Life with osteoporosis œ a cohort and qualitative study

PhD Thesis

Life with osteoporosis � a cohort and qualitative study

Carrinna Hansen

Research Unit of Nursing

Institute of Clinical Research

Faculty of Health Sciences

University of Southern Denmark

2014

PhD thesis

Life with Osteoporosis - a cohort and qualitative study

Carrinna Hansen, PhD student, MPH, nurse

Print Ph.d.-afhandling fra Enheden for Sygeplejeforskning, Klinisk Institut Det Sundhedsvidenskabelige fakultet Serietitel 2014 Rapport nr. 1 ISSN 2244-9302

This thesis has been accepted for defense of a PhD in nursing by the Faculty of Health Science, University of Southern Denmark and was defended 28th of January 2014.

Official opponents

Nina Emaus, Professor, Dr. Science, Department of Health and Care Sciences, Faculty of Health Sciences, UIT the Artic University of Norway, Tromsø

Erik Elgaard Sørensen, Research Manager, Associate Professor, PhD, Research Unit of Clinical Nursing, Aalborg University and Institute of Clinical Research, Faculty of Health Sciences, University of Aalborg

Niels Christian Hvidt, Associate Professor, Master of Theology, PhD, Health, Man and Society, institute of Public Health, Faculty of Health Sciences, University of Southern Denmark & Professor für Spiritual Care, LMU, München (chairman)

Supervisors

Birthe D. Pedersen, Associate Professor, PhD, Research Unit of Nursing, Institute of Clinical Research, Faculty of Health Sciences, University of Southern Denmark

Hanne Konradsen, Research Manager, PhD, Research Unit of Copenhagen University Hospital, Gentofte

Bo Abrahamsen, Professor, Department of Medicine F, Copenhagen University Hospital, Gentofte & Institute of Clinical Research, Faculty of Health Sciences, University of Southern Denmark

Research Unit of Nursing, Institute of Clinical Research, Faculty of Health Sciences, University of Southern Denmark & Department of Medicine C, Copenhagen University Hospital, Gentofte.

Financial support

University of Southern Denmark

Copenhagen University Hospital, Gentofte

The Capital Region of Denmark Research Foundation for Health Research

Aase and Ejnar Danielsens Foundation

Osteoporosis society

Cabinetmaker Sophus Jacobsen and wife Astrid Jacobsens Fond

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PREFACE

This thesis addresses issues related to life with osteoporosis. On the one hand, it deals with risk

groups based on refill compliance and persistence of anti-osteoporotic therapy among men and

women in Denmark; and on the other hand, lived experiences of newly-diagnosed women during

the first year after diagnosis. As part of the first objective, a register-based nationwide cohort study

of new users of oral anti-osteoporotic therapy was conducted to determine the distribution and

determinants of compliance and persistence to oral anti-osteoporotic treatment in order to gain

further understanding of the phenomenon of low compliance and persistence, a major and an on-

going worldwide challenge. As part of the second objective, a qualitative interview study was

conducted to investigate the �lived experience� of osteoporosis. Living with a chronic condition

often affects the entire life of an individual and their behaviour, emotionally and existentially; every

person experiences this in a way unique to him or herself. �Lived experiences� of osteoporosis are

important as perception, interpretation, acceptance and the development of adaptation to living

with osteoporosis may be closely related to the phenomena of compliance and persistence to anti-

osteoporotic therapy. The two study methods used in the thesis provide an extended

understanding of factors affecting life with osteoporosis.

The inspiration for the study was based on a few lines found in the Framework for Nursing

Research "Patient participation", a 2008 program of research and research training at Gentofte

Hospital. Within this framework, it was suggested that potential research in the nursing field could

be: �Osteoporosis Patient compliance and non-compliance in relation to the fracture preventive

treatment - Reasons for discontinuation of treatment� (Gentofte Hospital, 2008) pp. 8. Osteoporosis

was in keeping with my nursing experiences with musculoskeletal disorders, elderly patients as

well as health promotion and prevention. I have always given priorities to the patient's own

experience of illness, as I believe that patient involvement is important. Within these few lines of

the Framework for Nursing Research I saw an opportunity to immerse in an individual's experience

of having to live with a chronic condition like osteoporosis, as well as the opportunity to investigate

these questions in the Danish population.

Gentofte Hospital, 2008. Framework for Nursing Research 'Patient participation' - A program of

research training 2008. http://www.gentoftehospital.dk/NR/rdonlyres/513BF53A-4964-400A-9DC4-

05617B87A171/0/0811_rammeprogram_i_nyt_layout.pdf.

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ACKNOWLEDGEMENTS

The thesis was carried out at the Research Unit of Nursing, Institute of Clinical Research, Faculty

of Health Sciences, University of Southern Denmark and Department of Medicine C, Copenhagen

University Hospital, Gentofte, The Capital Region of Denmark. I wish to express my sincere

gratitude to everyone who made it possible to conduct the study and the project. In particular I wish

to thank:

University of Southern Denmark: thank you for assigning me one-year grants and for an

inspiring study environment at the Research Unit of Nursing

Copenhagen University Hospital, Gentofte for establishing a wage agreement with me and

providing an inspiring working environment at the Research Unit

The participating women from the Capital Region of Denmark and from the Region of

Zealand; Thank you for participating in the study

Department of Nuclear Medicine, Copenhagen University Hospital, Gentofte, in particular

Bente Krogsgaard Schaadt, Thora Buhl, the secretary and all the scanning staff for being

very helpful during patient recruitment

Department of Medicine, Koege Hospital, the Region of Zealand and in particular Lisbeth

Kjærgård Hjort Medical Laboratory Technologist radiography at Koege Hospital, for being

very helpful and thorough with patient recruitment

My supervisors:

Birthe D. Pedersen: Thank you for: believing in me and supporting me all the way through;

for sharing broad scientific knowledge within the field of qualitative research,

phenomenological-hermeneutic research and the interpretation theory guided by Ricoeur

as well as many opportunities for networking; for giving me a thorough and rapid

response/feedback whenever I needed it; for being patient and sharing your knowledge on

analyzing qualitative data; for providing me with very useful feedback concerning the

papers and the thesis. Last but not least, thank you for your pleasantness and warmth.

Hanne Konradsen: Thank you for being very helpful and focused in the initial phase of my

PhD study � without you, I do not think that I would have gotten started on my PhD project;

thank you for help with fundraising and many practical issues; for being an interesting

sparring partner and for sharing your knowledge and experiences, in particular for sharing

your great knowledge of longitudinal studies.

Bo Abrahamsen: Thank you for: 'sowing the first seeds "of ideas for the project; for giving

me a thorough and rapid response/feedback whenever I needed it; for being patient and

sharing your knowledge of analyzing data in a register based study; for providing me with

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very useful feedback concerning the statistical analyses, the papers and the thesis; for

sharing broad scientific knowledge within the field of osteoporosis and register based

studies and many networking opportunities from the very first beginning.

Tove Lindhardt: Thank you for believing in me and supporting me from the very first

beginning before and when I was a PhD sprout; thank you for paving the way for a great

PhD study environment when you were at Gentofte Hospital

Torben Laurén: Thank you for being advocating nursing research and thank you for making

the economic and employment path for a PhD possible

Transcription support: Janni Mathiasen, Kirsten Rye, Jakob Konradsen, Christina Louise

Hagen Olsen and Lena Boye. Thank you for carefully transcription of the interviews.

Fellow students in the Research Unit of Nursing, University of Southern Denmark, for

sharing rewarding discussions and fun over the years

Fellow students in the Research Unit, Copenhagen University Hospital, Gentofte in

particular: Connie Berthelsen, Lone Skov Jensen and Dorthe Brask-Lindemann. Thank you

for sharing fun, frustrations and rewarding professional discussions with and joint

reflections on both specific and metaphorical levels over the years

Fellow students at the European Academy Nursing Sciences for sharing experiences,

rewarding discussions and fun during the three years of EANS Summer school and in our

group on Facebook. In particular Eva Sving my friend and soulmate

Rebeka Sujic, St. Michael's Hospital, Toronto, Canada: Thank you for your great work on

proofreading paper II and the thesis; for being an inspirational colleague overseas

Lone Falck, secretary at the Research Unit, Gentofte Hospital: Thank you for your ever

present helpfulness. You have repeatedly over the years almost "saved" me

My family, in particular the two stars of my life Johannes and Linnea my two children who

have been very understanding and supportive all the way through despite my periodic

absences. Thank you to their father Ulf Olofsson � I am very grateful for all you help and

support and taking care of our children

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ABBREVIATIONS

BMD Bone mineral density.

CPR Civic Person Register

DDD Defined daily dose (a statistical measure of consumption, defined by the World

Health Organization: "The DDD is the assumed average per day for a drug used for

its main indication in adults�)

DXA Dual-energy X-ray absorptiometry (previously DEXA); is a means of measuring BMD.

GP General Practitioner.

MPR Medication Possession Ratio.

RCT Randomized controlled trial

SOC Sense of Coherence

STROBE Strengthening the Reporting of Observational Studies in Epidemiology

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DEFINITIONS

Osteoporosis is a systemic skeletal disease characterized by low bone density and micro

architectural deterioration of bone tissue. Osteoporosis is defined in Caucasian women as a bone

mineral density (BMD) that lies 2.5 standard deviations or more below the average for the young

healthy female population(T-score <-2.5 S.D.).

Persistence may in a medical context be defined as the accumulation of time from treatment

initiation to discontinuation of therapy (i.e. in time on treatment). In this study, the term is

operationalized as the number of days on treatment.

Compliance refers to the extent to which the patient acts in accordance with the prescribed

treatment regimen. In this study, compliance is interpreted according to filled prescriptions. Ideally

the term should include aspects such as if the drug was taken correctly (e.g., after fasting

overnight), at the correct time, whether large doses were taken to compensate for forgotten doses,

et cetera. Due to the nature of historical data, the only part of compliance that may be measured in

retrospective studies is unplanned drug holidays (i.e. when the patient does not have medication

available). In this study, compliance is quantified as medication possession ratio (MPR). A MPR

value of �75% was predefined as acceptable refill compliance; measured only during the period of

time patients were on treatment.

In this study, compliance is not understood in a positivistic perspective. The term has been chosen

since it is frequently used in the field of osteoporosis research.

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LIST OF PAPERS

The present thesis is based on the following papers, referred to in the text by their Roman

numerals:

Paper I Anti-osteoporotic therapy in Denmark � Predictors and demographics of poor

refill compliance and poor persistence

Hansen C, Pedersen BD, Konradsen H, Abrahamsen B

Osteoporosis International 2013; 24 (7), 2079-2097.

DOI: 10.1007/s00198-012-221-5

Paper II Women�s experiences of their osteoporosis diagnosis at the time of

diagnosis and six months later: a Phenomenological Hermeneutic Study

Hansen C, Konradsen H, Abrahamsen B, Pedersen BD

International journal of Qualitative Studies on Health and Wellbeing, submitted

Paper III Women�s Lived Experiences of the first year learning to live with diagnosed

Osteoporosis: A Longitudinal Qualitative study

Hansen C, Abrahamsen B, Konradsen H, Pedersen BD

Manuscript

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LIST OF TABLES AND FIGURS

Table 1 Example of data grouping

Table 2 Example of converting variable to binary exposure variables

Table 3 Overview: Self-reported socio-demographics of the informants

Table 4 An example of the structural analysis and themes

Table 5 Overview of selected results from three different analysis showing only

significant predictors

Table 6 An overview of key themes and sub themes

Figure 1 Analytic levels in the interpretation of findings

Figure 2 The interview path

Figure 3 Flowchart of New users of anti-osteoporosis drugs 1996-2006

Figure 4 Flowchart of inclusion of informants; and interview rounds

Figure 5 Model of study results within aspects of medical treatment and chronic condition

PhD�thesis�by�Carrinna�Hansen�

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Contents

Financial support ii

Preface iii

Acknowledgements iv

Abbreviations v

Definitions vi

List of papers vii

List of Tables and Figures viii

1. INTRODUCTION 3

1.1. Osteoporosis definition and diagnosis 3

1.2. Epidemiology and fracture 4

1.3. Compliance and persistence 5

1.4. Living with osteoporosis 6

2. AIMS AND OBJECTIVES 9

3. THE METHODOLOGICAL FRAME 11

3.1. Epidemiological method 11

3.1.1. Register-based study 12

3.1.2. Statistics Denmark 13

3.2. Phenomenological-hermeneutics 14

3.2.1. Narrative 15

3.2.2. Interpretation 16

4. METHODS AND MATERIALS 19

4.1. Epidemiological method 19

4.1.1. Data � study A 19

4.1.2. Statistical analysis 22

4.2. Phenomenological-hermeneutic study 24


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4.2.1. Data � study B 24

4.2.2. Analysis � Interpretation 26

5. ETHICAL CONSIDERATIONS 29

6. RESULTS 31

6.1. Paper I � Study A 31

6.2. Paper II and III � Study B 35

6.3. Model comprising the overall study results 40

7. DISCUSSION 43

7.1. Discussion of the results 43

7.2. Methodological considerations 55

7.2.1. Methodological considerations of the quantitative study A 56

7.2.2. Methodological considerations of the qualitative study B 58

8. CONCLUSION 61

9. PERSPECTIVES AND IMPLICATIONS 63

10. SUMMARY 65

11. RESUMÉ PÅ DANSK (SUMMARY IN DANISH) 67

12. REFERENCES 69

13. APPENDIX

14. PAPER I � II � III


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1. INTRODUCTION

Osteoporosis is an increasing major public health problem (Cadarette and Burden, 2010;

Genant et al., 1999; Kanis et al., 2013) affecting hundreds of millions of people worldwide

(Reginster and Burlet, 2006). Osteoporosis is underdiagnosed and undertreated (Kanis et

al., 2013; Reginster and Burlet, 2006; Vestergaard et al., 2005). In light of this, there is an

increased need for early detection, treatment and osteoporosis education to prevent

deterioration and disability (Reginster and Burlet, 2006; Vestergaard et al., 2005), improve

the prognosis (Cooper and Harvey, 2012) and quality of life (Cockerill et al., 2004; Weston

et al., 2011) as well as to prevent premature death (Abrahamsen et al., 2009; Cooper et

al., 1993; Kanis et al., 2013). Osteoporosis is characterized as a chronic condition that can

be treatable in otherwise healthy individuals but it can also be severely debilitating if left

untreated (Cooper et al., 2009; Genant et al., 1999).

1.1. Osteoporosis definition and diagnosis

The definition of osteoporosis for post-menopausal women as used today was proposed in

1944 by a Task Force under the World Health Organization (WHO) (Kanis et al., 1994):

�Severe osteoporosis (established osteoporosis). A bone mineral density (BMD) that is

more than 2.5 standard deviation (SD) below the young adult mean in the presence of one

or more fragility fractures� pp. 6 (World Health Organization, 1994).

The diagnosis is made on the basis of a Dual-energy X-ray absorptiometry (DXA) scan

and a quantitative assessment of the BMD which is a determinant of bone strength. BMD

is measured as a T-score in lumbar spine or hip. The meaning of low BMD may be clearer

to understand when comparing osteoporosis with other chronic conditions as for example

hypertension - which is diagnosed on the basis of blood pressure - may lead to stroke but

not in every patient. Likewise, low BMD can be a risk factor for fractures but does not

always lead to fractures. The risk of fracture is highly modified by other factors such as

age, heredity, BMI, weight-bearing exercise and lifestyle (Kanis et al., 2013; World Health

Organization, 2003b) which need to be taken into account. The most common clinical

consequences of osteoporosis are compression fractures of the spine, fracture at the hip,

distal forearm and proximal humerus.


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1.2. Epidemiology and fracture

According to the WHO, the prevalence of female osteoporosis in the five largest countries

in Europe (France, Germany, Italy, Spaine and UK) is high, with 21% of women aged 50-

84 years (representing more than 12 million) being affected in these countries (Kanis et al.,

2013). In Denmark, there are no accurate figures of how many individuals are affected by

osteoporosis, partly because the disease is often asymptomatic in the early stages and

because there are no systematic screening initiatives (Abrahamsen and Vestergaard,

2010; Genant et al., 1999; Kanis and Gluer, 2000). In 1995, Vestergaard et al estimated

that 40.8% of women and 17.7% of men over 50 years of age have osteoporosis in

Denmark (Vestergaard et al., 2005). The number of men and women with osteoporosis

has increased in the last decades. This increase may on one hand be due to increased

attention towards the condition and fracture risk. On the other hand, the increased

prevalence may be affected by the increase in aging population (Statistics Denmark, 2008;

World Health Organization, 2003b).

In 2000, Europe was estimated to contribute with approximately 34.8% of osteoporotic

fractures in the world among men and women � 50 years of age. 620,000 new fractures at

the hip, 574,000 at the forearm, 250,000 at the proximal humerus and 620,000 clinical

spine osteoporotic induced fractures were registered (Kanis et al., 2013). In comparison, in

1999, the annual number of fractures potentially attributable to osteoporosis in Danish

patients � 50 years of age (40.8% of women and 17.7% of men) was estimated to include

approximately 10,000 hip fractures, 7,000 distal forearm, and more than 2000

compression fractures, fragility fractures is to be added in this calculation (Vestergaard et

al., 2005). The consequences of osteoporotic fractures may include hospitalization, need

for rehabilitation, lost earnings, early retirement, chronic pain, lost mobility and impaired

quality of life (Christensen et al., 2005; Genant et al., 1999), as well as high morbidity and

mortality (Abrahamsen et al., 2009). In 2010, 43,000 deaths were estimated to be causally

related to osteoporotic fractures in the European Union with osteoporosis accounting for

more disability and life years lost than rheumatoid arthritis (Kanis et al., 2013). Death may

happen within the first year in 20-30 % of the cases after hip fracture (Abrahamsen et al.,

2009) and most deaths occur during the first 3-6 months (Kanis et al., 2013). Studies have

demonstrated that anti-osteoporotic treatment in 3-5 years prevents osteoporotic fractures

in approximately 50% of the cases (Abrahamsen and Vestergaard, 2010; Roerholt et al.,


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2009; Siris et al., 2006) and the available evidence indicates that prevention of

deterioration and improvement of prognosis by early detection and treatment is important

(Abrahamsen et al., 2009; Cooper and Harvey, 2012; Landfeldt et al., 2012). Barriers to

early detection and treatment are numerous. Research has shown that many patients find

it difficult to perceive and interpret the diagnosis, their current risk as well as managing

everyday life with osteoporosis (Besser et al., 2012; Nielsen et al., 2012; Sale et al., 2011;

Weston et al., 2011; Hooven et al., 2009). One reason may be that some patients are

diagnosed before they experience osteoporotic fractures; however, most individuals with

osteoporosis are unaware of the disease until bone fracture occurs, as osteoporosis is

usually an asymptomatic condition (which is why osteoporosis is often referred to as �a

silent disease�) (Reventlow et al., 2006; Weston et al., 2011; de Souza et al., 2010).

Because of the asymptomatic nature of osteoporosis, there is an increased need for health

care professionals to be able to communicate fracture risk in an easy, understandable and

meaningful way.

1.3. Compliance and persistence

For the purpose of this thesis, literature was searched regarding compliance and

persistence of anti-osteoporotic treatment: literature published between 2000 to October

2012 was searched using the databases CINAHL, Cochrane Library, EMBASE, ERIC and

PubMed (which includes citations from Medline). Osteoporosis was used as the search

term combined with the following terms: adherence, compliance, drug/medical therapy and

persistence. Inclusion criteria were: studies reporting non-institutionalised patients

adherence, compliance or persistence to anti-osteoporotic treatment compricing weekly

oral bisphosponate. The search was limited to �full text� and English. In total, 20 papers

complimented with The WHO report �Adherence to Long-Term Therapies, Evidence for

Action� and �The Global Longitudinal Study of Osteoporosis in Women (GLOW): Rationale

and study design� were included.

Low compliance and persistence towards medical therapy of chronic diseases are and

have been a widespread challenge for decades (Dimatteo, 2004b; World Health

Organization, 2003a). This is also the case in relation to osteoporosis as shown in several

studies (Abrahamsen et al., 2009; Cooper and Harvey, 2012; Landfeldt et al., 2012;

Roerholt et al., 2009; Papaioannou et al., 2007; Seeman et al., 2007). These studies,


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among others, have shown that compliance and persistence with weekly or daily

bisphosphonate therapy are suboptimal and that this may impact fracture risk.

Bisphosphonate therapy on a weekly or daily basis is the most widely used treatment for

osteoporosis worldwide. Systematic reviews have reported that approximately 50% of

patients or more stop treatment during the first year (Imaz et al., 2010; Kothawala et al.,

2007). However, the findings depend somewhat on the study�s methodology. Thus, the

length of treatment pauses (or �grace periods�) permitted by the analysis influences

findings as some patients who stop bisphosphonate therapy have been found to reinitiate

treatment even after extended gaps (drug �holidays�) (Cadarette and Burden, 2010). This

is in line with register-based studies in Denmark, Sweden and USA which have all

reported non-compliance. A Danish population study of 152,777 male and female patients

with recent fracture showed that few patients began osteoporosis treatment, and that one

in four patients stopped therapy within the first year (Roerholt et al., 2009); A Swedish

population study of 56,586 treatment-naïve male and female patients showed that half of

the patients stopped treatment within the first year (Landfeldt et al., 2012); while an

American population study of 35,537 women reported a refill compliance of approximately

50% (Siris et al., 2006).

We know that patients compliance commonly stabilizes about six months after treatment

prescription, and patients tend to increase compliance just before and a short time after

consultation with the physician (Lindberg et al., 2008; World Health Organization, 2003a).

We also know that there may be many different explanations for non-compliance such as

side-effects or potential treatment imprecision (Dimatteo, 2004b), but the determinants of

low compliance and persistence to treatment are still not well understood (Landfeldt et al.,

2012; Silverman and Gold, 2010). Studies have been carried out in order to gain more

insight into compliance and persistence with medication for osteoporosis (Block et al.,

2008; Cadarette and Burden, 2010; Cortet and Benichou, 2006; McLeod and Johnson,

2011). These studies suggest that personal networks, psychological factors and a

relationship with the physician are important factors (Balkrishnan, 1998; Barat et al., 2001;

Block et al., 2008; Dimatteo, 2004a; Martin et al., 2005; Zolnierek and Dimatteo, 2009) as

well as motivation and health beliefs (McLeod and Johnson, 2011). Patients�

understanding of the effect of the medication (Cadarette and Burden, 2010) may also be

important key factors in relation to compliance and persistence towards anti-osteoporotic

therapy. One systematic review on this topic was found. This review assessed six


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randomized controlled trials (RCT�s) and one nonrandomized trial. These (six studies)

reported considerable efforts towards improving compliance and persistence of anti-

osteoporotic therapy, however, the systematic review showed that the effects of the

reported interventions were limited (Gleeson et al., 2009). This may strengthen the

assumption that the understanding of the phenomenon of low compliance to osteoporosis

therapy is still incomplete (Silverman and Gold, 2010). The �missing link� may possibly be

found in qualitative studies of patients� own descriptions of factors influencing their daily

experiences with osteoporosis treatment.

1.4. Living with osteoporosis

For the purpose of this thesis, literature was searched regarding �lived experience� of

osteoporosis: the literature published before February 2013 using CINAHL, ERIC,

EMBASE and PubMed databases. Osteoporosis was used as the search term combined

with the following terms: lived experience(s), life experiences, phenomenological,

qualitative studies/research. Inclusion criteria were: qualitative studies investigating

patients' experiences or thoughts related to osteoporosis; in an aspect of not only

predefined to investigate experiences related to calcium and Vitamin C, exercise, DXA

scan results ect. The search was limited to �full text�, language: English, German,

Norwegian, Swedish or Danish . In total 15 papers as well as �A systematic review of

osteoporosis health beliefs in adult men and women� were included.

It is well known that being diagnosed with osteoporosis may lead to psychological and

physical consequences for the individuals and it may impact quality of life (Nielsen et al.,

2012; Nielsen et al., 2011; Reventlow and Bang, 2006; Reventlow et al., 2006; Reventlow,

2007; Roberto and Reynolds, 2001; Solimeo et al., 2011; Weston et al., 2011; Wilkins,

2001b). In addition to this, patients may find it difficult to make sense of the diagnosis and

the implications for current and future health (Weston et al., 2011). These difficulties may

be associated with challenges of emotional handling of the knowledge of the everyday

fracture risk. Some patients were affected by this knowledge in a more negative way than

others (Nielsen et al., 2012). Moreover, elderly women�s thoughts about osteoporosis and

risk perception has been found to be influenced by stereotypes of bodily decay and

founded on worst-case scenarios (Reventlow and Bang, 2006). After DXA scan for

osteoporosis, has women�s self-awareness been found to be promoted by the information


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derived from the scan; the women appeared to interpret the result as body fragility and to

plan their life according to this with preventive actions, caution and new symptom

interpretations (Reventlow et al., 2006). There is a diversity of ways in which women with

osteoporosis perceive themselves and manage their aging and chronic illness (Wilkins,

2001a). A systematic review of 22 papers evaluating health behavior and health belief of

osteoporosis (McLeod and Johnson, 2011) has found that structural and psychological

determinants of health behavior need to be understood in order to better understand and

manage the disease. This thesis will therefore focus on further exploration of women�s

experiences of living with osteoporosis.


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2. AIMS AND OBJECTIVES

The aim of the study was to gain a deeper understanding of patients� life with osteoporosis

by investigating both the refill compliance and persistence of new users of oral anti-

osteoporotic therapy and the experiences of newly diagnosed women in terms of living

with osteoporosis after pharmaceutical osteoporosis treatment for fracture prevention had

been prescribed.

The objectives were:

1) To investigate epidemiological characteristics of men and women on anti-osteoporotic

therapy including treatment related, demographic and socioeconomic factors in Denmark.

Furthermore, to investigate compliance and persistence based on medication patterns of

refill compliance in order to assess whether it is possible to identify potential risk groups.

Hypothesis: patients who stop treatment very early differ in socioeconomic demographics,

co-medications and comorbid conditions from other patients with poor compliance. (Paper

I and thesis).

2) To investigate experiences of life with osteoporosis for newly diagnosed women; during

the first six months after diagnosis and preventive treatment was prescribed (Paper II and

thesis).

3) To investigate individual concerns and perceptions of women; during time when they

are adapting to life with osteoporosis and preventive treatment (Paper III and thesis).

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3. THE METHODOLOGICAL FRAME

This thesis examines life with osteoporosis and includes both a quantitative and a

qualitative study to provide a better understanding of this topic. First, we conducted an

epidemiological register-based study conducted in order to quantitate the magnitude of the

problem and investigate patterns and causes of refill compliance and persistence in a

group of new users of anti-osteoporotic treatment in Denmark. In general terms,

epidemiology is considered the cornerstone of public health because it can be used to

identify risk factors for disease and targets for preventive medicine which may inform the

basis of policy decisions and evidence-based medicine. Epidemiological observation

studies address the �who, what, where and when� of the occurrence of health-related

conditions (Saracci, 2010). Second, a qualitative method with a phenomenological-

hermeneutic approach and longitudinal design was chosen in order to be able to gain in-

depth knowledge of newly-diagnosed women�s lived experiences of osteoporosis. A

longitudinal design was chosen because it allows for investigation of the evolvement or

patterns of changes over time; and can potentially provide more complete information

about individual experiences (Saldaña, 2003). The approach was inspired by Paul

Ricoeur's theory of narrative and interpretation (Ricoeur, 1976). Qualitative research aims

to investigate an in-depth understanding of human experience and the �why� and �how� of

decision making (Sandelowski et al., 2006).

The two selected methods complemented each other as the issues related to life with

osteoporosis were investigated in a general, population-level and in a specific, individual

level. Both will be presented below and the results will be discussed in conjunction with

one another which rendering a deeper understanding than it would have been achieved if

the two studies had been interpreted separately.

3.1. Epidemiological method

The epidemiological study is located in the field of public health epidemiology which deals

with studies of the transition from health to disease (Silva, 1999; Vandenbroucke et al.,

2007).


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Historically, epidemiology may be divided into three periods: 1) early epidemiology (before

1830); 2) classical epidemiology (1830 � 1940); 3) new epidemiology (1940 � present)

(Saracci, 2010).

Hippocrates is considered to be the �founder� of the early epidemiology as he developed

the method by providing concise, accurate and complete descriptions of actual clinical

cases, including disease. Further, epidemiological theories were elaborated to explain the

spreading of disease. Early epidemiology contained three basic trends: medical,

demographic and theoretical. These trends became the foundation of the epidemiology as

it is known today: an investigation of diseases and their etiology at the population level

(Saracci, 2010).

The classical epidemiology was influenced by the industrial development in Western

Europe. In the UK medical death registration was introduced as early as 1801 and causes

of death were recorded as of 1838 (Saracci, 2010). In Denmark, death certificate became

mandatory by law in 1871 and causes of death have been registered since 1875 (Helweg-

Larsen, 2011). One of the most important epidemiologists from the period of classic

epidemiology may be John Snow (1813�1858). He is known for his analytical investigation

of the water supply and spreading of cholera during the epidemic in London in 1849 and

1854 and he discovered how the polluted water influenced the development of the

epidemic. In 1849, a Danish physician and epidemiologist, Peter Anton Schleisner (1818�

1900), discovered the correlation between mortality among newborns and droplet infection

(direct body contact or poor hygiene), the discovery of which led to neonatal mortality rate

in Vestmanna Islands in Iceland decreasing from 60-74% to five percent. Further, Louis

Pasteur (1822�1895) discovered microorganisms and agents of several diseases such as

tuberculosis and he contributed to formulating criteria for establishing causality in

epidemiological studies (Saracci, 2010; Silva, 1999).

The period around the Second World War was a turning point for new epidemiological

studies investigating life expectations for smokers as compared to non-smokers and the

risk of lung cancer, leading to many studies related to tobacco smoking and the

development of various diseases. Epidemiology of today is applied to description and

causation within population based investigations of health and disease as well as other

health-related conditions (Silva, 1999). Thus, this method is based upon how the patterns


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13

of the disease cause changes in the function of living beings. Because of the aim of this

thesis, a register-based approach was chosen.

3.1.1. Register-based study

The chosen approach was observational descriptive register-based, using the Danish

National Registers (Statistics Denmark, 2008). The reporting of the register-based study in

this thesis has sought to meet the recommendations of Strengthening the Reporting of

Observational Studies in Epidemiology (STROBE) (Vandenbroucke et al., 2007) (STROBE

checklist is provided as appendix A to the thesis). The Danish National Registers are from

an international point of view unique nationwide data sources (Frank, 2000); all admissions

and diagnosis have been registered since 1977 in the National Hospital Discharge

Register; outpatient diagnoses have been registered since 1995; prescribed use of

medication dispensed from pharmacies has been recorded since 1995 in the National

prescription Database, and demographic information (e.g., income, education, family,

housing etc.) has been recorded in the Socio-Economic Database since 1966 (Statistics

Denmark, 2008). These databases are linked by personal identification numbers (Civic

Person Register; CPR) which were introduced in 1968; this provides the opportunity to

collate information about the same individual from different databases without revealing

the identity of the individual person to the researchers. Due to such comprehensive

nationwide databases containing key medical, health care and socio-economic information

concerning citizens in Denmark (approximately 5,6 mill individuals ) (Statistics Denmark,

2008), a long tradition of register-based studies has been developed (Frank, 2000; Furu et

al., 2010), in addition to the long tradition of keeping comprehensive statistics on drug use

of the entire population (Hallas and Stovring, 2006). Registry-based studies in Denmark

have taken advantage of the wealth of health-related information in registries for several

decades.

Retrospective register-based cohort studies investigate population-based events that took

place over a specific time period. Such observational studies are commonly placed behind

Randomized Controlled Trials (RCTs) in terms of the strength of the evidence (Gliklich and

Dreyer, 2010; Silva, 1999). because of the limitations such as over-and under- coding,

poor confounder control, referral and prescription bias and variable internal validity),

Despite these limitations, observational studies have several advantages over RCT


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14

studies because of the power of long time follow-up and real-world validity in assessing the

advantages and disadvantages of preventive medicine, all of which can be valuable when

informing policy decisions and evidence-based medicine (Abrahamsen, 2010). Register-

based studies also include patients that RCTs would not traditionally include, such as

patients of all ages, with multiple confounding complications and various socioeconomic

backgrounds (Gliklich and Dreyer, 2010).

3.1.2. Statistics Denmark

Statistics Denmark was founded in 1850 as an extension of the establishment of the

government by the people; with the Constitution of 1849. Statistics Denmark is the central

authority for the Danish statistics. They collect, compile and publish statistics of the Danish

society (Statistics Denmark, 2008).

For the purpose of the study, were access required to The National Patient Database, the

National prescription Database, the Socio-Economic Database and The Danish Death

Register. Statistics Denmark approved the application and gave access to data through

permit 702538.

This permit allowed for identify, characterize and follow Danish citizens who were new

users of oral bisphosphonates.

3.2. Phenomenological-hermeneutics

The theoretical framework for the qualitative study was based on the French philosopher

Paul Ricoeur's description of a phenomenological-hermeneutic theory - the Interpretation

theory and time and narrative (Ricoeur, 1984; Ricoeur, 1985; Ricoeur, 1988; Ricoeur,

1976). This theory is commonly regarded as a bridge between the philosophies of

phenomenology and hermeneutic (Kemp, 1999; Hermansen and Rendorff, 2002).

Phenomenology concerns the philosophical study of the structures of subjective

experience and consciousness. As a philosophical movement it was founded in the early

20th century by Edmund Husserl. Husserl was concerned with systematic consideration

and investigations of structures of consciousness and the phenomena which appear in

actions of consciousness; thus, Husserl intended to provide a pure description of the

phenomena as they appear from the consciousness (Hermansen and Rendorff, 2002).

Paul Ricoeur analyzed the relationship between phenomenology and hermeneutic (Kemp,


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15

1999; Pedersen, 1999; Hermansen and Rendorff, 2002). He moved through his authorship

from an existential - to a structural - to a phenomenological hermeneutic way of thinking.

The hermeneutic philosophy is primarily based on the philosophers Heidegger and

Gadamer�s work from the early to mid-twentieth century. Heidegger investigated the

question of self or human realization of themselves and the world - with the interpretation

as an ontologically basic condition for the self and its realization where an interpretation

follows a circular structure; the hermeneutic circle. Gadamer (Heidegger�s student)

elaborated and further developed the doctrine of pre-understanding and the hermeneutic

circle (Pahuus, 2001). According to the hermeneutic interpretation, Ricoeur argued for the

essential of listen for and grasps phrases that describe how the world and reality is. In this

manner, he sought, reflected and argued, throughout his authorship in a dialectical

process to reconcile his thinking and thus bridge the gap between different perceptions

and sciences, not only as combining these, but as a new way of thinking, a so-called third

way (Kemp, 1999; Pedersen, 1999; Hermansen and Rendorff, 2002).

The phenomenological and hermeneutic theoretical basis of this thesis is inspired by Paul

Ricoeur's manner of viewing the world and his interpretation of individuals in the world.

3.2.1. Narrative

In order to be able to gain a deeper understanding of the aspects affecting newly

diagnosed women with osteoporosis; a story-telling / narrative approach was chosen.

Ricoeur argued that narrations are central to human lives. He perceived the language and

the way we experience the society as central and considered that language and

conversation are where the culture is formed. Ricoeur was interested in the cultural

aspects which are difficult to explain and which may seem irrational. He believed that

individual identity arises in connection with others (Hermansen and Rendorff, 2002;

Ricoeur, 1976).

A story may either be verbal (e.g., �live� speech) or in writing. Ricoeur describes three

aspects of the spoken language: 1) a sensuous aspect where the voice, tone and body

language give meaning to the words, 2) an ethnic cultural aspect, which refers to how one

uses language from the collective experience of the use of everyday language as a

common language, on an everyday basis 3) an ethical and moral aspect which affects the

individual's involvement in the discourse (Pedersen, 1999; Hermansen and Rendorff,


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16

2002). Ricoeur describes a discourse as an event where �someone says something to

someone about something�. Events become history when they are told or narrated, and

they leave a trail. Every story has a theme, idea or point, what is of concern in the story as

we followed the action, recalled or retold it (Kemp, 1999; Hermansen and Rendorff, 2002).

As previously mentioned, the �theme, idea or point� of the current study is newly-

diagnosed women�s experiences of living with osteoporosis they were prescribed

treatment for. Ricoeur describes how the narrator establishes a sequence order: a

beginning, middle and an ending to the story. A narration can be viewed as an ongoing

coherence in a meaningful whole which helps us to understand something different from

ourselves, leading to increased self-understanding (Ricoeur, 1976). Ricoeur further states

that to retell an event means to bring the past into the present in order to shape the future.

Ricoeur believed that an individual becomes aware of her participation in the world by

talking about it (Kemp, 1999; Pedersen, 1999; Ricoeur, 1976; Hermansen and Rendorff,

2002). Therefore this approach was chosen to describe women�s experiences living with

osteoporosis.

3.2.2. Interpretation

Essential to Ricoeur�s Interpretation theory, is his understanding of text and the concept of

distanciation - being objective or standing separate from the text. He argued that text is

discourse which is fixed in writing, in relation to four themes: 1) text as speech in writing, 2)

text as a structured work, 3) text as a projection of a world, and 4) text as the mediation of

self-understanding (Pedersen, 1999; Ricoeur, 1976; Hermansen and Rendorff, 2002).

A triple Mimesis, is used by Paul Ricouer as a method to understand any textual written

document of human action. Mimesis describes the time-related action and the shape of the

temporal lived life (Kemp, 1999; Ricoeur, 1984; Ricoeur, 1985; Ricoeur, 1988; Hermansen

and Rendorff, 2002). Mimesis1 �Pre-figuration� is a practical pre-understanding prior to

action in life comprising the ability of, in an expressive manner to be able to use the

concepts of actions in life, by using symbols, rules and norms based on an understanding

of time in action in life and speech (Hermansen and Rendorff, 2002). In the study,

mimesis1 are the women�s understandings and basic narratives of life based on the

historical and existential reality of the human life, prior to the tale about their experiences

and life lessons with osteoporosis.


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17

Mimesis2 is �configuration�; the creative composing activity which creates the action

(Hermansen and Rendorff, 2002). Mimesis2 will thus be the woman's individual stories

about life experiences with osteoporosis.

Mimesis3 completes the process; �configuration� of the action is not experienced as a story

unless we acquire and integrate it. We are affected when we follow the story thereby we

will become a �new person�. A �new-figuration� of our lives opens a reconstructed action

world, which we can live in. Mimesis3 is the process of comprehension - the researcher's

reflective work in order to interpret the hidden meaning in the obvious (Hermansen and

Rendorff, 2002; Ricoeur, 1976). The researcher �receives� the woman's narration, which is

given a meaning based on the researcher�s current situation and the historical period. The

tension between the researcher and the narrative world is recognized and new knowledge

is created (Kemp, 1999; Pedersen, 1999; Ricoeur, 1976; Ricoeur, 1984; Ricoeur, 1985;

Ricoeur, 1988; Hermansen and Rendorff, 2002).

According to Ricoeur, the interpretation of this study had three levels: a naïve reading, a

structural analysis as well as a critical interpretation and discussion (Ricoeur, 1976)

illustrated in Fig. 1 below and elaborated in section 4.2.2.

Fig. 1. Analytic levels in the interpretation of findings

�


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18

Naïve reading is the first reading and re-reading of the text in order to grasp its meaning as

a whole. Through the naïve reading the first interpretation of �what is spoken about� arises

where the interpreter tries to read the text with openness and with a phenomenological

attitude. The naïve reading is regarded as the first presumption (Lindseth and Norberg,

2004; Pedersen, 1999).

A structural analysis is carried out to validate and adjust the naïve interpretation and to

reach a deeper understanding. According to Ricoeur, the structural analysis addresses

identification of meaningful context: quotations as �what is said� and the primary

interpretation �what is spoken about� (Lindseth and Norberg, 2004; Pedersen, 1999). In the

movement from what the text says to what it talks about, significant units are identified and

formulated leading to emission of key themes, sub themes and patterns (Pedersen, 1999).

The analyzing process takes place through a dialectical movement between the parts and

the whole, performed in a helical process.

Critical interpretation and discussion is the final level of analysis and comprehension;

relevant literature and other research findings are incorporated in a discussion of the

benefit of one or more appropriate interpretations (Pedersen, 1999; Ricoeur, 1976;

Lindseth and Norberg, 2004). The interpretation is preceded by the critical analysis in a

dialectic process, between explanation and comprehension as a discussion of relevant

literature and other research findings within findings from the current study by relating to:

the naïve understanding; text as a whole; quotes from the interviews; significant units as

well as key themes and sub themes.


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19

4. METHODS AND STUDY POPULATION

Because different research questions require different type of data and different

approaches, the thesis contains both an epidemiological and a phenomenological-

hermeneutic study (cf. aim chap. 3). Methodologically, the thesis consists of two distinct

phases.

4.1. Epidemiological method

As the focus of the first study was to determine the distribution and determinants of

compliance and persistence to oral anti-osteoporotic treatment, an observational cohort

study with a register-based approach was used (also presented in section 3.1)

(Vandenbroucke et al., 2007). Baseline data were collected from The Danish National

Registers: data of admissions back to 1977, use of drugs back to 1995 these data were

obtained until the year before the index year; further were data from the socio-economic

dataset permitted from 1996, these data were obtained the year before index year and for

those entering the study in 1996 data were obtained 1996. Data of anti-osteoporotic drug

were obtained the year before index year, at baseline and follow-up during at least one

year after purchase prescription or until end of study. Additionally, were data from the

Danish Death Register obtained in the study period.

4.1.1. Data � study A

Data were obtained from the following Danish National Registers: the National Patient

Database (all admissions from 1977), the National prescription Database (use of drugs

from 1995), the Socio-Economic Database as well as the Danish Death Register. Based

on personal identification numbers data of the same individual were collated from these

databases (cf. section 3.1.1.) this was possible due to access from Statistics Denmark (cf.

section 3.1.2.). The participants whose data were collected between 1/1/1996 and

31/12/2006 were included in the study; follow-up was completed by the end of 2008.

Register based data are sensitive to the accuracy of the individual registration.


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20

Participants and descripting variables

All individuals 35 years or older who initiated osteoporosis pharmacotherapy were

identified and included based on at least one prescription for an osteoporosis drug by

using ATC codes: M05BA, M05BA01, M05BA02, M05BA04, M05BA06, M05BA07,

M05BB, M05BB01, M05BB02 (oral bisphosphonates), M05BX03 (strontium ranelate),

G03XC01 (raloxifene), H05AA01 and H05AA02 (PTH analogues). Several initiatives were

performed in order to adress potential souces of bias. Only participants with filled

prescription of osteoporosis medication were included; participants who exclusively used

intravenous osteoporosis medication were excluded. The participants had not filled

prescriptions for anti-osteoporotic drugs during one year before inclusion. Excluded were

participants who were not able to be followed up for a year, minimum one year of follow-up

was required as measured by filled prescriptions. Participants who were included in the

study comprised of 100,556 new users of anti-osteoporosis treatment, all with primary

osteoporosis.

Variables describing the population were grouped according to previous grouping and

meaningfulness of groups. Indexing variable was oral osteoporosis medication as

described above. Osteoporosis treatment was grouped in three categories: alendronate,

other bisphosphonate and non-bisphosphonate. Patients� age ranges from the index year

were grouped in quartiles: 1st: 35.0 � 70.1 years; 2ond: 70.2 � 71.8 years; 3rd: 71.9 � 79.0

years; 4th: 79.1 � 103 years. Number of comorbidities (0, 1, 2 and 3+) was calculated and

described as related to any registered diagnosis since 1977 until the year before the index

year. The following comorbidities were included: pulmonary, collagen and malignant

diseases, hemiplegia, dementia and heart failure. Registered fractures after age 50 were

grouped in fracture of the spine, hip, forearm, humerus, or any other fracture as one group.

Numbers of medications that participants had filled prescriptions for were grouped into four

categories: 0, 1-3, 4-9 and 10+, based on filled prescriptions from 1995 until the year

before the index year. The following medications were included: angiotensin-converting-

enzyme (ACE) inhibitor, anticoagulation (AC), antiarrhythmics, Angiotensin II (AT2)-

antagonists, beta blockers, anti-diabetics, opposed and unopposed hormone replacement

therapy (HRT), lipid lowering, prednisolone, analgesics, anti-epileptics, parkinson

medications, selective serotonin re-uptake inhibitors (SSRI) and other antidepressants.

Further, descriptions related to living conditions (socioeconomic and demographic

variables) from the year before the patient received the first prescription (or most recent)


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21

were included. Education level was classified into three categories: primary school �10

years, secondary school >10 years and bachelor degree or higher. Information on

household income (i.e., total family income) was available until the end of 2003: income

was divided into tertiles, further were the lowest income quintile also used. Retirement was

grouped into two categories: full retirement and retirement with limited earned income.

Information on housing was available until the end of 2004; this information was grouped

into: single family house, apartment, caravan site/allotment and institution/other.

Residential area was grouped as shown in the example in Table 1.

Table 1 Example of data grouping

codes from Statistics Denmark New code description

01 The metropolitan, Copenhagen 1 1.500.000 �

02 City with over 100,000 inhabitants 2 < 500.000 �

03 City with 50,000-99,999 inhabitants

04 City with 20,000-49,999 inhabitants

3 < 99.999 �

05 City with 10,000-19,999 inhabitants 06 City with 5,000-9,999 inhabitants 07 City with 1,000-4,999 inhabitants

4 < 19.999 �

08 City with 500-999 inhabitants 09 City with 250-499 inhabitants 10 City with 200-249 inhabitants

5 < 999 �

countryside or villages

�13 rural Area 6 < 199

Additionally, living distance from a university hospital was arbitrarily classified in our study

as under 50km/over 50km. Living with or without a spouse was estimated based on

whether another adult >25 years of age was registered at the same address. Data on

dependants (children under 25 years of age living at home) were also taken - these data

were available from January 1996.

Data from the National Prescription Database were used to calculate the number of doses

of oral osteoporotic medication. Purchases made by each patient were recorded in the

database and used to calculate a specific number of tablets bought and doses available

for each patient. The doses available were the basis for calculations of compliance and


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22

persistence to the medical treatment, which was quantified as medication possession ratio

(MPR) describing the extent to which the patients purchased enough daily doses during

the treatment period. The minimum of acceptable refill compliance was defined as an MPR

value of 75% (Siris et al., 2006) with patients allowed to change to other osteoporosis

medications without impact on compliance or estimates of persistence, the criterion

comprised gaps of medication available which was not allowed to exceed 56 days (8

weeks) (Landfeldt et al., 2012).

Potential bias may have occured according to the exposure, since information on the

dispensing date and medication dosage for each patient were available, it was although

not possible to know if and to what extend the patients took the dispensed medication.

This leads to a possible overestimation of the medication use. On the other hand, an

underestimation is possible as well as the information on medication obtained through

hospitals and other institutions were not available for this study. In addition, it is possible

that some patients may have purchased the medication abroad; this lack of information

may lead to an underestimation of drug use.

Another potential bias could have occurred in relation to the calculation of comorbid

conditions, because there is a risk that we over- or under- coded comorbid conditions

since diagnostics derived from general practice was not available in cases where inpatient

or outpatient hospital treatment was not required. This source of uncertainty may however

have been reduced through the indirect information of comorbid conditions by data

available of all filled prescriptions issued by general practitioners. For example, a

diagnosis of diabetes or hypertension made by the family physician in the absence of

hospital contacts would not be available for the statistical analysis, but any use of drugs for

diabetes or hypertension prescribed by the family physician would still be on record for

study.

4.1.2. Statistical analysis

All analyses were carried out using the Statistical Package for Social Sciences (SPSS) by

Statistics Denmark.

In order to describe baseline variables, crosstabs of gender and all variables were

displayed, with frequencies, percentages and P-values. The analysis included X2 test and

Fisher�s exact test to test the normal distribution of numerical (e.g. age and number of co-


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23

medications) and categorical variables (e.g. gender, comorbid conditions and prior

fracture).

Treatment persistence was estimated by non-parametric survival analysis - Kaplan Meier

survival analysis (normal approximation based on the standard error of the median)

(Barker, 2009; Silva, 1999); incorporating a new variable which was created to describe

the �end of treatment/end of study�. Gaps of more than 56 days (8 weeks) were

considered as non-persistence (Landfeldt et al., 2012).

7,191 individuals died before one year of follow-up and were therefore considered �lost to

follow up� and excluded before three non-overlapping outcome groups A, B and C were

defined based on refill compliance and persistence (cf. Fig. 3 in section 6.1.). Group A

(early quitters), who were persistent for less than one year consisted of individuals who

discontinued their medication prematurely (� 84 DDD during the first 6 months and no

further prescriptions). Group B (suboptimal), were persistent for more than one year but

exhibiting a refill compliance of MPR < 75% (less compliant but persistent users). Group C

(compliant and persistent users), were persistent for more than one year with compliance

> 75%.

Because logistic regression was used, binary response variables were created (1 and 0)

(Bewick et al., 2005; Silva, 1999) for the following variables: quartiles of age at index,

osteoporosis treatment (cf. example in Table 2), Charlson index, number of co-medication,

education level and household income. This leads to that only one position within each of

these variables is possible for each individual patient.

Table 2 Example of converting variable to binary exposure variables �

alendronate dummy_is_aln 1 0 0

other bisphosphonate dummy_is_non_aln_BP 0 1 0

Non-bisphosphonate dummy_is_non_BP 0 0 1

To identify independent factors predicting each of the three risk groups, logistic regression

analyses were conducted. Multivariate Cox proportional hazard regression model (model

A, B and C) with a backward stepwise procedure was applied (Lu Xu and Wen-Jun Zhang,


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24

2001) and analyses were used to calculate hazard ratios, 95% confidence intervals and P-

value. Five analyses using this model were conducted: analyses of group A (Early quitters)

versus group B+C (persistent users); group B (Persistent user with MPR<75%) versus

group C (Persistent and compliant users with MPR>75%); and group A (Early quitters)

versus group B (Persistent user with MPR<75%). Since the cohort consisted of mostly

women (86%) it was assumed that these results would be generalizable to the female

population only. An analysis using the same model but entirely based on the smaller male

population was also conducted (group A (Early quitters) versus group B+C (persistent

users) and group B (Persistent user with MPR<75%) versus group C (Persistent and

compliant users with MPR>75%).

Due to the hypothesis explanatory variables based on biological and demographic grounds

was prioritised in model A and B. Explanatory variables were mutually adjusted in all three

models A, B and C. Baseline characteristics were used as explanatory variables as

follows: Model A: index year (numeric), education level, housing, children, living without a

spouse/single, household income and osteoporosis treatment. Model B: prior explanatory

variables in addition to gender and age. Model C: prior variables added explanatory

variables selected by mutually adjusting for all other significant or borderline significant

explanatory variables from remaining baseline variables selected from using stepwise

analysis with a critical P<0.20. This led to a minor difference in the explanatory variables

selected for model C in the analyses of risk groups. All analyses included the following

variables: number of drugs; type of drugs; numbers of comorbidities; comorbidity; fracture;

and hormone replacement therapy (opposed). Three analyses, group A versus B for the

total cohort as well as the two analyses based on the male population included the

following variables: retirement status, retirement with maintained limited work income,

living close to university hospital and living in the countryside or in a village.

Sensitivity tests (Bewick et al., 2004; Silva, 1999) were performed according to compliance

and persistence as some patients who stop bisphosphonate therapy have been found to

reinitiate treatment even after extended gaps (Cadarette and Burden, 2010). Different

outcomes were tested by using time gaps of 84 days (12 weeks) and 56 days (8 weeks)

(Landfeldt et al., 2012), the latter of which was chosen in the study as no difference was

found. Further, two sensitivity tests were performed regarding the 0.1 % (N=131) who

lacked education and / or income information - these participants were subsequently


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25

excluded in the primary analysis. In the first test, the individuals with missing information

on education were assigned a value equal to the mean education level for the entire

group. In the second sensitivity analysis, individuals with missing data on income were

assigned a value equal to those participants who had the same level of education. If this

information was missing, median income of the study cohort were used. These sensitivity

tests yielded the same conclusions as the primary analysis. No other imputation was used

for missing data in the study.

4.2. Phenomenological-hermeneutic method

Study B was based on a phenomenological-hermeneutic approach and the theory of

interpretation (Ricoeur, 1976) presented in section 3.2., as the focus was women�s

experiences of life with osteoporosis. The design was longitudinal because longitudinal

data allow for investigation of the evolvement of patterns and changes over time and can

potentially provide fuller information about individual experiences (Saldaña, 2003).

4.2.1. Data � study B

The study was conducted in relation to Gentofte Hospital and Koege Hospital which are

located in two different regions of Denmark.

15 newly-diagnosed women were enrolled in the study. The women were contacted when

they were at the hospital to undergo a DXA scan, and included consecutively and

according to the inclusion criteria between January and April 2011.

Inclusion criteria were: a DXA scan at one of the participating hospitals showing a T-score

below -2.5 (Lower back or hip) (Kanis et al., 1994); female gender, age > 65 years; no

previous known osteoporotic fracture; at least one of the known risk factor�s (Genant et al.,

1999); and prescription of anti-osteoporotic treatment. Exclusion criteria were: signs of

cognitive impairment which would interfere with the interview; previous osteoporosis

diagnosis or previous treatment with anti-osteoporotic medication.

Inclusion and exclusion were based on information provided by participants at the first

telephone contact shortly after the participants had received the scan result and were

diagnosed.


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26

Data were obtained using individual interviews three times during one year; first interview

shortly after the DXA-scan and diagnosis was obtained during early spring 2011; second

interview after six months in the fall 2011; and the third interview after approximately one

year, these were obtained during spring and summer 2012. These intervals were chosen

since studies have shown that individuals compliance stabilizes around six months after

medical treatment is initiated (Lindberg et al., 2008; World Health Organization, 2003a)

and since adjustments to living with a chronic condition often take a long time period

(Holloway, 2007). An open interview guide (the interview guide is provided as appendix B

to the thesis) was used to ensure consistency and encourage openness and flexibility

during the interviews and the interview guide was adjusted between interview-rounds

(Kvale and Brinkmann, 2009). The participants were first asked to describe their socio-

demographic basics as age, marital status, children etc. these are presented in Table 3.

The opening question was: "Will you please tell about your experiences of living with

ostoporosis?"

Table 3 Overview: Self-reported socio-demographics of the informants �

age 65 � 79 years, mean age 71.9 years living with spouse 8 children 0 � 4 (2 women had no children) retired 13 comorbidity cancer / hysterectomy: 6 / 2

collagen disease: 3 fracture: 1 cardiac / hyper cholesterol: 2 / 2 COPD: 4 metabolism / Scheuermann's disease: 1 / 1

BMI < 19 3 menopause <45y 4 heredity 10 daily pain 8 smoking / alcohol 6/1 referral to DXA scan General practitioner (GP) / own

request to GP: 7 / 4 medical specialist: 4

PhD�thesis�by�Carrinna�Hansen

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27

Participants were encouraged to tell stories of their personal experiences of living with

osteoporosis, encouraged by the interviewer saying "yes continue" or "will you elaborate

on what you said about ... little bit more" and similar; the interviewer was open and

attentive to ensure rigor in catching the individual woman's perspective and unfold the

meaning attached to the informants� lived experiences and life-world (Hall, 2001) so that

the participants were able to tell their stories (cf. section 3.2.1.). Most interviews took place

in the participants� homes. Three women chose to give their interview at the hospital, and

five telephone interviews were conducted as the third interview at the convenience of the

participants to ensure continuous participation. One participant wished to withdraw from

the study when contacted prior to the second interview-round due to personal reasons and

another participant was not reachable for the third interview-round. The path and length of

interviews are illustrated in figure 2 below.

�

Fig. 2 The interview path

�

The interviews were taped and transcribed verbatim and fully, totalling a text of 656 pages

(Times new roman, font size 12, spaced 1).

4.2.2. Analysis - Interpretation

The data analysis was inspired by Paul Ricoeur's theory of interpretation (Lindseth and

Norberg, 2004; Pedersen, 1999; Ricoeur, 1973; Ricoeur, 1976) as described in section

3.2.2.

The level of naïve reading was achieved by reading and rereading the 656 pages several

times to create an overview and an interconnected understanding of what the text was

about. In this process notes were made: immediate thoughts and questions to the

Fifteen face to face interviews shortly after

diagnosis

Fourteen face to face interviews after six month

Eight face to face interviews and five

telephone interviews after approx. one year

length of interview 21-72 min.




__________________________________________________________________________________________________________________________________�

28

understanding of what the text was telling. Reading the text as a whole led to a

distanciation from individual stories to the perception of the text as a whole.

The level of structural analysis was performed as the first interpretation when identifying

significant units of what was spoken about in the text; these units validated and adjusted

the naïve understanding. Next, significant units were interpreted and key themes and sub

themes emerged, these were validated in relation to the naïve understanding, meaningful

units and significant units. As according to Ricoeur (Ricoeur, 1976); the structural analysis

was performed within a dialectical movement between the parts and the whole of

interpretation, in a helical process this as described in section 3.2.2. Specifically, the

process of the structural analysis was as concretized in the illustrated example in Table 4.

Table 4 An example of the structural analysis and themes

Meaningful units

what is said �Quotes�

�

Significant units

what is spoken about (primary interpretation)

�

Sub themes

�

Themes

emissions of key themes

�Yes, well I've always bicycled a lot in my life, but now I've found out that � walking is actually better (�) so now I am walking a lot�

To have gained new insight of the type of physical activity that are especially suited for strengthening of bones

Lifestyle Changes

Being on the path of learning to live with osteoporosis

�

The structural analyses led to a deeper understanding of the experiences of living with

osteoporosis which was refined and finally interpreted through a critical analysis and

discussion. A continued interpretation was performed through a dialectic process between

explanation and comprehension as a discussion. Benefits of one or more appropriate

interpretations were combined by incorporating relevant literature and other research

findings in the discussion of the findings. This involved the text as a whole; naïve

understanding; quotes from the interviews; significant units as well as key themes and sub

themes. Thus the new interpretation was created and a new understanding of life with

osteoporosis was derived.


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29

5. ETHICAL CONSIDERATIONS

The National Committee on Health Research Ethics of the capital region confirmed that

approval for the study was not required according to Danish law due to the non-biological

character of the study (J.no. H-C-FSP-2011_01, cf. appendix C). Statistic Denmark

provided the access to micro data from the Danish National Registers for the purpose of

the epidemiological study was given by Statistics Denmark through permit 702538. Data

processing is done via Statistics Denmark. Data cannot be transferred from Statistics

Denmark to the researcher�s computer but are analysed using a secure, encrypted Internet

connection. The identities of individual patients were blinded to the investigators.

The study was approved by the Danish Data Protection Agency (J.no. 2012-41-0875)

following the directives for data retention (cf. appendix D). The study also followed the

ethical principles of the World Medical Association Declaration of Helsinki (World Medical

Association, 2008).

As part of the qualitative study, pursuant to the rules of the Helsinki Declaration, the

participating women received both oral and written information about the study (the written

information is provided as appendix E1 and E2 to the thesis). Researchers did not have

access to patients� personal identification numbers or medical files. The participating

women gave written informed consent to be contacted for further participation in the study

and oral informed consent was obtained before each interview. All forms of reporting and

publication of the results are in an anonymized form.

Individual interviews were chosen in order to explore the individual perspective in depth.

My role as an interviewer may have affected the interviews because the informants knew

that I was a nurse. The informants expressed expectations of getting the opportunity to get

advice during the interview, however their questions were kindly circumvented until after

the interview was completed. After the interview was done, the informants had an

opportunity to obtain my advice as a nurse: in most cases I encouraged the women to

contact their GPs or other relevant healthcare professional for clarification of the

questions, as well as in cases of any other health related problem which I considered

needed to be examined by the physician; as it is my ethical responsibility as a nurse. On

the other hand, it is also possible that the informants were more open during the interviews

due to my healthcare profession, or due to the interview performed in their home (Herzog,

2005). However, my role as a nursing-researcher may have been diminished by my


__________________________________________________________________________________________________________________________________�

30

conducting the interviews in the women�s homes, where the interaction was less affected

by my professional status as I was a guest in the women�s homes (Hall, 2001).

Some of the interviews in the third interview-round were conducted as telephone

interviews at the convenience of the informants in order to ensure participation. Telephone

interview can be an effective method of data collection when the interviewer is aware of

the challenges involved (Musselwhite et al., 2007). These challenges include the lack of

control over the extraneous information, challenges regarding maintenance of the

informants� involvement and the lack of the ability to observe non-verbal communication.

Before the telephone interview, I had met and interviewed the informants twice and we

were therefore acquainted with each other. Based on the previous interactions, I did not

get the impression that the interviews in a telephone format affected the data.

Newly diagnosed patients� are in a vulnerable situation and the experience of participation

in a research study may possibly add an additional burden. When individuals have an

opportunity to talk about their own experiences, they may gain meaning and understanding

through telling their story which may be helpful when adapting to difficult situations

(Holloway, 2007). This may have been the case for several of the participating women, as

they spontaneously described that they had chosen to participate in the project in order to

have the opportunity to gain a better understanding of osteoporosis.

Throughout the data collection the informants� needs were looked after through

attentiveness to the individual needs and wishes in relation to interview time, location and

in other contexts.


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31

6. RESULTS

The results of the two studies in this thesis are presented in three papers. Paper I � study

A named Anti-osteoporotic therapy in Denmark � predictors and demographics of poor

refill compliance and poor persistence; paper II � study B named Women�s experiences of

their osteoporosis diagnosis at the time of diagnosis and six months later: a

Phenomenological Hermeneutic Study and paper III � study B named Women�s Lived

Experiences of learning to live with Osteoporosis: A Longitudinal Qualitative study. The

results of the quantitative study (paper I) and the qualitative study (paper II and III) are

summarized below.

6.1. Paper I - study A

The results from paper I expand on the current knowledge of compliance and persistence

by identifying the determinants for potential risk groups based on a Danish cohort

consisting of N=100,949 individuals. As shown in Fig. 3. Lost to follow-up: 393 emigrated

before one year and 7,191 died before one year of follow-up leading to the final cohort of

N=93,365. The cohort comprised 86 % women, mean age were 70.4 years, mean time of

follow-up 5.2 years with a total of 520,604 person years of observation time. A significant

linear time trend was observed, which shows fewer individuals stopping treatment early

(OR 0.92, 95 CI 0.92-0.93) and fewer with suboptimal MPR in later years (OR 0.98, 95 CI

0.96-0.99) (cf. Table 5 below). Information of education and/or income were missing for

0,1 % (N = 131) of the population.

A summary of the baseline characteristics: alendronate was prescribed for 54% of the

individuals, 40% were prescribed other bisphosphonates and 6% were prescribed other

anti-osteoporotic treatment mainly raloxifene. Hospital treated comorbid conditions were

not recorded for 52% of the individuals; however, 73 % had used more than three different

medications the year before index. Prior fracture was recorded for 39 % of the cohort; and

almost one quarter had been treated with prednisolone the year before index. Educational

level was distributed as follows: 65% with primary school education, 21% with secondary

school education and 14% had a bachelor degree or higher education. Almost 75% were

retired, approximately 48% lived without a spouse, and only 3% of the individuals had

children living at home. More than two thirds lived less than 50 kilometres from a university

hospital (cf. Table 1: paper 1).


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32

Persistence � the estimated length of treatment was estimated with different anti-

osteoporotic treatments. Mean treatment with alendronate was found to be four years with

a median treatment of almost 3 years. Treatments with other bisphosphonates were of

considerable shorter duration except for ibandronate and risidronate which were

associated with almost as long treatment duration as alendronate. There was only a

negligible difference between treatment duration of alendronate and non-bisphosphonates

(cf. Table 2: paper 1).

�

The cohort of 93,365 individuals were found to be distributed in the three outcome groups

A, B and C (described in section 4.1.2.) with 38.7% terminating treatment within the first

New users of anti-osteoporosis drugs 1996-2006, age � 35 years, lived in the country at least 3 years before treatment:

N = 100,949 female: 85,581 (84.8%) - male: 15,368 (15.2%)

7,191 died before one year of follow-up:

N = 93,365 female: 80,242 (86.0%) male: 13,123 (14.0%)

393 emigrated before one year of follow-up was complete:

N = 100,556 female: 85,258 (84.5%) male: 15,298 (15.2%)

Fig. 3

Group A) terminating treatment during 1st year (early quitters)

N = 36,156 (38.7%)

female: 30,579 (84.6%) male: 5,577 (15.4%)

Group C) persistent and compliant MPR � 75% (compliant)

N = 52,841 (56.6%)

female: 45,896 (86.9%) male: 6,945 (13.1%)

Group B) persistent but compliant with MPR <75% (suboptimal)

N = 4,368 (4.7%)

female: 3,767 (86.2%) male: 601 (13.8%)


__________________________________________________________________________________________________________________________________�

33

year, 4.7% persistent with low refill-compliance, while 56.6% were persistent and

compliant (cf. Fig. 3).

Characteristics of �early quitters�

Within the first year 38.7% of anti-osteoporotic users stopped treatment as defined above

(MPR in section 4.1.1. and outcome groups in section 4.1.2.). Statistically significant key

factor characteristics of individuals who were persistent for less than one year (outcome

group A) compared with those who were refill compliant for more than one year (group

B+C) are presented in Table 5 below as well as in paper I.

A decreased risk of quitting during the first year of treatment was associated with

household income level above lowest tertile, age in the second and third quartile (age 70.2

� 71.8 and 71.9 � 79.0 years), retirement both with and without maintaining limited work

income, living in the countryside or in village, previous fracture of the forearm, other

medication such as ACE inhibitors, lipid lowering and analgesics and a diagnosis of

dementia. An increased risk of quitting during the first year of treatment was demonstrated

to be associated with previous fractures overall, as one category. Other factors were: living

in an apartment, having children who are living at home, living without a spouse,

osteoporosis treatment with other bisphosphonates or non-bisphosphonates, male gender,

living close to a university hospital, number of drugs taken concurrently, and taking other

medications such as: hormone replacement therapy, prednisolone and anti-depressants.

Other factors associated with an increased risk of quitting early were: the presence of

more than one comorbidities (particularly 3 or more), and presence of pulmonary or

collagen diseases.

As hypothesized, the differences between the two groups with low refill compliance

(group A �early quitters� versus group B �suboptimal MPR� (cf. Table 5 and paper I)) were

also found in terms of socioeconomic and demographic factors as well as some concurrent

medications and comorbid conditions predicting a decreased or an increased risk of

stopping treatment prematurely (further description cf. paper I pp. 2086 and 2088). The

overwhelming health preventive problem was found within the group of individuals

terminating treatment prematurely and not the smaller group that persisted with treatment

but had low refill compliance.

In the interests of clarity, are three analyses of the risk groups A, B and C summarized in

Table 5, presenting the statistically significant key predictors of �early quitters�, �suboptimal


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34

compliance� and comparison of the large group of individuals who terminated treatment

prematurely and the smaller group of individuals who persisted with low refill compliance.

Table 5 Overview of selected results from three different analysis showing only significant predictors

Model C Backward stepwise adjusted for hemiplegia, malignant disease, cardiovascular diseases, hip fractures , humerus fractures, hormone replacement therapy unopposed, analgesics, anti-epileptics, anti-parkinson, other anti-depressants

group A versus group B+C

(cf. Table 3: paper I) N = 93,365

male: 13,123 female: 80,242

group B versus group C

(cf. Table 4: paper I) N = 57,209 male: 7,546

female: 49,663

group A versus group B

(cf. Table 8: paper I) N = 40,524 male: 6,178

female: 34,346

Effect of the index year � time trend 0.92 (0.92-0.93)*** 0.98 (0.96-0.99)*** 1.07 (1.05-1.08)*** Education level; Prim. school (ref) Secondary school

1 0.90 (0.82-0.98)*

Housing; Single family house(ref) Apartment

1 1.10 (1.07-1.14)***

1 0.90 (0.83-0.97)*

Children at home (< 25 years) 1.47 (1.35-1.59)*** 0.68 (0.56-0.82)*** living without a spouse/single 1.18 (1.14-1.22)*** 1.12 (1.03-1.21)** Household income; Low. tertile(ref) Middle tertile Highest tertile

1 0.94 (0.91-0.98)** 0.86 (0.83-0.90)***

1 1.24 (1.13-1.37)***

Osteoporo. treat.; Alendronate(ref) Other bisphosphonate Non-bisphosphonate

1 1.73 (1.68-1.79)*** 1.15 (1.09-1.22)***

1 0.10 (0.09-0.11)*** 0.82 (0.73-0.93)**

1 0.06 (0.06-0.07)*** 0.73 (0.65-0.83)***

Sex male 1.33 (1.27-1.38)*** 0.78 (0.71-0.86)*** Age at treat; 1st quartile (ref) 2ond quartile (70.2 � 71.8 years) 3rd quartile (71.9 � 79.0 years)

1 0.85 (0.81-0.89)*** 0.84 (0.80-0.88)***

1 0.84 (0.75-0.93)**

1 1.14 (1.02-1.27)*

Retired 0.94 (0.90-0.97)*** 0.90 (0.83-0.97)* Retirement (limited work income) 0.93 (0.88-0.98)** Living close to university hospital 1.09 (1.06-1.12)*** 0.88 (0.82-0.94)*** Living in countryside or in village 0.97 (0.93-1.00)* 1.09 (1.01-1.18)* 1.12 (1.03-1.21)** Number of drugs; 0 (ref) 1-3 4-9 10+

1 1.10 (1.03-1.18)** 1.23 (1.16-1.32)*** 1.33 (1.23-1.43)***

1 0.84 (0.72-0.98)* 0.73 (0.63-0.85)*** 0.69 (0.58-0.82)***

Type of drugs ACE inhibitors Hormone replacement (opposed) Lipid lowering therapy Prednisolone Analgesics Anti-parkinson Anti-depressants (ssri)

0.90 (0.86-0.94)*** 1.09 (1.02-1.17)** 0.94 (0.89-1.00)* 1.08 (1.04-1.12)*** 0.97 (0.94-1.00)* 1.08 (1.03-1.13)**

1.10 (1.03-1.19)**

1.13 (1.05-1.22)** 1.39 (1.09-1.77)**

Charlson index; 0 (ref) 2 3+

1 1.05 (1.01-1.1)* 1.14 (1.08-1.21)***

1 1.16 (1.05-1.27)** 1.20 (1.05-1.37)**

Comorbidity Pulmonary disease Collagen disease Dementia

1.05 (1.00-1.10)* 1.19 (1.15-1.23)*** 0.83 (0.72-0.95)**

0.89 (0.79-1.00)* 0.87 (0.80-0.95)** 1.58 (1.17-2.13)**

Fracture Forearm Any

0.92 (0.88-0.97)** 1.07 (1.03-1.11)**

* p<0.05, ** p<0.01, *** p<0.001 Model A, B and C, prior covariates due to hypothesis: education, housing, children, living without a spouse/single, income, and treatment

Model B and C, priori covariates: sex and age


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35

The cohort comprised of 13,123 male individuals. Male �early quitters� compared to male persistent

users group B+C (cf. Table 5: paper 1) showed a decreasing risk of quitting during the first year of

treatment, was associated with: middle (OR 0.88, 95 CI 0.80�0.98) and highest tertile of household

income (OR 0.79, 95 CI 0.71�0.87), second age quartile (70.2 � 71.8 years) (OR 0.79, 95 CI 0.71�

0.89) and third age quartile (71.9 � 79.0 years) (OR 0.74, 95 CI 0.66�0.83). Other factors

statistically associated with decreased risk of stopping treatment prematurely were prior use of

ACE inhibitors (OR 0.87, 95 CI 0.78�0.98), dementia (OR 0.63, 95 CI 0.45�0.88) and fracture of

the forearm (OR 0.80, 95 CI 0.68�0.95). An increased risk of quitting during the first year of

treatment was found to be associated with: living in an apartment (OR 1.16, 95 CI 1.06�1.28),

having children at home (OR 1.35, 95 CI 1.15�1.60), living alone (OR 1.22, 95 CI 1.11�1.33),

osteoporosis treatment with other bisphosphonates than alendronate (OR 2.12, 95 CI 1.95�2.31)

and non-bisphosphonate (OR 1.37, 95 CI 1.03�1.83), living close to a university hospital (OR 1.14,

95 CI 1.05�1.23), prior treatment with prednisolone (OR 1.12, 95 CI 1.03�1.22), as well as

collagen disease (OR 1.10, 95 CI 1.01�1.20).

In the smaller population of 7,546 men who were persistent beyond one year; the group of

persistent male users with low refill compliance were compared with compliant and persistent

users with MPR>75% (cf. Table 7: paper 1). Other bisphosphonate than alendronate (OR 0.19, 95

CI 0.14�0.26) was found to be the only decreasing factor associated with suboptimal MPR;

additionally, factors associated with an increased risk were found to be: having a bachelor degree

or higher education (OR 1.36, 95 CI 1.06�1.75), prior treatment with anti-parkinson medication (OR

2.05, 95 CI 1.28�3.27) and Charlson comorbidity index 1 (OR 1.38, 95 CI 1.01�1.88).

6.2. Paper II and paper III - Study B

Study B is reported in paper II and III. It comprised of 15 women with osteoporosis in the age range

of 65 to 79 years (at the first interview). The informants were recruited from two university hospitals

(cf. Fig. 4 below). Nearly half of the women reported living alone; a majority was retired when

diagnosed; with two to four children on average except two who did not have any children. The

majority had a history of hereditary osteoporosis, and more than half of the women had daily back

pain and all except three reported other comorbidities (mainly cancer, COPD and collagen

disease) (cf. Table 3 in section 4.2.1).

Two informants were lost to follow up all the way during the one-year study period. One informant

wished to withdraw from further participation in the study prior to the second interview round and

another informant was deemed not reachable after multiple attempts to reach her in relation to the


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36

third interview. As illustrated in Fig. 4, the first interview round comprised of 15 interviews, the

second interview round comprised of 14 interviews and the third interview round comprised of 13

interviews. Characteristics of the study population and the material used in the interview are

described in section 4.2.1.

�

�

�

�

�

�

�

�

The results from the two papers will be briefly described below. Themes which emerged during the

analysis are presented in relation to the structure in Table 6.

The interpretation arose through three levels of analysis (cf. section 3.2.2). When it came to the

experiences of living with osteoporosis, the first interpretation of �what is spoken about� appeared

to be closely related to the process of diagnosis, the understanding of the medical treatment and

the considerations concerning adaption to living with osteoporosis. Further development of this

understanding led to coming to terms with the medical treatment with choices being based on

individuals� interpretations and reflections related to osteoporosis.

These topics were subject of further analysis and interpretation, which led towards the necessity of

understanding and find meaning in the diagnosis, the medical treatment along with the need to

adapt to live with osteoporosis. The structural analysis led to three key themes emerged: 1) �being

diagnosed�; 2) �to be prescribed medical treatment�; 3) �being on the path of learning to live with

osteoporosis� (cf. paper II); Further the structural analysis led towards an understanding of patterns

of living with osteoporosis one year after the diagnosis this led to two key themes related to life

Fig. 4

University hospital A)

N = 8

University hospital B)

N = 7

15 informants

First interview (15)

Second interview (14)

Third interview (13)

Paper II

Paper III


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37

with osteoporosis during time: 1) �to become influenced by the treatment� and 2) �daily life with

osteoporosis� (cf. paper III). The key themes and sub themes are summarized in Table 6 below.

Table 6 An overview of key themes and sub themes (paper II and paper III)

� Key themes Sub themes

�

�

Paper II

Being diagnosed - to be taken seriously

- acceptance

Being prescribed medical treatment - decision against the medical

treatment

- decision to pursue the medical

treatment

Being on the path of learning to live with

osteoporosis

- the need to adapt

- lifestyle changes

Paper III

To become influenced by the treatment - taking the medication

- stops taking the medication

Daily life with osteoporosis - interpretation of symptoms

- interpretation of scanning result

- life style reflections

�

Being diagnosed was described as a process, rather than a discrete event, which could extend

over a longer period of time. Another central aspect of this theme is the experience of being �taken

seriously� and cared of. When the physician is aware of a patient�s increased risk of osteoporosis

(for instance due to previous illness or hereditary predisposition) and acts upon this awareness,

this fosters the feelings of �being taken cared of� and �taken seriously�. On the contrary, needing to

be a persistent advocate for one�s own health and having to convince the physician of the need for

a thorough examination due to symptoms or a feeling of something needs to be done produces the

opposite feelings (cf. paper II). Six month after diagnosis, the negative experiences remained

central to living with osteoporosis, although they seemed to have undergone a sort of

transformation or resignation affecting the basic trust towards one�s physician and the health care

system in general and to some extent mediating uncertainty related to the diagnosis, fracture risk

and implications for future health (cf. paper II); this development was found to be highly dependent

on earlier negative experiences based on the previous contact with the health care system (cf.

paper III). In addition, lack of sufficient information tends to be a mediating factor both related to


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38

accepting the diagnosis and to the understanding of being at an increased risk of osteoporotic

fracture; irrespective of negative or positive experience of being taken seriously (cf. paper III). The

need for thorough and more detailed information are especially important when the patient has had

a critical illness and experiences of not being taken seriously as well as an asymptomatic

osteoporosis (cf. paper II). Further, appeared difficulties with accepting the diagnosis to be

multifaceted; these difficulties turned out in several ways, frequently with a considerable element

related to the conditions invisibility, but also a matter of an immediate decision, a decision made

over a longer period or a matter of trust towards the physician and health care system or as fear of

being dependent on someone else�s assistance or the fear �looking like a mother or a grandmother

with visible signs of osteoporosis� (cf. paper II). An awareness of the impact of osteoporosis was

developed and came into view in the second interview round and presented in a form of a struggle

to accept the diagnosis, which emerged in relation to the impact that osteoporosis had on social life

and life in general: altered appearance and vanity, visible limitations, physical and mental energy

limitations affecting the ability to care for ill spouse. On one hand, this was found to lead to anxiety

regarding future expectations and, on the other hand, it also lead to feelings of being satisfied but

slightly alarmed as long as it did go well (cf. paper II).

Being prescribed medical treatment appeared to be a cognitive process of comprehension and

meaning creation reflected in a decision-making regarding whether to pursue or reject medical

treatment (cf. paper II and paper III). Shortly after diagnosis, patients focused on practical issues

(such as how to take the medication) which are a part of the cognitive process, together with the

individual�s thoughts and perception this appeared to be a mediating factor. Thoughts of being

worried and anxious regarding side effects occurred in relation to the comprehensive package

leaflet in the medication package, information from other sources as others' experiences or

information through the internet and likewise; but also due to lack of information from the GP and

other healthcare professionals (cf. paper II).

Being on the path of learning to live with osteoporosis appeared to be closely linked to making

a decision not to let the illness control one�s life but having hope and bright ideas and focusing

instead on enjoyable things in life such as new challenges and ways of being physically active (cf.

paper II). Adapting to live with osteoporosis was raised as developing acceptance in the beginning

of the process of decision-making (cf. paper II).

To become influenced by the medical treatment did throughout the second round of interviews

occurred as continued focus on side effects and seeking information and advice on this topic.

Focus was also own experiences of side effects or discomfort, in some cases, this was followed by


__________________________________________________________________________________________________________________________________�

39

patients becoming skeptical of medical treatment due to lack of meaningfulness of taking

medication giving side effects or discomfort when treating a not noticeable condition (cf. paper II).

Primarily in the third round of interviews did the informants come to terms with the medical

treatment (cf. paper III). Discontinuation of the medical treatment was influenced by the

interpretation of lack of information, experiences of not �being taken seriously� and experiences of

side effects or discomfort, together with a pronounced reserved attitude towards taking medication

generally. Moreover, central to the decision of start to take and continue taking the medication was

the experience of trusting the validity of the diagnosis, and the soundness of the treatment plan,

belief in the ability to prevent deterioration and disability when following the medical treatment as

well as faith in the GP; and to some extent anxiety of what would happen if the medication

treatment was discontinued (cf. paper III). Other factors influencing the decision around one�s

medical treatment were: feelings of responsibility to make the right choice, respect for authority and

worry about forgetting to take one�s medications (cf. paper II and paper III). Patients who did not or

almost did not describe these processes tended to be preoccupied with other issues such as taking

care of an ill spouse, grief or things as having other extensive comorbidities, having trusting the

validity of medical treatment and trust in the GP; or a combination of these factors (cf. paper III).

Daily life with osteoporosis was further elaborating learning to live with osteoporosis as a

trajectory and evolvement of experiences related to osteoporosis as interpretation of symptoms

(back pain, side effects or discomfort of the medical treatment) as well as efforts to explain and

accept the diagnosis and the medical treatment (cf. paper III). Six month after diagnosis, some

degree of lifestyle changes was shown to be recurrent, mainly in relation to diet and considerations

to be taken as an on-going need for planning daily activities; these were interpreted to be based on

new knowledge obtained through various sources and reflected in a sense of pride in carrying out

physical activities, changing diet, planning daily activities and leading an independent life (cf. paper

II). This process of acceptance and adaptation to live with osteoporosis appeared mainly at the first

interview round. It focused on information and/or perceptions of lifestyle advice when living with

osteoporosis. At the second interview round the focus were mainly on testing diet and physical

activity changes (cf. paper II); these factors were exposed to a deeper reflection together with

reflections on prior lifestyle at the third interview round (cf. paper III). Life with osteoporosis was, in

many cases, connected to a development related to understanding of the scanning result as well

as the interpreted value and the expected results of a future scan, which is often planned two years

after the diagnosis (cf. paper III).

Exceptions from being involved in this process of changing and adapting to living with osteoporosis

was found, in cases of being occupied with other things such as taking care of an ill spouse, grief,


__________________________________________________________________________________________________________________________________�

40

other comorbidities or a combination of these factors (cf. paper III). To some extent the

experiences of living with osteoporosis were found to be described in terms of being �intrusive�,

and in some cases described as completely non-intrusive to the point of �forgetting about the

disease� (cf. paper II and paper III).

6.3. Model comprising the overall study results

The study results expand the current knowledge of compliance and persistence in defined risk

groups, provide a deeper understanding and identify overarching themes related to life with

osteoporosis during time. The overall results are illustrated in a model (Fig. 5), which visually

represents a suggestion of how living with osteoporosis is influenced by the medical treatment and

decision-making based on comprehension and new meaning creation through a mutual interplay

when development of adaptation to life with a chronic condition.

Summary of the model:

Daily life with osteoporosis as a chronic condition is sought to be illustrated as a process of

comprehension and meaning creation reflected in a decision related to medical treatment. �Being

diagnosed� is partly outside the model, because being diagnosed was interpreted as a process

which could take long time, sometimes years, during which the diagnosis was uncertain or

unknown. "Being diagnosed" was both a part of patient�s life before diagnosis and a part of daily

life with osteoporosis when it was diagnosed (cf. paper II).

�To be prescribed medical treatment� was found to be closely related to �being diagnosed�: the two

events were positioned in conjunction with one another, but also as separate events.

The direction arrows follow the time flow of adapting to live with osteoporosis, but also back and

forth when negotiating how to live with a chronic condition requiring pharmaceutical treatment. One

direction arrow in the model pointing from �to be prescribed medical treatment� to all three outcome

groups demonstrated in study A (cf. paper I) as a connection to the results of study B (cf. paper II).

This connection is interpreted since first step of decision-making about the medical treatment were

in most cases taken at this time but also highly mediated by �to be influenced by the medical

treatment�, which is illustrated with a similar arrow leading to the three outcome groups of study A;

further, the shorter direction arrow between this stage and �to be prescribed medical treatment�

symbolises the contiguous and nearby relation. In the model, �to be influenced by the medical

treatment� and �to be on the path of learning to live with osteoporosis� are represented as


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41

impacting each other. The same relationship characterizes the theme of �to be on the path of

learning to live with osteoporosis� and the outcome group �early quitters� illustrating that treatment

may in some cases be resumed almost one year after diagnosis, as described in study B (paper

III), and also mentioned in study A (paper I). Another result of the process is a decision to

terminate the treatment because of discomfort and instead perform other preventive actions while

waiting for the next scan result (cf. paper III). The �persistent and compliant group� of study A, is to

be seen as a final stage of the decision related to implementing and maintaining the medical

treatment in light of coming to terms with living with the preventive medical treatment. �Being

persistent and compliant� theme had a mediating effect on �learning to live with osteoporosis� as

patients experiences found confidence in their reactions and ability to pursue the treatment (cf.

paper III).

�

�

�

�

�

�

�

�

�

�

�

�

�

�

The following discussion comprises selected results from the current study. They are discussed in

relation to one another based on the model (Fig. 5) incorporating literature and other research

Fig. 5 Model of study results within aspects of medical treatment and chronic condition

To be prescribed medical treatment Being

diagnosed

To be influenced by the treatment

�Early quitters�

Persistent with MPR<75%

To be on the path of learning to live with osteoporosis

Daily life with osteoporosis - as a chronic condition

Persistent and compliant with

MPR>75%


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42

findings related to persistence and compliance with anti-osteoporotic medical treatment,

acceptance/decision-making, chronic illness, coping and nursing perspectives.


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43

7. Discussion

The thesis provided information on life with osteoporosis based on quantitative and qualitative

study results. The discussion is carried out based on selected results from these current studies,

related to the model presented in Fig. 5 section 6.3. The discussion of the results is structured in

three parts: 1) risk of terminating treatment early; 2) acceptance; 3) decision-making, nursing

perspectives and learning to live with chronic illness. Subsequent is methodological considerations

discussed.

Central to the discussion of the results, were following areas chosen:

Aspects of medical treatment mainly related to the group of �early quitters�

Aspects of living with a chronic condition

7.1. Discussion of the results

The investigation showed how aspects of medical treatment are multifaceted, both in a quantitative

way as demonstrated in study A and in a qualitative manner of influencing life with osteoporosis as

shown in study B. An observational cohort study with a register-based approach, using the Danish

National Registers was chosen for study A. The hypothesis that patients who stop treatment very

early differ in socio-economic demographics, medication regimen and comorbid conditions from

other patients with poor compliance, was confirmed. It was also demonstrated that the

overwhelming health problem consisted of terminating treatment prematurely and not persisting

with treatment but having low refill compliance. In addition, were a phenomenological-hermeneutic

approach inspired by Paul Ricoeur's theory of narrative and interpretation conducted. The aim was

to gain insight into women�s experiences of living with osteoporosis and the evolvement of changes

during the first year after diagnosis. Learning to live with osteoporosis was found to be a

multifaceted process highly influenced by the medical treatment. Medical aspects obviously have

an important role in life with osteoporosis and maybe especially before known osteoporotic fracture

occurs, since the medication is concrete and have to be handled in one way or another. The

medical (pharmaceutical) treatment as a central issue in relation to osteoporosis has been found in

several other studies dealing with treatment required osteoporosis, initiation of medical treatment

as well as persistence and compliance of anti-osteoporotic medication (Roerholt et al., 2009;

Nielsen et al., 2012; Landfeldt et al., 2012; Weston et al., 2011; Kothawala et al., 2007). The thesis

adds knowledge of predicting factors demonstrated to increase or decreased the risk related to

three non-overlapping risk groups (cf. Fig. 3 in section 6.1.) of which mainly one group will be of

focus in the discussion together with aspects of the medical treatment�s influence on life with

osteoporosis during the first year after diagnosis (cf. Table 6 in section 6.2.).


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44

Risk of terminating treatment early

It was demonstrated that 38.7% of all new users of oral bisphosphonate beginning treatment

between 1/1/1996 and 31/12/2006 in Denmark stopped treatment within the first year. Thus, there

were 4.7% who were persistent but with suboptimal compliance and 56.6% of the patients were

persistent and compliant with treatment (MPR �75%) for more than one year; in comparison, a

two-year persistence as high as 84% - 88% was reported in a Danish survey from three different

clinics (Nielsen et al., 2010); overall these results may lead to the assumption that persistence and

compliance may be fairly good in Denmark compared with studies from other countries: in Norway

it is reported that adherence during a study period of 4.2 years were 45.4% (Devold et al., 2012); a

Swedish study reported 23% of osteoporosis patients stopped treatment early and 51.7% were

persistent with alendronate treatment after one year (Wastesson et al., 2013); another Swedish

study demonstrated about half of the patients stop treatment within the first year (Landfeldt et al.,

2012) and similar findings was confirmed from the USA (Siris et al., 2006). These results; together

with the finding of 38.7% early quitters in the current study, evident a general need to investigate

the reasons for stopping treatment early. The continued discussion will therefore concentrate on

this group. The finding of 38.7% patients stopping treatment within the first year together with the

observed significant improvement in compliance and persistence (time trend, cf. Table 5 in section

6.1. and paper I) in patients who began treatment in later study years compared with those who

began earlier could be related to the relatively strict criteria for reimbursement of treatment which

may select patients with a higher motivation for treatment, moreover may the reimbursement letter

from the Medicines Agency reinforce patient beliefs in the appropriateness of treatment in their

individual case. Further, more convenient dosing opportunities and the increased patient education

specifically targeting osteoporosis could be contributing to this trend. Finally, it is conceivable that

there may be some cultural factors to be considered together with the current findings of

socioeconomic and demographic factors, some co-medications and comorbid conditions to predict

increased or decreased risk of terminating treatment prematurely (cf. Table 5 in section 6.1. and

paper I), potential cultural factors, is not possible to indicate in the current study, further studies are

needed.

As mentioned, more convenient dosing and treatment options have been available in later years.

Anti-osteoporotic treatment compricing weekly oral bisphosponate are by far the most widely used

treatment for osteoporosis worldwide including Denmark. Terminating treatment early could

possibly be effected by the challenges regarding consuming medication on weekly basis, since

there is a risk of forgetting to take the medicine as it was found in study B; several initiatives were

described in the form of routines and strategies in order to remember to take the medication. This

was problematized consistently throughout the study as worries of forgetting to take the medicine.

Routines of medication intake is substantially when patients are prescribed oral bisphosphonate


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45

since treatment requires that the patient is able to comply with the special requirements for tablet

intake, requirements that may have an impact on persistence and compliance if they are found

difficult. Lau et al found that those patients who had difficulties with rearranging their daily routines

preferred to take the medication on weekly basis, and those who successfully had integrated taking

medication into their daily routines preferred the daily dosing option (Lau et al., 2008). It seems

plausible to think that routines and strategies of drug intake may be a factor contributing to our

finding in study A related to the comorbid condition dementia; dementia were found to predict

decreased risk of stopping treatment early; this assumption may be strengthening by the fact that

medication for individuals with dementia in many cases are assisted with co-administration with

help from others or prescriptions are being delivered to the home.

Another aspect of the group of "early quitters" and which also emerged in study B; is the fact that

some patients redeem the prescription and then put the medication aside; it is therefore argued in

paper I (study A), that one of the shortcomings of a register-based study, is that it is not known

whether the individual takes the dispensed drugs or to what extend they are using them, the

information derived from a register-based study is in this particular case only based on the date of

dispensing and dosage of drugs dispensed to each individual from pharmacies in Denmark only. In

analysis of anti-osteoporotic treatment it was demonstrated that treatment other than alendronate

was associated with an increased risk of stopping treatment early, but also a predictor of reduced

risk of subsequent low refill compliance. This may possibly be explained by physicians focus on

medical precaution related to for example pre-existing upper gastrointestinal symptoms. Further,

were prior use of co-medications as hormone replacement therapy, prednisolone and anti-

depressants found to be associated with increased risk of stopping treatment early, as well were

an increasing numbers of drugs used the year before index. Similar findings were found in the

Norwegian study (Devold et al., 2012).

Regarding comorbidity, Charlson comorbidity index overall and especially 3+ was found to

increase the risk of stopping treatment early specifically were comorbid conditions as pulmonary

disease and collagen disease, as well as dementia mentioned earlier; these findings are in line

with general findings in the literature of comorbidity predicting decreased persistence and

compliance (Landfeldt et al., 2012).

Less than half of the population, about 40% had a prior hospital treated fracture; this low number

may be seen as supporting the appropriateness of conducting a study in line with study B, as a

large proportion of patients treated with osteoporosis medications may be considered not to have

had a known osteoporotic fracture (Landfeldt et al., 2012; Nielsen et al., 2010; Siris et al., 2006;

Wastesson et al., 2013). Statistically, it was surprisingly found that a prior fracture in general, had a

tendency to increase the risk of terminating treatment early. However, fracture of the forearm was

found to predict a lower risk of stopping treatment early. These findings may be related to patients�


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46

motivation to begin treatment after having had a fracture; since, it may be presumed that patients

who begin treatment after forearm fracture � often the first osteoporotic fracture experienced - may

be more motivated than those who had a more potentially dangerous fractures such as those of

the spine and of the hip before beginning treatment. However, further research is necessary to

obtain deeper understanding of this phenomenon; no other studies, investigating risk of premature

termination of treatment or low refill compliance and the association with pre-existing osteoporotic

fractures were found.

Highest income level was found to be associated with a decreased risk of stopping treatment early.

Since the health care system in Denmark is financed by taxes with free access to healthcare for all

citizens, this finding were not initially expected, although reimbursement of osteoporosis drugs is

typically only 50% of the price of the medication; reimbursement of osteoporosis drugs is not

universal in Denmark, other mechanisms of income level than medical expenditures may also be

taken into account. It is possible to assume that income level to some extent may serve as an

indicator of individual health beliefs and perceptions; this assumption may be strengthen by the

findings in a Canadian study of patients with fragility fractures which were found to interpret

information concerning osteoporosis in ambiguous ways (Sale et al., 2010); as well as by findings

in a systematic review of health beliefs and preventive health behaviour of osteoporosis (McLeod

and Johnson, 2011). In addition, the current study did not demonstrate any statistical significance

of household income in predicting low refill compliance after treatment was established; this finding

are somewhat supported in a Norwegian investigation of the influence of socioeconomic factors to

alendronate treatment of osteoporosis (Devold et al., 2012). Furthermore, in study B it was not

highlighted that drug costs was an issue, however, household income did not appear in this study;

although, most women were retired and almost half of the women lived alone (cf. Table 3 section

4.2.1.). Further research is needed to gain a deeper understanding of these issues.

Regarding age at treatment, reduced risk of stopping treatment early were statistically associated

with 2ond age quartile (70.2 � 71.8 years) and 3rd age quartile (71.9 � 79.0 years); the Norwegian

study reported higher odds of adherence for women �60 years of age (Devold et al., 2012),

meanwhile both the Swedish study and the Danish survey did not find any association between

age and persistence and compliance (Landfeldt et al., 2012; Nielsen et al., 2010).

An association with housing condition were found. Living in apartment were found to increase the

risk of stopping treatment early; several explanations may be speculated but cannot be clearly

identified in a register based study. It is possible to imagine that this finding may describe

differences in the quality of health care in various types of housing, such as general practitioners,

home care schemes and other municipal systems; or simply that individuals of the same sex,

income, age and education might function in different ways: socially, mentally, health belief and in

relation to social support. Only one of the three women in study B, who probably could be


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47

considered to be �included� in the risk group of �early quitters� lived in an apartment (cf. Table 3

section 4.2.1.).

Young adults up to age 25 � children living at home, were included in the analysis and found to

increase the risk of terminating treatment early for the few patients with children still at home and

therefor maybe have to be seen as a chance finding. Only one prior study was found addressing

the predictive impact of children in the home; the cross-sectional study of the National Health and

Wellness Survey of women with osteoporosis or osteopenia; the researchers did not find any

association related to children <18 years at home (Meadows et al., 2007). None of the women in

study B had children living at home; otherwise it may be in qualitative studies with focus on the

individual perspective that would be able to provide insight to the mechanism of children living at

home.

Living in the countryside or in a village were found to predict a slightly lower risk of stopping

treatment early but also a factor associated with increased risk of persistence with low refill

compliance; these findings were somewhat in contrast to findings in the Swedish study (Landfeldt

et al., 2012). Further, it was found that proximity to a university hospital were associated with a

noteworthy increased likelihood of stopping treatment early. Together, these findings may be

considered as contributing to the findings but as so far not explained proxy to refill persistence and

compliance, since explanations cannot be identified in a register based study. Treatment

thresholds could be lower for patients with easy access to specialist care and this may lead to

patients with lower motivation beginning treatment. Further studies are needed, no other study

investigating the association between risk of stopping treatment early and proximity to a university

hospital were found.

Lau et al argues that strategies to improve persistence and compliance to anti-osteoporotic

medication should be individualized, since reasons for non-compliance may be different for each

patient related to individual perceptions and experiences as well as individual beliefs and

circumstances (Lau et al., 2008). They did comparatively in line with the current study B; find that

some women believed that medical treatment should be the last resort due to the women�s belief

that lifestyle modification would be enough to prevent osteoporosis.

Acceptance

To be prescribed medical treatment and initiation of the medical treatment seems to be influenced

by the acceptance and understanding of the diagnosis, and understanding of being at risk of

osteoporotic fracture, as it appeared in study B.


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48

In a grounded theory study of healthy risk awareness the authors argues that the acceptance of

the new condition of being a woman with fragile bones is the first phase of the emotional and

cognitive process of being motivated for acting in a preventive way (Hjalmarson et al., 2007).

Similar findings occurred in the current study B; where it was found that acceptance in many cases

were a struggling process characterized by thoughts and worries, as indicated by alternation

between the two levels of �being influenced by the medical treatment� and �learning to live with

osteoporosis� as illustrated in Fig. 5 section 6.3. Hjalmarson et al found that difficulties of

acceptance did undergo a development through the fear created by these difficulties. Their

interpretation is that fear impedes learning, which may lead to absence of risk awareness,

increased fear as well as physical inactivity and social isolation unless the desire to overcome the

fear is used to initiate a learning process that facilitates physical and social activities (Hjalmarson

et al., 2007). In the current study B; the women expressed fear and worries of making the right

choice related to medical treatment and for some were fear and worries related to lifestyle also an

issue; fear of what if breaking something were also present together with the desire of living as

normal as possible, which were found to be handled in a way of thinking of strategies and adaptive

actions, testing these actions and reject some but also incorporate others in everyday life, which

may be seen as a way of using the fear to adapt and generate risk awareness as found by

Hjalmarson et al. Acceptance described as developing strategies is also found in the study of Lau

et al, they found strategies to be as when the patient has accepted the need of treatment, they

devised strategies according to taking the medication, strategies that worked best for the individual

patient according to their daily routine (Lau et al., 2008). These findings was consistent with the

findings of Weston et al, who found that strategies was a way of taking control of feared negative

outcome, to be careful not to fall and break a bone and to keeping things in perspective and

focusing on the moment rather than look into the future, but also as taking the medication and to

manage emotional impact of the diagnosis (Weston et al., 2011). In addition, has several studies

reported that the relationship with the physician and other healthcare professional has an important

impact on patients persistence and compliance to medical treatment (Balkrishnan, 1998; Barat et

al., 2001; Block et al., 2008; Dimatteo, 2004a; Zolnierek and Dimatteo, 2009; Martin et al., 2005);

in the current study B, the importance of the relationship with the physician was widely described in

relation to the process of being diagnosed; it was found that the women�s basic trust towards the

physician and the health care system in general in some extend were mediated through

experiences of not feeling taken seriously or cared for; further, were lack of sufficient information

related to the diagnosis and thoughts and worries about the medical treatment found to effect the

acceptance of the diagnosis and the understanding of being at risk of osteoporotic fracture. These

findings are in accordance with other studies, for instance did Lau et al found that trust in the

physician was a key factor for patients to accept osteoporosis medication, further they found the

information given by health care providers was lacking and not always on the level or of the extent


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49

which was needed (Lau et al., 2008). In addition, has Martin et al reported in a major review of

patient adherence; that the physician�patient partnership are the core when choosing amongst

various therapeutic options to increase adherence to medical treatment; this is due to the

significance of engagement a mutual collaboration in which patients take responsibility, which has

been found to foster greater patient satisfaction, reducing the risks of non-adherence, and

improving the health care processes and outcomes for patients� (Martin et al., 2005).

The information and communication in a physician�patient relationship are naturally influenced by

many factors, it is crucial that the patient is cognitive able to understand and receive information,

attitude and willingness for cooperation is also essential. Health care professional communication

skills and credibility has been found in several studies to impact on decision-making related to

medical treatment (Mazor et al., 2010; Lewiecki, 2010) as well as being at focus in relation to

shared decision-making (McHorney et al., 2007; McHorney and Spain, 2011; Weiss et al., 2006;

Yood et al., 2008).

The need for information related to osteoporosis may be particularly necessary because of the

invisibility of the condition (Reventlow et al., 2006; Weston et al., 2011; de Souza et al., 2010). In

the current study B, the invisibility was found to be a factor related to the acceptance but also as an

argument for the need of information and understanding of the scanning result, in some cases the

women had had a print of the scanning result visualized by numbers reflecting the bone calcium

content. These numbers are mapped on a curve in a diagram with green, yellow and red collars

and are compared with the mean within the same age group as well as that of young healthy

adults, combined with a small radiographic picture (of the hip and/or back); most women had

difficulties of understanding and interpreting the scanning result and expressed confusion instead

of clarity. This may be contrasted to Reventlow et al findings when incorporating the visualization

of scanning result in a study of women�s bodily experiences of osteoporosis diagnosis; the

researchers found that a visual image of the women�s bone loss formed their body image in a

cultural interpretation of osteoporosis as having a fragile and easily damaged body, which drew the

attention to avoid fractures� her and now. The authors argue that the women's experience of bodily

sensations together with their interpretations of these experiences was seen as the process of

osteoporosis (Reventlow et al., 2006). This may be an example of the significance and benefits of

a professional interpretation communicated by visualization of osteoporosis; and as such, it may be

assumed to be reflected and elaborated based on the findings in the current study B, together with

the findings of Weston et al who reported a significant barrier of being able to fully accept the

diagnosis was the invisible and asymptomatic nature of the disease when the women received a

verbal diagnosis from their GP, which seemed to make it difficult to give meaning to the condition

(Weston et al., 2011).


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50

Decision-making, nursing perspectives and learning to live with chronic illness

Acceptance emerged to be a mediating factor of decision-making related to anti-osteoporotic

treatment in a manner of worries and uncertainty as well as difficulties of deciding on whether to

pursue medical treatment and thoughts of making the right choice; this leads to the conceivable

argument that the acceptance and understanding of the diagnosis impacts on the decision-making

related to the medical treatment and life in general when living with osteoporosis in ways as the

illustration in Fig. 5 section 6.3. is suggesting.

Other studies investigating decision-making in relation to osteoporotic treatment has also found

this to be a process; Hjalmarson et al found the decision-making process to be in relation to an

interaction of emotional and cognitive learning process of being aware of having osteoporosis

when developing a healthy risk awareness, which were considered to be a key to fracture

prevention (Hjalmarson et al., 2007); in continuation of this finding Weston et al found that the

women did go through a sense-making process of believing that osteoporosis is treatable (Weston

et al., 2011); Lau et al demonstrated that the women were going through a process of trial and

error to find strategies that worked best for them (Lau et al., 2008). Among others, these qualitative

studies which claim to investigate processes related to osteoporosis, medical treatment and beliefs

and likewise, seems to commonly not investigate the process in longitudinal study designs (as it

was chosen in relation to study B); despite this limitation the study of Sale et al investigated

patients actual decision to take prescribed osteoporosis medication after having had sustained a

fragility fracture, the interviews were based on recall and predictions about future behaviour. Out of

the 14 patients taking bisphosphonate and 7 patients not taking osteoporosis medication, it was

found that for 12 patients the decision regarding the medication were easy without profound

reflections and occurred at the time of prescription mainly based on trust towards the health care

professional (10 had decided to take osteoporosis medication). Those who experienced difficulties

of the decision were worried about side effects, sought information from different sources and were

engaged in risk-benefit analysis and did not feel convinced by their health care provider (Sale et

al., 2011). These findings is somewhat in line with the current study where influencing factors of

decision-making were found to be feeling taken seriously and cared for, trust and fear as well as

the development of an awareness of the impact of osteoporosis on social life and life in general.

Moreover the authors found that more than half of the patients indicated they might changes their

decision of taking or not taking medication (Sale et al., 2011). This later finding may together with

findings in the current study strengthen the assumption that the decision of initiating medical

treatment is multifaceted and therefor for some a matter of an immediate decision and for others a

process of a decision made over a longer period of time.

There seems to be some natural lines that can be drawn from the current study to the salutogenic

model of health developed by Antonovsky (Antonovsky, 1979). He was occupied with the question


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51

of how humans survive, adapts and overcome life-stress experiences by having a strong focus on

how humans manage to stay well, despite a lack of control in their lives. This was conceptualized

in the �Sense of Coherence� (SOC), which is strongly related to perceived health, in particular

perceived mental health (Eriksson and Lindstrom, 2006). SOC is a sort of life-orientation and has

been operationalized in three components: comprehensibility, reflecting the degree of life events

experienced as rationally understandable, ie. an inner and outer information is orderly, coherent

and structured in a predictable way; managreability, reflecting beliefs of having necessary

resources available when meeting the requirements in life, which are manageable and within

control; and finally meaningfulness, reflecting the inner experience of being motivated by problems

and demands since life is predominantly interesting and worth fighting for (Antonovsky, 1979).

To accept a life with osteoporosis, to adapt and to make decisions in relation to these new life

circumstances may be seen as a learning process filled with stressors in a salutogenic perspective.

According to Antonovsky, SOC develops from a variety of learning processes throughout life.

Besides SOC in itself probably increases the quality of life; Antonovsky hypothesis SOC to be

relevant to human health behavior, due to humans with high SOC seems to be less likely to enter

situations that causes health threatening stress, and they appears to change habits easier when

indications of health problems arises (Antonovsky, 1979). The women in the current study was

found to undergo a process of interpreting and finding meaning of their diagnosis and the medical

treatment; Antonovsky would probably say that a cognitive process of comprehension, managing

and meaning creation were activated as an ongoing development of SOC in life circumstances of

osteoporosis, this were reflected in the findings of the women�s concerns and need of information

related to handle the stressors of acceptance and decision-making.

In addition, did Hjalmarson et al draw a line to the salutogenetic perspective: between the cognitive

learning process of healthy risk awareness, viewing fear as a stressor when accepting the risk of

bone fragility (Hjalmarson et al., 2007).

It is argued that nursing is implicit rooted in the salutogenic orientation while caring according to a

specific health problem is considered as strengthen resources for the patient when coping with

stressors according to illness and well-being (Benner and Wrubel, 1989; Sullivan, 1989). The value

of nursing is to pay attention to pain and suffering as a valuable purpose in itself; to listen, to

confirm, to guide and to comfort is ways of relating to the human responses and provide a

respectful and caring atmosphere (Benner and Wrubel, 1989). Further, Benner and Wrubel

emphasizes, that nurses is in a unique position, of being able to understand illness experiences

and the meanings of the experience supplied by the patients. Nurses establish a healing relation to

the patient, by helping with mobilizing hope and active healing using the social, emotional and

spiritual resources available; this helps the patient to form the illness experience and overcome


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52

stressors due to illness and gain well-being, through nurse led guidance, interpretation and

�training� of possible ways of approaching the unknown context (Benner and Wrubel, 1989).

Benner and Wrubel argue that a phenomenological understanding of health must incorporate what

one is and what one can become. It must be based on an integrated view of mind, body and soul,

and shall be based on the opportunities that the situation offers. The intention of caring science is

to uncover the subjective understanding of lived experience of the patient's; therefore, it is

preferred to use the term well-being rather than health because well-being includes the lived

experience; thus, they takes the view that nursing perspective should be focused at stress and

coping based on the patient's personal experience related to well-being and when basis for health

behavior changes are present, consideration should be given according to the person it relates to,

experiencing the changes meaningfully and integrates them within the social contexts (Benner and

Wrubel, 1989). Health and disease is viewed as a whole and as continuous instead of in a

dichotomous sense (Antonovsky, 1979; Benner and Wrubel, 1989). With the focus: what keeps

individuals healthy, rather than on what makes them ill; what strengthen health and sustaining

competences to act in order to sufficiently manage the challenges of everyday life (Antonovsky,

1979; Sullivan, 1993; Sullivan, 1989), hereby it is believed that every individual person to some

extent, possess personal and healing resources regardless of the current state of illness and well-

being.

Challenges of everyday life with the often invisible (de Souza et al., 2010; Reventlow et al., 2006;

Weston et al., 2011) and inherently chronic condition osteoporosis has been the subject of some

research; since the interest in health promotion and reduction of risk factors for chronic diseases in

healthy populations has expanded (Reventlow and Bang, 2006; Wilkins, 2001b; Roberto and

Reynolds, 2001). Based on the findings of the women�s difficulties of coping with challenges in

everyday life with osteoporosis which might conceivable be influencing on persistence and

compliance and may be speculated to be some of the reasons of individuals terminating treatment

as illustrated in Fig. 5 section 6.3. To learn to live with new life circumstances due to osteoporosis

seems to be a cognitive process of grasping the comprehension, manageability and gain

meaningfulness challenged by stressors in a salutogenic perspective.

A picture of everyday life with osteoporosis seems to be added up when current findings of

learning to live with osteoporosis as a prolonged process, in accordance with similar findings like

challenges of emotional handling the knowledge of the fracture risk (Nielsen et al., 2012); the self-

concept as the way of thinking of oneself and prepare for the future (Roberto and Reynolds, 2001);

and the diversity of self-concept related to meanings of ageing and chronic illness which providing

an understanding of the strategies chosen to manage life on a day-to-day basis (Wilkins, 2001b). It

seems to make sense to complete the picture of the understanding of everyday life with

osteoporosis by adding theoretical reflections of osteoporosis as a chronic condition; since life with


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53

osteoporosis appears to generate recognizable patterns which may be equated with many other

chronic conditions. Living with a chronic disease may initially cause feelings of being different.

Finding new meaning in life with chronic illness and suffering can be a demanding and threatening

process related to the changed life situation; this seems to take time to adjust. The suffering, is a

phenomena of life and the emotional life is an important part of meaningfulness when chronically ill

(Sonder et al., 2007). Based on theoretical reflections Morse has developed the theory

�Responding to Threats to Integrity of Self�, which is a model based on a need for a more general

theory of the experienced processes of recovery following the threat of illness or injury, with focus

on the meaning and changes to the person-self rather than on a particular illness, disease process

or the disruption to physical health (Morse, 1997). The model is defined to comprise five stages

based on commonalities despite the onset of illness: Vigilance; Disruption: enduring to survive;

Enduring to live: striving to regain self; Suffering: striving to restore self; and Learning to live with

altered self. This model is meant to be seen as a revision and combination of the two former

models �The Illness-Constellation Model� developed by Morse and Johnson (Morse and Johnson,

1991) and �Preserving Self� developed by Morse and O�Brien (Morse and O'Brien, 1995). The

process of recovery is to be seen as mutable throughout the five stages in varying degrees for

individuals experiencing threat to integrity of self. In relation to the current study where

osteoporosis often is a condition without symptoms and �warning signs� the diagnosis may come

as a surprise. Vigilance (activated by symptoms) of a �threat to integrity self� always comes as a

surprise when the symptom has been silent and unnoticed, with a feeling of the body had betrayed

the person, as the body moments ago were considered healthy; this makes the individual more

aware of the body, symptoms and of the self. Safety feelings related to the caregivers

competences, vigilance and trustworthiness, is required before the individual will be able to accept

and receive care (Morse, 1997). This may be some of the explanation of why being taken seriously

and taken care for appears as an important factor in life with osteoporosis. The ordinary life is

interrupted and as Morse describes this disruption (enduring to survive) a situation of �holding on�

arises and the individual anchors to significant others which will be a sort of buffer for unnecessary

worries and protecting the person (Morse, 1997). In the current study, this stage is specifically

recognized in relation to those women who had daily pain of the back, and were vaguer in relation

to those without or with few physically symptoms, which might be due to some difficulties of talking

with spouses about the mental impact of osteoporosis. The latter individuals seemed more to try to

bear it and very often alone without particular interference of spouses. Enduring to live (striving to

regain self) is when the individual is trying to learn to take it by developing strategies, suppress

emotions and when appearing to accept what had happened. Considerations of the implications of

the illness are made and the individual are moving towards suffering and maybe planning for the

future. Relatives may still play a significant role (Morse, 1997). Accepting and adjusting to live with

osteoporosis appeared, in the current study, as a varying and sometimes a lengthy process. In


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some cases it was found to be a process of to pull oneself together and convincing oneself of that

it will be okay, as well as thinking out strategies to ensure this. Worries of a future life with

osteoporosis occupied some and seemed not to be a subject to others. When an individual

becomes able to acknowledge the illness a struggle related to losses and altered future begins as

a suffering (striving to restore self); emotions are being handled and blaming, guilty feelings and

experiences of despair will occur. This is the healing stage, with new hopes and realistic goals;

suffering is resolved through the work of making sense of the experience (Morse, 1997). The

healing process, in the current study, was in some cases found to be clear and pronounced when

struggling to accept and sometimes seeking explanations through interpretation of symptoms and

life style reflections, often together with testing different adjustments to life with osteoporosis. For

others the acknowledgment of osteoporosis was sort of put aside due to being occupied with other

things. Adjustment to the new life circumstances with limitations by trusting the altered body is

basic when learning to live with altered self; acceptance of the experience will develop and the

suffering will be put aside (Morse, 1997). In the current study most women expressed a pride of

leading an independent life, at different time during the first year after diagnosis, they all described

ways of coming to term with a life with osteoporosis.

Morse argues that by moving away from medical diagnosis and focusing on the individual

responses, the model provides a more useful framework for patient care (Morse, 1997).

In a future perspective of prevention and health promotion initiatives of the growing public health

problem osteoporosis, it might be beneficial to include perspectives of risk assessment and

handling everyday life with osteoporosis, as the current study.

7.2. Methodological considerations

The twofold method consists of two distinct phases. In one of the phases, the qualitative data

provided an enriched and deeper understanding of life with osteoporosis, thereby complementing

and helping explain the quantitative data and vice versa (Sandelowski et al., 2006). The interviews

were conducted before and along with the statistical analysis, which means that the results of the

quantitative data were not available to guide the interviews or the other way around. The findings

from both the quantitative and qualitative study were interpreted separately and thereafter

discussed.

In this section, methodological considerations of study A will be discussed followed by discussion

of the methodological considerations regarding study B.


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7.2.1. Methodological considerations of the quantitative study A

In this section, methodological considerations of strengths and limitations of study A will be

discussed based on the STROBE recommendations (Vandenbroucke et al., 2007).

The study comprised of 100,949 individuals (84.8% female) with primary osteoporosis. The cohort

consisted of those patients who filled at least one prescription of oral anti-osteoporotic drug. It is

therefore necessary to keep in mind that the study findings may not generalize to all individuals

diagnosed with osteoporosis in Denmark in the same time- period, since not all patients with

osteoporosis are prescribed medical treatment due to various reasons, such as other risk factors or

more severe comorbidity (Kanis et al., 2013; World Health Organization, 2003b). Untreated

patients with osteoporosis are difficult to identify through health databases since the disease is

heavily underdiagnosed and under coded - most physicians do not assign ICD codes for

osteoporosis to a patient they do not prescribe treatment for (Abrahamsen, 2010). In addition,

some patients who are prescribed medical treatment may choose not to fill their prescription (Siris

et al., 2006; Roerholt et al., 2009). These patients are not included in the study and this may

underestimate the overall extent of low compliance and persistence to treatment.

In order to estimate the risk of low refill compliance and persistence, three non-overlapping

outcome groups were defined based on refill compliance (cf. section 4.1.2.).

A critical MPR value of 75% was predefined as the lower limit of acceptable refill compliance, since

compliance commonly is calculated as MPR �75% or �80% (Siris et al., 2006). An MPR value of

�75% were chosen for the study, because the fracture probability has been found to greatly decline

from MPR 75% and over and the probability of fractures also declines but only confined for MPR

values between 50% - 75% (Siris et al., 2006). In addition, most bisphosphonates are sold in

packages of 3 months and a delayed redemption of one packet would easily bring the annual MPR

below the 80% mark (Roerholt et al., 2009).

Persistence of treatment was defined as duration of time maintaining an MPR �75%. Patients were

allowed to have gaps between filled prescriptions, but were defined as non-persistent if they had a

gap of more than 56 days (8 weeks). Gaps of refill compliance are commonly used in analysis of

persistence of treatment (Landfeldt et al., 2012; Siris et al., 2006; Cadarette and Burden, 2010).

Treatment persistence was estimated by Kaplan Meier survival analysis (Barker, 2009; Silva,

1999). Sensitivity tests were performed to test different outcomes by using time gaps of 84 days

(12 weeks) and 56 days (8 weeks) and the length of persistence with treatment was found to be

fairly insensitive to the length of the gaps and comparable to the study of Landfeldt et al (Landfeldt

et al., 2012). It is also found that treatment may be reinitiated even after extensive gaps (Cadarette


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and Burden, 2010). Gaps of treatment could be due to hospitalisation where the patient would be

supplied with medication from the hospital, however, the hospitalisation even in relation to severe

hip fracture would only in few cases be longer than 56 days in Denmark. Therefore, the gap of 56

days was chosen as a measurement.

A register-based study has advantages and some shortcomings. The benefits of a register based

study (which also applied to the current study) were: long follow-up, good external validity, low cost

and less labour intensive work (compared to prospective study design) as well as the ability to

include patients of all ages, with multiple complications and various socioeconomic backgrounds.

Further, a major advantages of register based studies is that patients are not affected by the study

and do not change their behaviour because they are being studied. Because of these benefits, the

register based approach has been commonly used in studies of prescription patterns

(Abrahamsen, 2010; Landfeldt et al., 2012; Furu et al., 2010; Hallas and Stovring, 2006; Gliklich

and Dreyer, 2010).

Register based studies also have some shortcomings. Limitation of the current study was that it

was not possible to know if and to what extent the patients took the dispensed medication,

although the dispensing date and drug dosage for each patient was available. Thus,

overestimation of the medication use is a possible bias of this study. On the other hand, an

underestimation is also possible, since information about medication obtained through hospitals,

outpatient clinics and other institutions was not available for this study, and neither was information

about medication purchased abroad. These types of issues normally affect the results, since they

impact the control of exposures and thereby the internal validity of the results. However, the

impression in the current study is that this may be a minor issue due to the large study population,

since the effect is considered to be of a lesser extent and since it is possible that the

underestimation and overestimation of medication refill will almost offset each other. Nevertheless,

these are possible biases. Another limitation is in regards to the information about comorbid

conditions when diagnosed in general practice and when the patient did not need an in- or out-

patient hospital treatment. These diagnoses are not registered in national registry. This bias was

addressed through indirect information about comorbid conditions through obtaining of all filled

prescriptions issued by general practitioners.

The lacking socio economic data (education level and household income) for 131 individuals were

addressed by preforming two sensitivity tests (cf. section 4.1.2.). Missing data generally affect the

internal validity of the study, but since these sensitivity tests yielded the same conclusions as the

primary analysis and since the individuals with missing data only comprised 0.1% of the large

study population, the conclusion was that the internal validity was not affected.


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There are other variables that could have been of interest for the study but were not available

because of the register based approach. For instance it would have been interesting to control for

bone mineral density, life style factors or patients health beliefs, but this was not possible.

The generalizability of a study is a very important issue, since the accuracy of prognostic models

tends to be lower when applied to data other than those used to develop them (Vandenbroucke et

al., 2007). Any explanation for an association between risk groups of low compliance and

persistence in relation to socioeconomic demographics, concurrently-taken medications and

comorbid conditions may be conjectural. No published studies on risk groups were found, although

other epidemiologic studies with relevant results of low compliance and persistence were found.

The results may be generalized to individuals with primary osteoporosis in Denmark and probably

also to other comparable countries. The strength of the large health database was in providing the

�real-world� validity for prediction of low compliance and persistence to osteoporosis medical

treatment. Despite the population size and the extensive observation time of 520,604 person years

(mean time of follow-up 5.2 years), more evidence on the subject is needed, particularly because

of the lack of comparable evidence.

7.2.2. Methodological considerations of the qualitative study B

A phenomenological-hermeneutic approach inspired by Paul Ricoeur's theory of narrative and

interpretation were chosen to gain insight into women�s experiences of life with osteoporosis; a

longitudinal design was chosen to explore the evolvement of changes during the first year after

diagnosis. The methodology allowed entering the women�s subjective world of osteoporosis

through their stories of experiences; thus, the opportunity to explore their main concerns when

adjusting to live with osteoporosis was achieved. In this section, I will discuss the considerations of

strengths and limitations of the qualitative study.

The qualitative research process has constraints and requires continuous choices. Different

choices would therefore lead to different perspectives which would generate different empirical

data and results. The qualitative interview in this study allowed for open, nuanced descriptions of

various aspects of women�s life after the diagnosis when medical treatment was prescribed. The

appearance of the phenomenon �life with osteoporosis� was enabled; in-depth aspects of this

phenomenon would not have been captured by using other methods such as e.g. a questionnaire

survey.

The participating women were included according to inclusion and exclusion criteria, ensuring that

they all were diagnosed with osteoporosis and were eligible for the study. The women were


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consecutively included, to ensure a comprehensive and purposeful sample. They were ordinary

women who were good at describing their experiences of living with osteoporosis. The study

comprised of 15 informants. This limited number is seen as a strength of the study, since an

excessive number of informants in a qualitative study may lead to superficial results without an in-

depth investigation (Kvale and Brinkmann, 2009; Malterud, 2001). The longitudinal design is

considered to provide a thorough exploration of the research question (Saldaña, 2003). Most

interviews took place in the women�s homes which could have affected the interview results

through my role as a nursing-researcher (Hall, 2001). However, my role also may have been

diminished by conducting the interviews in the women�s homes, as I was a guest in their homes

(Hall, 2001). Further, interviews conducted at the informants� homes may enrich the information

gained through the interviews. Herzog reported finding a distinct difference in the story depending

on whether the interview was conducted in the informant's home or elsewhere (Herzog, 2005).

According to Holloway (Holloway, 2007), a relaxed environment when telling about personal

experiences could conceivably be affecting the women�s own interpretation and understanding

gained when they were telling stories of their experiences of osteoporosis. Further, it is possible to

think that the interviews would lead to therapeutic effect it self, which obviously should lead to the

question of the connection between the participant�s reality and the study results, in other words

whether the findings were �real� and �untouched�. No qualitative study is without the �touch� or

influence of the investigation itself or the researcher as an �interacting� individual. When doing

qualitative research, it is crucial to reflect upon possibilities of how the researcher affects the

process or whether such an effect can be prevented, both during data collection, during analysis

and interpretation (Malterud, 2001). During interviews I sought to be aware of my own

preconception, not posing leading questions but giving the women the opportunity to tell their story

and give them time to pause during the storytelling, before I encouraged them to elaborate or tell

more about something they had mentioned with the purpose of unfolding the meaning attached to

the individual experience. This was a challenge, even though I had some experiences with

performing interviews. It was a learning process as a researcher, which is also reflected in the

interviews, as the later interviews largely and exclusively comprise the women's stories. Also, we

became acquainted with each other, since I interviewed the women three times. I saw this as an

advantage when conducting telephone interviews at the time of the third interview. These

interviews were naturally shorter, but did not leave the impression of something not told. The

women were interviewed at three predetermined times: shortly after diagnosis, six month later and

approximately one year after diagnosis. It is worth reflecting upon these points of time where the

interviews were conducted. Would the results have been significantly different if the time of

interviews had been in shorter intervals or concentrated specifically on the initial faze? Hard to tell,

but as it was found in the current study, the process of adapting to life with osteoporosis, was


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different among the women. Although, the three predetermined times of interview were chosen

since studies have shown individuals compliance to treatment stabilizes about six month after

medical treatment is initiated (Lindberg et al., 2008; World Health Organization, 2003a) and since

adjustments to living with a chronic condition often take a long time period (Holloway, 2007). The

longitudinal design made it possible to detect the developmental and changeable aspects of the

findings.

Analysis and interpretation were discussed with fellow researchers/supervisors during all three

levels of the interpretation to enhance trustworthiness. Involvement of multiple researchers is

recommended when conducting qualitative research, as this might strengthen the design of a

study. During the analysis process and interpretation multiple researchers may supplement and

contest each other�s statements which may enrich and qualify the analysis (Kvale and Brinkmann,

2009; Malterud, 2001). It is crucial to present the prerequisites for analysis and the results, to make

the process transparent in order to inform the reader of the perspective of being able to understand

why the presented interpretation appears as the most relevant. To address this argument, the

analysis and interpretation attempted was made transparent and comprehensively described in the

method and the process in section 3.2 and 4.2. Further, the interpretation of meaning was, in a

sense empirically tested by the longitudinal design, since the women had the opportunity to

deepen and expand their stories three times during one year. The informants were not asked if the

interpretation was correctly perceived, they were asked to elaborate on issues they had mentioned

or talked about earlier. Findings were subjected to a type of empirical elaboration, through the

longitudinal design with several interviews; this in combination with the connection between

research question and methodology are described by Morse et all as responsiveness (Morse et al.,

2002), similar strategies of verification are Malterud describing as reflexivity and transferability

(Malterud, 2001).

The aim of this qualitative study was not to generate generalizable results, but to explore a specific

area that was unknown. However, the findings have gained strength from the international

research literature by coinciding with the project�s findings. This points to the results being likely

transferable to similar contexts such as women with osteoporosis in Denmark and maybe other

Western countries, as the methods used are broadly recognized and well documented.

Transferability to women with other �invisible� conditions such as hypertension, type II diabetes and

hyper cholesterol, might be an option, but further studies would be required.


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8. CONCLUSION

Two methodological approaches were used in the studies described in this thesis. The overall aim

was to attain an increased understanding of patients� life with osteoporosis. There were two

objectives: to quantitate the magnitude of the problem of low refill compliance and persistence and

investigate patterns and causes in a population of new users of anti-osteoporotic treatment in

Denmark and to explore newly diagnosed women�s experiences of living with osteoporosis after

pharmaceutical osteoporosis treatment for fracture prevention had been prescribed.

New risk groups based on refill compliance and persistence were identified - these may be

candidates for targeted actions. The findings highlighted the pronounced health problem of

patients� premature termination of pharmaceutical osteoporosis treatment: the group of those

patients who remains on treatment but have poor refill compliance were found to be a smaller

group although also important to identify for fracture prevention. These results leading to

suggestion of health preventive actions supporting patients to facilitate the interpretation of the

disease and the fracture preventive therapy, in order to reduce the risk of suboptimal or termination

of treatment to increase fracture prevention. The study adds new knowledge useful to identify

subgroups for targeted action by subsequent intervention studies. Further research including

socioeconomic and demographic differences in osteoporosis treatment and prevention are needed

to identify the underlying pathways.

Additionally, it is concluded that women handle osteoporosis in different ways, depending on the

interpretation of osteoporosis; acceptance and adjustment to a life with osteoporosis, which is a

multifaceted process highly influenced by the medical treatment both in relation to healthcare

professionals/healthcare system and decision-making of the pharmaceutical therapy, as well as in

relation to other related life events and experiences. Lived experiences of osteoporosis were

described based on the emerging key themes which were: �being diagnosed�, �being prescribed

medical treatment�, �being on the path of learning to live with osteoporosis�, �to become influenced

by the treatment� and �daily life with osteoporosis�. The study adds new knowledge and enriches

the understanding of life with osteoporosis which may be useful for healthcare professionals when

supporting individuals with osteoporosis. Further research is needed to explore specific

components of the multifaceted process in-depth, for instance the decision-making process,

interpretation of self-experienced side effects or discomfort of the medical treatment, adaptation

and life style changes.

Findings based on two different study approaches can be beneficial when investigating life with

osteoporosis as illustrated in the model �Daily life with osteoporosis � as a chronic condition�.

Similarities were found and lines were drawn between the three risk groups of refill compliance and

persistence in the quantitative study to themes emerging from qualitative interviews regarding


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women�s experiences of living with osteoporosis. Confirmation and similarities were shown when

incorporating former research findings. This lead to the conclusion that health preventive actions

targeted at osteoporotic fracture prevention would benefit from incorporating findings from this

thesis.


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9. PERSPECTIVES

In this thesis life with osteoporosis was studied by using a quantitative and a qualitative approach.

Several factors of varying degrees are indicated to be significant and impacting on life with

osteoporosis and the adjustments to live with this preventable chronic condition.

Since findings are obtained from a large population-based study and from a longitudinal study on

an individual level, it is the impression that these findings may be useable indicators of the

direction of development of new interventions targeting osteoporotic fracture prevention. The

findings suggest that interventions should be targeted on a joint healthcare professional level and

the individual patient level to be able to specifically support the individual patient according to

difficulties of everyday life with osteoporosis.

In a future perspective of prevention and health promotion initiatives of the growing public health

problem of osteoporosis, the study may contribute by below-described suggestions.

Interventions developed with inspiration from Wagner�s Chronic Care Model (Wagner et al., 2001),

(which is based on a rewarding cooperation between informed active patients and well-prepared

healthcare teams) could be implemented and tested in accordance with the need for patient

support. The Chronic Care Model is a widely adopted approach to improving outpatient treatment

around the world and is proven to support well-considered decision-making within healthcare

systems worldwide (Coleman et al., 2009; de Bruin et al., 2011). The strength of this approach may

be the considerations of incorporating the necessary levels from resources and policies at the

community level and the organization of the health care within the health system (including e.g.

development of clinical practice, educating healthcare providers etc.) in order to be able to

generate productive interaction leading to improved outcome (Wagner et al., 2001; © 2006-2013

Improving Chronic Illness Care, 2013). The model has been adjusted to several contexts and

societies. For instance the Danish CCM �Model for behandling af kronisk syge�, which could be

used as a template for the efforts to improve patient care and treatment, and enhanced support for

patients with osteoporosis according to their interpretation, management and decision-making. The

Chronic Care Model is constructed to capture healthcare across the borders of primary and

secondary healthcare system which is very much needed in the Danish healthcare system, since

the trend over the past decade has been going towards fewer and shorter hospitalizations with

increased responsibility for the primary healthcare as well as for outpatient clinics. Further, more

specialist nursing activities have been required over the past decade. As discussed in the thesis

and emphasized by Benner and Wrubel, nurses are in a unique position of being able to

understand illness experiences and the meanings of the experience supplied by the patients

(Benner and Wrubel, 1989). According to the findings, when seeking to support patients with


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osteoporosis, future interventions should be planed as a collaborative effort between the treating

physician and specialist nurses.

Further, interventions targeting osteoporotic fracture prevention and encouraging collaboration

between patients� and healthcare professionals may incorporate approaches of shared decision-

making. Shared decision-making has been found to be the most effective therapeutic option to

increase compliance and persistence to medical treatment, together with fostering greater patient

satisfaction and improving the health care process and outcomes for patients (Martin et al., 2005).

However, such approaches have mainly been performed as informing the patients of the decision

made by an �imposing� healthcare professional. A Person-Centered Communication and Reflection

Model emphasizes that shared decision-making in chronic care is more about professionals

gaining insight into patients� decisions, rather than the opposite, as it improves the opportunities for

supporting each patient in developing illness-specific self-management skills (Zoffmann et al.,

2008). Since this model is expected to be applicable across chronic conditions, it would be

interesting to conduct an intervention testing the applicability and outcome in an osteoporosis

outpatient clinic. The essence of shared decision-making is to be seen as the healthcare

professionals inviting �the owner� of a problem to participate in problem solving and decision

making. This shared decision making happens when the patient is given an opportunity to benefit

from professional competences through discussing difficulties of the self-management of their

illness. It may be difficult to implement this model as healthcare professionals may find it hard to

take a health-oriented stance and focus on the particular patient�s reaction to the illness in daily

life, different from the disease-oriented perspective (Zoffmann et al., 2008). Implementing and

testing this Person-Centered Communication and Reflection Model would be in accordance with

the theory of the experienced processes of recovery: Responding to threats to integrity of self

(Morse, 1997). Within such intervention it would be appropriate to draw on the findings in this

thesis as life with osteoporosis is a matter of a multifaceted adapting process of varying length for

the individual patient, which is highly influenced by decisions regarding the medical treatment.

Additionally, the study found some answers in the Sense of Coherence Theory according to the

new life event - being diagnosed with osteoporosis and being compelled to live with it, as coping

with stressors in a salutogenic perspective. Also, new questions arise. In a health promotion

perspective, it would be interesting to further investigate the process of acceptance of

osteoporosis, as well as the process of decision-making within a longitudinal design and in light of

exploring the relationship to sense of coherence.


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10. SUMMARY

Background: Osteoporosis is considered a major worldwide public health problem. Men and

women with osteoporosis are mostly unaware of the illness, until bone fractures occur. It is

estimated that more than one in three European women and one in five men over 50 years will

experience osteoporotic fractures followed by considerable morbidity and mortality. In line with

many other chronic illnesses, one of the pronounced challenges related to osteoporosis is

considered to be compliance and persistence to medical treatment. The causes of low compliance

and persistence to treatment remain unclear. Living with a chronic illness often affects the entire

human life situation, specifically, emotionally and existentially. How this is experienced personally

and how this affects the individual�s life is still unknown.

Aim: To gain a deeper understanding of patients� life with osteoporosis by determining the

distribution and determinants of refill compliance and persistence to oral anti-osteoporotic

treatment among new users of oral anti-osteoporotic therapy in Denmark as well as to explore the

experiences of newly diagnosed women in terms of living with osteoporosis after pharmaceutical

osteoporosis treatment for fracture prevention had been prescribed.

Method: The quantitative study was a register-based national open cohort study of compliance

and persistence of anti-osteoporotic therapy, comprising of 100,949 men and women. Statistical

analysis including backward stepwise logistic regression analysis was used to explain causes of

treatment failure and Kaplan Meier survival analysis to estimate persistence of treatment.

The qualitative study was a longitudinal study with a phenomenological-hermeneutic approach.

Fifteen women with osteoporosis prescribed pharmaceutical preventive treatment were included.

Data were obtained using individual interviews three times during one year. Data were analysed on

three levels: Naïve reading, structural analysis and critical interpretation and discussion.

Ethical considerations: The study was approved by the Danish Data Protection Agency (J.no.

2012-41-0875) and The National Committee on Health Research Ethics (J.no. H-C-FSP-2011_01).

Results: The register-based study (paper I) showed that 56.6% of the 93,365 patients were

persistent and compliant, 4.7% were persistent with low refill-compliance, while 38.7% terminated

treatment within the first year (�early quitters�). �Early quitters� were found to differ in socio-

economics demographics from other patients with low refill compliance. An increased risk of

quitting during the first year of treatment compared with those who were refill-compliant for more

than one year was found to be associated with: fractures overall (as one category), living in an

apartment, children living at home, living alone, other bisphosphonates or non-bisphosphonates,

male gender, living close to a university hospital, number of drugs, hormone replacement therapy,

prednisolone and anti-depressants, comorbidities 3+, pulmonary and collagen diseases.


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In the interview study of �lived experiences� of osteoporosis the first six month three key themes

emerged as well as sub themes: 1) �being diagnosed�: to be taken seriously, accepting the

diagnosis. 2) �being prescribed medical treatment�: decision against the medical treatment,

decision to pursue the medical treatment. 3) �being on the path of learning to live with

osteoporosis�: acceptance � the need to adapt, lifestyle changes. Furthermore, in the one-year

longitudinal interview study found two key themes on to life with osteoporosis: 1) �to become

influenced by the therapy�: taking the medication, stopping to take the medication. 2) �daily life with

osteoporosis�: interpretation of symptoms, interpretation of scanning result and life style reflections.

Conclusion: These quantitative and qualitative findings together contribute to the current

knowledge regarding compliance and persistent, determinants for potential risk groups as well as

topical thematic areas central to newly-diagnosed women and main strategies for handling life with

osteoporosis. These findings point to a new direction of health promotion areas which may be used

to suggest target actions when seeking to establish improved patient care and treatment. Patients

need to be supported to interpret the diagnosis, fracture risk and decisions around medical

treatment as related to life with osteoporosis to prevent osteoporotic fractures and promote bone

health. Further research is needed to explore specific components of the multifaceted process of

learning to live with osteoporosis in an in-depth fashion.


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11. RESUMÉ PÅ DANSK (SUMMERY IN DANISH)

Baggrund: Osteoporose betragtes som et verdensomspændende folkesundhedsproblem. Mænd

og kvinder som har osteoporose er for det meste ikke bevidste om dette før knoglebrud opstår. Det

anslås at mere end en af tre europæiske kvinder og hver fjerde mand over 50 år vil opleve at få

osteoporotiske brud med betydelig sygelighed og dødelighed til følge. I lighed med mange andre

kroniske lidelser, betragtes en af de største udfordringer vedrørende osteoporose at være

compliance og vedholdenhed i forhold til den medicinske behandling. Årsagssammenhænge i

forbindelse med lav compliance og vedholdenhed af den farmaceutiske behandling er fortsat

uklare. At leve med en kronisk tilstand påvirker oftest menneskets livssituation, både specifikt,

emotionelt og eksistentielt, hvordan dette opleves er individuelt og det er endnu uvist hvordan det

påvirker den enkeltes livssituation.

Formål: At opnå en dybere forståelse af patienters liv med osteoporose ved at bestemme

omfanget af, samt beskrive faktorer vedrørende receptindløst compliance og vedholdenhed af oral

anti-osteoporotisk behandling blandt nye brugere af oral anti-osteoporotisk medicin i Danmark.

Tillige at udforske ny-diagnosticerede kvinders levede erfaringer med osteoporose når

farmakologisk fraktur forebyggende behandling var ordineret.

Metode: Det kvantitative studie var en registerbaseret national åben kohorte undersøgelse af

compliance og vedholdenhed af anti-osteoporotisk behandling. Undersøgelsen omfattede 100.949

mænd og kvinder. Statistiske analyser blev anvendt til at forklare årsager til behandlingssvigt,

herunder backward stepwise logistic regression analyse, samt Kaplan Meier overlevelsesanalyse

til estimering af behandlingslængde.

Det kvalitative studie havde en fænomenologisk-hermeneutisk tilgang, samt et longitudinelt design.

Femten kvinder blev inkluderet, de havde diagnosticeret osteoporose og farmakologisk

forebyggende behandling var ordineret. Individuelle interviews blev gennemført tre gange i løbet af

det første år. Dataanalysen omfattede tre niveauer: naïv læsning, strukturanalyse, samt kritisk

fortolkning og diskussion.

Etiske overvejelser: Studiet var godkendt af Datatilsynet (J.no. 2012-41-0875) og

Videnskabsetisk komité havde ingen indvendinger til studiet (J.no. H-C-FSP-2011_01).

Resultater: Det registerbaserede studie (artikel I) viste at 56,6% af de 93.365 patienter var

vedholdende og compliant, 4,7% var vedholdende men med lav receptindløst-compliance, mens

38,7% stoppede med behandlingen i løbet af det første år (�early quitters�). �Early quitters� viste sig

at adskille sig socioøkonomisk og demografisk fra andre patienter med lav receptindløst-

compliance. Studiet viste at der var en øget risiko for at stoppe behandlingen det første år, når der


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blev sammenlignet med dem der compliant udover et år (baseret på indløste recepter), disse

forskelle var associeret med: Fraktur (som en kategori), at bo i lejlighed, at have hjemmeboende

børn, at bo alene, behandling med andre bisphosphonater eller ikke- bisphosphonater, at være

mand, at bo tæt på universitets hospital, antal af andre præparater, hormon behandling,

prednisolon og anti-depressiva, ko-morbiditeter 3+, lunge og reumatologisk lidelser.

I interviewundersøgelsen af erfaringer med at leve med osteoporose de første seks måneder blev

tre centrale temaer udledt: 1) "at blive diagnosticeret": at blive taget alvorligt, acceptere diagnosen.

2) "at blive ordineret medicinsk behandling": beslutning mod den medicinske behandling,

beslutning om at følge den medicinske behandling. 3) "at være på vej til at lære at leve med

osteoporose": accept - behovet for at tilpasse sig, livsstilsændringer. Derudover blev der i den

etårige longitudinelle interviewundersøgelse fundet to centrale temaer beskrivende livet med

osteoporose: 1) "til at blive påvirket af den medicinske behandling": at tage medicinen, stoppe med

at tage medicinen. 2) "det daglige liv med osteoporose": fortolkning af symptomer, tolkning af

scanning resultat og refleksioner vedr. livsstilsfaktorer.

Konklusion: Tilsammen bidrager de kvantitative og kvalitative fund til den aktuelle viden om

compliance og vedholdenhed, ved at beskrive afgørende faktorer for potentielle risikogrupper,

samt aktuelle temaer centrale for ny-diagnosticerede kvinder og betydningsfulde strategier til

håndtering af livet med osteoporose. Disse resultater peger i en ny retning vedr.

sundhedsfremmende områder, som kan anvendes til at målrette interventioner, med henblik på at

etablere forbedret patientpleje og behandling. Patienterne har behov for støttet til at forstå

diagnosen og fraktur risiko, samt støtte vedr. beslutninger omkring den medicinske behandling,

med henblik på at opnå accept af diagnosen, at leve med osteoporose, samt at mindske risikoen

for knoglebrud og fremme knoglesundheden. Yderligere forskning er nødvendig for at undersøge

specifikke komponenter af den mangesidede proces med at lære at leve med osteoporose.


__________________________________________________________________________________________________________________________________�

69

12. REFERENCES

Abrahamsen,B., 2010. Bisphosphonate adverse effects, lessons from large databases. Curr. Opin. Rheumatol. 22 (4), 404-409.

Abrahamsen,B., van,S.T., Ariely,R., Olson,M., Cooper,C., 2009. Excess mortality following hip fracture: a systematic epidemiological review. Osteoporos. Int. 20 (10), 1633-1650.

Abrahamsen,B., Vestergaard,P., 2010. Declining incidence of hip fractures and the extent of use of anti-osteoporotic therapy in Denmark 1997-2006. Osteoporos. Int. 21 (3), 373-380.

Antonovsky,A., 1979. Health, Stress and Coping. Jossey-Bass, San Francisco.

Balkrishnan,R., 1998. Predictors of medication adherence in the elderly. Clin. Ther. 20 (4), 764-771.

Barat,I., Andreasen,F., Damsgaard,E.M., 2001. Drug therapy in the elderly: what doctors believe and patients actually do. Br. J. Clin. Pharmacol. 51 (6), 615-622.

Barker,C., 2009. The Mean, Median and Confidence Intervals of Kaplan-Meier Survival Estimate -Computationa and Applications. The American Statistician (TAS) 63 (1), 78-80.

Benner,P., Wrubel,J., 1989. The primacy of Caring: Stress and Coping in Health and Illness. Addison Wesley Publishing Company, Menlo Park, California.

Besser,S.J., Anderson,J.E., Weinman,J., 2012. How do osteoporosis patients perceive their illness and treatment? Implications for clinical practice. Arch. Osteoporos. 7 (1-2), 115-124.

Bewick,V., Cheek,L., Ball,J., 2004. Statistics review 8: Qualitative data - tests of association. Crit Care 8 (1), 46-53.

Bewick,V., Cheek,L., Ball,J., 2005. Statistics review 14: Logistic regression. Crit Care 9 (1), 112-118.

Block,A.E., Solomon,D.H., Cadarette,S.M., Mogun,H., Choudhry,N.K., 2008. Patient and physician predictors of post-fracture osteoporosis management. J. Gen. Intern. Med. 23 (9), 1447-1451.

Cadarette,S.M., Burden,A.M., 2010. Measuring and improving adherence to osteoporosis pharmacotherapy. Curr. Opin. Rheumatol. 22 (4), 397-403.

Christensen,P.M., Brixen,K., Gyrd-Hansen,D., Kristiansen,I.S., 2005. Cost-effectiveness of alendronate in the prevention of osteoporotic fractures in Danish women. Basic Clin. Pharmacol. Toxicol. 96 (5), 387-396.

Cockerill,W., Lunt,M., Silman,A.J., Cooper,C., Lips,P., Bhalla,A.K., Cannata,J.B., Eastell,R., Felsenberg,D., Gennari,C., Johnell,O., Kanis,J.A., Kiss,C., Masaryk,P., Naves,M., Poor,G., Raspe,H., Reid,D.M., Reeve,J., Stepan,J., Todd,C., Woolf,A.D., O'Neill,T.W., 2004. Health-related quality of life and radiographic vertebral fracture. Osteoporos. Int. 15 (2), 113-119.

Coleman,K., Austin,B.T., Brach,C., Wagner,E.H., 2009. Evidence on the Chronic Care Model in the new millennium. Health Aff. (Millwood. ) 28 (1), 75-85.

Cooper,C., Atkinson,E.J., Jacobsen,S.J., O'Fallon,W.M., Melton,L.J., III, 1993. Population-based study of survival after osteoporotic fractures. Am. J. Epidemiol. 137 (9), 1001-1005.


__________________________________________________________________________________________________________________________________�

70

Cooper,C., Harvey,N.C., 2012. Osteoporosis risk assessment. BMJ 344 , e4191.

Cooper,C., Rizzoli,R., Reginster,J.Y., 2009. The International Osteoporosis Foundation: history, objectives and achievements. Osteoporos. Int. 20 Suppl 3 , S241-S242.

Cortet,B., Benichou,O., 2006. Adherence, persistence, concordance: do we provide optimal management to our patients with osteoporosis? Joint Bone Spine 73 (5), e1-e7.

de Bruin,S.R., Heijink,R., Lemmens,L.C., Struijs,J.N., Baan,C.A., 2011. Impact of disease management programs on healthcare expenditures for patients with diabetes, depression, heart failure or chronic obstructive pulmonary disease: a systematic review of the literature. Health Policy 101 (2), 105-121.

de Souza,L.B., Da Silva Mazeto,G.M., Bocchi,S.C., 2010. Self-managing osteoporosis treatment for well-being recovery mediated by the (in)visibility of the disease signs. Rev. Lat. Am. Enfermagem. 18 (3), 398-405.

Devold,H.M., Furu,K., Skurtveit,S., Tverdal,A., Falch,J.A., Sogaard,A.J., 2012. Influence of socioeconomic factors on the adherence of alendronate treatment in incident users in Norway. Pharmacoepidemiol. Drug Saf 21 (3), 297-304.

Dimatteo,M.R., 2004a. Social support and patient adherence to medical treatment: a meta-analysis. Health Psychol. 23 (2), 207-218.

Dimatteo,M.R., 2004b. Variations in patients' adherence to medical recommendations: a quantitative review of 50 years of research. Med. Care 42 (3), 200-209.

Eriksson,M., Lindstrom,B., 2006. Antonovsky's sense of coherence scale and the relation with health: a systematic review. J. Epidemiol. Community Health 60 (5), 376-381.

Frank,L., 2000. Epidemiology. When an entire country is a cohort. Science 287 (5462), 2398-2399.

Furu,K., Wettermark,B., Andersen,M., Martikainen,J.E., Almarsdottir,A.B., Sorensen,H.T., 2010. The Nordic countries as a cohort for pharmacoepidemiological research. Basic Clin. Pharmacol. Toxicol. 106 (2), 86-94.

Genant,H.K., Cooper,C., Poor,G., Reid,I., Ehrlich,G., Kanis,J., Nordin,B.E., Barrett-Connor,E., Black,D., Bonjour,J.P., Dawson-Hughes,B., Delmas,P.D., Dequeker,J., Ragi,E.S., Gennari,C., Johnell,O., Johnston,C.C., Jr., Lau,E.M., Liberman,U.A., Lindsay,R., Martin,T.J., Masri,B., Mautalen,C.A., Meunier,P.J., Khaltaev,N., ., 1999. Interim report and recommendations of the World Health Organization Task-Force for Osteoporosis. Osteoporos. Int. 10 (4), 259-264.

Gleeson,T., Iversen,M.D., Avorn,J., Brookhart,A.M., Katz,J.N., Losina,E., May,F., Patrick,A.R., Shrank,W.H., Solomon,D.H., 2009. Interventions to improve adherence and persistence with osteoporosis medications: a systematic literature review. Osteoporos. Int. 20 (12), 2127-2134.

Gliklich,R.E., Dreyer,N.A., 2010. Registries for Evaluating Patient Outcomes: A User's Guide. Chapter 14: Evaluating Registries. AHRQ Publication NO. 10-EHC049. Agency for Healthcare Research and Quality, Rockvill. 2ond ed. 305-315.

Hall,E., 2001. Relationen mellem interviewer og informant i det kvalitative forskningsinterview. In: Lunde,I.M., Ramhøj,P. (Eds.), Humanistisk forskning inden for sundhedsvidenskab. Akademisk Forlag, København, pp. 182-200.

Hallas,J., Stovring,H., 2006. Templates for analysis of individual-level prescription data. Basic Clin. Pharmacol. Toxicol. 98 (3), 260-265.


__________________________________________________________________________________________________________________________________�

71

Helweg-Larsen,K., 2011. The Danish Register of Causes of Death. Scand. J. Public Health 39 (7 Suppl), 26-29.

Hermansen,M., Rendorff,J.D., 2002. En Hermenutisk Brobygger - tekster af Ricoeur [A Hermenutic Bridging - texts by Ricoeur]. Forlaget Klim, Århus.

Herzog,H., 2005. On Home Turf: Interview Location and Its Social Meaning. Qualitative Sociology 28 (1), 25-47.

Hjalmarson,H.V., Straudmark,M., Klässbo,M., 2007. Healthy risk awareness motivates fracture prevention behavior: A grounded theory study of women with osteoporosis. Int J. Qual. Stud. Health Well. -being. 2 , 236-245.

Holloway,I., 2007. Vulnerable story telling: narrative research in nursing. Journal of Research in Nursing 12 (6), 703-711.

Hooven,F.H., Adachi,J.D., Adami,S., Boonen,S., Compston,J., Cooper,C., Delmas,P., Diez-Perez,A., Gehlbach,S., Greenspan,S.L., LaCroix,A., Lindsay,R., Netelenbos,J.C., Pfeilschifter,J., Roux,C., Saag,K.G., Sambrook,P., Silverman,S., Siris,E., Watts,N.B., Anderson,F.A., Jr., 2009. The Global Longitudinal Study of Osteoporosis in Women (GLOW): rationale and study design. Osteoporos. Int. 20 (7), 1107-1116.

Imaz,I., Zegarra,P., Gonzalez-Enriquez,J., Rubio,B., Alcazar,R., Amate,J.M., 2010. Poor bisphosphonate adherence for treatment of osteoporosis increases fracture risk: systematic review and meta-analysis. Osteoporos. Int. 21 (11), 1943-1951.

Kanis,J.A., Gluer,C.C., 2000. An update on the diagnosis and assessment of osteoporosis with densitometry. Committee of Scientific Advisors, International Osteoporosis Foundation. Osteoporos. Int. 11 (3), 192-202.

Kanis,J.A., McCloskey,E.V., Johansson,H., Cooper,C., Rizzoli,R., Reginster,J.Y., 2013. European guidance for the diagnosis and management of osteoporosis in postmenopausal women. Osteoporos. Int. 24 (1), 23-57.

Kanis,J.A., Melton,L.J., III, Christiansen,C., Johnston,C.C., Khaltaev,N., 1994. The diagnosis of osteoporosis. J. Bone Miner. Res. 9 (8), 1137-1141.

Kemp,P., 1999. Tid og fortælling. Introduktion til Paul Ricoeur [Time and narrative. Introduction to Paul Ricoeur]. Århus Universitets Forlag, Århus.

Kothawala,P., Badamgarav,E., Ryu,S., Miller,R.M., Halbert,R.J., 2007. Systematic review and meta-analysis of real-world adherence to drug therapy for osteoporosis. Mayo Clin. Proc. 82 (12), 1493-1501.

Kvale,S., Brinkmann,S., 2009. Interviews - Learning the Craft of Qualitative Research Interviewing. SAGE Publications, Inc., Thousand Oaks, California.

Landfeldt,E., Strom,O., Robbins,S., Borgstrom,F., 2012. Adherence to treatment of primary osteoporosis and its association to fractures--the Swedish Adherence Register Analysis (SARA). Osteoporos. Int. 23 (2), 433-443.

Lau,E., Papaioannou,A., Dolovich,L., Adachi,J., Sawka,A.M., Burns,S., Nair,K., Pathak,A., 2008. Patients' adherence to osteoporosis therapy: exploring the perceptions of postmenopausal women. Can. Fam. Physician 54 (3), 394-402.

Lewiecki,E.M., 2010. Risk communication and shared decision making in the care of patients with osteoporosis. J. Clin. Densitom. 13 (4), 335-345.


__________________________________________________________________________________________________________________________________�

72

Lindberg,M.J., Andersen,S.E., Christensen,H.R., Kampmann,J.P., 2008. [Compliance to drug prescriptions]. Ugeskr. Laeger 170 (22), 1912-1916.

Lindseth,A., Norberg,A., 2004. A phenomenological hermeneutical method for researching lived experience. Scand. J. Caring. Sci. 18 (2), 145-153.

Lu Xu, Wen-Jun Zhang, 2001. Comparison of different methods for variable selection. Analytica Chemica Acta 446 (1), 477-483.

Malterud,K., 2001. Qualitative research: standards, challenges, and guidelines. Lancet 358 (9280), 483-488.

Martin,L.R., Williams,S.L., Haskard,K.B., Dimatteo,M.R., 2005. The challenge of patient adherence. Ther. Clin. Risk Manag. 1 (3), 189-199.

Mazor,K.M., Velten,S., Andrade,S.E., Yood,R.A., 2010. Older women's views about prescription osteoporosis medication: a cross-sectional, qualitative study. Drugs Aging 27 (12), 999-1008.

McHorney,C.A., Schousboe,J.T., Cline,R.R., Weiss,T.W., 2007. The impact of osteoporosis medication beliefs and side-effect experiences on non-adherence to oral bisphosphonates. Curr. Med. Res. Opin. 23 (12), 3137-3152.

McHorney,C.A., Spain,C.V., 2011. Frequency of and reasons for medication non-fulfillment and non-persistence among American adults with chronic disease in 2008. Health Expect. 14 (3), 307-320.

McLeod,K.M., Johnson,C.S., 2011. A systematic review of osteoporosis health beliefs in adult men and women. J. Osteoporos. 2011 , 197454-197465.

Meadows,L.M., Mrkonjic,L.A., O'Brien,M.D., Tink,W., 2007. The importance of communication in secondary fragility fracture treatment and prevention. Osteoporos. Int. 18 (2), 159-166.

Morse,J.M., 1997. Responding to threats to integrity of self. ANS Adv. Nurs. Sci. 19 (4), 21-36.

Morse,J.M., Barrett,M., Mayan,M., Olson,K., Spiers,J., 2002. Verification Strategies for Establishing Reliability and Validity in Qualitative Research. International Journal of Qualitative Methods 1 (2), 13-22.

Morse,J.M., Johnson,J.L., 1991. Toward a Theory of Illness: The Illness Constellation Model. In: Morse,J.M., Johnson,J.L. (Eds.), The Illness Experience - Dimensions of Suffering. SAGE Publication, Inc., Newbury Park, California, pp. 315-342.

Morse,J.M., O'Brien,B., 1995. Preserving self: from victim, to patient, to disabled person. J. Adv. Nurs. 21 (5), 886-896.

Musselwhite,K., Cuff,L., McGregor,L., King,K.M., 2007. The telephone interview is an effective method of data collection in clinical nursing research: a discussion paper. Int. J. Nurs. Stud. 44 (6), 1064-1070.

Nielsen,D., Huniche,L., Brixen,K., Sahota,O., Masud,T., 2012. Handling knowledge on osteoporosis - a qualitative study. Scand. J. Caring. Sci., 0238-9318.

Nielsen,D.S., Brixen,K., Huniche,L., 2011. Men's experiences of living with osteoporosis: focus group interviews. Am. J. Mens. Health 5 (2), 166-176.


__________________________________________________________________________________________________________________________________�

73

Nielsen,D.S., Langdahl,B.L., Sorensen,O.H., Sorensen,H.A., Brixen,K.T., 2010. Persistence to medical treatment of osteoporosis in women at three different clinical settings--a historical cohort study. Scand. J. Public Health 38 (5), 502-507.

Pahuus,M., 2001. Hermenutik. In: Collin,F., Køppe,S. (Eds.), Humanistisk Videnskabsteori. DR Multimedie, pp. 109-137.

Papaioannou,A., Kennedy,C.C., Dolovich,L., Lau,E., Adachi,J.D., 2007. Patient adherence to osteoporosis medications: problems, consequences and management strategies. Drugs Aging 24 (1), 37-55.

Pedersen,B.D., 1999. Sygeplejepraksis.Sporg og erkendelse [Nursing practice Language and cognition]. PhD thesis, The Faculty of Health Science, University of Aarhus, Denmark. (3 ed.2005).

Reginster,J.Y., Burlet,N., 2006. Osteoporosis: a still increasing prevalence. Bone 38 (2 Suppl 1), S4-S9.

Reventlow,S., Bang,H., 2006. Brittle bones: ageing or threat of disease exploring women's cultural models of osteoporosis. Scand. J. Public Health 34 (3), 320-326.

Reventlow,S.D., 2007. Perceived risk of osteoporosis: restricted physical activities? Qualitative interview study with women in their sixties. Scand. J. Prim. Health Care 25 (3), 160-165.

Reventlow,S.D., Hvas,L., Malterud,K., 2006. Making the invisible body visible. Bone scans, osteoporosis and women's bodily experiences. Soc. Sci. Med. 62 (11), 2720-2731.

Ricoeur,P., 1973. The hermeneutical function of distanciation. Philosophy Today 17 , 129-141.

Ricoeur,P., 1976. Interpretation Theory: Discourse and the surplus of meaning. Christian University Press, Fort Worth, Texas.

Ricoeur,P., 1984. Time and Narrative Volume I. The University of Chicago, Translated by Kathleen MaLaughlin and David Pellauer from 'Temps et récit Tome I', Paris 1983.

Ricoeur,P., 1985. Time and Narrative volume II. Translated by Kathleen MaLaughlin and David Pellauer from 'Temps et récit. La configuration du temps dans lerécit de fiction Tome II', Paris 1985.

Ricoeur,P., 1988. Time and Narrative volume III. Translated by Kathleen MaLaughlin and David Pellauer from 'Temps et récit Tome III', Paris 1985.

Roberto,K.A., Reynolds,S.G., 2001. The meaning of osteoporosis in the lives of rural older women. Health Care Women Int. 22 (6), 599-611.

Roerholt,C., Eiken,P., Abrahamsen,B., 2009. Initiation of anti-osteoporotic therapy in patients with recent fractures: a nationwide analysis of prescription rates and persistence. Osteoporos. Int. 20 (2), 299-307.

Saldaña,J., 2003. Longitudinal Qualitatve Research: analyzing changes through time. AltaMira Press.

Sale,J.E., Beaton,D.E., Sujic,R., Bogoch,E.R., 2010. 'If it was osteoporosis, I would have really hurt myself.' Ambiguity about osteoporosis and osteoporosis care despite a screening programme to educate fragility fracture patients. J. Eval. Clin. Pract. 16 (3), 590-596.


__________________________________________________________________________________________________________________________________�

74

Sale,J.E., Gignac,M.A., Hawker,G., Frankel,L., Beaton,D., Bogoch,E., Elliot-Gibson,V., 2011. Decision to take osteoporosis medication in patients who have had a fracture and are 'high' risk for future fracture: a qualitative study. BMC. Musculoskelet. Disord. 12 , 92-101.

Sandelowski,M., Voils,C.I., Barroso,J., 2006. Defining and Designing Mixed Research Synthesis Studies. Res. Sch 13 (1), 29.

Saracci,R., 2010. Introducing the history of epidemiology. In: Olsen,J., Saracci,R., Trichopoulos,D. (Eds.), Teaching Epidemiology: A guide for teachers in epidemiology, publich health and clinical medicine. Oxford University Press, Oxford.

Seeman,E., Compston,J., Adachi,J., Brandi,M.L., Cooper,C., Dawson-Hughes,B., Jonsson,B., Pols,H., Cramer,J.A., 2007. Non-compliance: the Achilles' heel of anti-fracture efficacy. Osteoporos. Int. 18 (6), 711-719.

Silva,I., 1999. Cancer Epidemiology: Principles and Methods. International Agency for Research on Cancer, WHO.

Silverman,S., Gold,D.T., 2010. Compliance and persistence with osteoporosis medications: a critical review of the literature. Rev. Endocr. Metab Disord. 11 (4), 275-280.

Siris,E.S., Harris,S.T., Rosen,C.J., Barr,C.E., Arvesen,J.N., Abbott,T.A., Silverman,S., 2006. Adherence to bisphosphonate therapy and fracture rates in osteoporotic women: relationship to vertebral and nonvertebral fractures from 2 US claims databases. Mayo Clin. Proc. 81 (8), 1013-1022.

Solimeo,S.L., Weber,T.J., Gold,D.T., 2011. Older men's explanatory model for osteoporosis. Gerontologist 51 (4), 530-539.

Sonder,H., Boje,T., Delmar C., Dylmer,D., Forup,L., Jakobsen C., Moller,M., Pedersen,B.D., 2007. Lidelse som en skygge? - et aspekt i livet med krinisk sygdom [Suffering as a shadow? - An aspect of life with chronic illness. Klinisk Sygepleje 3 , 69-80.

Statistics Denmark, 2008. http://www.statistikbanken.dk/statbank5a/default.asp?w=1280

Sullivan,G.C., 1989. Evaluating Antonovsky's Salutogenic Model for its adaptability to nursing. J. Adv. Nurs. 14 (4), 336-342.

Sullivan,G.C., 1993. Towards clarification of convergent concepts: sense of coherence, will to meaning, locus of control, learned helplessness and hardiness. J. Adv. Nurs. 18 (11), 1772-1778.

Vandenbroucke,J.P., von,E.E., Altman,D.G., Gotzsche,P.C., Mulrow,C.D., Pocock,S.J., Poole,C., Schlesselman,J.J., Egger,M., 2007. Strengthening the Reporting of Observational Studies in Epidemiology (STROBE): explanation and elaboration. Epidemiology 18 (6), 805-835.

Vestergaard,P., Rejnmark,L., Mosekilde,L., 2005. Osteoporosis is markedly underdiagnosed: a nationwide study from Denmark. Osteoporos. Int. 16 (2), 134-141.

Wagner,E.H., Austin,B.T., Davis,C., Hindmarsh,M., Schaefer,J., Bonomi,A., 2001. Improving chronic illness care: translating evidence into action. Health Aff. (Millwood. ) 20 (6), 64-78.

Wastesson,J.W., Ringback,W.G., Parker,M.G., Johnell,K., 2013. Educational level and use of osteoporosis drugs in elderly men and women: a Swedish nationwide register-based study. Osteoporos. Int 24 (2), 433-442.


__________________________________________________________________________________________________________________________________�

75

Weiss,T.W., Gold,D.T., Silverman,S.L., McHorney,C.A., 2006. An evaluation of patient preferences for osteoporosis medication attributes: results from the PREFER-US study. Curr. Med. Res. Opin. 22 (5), 949-960.

Weston,J.M., Norris,E.V., Clark,E.M., 2011. The invisible disease: making sense of an osteoporosis diagnosis in older age. Qual. Health Res. 21 (12), 1692-1704.

Wilkins,S., 2001a. Aging, chronic illness and self-concept: a study of women with osteoporosis. J. Women Aging 13 (1), 73-92.

Wilkins,S., 2001b. Women with osteoporosis: strategies for managing aging and chronic illness. J. Women Aging 13 (3), 59-77.

World Health Organization, 1994. Assessment of fracture risk and its application to screening for postmenopausal osteoporosis. Report of a WHO Study Group 843 , 1-129.

World Health Organization, 2003a. Adherence to Long-Term Therapies, Evidence for Action. http://www.who.int/chp/knowledge/publications/adherence_full_report.pdf

World Health Organization, 2003b. Prevention and Management of Osteoporosis. Report of WHO Scientific Group. http://whqlibdoc.who.int/trs/who_trs_921.pdf

World Medical Association, 2008. World Medical Association Declaration of Helsinki, Ethical Principles for Medical Research Involving Human Subjects. http://www.who.int/bulletin/archives/79(4)373.pdf

Yood,R.A., Mazor,K.M., Andrade,S.E., Emani,S., Chan,W., Kahler,K.H., 2008. Patient decision to initiate therapy for osteoporosis: the influence of knowledge and beliefs. J. Gen. Intern. Med. 23 (11), 1815-1821.

Zoffmann,V., Harder,I., Kirkevold,M., 2008. A person-centered communication and reflection model: sharing decision-making in chronic care. Qual. Health Res. 18 (5), 670-685.

Zolnierek,K.B., Dimatteo,M.R., 2009. Physician communication and patient adherence to treatment: a meta-analysis. Med. Care 47 (8), 826-834.

© 2006-2013 Improving Chronic Illness Care, 2013. The Chronic Care Model. http://www.improvingchroniccare.org/index.php?p=The_Chronic_Care_Model&s=2

13. APPENDIX

Appendix�A�

Strengthening�the�Reporting�of�Observational�Studies�in�Epidemiology�(STROBE)��

checklist�

Section/Topic Item #

Recommendation Reported on page #

Title and abstract

1 (a) Indicate the study�s design with a commonly used term in the title or the abstract cohort

Titel / summary

(b) Provide in the abstract an informative and balanced summary of what was done and what was found

Summary

Introduction

Background/ rationale

2 Explain the scientific background and rationale for the investigation being reported

Introduction

Objectives 3 State specific objectives, including any prespecified hypotheses

9

Methods

Study design 4 Present key elements of study design early in the paper

12 section 3.1.1. and 18 section 4

Setting 5 Describe the setting, locations, and relevant dates, including periods of recruitment, exposure, follow-up, and data collection

18-19

Participants 6 (a) Give the eligibility criteria, and the sources and methods of selection of participants. Describe methods of follow-up

19, 20

(b)For matched studies, give matching criteria and number of exposed and unexposed

/

Variables 7 Clearly define all outcomes, exposures, predictors, potential confounders, and effect modifiers. Give diagnostic criteria, if applicable

21, 22

Data sources/ measurement

8* For each variable of interest, give sources of data and details of methods of assessment (measurement). Describe comparability of assessment methods if there is more than one group

Data sources: pp 12, asses-sement pp 19-20. variable 19

Bias 9 Describe any efforts to address potential sources of bias

21

Study size 10 Explain how the study size was arrived at 18 Quantitative variables

11 Explain how quantitative variables were handled in the analyses. If applicable, describe which groupings were chosen and why

21-23

Statistical methods

12 (a) Describe all statistical methods, including those used to control for confounding

21-23

(b) Describe any methods used to examine subgroups and interactions

21

(c) Explain how missing data were addressed 21-23 (d) If applicable, explain how loss to follow-up was addressed

22

(e) Describe any sensitivity analyses 24

Results

Participants 13* (a) Report numbers of individuals at each stage of study�eg numbers potentially eligible, examined for eligibility, confirmed eligible, included in the study, completing follow-up, and analysed

31-32

(b) Give reasons for non-participation at each stage

31

(c) Consider use of a flow diagram 32 Descriptive data

14* (a) Give characteristics of study participants (eg demographic, clinical, social) and information on exposures and potential confounders

Table 5 pp 34

(b) Indicate number of participants with missing data for each variable of interest

31

(c) Summarise follow-up time (eg, average and total amount)

31

Outcome data 15* Report numbers of outcome events or summary measures over time

Table 5 pp 34

Main results 16 (a) Give unadjusted estimates and, if applicable, confounder-adjusted estimates and their precision (eg, 95% confidence interval). Make clear which confounders were adjusted for and why they were included

Table 5

(b) Report category boundaries when continuous variables were categorized

20

(c) If relevant, consider translating estimates of relative risk into absolute risk for a meaningful time period

/

Other analyses

17 Report other analyses done�eg analyses of subgroups and interactions, and sensitivity analyses

35

Discussion Key results 18 Summarise key results with reference to study

objectives 42

Limitations

Interpretation 20 Give a cautious overall interpretation of results considering objectives, limitations, multiplicity of analyses, results from similar studies, and other relevant evidence

55

Generalisability 21 Discuss the generalisability (external validity) of the study results

56

Other information

Funding 22 Give the source of funding and the role of the funders for the present study and, if applicable, for the original study on which the present article is based

ii

Appendix�B�In

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Appendix�C

Datatilsynet har den 2. august 2013 modtaget din anmodning om forlængelse

af tilladelsen til at behandle personoplysninger i det videnskabelige projekt

med ovennævnte journalnummer.

Projektets titel er: �At leve med knogleskørhed - et epidemiologisk og kvalita-

tivt studie�.

Det fremgår, at tilladelsen ønskes forlænget til 1. maj 2014.

Anmodningen giver ikke Datatilsynet anledning til bemærkninger.

Datatilsynets tilladelse forlænges hermed til: 1. maj 2014.

Behandlingen af personoplysningerne kan fortsætte indtil denne dato på de af

Datatilsynet tidligere fastsatte vilkår.

Ved tilladelsens udløb skal du særligt være opmærksom på følgende:

Hvis du ikke inden denne dato har fået tilladelsen forlænget, går Datatilsynet

ud fra, at projektet er afsluttet, og at personoplysningerne er slettet, anonymi-

seret, tilintetgjort eller overført til arkiv, jf. de tidligere fastsatte vilkår om

projektets afslutning. Anmeldelsen af projektet fjernes derfor fra fortegnelsen

over anmeldte behandlinger på Datatilsynets hjemmeside.

Datatilsynet gør samtidig opmærksom på, at al behandling (herunder også

opbevaring) af personoplysninger efter tilladelsens udløb er en overtrædelse

af persondataloven, jf. § 70.

Den ændrede anmeldelse offentliggøres i Datatilsynets fortegnelse over an-

meldte behandlinger på tilsynets hjemmeside.

Med venlig hilsen

Anne-Marie Müller

Ph.d.-studerende Carrinna Hansen

Forskningens Hus

Ledreborg Allé 8, 3. sal

2820 Gentofte

Sendt til: [email protected]

9. september 2013 Forlængelse af tilladelse fra Datatilsynet � j.nr. 2012-41-0875

Datatilsynet

Borgergade 28, 5.

1300 København K

CVR-nr. 11-88-37-29

Telefon 3319 3200

Fax 3319 3218

E-mail

[email protected]

www.datatilsynet.dk

J.nr. 2012-41-0875 Sagsbehandler

Anne-Marie Müller

Direkte 3319 3252

Appendix�D

Information Januar 2011

Dato Navn

Dit tlf. nr. hvor jeg kan kontakte dig for nærmere aftale:_______________________________

I forbindelse med forskningsprojektet: �At leve med knogleskørhed � et epidemiologiske og kvalitativt

studie�, har jeg brug for at tale med nogle mennesker, som har fået stillet diagnosen knogleskørhed. Derfor

vil jeg bede dig om at tage stilling til, om du ønsker at deltage i projektet, hvis din scanning viser at du har

knogleskørhed.

Formålet med undersøgelsen er at opnå øget indsigt i menneskers oplevelser og erfaringer med at leve med

knogleskørhed. Formålet med denne nye viden er, at den i fremtiden bliver brugt til at yde bedre støtte og

behandling til mennesker med knogleskørhed.

Interviewene: Hvis du har knogleskørhed og ønsker at deltage i undersøgelsen, vil jeg interviewe dig kort

tid efter diagnosetidspunktet(scanningen) og igen efter ca. 6 måneder og et år. Jeg vil meget gerne interviewe

dig i dit hjem. Men ønsker du ikke det, er det også muligt at gennemføre interviewene på hospitalet.

Interviewene vil handle om, hvordan du oplever at leve med knogleskørhed i din hverdag, og hvordan det

påvirker dig. Jeg vil være interesseret i spørgsmålene:

· Hvordan oplever du at leve med knogleskørhed?

· Hvilke erfaringer har du gjort indtil nu?

· Er der noget du oplever som særligt udfordrende i forhold til at leve med knogleskørhed?

Fortrolighed og rettigheder

Interviewene vil blive optaget på bånd, disse vil blive opbevaret aflåst og udelukkende være tilgængelige for

undertegnede, de vil blive destrueret efter endt databearbejdning. Dette er i overensstemmelse med

persondataloven. Datatilsynet, som er en offentlig instans, har givet mig tilladelse til at gennemføre

undersøgelsen. Tilladelse er også givet af Videnskabs Etisk Komite.

Det er frivilligt at deltage. Du vil være sikret fuld anonymitet, og du kan på ethvert tidspunkt trække dit

ønske om deltagelse tilbage både før, under og efter de tre interviews. Din videre behandling vil ikke blive

påvirket af om du vælger at deltage eller ej.

Jeg har tavshedspligt og må ikke udtale mig til andre om dig og dine oplysninger.

Mange tak for din tid. Du er altid velkommen til at kontakte mig

Med Venlig Hilsen Carrinna A. Hansen. Mail: [email protected] eller mobil tlf. 20974971

RIV��RIV�� RIV

Jeg ønsker at deltage i ovenstående undersøgelse, hvis undersøgelsen viser, at jeg har knogleskørhed.

JA ! Nej !

________ _____________________________________________________________________

Appendix�E1

Information

Dato Navn

Dit tlf. nr. hvor jeg kan kontakte dig for nærmere aftale:_______________________________

I forbindelse med forskningsprojektet: �At leve med knogleskørhed � et epidemiologiske og kvalitativt

studie�, har jeg brug for at tale med nogle mennesker, som har fået stillet diagnosen knogleskørhed. Derfor

vil jeg bede dig om at tage stilling til, om du ønsker at deltage i projektet. Jeg vil bede dig sende dit svar i

den frankerede svarkuvert, også selvom du ikke ønsker at deltage, og da udelukkende med et kryds i Nej.

Formålet med undersøgelsen er at opnå øget indsigt i menneskers oplevelser og erfaringer med at leve med

knogleskørhed. Formålet med denne nye viden er, at den i fremtiden bliver brugt til at yde bedre støtte og

behandling til mennesker med knogleskørhed.

Interviewene: Hvis du har knogleskørhed og ønsker at deltage i undersøgelsen, vil jeg interviewe dig kort

tid efter diagnosetidspunktet(scanningen) og igen efter ca. 6 måneder og et år. Jeg vil meget gerne interviewe

dig i dit hjem. Men ønsker du ikke det, er det også muligt at gennemføre interviewene på hospitalet.

Interviewene vil handle om, hvordan du oplever at leve med knogleskørhed i din hverdag, og hvordan det

påvirker dig. Jeg vil være interesseret i spørgsmålene:

· Hvordan oplever du at leve med knogleskørhed?

· Hvilke erfaringer har du gjort indtil nu?

· Er der noget du oplever som særligt udfordrende i forhold til at leve med knogleskørhed?

Fortrolighed og rettigheder

Interviewene vil blive optaget, disse optagelser vil udelukkende være tilgængelige for undertegnede, de vil

blive destrueret efter endt databearbejdning. Dette er i overensstemmelse med persondataloven. Datatilsynet,

som er en offentlig instans, har givet mig tilladelse til at gennemføre undersøgelsen. Tilladelse er også givet

af Videnskabs Etisk Komite.

Det er frivilligt at deltage. Du vil være sikret fuld anonymitet, og du kan på ethvert tidspunkt trække dit

ønske om deltagelse tilbage både før, under og efter de tre interviews. Din videre behandling vil ikke blive

påvirket af om du vælger at deltage eller ej.

KLIP��..��KLIP�� KLIP

Jeg ønsker at deltage i ovenstående undersøgelse, hvis undersøgelsen viser, at jeg har knogleskørhed.

JA ! Nej !

________ _____________________________________________________________________

Jeg har tavshedspligt og må ikke udtale mig til andre om dig og dine oplysninger.

Mange tak for din tid. Du er altid velkommen til at kontakte mig

Med Venlig Hilsen Carrinna A. Hansen. ____________________________________

Mail: [email protected] eller mobil tlf. 20 97 49 71

Appendix�E2

12. PAPER � I � II - III

Artikler:

(1)

1. Hansen C, Pedersen BD, Konradsen H, Abrahamsen B. Anti-osteoporotic therapy in Denmark--

predictors and demographics of poor refill compliance and poor persistence. Osteoporos.Int.

2013;24[7]:2079-2097.

2. Carrinna Hansen, Hanne Konradsen, Bo Abrahamsen, Birthe D. Pedersen. Women’s experiences

of their osteoporosis diagnosis at the time of diagnosis and six months later: a Phenomenological

Hermeneutic Study, udkast

3. Carrinna Hansen, Hanne Konradsen, Bo Abrahamsen, Birthe D. Pedersen. Women’s Lived

Experiences of learning to live with Osteoporosis – A Longitudinal Qualitative study. Osteoporosis

International, udkast

Documents

Life with osteoporosis œ a cohort and qualitative study