Reducing endocrine metabolic disease risk in adults with chronic spinal cord injury: strategic activities conducted by the Ontario-Quebec RIISC team

Abstract

Purpose

The Rehabilitation Interventions for Individuals with a Spinal Cord Injury in the Community (RIISC) team aimed to develop and evaluate innovative rehabilitation interventions to identify endocrine metabolic disease (EMD) risk, intending to reduce the frequency and severity of EMD related morbidity and mortality among adults living with chronic spinal cord injury or disease (SCI/D).

Materials and Methods

An interprovincial team from Ontario and Quebec reviewed available EMD literature and evidence syntheses and completed an inventory of health services, policies and practices in SCI/D care. The review outcomes were combined with expert opinion to create an EMD risk model to inform health service transformation.

Results

EMD risk and mortality are highly prevalent among adults with chronic SCI/D. In stark contrast, few rehabilitation interventions target EMD outcomes. The modelled solution proposes: 1) abandoning single-disease paradigms and examining a holistic perspective of the individual’s EMD risk, and 2) developing and disseminating practice-based research approaches in outpatient community settings.

Conclusions

RIISC model adoption could accelerate EMD care optimization, and ultimately inform the design of large-scale longitudinal pragmatic trials likely to improve health outcomes. Linking the RIISC team activities to economic evaluations and policy deliverables will strengthen the relevance and impact among policymakers, health care providers and patients.

IMPLICATIONS FOR REHABILITATION

• Living with a spinal cord injury or disease (SCI/D) increases endocrine metabolic disease (EMD) risk.

• EMD-related outcomes include fracture; type II diabetes; and cardiovascular disease (myocardial infarction, sudden cardiac death and stroke), directly contributing to higher morbidity and mortality.

• Single-disease paradigms are not the ideal strategy to address multimorbidity contexts experienced in SCI/D.

• Practice-based research could be an alternative/adjunct to randomized control trials at generating evidence on current and emerging rehabilitation approaches.

Keywords:

• Spinal cord injury
• endocrine metabolic disease
• cardiometabolic disease
• osteosarcopenia
• prevention
• rehabilitation

Introduction

Living with a spinal cord injury or disease (SCI/D) poses an increased health risk and propensity for developing endocrine metabolic disease (EMD) specifically, fracture, type II diabetes, and cardiovascular disease [1]. EMDs are associated with increased morbidity and mortality [1–3], yielding a spectrum of disease and co-morbid conditions that affect the quality of life and longevity of adults living with SCI/D, and their families and caregivers, in addition to the economic burden on an overwrought public health system [4].

An elevated EMD risk is defined as the presence of three or more EMD risk factors such as obesity, insulin resistance, and hypertension [5–8]. To understand the concept of EMD risk, however, one needs to consider how early, dramatic changes in metabolic function and body composition manifest as downstream or late health complications when combined with systemic inflammation and inactivity [9]. Such changes include reductions in bone and muscle mass, and increases in fat mass and inflammatory stress (a high serum c-reactive protein) [10,11], which impose a significant EMD risk and related disease burden that can last several years after the onset of SCI/D (See Figure 1). In fact, because chronic inflammation amplifies the risk of cardiometabolic disease, countermeasures should be considered from the point of initial injury, and throughout the lifespan of people living with SCI/D[10]. Thus, Understanding the complex interactions between changes in body composition, biomarkers, and inflammatory stress is an important initial construct [12]. These processes are further accelerated by impairment-related physical inactivity [13,14]; poor diet [15]; the individuals’ personal and environmental contexts [3]; and their degree of community participation [16]. Altogether, the integration of these factors over many years leads to the often undetected elevation in EMD risk, which manifests in diverse EMD expression [e.g., fracture, diabetes, dyslipidemia, cardiovascular disease or combination(s)] and related adverse personal health, well-being [17,18], and participation outcomes [16]. Moreover, it has been suggested that SCI/D related comorbidities with more explicit symptoms (e.g., neurogenic bladder) often draw attention away from the risks related to the so-called “silent” metabolic dysfunctions [6]. Thus, continuous EMD surveillance is encouraged even when explicit symptoms are not present.

Figure 1. A model for understanding EMD risk and the adverse outcomes of EMD expression over the course of an individual’s lifetime.

A four-panel model linked through double-headed arrows to show interconnected domains relevant to understanding EMD risk. Each panel holds a domain, presented from left to right as list of Impairments, Activity Limitations, Disease Expression and Outcomes.

Despite the multifactorial nature of EMD risk, many SCI/D-centered health services are clustered around specific health complications or a single disease states, such as a diabetes or heart disease clinic and do not consider the complex needs of adults living with SCI/D who report a median of 7 concurrent secondary health conditions at 10 years post injury [19]. To date, single-disease paradigms have failed to adequately serve adults with SCI/D and multimorbidity. For example, heart disease clinics are not prepared to do arm ergometry stress tests for those with SCI/D, or diabetes clinics do not have accessible scales, or general internists have not considered the implications of routine antihypertensive therapy for elevated blood pressure on autonomic dysfunction or orthostatic hypotension. Consequently, adults living with SCI/D who are prone to multiple health conditions with competing symptoms are often provided treatment plans which can be challenging to implement, whilst not integrated to their overall care plan. Multimorbidity and high health care utilization are interrelated and associated with lower health status, which in turn is associated with lower quality of life [20].

The co-occurrence of EMD risk (elevated fracture risk and elevated risk of developing diabetes or heart disease) are strong drivers for healthcare utilization that often go undetected prior to disease onset and influence health services and systems on a global scale [2]. Thus, strategies to identify an elevated EMD risk among adults with SCI/D, and to develop multi-modal interventions to reduce the incidence and severity of EMD, are warranted and presented herein as a focus of this white paper.

A prior decade old call to action and plea for health system transformation, “The SCI Manifesto” (https://www.cravenlab.ca/_files/ugd/078cd4_b6d4221b1eb04322ab144ff6b88abaf4.pdf), was made to Canadian health care professionals and endorsed by many SCI/D consumer and stakeholder agencies (including the Ontario Neurotrauma Foundation (ONF), the Quebec Rehabilitation Research Network (REPAR), Spinal Cord Injury Ontario (SCIO), and the Ontario Spinal Cord Injury Solutions Alliance, to address the health consequences of fracture, cardiovascular disease, and pressure sores among adults living with SCI/D. This plea for change was issued following an October 2013 consensus meeting of 23 international experts in SCI/D rehabilitation, who reviewed extensive data on health care, service and policy practices, and research activities. The experts advised that reducing morbidity and mortality related to the aforementioned health conditions was necessary to transform the health of Canadians living with chronic SCI/D in the community. Prior and current research conducted by the ONF and REPAR funded SCI/D rehabilitation research team(s) are relevant to this plea for health system transformation. This manuscript is a direct response to the manifesto for change from the RIISC inter-provincial SCI/D collaborative initiative which aims to reduce the frequency and severity of EMD risk in adults living with SCI/D, and the associated incidence and prevalence of fracture, diabetes and heart disease, by promoting the implementation of interprofessional rehabilitation interventions and multiple disease paradigms for evaluating health service delivery among adults living with SCI/D in the community.

Incidence/prevalence of EMD risk

Identifying the impact of Declines in bone quality

Traumatic SCI leads to rapid onset of osteoporosis that threatens skeletal integrity, particularly below the level of injury [21]. Declines in bone mass and bone quality of the hip and knee regions after SCI are rapid (3%/month) in the acute stages, establishing a lower steady-state bone mass 1–2 years after injury [22]. Losses in both trabecular and cortical bones at the tibia, the most common site of open fractures [23], persist for more than three years after SCI, although there is significant inter-individual variability in the pattern of bone loss [24]. The lifetime incidence of lower extremity (LE) fragility fracture among adults with chronic SCI ranges between a quarter to half of all people living with SCI/D [23,25]. While this incidence is much higher than the 10-year fracture projections suggested for the general population [26,27], agreement on the best strategies to prevent and manage LE fractures in SCI has been lacking [21,23]. In fact, it was not until recently that consensus recommendations regarding fracture diagnosis and management were developed to guide general clinical practice, the involvement of rehabilitation professionals and of specialized physicians [23]. For instance, only in 2019 the International Society for Clinical Densitometry (ISCD) released a position statement on Bone Mineral Density Testing after SCI. That document indicated that physiatrists and therapists should review fracture risk with patients before prescribing them rehabilitation therapies. It was further stated, however, that risk assessment prior to exercise or rehabilitation interventions should be assigned to rehabilitation professionals [21].

SCI-specific risk factors for LE fragility fracture include: history of fragility fracture [28], family history of fragility fracture [26], body mass index ≤19 [29], duration of injury ≥10 years [29], female sex [29,30], age at injury ≥ 16 years [31], motor complete injury (i.e., AIS A or B) [32], paraplegia [33], alcohol use ≥ 5 servings per day [34], and use of opioid analgesia [35], or benzodiazepines [36], spasticity medications, or unfractionated heparin [35]. Of note, the latter five risk factors are modifiable to intervention. Another significant risk factor that can be used to quantify fracture risk is low bone mineral density (BMD) of the hip and knee regions. For example, every one standard deviation (i.e., 0.1 g/cm²) decrease in femoral neck BMD below the young adult mean is associated with a 2.2 times increased risk of fracture [37]. In addition, for every one standard deviation (i.e., 0.1 mg/cm²) increase in distal femur BMD there is a 1.2% decrease in fragility fracture risk (after adjusting for completeness of injury and bisphosphonate use OR = 0.988, 95% CI: 0.978 to 0.998, p = 0.0226) [38]. The clinical links between absolute BMD and fracture risk suggest that rehabilitation therapies capable of increasing hip and knee region bone mass have the potential to reduce fracture incidence.

Data from the Canadian Community Health Survey (CCHS) of individuals with SCI/D living in the community, found that one in ten Canadians with SCI/D reported a fracture in the last calendar year [39]. A single fracture significantly increases morbidity due to delayed healing or non-union, and fracture-related complications (venous-thromboembolism, cellulitis, pressure sore) [21]. In addition, increased attendant care requirements, time off work and reconditioning following fracture healing are anticipated in association with adults living with chronic SCI/D hospitalized for fracture staying seven times longer than those without fracture [34]. Further, and most concerning, there is an increased five–year risk of mortality following a lower-extremity fragility fracture reported in men with motor complete injury [40,41].

Identifying the impact of sarcopenic obesity

Following SCI/D, additional unique changes in body composition are observed. Declines in muscle mass occur with an associated 50% decrease in muscle cross-sectional area, preferential atrophy of type I muscle fibers, predominance of type IIx fibers, and increased fatty infiltration of muscle [42]. Additionally, there are increases in visceral and abdominal adipose tissue deposition. Increased visceral adipose tissue (VAT) is a known risk factor for insulin resistance, glucose intolerance, and cardiovascular disease [43–45]. There are several challenges associated with the measurement of VAT after SCI/D. The standard cut-off values for body mass index (BMI) and waist circumference used in the non-injured population underestimate obesity in the SCI/D population [46]. Optimal cut-off values for whole-body adiposity in the SCI/D population for waist circumference and BMI are 94 cm and 22 kg/m², respectively [46–48]. Further, waist circumference measurement does not distinguish visceral adiposity and subcutaneous adiposity. Consequently, BMI, frequently used as a surrogate measure of adiposity, drastically underestimates fat mass and does not consider regional adipose tissue distribution [49]. While the amount of VAT is correlated with waist circumference in non-injured individuals, an increase in VAT in adults with spinal cord injury is often not correlated with an increase in waist circumference [50]. Conversely, in a recent cross-sectional study using population-specific waist circumference measures, 74% of adults (n = 136) with chronic SCI/D had elevated VAT and met diagnostic criteria for obesity [42]. As an abnormal amount of VAT is linked with all-cause mortality, urgent attention targeting VAT reduction is warranted [51,52]. A challenge to which potential solutions can ideally be offered through the implementation of multimodal rehabilitation interventions. For instance, it has been proposed that combining a Mediterranean diet and high intensity interval training exercise is a promising strategy against neurogenic obesity [53] – the paralysis-induced adipose tissue accumulation that results in chronic, systemic inflammation and metabolic dysfunction – a hallmark of chronic SCI/D [54].

Identifying the impact of cardiometabolic risk

Cardiometabolic disease (CMD) occurs with increased frequency among adults living with SCI/D and represents the leading cause of mortality in that group [55]. CMD risk factors include abdominal obesity [47,56–58], low high-density lipoprotein (HDL) [59–66], physical inactivity and deconditioning [3,67], diabetes [65,68–71], systemic hypertension (in paraplegics) [72,73], arterial stiffness [74,75] and elevated pro-atherogenic inflammation [59,76–79]. Clustering of these risk factors elevates the CMD hazard in the SCI/D population [5,68,80]. Chopra and colleagues [55] reported that in addition to presenting with multiple CMD risk factors at the same time (∼25%), adults with chronic SCI/D are not screened for such risk factors. This pattern was evidenced by the reported insufficient adherence to established Canadian hypertension, dyslipidemia, and diabetes treatment guidelines. Conversely, there is presently insufficient emphasis in the SCI/D community on routine screening for CMD risk, especially considering that adults with chronic SCI/D are more prone to one or more of these risk factors [73,81,82].

The aforementioned challenges to health services are further compounded by poor physical or environmental access to primary care settings, limited family physician awareness of the elevated CMD risk, infrequent participation in general health screening, scarce SCI/D-focused treatment guidelines and screening protocols, inability to perform follow-up evaluations and inaccessibility to prescribed medication and supplements [83,84]. These data highlight the need for targeted knowledge translation (KT) activities to address the gaps between low rates of screening, poor guideline adherence, and high disease rates, likely to minimize the fatal nature of CMD in chronic SCI/D. Indeed, the urgent demand for such activities lies in the fact that the incidence of cardiovascular disease in people with SCI/D is as high as 70% [59]. This proportion agrees with the Canadian Community Health Survey (CCHS) reporting that SCI/D is associated with significantly higher odds of cardiovascular disease [OR] = 2.72, 95% [CI] 1.94–3.82) compared to non-injured individuals [85]. Moreover, evidence of frequent silent ischemia and sudden death after SCI/D further highlight the need for interventions to reduce CMD risk [79].

Altogether, interventions that modify EMD risk are needed to change the health and well-being of adults living and aging with SCI/D in the community. Helping adults with SCI/D to live longer and healthier by reducing the frequency and severity of fractures, diabetes and cardiometabolic disease is of particular relevance to them, their families and members of their support network, including healthcare providers. Among the modifiable risk factors presented, the RIISC team focused on multimodal and inter-professional community-based rehabilitation solutions targeting obesity, inflammatory stress, and physical inactivity. Next, the team plans to introduce primary EMD prevention strategies, followed by stratification of individuals based on EMD risk, and ensure the provision of multimodal interventions to mitigate EMD expression in those with high EMD risk.

Aim statement

The overall aim of the RIISC team is to reduce the frequency and severity of EMD risk, specifically fracture, diabetes, and cardiovascular disease among adults with chronic SCI/D living in the community. Thereby, morbidity and mortality are expected to reduce, while the health, functional abilities and well-being of adults with SCI/D are enhanced. Ultimately, the focus of the RIISC team activities in knowledge creation, clinical implementation, and policy change are to modify the natural course of disease by intervening before EMD expression.

Selecting and evaluating EMD interventions

It is currently unclear the extent to which community-based rehabilitation interventions can modify EMD risk and improve health status in adults with chronic SCI/D. This was the conclusion of the RIISC team’s recent systematic review and scoping synthesis of the data characterizing these interventions and summarizing evidence of their efficacy/effectiveness to modify precursors to EMD (e.g., fractures, diabetes, and heart disease) in community-dwelling adults with chronic SCI/D [1,86]. Systematic searches (2000-November 2017) of MEDLINE PubMed, EMBASE Ovid, CINAHL, Cochrane Database of Systematic Reviews, and PsychInfo were completed. All randomized controlled trials and quasi-experimental/prospective controlled trials comparing rehabilitation/therapeutic interventions with control/placebo interventions in adults with chronic SCI/D were eligible for inclusion. Two authors independently selected studies and abstracted data. Disagreements were resolved by consensus, or third-party adjudication. The Cochrane Collaboration’s Risk of Bias tool was used to evaluate each study.

Of 489 articles identified, 16 articles (11 studies; 396 participants) were eligible for inclusion. Seven studies implemented an exercise intervention (aerobic with/without electrical stimulation (ES), N = 5 [87–95]; ES-assisted resistance training and dietary intervention, N = 1 [96]; physical activity guideline counseling, N = 1) [97], which were performed 2-3 times/week for 18-60 min/session. Four studies implemented dietary intervention or supplementation (polyunsaturated fatty acid/omega-3 supplementation, N = 2 [98–100]; anti-inflammatory diet, N = 1 [101]; alpha-lipoic acid supplementation, N = 1 [102]). No studies included involved whole-body vibration, robotic treadmill or overground interventions. Seven studies included women (60/396 participants) [87,89,95,97–101] and most studies (8/11) were ≤ 24 weeks in duration. No studies assessed the effects of rehabilitation interventions on incident fractures, diabetes and/or heart disease. Individual studies reported that exercise and/or nutrition interventions could improve anthropometric indices, body composition/adiposity, and biomarkers. However, there were also reports of non-significant between-group differences for the above-mentioned outcomes. Low risk of bias was found across most domains for seven articles, and unclear/high risk of bias across most domains for nine articles.

Despite the plethora of exercise interventions, there was very low-quality evidence that rehabilitation interventions can improve EMD risk-related precursors, especially bone mineral density and bone turnover markers. Additionally, there was no evidence that rehabilitation interventions can alter the incidence of EMD clinical endpoints (fractures, diabetes, and cardiovascular disease) in community-dwelling adults living and aging with chronic SCI/D [1]. Further, most of these rehabilitation interventions were tested among relatively small and heterogeneous samples of men with paraplegia over relatively short periods within well-organized clinical or research environments and not community settings.

The small number of studies, and variability across studies limited our ability to make generalizable conclusions. Few studies have been adequately powered or of sufficient duration to determine their efficacy for reducing EMD risk in the chronic phase (≥1-year) after injury, with a few exceptions [85,103,104]. Of note, three of the higher-quality studies were led by active or affiliate members of the RIISC team [87,97,101]. Based on their work, a high-quality longitudinal intervention trial, preferably an RCT or pragmatic clinical trail, greater than 24 weeks in duration, in a representative sample of adults with chronic SCI/D and EMD risk is needed to inform community-based exercise and nutrition rehabilitation strategies for EMD risk. To be of the greatest clinical use, superiority, equivalence, and non-inferiority trial designs need to be completed, which is rarely the case at present. Decision tools are needed to identify the best possible intervention and how to progress it for a given individual with a specific cluster of EMD risk factors at a given time point.

Practice-based research

Conventionally, randomized controlled clinical trials are used to evaluate the efficacy of new treatments in healthcare, as they are perceived to be the “gold standard” for producing evidence that is then used in knowledge products and Clinical Practice Guidelines. However, it has been increasingly recognized that RCTs may not be the most appropriate option for certain clinical situations [105,106], for instance, those involving complex interventions and/or combinations of various approaches, and those involving activity-based therapies, physical activity, nutrition and other lifestyle modification or behaviour change strategies alone or in combination [107].

Challenges to conducting traditional clinical trials include: 1) small numbers of patients in individual clinical research sites, which limits the available sample size and thus the statistical power to detect meaningful change; 2) inability to achieve “blinding”; 3) high drop-out rates, especially when participants are not assigned to the experimental group; 4) lack of understanding regarding the details of “conventional” practice often used as a control; 5) difficulties introducing expensive technologies into clinical practice (particularly when high-level evidence is lacking); and 6) the complex nature of multiple therapeutic interventions as is typical in an interdisciplinary outpatient rehabilitation setting [108]. For example, it may be considered unethical to perform an RCT where the control group would not receive the proposed intervention – should it be already available for consumer use at rehabilitation centers.

Furthermore, adults with SCI/D may be reluctant to participate in a conventional RCT if there is a potential for randomization to the control condition, as they may not necessarily see how the RCT directly adds to their care [109]. Although RCT designs are certainly the gold standard, their very nature results in approaches that emphasize bias reduction through restrictive inclusion and exclusion criteria, often coupled with constraints to maintain the most parsimonious intervention possible for the sake of attribution and feasibility [110]. These factors may result in findings that at their best are not necessarily generalizable to the actual “average” patient in the “real world,” and at their worst may be completely irrelevant to what might be better, more customizable approaches in clinical practice. Given these factors and challenges, one needs to look beyond the conventional RCT design to further study and generate the evidence for efficacy, effectiveness and acceptability of the already available and emerging rehabilitation approaches, which may act to mitigate EMD risk. Pragmatic clinical trials conducted in routine clinical setting using practice based evidence approaches offer some advantages of conventional clinical trials [111]. Two promising alternatives to overcome some of these limitations, and which provide a complementary approach to RCTs, lie in the establishment of self-controlled intervention and practice-based research (PBR) methodologies. While the first method adds to personalized care as per individuals’ perceived affordance [112], PBR designs are characterized by implementing real-world interventions within practice settings [105,106] along with many of the following features:

• All interventions deemed relevant may be included, with the details of each captured systematically.

• Minimal exclusion criteria.

• Multiple outcome measures.

• High generalizability and external validity.

• Especially amenable to subgroup analyses (i.e., identification of which types of patients might respond more to a given intervention than others).

These designs are particularly useful when there are population-based samples, with variability in interventions and outcomes, so that specific practices can be linked to better outcomes. This allows knowledge to be gained beyond the confirmatory evidence obtained from RCTs [113] – rather, the information obtained from PBR could lead to additional insight and “discovery” of new approaches. In this way, the findings can be very complementary to traditional RCT designs in that they can be used to identify new approaches or particular aspects of an intervention that might reflect an appropriate target for a future RCT [114,115]. Widespread adoption of pragmatic trials using practice-based evidence are a key building block of a learning health system and are particularly impactful when the field has a common vision and many stakeholders are focused on the same goals and priorities.

Implementation consideration – behaviour change

Despite knowing the benefits of specific health behaviours (e.g., physical activity, regular exercise and proper nutrition) on EMD risk from a longitudinal cohort study [103,116], the majority of adults with SCI/D do not consistently participate in these health behaviours [117,118]. To enhance engagement and adherence to EMD risk-related health behaviours, the SCI/D field should focus on identifying the best (a) techniques to build health behaviour change interventions; and (b) methods to deliver these types of rehabilitation interventions, while also considering their cost-effectiveness and reach.

Health behaviour change techniques

There is currently limited understanding of the active ingredients that facilitate behaviour change [119] that are optimal for utilization within EMD primary prevention strategies. It has been reported that behaviour change techniques such as action and coping planning [120,121] are effective and person-centred relational techniques can facilitate behaviour change [122–124]. However, without a combined effort to better characterize the active ingredients of health behaviour change interventions, it remains unclear how to best promote and/or modify health behaviours aimed to reduce EMD risk among adults with SCI/D.

Modes of delivery

To broaden the reach of health behaviour interventions, SCI/D-based interventions will need to identify and utilize alternative modes of delivery. Technological tools can be used to deliver and assess behaviour change interventions to help overcome transportation barriers and increase our reach to remote areas. Tele-health approaches (e.g., telephone, video-based communication, video games) and wearable technologies [cameras, smart clothing and accelerometers [125,126]] are viable alternative methods [104,124,127,128] that need further examination among the SCI/D field. Additionally, emerging research shows that peers with motivational interviewing skills could help deliver health behaviour interventions such as physical activity [123,129]. Overall, interventions aimed to reduce EMD risk need careful consideration of new, alternative modes of delivery towards broad-reaching, sustainable interventions to reduce EMD risk among adults with SCI/D.

Characterizing non-active sitting time

Despite growing evidence supporting the benefits of physical activity as part of living with a SCI/D, adults in this population are among the most inactive in today’s society. Current physical activity guidelines for SCI/D recommend that adults perform at least 20 min of moderate-to-vigorous aerobic activity and strength training twice-weekly for each major muscle group to maintain their physical and cardiorespiratory fitness, and muscle strength [3,130]. Despite efforts to adhere to these physical activity guidelines, there are many waking hours during which adults with SCI/D are entirely sedentary. There has been rapid development of evidence suggesting that a whole-day approach to physical activity promotion is ideal, rather than the traditional focus on specific time thresholds each week for engaging in moderate-to-vigorous physical activity [131]. Evidence has accumulated in the general population demonstrating distinct health risks associated with non-active sitting time (NAST) [132], even if an individual is meeting the current physical activity recommendations.

Uninterrupted periods of prolonged sedentary behaviour, or NAST, are associated with metabolic dysfunction, inflammation, and deleterious changes in vascular function, significantly increasing the risk of type 2 diabetes and cardiovascular disease [133,134]. While the resultant public health message from the sedentary physiology literature is to simply stop sitting so much, this same message cannot be applied to adults with SCI/D, many of whom are wheelchair dependent. For community members living with SCI/D, it is time to consider a redirection of focus from recommendations towards the sole achievement of certain physical activity standards at the moderate-to-vigorous intensity range. In fact, there is also a need to move towards recommendations focused on light intensity activities and reduced NAST in people who depend on wheelchairs for household and community mobility. It is hypothesized that the amount of NAST in wheelchair-dependent adults with SCI/D will be directly correlated with EMD risk factors, and that the incorporation of regular bouts of light intensity physical activity throughout the day for people in wheelchairs will share similar cardiometabolic health benefits to regular interruptions in sitting time in the non-injured population [135].

Role and importance of risk stratification

A key tenant of public health policy is risk stratification and appropriate allocation of resources to those with the highest risk. Similarly, adults with SCI/D will require routine EMD risk screening and stratification into low, moderate and high EMD risk groups. Primary prevention strategies will target al.l adults living with SCI/D in the community through community-based interventions. Secondary and tertiary prevention efforts, however, should be directed towards those with a high EMD risk requiring specialty services and expertise to ameliorate EMD. Conventional EMD risk stratification is done through the combined outcomes of the Fracture Risk Assessment Tool – FRAX (https://www.shef.ac.uk/FRAX/tool.jsp_) [136] and the Framingham Risk Score for Hard Coronary Heart Disease(https://www.cvdriskchecksecure.com/framinghamriskscore.aspx) [137] risk assessment tools. Recent reviews of these tools indicate they are poor screening tools for EMD risk after SCI/D, as they tend to underestimate fracture or heart disease risk and/or fail to consider potent SCI/D-specific specific EMD risk factors in their predictive models [55]. One could also argue that regardless of accuracy, stratified recommendations are too absolute for a population as variable as adults living with SCI/D. It should be highlighted, however, that stratification allows for narrowing the spectrum of alternatives available for SCI/D care. That could facilitate the identification of targeted strategies and foster more personalized care as per individual capacities.

Developing-refining process and outcome measures

There is a strong need to develop robust measurement tools and indicators of EMD risk to ensure that people with SCI/D receive appropriate, quality interventions allowing for the best possible results at the individual and societal level in terms of reduced EMD burden and cost. Early after SCI/D onset, and at multiple time points across the lifespan, adults with SCI/D should be screened with robust and sensitive EMD risk assessment tools. Given the impact of EMD risk on the morbidity and mortality of the SCI/D population, these tools must be sensitive to the risk factors most prevalent and most predictive of adverse outcomes. These outcome measures must be developed and validated specifically for adults with chronic SCI/D [3,55], and should be informative for both patients and health care providers.

Further, structure, process and outcome indicators are needed to describe and evaluate the implementation of quality EMD practice and secondary/tertiary EMD prevention interventions [3] and to optimize community participation [138]. With the introduction of routine use of EMD indicators, health care providers and decision-makers will be able to make informed decisions regarding population health strategies for those with low and high EMD risk, that is a risk score < 10 (%) or > 20 (%), respectively – as per both the Canadian FRAX (https://osteoporosis.ca/frax/) [136] and the Framingham Risk Score (FRS) (https://ccs.ca/app/uploads/2020/12/FRS_eng_2017_fnl_greyscale.pdf) [139]. Systematic collection, along with storing of outcomes and indicators will allow monitoring and evaluation of EMD interventions, in addition to comparisons across settings and provinces.

Changing the dialogue to inform policy change

Through its primary prevention strategies, the RIISC team aims to change behaviors on how the role of exercise is viewed in the health and well-being of adults living with SCI/D. Through this research platform, the team plans to host a meeting for consensus on terminologies and definitions involved in SCI/D care, improving communication and clearly describing how different forms of exercise influence neuro-recovery, functional restoration, injury repair, and health maintenance over the lifetime of an individual with SCI/D. The new terminology and related taxonomy of strategies will describe how the type, intensity, and duration of exercise can modulate EMD risk, well-being, health, and participation.

The RIISC knowledge mobilization plan will:

1. Develop specific educational materials to target the SCI/D community and relevant stakeholders.

2. Describe specific business cases to target resources from the Ontario/Quebec Government to enable knowledge development and implementation.

3. Promote prescription of “Exercise Is Medicine” to the SCI/D community and relevant stakeholders including caregivers.

Through these knowledge mobilization activities, a public policy plan will be developed to change the way government views exercise and neuro-recovery in the community:

Neuro-recovery and physical activity within the first two years of SCI/D

Following a SCI/D, rigorous intensity rehabilitation and time are required to maximize the individual’s neuro-recovery and independence. It is established that in-patient rehabilitation length of stay in Canada is not sufficient to address long-term health of individuals with SCI/D [16,140]. More defined neuro-recovery therapy and access to behaviour change programs (e.g., physical activity, diet, and mobility) are required in the community to enable a person to achieve their full potential after SCI/D [1,3]. Essential mechanisms, knowledge, capacity, and wherewithal on how to serve a person’s neuro-recovery with SCI/D in the community are lacking, particularly in regions far from dedicated SCI/D Rehabilitation Centers. Extending rehabilitation services and offering behaviour change programs, including prescribed physical activity in the community, will reduce EMD risk, the overall cost burden of EMD and prepare adults with SCI/D to live independently. These initiatives will pave the way for this population to become self-reliant, while preventing EMD related complications and inspiring full community participation.

Long-term EMD prevention & well-being

People with SCI/D require specialized and defined rehabilitation interventions to manage their chronic EMD risk and achieve health and well-being [3]. These activities will target family physicians, as well as Home and Community Support Services. Presently, the healthcare system does not equitably support people with SCI/D, regardless of whether they live in Ontario or Quebec. The RIISC team’s public policy plan will address the long-term maintenance needs in terms of fracture, diabetes and cardiovascular disease risk reduction and encourage the Ontario and Quebec governments to invest in long-term health maintenance strategies specific to SCI/D.

EMD roadmap to success

The International Classification of Functioning, Disability, and Health (ICF) conceptualizes the dynamic and bidirectional interactions between the body structure and function, and how activity affects the extent of an individual’s participation [141]. The ICF highlights how contextual factors, specifically personal and environmental factors, impact a person’s capacity and performance. The ICF acknowledges the multiple dimensions that contribute to participation restrictions concurrent with EMD risk, and that multiple intervention goals relative to body structure, function, activity and participation should be addressed within an individual context [142]. These complex interrelationships are illustrated in Figure 1.

The RIISC team’s roadmap for successful amelioration of EMD risk and EMD expression seen in Figure 2 is based on the current understanding of the complex interactions between an individual’s impairment, changes in body composition and biomarkers, inactivity and the need for both primary and secondary EMD risk prevention strategies. In this context, primary prevention refers to preventing the onset of diseases via risk reduction and behaviour alteration; secondary prevention refers to procedures/processes that detect and treat pre-clinical pathological changes and seek to restrict disease progression; and, tertiary prevention, not shown in Figure 2, seeks to lessen the impact caused by disease on the patient’s function, participation, longevity, and quality of life.

Figure 2. An overview of the RIISC team’s roadmap to successful amelioration of EMD risk.

A five-panel model showing the RIISC team’s sequence of strategies to ameliorate EMD risk. From left to right, the panels list Baseline characteristics of populations (first), primary prevention/community health promotion (second), metrics (third), secondary prevention/rehabilitation (fourth) and outcomes (fifth).

Invitation to essential partners and measures of success

The RIISC team invites the assistance of essential partners, including but not limited to Osteoporosis Canada, the Canadian Diabetes Society, the Heart and Stroke Foundation and the many provincial community service partners to assist in implementing the proposed roadmap to success, and in reducing the frequency and severity of EMD through primary, secondary and tertiary prevention initiatives. Effective implementation of the EMD Roadmap to Success will require sustained KT efforts with multiple outputs reflected in the form of KT indicators. As shown in Figure 3, gaps or the so called “valleys of death” mainly exist when conveying knowledge from basic to clinical research, and from clinical research to its application in healthcare practice [143]. Such valleys encompass obstacles that directly challenge the various success indicators presented in this manuscript and which justify the relevance of ongoing, integrative initiatives warranted to both monitor and optimize the proposed information continuum as an attempt to minimize loss in translation [144]. In fact, recent findings specific to Physical Medicine and Rehabilitation showed that to date, considerable financial resources are still put to waste due to expensive healthcare investigation without practical implementation, and that in addition to evidence publication, several activities (e.g., multicomponent KT interventions) are needed for successful implementation of evidence-based practices [145]. These findings reinforce the relevance of our Roadmap for the establishment of rehabilitation strategies in SCI/D beyond the second valley – that is when solutions probed in clinical research settings finally “reach the bedside.”

Figure 3. RIISC team’s knowledge translation indicators of success. (clinical translational continuum modified from steven reis, University of pittsburgh and harold pincus, columbia University and knowledge translation indicators modified from the University of Toronto, department of Medicine academic Strategic plan 2011–2016).

A two-part figure. Above, a wave graph adapted from Steven Reis Clinical Translational Continuum. It shows Basic Biomedical Research, Clinical Science & Knowledge, and Clinical Practice & Health Decision Making as three decreasing wave peaks separated by two valleys over a rightward arrow. Below, an eight-panel model showing a list of knowledge translation indicators of success including, from left to right, Reach, Usefulness, Use, Partnership & Collaboration, Policy, Mentorship and Consumer Perspective.

Conclusions

Altogether, this paper covered relevant themes associated with EMD in SCI/D, all depicted in a framework that is essentially plausible, albeit yet to be fully developed. The presented model warrants reframing in respect to both the multiple contexts it aims to address, and the future steps which will secure the model’s successful implementation – a process that must equally consider the competing priorities of the two provinces (health systems) involved, the needs of adults living with SCI/D, and our joint capacity to deliver the expected outcomes.

This manuscript further underscores the current disconnect between rehabilitation service provision and the evaluation of their impact on long term health outcomes and cost-effectiveness. At this stage, a prerogative of our interprovincial initiative is the understanding that single-disease paradigms neither effectively improve the lifestyle nor the health course of adults living with SCI/D. For this population, health complexity is significantly increased due to multimorbidity that yields a pressing need for multimodal responses. The latter should include patients as co-designers, particularly in respect to therapeutic strategies and aims (e.g., disease prevention, mitigating disease severity, general wellness). The MOH-REPAR collaborative offers the opportunity to create a platform to foster patient engagement, and to build capacity in leading holistic rehabilitation models with the potential to mitigate EMD risk, gather investment funding, leverage health system priorities, and meet the needs of those who live with SCI/D in Canada and ultimately – should proper contextual adaptations be made – all over the world.

In addition to adults living with SCI/D, clinicians, policy makers and other stakeholders are invited to work as partners to best disseminate the RIISC team’s products and advocate for the implementation of programs and community services that could equitably address the goals of the various parties involved in the complexity of SCI/D care, including specific population needs in terms of healthcare logistics (e.g., acute, sub-acute and chronic rehabilitation settings) and health outcomes (e.g., endocrine metabolic function, motor control and health-related quality of life).

Acknowledgments

The RIISC team is thankful to Laurent Boyer, David Ditor, Isabelle Coté, Mir Hatef Shojaei, Jawad Christie, Stephanie Marrocco, Mohammad Alavinia and Jerry Mings for their insightful academic contributions to the RIISC model, and hence to this manuscript.

Disclosure statement

Dr Craven is the Toronto Rehab/University of Toronto Chair in SCI Rehabilitation. Dr Craven is the Chair of the Canadian Spinal Cord Injury - Rehabilitation Association and a Member of the Osteoporosis Canada Scientific Advisory Council. Dr Craven received consulting fees from Praxis in the past. No potential conflict of interest was reported by the authors.

Additional information

Funding

This study was funded in part by interprovincial Grants (MOH-REPAR Grant # 719-D and ONF-REPAR Grant # 2018 – REPAR − 1026). The opinions, results and conclusions in this paper are those of the authors and are independent from their Province.