Feasibility of RetinoQuest: e-health application to facilitate and improve additional care for retinoblastoma survivors

Retinoblastoma (Rb) is a malignant intraocular pediatric cancer. It occurs in the first years of childhood, usually between the ages of 0 and 5 years. The incidence is 1:17,000 newborns (10–12 new patients in the Netherlands each year) [1], which represents approximately 3% of all pediatric malignancies.

In spite of good survival chances, the psychosocial effects of Rb vary according to the severity of the disease and the treatment, late sequelae and coping mechanisms of the patient and parents [2]. Many survivors experience distress regarding their cosmetic appearance and the fear of blindness. Other concerns relate to recurrence of the disease, passing Rb to offspring, second primary tumors, restrictions in education and professional career, mobility, self-care, and relationships [3‐8]. Furthermore, the parents’ ability to cope with the stress associated with diagnosis, treatment, and heredity impacts the psychosocial functioning and development of their affected child [2, 7, 9‐12].

Van Dijk et al. showed that 30% of Rb survivors develop behavioral problems [8]. Adult survivors have 20% more psychological problems like anxiety or depression than a healthy reference group [3]. The study by Van Dijk et al. also noted that a history with Rb influenced the health-related quality of life (HRQoL) and daily living negatively in 50% of Rb survivors [3].

Though psychosocial problems are thus common among Rb survivors, they are infrequently discussed during the annual follow-up visits at the outpatient clinic. Adequate psychosocial care should include an initial survey of the psychosocial background and early detection of psychosocial problems. If necessary, survivors should be referred to one of the specialized psychosocial disciplines. In the past 10 years, interest in using patient-reported outcome measures (PROMs) to screen for psychosocial problems and the need for supportive care in routine clinical practice has increased [13‐19]. Several studies have shown that using PROMs in clinical practice facilitates open communication [20, 21] regarding HRQoL between doctors and patients and offers the possibility of early detection of psychological problems [22, 23]. In the Netherlands, we already use validated questionnaires for cancer survivors such as OncoQuest, QLIC-ON PROfile, and KLIK [14, 15, 24]. However, these questionnaires do not highlight Rb-specific distress such as cosmetic appearance and the fear of blindness. Furthermore, OncoQuest is a HRQoL-monitoring computer-assisted system for adult cancer survivors, whereas our population consists of both adults and children. Thus, currently existing validated questionnaires are not fully applicable to monitor HRQoL of Rb survivors. Therefore, we decided to develop Rb-specific questionnaire which consists of a combination of OncoQuest software with questions from Strengths and Difficulties Questionnaire (SDQ) and General Health Questionnaire (GHQ) [25‐28]. RetinoQuest is a touch screen computer program to monitor HRQoL of retinoblastoma survivors via three age-specific PROMs (Table 1). The objective of the present study was to evaluate the feasibility of RetinoQuest in clinical practice, from the survivors’ and health care professionals’ (HCPs’) point of view.

The current study investigated the feasibility of RetinoQuest in clinical practice. Earlier studies have shown that computer-assisted data collection on HRQoL data is feasible in daily clinical practice [4, 5, 12‐17, 19, 22, 29]. In line with the findings of de Bree et al. [14], we showed that RetinoQuest is a feasible e-health application to monitor HRQoL in Rb survivors in clinical practice. Ninety percent of the participants (n = 96) stated that the system was fast and easy to use. Furthermore, the system was rated with an overall satisfaction rate of 8.0 (scale 1–10).

The use of PROMs in pediatrics is scarce [16, 22, 24], while children with a chronic disease are at a greater risk of quality-of-life problems than a healthy reference group [3, 30]. Since RetinoQuest is developed for survivors of different age groups, the system uses three different validated questionnaires depending on the age group: the SDQ parent 4-10, SDQ 11-18, and the GHQ-28 [25‐28]. Therefore, participants were divided into three evaluation groups: parent participants, adolescent participants, and adult participants. The parents of the children between 4 and 10 years were most often satisfied with the system. Meanwhile, adolescents rated the questionnaire the highest. The majority of this group (28 out of 38 respondents) stated that the questions were clear. The majority of the adolescent participants (63%) stated there were no redundant questions. A large group of the adult participants (41%) were dissatisfied with questions about severe depression, suicide, and death. Because GHQ-28 is a validated questionnaire, we chose not to omit or change items of this questionnaire. Currently, we inform the survivors and their parents about the fact that RetinoQuest includes a wide variety of questions (also regarding depression) due to the large heterogeneity in the survivor group.

Special attention was given to the visibility of RetinoQuest because many retinoblastoma survivors are visually impaired [31]. Questions were presented on a large screen in clear, large characters with bright colors. The majority (88%) of participants were able to read the questions adequately. Four blind adult participants were unable to read the screen and were assisted by a trained staff member who read out the questions. These findings indicate that computer-assisted data collection on HRQoL data should be tailored to the target group.

RetinoQuest facilitated the medical team in starting and having a structured conversation about the psychosocial condition and HRQoL of a patient, in addition to the medical follow-up. Although this study had a cross-sectional design, our clinical experience is that RetinoQuest also helps to provide clear insight in how specific problems improve or deteriorate over time. Podmore et al. [32] found that if clinicians rely on patients to initiate a discussion about psychosocial issues, patients’ problems may go unaddressed. Many patients do not spontaneously confess to doctors that they are suffering from psychosocial difficulties during consultation [17, 33].

From the structured interviews with the two HCPs at the end of the study, it was clear that RetinoQuest supports HCPs to communicate relevant psychosocial issues with survivors and their families. The interviews also revealed that HCPs appreciate the use of RetinoQuest in clinical practice. The discussion with survivors about their well-being profile is efficient and usually does not prolong the consultation time, which is in line with the study from Engelen et al. [23]. However, when problems are detected, it does take extra time to adequately support the patient, and it was emphasized that special psychological care should be available when necessary. The ophthalmologists’ observations confirmed that the PROMs included in RetinoQuest are satisfactory for clinical use, except for the questions about depression and suicide. Furthermore, they had the impression that the well-being profile matches the psychosocial situation of patient. The main flaw in the use of RetinoQuest was that the HCPs did not always have access to the well-being profile due to technical or logistical issues.

In the present study, the ophthalmologists used RetinoQuest in 80% of the visits to discuss the quality-of-life issues with the Rb survivors. Other authors found [14, 22, 29] that survivors and HCPs are willing to discuss the results of PROMs in clinical practice and that the possibility of data collection is depending on the technical and logistical capabilities of the organization.

An essential factor for implementing systems as RetinoQuest in clinical practice is having dedicated manpower (e.g., staff members) responsible for this task in supporting survivors to use the system in the hospital. Web-based data collection has shown to be logistically feasible [24]. This would allow visually impaired survivors to administer the tests to their own vision aid at home, thereby ensuring that no survivors are missed in hospital. Furthermore, it would give the HCPs the opportunity to screen the results in advance. We are currently working on a web-based version of RetinoQuest, so survivors can complete the questionnaire at home before the clinical visit.

One of the limitations of the current study is that the added value of the use of RetinoQuest during consultation was only evaluated from the HCPs’ point of view. Future studies should also focus on the question if survivors feel that the program adds value to their consultation with the HCP. Furthermore, it should also be investigated if RetinoQuest increases referrals for supportive care or counseling at clinical psychology.

This study reveals that RetinoQuest is a feasible e-health application to monitor HRQoL in retinoblastoma survivors in clinical practice. Survivors as well as ophthalmologists are satisfied with the system as a tool to improve communication on quality of life and psychosocial difficulties. However, the use of the system is dependent on a good logistic organization.