Benefits and Disadvantages of Electronic Patient-reported Outcome Measures: Systematic Review

Background Patient-reported outcome measures (PROMs) are important in clinical practice and research. The growth of electronic health technologies provides unprecedented opportunities to systematically collect information via PROMs. Objective The aim of this study was to provide an objective and comprehensive overview of the benefits, barriers, and disadvantages of the digital collection of qualitative electronic patient-reported outcome measures (ePROMs). Methods We performed a systematic review of articles retrieved from PubMED and Web of Science. The Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines were followed during all stages. The search strategy yielded a total of 2333 records, from which 32 met the predefined inclusion and exclusion criteria. The relevant ePROM-related information was extracted from each study. Results Results were clustered as benefits and disadvantages. Reported benefits of ePROMs were greater patient preference and acceptability, lower costs, similar or faster completion time, higher data quality and response rates, and facilitated symptom management and patient-clinician communication. Tablets were the most used ePROM modality (14/32, 44%), and, as a platform, Web-based systems were used the most (26/32, 81%). Potential disadvantages of ePROMs include privacy protection, a possible large initial financial investment, and exclusion of certain populations or the “digital divide.” Conclusions In conclusion, ePROMs offer many advantages over paper-based collection of patient-reported outcomes. Overall, ePROMs are preferred over paper-based methods, improve data quality, result in similar or faster completion time, decrease costs, and facilitate clinical decision making and symptom management. Disadvantages regarding ePROMs have been outlined, and suggestions are provided to overcome the barriers. We provide a path forward for researchers and clinicians interested in implementing ePROMs. Trial Registration PROSPERO CRD42018094795; https://www.crd.york.ac.uk/prospero/display_record.php?RecordID=94795


Introduction
In patient-centered care, patient-reported outcome measures (PROMs) are the gold standard for efficiently evaluating patients' feelings, thoughts, and complaints about a clinical intervention or disease [1].
Clinicians use PROMs to guide and audit routine care and support patient-centered care. Standard intake procedures already include many questionnaires such as generic quality of life questionnaires administered before arthroplastic surgeries [2]. At the patient level, the data can be used to monitor individual progress, investigate the effects of medical and surgical interventions [2], and improve communication between patients and caregivers [3]. On a larger scale, PROM data can be used to screen for health problems, compare outcomes between populations, and assess quality of care. They are widely implemented in clinical research [1,4], with positive effects on patient-clinician communication and mutual decision making. PROMs are traditionally measured using pen-and-paper questionnaires. We aimed to investigate whether pen-and-paper methods are the best option because unsupervised paper-based PROM data collection in clinical trials has resulted in unreadable, missing, or faulty data [5].
The growth of electronic health (eHealth) technologies provide unprecedented opportunities to systematically collect information via PROMs. Patients of all ages and sociodemographic backgrounds worldwide are comfortable using digital networks and services [6]. Furthermore, smartphones and lightweight computers or tablets with touchscreens are omnipresent. Supposed advantages of electronic PROMs (ePROMs) include more complete data capture and lower cost but it is unknown if the advantages of ePROM outweigh the disadvantages. Various research groups in different medical fields have investigated the use of electronic questionnaires in different patient groups; however, the benefits and disadvantages of ePROM collection have not yet been systematically explored. When transferring questionnaires from paper to electronic format, comparability is questioned. Many individual studies and several meta-analyses [7][8][9][10] have concluded that scores derived from ePROMs are equivalent to their original paper versions. In other words, scores derived from a computerized measure do not differ from scores derived from the pencil-and-paper version. The International Society for Pharmacoeconomics and Outcomes Research (ISPOR) reported 3 levels of modification (minor, moderate, and substantial) for the migration from original paper-based PROM to ePROM. The ISPOR also provides an effective strategy for testing measurement equivalence (reliability and validity). Minor modification means simply placing a paper-based scale form into a screen-based format without changing font size or altering items. Then, only a cognitive interview with 5-10 patients and a usability test is recommended. Moderate modifications are changes such as splitting single items into multiple screens, requiring the patient to use a scroll bar to see all the items or responses, or changing the order of items. With moderate modifications, equivalence testing with a randomized parallel group or randomized crossover design is advised in addition to usability testing. Major changes include removing items. With major modifications, full psychometric evaluation and large-scale usability testing in the target population are required [11]. However, recent evidence suggests that previous usability evidence in a representative group is sufficient to assume equivalence [12].
The ISPOR's electronic patient-reported outcome (ePRO) System Validation Task Force also developed recommendations on the validation of electronic systems used to collect PRO data in clinical trials [13]. This report enhances the understanding of different steps needed to develop ePROM. Both reports, based on expert opinion, give important insights in the development of ePROM based on the paper-version counterpart.
Hence, there is growing emphasis on ePROMs with a clear shift towards electronic data capture driven by regulatory and practical considerations [14], and patients seem motivated to use these tools as long as they provide added value and quality of care [15]. While a number of reviews have summarized the equivalence of digital questionnaires, none of these reviews systematically assessed the benefits and disadvantages of ePROM. Since more people have gained access to the internet via many types of devices, many opportunities have arisen in the eHealth ecosystem. Weighing the advantages against the disadvantages is necessary and imperative for clinical practice and research purposes. This systematic review aimed to evaluate the scientific evidence for the use of digital questionnaires to assess PROMs and more particularly describe the benefits and disadvantages.

Methods
The protocol for this review was accepted in the PROSPERO systematic review database (ID: CRD42018094795) [16]. This systematic review was conducted and reported following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines [17].

Inclusion and Exclusion Criteria
The PICO model was used to define the criteria to assess study eligibility. To be included in this review, studies had to report about questionnaires that evaluated PROMs. These questionnaires had to be in digital format (ie, tablet, computer, or mobile app). The criteria did not include a comparison; both studies comparing digital against paper formats and studies solely reporting about a digital questionnaire were included. The outcome measures described either benefits or disadvantages of digital questionnaires. This systematic review focused on the use of digital questionnaires. The scope of digital questionnaires was broad, including any web-, tablet-, computer-, or mobile-based method to assess PROMs.
To be included, articles had to evaluate ePROMs, preferably those used by general practitioners, doctors, occupational therapists, physiotherapists, or other health care workers; assess questionnaires in a digital format; compare a digital questionnaire with a paper-based method; describe either benefits or disadvantages of a digital questionnaire; or describe a randomized controlled trial or cohort, case-control, longitudinal, descriptive, or qualitative research.
Articles were excluded when the questionnaire was not used in the health care setting, it did not describe one of the listed aspects or clinical parameters mentioned in the keywords, or it described a review, meta-analysis, case study, or case report.

Information Sources and Search Strategy
A systematic computerized search strategy was performed in PubMed and Web of Science in October 2017. Additionally, manual screening of reference lists of relevant published literature occurred in November 2017. Neither filters nor limitations on the query were used. We searched for articles using the keywords patient related outcomes, self-management, self-reported, self-administered, questionnaire, survey, PRO, ePRO, PROM, ePROM, electronic, web-based, tablet-based, and digital questionnaires in combination with the keywords advantages, disadvantages, benefits, efficacy, acceptability, feasibility, validity, reliability, reproducibility, and response rate.

Study Selection
Two reviewers (JM and NH) searched and screened the identified records based on the eligibility criteria. Screening and selection were performed first on the title and abstract and second on the full text. Only published full-text articles in English were included.

Data Collection
The following relevant information was extracted: study description, examined ePROMs, outcome measures, and main results.

Methodological Quality
Two researchers (NH and JM) independently assessed the methodological quality. Both researchers were not aware of the other's evaluation before holding a consensus meeting. Methodological quality of the experimental studies was assessed with a 10-item checklist provided by the Dutch Cochrane Centre [18]. Observational studies were assessed with the 14-item Quality Assessment Tool for Observational Cohort and Cross-Sectional Studies [19]. Studies with high methodological quality were given more value when making final conclusions about the advantages and disadvantages of ePROMs.

Study Selection
The results of the literature search and study selection are shown in Figure 1. In summary, 2333 records were identified after removing duplicates. After screening the titles and abstracts, 100 eligible studies remained, and the full-text versions were screened. After reading the full text, 32 articles that met the predefined inclusion and exclusion criteria were included in this systematic review. Two reviewers (NH and JM) screened the identified records using the eligibility criteria. Screening was first performed based on the titles and abstracts. Full-text articles were retrieved when a record was assessed as eligible. Each full-text article was once again assessed against the inclusion criteria. Disagreements were discussed between the researches, and consensus was always achieved. The intervention of a third reviewer (UVD) was not necessary.
Overall, the included studies represented 11,006 individuals (mean age 49 years, range 13-93 years) exposed to an ePROM or asked their opinion about it. Not all studies [30,31,38,51] reported the ratio between male and female participants, meaning the sex of 3038 of the 11,006 participants was unknown. Based on the available data, 61% (4827/7968) of the subjects were female, and 39% (3141/7968) were male.
The characteristics of the studies are presented in Table 1, and the results are presented in Table 2.   [26] 92.3% of those exposed to both electronic and paper vs 59% of those exposed only to paper (P=.001) were willing; patients exposed only to paper more likely to report barriers: data privacy ( Mean (SD) pleasantness: 2.7 (0.9) for pencil and paper vs 3.0 (0.8) for internet (P<.01); mean (SD) difficulty: 3.6 (0.7) for pencil and paper vs 3.9 (0.7) for internet (P<.01) Preference CSGA ll Tablet PC [27] ≥50% unable complete without assistance (reason: computer illiteracy) Feasibility in older patients CHQ-CF mm PC Web [39] 0.54% with paper vs 0.04% with internet (P<.01) Data completion, missing data EQ-5D nn , PHQ-9 oo PC and tablet Web [33] 92.3% found it easy to use, 87.6% thought it time appropriate, 77.3% saw a perceived benefit  7 None mentioned in particular.

Methodological Quality
The risk of bias scores and the level of evidence, based on the classification of the Dutch Centraal BegeleidingsOrgaan-classificatiesysteem [52], are reported in Table 1
The satisfaction with and attitude towards ePROMs were reported in 7 studies. Most patients who were exposed to an ePROM found it easy to learn, easy to use, would recommend it to other patients, and would like to continue using it [20,21,33,34,43,47]. In a feasibility and acceptability study of a smartphone app for seizure self-management, patients with epilepsy thought ePROMs would reduce medical visits and health-related costs. Positive satisfaction levels with ePROMs were found for people who were younger (P=.002), lived in a city (P<.001), had higher education levels (P=.001), had stable employment (P<.001), had more frequent seizures (P=.01), had poor medication adherence, and owned a smartphone (P=.001) [21]. In breast cancer patients, willingness to use ePROM was higher in the group with previous experience with ePROM than in the group with previous experience with only paper PROM (92.3% and 59%, respectively, P=.001) [26]. Finally, reviewing the results with a health care professional was associated with 6.6-fold increased odds (P<.001) of perceiving systematic ePROMs as a benefit [33].

Completion Time
Time to complete electronic and paper-based questionnaires was reported in 9 studies [35][36][37]40,43,[45][46][47][48], and 3 of these studies reported no significant differences in completion time [35,47]. In one study, however, subjects reported that the completion time for the electronic variant was more acceptable (P=.02) and was perceived as less of a barrier (P=.003) compared to the paper version [36]. Significantly lower times for the electronic variant were reported in 3 other studies [37,40,43]. Only 2 of the 9 studies reported significantly lower completion times for the paper version [45,46], owing to the longer log-on procedure required for the ePROM [45]. One study was indecisive. A detailed overview of the completion times can be found in Table 4. Overall, the completion times for ePROMs were at least equal to or faster than those for paper forms.

Cost
Engan et al [23] calculated and compared the human resource (HR) costs, specifically the time spent by an employee preparing, receiving, and handling data, of web-based and paper-based questionnaires. The mean HR cost for the web version was 9.5 minutes, whereas the mean HR cost for the paper version was 24 minutes.
Based on a cohort of 500,000 subjects [36], the financial costs of a paper-based questionnaire were calculated, including printing, mailing, returns, and double data entry. In total, it cost €4,965,833 (€9.94/subject) to use a paper-based version. In comparison, the development of a web-based tool by professionals was estimated to cost only €150,000 (€0.3/subject) or just 3% of the amount of the paper version.
Overall, these results indicate that digital data collection is less expensive, especially with large sample sizes, and it reduces HR-related costs.

Data Quality and Completion
Of the 10 studies [23,31,35,36,38,39,41,42,45,50] that reported on missing and incomplete data, 7 studies [23,31,36,38,39,41,50] indicated that electronic methods are associated with less missing data and more complete data. Integrated controls embedded in their ePROM administration was reported by 3 articles [23,35,36]. When a question wasn't answered, an alert message provided the option to revise the answer prior to submission. As such, data entry mistakes in the form of missing, inconsistent, or abnormal values could theoretically be reduced to zero [23,36]. Regarding unanswered questions or incomplete questionnaires, 2 studies reported no statistically significant differences between ePROMs and paper PROMs [35,45]. One study [42] found significantly more missing items in the electronic version. And, one study reported that the answers were more detailed in 4 of 5 open questions on their electronic questionnaire (P<.05) [31]. Details of these results can be found in Table 5. Based on these results, we conclude that data quality is higher with ePROMs.

Response Rate, Adherence, and Compliance
A retrospective cohort analyzed the annual data from PROM non-completers. PROM monitoring was completed via paper until 2010, and in 2011, ePROMs were implemented. The initial rate of PROM non-completers was 43%-58%. This decreased to less than 20% since the implementation of ePROMs in 2011 [28]. One randomized controlled trial reported response rates of 17.9% in the internet group and 73.2% in the paper group. After sending a reminder, response rates were 64.2% and 76.5%, respectively (risk difference 12.2%, P=.002) [50]. Another study found no differences in completion rates between ePROMs and paper PROMs (P=.208) [35].
There is conflicting evidence on the effect of electronic data collection on response rates and adherence. Adherence to ePROM declines over time [20,29]. The opportunity to send automated reminders (eg, email or notification) to subjects can improve response rates and compliance [20,50].

Other Benefits
The role of ePROMs in symptom management and decision making was acknowledged in multiple studies. Andikyan et al [20] and Schnall et al [22] reported that electronic symptom self-reporting was important in clinical decision making. Automated data collection and processing via ePROM can generate automated alerts to health care professionals when a patient reports disturbing or severe symptoms [20]. It allows early detection of complications, immediate action, and potentially reduction in symptom burden, complications, and readmissions to the hospital. Furthermore, it empowers patients and improves patient-clinician communication [22,24,42]. This is facilitated by the opportunity to plot results visually with a graph or visual aids and gives both the patient and clinicians better insight in the evolution of the patient's health status [25,34,43]. ePROMs have the advantage of always being available [24,25]. There is no paper waste [34,41], and ePROMs are portable and can be used to measure across multiple devices [42,46,49]. These reported 'other benefits' originate from studies with the lowest methodological quality.

Disadvantages
As of May 25, 2018, all European organizations are expected to be compliant with the General Data Protection Regulations. This is reassurance for patients that the law is on their side when it comes to the use of their personal health data. All included articles and studies were performed before the implementation of the General Data Protection Regulations. However, privacy concerns were reported in 2 studies [21,26]. Liu et al [21] reported that the majority of patients (71.7%) thought their privacy should be adequately protected. In another study, patients were asked whether there were any barriers related to privacy and technology that would negatively influence their willingness to use ePROMs, and 30% were concerned about privacy issues. The study showed that barriers can be overcome by exposing the patients to an ePROM, which significantly influenced the willingness to participate in electronic assessments [26].
Disadvantages due to technical issues were addressed in 5 articles. The difficulty of or problems with login procedures were addressed in 3 studies [24,45]. Furthermore, technical difficulties adversely impacted compliance; patients who experienced technical difficulties completed fewer daily symptom entries (41.0%) than those who did not (76.0%) [29]. In another study, the needs and possible technological support structures were investigated. The importance of different possible support services to help complete a web-based questionnaire was assessed. Onsite support services were rated as being moderately or highly important by 38%. Technical telephone support was rated as moderately important or very important by 52%. At least 61% would appreciate receiving direct feedback after using the ePROM app [26].
Electronic data collection may require a large initial financial investment (eg, to purchase tablets or computer infrastructure and software, equipment costs, hiring computer programmers, or accessing cellular internet) [30,36,45].
A major disadvantage of ePROM is the potential of a 'digital divide'. People who are computer illiterate, are older, or have no access to infrastructure could be disadvantaged. One study reported that more than 50% of >70 year olds were not able to complete the electronic version without assistance due to computer illiteracy; less assistance was required for patients completing the paper version [27]. In a second study, patients who needed support were significantly older [42]. The digital divide was also illustrated in another study with cancer patients. Patients who refused ePROM or chose phone calls over (home-based) ePROMs were approximately 10 years older. Patients may differ in terms of available internet, user experience, and affinity for new media. Older or computer-illiterate patients need opportunities to familiarize themselves with the devices [24]. Older patients with poorer health-related quality of life and fewer pre-existing technical skills reported barriers for ePROMs more frequently [26]. Wintner et al [24] reported that patients found ePROMs too impersonal.

Suggestions
Suggestions and tips for ePROM apps were extracted from 12 studies [20,21,24,27,29,30,[32][33][34][35]38,42]. ePROMs should be free, simple, and minimalistic. They should have a good design, good user experience, adjustable font size, and adaptable user interface. When you start implementing ePROMs, provide educational sessions or support, think of the link with electronic health records, and review the results of the ePROMs with the patients because of the increased perception of benefit. ePROMs should provide positive reinforcement for the patients. Based on our results and discussion, we created a comprehensive overview of the benefits, disadvantages, and suggestions for ePROMs (see Figure 3).

Principal Findings
The goal of this systematic review was to systematically and critically summarize the evidence on the use of ePROMs and find the potential benefits and disadvantages. We conclude that ePROM collection is feasible and accepted in healthy people and a wide range of patients with different conditions. Taking into account the results from the strongest methodological studies and the items that were reported in multiple studies, electronic data collection is preferred over paper-based collection, costs less, improves data quality, results in similar or faster completion times, and requires less administration time. Clinical decision making in combination with adequate symptom management can be facilitated. Expressed opinions reflected positive thoughts and attitudes towards ePROMs.
Overall, participants found it easy to use, found it easy to learn, and would recommend it to others.

Strengths and Limitations
Although our findings are generally favorable towards ePROMs, we cannot ignore the potential disadvantages. Aspects to consider are privacy protection, the one-time large financial investment, and exclusion of certain populations. Patients may be unwilling or unable to complete ePROMs due to higher age or computer illiteracy. Some patients have no internet access, do not have technological devices, or are not acquainted with technological devices. These reported disadvantages and barriers need to be considered when implementing a digital data collection tool in any population. Potential solutions may include an educational session on the use of the digital app and providing sufficient support [24,27,42]. It is also useful to at least provide back-up pen-and-paper data collection to avoid excluding segments of the population from receiving the best possible health care [20,32]. Several suggestions to keep in mind when creating an ePROM are also mentioned in this literature review, which could increase patient experience, usability, and acceptability.
Considering the influence of age, 2 studies suggest that it is an important factor that could potentially increase completion time [40,46]. In contrast, one study found no relationships between completion time and computer skills, age, or education [37]. Older people in particular have reservations concerning modern computer technology and need to be properly approached, especially since we found that younger people had a significantly greater preference for ePROMs [23,32,42]. In our systematic review, we found that various groups of patients with a chronic disease preferred ePROMs over paper versions. On the aspect of completion time, only the time for the patient to complete the questionnaire was measured in the included articles. However, one of the greatest reported advantages of electronic data collection is automated data processing [36,38,[41][42][43]45,49], which subsequently reduces HR time [23], and data are less prone to administration errors. Clinical-based decision-making models using daily registration of PROMs can thus be created.
The strengths of this literature review are that 32 studies concerning the research question were retrieved. Not all studies were comparative trials but assessed patient satisfaction or attitude towards a single ePROM [21,24,25,32,33]. These studies, although not methodologically the strongest, provided capital insights for the research question.
In this systematic literature search, we only searched two databases. It is, therefore, possible that we missed some clinical studies. Moreover, the limited methodological quality of some of the included studies diminished the power of the recommendations.
The overall methodological quality of the included articles was moderate. Disadvantages were a lack of blinding of participants, heterogeneity of outcome measures, heterogeneity of patient populations, different ePROM questionnaires, and different ePROM modalities/formats. Generalizing or comparing results is therefore more difficult, and the results should be interpreted with caution.
The most frequently used screen-based device was tablets. This may be because tablet screens are larger than traditional handheld devices, are easy to use, and can be used for device-based systems. They can provide access to web-based portals or can be used with downloadable apps, which makes them the primary platform for site-based (ie, hospital, care centers) ePROM collection. On the contrary, desktops usually lack touch screen functionality and require the use of a keyboard and/or mouse to respond to questions [14]. Different electronic modes were used in the different articles. The advantages and disadvantages of the different electronic modes are difficult to conclude from this study. Contrasting evidence was found in previously published literature. Two reviews reported their concerns of equivalence between different electronic modes [8,10]; however, White et al [10] found small differences in the correlations, which were not significant regardless of the electronic mode used. In clinical trials, multiple modes of administration may be used, and new findings may be compared to findings that used a different electronic mode of data collection. Further research is warranted regarding the influence of the electronic mode on measurement equivalence. Our findings predominantly complement those from other published literature. Belisario et al [53] conducted a review to assess the impact of apps on the quality of survey questionnaire responses and reported contradictory results regarding completion times but acknowledged that apps might improve data completeness with more complete records than paper administration. Similar to our findings, they reported that there is not enough evidence that apps impact adherence to sampling protocols. Muehlhausen et al [9] conducted a meta-analysis on the equivalence of electronic and paper administration of PROMs and showed that ePROMs yielded comparable results to those of the paper-based variant. Their findings also confirmed the ISPOR taskforce's conclusion that full psychometric testing of new ePROMs is not necessary for migrations with minor changes only [12]. For researchers and sponsors, this is a clinically and financially reassuring aspect that might facilitate the decision-making process to migrate from paper to digital data collection. The bring-your-own-device (BYOD) approach for ePROM data collection shows potential. BYOD allows participants to use their own computer device (eg, smartphone, tablet, laptop) to access and complete ePROMs [14]. However, there are still a number of issues (eg, software, security, ownership) that need to be resolved before BYOD becomes widely used.

Future Work
The importance of PROMs is widely accepted. Collecting PROMs with paper-based questionnaires requires many subsequent time-consuming steps [45] that hamper wide implementation in daily care. Electronic collection of PROMs overcomes many of these steps. The potential to collect, score, analyze, visualize, and almost instantly review the results may facilitate workflow. Clinically, we believe ePROMs will improve the interchangeability of information between health care workers, patient-clinician communication, and patient care due to its always available nature. In addition, automated data processing in combination with targeted strategies (eg, automated alerts when patients report disturbing symptoms) has major clinical implications. Clinicians and researchers will also benefit from digital data collection since it reduces administration time. Furthermore, integration of ePROMs into electronic health records may be fundamental to advancing clinical care to improve patient engagement and health outcomes.

Conclusion
Based on this study, we found multiple advantages for the use of ePROMS in several fields of care. ePROMs are preferred over paper-based forms, cost less, improve data quality, result in similar or faster completion times, reduce administration times, and facilitate clinical decision making in combination with adequate symptom management. Subjects expressed positive thoughts and attitudes towards electronic data collection. Potential disadvantages have been mapped but they are not of the magnitude to disregard ePROMs. Furthermore, suggestions have been provided to counteract the disadvantages.