Integrated Care Intervention Supported by a Mobile Health Tool for Patients Using Noninvasive Ventilation at Home: Randomized Controlled Trial.

Background Home-based noninvasive ventilation has proven cost-effective. But, adherence to therapy still constitutes a common clinical problem. We hypothesized that a behavioral intervention supported by a mobile health (mHealth) app could enhance patient self-efficacy. It is widely accepted that mHealth-supported services can enhance productive interactions among the stakeholders involved in home-based respiratory therapies. Objective This study aimed to measure changes in self-efficacy in patients with chronic respiratory failure due to diverse etiologies during a 3-month follow-up period after the intervention. Ancillary objectives were assessment of usability and acceptability of the mobile app as well as its potential contribution to collaborative work among stakeholders. Methods A single-blind, single-center, randomized controlled trial was conducted between February 2019 and June 2019 with 67 adult patients with chronic respiratory failure undergoing home-based noninvasive ventilation. In the intervention group, a psychologist delivered a face-to-face motivational intervention. Follow-up was supported by a mobile app that allowed patients to report the number of hours of daily noninvasive ventilation use and problems with the therapy. Advice was automatically delivered by the mobile app in case of a reported problem. The control group received usual care. The primary outcome was the change in the Self Efficacy in Sleep Apnea questionnaire score. Secondary outcomes included app usability, app acceptability, continuity of care, person-centered care, and ventilatory parameters. Results Self-efficacy was not significantly different in the intervention group after the intervention (before: mean 3.4, SD 0.6; after: mean 3.4, SD 0.5, P=.51). No changes were observed in adherence to therapy nor quality of life. Overall, the mHealth tool had a good usability score (mean 78 points) and high acceptance rate (mean score of 7.5/10 on a Likert scale). It was considered user-friendly (mean score of 8.2/10 on a Likert scale) and easy to use without assistance (mean score of 8.5/10 on a Likert scale). Patients also scored the perception of continuity of care and person-centered care as high. Conclusions The integrated care intervention supported by the mobile app did not improve patient self-management. However, the high acceptance of the mobile app might indicate potential for enhanced communication among stakeholders. The study identified key elements required for mHealth tools to provide effective support to collaborative work and personalized care. Trial Registration ClinicalTrials.gov NCT03932175; https://clinicaltrials.gov/ct2/show/NCT03932175


2a-i) Problem and the type of system/solution
"Despite its proven cost-effectiveness, patients' adherence to home-based NIV has still potential to improve, which should further enhance healthcare efficiencies of the intervention". "Nevertheless, improvement of behavioral aspects such as patient motivation and empowerment for self-management are important factors to consider when addressing adherence to respiratory therapies."."The current study seeks to explore the transfer of previous positive experiences on behavioral interventions in other fields (i.e. physical activity) into home-based NIV. Specifically, we will address the concept of selfefficacy,"."We propose the use of a behavioral mHealth intervention to support changes in self-efficacy,". "information and communication technologies (ICT) have been identified as promising to generate efficiencies by enhancing coordination between stakeholders and contributing to improve health outcomes [26,27]. Nonetheless, it is acknowledged that the scenario is not still mature [28]. Mainly, because of lacking evidence in real-world scenarios for the capacity of ICT to escort behavioral changes including self-efficacy in chronic complex patients."."The principal objective of the current study is to explore the capacity of a behavioral mHealth intervention to increase patient empowerment for self-management and adherence to therapy; whereas, the secondary aim is to learn, based on professionals' and patients' experience, how the mHealth tool should evolve to support collaborative work." 2a-ii) Scientific background, rationale: What is known about the (type of) system " Besides self-efficacy as a way to influence behavioral change, previous reports by Hernandez et al [24] and Cano et al [25] have identified two commonalities usually hindering effective implementation of complex respiratory therapies (i.e. long-term oxygen therapy, continuous positive airway pressure therapy, home NIV and home-based nebulizer therapy). Firstly, is the need for interaction and communication among several stakeholders, namely: health professionals at different healthcare tiers (primary care, specialized care, etc.), patients and carers, companies undertaking maintenance of the equipment, and others, which may greatly benefit from digital tools supporting collaborative work. Secondly, is the improvement in therapeutic adherence that should be achieved through patients' empowerment for self-management. " Does your paper address CONSORT subitem 2b?
"The principal objective of the current study is to explore the capacity of a behavioral mHealth intervention to increase patient empowerment for self-management and adherence to therapy; whereas, the secondary aim is to learn, based on professionals' and patients' experience, how the mHealth tool should evolve to support collaborative work." METHODS 3a) CONSORT: Description of trial design (such as parallel, factorial) including allocation ratio "A single-blinded, single center, randomized controlled trial with two parallel arms (1:1 ratio) was conducted" 3b) CONSORT: Important changes to methods after trial commencement (such as eligibility criteria), with reasons There were no changes in the methodology once the study started 3b-i) Bug fixes, Downtimes, Content Changes 4a) CONSORT: Eligibility criteria for participants "Inclusion criteria were defined as follows: all adult patients with hypercapnic ventilatory failure due to chest wall, neuromuscular, lung parenchyma and/ or airway disease already receiving treatment with NIV irrespective of treatment duration and having a mobile phone, tablet or personal computer that could support the use of the mobile health application (MyPathway®). MyPathway® [29] is a secure, digital communications channel connecting patients to clinicians and services. It is an app-based application for both patients and clinicians to use on phones and/or tablets. See Multimedia Appendix 1 for more details. Patients with severe psychiatric and/or neurological diseases where excluded, as well as those hospitalized at time of assessment. " 4a-i) Computer / Internet literacy 4a-ii) Open vs. closed, web-based vs. face-to-face assessments: "All the eligible patients were contacted by telephone in order to briefly explain the study and invite them to participate. Those showing interest were invited to the hospital outpatient clinics. Study investigators (EB, EA and MM) explained the study face-to-face and in case of acceptance, consent was signed". 4a-iii) Information giving during recruitment 4b) CONSORT: Settings and locations where the data were collected "At time of enrollment, semi-structured motivational interviews were conducted individually. Participants were asked about the following topics: (i) Treatment adaptation experience, (ii) Lifestyle (physical activity and food habits) and (iii) Use of information and communication technologies. In each session, field notes were taken anonymously, and no recordings were made. The intervention consisted of a 10 to 50-minutes face-to-face session at the hospital or at the participants' home". "During follow-up, the MyPathway® app was used by study participants for bi-directional interaction with the research team. It consisted of positive feedback or reinforcement messages in response to the number of hours of NIV use filled by the patient in a daily basis.". "A webbased clinical portal enabled the research team to monitor the NIV hours of use and clinical problems reported by the patients". "a dedicated nurse (one of the authors, MM) took the role of case manager, with clinical and technical knowledge, in order to support collaborative work. She used the web-based portal to identify adherence problems and, accordingly, she contacted the participants via telephone or at home (for those with severe mobility problems) in order to enquire about potential problems, either clinical or technical and solve them." 4b-i) Report if outcomes were (self-)assessed through online questionnaires "All assessments were done at baseline and at three months in the final visit programmed in the outpatient clinic for the control group. For the intervention group, the follow-up was done remotely, by the nurse case manager (MM), using the MyPathway app® and its clinical portal. When deemed necessary, the nurse case manager visited the patient at home or a visit was programmed at the outpatient clinics. There was no active follow-up for the control group." 4b-ii) Report how institutional affiliations are displayed 5) CONSORT: Describe the interventions for each group with sufficient details to allow replication, including how and when they were actually administered 5-i) Mention names, credential, affiliations of the developers, sponsors, and owners 5-ii) Describe the history/development process 5-iii) Revisions and updating 5-iv) Quality assurance methods 5-v) Ensure replicability by publishing the source code, and/or providing screenshots/screen-capture video, and/or providing flowcharts of the algorithms used

5-vii) Access
"Free access was granted after receiving an invitation via the hospital Health Information System (SAP®), which prompted the participant to register using an email address as a username. The application could also be downloaded to the carers' phone in case the patient did not have a smartphone " 5-viii) Mode of delivery, features/functionalities/components of the intervention and comparator, and the theoretical framework "At time of enrollment, semi-structured motivational interviews were conducted individually. Participants were asked about the following topics: (i) Treatment adaptation experience, (ii) Lifestyle (physical activity and food habits) and (iii) Use of information and communication technologies. In each session, field notes were taken anonymously, and no recordings were made. The intervention consisted of a 10 to 50-minutes face-to-face session at the hospital or at the participants' home, following the principles of collaborative and evocative motivational interview [27][28][29][30], favoring the participant's autonomy. The techniques used were open questions, active listening, empathy, returning reflected thoughts, exploring change in goals, summarizing and giving feedback. Also during the enrollment visit, patients were given verbal and written explanation on how to use the app". "During follow-up, the MyPathway® app was used by study participants for bi-directional interaction with the research team. It consisted of positive feedback or reinforcement messages in response to the number of hours of NIV use filled by the patient in a daily basis. In addition, general advice on specific NIV clinical problems was automatically provided by the app according to patients' weekly input. Additional educational material on physical activity, diet and sleep hygiene could be accessed at any time from a dedicated link". See also Multimedia appendix 1 for more technical details 5-ix) Describe use parameters 5-x) Clarify the level of human involvement 5-xi) Report any prompts/reminders used "the MyPathway® app was used by study participants for bi-directional interaction with the research team. It consisted of positive feedback or reinforcement messages in response to the number of hours of NIV use filled by the patient in a daily basis" after getting an SMS remainder 5-xii) Describe any co-interventions (incl. training/support) "The behavioral mHealth intervention assessed in the current manuscript included: i) a face-to-face motivational interview by a psychologist (EA) to assess patient's adherence profile and lifestyle habits;" 6a) CONSORT: Completely defined pre-specified primary and secondary outcome measures, including how and when they were assessed "The primary outcome was a change in self-efficacy measured by the Self Efficacy in Sleep apnea (SEMSA) questionnaire. The SEMSA is a US-designed self-report questionnaire comprising 26 items rated from 1 to 4 on a 4-point Likert scale. The arithmetic mean of the Likert rating for each participant is computed for the overall SEMSA score and for each of the three factors. The total score ranges from 1 to 4. Higher scores indicate greater risk perception, higher benefit expectancy with treatment and greater perceived self-efficacy. Secondary outcomes included: i) usability of the ICT tool measured by the System Usability Scale; ii) patient satisfaction measured using the Net Promoter Score alongside three custom made general satisfaction questions using a Likert scale; iii) continuity of care using the Nijmegen continuity of care questionnaire; and, iv) the Person centered coordinated experience questionnaire as described by Leijten et al. Moreover, ventilator-specific data such as mean hours of use per day, unintentional leaks (L/s), minute ventilation (L/min), tidal volume (mL) and back-up rate (breaths/min) were directly downloaded from the NIV machine. Tertiary outcomes included mortality, health-related quality of life (using the EuroQol 5D questionnaire and sleepiness (using the Epworth Sleepiness Score). The impact of the motivational mHealth tool recommendations on diet and exercise was indirectly measured by body weight changes.
All assessments were done at baseline and at three months in the final visit programmed in the outpatient clinic" 6a-i) Online questionnaires: describe if they were validated for online use and apply CHERRIES items to describe how the questionnaires were designed/deployed 6a-ii) Describe whether and how "use" (including intensity of use/dosage) was defined/measured/monitored 6a-iii) Describe whether, how, and when qualitative feedback from participants was obtained 6b) CONSORT: Any changes to trial outcomes after the trial commenced, with reasons "At time of enrollment, semi-structured motivational interviews were conducted individually. Participants were asked about the following topics: (i) Treatment adaptation experience, (ii) Lifestyle (physical activity and food habits) and (iii) Use of information and communication technologies. In each session, field notes were taken anonymously, and no recordings were made. The intervention consisted of a 10 to 50-minutes face-to-face session at the hospital or at the participants' home". "During follow-up, the MyPathway® app was used by study participants for bi-directional interaction with the research team. It consisted of positive feedback or reinforcement messages in response to the number of hours of NIV use filled by the patient in a daily basis.". "A webbased clinical portal enabled the research team to monitor the NIV hours of use and clinical problems reported by the patients". "a dedicated nurse (one of the authors, MM) took the role of case manager, with clinical and technical knowledge, in order to support collaborative work. She used the web-based portal to identify adherence problems and, accordingly, she contacted the participants via telephone or at home (for those with severe mobility problems) in order to enquire about potential problems, either clinical or technical and solve them." 7a) CONSORT: How sample size was determined 7a-i) Describe whether and how expected attrition was taken into account when calculating the sample size 7b) CONSORT: When applicable, explanation of any interim analyses and stopping guidelines "The primary outcome was a change in self-efficacy measured by the Self Efficacy in Sleep apnea (SEMSA) questionnaire. The SEMSA is a US-designed self-report questionnaire comprising 26 items rated from 1 to 4 on a 4-point Likert scale. The arithmetic mean of the Likert rating for each participant is computed for the overall SEMSA score and for each of the three factors. The total score ranges from 1 to 4. Higher scores indicate greater risk perception, higher benefit expectancy with treatment and greater perceived self-efficacy. Secondary outcomes included: i) usability of the ICT tool measured by the System Usability Scale; ii) patient satisfaction measured using the Net Promoter Score alongside three custom made general satisfaction questions using a Likert scale; iii) continuity of care using the Nijmegen continuity of care questionnaire; and, iv) the Person centered coordinated experience questionnaire as described by Leijten et al. Moreover, ventilator-specific data such as mean hours of use per day, unintentional leaks (L/s), minute ventilation (L/min), tidal volume (mL) and back-up rate (breaths/min) were directly downloaded from the NIV machine. Tertiary outcomes included mortality, health-related quality of life (using the EuroQol 5D questionnaire and sleepiness (using the Epworth Sleepiness Score). The impact of the motivational mHealth tool recommendations on diet and exercise was indirectly measured by body weight changes.
All assessments were done at baseline and at three months in the final visit programmed in the outpatient clinic" 8a) CONSORT: Method used to generate the random allocation sequence "The randomization scheme was generated by using the website Randomization.com by one of the researchers (EB), prior to patient enrollment. Blocks of 4 were used. Only after the participant signed the informed consent, the investigator opened the envelope with the allocated study group." 8b) CONSORT: Type of randomisation; details of any restriction (such as blocking and block size) "The randomization scheme was generated by using the website Randomization.com by one of the researchers (EB), prior to patient enrollment. Blocks of 4 were used. Only after the participant signed the informed consent, the investigator opened the envelope with the allocated study group." 9) CONSORT: Mechanism used to implement the random allocation sequence (such as sequentially numbered containers), describing any steps taken to conceal the sequence until interventions were assigned "The randomization scheme was generated by using the website Randomization.com by one of the researchers (EB), prior to patient enrollment. Blocks of 4 were used. Only after the participant signed the informed consent, the investigator opened the envelope with the allocated study group." 10) CONSORT: Who generated the random allocation sequence, who enrolled participants, and who assigned participants to interventions "The randomization scheme was generated by using the website Randomization.com by one of the researchers (EB), prior to patient enrollment. Blocks of 4 were used. Only after the participant signed the informed consent, the investigator opened the envelope with the allocated study group." 11a) CONSORT: Blinding -If done, who was blinded after assignment to interventions (for example, participants, care providers, those assessing outcomes) and how 11a-i) Specify who was blinded, and who wasn't "Due to the nature of the intervention, neither the participants nor the investigators in direct contact with them were blinded. Only the investigator in charge of data analysis was blinded." 11a-ii) Discuss e.g., whether participants knew which intervention was the "intervention of interest" and which one was the "comparator" 11b) CONSORT: If relevant, description of the similarity of interventions Not relevant 12a) CONSORT: Statistical methods used to compare groups for primary and secondary outcomes "Accepting an alpha risk of 0.05 and a beta risk of 0.2 in a two-sided test, 31 subjects were necessary in the intervention group and 31 in the control group to recognize as statistically significant a difference greater than or equal to 0.35 units in the SEMSA overall score. The common standard deviation was assumed to be 0.46 [37]. It was anticipated a drop-out rate of 10%. Baseline and end-of-study data (questionnaires) were collected face-to-face at the outpatient clinic by the investigators (EB, EA and MM). Study data were collected and managed using the REDCap electronic case report form [38,39] hosted at Hospital Clínic of Barcelona. Data on NIV use and clinical problems with NIV was collected on-line during the study period as reported by the participants using MyPathway®. Results are presented as mean (SD) or n (%) when indicated. Comparisons were done using chi-square or Fisher exact tests for categorical variables, and Student' or Wilcoxon tests, depending on the distribution of the variables, for numerical variables." 12a-i) Imputation techniques to deal with attrition / missing values Attrition rate expected was very low since all were long-term patients in the clinical service, known well by the physcians participating in the study. This was reflected by the fact that only 1 patient in the intervention group discontinued due to deteriorating health condition not related to the intervention or respiratory pathology but from a surgical complication.

12b) CONSORT: Methods for additional analyses, such as subgroup analyses and adjusted analyses
We did not do any additional analysis RESULTS 13a) CONSORT: For each group, the numbers of participants who were randomly assigned, received intended treatment, and were analysed for the primary outcome As seen in the CONSORT flow diagram all patients in the intervention group (except for one drop-out) downloaded the app and used it. All received face-toface motivational interview at enrolment. All were actively followed by the dedicated nurse using the web portal, and if deemed necessary, phone contact and/or home visit. 13b) CONSORT: For each group, losses and exclusions after randomisation, together with reasons In Multimedia Appendix 3 -CONSORT flow diagram the exclusion and reasons are stated. In the control group there were no loses. In the intervention group only one lost due to deteriorating health condition.  Table 1 and Multimedia Appendix 2."

15-i) Report demographics associated with digital divide issues
In table 1 and multimedia appendix 2, the following demographics are reported: sex, gender, educational level 16a) CONSORT: For each group, number of participants (denominator) included in each analysis and whether the analysis was by original assigned groups 16-i) Report multiple "denominators" and provide definitions In multimedia appendix 2, "NIV usage reported by patients using MyPathway" details the use of the app for reporting and "MyPathway logobook analysis" report the absolute and relative numbers for different aspects of the usage.

16-ii) Primary analysis should be intent-to-treat
17a) CONSORT: For each primary and secondary outcome, results for each group, and the estimated effect size and its precision (such as 95% confidence interval) "For the primary outcome, there was no statistically significant difference after intervention in the SEMSA score for self-efficacy (mean[SD]=3.4[0.6] vs 3.4 [0.5], P=.51). For the perceived risks, outcome expectancies, Epworth Sleepiness Score and EuroQol 5Q-5D questionnaires, there were no statistically significant differences neither (see Multimedia Appendix 2). As for the patient experience questionnaires, neither the Nijmegen continuity of care questionnaire nor the Person centred coordinated experience questionnaire showed statistically significant differences between groups (see Multimedia Appendix 2)". "Adherence

20) CONSORT: Trial limitations, addressing sources of potential bias, imprecision, multiplicity of analyses 20-i) Typical limitations in ehealth trials
"We do acknowledge that by using an already existing app, the co-design phase was skipped. Also we did not measure technological literacy in our older population (average age 69 years)". "a clear limitation of our study was the exclusion of new NIV patients, were the behavioral intervention may have had more impact" 21) CONSORT: Generalisability (external validity, applicability) of the trial findings 21-i) Generalizability to other populations "Our study considered the whole population of patients attending the clinic, making it a realistic clinical scenario" 21-ii) Discuss if there were elements in the RCT that would be different in a routine application setting 22) CONSORT: Interpretation consistent with results, balancing benefits and harms, and considering other relevant evidence 22-i) Restate study questions and summarize the answers suggested by the data, starting with primary outcomes and process outcomes (use) "We report the results of a behavioral mHealth intervention based on a face-to-face interview and the use of a mHealth tool (MyPathway® app), during a follow-up period of three months, on patients with hypercapnic chronic respiratory failure under home-based long-term noninvasive ventilation. To the best of our knowledge, this is the first randomized controlled trial using digital tools to support behavioral changes in this population"."In the current study, the self-efficacy mean score was already high at baseline (Table 1) and we did not find any significant effect on behavioral changes after the intervention". "Notwithstanding the clinical results, it is important to note that the mHealth tool was well appreciated by the patients and their family/caregivers. "."In this respect, we measured two process outcomes [50] related to patient experience [33], i.e. continuity of care and person-centered care. Both parameters were very well evaluated by all of our study population, which included not only patients, but also their family and carers in a third of cases in the intervention group.