Fatigue is known to be an everyday problem for most individuals with visual impairment (VI) [1-4]. Consequences of fatigue on emotional and cognitive functioning, social roles, and participation in society have been reported to be problematic [5,6]. To address this difficulty, an intervention called E-nergEYEze was developed tailored to the unique aspects of individuals with vision loss who experience severe fatigue and investigated in a randomized controlled trial (RCT) [7]. E-nergEYEze is a blended eHealth intervention based on cognitive behavioral therapy (iCBT) and self-management. Professionals from low vision services offer online support and motivate patients to follow the intervention as intended. The goal is to teach patients how to cope with fatigue by focusing on understanding vision-specific fatigue, which makes them perceive it as more controllable and to reduce its consequences on daily life [5,8].

Considering the increasing demand for support in health care, eHealth offers a solution to reach a broader and larger audience. It can be offered independently of time and place, may stimulate patient empowerment, and requires less professional effort [9,10]. However, the challenge is to ensure that an intervention is consistent with patients’ needs and is easy for individuals with VI to use [11-13]. User experiences are of essential importance in all development and testing steps of iCBT to identify factors that drive participant behavior or affect outcomes and to gain a deeper understanding of participant experiences and needs [14]. Moss-Morris et al [15] conducted a pilot-RCT of a blended iCBT for individuals with multiple sclerosis (MS) in which qualitative feedback was reported on content and technical difficulties. Muijs et al [16] conducted a self-help iCBT intervention to prevent and reduce fatigue in adults with diabetes and reported the relevance of user experiences, stating that content and features need to be further personalized to preferences and needs. Finally, Babbage et al [17] studied a self-management app for fatigue in individuals with MS. Users reported challenges with the quantity and complexity of the text-based content. Results confirmed the relevance of qualitative feedback and the perceived value of fatigue-reducing iCBT tools.

The perspective of professionals is equally important to consider, especially because Dijksman et al [18] reported that 30% of psychologists did not believe that blended care had advantages in effectiveness and time savings compared to treatment as usual. However, overall, professionals were positive toward blended care, with significant differences in favor of active users versus nonusers. Because guidance skills, time, and input could influence outcomes [15,19], educational activities regarding eHealth, patient perceptions, and liability are considered necessary before using a new blended care intervention [18].

E-nergEYEze was designed in collaboration with all stakeholders (ie, patients, professionals, and researchers in low vision, fatigue, and eHealth). To maintain involvement in future iterations, this newly developed iCBT intervention was first pilot-tested in a usability and feasibility study by considering fidelity. Results showed a promising trend to reduce severity and impact of fatigue. The study methods were feasible, and the fidelity of the intervention protocol was suitable [20]. As the intervention was only studied partially to refine E-nergEYEze, we planned to further explore stakeholder experiences by offering the full intervention while focusing on its fatigue-reducing accessibility, content, and guidance. To complement the future cost-effectiveness outcomes, our aim was to evaluate the user experiences of the E-nergEYEze intervention from the perspective of patients and professionals.

This study was conducted according to the E-nergEYEze protocol [7].

Recruitment with information letters was performed by low vision service organizations in the Netherlands (Royal Dutch Visio and Bartiméus) and additionally through social media, patient associations, and 2 companies that provide low vision aids. Patients with VI and severe fatigue were included (N=98) in a single-blinded RCT and randomized to the intervention group (n=51, 52%) or control group (n=47, 48%). Patients were screened on the inclusion criteria having a mild VI (logarithm of the minimum angle of resolution visual acuity 0.30-0.40 or peripheral vision loss of <45°) or worse caused by an ophthalmic disorder; experiencing severe fatigue (a score on the Checklist Individual Strength questionnaire’s fatigue severity subscale of ≥35) [21]; understanding the Dutch language; having access to the internet; and being aged ≥18 years. Exclusion criteria were having severe cognitive impairment, as assessed with a 6-item screener (the validated 6-item Mini-Mental State Examination, with a cutoff score of >3) [22] and receiving treatment or having received treatment in the past 12 months from a medical specialist for a comorbid disease that could explain the presence of fatigue (eg, MS, cancer, or psychiatric disorder). A process evaluation was performed in patients who received the intervention (n=51, 52%) and professionals (n=11) of low vision service organizations who provided support. Both social workers (n=7) and computer trainers (n=4) kept time records, and the eHealth platform (e-platform) provided data on user engagement. A mixed methods design was used.

Patients were invited for a face-to-face session with a social worker and a computer trainer. Access was given to the e-platform Minddistrict [23] to follow an introduction module and 8 thematic modules with information and assignments on how to cope with fatigue based on cognitive behavioral therapy and self-management. The module themes and content are provided in Textbox 1.

Patients were able to log in to their computer, tablet, or smartphone at their preferred place. Content was represented in module steps: the steps within a module that contained information or assignments. The e-platform logged how often, what content, and for how long patients were active over a period of 12 months. Social workers gave online feedback on assignments, and psychologists (n=2) were available for peer consultation.

A survey was conducted using the Dutch Mental Health Care Thermometer (MHT) questionnaire [24] after 6 months by telephone to evaluate patients’ experiences. This questionnaire consisted of yes or no questions with regard to dimensions specified in the subscales: appreciation of information, appreciation of decision-making, and appreciation of professional and treatment results. Internal consistency of the subscales ranges between 0.70 and 0.86 [25]. Researchers (PhD candidates) from Amsterdam University Medical Centers conducted the assessments by telephone with registration into data-entry software called Castor [26]. There was no relationship established between researchers and patients before the study commencement. The e-platform provided data on user engagement.

Blended care is a relatively new way of providing guidance for professionals of low vision services in the Netherlands. The exploration of the experience of social workers and computer trainers with blended care and with guiding patients who follow the E-nergEYEze modules enabled a deeper understanding of insight into the professional perspective toward the digital aspect and content of the E-nergEYEze intervention within this target group. Therefore, social workers (n=7) completed the therapist evaluation for each patient. This questionnaire included questions on their experience with the intervention, online guidance, and compliance of patients to the intervention. Computer trainers (n=4) kept time records and evaluated the computer training. To complement the therapist evaluation outcomes, a focus group meeting was held with the involved social workers (n=4), computer trainer (n=1), and psychologists (n=2).

Careful consideration was given to information related to whether the intervention was completed. We decided to distinguish between patients who completed most of the module steps (≥80%) and patients who discontinued earlier (<80%). Quantitative data were analyzed using descriptive statistics, including baseline fatigue severity. Data were not normally distributed; therefore, results were reported with medians and IQR. The Mann-Whitney U test was performed to examine group differences in continuous variables and the chi-square test for nominal and ordinal variables. Qualitative data were analyzed by applying a thematic approach in MaxQDA2020 (VERBI Software) [27], a method to identify, analyze, and report patterns within data. To strengthen the reliability and validity of the analyses, MV (medical doctor and PhD candidate) and HvdA (senior researcher) separately analyzed answers to open questions of 3 evaluation forms to create thematic codes for either perspective, after which 2 code trees were created. All evaluations were then coded by MV, and data were analyzed by interpreting patterns within and between themes. Coding, analyses, and results were discussed in detail with HvdA.

Results from the professional perspective formed the content of the focus group meeting which was organized with 3 moderators (RvN, HvdA, and MV). Interaction among the professionals was encouraged with the leading questions “What goes well?” and “What can be improved and how?” This session was audio-recorded and transcribed verbatim by MV. Data were coded in vivo and analyzed thematically, using the preexisting code tree as a guide, and returned to the professionals for additional comments.

Ethics approval was obtained by the Medical Ethical Committee of the VU University Medical Center Amsterdam, the Netherlands (reference no. 2019/027, NL67802.029.18). The COREQ (Consolidated Criteria for Reporting Qualitative Research) checklist was applied in this manuscript [28]. Data was anonymized during data collection and analysis. Verbal consent was given by all professionals to record and transcribe the focus group meeting. All participants signed an informed consent form to participate in the study. Participants did not receive compensation for participation in the study.

Demographics and clinical characteristics of social workers and patients are presented in Tables 1 and 2, respectively. None of the variables were statistically different between patient subgroups. Clinically relevant differences were noticed in the subgroup who discontinued the intervention, including longer duration of the VI, less paid work, a higher percentage of participants living alone, and a history of psychiatric illness and comorbidities.

This study evaluated the user experiences of the E-nergEYEze intervention, an iCBT intervention to reduce severe fatigue [14], from the perspective of patients with VI and professionals who used E-nergEYEze [7]. These firsthand experiences showed that patients completed a median of almost 90% of the module steps, almost two-thirds of the participants completed most of the intervention, and some were active up until 12 months from baseline. More than half of the patients were active on the e-platform at least 1 week after individually completing the last module step. This does not suggest that every patient finished all module steps but confirms that patients were actively engaged in the intervention and the potential of repetition offered by eHealth was noticed and applied to achieve long-term progress. Continuing E-nergEYEze was related to the presence of digital proficiency; having the appropriate expectation; content that matches personal preferences and life context; and the absence of impeding personal circumstances, mental health issues or concurrent other rehabilitation programs. However, it is challenging to find or develop an e-platform that is all-encompassing for the accessibility needs of individuals with VI.

E-nerEYEze was perceived as valuable to patients, especially personally relevant topics that triggered a positive change in dealing with fatigue. iCBT seems to be equally effective as face-to-face cognitive behavioral therapy [10], though we noticed an adherence drop in the subgroup that discontinued after module 2 or during module 3. Module 3, on graded activity, proved to be especially important in the reduction of fatigue [29,30]; however, this module is also relatively long and intensive, which may have caused patients to drop out. Adherence could be increased by motivating patients, embedding a midterm evaluation, or considering changing the order of modules. Reevaluating the difficulty of the content with professionals in cognitive behavioral therapy could be useful as well, for example, by reconsidering content of module 3 on the essence of physical activity to increase physical health and reduce fatigue. The examples of physical activities could be reconsidered in consultation with patient representatives to increase the feasibility of this module for individuals with VI and to lower the barrier to starting physical activities. The length of a module affects concentration and fatigue symptoms; however, a module does not have to be completed all at once. Practical alternatives to deal with length and intensity of content suggested by Babbage et al [17] were already implemented in the E-nergEYEze intervention, such as the use of voice-overs, read-aloud audio, and videos. We chose a blended design for E-nergEYEze design in consideration of the benefits of eHealth [9,10] and the more favorable outcomes that have been reported for guided iCBT versus unguided iCBT [31-33]. Our results showed that guidance was essential for patients; we underestimated the influence of and need for frequent and substantive support. Social workers were instructed to follow, motivate, and confirm patients’ progress in feedback moments, without extensive conversations on the costs and benefits of eHealth; this was seen as a discrepancy with patients’ needs. Worm-Smeitink et al [34] suggested including more flexibility in the intensity of guidance, a suggestion that we echo with respect to remote support. Our social workers had no experience in providing online support, and some struggled with the remote nature of eHealth, especially because of the limited interaction. Online interaction calls on other skills; therefore, skill, time and input could have influenced the treatment effects [15,19]. Precisely because eHealth is a new mode of treatment in this target population, it is important to pay adequate attention to the training of professionals providing low vision services and arrange prescheduled guidance time and peer consultation moments. In accordance with Dijksman et al [18], our results indicated that professionals need more support in providing substantive remote support.

The major strengths of this study are presented in Textbox 3.

We considered it a strength that these user experiences were analyzed ahead of the cost-effectiveness results, which confirm an independent analysis and interpretation of data. E-nergEYEze was developed by various professionals and patients, based on evidence-based iCBT [35,36] and was pilot tested [20]. Patients gave positive feedback on improvements made based on the pilot study, reflecting the value of pilot testing before conducting an RCT. The content variety gave patients the possibility to personalize their treatment by applying coping strategies that appealed to them in daily life, as well as broadening their horizon outside their comfort zone. E-nergEYEze has been implemented in compliance with the study protocol [7], ensuring the internal validity of these results with respect to the intervention. However, the reliance of self-reported time records of professionals could limit actual or perceived validity. Another limitation that should be considered is the possibility of self-assessment bias, that is, the overestimation of one’s own performance. The mixed method design included qualitative analysis; therefore, discussing reliability is challenging, but we have striven to be consistent in the analysis. Internal validity was enhanced by triangulation of research methods and verification of qualitative analysis. The sample size of this study was relatively small, as we zoomed in on the user experiences of the intervention group and the professionals who were involved. This could limit the generalizability of the results. In addition, attrition bias could have influenced the results, as 5 patients did not complete the Dutch MHT questionnaire.

Our results respond to the knowledge gap regarding user experiences with blended iCBT in adults with VI [37]. To determine whether a patient receiving low vision services is suitable for E-nergEYEze, the difference in patient characteristics between the subgroups that we investigated may be of clinical relevance. Patients who discontinued the intervention could have benefited from receiving more support, for which training of professionals is essential. Guided iCBT has increasingly been investigated, with varying guidance intensity [15,36,38,39], and it would be valuable to examine what frequency and type of guidance can best be offered in iCBT, as well as the amount of content and how to match the difficulty to the skills and opportunities of patients. Taking our results into consideration, individuals with VI who experience severe fatigue, who are digitally sufficient, and who are willing to receive remote treatment with the ability and motivation to work at self-set times are likely to benefit from the E-nergEYEze intervention. Individuals with impeding personal circumstances, mental health issues, or who are participating in other concurrent rehabilitation programs are less likely to benefit or more likely to discontinue the E-nergEYEze intervention.

The potential of the E-nergEYEze intervention to reduce fatigue severity in individuals with VI was underlined by user experiences. These results provide information to learn from and optimize E-nergEYEze into a useful, available, and affordable tool to reduce severe fatigue in individuals with VI. This research offers future prospects for the development of blended eHealth interventions for other conditions in this target group with specific needs regarding accessibility of e-platforms and guidance with an affinity for providing digital care.