above studies (55%, 64%, 69%, respectively), the proportion of patients receiving treatment in a specialised centre in this author’s study is also similar, at 54%

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(6/11). Of the 54% (n=6) who were treated in a specialised lymphoedema service, 50% (n=3) were treated by a private therapist.

Moffatt et al. (2003) also identified that skin care and elastic hosiery were the most predominant treatments provided at 88% and 82% consecutively. However, patients treated by community services would only have access to hosiery available through the Drug Tariff and not the specialist garments required for lymphoedema care. Despite the value of exercise in the control of lymphoedema, only 53% patients were using exercises in their management. The participants in this study did not report either skin care or exercise as part of their treatment, and compression hosiery was worn by 83% (n=10) of the participants; however, only 50% (n=5) of those participants would have received appropriate lymphoedema hosiery.

Moffatt et al (2003) further reported that only 13% of her patient population were receiving bandaging, whereas 45% (n=5) of the participants in this study had received compression bandaging as an element of their treatment. Manual lymph drainage was rarely used in the Moffatt et al (2003) study, with only 4% having had MLD as a part of their treatment, however, 54% (n=6) of this studies participants had received MLD. Diuretics were used with 8% of patients in the Moffatt et al (2003) study whereas a remarkable 85% (n=17) of the participants in this study were prescribed diuretics by their General Practitioner despite their lack of effect in the treatment of lymphoedema.

Comparisons between this author’s study and the Moffatt et al (2003) study must be examined with caution, as the sample size in this study is small, from a quantitative perspective; however, in combination with the complementary qualitative data, the findings have tentatively more strength.

7.3 Key Findings of the research study

The key findings of this study will be presented with reference to each research question, the methods employed to answer the questions and the resulting key findings. The research questions were as follows:

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1. What is the impact of Complex Decongestive Therapy (CDT) as a treatment for primary and secondary lower limb lymphoedema during the intensive and maintenance phases of CDT, in relation to; a. quality of life, b. limb circumferential and volume changes 2. What are the patients’ experiences of living with lymphoedema? 3. What are the patients’ experiences of the four elements of CDT during the intensive and maintenance phases of treatment?

7.3.1 Research Question 1 and key findings

Complex Decongestive Therapy, which included manual lymphatic drainage, skin care, compression bandaging, and exercise was initiated with a purposive sample of 20 participants following their informed consent. The intensive therapy program ran for 3 days per week for 4 weeks, followed by maintenance therapy twice weekly for 4 weeks until the participant was fitted with compression hosiery.

The methods employed to answer the research question included;

 circumferential limb measurement at 4 cm intervals from a fixed point on the lower limb  a disease specific quality of life questionnaire called LYMQOL LEG  a self-efficacy tool to measure the participants confidence in managing lymphoedema self care.

7.3 2 Complex Decongestive Therapy does improve quality of life and reduce limb volume:

7.3.2.1. Quality of life scoring varies over time depending upon changes in limb volume.

Quality of life scoring was recorded over 3 timelines; baseline, week 8 at the completion of the intensive phase of CDT and week 24, which was 4 months post the self-maintenance period. LYMQOL LEG developed by Keeley (2010) divides the quality of life scoring into 4 domains, function, appearance, mood, and symptoms. There is also an overall score rated from 0 (poor) to 10 176

(excellent). For overall quality of life scoring, the mean QoL scoring at baseline was 4.35 (ranging from 0 to 9). Three participants (15%) rated their QoL as 0 (poor) and only one participant rated their QoL at 9. All of the participants had an improved QoL over the 8 week period. From week 8 to week 24, a reduction in QoL scoring was noted for three participants, due to one participant having developed a cellulitis and another participant having difficulties with consistently wearing the lymphoedema garment, thus the oedema was not controlled adequately and had increased. The third participant had left the study before week 24. The mean percentage change from week 1 to week 24 was 48%. Therefore, 90% of the participants had an improved QoL overall, from baseline to week 24.Likewise, QoL scoring in all domains varied over the 24 week period.

 In the domain of function, ninety percent of the participants (n=18) had an improvement in QoL from baseline to week 8. However, one participant reported a reduced QoL at week 24 in comparison to week 8 due to an episode of cellulitis. All participants had a reduced score and therefore an improved QoL in the function domain from baseline to week 24.  In the domain of appearance, all participants scored a reduced figure at week 8, thus indicating an improved QoL. A further 30% (n=6) of the participants continued to score improvement in QoL into week 24. However, 5 of the participants had an elevated scoring and thus a reduction in QoL reported due to exacerbation of the oedema.  In the domain of mood, from baseline to week 24, 75% (n=15) of the participants had an improved QoL. One participant did not complete the program to week 24. The subsequent 20% (n=4) of participants remained unchanged and had originally scored 1, therefore they were not affected by lymphoedema.  In the domain of symptoms, from baseline through to week 24, 85% (n=17) of the participants all had a reduction in scores and therefore an improvement in QoL. One participant did not complete the program and the remaining participants remained unchanged and symptom free.

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The overall QoL findings of this study concur with Greene and Meskell (2017) who identified that while lower limb chronic oedema has a significantly negative impact on QoL, the overall QoL score had a mean of 5.57 (SD 2.52), thus the patients rated their overall QoL as fairly good. The baseline overall QoL score in this study was 4.35, which indicates an average QoL. Likewise, Greene and Meskell (2017) aimed to examine the impact that chronic oedema can have on health related QoL with it being measured using the LYMQOL LEG questionnaire. However, only one timeline was used to collect QoL data. The inclusion criteria was patients with oedema present for greater than 3 months attending lymphoedema clinics, public health nursing, and vascular clinics, therefore it is unclear as to what stage of treatment each of the participants were progressing through, which indeed would have had considerable influence on the scoring of QoL at that point in time. It may be fair to assume that the slightly higher score of 5.57 for overall QoL in the Greene and Meskell (2017) cohort of patients may be due to the fact that they have been actively in treatment and thus their QoL is also improving.

The overall QoL score in this study improved from a baseline mean score of 4.35, to a mean score of 8 at week 8. Similarly, Moffatt et al. (2003), using a survey to evaluate the prevalence of chronic oedema in a community area of the U.K., also proposed to determine the impact of chronic oedema on the QoL of that same population. The Short Form 36, a questionnaire widely used for self-assessment of health and QoL, examined a population of 228 patients with lymphoedema and reported a significant difference in role physical, role emotional, social functioning, and physical functioning. Notably, only 64% of these patients were receiving any treatment and, of these, 54% were accessing specialist lymphoedema care and 12% were receiving treatment from community services alone. Once again, because this cohort of patients came from different health services and had varying treatments provided, if at all, the QoL scoring would be quite diverse and not specific to a particular timeline in treatment. Therefore, comparison of QoL scoring becomes quite difficult and consideration needs to be given to timeframes, questionnaires, and population when drawing comparisons.

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Herberger et al. (2017) also used a population based study to determine QoL scores of patients with lymphoedema in the community. The Short Form 36 was used to collect QoL data that could be compared to the wider population, along with the FLQA-LK, a disease specific QoL tool in the German language. A sample size of 301 patients with oedema for greater than 3 months were recruited from health care providers in all sectors of lympoedema care, including GPs’, hospitals, nursing homes, self-referral and specialised wound care and lymphology clinics. This study revealed a marked impairment of QoL in comparison to the general population, and in particular in the specialist centres, where a lower QoL rating and social impairment was demonstrated.

Many other studies have been quite specific in population and timing of questionnaire (Huggenberger et al. (2015) Noh et al. (2015) Kim and Park (2008). Both Huggenberger et al. (2015) and Noh et al. (2015) included patients from dedicated lymphoedema and physiotherapy services, and all patients were treated with Complex Decongestive Therapy, with quality of life measured at designated time lines post treatment. The Short Form 36 was used to collect data on QoL in both studies. On the basis of similar populations, treatments and measurement tools, the comparison of results can be more easily qualified. The benefit of using a generic QoL tool is that comparisons can be made also with population norms in which the SF 36 used. Kim and Park (2008) and Noh et al. (2015) both displayed improved QoL scoring post CDT, and Huggenberger et al. (2015) demonstrated that, within the population of primary and secondary lower limb lymphoedema patients, mostly all domains were on par with the population norm; however, physical functioning was reduced. The quantitative findings of this researchers’ study, in isolation, should be regarded with caution, as the sample size is quite small, at 20 participants;, however, these findings are strengthened by the narrative provided by the diary which explains the impact that increases in limb volume have had on the participants and their QoL.

179 greatest negative changes, with increases in limb volume from week 8 to week 24.

In the overall QoL scoring from week 8 to week 24, participant No. 9 had a minus 44% reduction in QoL. In the domain of function, there was a 94% increase in score, while in the domain of appearance there was a 22% increase in score. In the domain of symptoms, there was an 80% increase in score and, finally, in the domain of mood there was a 100% increase in score. Therefore, all domains indicated a reduction in QoL In addition with this reduction QoL, a 14.51% increase in limb volume on the right leg and a 9.91% increase in volume on the left leg was noted within this same time period. Following analysis of the diary of participant No. 9, the changes in QoL and limb volume can be explained. This participant developed a cellulitis to the right leg and required hospitalisation at week 12; the compression was discontinued while the cellulitis was being actively treated and this participant was discharged from hospital without compression.Todd(2018) noted that indeed, compression garments are often removed during the active and painful initiation of cellulitis, however, this then exacerbates swelling and quite often the compression is not recommenced as the pain starts to abate and the cellulitis is reducing. Cox (2006) highlights the fact that obesity is a known fact for increased risk of cellulitis. Participant No. 9 had a BMI of 60kg/m2 , however, as reported in her diary, she was capable of completing her skin care daily and was becoming more active and indeed compliant with her compression hosiery. She describes feelings of disappointment due to the fact that she developed cellulitis despite being concordant with all cornerstones of self-care.

Deng et al. (2014), in a cross-sectional survey study of patients with lower extremity primary lymphoedema in the United States and worldwide, concluded that patients with bilateral lower limb lymphoedema are more likely to have an episode of infection when compared to the population of patients with unilateral lower limb lymphoedema despite the fact that they are more likely to be compliant with regular skin care.

For participant no.10 the overall QoL scoring from week 8 to week 24 showed a minus 14% reduction in QoL, there was a 15.46% increase in limb volume on the right leg from week 8 to week 24, and a 6.15% increase in limb volume on 180 the left leg. However, overall, from week 1 to week 24, there was a total limb volume reduction of minus 11.96% on the right leg and a minus 1.63% reduction on the left leg, Participant No. 10 had difficulties with consistently wearing the lymphoedema garment, thus the oedema was not controlled adequately. Brown (2011) discusses the many suggested reasons for non- concordance with compression hosiery which include improper or inappropriate provision of hosiery, poor body image and poor understanding of the dynamics of compression and how compression works to maintain oedema reduction. Todd (2018) also debates that some patients are just not ready to change poor lifestyle poor lifestyle choices in order to improve swelling.

7.3.2b. Lower limb lymphoedema has the greatest negative influence in the domain of function and appearance.

The impact of lower limb lymphoedema on QoL, in the domains of function and appearance, are considerable when compared to the domains of mood and symptoms. At baseline, between 40% (n=8) and 45% (n=9) of participants reported that lymphoedema did not affect, or only affected a little, their mood or symptoms. Whereas, between only 15% (n=3) and 30% (n=6) of participants reported that lymphoedema did not affect, or only affected a little, their appearance or function. A total of 55% (n=11) of participants reported that lymphoedema affected their appearance a lot and 35% (n=7) reported that lymphoedema affected their function a lot. While QoL scoring in appearance and function did improve at week 8 for all participants, by week 24, 30% (n=6) of the participants had a reduced score, thus a reduced QoL in the domain of appearance. These findings are due to the fact that 6 participants had an increase in limb volume; one participant developed a cellulitis and the other participants were non-concordant with wearing their compression hosiery due to warm weather.

Greene and Meskell (2017) also concurs that lymphoedema has a significant impact in the domain of appearance, however, they did not find that the domain of function was as greatly affected by lymphoedema unlike the findings of this study. Further to this Gethin et al (2012), using the SF-36 to determine the QoL scores of persons diagnosed with lymphoedema and attending an Acute care vascular clinic, found that lower limb lymphoedema significantly affected QoL in 181 the domain of physical functioning, with emotional well-being being the least affected. However, a limitation of this evidence is that only 13 patients completed the questionnaire.

Although it would appear from the LYMQOL data that the domains of function and appearance are more significantly affected than the domain of emotion, however, the qualitative interviews generate rich data which appears to indicate a different story in some situations. Emotional reactions to the situation appear strong and painful in the qualitative data and yet do not emerge so strongly in the LYMQOL results. This is an indication of the value of mixed methods approach and that even a disease specific tool may not capture the true patient experience.

7.3.3 Summary

Complex Decongestive Therapy does improve QoL and reduce limb volume and, more specifically,

 Quality of life scoring varies over time depending upon changes in limb volume,  Lower limb lymphoedema has the greatest negative influence in the domain of function and appearance.

Several studies have been presented which in general have had similar findings (Kim and Park, 2008, Huggenberger et al., 2015, Noh et al., 2015, Greene and Meskell, 2017, Gethin et al., 2012, Moffatt et al., 2003). Varying tools have been used to assess QoL, with the SF-36 being the most predominant. Most studies have shown improved QoL scores for participants involved in CDT programs (Huggenberger et al., 2015, Kim and Park, 2008, Noh et al., 2015).The remaining studies have examined QoL in a particular population (Moffatt et al., 2003, Herberger et al., 2017, Greene and Meskell, 2017, Gethin et al., 2012). Overall, it can be concluded that QoL is not a constant value and may improve or dis-improve in the lymphoedema population, depending on oedema level and control.

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7.4 Research Question No. 2 and No.3 and Key Findings

Both research questions were examined using qualitative methods, which included semi-structured interviews and a 4 month maintenance diary.

Semi-structured interviews were conducted with 18 participants using an interview schedule of 10 open-ended questions. Following thematic analysis of the interview transcripts, 3 main themes emerged:

 Health professionals lack of knowledge in the field of lymphoedema  Impact of Lymphoedema: Psychosocial, Physical, Financial.  Therapeutic benefits of CDT

Three key findings emerged from this part of the study.

7.4.1 The patient perspective in 2018 in Ireland remains unchanged since it was last explored in 2010

Murray et al. (2010) conducted a survey based study, with the overall aim of investigating current lymphoedema service provision from both service providers and patients’ perspectives, in addition to exploring patients’ experiences of living with lymphoedema in Ireland. The first phase of the study examined the perspective of service providers. The second phase of the study employed 5 focus groups, consisting of 5-8 participants per group to explore patients’ experiences of accessing treatment and living with lymphoedema. Finally, the third phase of the study involved a postal survey of lymphoedema patients to investigate the experiences of a wider group of lymphoedema patients. The findings of the postal survey in the third phase is more closely related to the perspectives of the participants in this study, as the lymphoedema population is broader and not so predominantly cancer related. The postal survey was sent to 1,539 patients whose names were accessed via hospitals and support services. A total of 735 were returned for analysis. A sum of 6.5% of participants had primary lymphoedema, 5.5% had non-cancer-related secondary, and all other lymphoedemas were cancer related. Following analysis of the data, the findings were summarised into two broad themes:

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1. Inequitable service provision, 2. Impact on daily life.

Both of themes in the Murray et al (2010) study concur with the findings of this study. Inequality in service provision was expressed by all of the participants in this study with it being clear that there is a sparsity of lymphoedema clinics, in particular for patients with non-cancer related lymphoedema, in Ireland. Participants said that health care professionals do not know where to refer them for treatment and most often their only option is for private treatment, which has profound financial implications. Murray et al (2010) reiterate the same statements from their participants who present a service provision that is based on aetiology rather than need. They discuss the fact that patients with primary lymphoedema are waiting up to six years for a diagnosis and then wait a longer time again between diagnosis and first treatment than those with a cancer diagnosis. Another common theme between the two studies is the availability of garments to those with a medical card in comparison to patients without a medical card who will have to privately purchase their own garment, costing up to 1000 euro per year. Participants of both studies also discuss the need to travel for treatment because of a lack of services within their local towns and cities.

The impact of living with lymphoedema on daily life has also remained unchanged over the last 8 years. Participants in both studies report the psychological impact of altered body image and resulting social isolation, the physical impact that causes reduced mobility, and the resulting inability to complete daily tasks such as household chores and self-care. Unfortunately, these themes are not just to be found in Ireland. According to Hardy (2012), following an investigation of the experiences of young people in the U.K. with primary lymphoedema, it was found that the participants reported struggling with feelings of low self-esteem, poor self-confidence and alteration in body image. Similarly, Williams et al. (2004), using a phenomenological approach, explored the lived experiences of 15 patients with lymphoedema, with a particular emphasis on primary lymphoedema and found that the patients expressed feelings of depression and anxiety, altered body image, and feelings of isolation. The participants of both studies stated that health professionals

184 were lacking in knowledge with regard to lymphoedema and they all described a prolonged journey from diagnosis to actually accessing a specialist service.

Moffatt and Murray (2010) examined the journey of children with lymphoedema and their families as they lived with lymphoedema and navigated the health care system in the U.K. for diagnosis and treatment. They employed a qualitative methodology using semi-structured interviews with the children (n=20) and their parents. Once again, common themes arose which included the struggle for diagnosis and a lack of knowledge about lymphoedema by health professionals. In addition, they report the difficulty in finding information about lymphoedema and the fear experienced by parents when they do access information on lymphoedema and are horrified at the prospect of what the future may be. This study also identified a similar theme. The participants found it difficult to locate information and then when they did find books or websites they were upset by the images of lymphoedema that were portrayed. The lack of easily accessible and appropriate information for patients and families is evident and is something that must be addressed.

Moffatt and Murray (2010) also discussed the effect of lymphoedema on the extended family because the child with lymphoedema requires so much of the parents’ time and energy to maintain oedema reduction. This study had findings in relation to the extended family. Four participants reported that they felt guilty as mothers because they felt that their lymphoedema had stopped them from bringing their children on outings, that family holidays were never abroad as flying was an issue, and the beach was never an option due to body image and fear of sunburn. They also spoke about their relationships with their husbands and felt that lymphoedema affected their intimacy and caused self- consciousness.

7.4.2 Compression bandaging, a means to an end.

Compression bandaging is one of the cornerstones of lymphoedema management and may indeed play a part in maintenance. However, compliance with compression bandaging has frequently been reported as a challenge (Cooper, 2013, Todd, 2013). Quite often, and quite quickly in clinical practice, a patient is labelled as non-compliant when they are not following practitioners’

185 recommendations. However, many studies have examined non-concordance and the many reasons why a patient may be non-concordant with compression bandaging and hosiery (Brown, 2011, Gray, 2013, Morgan et al., 2011, Anderson, 2015). The following rationales have been offered including poor understanding of compression, body image, difficulties with applying and removing hosiery, and inexperienced bandaging by practitioners (Brown, 2011).

Much of the reported research on the topic of compression and compliance is in the area of venous leg ulceration and there is a sparsity of evidence in the area of lymphoedema bandaging. One study by Morgan et al. (2011) does examine the experiences of patients with lymphoedema undergoing compression bandaging. They utilised a qualitative methodology with semi-structured interviews of 12 patients undergoing compression bandaging. This study also used semi-structured interviews to assess the participants’ perspective of the 4 elements of Complex Decongestive Therapy. Both studies are using a 2 layer compression bandaging system, however, the patients in the Morgan et al. (2011) study had previous experience of multi-layer compression bandaging and were comparing the experience of wearing a 2 layer system versus a multilayer system. However, in general, the participants in both studies were reporting on the 2 layer system. The participants in the Morgan et al. (2011) study reported improved mobility, speed of bandage application, and speed of oedema reduction with the 2 layer system. One participant with hand oedema was amazed that “they could see their knuckles” when they removed the bandages.

This key finding in this study was voiced by all of the participants with regard to compression therapy, and the message that they relayed through their interviews was that, while they did not necessarily enjoy or like compression bandaging, they were so pleased with the resulting oedema reduction that they observed when the bandages were removed that they were happy to continue with the therapy. Therefore, the resounding message from the participants of this study was: “Compression bandaging, a means to an end”. Participants gave varying statements to confirm this statement, such as:

P9 “when the leg went smaller and smaller and you get used to it, and you just like feel that it is working, and you are not so stressed”

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P14 “It works, okay and that is what makes you persevere through it, I see my ankles”.

P17 “I look at the bandage and I see a means to an end and a future again”

This study can provide evidence to strengthen the opinion of many other authors (Gray, 2013, Cooper, 2013, Brown, 2011), namely, that improved understanding of the dynamics of compression can enhance patients’ compliance with compression therapy.

7.4.3 Heat can be a factor that causes patients with lower limb lymphoedema to be non-concordant with wearing compression hosiery

The participants in this study also spoke about the perceived challenges of self- care and all voiced concerns about compression hosiery for a variety of reasons:

 Heat, especially in the summer (n=18),  Applying and removing compression hosiery (n=5),  Cost of compression hosiery (n=4),

The challenges that were perceived by the participants were outlined in their interviews and described narratively in their diary reflections.

Of the 18 participants who completed their diary, 15 participants wore their compression hosiery from between 12 and 14 hours each day.

Five participants actually required the assistance of a person to get the socks on and off. These participants were in the age group range of 45-60 years old; however, they were all in the morbidly obese category and physically could not reach their toes or manipulate a device. Therefore, the success or failure of their maintenance care depended upon another person. Tandler (2016) conducted a survey based study to explore professionals’ knowledge about and opinions on patients’ compliance with compression hosiery. She sent a questionnaire to 26 health professionals who were working in a tissue viability team in the UK. The findings indicated that application difficulties and discomfort were the main reasons for non-compliance with compression hosiery. Professionals also felt that they needed to be supported in the pursuit of training 187 in compression hosiery in order to provide the patient with the most therapeutic and suitable choice of stocking.

Thematic analysis of the 18 interviews also identified that many of the participants had a concern with wearing compression hosiery in the summer and whether they would be able to tolerate the combination of heat and compression. Analysis of the diaries confirmed this with seven participants reporting non-concordance with wearing their compression hosiery during the summer or while on holidays abroad.

Many studies discuss the concept of non-concordance with compression hosiery and offer many explanations as to why patients may be non-concordant, such as poor understanding, body image, pain, inability to apply and remove compression sock (Brown, 2011, Gray, 2013, Cooper, 2015, Williams, 2010, Martin et al., 2011). Few studies discuss the fact that heat and summer can be an issue, however, in this author’s study it was the participants’ first instinct to worry about how they would be able to continue to wear their stockings in the warm weather and what would happen if they went on holidays to a warmer climate. Indeed, Greene and Meskell (2017), who also examined an Irish population of patients with chronic oedema, reported that the participants in their study were concerned that they might not be able to tolerate compression during the summer heat, which could possibly indicate an Irish cultural trait.

Clinicians need to be aware that because lymphoedema is a chronic condition, whereby management of oedema reduction and skin is a daily task, which requires a lot of time on the part of the patient or their family member, that there will be times when the patient may not feel like doing self-massage or doing skin care. There may be days when the thought of putting on compression hosiery is just too much. As one participant stated in their interview on the topic of maintenance,

This study has highlighted that concordance with wearing compression is also not necessarily a constant. In this study, all participants felt that warm weather

188 could influence their level of concordance and not every day will be a “concordant day”. Therefore, the researcher sees a need for therapists to help patients live with the reality of an everyday condition and be able to problem- solve by self-bandaging or using wraps when they have had a day when applying compression hosiery was just not possible.

The use of a wrap as an alternative to either bandaging or hosiery in the case of stubborn, rebound oedema can provide an efficient, user-friendly, and cost effective option to patients. The proposal that the stiffness of a garment is of more significance than the mmHg applied from an elastic garment material has seen increased support for inelastic bandaging and the development of the Velcro wrap in the treatment and management of lymphoema (Partsch, 2007). Short stretch materials act by exerting high pressure peaks during walking and low pressures when the patient is resting, thus the materials have a massaging effect and are well tolerated because of the reduced pressure during inactivity (European Wound Management Association, 2005).

Tidhar et al. (2014) explored the application of a self-bandaging programme for patients with lower limb lymphoedema in Canada. The Canadian system of health care also has difficulties with the provision of compression bandaging and many patients have to access treatment in a private clinic capacity. The methods used included a retrospective chart review and telephone interviews with 30 patients who had attended a self-bandaging clinic in a physiotherapy centre. The findings were quite positive with regard to oedema reduction, self- efficacy, and self-care. All patients achieved significant oedema reduction in the range of 48%-92%. Interview themes included statements on feelings of being in control and empowerment.

This study demonstrates that self-maintenance in daily life poses many challenges and it is important that the patient has the tools to problem solve when situations occur, such as holidays, which results in changes in oedema. The addition of a wrap into the toolbox of lymphoedema self-care could allow the patient to reduce oedema independently and then be able to reapply the compression hosiery. Self-bandaging requires that the patient has good flexibility, strength, and dexterity therefore the participants of this study, who predominantly had varying levels of obesity (n=12), and who had difficulty 189 reaching down to their toes, would not have been able to self-bandage. One of the participants did practice self-bandaging and found it quite useful when an increase in oedema had occurred. A second participant used a full leg wrap as an alternative to full leg bandaging during the intensive therapy phase and into the maintenance phase. This participant had a particular skin condition and, because the skin required regular monitoring, the wrap was chosen, as it allowed daily inspection of the skin without interfering with the consistency of compression. The facility to regularly inspect the skin reduced the participant’s anxiety and allowed the participant to be independent in managing their compression throughout the therapy phase. While a wrap had not been introduced to the remaining participants, the findings of the study have highlighted the potential for improved self-efficacy with the use of a wrap.

The qualitative analysis of the 4 month maintenance diary followed the progress of 18 participants through the 4 month self-maintenance period. Each participant recorded daily activities and reflections under the headings of, maintenance self-care, skin care, compression, simple lymphatic drainage and exercise. A key finding emerged from diary data and will be now discussed.

7.4.4 Elevated BMI has a negative influence on self-efficacy in lymphoedema self-care

Worldwide obesity has more than doubled since 1980 (World Health Organisation, 2014). Data from the Department of Health (2015) shows that 60% of the Irish population aged 15 years and over are either overweight or obese, 37% are overweight, and a further 23% are obese. Obesity is a key risk factor for chronic conditions such as diabetes, cardiovascular disease, cancer and arthritis and mental health (Department of Health, 2015). Lymphoedema is not referred to in any departmental documents as a complication of obesity, however, a direct link between obesity and lymphoedema has been established (Greene et al., 2012, Lewis and Morgan, 2008).

Obesity leads to reduced mobility and has many associated co-morbidities, which include hypertension, heart failure, diabetes, and sleep apnoea. Many of the complications of obesity and lymphoedema can have serious implications on a persons’ health, however, the link between both conditions is rarely

190 discussed (Ching and Kumarasinghe, 2015). Obesity is present in 70% of patients with obstructive sleep apnoea (Tkacova, 2018) .Sleep apnoea causes the patient to feel more comfortable sleeping in a chair, as breathing and chest expansion is facilitated by being upright. The dependency of the limbs then results in pooling of fluid in the interstitial spaces and, if the dependency is prolonged for a lengthy period, eventually the tissue becomes non-pitting and lymphovenous oedema occurs (Stigant, 2009).

Qualitative analysis of diary data in this study demonstrated a cross over between poor self-efficacy scoring and an elevated BMI. As previously discussed in the demographic data section, only 15% (n=3) had a healthy BMI, 25% (n=5) were overweight, and the remaining 60% (n=12) were in the category of obese. Of those deemed to be obese, 20%( n=4) were moderately obese, 10% (n=2) were severely obese, 15% (n=3) were very severely obese and, of note, 15% (n=3) were super obese, indicating a BMI between 50-60 kg/m2.

Each participant recorded daily in the diary, for a period of 4 months, their account of self-care activities in relation to lymphoedema management, which included skin care, exercise, compression hosiery, and simple lymphatic drainage. Then all participants, at week 24, recorded their self-efficacy scoring, which indicated their level of confidence in the completion of lymphoedema self- care as an objective measure. Many of the participants progressed through the 4 month self-care period without an issue (n=13) and scored moderate to high in all 4 self-efficacy short forms (between 10 to 20). Self-efficacy scoring and BMI of the five participants who had difficulty during the 4 month maintenance period are displayed in the table below;

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Table 7.1 Self-efficacy and BMI of 5 participants

Participant Self- Self- Self- Self-efficacy BMI efficacy efficacy efficacy Daily activity Kg/m2 social emotional symptoms 5 16 12 11 16 40 6 20 9 12 4 43 9 12 11 12 11 60 10 20 10 14 15 54 17 15 6 9 5 60

Participant 5 scored an 11 in symptoms and 12 in emotions, therefore, a little confident in dealing with her oedema yet needing the support and assistance of others. This participant lived with her parents and required her mother’s help to get on her stockings in the morning. However, once her stockings were applied she could go about her day reasonably independently, therefore her self- efficacy scorings were not too low, ranging from 11-16. This participant’s BMI was 40kg/m2. She was only 41 years old and was still able to mobilise quite well, but she could not reach her toes to apply or remove her stockings.

Participant 9 scored 11 in emotion and daily activities and 12 for social and symptoms. This participant had been able to independently apply and remove her compression hosiery with the use of an aid, however, possibly due to poor skin integrity associated with skin folds and obesity, she developed a cellulitis during the 4 month maintenance period. This resulted in a rebounding of oedema, as compression hosiery was discontinued for a period and therefore her confidence was reduced, as she felt that she could not deal with her swelling and symptoms. Her mobility and activity level had reduced again, and she was only a little confident that she could complete her daily activities. In her diary, she described how much more confident she had become when the oedema had reduced, she was able “to mop the floor, climb the stairs”, but subsequent to the cellulitis and increased oedema, she was “disappointed”.

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Swelling of the limb and obesity cause overlapping skin folds and stretching of the skin. The function of the skin as a barrier to infection is compromised by minor skin breakdowns caused by the stretching of skin. The moisture and heat provided by the skin folds provide incubation for bacteria which in turn results in chronic recurrent cellulitis (Ching and Kumarasinghe, 2015).

Participant 10, who had a BMI of 54kg/m2, only scored low in emotions because her functional ability was still good despite her level of obesity. She had made a decision that she would no longer wear the stockings and therefore didn’t have to battle with getting them on. However, she stated in her diary that she was “disappointed” that she could not continue to manage the compression and also stated that she “worries” about the future.

Two remaining participants scored quite low in the self-efficacy tool, participant ID 6 and 17. Participant 6 had a BMI of 43kg/m2 and was a 77 year old lady, who had a stage 3 lower limb lymphoedema and other co-morbidities including diabetes and renal insufficiency. This participant’s level of mobility was quite poor and she required the assistance of her carer for all activities of daily living. This participant did very well, reduced oedema and improved her QoL. However, her success in maintenance was dependant on her carer and thus she scored a 4 in daily activities and a 9 in emotions. Her social score was a 20 because she knew she had all the help she needed. This participant’s symptom score was 12, as she had the knowledge to deal confidently with her oedema but needed the physical assistance of her carer.

Finally, participant 17 was in the super obese category and as a result her mobility was reduced and she required the assistance of a carer to assist with general household activities. All of her self-efficacy scores were quite low except for social functioning (5 in daily activities, 9 in symptoms, 6 in emotions). She scored 15 in social functioning, as she had good family and community support. This participant had an oedema reduction and had improved her quality of life during the study. She reports in her diary that she could apply and remove her compression hosiery and complete her skin care. Her activity level, though low, had also improved and she reported taking walks in her back garden.

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Wilkins et al.(2014) conducted a prospective survey of patients who attended a bariatric Lymphoedema clinic over a one year period in the United States. Thirty patients were referred to the clinic. The interventions provided at the clinic over a 4 week period included a multidisciplinary, holistic approach and included a session dedicated to the benefits of weight loss and exercise. The findings of this survey included a high level of co-morbidities in the obese population such as cardiac failure which excluded them from the criteria of treatment. Of the 30 patients who attended the clinic initially, 15 of those patients were discharged after the first consultation appointment due to co-morbidities.A high rate of non- attendance to clinic appointments, of up to one third, was highlighted with the remaining patients. The poor uptake of dietician referral was also seen as a barrier to improvement and the waiting lists were so long, up to six months, which was also seen as a barrier to treatment.

Quite possibly, had there been a disease specific self-efficacy tool that the participant scoring would have had greater relevance to the condition of lymphoedema. While "classic secondary" lymphoedema after oncological surgery is decreasing due to improved surgical techniques, the number of patients with obesity-associated lymphoedema is increasing at an exceptional rate. Obesity can cause lymphoedema, and worsen pre-existing lymphoedema in obese patients with lymphoedema. The management of obesity related lymphoedema requires a multidisciplinary approach, including CDT, psychology, dietician, and medical assessment for surgical options (Bertsch, 2018).

7.5 Summary

Five key findings have emerged:

1. Complex Decongestive Therapy does improve quality of life and reduce limb volume and more specifically; a. Quality of life scoring varies over time depending upon changes in limb volume.

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b. Lower limb lymphoedema has the greatest negative influence in the domain of function and appearance. 2. The patient perspective in 2018 in Ireland remains unchanged since last explored in 2010. 3. Compression bandaging was seen as a means to an end. 4. Heat can be a factor that causes patients with lower limb lymphoedema to be non-concordant with wearing compression hosiery. 5. Elevated BMI has a negative influence on self-efficacy in lymphoedema self-care.

Key finding No. 1 is not surprising, as it would have been anticipated that CDT would indeed reduce oedema and, as a result, improve QoL. This finding is consistent with the majority of studies that examine lymphoedema treatment and outcomes (Huggenberger et al., 2015; Kim and Park, 2008; Noh et al., 2015).

Key finding No. 2, which related to the participants’ experience of living with lymphoedema, is unfortunate, as for people living with lymphoedema in Ireland there have been no changes to the challenges faced by this population in their pursuit of treatment and care in Ireland.

Key findings No. 3 and No. 4 relate to the experiences of self-care in lymphoedema and the personal challenges experienced by this population. Although it is well recognised from many research studies (Brown, 2011, Gray, 2013, Morgan et al., 2011, Anderson, 2015) that compression bandaging and hosiery can pose physical and psychological barriers to the patient, the researcher has discovered a different view point on compression than has not been reported in research studies until now. The participants in this study developed the theme of “a means to an end”, indicating the way in which they view compression and the “trade-off” of therapeutic value for concordance. The challenge of heat and summer months as a threat to concordance with compression is also rarely reported in research, except for one other Irish study (Greene and Meskell, 2017).

Key finding no. 5 was about the participants’ journey through self-maintenance and the influence that an elevated BMI can have on the day to day physical,

195 social, and emotional aspects of self-care. The subjective reporting in the participants’ diary is strengthened by the objective scoring of the self-efficacy tool. The area of self-maintenance and self-efficacy in lymphoedema is poorly researched and this study introduces the concept that an elevated BMI does have a negative influence on self-efficacy in lymphoedema self-care.

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Chapter 8 Conclusion

8.1 Introduction

The purpose of this chapter is, firstly, to make recommendations for clinical practice and future research. The strengths and limitations of the study will also be presented, followed by the plans for dissemination of the study outcomes. Following this, a conclusion to the thesis will be presented, whereby the key points from the literature review and the research design chosen are highlighted, with the main results of the thesis briefly discussed. Following this, an overall conclusion is presented, including the main recommendations arising from this work.

8.2 Strengths of the study

Following an extensive literature review, it was identified that the area of lower limb, primary and secondary, non-cancer related lymphoedema is poorly researched. Although there is a predominance of research literature in the area of breast cancer related lymphoedema, this body of knowledge cannot be truly transferred to the field of lower limb lymphoedema. In addition, while there are many opinion articles on lymphoedema self-maintenance, there is a paucity of original research exploring the challenges of lymphoedema self-care. The researcher also identified a research gap in the examination of patients’ experiences of each of the elements of CDT, namely, compression bandaging, manual lymphatic drainage, skin care, and exercise. Therefore, a strength of this study is that it set about to address real life clinically relevant research gaps.

A mixed methods approach was chosen. Zhang and Creswell (2013) state that the integration of the data from mixed methods studies gives strength and greater clarity to the research findings than if one set of methods were used. Traditionally, nursing research has largely focused on the use of qualitative or quantitative studies, however, in the last ten years, the complexity of health sciences and the need for research to provide the answers that will assist 197 clinicians to utilise an evidence base to develop services has seen an increase in the use of mixed methods research to answer these complex, multi- dimensional research questions. In this study, had quantitative data been collected in isolation, the changes in limb circumference, QoL and self-efficacy could not have been explained. However, the integration of both quantitative and qualitative data has explained the changes in measurements, QoL and self- efficacy scoring based on the participants’ experiences in treatment and self- maintenance.

The 20 person sample chosen for this study provides a good representation of the population of patients with non-cancer related lower limb lymphoedema, the experiences of these patients living with lymphoedema and also their experiences of self-maintenance. The use of circumferential measurement at 4cm intervals is documented as reliable and has good inter-rater reliability (Williams and Whitaker, 2015). The QoL questionnaire is disease-specific and has been tested for reliability (Keeley et al., 2010). The self-efficacy tool, which was applied at week 24, was also tested for validity and reliability by other authors (Gruber‑Baldini et al., 2017). Therefore, strength of this study was the use of tools that are proven to be both reliable and valid.

8.3 Limitations of the study

A limitation of the study could be the sample size of 20 participants, particularly from a quantitative point of view.

The follow-up period of 4 months in the self-maintenance phase of the study is another limitation. A greater insight into the challenges and self-efficacy in lower limb lymphoedema care could have been provided had the follow-up timeline been longer. However, the study timeframe did not allow for a duration any greater than the 4 month follow-up that was utilised.

The fact that the researcher was also the therapist in the study may have allowed bias to arise by virtue of the possibility that the researcher may have allowed her prior knowledge of this particular population to influence the interpretation of data. However, methods used by the researcher included 198 the use of a self-efficacy tool as a means of objective assessment of lymphoedema self care. Also, the presentation of multiple direct quotes during the qualitative data analysis, in order to demonstrate that the researcher was not influenced by her own perception of what the participant was saying but was analysing the data as it was presented by the participant using a specific thematic framework to guide the process of data analysis.

Another limitation was the unavailability of a disease-specific self-efficacy tool for lower limb lymphoedema. However, the self-efficacy tool that was used was specific to chronic disease management across a broad spectrum of chronic diseases and it was a validated and reliable tool. The researcher also used cognitive interviewing with 2 participants to assess their understanding of the statements in the 4 Short Forms and adjustments were made to the use of “symptoms” and “doctor” as terms in the tool, which the participants translated as “swelling” and “therapist” accordingly to facilitate their interpretation of the statements.

Despite the limitations discussed above, this study does provide insight into the therapeutic effects of CDT on limb volume and QoL, the everyday experiences of patients with non-cancer related lymphoedema, and the challenges that lymphoedema self-care can present.

8.4 Plans for dissemination

The findings of this research will be implemented locally, nationally, and internationally. The process has already begun, as outlined by the list of oral and poster presentations. The research has been presented locally to a regional Research Group within the Health Service Executive (HSE) and nationally at the Royal College of Surgeons (RCSI) annual conference, the Institute of Community Health Nursing (ICHN) conference, the Nursing and Midwifery Planning and Development Unit (NMPDU) conference, and the Wound Management Association of Ireland (WMAI) conference. Internationally, the research has been presented at the European Wound Management Association (EWMA) conference in Poland (2018), and Belfast (2017), and the Wounds UK

199 conference in Harrogate (2016). This researcher has also been accepted for an oral presentation of the study findings at the next Wounds UK Conference in Harrogate, 2018.

It is the researcher’s intention to publish the findings of the study in a leading international journal. Arising from this work, the researcher has been involved in a national project to develop a Model of Care for Lymphoedema services in Ireland and it is hoped that this study will add to the small body of research evidence in existence in the Irish setting. The researcher is also involved on a national basis in the National Lymphoedema Framework of Ireland (NLFI), whose goal is to develop a program of education in Lymphoedema diagnosis, treatment, and management for health professionals in Ireland. The researcher was also involved in the publication of a Systematic Review on Community Tissue Viability Services and Recommendations for Practice (2018).

The author has also provided talks on lymphoedema treatment and management for nurses in all areas of care, including acute care, nursing homes and public health in order to improve the awareness of lymphoedema and chronic oedema. The publication of this study will improve the researcher’s creditability as a knowledgeable practitioner and researcher in the area of lymphoedema treatment and management.

8.5 Recommendations for Nursing Practice

The recommendations for Nursing Practice are as follows:

1. A focus on prevention and early treatment of lower limb oedema.

This study has highlighted the plight of patients with non-cancer related lower limb lymphoedema in the Irish health service, the delay and sometimes absence of diagnosis of the condition, and following on from that the absence of referral pathways and appropriate treatment services. The author would recommend the introduction of lower limb oedema clinics to complement the present practice of Nurse led leg ulcer clinics, with the aim of preventing

200 patients with lower limb oedema and lymphoedema from progressing to the stages of ulceration before any therapeutic action is taken.

Although the true potential for using compression to prevent complications and deterioration in subclinical lymphoedema has not been fully examined, low levels of compression (14-21mmHg) are recommended in patients who are known to be at risk of developing lower limb lymphoedema (Doherty et al.,2006). Also, having significant co-morbid conditions such as obesity and cellulitis, is generally thought to add to the risk of developing secondary lymphoedema (Clinical Resource Efficiency Support Team (CREST), 2008).

The provision of early and co-ordinated intervention, including skin care awareness, improving general healthcare and maintaining limb function through exercise and normal function can contribute to better disease management and support optimal self-care.

In reality, for the short-term foreseeable future, not all clinics will have a lymphoedema therapist who can provide CDT. However, the introduction of lower limb oedema clinics, with the aim of early commencement of compression therapy to prevent advancement of the condition, would be a reactive strategy in the prevention of chronic oedema and many secondary non-cancer related lower limb lymphoedema.

2. The identification and onward referral of high risk groups for the development of secondary lower limb lymphoedema.

In this study, the influence of obesity on lower limb lymphoedema has been identified. Obesity then results in reduced mobility, which in turn then increases the risk of oedema. Of the 20 participants in this study, 10 participants had a secondary lymphoedema either related to chronic venous insufficiency (n=2) or obesity (n=4) or a combination of both CVI and obesity (n=4). An early identification of lower limb secondary lymphoedema as a risk factor of their condition and the appropriate treatment at that time could have prevented the further progression of the oedema. The identification of groups within the health care setting who are at risk of lower limb secondary lymphoedema due to reduced mobility or immobility, obesity and chronic venous insufficiency and the

201 early intervention of compression therapy in dedicated clinics would provide a positive therapeutic impact for this patient population.

8.6 Recommendations

8.6.1 Recommendations for Education

The recommendations for education are as follows;

 The qualitative interviews of all the participants (n=18) led to the development of the theme of poor knowledge in the health care professions about lymphoedema. This researcher would like to use the evidence from this study and from that of Murray et al. (2010) to address the issue of poor knowledge within the health professions. The International Lymphoedema Framework (2016) and Lymphoedema Education Benchmark Statements(LEBS) have standardised the requirements of an educational program for health practitioners in the area of lymphoedema. The researcher would like to collaborate with other health educators in the development of an educational program that incorporates the recommendations of the LEBS for health professionals at all levels, both undergraduate and postgraduate.  The development of educational material for patients with lymphoedema and their families. Lower limb lymphoedema self-care requires the patient to be competent in the 4 cornerstones of lymphoedema self- maintenance, which include skin care, exercise, simple lymphatic drainage, and compression hosiery, and sometimes self-bandaging. This study followed the participants through 4 months of self-maintenance care. The participants had all received both written and verbal one to one instruction on each of the 4 cornerstones of self-care. One participant’s diary reflected the need for more visual, diagram style educational material, with regard to simple lymphatic drainage. All participants reported the use of daily emollients in skin care, but none of the participants reported foot care or inspection of skin folds, which the

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researcher acknowledges was a deficit in her verbal education to the participants.  There is an abundance of educational literature for patients with cancer- related lymphoedema, mostly upper arm lymphoedema secondary to breast cancer; however, there is a lack of educational material for patients with lower limb lymphoedema. In addition, as highlighted by the participants in this study, there is a need for more visual educational material, through diagram, video, and pictures. The participants also discussed the sources of information and the need to be sure that the information that they can access is from a reliable source appropriate to their learning needs and literacy. Educational material also needs to be at the level of the patients, in easy to understand plain English.

8.6.2 Recommendations for future research

The need for further research in the area of non-cancer related lower limb lymphoedema is significant. The vast area of self-maintenance care needs further research studies that are longitudinal and follow the participants for periods greater than 1 year in order to get a clear picture of the successes, barriers, and challenges of lymphoedema self-care. In addition, the investigation of QoL changes over a prolonged period of self-care would be of interest. The identification of the absence of a disease specific self-efficacy tool for lower limb lymphoedema highlights the need for the development and validation of such a tool for use in future self-maintenance studies.

Once again, without evidence of the prevalence and burden of this disease, the development of policy and services to assess and treat this condition will not be funded. Presently, in Ireland, there is only one prevalence study, which looked at the prevalence of lower limb lymphoedema within a vascular clinic setting (Gethin et al., 2012). Many more prevalence studies are required from a population perspective such as Moffatt et al. (2003) and Moffatt et al. (2017), also looking at specific aetiologies such as obesity or wheelchair bound patients. Internationally, studies such as LIMPRINT (Lymphoedema Impact and Prevalence –International Lymphoedema Framework, 2014-2017) aim to evaluate the size and impact of chronic oedema in different countries.

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Presently, LIMPRINT is at the stage of initial data analysis and the findings of the study will hopefully be published later this year.

8.6.3 Recommendations for Policy

Presently, in Ireland, there is provision for services in the area of cancer-related lymphoedemas and these services are governed by Department of Health. However, there is no policy or governance surrounding the area of non-cancer related lower limb or primary lymphoedemas. The initial preparation for policy begins with a model of care, which then needs to be presented at government level in order to highlight the need for such a policy and then the further development of services. The U.K. have implemented such policy and have developed lymphoedema clinics in many areas. However, as stated prior to this, the development of policy and services is reliant on evidence of prevalence and impact. Presently, in Ireland, due to a lack of such research, it is only possible to predict the prevalence of lymphoedema and chronic oedema in Ireland based on the prevalence figures produced by other countries. Therefore, there is an urgent need for prevalence studies in Ireland in order to influence Irish policymakers.

8.7 Reflection

The commencement of my journey began in the nurse led leg ulcer clinics as a tissue viability nurse, where many patients presented with significant problems due to ongoing years of unresolved, undiagnosed lower limb oedema. I did not feel that I was equipped with the expertise or knowledge needed to treat these patients. I also realised that there was no referral pathway for these patients. These were the patients who had repeated hospitalizations for cellulitis and whose condition worsened on each admission. This was my impetus to acquire more knowledge. As a part fulfillment of a Masters in Advanced Practice, I conducted a systematic review on the role of manual lymphatic drainage in the treatment of lower limb lymphoedema. The findings of this systematic review established that most of the evidence was anecdotal. Therefore, my next step was to train as an MLD Therapist and bring this back to my practice. After

204 practicing for over a year, the stage was then set for the next step, which was this research study, in order to provide evidence around the intensive and maintenance phases of treatment and patient outcomes, the patients’ experiences of living with lymphoedema, and self-care. I also used this study as an opportunity to achieve an academic qualification that I suppose would give strength to my voice in the world of lymphoedema.

I found the process of conducting a research study a formidable challenge, however, the support and direction of my supervisors in the Royal College of Surgeons in Ireland gave me the confidence to carry it through. Anyone who works in the area of lymphoedema treatment will concur that this field of practice is physically and mentally quite draining on the therapist. As the findings of the study will support, these patients have immense psychological burden from years of unresolved oedema, feelings of hopelessness and, quite often, social isolation. Many of the participants had never shown their affected limb to anyone and, as their interviews show, they hid their limbs for years under big trouser legs. So for them the possibility of actually talking about their condition and their experiences was “consoling” or therapeutic. However, as the researcher and the therapist, it required constant reflection on each role in order to be objective and yet be empathetic to the participants. So, essentially, on many occasions I felt both physically and mentally challenged myself.

The requirement to use databases, that I was unfamiliar with, was a challenge also for me. These included reference managing systems such as Endnote and data analysis using SPSS. However, training programs and individualized support from my supervisors helped me to overcome my fear of databases.

I would pride myself as being an advocate for my patients and essentially a clinician at heart. However, as the research study has progressed and as having presented my work at different stages to varying academic and clinical audiences, I find that I have begun to think more critically about research and my peers tend to look to me for academic advice. Therefore, I also feel that the process of progressing through a research study has allowed me to show other nurses that it is possible to do the same and hopefully some will follow the same pathway.

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8.8 Conclusion to the Thesis

This study set out to explore the experiences of patients with primary and secondary lower limb lymphoedema during the intensive and maintenance phases of Complex Decongestive Therapy and its impact on their lives. This was a mixed methodology study. The quantitative data examined changes in limb volume and QoL during the intensive and maintenance phases of CDT. The qualitative data examined the participants’ experience of living with lymphoedema, their experiences of the CDT treatment, and the challenges of self-caring for lower limb lymphoedema.

The findings included reduced limb volume from baseline to week 8 during intensive therapy and then a fluctuation of oedema for 6 of the participants during the 4 month self-maintenance period due to reasons such as cellulitis, non-concordance with compression hosiery, and difficulty with applying and removing compression hosiery. The participants’ QoL improved from baseline to week 8 for all participants and then the participants, who had increased oedema from week 8 to week 24, had slight reductions in QoL but not reduced back to baseline scoring.

The participants’ experiences of living with lymphoedema in Ireland developed themes of delayed diagnosis, poor knowledge within the health care system, the negative physical, psychological and financial impact of lymphoedema, and indeed the positive impact of treatment. All participants reported a positive experience from all the elements of CDT, however, compression bandaging was the most challenging aspect. Nevertheless, adherence to the program was influenced by the visual experience of seeing the limb reduce in size. Lymphoedema self-care proved the most challenging for those participants with an elevated body mass index due to difficulties applying hosiery and reduced mobility.

This research study has been successful in providing valuable information in relation to treatment outcomes, living with lower limb lymphoedema in Ireland, and providing an insight into the challenges that lymphoedema self-care can offer.

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Appendices

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Appendix 1: Ethical Approval Letter

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Appendix 2: Ethical Approval RCSI January 2016

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Appendix 3: Ethical Approval RCSI April 2017

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Appendix 4: Patient Information Leaflet

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229

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Appendix 5: Patient Consent Form

Version: 1 Date: 11/11/2015

Title of Study:

Exploring the experiences of patients with primary lymphoedema during the intensive and maintenance phases of Complex Decongestive Therapy (CDT) and its impact on their lives.

I have read and understood the Information Leaflet about this research project. The information has been fully explained to me and I have been Yes No able to ask questions, all of which have been answered to my   satisfaction.

I understand that I don’t have to take part in this study and that I can opt out at any time. I understand that I don’t have to give a reason for Yes No opting out and I understand that opting out won’t affect my future   medical care.

I am aware of the potential risks of this research study. Yes No   I understand that an audio recording will be made and that I have the Yes No right to review and edit any transcripts to which I have contributed.   I have been given a copy of the Information Leaflet and this completed Yes No consent form for my records.   Storage and future use of information:

no yes  I give permission for material/data to be stored for possible future  research related to the current study without further consent being required subject to research ethics committee approval.

Participant Name (Block Capitals): ______Participant Signature: ______Date: ______

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I the undersigned have taken the time to fully explain to the above patient the nature and purpose of this study in a manner that they could understand. I have explained the risks involved as well as the possible benefits. I have invited them to ask questions on any aspect of the study that concerned them.

Principal Investigator Name & Qualifications (Block Capitals): ______

Signature: ______Date: ______

3 copies will be made: 1 for patient, 1 for PI and 1 for practice records (if relevant).

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Appendix 6: Letter to Director of Public Health Nursing

Primary Care Centre Laois 11/11/2015 Mrs Y Director Public Health Nursing Tullamore Laois

Dear Mrs Y, As you are aware I am progressing through my studies , PhD through Research in the Royal College of Surgeons in Ireland and I want to take this chance to thank you for your support so far. A requirement of the programme is that I conduct a research study in my area of practice. I plan to do a study on ‘ Exploring the experiences of patients with primary lymphoedema during the intensive and maintenance phases of Complex Decongestive Therapy (CDT) and its impact on their lives. ‘ I have applied to the Ethics committee for permission to submit my study for approval. My hope is to conduct the research study in Portarlington Primary Care Centre, Laois with your approval. I look forward to hearing from you,

Thanking you, Mary Costello, TVN, MLD Therapist

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Appendix 7: Response to query from Tullamore Ethics Committee

Mary Costello

Tissue Viability Nurse

Mountmellick Primary Care Centre

Mountmellick

Laois

Ref: 021215MC Re: Exploring the experiences of patients with primary and secondary lower limb lymphoedema during the intensive and maintenance phases of Complex Decongestive Therapy and its impact on their lives

Clarifications as follows;

1. Steps in place for patients should this particular bandage slip or move from its site

 The lymphoedema therapist is specifically trained in the application of a full-leg bandage during the Dr. Vodder Certification Course in order to promote competence in bandage application.

 This particular style of bandaging uses a polyester padding under the bandage to protect the skin and prevent pressure damage. The therapist will also use ‘pillows’ of padding to protect skin folds.

 The fact that the bandage is changed three times weekly also protects from slippage as the regularity of bandage change compensates for limb volume changes.

 The skin is inspected on each visit.

 The client or carer is also instructed that if they have any concerns or feel that the bandage is causing trauma, the client can remove it themselves.

2. In the case where a statistician may be required, the data will be anonymous and only the researcher will have the identifiable codes.

3. Retention of data; The proposed timeframe for retention of data will be 2years and 6 months, as suggested in case of any queries after publishing.

I hope that this information will add the clarity required.

Thanking you, Regards,Mary Costello

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Appendix 8: Research Ethics Approval

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Appendix 9: Lymphoedema Handout

HANDOUT :

BREATHING EXERCISES

Exercise 1:

Procedure: Lay your arms on the table in front of you, making a U shape and with your head turned to the right. While breathing in, lift both arms simultaneously and hold for 7 secs. Then lower them as you breathe.

Variation: Breathe in while you extend and breathe out while you flex both arms at the shoulder.

Exercise 2:

Position: Side lying on the left side

Procedure: Extend the left leg and lay the flexed right leg over it. The right knee touches the bed.

Extend the right arm at the shoulder, above the head and back behind the right ear.

Rotate the upper body as if moving towards a lying position without losing contact with your right knee on the table. Be aware of which part of your lungs you breathe into for about 20 – 30 secs.

Relax and move onto your back and notice once again which part of your lungs you breathe into.

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HANDOUT :

OEDEMA EXERCISES : for patients with a leg oedema

Each exercise should be repeated 10 times with appropriate pauses. Repeat exercises 2 – 3 x per day and never over exert. Breathing exercises can be interspersed in the breaks.

1. Position: Standing Procedure: “Roll on the soles”: change position from heel to toes

2. Position: Standing Procedure: With feet angled out , lift a knee until the hip is in 90 degrees flexion while pointing the toes down to the floor.

3. Position: Sitting with legs outstretched Procedure: Bend the knees then “roll the soles” from heel to toes.

4. Position : lying on the back Procedure: “Bicycle” with the legs in the air while flexing and extending the ankle. Extend against an imagined resistance.

5. Position: Lying on back with knees bent Procedure: One after the other, extend and hold the right and left legs in the air. Against an imagined resistance, make circles to the right and to the left with the feet then circle inwards, then outwards.

6. Position: Lying on back with knees bent

Procedure Raise the head, right shoulder and arm and try to stretch past the left knee. Repeat on the opposite side with the left shoulder and arm.

Variation: Raise the upper body and press both knees together (hold for 7 secs). Repeat while pushing the legs apart.

HANDOUT : Recommendations to patients with lymphoedema 238

Try to sleep so that your body weight is not pressing on the limb (slows circulation).

Exercise is good but for shorter rather than longer periods so as not to over-tire the limb.

Use the unaffected limb for blood pressure measurements, injections and avoid all injury to the limb.

Have any infections and unusual redness (inflammation) attended to immediately by a medical doctor.

Have MLD done immediately on recent bruising..

Clothing should be loose and comfortable with no tight constriction on the affected side e.g. elasticised socks or under garments.

Keep the affected limb as clean and supple as possible by using emollients to wash and moisturise.

When taking a shower or bath use tepid-warm rather than hot-warm temperatures as heat will make the edema worse.

Report any chaffing, redness, indentation, looseness or tightness of the compression hosiery to your therapist.

Keep your weight down as much as possible and try to lose weight if you are overweight.

A low-salt diet is recommended.

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Appendix 10: Simple Lymphatic drainage handout

SIMPLE LYMPHATIC DRAINAGE

SELF MASSAGE

This self massage document applies to clients who has completed intensive and maintenance manual lymphatic drainage witha certified MLD Practitioner

Adapted from Lymphoedema, Twycross, Jenns and Todd,2004

SELF MASSAGE

A warm, quiet and comfortable room will provide a suitable environment to start the massage.

Ideally, you should be lying down in order for the lymph vessels of the groin to be free.

The massage should be light, slow and rhythmic and should not cause pain or discomfort.

Oils or moisturisers should not be used prior to the massage as this can cause the hand to slip.

For best benefit from the massage, it is preferable that you do the massage every day for 15-20 minutes.

1.DEEP BREATHING

Start the session with a breathing exercise. Lying flat on the back with your knees bent, place your hands flat just below the ribs to offer a little resistance. Without arching the back, take a slow, deep breath in through the nose. Your hands should rise on breathing in (abdomen ballooning out).

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Hold for a count of 2. Then slowly let the breath out through the mouth. The hands should lower on breathing out (abdomen sinking in). The upper chest should not move. Repeat the deep breathing 3 – 5 times, taking care not to become dizzy.

2. NECK MASSAGE

First position

Place your fingers on either side of the neck, keeping the fingers together and straight. The little finger of both hands should be just below the ear lobes. Keeping your hands as near as possible to the perpendicular, take the skin back and then circle down towards the shoulders. Release the light pressure and let the skin bring your hands back to the start position so completing the circle. Repeat the movement 5 times in the same position.

Second position

Move your hands down on both sides of the neck to a second position, so they are approximately half way between the ears and the shoulders. Repeat the movement as described for the first position 5 times. The skin will usually move more easily here.

Third position

Move both of your hands down to the base of the neck to a comfortable position between the collarbone and the large shoulder muscle. Relax your wrists and fingers. On both sides, using the pads of your index and middle fingers bring the skin forwards towards the collarbone and then circle in towards the neck. Release the light pressure and let the skin bring your fingers back to the start position so completing the circle. Repeat the movement 5 times in the same position .

Repeat all of the neck massage on both sides 3 times.

3. GROIN LYMPH NODES MASSAGE

First Position

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Place your fingers on the inner aspect of the upper thigh, keeping them together and straight. Take the skin towards the inner thigh and then circle up towards the groin. Release the light pressure and let the skin bring your hand back to the start position so completing the circle.

If both legs affected repeat on both right and left groin.

Second position

Move your hands to the top of the thigh to a second position. Take the skin towards the inner thigh and then circle up towards the groin. Release the light pressure and let the skin bring your hands back to the start position so completing the circle. Repeat the movement 5 times in the same position.

Repeat all of the groin lymph nodes massage 3 times.

4. DEEP BREATHING

Finish the session with the breathing exercise . Relax and remain lying down for a couple of minutes before getting up.

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Appendix 11: Lymphoedema Measurements – Week 1

Progress report: circumference and volume of limb: Leg: Right/Left

Patient: ______Therapist: ______Bandages: ______

History: ______

INITIAL

1st week of treatments 2nd week of 3rd week of 4th week of treatments treatments treatments

Date: Weight: Date: Weight: Date: Weight: Date: Weight:

Posn. Circ Circ. Circ. Circ. Diff. Circ. Circ. Diff. Circ. Circ. Diff. . 2 2 2 2 Cm Rt. Rt Rt Rt Lt. Lt Lt Lt

-4

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1st week of treatments 2nd week of 3rd week of 4th week of treatments treatments treatments

Date: Weight: Date: Weight: Date: Weight: Date: Weight:

100

Volum e

INITIAL VOL: ml. FINAL VOL: ml.

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Appendix 12: LYMQOL LEG

Lymphoedema Quality of Life Tool

This questionnaire has been designed and validated for patients with chronic oedema/ lymphoedema of one or both legs to measure quality of life.

Please tick the box that best describes how you feel about each of the questions.

Name: ……………………………………………….. Hospital Number:………………………

Date: …………………………

(Q1) How much does your swollen leg affect the

following activities? If any of the items are not applicable to you, please Not A Quite A lot write N/A in the relevant answer box(es) at all little a bit a) your walking b) your ability to bend, e.g. to tie shoelaces or cut toenails c) your ability to stand. d) your ability to get up from a chair. e) your occupation f) your ability to do housework

(Q2)Does the swelling affect your leisure activities/ social life?

Please give examples of this ......

(Q3) How much do you have to depend on other people?

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Not A Quite A lot at all little a bit (Q4) How much do you feel the swelling affects your appearance? (Q5) How much difficulty do you have finding

clothes to fit? (Q6) How much difficulty do you have finding clothes you would like to wear? (Q7) Do you have difficulty finding shoes to fit?

(Q8) Do you have difficulty finding socks/ tights/ stockings to fit? (Q9) Does the swelling affect how you feel about yourself? (Q10)Does it affect your relationships with other people?

Not A Quite A at all little a bit lot (Q11) Does your lymphoedema cause you pain? (Q12) Do you have any numbness in your swollen leg(s)? (Q13) Do you have any feelings of "pins & needles" or tingling in your swollen leg(s) (Q14) Does (do) your swollen leg(s) feel weak? (Q15) Does (do) your swollen leg(s) feel heavy?

In the past week…. Not A Quite A at all little a bit lot (Q16) Have you had trouble sleeping? (Q17) Have you had difficulty concentrating on things,

e.g. reading? (Q18) Have you felt tense? (Q19) Have you felt worried? (Q20) Have you felt irritable? (Q21) Have you felt depressed?

(Q22) Overall, how would you rate your quality of life at present?

Please mark your score on the following scale:

0 1 2 3 4 5 6 7 8 9 10

poor excellent

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Thank you for completing this form.

If you have any comments or queries about it, please discuss these with

Dr V L Keeley, Consultant

Questions 16 to 21 have been reproduced with permission from the EORTC.

These questions are only a part of the QLQ-C30 Questionnaire. Copyright November 2007 Ref LEG V II

All rights reserved. This document can be used or reproduced freely provided that this copyright statement is left intact, that the source is acknowledged, that the user registers and that no changes are made without permission of the author. Application for permission and for registration should be forwarded in writing to Dr Vaughan Keeley, Consultant in Palliative Medicine, Lymphoedema Clinic, Royal Derby Hospital, Uttoxeter Rd, Derby. DE223NE

247

Appendix 13: LYMQOL LEG scores

LYMQOL LEG- Scoring System

Lymphoedema Quality of Life Tool

The score for the individual responses are given below. If the item is not scored and left blank or not applicable this is scored with a 0. Domain totals are calculated by adding the individual scores and dividing the total by the number of questions answered. (If >50% of questions per domain are not answered this cannot be calculated and =0).

The four domains and their corresponding questions are: Function 1 (a-f), 2,3

Appearance 4,5,6,7,8,9,10 Symptoms 11,12,13,14,15 and Emotion 16,17,18,19,20,21.

Overall quality of life (Q22) is scored as the value marked by the patient, between 0-10.

(Q1) How much does your swollen leg affect the

following activities? If any of the items are not applicable to you, please Not A Quite A lot write N/A in the relevant answer box(es) at all little a bit a) your walking 1 2 3 4 b) your ability to bend, e.g. to tie shoelaces or cut 1 2 3 4 toenails c) your ability to stand. 1 2 3 4 d) your ability to get up from a chair. 1 2 3 4 e) your occupation 1 2 3 4 f) your ability to do housework 1 2 3 4

(Q2)Does the swelling affect your leisure 1 2 3 4 activities/ social life?

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Please give examples of this ......

......

(Q3) How much do you have to depend on other 1 2 3 4 people?

Not A Quite A lot at all little a bit (Q4) How much do you feel the swelling affects 1 2 3 4 your appearance? (Q5) How much difficulty do you have finding 1 2 3 4 clothes to fit? (Q6) How much difficulty do you have finding 1 2 3 4 clothes you would like to wear? (Q7) Do you have difficulty finding shoes to fit? 1 2 3 4 (Q8) Do you have difficulty finding socks/ tights/ 1 2 3 4 stockings to fit? (Q9) Does the swelling affect how you feel about 1 2 3 4 yourself? (Q10)Does it affect your relationships with other 1 2 3 4 people?

Not A Quite A at all little a bit lot (Q11) Does your lymphoedema cause you pain? 1 2 3 4 (Q12) Do you have any numbness in your 1 2 3 4 swollen leg(s)? (Q13) Do you have any feelings of "pins & 1 2 3 4 needles" or tingling in your swollen leg(s) (Q14) Does (do) your swollen leg(s) feel weak? 1 2 3 4 (Q15) Does (do) your swollen leg(s) feel heavy? 1 2 3 4

In the past week…. Not A Quite A at all little a bit lot (Q16) Have you had trouble sleeping? 1 2 3 4

249

(Q17) Have you had difficulty concentrating on 1 2 3 4 things,

e.g. reading? (Q18) Have you felt tense? 1 2 3 4 (Q19) Have you felt worried? 1 2 3 4 (Q20) Have you felt irritable? 1 2 3 4 (Q21) Have you felt depressed? 1 2 3 4

(Q22) Overall, how would you rate your quality of life at present?

Please mark your score on the following scale:

0 1 2 3 4 5 6 7 8 9 10

poor excellent

Thank you for completing this form.

If you If you have any comments or queries about it, please discuss these with

Dr V L Keeley, Consultant

Questions 16 to 21 have been reproduced with permission from the EORTC.

These questions are only a part of the QLQ-C30 Questionnaire.

All rights reserved. This document can be used or reproduced freely. If all or part of the documents be reproduced, we request that the source and copyright owners be acknowledged. Application for the copyright owners permission should be forwarded in writing to Dr Vaughan Keeley, Consultant in Palliative Medicine, Nightingale Macmillan Unit, 117A London Road, Derby DE1 2QS.

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Appendix 14: PROMIS Item Bank v1.0 Short Form

Self-Efficacy for Managing Emotions – Short Form 4a

Please respond to each question or statement by marking one box per row. CURRENT level of I am not at I am a little I am I am quite I am very confidence all confident somewhat confident confident confident confident I can handle negative feelings I can find ways to manage stress I can avoid feeling discouraged I can bounce back from disappointment

Self-Efficacy for Managing Social Interactions – Short Form 4a

Please respond to each question or statement by marking one box per row. CURRENT level of I am not at I am a little I am I am quite I am very confidence all confident somewhat confident confident confident confident I can talk about my health problems with someone If I need help I can find someone to take me to the doctor’s office I can get emotional support when I need it I can ask for help when I don’t understand something

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Self-Efficacy for Managing Symptoms – Short Form 4a

Please respond to each question or statement by marking one box per row. CURRENT level of I am not at I am a little I am I am quite I am very confidence all confident somewhat confident confident confident confident I can manage my symptoms during my daily activity I can keep my symptoms from interfering with relationships with friends and family I can manage my symptoms in a public place I can work with my doctor to manage my symptoms

Self-Efficacy for Managing Daily Activities– Short Form 4a

Please respond to each question or statement by marking one box per row. CURRENT level of I am not at I am a little I am I am quite I am very confidence all confident somewhat confident confident confident confident I can perform my household chores I can go shopping and Run errands I can walk around inside my house

I can maintain a regular exercise program

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Appendix 15: Grant Approval Tullamore

1st April 2016

Ms Mary Costello

Mountmellick Primary Care Centre

Connolly Street

Mountmellick

Co. Laois

Dear Mary

Re: Research PHD – RCSI

I refer to your recent correspondence requesting funding in respect of the above programme.

I am pleased to inform you that funding will be provided to cover the fees associated with course in accordance with the Health Service Executive Sponsorship Scheme (Circular 020/2014). You will be covered for 100% of the fees associated with year two.

 Please note as a condition of funding you will be required to facilitate oral/poster presentations at NMPD conferences and local services within 1 year of programme completion.

 Please note also that you are required to submit a copy of your results to this office on completion of the course.

 If you do not proceed with this course you MUST inform the NMPD by contacting [email protected] or by written confirmation.

I would like to take this opportunity to wish you all the very best with your studies.

Please note: Third Level College must Invoice NMPD directly, as re-imbursement directly to course participant is not possible.

253

Yours sincerely

Mary Manning, Director / National Nursing Lead for Dementia Nursing & Midwifery Planning & Development

cc; Joan Bourke, Director of Public Health Nursing, Health Centre, Tullamore

254

Appendix 16: Grant Amendment

Research Ethics Committee Appendix 11: HSE – Midland Area

HSE Area Offices

st 31 March 2017 Ref: 021215MCArden Road

Tullamore

Ms. Mary Costello Co. Offaly

Tissue Viability Nurse

Mountmellick Primary Care Centre Telephone: 057 9359894 Fax: 057 9359906

Mountmellick

County Laois

Re: Exploring the experiences of patients with primary and secondary lower limb lymphoedema during the intensive and maintenance phases of Complex Decongestive Therapy and its impact on their lives

Dear Ms. Costello,

Thank you for your recent correspondence regarding a proposed amendment to your research study.

The Chairperson has reviewed your amendment and has provided a Favourable Opinion.

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Best wishes with your research.

Yours Sincerely,

______

Paul Marsden

Secretary – Research Ethics Committee

On behalf of

Dr. Una Fallon MCRN 014313

Chairperson – Research Ethics Committee

256

Appendix 17: Interview Sample

ID 13

Interview Details

PI: Interview date is the 21st of April, 2017. Interview start time is 11.10 a.m. Interviewer: Principal Investigator Mary Costello. Interviewee: Interview Identifier Number –“13”. Living with Lymphedema. When did you first notice that there was a problem with your legs?

ID 13: I first noticed in about 1987, when I was twenty-one. I thought I had sprained my ankle , with high-heel shoes, it wasn’t sore it was swollen so I thought I had twisted my ankle on the shoes, and then maybe a while later I’d notice, no it wasn’t that ankle it was the other ankle. And from then on my ankles would have been kind of swollen.

PI: So both ankles were involved?

ID 13: Yes, but it was never painful and it would go. First of all I thought it was one leg, then I thought it was the other. It just kind of swapped, and it would depend on how much attention I would put onto it, whether I noticed it or not. So wearing jeans and runners I didn’t notice it, but if I was wearing high heels and a short skirt I noticed it, but it never caused me any pain. And even then in 1992 I did a reflexology course over about six or nine months and I had people at my feet all the time, you know as they were practicing but it was very mild, very very mild.

PI: Was the swelling at that stage, something that would be gone in the morning but worse in the evening? 257

ID 13: Yes always down in the morning.

PI: Yes, but yet by the end of the day, particularly if you had a day where you were on your feet or standing or whatever……

ID 13: Yes, I mean in the beginning it mightn’t have been every day, it wouldn’t have been something that I would have been terribly conscious of, it only bugged me as a part of vanity, do you know, and nobody else would have noticed it probably.

PI: And, as you said, you were twenty one at this stage. So, at what stage did the swelling progress?

ID 13: And when I was doing the reflexology that was in 1992, like I know now there was no pitting, even when people were massaging my feet, whatever was there might move around, it wasn’t an issue. Then kind of as I got older then, It was in the summer of 1999 I think, I had gone to my Doctor and maybe over the previous year or so say 1998, or 1999 she put me on diuretics for a while.

PI: You were seeking medical advice from you own G.P. so was the swelling becoming a bigger problem?

ID 13: Yes it was, but mostly only from a vanity point of view. This wasn’t painful or anything like that.

PI: You said that you were started on diuretics, did your G.P give you a diagnosis?

ID 13: No. As far as I know they didn’t really know what it was and they weren’t particularly concerned about it as far as I can remember, but they did send me to a vascular consultant and I had a consultation with him and had an ultrasound and then he gave me a letter then to say I was diagnosed with Lymphedema.

PI : And what age were you at this stage?

258

ID 13: I was thirty in 1996, so I was thirty three.

PI: Okay, so you would have had ten, twelve years with slow progression shall we say, but you had a diagnosis at that stage.

ID 13: Yes.

PI: So you were diagnosed as you said, by a Vascular Consultant, and with the diagnosis were you given a recommendation for a particular treatment ?

ID 13: No, no. I was just told it was one of those things and I think at that time, I can’t rightly remember but I think that’s when I started wearing stockings, but I think I only went to a Chemist for them, do you know what I mean, they weren’t measured, they were very big.

PI: So there was no distinct treatment plan?

ID 13: No, no, no.

PI: And the next progression, where did it go from there?

ID 13: So that was 1999 and I had these compression socks that I bought in a Chemist. I got married in 2002 and like I was working as a physical therapist at this stage and I was on my feet a lot but I was in runners and tracksuit, and they would still be swollen but it still wasn’t a problem. Like it wasn’t that my feet would be tired the only time they would really be swollen was like if you were away on holidays, they could get quite puffy, but then lots of people’s do that. So therefore it wasn’t a problem. Then in 2005 I was pregnant, my first pregnancy, and I had been to the Doctor and when I decided to get pregnant I went with a list of questions to the Doctor because I was going to be thirty eight, thirty nine and I had this, you know and my mother had a history of varicose veins which she said always got worse on her, or really bad on her when she was pregnant, but then she was pregnant at twenty-two, do you know what I mean. So really my big concern around the pregnancy was that I wouldn’t get varicose veins. So I was very good, I wore the socks the whole way through the pregnancy whereas up to that point I had only been wearing them on and off if it suited me, and that was fine. I didn’t really start noticing them much anymore after that and then I had my second pregnancy which was in 2007, and that was fine I wore them again after that and then after that I really stopped wearing them because I was in my tracksuit and mummy runners and stuff and I

259 stopped wearing them. I imagine or I think around that stage the swelling, like I think it would still be down in the morning. I would go through phases of having my pillows under the mattress over those numbers of years, and I would have the bed elevated and they would always be lower in the morning. I think it was around that time maybe that I started noticing the pitting, that if you put your finger into it, that it would stay, but I could be imaging that too, but then the big thing was in February 2010 I got Cellulitis.

PI: Your first bout of Cellulitis?

ID 13: Yes. So I woke up one morning feeling terrible, had a temperature, feeling sick, I rang a friend of mine to take the kids for me and rang my mother to come down, I thought I had food-poisoning or something and she was like, “there is something wrong here”. We went to the Doctor and he couldn’t figure out what it was.

PI : And your leg, were there signs of cellulitis?

ID 13: No. There was nothing on my leg at that time but I was feeling terrible. So they kind of said go to bed, take paracetamol and drink glasses of water and the following morning I was still feeling fairly rotten and when I got out of the shower, I noticed I had a red foot up as far as my ankle, and as soon as I told my mother that she was like “get to the Doctor that is something in your blood”, she said, and when I got to the Doctor then, they just “oh up to the hospital with it”. So I went up to the hospital and I was there for, oh I don’t know, ten days with it.

PI: Did you have I.V antibiotics?

ID 13: Yeah I.V antibiotics and I remember we were kind of saying to one of the Doctors “well why did I get this” and he said “oh you’ll get it again, you’ve Lymphedema”. But that was the sole amount of care that I got.

PI : So once again, was there any follow on or recommendation for treatment?

ID 13: No, no follow on. So maybe I kind of went back to wearing the socks again and you know maybe putting pillows in under the bed, but that was all. There was never any problems with my feet, in that there was never any broken skin, or I didn’t have poor quality skin, so that didn’t come into my, the following year, I remember about August of the following year, I got a bite on my foot and

260

I went to the Doctor and I took an oral course of antibiotic but I know now that if I’d had Cellulitis that oral antibiotics wasn’t going to be doing an awful lot for me. That year, after I got it I had oral antibiotics and if we went on holidays I brought them with me. So that was the level of care I was taking.

PI : So your plan of care now included having antibiotics available should you see any signs of cellulitis?

ID 13: Yes, but I know now, that they wouldn’t have done anything really. So I was getting on fine and then ignored the socks again, and yeah they were getting puffy and even when I was wearing them they were getting more puffier, I think, but it was always focused around the ankle. So when I’d be buying shoes, I couldn’t buy shoes with straps or, anything like that.

PI : So, at this stage you were buying specific types of footwear?

ID 13: I had kind of really been doing that from about 1999 onwards, do you know. And then once of course, you wear boots you don’t notice it at all and that was all fine. And then in August 2nd last year, we went to the National Aquatic Centre with my two daughters on a busy summers day, lovely microbial pool we were there for about three and a half hours in this water, surrounded by all sorts of people going up and down slides, running around in my bare feet and kind of thinking “ah they don’t look too bad”, you know, but I was aware that I had a bit of hard skin behind my small toe and I was aware that there was a kind of a crack in that, but I just ignored it entirely. And the following morning oh god, that night I woke up feeling rotten, and I was frozen and I was roasting, trying to burrow in under my husband, and I rang the GP oncall service and I was kind of in denial, I was kind of like could this be cellulitis, or maybe it’s just kind of a gastro-flu, gastro-virus and I still denied it and the following morning again I got out of bed and came down and by two o’clock my mother said “no go to the Doctor, you’re still not right”. And I kind of went to the Doctor saying “no, there’s nothing on my foot”, but no by the time I made the appointment with the Doctor and by the time I got the Doctor’s appointment and was actually in the Doctor’s office, the foot was actually starting to heat. So there was no discussion then, you know, straight into hospital. The doctor asked if I had private health insurance, which I did, so the doctor sent me to a private hospital A& E.. I came home got into the car and my father drove me up to the hospital and they started giving me the antibiotic at that time and they had to change my antibiotic over the next few days before we actually got a handle on it.

PI : So was the cellulitis quite severe?

261

ID 13: Yes, they were pumping the stuff into me and I was fairly wishy-washy for the first number of days. And it was always on the left leg, the cellulitis the first time was on the left leg. The first time the foot really swelled and the all the skin kind of broke outside it. It didn’t seem to be as bad this time. I was under an infection control person, I have his report there, I’ll give you that too, and I think it finally kind of dawned on them that it was the Lymphedema that was the problem. And then he got a vascular consultant and she came in and she talked to me for a long time about it (lymphoedema) and that was the first time it really hit home, even though I had known an awful lot of it but for her to actually say “yes it is primary Lymphedema” and that she deals with “lots of people with it”, I had never met anybody who had had it before and that it wasn’t just as a consequence of cancer and kind of what I needed to be doing and I was shown that there was a stronger level of sock and that I needed the manual lymphatic drainage and all the skincare and all of that.

PI : So what was the next step?

ID 13 : The Lymphedema kind of went half way up the calf and there was bit of broken skin in behind the calf muscle here really kind of where I had been lying on it and she had me come back to Out-Patients for two, or three times and she actually sent me home with it, with kind of this bandage on it that had a kind of a putty like substance on it, so there was kind of this soft squidgy stuff on a bandage and then that was wrapped around and I had that replaced, twice or three times to heal the skin, and it was really then on that I was given the name of a therapist to contact, because I was kind of thinking I wanted to be under somebody or do I go back to the consultant, do you know.

PI: And that was probably the next part of the puzzle, now you know what you have to do but where do you find it, isn’t it?

ID 13: Yes, well one, at least I had the name of a consultant who knew how to deal with my condition and the other thing was like when we took off the bandage, my skin was gone brown on my leg, because this was like a physical manifestation, what was after happening.

PI: So you could actually see……

ID 13: Yes there was a stain on my leg and kind of, she said “the veins could be damaged or the lymph vessels could be damaged”, so it was the first time I realised, you know if I get this again, this isn’t just a matter of just going to hospital for a week and just being pumped full of antibiotics and going home again. You know, this is going to, like if it happens again it could really make the lymph vessels more damaged, so that then the damage becomes more and

262 more, so you know the damage is building up on it. So that’s kind of what I wanted to stop, but finding the information was tricky, because this consultant I knew she treated primary Lymphedema, but how I was going to get into her system, I didn’t know.

PI: You said that you had been given the name of a therapist, did you contact her yourself?

ID 13: Yes, she was a private therapist, I couldn’t find anyone doing manual lymphatic drainage in the health service. It was quite expensive but I knew that I had to do this. Up until then I was wearing light socks that the chemist recommended., they were only like flight stockings and I was thrilled with these new stockings because they were so light and so easy. And it was only really when I saw the therapist and she said “oh look at your poor toes” and that was the first time that I really noticed my swollen toes, I was always fixated on the ankles.

PI: In what way would you say that Lymphedema has affected your everyday life, physically has it affected you?

ID 13: No, I’m actually quite fit.

PI: Would you think from a social, psychological perspective has it affected you in any way?

ID 13: Well not really, well no, really in the beginning as I said it was really only down to vanity really. It has affected my choice in shoe wear, do you know.

PI: Your level of oedema is quite mild, more so affecting the foot and the toes, so as you said it hadn’t got to a stage where it was visible to others.

ID 13: No not particularly. My family would notice but only because I was going on about it so much.

EXPERIENCE OF TREATMENT

263

PI: You have been through the manual lymphatic drainage now, your experience of it, is it positive or negative or what would you have to say about manual lymphatic drainage?

ID 13: Well the first time I went through it with a private therapist as a part of an overall compression treatment, so I suppose on that, I was more focused on the compression treatment which was uncomfortable but we got through it. It was time-consuming and uncomfortable but you could see that it was doing something. So it needed the manual lymphatic drainage to get that done. The manual lymphatic drainage was fine. When I was coming down here and started the treatment plan and I had gotten my manual lymphatic drainage it was kind of a more long term thing, you wouldn’t see or at that stage you wouldn’t see a noticable difference from one day to the next but it was the more long term for just keeping a level.

PI :Any negatives about it, anything that you didn’t like about manual lymphatic drainagel?

ID 13: No.

PI: Compression bandaging? Tell me about your thoughts, both positive and negative.

ID 13: Compression bandaging was challenging. I did find that it was uncomfortable but in the greater scheme of things, when you saw that they were working you got on with it. Oh absolutely, and you could see that there was a difference, like the first time you could always be seeing ankle bones and the other thing was like, when I took it off my husband kind of looked at me and said, I never noticed that vein running down your leg. And he probably never did and there are now little bubbly veins running down the front of my shins, which is probably a varicose vein and also the fact that I’m fifty and all of that kind of thing, but they were probably never noticeable before because there was kind of a swelling on it. So that kind of just shows what kind of swelling I had.

PI : And say now, the compression hosiery that you are in, you wear knee-high socks with the toe-caps. How do you find them to wear?

ID 13: The knee-high socks are fine, I manage them no problem. My only problem was with the toe-caps and the discomfort that was with them was

264 erratic, the same pair of shoes could be comfortable one day and not comfortable the next and it was just focused in around the toes and there was no discomfort up around the leg, or the ankle or anything like that. It was just in around the toes. And some days then, like the relief of taking of those toe-caps was just, like some days they were just excruciating. I know really I suppose how important the socks were and like this is the first time I had toe-caps aswell, so even what I was doing with socks was fine, but it wasn’t helping, like the Lymphedema was still staying down on the front of the foot. Both times that I got cellulitis, they told me that I had Athletes Foot between my toes, but I wasn’t aware of that, so it must of only been a very small amount of it, and that’s all it took. I suppose the fact that the toes, like I never particularly noticed it that the toes were swollen, but like if the toes were swollen, there was no air getting in between the toes. The first time the bandages were taken off the toes after the first compression thing, I’m looking at these skinny toes, and I’m gobsmacked because I know I haven’t seen spaces between my toes in an awfully long time.

PI : Now that you know that this is a lifelong pursuit, does that bother you or is that a challenge for you in some way?

ID 13: I think maybe it might have bothered me twenty years ago, now it doesn’t. It really doesn’t like I went through my summer wardrobe last week and I took out the shorts and the shorts down to the knees, the lovely white ones I had, and I gave them all to my sister-in-law. It’s not as if anybody else would notice, and at this stage I now have bubbly legs anyway so I wasn’t going to be sticking those legs out under a mini skirt at this age anyway.

PI : Do you think then that the age at which you are diagnosed and treated, would have challenges?

ID 13 : Oh yes, like I mean if I was thirty and kind of conscious of bubbly legs under a bikini or something, but then, people have other body issues and to everybody else they can look perfect, do you know.

PI : Do you exercise?

ID 13 : Exercise, I don’t do enough of it really but I do enough running around, my big thing is that I am keeping my weight in order and that’s through slimming world.

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PI : Do you think that weight has an impact on Lymphedema?

ID 13: Yeah and my big thing was kind of turning fifty, like heading into the menopause and all of that and just try and keep the weight off the legs, and certainly now that I am slimmer, now not that I was a whole lot heavier, I think it’s easier on my legs. But I would have an understanding like from physical therapy like for your knee joints and your hip joints and everything, like weight just puts so much stress on it. I’d go for walks, and take notions every now and again doing exercise classes, like I’ve done them all.

PI : Your skincare, what is your routine?

ID 13 : I keep the legs, I moisturise every night with Aveeno moisturiser and then I started using a paraffin gel on my feet at night-time because I don’t want to be putting it on the socks, so I’d put on a good old lash of the paraffin stuff on my feet and put on pair of soft socks and get into bed, every second or third day, but I’d be very cautious of. If I got a cut, but I haven’t had any. but I would just watch it, put antiseptic cream on it but the fact that that both times I had an overwhelming reaction like, I went from being fine to waking up with a roaring temperature within twelve hours, I had no warning signs of the Lymphedema or anything like that, it was literally that I woke up feeling, or how I denied the symptoms the second time around, I don’t know.

PI : Now moving forwards, do you have any fears of concerns about maintaining your legs independently?

ID 13 : No. What I’ll do now is I’ll make sure that I get the right socks., I mean like I have to get replacements for them in another while because they are only supposed to be worn for the six months or whatever, but you know I’ll have two pairs or whatever. I think I will schedule more manual lymphatic drainage for myself as well. I suppose compared to this time last year, I would be very conscious of my weight and I would be very confident that my diet is very good. I have an old break out every now and again, but that’s life, but you know I am confident that my diet is good and that’s the first time where I know this. I suppose I am only looking to mind it, on the premise of also having a healthy lifestyle. I started looking up websites and stuff and I could read about lymphoedema, but if I had of read about that, let’s say, without you telling me about it, I would have assumed that, that was for someone else that was a lot worse off than me because you have no reference points on these websites as to who they are talking to, and then getting the right compression stockings, the ones I had been

266 wearing before were useless but you presume that the pharmacist knows best but now I know otherwise. PI : Do you think that having the knowledge about your condition is important?

ID 13: Yeah if you don’t know, and like no website is going to give you what you need to know. I’d have just gone on like I was before waiting for the next one to happen because even I have a great G.P and that, but like she doesn’t know any more about it. I know more about lymphoedema than what she does. I thought it was a very rare thing, I assumed primary Lymphedema was kind of a small little area and most people dealt with secondary Lymphedema and also the insurance companies think the same because I couldn’t get my manual lymphatic drainage paid for because I didn’t have an Oncologist’s report, do you know, so that’s something else.

PI : Very good, look it I want to say just thanks for doing the interview and we are going to conclude at about 12 o’clock now.

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Appendix 18: Searching for Themes

KNOWLEDGE DEFICIT

IMPACT

THERAPEUTIC

INEQUALITY OF NO PRIVATE SHAME HOPE SERVICES INFORMATION TREATMENT

POOR FRUSTRATION FINANCIAL BODY IMAGE UNDERSTANDING KNOWLEDGE STRUGGLE

NOT HOPELESS EXPENSIVE ISOLATION POSITIVE IMPORTANT CHANGES

PUT UP LOST LOSS OF NO CONSISTENCY EARNINGS CONSISTENCY

GET ON STRUGGLE MAKE DO DIURETICS KNOWLEDGE

TIRED HEAVY EMPOWERED EDUCATION DIRECTION

POWERLESS BLAME LOOSE WEIGHT NO DIAGNOSIS ADVOCACY

NOTHING CAN NO THERAPISTS FAMILY COPING SECURITY BE DONE IN HSE AFFECTED

NO DIAGNOSIS NO ONE KNEW FEAR NO SUPPORT DIAGNOSIS

268

Appendix 19: Diary

DATE SOCKS SOCKS TOTAL ACTIVITY DURATION OF ON OFF HOURS ACTIVITY AM PM WORN

06-Oct- Irish 17 12 17 -5 Bike 5 mins socks Removed right sock uncomfortable 07-Oct- Irish 17 12 16 -4 Walked 10 mins socks Removed right sock uncomfortable 08-Oct- 17 12 23 -11 Walked 10 mins German socks 09-Oct- Right Leg Only German Left leg too sore to do 17 13 23 -10 Sock exercise 10-Oct- Right Leg Only German Left leg too sore to do 17 11 23 -12 Sock exercise 11-Oct- Right Leg Only German 17 0 0 0 Walked 5 mins Sock 12-Oct- 17 0 Washed Sock 13-Oct- Right Leg Only German 17 9 23 -14 Walked 5 mins Sock 14-Oct- Right Leg Only German 17 12 22 -10 Walked 5 mins Sock 15-Oct- 17 0 0 0 Washed Sock 16-Oct- Right Leg Only German 17 12 20 -8 Bike 5mins Sock 269

DATE SOCKS SOCKS TOTAL ACTIVITY DURATION OF ON OFF HOURS ACTIVITY AM PM WORN 17-Oct- Right Leg Only German 17 9 23 -14 Shopping Sock 18-Oct- Right Leg Only German 17 9 20 -11 Walked 10mins Sock 19-Oct- 17 0 0 0 Washed Sock 20-Oct- Right Leg Only German 17 9 24 -15 Bike 5mins Sock 21-Oct- Right Leg Only German 17 12 22 -10 Walked 10mins Sock 22-Oct- 17 0 0 0 Washed Sock 23-Oct- Right Leg Only German 17 12 23 -11 Bike 5mins Sock 24-Oct- Right Leg Only German 17 11 22 -11 Shopping Sock 25-Oct- 17 0 0 0 Washed Sock 26-Oct- Right Leg Only German 17 9 22 -13 bike 5mins Sock 27-Oct- Right Leg Only German 17 11 22 -11 Housework Sock 28-Oct- Right Leg Only German 17 12 22 -10 Walked 10mins Sock 29-Oct- 17 0 0 0 Washed Sock 30-Oct- Right Leg Only German 17 12 22 -10 Housework Sock 270

DATE SOCKS SOCKS TOTAL ACTIVITY DURATION OF ON OFF HOURS ACTIVITY AM PM WORN 31-Oct- Both 17 9 23 -14 Shopping Legs German Socks 01-Nov- Both 17 10 22 -12 Walked 10 mins Legs German Socks 02-Nov- Both 17 11 23 -12 Walked 10 mins Legs German Socks 03-Nov- 17 0 0 0 Washed Sock 04-Nov- Both 17 10 23 -13 shopping Legs German Socks 05-Nov- Both 17 11 22 -11 Walked 10mins Legs German Socks 06-Nov- 17 0 0 0 Washed Sock 07-Nov- Both 17 10 22 -12 Bike 5mins Legs German Socks 08-Nov- Both 17 0 0 0 Bike 5mins Legs German Socks 09-Nov- 17 0 0 0 Washed Sock 10-Nov- Both 17 11 22 -11 Walked 10 mins Legs German Socks 11-Nov- Both 17 9 22 -13 Shopping Legs German Socks TOTAL -288

271