key: cord-0753045-v93lk52a authors: Gromisch, Elizabeth S; Turner, Aaron P; Haselkorn, Jodie K; Lo, Albert C; Agresta, Thomas title: Mobile health (mHealth) usage, barriers, and technological considerations in persons with multiple sclerosis: a literature review date: 2020-12-15 journal: JAMIA Open DOI: 10.1093/jamiaopen/ooaa067 sha: 299be69a1f509e55c8eeed247ecb5314bee64cef doc_id: 753045 cord_uid: v93lk52a OBJECTIVES: Persons with multiple sclerosis (MS) can face a number of potential healthcare-related barriers, for which mobile health (mHealth) technology can be potentially beneficial. This review aimed to understand the frequency, current uses, and potential barriers with mHealth usage among persons with MS. METHODS: A query string was used to identify articles on PubMed, MEDLINE, CINAHL, and IEEE Xplore that were published in English between January 2010 and December 2019. Abstracts were reviewed and selected based on a priori inclusion and exclusion criteria. Fifty-nine peer-reviewed research studies related to the study questions are summarized. RESULTS: The majority of persons with MS were reported as using smartphones, although rates of mHealth utilization varied widely. mHealth usage was grouped into 3 broad categories: (1) disability and symptom measurement; (2) interventions and symptom management; and (3) tracking and promoting adherence. While there have been an increasing number of mHealth options, certain limitations associated with MS (eg, poor dexterity, memory problems) may affect usage, although including persons with MS in the design process can address some of these issues. DISCUSSION: Given the increased attention to mHealth in this population and the current need for telehealth and at home devices, it is important that persons with MS and healthcare providers are involved in the development of new mHealth tools to ensure that the end product meets their needs. Considerations for addressing the potential mHealth use barriers in persons with MS are discussed. Up to 1 million people in the United States are estimated to have multiple sclerosis (MS), a chronic neurological disorder. 1 Persons with MS can present with a wide array of symptoms, including difficulties with ambulation, cognition, vision, fine motor abilities, and fatigue. Effectively managing these symptoms and having access to appropriate healthcare services to address their MS-related needs can be challenging. For instance, cognitive dysfunction, which can affect up to 70% of persons with MS, can be progressive in nature and has been associated with greater difficulty completing daily activities, including functional tasks such as medication management, bill paying, decreased social engagement, and poorer health-related quality of life. [2] [3] [4] [5] [6] During the COVID-19 pandemic, persons with MS may not be able to get routine appointments because they are not considered urgent enough for face-to-face visits. In addition, persons with MS can face a number of healthcare-related barriers, including health insurance coverage, transportation, and accessibility. 7 Because of the potential barriers to healthcare that persons with MS can face, one option for improving access to care has been through mobile health (mHealth) technology. The National Institutes of Health (NIH) defines mHealth as "the use of mobile and wireless devices (cellphones, tablets, [wearable devices such as a smartwatch], etc.) to improve health outcomes, healthcare services, and health research." 8 Many persons with MS have access to mobile technology, with one study noting that over 86% use a smartphone, tablet, or both. 9 While technology holds promise to offer persons with MS alternate methods for tracking and managing symptoms, communicating with their healthcare providers, and improving their health-related quality of life, it is unclear if available mHealth tools are meeting the needs of their intended consumer. For instance, while Salimzadeh et al. 10 found 104 MS-related applications in iTunes and Google Play, they noted that there was no corresponding evidence regarding the usability and utility of these applications in persons with MS. This literature review aimed to gain a better understanding of mHealth usage among persons with MS by addressing the following 3 questions: (1) how frequently do persons with MS use mHealth; (2) what are the current uses of mHealth within the MS population, based on the literature; and (3) what are the potential barriers and technological considerations that may affect persons' with MS usage of mHealth. (2) between January 2010 and December 2019, resulting in 623 abstracts ( Figure 1 ). One additional publication that met inclusion criteria was identified through ancestral review of the references and added to the total. After removing duplicate entries (n ¼ 140), literature reviews, conference abstracts, protocols, letters to the editor, and corrections (n ¼ 121) were excluded. The titles and abstracts were then screened by the primary author, with another 180 studies removed due to them not being about persons with MS or mHealth. The remaining 183 articles were assessed by the primary author to evaluate whether they explicitly referred to mobile technology and were related to the study questions. A cross-check second review was not done. If participants could access an intervention, assessment, or symptom management tool with either a mobile device (eg, smartphone or tablet) or another electronic device (eg, a laptop or desktop computer), the study could be included in the review as long as it was noted in the methods or results section that some of the participants did use a mobile device. A total of 59 studies were included. For articles that addressed the second and third study questions, the uses of mHealth, barriers to mHealth usage, and technological considerations were coded by the primary author, and then reviewed by all members of the research team to identify themes. As this was a review of published literature, no ethical approvals were required. Regular smartphone use was noted to be common among persons with MS, with 86.9% reporting in one study that they use the device at least once a day. 11 Engagement in physical activity, greater income, having 3 or more co-occurring conditions, female gender, and being unmarried have been associated with an increased odds of smartphone usage among persons with MS. 9 Usage has been noted to be lower in older adults, 12 as well as persons with MS who smoke or have greater levels of disability. 9 Even though smartphone usage was high, a German-based survey found that most persons with MS (63%) have no experience using mHealth applications for their MS, with only 18% being current users of this technology. 13 Marrie et al. 9 noted that 46.2% of smart- Mobile health (mHealth) has the possibility to improve access to care for persons with multiple sclerosis (MS). This literature review aimed to understand the frequency and types of mHealth use, as well as any barriers to use, by persons with MS. While use of devices like smartphones were common, the use of mHealth applications varied. The different types of mHealth tools identified included symptom measurement (eg, level of physical disability), symptom management (eg, improving fatigue), and treatment adherence (eg, reminders to take medications). A number of potential barriers were identified, including ones related to MS symptoms (eg, difficulties with vision, fine motor control (or alternately dexterity), memory), which can be addressed by including patients and their providers in the design process. phone and tablet users, who were based in the United States, endorsed using a mHealth application at least once. Higher education, income, number of co-occurring conditions, and engagement in physical activity have been associated with greater likelihood of mHealth application use. 9 As with smartphone usage, general selfreported mHealth application use decreased with older age 9 ; however, that trend may differ with certain mHealth tools. For instance, Merlo et al. 14 found that older persons with MS were more likely to persist on repeat testing using the computerized MSReactor program. Disability and symptom measurement Several mHealth tools have focused on the measurement of persons' with MS symptoms and level of disability (Table 1) . A number of applications, both mHealth-based and web-based that are accessible with a mobile device, have included patient-reported outcomes as a feature, such as questionnaires [15] [16] [17] [18] [19] [20] and visual analogue scales. 21 Other efforts have focused on the use of mHealth tools to conduct objective measurements, such as remote Expanded Disability Status Scale (EDSS) evaluations using tele-video. 22 While there are several computer-based cognitive assessments used for persons with MS, [23] [24] [25] [26] there have been 2 studies 14,27 investigating multi-domain evaluations that could be conducted using a tablet, as well as one tabletbased application focusing on processing speed that is included as part of a composite disability assessment. 28 Smartphone-based suites have also included one or more measures of objective cognitive functioning. 15, 19 With mHealth-based cognitive assessments, there is an ongoing debate as to whether a technician needs to be present. Wojcik et al. 29 found largely similar performances on computerized assessments with and without a technician present, although participants were aware there was one nearby should problems with test administration arise. Smartphone-or tablet-based cognitive assessments have also been included in mHealth disability composite measures. There have been at least 2 mHealth programs 30-33 based on the MS Functional Composite (MSFC), which traditionally consists of the Paced Auditory Serial Addition Test (processing speed), Nine Hole Peg Test (manual dexterity), and 25-Foot Walk (walking speed), 44 although there have been modifications such as including a measure of low contrast visual acuity 45 and using the Symbol Digit Modalities Test instead as the cognition measure. 46 Both mHealth programs assessed 4 domains of functioning (walking speed, manual dexterity, low contrast visual acuity, and processing speed), with the MS Performance Test also including a balance test. [30] [31] [32] [33] In addition to being part of composite measures, there have been standalone applications examining visual acuity 34 and fine motor abilities. 35, 36 Given that ambulation is a significant component of disability measurements such as the MSFC, 44 EDSS, 47 and Patient Determined Disease Steps, 48-51 several studies [37] [38] [39] [40] have used triaxial accelerometers as a way of measuring the level of ambulationbased disability. Furthermore, objective step counts and activity monitoring may also provide more accurate representations of functional ability compared to patient reports. [41] [42] [43] Interventions and symptom management Using mHealth as an interventional or symptom management tool has become more common (Table 2) , with persons with MS noting that they use mHealth to improve their cognition, manage their stress and mood, and monitor their physical activity and diet. 13, 52 In 19 • Newland et al. 20 Visual Analogue Scales • Feasible to administer visual analogue scales for anxiety, fatigue, pain, and quality of life on a smartphone or tablet • Tablet-based administration did not have higher reliability, which may have been partially due to the exclusion of persons with MS with more significant levels of impairment Remote disability assessments Modified Tele-EDSS Evaluation a • Patient received an "in-home neuro kit" and followed verbal instructions from an examiner via video chat (73% completed with a smartphone and 15% with a tablet) • High level of acceptance • Good correlations between in-clinic and tele-EDSS evaluations, particularly for individuals with higher EDSS scores the literature, at least 2 mHealth-based cognitive training programs have been examined, 53,54 which were well-received by persons with MS. In addition, text messages, received through either a mobile phone or pager, have been explored as a compensatory strategy. 55 Besides using wearable devices to track physical activity via methods such as step counts (see Table 1 ), mHealth has been used as an option to deliver web-based physiotherapy programs. 56, 57 As fatigue is a common and often disabling symptom of MS, 58, 59 there have also been 4 mHealth-based applications that promote fatigue selfmanagement through various strategies, including cognitive behavioral therapy principles, 60 tele-monitoring and tele-coaching, 61 gamification, 62 and self-assessments and self-monitoring. 18, 63 Tracking and promoting adherence Another common use within the MS population has been tracking and promoting adherence: about 44% of persons with MS in one survey noted that they use mHealth to remind them about their medications or upcoming appointments. 13 Similarly, 37% of the commercially available MS-related self-management applications focused on medication management. 10 Besides using applications to communicate with their clinicians about their care, persons with MS have reported using text messages, although they noted using secure online portals and email more frequently than texts or applications. 9 Some persons with MS have opted to have text message reminders sent to their mobile phones (vs via email or through a home monitoring device) to take their disease modifying therapy (DMT) 64 or engage in physical activity. 65 Text messages have also been incorporated, along with phone calls and emails, as part of a telemedicinebased patient support program. 66 When given the choice, more than half of persons with MS will choose a digital diary over a paper diary to monitor their DMT adherence. 67 In addition, using a digital diary with reminders has been associated with a reduced risk of suboptimal adherence. 68 Tools such as MSdialog (web-and mobile-based) 17 and PatientConcept (mobile-based) 69 has been shown to help track DMT usage, and include other features that may promote adherence, such as health reports via patient-reported outcome measures, reminders, and appointment requests. For both applications, persons with MS have endorsed that they were useful in helping them communicate with their healthcare provider. It should be noted that several of the mHealth tools that have been developed for monitoring adherence are associated with specific DMTs, such as MSdialog and interferon beta-1a. 17 While mHealth is being used by persons with MS in a variety of contexts and for a variety of purposes, there are a number of potential barriers that may inhibit its use (Table 3) . Although any limitation in function may affect mHealth usage for persons with MS, difficulties related to vision, fine motor dexterity, and cognitive functioning may be more salient, depending on the MS presentation, severity, and course of progression. For instance, persistent visual difficulties are common in MS, 70 and as such, individuals with this symptom may struggle to use mHealth tools with small or hard-to-read text. 62, 71 Allowing for verbal cues or using larger text and buttons 11,72,73 may help compensate for these issues. Coupled with sensory and/or fine motor issues, 74 the size of the mobile device needs to be considered, particularly with applications with more complex components (eg, longer questionnaires). 18 In addition, given that up to 70% of persons with MS experience cognitive impairment, 2 including reminders in the application may be a valuable addition and assist with adherence to a mHealth program. 75, 76 Several of the problems identified in the literature are not unique to persons with MS. For instance, confusing interfaces have been noted to make it difficult for persons with MS to adopt mHealth tools, 63,77 which could influence mHealth usage in a variety of users. Other problems, such as cognitive impairment affecting users' abilities to remember tasks related to the mHealth tool, 41, 42 can occur in other chronic health conditions. Some of these potential issues can be addressed in the design process, such as allowing users to retake a timed task if something unforeseen interferes with their performance. 35 Furthermore, if persons with MS are included throughout the design process, potential limitations can be identified and corrected earlier in development. However, other barriers such as limited cellular network coverage and disruptions in the signal, 41,61 may be out of developers' control. Given the healthcare-related challenges that persons with MS can face 7 and the growing need for telehealth, mHealth offers an important option to access needed services and resources. However, in order for mHealth to be meaningful to persons with MS, it needs to be usable, useful, and satisfying. This literature review aimed to better understand persons' with MS use of mHealth and the potential barriers. Overall, the majority of persons with MS use devices like smartphones on a regular basis. Although rates of mHealth utiliza- Described as a "web-based program," but only access with a mobile device was noted. tion varied, it appears to be more common in the United States compared to parts of Europe, based on the reported data. 9, 13 Some of the factors noted to be associated with mHealth usage (eg, level of education, income, age) 9 may be related to some of the barriers identified in the literature. For instance, older adults may be less likely to use mHealth because of limited digital literacy. 52 Clinicians and researchers working with older persons with MS should consider providing a training session with the device or application to help encourage mHealth usage. 21, 56 The use of mHealth as a component of MS-related care has grown over the past decade, with increased sophistication and options for end users. For instance, while earlier work investigated the use of a personal digital assistant (PDA) as a compensatory tool (eg, setting alarms and reminders), 83 less than a decade later there are standalone, mHealth-based cognitive training programs that involve domain-specific exercises, 53, 54 allowing for persons with MS to engage in active training whenever and wherever it is convenient for them. The adoption of mobile devices as part of everyday life has also been reflected in individuals needing fewer instruments to engage in technology-based interventions. Rather than needing a home monitoring device connected through a landline to receive reminders, persons with MS can elect to get them sent directly to their cell phones. 65, 84 While several of the identified applications were web-based and could be accessed via a mobile or non-mobile device, 14, 16, 17, 22, 56, 57 a number of applications were designed specif-ically for smartphones, 15, 19, 20, 31, 35, 36, [60] [61] [62] tablets, 21, 27, 28, 30, [32] [33] [34] 53 or both devices. 54, 69 In terms of current uses of mHealth in the MS population, both mHealth-based applications and web-based applications that can be accessed with a mobile device, options ranged from assessment tools used by clinicians to quantify disability to programs that can help persons with MS improve their working memory. Many of these mHealth tools have been developed recently, demonstrating the increased attention and availability of technology in MS-related care. Of the 21 studies that discussed disability and symptom measurement, 72% (n ¼ 18) were initially published between 2017 and 2019, with half of those in 2019 alone. Similarly, 60% (n ¼ 6) of the studies detailing intervention and symptom management mHealth tools were published in 2018 and 2019. Furthermore, smartphoneand tablet-specific programs have become more common, with almost two-thirds of the studies (n ¼ 14) examining them initially published between 2018 and 2019. Although many of the mHealth applications focused on the provider (eg, disability evaluations), several of the identified tools can be used by persons with MS for tracking and improving their symptoms. While there are a growing number of mHealth options for persons with MS, several potential barriers have been identified that might negatively influence the adoption of mHealth. Although the symptom presentation varies from person to person, many persons with MS experience cognitive, sensory, and/or physical difficulties, NeuroPage • Participants could receive reminder messages via their mobile phone or a pager • Associated with reduced emotional distress and better recall of specific, intended tasks • Goodwin et al. 55 Physical activity Web-based Physiotherapy a • Program could be accessed through a tablet or a computer • Several benefits for persons with MS, including being able to engage in the program based on their schedules and not needed to travel • Improvement in self-reported physical impact of MS • 40% were still adherent at 6 months • Paul et al. 56 • Paul et al. 57 Fatigue selfmanagement • Developed using cognitive behavioral principles (eg, how thoughts and behaviors influence MS-related fatigue) • Babbage et al. 60 • Used a combination of tele-monitoring (utilizing the integrated accelerometers and having participants report their fatigue impact levels) and tele-coaching (goal setting and motivational messages) • D'hooghe et al. 61 More Stamina • Used gamification to promote behavior change related to fatigue management (eg, awarding points and achievements) • Giunti et al. 62 MSmonitor b • Used self-assessments (eg, questionnaires and inventories) and self-monitoring (eg, activity diaries) • Modest negative correlation between how frequently persons with MS were using the diary option and their level of fatigue impact • 46% of persons with MS using the application reported that they had an improved understanding of their symptoms Described as a "web-based program," but only access with a mobile device was noted. which can impede their usage of certain mHealth tools. 52, 62, 71, 74 Some barriers, such as digital literacy, costs, and interface design can apply to mHealth in general. Even some of the aforementioned disease-related barriers and their associated considerations, such as including reminders or adjusting the text size, 11, 72, 73, 75, 76 can apply to other populations. A number of studies included participants with other conditions besides MS, 41, 42, 74, 78, 80, 82 suggesting that these barriers need to be considered when designing mHealth for a wide variety of patient users. Some of these issues, such as factoring in the physical manifestations of MS, highlight the need to use user-centered design processes 85 during the development of new mHealth tools for persons with MS. Including persons with MS, as well as healthcare providers, during the design process can elucidate potential diseaserelated limitations and their solutions prior to the product being tested commercially. For instance, if a mHealth tool involves physical activity where falls or a cardiac event may be a possibility, a healthcare provider might bring up the need to include an emergency contact, while a person with MS may suggest ways of handling potential societal barriers. Furthermore, involving persons with MS throughout the development of new mHealth applications, particularly in the early stages, can increase the likelihood that these tools are meeting the needs of its end users and will be implemented in everyday life. It is important to note that while these procedures were largely used in the included studies, this may not always be the case in commercially available applications, as noted in Salimzadeh et al. review. 10 With the recent finalization by the Center for Medicare and Medicaid Services of the Interoperability and Patient Access Rule, 86 required as part of the 21st Century Cures Act, 87 all certificated Health Information Technology (IT) systems (electronic health records and Insurance IT systems) will be required to use a standard Application Programming Interface to allow developers to create mHealth tools for persons with MS that interoperate with their clinicians' systems. This will open up a broad array of new opportunities to develop new tools that meet specific needs. It would be imperative to consider the lessons learned already, as demonstrated by this literature review, to rapidly develop the most effective, user-friendly mHealth tools possible. Several caveats should be noted in the current review. Given that the review was limited to English language articles, studies published in other languages were not included. As such, mHealth usage may be different in other cultures and communities, potentially over-estimating or under-estimating the frequency of mHealth usage among persons with MS, and other types of mHealth applications and barriers may have been omitted from this review. In addition, as the current study was a literature review rather than a metaanalysis, the efficacy of these mHealth tools were not examined. As the number of mHealth tools available for persons with MS continue to grow, particularly interventional applications, the efficacy of technology-delivered programs compared to those delivered faceto-face will need to be closely examined to ensure patients are receiving efficacious services, even at a distance. Given the rapid development of telehealth services in response to COVID-19, there will likely be a plethora of new literature on mHealth's efficacy and effectiveness, including optimal delivery mechanisms and its impact on quality of life. Finally, as it was beyond the scope of the current review, other factors that may influence the utility of some of these mHealth tools (eg, costs, frequency of updates, and reliability) were not explored. In conclusion, the majority of persons with MS have access to smart devices, with several individuals using them for mHealth. While there has been an increasing number of mHealth options for persons with MS, certain physical, cognitive, and technological barriers may affect usage. As such, it is important that persons with MS are involved in the design and testing stages of mHealth development to ensure that the end product meets theirs and their healthcare providers' needs. ESG made substantial contributions to the conception, design, acquisition, and drafting of the literature review. All authors made substantial contributions to the interpretation of the data. APT, JKH, ACL, and TA made critical revisions of the manuscript. All authors provided final approval and agree to be accountable for all aspects of this work. 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ONC's Cures Act Final Rule The authors wish to thank Joe Pallis for his assistance with refining the PubMed query string, and Steven Demurjian for his review of an earlier draft of this manuscript. None declared. No new data were generated or analyzed in support of this research.