Search results
The database search yielded 933 titles and abstracts in addition to another two articles from hand searching. Following the removal of duplicates, 890 titles and abstracts were screened, after which 874 were excluded. The full-text articles of 13 references were obtained and considered against inclusion and exclusion criteria. Eight studies were excluded for different reasons, as shown in the PRISMA chart. These left five studies to be included in the final review (see Fig.1 for the PRISMA flowchart).
Study Characteristics
Five studies were retained as they met the inclusion criteria, including two studies from Jordan and three from Egypt. These were published between 2008 and 2018. In total, 344 dyads of patients and their caregivers were recruited into the studies. The designs were two RCTs (36, 37), two non-randomized trials (38, 39) and one qualitative study (40). See table 1 for the descriptive characteristics of the studies.
Quality Assessment
First, as shown in table (2), the methodological quality was good for Hasan et al. (2014) and poor for Rami et al. (2018), which has a higher risk of bias. Rami et al. (2018) stated that randomization was accomplished. However, they were not explicit about the method of randomization or allocation concealment, which makes it open to the introduction of selection bias. Furthermore, the study protocol was not available to assess the reporting bias, and the study did not provide a hypothesis or power calculation. They had multiple primary outcomes, including clinical, social, quality of life and attitude towards medications. The primary outcome should be the one that has the most existing evidence in direct association with the exposure of the intervention (41). Therefore, there should be one primary outcome to do a power calculation. All statistical differences between arms were reported, but there was no report of effort to minimise bias.
Second, for non-randomized trials, the two studies have a high risk of bias because two or more criteria are not met according to the JBI tools (34) (see Additional file 2 for Methodological Quality of Non-randomized Trials).
However, there is not enough data reported to judge the quality in many instances. The study, done by Soliman et al. (2018), was reported as a cross-sectional interventional study. However, it is more consistent with quasi-experimental design because of the lack of randomisation and inclusion of the control and intervention group. The study risked bias in selecting participants because the sample was not randomized. Furthermore, the drop out was not reported, which could have affected the analysis. The study by El-Shafei et al. (2008), was reported as a case-control design, but the elements of the control group and randomization make it more consistent with experimental studies. They did not report the difference in basic characteristics between participants in both groups, which may have introduced a selection bias. They did not report a sample calculation, and they included 30 participants only. Furthermore, no details about attrition, loss to follow-up or outcome measurement were reported.
Overall, the quality of the included studies is poor, and none of these studies, except Hasan, et al. (2014) had the statistical power to detect the benefit of family interventions. This indicates that the included studies have a risk of overestimating the effect of interventions.
Third, the qualitative study done by Al-HadiHsan et al. (2017) is consistent with good quality studies according to JBI tool (34). The study did not follow any methodological theory for qualitative research because the authors were trying to answer the research question and explain the quantitative data. Two questions in the appraisal tool were not reported. First, locating the researcher culturally or theoretically in the study. Second, the acknowledgement of the potential influence of the researcher in the study and vice versa. Although these two points were not reported, they are more applicable to different qualitative methodological theories that were not followed in this study.
Intervention Characteristics
The interventions in the five studies were delivered in Egypt (3) and Jordan (2). The qualitative study (40) was a second stage from another study in the review (37). Despite the difference in the content, all the studies shared the component of psychoeducation, and two included communication and problem-solving skills (36, 37), while ElShafei et al. (2006) used counselling sessions. Furthermore, they varied in term of intervention characteristics like mode of delivery, duration and numbers of the sessions. All studies were individual-family sessions and attended by patients and their caregivers. Three of the studies were delivered in a clinical setting in the outpatient department (36, 38, 39), and one was delivered using a booklet within patients home (37). The duration of the intervention ranged from 12 weeks to 6 months. The individual session duration was reported in one study as 60 minutes (Rami et al., 2018). Health care providers or researchers led all the interventions, and none of them was delivered in inpatient settings. All the studies compared family intervention to standard care. See table 3 for the Intervention Characteristics Table.
Contents and Components of the Interventions
Two studies reported the process of adaptation and modification of the original manuals. Hasan et al. (2014) used the framework of Atkinson and Coia, which covers Bloom's Taxonomy of Learning Domains, while Rami et al. (2018) used the Behavioural Family Therapy (BFT) manual by Mueser and Glynn (1999). However, for the component of psychoeducation, they adopted the program prepared by ElShafie and colleagues (2002), which was developed for Egyptian people.
First, psychoeducation components included signs, symptoms, aetiology, diagnosis, treatment, and relapse signs and management strategies for schizophrenia. Furthermore, it included truths and myths about schizophrenia and how these affect the persons' thoughts, emotions, and behaviour. The treatment component includes information about medication, its side effects, anticipated benefits of the medicine, adherence to treatment, the importance of follow-up, and information regarding prognosis. Furthermore, leaflets, which contain information about schizophrenia, high expressed emotions families, notes and homework assignments for the problem-solving and communication skills training, were distributed to participants during the sessions (Rami et al., 2018). Second, communication enhancement training included learning skills for active listening, delivering positive and negative feedback, and requesting changes in each other’s behaviours. Third, problem-solving skills training included identification of specific family problems and practical advice for solving them. Fourth, the stress vulnerability model discusses the role of the family, burden of care, and stress management skills and strategies.
Strategies Used to Adapt the Intervention
The strategies for adaptation included different themes, but the common themes in all studies were language, context and delivery, and family. First, language adaptation was reported in all studies, and the content was modified and translated into simple Arabic, and the complexity of psychoeducation was simplified. Second, context and delivery adaptation appeared in all the studies as they delivered the intervention in individual therapy sessions instead of groups to facilitate the cultural context of Arabs. Third, all the studies acknowledged the vital role of the family and the distinct Arab family structure and processes.
Rami et al. (2018) was the only study which reported a detailed process of cultural adaptation. They piloted the intervention before the actual study to assess the acceptability and linguistic accessibility, and they modified the intervention accordingly. Furthermore, the theme of concepts and illness models was incorporated by increasing the number of sessions regarding the biological basis of the illness from one in the original BFT manual to two sessions. They adapted the content to incorporate cultural norms and practices by including folk stories relevant to the cultural and religious beliefs of the participants. Further to these adaptations, the program in Rami, et al. (2018) was shortened to six months instead of nine because of practical and financial reasons that may influence adherence and attendance.
Feasibility and Acceptability of the Interventions
Feasibility included the assessment of recruitment, attendance, retention (the proportion of participants who complete therapy sessions) and the compatibility of the interventions with the available resources. All the studies reported a feasible recruitment process without significant barriers or difficulties. The attendance was also feasible because two of the studies (36, 38) delivered the interventions during the follow-up appointment, which ensured a high level of attendance. The third study by Hasan et al. (2014) was delivered via a booklet to patients’ homes. The study by El-Shafei et al. (2008) did not report attendance. The assessment of retention was reported in two studies only (36, 37). Rami et al. (2018) reported that four subjects from the case group and six subjects from the control group missed their regular sessions. The dropout in the Hasan et al. (2014) study was six from the intervention group and ten from the control group. All the studies reported compatibility of the intervention with the available resources. The study by Rami et al. (2018) reported that the intervention was applicable and accessible because of the brevity of the program. Furthermore, meeting the needs of caregivers enhanced the feasibility of the program.
Acceptability is defined as "a multi-faceted construct that reflects the extent to which people delivering or receiving a healthcare intervention consider it to be appropriate, based on anticipated or experienced cognitive and emotional responses to the intervention" (42). Hasan et al. (2014) followed his trial with a qualitative study to assess the acceptability of interventions. The qualitative interviews with service users and caregivers confirmed the acceptability of the interventions. They found that interventions using booklets was appropriate and valuable. No other studies examined acceptability.
Effect of Interventions
The outcomes reported across the studies vary a great deal, and most of them did not distinguish primary from secondary outcomes. The most frequently reported primary or secondary outcome is the severity of symptoms using the Positive and Negative Syndrome Scale (PANSS). The four studies found a statistically significant difference between the two groups concerning positive and negative symptoms experienced by service users, favouring the intervention group. Furthermore, Hasan et al. (2014) found a reduction in the severity of symptoms at three months follow-up. Other frequently reported outcomes were social functioning, adherence to medication, quality of life and knowledge of schizophrenia. One study only assessed family outcomes, including the family burden of care and carers’ quality of life. (See Table 4 for the results of each outcome).