Effectiveness of psychoeducation interventions in reducing negative psychological outcomes and improving coping skills in caregivers of children with cancer: A systematic review and meta‐analysis

Psychoeducation interventions (PEIs) have been used as an adjunct treatment for negative psychological outcomes in caregivers of children with cancer. This systematic review and meta‐analysis aimed to evaluate the evidence on the effectiveness of PEIs in reducing anxiety and depressive symptoms and improving health‐related quality of life (HRQoL) and coping skills in caregivers of children with cancer.


| INTRODUCTION
Cancer diagnosis in a child is stressful for caregivers and family members 1 ; that negatively impacts their psychological well-being. 2 Research has shown that alongside the burden of childhood cancer diagnosis, caregivers experience caregiving stress when caring for a sick child. 3While caregiving can be fulfiling, it can lead to psychological burnout due to the demands of providing care. 4Caregivers of children with cancer struggle with several stressors, including lack of knowledge about cancer and treatment, management of side effects, collaboration with healthcare workers, regular visits to the hospital for treatment, uncertainty regarding the child's prognosis, cancer communication, and multiple roles. 5,6Furthermore, caregivers of children with cancer reported unmet informational, emotional, and psychological needs. 7,8e stressors of child cancer diagnosis may be buffered by the coping skills adopted by the caregivers of children with cancer.
Coping is both an individual and an interactive process, and research shows that caregivers use adaptive and maladaptive coping skills. 9,10wever, other studies report that caregivers use more maladaptive coping skills, such as disengagement, self-blame, denial, and withdrawal. 11Prior findings have shown that maladaptive coping skills are associated with greater levels of psychological distress, such as anxiety and depressive symptoms. 12,13This confirms evidence in the literature that caregivers of children with cancer manifest signs of anxiety and depressive symptoms following a childhood cancer diagnosis, 14 which are known to negatively affect the caregivers' health-related quality of life (HRQoL). 15,16Therefore, interventions that support the psychological well-being of caregivers of children with cancer during the treatment phase of a childhood cancer diagnosis should be prioritised in paediatric oncology care. 17,18e psychoeducation intervention (PEI) is the therapy that aims to help individuals cope with the illness and improve psychological well-being through information giving, discussion of concerns, and cognitive-behavioural techniques. 19,20PEI has been categorised into counselling, cognitive-behavioural therapy, education, and social support. 21PEIs have previously been used as the adjunct treatment for managing negative psychological outcomes of psychological and physical illnesses and have been used in caregivers of children with cancer.One previous systematic review and meta-analysis was conducted to evaluate the effectiveness of PEIs in caregivers of children with cancer.Tang et al. 19 found that PEIs were not effective in reducing anxiety and depressive symptoms but effective in improving coping outcomes.However, the studies included in the meta-analysis compared psychoeducation interventions with usual care in combination with other alternative treatments. 19This may have affected the accurate determination of the effect of PEIs on caregivers of children with cancer over usual care.Further, the participants in the review were primarily caregivers of children newly diagnosed with cancer.The review did not identify an effective design and intervention components of PEIs.The current review will address these gaps by (1) identifying the active components and design of PEIs for caregivers of children with cancer undergoing cancer treatment and (2) evaluating the effectiveness of PEIs in reducing anxiety and depressive symptoms and improving coping skills and the HRQoL in caregivers of children with cancer.

| Protocol registration
A systematic review and meta-analysis of the randomised controlled trials was conducted using the Preferred Reporting Items for Systematic Review and Meta-analysis (PRISMA) guidelines to guarantee the quality of the review. 22The review was registered at the International Prospective Register of Systematic Reviews (PROSPERO) (registration number: CRD42022366229).

| Study selection
Two reviewers (LP and PGMC) independently selected the studies.
The titles and abstracts were first screened for eligibility based on the eligibility criteria.The studies that needed more details and full texts were retrieved and further reviewed.In case of discrepancies between the two reviewers, a consensus was reached after discussion.

| Data extraction
Two reviewer authors (LP and PGMC) extracted the data from all included studies.Any discrepancies in the extracted data were resolved through discussion, and the third reviewer was consulted if necessary.

| Assessment of risk of bias
Two reviewer authors (LP and PGMC) independently assessed the studies risk of bias using the revised Cochrane Risk of Bias (RoB 2). 23 assessed the risk of bias in the following domains of bias: bias from the randomisation process, deviation from the intended intervention, missing outcome data, measurement of the outcome, and bias in the selection of results.Based on the score for each domain, the studies were categorises as high risk of bias, some concerns of bias, or a low risk of bias.Any disagreements were resolved through discussion or consultation with third reviewer if necessary.

| Data analysis
To evaluate the effects of psychoeducation intervention, a mixed method analysis was conducted using meta-analysis and narrative synthesis.Meta-analysis was conducted to evaluate the effectiveness of PEIs on anxiety, depressive symptoms, and HRQoL.The analysis was performed using the random effects model because there was heterogeneity in the intervention components and dose of the interventions, and the statistical heterogeneity (I-squared) was >50%.
The standardised mean difference and a 95% confidence interval were used to determine the effect of PEIs considering that outcomes were measured differently among the included studies.The forest plots were used to present the results of the meta-analysis visually.
The extent and the impact of heterogeneity between studies was assessed by calculating the chi-squared and the I-squared, respectively.The interpretation of I 2 developed by Higgins et al. 24 was used.
Here 25%, 50%, and 75% are the cut-off points for low, moderate, and high heterogeneity.
The narrative synthesis was performed for coping outcome problem solving skill because of insufficient statistical data for metaanalysis (mean, standard deviation, and standard errors).The three stages stipulated by Popay et al. 25 were used to guide the data narrative synthesis.The stages included (1) developing an initial synthesis, (2) exploring relationships within and between studies, and (3) assessing the robustness of the synthesis.During the initial synthesis, the results of the included studies were organised in such a way to be able to describe patterns among them regarding the direction and magnitude of the effects reported in Table 1. 25 Relationships within and between the studies were then explored by grouping similar findings and identifying relationships between them.
In the next step, the robustness of the synthesis was assessed by reflecting on the synthesis process.The coping skills was analysed by looking at the reported significance level and calculating the effect size to quantify the magnitude of the effect of the intervention in that study.

| Study selection
The systematic literature search for the databases retrieved 9070 studies, of which 1739 studies were duplicates.After removing duplicates, the titles, and abstracts of 7334 studies were screened.
After screening titles and abstracts, 7291 studies were excluded, and 43 studies were assessed for eligibility, of which 14 studies met the inclusion criteria and were included in this review as shown in PRISMA flow diagram (Figure 1).

| Intervention characteristics
The key characteristics of the PEIs are summarised in Table 1.][34][35][36][37]39 Eight studies developed their interventions based on theoretical frameworks.The theories used were the transaction stress and coping model, 27,28 cognitive behavioural therapy, 32,39 resilient framework, 37 problem-solving therapy, 30,31 and a combination of resilient, stress, and coping theories. 29 this review, the contents of the interventions varied among the studies.However, based on the structured PEI, we can categorise the contents into four main components: health education, coping skills training, stress management techniques training, and psychological support. 40,41Almost all studies had a component of coping skills training.Seven studies had a component of stress management technique training [27][28][29]34,35,37,39 ; three studies had a component of cancer health education, [33][34][35] while two studies had a component of psychological support. 34,38 Tweve interventions were delivered individually, [26][27][28][29][30][31][32][33]35,[37][38][39] while two studies were delivered in groups.34,36 Nine studies were delivered through face-to-face format within the hospital 26,[29][30][31][32][34][35][36]38 ; two studies combined face-to-face and telephone follow-up sessions 28,35 whilst one study added web-based component in addition to face-to-face and telephone follow-up sessions. 27Three studies were delivered through the Internet. 33,37,39The interventions varied based on the intervention provider; in two studies, the interventions were delivered by a nurse, 36,37 whilst others were provided by a clinician, 27,28 a psychologist, 29 a social worker, 35,38 psychiatrists, 34 and two were self-administered through the internet. 37,39e intervention duration, frequency and sessions varied across the studies.The total intervention duration ranged from three weeks to 16 weeks, whereas the sessions ranged from three to 16 sessions.

| Risk of bias
Figure 2 summarises the findings of the quality assessment of the included studies.Overall, 12 studies were rated as having some concerns of bias; one was rated as having a low risk of bias 28 and a high risk of bias. 38Five studies were rated some concerns of bias in the randomisation process due to inadequate allocation concealment [27][28][29]33,35,36 whereas 12 studies were rated some concerns of Risk of bias summary.
PHIRI ET AL. -1521 31][32][33][34][35][36]39 Four studies were rated some concerns in the deviation from the intended intervention because the analysis did not follow the intention to treat analysis. 27,28,33Almost all the studies were rated as having some concerns in the risk of bias in the selection of reporting results due to the unavailability of protocol or statistical plan.One study was rated high risk on the randomisation process because they used card numbers that could easily depict which group will the next participant be allocated to. 38The funnel plots to detect publication bias were not conducted because the review included nine studies in the meta-analysis, less than the required number of studies. 42

| Depressive symptoms
8][39] The pooled analysis was performed in five studies that presented the relevant statistics for meta-analysis with 410 caregivers. 27,28,33,37,39The pooled analysis was also conducted on two studies with 199 caregivers greater than 6 months postintervention. 28,37The pooled results showed that PEIs significantly

| Coping
Three studies reported the effect of PEIs on coping outcomes. 30,31,36e to variations in how the outcome was measured and analysed, a meta-analysis was not performed.The coping outcomes reported were coping and problem-solving skills.Results showed that PEIs were effective in improving problem-solving skills 30,31 immediately post-intervention.The effect was maintained 3 months postintervention.Further the results showed that the PEIs were effective in improving the coping in all domains on coping health inventory for parents. 36The PEIs were effective on improving family integration (Effect size [ES] = 6.6, p < 0.001), Social support (ES = 9.2, p < 0.001) and understanding medical situation (ES = 5.05, p < 0.001) which was maintained at 3 months post-intervention.

| DISCUSSION
This review analysed RCT studies that evaluated the effect of PEIs in reducing negative psychological outcomes and improving coping skills in caregivers of children with cancer.The meta-analysis showed that PEIs were effective in reducing anxiety and depressive symptoms in the targeted caregivers.Thus, the current review supports the evidence that PEIs are effective in reducing anxiety levels and depressive symptoms in caregivers of children with cancer.This implies that PEIs are promising interventions that can be used by caregivers of children with cancer to improve their psychological well-being.The findings of this review are in line with the results of Cheng et al. 43 and Kusi et al. 44 In their meta-analysis of RCTs with PEI interventions on adult caregivers, both Cheng et al. 43 and Kusi et al. 44 found that PEIs reduced anxiety and depressive symptoms.
However, the findings of the current review are different from the findings of the meta-analysis by Tang et al. 19 Tang et al. 19 reported an insignificant effect of PEIs on anxiety and depressive symptoms in caregivers of children with cancer.It is important to note that in the systematic review and meta-analysis of Tang et al., 19 the findings were based on pilot studies with small sample sizes.The small sample sizes of the included studies may have underpowered the effect size. 19e effect of PEIs on reducing anxiety and depressive symptoms and improvement in HRQoL can be explained in line with the content of the interventions.For instance, the PEIs in the current review included health education, stress management technique, coping skills, and psychological support.This information may have helped the caregivers cope with caregiving challenges and psychological issues.Three of the included studies reported that the intervention-related improvement in caregivers' psychological functioning was associated with the development of stress management and coping skills 28,31,37 and the acquisition of knowledge of the disease, skills in managing the patient's symptoms and behaviour, and sharing of experiences with other caregivers. 34The acquisition of coping skills helped caregivers to manage their 1522 - emotions effectively. 45Furthermore, caregiver involvement in PEIs led to acceptance and better adjustment to the child's illness.
Therefore, it is crucial to incorporate PEI for caregivers early in child cancer diagnosis and treatment.
In this review, we found variations in the contents of the PEIs.
The structured psychoeducation model stipulates that PEI with multicomponent interventions are more powerful in affecting the expected outcome than those with single components. 46The structured psychoeducation model proposes four main components: health education, coping skills training, stress management techniques training, and psychological support. 46,47Evidence shows that incorporating these components into the PEI is critical in promoting psychological well-being. 48,49The analysis of our review showed that the included PEIs incorporated the components of health education, This may help caregivers to be resilient and cope with the stressors of childhood cancer diagnosis, thereby improving their emotional well-being.
Moreover, this review found that the most common interveners were healthcare workers who specialised in mental health, such as psychologists, psychiatrists, or mental health professionals.Only in two studies the intervention was delivered by the nurses.This suggests that PEIs provided by nurses as interveners to caregivers of children with cancer are in their infancy.Cheng et al. 43 and Chan et al. 20 49 They also reduce isolation and serve as a forum for recognising and normalising experience among participants and gives participants motivation, inspiration, and a sense of purpose. 36,49wever, group psychoeducation combined with individual consultation may be more beneficial and supportive for caregivers. 52The two modalities can be complementary in deepening and enriching the therapy. 53Therefore, the focus of future research should be evaluating the feasibility and effectiveness of group-based PEIs combined with individual consultations.
In this review, the variation in terms of the follow-up period for the intervention's effect was reported.The period varies from 2 weeks to 12 months, with 3 months being the most common.This variation shows that the follow-up period for the PEIs is not standardised.Further, based on the study by Foschiera et al. 54 , the followup period was divided into two categorises: <6 months for shortterm follow-up and >6 months for long-term follow-up; most studies included short-term follow-up.This may have affected the determination of the long-term follow-up of the effect of PEIs on anxiety and HRQoL.Therefore, more long-term follow-up studies are needed to enhance the understanding of the long-term effect of PEIs on caregivers' psychological outcomes and coping skills.
Confidence in the quality of the results of a systematic review depends on the methodological rigour of the included studies.
Despite showing substantial evidence of the effectiveness of the various PEIs, existing methodological flaws in the included studies may have reduced the quality of the evidence generated.For instance, some studies did not implement allocation concealment, blinding, and intention to treat analysis.According to Bezerra et al. 55 methodological flaws and heterogeneity affect the quality of evidence.Therefore, fixing current methodological pitfalls by adhering to allocation concealment, intention to treat analysis, blinding of outcome assessors needs to be the focus of future research in PEIs.

| Study limitations
The review has some limitations.First, the included studies are highly heterogenous, and some concerns of bias affected the quality of evidence, making it difficult to draw conclusions.Further, due to the insufficient number of studies on coping skill, a meta-analysis of the effect of PEI on this outcome was not performed.The included studies used the terms efficacy and effectiveness to determine the impact of psychoeducational interventions.Efficacy is how an intervention performs under ideal and controlled circumstances, whereas effectiveness is how an intervention performs under real-world conditions. 56Therefore, effectiveness in this regard can be encouraged to ensure more rigorous study designs.The review included pilot and feasibility studies that may reduce the validity of the overall results.Therefore, the findings need to be interpreted with great caution.

| Clinical implications and future research direction
This review highlights key points for clinical practice and research.
This review shows that PEIs can reduce anxiety and depressive numerous stressors when caring for their children with cancer.
Therefore, PEIs for caregivers are promising interventions that can be applied in clinical practice by different healthcare professionals.
The review proved that the PEIs for caregivers provided by nurses were as effective as PEIs given by other healthcare professionals with mental health expertise.Based on this finding, nurses are encouraged to develop skills in PEIs to provide psychological support to caregivers.
The review identified significant gaps in the existing literature on PEIs.One of the significant gaps is the need for well-designed RCTs.
The quality of the current evidence may still be subjected to scepticism to some extent due to the methodological gaps identified in the included studies.Future researchers will need rigorous designs with clear allocation concealment, blinding of outcome assessors, and appropriate data analysis methods to improve the quality of evidence.Furthermore, studies assessing the effect of PEIs in devel- The search results were limited to studies published in English.A manual search was performed to identify additional studies by screening eligible studies' reference lists and other reviews.All the searches were conducted in August 2022, and the search was updated in November 2022 with no new studies added.The complete search strategy is included in Supporting Information S1: Appendix A. The Population Intervention Comparison Outcome (PICO) framework was used to define the inclusion and exclusion criteria for the review.The studies which met the following criteria were included: (1) participants who were caregivers of children aged less PHIRI ET AL. than 18 years on cancer treatment within 1-year post-diagnosis; (2) evaluated psychoeducation interventions and their effect on anxiety, depressive symptoms, HRQoL, and coping outcomes.The psychoeducation interventions were defined as any therapy that used education or counselling, or behavioural intervention to help caregivers cope with the child's diagnosis: (3): used usual care, waitlist, or active control group as a control group; (4) studies which were Feasibility, pilot, and hypothesis testing Randomised Controlled Trials (RCTs).

F I G U R E 1
PRISMA flow diagram showing the screening process.

47 F I G U R E 3 F I G U R E 4 F I G U R E 5
stress management technique training, coping skills training, and psychological support; nevertheless, none of the included studies had all four components.According to structured psychoeducation model, one component of the intervention may be effective, but the variance accounted for each component is small.Combining various components into PEIs considers the different needs of caregivers and leads to better adaptation to stressful situations. 48Future studies should test the PEIs with the components of health education, coping skills, stress management, and psychological support as stipulated in the structured psychoeducation model. 46,Effectiveness of psychoeducation interventions on anxiety immediately post-intervention.Effectiveness of psychoeducation interventions on depressive symptoms immediately post-intervention and greater than 6 months follow-up.Effectiveness of psychoeducation interventions on health-related quality of life immediately post-intervention.PHIRI ET AL.Although the contents of the interventions in the included studies varied, the focus was stress management and promoting resilience in caregivers, thereby enabling caregivers to adapt to the stressful encounters of a childhood cancer diagnosis.Resilience provides an individual with the strength to cope with stressful situations.50Resilience is based on resilience factors such as positive emotions, self-esteem, active coping, optimism, social support, cognitive flexibility, spirituality, and thus resilience interventions can strengthen these factors.51These were the main contents in most of the included studies.For example, in this review, interventions with a problem-solving skills component appeared to be effective in promoting an individual's positive problem orientation and problemsolving skills.Stress management interventions helped to facilitate individual adjustment to stressful situations and strengthen individual coping abilities and confidence in using the coping collection.51 symptoms and improve HRQoL and coping skills in caregivers of children with cancer.Caregivers experience caregiving stress and 1524 -PHIRI ET AL.
oping countries have not been well documented.Therefore, additional evaluation studies, especially from developing countries such as Africa, are needed to expand the understanding of PEIs effectiveness.The review identified variations in the contents, dose, and form of the interventions.Future research is necessary to determine the most effective composition and dose of PEI, as intervention effects depend on the characteristics of the intervention.In addition, few interventions were delivered by the nurses, and future research should focus on nurses as intervention providers.Moreover, only one study evaluated the effect of coping, therefore more studies are needed to validate the effect of PEI on coping among caregivers of children with cancer.5 | CONCLUSIONThe current systematic review and meta-analysis support the evidence that PEIs have the potential to reduce anxiety and depressive symptoms and improve the HRQoL and coping skills in caregivers of children with cancer.However, methodological challenges in the included studies have the potential to affect the quality of the current evidence.In addition, the review found variations in the components and dose of the interventions.Therefore, more research is needed to determine the most effective combination of components and equivalent dosage, frequency, and duration of PEI.In addition, future research should determine the most effective modes of delivering PEI among caregivers of children with cancer.
found that nurses were the majority of the PEIs providers among the caregivers of adult patients with cancer.Nurses are considered appropriate interveners, and the PEIs delivered by nurses