Included studies

Sixteen studies were included for this review, 1 systematic review (Tellegen 2013), 12 randomised controlled trials (Benn 2012, Bilgin 2009, Chacko 2009, Chacko 2018, Giallo 2008, Haakonsen Smith 2018, Hoath 2002, Lange 2018, Plant 2007, Sonuga-Barke 2001, Treacy 2005, Taylor 2017), and 3 non-randomised studies (Costich 2009, Harris 2016, and Truesdale Kennedy 2006).

The included studies are summarised in Table 2.

Eight studies compared parent training to either a waitlist (n=5: Bilgin 2009, Benn 2012, Chacko 2018, Giallo 2008, and Treacy 2005); to standard care (n=2: Haakonsen Smith 2018, and Lange 2018); or to both a waitlist and standard care (n=1: Sonuga-Barke 2001). Three studies compared an enhanced parent training program to either a waitlist (n=1: Hoath 2002); or to standard parent training (n=2: Chacko 2009, Plant 2007). One study compared advocacy training to a waitlist (Taylor 2017), and 1 study compared person centred planning to standard care (Truesdale Kennedy 2006). Three studies compared before and after a parenting program called Stepping Stones Triple P: Positive Parenting Program (n=1: Tellegen 2013); a community health worker service (n=1: Costich 2009); or digital health technology (n=1: Harris 2016).

See the literature search strategy in appendix B and study selection flow chart in appendix C.

Excluded studies

Studies not included in this review are listed, and reasons for their exclusion are provided in appendix J.

Included studies

Two economic studies were identified which were relevant to this question (NEF Consulting 2009, Copps 2007). A single economic search was undertaken for all topics included in the scope of this guideline. See Supplement B for details.

Excluded studies

Economic studies not included in this review are listed, and reasons for their exclusion are provided in appendix J.

Economic

• There was mixed evidence from two cost-offset analyses based on modelling. One study showed that a short break service for disabled children and their families resulted in financial benefits. The other study showed that a short break service resulted in a cost increase. Although derived from UK studies, this evidence is partially applicable to the NICE decision-making context as it was unclear from the definitions provided how applicable the populations were. Both studies were characterised by potentially serious limitations. One study has not considered intervention costs. In the other study, some model inputs were based on authors’ assumptions, and it has not considering non-financial benefits.
• There was evidence from one cost-offset analysis based on modelling showing that a key worker service resulted in a cost increase in severely disabled children. This evidence, although derived from a UK study, is partially applicable to the NICE decision-making context as it was unclear from the definition provided how applicable the population was, and is characterised by potentially serious limitations, including some model inputs based on authors’ assumptions and not considering non-financial benefits.

The outcomes that matter most

Participation and inclusion of families and carers in the design, planning and delivery of care was prioritised by the committee as a critical outcome because it measured the level to which the role of families and carers are supported, which was the objective of this review question. Satisfaction with services was selected as a critical outcome due to the importance of providing person-centred services. Communication was selected as a critical outcome due to the importance of communication in enabling families and carers to interact with the child or young person, and subsequently be involved in the planning and delivery of care.

Quality of life (both health- and social-related quality), capability and confidence to meet the needs of the child or young person, social capital, and employment were agreed as important outcomes. Quality of life was considered an important outcome due to the importance of providing person-centred services. Capability and confidence to meet the needs of the child or young person was considered an important outcome due to the fact that when parents and carers feel capable and confident, they are more likely to be involved in the planning and delivery of care. Social capital was considered an important outcome because of the importance of access to social, emotional or practical support in supporting the role of families and carers. Employment was considered an important outcome due to the fact that many parents will give up employment in order to care for their disabled child or young person with severe complex needs.

No evidence was found for the outcomes of participation and inclusion of families and carers, quality of life and employment.

The quality of the evidence

The quality of the evidence was assessed with GRADE and was rated as very low to high. Concerns about the risk of bias ranged from “no serious” to “very serious”. The most serious concerns for the RCTs was bias arising from randomisation, missing outcome data, measurement of the outcome, and selection of reported results. The most serious concerns for the non-randomised studies were biases arising from confounding and measurement of outcomes. There were no serious concerns arising from the systematic review. Concerns about inconsistency ranged from “no serious” to “serious”. The serious concern related to the outcome capability and confidence to meet the needs of the child or young person when parent training was compared to waitlist, and was due to heterogeneity. Concerns about indirectness ranged from “no serious” to “serious” indirectness”. For all outcomes rated as “serious”, this was due to an indirect aspect of the PICO (population) in 1 study. Concerns about imprecision ranged from “no serious” to “very serious”. This was due to 95% confidence intervals crossing boundaries for minimally important differences.

Benefits and harms

There was evidence of a benefit of parent training in terms of communication, capability, and confidence to meet the needs of the child or young person, and social capital. However, important differences were demonstrated in the evidence when outcomes were measured with some scales, but not for all scales indicating there is some uncertainty in the results. Specifically, whilst a benefit was evident in parent training (compared to waitlist and standard care) for capability and confidence to meet the needs of the child or young person when measured with the Parenting Scale (PS) Overall, PS Verbosity, Parenting Sense of Competence (PSOC) Scale Satisfaction and efficacy subscales, and Everyday Parenting Scale, there was no evidence of a benefit in other measures including the PS Laxness, PS Over reactivity, Personal success subscale of the Duygun version Maslach Burnout Inventory, PSOC Efficacy subscale, coping self-efficacy and Parental Locus of Control Scale Efficacy subscale. Similarly, the benefit in social capital was evident when measured with the Seeking Social Support subscale of the Self-Report Coping Scale, whilst there was no evidence of a benefit in other measures including the Brief Social Support Questionnaire (BSSQ) Size and BSSQ Satisfaction. Given these differences, the committee interpreted the results with caution and used the evidence to recommend general elements of the parent training intervention rather than recommending any specific intervention. Further, the quality of evidence was variable and ranged from very low to high quality, however since the majority of evidence was low quality, the committee agreed that a strong recommendation could not be made. The parent training interventions focussed around psychoeducation and included stress management, coping effectiveness and behavioural strategies aimed at increasing the understanding of the child’s needs, providing tools for parents or carers to effectively support their child’s communication, and positively respond to their child’s behaviours. Therefore, the committee made recommendations that education, health and social care services consider jointly developing training, that is co-produced with parents carers, particularly in these areas [1.6.5]. Further, qualitative evidence reported service providers feeling that families needed more support to provide care for their disabled child or young person with severe complex needs (see evidence report M, sub-theme 3.3), so the committee included in the recommendation that training be designed to help parents and carers understand how services work and what support they can provide. There was also low and moderate quality evidence of a benefit of advocacy training where parents who were educated and enabled to advocate for services increased their capability and confidence to meet the needs of the child or young person. Therefore the committee recommended that training be designed to help parents and carers advocate for their child [1.6.6]. Some of the parent training interventions were conducted in group format and subsequently provided parents and carers the opportunity to engage with, and share experiences with other parents and carers of children and young people with similar disabilities and severe complex needs. The committee agreed that providing such opportunities would have a positive impact on parents and carers by helping them to learn from each other and develop support networks. [1.6.9]

There was evidence of a benefit of enhanced parent training in terms of satisfaction with services, and capability and confidence to meet the needs of the child or young person. However, the benefit of enhanced parent training (compared to waitlist) for capability and confidence to meet the needs of the child or young person was evident when measured with the PS Verbosity, whilst there was no evidence of a benefit in other measures including PS Laxness and PS Over reactivity. Similarly, when compared to standard parent training, the benefit in satisfaction with services was evident when measured with the Therapy Attitude Inventory (TAI) satisfaction with process and TAI satisfaction with outcome, whilst there was no evidence of a benefit in other measures including the Client Satisfaction Questionnaire. The evidence was very low and moderate quality and the intervention consisted of enhancements to the format, delivery and content of traditional parent training including training that was targeted at specific diagnoses. Given these differences, and the quality of the evidence, the committee interpreted the results with caution and used the evidence to recommend general elements of enhanced parent training interventions rather than recommending any specific intervention. The committee discussed that typically training should begin with basics and progress to become more detailed, but that training should be focussed to suit the individual as some parents and carers might require more in-depth training straight away, whilst others will not. Further, the committee agreed that practitioners who lead the training need to have skills that are appropriate for the content of the training so that people are not going beyond their skillsets [1.6.7]. If these things are not done, the committee agreed that the training is likely to be less effective. The same evidence also provided data on the availability of afterhours appointments and home-based sessions to encourage attendance. The committee discussed the difficulty for parents or carers to receive training due to conflicting commitments and/or circumstances, thus the need for training to be more flexibly delivered. There was evidence of a benefit of digital health technology in terms of capability and confidence to meet the needs of the child or young person. Although the evidence was of low quality, the committee agreed on the usefulness of digital technology (for example videoconferencing) as a format to deliver training, particularly in the current climate but they recommended using a variety of formats because not everyone will have access to digital technology. They also recommended considering aligning the timing of training with when the child or young person will be receiving prearranged care so that parents are available to attend [1.6.11]. Based on their experience, the committee felt the inclusion of parents and carers in the training could be facilitated further by considering the application of different teaching styles during the training, such as focus groups, role playing and problem based learning with these being tailored to the understanding and learning styles of those individuals attending. This would be particularly relevant for those where English is not their first language or who have poor literacy [1.6.8]. Based on their experience, the committee discussed that training may not always be offered to parents and carers at the optimal time. Whilst some parents and carers are eager to learn as much possible and as quickly as possible, others may need a period of time to deal with emotions such as grief or denial before embarking on training. Given the central importance of parents and carers receiving training to enable them to support their child, the committee felt strongly that parents and carers who decline training when first offered should not be disadvantaged, and should have the option to attend training at a later time when they are ready [1.6.10].

The committee acknowledged that different learning styles existed amongst parents and carers and it is important to involve parents and carers in the development of awareness training programmes (that are either for them or help practitioners work more effectively with them and their children), particularly in relation to the content, structure and evaluation to ensure that training is targeted and relevant and will result in the best possible outcomes. This was supported by qualitative evidence which reported parents and carers feeling positively when given the opportunity to provide their views, and expressing praise for practitioners that had recognised their expertise as parents and carers of the child or young person, whilst working within the boundaries of responsibility (see evidence report A, sub-themes 9.1 and 9.4). The committee felt that currently the development of training programmes did not always involve parents and carers or children and young people and therefore a recommendation was needed [1.15.21]. Further, the committee felt strongly that parents and carers should evaluate training against it’s objectives to ensure it is effective (otherwise providing it is would not be an efficient use of resources) [1.6.12]. As the objectives of training will all differ the committee did not specify what these were in the recommendation.

There was no evidence on psychological input and support at diagnosis. However, based on their knowledge and experience, the committee discussed that parents or carers may require emotional and practical support to enable them to come to terms with their child’s diagnosis and subsequently be effectively involved in the planning and delivery of care. In the committee’s experience, families often report a desire to have been signposted to these services earlier [1.6.1].

There was evidence of a benefit of person centred planning in terms of social capital. The committee agreed that a person centred planning approach would be beneficial in helping to include parents and carers in care planning but due to the low and very low quality of the evidence and that it only reported on one outcome they only made a weak recommendation [1.6.2].

No evidence was found on the effectiveness of short breaks to enable families/carers to be involved in the planning and delivery of care. However, the committee agreed, based on their experience, that short breaks were beneficial to involving parents and carers in the planning and delivery of care by providing needed respite. This was supported by qualitative evidence from parents who viewed short breaks as beneficial to the whole family as they allowed them to spend time with, and provide attention to other children in the family, and to address other practical demands such as household chores (see evidence report A, sub-theme 13.1). They noted that the Breaks for Carers of Disabled Children Regulations 2011 requires the provision of a range of short breaks but noted that in practice services can be constrained by the resources currently available in that area, whereas the most successful short breaks are those done in consultation with the parents or carers and tailored to their specific needs. The committee agreed that parents only want care as good as that which they provide themselves for their child or young person, and it is the bespoke offer rather than the ‘off the shelf’ offer that provides the most value to them. Therefore more needs to be done to provide short break options for children and young people and, the committee re-emphasised the duty in the Regulations for local authorities to provide a range of short breaks [1.17.13]. Due to a lack of evidence on the effectiveness of short breaks, it remains unclear which aspects of short breaks make them most effective, and why some short breaks are valued over others by disabled children and young people with severe complex needs and their families or carers. Therefore, the committee agreed a research recommendation was needed to determine which components of short break services provide the most effective short breaks for disabled children and young people with severe complex needs, their families and carers.

Cost effectiveness and resource use

The committee discussed the recommendations around general support to parents and carers coming to terms with a child’s needs and diagnosis. It was noted that since support services already exist and the recommendations are only about signposting to such services / organisations, not providing new support, this recommendation is not expected to result in a resource impact.

There was conflicting existing economic evidence from two studies on short break services. One study found that short break services result in substantial financial benefits but it did not consider the costs associated with providing a short break service. However, assuming intervention costs are the same as reported in Copps 2007 the financial benefits associated with the short break service would far outweigh the provision costs. Evidence from another study on short break services concluded that financial benefits gained were not sufficient to cover the provision costs. The committee acknowledged that both studies were only partially applicable to the NICE decision making context as it was unclear from the definitions provided how applicable the population was. The committee noted that the provision of short breaks is essential to improving outcomes for parents and supporting them to continue in their caring role. Also, whilst not the focus of this question, short breaks can improve outcomes for children and young people by improving their independence and confidence, which can ultimately mean less pressure on parents and carers to provide care. The existing economic analyses did not capture benefits on the child’s/ young person’s outcomes and therefore may have underestimated the cost-effectiveness of short break services. Given the uncertainties with the economic evidence and the lack of effectiveness evidence, the committee did not make a recommendation about short break services but re-emphasised the duty for local authorities to provide a range of short breaks as set out in the Breaks for Carers of Disabled Children Regulations 2011.

There was economic evidence from one study on key worker services. The analysis showed that key worker services costed more to provide than the value of the financial gains. However, under a certain set of assumptions, key worker services could potentially be cost-saving. It was acknowledged that this study was only partially applicable to the NICE decision making context because it was unclear from the definition provided in the study how applicable the population was. Also, this study was characterised by potentially serious limitations. As a result, and given the lack of effectiveness evidence, the committee could not draw any firm conclusions from this evidence.

The committee explained that person-centred approaches are currently used in planning care and so the recommendation about this was unlikely to result in any significant resource implications. This aligns with the committee’s understanding of the SEND Code of Practice (2015) that when commissioning training, consideration should be given to meeting the needs of children and young people with SEN or disabilities in a more personalised way.

Similarly, the committee discussed the recommendations around parent / carer training. The committee explained that, at present, services commission such training in isolation or only two services working together (i.e. local authorities with support from health services or health services could lead with the backing from local authorities). Parents and carers could also access such training through children’s services, parent / carer forums, and the disabled children’s teams. There is no dominant route to this. The committee agreed there is a need for a coordinated approach to training across all three sectors. The committee explained that there might be some costs associated with setting up the framework for such a collaborative approach. However, collaborative working will make the training approach coordinated, cut out duplication of effort and result in efficiencies and cost savings to the organisations involved. Similarly involving parents and carers in the development of such training will result in improved, more relevant content but there is unlikely to be a significant resource impact from doing so. The committee also recommended that the timing of training is aligned with times when the child or young person will be receiving prearranged care, education or familial care so there is unlikely to be a significant resource implication from providing care whilst parents attend training.

The recommendation on information provision is unlikely to have any significant additional resource implications as it is a requirement in the Children and Families Act 2014 for local authorities to make arrangements for providing parents with advice and information about matters relating to special educational needs and disability.

Effectiveness studies

Table 16. Excluded studies and reasons for their exclusion

Economic studies

See Supplement B for the list of excluded studies across all reviews.