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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
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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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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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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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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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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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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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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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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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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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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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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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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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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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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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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
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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.
length of interview 15-67 min.
length of interview 12-78 min.
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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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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
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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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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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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%)
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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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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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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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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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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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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
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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,
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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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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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findings related to persistence and compliance with anti-osteoporotic medical treatment,
acceptance/decision-making, chronic illness, coping and nursing perspectives.
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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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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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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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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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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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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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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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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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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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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.
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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
terv
iew
gu
ide
: a
t le
ve
me
d k
no
gle
skø
rhe
d
An
de
n o
g t
red
je i
nte
rvie
w r
un
de
Hv
ord
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lev
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at
lev
e m
ed
kn
og
lesk
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ed
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Hv
ilk
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ar
du
gjo
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nd
til
nu
?
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de
r i
forh
old
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dit
liv
me
d k
no
gle
skø
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ke
t
no
gle
væ
sen
tlig
e æ
nd
rin
ge
r si
de
n v
i si
dst
ta
lte
sam
me
n?
Leve
r d
u d
it l
iv a
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erl
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nu
en
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u g
jord
e f
or
et
ha
lvt
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sid
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ilk
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ke
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·
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me
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kn
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·
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er
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-dia
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·
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er
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u g
ern
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il u
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lle
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rtæ
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ind
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afs
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inte
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om
råd
e
Ind
led
en
de
: In
terv
iew
et
vil h
an
dle
om
hvo
rda
n d
u o
ple
ve
r a
t le
ve
me
d k
no
gle
skø
rhe
d i
din
hv
erd
ag
,
og
hv
ad
du
op
lev
er
som
be
tyd
nin
gsf
uld
t i
din
hve
rda
g.
Du
ha
r fo
r n
yli
gt
fåe
t st
ille
t
dia
gn
ose
n k
no
gle
skø
rhe
d (
ve
d
førs
te i
nte
rvie
w r
un
de
).
Liv
et
me
d k
no
gle
skø
rhe
d
Afs
lutt
en
de
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
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