Improving osteoarthritis care by digital means - Effects of a digital self-management program after 24- or 48-weeks of treatment

Leif E Dahlberg; Andrea Dell'Isola; L Stefan Lohmander; Håkan Nero

doi:10.1371/journal.pone.0229783

. 2020 Mar 4;15(3):e0229783. doi: 10.1371/journal.pone.0229783

Improving osteoarthritis care by digital means - Effects of a digital self-management program after 24- or 48-weeks of treatment

Leif E Dahlberg ^1,², Andrea Dell'Isola ¹, L Stefan Lohmander ^1,², Håkan Nero ^1,^2,^*

Editor: Kelly Naugle³

PMCID: PMC7056265 PMID: 32130276

Abstract

Background

Osteoarthritis (OA) is highly prevalent in older adults and a growing cause of disability. Easily accessible first-line treatment of OA is increasingly important. Digital self-management programs have in recent years become available. Evidence of short-term effects of such programs are abundant, yet reports on long-term benefits and adherence to treatment are scarce. The current study’s objective was to investigate the long-term pain and function outcomes of people with hip or knee OA participating in a digital self-management programme.

Methods and findings

In this longitudinal cohort study, individuals with hip and knee OA, from the register of a digital self-management program and with 0-24-week (n = 499) or 0-48-week adherence (n = 138), were included. The treatment effect in terms of monthly pain (NRS, 0–10 worst to best) and physical function (30-second chair stand test (30CST), number of repetitions) change were investigated using a mixed model, controlling for the effect of age, body mass index (BMI), gender and index joint. For the 24-week sub-sample, pain NRS decreased monthly by -0.43 units (95% CI -0.51, -0.35, mean knee pain from 5.6 to 3.1, and hip pain from 5.9 to 3.8) and 30CST repetitions increased monthly by 0.76 repetitions (95% CI 0.64, 0.89 mean for knee from 10.0 to 14.3, and for hip from 10.9 to 14.8). For the 48-week sub-sample, pain decreased monthly by -0.39 units (95% CI -0.43, -0.36, mean knee pain from 5.7 to 3.2, and hip pain from 5.8 to 3.8), and repetitions increased by 0.72 repetitions (95% CI 0.65, 0.79, mean repetitions for knee from 10.3 to 14.4, and for hip from 11.1 to 14.9). There were no clinically relevant effects on the improvement of pain or function by any covariate (age, sex, index joint). The lack of a control group and randomization limit our ability to explain the mechanisms of the observed results.

Conclusions

Continuously participating in a digital OA treatment program for 6 or 12 months was associated with a clinically important decrease in joint pain and increased physical function, in hip and knee OA. Future research should follow OA-related outcomes in participants that end their treatment to explore when and why that decision was made.

Introduction

Osteoarthritis (OA) is the most common joint disease and one of the main causes of musculoskeletal disability worldwide [1]. More than 10% of men and 18% of women aged older than 60 suffer from symptomatic OA [2]. Driven by the increasing longevity of the population combined with the rise of obesity, joint injuries, and physical inactivity, OA is now the fastest growing cause of disability worldwide [3–5].

National and international guidelines recommend education and exercise as first-line treatment for OA due to the effectiveness in reducing pain and disability [6–12]. However, reports show a low uptake of these recommendations with only 36% of patients receiving a joint replacement having been provided a first-line intervention, and less than 50% receiving appropriate care for their OA [13, 14]. Without immediate action, the rising OA prevalence will be a challenge without solution for the health care systems and for society worldwide [7, 15].

To tackle the increasing burden of OA and increase uptake of recommended first-line treatment, self-management programmes have been initiated in several countries [16–18]. Despite their success in improving patients’ symptoms, the high prevalence of OA and difficulties related to their implementation, these programmes reach only a minority of the suffering OA population that would benefit from education and exercise [17, 19, 20].

Digital self-management programmes have been developed to further facilitate access to first-line treatment for OA and to aid patients in maintaining a long-term exercise regime [21, 22]. Early reports have shown a reduction in pain, disability and desire for surgical treatment in patients with OA after six weeks in a digital self-management programme [23, 24]. However, there is a dearth of knowledge surrounding the long-term effectiveness of such programmes, or adherence and effectiveness of the face-to-face counterparts [18, 19]. Hence, the main objective of the current study was to report on the long-term outcomes (24 and 48 weeks) of people with hip or knee OA participating in a digital self-management programme delivering first-line OA management, and statistically investigate the mean treatment effect of duration on pain and physical function, as well as differences in pain over time between hip and knee OA with or without additional covariates.

Methods

This was an observational longitudinal cohort study approved by the regional ethics committee of Lund University and the Swedish Ethical Review Authority (Dnr: 2018/650 and 2019–02232). Written informed consent from participants was obtained during registration. The study adheres to the STROBE guidelines for observational studies [25] (S1 Checklist).

Setting and participants

Participants joined the digital OA self-management and education programme (see Intervention below for details), through recommendation by their local orthopaedic surgeon or physiotherapist, and via online advertisements and campaigns placed on search engines and social networks. Included participants had a radiographic and or clinical diagnosis of hip or knee OA from a physical therapist or physician (95% of all patients at the date of data extraction). Individuals without a prior diagnosis had clinical OA confirmed by an orthopaedic surgeon or physiotherapist via telephone (diagnosis according to NICE criteria and Swedish National Guidelines, and confirming the absence of any red flag symptoms), or if deemed necessary were recommended to seek face-to-face care before inclusion in the programme.

Data was extracted from the digital self-management programme registry on the 13th of March 2019. At that time point, the register contained data from 1709 Swedish participants (710 individuals had started ≥48 weeks before data extraction) that had reported one of their knees or hips as their most symptomatic joint (index joint), had been treated in the programme for at least three weeks with a minimum adherence of 70% and had registered ≥24 weeks before data extraction. Adherence was defined as the percentage of completed activities (exercises, text or video lessons on OA, and quizzes on lesson material) per the pre-defined period (the cut-off of 70% for the initial three weeks represents about 5 out of 7 days per week performing recommended activities), and mean adherence was defined as the group mean for the period of interest. Outcome analysis was made in two separate sub-samples; participants with a pain report at start and at week 24 or adjacent week (+- 4 weeks) and at start and at week 48 or adjacent week (+- 6 weeks), respectively. Adjacent weeks were added to collect the maximum amount of data (around 50% of active participants had reported pain at week 24 and 48, respectively). Hence, if a pain report was missing at week 24 or 48, the report from the closest week available was used (if two pain reports were available at the two most adjacent weeks, e.g. week 25 and 23, mean pain for these two was used). Those included in the 48-week sub-sample were excluded from the 24-week sub-sample, to enable comparison.

Intervention

The intervention consisted of a digital, structured and individualized treatment programme for people with hip or knee OA (Joint Academy®; www.jointacademy.com). The programme consists of instructions for neuromuscular exercises appropriately adjusted to each patient in regard to degrees of complexity and difficulty. Exercises are distributed daily during the whole participation period, in general, two per day. While rating perceived difficulty and adding comments, patients also indicated when exercises were completed. Program continuation was halted until all exercises for the day were marked as completed. Information (based on current OA management guidelines and research) in the form of text or video lessons (with quizzes on the material after each episode) on subjects related to OA, OA symptoms and its management is also distributed to each participant. The lessons come packed in themes, with each theme containing 1–5 lessons where participants receive a theme per week the first six weeks, and then every other week, for a total of 70 lessons over a 48-week period. Completion of a lesson was indicated by the patient answering the quiz correctly. Additionally, continuous access to and dialogue with a physiotherapist through an encrypted chat function, and/or telephone, is provided.

Outcome measures

In this program, several measures are collected at separate time points. There are weekly measures (joint pain), bi-weekly measures (physical function), and quarterly measures (other instruments not relevant for this study). Joint pain was assessed at baseline, and weeks 12, 24 and 48 using the Numerical Rating Scale (NRS, discrete boxes 0–10) with the instruction [26]; Mark on this scale how much pain you had the last week in your hip/knee, followed by a 0–10 scale where 0 was defined as No pain and 10 was defined as Maximum pain. Minimal clinically important change (MCIC) of pain was defined as an improvement of 20% (slightly or moderately important improvement according to Tubach et al., 2012) [27]. As a measure of physical function, the 30CST from week 12, 24 and 48 were used [28], performed by the participant with the help of an instruction video with a coupled visual timer. The patient entered the performed number of repetitions after each test. Physical function data was handled similarly to the NRS, with week 24 ±4 adjacent weeks and week 48±6 adjacent weeks included for those with available NRS data. All outcomes were self-assessed and self-entered using the digital programme interface and chosen based on the International Consortium for Health Outcomes Measurement Standard Set for Hip & Knee Osteoarthritis (ICHOM) [29]. Data on the participants’ overall health and characteristics (age, gender, BMI), as well as OA-related factors (most painful joint, afflicted side), were collected at inclusion.

Statistical analysis

Summary data are described by the mean value, standard deviation and number of observations or the number and percent of the categories of interest. Comparisons of baseline data between the 24-week sub-sample and excluded participants (with missing data due to ending treatment or not reporting pain) were performed using independent samples t-test and the Fisher’s Exact test (for dichotomous variables). The group-specific mean treatment effect of duration on pain and repetitions (30CST—physical function), as well as differences in pain over time between hip and knee OA with or without additional covariates, were estimated and tested using random slopes and intercepts models. Pain development over time was plotted for hip and knee OA, respectively.

To describe patients adhering for six months with contrasting pain severity at baseline, participants were divided into same-sized tertiles based on reported baseline pain, and mean pain per time point was calculated and plotted for each group (not performed for the 48-week sub-sample due to small numbers per group).

Significance level was set to p<0.05, and p-values and 95% confidence intervals were reported when applicable. Statistical calculations were performed in SPSS Version 25 (IBM Corporation, New York, USA) and Stata 15.1 (StataCorp LLC, Texas, USA).

Role of the funding source

The funding source had no role in any part of this study. The in-kind support sponsor, Arthro Therapeutics, aided in the collection and extraction of data from the registry, otherwise was not in any way involved in design, analysis or interpretation of data, writing of the text or submitting the paper for publication.

Results

After identifying active users at week 24, a total of 920 individuals were found, whereof 290 individuals had not reported their pain into the register at or around week 24. Excluding those with 48-week data, 499 individuals reporting pain were included in the 24-week sub-sample. For the 48-week sub-sample, a total of 138 individuals (n = 7 missing 24-week data included) with pain data at 48 weeks were included (Fig 1).

24-week sub-sample

There were no differences between the 24-week sub-sample (n = 499) and excluded participants (combining those ending treatment, not reporting pain at 24 weeks or excluded due to having reported pain at 48 weeks also, n = 1210) in regard to age, BMI, baseline pain, baseline physical function, or the distribution of gender and index joint (for details, please see S1 Table).

Percentage of those reaching MCIC in pain after 24 weeks was 72% while mean adherence (SD) was 75% (18%). Descriptive data separated by index joint is reported in Table 1. Pain change over time per index joint showed continuous pain relief over 6 months of adherence for both joints (Fig 2). Visual inspection of plotted mean pain per pain severity group suggested a difference in absolute change, yet similar pain relief in terms of relative change for both hip and knee (Fig 3).

Table 1. Descriptive data for knee and hip OA in the 24-week sub-sample (n = 499).

Characteristic	Knee (n = 301)	Hip (n = 198)
Age, mean (SD)	64 (9)	63 (9)
Female, n (%)	220 (73)	152 (77)
BMI, mean (SD)	28.2 (4.8)	26.5 (4.9)
Weight status, n (%)
• Underweight (BMI <18.5)	1 (0)	1 (1)
• Normal weight (BMI 18.5–24.9)	67 (22)	83 (42)
• Overweight (BMI 25–29.9)	141 (47)	79 (40)
• Obese (BMI ≥30)	92 (31)	35 (18)
Baseline pain per severity group
• Low, mean (SD)	3.3 (1.3)	4.1 (0.9)
• Moderate, mean (SD)	5.6 (0.5)	6.4 (0.5)
• Severe, mean (SD)	7.6 (0.8)	7.7 (0.8)

Open in a new tab

BMI = body mass index. NRS = Numeric rating scale. OA = osteoarthritis.

Fig 2 — Symbols show mean pain value at 0, 12 and 24 weeks for patient subgroups with index joint knee (n = 301) or hip (n = 198). Error bars represent 95% confidence intervals.

Fig 3 — Symbols show mean pain value at 0, 12 and 24 weeks for patient subgroups with index joint knee (n for tertiles low 100, mid 99, high 102) or hip (n for tertiles each 66). Error bars represent 95% confidence intervals.

48-week sub-sample

Percentage of those in the 48-week sub-sample reaching MCIC in pain was 67% while mean adherence (SD) was 74% (21%). Descriptive data separated by index joint is reported in Table 2. Plotted pain change over time per index joint showed maximum pain relief achieved at around 6 months, and no signs of worsening pain for either joint, for up to 12 months (Fig 4).

Table 2. Descriptive data for knee and hip OA in the 48-week sub-sample (n = 138).

Characteristic	Knee (n = 78)	Hip (n = 60)
Age, mean (SD)	65 (9)	64 (8)
Female, n (%)	54 (69)	42 (70)
BMI, mean (SD)	27.0 (4.4)	26.0 (3.8)
Weight status, n (%)
• Underweight (BMI <18.5)	0 (0)	0 (0)
• Normal weight (BMI 18.5–24.9)	29 (37)	30 (50)
• Overweight (BMI 25–29.9)	28 (36)	17 (28)
• Obese (BMI ≥30)	21 (27)	13 (22)
Baseline pain per severity group
• Low, mean (SD)	2.7 (1.0)	3.8 (1.1)
• Moderate, mean (SD)	5.5 (0.7)	6.5 (0.7)
• Severe, mean (SD)	7.6 (0.7)	8.4 (0.7)

Open in a new tab

BMI = body mass index. NRS = Numeric rating scale. OA = osteoarthritis.

Fig 4 — Symbols show mean pain value at 0, 12, 24 and 48 weeks for patient subgroups with index joint knee (n = 78) or hip (n = 60). Error bars represent 95% confidence intervals.

Treatment effect over time

The treatment effect was modelled in terms of monthly pain change and monthly change in repetitions as a function of treatment duration for 24- and 48-week sub-samples using a mixed model with random slopes and intercepts. The models yielded marginally different estimates for pain with a decrease of -0.43 units (95% CI -0.51, -0.35) and -0.39 units (95% CI -0.43, -0.36) per month for the 24- and 48-week sub-sample, respectively. Similarly, physical function increased by 0.76 repetitions (95% CI 0.64, 0.89) and 0.72 repetitions (95% CI 0.65, 0.79) per month, for the 24- and 48-week sub-sample respectively. The group difference (comparing 24- and 48-week sub-samples) in pain change did not reach statistical significance.

BMI, index joint or gender did not importantly influence pain or physical function over time. Pain improvement was not affected by age, yet with increasing age, there was a statistically significant but not clinically relevant decrease in the improvement in physical function (-0.003 per year; 95% CI -0.005, -0.002; p<0.01). Mean and median values of pain and physical function, for each time point and per index joint and sub-sample, are reported in Table 3.

Table 3. Pain and physical function at baseline and follow-up.

Time	Mean (95% CI)	Median (IQR)	Mean (95% CI)	Median (IQR)
24-week sub-sample (n = 499)
	Knee OA (n = 301)		Hip OA (n = 198)
Pain
Baseline	5.6 (5.4–5.8)	6 (2)	5.9 (5.7–6.2)	6 (2)
Week 12	3.4 (3.2–3.7)	3 (3)	3.9 (3.7–4.3)	4 (4)
Week 24	3.1 (2.9–3.4)	3 (3)	3.8 (3.4–4.1)	3 (3)
Physical function
Baseline	10.0 (9.6–10.4)	10 (4)	10.9 (10.4–11.5)	10 (3)
Week 12	13.7 (13.2–14.2)	13 (6)	14.2 (13.4–14.9)	13 (6)
Week 24	14.3 (13.7–14.8)	14 (6)	14.8 (13.9–15.5)	14 (7)
48-week sub-sample (n = 138)
	Knee OA (n = 78)		Hip OA (n = 60)
Pain
Baseline	5.7 (5.2–6.1)	6 (3)	5.8 (5.2–6.3)	6 (3)
Week 12	3.2 (2.8–3.7)	3 (3)	3.9 (3.3–4.4)	4 (4)
Week 24	2.9 (2.4–3.5)	2 (3)	3.4 (2.8–3.9)	3 (3)
Week 48	3.2 (2.7–3.8)	3 (4)	3.8 (3.1–4.4)	4 (4)
Physical function
Baseline	10.3 (9.6–10.9)	10 (4)	11.1 (10.2–12.1)	11 (4)
Week 12	13.7 (12.8–14.6)	14 (5)	14.6 (13.5–15.7)	14 (6)
Week 24	15.1 (13.9–16.3)	15 (6)	15.4 (14.1–16.7)	15 (7)
Week 48	14.4 (13.1–15.7)	14 (6)	14.9 (13.7–16.2)	15 (5)

Open in a new tab

^aMeasured using the Numerical Rating Scale (NRS 0–10).

Discussion

This is, to the best of our knowledge, the first study investigating the long-term benefits of patients adhering to a digital OA self-management programme. Results in two groups of 499 and 138 individuals being adherent to treatment for about 5 out of 7 days per week for 24 or 48 weeks showed a substantial reduction in the level of pain and an increase in physical performance per month of treatment. Based on available group-level data, there were no signs of worsening of symptoms during participation. Around 70% of those undertaking the programme reached a clinically relevant pain reduction at both follow-ups, and could thus be characterized as responders to treatment, with no clear difference between people with knee or hip OA. This result suggests that taking part in and adhering to a digital self-management programme is beneficial for reducing pain intensity and increasing physical function for older adults with knee or hip OA, and that results are maintained for up to one year.

In the present study, we observed greater improvements in both pain and physical function when compared to results from systematic reviews summarizing studies on people with knee and hip OA undergoing first-line interventions, where a decrease in pain of 6 points out of 100, at six months, was reported for knee OA [6, 7]. Yet these results are not directly comparable since the meta-analysis reported results several months after the close of active treatment, while the current study reports on a continuous intervention, but also due to potential differences in study population and intervention delivery.

The reduction in pain and increase in physical function in people with OA participating in first-line interventions has been shown to decline over time [6, 7, 18], probably due to the limited treatment duration (usually between 8 and 12 sessions over 8 weeks). Available evidence suggests that undertaking exercise for longer periods is associated with better outcomes [6, 30], regardless of the duration of a single session, potentially explaining the larger improvement experienced by people undergoing the present digital programme [31, 32].

The results of this study are comparable to, or somewhat better than shown in previous studies analysing the effect of digital self-management programmes. Bossen et al. showed a 2-point reduction on the WOMAC pain subscale (equivalent to a 10-point reduction on a 0–100 scale) after 8 months of providing a fully automated web-based programme aiming to increase patients’ physical activity (n = 199) [21]. The intervention consisted of a 9-week gradually increasing program. The presence of targeted joint-specific exercises in the present digital self-management programme performed 5–10 minutes daily, combined with the inclusion of human supervision with continuous follow-up of goals and outcomes, may explain the greater pain reduction showed in the current study. Another self-management programme including education and exercise delivered digitally, showed a similar pain reduction after 6 months in comparison to the current study, although results were based on a small sample (n = 41), and the program ended after 12 weeks [22].

Participants in the present digital self-management programme with more severe pain experienced larger absolute pain reductions compared to those with less severe pain, yet the relative reduction was comparable. This suggests that the programme has the potential to yield similar benefit regardless of pain experienced by the participant at baseline. Hence it may be argued that the selection of patients for an exercise-based intervention should not be based solely on pain, and confirms that exercise is effective in reducing pain regardless of symptom severity [12].

Results from some exercise-based OA interventions suggest that knee OA patients experience a larger pain reduction, in comparison to those suffering from hip OA [18, 19]. We were unable to confirm this differential response to exercise of the knee and hip in the present study. Differences in participant’ characteristics (e.g. age, disease severity), outcomes used, treatment delivery and exercise dose may explain these different study results.

Some limitations of our study need to be addressed. The lack of a control group and randomization limit our ability to explain the mechanisms of the observed reduction in pain and increased physical function in these OA patients, or to claim a direct cause and effect relationship. However, previous systematic reviews and meta-analyses of controlled trials provide evidence of a cause and effect relationship between exercise and patient benefits [33, 34].

In the current study pain and physical function were both shown to improve over time. Furthermore, patients of all pain levels at baseline improved (high variability in baseline pain was found). Even so, some contribution to outcomes by context effects and regression to the mean would be expected, although the magnitude is uncertain. Only a randomized controlled trial can provide a specific answer to the degree of regression to the mean.

Since results are based on a register of patients voluntarily choosing whether to report their outcomes and when to end treatment, reflecting clinical reality, some data is missing. Although the results suggest that those ending treatment do not importantly differ from included participants in terms of descriptive factors at baseline, for future studies it would be of value to interview and follow those ending treatment, and their OA-related outcomes. Specifically, it would be valuable due to the current program being continuous and having no defined ending. The participant sample was drawn from a register and inclusion criteria comprised of a hip or knee OA diagnosis and having initiated treatment with a 70% adherence for the initial weeks, supporting generalisability. Hence, the sample should reflect the population to a greater extent than randomized controlled trials that commonly have more stringent inclusion criteria. Adding on, the 30 CST was instructed using a video and the patient thus performed the test accordingly without the supervision of a physiotherapist, hence the result from the measure should be interpreted with caution when compared to other studies. Finally, we could not control for other treatments undertaken by the participants during the study period, therefore use of concurrent non-pharmacological or pharmacological treatments may have influenced the results.

The challenges and barriers of delivering exercise and education based self-management programmes to the growing OA population are substantial. Considering the positive results showed in this and previous studies, digital interventions may represent a viable alternative for patients without access to or not interested in participating in traditional face to face programmes. Digital interventions such as the present one may also complement traditional programmes to enhance long-term adherence to treatment. Yet further investigation into specific components of digital interventions outside of exercise is warranted, to more clearly understand the mechanics and benefits of specific parts of digital OA treatment programs.

Supporting information

S1 Table. Comparative data for the 24-week sub-sample (n = 499) and excluded participants (n = 1210).

(PDF)

Click here for additional data file.^{(58KB, pdf)}

S1 Checklist. STROBE statement—checklist of items that should be included in reports of cohort studies.

(DOC)

Click here for additional data file.^{(122KB, doc)}

Acknowledgments

The authors would like to thank the participants of the study for contributing essential data. We express our gratitude to Vinnova, the Swedish Innovation Agency, for their financial support. Also, thanks to Jonas Ranstam for statistical guidance and advice, and to Daniel Kloek for graphical assistance.

Data Availability

Data from this registry contains potentially identifying and sensitive patient information and cannot be shared publicly. Anonymized data will be shared with researchers completing a data request with an approved research proposal and a data access agreement, signed by all parties. Contact data@jointacademy.com for more information.

Funding Statement

Funding was received by LED from Vinnova - Sweden’s Innovation Agency (grant number: 2016-04187, www.vinnova.se) and Stiftelsen för Bistånd åt Rörelsehindrade i Skåne (grant number: 2019-01-20, www.stiftbistandskane.se) to the Department of Clinical Sciences Lund, Orthopaedics, Lund University, Sweden. In kind support (data gathering and extraction) was provided by Arthro Therapeutics Inc. The funder provided support in the form of consulting fees for authors [LED, LSL, HN], but did not have any additional role in the study design, data collection and analysis, decision to publish, or preparation of the manuscript. The specific roles of these authors are articulated in the ‘author contributions’ section.

References

1.Murray CJL, Vos T, Lozano R, Naghavi M, Flaxman AD, Michaud C, et al. Disability-adjusted life years (DALYs) for 291 diseases and injuries in 21 regions, 1990–2010: a systematic analysis for the Global Burden of Disease Study 2010. The Lancet. 2012;380(9859):2197–223. 10.1016/s0140-6736(12)61689-4 [DOI] [PubMed] [Google Scholar]
2.Willett M, Duda J, Gautrey C, Fenton S, Greig C, Rushton A. Effectiveness of behavioural change techniques in physiotherapy interventions to promote physical activity adherence in patients with hip and knee osteoarthritis: a systematic review protocol. BMJ Open. 2017;7(6):e015833 Epub 2017/07/02. 10.1136/bmjopen-2017-015833 [DOI] [PMC free article] [PubMed] [Google Scholar]
3.Kingsbury SR, Gross HJ, Isherwood G, Conaghan PG. Osteoarthritis in Europe: impact on health status, work productivity and use of pharmacotherapies in five European countries. Rheumatology (Oxford). 2014;53(5):937–47. Epub 2014/02/04. 10.1093/rheumatology/ket463 . [DOI] [PubMed] [Google Scholar]
4.Turkiewicz A, Petersson IF, Bjork J, Hawker G, Dahlberg LE, Lohmander LS, et al. Current and future impact of osteoarthritis on health care: a population-based study with projections to year 2032. Osteoarthritis and cartilage / OARS, Osteoarthritis Research Society. 2014;22(11):1826–32. Epub 2014/08/02. 10.1016/j.joca.2014.07.015 . [DOI] [PubMed] [Google Scholar]
5.Wallace IJ, Worthington S, Felson DT, Jurmain RD, Wren KT, Maijanen H, et al. Knee osteoarthritis has doubled in prevalence since the mid-20th century. Proceedings of the National Academy of Sciences of the United States of America. 2017;114(35):9332–6. Epub 2017/08/16. 10.1073/pnas.1703856114 [DOI] [PMC free article] [PubMed] [Google Scholar]
6.Fransen M, McConnell S, Harmer AR, Van der Esch M, Simic M, Bennell KL. Exercise for osteoarthritis of the knee. In: Fransen M, editor. Chichester, UK: John Wiley & Sons, Ltd; 2015. [Google Scholar]
7.Fransen M, McConnell S, Hernandez-Molina G, Reichenbach S. Exercise for osteoarthritis of the hip. Cochrane Database Syst Rev. 2014;(4):CD007912 Epub 2014/04/24. 10.1002/14651858.CD007912.pub2 . [DOI] [PMC free article] [PubMed] [Google Scholar]
8.Fernandes L, Hagen KB, Bijlsma JW, Andreassen O, Christensen P, Conaghan PG, et al. EULAR recommendations for the non-pharmacological core management of hip and knee osteoarthritis. Ann Rheum Dis. 2013;72(7):1125–35. Epub 2013/04/19. 10.1136/annrheumdis-2012-202745 . [DOI] [PubMed] [Google Scholar]
9.Hochberg MC, Altman RD, April KT, Benkhalti M, Guyatt G, McGowan J, et al. American College of Rheumatology 2012 recommendations for the use of nonpharmacologic and pharmacologic therapies in osteoarthritis of the hand, hip, and knee. Arthritis Care Res (Hoboken). 2012;64(4):465–74. Epub 2012/05/09. 10.1002/acr.21596 . [DOI] [PubMed] [Google Scholar]
10.The Swedish National Board of Health and Welfare—National guidelines for orthopaedic conditions. Available from: http://www.socialstyrelsen.se/nationalguidelines/nationalguidelinesfororthopaedicconditions
11.Bannuru RR, Osani MC, Vaysbrot EE, Arden NK, Bennell K, Bierma-Zeinstra SMA, et al. OARSI guidelines for the non-surgical management of knee, hip, and polyarticular osteoarthritis. Osteoarthritis Cartilage. 2019. Epub 2019/07/07. 10.1016/j.joca.2019.06.011 . [DOI] [PubMed] [Google Scholar]
12.Juhl C, Christensen R, Roos EM, Zhang W, Lund H. Impact of exercise type and dose on pain and disability in knee osteoarthritis: a systematic review and meta-regression analysis of randomized controlled trials. Arthritis Rheumatol. 2014;66(3):622–36. Epub 2014/02/28. 10.1002/art.38290 . [DOI] [PubMed] [Google Scholar]
13.Basedow M, Esterman A. Assessing appropriateness of osteoarthritis care using quality indicators: A systematic review. Journal of Evaluation in Clinical Practice. 2015;21(5):782–9. 10.1111/jep.12402 [DOI] [PubMed] [Google Scholar]
14.Runciman WB, Hunt TD, Hannaford NA, Hibbert PD, Westbrook JI, Coiera EW, et al. CareTrack: assessing the appropriateness of health care delivery in Australia. Med J Aust. 2012;197(2):100–5. Epub 2012/07/17. 10.5694/mja12.10510 . [DOI] [PubMed] [Google Scholar]
15.Caneiro JP, O'Sullivan PB, Roos EM, Smith AJ, Choong P, Dowsey M, et al. Three steps to changing the narrative about knee osteoarthritis care: a call to action. British journal of sports medicine. 2019. Epub 2019/09/06. 10.1136/bjsports-2019-101328 . [DOI] [PubMed] [Google Scholar]
16.Allen KD, Choong PF, Davis AM, Dowsey MM, Dziedzic KS, Emery C, et al. Osteoarthritis: Models for appropriate care across the disease continuum. Best Practice and Research: Clinical Rheumatology. 2016;30(3):503–35. 10.1016/j.berh.2016.09.003 [DOI] [PubMed] [Google Scholar]
17.Thorstensson CA, Garellick G, Rystedt H, Dahlberg LE. Better Management of Patients with Osteoarthritis: Development and Nationwide Implementation of an Evidence-Based Supported Osteoarthritis Self-Management Programme. Musculoskeletal Care. 2015;13(2):67–75. Epub 2014/10/28. 10.1002/msc.1085 . [DOI] [PubMed] [Google Scholar]
18.Dell'Isola A, Jonsson T, Ranstam J, Dahlberg LE, Hansson EE. Education, home exercise and supervised exercise for people with hip and knee osteoarthritis as part of a nationwide implementation programme: data from the BOA registry. Arthritis Care Res (Hoboken). 2019. 10.1002/acr.24033 . [DOI] [PubMed] [Google Scholar]
19.Skou ST, Roos EM. Good Life with osteoArthritis in Denmark (GLA:D): evidence-based education and supervised neuromuscular exercise delivered by certified physiotherapists nationwide. BMC Musculoskelet Disord. 2017;18(1):72 Epub 2017/02/09. 10.1186/s12891-017-1439-y [DOI] [PMC free article] [PubMed] [Google Scholar]
20.Hurley MV, Walsh NE, Mitchell H, Nicholas J, Patel A. Long-term outcomes and costs of an integrated rehabilitation program for chronic knee pain: a pragmatic, cluster randomized, controlled trial. Arthritis Care Res (Hoboken). 2012;64(2):238–47. Epub 2011/09/29. 10.1002/acr.20642 . [DOI] [PubMed] [Google Scholar]
21.Bossen D, Veenhof C, Van Beek KE, Spreeuwenberg PM, Dekker J, De Bakker DH. Effectiveness of a web-based physical activity intervention in patients with knee and/or hip osteoarthritis: randomized controlled trial. J Med Internet Res. 2013;15(11):e257 Epub 2013/11/26. 10.2196/jmir.2662 [DOI] [PMC free article] [PubMed] [Google Scholar]
22.Smittenaar P, Erhart-Hledik JC, Kinsella R, Hunter S, Mecklenburg G, Perez D. Translating Comprehensive Conservative Care for Chronic Knee Pain Into a Digital Care Pathway: 12-Week and 6-Month Outcomes for the Hinge Health Program. JMIR Rehabil Assist Technol. 2017;4(1):e4 Epub 2017/06/06. 10.2196/rehab.7258 [DOI] [PMC free article] [PubMed] [Google Scholar]
23.Nero H, Dahlberg J, Dahlberg LE. A 6-Week Web-Based Osteoarthritis Treatment Program: Observational Quasi-Experimental Study. Journal of medical Internet research. 2017;19(12):e422 Epub 2017/12/20. 10.2196/jmir.9255 . [DOI] [PMC free article] [PubMed] [Google Scholar]
24.Cronstrom A, Nero H, Dahlberg LE. Factors associated with patients' willingness to consider joint surgery after completion of a digital osteoarthritis treatment program: A prospective cohort study. Arthritis Care Res (Hoboken). 2018. Epub 2018/10/10. 10.1002/acr.23772 . [DOI] [PMC free article] [PubMed] [Google Scholar]
25.Von Elm E, Altman DG, Egger M, Pocock SJ, Gøtzsche PC, Vandenbroucke JP. The Strengthening the Reporting of Observational Studies in Epidemiology (STROBE) statement: guidelines for reporting observational studies. Annals of internal medicine. 2007;147(8):573–7. 10.7326/0003-4819-147-8-200710160-00010 [DOI] [PubMed] [Google Scholar]
26.Williamson A, Hoggart B. Pain: a review of three commonly used pain rating scales. Journal of clinical nursing. 2005;14(7):798–804. Epub 2005/07/08. 10.1111/j.1365-2702.2005.01121.x . [DOI] [PubMed] [Google Scholar]
27.Tubach F, Ravaud P, Martin-Mola E, Awada H, Bellamy N, Bombardier C, et al. Minimum clinically important improvement and patient acceptable symptom state in pain and function in rheumatoid arthritis, ankylosing spondylitis, chronic back pain, hand osteoarthritis, and hip and knee osteoarthritis: Results from a prospective multinational study. Arthritis Care Res (Hoboken). 2012;64(11):1699–707. Epub 2012/06/08. 10.1002/acr.21747 . [DOI] [PubMed] [Google Scholar]
28.Dobson F, Hinman RS, Roos EM, Abbott JH, Stratford P, Davis AM, et al. OARSI recommended performance-based tests to assess physical function in people diagnosed with hip or knee osteoarthritis. Osteoarthritis and cartilage / OARS, Osteoarthritis Research Society. 2013;21(8):1042–52. Epub 2013/05/18. 10.1016/j.joca.2013.05.002 . [DOI] [PubMed] [Google Scholar]
29.Rolfson O, Wissig S, van Maasakkers L, Stowell C, Ackerman I, Ayers D, et al. Defining an International Standard Set of Outcome Measures for Patients With Hip or Knee Osteoarthritis: Consensus of the International Consortium for Health Outcomes Measurement Hip and Knee Osteoarthritis Working Group. Arthritis Care Res (Hoboken). 2016;68(11):1631–9. Epub 2016/10/27. 10.1002/acr.22868 [DOI] [PMC free article] [PubMed] [Google Scholar]
30.Young JL, Rhon DI, Cleland JA, Snodgrass SJ. The Influence of Exercise Dosing on Outcomes in Patients With Knee Disorders: A Systematic Review. J Orthop Sports Phys Ther. 2018;48(3):146–61. Epub 2018/01/13. 10.2519/jospt.2018.7637 . [DOI] [PubMed] [Google Scholar]
31.Nicolson PJA, Hinman RS, Kasza J, Bennell KL. Trajectories of adherence to home-based exercise programs among people with knee osteoarthritis. Osteoarthritis and cartilage / OARS, Osteoarthritis Research Society. 2018;26(4):513–21. Epub 2018/01/24. 10.1016/j.joca.2018.01.009 . [DOI] [PubMed] [Google Scholar]
32.Lee AC, Harvey WF, Han X, Price LL, Driban JB, Bannuru RR, et al. Pain and functional trajectories in symptomatic knee osteoarthritis over up to 12 weeks of exercise exposure. Osteoarthritis Cartilage. 2018;26(4):501–12. Epub 2018/02/03. 10.1016/j.joca.2018.01.016 [DOI] [PMC free article] [PubMed] [Google Scholar]
33.Edge R, Farrah K. Exercise for the Management of Knee Osteoarthritis: A Review of Clinical Effectiveness. CADTH Rapid Response Reports. Ottawa (ON)2017. [PubMed]
34.Wellsandt E, Golightly Y. Exercise in the management of knee and hip osteoarthritis. Curr Opin Rheumatol. 2018;30(2):151–9. Epub 2017/12/19. 10.1097/BOR.0000000000000478 . [DOI] [PubMed] [Google Scholar]

PLoS One. doi: 10.1371/journal.pone.0229783.r001

Decision Letter 0

Kelly Naugle

Transfer Alert

This paper was transferred from another journal. As a result, its full editorial history (including decision letters, peer reviews and author responses) may not be present.

20 Dec 2019

PONE-D-19-29313

Improving osteoarthritis care by digital means - Effects of a digital self-management program after 24 weeks of adherence

PLOS ONE

Dear Dr. Nero,

Thank you for submitting your manuscript to PLOS ONE. After careful consideration, we feel that it has merit but does not fully meet PLOS ONE’s publication criteria as it currently stands. Therefore, we invite you to submit a revised version of the manuscript that addresses the points raised during the review process.

Please address the minor concerns of both reviewers when resubmitting your manuscript.

We would appreciate receiving your revised manuscript by Feb 03 2020 11:59PM. When you are ready to submit your revision, log on to https://www.editorialmanager.com/pone/ and select the 'Submissions Needing Revision' folder to locate your manuscript file.

If you would like to make changes to your financial disclosure, please include your updated statement in your cover letter.

To enhance the reproducibility of your results, we recommend that if applicable you deposit your laboratory protocols in protocols.io, where a protocol can be assigned its own identifier (DOI) such that it can be cited independently in the future. For instructions see: http://journals.plos.org/plosone/s/submission-guidelines#loc-laboratory-protocols

Please include the following items when submitting your revised manuscript:

A rebuttal letter that responds to each point raised by the academic editor and reviewer(s). This letter should be uploaded as separate file and labeled 'Response to Reviewers'.
A marked-up copy of your manuscript that highlights changes made to the original version. This file should be uploaded as separate file and labeled 'Revised Manuscript with Track Changes'.
An unmarked version of your revised paper without tracked changes. This file should be uploaded as separate file and labeled 'Manuscript'.

Please note while forming your response, if your article is accepted, you may have the opportunity to make the peer review history publicly available. The record will include editor decision letters (with reviews) and your responses to reviewer comments. If eligible, we will contact you to opt in or out.

We look forward to receiving your revised manuscript.

Kind regards,

Kelly Naugle, PhD

Academic Editor

PLOS ONE

Journal Requirements:

When submitting your revision, we need you to address these additional requirements.

1. Please ensure that your manuscript meets PLOS ONE's style requirements, including those for file naming. The PLOS ONE style templates can be found at

http://www.journals.plos.org/plosone/s/file?id=wjVg/PLOSOne_formatting_sample_main_body.pdf and http://www.journals.plos.org/plosone/s/file?id=ba62/PLOSOne_formatting_sample_title_authors_affiliations.pdf

2. Please include additional information regarding the survey or questionnaire used in the study and ensure that you have provided sufficient details that others could replicate the analyses. For instance, if you developed a questionnaire as part of this study and it is not under a copyright more restrictive than CC-BY, please include a copy, in both the original language and English, as Supporting Information.

3. We note that you have indicated that data from this study are available upon request. PLOS only allows data to be available upon request if there are legal or ethical restrictions on sharing data publicly. For information on unacceptable data access restrictions, please see http://journals.plos.org/plosone/s/data-availability#loc-unacceptable-data-access-restrictions.

In your revised cover letter, please address the following prompts:

a) If there are ethical or legal restrictions on sharing a de-identified data set, please explain them in detail (e.g., data contain potentially identifying or sensitive patient information) and who has imposed them (e.g., an ethics committee). Please also provide contact information for a data access committee, ethics committee, or other institutional body to which data requests may be sent.

b) If there are no restrictions, please upload the minimal anonymized data set necessary to replicate your study findings as either Supporting Information files or to a stable, public repository and provide us with the relevant URLs, DOIs, or accession numbers. Please see http://www.bmj.com/content/340/bmj.c181.long for guidelines on how to de-identify and prepare clinical data for publication. For a list of acceptable repositories, please see http://journals.plos.org/plosone/s/data-availability#loc-recommended-repositories.

We will update your Data Availability statement on your behalf to reflect the information you provide.

4. Thank you for providing the following Funding Statement:

'Funding was received by LED from Vinnova - Sweden’s Innovation Agency (grant number: 2016-04187, www.vinnova.se) and Stiftelsen för Bistånd åt Rörelsehindrade i Skåne (grant number: 2019-01-20, www.stiftbistandskane.se) to the Department of Clinical Sciences Lund, Orthopaedics, Lund University, Sweden. In kind support (data gathering and extraction) was provided by Arthro Therapeutics Inc. The funders or supporter had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.'

a. We note that one or more of the authors is affiliated with the funding organization, indicating the funder may have had some role in the design, data collection, analysis or preparation of your manuscript for publication; in other words, the funder played an indirect role through the participation of the co-authors.

If the funding organization did not play a role in the study design, data collection and analysis, decision to publish, or preparation of the manuscript and only provided financial support in the form of authors' salaries and/or research materials, please review your statements relating to the author contributions, and ensure you have specifically and accurately indicated the role(s) that these authors had in your study in the Author Contributions section of the online submission form. Please make any necessary amendments directly within this section of the online submission form. Please also update your Funding Statement to include the following statement: “The funder provided support in the form of salaries for authors [insert relevant initials], but did not have any additional role in the study design, data collection and analysis, decision to publish, or preparation of the manuscript. The specific roles of these authors are articulated in the ‘author contributions’ section.”

If the funding organization did have an additional role, please state and explain that role within your Funding Statement.

b. Please also provide an updated Competing Interests Statement declaring this commercial affiliation along with any other relevant declarations relating to employment, consultancy, patents, products in development, or marketed products, etc.

Within your Competing Interests Statement, please confirm that this commercial affiliation does not alter your adherence to all PLOS ONE policies on sharing data and materials by including the following statement: "This does not alter our adherence to PLOS ONE policies on sharing data and materials.” (as detailed online in our guide for authors http://journals.plos.org/plosone/s/competing-interests). If this adherence statement is not accurate and there are restrictions on sharing of data and/or materials, please state these. Please note that we cannot proceed with consideration of your article until this information has been declared.

c. Please include both an updated Funding Statement and Competing Interests Statement in your cover letter. We will change the online submission form on your behalf.

Please know it is PLOS ONE policy for corresponding authors to declare, on behalf of all authors, all potential competing interests for the purposes of transparency. PLOS defines a competing interest as anything that interferes with, or could reasonably be perceived as interfering with, the full and objective presentation, peer review, editorial decision-making, or publication of research or non-research articles submitted to one of the journals. Competing interests can be financial or non-financial, professional, or personal. Competing interests can arise in relationship to an organization or another person. Please follow this link to our website for more details on competing interests: http://journals.plos.org/plosone/s/competing-interests

[Note: HTML markup is below. Please do not edit.]

Reviewers' comments:

Reviewer's Responses to Questions

Comments to the Author

1. Is the manuscript technically sound, and do the data support the conclusions?

The manuscript must describe a technically sound piece of scientific research with data that supports the conclusions. Experiments must have been conducted rigorously, with appropriate controls, replication, and sample sizes. The conclusions must be drawn appropriately based on the data presented.

Reviewer #1: Yes

Reviewer #2: Yes

**********

2. Has the statistical analysis been performed appropriately and rigorously?

Reviewer #1: Yes

Reviewer #2: Yes

**********

3. Have the authors made all data underlying the findings in their manuscript fully available?

The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception (please refer to the Data Availability Statement in the manuscript PDF file). The data should be provided as part of the manuscript or its supporting information, or deposited to a public repository. For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified.

Reviewer #1: No

Reviewer #2: Yes

**********

4. Is the manuscript presented in an intelligible fashion and written in standard English?

PLOS ONE does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here.

Reviewer #1: Yes

Reviewer #2: Yes

**********

5. Review Comments to the Author

Please use the space provided to explain your answers to the questions above. You may also include additional comments for the author, including concerns about dual publication, research ethics, or publication ethics. (Please upload your review as an attachment if it exceeds 20,000 characters)

Reviewer #1: Thank you for the opportunity to review this longitudinal cohort study exploring the effects of a digital self-management program, and the impact of adherence at 6 and 12 months on outcomes. The study is sound in both methodology and reporting, however I would recommend a small number of minor considerations.

General comment/query – the use of the phrase ‘long-term’ is usually reserved for follow-up periods of 12 months or more?

Methods

Setting and Participants

‘About 95%’ – please make this a specific figure

E-questionnaire details – these would be better in the Outcome measures section

Adherence – was exercise completion logged by participants online? This is self-reported whereas the completion of quizzes/interaction with online content is objectively measured. Is this accounted for? Was there justification for including only participants who had adherence of 70% or more in the initial weeks?

Pain reports – was there justification for the choice of the window of +/- 4 or 6 weeks either side of the 24 and 48 weeks timepoints?

Outcome measures – joint pain - was this average pain over the past week?

Figures

Would benefit from formatting (currently directly taken from statistical package)

Discussion

The discussion focuses on comparison to other existing studies. A stronger case for the impact of the findings of this study would be beneficial.

Drop-outs between week 24 and week 48 are significant (n=400) – what are the implications of this for the results and also the programme moving forward (how might these participants be encouraged to remain in the programme).

Reviewer #2: General comments

This is a well-conducted registry-study, providing interesting insights in digital self-management of OA. The manuscript is clearly written and well-reported.

I agree that one of the great challenges in providing evidence-based first-line care for people with OA is findings ways to ensure both appropriate timing and order of treatments (exercise, weight management and education as first-line) as well ensuring treatment retention. Digital self-management may help support appropriate self-management for those people, who we fail to reach through traditional avenues. Digital self-management and better access to professional support (via chat or phone) used in conjunction with treatment strategies may also improve treatment retention. Thus, digital self-management may have several clinically important perspectives in securing optimal care for OA patients.

Apart from knowing very little about the effectiveness of non-surgical treatment options, regarding different exercise types and content of education and self-management, there is a big knowledge gap regarding the dosing of non-surgical treatment strategies for optimal effects.

Specific comments

Abstract:

Results and conclusion:

It is unclear to me, how big the effect on pain and function was. Is the reader supposed to sum up the monthly decrease in pain for 24 weeks and 48 weeks, respectively? I would prefer a more clear presentation of your findings at the two follow-ups.

“There were no clinically relevant effects on the improvement of pain or function

by any of the controlled factors” – what do you mean by controlled factors?

In the conclusion you change the time-period to over 6 months – which of the two follow-ups do you relate this to? Also, you state important decrease in pain and increased function. Do you mean clinically important? Above a certain threshold? Please clarify.

Methods:

Settings and participants

- Which diagnostic criteria did you use to confirm clinical OA via telephone?

- Please clarify the use of +/- 4 week intervals for 24-week follow-up and +/- 6 week intervals for 48-week follow-up. This seems a bit random and means that could essentially be up to 12 weeks difference when follow-up for 48-weeks was reported. This interval alone equals the length of most non-surgical treatment interventions. Please clarify.

- I cannot really understand the choice to divide subjects into the two subsamples (24-week and 48-week). Why not keep participants in one group? In relation to this, you do not really spend much time comparing the two groups anyway.

Outcome measures:

- The 30 s chair-stand test was originally developed as an objectively assessed measure of performance-based physical function. In this study, it was performed as a self-assessed measure. I would recommend extreme caution when interpreting findings from the test. Please discuss this in the context of the findings.

Results:

- The number of participants included in the 24-week subsample do not add up. 920 – 290 = 630. 630 – 138 = 492.

Discussion:

Most of the discussion concerns the exercise component of the digital self-management program. However, it seems unclear to me how much of the total program comprised of exercise (two daily exercises is not much) and how much comprised of self-management strategies. I would recommend a careful interpretation of exercise effects while also appreciating other elements of the program, which might have at least as important. A more in-depth discussion of the content of the program help the reader understand what was actually delivered.

- In relation to this, please clarify more specifically how this digital self-management program is different from other cited digital self-management programs.

- Please comment on the dosage of the program with regards to exercise and self-management compared to other digital and face-to-face programs. This will help clarify the clinical feasibility and sustainability of this intervention.

- Please comment on the clinical importance of the improvements in 30 s chair-stand.

- I would recommend the authors to refrain from discussing potential factors speaking against the potential involvement of regression to the mean. Patents typically seek care when their symptoms are elevated compared to their normal levels (this current sample). The fact that patients improve regardless of baseline pain levels does not speak against involvement of the regression of the mean phenomenon, since the pain levels are individually relative. Only randomization can provide a specific answer to the degree of regression to the mean.

- Please provide some perspectives on how we should move on from here, i.e. how do we explore working mechanisms of the program?

**********

6. PLOS authors have the option to publish the peer review history of their article (what does this mean?). If published, this will include your full peer review and any attached files.

If you choose “no”, your identity will remain anonymous but your review may still be made public.

Do you want your identity to be public for this peer review? For information about this choice, including consent withdrawal, please see our Privacy Policy.

Reviewer #1: No

Reviewer #2: No

[NOTE: If reviewer comments were submitted as an attachment file, they will be attached to this email and accessible via the submission site. Please log into your account, locate the manuscript record, and check for the action link "View Attachments". If this link does not appear, there are no attachment files to be viewed.]

While revising your submission, please upload your figure files to the Preflight Analysis and Conversion Engine (PACE) digital diagnostic tool, https://pacev2.apexcovantage.com/. PACE helps ensure that figures meet PLOS requirements. To use PACE, you must first register as a user. Registration is free. Then, login and navigate to the UPLOAD tab, where you will find detailed instructions on how to use the tool. If you encounter any issues or have any questions when using PACE, please email us at figures@plos.org. Please note that Supporting Information files do not need this step.

PLoS One. 2020 Mar 4;15(3):e0229783. doi: 10.1371/journal.pone.0229783.r002

Author response to Decision Letter 0

3 Feb 2020

Addressing journal Requirements

Journal Requirements:

When submitting your revision, we need you to address these additional requirements.

1. Please ensure that your manuscript meets PLOS ONE's style requirements, including those for file naming. The PLOS ONE style templates can be found at

Action: Removed the capital E in the title.

Response: The digital program contains several questions and questionnaires at different time points, hence there is no one single questionnarie available. We will provide additional information regarding relevant questions and data.

Action: Added information on outcomes and questionnaires under Outcome measures, on page 7, at the beginning and the end of the paragraph.

Response: Data from this registry study contains potentially identifying and sensitive patient information. Since this is a registry study and we are unable to ask for consent from participants before sharing data (contact information of participants ending treatment is discarded, according to Swedish law), we limit the data sharing to be based upon a reasonable request via email.

Action: The above text has been added to the Data Sharing statement, page 16, to further clarify why we take extra precautions in terms of data.

In your revised cover letter, please address the following prompts:

We will update your Data Availability statement on your behalf to reflect the information you provide.

Action: We have updated the cover letter.

4. Thank you for providing the following Funding Statement:

If the funding organization did have an additional role, please state and explain that role within your Funding Statement.

Action: We have added the recommended statement to the Financial Disclosure Statement, please see page 3 in the manuscript.

...” The funder provided support in the form of consulting fees for authors [LED, LSL, HN], but did not have any additional role in the study design, data collection and analysis, decision to publish, or preparation of the manuscript. The specific roles of these authors are articulated in the ‘author contributions’ section.”..

Action: The Competing Interests statement has been updated accordingly, please see page 15 in the manuscript.

c. Please include both an updated Funding Statement and Competing Interests Statement in your cover letter. We will change the online submission form on your behalf.

Action: The cover letter has been updated accordingly.

Rebuttal letter PONE-D-19-29313

To the editor and reviewers,

Thank You for the opportunity to improve our manuscript Improving osteoarthritis care by digital means - effects of a digital self-management program after 24 weeks of adherence, referenced PONE-D-19-29313. We have addressed the comments by the reviewers and hope that our revision will be satisfactory. Please find below our point-by-point responses and actions to each of the editors and reviewers’ concerns. The reviewer comments are written in italics and changes are referred to in the manuscript based on page number and section as well as quoted after each described action.

1. General comment/query – the use of the phrase ‘long-term’ is usually reserved for follow-up periods of 12 months or more?

Response: Thank you for the taking the time to review our manuscript. We used the phrase long-term since the most common treatment period in terms of first-line treatment for osteoarthritis (OA) is from 6 weeks to 3 months, and to the best of our knowledge no other study has reported results from continuous treatment for 6 months or more.

Action: None.

2. Methods

Setting and Participants

‘About 95%’ – please make this a specific figure

Response: Thank you for the suggestion, we will specify it as 95%, as this was the percentage at the time of data extraction.

Action: Removed the word “About” before “95%” below Setting and participants, page 5.

..”Included participants had a radiographic and or clinical diagnosis of hip or knee OA from a physical therapist or physician (95% of all patients at the date of data extraction).”…

3. E-questionnaire details – these would be better in the Outcome measures section

Response: Thanks, the section has been moved accordingly, and also altered to clarify details on outcomes (as suggested by the editor).

Action: Moved text from under Setting and participants page 5, to Outcome measures, page 7 in the manuscript.

..”Data on the participants’ overall health and characteristics (age, gender, BMI) as well as OA-related factors (most painful joint, afflicted side) were collected at inclusion.”…

4. Adherence – was exercise completion logged by participants online? This is self-reported whereas the completion of quizzes/interaction with online content is objectively measured. Is this accounted for? Was there justification for including only participants who had adherence of 70% or more in the initial weeks?

Response: Exercise completion was indicated by patient actions through the smartphone app. When exercises (in video format with explanatory graphics) were distributed to the patient, the patient was asked to rate the perceived difficulty of the exercise. Entering the perceived difficulty was used as indicator that the exercise was performed. Completion of quizzes/lessons was indicated by the patient answering the quizzes correctly. Program continuation was halted until these tasks were completed. Agreed, there is some difference in terms of how lessons and exercises are handled, and we have made a slight change in the manuscript to clarify this.

We intended to monitor the effect of participation in the digital treatment, and arbitrarily chose the 70% cut-off as a threshold for what could reasonably be considered participation in treatment.

Action: Information on collection of information on completed exercises and lessons has been added to the Intervention section, please see page 6 and 7 of the manuscript.

…“While rating perceived difficulty and adding comments, patients also indicated when exercises were completed. Program continuation was halted until all exercises for the day were marked as completed.”…

…“Completion of a lesson was indicated by the patient answering the quiz correctly.”…

5. Pain reports – was there justification for the choice of the window of +/- 4 or 6 weeks either side of the 24 and 48 weeks timepoints?

Response: The weekly reporting of pain is not mandatory, to alleviate patient strain and add more flexibility. Around 54% of the 24 week sub-sample had reported pain at week 24. Similarly, around 50% of participants active at week 48 had reported pain this specific week. Hence we opted for the +/- 4 weeks and +/- 6 weeks window to collect the maximum amount of data. We will clarify this in the manuscript.

Action: Clarified information on time windows on page 6, Setting and participants:

..”Outcome analysis was made in two separate sub-samples; participants with a pain report from week 24 or adjacent week (+- 4 weeks) and week 48 or adjacent week (+- 6 weeks), respectively. Adjacent weeks were added to collect the maximum amount of data (around 50% of active participants had reported pain at week 24 and 48, respectively).”..

6. Outcome measures – joint pain - was this average pain over the past week?

Response: Yes, as stated under Outcome measures: ..”Mark on this scale how much pain you had the last week in your hip/knee”…

Action: None.

7. Figures

Would benefit from formatting (currently directly taken from statistical package)

Response: Thank you for the suggestion, the figures have been formatted/updated. Please see new figure 2, 3 and 4 below.

Action: Formatted figures and updated the manuscript files, also attached new figures below:

8. Discussion

The discussion focuses on comparison to other existing studies. A stronger case for the impact of the findings of this study would be beneficial.

Response: Thanks for highlighting these issues. We would like to offer an alternative view on the matter. We believe that, in the current scientific community, you must argue for the impact of your study by comparing it to the work of other researchers. If no comparison is available, an evaluation is hard. We also believe that we have in fact argued for the impact of our findings, by first comparing to previous work and then highlighting our unique findings, or our confirmatory findings, and their potential impact.

For example, from page 14, second paragraph: ...” Participants in the present digital self-management programme with more severe pain experienced larger absolute pain reductions compared to those with less severe pain, yet the relative reduction was comparable. This suggests that the programme has the potential to yield similar benefit regardless of pain experienced by the participant at baseline. Hence it may be argued that the selection of patients for an exercise-based intervention should not be based solely on pain, and confirms that exercise is effective in reducing pain regardless of symptom severity [12].”...

In regard to drop-outs, it is more complicated to define a drop-out when you administer a program that has no defined ending. The treatment ends when the patient considers it appropriate, or when the physiotherapist deems it suitable. Our previous qualitative studies indicated several reasons for the patient quitting the program, such as: they had improved to an extent where continued treatment seemed unnecessary; they had not improved sufficiently and was seeking other options; or that the patient continued exercising on their own without the smartphone application (1). Interestingly, the first pilot study based on this program showed no difference between those ending treatment and those continuing, in terms of level of pain (2). Unfortunately for the current study we do not have data (on a group level) on what was the specific reason for patients to quit the program. Adding on, patients in the register have started the treatment sequentially, hence not all participants in our sample have had the opportunity to be in the program for 48 weeks. In fact, only 710 individuals of the 1709 had started more than or equal to 48 weeks before data extraction.

Since OA is a chronic disease the treatment needs to be continuous yet changing life-style routines and habits in patients is, as we know, hard. Research on our end is ongoing, trying to understand when and why patients stop adhering, and we are also interested in scientifically testing some different behavioral strategies to prolong adherence.

To further clarify the matter of drop-outs, we have edited some text in the Method section, added some information to the topic in the Discussion and edited Figure 1.

Action: Added/edited the following text in the Discussion, page 15:

..” Since results are based on a register of patients voluntarily choosing whether to report their outcomes and when to end treatment, reflecting clinical reality, some data is missing. Although the results suggest that those ending treatment do not importantly differ from included participants in terms of descriptive factors at baseline, for future studies it would be of value to interview and follow those ending treatment, and their OA-related outcomes. Specifically, it would be valuable due to the current program being continuous and having no defined ending.”...

Added the following text to the Method section, page 6:

..”At that time point, the register contained data from 1709 Swedish participants (710 individuals had started ≥48 weeks before data extraction) that had reported one of their knees or hips as their most symptomatic joint (index joint), had been treated in the programme for at least three weeks with a minimum adherence of 70% and had registered ≥24 weeks before data extraction.”..

Edited Figure 1 to clarify how many had started the program 48 weeks before data extraction, and how many were excluded from the 24-week sub-sample due to data at week 48 or adjacent:

Reviewer #2: General comments

1. This is a well-conducted registry-study, providing interesting insights in digital self-management of OA. The manuscript is clearly written and well-reported.

Response: Thank you for your help in improving our manuscript. Agreed, there are unknown aspects about optimal dosage or dose-response in terms of first-line treatment for osteoarthritis. What we do know is that most programs reporting beneficial results contain a minimum of 3 sessions per week, focus on single-type exercises (e.g. strengthening muscles surrounding the painful joint), and are supervised (3). Our study adds to the evidence in terms of duration, showing that treatment for a minimum of 6 months leads to substantial improvements in terms of pain.

Action: None.

Specific comments

Abstract:

2. Results and conclusion:

Response: We were interested in reporting the effect per month of duration to add to the evidence of exercise duration and its effects on pain and function. We felt this adds more valuable information than just a mean difference, seeing as the data underlying the calculation was weekly reports. Adding on, means for baseline and follow-ups can be found in table 3. However, we concur that the results in the abstract can be clearer in this aspect and will revise the abstract accordingly.

Action: Mean numbers for baseline and follow-up was added to the Methods and Findings section of the abstract, and the results were further clarified:

..” For the 24-week sub-sample, pain NRS decreased monthly by -0·43 units (95% CI -0·51, -0·35, mean knee pain from 5.6 to 3.1, and hip pain from 5.9 to 3.8) and 30CST repetitions increased monthly by 0·76 repetitions (95% CI 0·64, 0·89 mean for knee from 10.0 to 14.3, and for hip from 10.9 to 14.8). For the 48-week sub-sample, pain decreased monthly by -0·39 units (95% CI -0·43, -0·36, mean knee pain from 5.7 to 3.2, and hip pain from 5.8 to 3.8), and repetitions increased by 0·72 repetitions (95% CI 0·65, 0·79, mean repetitions for knee from 10.3 to 14.4, and for hip from 11.1 to 14.9).”...

3. “There were no clinically relevant effects on the improvement of pain or function

by any of the controlled factors” – what do you mean by controlled factors?

Response: Thanks for highlighting this, in fact the proper term should be covariates, as described in the Statistical analysis. This needs to be clarified.

Action: Added text in the Abstract – Methods and findings:

..” There were no clinically relevant effects on the improvement of pain or function by any covariate (age, sex, index joint).”...

4. In the conclusion you change the time-period to over 6 months – which of the two follow-ups do you relate this to? Also, you state important decrease in pain and increased function. Do you mean clinically important? Above a certain threshold? Please clarify.

Response: The word over may confuse hence it will be changed to for. In terms of important increase, we refer to the fact that it was a clinically relevant improvement, and as referred to in the manuscript, on page 7 under Outcome measures, above what was previously defined as minimal clinically relevant change (20%) for pain. We will clarify this.

Action: Changed the text in the Conclusion of the Abstract, page 3:

..” Continuously participating in a digital OA treatment program for 6 months was associated with a clinically important decrease in joint pain and increased physical function, in hip and knee OA.”...

Methods:

Settings and participants

5. - Which diagnostic criteria did you use to confirm clinical OA via telephone?

Response:

The NICE guidelines were used as diagnostic criteria, also acknowledged in the Swedish National Guidelines, stating that OA can be diagnosed without further evaluation if the person is 45 years old or over, has activity-related joint pain, has either no morning stiffness or morning stiffness that lasts no longer than 30 minutes. It was also confirmed that no atypical features, or red flags, were present.

Information relating to symptoms is available to the physiotherapist before the initial telephone meeting. The physiotherapist is obliged during the telephone call to confirm all collected data and ask clinically relevant questions to rule out differential diagnosis. After including more than 12.000 patients into the program, there has been no problem related to improper diagnosing or adverse events.

We have added a small paragraph under Methods, Setting and participants, page 5 to clarify.

Action: Added some information on red flags under Settings and participants, page 5:

..”Individuals without a prior diagnosis had clinical OA confirmed by an orthopaedic surgeon or physiotherapist via telephone (diagnosis according to NICE criteria and Swedish National Guidelines, and confirming the absence of any red flag symptoms), or if deemed necessary were recommended to seek face-to-face care before inclusion in the programme.”..

6. - Please clarify the use of +/- 4 week intervals for 24-week follow-up and +/- 6 week intervals for 48-week follow-up. This seems a bit random and means that could essentially be up to 12 weeks difference when follow-up for 48-weeks was reported. This interval alone equals the length of most non-surgical treatment interventions. Please clarify.

Response: As stated in the response to Reviewer 1, question number 5, the weekly reporting of pain is not mandatory, to alleviate patient strain and add more flexibility. After inspecting the data, around 54% of the 24 week sub-sample had reported pain at week 24. Similarly, around 50% of participants active at week 48 had reported pain this specific week. Hence we opted for the +/- 4 weeks and +/- 6 weeks window to collect the maximum amount of data.

Action: None.

7. - I cannot really understand the choice to divide subjects into the two subsamples (24-week and 48-week). Why not keep participants in one group? In relation to this, you do not really spend much time comparing the two groups anyway.

Response: If we would merge the 2 groups, we would increase the number up to 24w, but would have a larger dropout after this, and the patients responding at 48w would not be the same as those responding at 24w. We were interested in reporting results of prospective longitudinal cohorts with minimum loss to follow-up. Adding on, in a Swedish face-to-face program with published data from its registry, there is a worsening of symptoms (pain e.g.) between 3-12 months (4), hence it was interesting for us to see if this was also evident for a digital continuous program, and if so when the deterioration occurred. Hence we wanted separated cohorts in terms of duration of adherence. We clarified this in the title of the manuscript.

Action: Edited the title on page 1:

..”Improving osteoarthritis care by digital means - effects of a digital self-management program after 24- or 48-weeks of treatment.”..

8. Outcome measures:

Response: The performance of the test is pedagogically instructed using a video with text that can be played as many times as needed. The instructions are according to those published by the Swedish Physiotherapy Association. Hence the instructions are the same that a physiotherapist interprets in the clinic, where there may also be individual differences. Furthermore, the test and its result are used intra-person, hence we do not use the result to compare with other studies or individuals. Following your comment, we have added a paragraph on the interpretation of the test in the Discussion.

Action: Added a paragraph in the Discussion, page 15:

..”Adding on, the 30 CST was instructed using a video and the patient thus performed the test accordingly without the supervision of a physiotherapist, hence the result from the measure should be interpreted with caution.”..

9. Results:

- The number of participants included in the 24-week subsample do not add up. 920 – 290 = 630. 630 – 138 = 492.

Response: Following the flow chart in figure 1, it does add up. Most likely, from your calculation above, the 7 participants missing in your calculation are individuals with 48 week data that were missing data at week 24. Hence counting backwards, it does not seem to add up. We clarified this in the Results, thank you for pointing this out.

Action: Added information in the Results, page 9:

..” For the 48-week sub-sample, a total of 138 individuals (n=7 missing 24-week data included) with pain data at 48 weeks were included (Figure 1).”...

10. Discussion:

Response: Reading through the discussion once again, we do not completely agree that most of the discussion concern the exercise component only. Most of the discussion, we suggest, concerns program specific components, one of them being exercise. There is one paragraph on page 14 that focuses primarily on exercise, this due to the fact that there is an abundance of studies investigating the effects of exercise, as compared to the other components such as education, physiotherapy support, automatic notifications, etc. It would be highly interesting to investigate specific effects of these individual components, but that would be out of scope for this study. If self-management is interpreted as theoretical education about the disease and how to self-manage in daily life, we know from previous studies based on face-to-face self-management programs, that the theoretical part of the program (patient education) alone was associated with less improvement of symptoms than education combined with exercise (4). Whether this applies to digital programs as well, is still unknown.

We have added some text in the Discussion concerning the content of the program.

Action: Added text to the Discussion, page 16:

..” Yet further investigation into specific components of digital interventions outside of exercise are warranted, to more clearly understand the mechanics and benefits of specific parts of digital OA treatment programs.”...

11. - In relation to this, please clarify more specifically how this digital self-management program is different from other cited digital self-management programs.

Response: To the best of our understanding, the unique aspects of Joint Academy are that each patient is connected to a dedicated personal physiotherapist which the patient can contact at any time of day for advice or discussion. Furthermore, the program necessitates no added equipment to perform and start exercising. Thirdly, according to our knowledge, this is the only digital self-management program that is based on a nationally implemented face-to-face initiative that has been delivered to more than 120.000 individuals in Sweden.

The study from Bossen et al cited in the manuscript describes a web-based intervention focusing on increasing patient-selected physical activity, in increments. The intervention lasts for 9 weeks and for the study n=199 individuals were recruited. The other cited study describing a digital program, by Smittenaar et al, is based on n=41 individuals participating in a 12 week program. Participation necessitates wearing several sensors while exercising.

Action: Added some info on the two cited studies on digital programs, in the Discussion, page 14:

..”Bossen et al. showed a 2-point reduction on the WOMAC pain subscale (equivalent to a 10-point reduction on a 0-100 scale) after 8 months of providing a fully automated web-based programme aiming to increase patients’ physical activity (n=199) [21]. The intervention consisted of a 9-week gradually increasing program.”..

..”Another self-management programme including education and exercise delivered digitally, showed a similar pain reduction after 6 months in comparison to the current study, although results were based on a small sample (n=41), and the program ended after 12 weeks [22].”..

12. - Please comment on the dosage of the program with regards to exercise and self-management compared to other digital and face-to-face programs. This will help clarify the clinical feasibility and sustainability of this intervention.

Response: It is hard to access or even find other digital programs similar to Joint Academy offering treatment for osteoarthritis of the hip or knee. As previously noted in the response to Question 11, we have referenced to two other programs focusing on knee pain or OA. There is also a digital program called Kaia that distributes exercises every day at about the same amount as Joint Academy but focusing on unspecified lower back pain. In terms of face-to-face programs, due to the effort of frequent visits to a clinic, most programs offer exercise sessions a few times per week, but with longer session duration. These programs also have a specified duration (3 months for BOA and the GLA:D programs) and thereby data on how much exercise is performed after the program may be sparse. We hypothesize, as mentioned in the Discussion on page 13-14, that the greater mean improvements reported in the current study is due to the constant availability of support from a physiotherapist, and the high frequency of training.

Action: Added a few words on high frequency of exercises, in the Discussion, page 14:

..”The presence of targeted joint-specific exercises in the present digital self-management programme performed 5-10 minutes daily, combined with the inclusion of human supervision with continuous follow-up of goals and outcomes, may explain the greater pain reduction showed in the current study.”..

13. - Please comment on the clinical importance of the improvements in 30 s chair-stand.

Response: There are published thresholds for what constitutes a major clinically important improvement in people with hip osteoarthritis performing the 30 CST (5). These vary between an increase of 2.0 - 2.6 repetitions, depending on method. Hence our result of an increase of 3.8 – to 3.9 (depending on sub-sample) for the hip OA patients would constitute a clinically important improvement.

Action: None.

14. - I would recommend the authors to refrain from discussing potential factors speaking against the potential involvement of regression to the mean. Patents typically seek care when their symptoms are elevated compared to their normal levels (this current sample). The fact that patients improve regardless of baseline pain levels does not speak against involvement of the regression of the mean phenomenon, since the pain levels are individually relative. Only randomization can provide a specific answer to the degree of regression to the mean.

Response: Thanks for highlighting this issue. OA can be intermittent in regard to symptoms such as pain, so a variation over time would not be surprising. The variation in pain at baseline for the different severity groups is; low: 0-5 (minimum-maximum), moderate: 5-7 (min-max) and severe: 7-10 (min-max). This would indicate quite a spread in terms of pain at baseline, although not disproving the effect of regression to the mean it indicates that perhaps not all patients seek treatment mainly due to severe pain. In fact, it has been suggested that care seeking behavior seems to be based on a mix of social and psychological factors and not simply based on the absence or presence of medical problems (6). Whether care seeking behavior in relation to digital programs differs from face-to-face treatment, is unknown. For certain societal groups and diagnoses digital care may be more accessible and hence care seeking may be related to other factors.

Furthermore, regression to the mean typically constitutes a value closer to the mean following an extreme measure, and as mentioned we cannot disprove this effect in the current study. However, we can argue that the effect of regression to the mean is reduced when taking serial measurements and calculating the average change (7).

Yet after a brief discussion of this phenomena, we agree that randomization in a controlled study is what can provide a specific answer to the degree of the regression to the mean, and we will edit the manuscript to this effect.

Action: Added and edited a paragraph in the Discussion, page 15:

..” In the current study pain and physical function were both shown to improve over time. Furthermore, patients of all pain levels at baseline improved (high variability in baseline pain was found). Even so, some contribution to outcomes by context effects and regression to the mean would be expected, although the magnitude is uncertain. Only a randomized controlled trial can provide a specific answer to the degree of regression to the mean.”…

15. - Please provide some perspectives on how we should move on from here, i.e. how do we explore working mechanisms of the program?

Response: With digital programs and the possibility of collecting vast amounts of data on a scale not possible with face-to-face programs, the option of using machine learning algorithms to test and examine what mechanisms are most important for specific sub-groups is available. In a digital setting it is possible to rather easily alter parts of the program to a randomized set of participants, and this is what we are hoping to achieve in the near future.

We have added a comment in the discussion on this subject as mentioned in the response to question number 10 above, but not specified further, as we feel it is outside of the scope of this study.

Action: On page 16 in the Discussion, some text was added:

References

1. Cronström A et al. 'I Would Never Have Done It if It Hadn't Been Digital': A Qualitative Study on Patients' Experiences of a Digital Management Programme for Hip and Knee Osteoarthritis in Sweden. BMJ Open, 9 (5), e028388. 2019.

2. Dahlberg LE et al. A Web-Based Platform for Patients With Osteoarthritis of the Hip

and Knee:A Pilot Study. JMIR Res Protoc. 5 (2), e115. 2016.

3. Juhl C et al. Impact of Exercise Type and Dose on Pain and Disability in Knee Osteoarthritis. Arthritis Rheumatol, 66 (3), 622-36. 2014.

4. Jönsson T et al. The Better Management of Patients With Osteoarthritis Program: Outcomes After Evidence-Based Education and Exercise Delivered Nationwide in Sweden. PLoS One. 14 (9), e0222657. 2019.

5. Wright AA et al. A Comparison of 3 Methodological Approaches to Defining Major Clinically Important Improvement of 4 Performance Measures in Patients With Hip Osteoarthritis. J Orthop Sports Phys Ther. 41 (5), 319-27. 2011.

6. Campbell SM et al. Why Do People Consult the Doctor? Fam Pract. 13 (1), 75-83. 1996.

7. Morton V et al. Effect of regression to the mean on decision making in health care. BMJ;326:1083. 2003.

Attachment

Submitted filename: Rebuttal letter.docx

Click here for additional data file.^{(278.7KB, docx)}

PLoS One. doi: 10.1371/journal.pone.0229783.r003

Decision Letter 1

Kelly Naugle

14 Feb 2020

Improving osteoarthritis care by digital means - effects of a digital self-management program after 24- or 48-weeks of treatment

PONE-D-19-29313R1

Dear Dr. Nero,

We are pleased to inform you that your manuscript has been judged scientifically suitable for publication and will be formally accepted for publication once it complies with all outstanding technical requirements.

Within one week, you will receive an e-mail containing information on the amendments required prior to publication. When all required modifications have been addressed, you will receive a formal acceptance letter and your manuscript will proceed to our production department and be scheduled for publication.

Shortly after the formal acceptance letter is sent, an invoice for payment will follow. To ensure an efficient production and billing process, please log into Editorial Manager at https://www.editorialmanager.com/pone/, click the "Update My Information" link at the top of the page, and update your user information. If you have any billing related questions, please contact our Author Billing department directly at authorbilling@plos.org.

If your institution or institutions have a press office, please notify them about your upcoming paper to enable them to help maximize its impact. If they will be preparing press materials for this manuscript, you must inform our press team as soon as possible and no later than 48 hours after receiving the formal acceptance. Your manuscript will remain under strict press embargo until 2 pm Eastern Time on the date of publication. For more information, please contact onepress@plos.org.

With kind regards,

Kelly Naugle, PhD

Academic Editor

PLOS ONE

Additional Editor Comments (optional):

Reviewers' comments:

Reviewer's Responses to Questions

Comments to the Author

1. If the authors have adequately addressed your comments raised in a previous round of review and you feel that this manuscript is now acceptable for publication, you may indicate that here to bypass the “Comments to the Author” section, enter your conflict of interest statement in the “Confidential to Editor” section, and submit your "Accept" recommendation.

Reviewer #2: All comments have been addressed

**********

2. Is the manuscript technically sound, and do the data support the conclusions?

Reviewer #2: Yes

**********

3. Has the statistical analysis been performed appropriately and rigorously?

Reviewer #2: Yes

**********

4. Have the authors made all data underlying the findings in their manuscript fully available?

Reviewer #2: No

**********

5. Is the manuscript presented in an intelligible fashion and written in standard English?

Reviewer #2: Yes

**********

6. Review Comments to the Author

Reviewer #2: I appreciate the authors efforts in accommodating the manuscript according to comments and concerns raised. I have no further concerns regarding the reporting of this study.

I look forward to learn more about digital self-management in the treatment of OA in future work from your end.

**********

7. PLOS authors have the option to publish the peer review history of their article (what does this mean?). If published, this will include your full peer review and any attached files.

If you choose “no”, your identity will remain anonymous but your review may still be made public.

Do you want your identity to be public for this peer review? For information about this choice, including consent withdrawal, please see our Privacy Policy.

Reviewer #2: No

PLoS One. doi: 10.1371/journal.pone.0229783.r004

Acceptance letter

Kelly Naugle

19 Feb 2020

PONE-D-19-29313R1

Improving osteoarthritis care by digital means - effects of a digital self-management program after 24- or 48-weeks of treatment

Dear Dr. Nero:

I am pleased to inform you that your manuscript has been deemed suitable for publication in PLOS ONE. Congratulations! Your manuscript is now with our production department.

If your institution or institutions have a press office, please notify them about your upcoming paper at this point, to enable them to help maximize its impact. If they will be preparing press materials for this manuscript, please inform our press team within the next 48 hours. Your manuscript will remain under strict press embargo until 2 pm Eastern Time on the date of publication. For more information please contact onepress@plos.org.

For any other questions or concerns, please email plosone@plos.org.

Thank you for submitting your work to PLOS ONE.

With kind regards,

PLOS ONE Editorial Office Staff

on behalf of

Dr. Kelly Naugle

Academic Editor

PLOS ONE

Associated Data

This section collects any data citations, data availability statements, or supplementary materials included in this article.

Supplementary Materials

S1 Table. Comparative data for the 24-week sub-sample (n = 499) and excluded participants (n = 1210).

(PDF)

Click here for additional data file.^{(58KB, pdf)}

S1 Checklist. STROBE statement—checklist of items that should be included in reports of cohort studies.

(DOC)

Click here for additional data file.^{(122KB, doc)}

Attachment

Submitted filename: Rebuttal letter.docx

Click here for additional data file.^{(278.7KB, docx)}

Data Availability Statement

[pone.0229783.ref001] 1.Murray CJL, Vos T, Lozano R, Naghavi M, Flaxman AD, Michaud C, et al. Disability-adjusted life years (DALYs) for 291 diseases and injuries in 21 regions, 1990–2010: a systematic analysis for the Global Burden of Disease Study 2010. The Lancet. 2012;380(9859):2197–223. 10.1016/s0140-6736(12)61689-4 [DOI] [PubMed] [Google Scholar]

[pone.0229783.ref002] 2.Willett M, Duda J, Gautrey C, Fenton S, Greig C, Rushton A. Effectiveness of behavioural change techniques in physiotherapy interventions to promote physical activity adherence in patients with hip and knee osteoarthritis: a systematic review protocol. BMJ Open. 2017;7(6):e015833 Epub 2017/07/02. 10.1136/bmjopen-2017-015833 [DOI] [PMC free article] [PubMed] [Google Scholar]

[pone.0229783.ref003] 3.Kingsbury SR, Gross HJ, Isherwood G, Conaghan PG. Osteoarthritis in Europe: impact on health status, work productivity and use of pharmacotherapies in five European countries. Rheumatology (Oxford). 2014;53(5):937–47. Epub 2014/02/04. 10.1093/rheumatology/ket463 . [DOI] [PubMed] [Google Scholar]

[pone.0229783.ref004] 4.Turkiewicz A, Petersson IF, Bjork J, Hawker G, Dahlberg LE, Lohmander LS, et al. Current and future impact of osteoarthritis on health care: a population-based study with projections to year 2032. Osteoarthritis and cartilage / OARS, Osteoarthritis Research Society. 2014;22(11):1826–32. Epub 2014/08/02. 10.1016/j.joca.2014.07.015 . [DOI] [PubMed] [Google Scholar]

[pone.0229783.ref005] 5.Wallace IJ, Worthington S, Felson DT, Jurmain RD, Wren KT, Maijanen H, et al. Knee osteoarthritis has doubled in prevalence since the mid-20th century. Proceedings of the National Academy of Sciences of the United States of America. 2017;114(35):9332–6. Epub 2017/08/16. 10.1073/pnas.1703856114 [DOI] [PMC free article] [PubMed] [Google Scholar]

[pone.0229783.ref006] 6.Fransen M, McConnell S, Harmer AR, Van der Esch M, Simic M, Bennell KL. Exercise for osteoarthritis of the knee. In: Fransen M, editor. Chichester, UK: John Wiley & Sons, Ltd; 2015. [Google Scholar]

[pone.0229783.ref007] 7.Fransen M, McConnell S, Hernandez-Molina G, Reichenbach S. Exercise for osteoarthritis of the hip. Cochrane Database Syst Rev. 2014;(4):CD007912 Epub 2014/04/24. 10.1002/14651858.CD007912.pub2 . [DOI] [PMC free article] [PubMed] [Google Scholar]

[pone.0229783.ref008] 8.Fernandes L, Hagen KB, Bijlsma JW, Andreassen O, Christensen P, Conaghan PG, et al. EULAR recommendations for the non-pharmacological core management of hip and knee osteoarthritis. Ann Rheum Dis. 2013;72(7):1125–35. Epub 2013/04/19. 10.1136/annrheumdis-2012-202745 . [DOI] [PubMed] [Google Scholar]

[pone.0229783.ref009] 9.Hochberg MC, Altman RD, April KT, Benkhalti M, Guyatt G, McGowan J, et al. American College of Rheumatology 2012 recommendations for the use of nonpharmacologic and pharmacologic therapies in osteoarthritis of the hand, hip, and knee. Arthritis Care Res (Hoboken). 2012;64(4):465–74. Epub 2012/05/09. 10.1002/acr.21596 . [DOI] [PubMed] [Google Scholar]

[pone.0229783.ref010] 10.The Swedish National Board of Health and Welfare—National guidelines for orthopaedic conditions. Available from: http://www.socialstyrelsen.se/nationalguidelines/nationalguidelinesfororthopaedicconditions

[pone.0229783.ref011] 11.Bannuru RR, Osani MC, Vaysbrot EE, Arden NK, Bennell K, Bierma-Zeinstra SMA, et al. OARSI guidelines for the non-surgical management of knee, hip, and polyarticular osteoarthritis. Osteoarthritis Cartilage. 2019. Epub 2019/07/07. 10.1016/j.joca.2019.06.011 . [DOI] [PubMed] [Google Scholar]

[pone.0229783.ref012] 12.Juhl C, Christensen R, Roos EM, Zhang W, Lund H. Impact of exercise type and dose on pain and disability in knee osteoarthritis: a systematic review and meta-regression analysis of randomized controlled trials. Arthritis Rheumatol. 2014;66(3):622–36. Epub 2014/02/28. 10.1002/art.38290 . [DOI] [PubMed] [Google Scholar]

[pone.0229783.ref013] 13.Basedow M, Esterman A. Assessing appropriateness of osteoarthritis care using quality indicators: A systematic review. Journal of Evaluation in Clinical Practice. 2015;21(5):782–9. 10.1111/jep.12402 [DOI] [PubMed] [Google Scholar]

[pone.0229783.ref014] 14.Runciman WB, Hunt TD, Hannaford NA, Hibbert PD, Westbrook JI, Coiera EW, et al. CareTrack: assessing the appropriateness of health care delivery in Australia. Med J Aust. 2012;197(2):100–5. Epub 2012/07/17. 10.5694/mja12.10510 . [DOI] [PubMed] [Google Scholar]

[pone.0229783.ref015] 15.Caneiro JP, O'Sullivan PB, Roos EM, Smith AJ, Choong P, Dowsey M, et al. Three steps to changing the narrative about knee osteoarthritis care: a call to action. British journal of sports medicine. 2019. Epub 2019/09/06. 10.1136/bjsports-2019-101328 . [DOI] [PubMed] [Google Scholar]

[pone.0229783.ref016] 16.Allen KD, Choong PF, Davis AM, Dowsey MM, Dziedzic KS, Emery C, et al. Osteoarthritis: Models for appropriate care across the disease continuum. Best Practice and Research: Clinical Rheumatology. 2016;30(3):503–35. 10.1016/j.berh.2016.09.003 [DOI] [PubMed] [Google Scholar]

[pone.0229783.ref017] 17.Thorstensson CA, Garellick G, Rystedt H, Dahlberg LE. Better Management of Patients with Osteoarthritis: Development and Nationwide Implementation of an Evidence-Based Supported Osteoarthritis Self-Management Programme. Musculoskeletal Care. 2015;13(2):67–75. Epub 2014/10/28. 10.1002/msc.1085 . [DOI] [PubMed] [Google Scholar]

[pone.0229783.ref018] 18.Dell'Isola A, Jonsson T, Ranstam J, Dahlberg LE, Hansson EE. Education, home exercise and supervised exercise for people with hip and knee osteoarthritis as part of a nationwide implementation programme: data from the BOA registry. Arthritis Care Res (Hoboken). 2019. 10.1002/acr.24033 . [DOI] [PubMed] [Google Scholar]

[pone.0229783.ref019] 19.Skou ST, Roos EM. Good Life with osteoArthritis in Denmark (GLA:D): evidence-based education and supervised neuromuscular exercise delivered by certified physiotherapists nationwide. BMC Musculoskelet Disord. 2017;18(1):72 Epub 2017/02/09. 10.1186/s12891-017-1439-y [DOI] [PMC free article] [PubMed] [Google Scholar]

[pone.0229783.ref020] 20.Hurley MV, Walsh NE, Mitchell H, Nicholas J, Patel A. Long-term outcomes and costs of an integrated rehabilitation program for chronic knee pain: a pragmatic, cluster randomized, controlled trial. Arthritis Care Res (Hoboken). 2012;64(2):238–47. Epub 2011/09/29. 10.1002/acr.20642 . [DOI] [PubMed] [Google Scholar]

[pone.0229783.ref021] 21.Bossen D, Veenhof C, Van Beek KE, Spreeuwenberg PM, Dekker J, De Bakker DH. Effectiveness of a web-based physical activity intervention in patients with knee and/or hip osteoarthritis: randomized controlled trial. J Med Internet Res. 2013;15(11):e257 Epub 2013/11/26. 10.2196/jmir.2662 [DOI] [PMC free article] [PubMed] [Google Scholar]

[pone.0229783.ref022] 22.Smittenaar P, Erhart-Hledik JC, Kinsella R, Hunter S, Mecklenburg G, Perez D. Translating Comprehensive Conservative Care for Chronic Knee Pain Into a Digital Care Pathway: 12-Week and 6-Month Outcomes for the Hinge Health Program. JMIR Rehabil Assist Technol. 2017;4(1):e4 Epub 2017/06/06. 10.2196/rehab.7258 [DOI] [PMC free article] [PubMed] [Google Scholar]

[pone.0229783.ref023] 23.Nero H, Dahlberg J, Dahlberg LE. A 6-Week Web-Based Osteoarthritis Treatment Program: Observational Quasi-Experimental Study. Journal of medical Internet research. 2017;19(12):e422 Epub 2017/12/20. 10.2196/jmir.9255 . [DOI] [PMC free article] [PubMed] [Google Scholar]

[pone.0229783.ref024] 24.Cronstrom A, Nero H, Dahlberg LE. Factors associated with patients' willingness to consider joint surgery after completion of a digital osteoarthritis treatment program: A prospective cohort study. Arthritis Care Res (Hoboken). 2018. Epub 2018/10/10. 10.1002/acr.23772 . [DOI] [PMC free article] [PubMed] [Google Scholar]

[pone.0229783.ref025] 25.Von Elm E, Altman DG, Egger M, Pocock SJ, Gøtzsche PC, Vandenbroucke JP. The Strengthening the Reporting of Observational Studies in Epidemiology (STROBE) statement: guidelines for reporting observational studies. Annals of internal medicine. 2007;147(8):573–7. 10.7326/0003-4819-147-8-200710160-00010 [DOI] [PubMed] [Google Scholar]

[pone.0229783.ref026] 26.Williamson A, Hoggart B. Pain: a review of three commonly used pain rating scales. Journal of clinical nursing. 2005;14(7):798–804. Epub 2005/07/08. 10.1111/j.1365-2702.2005.01121.x . [DOI] [PubMed] [Google Scholar]

[pone.0229783.ref027] 27.Tubach F, Ravaud P, Martin-Mola E, Awada H, Bellamy N, Bombardier C, et al. Minimum clinically important improvement and patient acceptable symptom state in pain and function in rheumatoid arthritis, ankylosing spondylitis, chronic back pain, hand osteoarthritis, and hip and knee osteoarthritis: Results from a prospective multinational study. Arthritis Care Res (Hoboken). 2012;64(11):1699–707. Epub 2012/06/08. 10.1002/acr.21747 . [DOI] [PubMed] [Google Scholar]

[pone.0229783.ref028] 28.Dobson F, Hinman RS, Roos EM, Abbott JH, Stratford P, Davis AM, et al. OARSI recommended performance-based tests to assess physical function in people diagnosed with hip or knee osteoarthritis. Osteoarthritis and cartilage / OARS, Osteoarthritis Research Society. 2013;21(8):1042–52. Epub 2013/05/18. 10.1016/j.joca.2013.05.002 . [DOI] [PubMed] [Google Scholar]

[pone.0229783.ref029] 29.Rolfson O, Wissig S, van Maasakkers L, Stowell C, Ackerman I, Ayers D, et al. Defining an International Standard Set of Outcome Measures for Patients With Hip or Knee Osteoarthritis: Consensus of the International Consortium for Health Outcomes Measurement Hip and Knee Osteoarthritis Working Group. Arthritis Care Res (Hoboken). 2016;68(11):1631–9. Epub 2016/10/27. 10.1002/acr.22868 [DOI] [PMC free article] [PubMed] [Google Scholar]

[pone.0229783.ref030] 30.Young JL, Rhon DI, Cleland JA, Snodgrass SJ. The Influence of Exercise Dosing on Outcomes in Patients With Knee Disorders: A Systematic Review. J Orthop Sports Phys Ther. 2018;48(3):146–61. Epub 2018/01/13. 10.2519/jospt.2018.7637 . [DOI] [PubMed] [Google Scholar]

[pone.0229783.ref031] 31.Nicolson PJA, Hinman RS, Kasza J, Bennell KL. Trajectories of adherence to home-based exercise programs among people with knee osteoarthritis. Osteoarthritis and cartilage / OARS, Osteoarthritis Research Society. 2018;26(4):513–21. Epub 2018/01/24. 10.1016/j.joca.2018.01.009 . [DOI] [PubMed] [Google Scholar]

[pone.0229783.ref032] 32.Lee AC, Harvey WF, Han X, Price LL, Driban JB, Bannuru RR, et al. Pain and functional trajectories in symptomatic knee osteoarthritis over up to 12 weeks of exercise exposure. Osteoarthritis Cartilage. 2018;26(4):501–12. Epub 2018/02/03. 10.1016/j.joca.2018.01.016 [DOI] [PMC free article] [PubMed] [Google Scholar]

[pone.0229783.ref033] 33.Edge R, Farrah K. Exercise for the Management of Knee Osteoarthritis: A Review of Clinical Effectiveness. CADTH Rapid Response Reports. Ottawa (ON)2017. [PubMed]

[pone.0229783.ref034] 34.Wellsandt E, Golightly Y. Exercise in the management of knee and hip osteoarthritis. Curr Opin Rheumatol. 2018;30(2):151–9. Epub 2017/12/19. 10.1097/BOR.0000000000000478 . [DOI] [PubMed] [Google Scholar]

PERMALINK

Improving osteoarthritis care by digital means - Effects of a digital self-management program after 24- or 48-weeks of treatment

Leif E Dahlberg

Andrea Dell'Isola

L Stefan Lohmander

Håkan Nero

Roles

Abstract

Background

Methods and findings

Conclusions

Introduction

Methods

Setting and participants

Intervention

Outcome measures

Statistical analysis

Role of the funding source

Results

Fig 1. Flowchart describing the disposition of patients.

24-week sub-sample

Table 1. Descriptive data for knee and hip OA in the 24-week sub-sample (n = 499).

Fig 2. Pain change over time, per index joint, for the 0 to 24-week subgroup of patients.

Fig 3. Joint pain change over 24 weeks stratified in tertiles by baseline pain severity for the 0 to 24-week subgroup of patients.

48-week sub-sample

Table 2. Descriptive data for knee and hip OA in the 48-week sub-sample (n = 138).

Fig 4. Pain change over time, per index joint, for the 0 to 48-week subgroup of patients.

Treatment effect over time

Table 3. Pain and physical function at baseline and follow-up.

Discussion

Supporting information

Acknowledgments

Data Availability

Funding Statement

References

Decision Letter 0

Kelly Naugle

Roles

Transfer Alert

Author response to Decision Letter 0

Decision Letter 1

Kelly Naugle

Roles

Acceptance letter

Kelly Naugle

Roles

Associated Data

Supplementary Materials

Data Availability Statement

ACTIONS

PERMALINK

RESOURCES

Similar articles

Cited by other articles

Links to NCBI Databases