Patients with lymphoma are at risk of experiencing adverse effects of cancer and its treatment, such as fatigue, cognitive problems, and neuropathy [1-4]. In addition, up to a quarter experience persistent levels of anxiety, depressive symptoms, and fear [5], also known as psychological distress. Both symptoms and psychological distress may be exacerbated when information and supportive care are unavailable [6,7]. This may, in turn, lead to increased health care use [8]. Regular screening of physical and psychosocial symptoms using patient-reported outcomes (PROs) could increase awareness and recognition of symptoms and can contribute to their management [9-13].

Since the past decade, there has been a cultural shift toward patient engagement in health, with a growing demand from patients to access their individual results [14-17]. Returning individual PRO results enables patients to monitor their functioning and create awareness of symptoms. Furthermore, it offers patients the opportunity to compare their scores with peers [15] to evaluate if their scores are normal and to incorporate this information into personal decision-making [16]. However, some clinicians have expressed reservations about disclosing PRO research results to patients, as it may cause patients to become more concerned and lead to increased health care use by patients and higher workload for clinicians. Therefore, it is important to investigate whether patients will be in a better or worse condition with their PRO results being disclosed and which patients wish to receive their PRO results.

To improve health outcomes, subsequent steps such as self-management interventions are expected to be necessary in addition to monitoring and returning PRO results [18]. Self-management interventions intend to enhance patients’ knowledge and skills and empower them to play an active role in the management of their disease and its consequences [19,20]. Studies on the outcomes of self-management are mainly based on patients with solid cancers and are not consistent, with some randomized controlled trials (RCTs) showing no effects [21-23]. In addition, most self-management interventions are not specifically aimed at patients with lymphoma and are found to be effective among selected groups of patients. Little is known about the effects of such interventions in a population-based setting within an unselected group of patients.

To investigate the effect of (1) return of individual PRO research results, including comparison with peers [24], and (2) a web-based self-management intervention Living with lymphoma, the Lymphoma Intervention (LIVE) trial was performed. The primary objective of the LIVE RCT was to examine the effects of return of PRO results to patients with or without access to Living with lymphoma on self-management, satisfaction with information, and psychological distress in a population-based setting of patients with lymphoma [24]. We hypothesized that those with access to the return of PRO results or access to LIVE would have higher levels of self-management and satisfaction with information and lower levels of psychological distress. On the basis of new insights that psychologically distressed patients reported increased health care use [8], we also investigated the effects of return of PRO results to patients with or without access to Living with lymphoma on health care use (secondary objective). Furthermore, we explored sociodemographic, clinical, and psychological differences between patients who viewed their individual PRO results and those who did not.

This RCT was embedded in the population-based Patient-Reported Outcomes Following Initial Treatment and Long-Term Evaluation of Survivorship (PROFILES) registry [25]. PROFILES enables PRO data collection management and links PRO data to clinical data from the Netherlands Cancer Registry (NCR).

Between October 2015 and February 2019, patients diagnosed with lymphoma (Hodgkin lymphoma, non-Hodgkin lymphoma, or chronic lymphocytic leukemia), as defined by the International Classification of Diseases for Oncology-3 codes [26], from 13 hospitals in the Netherlands were selected for participation 9 to 18 months after diagnosis. The NCR registers all newly diagnosed patients with cancer in the Netherlands within the first year after diagnosis and routinely collects detailed data on sociodemographic and clinical characteristics (eg, age and sex, date of diagnosis, cancer type, and primary treatment). Treating hemato-oncologists were asked to verify the eligibility of the patients. As we aimed to keep a population-based approach, we only defined a few exclusion criteria, such as presence of severe psychopathology or dementia, being in transition to terminal care, and not being able to complete a Dutch questionnaire. Patients were informed that completion of the web-based questionnaire resulted in RCT enrollment with automatic randomization to 1 of the 3 study arms. Paper respondents were not eligible for the RCT (as the return of PRO results and Living with lymphoma were web-based) and were observationally followed within the PROFILES lymphoma registry. A reminder mail was sent after 3 weeks. Respondents received follow-up questionnaires at 4 months (T1), 12 months (T2), and 24 months (T3) after the baseline questionnaire. More details about the study design, enrollment, and sample size calculation have previously been published in the research protocol [24]. The RCT was centrally and locally approved by a medical research ethics committee [24].

Randomization was performed using block randomization to ensure a balance in sample size across arms over time [27]. Participants were equally randomized to (1) care as usual (CAU), (2) CAU plus return of PRO results, or (3) CAU plus return of PRO results and Living with lymphoma.

In total, 1193 patients were selected from the NCR and 892 patients were invited to participate. Of these, 51.1% (456/892) participated, of which 25.7% (229/892) were excluded for the RCT as they completed the questionnaire on paper (CONSORT [Consolidated Standards of Reporting Trials] diagram; Figure 1).

Overall, 25.4% (227/892) completed the web-based questionnaire and were included in the RCT. They were randomly assigned to CAU (control group; n=76), return of PRO results (n=74), or return of PRO results and access to Living with lymphoma (n=77). Completion rates of follow-up questionnaires were 84.1% (191/227) on T1, 74.9% (143/191) on T2, and 68.5% (98/143) on T3, and did not differ significantly among groups.

Those who declined participation or completed the questionnaire on paper were analyzed as nonparticipants in this study. All participants provided written informed consent.

RCT participants were younger than nonparticipants (60.7 vs 65.3 years; P<.001), and more often men (161/227, 70.9% vs 377/666, 56.6%; P<.001). RCT participants were on average 14.0 months after diagnosis (SD 3.2 months). The majority of RCT participants had a partner (190/227, 83.7%). Of the RCT participants, 75.8% (172/227) received active treatment, mostly chemotherapy (137/227, 60.4%), whereas 23.3% (53/227) were on active surveillance. The majority of patients had Ann Arbor stage IV disease at the time of diagnosis. Other baseline sociodemographic and clinical characteristics did not differ across groups (Table 1).

The use of self-management intervention was very low (3/76, 4%). Therefore, the effects of Living with lymphoma could not be determined within this RCT and were not included in the results. The analyses were performed with the three original RCT arms to maintain a power of 90%.

As we observed that adherence to Living with lymphoma intervention was very low one year after starting patient inclusion, research assistants sent an email for inquiry. A random sample of 5 patients who had access to the intervention and signed up were asked to respond without any obligation and were asked for their reasons for nonadherence. Two patients responded that they felt well and still had regular appointments with their hematologist, and therefore were not in need of an intervention:

No statistically significant differences were observed for psychological distress, self-management subscales, and satisfaction with information provision between patients who received their individual PRO results and those who did not (Table 2). In addition, the return of PRO results did not have a significant effect on health care use. As no significant overall group-by-time interaction was found for the outcome variables, we were not allowed to explore specific contrasts.

Of the 150 patients who were randomized to arm 2 or 3 and had access to individual PRO results, 115 (76.7%) patients viewed their PRO results. The majority (79/115, 68.7%) viewed their PRO results more than once, and 16% (13/79) viewed it more than 5 times.

Patients with lymphoma who viewed their PRO results were more recently diagnosed (13.6 vs 15.0 months; P=.03), had a more conscientious personality (3.8 vs 3.6; P=.01), and had a less fatalistic coping style than those who did not (2.1 vs 2.2; P=.02; Table 3). In addition, patients who did view their PRO results more often wished to receive all available information about the disease compared with those who did not (68/115, 59.1% vs 10/35, 29%; P=.004).

Of those who viewed the PRO results, 91.3% (105/115) wished to compare their individual results to other patients with lymphoma and 80.8% (93/115) to a normative population without cancer. Only 7.8% (9/115) solely wanted to view their individual results.

The results of this RCT demonstrated that patients were neither in a better nor in a worse situation when their individual PRO results were disclosed, as no effects of return of PRO results were found on psychological distress, self-management skills, and satisfaction with information provision. In addition, patients who received their PRO results did not report more contact with their general practitioner or medical specialist compared with those receiving CAU.

Return of individual PRO research results seems to meet patients’ wishes, as the majority of those with access viewed their individual results, of whom two-thirds viewed it more than once. The possibility of comparing their scores with peers was most often chosen, indicating the importance of including normative data to place outcomes in perspective. Almost a quarter chose not to receive their results. Therefore, patients should have the choice as to whether they would like to receive their outcomes [14].

We observed little statistical differences in characteristics of patients who did view their PRO results and those who did not. Patients who viewed their PRO results more frequently wanted to receive all available information about the disease compared with those who did not view their results. This is in line with the literature that patients with a monitoring (information seeking) coping style tend to benefit from more provided information, whereas patients with a blunting (information avoiding) coping style benefit from less information [48,49].

There is increasing interest in integrating the collection of PROs in routine practice to enhance clinical care [50]. Weekly measurement of symptoms by patients during active treatment has proven to be effective, as 2 landmark studies on advanced solid tumors showed improvement in HRQoL and survival [51-53]. In our RCT, return of PRO results took place after treatment completion, including patients without advanced cancer stage, not specifically focusing on symptoms and was returned to patients only and not to their treating physician, which all may have contributed to differences in effects. With the continuing development of new and often expensive therapies for patients with cancer (eg, immunotherapy), further research and monitoring of the early onset and course of symptoms remains highly needed [54].

We could not compare our results to the evidence-based BREATH intervention for breast cancer [32,33], the original web-based self-management intervention from which Living with lymphoma intervention was derived, as uptake in our RCT was too low to study the effect. With respect to the design of the study, it was our explicit intention to examine the uptake and effects of a self-management intervention (without personal contact with a therapist) in a population-based setting (where all patients were asked, and no screening took place) to evaluate if this would be a possibility to provide low-intensity care to the continuously growing group of cancer survivors. This may have contributed to the much lower uptake compared with that of BREATH. Therapist guidance may improve patient engagement with a self-management intervention [55]. An important aspect of engagement is self-efficacy, which is a central element of several therapies, such as behavior change theory and planned behavior as a health access approach.

With respect to the Living with lymphoma intervention, we were not able to determine an effect as uptake was too low (3/76, 4%). Only 2 patients opened several components of the cognitive behavioral therapy parts, and various items from the library were viewed: Reliable information, Fatigue, Emotional counselling, and Nutrition and cancer, Exercise, Physical counselling, Sexuality, and Reintegration. As we observed that adherence was very low, we asked patients about reasons for nonadherence, and they indicated they felt well and still had regular appointments with their hematologist and therefore were not in need of an intervention. Furthermore, the majority of participants were men, and poor engagement in self-management was more common for men, as men may be more reluctant to seek help [56]. In addition, the timing of the intervention may not have been optimal, as participants were on average 14 months after diagnosis and patients appeared more receptive to interventions offered near diagnosis [57]. There is evidence that unguided self-management interventions could be more effective when targeted to those in the greatest need of an intervention, such as patients with low distress [32,58]. This might suggest that the need for intervention in our sample may be low and we may have not reached the right group, despite the invitation of a population-based sample and limited exclusion criteria.

This is the first RCT to study the effect of the return of individual PRO results to patients with lymphoma or to patients with cancer in general. The strengths of this study include the design and linkage of PROs with clinical data from the NCR. Owing to the design of our RCT, within a population-based observational cohort, we had access to sociodemographic information about nonparticipants. This provides a unique opportunity to make clear statements about the representativeness of the sample. Participants of the RCT were younger, more often male, and more often highly educated [59]. Ideally, the RCT sample will be representative of the entire target population so that generalizations about the population can be made. More research is needed to understand why underrepresented patients were not reached and how they could be reached in the future.

In conclusion, the return of individual PRO results seems to meet patients’ wishes, even though it had no beneficial effects on patient outcomes; it did not have negative effects either. Therefore, we decided to include and implement the return of individual PRO results in the PROFILES registry. In addition, at this moment, the return of individual PRO results is extended to other cancer types, such as colorectal cancer, and is used in daily clinical practice. No conclusions could be drawn about the effectiveness of the self-management intervention because the uptake was too limited.