Many previous studies have found that parents and carers of children with complex needs (such as disability, developmental delay or learning difficulties, or other chronic or complex conditions such as autism spectrum disorder) experience exceptional pressure to meet the emotional and physical needs of the affected child or children, while at the same time maintaining family functioning (Cheshire 2010; Lee 2007; McGuire 2004; Resch 2010; Strunk 2010). Parents of children with complex needs often show poor results on markers of psychosocial well-being such as quality of life and life satisfaction, and show elevated levels of psychological distress such as depression, anxiety, or stress (Cheshire 2010; McGuire 2004; Resch 2010). The daily care-giving activities and responsibilities of parents of children with complex needs can require extensive amounts of time, and can be physically and emotionally demanding (McGuire 2004; Resch 2010). These demands on the parent’s/carer’s time and energy can reduce the resources available for other meaningful and health-protective activities such as employment, social activities, and hobbies. Family and social relationships can be disrupted and parents left feeling overwhelmed, isolated and lacking support (McGuire 2004; Resch 2010; Strunk 2010).
Description of the condition
Families of children with complex needs report experiencing more stress than families of children who do not have complex needs, regardless of the child's particular condition (Van Riper 1992; Tak 2002).
Demands of caregiving
Parents caring for children with complex needs experience anxiety about their child's diagnosis and prognosis, and may also experience short-term emotional distress, loneliness, uncertainty and symptoms of depression (Barlow 2006). Physical caregiving activities, supporting the provision of therapy, and advocating for the child can prove extremely time-consuming (McGuire 2004). Parents may have difficulty gaining access to the services and resources they need (Banach 2010).
Behavioural problems may cause stress for families regardless of the underlying condition. For example, in families where children have a developmental delay, behavioural problems resulting from the delay were a greater contributor to increased parenting stress than was the developmental delay itself (Baker 2002). Parents may feel a lack of confidence in dealing with behavioural issues and suffer from a paucity of support services (Twoy 2007).
Changes to family life
A recent review found that chronic diseases in children interfere with daily life in the family, increasing the parents' burden of care (Barlow 2006). Balancing the healthcare needs of the affected child against other family needs, and the reduced time for other necessary or enjoyable activities, is a source of family stress (Banach 2010). In addition to stresses directly relating to the child's condition, families of children with complex needs must adapt to new roles, adjust their lives to cope with the needs of the child, and accommodate increased strain on family resources. As well as managing the needs of the affected child and any other children in the family, parents must cope with their own chronic stress and periodic family crises (Bourke-Taylor 2010; Dellve 2006).
Social stigma and isolation
Families in which a child has a chronic illness are at increased risk of isolation from formal and informal social support mechanisms (Tak 2002).
Challenging behaviours may make social outings difficult, a problem exacerbated by a lack of understanding in the community of the underlying condition (Twoy 2007), meaning parents may choose isolation over the frustrations of taking their child out in public (Tak 2002). Physical frailty of the child may similarly restrict parents' ability to maintain social networks.
Parents often feel a need to assist family and friends in handling their feelings about their child's condition, and to educate others about the condition (Dellve 2006). Parents can feel stigmatised (either through the direct actions and comments of others, or indirectly through their own attributions and anxieties about what others might be thinking). As a result, they may restrict social activities or may socialise only with other families whose children have a similar diagnosis. In some cases families may be excluded from social gatherings by others (Gray 2002).
Description of the intervention
The intervention of interest is the creation and maintenance of peer support networks or groups for parents and carers of children with complex needs. The intervention may encompass peer-led and facilitator-led interventions, where the focus is on fostering peer-to-peer interactions and increasing social support. One-to-one, group, face-to-face, and technology-assisted (e.g. conducted by telephone or Internet) interventions will be included in this review.
The aim of peer support interventions, for the purposes of this systematic review, is to enhance the social support (perceived and/or actual) available to participants; and via this means to improve the well-being of parents and carers across a range of psychological and psychosocial indicators.
'Children with complex needs' will be defined in the broadest possible terms to include children with any acute or chronic medical or psychological condition with relatively long-lasting course or sequelae. Examples of conditions for which peer support may be appropriate for parents and carers include but are not limited to: autism spectrum disorders (Whitaker 2002), cerebral palsy (Palit 2006), childhood cancers (Papaikonomou 2007), developmental delay (Buchan 1988), and juvenile rheumatic disease (Daniels 1987).
Peer support is defined in this review as the existence of a community of common interest where people gather (in person or virtually by telephone or computer) to share experiences, ask questions, and provide emotional support and self-help (Eysenbach 2004; Iscoe 1985). This is consonant with definitions used in published Cochrane reviews, with the additional stipulation that the specific knowledge possessed by the peer group is concrete, pragmatic, and derived from personal experience rather than formal training; and that the group consists of individuals who are perceived to be equal (Dale 2008). Peer support interventions can range from the purely emergent, informal, and member-driven, to those that are mandated, professionally-driven, and formal (Doull 2005).
This review will consider a continuum of interventions of various degrees of formality, but which all emphasise the role of personal experience in the provision of peer support. Interventions which utilise a formal or professional facilitator may be included, provided the facilitator's role is to manage group interpersonal processes rather than to provide counselling or psycho-education.
How the intervention might work
Peer support interventions are postulated to work by increasing the amount of social support available to parents and carers of children with complex needs, and providing that support in a form which is most useful and acceptable to participants. Current or prior affiliation with a support group (together with time and resources to adjust to diagnosis) has been suggested as a reason why families of children with complex needs displayed similar levels of function to families of children without such needs (Van Riper 1992). The perceived availability of support may play as great a role in determining stress levels in affected families as the actual support provided (Duarte 2005).
Peer support interventions are intended to supplement parents' existing social networks and reduce feelings of isolation and stigma by introducing individuals, who might otherwise not meet, to others who can appreciate and understand their experiences. Participants' circumstances should be similar but need not be identical. For example, parents who are in the early stages of adjusting to a diagnosis may benefit from the expertise of parents who have been coping with a diagnosis for longer; parents who have been living with their child's condition for some time benefit from feeling that their experiences have meaning for others and from taking on an expert role.
While social support is the primary goal, peer support interventions may additionally increase instrumental (tangible) support for parents by increasing access to local social and health services, and improve parents' knowledge about and confidence in managing their child's illness and other family issues.
Benefits of social support
Social support (defined as a combination of emotional concern, instrumental (tangible) aid, information, and appraisal) may mediate the stress experienced by families of children with serious physical, emotional, or behavioural challenges by contributing to coping resources (Lazarus 1987; Dunkel-Schetter 1987). It reduces stress in, for example, parents of children with severe learning difficulties (Quine 1991). Social integration and support protect against the potentially harmful effects of stressful family circumstances, but also have beneficial effects on well-being whether or not a person is currently under stress (Armstrong 2005).
Emotional support and hope
Reports from practitioners working with parents of children with complex needs reveal that these parents want emotional support (for example, someone to listen and understand), knowledge of others in a similar situation who are doing well, and to hear stories from others that give them hope for the future and make them feel less alone (Santelli 1996).
Reduction of isolation and stigma
Stigma, either experienced or feared, can lead parents to avoid contact with others. Combined with the time-consuming care tasks undertaken by these parents, this may mean an increased risk of isolation for the families of children with complex needs. Social support can be an effective buffer against isolation (Kerr 2000).
As well as buffering against stress, social support can have a direct effect on parenting stress by increasing exposure to incidental learning opportunities and competence-promoting social interactions: parents can benefit from the experience and knowledge of their peers without taking part in overt training and information sessions. A general survey of interactions between socio-economic status, positive and negative parenting behaviours, and child difficulties recommended interventions to strengthen parents' social relationships in order to reduce stress and create opportunities for parents to learn from and affirm one another (McConnell 2011).
Advocacy and self efficacy
Social support has been linked with enhanced advocacy skills and confidence in parents of children with complex needs (Banach 2010).
Valued instrumental (tangible) support includes information about specific disabilities and caring for children with complex needs, and ways to find and get access to services and community resources (Santelli 1996).
Social support provided by peers is suggested to provide reciprocal benefits: those receiving support gain the advantages described above, while people providing support also report enhanced quality of life and a validation of their previous experiences (Santelli 1997; Schwartz 1999). It has been suggested that social support must be reciprocal (or the possibility of reciprocity must at least exist) in order to be maximally effective (Hogan 2002). A group of peers of similar status provides a plausible arena for this egalitarian give-and-take of mutual support, in contradistinction to the power imbalance which may exist between service provider and service recipient.
Why it is important to do this review
There is a large number of self-help or peer support groups and programs which target parents and carers of children with complex needs (Canary 2008; Davies 2005; Hastings 2004; Law 2001). These groups aim to provide “social support, practical information, and a sense of shared purpose or advocacy” (King 2000, p. 226). It is widely believed that these groups or programs improve parental well-being through the social support mechanisms and peer support provided through the sharing of experiences, information, and understanding, and the provision of adaptive and credible models of coping (Davies 2005; King 2000; Lee 2007; McGuire 2004). However, despite much anecdotal evidence that these benefits do come from participation in such groups and programs (Ainbinder 1998; Davies 2005; Hartman 1992; Law 2001), little research has been undertaken to investigate the outcomes of participation in these groups and programs for the parent/carer, the child, and the family in general.
Social support networks are not always uniformly positive in effect (Ortega 2002). Peer support groups have the potential to damage self-esteem by reinforcing parents' self-image as a member of a stigmatised group, and social comparison can lead to negative affect (Hogan 2002); so it is important to find out when and how peer support interventions help, what barriers might exist to people's access to peer support, and if there are known negative effects.
Existing Cochrane titles in this area are:
- Peer support strategies for improving the health and well-being of individuals with chronic diseases (Doull 2005)
- Peer-based interventions for reducing morbidity and mortality in HIV-infected women (Doull 2004)
- Peer support telephone calls for improving health (Dale 2008)
This review will create a point of difference from the existing Cochrane reviews through its focus on the target population of parents and carers of children and adolescents with complex needs, rather than focusing on the individual with special needs themselves, or parents/carers of adults with special needs. This is an important and urgent area of investigation due to the exceptional pressures these parents/carers can be under to meet the needs of the child, often while maintaining a number of other roles. There is a large number of support groups and programs targeting parents/carers of children with complex needs, and it is important (given the competing demands on parents' time) to rigorously evaluate the outcome of these interventions.
If peer support programs are effective for parents and of children with complex needs, it is also important to know if particular subgroups receive more or less benefit than others, or if particular settings and modes of delivery are more effective than others. Some variables which may influence the effectiveness of peer support groups (and which should be investigated via subgroup analysis) include:
- How socially connected are parents before commencing the intervention?
While all parents may benefit from contact with parents in situations very similar to their own, those who are already well supported with social connections might be expected to benefit less. Alternatively, it may be that peers (as defined in this review) provide a benefit regardless of existing social connectedness.
- How is the peer support delivered: in pairs, in a group, peer-led, facilitator-led?
For example, do parents of newly-diagnosed children benefit more than parents who have been living with a diagnosis for longer, or vice versa; or is it best to match 'novice' with 'veteran' parents? Parents who have more experience living with a condition are well placed to provide information and tangible support to less experienced parents, and having gained some distance on the emotions associated with diagnosis may be better placed to provide emotional support than another parent at the same emotional stage. On the other hand such a matching process is administratively costly and may provide no extra benefit. It may be sufficient to assemble peer support groups with no matching process. Would such groups benefit most from being left to form their own connections and ways of operating, or would provision of a facilitator--a more expensive alternative--be of benefit?
- Should peer support be delivered face-to-face, or assisted by technology?
Must a group or pair meet physically, or are telephone and internet 'virtual' groups equally helpful? It may be that there is no substitute for interacting with peers in a physical setting; but on the other hand the benefits of reduced cost, travel burden, and time requirements may be sufficient to outweigh any reduction in the effectiveness of virtual peer support.
- Is there an optimum group size (apart from the support pairs considered above)?
More group members increases the resources available to the group, but there may be a point at which numbers become so large that the group fragments.
- Is there an optimum duration, either of individual sessions or of the intervention?
It may be that after a minimum time for support structures to develop and be felt by members, there is a duration after which no further benefits accrue. Alternatively, benefits may continue to be felt by members, but permanent peer support networks have been developed which no longer require participation in the intervention to be maintained.
- Is there an optimum timing for peer support interventions?
The need for support and the ability both to benefit from it and to offer it to others may differ between those with newly-diagnosed children and those who have been living with their child's condition for some time.
- Is it better for peer groups or pairs to be homogeneous (that is, age- or condition-specific)?
Parents of children with very similar conditions may have heightened potential to offer peer support due to having greater knowledge and experiences in common. In some cases, for example when located in hospitals, they may be relatively easy to set up. In most cases, however, matching parents on particular criteria will be administratively burdensome, and hence costly, in comparison with providing a venue and a referral mechanism only. It may also be the case that homogenous groups are unnecessary because the experience of living with any chronic or complex condition in a child is sufficient to create a peer group for the purposes of support.
We will, where possible, conduct subgroup analyses in order to answer these questions.
To assess the effects of peer support interventions on a range of psychological and psychosocial outcomes for parents and other family carers of children with complex needs, compared with control.
Attention will be given to both positive or negative outcomes for parents and carers associated with participation in such groups or programs. As caring and financial demands on these parents and carers are high, a secondary objective of this review is to collect and report data related to barriers to participation, as documented in qualitative research reported as part of the included studies on intervention effectiveness.
Criteria for considering studies for this review
Types of studies
Studies will not be limited to randomised controlled trials as, perhaps due to the self-directed nature of participation in peer support interventions, our preliminary searches of the literature have found that few if any randomised controlled trials have been performed in this area.
We will include:
- randomised controlled trials (RCTs) and cluster RCTs,
- controlled before and after (CBA) studies, and
- interrupted times series (ITS) studies.
To be included in this review, CBA and ITS studies must meet the Cochrane Effective Practice and Organisation of Care Review Group (EPOC) criteria (Ryan 2011). These specify, for CBA designs: at least two intervention sites and two control sites, comparable timing of measurements for control and intervention groups, and comparable key characteristics for control and intervention groups; and for ITS designs: a clearly specified intervention time-point and at least three data collection points before and after that intervention.
We will include studies with a broad range of control groups, including no-treatment, wait list, and usual care controls.
We will enhance this review by incorporating evidence from qualitative research which we may identify whilst searching for evidence of effectiveness (Noyes 2011b). We will include qualitative data associated with the studies of effectiveness described above, but will not conduct a separate search for qualitative studies and will therefore not include either qualitative-only studies, or conduct an exhaustive review of mixed-methods studies.
Types of participants
Parents and carers of children with complex needs, where complex needs include chronic or severe acute illness, disability, or delayed/atypical development in the physical, psychological, developmental, or intellectual domains.
Children will be defined as individuals aged 18 years or younger.
Types of interventions
The target intervention will be the provision of peer support through networks or groups. This will include peer-led and facilitator-led interventions where the focus is on peer-to-peer interactions and support. We will include one-to-one and group interventions, and both face-to-face interventions and those which are technology-assisted (i.e. conducted over the telephone or Internet).
We will include the following comparisons in the review:
- Any peer support intervention delivered to parents or carers of children with complex needs versus control (no-treatment, wait list, or usual care); and
- One peer support intervention versus another.
We will exclude studies if the effects of the peer intervention are not able to be separated from those of other intervention components. For example, studies will be excluded where the primary focus is other than developing and supporting peer networks (e.g. where professionals deliver an educative component or formal therapy), and improved peer networks are an incidental outcome.
Studies where the client or focus of the intervention is the child will only be included if direct outcomes for parents and carers are measured. For example, an intervention where the child is the primary client (such as play groups and early intervention programs) and parent peer support is an incidental putative outcome will be excluded from the review, unless this support and other parental outcomes are directly measured.
Types of outcome measures
The primary outcome of interest is the psychosocial well-being (assessed via a range of psychological, psychosocial and skills acquisition outcomes) of parents or other family carers following the intervention. We will assess outcomes for intervention participants, and for other non-participating family members where these are measured.
As social support (the target of the intervention) and well-being (the primary intervention outcome) are somewhat nebulous concepts, we anticipate that included studies will evaluate a broad range of measures, and limited overlap between measures may prove problematic. We will operationalise the primary outcome using measures developed by the Cochrane Consumers and Communication Review Group (CHCP 2012).
The following outcome categories are relevant to this review:
- Psychological health outcomes:
- Level of anxiety and depression
- Level of confidence and self-esteem
- Perception of coping
- Psychosocial outcomes:
- Quality of life, life satisfaction
- Family functioning
- Perceived social support
- Skills acquisition outcomes:
- Confidence and skill at navigating medical, community, and service support networks
- Knowledge of local resources
- Adverse outcomes:
It is generally assumed that increased social support (such as that putatively provided by peer support groups), improves psychological health and psychosocial outcomes. It may be, however, that psychological health of participants or particular subgroups of participants decreases following participation in peer support groups, or that perception of available social support worsens. This might be through mood contagion (anxiety or depression from hearing only negative experiences from other participants (Wojciszke 2009)), or through increased feelings of stigma from group membership, or from negative group interactions (such as difficulty fitting in or bullying within the group). If studies find that measures in the outcome categories listed above show a worsening of outcomes following participation, these will be treated as adverse outcomes. As the measures for adverse outcomes fit the same broad categories as those for beneficial outcomes, we will adopt the strategy outlined in the Cochrane Handbook, of assessing beneficial and adverse effects together using the same method, with common eligibility criteria for included studies (Higgins 2011a).
Each outcome category may include multiple outcomes that can be measured and included in that category. A risk is that a single trial might contribute multiple times to the same outcome category. We will adopt the approach common to the Cochrane Effective Practice and Organisation of Care Review Group (EPOC), as follows:
- Select the primary outcome identified by the publication authors,
- Where no primary outcome has been identified, select the outcome specified in the sample size calculation,
- If there are no sample size calculations, rank the effect estimates and select the median estimate. Where there is an even number of outcomes, we will select the outcome whose effect estimate is ranked n/2, where n is the number of outcomes.
The Cochrane Handbook (Higgins 2011a) suggests grouping into short-term, medium-term, and long-term outcomes, and taking no more than one of each from each study. We will include outcomes measured for all time frames for which data are available, subject to this constraint. If we retrieve studies whose primary outcomes are measured at the short-, medium-, and long-term, we will prefer longer over shorter-term outcomes.
Outcomes for 'Summary of findings' table
We will report primary outcomes using a 'Summary of findings' table (template provided in Higgins 2011a). We consider that the consumer-important outcomes essential for decision making regarding intervention suitability are:
- quality of life,
- perceived social support,
- confidence and skill at navigating support networks,
- adverse outcomes (in any of categories 1 to 5).
In addition to primary intervention outcomes, we will consider (where data are available):
- Parent/carer satisfaction with intervention
- Parent/carer incidental learning/improved knowledge
In addition to primary and secondary outcomes, we will consider process factors which may influence these outcomes:
- Contextual factors influencing the development of peer support: facilitators and barriers to uptake of peer support interventions,
- User and provider experiences of peer support interventions.
As these factors will be evaluated primarily through qualitative data identified in our search for the four study types identified above (mixed methods studies), this will add insight into how or why the intervention works (if it does in fact do so), but will not be completely comprehensive or systematic. The qualitative component of this review is intended to provide insight into why particular interventions are or are not effective for particular participants or particular outcomes.
Search methods for identification of studies
We will search:
- Cochrane Central Register of Controlled Trials (CENTRAL, The Cochrane Library, latest issue)
- MEDLINE (OvidSP) (1966 to present)
- EMBASE (OvidSP) (1974 to present)
- PsycINFO (OvidSP) (1887 to present)
- BiblioMap (EPPI-Centre, Health Promotion research database)
We will also search the Science Citation Index and the Social Sciences Citation Index.
We present the MEDLINE (OvidSP) strategy in Appendix 1. Strategies will be tailored to other databases and reported in the review.
There will be no date restrictions. We will seek translation of potentially-relevant studies in languages other than English.
Searching other resources
We will handsearch the reference lists of relevant retrieved articles to locate studies that are not identified by the database search.
We will search www.clinicaltrials.gov and the World Health Organisation clinical trials registry (www.who.int/ictrp) for relevant studies.
We will search Scopus conference proceedings. We will also consult databases at http://www.evidence.nhs.uk/ (Evidence in Health and Social Care), the New York Academy of Medicine Grey Literature report, and the OpenGrey system for information on grey literature in Europe for other avenues leading to unpublished studies.
We will consult advocacy and support groups, and contact colleagues and key investigators identified through other searches, for advice on identifying other unpublished data. We will also make use of existing professional connections with disability support and early childhood agencies in order to identify results which might otherwise be missed.
We will check trial registries (e.g. the International Clinical Trials Registry Platform Search Portal (www.who.int/trialsearch), and mRCT (www.controlled-trials.com/mcrt)) for trials commenced in recent years, for which data may exist which have not yet been published.
Identification of qualitative studies
As qualitative data will be captured from studies identified in the search outlined above (that is, we will not conduct a separate search for all qualitative studies on this topic), we anticipate retrieving only a subset (relating to our process outcomes) of the total qualitative studies conducted on this question.
Data collection and analysis
Selection of studies
Search strategies will be reviewed by the Cochrane Consumers and Communication Review Group before being executed. Bibliographic details for all search results will be consolidated and duplicate records removed using reference management software such as EndNote.
Abstracts will be reviewed and rated for inclusion independently by two authors based on the Criteria for considering studies for this review. Differences in rating will be resolved by a third author. We will record details of abstracts and decisions about inclusion, exclusion, or need to request further information from authors in an electronic database. Studies will not be excluded on the basis of outcomes; if an included study fails to report on an outcome of interest this will be reported in the review.
We will retrieve in full text all studies assessed as possibly relevant by one or more authors, based on assessment of title and abstract. Full text studies will be assessed against the Criteria for considering studies for this review, and any excluded at this stage will be listed in the Characteristics of Excluded Studies table, together with a reason for exclusion. We will use an adapted PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) flow-chart to describe the study selection process (Liberati 2009).
Data extraction and management
We will use the data extraction template provided by the Cochrane Consumers and Communication Review Group (Ryan 2011) for quantitative data relating to intervention effectiveness, revising where needed (for example, any ITS studies will need to be assessed using the Review Group's amended risk of bias tool (Ryan 2011), and the presence of any qualitative component addressing review outcomes will be noted) and piloting any revised version.
Data to be extracted will include the following items: details of the study (aim, design, recruitment method, statistical method); characteristics of the participants; characteristics of the intervention(s) including control/usual care; outcome measures; and results.
We will extract qualitative data identified during study search and selection using a template similar to that used by Noyes and Popay (2007, cited in Noyes 2011a). This template will be modified to enable the extraction of data relating to the process outcomes described above (Types of outcome measures). We will take the inclusive approach outlined in Noyes 2011a where all text or tables labelled as results in study reports will be included if they relate to our specified outcomes, not only direct quotations from respondents.
Data will be extracted by one author and will be cross-checked and confirmed by another; where necessary, consensus will be reached through discussion and consultation with a third author.
All extracted data will be entered into RevMan software (RevMan 2012) by one review author, and will be checked for accuracy against the data extraction sheets by a second review author working independently.
Assessment of risk of bias in included studies
We will assess and report on the risk of bias of included studies using the tool provided in the Cochrane Handbook (Higgins 2011b) and the guidelines of the Cochrane Consumers and Communication Review Group (Ryan 2011), assessing the following domains: random sequence generation; allocation sequence concealment; blinding (participants, personnel); blinding (outcome assessment); completeness of outcome data (attrition), selective outcome reporting; other sources of bias. This latter category will cover risks specific to non-randomised studies, such as selection bias (Higgins 2011a) and sources of bias associated with cluster-randomised trials (Higgins 2011c). We will judge each item as being at high, low, or unclear risk of bias as set out in the data extraction template adapted by the Cochrane Consumers and Communication Review Group (Ryan 2011, adapted from Higgins 2011a), and describe the relevant supporting information provided by the study authors for each item. Risk of bias will be addressed at study level and at outcome level.
RCTs will be deemed to be at the highest risk of bias if they are scored as being at high risk of bias on the sequence generation, allocation concealment, or incomplete outcome data domains. RCTs will be rated as at unclear risk of bias if they are rated as unclear on at least one of these three domains; while low risk of bias studies will be defined as those receiving a low risk of bias rating on all three of the sequence generation, allocation concealment and incomplete outcome data domains of the tool.
If quasi-RCTs are included in this review we will assess and report them as being at high risk of bias on the random sequence generation item of the 'Risk of bias' tool.
If cluster RCTs are included in this review we will also assess and report the risk of bias associated with an additional domain: selective recruitment of participants (described in Ryan 2011).
If CBA studies are included in this review, we will assess their risk of bias systematically using the adaptations developed by the EPOC Group, outlined in Ryan 2011. CBA studies will be assessed against the same criteria as RCTs, but will be reported as being at high risk of bias on both the random sequence generation and allocation concealment items. We will exclude these studies from the review if intervention and control groups are not reasonably comparable at baseline.
If ITS studies are included in the review, we will assess their risk of bias systematically using the adaptations developed by the EPOC Group, outlined in Ryan 2011. Specifically, we will assess and report the following individual items for ITS studies: intervention independence of other changes; pre-specification of the shape of the intervention effect; likelihood of intervention affecting data collection; blinding (participants, personnel); blinding (outcome assessment); completeness of outcome data; selective outcome reporting; and other sources of bias.
In all cases, two authors will independently assess the risk of bias of included studies, with any disagreements resolved by discussion and consensus. We will contact study authors for additional information about the included studies, or for clarification of the study methods as required.
Reporting and applying the 'Risk of bias' assessment
We will incorporate the results of the 'Risk of bias' assessment into the review through standard tables and systematic description and commentary about each of the elements, to obtain an overall assessment the risk of bias of included studies and a judgement about the internal validity of the review’s results. We will discuss the risk of bias for quasi-RCT, CBA, and ITS studies separately to the risk of bias for RCTs.
We will make use of the summary table suggested in the Cochrane Handbook (Higgins 2011b, table 8.7.a) to report the risk of bias across studies as well as within studies.
Other variables will be considered in our assessment of the reliability of findings, particularly: intervention setting and delivery mode and their likely effect on under- or over-estimation of effect; possible differences in treatment of different participant groups which might affect outcomes; and any limitations which affect many of the included studies that might render results less believable and generalisable (Ryan 2011).
All studies meeting inclusion criteria (including those relating specifically to CBAs and ITSs described above) will be included in our data synthesis, regardless of the outcome of the 'Risk of bias' assessment. These overall assessments of bias will be used to group included RCT and cluster and quasi-RCT studies for stratified meta-analyses (where relevant) as described in the Cochrane Handbook (Higgins 2011b, section 126.96.36.199). Data from CBA and ITS studies will be presented descriptively.
Other information which might affect judgements of study quality, but which do not constitute a source of bias (such as the reliability and validity of outcome measures and whether study authors obtained ethical approval), will be collected under 'Other measures of study quality' in the quantitative data extraction template.
Assessment of qualitative data
The qualitative data extraction template given as an example in Noyes 2011a will be adapted for use in this review, and quality will be assessed on the following domains: credibility, transferability, dependability, and confirmability. The template gives suggestions as to how these domains should be operationalised (for example, credibility is assessed using the prompts such as: Were there external auditors? Was attention paid to negative cases? Was analysis conducted independently by more than one researcher? Are verbatim quotes available to support findings?). Critical appraisal of these domains will use criteria suggested by Popay 1998, which are specifically concerned with the appropriateness of care and with lay and professional behaviour in a healthcare setting. In particular, we will look for evidence of responsiveness of research design to the real-life social settings met in the study, adequacy of sampling of participants, and evidence of moving from data description to analysis and interpretation.
Measures of treatment effect
Where both intervention and control baseline data are available, we will report pre- and post-intervention means.
We will present dichotomous outcomes as risk ratios (relative risk) and 95% confidence intervals (CI), based on the number of events and numbers of people in the control and intervention groups. We will also calculate risk difference, along with the number needed to treat to achieve the desired outcome. As advised in the Cochrane Handbook (Higgins 2011a, section 188.8.131.52) any results reported as odds ratios will be converted to risk ratios before interpretation.
For continuous outcomes, we will use final values scores in preference to change-from-baseline scores. If only change scores are reported in a study, and if it is not possible to extract the final score, mean and standard deviation, or other data necessary to calculate a standardised mean difference (SMD), we will report change scores in a separate table.
We will assume that included studies will use a variety of measures to assess the same outcome. We will therefore calculate the SMD and 95% CI using the mean, standard deviation and number of people assessed in control and intervention groups using the inverse variance method in Review Manager 5 software (Higgins 2011a, section 184.108.40.206; RevMan 2012).
If reporting CBA studies, we will report dichotomous and continuous outcomes as described above.
If reporting ITS studies, we will use the change in the level of outcome (extrapolating pre-intervention regression line to the first point post-intervention) or change in trend (post-intervention regression slope minus pre-intervention regression slope) as determined by the included study (Ramsay 2003).
Trichotomous outcomes may be relevant to this review (for example, classification of distress as mild/moderate/severe). The review is also very likely to encounter ordinal outcome data in the form of various scales. We will check such scales for established cut-points for conversion to dichotomous data; we will treat larger ordinal scales as continuous data.
Count measures may be relevant to this review (for example, numbers of social connections established). However, they are likely to be counts of common events and hence will be treated in the same way as continuous data; i.e. treatment effect will be measured via mean difference.
Qualitative outcomes may be reported in the studies included in this review (as described above), and insight from participants into the effects and meaning of the intervention is particularly relevant to this review topic. If sufficient qualitative data are identified in our search for relevant studies, we will undertake a qualitative evidence synthesis to supplement our main data synthesis. This qualitative data may be extracted from studies which are excluded from the quantitative synthesis on methodological grounds.
Our qualitative synthesis will explore process factors affecting implementation, such as facilitators and barriers to participation in peer support interventions, and such evidence is likely to be associated with trials identified in our search strategy as mixed methods research (Higgins 2011a, section 20.2.3). We will also contact trial principal investigators to identify unpublished qualitative data associated with trials. If identified trials do not meet eligibility criteria for inclusion but are accompanied by qualitative data of high quality as determined by the qualitative data extraction template (Noyes 2011a), this latter data will be incorporated into the review.
Unit of analysis issues
If the assessment of bias of individual RCT studies outlined above identifies sufficient studies for a meta-analysis to be appropriate (Higgins 2011a, section 9.1.4), the analysis will take into account the level at which randomisation occurred (e.g. individual, cluster, cross-over) in included studies.
If cluster-randomised trials are included, we will check that the original analysis was conducted appropriately for clusters as units of randomisation (rather than, incorrectly, for individuals). If an inappropriate analysis was performed, we will attempt to extract: (1) the number or mean size of clusters randomised to each intervention group; (2) outcome data for the total number of individuals, ignoring cluster design, and (3) an estimate of the intracluster correlation coefficient (ICC). This estimate will be sourced using the Health Services Research Unit 2012 online database and Ukoumunne 1999.
Once an appropriate ICC is obtained, we will follow the procedure for obtaining an effective sample size outlined in Higgins 2011a (section 16.3.4).
If it is not possible to obtain sufficient information to reanalyse the data, we will report effect estimates and annotate 'unit-of-analysis error'.
Dealing with missing data
In the first instance, we will attempt to contact authors of studies with missing data to request the missing data. If missing data cannot be obtained, we will seek the advice of a biostatistician and impute the missing data, with appropriate adjustments to the standard error to account for the added uncertainty in the results. If there are significant missing data, we will also consider how sensitive our results are to changes in the assumptions made to impute the data, and address the implications of any sensitivity in the Discussion section.
Assessment of heterogeneity
Where studies are considered similar enough, based on consideration of populations and intervention settings to allow pooling of data using meta-analysis, we will assess the degree of heterogeneity by visual inspection of forest plots and by examining the Chi
Where we detect substantial clinical, methodological, or statistical heterogeneity across included studies we will not report pooled results from meta-analysis but will instead use a narrative approach to data synthesis. In this event we will attempt to explore possible clinical or methodological reasons for variation by grouping studies that are similar in terms of the variables listed under Subgroup analysis.
Assessment of reporting biases
The existence of reporting bias will be assessed by testing for asymmetry of the funnel plot of intervention effect estimates against standard error of the intervention effect estimates. We will only proceed with these tests if there are least 10 studies included in the meta-analysis (Higgins 2011a; Sterne 2011).
The decision to meta-analyse or not will be based on an assessment on whether participants, settings, intervention, comparison, and outcomes are sufficiently similar to ensure a clinically-meaningful result. We will use a random-effects model, as we expect our included studies to be clinically heterogeneous.
We will meta-analyse RCT (including cluster RCT and quasi-RCT) studies. Meta-analyses will be stratified into low versus high or unclear risk of bias according to the methods described at Assessment of risk of bias in included studies.
If our 'Risk of bias' assessment indicates that many studies located are at generally high risk of bias, or if the risk is unclear for all, we will present an estimated intervention effect with a tabulated discussion of risk of bias.
If included RCTs are not suitable for meta-analysis (because they are insufficiently similar to ensure clinically-meaningful conclusions), we will follow the recommendation of Higgins 2011a (section 220.127.116.11) and display their individual results in a forest plot while suppressing a pooled estimate.
We will present data from CBA and ITS studies descriptively (for example as median effect sizes) and graphically where possible.
We will present tables of the results for studies included in the review but not suitable for meta-analysis, which will be grouped according to outcome, and will summarise study type, intervention sub-type (if relevant), participant characteristics, and the findings of the study. We will use the summary of the risk of bias of an outcome across studies to judge the robustness of this evidence (Cash-Gibson 2012). Tables of results will be used to form a narrative assessment of the evidence, clustering studies by intervention type and setting. For each study, we will report the same elements of information in the same order (Higgins 2011a, section 11.7.2).
We will conduct statistical analyses using the latest version of RevMan software (RevMan 2012).
Synthesis of qualitative data
If qualitative data are available for review, we will summarise them in a matrix and relate them to quantitative data in a narrative analysis. For example, qualitative findings may shed light on how easy or difficult people find it to participate in peer support interventions, which may influence primary outcomes.
Subgroup analysis and investigation of heterogeneity
As discussed in the Background, subgroups of the population may differ in their capacity to benefit from the intervention, and intervention mode and setting may also influence outcomes for different subgroups.
If our assessment of heterogeneity suggests that substantial heterogeneity is present in meta-analysed data, we will conduct subgroup analyses to investigate the variables (described in detail in the Background) which may influence the effectiveness of the intervention, such as:
- existing social connectedness of participants (where this is measured using scales with established thresholds for high versus low connectedness);
- delivery mode:
- paired versus group;
- peer-led versus facilitator-led;
- setting (e.g. face-to-face versus technology-assisted);
- duration and size. Preliminary investigation suggests that peer support group duration, frequency of meeting, and size vary considerably where details are specified (which is not universal) (Bywater 1984; Campbell 2004; Castelein 2008; Doull 2005). Cut-points for duration, frequency, and size comparisons will be determined following retrieval of studies. Typical ranges identified in the preliminary searches are:
- Duration of program: 10 weeks to 8 months;
- Duration of session: 60 to 90 minutes;
- Number of sessions: 8 to unbounded;
- Size of groups: 4 to 16 people;
- How recently the child's condition was diagnosed (e.g. parent referred to peer support intervention as part of diagnosis/initial medical intervention versus later referral);
- homogeneous (that is, condition-specific) versus heterogeneous conditions affecting children.
While some sensitivity analyses can be pre-specified in this protocol, many issues which could be suitable for sensitivity analysis will only become apparent during the review process when the individual peculiarities of the studies under investigation are identified (Higgins 2011a). Questions which may be relevant to this review are:
- For dichotomous outcomes, should odds ratios, risk ratios, or risk differences be used?
- For continuous outcomes, should results be analysed as a standardised mean difference across all scales or as individual mean differences across all scales? (Higgins 2011a)
- Where ordinal outcome measures have been converted to dichotomous or trichotomous outcomes, and where there is no consensus on cut-points for making such conversions, do different cut-points affect the results of the analysis?
- If missing data were imputed, do changes in the assumptions made for the imputation affect the results of the analysis?
'Summary of findings' table
We will prepare a 'Summary of findings' table to present the results of our meta-analysis, based on the methods described in Schünemann 2011. We will present the results of meta-analysis for the major comparisons of the review for each of the major primary outcomes, including potential adverse outcomes, as outlined in the Types of outcome measures section. We will provide a source and rationale for each assumed risk cited in the table(s), and will use the GRADE system to rank the quality of evidence using the GRADEprofiler software (Schünemann 2011). The standard elements of this type of table are: outcome, illustrative comparative risk (intervention versus control), absolute/relative measure of effect (as appropriate), numbers of participants and studies addressing the outcome, rating of the quality of the evidence for the outcome, and comments as necessary.
If meta-analysis is not possible, we will present results in a narrative summary organised by intervention type, population group, setting etc. as suggested in Schünemann 2011 (section 11.7.2). We will take Chan 2011 as the model for our 'Summary of findings' table.
The authors have strong links with early childhood and disability advocacy agencies in Australia. We will send our contacts in these agencies drafts of the review and seek their comment, especially on the discussion of and recommendations regarding the consumer-important outcomes reported in the summary of findings. We will also use our connections to work with local consumers and will follow up agencies' international networks to seek input from consumers overseas. Input from consumers may be in the form of comments on review drafts or, with the help of our collaborators, may be obtained from local focus groups convened to discuss the findings and their implications for consumers and future policy.
We will also invite facilitators of local peer support groups to comment on the review findings (in particular, the presentation of the 'Summary of findings' table), and seek their advice and the advice of advocacy agencies on the best way to circulate the review findings to other professionals in a position to refer consumers to peer support groups.
The authors thank Ms Danielle Bullen for assistance with initial scoping and preparation of our application to register the Cochrane review title. We also thank Dr Megan Prictor and Dr Rebecca Ryan for their extensive assistance in the preparation of this protocol, and Mr John Kis-Rigo for his help in drafting our MEDLINE search strategy. Dr Prictor, Dr Ryan, and Mr Kis-Rigo are from the Cochrane Consumers and Communication Review Group, based at La Trobe University's Centre for Health Communication and Participation.
Appendix 1. MEDLINE search strategy
1. exp child/
2. exp infant/
6. (child* or infant* or newborn* or baby or babies or neonat* or perinatal or adolescen* or youth* or juvenile or teen* or pediatric*).tw,hw.
8. exp parents/
9. exp parent child relations/
11. child rearing/
12. foster home care/
13. (parent? or parental or parenting or mother* or maternal or father* or paternal).tw.
15. (carer* or caregiver* or care giver* or guardian* or foster care or foster home*).tw.
17. 7 and 16
19. ((persistent or long* term or ongoing or degenerative) adj3 (disease* or ill* or condition* or insufficienc* or disorder*)).tw.
20. long term care/
21. long* term care.tw.
22. exp cardiovascular diseases/
23. (heart disease* or heart failure or myocardial ischemia or coronary disease* or coronary artery disease* or myocardial infarction or hypertension or high blood pressure).tw.
24. sickle cell.mp.
25. exp lung diseases obstructive/
26. (obstructive lung disease* or obstructive pulmonary disease* or copd or asthma or bronchitis).tw.
27. exp emphysema/
28. exp pulmonary emphysema/
30. (cystic fibrosis or respiratory distress).mp.
31. exp nervous system diseases/
32. (brain adj (disease* or damage* or injur*)).tw.
33. (cerebrovascular or brain ischemia or cerebral infarction or carotid artery disease* or stroke or epilep* or seizure*).tw.
34. (neurodegenerative or Huntington* or Parkinson* or amyotrophic lateral sclerosis or multiple sclerosis or motor neuron disease).tw.
35. (paralys* or quadriplegi* or tetraplegi* or paraplegi* or locked-in syndrome).tw.
36. ((communication or learning or consciousness or perceptual or speech or voice or vision or hearing or psychomotor) adj disorder*).tw.
37. (hearing loss or hearing aid* or deaf* or blind* or stutter*).tw.
38. down* syndrome.tw.
39. cerebral palsy.tw.
40. exp gastrointestinal diseases/
41. (gastroenter* or intestinal or bowel or colonic).tw.
42. renal insufficiency/
43. ((renal or kidney) adj (failure* or insufficienc*)).tw.
44. diabetes mellitus/
45. (diabetes or diabetic*).tw.
46. exp nutrition disorders/
47. (underweight or malnutrition or malnourished or overweight or obes*).tw.
48. exp arthritis/
49. exp rheumatic diseases/
50. (arthritis or osteoarthritis or rheumati* or fibromyalgia).tw.
51. ((back or neck) adj pain).tw.
52. exp thyroid diseases/
54. exp hypersensitivity/
55. (hypersensitivit* or allerg* or intolerance or anaphyla*).mp.
56. exp neoplasms/
57. (cancer* or oncolog* or neoplasm* or carcinom* or tumo?r* or malignan* or leuk?emia).tw.
58. exp hiv infections/
59. (hiv infect* or hiv disease*).tw.
60. exp mental disorders/
61. exp behavioral symptoms/
62. ((mental* or psychiatr* or psychological*) adj (ill* or disorder* or disease* or distress* or disab* or problem* or health* or patient* or treatment)).tw.
63. ((personality or mood or dysthymic or cognit* or anxiety or stress or eating or adjustment or reactive or somatoform or conversion or behavior or perception or psycho* or impulse control or development* or attention deficit or hyperactivity or conduct or motor skills or movement or tic or substance related) adj disorder*).tw.
64. (psychos#s or psychotic* or paranoi* or schizo* or neuros#s or neurotic* or delusion* or depression or depressive or bipolar or mania or manic or obsessi* or compulsi* or panic or phobic or phobia or anorexia or bulimia or neurastheni* or dissociative or autis* or Asperger* or Tourette or dyslex* or affective or borderline or narcissis* or suicid* or self injur* or self harm or adhd).tw.
65. (((substance or drug or alcohol) adj abuse) or "substance use" or "illegal drug use" or addict* or alcoholism or (problem* adj1 drinking)).tw.
66. exp "Congenital, Hereditary, and Neonatal Diseases and Abnormalities"/
67. (congenital* or abnormalit*).mp.
68. exp chromosome aberrations/
69. ((genetic or hereditary or chromosome) adj (disease* or disorder*)).mp.
70. exp disease susceptibility/
71. exp infant low birth weight/
72. infant premature/
73. (preterm or low birth weight).tw.
74. growth disorder*.mp.
75. exp disabled persons/
76. (disabled or disabilit* or handicapped or impaired or impairment* or dysfunction*).tw.
77. developmental delay*.tw.
78. ((behavio* or emotion*) adj1 (problem* or disorder*)).mp.
79. exp education special/
80. ((complex or special) adj3 need*).tw.
82. 17 and 81
83. ((lay or user*) adj2 (led or run)).tw.
84. (lay adj2 (expert* or person* or worker* or advisor* or consultant* or leader* or educator* or tutor*)).tw.
85. (layperson* or expert patient* or non professional* or nonprofessional* or non medical or nonmedical).tw.
86. peer group/
88. self help groups/
89. (((self help or support*) adj (group* or network*)) or ((parent* or carer* or caregiv* or care giv*) adj2 (group* or network*)) or (mutual adj (aid or support)) or ((social or community) adj2 network*)).tw.
90. parent to parent.tw.
91. community networks/
93. (mentor* or befriend* or buddy or buddies).tw.
94. voluntary workers/
95. ((trained or aide*) adj1 volunteer*).tw.
96. ((voluntary or volunteer) adj (work* or care* or service* or involvement or health* or help* or counsel* or staff or personnel or provider* or group* or organi#ation* or agenc* or sector)).tw.
97. ((online or on-line or internet or web or electronic or virtual) adj communit*).tw.
99. 82 and 98
100. randomized controlled trial.pt.
101. controlled clinical trial.pt.
102. clinical trial.pt.
103. evaluation studies.pt.
104. comparative study.pt.
108. research design/
109. follow up studies/
110. prospective studies/
111. cross over studies/
112. (experiment* or intervention*).tw.
113. (pre test or pretest or post test or posttest).tw.
114. (preintervention or postintervention).tw.
115. time series.tw.
116. (cross over or crossover or factorial* or latin square).tw.
117. (assign* or allocat* or volunteer*).tw.
118. (control* or compar* or prospectiv*).tw.
119. (impact* or effect? or chang* or evaluat*).tw.
121. exp animals/ not humans.sh.
122. 120 not 121
123. 99 and 122
Contributions of authors
GS conceived the review question, coordinated the submission of the protocol, and prepared the first draft of the protocol.
RM and VL provided advice on formulating the review question and assisted with protocol revision.
Declarations of interest
The first author is the manager of a federally-funded peer support program for parents and carers of children with complex needs in Australia. All three authors work at The Parenting Research Centre, which administers the program on behalf of the Australian government. A study of the effectiveness and acceptability of this program is in progress, conducted by the first and third authors and their colleagues. If this study is eligible for inclusion in the review, we will seek an external check of our study selection, data extraction, and data entry and analysis procedures by an independent third party.
Sources of support
- Parenting Research Centre, Australia.Staff time
- No sources of support supplied