Current situation of shared decision making in osteoporosis: A comprehensive literature review of patient decision aids and decision drivers

Abstract Background and Aims Osteoporosis is a systemic skeletal disease characterized by low bone mass and microstructural deterioration of bone tissues, resulting in bone fragility and increased fracture risk. It is the most common bone‐related disease in the population. However, the proportion of patients who start treatment but discontinue it during the first year is very high (around 50%). Endeavors are made to promote patient participation in treatment by implementing patient decision aids (PDA), whose function is to help the patient make disease‐related decisions. We aim to summarize the characteristics of the currently available PDA for osteoporosis, as well as deciding factors. Methods Comprehensive review of the literature. Results Currently, eleven PDAs can be found for osteoporosis. These PDA have different characteristics or options such as information about treatments tailored to patient needs, graphic information of the results (to facilitate understanding), personal histories (learning), tests to check the knowledge acquired, provision of evidence, clinical practice guidelines or a final summary to share with their doctor. Only five of these PDAs can be considered complete since they provide relevant disease information and therapeutic options to the patient, promote patient's reflection and foment patient‐physician discussion. Conclusions This study provides an update on the current state of decision making on osteoporosis and available PDA, which can help engage the patient through shared decision‐making by considering, among other things, patient preferences. Physicians should consider PDA, as it may promote adherence and effectiveness of treatment.


| BACKGROUND AND AIMS
Osteoporosis is a systemic skeletal disorder characterized by low bone mass and microarchitectural deterioration of bone tissue that leads to bone fragility and increased fracture risk. 1,2 It is the most common bone disease affecting predominantly women. 1,3 About 27 million people are affected by the disease in the European Union (EU) 4 and, moreover, its prevalence is expected to rise in the coming years. 5 Since osteoporosis is a silent disease without any pathognomonic clinical signs, 6 it usually remains undiagnosed until a low-trauma fracture occurs. 1 Osteoporotic fracture, commonly involving the hip and spine, may affect an individual's ability to function independently, leading to chronic pain and prolonged rehabilitation. 7 It is widely recognized that osteoporosis-related fractures are associated with increased mortality, with the exception of forearm fractures. 8 Thus, between 20% and 40% of individuals suffering hip fractures die within a year. 9 The economic burden of osteoporosis is also high. In the EU it is estimated that the cost of osteoporotic fractures in 2010, including pharmacological and long-term disability, reached 37,000 million euros. 10,11

| Strategies to prevent osteoporosis fractures
To prevent fractures in osteoporotic patients, both nonpharmacological and pharmacological interventions are recommended. Nonpharmacological strategies include a healthy lifestyle, such as a balanced diet, regular physical exercise, no smoking, limited alcohol consumption, and implementation of fall prevention measures. 1,12 Pharmacological treatments, such as antiresorptive and anabolic drugs, target patients with high or very high fracture risk.
Both therapies have been shown to increase bone strength. 2,13 However, their mechanisms of action differ, as antiresorptive therapies inhibit bone resorption by suppressing osteoclasticmediated bone breakdown and bone turnover, whereas anabolic agents restore bone mineral content. 2,14,15 Main pharmacological agents currently available for osteoporosis are: bisphosphonates, anti-RANKL antibodies (denosumab), selective estrogen receptor modulators, estrogen replacement, monoclonal antisclerostin antibodies (romosozumab), strontium ranelate and parathyroid hormone analogs. 16,17 The latest updates in clinical practice guidelines recommend teriparatide, abaloparatide, or romosozumab for patients with a very high risk of osteoporosis fracture. 17,18 Despite the wide range of pharmacological options available, there is a large gap between the number of women receiving treatment and those that could be considered eligible for treatment based on their fracture risk. 19 Several studies reveal that less than 20% of patients suffering from a fragility fracture receive therapy to reduce future fractures within the following year. 19 Moreover, many women at high risk of fractures choose not to initiate therapy, and, of those who do, up to 50% discontinue treatment in less than 1 year. 20 These low rates of treatment compliance and persistence fail to reduce the risk of osteoporotic fractures, which in turn can increase healthcare costs and greatly decrease patients' quality of life 21 and life expectancy. 8,22 Previous studies have shown that patient preferences play a key role in accepting or rejecting osteoporosis treatment. 23 Accordingly, in addition to choosing the treatment based on the patient's characteristics and their risk of fractures, 7 it is fundamental to align treatment choice with patients' preferences and engage them in treatment decisions.

| Patient-centered care and patient decision aids
In broad terms, there are three models of doctor-patient interaction regarding clinical decisions: (1) paternalism, where the physician has all the relevant information and is the sole decision maker; (2) informed model, where the physician presents "the facts" and the patient makes all decisions; and (3) shared decision-making (SDM), where the physician and patient share information, discuss options using the best evidence and reach a collaborative decision that takes into account the patient's context, values, and preferences. [24][25][26][27] The SDM is a key component of patient-centered care. In this model, the physician offers a recommendation and shares information about the benefits, drawbacks, and burdens of the therapeutic options available. At the same time, patients are encouraged to become involved in the decision and to express their feelings and treatment expectations. 25,26,28 This process encompasses five steps: (1) understanding the patient's experience, preferences, and expectations; (2) building partnerships; (3) providing treatment evidence, including uncertainties; (4) giving recommendations, and (5) checking for understanding and agreement. 25 SDM does not advise the patient to choose one option over another, it provides structured guidance in the decision-making steps and helps patients to make informed value-based decisions together with their physician. 29,30 The final choice depends on how a patient evaluates the drawbacks and benefits of the different treatment options. 31 In 1989 a paper was published describing clinical strategies when a patient has decisional needs. 32 Subsequently, in 1995 the Patient Decision Aids Research Group developed the Decisional Conflict Scale, the first scale to measure changes in decisional requirements following counseling. 33 In the following years, different guidelines for developing PDAs were published. Also, in 2003 the International Patient Decision Aid Standards (IPDAS) was founded. It aimed to improve the quality and effectiveness of patient decision aids by establishing a shared evidence-based framework with a set of criteria to improve their content, development, implementation, and evaluation. In this sense, IPDAS established a set of quality criteria for PDA that works as a checklist for developers and users 29,30 On the other hand, the European League Against Rheumatism and the European Federation of National Associations of Orthopaedics and Traumatology recommend the inclusion of patient education programs to prevent fractures. 20 In this respect, PDAs play a key role in providing evidence-based information about conditions, treatment options, outcomes, probabilities, and an opportunity for patients to ponder their preferences. 20 PDA facilitate SDM by improving the patient's knowledge of the disease and encouraging reflection, generating more realistic expectations about options, reducing decisional conflict, and helping patients to clarify their preferences. 26,27,34 PDA also help patient to share information with the physician, allowing a consensus to be reached on the best treatment option. 24 Although further research is required to determine the effect of adherence, involving patients in decision-making could lead to improved disease management. 35,36 This becomes particularly relevant when there is more than one therapeutic option, each with its benefits and drawbacks, as is the case for osteoporosis. Previous studies have shown that the use of PDA contributes to reducing decisional conflict (i.e., uncertainty about the choice, ignorance about the pros and cons of each option, pressure to make a particular choice, and effectiveness of the decision). [37][38][39][40][41] In addition, the use of a PDA can also contribute to reducing the variation of the clinical practice in preference-sensitive options, and improve care more broadly. 27,28,30,34 This study aims to summarize the characteristics of the currently available PDA for osteoporosis and decision factors.

| METHODS
Two literature reviews were conducted: (1) a review of studies on physician and patient preferences for osteoporosis treatments; and (2) an ordered review of the literature on PDA for patients with Osteoporosis.

| Data sources and search strategies
The international databases PubMed/Medline and Cochrane library, and the national Medicina en Español (MEDES) and the Índice Bibliográfico Español en Ciencias de la Salud (IBECS) databases, were searched to identify relevant publications on treatment preferences.
PubMed/Medline and manual sources (e.g., google, google scholar, Ottawa Hospital) were searched to identify available PDAs for Osteoporosis.

Databases were searched using both MeSH (Medical Subject
Headings) and free-text terms, combined with the Boolean connectors "OR" and "AND." The treatment preferences search was conducted in English (international database) and Spanish (national database).

| Eligibility criteria
Clinical trials, observational studies, and narrative or systematic

| PDA
Eleven PDAs are currently available worldwide for osteoporosis.
IPDAS establish a set of quality criteria for PDA that works as a checklist for developers and users The IPDAS criteria provided by each PDA are detailed in Table 1. On the other hand, considering that a complete PDA explains the decision; provides information on options, benefits, and harms; and helps patients clarify which benefits and harms matter most, only five of the identified tools can be considered as complete PDA, since they are the only ones that provide information, explore patients' preferences and facilitate physician-patient discussion (Table 1). [41][42][43][44][45] Concerning the treatment options, some of the PDA support patients in the decision to choose a specific drug. [36][37][38][39][41][42][43][44][45][46][47] Most tools are focused on bisphosphonate treatment while only three display most of the available therapies. In relation to the format, four of the PDA are on paper, 37,43-45 four on online platforms 36,39,41,47,48 and three are in both formats. 38 Complete Decision-making support tool, which must provide relevant disease information and therapeutic options to the patient, promote patient's reflection and foment patient-physician discussion.
During its development, a PDA should be validated by conducting an alpha and/or beta test. An alpha test is an acceptability test that aims to identify all possible issues before launching a product to users, while a beta test is a user utility test in which a sample of the intended audience tries the product out. 49,50 Only some of the identified PDA have undergone alpha [37][38][39]41,48 and/or beta tests 37,39,41,48 (Table 2).  (Table 3).
Since patient profile in osteoporosis is variable, adaptability is a The option of performing a patient knowledge test helps evaluate whether the patient has understood the information provided in the PDA related to the disease and its treatment. Five PDA includes a test that evaluates the knowledge acquired by patients. [42][43][44][45]48 The level of evidence provided for the information presented in the PDA demonstrates the validity and integrity of that information.
Four PDAs include an acceptable level of evidence for the information provided. [43][44][45]47 Clinical practice guidelines provide evidence-based recommendations founded on rigorous systematic reviews and synthesis of published research in academic, governmental, and private sectors. 27 Knowing It is very important for osteoporosis patients to adopt healthy lifestyles, including a varied and balanced diet, which guarantees the supply of essential nutrients for bone health and the amelioration of osteoporosis. 54 One of the best ways to build and maintain healthy bones is through exercise. Exercise improves disequilibrium and reduces the risk of falls. 55 None of the PDA available includes adaptations to the patient's exercise program, which should address flexibility, strength, core stability, cardiovascular fitness, and equilibrium.
Patients expressed a positive attitude towards the use of these PDAs as they improved their preparation for decision-making and decreased decisional conflict. These PDAs improved knowledge transfer and patient involvement in decision-making with adequate patient and physician satisfaction, but with a weak or null effect on medication adherence. 36,39 Interestingly the effectiveness of four PDAs has been evaluated. In three studies, 36 59 In addition to these issues, to reach an informed decision, patients demand further information about osteoporosis disease, drug-specific detail (e.g., whether it is solid or liquid, or whether it should be stored refrigerated), and information related to healthy lifestyles (exercise and/or nutrition). 67

| CONCLUSIONS
This study provides an update on the current status of shared decision making in Osteoporosis treatment and the PDA currently available. The results highlight that patient preferences should be considered by physicians since they can impact adherence to the treatment and its efficacy. Currently, available PDAs can help to engage patients through shared decision-making. Since the purpose of a PDA is to help patients in the decision-making process there is certain information that must necessarily be included in the PDA. The information gathered in this review regarding the decision drivers may help to define which content should be included in a PDA.

ACKNOWLEDGMENTS
The study has been supported by Amgen Spain.

DATA AVAILABILITY STATEMENT
Data sharing is not applicable to this article as no datasets were generated or analyzed during the current study.

TRANSPARENCY STATEMENT
Luis Lizán affirms that this manuscript is an honest, accurate, and transparent account of the study being reported; that no important aspects of the study have been omitted; and that any discrepancies from the study as planned (and, if relevant, registered) have been explained.