https://orcid.org/0000-0002-4749-7611
Figures
Abstract
This review aimed to systematically review and meta-analyze the effects of interventions in improving bracing compliance among adolescent idiopathic scoliosis (AIS) patients. Eight databases were searched from their inception to April 2022. The eligibility criteria included controlled studies that used any type of intervention to enhance bracing compliance in braced AIS patients. Two researchers independently screened articles and extracted data based on the PICO (participant, intervention, comparator, and outcome) framework. Quality appraisal of included studies was performed using GRADE (overall assessment), and the risk of bias was assessed with Cochrane RoB Tool 2 for randomized controlled trials (RCT) and ROBINS-I for non-RCT studies. The primary outcome was bracing compliance and secondary outcomes included Cobb Angle and measurements for quality of life. Six eligible studies involving 523 participants were included. All studies were evaluated as low or very low quality with a high risk of bias. Four types of interventions were identified, including sensor monitoring (n = 2, RCTs), auto-adjusted brace (n = 1, RCT), more intensive or collaborated medical care (n = 2), and psychosocial intervention (n = 1). A meta-analysis of 215 patients from the three RCTs suggested that the compliance-enhancing intervention group had 2.92 more bracing hours per day than the usual care control (95%CI [1.12, 4.72], P = 0.001). In subgroup analysis, sensor monitoring significantly improved bracing wearing quantity compared to usual care (3.47 hours/day, 95%CI [1.48, 5.47], P = 0.001), while other aforementioned interventions did not show a significant superiority. Compliance-enhancing interventions may be favorable in preventing curve progression and promoting quality of life, but the improvements cannot be clarified according to limited evidence. In conclusion, although the results of this study suggested that sensor monitoring may be the most promising approach, limited high-quality evidence precludes reliable conclusions. Future well-designed RCTs are required to confirm the actual benefit of compliance-improving interventions in clinical practice.
Citation: Li X, Huo Z, Hu Z, Lam TP, Cheng JCY, Chung VC-h, et al. (2022) Which interventions may improve bracing compliance in adolescent idiopathic scoliosis? A systematic review and meta-analysis. PLoS ONE 17(7): e0271612. https://doi.org/10.1371/journal.pone.0271612
Editor: Alessandro de Sire, University of Catanzaro: Universita degli Studi Magna Graecia di Catanzaro, ITALY
Received: April 26, 2022; Accepted: July 1, 2022; Published: July 20, 2022
Copyright: © 2022 Li et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
Data Availability: All relevant data are within the manuscript and its Supporting information files.
Funding: This work was supported by the General Research Fund of the The Research Grants Council of Hong Kong (https://www.ugc.edu.hk/eng/rgc/funding_opport/grf/; Grant Number: 14614416 [to BHKY]) as part of the project, “The use of mindfulness-based intervention for improving bracing compliance for adolescent idiopathic scoliosis patients: A randomized controlled trial”. The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.
Competing interests: The authors have declared that no competing interests exist.
Introduction
Adolescent idiopathic scoliosis (AIS) is the most common form of scoliosis affecting about 1–4% of adolescents [1]. Several long-term disabling complications of curve progression have been noted, including back pain, cardiopulmonary problems, spinal cord injury, and psychosocial concerns, which highlighted the needing for tailored strategies and precise management in AIS treatment [2–5]. Even though few studies suggested that spinal fusion surgery may not generate severe impairment afterward [6, 7], the benefit of surgery is still controversial and more than 500 million dollars per year are spent in the United States alone on AIS surgeries [7, 8]. Thus population-based screening and proper treatment at the onset of disease to prevent curve progression will not only improve AIS patients’ health outcomes but also save healthcare dollars [9–13].
Bracing, which was suggested to be effective in preventing curve progression to surgery threshold [8, 14, 15], is the most commonly used conservative treatment beyond monitoring for AIS patients with an immature skeleton of Cobb angle between 25–45 degrees [3]. To achieve the therapeutic potential of brace treatment, patients’ compliance with bracing is crucial. However, poor compliance with bracing is widely reported in braced AIS patients (33%~77% of prescribed hours) [16–18]. Reasons for non-compliance are associated with the adverse effects of brace treatment, such as the negative cosmetic appearance [19], functional discomfort resulting from pressure points [8], irritation in hot weather [15], and restriction of movement [15]. Emotional discomfort and effects on quality of life are also considered to be important potential psychosocial determinants of compliance [20, 21].
Although the International Society on Scoliosis Orthopaedic and Rehabilitation Treatment (SOSORT) guideline has emphasized the importance of bracing [22], there are currently no clinical guidelines or recommendations on interventions to improve bracing compliance, and there remains a dearth of reviews focusing on the discussion of the behavioral aspects on how to improve post-bracing clinical outcomes. Furthermore, despite that recent studies have reported hypothetical factors that may influence compliance [23], thus far, no systematic review has been conducted to synthesize all existing evidence on the effectiveness of interventions in improving compliance with the bracing regimen. This systematic review aims to fill this knowledge gap by synthesizing the effectiveness of compliance-enhancing interventions for AIS patients.
Materials and methods
This systematic review was reported following the PRISMA statement for systematic review and meta-analysis [24] (S1 Table).
Criteria for considering studies for this review
The eligibility criteria were constructed according to the Participants (P), Interventions (I), Comparator (C), and Outcomes (O) (PICO) framework.
Study designs.
Randomized controlled trials (RCTs), controlled clinical trials without randomization (CCTs) (definition of CCT: investigators had direct control over study conditions but interventions were not randomly assigned [25]), and controlled observational studies (cohort studies, and case-control studies) were included because it was anticipated that very few RCTs would be identified.
Population.
We included studies in which all patients, following a confirmed clinical and radiological diagnosis of AIS, were prescribed bracing treatment.
Interventions and comparators.
The experimental interventions in this review included all types of interventions that were considered to aim at improving bracing compliance. Controls were usual care of bracing treatment without any component to increase bracing compliance.
Outcomes.
The primary outcome is bracing compliance, as measured by the percentage of prescribed regimen, the number of daily bracing hours, or the proportion of compliant patients [8, 26]. Binary outcome variables (good compliance vs poor compliance) were presented as risk ratio (RR) or odds ratio (OR), and continuous outcomes were presented as means, standard deviations (SD), and/or standard errors (SE). We did not set the restriction on the assessment method of bracing wearing compliance (i.e., self-report or sensor records).
Secondary outcomes are scoliosis parameters, as measured by Cobb angle in degrees, angle of trunk rotation (ATR) in degrees, the number of patients who have progressed by more than 5° Cobb, or the number of subjects for whom surgery was prescribed. Quality of life data, as measured by specific validated questionnaires, e.g. SRS-22, SF-36 [27], BrQ [28] was also extracted.
Search methods for identification of studies
Online literature searches were performed on the six international databases and two Chinese databases. The starting year is the inception for each database, e.g. 1946 for Medline, and the search was conducted in April 2022. The terms of participants (AIS patients) and primary outcome (bracing compliance) were intentionally applied to achieve a comprehensive retrieval of records from databases without the restriction of publication status (Fig 1). The search strategy for each database is detailed in the S2 Table in the appendix. The reference lists of relevant reviews were scrutinized for further articles. Searching of the main online sources of ongoing trials (National Research Register, meta-Register of Controlled Trials; Clinical Trials) for available data was conducted, including grey literature, including conference proceedings and Ph.D. theses. Authors of registered trials were contacted for possible available data to identify any further studies we could include.
Data collection and analysis
Selection of studies.
Two review authors (XL, ZSH) independently screened the search results based on titles and abstracts, followed by an independent full-text assessment of potentially relevant studies. The pre-specified inclusion and exclusion criteria of eligibility which were drafted according to the PICO framework were adopted for studies selection. Disagreements between the two authors were resolved by discussion until consensus was reached or through discussion with a third researcher (BHKY).
Data extraction and management.
The data of the included papers were extracted in predesigned data extraction form by two independent reviewers (XL and ZHH). Extracted variables included: (1) study characteristics (study design, country, year, recruitment modality, risk of bias); (2) patient characteristics (number of participants, age, sex, baseline compliance); and (3) description of the experimental and comparison interventions, co-interventions, adverse effects, duration of follow-up, outcomes assessed, and results. Disagreements were resolved through discussion. Key findings were summarized in a narrative format and then assessed for inclusion in a meta-analysis where possible.
Assessment of risk of bias and methodological quality.
We assessed the risk of bias in RCTs in this review using the Cochrane risk-of-bias tool for randomized trials (RoB 2) [29] according to the following domains: bias arising from the randomization process; bias due to deviations from intended interventions; bias due to missing outcome data; bias in the measurement of the outcome; and bias in the selection of the reported result. Observational studies and CCTs were assessed using the Risk Of Bias In Non-randomised Studies-of Interventions (ROBINS-I) tool [30, 31]. The ROBINS-I assesses four broad areas: confounding, selection bias, information bias, and reporting biases.
The overall quality of the evidence for the primary outcome was assessed with the adapted GRADE approach [32, 33]. Domains that may decrease the quality of the evidence are study design and implementation (risk of bias), inconsistency (heterogeneity), indirectness (inability to generalize), imprecision (insufficient or imprecise data), and publication bias across all studies that measure that particular outcome. The quality of the evidence on a specific outcome is based on the performance against six factors: study design, risk of bias, consistency, and directness of results, the precision of the data, and publication bias across all studies that measured that particular outcome.
Two reviewers (XL, ZHH) appraised each study independently and disagreements were resolved through discussion with a third reviewer (BY).
Data synthesis
The primary analysis was comparisons of compliance-enhancing interventions versus no intervention or other interventions for two or more studies with the same study design. Standard deviations were calculated for meta-analyses purposes if they weren’t provided. The mean differences (MD) with 95% confidence intervals were calculated for all continuous variables. Multiple subgroups classified according to the dose of interventions were combined into a single intervention group [34]. In subgroup analysis, pooled estimates were conducted where two or more studies were adopting a similar intervention. The random-effects inverse variance model was used due to the possible variation in study methodology and bracing regimen applied (brace type, recommended wearing hours). Heterogeneity was assessed using the I2 value. Review Manager Software, version 5.3 was used for the analysis.
Results
Results of the search
From the bibliographic search, we identified 807 references. After removing duplicates, we identified 543 potentially relevant references; 517 were excluded based on title and abstracts, leaving 26 studies that were acquired in full text or study report with available information for further evaluation. (Fig 1). After conducting a full-text review, six studies [35–40] were included in our systematic review. A hand-search of references of the included studies revealed no further relevant publications. Substantive descriptions of the included studies can be seen in Table 1, while the reasons for excluding studies after full-text review are listed in S3 Table.
Included studies
The six included studies were published between 2012 and 2020. They consisted of three RCTs [35, 36, 40], one non-randomized controlled trial [37], one retrospective cohort study [39], and one retrospective case-control study [38] (Table 1). The median sample size was 31 participants (range: 21–246 participants). Two studies were conducted in the USA [35, 36], two in Italy [38, 39], one in Denmark [37], and one in Hong Kong [40]. The mean age of participants across 6 studies ranged from 11.9 to 15.8 years and the proportion of girls in each study ranged from 68.4% to 100%. Bracing compliance was assessed through self-reporting [37, 38] and sensor monitoring [35, 36, 39, 40].
Risk of bias and quality assessment
The overall risk of bias was high for three RCTs (Fig 2), and in particular, there were some concerns or high risk of bias due to missing outcome data and some concerns or high risk of bias due to following modified intention -to treat principle. In Karol et al study [41], only patients with complete data were included in the final analysis, which increased the risk of bias toward favoring the intervention. In Miller et al. trial [35], the severe drop-out rate and the limited sample size are also a worry. Furthermore, none of the RCTs collected baseline compliance data and as a consequence not be able to adjust it in their analyses to prevent potential regression-to-mean results. Fig 3 illustrates the results of the risk of bias assessment of the observational studies and non-randomized controlled studies. Overall, all three studies suffer from a high risk of bias, particularly bias due to confounding and the measurement of outcomes. Details of the quality ratings of GRADE are presented in the S4 Table.
Interventions
Sensor monitoring [35, 36], psychosocial intervention [39], interventions of the change of medical care [37, 38], and auto-adjusted brace [40] were identified in included studies (Table 1).
Two RCTs [35, 36] investigated sensor monitoring vs. not monitoring. Patients in the intervention group were monitored using electronic sensors, which were installed on the braces and consisted of a temperature probe and can store compliance data measured by temperature [35]. In addition to an embedded temperature sensor, Karol et al. [36] provided patients with feedback counseling according to records retrieved from sensors at clinical visits, while standard clinical service was provided to the control group.
In a non-randomized trial, Al-Aubaidi et al. [37] compared an intensive hospitalization approach, which provided more prompt adjustments for participants for bracing adaptation in a few days, with a less intensive approach that was conducted in an outpatient clinic. Noteworthy, in Denmark, the intensive hospitalization approach is the usual practice, while the outpatient clinic approach was the intervention. This is different from other countries, at least those that were included in this systematic review, where outpatient service is more widely used as usual care for braces adaption and adjustment in scoliosis clinical practice. Using a retrospective case-control design, Tavernaro et al. evaluated the compliance-enhancing effect of a complete, multi-professional expert rehabilitation team [38], which involved parents and patients, collaborating closely through the aggregation of physiotherapists.
In a cohort study, the effect of the Cognitive Behavioural Approach dispensed during Physiotherapic Scoliosis Specific Exercises (CBA-PSSE) was studied [39]. Bracing compliance (0–4 months) was compared among three patient groups with different levels of adherence to CBA-PSSE intervention (good adherence to the intervention group (I):≧2 sessions, poor adherence group (P):1 session, control group (C): 0 sessions).
One RCT [40] was found to investigate the effectiveness of a newly developed automated pressure-adjustable orthosis, which could maintain a more consistent interfacial corrective effect at the prescribed level by inflation and deflation of the air bladder. Compliance monitoring sensors of similar size compared to the smart device for the intervention group were installed in the same area in the conventional rigid braces for the control group.
Intervention effects on bracing compliance
The results of the effects on bracing compliance for included studies are summarized in Table 2. The patients in groups with good (number of attending sessions>1) (I) and poor (number of attending sessions = 1) (P) adherence to the CBA intervention in the Negrini et al. study [39], were combined into a single intervention group in the meta-analysis. To be comparable with other studies, we treated outpatient clinics in Al-Aubaidi et al. study [37] as the control group in the meta-analysis. Three studies reported average wearing hours per day [35, 36, 40], while five studies reported the percentage of prescription wearing time as the outcome was reported or could be calculated [35, 37–40]. Using the former as the outcome measure, a meta-analysis was conducted with 215 patients from three RCTs [35, 36, 40] (intervention: n = 114, control: n = 101), and it indicated that higher bracing compliance can be achieved through interventions, i.e., the interventions group had on average 2.92 more bracing hours per day (95%CI [1.12, 4.72], P = 0.001). Low inconsistency was found in effect size (P = 0.35, I2 = 4%). (Fig 4).
Sensor monitoring vs non-monitoring.
Two RCTs which consisted of 192 participants, reported average bracing hours per day as the outcome [35, 36]. The meta-analysis revealed that bracing compliance significantly improved with monitoring when compared to no monitoring (3.47 hours/day, 95%CI [1.48, 5.47], P < 0.001). There was no evidence of heterogeneity (P = 0.37, I2 = 0%) (Fig 5A).
Intervention of medical care vs usual care.
Compliance data, which was defined as the percentage of the prescription wearing time, from 2 studies [37, 38] and 62 participants were available (Tables 1 and 2). The results favored the change in medical care, but the improvement in compliance was not significant (5.18%, 95% CI [−19.28, 29.64], P = 0.68) (Fig 5B). High inconsistency in effect size was observed (P = 0.007, I2 = 86%). For the result in single studies, the percentage (MD = 17%, 95%CI [5.05, 28.95], P = 0.03) and the odds ratio of achieving good compliance (OR = 5.5, 95%CI [3.6, 7.4], P<0.05) were reported to be significantly higher for a collaborated medical team approach as compared with usual care [38], while no superiority was detected for the hospitalization intervention with more intensive medical care [37].
Psychosocial intervention vs no intervention.
For the Cognitive Behavioural Approach versus no intervention comparison, data from only one study was available (Table 2). The percentage of prescription wearing time favored of intervention groups more than the control but did not reach the significance (I vs C: MD = 0.97%, 95%CI [-3.76, 5.70]; P vs C: MD = 3.96%, 95%CI [-1.26, 9.18]) (Table 2). The combined intervention group also did not present a significant superiority compared with the control (MD = 1.77%, 95%CI [-2.84, 6.38]).
Auto-adjusted brace vs usual care (conventional orthosis).
In the automated pressure-adjustable brace versus conventional rigid orthosis analysis, there was only one RCT [40] with 23 participants where data on bracing compliance was available. There was no significant difference between the effect of the new brace and the conventional brace (MD = 1.10 h/day, 95%CI [-2.53, 4.73], P = 0.55). (Table 2, Fig 4).
Radiographic outcome
The rate of bracing success (curves progression< 6°) was higher in the sensor monitored group than that in the non-intervention group (55/93 vs 36/78, RR 1.28, 95% CI 0.96–1.72, P = 0.098), and quite the opposite for the rate of failure (progression to cobb angel> 50°or surgery) (23/93 vs 28/78, RR 0.69, 95% CI 0.43–1.09, P = 0.114) [36]. Cobb angle data from one trial [40] revealed that at 1-year follow-up 4 out of 11 patients who were treated with an auto-adjusted brace had a curve reduction of more than 5°; while the number was 2 out of 12 in the control group with rigid braces (P = 0.156). The results of radiographic outcomes favor interventions in both studies although not significantly so.
Quality of life
A better level of quality of life measured by SRS-22 was found to be promoted by team approach [38] (4.13±0.46 vs 3.39±0.60, MD = 0.74, 95%CI [0.26, 1.22], P = 0.01), but not by auto-adjusted brace [40] (4.3±0.2 vs 4.3±0.4, MD = 0.00, 95%CI [-0.26, 0.26], P = 1.00). There was no significant difference concerning the score of the scoliosis Quality of Life Index (SQLI) questionnaire [42] between the outpatient service contrast the hospitalization in the Al-Aubaidi et al. study [37] (median = 77, IQR:73–87 vs median = 78, IQR:69–88).
Discussion
This is the first review that has systematically analyzed the effectiveness of compliance-enhancing interventions in braced AIS patients. We identified four approaches that were studied in the AIS population, including sensor monitoring [35, 36], more intense or collaborated medical care [37, 38], psychosocial intervention [39], and auto-adjusted brace [40]. Among the identified interventions, sensor monitoring may be the most promising approach. Interventions may be favorable concerning the effect of preventing curve progression [36] and promoting an improved quality of life [37, 38]. However, the clinical importance of the improvements still cannot be clarified according to limited evidence and limitations in the methodology quality of current studies.
In this study, electronic monitoring is considered to be most promising given its effect size on bracing compliance improvement and it has been demonstrated to be effective in improving medication adherence for patients with chronic diseases [43–46]. More optimal compliance could be observed when patients were informed to be objectively monitored, which may be a good use of the Hawthorne effect [47, 48]. Besides, as an accurate assessment of brace wearing is the basic necessary information when we try to improve bracing compliance, objective sensor monitoring should be considered as the routine method in the management of braced patients with AIS in clinical practice to ensure the effect of bracing treatment.
The positive influence of the integrated clinical team approach is also in line with evidence supporting the use of innovative, modified health care teams in enhancing patients’ compliance rather than traditional, independent physician practice and minimally structured systems [49, 50]. The underlining reason could be that good communication among patients and health professionals may contribute to a better flow of important clinical and psychosocial information, building trust, and providing support [51].
AIS patients may experience issues of low self-esteem, body image, social role definition, and stress [52], which may cause them to rebel against the regimen [53]. Therefore, although the certainty of the evidence is limited for confirming the effectiveness of CBT+PSSE intervention with meta-analysis in this review, it is still a considerable attempt to involve social behavior theory models (e.g., social cognitive theory self-regulation model, and social support theory) in the development of future effective interventions, since the wide application on treatment adherence in other diseases [54–57].
According to previous evidence, bracing compliance of AIS patients can be affected by age [58], gender [59], BMI index [60], the type of braces (structure and appearance) [15, 61–64], and brace wear pattern (daytime/ nighttime or part-time/ full-time) [65, 66]. As the core device of brace treatment, the optimal brace is the primary consideration. Braces promote proper spinal growth and motor behaviors by reducing unnatural loading and asymmetrical movements, which can be brought about by mechanical forces and external and proprioceptive inputs [67, 68]. Adequate wearing quantity could contribute to orthotic treatment effectiveness [8, 69]. Compared with traditional rigid spinal orthosis (e.g., Boston brace), elastic or flexible braces, in which movement is only partially restricted, were suggested as being more acceptable but with considerably higher rates of curve progression [15, 17, 70, 71]. Orthosis stabilization power and patient experience need to be balanced to achieve the best in quality and quantity of brace wearing. The relative efficacy of different types of braces on bracing compliance cannot be confirmed in this review, due to only one study being identified that provided comparative data. Compliance data for different types of braces need to be further collected and compared in future studies.
Only one of the included studies examines radiographic outcomes (e.g., cobb curve progression) [36], and 2 studies evaluated patient-relevant outcomes (e.g., quality of life) [37, 38]. Given the limited number and quality of studies, an evaluation of the actual benefit of the compliance-improving interventions is difficult. Previous evidence suggested that poor bracing compliance is associated with poorer quality of life and a higher risk of progression [8, 72]. Additional well-controlled research establishing whether the current findings generalize to bracing compliance, patient-related outcomes, and clinical outcomes is important for establishing the viability of hypnosis as an effective intervention.
Limitation
Although promising insights have emerged from this review, the current findings have several important limitations. The largest challenge is that only 6 eligible studies have focused on this topic. The lack of studies makes it difficult to analysis by each sub-intervention and derive a definitive reliable conclusion. Given high heterogeneity in methodologies, the included studies are difficult to be compared directly. There are differences in regimens applied (recommended wearing hours) and baseline characteristics (age and gender) across the included studies which may influence adherence [23]. Furthermore, for self-reporting assessments, which were adopted by two included studies [37, 38], a higher estimation of intake rather than the true adherence rate has been shown [73]. Objective assessment methods (e.g., electronic temperature monitoring) need to be considered in future studies. In addition, the possible confounding factors of environmental temperature and unfit braces should be noted in the objective assessment. Besides, the follow-up time of three out of six studies was shorter than 1.5 years [35, 37, 39], and only Karol et al. study covered the entire bracing treatment period [36]. It is still uncertain whether the intervention effect was sustained throughout the long treatment period, which consists of an average of 2.5 years of bracing [1]. Future studies might incorporate a longer follow-up period to clinical endpoints into their study designs while being mindful of the greater attrition rates with increased study lengths.
The methodological quality of the included trials also needs to be considered when interpreting these results. Firstly, only one study [37] included sample size calculations to detect statistically significant differences in their methodology. Adequate power is needed to reduce the risk of random error and false-positive results [74]. Secondly, in all four included trials [35–37, 40], the data were not analyzed according to intention-to-treat principles. This criterion is a source of bias because it could be assumed that patient groups that are at risk of being nonadherent are also more likely to be lost to follow-up. Thirdly, 5 studies reported without a baseline assessment of compliance outcomes [35–39] and all of the included studies did not conduct difference in difference analysis to evaluate the effectiveness of interventions [75]. The effect of interventions can be overestimated or underestimated when the sample size is limited. The likelihood of finding only slight differences between groups can be increased by a high baseline adherence level, which may result in a ceiling effect that limits room for improvement and a marginal group effect.
Impact on future practice and research
The modest effect sizes found in this review demonstrate the difficulty in changing compliant behavior which was also reported in the management of other diseases [76, 77]. In the future intervention design, clinicians need to consider the challenges in changing adherence behavior and make this a priority. Other add-on medical care, such as Schroth physiotherapeutic exercises, also can be recommended to individuals who are not compliant with bracing treatment as compensation for the standard treatment [78]. Furthermore, we are making the following recommendations for future brace compliance intervention trials in their study design to address the aforementioned limitations and heterogeneity we found in this systematic review. Firstly, a standardized objective assessment of bracing compliance (e.g. sensor monitoring) is essential for an accurate evaluation of the intervention effect. Secondly, to avoid regression-to-mean findings, due to an imbalance in baseline bracing measure, we strongly recommend that future RCTs should collect baseline compliance data (using the objective measure mentioned above) and adjust it by using the analysis of covariance (ANCOVA) principle, which is the preferred method for RCT when the outcome is continuous. ANCOVA also provides a greater statistical power to detect a true treatment effect than other approaches (post-only, pre vs post, or percentage change) [79, 80]. Furthermore, the follow-up time of all identified studies is limited to assess the sustainability of the intervention and its impact on clinical outcomes, e.g., curve progression. A longer follow-up time designed according to the 2.5 years of bracing treatment time on average could be considered [1]. Lastly, there are likely other factors that were not captured in these intervention techniques and could impact adolescents’ decision to wear their braces or not. Few interventions included in this review have attempted to tailor intervention approaches to patients’ barriers to good bracing compliance, e.g., negative cosmetic appearance [19], discomfort, and restriction of braces resulting from pressure points [8, 15], emotional problems, or poor quality of life [20, 21]. Future interventions can be designed using multiple strategies targeting these barriers to increase the likelihood of addressing the reasons for non-compliant for any given AIS patient.
Conclusion
Interventions of sensor monitoring, more intense or collaborated medical care, psychosocial intervention, and auto-adjusted brace have been studied for improving bracing compliance in AIS patients. Of these, sensor monitoring may be the most promising approach. The evidence is, however, not conclusive due to various limitations. We recommend future randomized controlled trials of bracing compliance intervention to have an adequate sample size, with longer follow-up to clinically relevant endpoints, and using objective measurements of compliance outcomes at baseline and post-intervention.
Supporting information
S1 Table. Preferred reporting items for systematic reviews and meta-analysis (PRISMA) checklist.
https://doi.org/10.1371/journal.pone.0271612.s001
(DOCX)
S3 Table. List of excluded studies and reasons for exclusion.
https://doi.org/10.1371/journal.pone.0271612.s003
(DOCX)
S4 Table. Overall GRADE quality assessment for the primary outcome (bracing compliance).
https://doi.org/10.1371/journal.pone.0271612.s004
(DOCX)
References
- 1. Cheng JC, Castelein RM, Chu WC, Danielsson AJ, Dobbs MB, Grivas TB, et al. Adolescent idiopathic scoliosis. 2015;1:15030.
- 2. Weinstein SL, Dolan LA, Cheng JCY, Danielsson A, Morcuende JA. Adolescent idiopathic scoliosis. The Lancet. 2008;371(9623):1527–37.
- 3. Weinstein SL, Ponseti IV. Curve progression in idiopathic scoliosis. J Bone Joint Surg Am. 1983;65(4):447–55. pmid:6833318
- 4. Weinstein SL, Dolan LA, Spratt KF, Peterson KK, Spoonamore MJ, Ponseti IV. Health and function of patients with untreated idiopathic scoliosis: a 50-year natural history study. JAMA. 2003;289(5):559–67. pmid:12578488
- 5. Lippi L, de Sire A, Desilvestri M, Baricich A, Barbanera A, Cattalani A, et al. Can scoliosis lead to spinal cord ischaemia? Early diagnosis and rehabilitation: A paradigmatic case report and literature review. Journal of Back and Musculoskeletal Rehabilitation. 2021;34:43–7. pmid:33164924
- 6. Byun YM, Iida T, Yamada K, Abumi K, Kokabu T, Iwata A, et al. Long-term pulmonary function after posterior spinal fusion in main thoracic adolescent idiopathic scoliosis. PloS one. 2020;15(6):e0235123. pmid:32584916
- 7. Martin CT, Pugely AJ, Gao Y, Mendoza-Lattes SA, Ilgenfritz RM, Callaghan JJ, et al. Increasing hospital charges for adolescent idiopathic scoliosis in the United States. Spine. 2014;39(20):1676–82. pmid:24983937
- 8. Weinstein SL, Dolan LA, Wright JG, Dobbs MB. Effects of bracing in adolescents with idiopathic scoliosis. The New England journal of medicine. 2013;369(16):1512–21. pmid:24047455
- 9. Workman JK, Wilkes J, Presson AP, Xu Y, Heflin JA, Smith JT. Variation in Adolescent Idiopathic Scoliosis Surgery: Implications for Improving Healthcare Value. J Pediatr. 2018;195. pmid:30448013
- 10. Lee CF, Fong DY, Cheung KM, Cheng JC, Ng BK, Lam TP, et al. Costs of school scoliosis screening: a large, population-based study. Spine. 2010;35(26):2266–72. pmid:20531070
- 11. Lee CF, Fong DY, Cheung KM, Cheng JC, Ng BK, Lam TP, et al. Referral criteria for school scoliosis screening: assessment and recommendations based on a large longitudinally followed cohort. Spine. 2010;35(25):E1492–8. pmid:21102278
- 12. Scaturro D, Costantino C, Terrana P, Vitagliani F, Falco V, Cuntrera D, et al. Risk Factors, Lifestyle and Prevention among Adolescents with Idiopathic Juvenile Scoliosis: A Cross Sectional Study in Eleven First-Grade Secondary Schools of Palermo Province, Italy. International journal of environmental research and public health. 2021;18(23). pmid:34886069
- 13. Scaturro D, de Sire A, Terrana P, Costantino C, Lauricella L, Sannasardo CE, et al. Adolescent idiopathic scoliosis screening: Could a school-based assessment protocol be useful for an early diagnosis? Journal of back and musculoskeletal rehabilitation. 2021;34(2):301–6. pmid:33285626
- 14. Nachemson AL, Peterson LE. Effectiveness of treatment with a brace in girls who have adolescent idiopathic scoliosis. A prospective, controlled study based on data from the Brace Study of the Scoliosis Research Society. J Bone Joint Surg Am. 1995;77(6):815–22. pmid:7782353
- 15. Wong MS, Cheng JC, Lam TP, Ng BK, Sin SW, Lee-Shum SL, et al. The effect of rigid versus flexible spinal orthosis on the clinical efficacy and acceptance of the patients with adolescent idiopathic scoliosis. Spine. 2008;33(12):1360–5. pmid:18496349
- 16. Vandal S, Rivard CH, Bradet R. Measuring the compliance behavior of adolescents wearing orthopedic braces. Issues in comprehensive pediatric nursing. 1999;22(2–3):59–73. pmid:10786513
- 17. Hasler CC, Wietlisbach S, Büchler P. Objective compliance of adolescent girls with idiopathic scoliosis in a dynamic SpineCor brace. Journal of children’s orthopaedics. 2010;4(3):211–8. pmid:21629374
- 18. Lou E, Hill D, Hedden D, Mahood J, Moreau M, Raso J. An objective measurement of brace usage for the treatment of adolescent idiopathic scoliosis. Medical engineering & physics. 2011;33(3):290–4. pmid:21112234
- 19. Clayson D, Luz-Alterman S, Cataletto MM, Levine DB. Long-term psychological sequelae of surgically versus nonsurgically treated scoliosis. Spine. 1987;12(10):983–6. pmid:3441826
- 20. Donzelli S, Zaina F, Negrini S. In defense of adolescents: They really do use braces for the hours prescribed, if good help is provided. Results from a prospective everyday clinic cohort using thermobrace. Scoliosis. 2012;7(1):12-. pmid:22651570
- 21. Ugwonali OF, Lomas G, Choe JC, Hyman JE, Lee FY, Vitale MG, et al. Effect of bracing on the quality of life of adolescents with idiopathic scoliosis. The Spine Journal. 2004;4(3):254–60. pmid:15125845
- 22. Negrini S, Donzelli S, Aulisa AG, Czaprowski D, Schreiber S, de Mauroy JC, et al. 2016 SOSORT guidelines: orthopaedic and rehabilitation treatment of idiopathic scoliosis during growth. Scoliosis and spinal disorders. 2018;13(1):3. pmid:29435499
- 23. Rahimi S, Kiaghadi A, Fallahian N. Effective factors on brace compliance in idiopathic scoliosis: a literature review. Disabil Rehabil Assist Technol. 2019:1–7. pmid:31248292
- 24. Page MJ, McKenzie JE, Bossuyt PM, Boutron I, Hoffmann TC, Mulrow CD, et al. The PRISMA 2020 statement: an updated guideline for reporting systematic reviews. BMJ. 2021;372:n71. pmid:33782057
- 25. Hartling L, Bond K, Harvey K, Santaguida PL, Viswanathan M, Dryden DM. Developing and testing a tool for the classification of study designs in systematic reviews of interventions and exposures. 2010.
- 26. Antoine L, Nathan D, Laure M, Briac C, Jean-François M, Corinne B. Compliance with night-time overcorrection bracing in adolescent idiopathic scoliosis: Result from a cohort follow-up. Medical engineering & physics. 2020;77:137–41. pmid:31992499
- 27. Asher M, Lai SM, Burton D, Manna B. The reliability and concurrent validity of the scoliosis research society-22 patient questionnaire for idiopathic scoliosis. Spine. 2003;28(1):63–9. pmid:12544958
- 28. Vasiliadis E, Grivas TB, Gkoltsiou K. Development and preliminary validation of Brace Questionnaire (BrQ): a new instrument for measuring quality of life of brace treated scoliotics. Scoliosis. 2006;1(1):7. pmid:16759366
- 29.
Higgins JPT S J, Page MJ, Elbers RG, Sterne JAC. Chapter 8: Assessing risk of bias in a randomized trial. In: Higgins JPT T J, Chandler J, Cumpston M, Li T, Page MJ, Welch VA editor. Cochrane Handbook for Systematic Reviews of Interventions version 60 (updated July 2019): Cochrane; 2019.
- 30. Sterne JA, Hernán MA, Reeves BC, Savović J, Berkman ND, Viswanathan M, et al. ROBINS-I: a tool for assessing risk of bias in non-randomised studies of interventions. 2016;355:i4919.
- 31.
Sterne JAC H M, McAleenan A, Reeves BC, Higgins JPT. Chapter 25: Assessing risk of bias in a non-randomized study. In: Higgins JPT T J, Chandler J, Cumpston M, Li T, Page MJ, Welch VA, editor. Cochrane Handbook for Systematic Reviews of Interventions version 60 (updated July 2019): Cochrane; 2019.
- 32.
Schünemann HJ HJ, Vist GE, Glasziou P, Akl EA, Skoetz N, Guyatt GH. Chapter 14: Completing ‘Summary of findings’ tables and grading the certainty of the evidence. In: Higgins JPT T J, Chandler J, Cumpston M, Li T, Page MJ, Welch VA editor. Cochrane Handbook for Systematic Reviews of Interventions version 60 (updated July 2019): Cochrane; 2019.
- 33. Furlan AD, Malmivaara A, Chou R, Maher CG, Deyo RA, Schoene M, et al. 2015 Updated Method Guideline for Systematic Reviews in the Cochrane Back and Neck Group. Spine. 2015;40(21):1660–73. pmid:26208232
- 34. Higgins JP, Li T, Deeks JJ. Choosing effect measures and computing estimates of effect. Cochrane Handbook for Systematic Reviews of Interventions. 2019:143–76.
- 35. Miller DJ, Franzone JM, Matsumoto H, Gomez JA, Avendaño J, Hyman JE, et al. Electronic monitoring improves brace-wearing compliance in patients with adolescent idiopathic scoliosis: a randomized clinical trial. Spine. 2012;37(9):717–21. pmid:22517480
- 36. Karol LA, Virostek D, Felton K, Wheeler L. Effect of Compliance Counseling on Brace Use and Success in Patients with Adolescent Idiopathic Scoliosis. J Bone Joint Surg Am. 2016;98(1).
- 37. Al-Aubaidi ZT, Tropp H, Pedersen NW, Jespersen SM. Comparison of in-and outpatients protocols for providence night time only bracing in AIS patients–compliance and satisfaction. Scoliosis. 2013;8(1):6. pmid:23587285
- 38. Tavernaro M, Pellegrini A, Tessadri F, Zaina F, Zonta A, Negrini S. Team care to cure adolescents with braces (avoiding low quality of life, pain and bad compliance): a case–control retrospective study. 2011 SOSORT Award winner. Scoliosis. 2012;7(1):17. pmid:22995590
- 39. Negrini A, Donzelli S, Lusini M, Minnella S, Zaina F, Negrini S. A cognitive behavioral approach allows improving brace wearing compliance: an observational controlled retrospective study with thermobrace. Scoliosis. 2014;9(S1):O79.
- 40. Lin Y, Lou E, Lam TP, Cheng JC-Y, Sin SW, Kwok WK, et al. The Intelligent Automated Pressure-Adjustable Orthosis for Patients With Adolescent Idiopathic Scoliosis: A Bi-Center Randomized Controlled Trial. Spine. 2020;45(20):1395–402. pmid:32453223
- 41. Karol LA, Virostek D, Felton K, Jo C, Butler L. The Effect of the Risser Stage on Bracing Outcome in Adolescent Idiopathic Scoliosis. J Bone Joint Surg Am. 2016;98(15):1253–9. pmid:27489315
- 42. Feise RJ, Donaldson S, Crowther ER, Menke JM, Wright JG. Construction and validation of the scoliosis quality of life index in adolescent idiopathic scoliosis. Spine. 2005;30(11):1310–5. pmid:15928558
- 43. Miremberg H, Ben-Ari T, Betzer T, Raphaeli H, Gasnier R, Barda G, et al. The impact of a daily smartphone-based feedback system among women with gestational diabetes on compliance, glycemic control, satisfaction, and pregnancy outcome: a randomized controlled trial. Am J Obstet Gynecol. 2018;218(4):453.e1–.e7.
- 44. Christensen A, Osterberg LG, Hansen EH. Electronic monitoring of patient adherence to oral antihypertensive medical treatment: a systematic review. Journal of hypertension. 2009;27(8):1540–51. pmid:19474761
- 45. Sabin LL, DeSilva MB, Hamer DH, Xu K, Zhang J, Li T, et al. Using electronic drug monitor feedback to improve adherence to antiretroviral therapy among HIV-positive patients in China. AIDS and behavior. 2010;14(3):580–9. pmid:19771504
- 46. Henriksson J, Tydén G, Höijer J, Wadström J. A Prospective Randomized Trial on the Effect of Using an Electronic Monitoring Drug Dispensing Device to Improve Adherence and Compliance. Transplantation. 2016;100(1):203–9. pmid:26588006
- 47. Robiner WN, Flaherty N, Fossum TA, Nevins TE. Desirability and feasibility of wireless electronic monitoring of medications in clinical trials. Translational behavioral medicine. 2015;5(3):285–93. pmid:26327934
- 48. Demonceau J, Ruppar T, Kristanto P, Hughes DA, Fargher E, Kardas P, et al. Identification and assessment of adherence-enhancing interventions in studies assessing medication adherence through electronically compiled drug dosing histories: a systematic literature review and meta-analysis. Drugs. 2013;73(6):545–62. pmid:23588595
- 49. Dimatteo MR. The role of effective communication with children and their families in fostering adherence to pediatric regimens. Patient Educ Couns. 2004;55(3):339–44. pmid:15582339
- 50. Horberg MA, Hurley LB, Towner WJ, Allerton MW, Tang BT-T, Catz SL, et al. Determination of optimized multidisciplinary care team for maximal antiretroviral therapy adherence. Journal of acquired immune deficiency syndromes (1999). 2012;60(2):183. pmid:22293551
- 51. Zolnierek KBH, DiMatteo MR. Physician communication and patient adherence to treatment: a meta-analysis. Medical care. 2009;47(8):826. pmid:19584762
- 52. Tones M, Moss N, Polly DW Jr. A review of quality of life and psychosocial issues in scoliosis. Spine. 2006;31(26):3027–38. pmid:17173000
- 53. Schwieger T, Campo S, Weinstein SL, Dolan LA, Ashida S, Steuber KR. Body image and quality of life and brace wear adherence in females with adolescent idiopathic scoliosis. Journal of pediatric orthopedics. 2017;37(8):e519. pmid:26886460
- 54. Khan A, Tansel A, White DL, Kayani WT, Bano S, Lindsay J, et al. Efficacy of psychosocial interventions in inducing and maintaining alcohol abstinence in patients with chronic liver disease: a systematic review. 2016;14(2):191–202. e4.
- 55. MacDonald L, Chapman S, Syrett M, Bowskill R, Horne RJJoAD. Improving medication adherence in bipolar disorder: A systematic review and meta-analysis of 30 years of intervention trials. 2016;194:202–21.
- 56. Jones CJ, Smith H, Llewellyn C. Evaluating the effectiveness of health belief model interventions in improving adherence: a systematic review. Health psychology review. 2014;8(3):253–69. pmid:25053213
- 57. Jones CJ, Smith HE, Llewellyn CD. A systematic review of the effectiveness of interventions using the Common Sense Self-Regulatory Model to improve adherence behaviours. Journal of Health Psychology. 2016;21(11):2709–24. pmid:25957228
- 58. Takemitsu M, Bowen JR, Rahman T, Glutting JJ, Scott CB. Compliance monitoring of brace treatment for patients with idiopathic scoliosis. Spine (Phila Pa 1976). 2004;29(18):2070–4; discussion 4. pmid:15371711
- 59. Yrjönen T, Ylikoski M, Schlenzka D, Poussa M. Results of brace treatment of adolescent idiopathic scoliosis in boys compared with girls: a retrospective study of 102 patients treated with the Boston brace. Eur Spine J. 2007;16(3):393–7. pmid:16909249
- 60. Karol LA, Wingfield JJ, Virostek D, Felton K, Jo C. The Influence of Body Habitus on Documented Brace Wear and Progression in Adolescents With Idiopathic Scoliosis. Journal of pediatric orthopedics. 2020;40(3):e171–e5. pmid:31259783
- 61. Law D, Cheung M-C, Yip J, Yick K-L, Wong C. Scoliosis brace design: influence of visual aesthetics on user acceptance and compliance. Ergonomics. 2017;60(6):876–86. pmid:27547883
- 62. Korovessis P, Syrimpeis V, Tsekouras V, Vardakastanis K, Fennema P. Effect of the Chêneau Brace in the Natural History of Moderate Adolescent Idiopathic Scoliosis in Girls: Cohort Analysis of a Selected Homogenous Population of 100 Consecutive Skeletally Immature Patients. Spine Deform. 2018;6(5):514–22. pmid:30122386
- 63. Banerjee P, Ghosh S. On the estimation of the incidence and prevalence in two-phase longitudinal sampling design. Biostatistics. 2020;21(2):202–18. pmid:30165583
- 64. Negrini S, Minozzi S, Bettany-Saltikov J, Chockalingam N, Grivas TB, Kotwicki T, et al. Braces for idiopathic scoliosis in adolescents. Cochrane Database Syst Rev. 2015(6):CD006850. pmid:26086959
- 65. Nicholson GP, Ferguson-Pell MW, Smith K, Edgar M, Morley T. The objective measurement of spinal orthosis use for the treatment of adolescent idiopathic scoliosis. Spine (Phila Pa 1976). 2003;28(19):2243–50; discussion 50–1. pmid:14520038
- 66. Helfenstein A, Lankes M, Ohlert K, Varoga D, Hahne HJ, Ulrich HW, et al. The objective determination of compliance in treatment of adolescent idiopathic scoliosis with spinal orthoses. Spine (Phila Pa 1976). 2006;31(3):339–44. pmid:16449908
- 67. Castro FP Jr. Adolescent idiopathic scoliosis, bracing, and the Hueter-Volkmann principle. The Spine Journal. 2003;3(3):180–5. pmid:14589197
- 68. Smania N, Picelli A, Romano M, Negrini S. Neurophysiological basis of rehabilitation of adolescent idiopathic scoliosis. Disability and Rehabilitation. 2008;30(10):763–71. pmid:18432434
- 69. Rowe DE, Bernstein SM, Riddick MF, Adler F, Emans JB, Gardner-Bonneau D. A meta-analysis of the efficacy of non-operative treatments for idiopathic scoliosis. J Bone Joint Surg Am. 1997;79(5):664–74. pmid:9160938
- 70. Brodaty H, Low L-F, Gibson L, Burns K. What is the best dementia screening instrument for general practitioners to use? The American Journal of Geriatric Psychiatry. 2006;14(5):391–400. pmid:16670243
- 71. Guo J, Lam TP, Wong MS, Ng BKW, Lee KM, Liu KL, et al. A prospective randomized controlled study on the treatment outcome of SpineCor brace versus rigid brace for adolescent idiopathic scoliosis with follow-up according to the SRS standardized criteria. European spine journal. 2014;23(12):2650–7. pmid:24378629
- 72. Rivett L, Rothberg A, Stewart A, Berkowitz R. The relationship between quality of life and compliance to a brace protocol in adolescents with idiopathic scoliosis: a comparative study. BMC Musculoskelet Disord. 2009;10:5. pmid:19144157
- 73. Gorenoi V, Schönermark MP, Hagen A. Interventions for enhancing medication compliance/adherence with benefits in treatment outcomes. GMS health technology assessment. 2007;3.
- 74. Brok J, Thorlund K, Gluud C, Wetterslev J. Trial sequential analysis reveals insufficient information size and potentially false positive results in many meta-analyses. Journal of clinical epidemiology. 2008;61(8):763–9. pmid:18411040
- 75. Pocock SJ, Assmann SE, Enos LE, Kasten LE. Subgroup analysis, covariate adjustment and baseline comparisons in clinical trial reporting: current practiceand problems. Statistics in medicine. 2002;21(19):2917–30. pmid:12325108
- 76. Conn VS, Ruppar TM. Medication adherence outcomes of 771 intervention trials: Systematic review and meta-analysis. Prev Med. 2017;99:269–76. pmid:28315760
- 77. Dean SE, Povey RC, Reeves J. Assessing interventions to increase compliance to patching treatment in children with amblyopia: a systematic review and meta-analysis. British Journal of Ophthalmology. 2016;100(2):159–65. pmid:26614629
- 78. Schreiber S, Parent EC, Khodayari Moez E, Hedden DM, Hill DL, Moreau M, et al. Schroth Physiotherapeutic Scoliosis-Specific Exercises Added to the Standard of Care Lead to Better Cobb Angle Outcomes in Adolescents with Idiopathic Scoliosis—an Assessor and Statistician Blinded Randomized Controlled Trial. PloS one. 2016;11(12):e0168746. pmid:28033399
- 79. Vickers AJ, Altman DG. Analysing controlled trials with baseline and follow up measurements. BMJ. 2001;323(7321):1123–4. pmid:11701584
- 80. Vickers AJ. The use of percentage change from baseline as an outcome in a controlled trial is statistically inefficient: a simulation study. BMC medical research methodology. 2001;1(1):1–4.