Introduction

Over past decades, major advances have been made in the effectiveness of treating osteoporosis or fragility fractures. However, a substantial treatment gap exists between recommended anti-osteoporosis medication (AOM) and the actual prescription initiation or patient adherence. Defined as the percentage of eligible individuals not receiving treatment with anti-osteoporosis medication, the treatment gap actually increased in Europe from 55% in 2010 to 71% in 2019 [1, 2]. Furthermore, only between 17 and 75% of patients eligible for treatment are adherent 1 year after initiation [3].

Medication non-adherence is multidimensional (comprising initiation, implementation, or persistence) and multifactorial. Non-adherence can be condition-related (e.g. polypharmacy and medical history), patient-related (e.g. lower education levels, lack of perceived benefits of therapy), therapy-related (e.g. medication side effects, complex medication administration instructions, and medication regimens), health system-based (e.g. fragmented care and lack of patient education and support services), and related to socio-economic factors (e.g. lower income level and lack of medical insurance coverage) [4]. Among the factors contributing to non-adherence, insufficient information and unaddressed patient values and preferences are specifically important [5,6,7]. Shared decision-making (SDM) with decision aids (DAs) offers incorporation of best available evidence on benefits and harms of treatment options whilst simultaneously accounting for patients’ values and preferences through patient-centred care [6,7,8,9].

Fracture liaison services (FLS) have been developed and implemented to identify and evaluate patients who had a recent fracture and have osteoporosis or prevalent vertebral fractures. It is well known that these patients are at high risk for recurrent fractures and pre-emptive treatment is both highly successful and cost-effective [2, 10]. One of the challenges is the high proportion of patients who do not respond to their FLS invitation [11]. If patients do respond and are eligible for AOM, treatment initiation and medication adherence remain low. A study among Dutch patients visiting the FLS and eligible for AOM revealed between 60 and 72% do not initiate or adhere to AOM [2, 12]. FLS therefore represents an opportunity to substantially reduce the subsequent fracture risk by improving treatment uptake and adherence [2, 3, 6, 10]. By integrating SDM using DAs within FLS, high-quality decisions can be established, as patients are fully informed by facts and best available evidence on treatment options and can assimilate those facts into their needs, values, and preferences [13, 14].

In 2016, Hiligsmann et al. developed a 4-page DA for post-menopausal women with osteoporosis, addressing (i) information about osteoporosis and the individualized fracture risk, (ii) clarification of the decision that should be taken with an explanation of the risk of future fracture without medication treatment (natural course), (iii) explanation of the reduction in fracture risk when accepting medication treatment, and (iv) an overview summary (option grid) of first and second line treatment options with medication information such as mode and frequency of administration [15]. Overall, healthcare providers (HCPs) and patients expressed a positive attitude towards the intended use of the proposed DA [15]. However, for use in FLS, adjustments to this DA are required, as the risk of a subsequent fracture without medication (natural course) is much higher in patients with recent fractures and osteoporosis or vertebral fractures. Additionally, the DA for post-menopausal women with osteoporosis had not yet included the potential harms of AOM or option grid and there was no specific page to allow patients to deliberate on the choice or for expression of personal preference and deliberation. The need for a high-quality DA for use in the FLS was further motivated by a planned experimental study to evaluate effectiveness and cost-effectiveness of SDM using a DA combined with motivation interviewing [15, 16]. The aim of this study is to report on the development and usability of a DA in a Dutch FLS setting.

Methods

Updating and adjustment of the existing DA were performed in concordance with the updated International Patient Decision Aid Standards (IPDAS) and the recent Dutch guidelines on SDM [17,18,19,20]. Whilst the final version of the DA will be used in an experimental study (referred to as the ‘beta test’), this paper describes all steps except the beta testing. The key elements of the development and testing process are shown in Fig. 1.

Fig. 1
figure 1

Development model of our decision aid based on Witteman et al. [18] and role of steering group and patients. *The Beta testing is performed in an ongoing trial. HCP, healthcare professionals

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Prior to initiating the scope phase, a steering group of ten persons was assembled, including HCPs (endocrinologists, rheumatologists, an osteoporosis nurse practitioner) and research experts (an epidemiologist, health technology assessment expert, implementation scientist, and a shared decision-making expert). We collaborated from the onset with the Dutch Osteoporosis Association (DOA), and had regular contact with the Associations’ chair to ensure the DA was aligned with their vision and mission. Although patients were not involved in the steering group, they were actively involved as participant or advisor at several stages of the study; in line with the IPDAS recommendations (Fig. 1) [17, 18].

Scope

From the outset, the SDM tool should support persons over the age of fifty (i) who had a recent fracture, (ii) attended the FLS, and (iii) had a treatment indication for AOM, in their decision to start AOM, and make lifestyle changes. In line with advices on SDM in the Netherlands, management options should follow the most recent Dutch management recommendations which contains general lifestyle recommendations as well as first and second line medication options for patient sub-groups [21]. The steering group agreed to develop a paper DA to be administered by an HCP during the FLS visit. A paper format was preferred by the steering group, as we learned from a previous developed DA that patients like to take the DA home, especially because it includes the notes by the HCP on personal risk factors, specific decision, and future appointment [22].

As a further context to the scope, unlike many other international osteoporosis treatment guidelines, the Dutch guideline advises anti-osteoporosis treatment in persons with osteoporosis and/or (prevalent) grade 3 (> 40% reduction in vertebral height) or grade 2 (25–40% reduction in vertebral height) vertebral fractures [23]. Finally, it is worth mentioning that in the Netherlands, FLS are run by qualified osteoporosis nurses that followed a 2-year specific training which ensures they are knowledgeable on fractures and osteoporosis and have the appropriate communication skills.

Design

The previously developed DA on AOM in post-menopausal women with osteoporosis served as a basis [16], although for FLS three aspects needed to be further clarified or adjusted: (a) the risk of subsequent fractures following a recent fracture, with or without initiation of AOM, (b) potential first and alternative medication options including potential barriers, such as treatment burden and risk of side effects, and (c) a section on formal deliberation on options and final decisions [15]. Several sources were identified to address these aspects, including (i) a systematic literature review (SLR) on the rate of subsequent fractures in patients with a recent fracture who did or did not visit FLS [24], (ii) the national Dutch guideline on osteoporosis and fracture prevention to identify medication options for the target population [21, 25], and (iii) a grey literature search to find DAs that provided examples of ways in which medication side effects can be presented and how personal preferences and values can be included in the final decision. The SLR was done to investigate the recurrent fracture rate in osteoporosis patients with recent fractures attending FLS and we used the fracture rate in the control group of studies with good quality as recurrence rate when no AOM would be started [24].

Furthermore, two focus groups were created with patients to understand their preferences about (i) communication of the potential harms of AOM and (ii) content of the section on final deliberation to start a specific treatment. Examples from DAs retrieved from the grey literature search were used to stimulate discussions. Information on the attributes/characteristics of the different AOMs to be included in the option grid had been collected in the context of the DA on post-menopausal osteoporosis [15]. Patients at risk of fractures were eligible to participate after attending a follow-up visit at the FLS of Maastricht University Medical Center (MUMC +), which serves as a regional and academic hospital. Efforts were made to include both males and females, and persons with diverse educational backgrounds. Oral informed consent was acquired. Due to time constraints only field notes were taken during the focus groups, which served as the basis to make adjustments in next steering group meetings. Results from focus group interviews were analyzed according to field notes. Before inclusion in the DA, information retrieved from the various sources was discussed repeatedly with HCPs who are involved in osteoporosis research and management.

Prototype

An initial prototype was developed by integrating the additional/updated information in the available DA. Prior to alpha testing, the prototype was piloted among three female medical support staff members.

Alpha testing

Alpha testing comprised readability, comprehensibility, and usability, and was assessed among (a) patients from the MUMC + and (b) members of the (lay) research panel of the Dutch Osteoporosis Association (DOA). Consecutive osteoporosis patients who attended the FLS for a follow-up visit after AOM initiation and agreed to participate in the individual interviews were included. The DOA chair invited the members of their panel of twenty experts by experience through our invitation and information letter. The first five persons that accepted the invitation received the DA and open-ended digital questionnaire for feedback. A digital questionnaire was chosen upon request by the Osteoporosis Society’s director. The intention was to include at least ten patients and at least five DOA members. This step was undertaken after adjusting the prototype based on feedback from the focus group patient interviews.

Interviews with patients from the FLS applied a cognitive debriefing approach and were executed by a research nurse and junior researcher (NR), who had been trained in interview methods. First, patients were asked to read each page of the DA prototype out loud and repeat the content of the page in his/her own words. Next, the interviewer asked about readability (difficult words or sentences) and comprehensibility of text and figures. At the end of the page-by-page debriefing, patients were asked whether the DA had improved their understanding of future fracture risk and the effect on this risk of both lifestyle changes and AOM. The patients were asked their opinion about the usability of several additional generic information folders. Following the open-ended questions, patients received a questionnaire and were asked to score five questions on amount of information, information clarity, design, patients’ comfort with decision responsibility, and DA recommendation on a 7-point Likert scale (1 being the lowest score and 7 being the highest). Interviews were audio-recorded and transcribed verbatim. Transcripts and field notes were summarized thematically per page and for the entire DA. After every three interviews, feedback from patients was discussed with at least two steering group members who decided whether and how to make changes. For major adjustments, agreement of the full steering group was sought.

The DOA members commented on the penultimate version (version 3) of the prototype. The DOA members were asked to score the same 7-point Likert scale questions and were invited in five open-ended questions to comment on overall readability, comprehensibility, and usability of the DA (Supplementary Table 1). Written comments were linked to the themes that had been identified in the patient interviews and new themes were added when applicable. In a final step, the steering group considered the remaining feedback and agreed on the final prototype.

Setting and ethical approval

This study was conducted in the Department of Internal Medicine, Division of Rheumatology at the MUMC + . The study was approved by the institutional review board from the MUMC + (2028–0507-A-10). All participants provided informed consent.

Results

Design

SLR on recurrent fracture rate

As part of the SLR, two retrospective studies described the risk of failing to treat recurrent fractures among patients older than 50 years, who suffered a recent fracture in the Netherlands [26, 27]. In line with the Dutch guideline on osteoporosis and fracture prevention, three DAs were developed depending on risk of recurrent fracture and the preferred first line AOM. The first DA applies to patients with a recent fracture that is not a ‘major’ fracture (i.e. not a hip, pelvis, or vertebral facture and not multiple fractures), indicating an intermediate risk for subsequent fractures (14% in the next 5 years) and for whom antiresorptives are recommended as first choice. The second DA is aimed to be used in patients with a major fracture (except for women with new or prevalent vertebral fractures), indicating a sub-group with a high subsequent fracture risk (25% in the next 5 years) and recommended to start antiresorptives as first choice treatment. The third DA was intended for women at high risk because of a major fracture including a new or prevalent vertebral fracture, for whom an anabolic agent is recommended as first choice of anti-osteoporosis treatment [21]. The treatment effectiveness displayed in all three DAs was 40% [8, 26].

Focus groups

Two focus groups revealed that patients preferred visualization of fracture risk with and without AOM by icon arrays, using broken bones over sad faces. Patients further agreed on the value of an option grid to provide an overview of AOM and information on benefits (i.e. effectiveness for the different fractures sites, medication administration, frequency, duration, and requirement of an HCP to administer the medication) and recommended to add information on potential harms (i.e. side effects and required precautions before and during treatment) to the option grid. For side effects, patients agreed they wanted to be informed on ‘common but less severe’ reactions (e.g. gastro-intestinal upset) as well as ‘rare but potentially severe’ side effects. For the final deliberation, patients demonstrated a preference for a simple interactive approach together with a specialized nurse guided by three questions: was the information provided clear, was additional information needed, and what was the final choice (whether to start with treatment or not, and if so, with which AOM). They liked information on the next steps (e.g. drug initiation, control visit) and on contact details for questions if anything were unclear before the new clinical visit/consultation.

Steering group

During the meetings of the steering group, it was emphasized the DA needed to be user-friendly, refrain from difficult medical terminology, and omit duplicate information. Suggestions were also given to balance the completeness of options whilst maintaining comprehensibility of their characteristics without too much detail (e.g. effectiveness, mode, and duration of administration).

Prototype

Each DA consisted of eight pages (Table 1). As an example, the third DA version can be found in Appendix 1. Wherever possible, text was accompanied by illustrative icons, e.g. the effect of medication on the risk of a subsequent fracture.

Table 1 Page and content of the prototype of the  decision aid. DA, decision aid; Ca++, calcium
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Alpha testing

The ten patients with an osteoporosis treatment indication who were recruited and who attended the FLS and five members of the DOA represented the population of interest (Supplementary Table 2). Of note, all participants had osteoporosis but one of the DOA members and one patient recruited from the FLS had not sustained a fracture. The FLS patient had a severely bruised finger, but revision of the X-ray could not confirm a fracture.

Figure 2 illustrates the answers to five usability questions asked of the patient group and DOA members per DA version. Overall, patients provided better scores compared to DOA members: patients were more satisfied, especially regarding the amount and clarity of the information (Fig. 2). Patients and DOA members recommended the use of this DA in clinical practice and scored this question the highest (7 versus 6.2). Of note, scores from the three last persons (in view of consecutive adjustment) demonstrated the highest scores, specifically increasing the most as more adjustments were made in response to the questions regarding design, patients’ comfort with decision responsibility, and DA recommendation. Interviewees often requested more detailed information, e.g. inclusion of physiotherapists in the region, inclusion of more rare health conditions as a risk factor for osteoporosis, specifications on lifestyle in relation to bone health.

Fig. 2
figure 2

Overview of results from five questions using a 7-point Likert scale, divided into a patient group (n = 10) and DOA members (n = 5). Results are illustrated with the mean per DA adaptation. Answers ranged on a scale from 1 to 7, with 1 being the lowest score and 7 being the highest. DA, decision aid; DOA, Dutch Osteoporosis Association

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Table 2 summarizes the results of the open-ended questions for the patients and DOA member group per theme, per DA adjustment version. Overall, the content was clear to the patients and some of the DOA members (Supplementary Table 1). The option grid with AOM information was especially appreciated and well-understood. Except for the interpretation of the figures presenting the risk of recurrent fractures with and without medication, the overall layout and design was experienced as clear, easy to read, and eye-catching. All ten FLS patients and five DOA members experienced the DA as useful for outpatient visits and would recommend that it be used routinely. Main points of improvement highlighted by the patient group included clarification of difficult terms such as ‘calcium intake’ and ‘corticosteroids’, and consideration of visual impairments (e.g. colour blindness) with regard to the figures. For all these suggestions, adjustments were made (e.g. colours in the icon array were changed from red and green into red and blue). Most patients and two DOA members felt more detailed information was needed to understand illustrations such as the illustration on page one regarding the cross-section of the bone, or types of medication on page six with their side effects. This confirms that the DA should not be provided outside the clinical visits.

Table 2 Overview results from open-ended questions of the 10 osteoporosis patients (interview) and 5 DOA members (survey). Numbers of patients are presented in frequencies per DA adjustment version. + , strong point; -, point of improvement; HCP, healthcare professional; DOA, Dutch Osteoporosis Association; AOM, anti-osteoporosis medication; DA, decision aid; FLS, fracture liaison services; MUMC+, Maastricht University Medical Center
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Other points of improvements for DOA members included the lack of information on the role of regional HCPs such as physiotherapists, inclusion of more extensive risk factors for fractures and osteoporosis (rare diseases), and additional information about medicines. With regard to extra information required on the role of specific diseases or lifestyle advice, the steering committee made a decision to limit the information, as most patients were already overwhelmed by the amount of information and HCPs can provide more details when needed. Contrary to persons participating in the focus groups, half of those participating in the alpha testing considered the page on deliberation as ‘scary’. This was likely a result of the context of the testing, where no real-life interaction could be simulated.

Discussion

This study reports on the development and usability of the DA by HCPs in the context of FLS for patients who have an indication for AOM and are over the age of 50. The strengths of this DA are its alignment with the new Dutch guideline on osteoporosis and fracture prevention, the involvement of relevant users (medical specialists, specialized nurses, the DOA members, and persons with a risk of fractures) and experts in shared decision-making, adherence to the IPDAS, and active involvement of patients throughout the process.

A challenge during the development related mainly to the data required to estimate the baseline risk of a subsequent fracture by recent fracture sufferers. Based on the Dutch guideline recommendations, three decision aids were created on the basis of future fracture risk and evidence of the effectiveness of treatment options (Appendix 2 illustrates the flowchart on selecting the most suitable DA for each patient) [21, 26]. Additional consultation with the specialized nurses indicated this would not be problematic in real life, as this distinction is representative of real practice. Along the same lines, communication of the rates of side effects was intensely discussed. The literature suggests a ‘best approach’ to communicate risk of side effects, chiefly because of the large differences in numeric literacy among patients [21, 26]. For this reason, and supported by patient preferences, the DA distinguishes between side effects that are ‘common but less severe’ and those that are ‘rare but potentially severe’.

All participants in the alpha testing recommended the usability of the DA in the FLS. They indicated that the preferences and needs of patients were adequately represented and emphasized the inclusion of lifestyle change as a strong point of the DA. During the initial interviews, a substantial number of textual enhancements were identified by patients and, due to subsequent adjustment, it was possible to evaluate these improvements during later interviews. Overall, DOA members were more demanding when answering questions about content, which is not surprising as they are trained to take a critical look at usual care. More often than patients, they suggested adding information on the potential diseases and causes underlying osteoporosis, and the need to change lifestyle behaviour by referral to HCPs such as physiotherapists to support behavioural change. However, the steering committee decided not to include such information, mainly to avoid diverting attention to aspects that are not within the scope of the DA. Importantly, our DA is professionally administered so that the specialized nurse is available to answer questions that might be appropriate or raised during consultation.

A systematic review and environmental scan performed by Paskins et al. analyzed the quality and effectiveness of osteoporosis treatment DAs [28]. At that time, there were only nine DAs available, all lacking several points of criteria later raised in the IPDAS development procedure. Despite this limitation, Paskins reported beneficial effects on decisional conflict, SDM, and accuracy of perceived fracture risk [28]. Our current DA is being tested at present in a trial including two hundred fifty patients from two Dutch FLS clinics and with adherence defined as an outcome [16]. As SDM alone is likely to be insufficient to ensure adherence, the trial will include motivational interviewing in the follow-up after 4 to 6 months, turning our intervention into a multi-component adherence intervention.

This study also has its limitations. The opinion of only one specialized osteoporosis nurse was included during the development of the DA. As we did not want to influence the opinion of the nurses before starting the intervention component of the ongoing trial, the opinion of the four osteoporosis nurses actively involved in the trial were not included in this development phase and their opinion will be identified during the process evaluation of the ongoing trial. Other limitations might be the lack of patient involvement as early as the steering group formation (IPDAS guidelines [17, 18]) but patients were actively involved in all other stages and had the opportunity to discuss all aspects of the DA. Furthermore, some patients are accustomed to the paternalistic model of the physician-patient relationship. This was also confirmed by previous studies [29, 30]. It should be noted that SDM is about collaborating with the patient to solve a problem; therefore, SDM should be considered with gradients that are congruent with patient values and preferences.

In addition, for the alpha testing, there was a difference in data collection methods between the two groups (patients were interviewed and received a survey, whilst DOA members only received a digital survey, albeit one more elaborate than the patients’ survey). These differences in collection methods probably contributed to discrepancies. As the DA was developed to be used as a HCP-administered tool, the more favorable feedback from those that had the face-to-face interview supports the importance of an administrator. To fully comprehend the feasibility and effects of the DA, the ongoing trial will report on the effectiveness and cost-effectiveness of the DA together with motivational interviewing [16]. Also, research should address difference in attitudes and preferences of persons towards SDM in order to enable decisions in line with (explicit/implicit) needs and preferences of these persons. Further limitations in the alpha testing were the relatively low sample size, partly related to COVID-pandemic, limiting generalizability, the lack of variation in ethnicity data, and use of educational level as surrogate for health literacy.

Before applying the DA in the FLS, nurses should be aware of the value of SDM and be trained in applying the DA during the clinical encounter. For the purpose of our ongoing trial, nurses will also apply the DA during a pilot study to identify and solve additional barriers and facilitators. This will include estimation of additional time for SDM. IT is well known that successful implementation of a DA will not only depend on the quality of the tool, but also on the support and self-efficacy of HCPs [31].

Conclusion

We established a DA specifically for Dutch osteoporosis patients who have an indication for AOM and are above the age of 50 through integration of patient needs and preferences, clinical expertise, user preferences, and iterative development. The tool’s utility during outpatient visits was recommended by patients, osteoporosis association members, and HCPs.