Journal of Clinical Epidemiology 66 (2013) 994e1005

A grounded patient-centered approach generated the Personal andSocietal Burden of Osteoarthritis model

Lucy Busijaa,e,*, Rachelle Buchbinderb,c, Richard H. OsbornedaMelbourne EpiCentre, Department of Medicine, University of Melbourne and Melbourne Health, 7 East, Royal Melbourne Hospital, Parkville,

Melbourne VIC 3050, AustraliabMonash Department of Clinical Epidemiology, Cabrini Hospital, Malvern, Suite 41 Cabrini Medical Centre, 183 Wattletree Road, Malvern,

VIC 3144, AustraliacDepartment of Epidemiology and Preventive Medicine, School of Public Health and Preventive Medicine, Monash University, Melbourne,

Victoria, AustraliadDepartment of Public Health, Public Health Innovation, Population Health Strategic Research Centre, Faculty of Health, Deakin University,

Burwood Campus, 221 Burwood Highway, Melbourne VIC 3125, AustraliaeDepartment of Public Health, Faculty of Health, Deakin University, 221 Burwood Hwy, Burwood, Melbourne, Victoria, Australia

Accepted 18 March 2013; Published online 15 June 2013

Abstract

Objective: To develop a conceptual model to describe the burden of osteoarthritis in individuals with this condition and on the broadercommunity.

Study Design and Setting: Six concept mapping workshops were undertaken: three with patients (n 5 26) and three with health pro-fessionals (n 5 27) in Australia and Sweden. The participants were asked to generate statements describing how osteoarthritis affects in-dividuals with this condition and those around them. The results were used to construct an integrated theoretical model of the personalburden of osteoarthritis (PBO) and its extended version, the personal and societal burden of osteoarthritis (PSBO) model.

Results: We identified eight potentially independent aspects of PBO, including physical distress, fatigue, physical limitations, psycho-social distress, physical deconditioning, financial hardship, sleep disturbances, and lost productivity. Physical distress and impaired physicalfunction combined to produce psychosocial problems, reduced work productivity, financial difficulties, and loss of physical fitness at theindividual level, and increased health and welfare cost at the societal level.

Conclusion: The PBO and PSBO models were developed directly from the views of patients and clinicians and provide new insights formanaging the individual and societal burden of osteoarthritis. � 2013 Elsevier Inc. All rights reserved.

Keywords: Theoretical model; Osteoarthritis; Burden of illness; Patient-centered outcomes; Quality of life; Mixed methods research

1. Introduction

Osteoarthritis is a large and increasing burden on soci-ety, accounting for 3% of total morbidity burden worldwide[1]. Already one of the most prevalent chronic conditions

Conflict of interest: The authors declare that they have no competing

interests in relation to this article.

Funding: The work by Dr L.B. was supported by a University of Mel-

bourne Postgraduate Research Scholarship, the Universitas 21 Solander

Travel Scholarship, and a postdoctoral fellowship jointly funded by theMel-

bourneCenter for Epidemiology,Biostatistics, andHealth ServicesResearch

and the Center for Research Excellence in Translational Neuroscience. Prof

R.B. is supported in part by a National Health andMedical Research Council

(NHMRC) Practitioner Fellowship. Prof R.H.O. was supported in part by an

NHMRC Population Health Career Development Award.

* Corresponding author. Faculty of Health, Deakin University, Mel-

bourne Campus at Burwood, Building F, Room F3.22, 221 Burwood High-

way, Burwood, Victoria 3125, Australia. Tel.: þ61-3-9244-6745x4409.

E-mail address: l.busija@deakin.edu.au (L. Busija).

0895-4356/$ - see front matter � 2013 Elsevier Inc. All rights reserved.

http://dx.doi.org/10.1016/j.jclinepi.2013.03.012

in developed [2e4] and developing [5,6] countries osteoar-thritis is expected to become even more prevalent in the fu-ture, largely because of the aging population [7] and raisingprevalence of obesity [8]. Estimation of needs for healthservices and the effectiveness of these services requires de-tailed knowledge of the ways in which individuals with os-teoarthritis are affected and the availability of measures thatcan capture these affects comprehensively and accurately.

Interest in the individual burden of osteoarthritis is notnew, and a number of qualitative and quantitative studieshave examined this topic. However, qualitative studies tendto have narrow scope, such as improvement of osteoarthritismanagement in primary care settings [9], personal meaningof osteoarthritis [10], and perceptions of pain [11,12] andfatigue [13]. Quantitative inquiries into the individual bur-den of osteoarthritis also provide limited insights into thenature of individual burden of osteoarthritis, either focusing

995L. Busija et al. / Journal of Clinical Epidemiology 66 (2013) 994e1005

What is new?

� This article has demonstrated how the applicationof a rigorous set of qualitative and quantitative re-search methods provided a firm foundation for thedevelopment of a novel, explicit, and testablemodel of the personal burden of osteoarthritis(PBO).

� The model was developed through genuineengagement with patients, clinicians, and policy-makers and identified eight potentially independentaspects of PBO, including physical distress, fa-tigue, physical limitations, psychosocial distress,physical deconditioning, financial hardship, sleepdisturbances, and lost productivity.

� The results will be used to guide the developmentand evaluation of a new questionnaire to measurethe individual burden of osteoarthritis.

on one specific area, such as financial impact [14], generalhealth [15], or disability [16], or on its global impact onquality of life [17,18]. Moreover, the choice of outcomesfor the assessment of the burden of osteoarthritis in quanti-tative studies is often guided by pragmatic considerations,such as availability of measures [19], or tradition-basedoutcomes rather than the empirical evidence of the rele-vance of outcomes to individuals with this condition.

A strong and increasingly recognized basis for selectingappropriate outcome measures is a conceptual model[20,21]. A conceptual model is preferred and helpful for se-lecting outcomes as it provides a means of determining theappropriateness of questionnaire content. Current lack ofa systematic approach to conceptualizing the individualburden of osteoarthritis limits the understanding of theoverall impact of this condition and can lead to miscommu-nication, fragmented service delivery, use of inappropriateoutcome measures, and misallocation of resources. Not sur-prisingly, a number of studies have reported that there isa mismatch between the expectations and the experiencesof people with osteoarthritis and those of the medical com-munity [9,11], with patients frequently reporting that theirneeds and priorities are not given sufficient considerationby the medical professionals [22,23]. These findings high-light the need, in clinical and health policy settings, fora unifying conceptual model of the burden of osteoarthritisthat accurately represents experiences of individuals withthis condition and integrates views of medical and patientcommunities.

The primary aim of this study was to identify majordimensions of the individual burden of osteoarthritis. How-ever, understanding of the consequences of a chronic con-dition at an individual level would not necessarily provide

information on how problems at a personal level exert theirinfluence at the community level. The secondary aim was toconduct preliminary exploration of how the individual bur-den of osteoarthritis impacts on the broader society. For thepurpose of this study, the individual burden of osteoarthritiswas defined as the perceived impact of this condition onany aspect of an individual’s life. The societal burden of os-teoarthritis was defined as the impact of this condition onthe patient’s social and community networks.

2. Methods

2.1. Ethics approval

This research was carried out in accordance with theDeclaration of Helsinki for Ethical Principles for MedicalResearch Involving Human Subjects. Approval for thisstudy was obtained from the Human Research EthicsCommittees of the Royal Melbourne Hospital, Melbourne,Victoria, Australia (approval number 2006.135); CabriniHospital, Melbourne, Victoria, Australia (approval number04-10-07-06); and the University of Melbourne (approvalnumber 060419). All participants provided written in-formed consent for the study.

2.2. Sample

In assembling workshop groups, care was taken to max-imize diversity both within and between groups. Individualswith osteoarthritis were recruited through the rheumatologyoutpatient clinic at one large community public hospital(Royal Melbourne Hospital) and one community-basedrheumatology practice (Cabrini Hospital) in Melbourne,Victoria, Australia. Patient groups were assembled to en-sure diverse representation of experiences, including jointaffected, osteoarthritis severity, socioeconomic and linguis-tic backgrounds, age, gender, and work status. Eligibilitycriteria were (1) existing clinical diagnosis of osteoarthritis,(2) English language proficiency, and (3) good cognitivefunctioning. Health professionals groups were assembledto include a diverse range of professional backgrounds.Two professional workshops were conducted in Melbourne,Australia, and one was conducted in Lund, Sweden. Partici-pants were purposefully selected from among individualswho had direct connections with the care of people with os-teoarthritis (orthopedic surgeons, rheumatologists, physio-therapists, occupational therapists, and aged care nurses)or planning, funding, and evaluation of osteoarthritis-specific health services and programs (at the State or Na-tional levels).

2.3. Procedure

Data were obtained directly from individuals usinga concept mapping (CM) approach [24,25] (described sub-sequently). Six workshops were conducted, including three

996 L. Busija et al. / Journal of Clinical Epidemiology 66 (2013) 994e1005

with patients and three with health professionals. Two ofthe workshops were facilitated by an external consultant ex-perienced in CM, three workshops were facilitated by L.B.,and one additional workshop was jointly facilitated by theconsultant and L.B. The decision to limit the number ofworkshops to six was based on thematic saturation. The-matic saturation was reached after the second workshopfor both patients and professionals, with very few conceptsemerging in the third session with either group ofparticipants.

3. Concept mapping

Each CM session had four major components [26]: (1)brainstorming, (2) sorting and rating, (3) data analysis,and (4) interpretation of the concept map.

3.1. Brainstorming

During the brainstorming session, a seeding statementwas used to guide generation of concepts by participants[24]. Separate seeding statements were developed for thepatients and professionals:

Patient seed: Thinking as broadly as you can, generatestatements about how osteoarthritis affects your life (con-sidering both yourself and those around you).

Professional seed: Thinking as broadly as you can, gen-erate statements about how osteoarthritis affects the life ofpeople with the condition and those around them.

Participants were instructed to form statements, eachcontaining one idea only. Statements were recorded verba-tim. There was no discussion or critical appraisal of thestatements during brainstorming, although the participantswere encouraged to seek clarification of unclear or ambig-uous statements [24]. Within each workshop, the process ofbrainstorming continued until no new statements emerged.

3.2. Sorting

Each statement was printed onto individual cards, andeach person was asked to sort the statements into piles, ina way that ‘‘made sense to them.’’ The only restrictionswere that (1) all statements should be sorted, (2) thereshould be more than one pile, (3) the number of pilesshould be less than the number of statements, and (4) thereshould not be a ‘‘miscellaneous’’ pile. The participantswere also asked to name each pile.

3.3. Data analysis

The Concept System software Version 1 [27] was usedfor data analysis. It uses two major analytic approaches:nondistributional two-dimensional multidimensional scal-ing (MDS) analysis and hierarchical cluster analysis.MDS analysis transforms the sorted data into distances,with smaller distances indicative of statements that were

sorted together more frequently (i.e., statements that wereperceived by the participants to be conceptually similar).Information on item distances generated using MDS wasthen used as input for cluster analysis to identify groupsof related statements. Although the clustering process canbe repeated until all statements are combined into a singlecluster, the Concept System software produces a default so-lution with the number of clusters equal to approximatelyone-fifth of the number of statements. This default solutionwas examined for interpretability by the research team, andif necessary, the number of clusters was amended to pro-duce a more coherent solution, which was then presentedback to the participants for naming and interpretation ofclusters.

3.4. Interpretation of the concept map

In the final stage of each CM workshop, participantswere presented with the cluster solution and asked to re-view the cluster composition for interpretability. Followingthis, a group discussion was held, with participants asked tosuggest a label for each cluster that best represented contentof this cluster. Group consensus was sought for each clusterlabel. Once cluster naming was complete, participants wereasked to reexamine the map to see whether some of theclusters could be grouped together into regions of concep-tually related clusters or whether some clusters should besplit. The participants were also asked whether they feltthere were some concepts missing from the map [This in-formation was used in the construction of a tool to measurePBO (described elsewhere)]. Finally, the participants wereasked to reflect on how well they felt the cluster map rep-resented their experiences and whether it ‘‘made sense’’overall.

3.5. Postworkshop analyses

Although two-dimensional concept maps tend to be eas-iest to display and interpret, previous studies reported thatthree-dimensional solutions are better able to capture simi-larities between statements and thus produce clusters thatare more homogeneous conceptually [25,28]. Postwork-shops, the sorted data from the individual workshops, weretransferred to Clustan software (Clustan, Ltd., Edinburgh,Scotland) [29], and cluster analysis was repeated usingthree-dimensional MDS as input. Beginning with the num-ber of clusters generated during a given workshop, solu-tions with progressively fewer clusters were produced andtheir content examined to determine whether cluster aggre-gation made sense. This was done to find the smallestmeaningful number of overarching ‘‘super clusters’’ inthe data, representing the most general concepts. In the nextstep, we repeated cluster analysis; this time splitting clus-ters from the initial solution into progressively smaller clus-ters with the solution reexamined at each split to determineif the split was conceptually meaningful. This step allowed

997L. Busija et al. / Journal of Clinical Epidemiology 66 (2013) 994e1005

us to determine the largest number of subdimensions pres-ent in the data.

The results of all workshops were then concatenated inconsensus workshops by the research team. This beganwith the statements from each workshop being printedout onto a set of cards [30] and sorted into the overarchingclusters derived from the three-dimensional cluster analy-ses. Two clusters from different workshops that were mostsimilar in content were then identified and paired off. Theprocess continued until all clusters had been included intoa synthesized group. The content of synthesized clusterswas reviewed and labels assigned. Where possible, clusterlabels were selected from the cluster names generated bythe participants during cluster interpretation task. If no suit-able label could be found to describe the aggregated cluster,the research team selected the most appropriate clustername from the pile names proposed by participants duringthe sorting process.

The next step in the development of a conceptual modelwas the exploration of putative interrelationships betweendifferent dimensions. In this study, the integration of differ-ent components of the model was accomplished using a rela-tional algorithm [31]. A relational algorithm is ‘‘a linguisticexercise that can identify unexamined relationships amongconcepts’’ ([31], p. 18). The algorithm was created by exam-ining the full set of statements generated during the work-shops to find statements that linked different aspect of theburden of osteoarthritis, as perceived by the participants.Concepts represented in these statements were then identi-fied and the nature of the relationship recorded. One exampleof a linking statement is ‘‘I get really depressed because Ican’t do my work around the house.’’ The statement containsreference to two concepts: depression and inability to dousual housework. The direction of effect suggests that inabil-ity to do housework contributes to depression. Linking state-ments were used to construct a flow chart of osteoarthritisburden. This step completed the development of the PBOconceptual model that was aimed to capture the burden ofthis condition at an individual level.

In the final step, we reviewed the content of synthe-sized clusters to identify statements describing factorsoutside osteoarthritis influencing the impact of this

Table 1. Summary of participation and major results of the workshops

Group number Location Number of a

Patient groups1 Cabrini Hospital 82 Royal Melbourne Hospital 83 Royal Melbourne Hospital 10

Total 26Professional groups4 Royal Melbourne Hospital, Melbourne 115 Rydges Hotel, Canberra 96 Lund University Hospital, Sweden 7

Total 27Grand total 53

a Two statements were later removed as repetitive; analyses for this gro

condition. The statements were coded according to thoseaffected (e.g., close others) and the type of impact (e.g.,emotional impact) and whether they were perceived bythe participants to exacerbate or reduce the burden of os-teoarthritis. Examples of contextual factors include socialsupport seeking (‘‘Important that you should find a supportgroup’’), accepting the situation (‘‘You need to accept thatpain is a part of the condition and that it is ok to take med-ication’’), and keeping a positive outlook [‘‘Past experi-ence can help you cope (helps keep things inperspective)’’]. Information on contextual factors wasused to represent a flow of osteoarthritis burden fromthe individual to community levels and construct a concep-tual model of the personal and societal burden of osteoar-thritis (PSBO).

4. Results

4.1. Participants

Of the 86 individuals with osteoarthritis invited to takepart in the study, 29 (34%) responded. During a screeningphone interview, two individuals were found to be ineligiblebecause of signs of cognitive impairment and one was ineli-gible because of insufficient English. The 26 eligible patientswho took part ranged in age between 51 and 80 years (M,63.9; SD, 8.3); 17 (65%) patients were female, and seven(27%) patients had English as their second language. Themajority (n 5 14) had osteoarthritis in the hip and/or knee,7 individuals had osteoarthritis of the hand, and the remain-ing 5 individuals had generalized osteoarthritis.

From 48 Australian-based professionals (health profes-sionals and policy makers) invited to participate, 20 (42%)took part. For the Swedish-based workshop, 11 health pro-fessionals were invited, with seven (64%) taking part.

4.2. Workshop statements and maps

The number of statements generated during the individ-ual CM workshops varied between 48 and 101, witha median of 68 statements (M, 71.7; SD, 20.5), and430 statements were generated in total (see Table 1).

ttendees Number of statements generated Number of clusters

48 990 12

101a 9239

72 1664 1255 8

191430 66

up were based on 99 statements.

Missing: • Joint deformity • Appearance

Mobility/disability/ movement/ walking

Pain effects on everyday activities

Limitations, loss of activities

Restrictions,

limitations of

activity

Pain, management of pain

Surgical effects

Adaptation and relationships

Age and economic effects (results, prior expectations) (life course/trajectory)

Frustration (items relate to other clusters)

Worries about causes and effects

Pain and its management and impact

Global

Fig. 1. Concept map generated during a concept mapping workshop with osteoarthritis patients (group 1). This concept map is based on 48 state-ments grouped into six major ideas or clusters. Each numbered point corresponds to a statement that was generated by a participant. The state-ments that are positioned closer to each other on the map are those that tended to be grouped together more frequently (perceived to beconceptually similar) by workshop participants, whereas those statements that are further apart tended to be grouped together less frequently (con-ceptually less similar). Solid black lines represent cluster borders, with cluster names appearing next to each cluster. Dotted red lines identifystatements that were perceived by the group to have poor conceptual fit with their cluster; the corresponding arrow for statement 37 from cluster5 indicates group’s consensus that this statement belongs to cluster 4. Bidirectional arrow between clusters 3 (limitations and loss of activities)and 4 (pain and management of pain) indicates a close association between these clusters. Broken red lines represent overarching dimensions ofthe burden of osteoarthritis, with names given in blue font. Text box in the bottom left hand corner identifies issues that did not come up duringbrainstorming but that were identified during group discussion as being important aspects of the osteoarthritis burden (For interpretation of thereferences to colour, please refer the web version of this article).

998 L. Busija et al. / Journal of Clinical Epidemiology 66 (2013) 994e1005

The number of clusters in each workshop varied bet-ween 8 and 16, with a median of 11 clusters (M, 11.0;SD, 3.0).

Examples of the concept maps generated in the individ-ual workshops are shown in Figures 1 and 2. A conceptmap generated by individuals with osteoarthritis (Fig. 1)contains nine clusters. The group also identified threebroader areas of osteoarthritis impact, including pain andits management and impact, restrictions/limitations of ac-tivity, and global effect. In an example of a concept mapfrom a workshop with professionals (Fig. 2), 12 clusterswere identified and named and two broader areas of osteo-arthritis impact: management and risks and psychosocialeffects. The remaining concept maps and lists of clusteredstatements generated from the individual workshops areavailable on request.

Cluster names from each workshop, together with thenumber of statements generated, are presented in Table 2(patient groups) and Table 3 (professional groups). Overall,there was substantial variability in the number and types ofdimensions of osteoarthritis burden across the workshops,indicating that it is a complex and multidimensionalconstruct. There were also some differences in the concep-tualization of osteoarthritis burden between patients andprofessionals. Specifically, statements related to pain andphysical limitations occurred more frequently in the patientthan in the professional groups, whereas in professionalworkshops, more emphasis was given to psychosocial ef-fects of osteoarthritis.

4.3. The PBO model

Cluster analysis based on three-dimensional MDSidentified eight major concepts within the combined datarelated to the PBO, including physical distress, psychoso-cial distress, physical limitations, fatigue, sleep distur-bances, physical deconditioning, financial impact, andlost productivity. Results are presented in Table 4. Distin-guishable subdimensions were also identified within physi-cal distress, physical limitations, and psychosocial distressclusters. The physical distress cluster included subdimen-sions that represented manageability of pain, treatmenthelpfulness (representing treatment efficacy and acceptabil-ity of side effects), health services use (need for pain con-trol), and symptoms and signs. Statements in themanageability of pain group represented emotive signifi-cance of living with unpredictable and uncontrollablepaindthe individual pain and suffering (‘‘Painful existence24/7’’). The treatment helpfulness subdimension capturedconcerns with lack of safe and effective pain control[‘‘No effective pain relief,’’ ‘‘Still have pain even afterthe operation (joint replacement surgery),’’ ‘‘Need medica-tion to deal with medication side effects’’]. The health ser-vices use subdimension represented increased use of healthcare related to the need for pain control [‘‘You’ll try any-thing (medications) to stop pain’’]. The symptoms andsigns subdimension represented direct manifestations ofosteoarthritis-related pathophysiology with five signs andsymptoms identified: (1) pain [e.g., ‘‘Pain affects ability

Fig. 2. Concept map generated during a concept mapping workshop with professionals (group 5). This concept map is based on 64 statementsgrouped into 12 major ideas or clusters. Each numbered point corresponds to a statement that was generated by a participant. The statementsthat are positioned closer to each other on the map are those that tended to be grouped together more frequently (perceived to be conceptuallysimilar) by workshop participants, whereas those statements that are further apart tended to be grouped together less frequently (conceptually lesssimilar). Solid black lines represent cluster borders, with cluster names appearing next to each cluster. Dotted red lines identify statements thatwere perceived by the group to have poor conceptual fit with their cluster; the corresponding arrows indicate group’s consensus on a cluster wherethe statement would fit best. Broken red lines represent overarching dimensions of the burden of osteoarthritis, with names given in blue font. Textbox in the bottom left hand corner identifies issues that did not come up during brainstorming but that were identified during group discussion asbeing important aspects of the osteoarthritis burden (For interpretation of the references to colour in this figure legend, the reader is referred to theweb version of this article).

999L. Busija et al. / Journal of Clinical Epidemiology 66 (2013) 994e1005

for physical activity’’ (Note: emphasis is added.)], (2) jointstiffness (e.g., ‘‘Embarrassing because of pain and stiffnesswhile traveling, especially plane travel’’), (3) muscularweakness in the affected joints (e.g., ‘‘Loss of strength injoints affected by arthritis’’), (4) joint deformities [e.g.,‘‘Body image problems (deformed joints, esp. hands)’’],and (5) reduced range of motion (e.g., ‘‘Difficulty withwriting due to pain and reduced movement in fingers’’).Subdimensions identified within the physical limitationscluster were activity limitations, participation restrictions,demands on time, and loss of self-reliance. The psychoso-cial distress cluster included statements describing relation-ship difficulties and emotional distress.

Examination of linking statements indicated that, asmight be expected with a painful chronic condition, phys-ical distress was central to osteoarthritis-related burden.One of the major effects of physical distress was impairedphysical function (physical limitations: ‘‘When pain is se-vere, I can’t even bend down to put my socks on’’). Phys-ical distress and physical limitations were linked to a rangeof problems, including reduced work productivity (‘‘Oste-oarthritis interferes with your paid workdcan’t stand forlong periods of time’’), increased expenses (‘‘Need to usehome help, which is extra financial burden’’), social prob-lems (‘‘Personal relationships are affected by not beingable to assist others’’), and psychological issues (‘‘Depres-sion due to pain’’ and ‘‘Frustration at not being able to dothings like you used to’’). The assessment of links alsoidentified a positive feedback loop (Fig. 3), in which

physical distress affected sleep (‘‘Your sleep pattern is to-tally disrupted because of pain’’), which then led to fatigue(‘‘Tiredness because of not enough restful sleep’’), whichthen exacerbated physical distress (‘‘Pain is worse when Iam tired’’). A bidirectional relationship between physicaldistress and physical deconditioning was also identified,whereby physical distresses lead to loss of fitness, whichin turn exacerbated joint pain. The major dimensions ofthe PBO and their interrelationships are summarized inFig. 3.

4.4. The PSBO

An integrated PSBO model representing the broader im-pact of osteoarthritis is shown in Fig. 4. The top panel sum-marizes the major aspects of the individual burden ofosteoarthritis (the PBO model). The middle panel presentsthe factors that either reduce (top part) or intensify (bottompart) specific aspects of the burden of osteoarthritis. Thebottom panel summarizes the wider consequences of oste-oarthritis on various sections of the community. The panelsrepresenting the PSBO are joined by two-way arrows to in-dicate reciprocal relationships between the levels of osteo-arthritis burden.

As Fig. 4 indicates, one aspect of the individual burdenof osteoarthritis with a broader impact on society appearsto be physical deconditioning. Participants noted that‘‘Decreased ability to maintain a healthy lifestyle [in-creases] risk of other conditions’’ leads to ‘‘greater risk of

Table 2. Clusters derived from concept mapping workshops with patient groups

Group Cluster Number of statements

1 Cabrini Hospital, Melbourne, Australia1.1 Adaptation and relationships 91.2 Pain and management of pain 61.3 Limitations and loss of activities 61.4 Age and economic effects (results, prior expectations, and life course) 61.5 Pain effects on everyday activities 51.6 Mobility/disability/movement/walking 51.7 Surgical effects 41.8 Worries about causes and effects 41.9 Frustration 32 Royal Melbourne Hospital workshop 1, Melbourne, Australia2.1 Physical ability/capability 162.2 Pain 152.3 Family 132.4 Emotional effects/personal feelings 132.5 Financial implications 102.6 Acceptance of osteoarthritis 92.7 Relationships 82.8 Depression 32.9 Medication issues 33 Royal Melbourne Hospital workshop 2, Melbourne, Australia3.1 Pain and medication 123.2 Impact of pain on leisure activities 113.3 Social problems 113.4 Impact of pain on physical activity 103.5 General ongoing difficulties of living with osteoarthritis 83.6 Mental health consequences of incomplete pain control 83.7 Impact of pain on social activities 83.8 Transport problems 73.9 Difficulties with doing things 73.10 Cost of assistive devices 63.11 Financial impact of osteoarthritis 63.12 Solutions, adjustingdnew ways of doing things 5

1000 L. Busija et al. / Journal of Clinical Epidemiology 66 (2013) 994e1005

falls or injuries’’ and affects management of comorbid con-ditions (‘‘Loss of ability to exercise ‘mucked up’ my con-trol of diabetes’’ and ‘‘Used to use exercise to cope withdepression, but can’t exercise now because of osteoarthri-tis’’). ‘‘Obesity arising from decreased physical activity’’also contributes to the increasing obesity problem in soci-ety, placing further demands on health resources.

5. Discussion

This study describes the development of a conceptualmodel representing the burden of osteoarthritis from theperspective of individuals with this condition. Eight majordimensions of the individual burden of osteoarthritis wereidentified, including physical distress, physical limitations,psychosocial distress, financial hardship, fatigue, sleep dis-turbances, physical deconditioning, and lost productivityusing CM methodology [32,33]. Another major outcomeof this study was the development of an integrated PSBOmodel, which summarizes the burden of osteoarthritis onthe wider community.

The PBO model indicates that the physical pain of oste-oarthritis is central to individual burden of this condition.Although several studies have highlighted the pivotal role

of pain in the individual experience of living with osteoar-thritis (e.g., [10,34]), the results of this study also indicatethat the individual burden of osteoarthritis pain results froman interplay of physical and emotional factors. The physicalsensation of pain, discomfort because of muscular weak-ness and joint stiffness, the desire to be free from pain,and limited pain relief combine to create a state of continu-ing inescapable sense of suffering. This state of physicaldistress is closely linked with impaired physical function,impacting on psychosocial well-being, work productivity,financial well-being, and physical fitness. Although theseresults may appear intuitive and unsurprising, this is thefirst time the individual burden of osteoarthritis has beenformally expressed in a conceptual model.

A further important outcome of this study was the devel-opment of the PSBO model, which summarizes the burdenof osteoarthritis on the wider community. There is some ev-idence that community-wide interventions can be effectivein the reduction of health-related community burden. Forexample, changing community attitudes toward back painthrough a media-based community education campaignwas effective in reducing time off work because of backpain [35]. Similarly, providing assistive devices to elderlycommunity members was shown to improve their indepen-dence and lead to reduced health services utilization [36].

Table 3. Clusters derived from concept mapping workshops with professional groups

Group Cluster Number of statements

4 Melbournea, Australia4.1 Limitations of treatment/health care issues 164.2 Psychological/emotional effects 154.3 Impact on relationships and roles 124.4 Financial impact 104.5 Physical effects of osteoarthritis 94.6 Maintaining health 44.7 Pain impact on daily life 35 Canberra, Australia5.1 Psychological: emotional impact 105.2 Participation in a quality life (physical emphasis) 85.3 Economic work impact on financial well-being 75.4 Psychological: social emphasis 75.5 Relationships 65.6 Diseases and health issues/treatment/comorbidities 65.7 Medical managementdperceived quality 55.8 Health system burden 55.9 Participation in management 45.10 Sense of isolation 35.11 Potential positive impact 36 Lund, Sweden6.1 Relations/participation 136.2 Leisure activities 126.3 Long-term health consequences 86.4 Psychological consequences 76.5 Pain 56.6 Economic consequences: society 56.7 Economic consequences: individual 46.8 Sexual function 1

a Cluster names are based on the results of three-dimensional analyses by the researcher; no cluster-naming task was conducted with this groupbecause of technical difficulties with processing the data during the workshop; cluster names were chosen from individual sort results.

1001L. Busija et al. / Journal of Clinical Epidemiology 66 (2013) 994e1005

The PSBO model offers a way to integrate potential inter-ventions and direct them at the specific end points thatare most likely to benefit people with osteoarthritis, thusproviding a targeted approach to reducing the societal bur-den of this condition.

Fig. 3. Conceptual model of the personal burden of osteoarthritis. Ellipses rerectangles represent subdimensions.

Some important differences between results of this studyand current conceptualizing of disease burden representedby the World Health Organisation’s International Classifi-cation of Functioning, Disability, and Health (ICF) model[37] should be noted. In comparison with the PBO model,

present major dimensions of the personal burden of osteoarthritis and

Fig. 4. Conceptual model of personal and societal burden of osteoarthritis. The top panel represents major dimensions of the personal burden ofosteoarthritis (note that sleep and fatigue were omitted as no statements were identified to link these dimensions with the specific sector of com-munity impacted by osteoarthritis), the bottom panel represents the societal burden of osteoarthritis, and the middle panel represents contextualmodifying factors that either reduce or increase the burden of osteoarthritis. The bidirectional arrows represent reciprocal relationships between thepersonal and the societal burden of osteoarthritis.

1002 L. Busija et al. / Journal of Clinical Epidemiology 66 (2013) 994e1005

the ICF covers a narrower set of dimensions. For example,in ICF, emotional difficulties are conceptualized as part ofthe general impairment of body functions rather than a sep-arate dimension. The PSBO, compared with the ICF, alsooffers a more explicit set of interrelationships betweenthe individual and the community burdens. In the ICF, suchrelationships are conceptualized as ‘‘a dynamic interactionbetween health conditions (diseases, disorders, injuries,traumas, etc.) and contextual factors’’ (p. 665 [37]). ThePBO and PSBO models can be used to complement theICF, especially when it is important to distinguish betweendifferent dimensions of the burden of osteoarthritis.

However, it is important to note that like any conceptualmodel, the models developed in this study offer a simplifiedschematic representation of complex reality. Evidence ofthe stability and repeatability of the dimensional structureof the models await further studies. The links between di-mensions of the PBO model and between individual andcommunity burdens in the PSBO model in particular needcorroboration as these were derived from secondary analy-sis of workshops data, hence not all pertinent interrelation-ships may have been captured.

One of the strengths of this study is that the emergentmodels were derived from the ground up, with participantsinvolved in data generation and analysis. The workshopsused a mix of qualitative and quantitative approaches todata collection and analysis. The use of mixed methodol-ogy enhances objectivity and potential repeatability of thefindings, at the same time allowing in-depth explorationof the meanings in the data [38,39]. The participants’ con-ceptual representations of the categories of osteoarthritisburden (reflected in their sort results [40]) were used asa basis for deriving dimensions of the PBO model, furtherdecreasing influence of the investigator on the results.

A potential limitation of this study is that two different fa-cilitators conducted the CM workshops. Consequently, thesubstantial variability in the number and composition of di-mensions that emerged in different workshops may reflectdifferences in personal styles of the facilitators. However,this could also be seen as strength because the differencesin the facilitators’ styles may have generated greater breadthand depth of information. An additional limitation of thisstudy was that only English-speaking individuals took partin the development of the PBO and PSBO models; hence,

Table 4. Major concepts and subdimensions of the personal burden of osteoarthritis generated from the synthesis of the results of structuredconcept mapping workshops

Concepts and their definitions

1. Physical distress. Captures ‘‘Pain and suffering’’ aspects of osteoarthritis. Major themes are1.1. Symptoms and signs: Direct manifestations of osteoarthritis. Five signs and symptoms were identified: pain, joint stiffness, muscle

weakness in the affected joints, reduced range of motion, and joint deformities. Example statementsPainLoss of strength in joints affected by arthritisLimited range of motion

1.2. Manageability of pain: ongoing physical discomfort and uncontrollable and unpredictable fluctuations in pain. Example statementsPainful existence 24/7Dealing with pain is difficult because it is constant and chronicNot being able to plan things in advance because you do not know how you are going to feel on the day

1.3. Treatment helpfulness: Perceptions of lack of helpfulness of the available pain management options, including poor effectiveness atcontrolling pain, inconvenience of use, and unacceptability of side effects. Example statementsNo effective pain reliefStill have pain even after the operation (joint replacement surgery)You need medication to deal with medication side effects

1.4. Health services use (need for pain control): Increased use of health care resources, driven by the need to be free from pain. ExamplestatementsYou will try anything (medications) to stop painHave to keep trying different medications to find one that will suitNeed for regular and ongoing commitment to participate in treatment

2. Psychosocial distress. This dimension represents emotional and interpersonal difficulties experienced in response to osteoarthritis. Majorthemes are2.1. Relationship difficulties: Difficulties in interpersonal relationships. Example statements

Osteoarthritis affects relationships with partnersCan be difficult for family members to cope with my mood swings (because of pain of osteoarthritis)Family feels cutoff from me when I am in extreme pain

2.2. Emotional distress: Emotional difficulties associated with having osteoarthritis. Example statementsDepression because of painFrustration and angerIt is degrading to always ask for help

3. Physical limitations. This domain assesses limitations in tasks of daily life because of osteoarthritis. Major themes are3.1. Activity limitations: Osteoarthritis-related limitations in tasks of daily life. Statements in this domain represent impairments in mobility and

self-care limitations. Example statementsDifficulty with shoppingDifficulty using public transport because of stepsHave to ask people to help with personal hygiene tasks

3.2. Participation restrictions: Difficulty with becoming involved in usual life situations, including leisure activities, and community anddomestic life. Example statementsLimitations in participation (type of activities)Restricts participation in volunteer workCannot take part in sporting activities and are reduced to being a spectator

3.3. Loss of self reliance: Increasing reliance on others for assistance with-day-to-day tasks. Example statementsBurden on time and effect for carersLoss of independence (have to ask others for help)Need to keep asking household members to help around the house

3.4. Demands on time: Extra demands on time from osteoarthritis-related physical limitations and treatment needs. Example statementsEverything takes more time, so less time for anything elseSpecialist and outpatient appointments for osteoarthritis are time consumingI do things really slowly because of osteoarthritis

4. Financial hardship: Osteoarthritis-related difficulties with paying for day-to-day and medical expenses. Example statementsIncreased costs because of health care expensesAdded expense of changing your furnitureIncreased costs for assistance

5. Fatigue: Tiredness and loss of energy. Example statementsFeel more tired (because of medication, pain, etc)Tiredness because of not enough restful sleepPain is worse when I am tired

(Continued )

1003L. Busija et al. / Journal of Clinical Epidemiology 66 (2013) 994e1005

Table 4. Continued

Concepts and their definitions

6. Sleep disturbances: This cluster reflects problems with falling asleep, waking up through the night, and reduced overall sleep quality because ofpain. Example statementsWorst part of osteoarthritis is that it affects ability to sleepPain wakes me up at nightYour sleep pattern is totally disrupted because of pain

7. Physical deconditioning: Loss of fitness and deterioration in general physical condition. Example statementsObesity arising from decreased physical activityVicious circledthe more weight you have, the worse you feel and the less able you are exercisePhysical deconditioning (e.g., leading to greater risk of falls or injuries)

8. Lost productivity: Reduced on-the-job effectiveness and work absences because of osteoarthritis. Example statementsReduce effectiveness of workIncreased sick leave costsLoss of productivity for people affected

1004 L. Busija et al. / Journal of Clinical Epidemiology 66 (2013) 994e1005

the applicability of these models to people from non-Englishspeaking backgrounds is not known. The applicability of thePBO and PSBO models outside Australia also requires ex-amination. The low participation rate for both patients andprofessionals can also be seen as a limitation. However, inqualitative studies, diversity of opinion and sample composi-tion are more important considerations than sample size andrepresentativeness. In assembling CM groups, care was takento maximize diversity both within and between groups. Re-sponse rates had no bearing on intended composition of CMgroups, with data collection continuing until theoretical sat-uration was reached.

This study presents a PBO model that was designed torepresent the burden of osteoarthritis on individuals with thiscondition as perceived by patients and professionals. The ex-tension of the PBO model, the PSBO model, proposes thepathways from the individual to the societal burden and iden-tifies clear targets for interventions. The information pre-sented in this article can greatly aid in the development ofpossible interventions to optimize outcomes for individualswith osteoarthritis and the broader society.

Acknowledgments

The authors thank patients and professionals who do-nated their time to this study; Roy Batterham, Plexus Con-sulting, Melbourne, who facilitated some of the CMworkshops; Professor Eva Roos who played a key role inorganizing the Sweden-based workshop. The authors alsothank Ms Kristina Theis and Dr Gerald Ellsworth for theirinsightful comments on earlier drafts and two anonymousreviewers whose insightful suggestions have helped to im-prove the quality and clarity of this manuscript.

Appendix

Supplementary data

Supplementary data related to this article can be found athttp://dx.doi.org/10.1016/j.jclinepi.2013.03.012

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