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Introduction, child development, developmental disability, early intervention for children with developmental disabilities, case studies of eci for children with developmental disabilities, the case for action, author's contributions, acknowledgements, competing interests, ethical approval.

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Early intervention for children with developmental disabilities in low and middle-income countries – the case for action

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Tracey Smythe, Maria Zuurmond, Cally J Tann, Melissa Gladstone, Hannah Kuper, Early intervention for children with developmental disabilities in low and middle-income countries – the case for action, International Health , Volume 13, Issue 3, May 2021, Pages 222–231, https://doi.org/10.1093/inthealth/ihaa044

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In the last two decades, the global community has made significant progress in saving the lives of children <5 y of age. However, these advances are failing to help all children to thrive, especially children with disabilities. Most early child development research has focussed on the impact of biological and psychosocial factors on the developing brain and the effect of early intervention on child development. Yet studies typically exclude children with disabilities, so relatively little is known about which interventions are effective for this high-risk group. In this article we provide an overview of child development and developmental disabilities. We describe family-centred care interventions that aim to provide optimal stimulation for development in a safe, stable and nurturing environment. We make the case for improving opportunities for children with developmental disabilities to achieve their full potential and thrive, including through inclusive early childhood development intervention. Finally, we call for the global research community to adopt a systematic approach for better evidence for and implementation of early interventions for children with developmental disabilities in low-resource settings.

Substantial global progress has been made in reducing child deaths since 1990 and the mortality rate of children <5 y of age has decreased in all world regions. However, non-communicable morbidities and disabilities have not been addressed to the same extent. This review discusses the urgency of taking actions to narrow the inequality gap in early childhood developmental care, especially for the 53 million children <5 y of age living with disabilities and developmental disorders such as epilepsy, intellectual disability, sensory impairments, autism spectrum disorder and attention deficit hyperactivity disorder. 1 A focus on supporting children with disabilities to thrive during their early years is important, as this period is critical for maximising their development. Furthermore, under the United Nations Convention of Rights for a Child and the United Nations Convention of the Rights of Persons with Disabilities, governments are duty-bound to provide early years services that are inclusive of and available to all children. 2 , 3 This article will describe child development and developmental disabilities and make the case for which equitable early childhood development (ECD) interventions may be optimal for helping children with developmental disabilities to achieve their potential.

Early childhood is a period of great opportunity for optimum brain growth, but it is also a period of vulnerability. Development in language, cognition, motor and socio-emotional domains occurs rapidly in these first years. These areas of development do not operate or develop in isolation, but enable each other and mutually interact as the child learns to become more independent. For instance, as a child learns to see, she will increasingly reach for and play with objects and thereby develop motor skills and coordination. Biological, psychosocial 4 , 5 and environmental factors also crucially affect the structure and functioning of the brain as it is developing. 6 For example, if a child experiences adequate nutrition and is provided with opportunities to play, she may progressively explore her environment and interact with her caregiver and by doing so, reinforce her psychosocial development. Furthermore, the time period when these factors influence brain growth are critically important, as there are particular early windows of opportunity that if not harnessed, may prevent optimal brain development and lifelong well-being. 7

It is increasingly apparent that optimal early child development has lifetime beneficial consequences for educational achievement, adult productivity and population health. 8–10 Conversely, exposure to biological and psychosocial risks negatively affects the developing brain and compromises the development of children. 5 Many structural factors determine these early child circumstances. These factors include a lack of nurturing care (nutrition, stimulation, good health) in the early years, as well as inadequate cognitive and psychosocial stimulation. 5 , 11 Children <5 y of age in low- and middle-income countries (LMICs) may be particularly at risk of poor development due to poor health and nutrition. 7

Child development can be encouraged through intervention in early childhood. 11 A number of mutually important elements are needed for maximising children's development. These include supporting responsive relationships, reducing sources of stress in the lives of children and families, building executive function and self-regulation skills and reinforcing contexts in which learning is most achievable across all developmental domains. 12 , 13 ECD interventions work to improve development through integrating family support, health, nutrition and educational services and providing direct learning experiences to young children and families. 14

The strategic focus of the World health Organization (WHO), United Nations Children's Fund (UNICEF) and World Bank ‘Nurturing Care Framework’ is therefore timely. 15–17 This action plan provides a framework for helping children survive and thrive through five strategic actions—lead and invest, focus on families and their communities, strengthen services, monitor progress and use data and innovate—and thereby aims to transform health and human potential. We know that urgent action is necessary to improve early childhood outcomes and ensure that all children reach their full potential as adults. Children with developmental disabilities must be included in this agenda, as they are a marginalised group with additional and specific needs and will otherwise be left behind.

Developmental delay and developmental disability are two distinct concepts. Developmental delay is often defined as a deviation from normative milestones; this may be in terms of delayed cognitive, language, motor and/or socio-emotional development. 18 The term developmental disabilities covers a range of childhood conditions and is used differently across different settings and cultures. 19 In this article we define developmental disability as a heterogeneous group of conditions that can impact on the development of children's function (e.g. sensory, cognitive, physical), with a very wide range of effects. 20 Developmental disability is the most common cause of childhood disability, with an estimated 53 million children <5 y of age living with developmental disabilities globally. 21 This estimate is based on only six conditions (epilepsy, intellectual disability, vision loss, hearing loss, autism and attention deficit hyperactivity disorder) and on present reporting of these conditions. It is likely therefore that the true number of children with developmental disability is much higher than this estimate, particularly if a broader age range is considered.

The majority of children with developmental disabilities live in LMICs, 21 and the prevalence is higher among families with high levels of poverty and low education. 27 However, there remain data gaps for the prevalence, epidemiology and causes of developmental disabilities in LMICs. 28 One reason for the uncertainty in the estimates is that identification of children with or at risk of developmental delay requires assessment using valid developmental evaluation tools to measure ECD 29 (Box 1 ), and these facilities are often not available in LMICs.

Identification of children with developmental disabilities

The impacts of developmental disabilities extend far beyond functional abilities. Children with developmental disabilities and their families are at high risk of social exclusion, exclusion from education and even stigma and violence. 30 Furthermore, looking after a child with developmental disabilities potentially places an enormous strain on families, and caregivers experience high levels of stress, anxiety, depression, physical exhaustion, stigma and discrimination. 31 This further increases the risk of mental ill health and social isolation in caregivers. A recent systematic review found caregivers of children with intellectual and developmental disabilities, when compared with caregivers of children without intellectual and developmental disabilities, experienced elevated levels of depressive symptoms (31% vs 7%, respectively) and anxiety symptoms (31% vs 14%, respectively). 32 There are also substantial costs to childhood disability, both the cost of additional services and resources required by the child and the lost income from parents who are caring for their child. Consequently, childhood disability may exacerbate poverty. 33 , 34 However, there is generally a lack of available services and support for children with disabilities and their families, especially in LMICs, which further compound these risks.

Evidence is limited, but growing, on the effectiveness of ECD interventions for children at risk of and with developmental delays, particularly in LMICs. 35 Indeed, many programmes and studies actively exclude children with developmental disabilities, as additional considerations may be required, and children with developmental disabilities may be unable to show progress when using developmental progress as the primary outcome 9 , 36–38 (Box 2 ).

Inclusion of children with developmental disabilities in clinical trials

Consequently, risks to delayed development are compounded for children with developmental disabilities, as they potentially receive less stimulation and fewer learning opportunities through other health service or care routes. 39 Exclusion of children with developmental disabilities from ECD thus perpetuates an already fragile cycle of development. We know that early childhood developmental intervention for these children is imperative, but we cannot inform planning and delivery of inclusive services for all children without better research in this area. For example, there are gaps in evidence-based approaches to monitoring and evaluation of ECD projects in LMICs, such as challenges in measurement of outcomes in routine programmes, which limit comparative understanding of impact, and in defining and monitoring quality and coverage. 25

Early identification of children with developmental disabilities, as well as early childhood intervention (ECI), improves children's opportunities to maximise their developmental potential and functioning as well as their quality of life and social participation. 40 , 41 Early identification and intervention are two distinct complementary strands; timely identification of children with developmental disabilities is required for early intervention, which strengthens the cumulative process of development, helping children acquire new skills and behaviours to reinforce and strengthen learning. In addition, some ECIs may have wider benefits for caregivers, such as through establishing support, thus helping build their knowledge, confidence and coping strategies, 32 with positive impacts for their mental health. However, data are lacking from LMICs and there is a paucity of implementation evidence to guide policymakers and donors. 33

ECI for children with disabilities can comprise a range of coordinated multidisciplinary services and can take many forms, including hospital- or clinic-based care, school-based programmes, parenting and community support and home-based childhood therapies. In high-resource settings, we know that family-centred interventions are more likely to result in the greatest satisfaction with services and improve psychosocial well-being for the child and caregiver. 42 With regards to impact, a systematic review of ECIs for children at risk of cerebral palsy demonstrated improved cognitive outcomes up to preschool age and improved motor outcomes during infancy, although variability in interventions limited the identification of which interventions are most effective. 43 Nevertheless, without such ECIs in LMICs, years lived with disability will be more than 3.3 million. 1

There are broadly two approaches to providing ECI for children with developmental disabilities, including children with disabilities in mainstream ECD interventions and targeted intervention programmes for children with disabilities. These approaches take many different forms, as they are used to support children and families with different needs. For example, universal programmes in the UK, such as the five mandated health visits for young children, are offered to all families. In contrast, targeted programmes, such as the Disabled Children's Outreach Service (DCOS), are aimed specifically at vulnerable families of children with a disability where the children are at higher risk of poor outcomes in later life. 44

While both inclusive and targeted efforts for children with disabilities at the level of early childhood centres have increased, 45 weak country health systems and conflict settings are major impediments to delivering high-quality services. 46 There remains a need for inclusive approaches for children with developmental disabilities in mainstream services, as well as within specialist ECIs. This means that the role of families can be particularly crucial to fill existing gaps in service availability.

A number of case studies have been identified for ECI for children with developmental disabilities. The following have been selected for description, as they illustrate different approaches for children with different developmental disabilities in several LMIC settings.

The WHO has developed Caregiver Skills Training (CST) for caregivers of children with intellectual disabilities. 47 , 48 The CST consists of nine group sessions and three home visits. The programme teaches strategies to promote communication and learning and address challenging behaviours. However, sustainable and scalable quality delivery of the group format by a lay facilitator remains a challenge due to limited integration in health systems. 49 Evidence of effectiveness is currently lacking, but randomised controlled trials are under way in Pakistan (Family Networks [FaNs] for Children with Developmental Disorders and Delays 50 ) and Italy, with future trials planned in China, Ethiopia and Kenya. 51

Interventions that aim to provide contextualised psychological support to caregivers of children with intellectual disabilities include ‘Titukulane’, a community group intervention that aims to reduce mental health problems among the parents of affected children. 52 This community-based intervention consists of eight modules that have been developed and piloted to help parents cope with the challenging role of caring for a child with intellectual disabilities.

Learning through Everyday Activities with Parents (LEAP-CP) is a family-centred intervention delivered peer to peer at home during 30 weekly 2h visits that aims to improve the mobility of children with cerebral palsy. 53 Visits include therapeutic modules (goal-directed active motor and cognitive strategies and LEAP-CP games) and parent education. Randomised controlled trials are currently under way in India. 54 The trial also provides nutrition and health support to all families in the study, which may influence the findings.

The London School of Hygiene & Tropical Medicine (UK) has developed three caregiver group interventions under the ‘Ubuntu’ umbrella (resources available from www.ubuntu-hub.org ). The interventions consist of 10 sessions, the content of which includes information about essential care practices, such as feeding, positioning, communication and play, offered through a local support group format. ‘Getting to know cerebral palsy’ was developed as a resource to empower families using a participatory approach at the community level. 31 , 55 The ABAaNA Early Intervention Programme (EIP) was developed in response to a recognised need to support families of very young children (<2 y) with an evolving developmental disability. 56 ‘Juntos’ was developed for children with congenital Zika syndrome and their families in Latin America and integrates a strengthened component on caregiver emotional well-being, arguably fundamental to a child's early development. 57–60

Interventions for children with autism spectrum disorder include PASS, a parent-mediated intervention for autism spectrum disorder in India and Pakistan. 61 The intervention uses video feedback methods to address parent–child interaction and was adapted for delivery by non-specialist workers. As PASS is focused on improving a child's social communication, common mental health comorbidities such as sleep difficulties will be important to integrate into wider intervention programmes.

These examples provide good case studies of diverse interventions for different children with developmental disabilities in different low-resource settings. These case studies indicate that in LMICs, the gap in meeting the holistic needs of children with developmental disabilities may be addressed through the use of community-based group interventions facilitated by trained and supervised health or peer support workers. Commonality is the focus on caregiver involvement, which is critical, particularly where there are few health services. Yet formal evaluation of their effectiveness and cost-effectiveness is lacking, in addition to limited implementation with education and social welfare, which hampers scaling of these services.

The number of children with developmental disabilities is large and the impacts on the child and family are extensive. There are valuable lessons learned from case studies, yet there remains insufficient progress in ECI for children with developmental disabilities and unmet needs are widespread. The causes of this gap are complex and diverse. An important reason is that in many settings health services are often fragile, poorly coordinated and overstrained, with concerns about the availability and quality of healthcare workers capable of delivering the intervention. Health systems gaps are particularly important in fragile states, including those affected by war and famine, as they experience many competing pressing needs. Furthermore, the policy agenda supporting a focus on children with developmental disabilities is weak internationally and nationally in many cases, limiting the priority given to this issue and the availability of funding for developing services. Ensuring inclusive education is a clear responsibility for United Nations member states under international treaties and Sustainable Development Goal 4, to ‘ensure inclusive, equitable quality education for all’. However, investing in inclusion prior to schooling is not mandated and consequently becomes optional. Cultural challenges also exist, such as widespread stigma and discrimination around children with disabilities and their families. 62 Finally, the evidence base on needs for and effectiveness of services is currently weak and needs to be strengthened. Enhancing environments that provide equal opportunities for children with developmental disabilities for ECI therefore requires a systems approach with global collaboration.

Accordingly, priorities for future research to ensure that all young children reach their development potential include assessment of the effect of interventions for children with developmental disability and their families in different low-resource settings. Further identification of barriers to accessing general services (e.g. primary healthcare) as well as specialist services is also required, as poverty remains a major issue for affected families in LMICs. Furthermore, studies that identify how to maximise the reach and cost-effectiveness of ECD interventions for children with developmental disabilities are warranted. Evaluation of how these interventions can be embedded within health systems are needed to strengthen the service delivery strategies. Global collaboration in these efforts are required in research, and critical steps include providing best evidence on practices to improve knowledge and skills at local levels to avoid children with developmental disabilities being turned away from existing services and evidence of ‘what works’ to provide sustainable, inclusive ECD interventions with impact in resource-constrained settings. We call for international research communities, including funders, to adopt a systematic approach for better evidence.

ECD interventions are aimed at improving the development of children. However, children with developmental disabilities are often excluded from these programmes, even though they have the greatest need for support. There is still a dearth of research about what interventions are effective in improving outcomes for this marginalised group and an even greater lack of evidence on cost-effectiveness and what can be successfully implemented at scale. A two-pronged approach is likely to be optimal, encouraging the inclusion of children with disabilities in mainstream ECD programmes, while also offering targeted approaches, most likely through caregivers. We call for global collaboration among international research communities, including funders, to adopt a systematic approach to strengthening the available evidence base of interventions for children with developmental disabilities and their families. We call for greater attention for this marginalised group, to prioritise public policies and hold governments accountable to ensure that multisectoral services centred around the child and his/her family are provided during this crucial time. This will contribute to ensuring that all children have an early foundation for optimal development, a key factor in equitable long-term health.

HK conceived the study. TS carried out the analysis and interpretation of case study data. TS and HK drafted the manuscript. MZ, CJT, MG and HK critically revised the manuscript for intellectual content. All authors read and approved the final manuscript. TS and HK are guarantors of the paper. The data underlying this article are available in the article and in its online supplementary material.

This work was supported by the Wellcome Trust and Department for International Development (grant 206719/Z/17/Z to HK). The funders had no role in the study design, data collection and analysis, decision to publish or preparation of the manuscript.

MG is a member of expert panels for the WHO and UNICEF on measurement of childhood development and disability. This research paper was undertaken outside and separate from these duties.

Not required.

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Completed clinical trials with a focus on developmental outcomes

• child development
• developmental disabilities
• disabled children
• early intervention (education)

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• Research article
• Open access
• Published: 02 February 2024

Strategies for addressing the needs of children with or at risk of developmental disabilities in early childhood by 2030: a systematic umbrella review

• Tracey Smythe   ORCID: orcid.org/0000-0003-3408-7362 1 , 2 ,
• Nathaniel Scherer 1 ,
• Carol Nanyunja 3 , 4 ,
• Cally J. Tann 3 , 4 , 5 &
• Bolajoko O. Olusanya 6  

BMC Medicine volume  22 , Article number:  51 ( 2024 ) Cite this article

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There are over 53million children worldwide under five with developmental disabilities who require effective interventions to support their health and well-being. However, challenges in delivering interventions persist due to various barriers, particularly in low-income and middle-income countries.

We conducted a global systematic umbrella review to assess the evidence on prevention, early detection and rehabilitation interventions for child functioning outcomes related to developmental disabilities in children under 5 years. We focused on prevalent disabilities worldwide and identified evidence-based interventions. We searched Medline, Embase, PsychINFO, and Cochrane Library for relevant literature from 1st January 2013 to 14th April 2023. A narrative synthesis approach was used to summarise the findings of the included meta-analyses. The results were presented descriptively, including study characteristics, interventions assessed, and outcomes reported. Further, as part of a secondary analysis, we presented the global prevalence of each disability in 2019 from the Global Burden of Disease study, identified the regions with the highest burden and the top ten affected countries. This study is registered with PROSPERO, number CRD42023420099.

We included 18 reviews from 883 citations, which included 1,273,444 children under five with or at risk of developmental disabilities from 251 studies across 30 countries. The conditions with adequate data were cerebral palsy, hearing loss, cognitive impairment, autism spectrum disorder (ASD) and attention-deficit/hyperactivity disorder. ASD was the most prevalent target disability ( n  = 8 reviews, 44%). Most reviews ( n  = 12, 67%) evaluated early interventions to support behavioural functioning and motor impairment. Only 33% ( n  = 10/30) of studies in the reviews were from middle-income countries, with no studies from low-income countries. Regarding quality, half of reviews were scored as high confidence ( n  = 9/18, 50%), seven as moderate (39%) and two (11%) as low.

Conclusions

We identified geographical and disability-related inequities. There is a lack of evidence from outside high-income settings. The study underscores gaps in evidence concerning prevention, identification and intervention, revealing a stark mismatch between the available evidence base and the regions experiencing the highest prevalence rates of developmental disabilities.

Peer Review reports

There are approximately 53 million children under 5 years of age with developmental disabilities worldwide [ 1 ]. Prevalence varies widely across regions and countries, with low- and middle-income countries (LMIC) experiencing a higher prevalence of developmental disabilities than high-income countries [ 2 ]. Developmental disabilities are a diverse group of conditions that affect a child’s physical, cognitive and social development [ 3 ]. These conditions encompass cerebral palsy, intellectual and learning impairments, epilepsy, hearing and vision impairment and autism spectrum disorder and attention deficit/hyperactivity disorder [ 4 ]. Typically, these conditions manifest during early childhood and can have a lifelong impact on children, their families and communities [ 5 ]. Children with developmental disabilities may experience delays in reaching developmental milestones, difficulty with social interactions and challenges in accessing and continuing education [ 6 ]. These challenges can have long-term consequences, such as decreased employment opportunities and increased dependence on caregivers [ 7 , 8 ]. Families of children with developmental disabilities may experience financial strain, social isolation and mental health issues [ 9 ]. Nevertheless, despite efforts to improve child health and well-being, children with developmental disabilities continue to experience health disparities, social exclusion and limited access to care, particularly in LMIC where the majority of affected children live [ 10 , 11 ].

In this context, the Sustainable Development Goals (SDGs) aim to achieve universal health coverage, reduce poverty and promote social inclusion, amongst other goals by 2030 [ 12 ]. SDG 4 is dedicated to early childhood development and care; specifically, Target 4.2 calls for actions to facilitate school readiness for children with disabilities towards inclusive education. These goals require the identification of children with or at risk of developmental disabilities in the first 5 years of age and the provision of services to address their needs before school entry [ 13 ]. However, despite the growing number of children with developmental disabilities, global funding schemes for early childhood development do not adequately address the challenges faced by these children and their families [ 14 ].

While services for children with and at risk of developmental disabilities (encompassing prevention, identification and rehabilitation interventions) are often perceived as highly specialised and costly, it is crucial to understand and provide evidence for comprehensive support that may not be so. For instance, evidence-based developmental screening tools integrated into regular early childhood check-ups can streamline identification of potential challenges early on, leveraging existing healthcare infrastructure [ 15 ]. This integration eliminates the need for extra appointments, ensuring timely support and contributing to intervention sustainability by utilising the existing network of healthcare professionals, making essential care accessible to a wider population and broadening their impact. Access to care and support should begin with ensuring that routine child health services and education are inclusive of children with disabilities [ 3 ]. By embedding inclusivity at this foundational level, we pave the way for a more equitable and supportive environment that can foster better developmental outcomes [ 16 ].

Consequently, amidst this drive for equitable access and comprehensive support, there is a growing interest in early identification of developmental disabilities, spurred by a global commitment to equity and inclusive education [ 17 ]. However, this poses practical and ethical challenges when suitable services are not available for identified children, particularly in LMIC. The goal of early identification is universal, and some methods and tools used in high-income countries can be beneficial without requiring significant adaptation, depending on the specific disabilities. For example, corrective glasses may not need adaptation to be prescribed in all populations. It is therefore essential to consider contextual differences and carefully assess how evidence-based interventions can be adapted and effectively implemented in various settings to ensure their relevance and effectiveness for the target population. Stigma, discrimination and exclusion further emphasise the need for a transformative approach to early care and support, because they perpetuate societal inequalities, hinder access to essential services and reinforce barriers that impede the holistic development and well-being of children with developmental disabilities [ 18 ].

In light of these considerations, this paper sets out to summarise available data on the prevalence of eight prominent developmental disabilities in children younger than 5 years, and the evidence-based interventions for prevention, early detection and rehabilitation. For the purpose of this review, we use the terms “early intervention” and “rehabilitation” for children under 5 with developmental disabilities to refer to timely and targeted strategies that address and mitigate challenges in physical, cognitive, communication and social development. These interventions may encompass a range of services, therapies and support systems designed to enhance their overall well-being, functional abilities and potential for successful integration into society as they grow.

This umbrella review was conducted following the Preferred Reporting Items for Overviews of Reviews (PRIOR) statement for conducting umbrella reviews [ 19 ]. The protocol for this systematic umbrella review was registered in the International Prospective Register of Systematic Reviews (PROSPERO), reference number CRD42023420099. A comprehensive search of electronic databases was conducted on 14th April 2023, including Embase, Medline, Cochrane Library and PsycINFO, to identify relevant systematic reviews and meta-analyses published in English in the last 20 years (from January 2003 to May 2023). The search strategy included relevant keywords and MeSH terms related to developmental disabilities, prevention, early detection, rehabilitation and children under 5 years of age.

For example: ("PREVENTION" OR "EARLY DIAGNOSIS" OR "EARLY DETECTION" OR "REHABILITATION" OR "EARLY INTERVENTION) AND ("DISABILITY" OR "IMPAIRMENT" OR "DISORDER") AND ("CHILD*" OR CHILD* UNDER FIVE OR CHILD* UNDER 5").

Inclusion and exclusion criteria

Meta-analyses that met the following criteria were included in this umbrella review:

Population: Children under 5 years of age diagnosed with or at risk of developmental disabilities, including autism spectrum disorder, attention deficit/hyperactivity disorder, cerebral palsy, epilepsy, hearing loss, intellectual disability, learning disabilities and vision loss. No distinction was made between reviews that evaluated population-based primary studies and those based on a random sample of participants.

Interventions: Evidence-based interventions for prevention, early detection and rehabilitation of developmental disabilities, including but not limited to medical, behavioural, educational and psychosocial interventions.

Study design: Systematic reviews and umbrella reviews that included meta-analyses and assessed the effectiveness of interventions for developmental disabilities using rigorous systematic review methodology, including comprehensive literature search, inclusion and exclusion criteria, and quality assessment of included studies.

Outcome measures: Meta-analyses that report a pooled effect size for child functioning outcomes related to prevention, early detection, or rehabilitation of developmental disabilities, including measures of developmental outcomes, cognitive function, social skills and quality of life.

Systematic reviews that did not meet the above inclusion criteria, such as narrative reviews, opinion pieces, or reviews with low methodological quality, were excluded.

Additional exclusion criteria are meta-analyses that:

Do not include results for children under 5 years of age

Address secondary health issues in children with disabilities (e.g. oral health for children with cerebral palsy)

Focus only on parents and do not include outcomes for children with disabilities

Focus on a specific population group such as children exposed to HIV or malnutrition

We also excluded studies that reported surgical interventions and all invasive medical procedures requiring hospitalisation (such as intrathecal baclofen, scoliosis correction, selective dorsal rhizotomy and umbilical cord blood cell therapy).

Data extraction

Two independent reviewers screened the titles and abstracts of identified articles for eligibility based on the inclusion and exclusion criteria (TS and either NS or CN). Full-text articles of potentially eligible reviews were retrieved and assessed for inclusion. Disagreements between reviewers were resolved through discussion or consultation with a third reviewer if necessary.

Data from studies retrieved through the systematic search were extracted using Rayaan.ai using pre-defined and piloted forms and exported to Microsoft Excel for analysis. Where studies included data with both child and adult information, only the child information was extracted. Extracted data included the characteristics of included reviews (e.g. authors, publication year, country of origin), population characteristics (e.g. sample size, age range for the meta-analyses undertaken), interventions assessed and outcomes reported. Disaggregated data were managed as follows: where data allowed for disaggregation by children under five, only these specific data were extracted. In cases where data were not disaggregated by age but included children under five, these data were extracted to a separate Excel sheet, and the age range was noted. Extracted data that were not disaggregated were presented as an appendix.

Quality assessment

The risk of bias (quality) in the included reviews was assessed by the lead author. The Assessment of Multiple Systematic Reviews (AMSTAR2) [ 20 ] tool, which is specifically designed for evaluating health intervention research, was utilised to evaluate relevant sources of bias in the reviews. The AMSTAR2 tool takes into consideration the quality of the primary studies included in the meta-analysis, rather than being limited to assessing only the technical aspects of the meta-analysis itself. The AMSTAR2 questionnaire comprises 16 criteria, and reviewers were required to respond with "Yes," "Partial Yes," "No," or "No Meta-analysis" options. The overall quality of the reviews was classified into categories of "critically low," "low," "moderate," or "high."

Data synthesis

Meta-analyses were grouped by target disability, tabulated and narratively synthesised. Data on effectiveness measures were summarised. Further quantitative meta-analysis was not performed, as studies reported a range of different measures, often in non-representative populations. We present the disaggregated data, with children under 5 years old, with nonaggregate data reported in an appendix.

Global burden of disease and prevalence of developmental disability

In addition to findings from the included meta-analyses, data were presented on the prevalence of developmental disabilities, as extracted from the most recent prevalence estimates reported by the Global Burden of Disease (GBD) study [ 21 ]. This is presently the only source of data on specific developmental disabilities in children under 5 years covering over 200 countries from all world regions [ 2 , 4 ]. We identified the world regions with the highest prevalence according to the classification of developmental disability and the top ten affected countries. The findings of high-quality reviews were then mapped to the conditions and tabulated.

We identified 883 citations in our umbrella review. Of these, 37 met inclusion criteria and three studies were included after manual review (Fig.  1 ). Amongst the 40 studies, 18 included disaggregated data for children under 5 years, while 22 reviews contained data for children under 5 years, but these were not disaggregated by age.

Study selection. *Full texts excluded with reasons provided in Additional File 1

Eighteen systematic and umbrella reviews explored evidence-based prevention, early detection and early intervention and rehabilitation for 1,273,444 children under five with or at risk of developmental disabilities from 251 studies in 30 countries. Amongst them, half of the reviews ( n  = 9) focused on interventions for children with behavioural disorders, including autism spectrum disorder (ASD) and attention deficit/hyperactivity disorder (ADHD) followed by six reviews (33%) that focussed on children with physical impairment, including cerebral palsy (CP) and neuromotor delay. One review looked at prevention and early intervention, while two focused solely on prevention, and three concentrated on early detection. The remaining 12 reviews (67%) were centred around early intervention (Table  1 ).

Out of the 30 countries represented in the studies included in the reviews, 20 (67%) were high-income countries, while 10 (33%) were middle-income countries. No low-income countries were represented in the reviews. The highest number of studies came from the USA with a total of 101 studies (40%), followed by the UK with 28 studies (11%) and China and Australia with 24 (10%) and 20 (8%), respectively. Four studies were undertaken in multiple countries. The middle-income countries represented included Bangladesh, China, Egypt, India, Iran, Pakistan, South Africa, Thailand, Tunisia and Turkey. The participant numbers varied across the included reviews, with sample sizes of the meta-analyses ranging from 58 participants with neuromotor delay [ 22 ] to 1,023,610 newborns evaluated for early screening for hearing loss [ 23 ].

Regarding quality review, this umbrella review includes a majority of reviews ( n  = 16, 89%) with high and moderate confidence (nine reviews and seven reviews respectively) and two reviews (11%) of low confidence (Additional File 2 show the results of the risk of bias assessment of each study with the AMSTAR tool, including the studies that were not disaggregated by age). The most common reasons for low confidence included a combination of the absence of an explicit statement regarding the establishment of review methods before conducting the review, the lack of a list detailing excluded studies and justifying these exclusions, and inadequate investigation of publication bias.

The outcomes and impacts varied across the studies, ranging from reduction in core symptoms for ASD, improved cognitive function and adaptive behaviour, to neuroprotection and improved sitting balance. Table 2 provides a summary of studies focusing on various disabilities and their corresponding evidence for children under 5 years [ 22 , 23 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 31 , 32 , 33 , 34 , 35 , 36 , 37 , 38 , 39 ].

Data that were not disaggregated are presented in Additional file 3  [ 40 , 41 , 42 , 43 , 44 , 45 , 46 , 47 , 48 , 49 , 50 , 51 , 52 , 53 , 54 , 55 , 56 , 57 , 58 , 59 , 60 , 61 ].

Cerebral palsy

Globally, approximately 8 million (95% uncertainty interval [UI] 7,113,334–9,231,577 children younger than 5 years had CP in 2019, with the highest burden being in the African Region (2.7million) and Southeast Asia (2.4million) [ 21 ]. Amongst the six (33%) reviews that examined prevention and early intervention for CP, only two [ 27 , 28 ] included data from a country ranking within the top ten highest prevalence countries, specifically China. Four reviews focussed on early intervention, one on prevention, and one on prevention and early intervention. Amongst preterm infants, antenatal corticosteroids, magnesium sulphate and prophylactic caffeine were all found to significantly reduce the risk of cerebral palsy when compared to placebo or standard care. Likewise, therapeutic hypothermia amongst term neonates with hypoxic-ischemic encephalopathy significantly reduced the risk of motor impairment at 18 months. Improved cognitive outcomes were seen during early childhood (age 2–3 years) following a variety of early developmental interventions, such as early rehabilitation (that included sensory stimulation, co-ordination training) and environmental enrichment. This effect continued to preschool age (4–5 years) (Table  3 ).

Cognitive impairment

Approximately 16 million (95% UI 11,515,194–20,980,652) children under 5 years had cognitive impairment worldwide, with the highest burden in Southeast Asia (6.3 million) and the African Region (3.3 million) [ 21 ]. China and the USA are the sole nations amongst the top ten with the highest prevalence of cognitive impairment represented in one systematic review that targeted prevention of cognitive impairment. This single systematic review explored prevention of cognitive impairment in preterm neonates and found prophylactic erythropoietin (rhEPO) reduced the risk of neurocognitive impairment at 18–26 months [ 29 ]. There were no studies disaggregated for children under five with cognitive impairment regarding early detection or inclusive early intervention and rehabilitation.

Hearing loss

There were over 14 million (95% UI 12,036,835–16,216,298) children under five with hearing loss, with the highest burden in Sub-Saharan Africa (4.4million) and South Asia (3.9million) [ 21 ]. Amongst the two reviews that examined prevalence, identification or intervention for hearing loss, only one was of high quality and neither included data from the regions with the highest prevalence. Infants with universal newborn hearing screening (UNHS) demonstrated a significantly elevated relative risk (RR) of identifying permanent bilateral hearing loss (PBHL) before 9 months, along with an average 13.2 months earlier age of PBHL identification compared to those without UNHS [ 23 ].

Attention deficit/hyperactivity disorder (ADHD)

Globally, approximately 1.4 million (95% UI 898,677–1,947,054) children under 5 years were affected by ADHD in 2019, and half of this cohort was situated within the regions of East Asia (0.5 million) and South Asia (0.2 million) [ 21 ]. There were no studies that examined prevention or early detection. The one review that examined early intervention included data from China, Iran and the USA which rank within the top ten highest prevalence countries [ 37 ]. The review determined that neurocognitive and behavioural interventions resulted in reduced ADHD symptoms and a positive effect on working memory.

Autism spectrum disorder (ASD)

Globally, the burden of ASD was estimated to be nearly 3 million (95%UI 2,418,074–3,461,585) children, with Sub-Saharan Africa accounting for approximately 0.8 million cases and the East Asia and Pacific region contributing 0.7 million cases each [ 21 ]. Amongst the seven moderate- to high-quality reviews that examined early identification and intervention for ASD, none included data from sub-Saharan Africa, the region with the highest burden. There were no studies on prevention of ASD. The review of 18 screening tests for early detection of ASD found that while diagnostic tools were helpful, their sensitivity and specificity varied [ 36 ]. Early intervention studies explored diverse approaches to enhance outcomes for children with developmental challenges and ASD. Spoken word interventions improved spoken language outcomes [ 32 ], and community-based interventions enhanced adaptive behaviour [ 33 ]. Parent-mediated interventions improved communication [ 34 ], although this review was of low quality. Intensive behavioural interventions improved adaptive behaviour [ 35 ] and behavioural and social communication interventions enhanced reciprocity of social interaction [ 38 ]. The Early Start Denver Model also demonstrated a significant effect on ASD symptoms [ 39 ], indicating the potential of these approaches in addressing ASD symptoms and improving outcomes.

We summarised findings from 18 systematic and umbrella reviews that explored evidence-based prevention, early detection, early intervention and rehabilitation for 1,273,444 children with or at risk of developmental disabilities from 251 studies in 30 countries. The majority of reviews ( n  = 12, 67%) focussed on evidence for early intervention. Half of the reviews ( n  = 9) focussed on behavioural disorders, with six (33%) focused on evidence for motor impairment such as cerebral palsy and developmental coordination disorder, and only two reviews (11%) targeted children with hearing impairment. The fewest number of studies were identified for children with cognitive impairment ( n  = 1). Of the 30 countries represented, 20 were high-income countries (67%), ten were middle-income countries (33%) and none were from low-income countries where the prevalence of developmental disabilities was frequently highest. The quality of included reviews was predominantly medium and high.

The synthesis of reviews on prevention for CP highlights the efficacy of interventions such as antenatal corticosteroids [ 26 ], magnesium sulfate [ 26 ], prophylactic caffeine [ 27 ] and neonatal therapeutic hypothermia [ 27 ] in reducing CP rates; additionally, early developmental interventions post hospital discharge [ 28 ] and environmental enrichment [ 26 ] demonstrate promising outcomes in enhancing motor skills and cognitive development for children under five. Moreover, cognitive impairment prevention in preterm infants found that prophylactic use of erythropoietin (rhEPO) [ 29 ] demonstrated a significant risk reduction, from 20 to 14%. With regard to hearing impairment, findings suggest that early hearing screening interventions, specifically UNHS, are associated with improved outcomes in identifying hearing loss in infants [ 23 ]. There were no meta-analyses for screening for vision, learning disabilities or epilepsy. Regarding ADHD, neurocognitive and behavioural interventions may reduce ADHD symptoms and positively influence working memory [ 37 ]. The findings suggest that diagnostic tools for ASD can be useful in early detection, but each test may have varying levels of sensitivity and specificity [ 36 ]. Early intervention studies encompassed a range of strategies aimed at enhancing outcomes for children with developmental challenges and ASD, including interventions focusing on improving adaptive behaviour [ 33 , 35 ], enhancing communication [ 32 , 34 ] and social interaction [ 38 ] and reducing ASD symptoms [ 31 , 39 ].

The results of this review highlight the disparity between high-income countries and LMICs in terms of evidence availability and applicability to different settings. We identified geographical and disability-related inequities. There is a lack of evidence from outside high-income settings. There was also an absence of data specifically for children with vision loss, even though at least 6 million children under five around the world have a vision impairment [ 62 ]. There are also large gaps in early detection. In addition, no developmental screenings during well-child visits were identified in our study. Efforts are therefore needed to gather more data on interventions in LMIC disaggregated by disability type, as this information is crucial to tailoring targeted and appropriate prevention, early detection and rehabilitation interventions.

Our study findings have implications for research. To address study quality, meta-analyses should include an explicit statement regarding the establishment of review methods before conducting the review, a list detailing excluded studies and justifying these exclusions, and investigate publication bias. More generally, there is a lack of data on children under five. Disaggregation by age group and studies that specifically target this age group to inform early interventions are required. Bolstering disability research capabilities across diverse settings is vital to tackle the challenges faced by children with and at risk of developmental disabilities and their caregivers worldwide. Inclusive research practices should emphasise representation and active engagement of children with disabilities and their caregivers to ensure pertinent, considerate and all-encompassing research outcomes.

Our results carry policy and practice implications. We expose gaps in evidence for prevention, identification and early intervention and rehabilitation, along with a disparity between evidence and regions with high prevalence. This underscores the absence of essential evidence for effective strategies in settings with the greatest burden. Importantly, this matter is even more urgent because global financing for rehabilitation, disability and assistive technology is largely not health-led amongst international agencies. A historical emphasis on combatting infectious diseases within the framework of development assistance for health (DAH) has created structures that disenfranchise other health needs—like those of children with disabilities—from core leadership and resources in the sector, including complementary programming. The principal contributor to DAH, the USA [ 63 ], largely directs disability-inclusive health investments away from the Global Health Bureau at the United States Agency for International Development (USAID), instead focussing on disproportionately small investments for rehabilitation through its Democracy, Human Rights and Governance sector [ 64 ]. It is therefore crucial to align funding strategies with the principles set forth in the Paris Declaration on Aid Effectiveness (2005) [ 65 ], including locally led health assistance and prioritisation of health system development, to bridge these disparities and ensure equitable access to appropriate care and interventions for all children. In addition, while the current included reviews have contributed valuable insights into prevalence, interventions and regional disparities, our examination reveals an opportunity for future research to explicitly focus on innovative strategies that challenge societal norms, promote inclusivity and foster a transformative shift in addressing stigma and discrimination associated with developmental disabilities in early childhood.

Supporting all children with disabilities will not be possible without a focus on the integration of evidence-based interventions, inclusive health systems and comprehensive education programmes that prioritise equity, empowerment and inclusion. Access to comprehensive care and support for children with disabilities is crucial for their well-being and overall development. This requires establishing inclusive child health services that cater to diverse needs. By harmonising evidence-based interventions within existing health systems, we can create sustainable and scalable solutions that benefit a larger population.

Further exploration of the interaction between current Early Childhood Development (ECD) programmes and disability support is required. It is evident that many ECD programmes often exclude children with disabilities, which is a missed opportunity for promoting disability-inclusive health and education [ 3 ]. However, these ECD initiatives can serve as potential platforms for promoting inclusivity and providing early support to children with disabilities. Finding effective ways to bridge the gap between ECD programmes and disability support could lead to better outcomes and more comprehensive care for all children, regardless of their abilities. This also raises the question of competing agendas, particularly between the focus on human capital development in ECD and the promotion of human rights for children with disabilities. ECD initiatives are often driven by a human capital approach, seeking to enhance children’s skills and abilities for future economic productivity. However, this approach might inadvertently leave behind children with disabilities, as their needs might not align with the productivity-driven goals of human capital development. It is crucial to find a harmonious way to integrate ECD goals with disability rights perspectives, ensuring that all children, including those with disabilities, receive the support they need to thrive and reach their full potential. This integration will require thoughtful policy and programme design, acknowledging and addressing the unique challenges faced by children with disabilities while promoting inclusivity and equity in early childhood development initiatives.

Strengths and limitations

This paper fills an important gap in the literature with a focus on high burden settings, which previous reviews have lacked. Strengths of this umbrella review include its adherence to standardised guidelines for conducting umbrella reviews and quality assessment, such as following the Preferred Reporting Items for Overviews of Reviews (PRIOR) statement and the AMSTAR2 tool, which has provided methodological rigour, transparency and replicability. The comprehensive search of electronic databases, including relevant broad keywords, helped ensure that a wide range of relevant systematic reviews was identified from 30 countries. Data extraction and quality assessment were conducted independently by two reviewers, reducing bias and enhancing the reliability of the findings. However, there are also limitations to consider. Despite the comprehensive search, it is possible that some relevant systematic reviews might have been missed, particularly as broad search terms were used. For example, parenting interventions. A limitation of the data about ADHD may have arisen from variations in age criteria across settings, where some countries adhere to a lower age cut-off of 4 or 5 years, while the DSM-5 lacks a specified lower age limit, which may potentially result in a lower number of articles available for analysis. Additionally, the absence of disaggregated data for this specific age group poses an issue, potentially resulting in overlooked interventions targeting a broader age range. The decision to exclude certain types of interventions and outcomes, such as surgical interventions and invasive medical procedures that require hospitalisation, may limit the scope of the findings and not fully capture the entire range of interventions available for developmental disabilities.

This paper summarises the evidence base on effective strategies for prevention, detection and early intervention and rehabilitation for children under 5 years with developmental disabilities globally. We identify a disparity between the settings from which this evidence base comes and the regions where the prevalence is highest. By highlighting the geographical inequities in evidence, we aim to foster a conversation on the allocation of resources and the direction of future research and interventions. Ultimately, this holistic approach has the potential to improve the lives of children with developmental disabilities and their families globally.

Availability of data and materials

All data generated or analysed during this study are included in this published article and its supplementary information files.

Abbreviations

Attention deficit/hyperactivity disorder

Assessment of Multiple Systematic Reviews

Autism spectrum disorder

Global Burden of Disease

Low- and middle-income countries

Permanent bilateral hearing loss

Sustainable Development Goals

Universal newborn hearing screening

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Tracey Smythe receives funding from the National Institute for Health Research (NIHR) [NIHR Global Research Professorship (Grant Reference Number NIHR301621)] awarded to Prof. Hannah Kuper. The views expressed in this publication are those of the author(s) and not necessarily those of the NIHR, NHS, the UK Department of Health and Social Care or FCDO. Nathaniel Scherer is funded by the United Kingdom’s Foreign, Commonwealth and Development Office (PENDA project, grant PO8073). Carol Nanyunja is funded by Seneca Trust. Cally Tann is funded by The Medical Research Council, the Bill and Melinda Gates Foundation, Grand Challenges Canada and the Seneca Trust. Bolajoko O. Olusanya declares no funding.

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Smythe, T., Scherer, N., Nanyunja, C. et al. Strategies for addressing the needs of children with or at risk of developmental disabilities in early childhood by 2030: a systematic umbrella review. BMC Med 22 , 51 (2024). https://doi.org/10.1186/s12916-024-03265-7

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Recent Trends and Future Directions in Research Regarding Parents with Intellectual and Developmental Disabilities

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Purpose of Review

This report updates research on parents with IDD and their children published since 2014.

Recent Findings

Since 2014, a plethora of studies using large administrative databases in different countries support a contextual approach to understand why parents with IDD and their children may have worse outcomes than other families. In most studies, increased risk of ill health in women with IDD and health and developmental problems in their children were fully or partially accounted for by socioeconomic and psychosocial hardships. New research has found that pregnant women with IDD tend to have risk factors for pregnancy, birth, and postpartum complications that may contribute to adverse child outcomes. Intervention research is gradually becoming more contextualized.

More studies are needed on multicultural aspects of parenting, programs that could help parents with IDD overcome social and health disadvantages, comprehensive and coordinated service models that start during pregnancy, innovative parent support arrangements, parenting education for teens and young adults with IDD, use of technology, and dissemination and implementation of evidence-based programs.

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Introduction

Research on parenting by persons with intellectual and developmental disabilities (IDD) Footnote 1 has gone through several stages as described by Llewellyn and Hindmarsh [ 1 ]. This report examines trends in this field in the 5 years since their report. Early research was concerned about effects on the children [ 2 , 3 ], the over-representation of children of parents with IDD in child protection [ 4 ], and teaching parenting skills to parents with IDD [ 5 ]. Then, through quantitative and qualitative analyses, the focus shifted to identifying socioeconomic and psychosocial determinants of parenting and child problems, including child protection involvement [ 6 ]. A more nuanced contextual view, such as the one proposed by Feldman [ 7 • ], has come to dominate the research focus. The contextual model has been expanded to include antenatal, birth, and postpartum factors that could affect initial parenting abilities and early child outcomes [ 8 ]. Intervention research has moved beyond parent education and is beginning to incorporate a contextual approach and addressing gaps in previous studies.

Extending Support for Contextual Models

Feldman’s [ 7 • ] contextual-interactional model (see Fig.  1 ) adapted well-established ecological viewpoints of family life by Bronfenbrenner [ 9 ], Sameroff [ 10 ], Belsky [ 11 ], and others for families headed by parents with IDD. For many years, researchers and advocates were dismayed by the dominance of a univariate model of parenting by persons with IDD [ 12 ]. This simplistic view argues that parents with IDD are inherently incapable of adequate parenting and would not benefit from parent education because of their low intelligence. The overshadowing of parental cognitive disability over other factors that may better predict parenting abilities, such as parent history of trauma [ 13 • , 14 ], parental mental health, and social support [ 15 , 16 ], has contributed to discriminatory child protection decision-making and legal outcomes regarding child custody when parents have IDD [ 17 , 18 ]. As can be seen in Fig. 1 , numerous variables potentially are involved and inter-related in determining parenting and child and family outcomes. Several specific variables are listed in each panel that can impede or support successful parenting, and parent, family, and child outcomes. For instance, societal factors such as stigmatization and discrimination of persons with disabilities (top left panel of the model), coupled with a parent’s history of successfully disguising their disability, may result in the parent eschewing needed services and supports to avoid admitting they need help. This action could reduce likelihood of parenting success, thereby adversely impacting child health and development. Child protection workers may identify the parent as uncooperative and blaming the parent’s lack of insight as to their support needs on their cognitive disability rather than a strong motivation to hide their disability. Another example of not looking beyond the parent’s disability would be to blame inadequate newborn care and apathy to the child on the mother’s IDD, rather than considering the possibility that she may have postpartum depression. At the time the model was first proposed in 2002 [ 7 • ], many of these relationships were hypothetical with respect to families with parents who have IDD. Since then, considerable evidence has emerged supporting the model. The remainder of this section summarizes recent research relevant to the model since the Llewellyn and Hindmarsh [ 1 ] report that provided evidence in support of a contextual approach to parenting by parents with IDD.

Interaction model of parenting for parents with learning difficulties (adapted from Feldman, 2002; reprinted with permission of NADD)

Evidence from large datasets has expanded support for a contextual model of parenting for these families. Emerson et al. [ 19 • ] used the UK Understanding Society longitudinal database of over 14,000 families to compare socioeconomic disadvantage and the health status of parents with and without IDD (several hundred parents had IDD with the exact sample size depending on age cohort). Parents with IDD had poorer health and socioeconomic hardship but controlling for socioeconomic factors eliminated the health differences between parents with and without IDD.

Powell and associates ran a series of secondary analyses of the US Fragile Families and Child Wellbeing Study subsamples of 1298 mothers without IDD and 263 mothers with IDD of 3-year old children. Like in the UK [ 19 • ], US mothers with IDD had significantly more health and socioeconomic hardships. As both poor parental health and low socioeconomic level are associated with inadequate parenting and child developmental problems [ 21 , 22 ], the authors surmised that these factors should be considered when trying to understand parenting issues in parents with IDD.

Several studies that included child outcome data provide further support for a contextual model [ 16 , 23 • , 24 , 25 •• ]. Wade et al. [ 16 ] showed that in a sample of 120 Australian parents with IDD, social support and parental mental health played key roles in parenting practices and child developmental outcomes, consistent with results reported in a randomized Canadian child protection sample of 1170 parents with IDD [ 15 ]. Wade et al. [ 16 ] found that social support mediated the relationship between measures of socioeconomic disadvantage and parent mental health. The relationship between parental mental health and child outcomes was mediated by (self-reported) parent’s warmth and child-care practices. Meppelder, Hodes, Kef, and Schuengel [ 23 • ] studied the interplay of parental stress, social support, and child behavior problems in 134 Dutch parents with IDD and their 1 to 7-year-old children. They found that large social support networks mediated the relationship between child-related parental stress and (teacher or worker) ratings of child behavior problems. Another study using the US Fragile Families and Child Wellbeing dataset reported no significant differences in having asthma or being overweight/obese in 3-year-old children of mothers with and without IDD [ 24 ]. While children of mothers with IDD were rated as being less healthy, controlling for family socioeconomic hardship, maternal health, stress, and social support eliminated the relationship of maternal IDD and child health problem scores. Hindmarsh, Llewellyn, and Emerson [ 25 •• ] conducted secondary analyses of the UK Millennium Cohort Study when children were 3 ( N  = 15,590), 5 ( N  = 15,246) and 7 years old ( N  = 13,857). Sample sizes of participating mothers with IDD were 64, 57, and 46 in the 3-, 5- and 7-year-old child age groups, respectively. Child wellbeing scores were significantly lower in the parental IDD group at 3 and 5 but not 7 years old. Adjusting for contextual variables such as child illness, poverty and maternal mental health significantly reduced risk to the children of parents with IDD and eliminated significant differences with children who had parents without IDD.

Wickström, Höglund, Larsson, and Lundgren [ 26 ] used Sweden birth registry information from 1999 to 2012 that included 478,577 children, 2749 of whom had birth mothers with IDD. Like Hindmarsh et al. [ 25 •• ], Wickström et al. [ 26 ] found risk reduction to children when controlling for parental factors such as mental health and substance use. However, Wickström et al. [ 26 ] still reported significant risk of injuries and child abuse to children of Swedish mothers with IDD even after adjusting for these maternal characteristics and some child factors (e.g., gestational age, epilepsy). It is interesting that these researchers did not control for external contextual variables included in other studies such as social support and economic disadvantage that found larger decreases in risk to children of parents with IDD [ 1 , 16 , 24 , 25 • ]. While caution must be exercised comparing results across studies from different countries using different databases and measures, the disparate findings of the Wickström et al. [ 26 ] study suggest, among other things, that social support and poverty may contribute important variance to child outcomes and/or there is a dose effect—the more disadvantages, the more risk [ 27 ].

Although the above studies show the influence of contextual factors on explaining differential outcomes of children with and without parents with IDD, there is still much unaccounted variance. Researchers have begun to look at earlier potential determinants of adverse child outcomes. Several independent research teams using birth cohort data in the US, Canada, UK, and Sweden have reported remarkably similar findings—mainly that compared to women without IDD, women with IDD smoke more during pregnancy, are younger, experience problems during pregnancy and delivery, and are less apt to access or benefit from prenatal care. Their newborns are at significant risk for adverse birth outcomes such as preterm, low birth weight, stillborn, perinatal death, and low Apgar scores [ 8 , 9 , 10 , 11 , 12 , 13 , 14 , 15 , 16 , 17 , 18 , 19 , 20 , 21 , 22 , 23 , 24 , 25 •• , 28 , 29 , 30 , 31 , 32 , 33 •• , 34 ]. Furthermore, women with IDD are more likely to have postpartum physical, neurological, and mental health problems than new mothers without IDD that could affect the care of their newborns [ 35 , 36 • , 37 ]. These recent large-scale studies are beginning to identify reasons why young children of mothers with IDD may show early developmental delay [ 3 , 15 , 16 , 17 , 18 , 19 , 20 , 21 , 22 , 23 , 24 , 25 •• , 38 ]. This research highlights the need for services that focus on prenatal and postnatal health and wellbeing of women with IDD [ 32 , 39 ]. Conceptual models should include prenatal maternal health, delivery, and postnatal complications as additional determinants of child outcomes.

Recent Trends in Interventions for Parents with IDD and Their Children

This section will briefly review some of the current directions of intervention research for these families. As evidence mounts supporting a contextual view of families headed by parents with IDD, are assessments and interventions adopting a more contextualized stance? Feldman and Aunos [ 40 ] have manualized a comprehensive, competence-based parenting capacity assessment approach based on Feldman’s [ 7 • ] contextual model coupled with direct observation of parenting skills and an empirical test of responsiveness to evidence-based parent training. In a case in Massachusetts in which a custody decision to remove a child of a mother with IDD was based primarily on her perceived intellectual inability to raise children, in 2015, the US Department of Justice and US Department of Health and Human Services [ 41 •• ] jointly issued guidelines which specified that contextually based parenting capacity assessments, emulating Feldman and Aunos’ model [ 40 ], for parents with IDD was necessary to meet the legal requirements of the Americans with Disabilities Act. Unlike typical parenting capacity assessments, recommendations from comprehensive and contextual assessments usually do not come to a definitive conclusion that the parent can or cannot parent. A contextualized assessment identifies impediments and supports for the parent to be successful and may make service and support suggestions beyond parent education [ 40 ]. For instance, recognizing the importance of social support, a contextual assessor may recommend teaching the parent to build a support network, be more socially engaged, and participate in activities that promote parent, child, and family wellbeing.

Unfortunately, few intervention studies have focused on building social support and wellbeing in parents with IDD. In one of the first studies attempting to decrease social isolation of mothers with IDD, McGaw, Ball, and Clark [ 42 ] conducted a control study of a 14-week social group for 12 British parents with IDD. They found that compared to the control group ( n  = 10), the training group increased their social networks, friendships, and self-confidence. However, these improvements did not impact the parents’ self-reported relationships with their children and there were no direct observations of the impact of the intervention on parent-child interactions. In Canada, McConnell et al. [ 43 ] employed an uncontrolled, pre-post, mix-methods design to evaluate the Social Learning Program (SLP) that is intended to increase community engagement and psychological health of parents with IDD. The SLP included individual goal setting, problem-solving, and feedback as well as group activities. Different from other intervention studies involving parents with IDD, McConnell et al. [ 43 ] primarily recruited from agencies supporting adults with fetal alcohol spectrum disorder (FASD) and included aboriginal parents. Of the 33 parents originally recruited, 18 mothers with IDD completed the full study. Pre-post scores on the Depression, Anxiety and Stress Scale [ 39 ] showed significant improvements in depression, anxiety, and stress, with moderate effect sizes. No pre-post differences were found on self-report measures of social support and personal control. Several themes emerged from qualitative analysis, including awareness there were other parents like them, making and helping new friends, being more socially engaged, enjoying social interactions, recognizing strengths, accomplishing goals, being more confident and having a more positive outlook. Gustavsson and Starke [ 40 ] qualitatively reported that a parenting group in Sweden reduced the parents’ social isolation, built friendships, and more trusting relationships with their social workers. The results of these preliminary group intervention studies to increase social supports and wellbeing in parents with IDD are encouraging, but more controlled studies are needed that include measures that demonstrate the impact of these approaches on parenting success, parent-child interactions, and child and family outcomes.

While there is now considerable evidence that behaviorally based parent training programs, using audio-visual materials, modeling, coaching, and positive reinforcement, can improve parenting skills in parents with IDD with corresponding benefits to the children [ 5 , 46 , 47 ], little research has been conducted on collateral effects of the intervention on the parents. Three recent parent training studies that focused on reducing child problem behavior and increasing positive parent-child interactions showed pre-post training decreases in parenting stress related to the child [ 48 • , 49 , 50 ]. Tahir et al. [ 50 ] showed increased parenting self-efficacy and decreased reported use of punishment in two mothers with IDD who received individualized game-based training.

Another trend in the intervention literature is to adapt existing parent training programs for parents with IDD. Recent studies have shown promising but variable results, for adaptations of Triple P—positive parenting program [ 51 ], parent-child interaction therapy [ 52 ], and positive behavior support [ 49 ]. An RCT of an adaptation of video-feedback intervention to promote positive parenting and sensitive discipline did not significantly improve ratings of targeted parenting skills [ 53 ]. Modifications to the original programs included making the material more concrete; less reliance on reading, writing, and detailed explanations; slower pace and extended sessions; and more frequent positive feedback. These features are already part of programs designed specifically for parents with IDD [ 54 ]. Variable findings of these program adaptations may be due to trying to adhere to original program components that do not work as well with parents with IDD (e.g., group sessions) or indirect measures of parent and child behaviors (e.g., self-report questionnaires, rating scales) rather than direct counts of parent and child behaviors).

An emerging trend is to start imparting parenting knowledge to teens and young adults with IDD before they become parents. Pearson, Chaisty, and Stenfert Kroese [ 55 ] recruited 25 students from special needs schools (mean age = 18 years, mean IQ = 59) to watch an 8-min informational DVD about parent-child attachment and ask questions. A seven-item questionnaire testing knowledge gleaned from the video was given pre-post viewing (same day). The participants then were given an information booklet to take home. Two to 3 weeks later, they were retested. There were significant increases in correct responses on the posttest and follow-up test from the pretest. The change scores were not significantly correlated with participant IQ score or other demographic characteristics. There was no test to see if the participants would interact with a baby any differently after training. A feasibility study in Sweden recruited six special needs high school students to take an 8-week parenting course using a parenting toolkit developed in the Netherlands by Marje Hodes [ 56 ]. The students also had to care for a programmable baby simulator for 3 days and nights. Taking care of the “baby” proved difficult for the participants, with only three of them being able to care for the baby for the 3-day trial, and then only at 54% correct care according to the data downloaded from the simulators. Scores on parenting attitude and self-efficacy questionnaires did not change significantly. Although the preliminary results were weak, the researchers concluded that it was feasible to mount a full-scale study using the simulator and toolkit. While it makes sense to give older teens and young adults with IDD some exposure to parenting, it remains to be seen how these programs affect teenage pregnancy and initial parenting knowledge and competence.

Future Directions

This section identifies some future research needs in expanding the contextual-interactional model and assessment and interventions based on recent findings in support of the model reviewed above.

Intergenerational Research

The contextual-interactional model as presented in Fig. 1 posits some intergenerational effects that are now supported by recent research—for example, the impact of parental childhood trauma on parenting, child, and family outcomes [ 13 • , 14 , 15 , 16 , 17 , 18 , 19 , 20 , 21 , 22 , 23 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 31 , 32 , 33 , 34 , 35 , 36 , 37 , 38 , 39 , 40 , 41 , 42 , 43 , 44 , 45 , 46 , 47 , 48 , 49 , 50 , 51 , 52 , 53 , 54 , 55 , 56 , 57 • , 58 ]. More research is needed to investigate mechanisms of intergenerational transmission and how children and grandchildren are impacted by positive and negative life events of the parent in question [ 59 ].

Assessment Research

The research on assessment practices involving parents with IDD suggests the system is fraught with bias and invalid methods [ 17 , 18 ]. The comprehensive, contextual parenting assessment approach [ 40 ] based on the contextual-interactional model is now recommended as best practice [ 41 •• ], but more research is needed to evaluate the validity and impact of these assessments. For instance, to what extent does implementation of the contextual assessment recommendations lead to better outcomes for families than the traditional assessment model that focuses on parental cognitive abilities as a proxy for parenting skills? Research is needed on developing a simple screening tool that can help workers quickly identify which parents may need accommodations such as pictorial rather than textual materials.

Multinational Collaboration and Diversification

Support for a contextual approach has benefited from secondary analyses of large administrative databases. However, these databases were not specifically designed to focus on families with parents with IDD and used different definitions, recruitment methods, measures and analyses. These differences make interpretation of results across studies problematic. It would be worthwhile to design a purpose-built multinational study with consistent objectives and research protocols. Perhaps, the Special Interest Research Group of the International Association for the Scientific Study of Intellectual and Developmental Disabilities could take the lead to plan such a study.

International comparative studies, in addition to national inquiries into minority cultures [ 60 ], could study the impact of distinct cultural practices. For instance, some cultures promote or even force marriage and pregnancy in young women while other cultures actively discourage or even prevent procreation by persons with disabilities. As such, the context of the pregnancy (e.g., choice, imposed, accidental, discouraged) might impact how parenting is perceived and supported. To inform policies and services, future research could examine the application of the interactional model [ 7 • ] to a broader population of parents that are viewed (usually wrongly) as being at risk for poor parenting. Such parents include those with other types of disabilities besides IDD (e.g., physical, sensory, mental illness) as well as inexperienced (e.g., teenage) parents.

Parent and Child Perspectives

The refinement of the contextual model would benefit from continuing inclusion of the parents’ and children’s voices. For instance, asking parents how they negotiate and choose their support network members could help organize supports that are more responsive to the parents’ needs and preferences. While numerous qualitative studies have either interviewed parents (usually mothers) with IDD or offspring [ 61 , 62 ], more studies are needed that interview mothers, fathers, and children from the same families to gain a better understanding of their views of their parent-child relationships and the impact of parental disability on family life.

Despite multiple adversities faced by parents with IDD, many of them are still parenting and living fruitful lives. The fields of positive psychology and resilience [ 63 ] may contribute to research on parenting and parents with IDD in determining how personal attributes and specific historical and current experiences build self-esteem, confidence, positive parent-child relationships, and more effective parenting practices. Research in this field should become more participatory, involving collaboration with the parents with IDD and their children not only as the subjects of studies, but also as research collaborators themselves [ 64 , 65 ].

Interventions

More than 40 years of research has demonstrated the effectiveness of behaviorally oriented parent training in increasing parenting skills in parents with IDD [ 5 , 46 , 47 , 54 ]. New well-designed studies could examine the use of technology (e.g., reminder texts, smartphone apps, video modeling and feedback, telehealth parent training, virtual reality). For instance, a parent could practice positive behavior support strategies to deal with child misbehavior using a virtual reality device and receive feedback from a parent trainer before trying the strategies with the child. Studies examining telehealth parent training [ 66 ] for parents with IDD are needed to reach parents living in remote areas, who prefer that nobody comes to their homes, or are in lockdown due to a pandemic. Parent training strategies, like general case training [ 67 ], should be tested that promote generalization and long-term impact of parent training programs on child and parent wellbeing and family preservation. More studies could focus on the consequences of contextually based comprehensive interventions to reduce economic and psychosocial hardships seen in these families. Research could explore how training might support women with IDD in establishing and maintaining healthy relationships and social support networks to reduce their risk of domestic violence and exploitation.

The notions of peer support [ 68 ] and co-parenting arrangements [ 69 ] need further exploration. In terms of parents with IDD, peer support involves other parents providing parent training in addition to instrumental and emotional support. Co-parenting involves another person or couple (related or unrelated to the mother and father with IDD) who share parenting responsibilities with the biological parents. Understanding the circumstances under which these relationships may work and what factors are needed for success might offer child protection authorities alternatives to permanent removal of the child while allowing the parents with IDD to raise their child. More research is needed on expanding the initial studies [ 55 ] of innovative approaches to educate teens and young adults with IDD about healthy and safe sex and relationships, family planning, pregnancy, and parent education before they become parents. This education may reduce current risk of adverse pre-, peri-, and postnatal mother and baby outcomes described above [ 24 , 28 , 30 ].

Implementation

While a more thorough understanding of parenting determinants and research to continue to build evidence-based intervention programs are necessary, further study is needed on the dissemination and implementation of this knowledge. We need to better understand how to make adapted and effective interdisciplinary services available and accessible in communities to those parents who need them. For instance, how can service pathways be developed and coordinated to include accessible family planning, perinatal care, maternal health, housing, and parent education in order to decrease the health disparities in mothers and their newborns [ 8 , 29 , 30 , 31 , 32 , 33 • ]? Parenting assessment procedures and intervention programs need to fit within current service systems, as well as be offered by skillful professionals. More studies are needed on training of social workers and clinical psychologists on assessing and intervening with parents with IDD. This topic could be embedded into social work curricula and CPS in-service training [ 70 ] so that workers become more comfortable interacting with these parents and are aware of evidence-based practices. Likewise, psychologists conducting parenting capacity assessments should be obliged to take workshops on adapting assessments for parents who have IDD, using a comprehensive, contextual approach [ 40 ] rather than the current emphasis on cognitive disability evaluations as a proxy for direct measures of parenting competence [ 18 ].

Evaluations of current child protection practices, the effects of contextually based parenting assessments (when used), and the decision-process of judges [ 40 ] may help to eliminate biases in the current system that result in premature and possibly unwarranted child removal and failure to provide necessary supports to keep the child with their biological parents. Certainly, to help guide court decisions towards family preservation, the availability of evidence-based services and supports would be essential, as judges might be more inclined to keep families together knowing they could receive ongoing supports that adjust to changing needs of the family.

Research in parenting by persons with IDD has grown in sophistication and design. We now have considerable knowledge of contextual variables affecting parent, child, and family outcomes. Blaming perceived and real parenting problems and removing the child based only on the parent’s IDD should be considered ethically objectionable. Yet, both authors as well as our colleagues in numerous countries frequently come across child protection cases that maintain this practice. Specific service pathways geared to supporting families with multiple impediments to parenting starting before conception might offer parents and children the opportunity to grow together in safe and nurturing families, as promoted by the United Nations Conventions on the Rights of Children, and Persons with Disabilities [ 71 , 72 ].

This term will be used to include persons with study labels of intellectual disability, intellectual or cognitive impairment, learning difficulties, learning disabilities (UK), or borderline intellectual levels. Many of the studies in this area use different definitions and measures to determine the intellectual level of their participants and rarely document formal ID diagnosis.

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Feldman, M.A., Aunos, M. Recent Trends and Future Directions in Research Regarding Parents with Intellectual and Developmental Disabilities. Curr Dev Disord Rep 7 , 173–181 (2020). https://doi.org/10.1007/s40474-020-00204-y

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Participation of children with disabilities in school: A realist systematic review of psychosocial and environmental factors

Donald maciver.

1 Occupational Therapy and Arts Therapies Subject Area, School of Health Sciences, Queen Margaret University, Edinburgh, Scotland, United Kingdom

Marion Rutherford

Stella arakelyan, jessica m. kramer.

2 Department of Occupational Therapy & PhD Program in Rehabilitation Sciences, Boston University, Boston, United States of America

Janet Richmond

3 School of Medical and Health Sciences, Edith Cowan University, Joondalup, Australia

Liliya Todorova

4 Occupational Therapy, Faculty of Public Health and Health Care, University of Ruse, Ruse, Bulgaria

Dulce Romero-Ayuso

5 Department of Physical Therapy, Occupational Therapy Division, Faculty of Health Sciences, University of Granada, Granada, Spain

Hiromi Nakamura-Thomas

6 Saitama Prefectural University, Graduate School of Health, Medicine and Welfare, Saitama, Japan

Marjon ten Velden

7 Amsterdam University of Applied Sciences, Faculty of Health, School of Occupational Therapy, Amsterdam, the Netherlands

Ian Finlayson

Anne o’hare.

8 Child Life and Health, SMC Research Centre, Edinburgh University, Edinburgh, Scotland, United Kingdom

Kirsty Forsyth

Associated data.

All relevant data are within the paper and its Supporting Information files.

In order to make informed decisions about how best to support children and young people with disabilities, effective strategies that facilitate active and meaningful participation in school are required. Clinical factors, diagnosis or impairments somewhat helpful in determining what should be provided in interventions. However, clinical factors alone will not offer a clear view of how to support participation. It is helpful then to look at wider psychosocial and environmental factors. The aim of this review was to synthesise evidence of psychosocial and environmental factors associated with school participation of 4–12 year old children with disabilities to inform the development of participation-fostering interventions.

A systematic search and synthesis using realist methods was conducted of published research. Papers had to include consideration of psychosocial and/or environment factors for school participation of children with disabilities. The review was completed in accordance with the Realist and Meta-narrative Evidence Syntheses: Evolving Standards (RAMESES) and Preferred Reporting Items for Systematic Review and Meta-Analysis (PRISMA) guidelines. Papers were identified via Boolean search of the electronic databases MEDLINE, CINAHL, PhycINFO and ERIC (January 2006-October 2018). Appraisal focussed on contributions in terms of whether the articles are appropriate for the review (relevance) and research quality (rigour). Data were analyzed using content and thematic analysis methods using a realist framework. A narrative synthesis of results was reported.

Results and implications

We identified 1828 papers in the initial search. Seventy two papers were included in the final synthesis. Synthesis of findings led to three overarching mechanisms representing psychosocial factors for children (1) identity (2) competence and (3) experience of mind and body. Environmental aspects (context) compromised five interrelated areas: (1) structures and organization, (2) peers, (3) adults, (4) space and (5) objects. Our synthesis provides insights on how professionals may organize efforts to improve children’s participation. Consideration of these findings will help to proactively deal with suboptimal participation outcomes. Development of theoretically determined assessments and interventions for management of school participation are now required.

Introduction

According to the World Health Organization’s World Report on Disability, an estimated 95 million children (5.1%) aged 0–14 years have some form of disability [ 1 ]. Common issues include Autism Spectrum Conditions, Developmental Delay, Behavioral Disorders and Learning Difficulties [ 1 ]. In many countries, these children have rights to be included in mainstream school [ 2 – 5 ]. For professionals who work with children this shift towards social and educational inclusion has meant that practices have had to evolve in tandem. Rehabilitation professionals now deliver a wide range of approaches to support early intervention and prevention for children with diverse needs. This includes school-based approaches alongside teachers and families to enable children’s full and active participation in school [ 6 ].

Participation or “involvement in life situations” [ 1 ] is a key outcome. Participation may take place anywhere. In this paper, we focus on the context of school. Participation in school includes unstructured activities (e.g friendships, play), organized activities (e.g. sports, clubs, arts), classroom based activities (e.g. group work, study) and engagement in social roles [ 7 ]. Children with disabilities are at significant risk for limited participation in school [ 8 ]. Such restrictions have significant lifetime consequences for achievement, quality of life and wellbeing [ 9 – 12 ]. There are several issues. Attendance for children with disabilities is reduced compared to peers [ 13 ]. Students with disabilities participate less in structured and unstructured activities, and experience reduced interaction and playground participation [ 14 ]. Children with disabilities additionally show less engagement in the wider school world, including clubs and organizations [ 13 , 15 ].

Whilst there is an urgent need to develop interventions that promote participation in school, there is limited understanding of processes that may enable it [ 16 ]. Research to date has recognized the importance of psychosocial factors, though conclusions have been hampered by heterogeneous populations and variability in design and outcome measures [ 17 ]. There is little in the way of specific school based research to guide practices. Moreover, a requirement remains for comprehensive theories/models, as research has primarily considered individual psychosocial factors in isolation. A trend is departure from “medical” and “social” models. Both positions have been challenged as limiting [ 18 ]. The World Health Organization’s International Classification of Functioning, Disability and Health (ICF) [ 1 ] has been foundational to discussion with its definition of participation as “involvement in a life situation” and its assertion that the environment is a key determinant of participation (an integration of the “medical” and “social” models). However, the ICF has also provoked confusion and inconsistency in the field [ 16 , 19 , 20 ]. Everything people do is “involvement in a life situation” and participation is classified together with “activities” giving rise to conflicting interpretations [ 19 , 21 ]. Driven by the ICF, there has also been a tendency to focus on a portfolio of actions done in everyday life [ 19 ]. Such indicators of “doing” say little about psychosocial drivers of participation such as motivation, social connection, preferences, choice and meaningfulness [ 19 , 22 , 23 ]. This paucity of theory leads to a situation whereby enhancement of participation outcomes is often an aspiration, but reliable, environmental or psychosocial interventions are not available.

To date only Imms et al. 2016 has conducted research which integrates various factors in a useful new direction [ 24 ]. Their narrative systematic review, although it did not focus on school specifically, concluded that the participation phenomenon is essentially dichotomous—requiring children to “attend” (be present) and also to be “involved” (engage, experience and so forth) [ 24 ]. A further insight has been to differentiate between participation and other influencing or “participation related constructs” which include preferences, sense of self and activity competence [ 24 ]. This work highlights the importance of careful definition, as well as identification of some import psychosocial factors. However, this work did not consider environment factors in detail, and was based on an analysis of randomized controlled trials (RCT) and intervention type studies only. Such restrictive inclusion criteria will have contributed to limiting the data that could have been available. RCTs rarely focus on context, detail on mechanisms of action or conceptual underpinnings [ 25 ]. Analysis of RCTs is less useful for answering conceptual or theory based questions [ 26 ]. Therefore, the present review employed a realist review approach to identify a broad range of environmental and psychosocial factors associated with participation, and to uncover the association between context, mechanisms and participation outcomes in school-aged children with disabilities to guide the development and implementation of interventions and assessments.

Materials and methods

The United Kingdom Medical Research Council’s (MRC) guidance on development of complex interventions argues that new interventions must be underpinned by a conceptual framework and a theoretical understanding of the key processes underpinning an intervention [ 27 ]. This study uses realist review to address the requirement for theory and conceptual framework development outlined by the MRC. The process drew on systematic review and realist review methods. For systematic searching of the literature, we followed the PRISMA guidelines [ 28 ], as far as was relevant for a realist review. Realist methods were completed in accordance with the Realist and Meta-narrative Evidence Syntheses: Evolving Standards (RAMESES) guidelines [ 26 ].

We selected realist review as it meets requirements for dealing with complexity of both topic and research methods [ 25 , 29 , 30 ]. Realist review is an interpretive, theory-driven narrative summary which applies realist philosophy of exploring context, mechanisms and outcomes [ 25 ]. Developed in response to the weaknesses of traditional systematic review, realist review focusses on refining and developing theory. Realist reviews are organized around Context-Mechanism-Outcome (CMO) configuration [ 25 ]. Review aims to identify what works for whom, in what circumstances, in what respects and how, by identifying processes (mechanisms) that lead to outcomes in context [ 25 , 29 , 30 ]. The identification of open, embedded, interactive systems is central to the process of analysis. These assumptions constitute a realist philosophical ‘lens’ [ 25 ]. The steps of realist review are: (1) identifying the review question; (2) formulating the initial theory; (3) searching for primary studies; (4) selecting the studies and appraise their quality; (5) extracting, analyzing and synthesizing data. The details are described below [ 25 ].

Identifying the question

The review question was: “What are the mechanisms and contexts which determine successful participation in 4–12 year old children with disabilities in school?” In developing the question, we drew from a range of perspectives. As the findings were indented for use internationally, the research team included professionals from several countries (Japan, the United Kingdom, Spain, Australia, Bulgaria, the Netherlands, and the United States of America). We ensured that team members represented a range of voices from those with an interest in participation in schools, representing expertise in psychology, rehabilitation, medicine, community pediatrics, neurodisability, community health sciences, education, occupational therapy, disability theory, and global health. The research team included professors, post-doctoral fellows and a range of senior academics and expert clinicians. A wider advisory group included rehabilitation, social care and educational practitioners and managers who provided regular input. Initial questions and review direction were discussed over email between the research team and advisory group. This included a discussion on realist informed approaches including an explanation of Context, Mechanism and Outcomes and the basics of realist theory. The research and advisory groups felt that the focus and question set was an authentic question which reflected curiosity about how schools were working and interest in understanding how to improve children’s participation.

Formulating the initial theory

In line with a realist review approach, our initial thinking was informed by factors identified in the literature as possible key drivers of participation outcomes in school. Key literature was identified and synthesized through a scoping search [ 1 , 10 , 16 , 17 , 19 – 21 , 23 , 24 , 31 – 37 ]. This initial scoping helped to identify theoretical areas, concepts and perspectives (a summary of the initial literature review is presented in Appendix A in S1 File ). Amongst the main ideas considered were Bronfenbrenner’s ecological systems model [ 31 ], the World Health Organization’s International Classification of Functioning, Disability and Health (ICF) [ 1 ] and practice models to support participation [ 34 ]. Using the above scoping review, discussion and analysis amongst the research team and advisory group led to the development of initial mechanisms, contexts and outcomes, and the target population. Initial mechanisms focused on personal psychosocial factors which may drive participation outcomes: (1) children’s choices, initiative, interests and skills and (2) patterning and performance of participation, including routines (e.g. going to school, eating lunch, playing with friends) and roles. Initial thinking also considered psychosocial factors related to common issues experienced by children with disabilities including pain, anxiety, stress, or fatigue. In considering the context, we drew on ecological systems theory, focusing on the “microsystem” as the system closest to the person and the one in which they have direct contact [ 31 ]. In this case, the characteristics of classrooms and schools, denoting circumstances within school that may be considered as enablers or barriers. This approach meant that issues pertaining to context outside the school (for example, the role of parents, home life, or government policies) were not considered.

Participation in school was the outcome of interest. The most common definition of participation originates in the World Health Organization’s International Classification of Functioning Disability and Health referring to a person’s “involvement in life situations” [ 1 ]. As noted, other authors have criticized this definition [ 17 , 38 ]. The definition used in the review builds on the ICF definition, but also implies that participation must be meaningful, with personal or social significance. Our definition reflects recent ideas [ 24 ] that participation has two essential components: attendance and involvement. The definition is presented in Table 1 .

The target population was defined as children who have a physical, developmental, behavioral, or emotional condition and who also require health and education services of a type or amount beyond that required by children generally [ 39 ]. Middle child hood (4–12 years) was selected as the target group. During middle childhood (defined as ages 4 to 12), a child’s mastery of developmental challenges is strongly influenced by school experiences, hence exploring participation in this context is important. Children are moving from nursery/kindergarten provision to increasingly formal education settings, but have not yet entered the adolescent phase where a number of other unique challenges appear.

Systematic searching process

Searches were conducted for English Language papers in MEDLINE, CINAHL, PhycINFO and ERIC databases covering the period January 2006 and November 2018. This span was chosen in order to capture a contemporary conceptualization of participation. Searching was completed by DM and SA. The search strategy utilized text word searching in the title or abstract along with database Subject Headings. Terms included disability “special needs”, “additional needs”, “disabled persons”, “motor disorders”, “developmental disabilities”, “intellectual disability”, “communication disorders”; age “child”, “children,” “pediatric”, “girl”, “boy”, “schoolchild”, “participation”, “inclusion,” “involvement”, “engagement”, “life situations,” “environment”, “surroundings”,”setting”, “context”, “school”, “education“, “class”, and “teacher”. Strategies were developed with support from an information professional (Appendix B in S1 File illustrates the strategy used in MEDLINE). In addition, a hand search compiled by DM and SA checked reference lists from relevant articles, including all those included in the review.

Selection and appraisal of studies

Members of the research team screened a portion of the titles and abstracts (DM, SA, MR). The potentially relevant records identified by individual members of the research team were then discussed with the other authors to confirm eligibility. This was followed by screening the full text of potentially relevant studies to determine eligibility for inclusion.

Overall, if papers contained evidence relevant to school participation (or related proxy outcome, e.g. school attendance), for children with a disability/special educational need, with discussion of environmental and/or psychosocial factors, the document was retained for further review. In keeping with realist methods, selection criteria regarding study design were not predominant [ 25 , 29 , 30 ]. Methodologically, papers could include any type of peer-reviewed paper including intervention studies, observational research, qualitative research and literature reviews. Literature reviews may be included in realist review if they provide relevant theoretical insights [ 25 , 29 , 30 ]. We did reject all purely descriptive accounts (e.g. opinion pieces or editorials) and grey literature as there was ample peer-reviewed material. We also excluded psychometrics focused papers, due to their general interest in identifying what participation was, rather than its influencing factors. Children with disabilities were identified via medical diagnosis or other support needs (e.g. identified as requiring “special” education). Papers focusing solely on community or leisure participation were rejected, although papers which discussed school participation amongst other settings were included. We aimed to identify studies of relevance to middle childhood which we defined as 4–12 years. Studies close to this age range were passed onto the next stage for further assessment if the findings were viewed by the team as potentially relevant and generalizable to middle childhood. In some cases the assessment of age was not necessary, as the participants were teachers, parents or health professionals, and in the case of some reviews. Initial screening criteria are in Table 2 .

In accordance RAMESES guidelines [ 26 ], final selection of papers focused on contributions in terms of whether articles were appropriate for the research question (relevance) and quality of evidence (rigour) [ 25 , 29 , 30 ]. This was an iterative process, and disagreements were dealt with via discussion (DM, SA and MR). Review of relevance was used to ensure a systematic process and to reduce selection bias. A system of questions was used to identify whether an article was relevant by examining content, insights provided by the study and focus (see Table 3 ). Assessment of rigour was used to judge quality, credibility and trustworthiness of evidence [ 25 ]. Each reviewer appraised each paper by asking key questions on research quality [ 40 ]. Papers could be excluded on the basis of relevance or rigour. Each paper was scored 0 (failed to meet criteria) or 1 (met criteria). Studies scoring 0 on either criteria were excluded.

Data extraction, analysis and synthesis

Data were extracted using predefined forms by DM, SA and MR, regularly checking each other’s work. Data were extracted on: country and author; sample characteristics: sample size; participants’ age and gender; diagnostic category (if available); key findings; relevance and rigor mechanisms, contexts, and outcomes.

Analysis were done by DM, SA and MR following a staged process based on careful review, coding and frequent return to primary studies as necessary. Broad aspects of context and mechanism were identified and coded first. The key analytic process in realist review involves iterative testing and refinement of theoretically based explanations for why outcomes happen, using research papers as data sources [ 25 , 29 , 30 ]. In our case we were focused on participation in the school, and we attempted to find and synthesize evidence to demonstrate that particular mechanisms were important in generating school participation outcomes and to identify which aspects of context mattered. Data were synthesized using qualitative methods (content and thematic analysis) [ 41 ], using realist concepts as a framework [ 25 , 26 ]. Context and mechanisms were operationalized using codes and sub-codes as in typical qualitative analysis [ 41 ]. In the early stages very many individual codes were created and grouped. For example all aspects relating to the child’s motivations were grouped into a broad “motivations” category and all aspects of the physical environment were grouped into a “physical environment” category. Specific aspects were then identified and coded with sub-codes, e.g. social aspects, physical access, or assistive devices. As analysis progressed, more refined codes were created and sorted and grouped to identify mutually exclusive categories of mechanisms and contexts which were coherent and could be designated a single unifying label.

As the analysis progressed, evidence of which mechanisms and context were important was carefully mapped against the emerging taxonomy. Tables were derived, including categories and sub-components, including each article relating to the sub-component. Regular meetings were held and interpretations shared across the research team and advisory group, including re-examination the original articles. Further refinement of the findings continued until agreement was reached. Following final assessment, two members of the research team reviewed once again the articles, and checked the findings. We also attempted to identify disconfirming data or data that might challenge or refute ideas. During this process there was a point at which no new categories of mechanisms or context emerged i.e. saturation was attained. Final labels were then assigned to each area and the narrative summary was written.

The electronic literature search and hand search identified 1828 papers, 1168 of which were removed at the title and abstract stage. Next, 172 papers were reviewed in full. On review, 100 papers were excluded, leading to 72 papers in the final synthesis ( Fig 1 ) (full details of all papers are provided in Appendices C and D in S1 File ). Type of disability was consistent with issues commonly seen in schools (including Autism Spectrum Disorders, Cerebral Palsy, Learning Disability, Learning Difficulty, Developmental Delay, and Physical Disabilities) (Appendices C and D in S1 File ). Forty-six percent (n = 33) of the research was quantitative in design (including trials, cross sectional studies, observational studies and quasi-experimental studies), with the remainder consisting of mixed-methods (n = 4, 5%), qualitative (n = 17, 24%) and review papers (n = 18, 25%). Sample size ranged from 6 to 47 participants in qualitative research, and 14 to 3,752 participants in quantitative (excluding two very large national studies ranging from 18,119 to 64,076 (weighted) participants) (Appendix C in S1 File ). Studies from Europe (n = 28), the USA and Canada (n = 22) accounted for 70% of papers with the remainder coming from Australia (n = 11), Brazil (n = 1), Chile (n = 1), Israel (n = 4), Japan (n = 1), New Zealand (n = 1), Taiwan (n = 1), India (n = 1) and Thailand (n = 1) (Appendices C and D in S1 File ).

The initial analysis identified 72 contexts and 79 mechanisms. These were the psychosocial child and environment factors driving participation outcomes in schools. Further analysis revealed three synthesized mechanisms, and five synthesized contexts. Based on the evidence, we constructed a conceptual framework that depicts mechanisms and contexts influencing school participation for children with disabilities ( Fig 2 ). Details on specific categories of mechanisms and context are provided below.

Context and mechanisms hypothesized to be vital intervening factors in predicting children’s participation. Context provides opportunities and constraints. Mechanisms drive participation outcomes. Participation as an outcome has two components: attendance and involvement [ 24 ]. As children participate, they experience feelings, sensations and perceptions which may be adaptive or maladaptive (e.g. enjoyment, boredom, amusement). There is a cyclical relationship between participation, context and mechanisms.

Synthesis of findings led to three overarching mechanisms representing psychosocial issues and the child’s experiences (supporting studies are presented in Table 4 and Appendix E in S1 File ).

• (1) Identity —these mechanisms were associated with “being”, or the thoughts and feelings the child had about themselves (e.g. believing in themselves, having confidence, understanding their roles or feeling a like a member of the school community) as well as perceptions of activities and tasks in school (e.g. interests, preferences or perceived enjoyment).
• (2) Competence —these mechanisms were associated with “doing” or what the child did in school (e.g. following rules, showing interest, being confident, or following a routine).
• (3) Experience of mind and body (symptoms) –these mechanisms were associated with issues commonly experienced by children with disabilities in schools: pain, anxiety, mood and fatigue/tiredness.

Analysis indicated that these mechanisms related to the child “being”, and how children perceived and made sense of their participation within school. Firstly, the information extracted from studies overwhelmingly and specifically demonstrated the relevance of mechanisms related to motivations, preferences, and interests. The key mechanisms were children’s own interests and preferences including selection of certain activities based on interests/preferences and perceptions around potential enjoyment (or not) of activities which motivated choices [ 14 , 24 , 42 – 46 , 46 – 52 ]. Participation was also strongly influenced by children’s self-perceptions, including self-esteem, self-efficacy, confidence, and perceived competence—all of which influenced children’s activity in-the-moment and over time, influencing current and future participation[ 24 , 46 , 48 – 58 ]. Perceptions around meaningfulness were also identified as an influencer of participation, including perceptions around activities that were valued or especially significant to children, as well as perceptions of personal satisfaction and pleasure associated with activities[ 14 , 21 , 24 , 45 , 50 , 51 , 59 , 60 ].

Children’s internalization and understanding of routines and habits emerged as conditions influencing participation in school. Studies highlighted children’s internalization of habit/routine, familiarity with habit/routine and automaticity of habit/routine were mechanisms for participation [ 44 , 53 , 62 – 64 ]. Knowing the steps involved in activities or routines led to reduced demands on the child to understand, process, or plan, and when internalized as patterns of actions, facilitated participation by providing a set of rules to navigate the school context. Routines of the school day were noted to shape children’s daily participation, with references to the fact that children’s participation in school was supported by structured activities and programs [ 44 , 64 ], and that regularized activities in the classroom supported participation for children with disabilities [ 44 ]. Children themselves perceived that rules, norms and routines are important in structuring their participation [ 62 ]. Parents also indicated that routines influence participation [ 44 , 53 ].

Children’s knowledge, understanding and subjective experience of roles influenced their participation. In the school, possible roles included being a pupil, friend or member of a club. Disabled children tended to occupy less “desirable” roles within the school. Roles considered desirable by children, especially those including being good at something (e.g. best in class) or “best friend” roles were seldom held by children with disabilities [ 61 ]. Children with disabilities also engaged in less “doing roles” (such as athlete, leader, helper and tutor) and more were likely to be classified into negative roles including “challenged learner”, victim or bully [ 61 ]. Mechanisms influencing participation were internalization of roles (either positive or negative roles), leading to positive or negative self-perceptions, and understanding/knowledge of role requirements [ 56 , 61 ]. Related mechanisms included self-perceptions relating to inclusion, focusing on children’s subjective experiences of social inclusion, sense of membership and sense “school” identity [ 57 ].

Competence mechanisms reflected “doing” or behavioral aspects and how children engaged in participation. Well-supported mechanisms enabling participation were children taking initiative, being proactive and acting on interests [ 14 , 24 , 44 , 52 , 53 , 65 – 68 ] Research also demonstrated that seeking independence and autonomy, showing responsibility and commitment, displaying persistence and perseverance were drivers of participation [ 45 , 47 , 51 – 53 , 57 , 68 – 72 ].

Other competence mechanisms related to following routines and having daily habits [ 55 , 62 ] as well as consistency of behavior, including being predictable, being systematic and preparedness for routines [ 55 , 62 ]. Also identified as important conditions for participation were children meeting teachers’ expectations and following the school’s rules [ 61 ]. Finally, patterns of behaviors that followed from particular roles were identified as shaping quality and quantity of participation, including patterns of behaviors associated with friendship roles and patterns of behaviors associated with school-based roles (for example sports team member) [ 57 , 61 , 70 , 71 , 73 ].

Studies exploring relationships between skills and participation were common. In total, 27 papers provided data. However, researchers are now concluding that deficits or improvements in skills, although related to participation, are not related in a direct or linear fashion. The evidence challenges the idea that an increase in skill equates to an increase in participation. Psychological characteristics, personality and preferences are also important [ 14 ]. The evidence did indicate, however, that skills were important for the completion of certain types of activities in certain situations. For example, social skills are often required to access play situations [ 57 ]. The mechanisms related to skills identified as important for participation were organisation and planning (e.g. sequencing, concentration and memory) [ 35 , 46 , 49 , 51 , 52 , 55 , 56 , 63 , 70 , 72 , 74 ]; communication/social skills [ 14 , 15 , 42 , 49 , 51 – 53 , 67 , 72 , 74 ] and motor skills [ 8 , 14 , 49 , 51 , 52 , 55 , 58 , 70 – 72 , 74 – 80 ].

Experience of mind and body

The literature provided good support for the influence of symptoms associated with disability on participation. These were pain, fatigue, anxiety and mood. Twenty-two papers provided data. Identified mechanisms were concerned with experiences related to symptoms. These were: pain (especially cognitions and catastrophizing) [ 8 , 14 , 52 , 71 , 75 , 79 , 81 – 83 ]; fatigue, including lowered energy, tiredness, and sleep disturbance[ 14 , 46 , 58 , 71 , 80 , 84 – 86 ]; anxiety and its consequences including fear, frustration, and anger [ 46 , 51 , 53 , 58 , 60 , 71 , 80 , 87 – 89 ], and low mood, sadness or depression [ 13 , 52 , 71 , 80 , 81 , 84 , 89 ].

Fundamental underpinnings were closely related across the different symptoms, drawing on social learning and cognitive-behavioural theory, suggesting that illness behaviours or responses generate negative behavioural patterns which may be maintained and strengthened over time [ 8 , 13 , 71 , 75 , 81 , 83 ]. These mechanisms lead to reduced participation through disengagement from activity and a cyclical pattern of attempts to control symptoms through increasing withdrawal from activities.

The next step was to explore how and which contexts facilitated or provided opportunities for participation versus contexts which restricted/constrained participation. This twofold role of context was evident throughout. Context comprised five interrelated areas: (1) structures and organization of the school, (2) peers, (3) adults, (4) physical spaces and (5) objects. Sub-components of each area were identified by the reviewers, focusing on opportunities (supports) or constraints (barriers) to school participation (supporting studies are presented in Table 5 and Appendix F in S1 File ).

Structure and organization

Structure and organization was a well-supported aspect focusing on the ways things were done in the school. Facilitative aspects were described as being tailored to the child, responsive to needs, individualized, and child led [ 10 , 15 , 17 , 21 , 24 , 42 – 44 , 49 , 52 , 54 , 57 , 58 , 62 – 65 , 67 – 70 , 80 , 90 – 94 , 96 ]. Facilitative structures/organization were also described as adaptable and flexible [ 10 , 14 , 36 , 44 , 52 , 64 , 65 , 68 , 69 , 80 , 96 , 97 , 98 ], predictable [ 44 ] and well-planned [ 10 , 68 , 69 , 93 , 96 , 98 ]. The most common constraint to participation identified was lack of individualization [ 15 , 20 , 42 , 44 , 49 , 53 , 57 , 58 , 65 , 66 , 69 , 71 , 72 , 80 , 91 , 97 , 98 ]. Other identified constraints included rigid routines [ 10 , 14 , 67 , 80 , 84 , 92 , 93 , 97 , 98 ] or routines which were unpredictable or disordered [ 70 , 72 ].

There was abundant evidence that adults (referring to teachers and other staff within school) were key in creating opportunities for participation and were also influential in shaping the quality, frequency and range of children’s roles [ 16 , 21 , 42 , 43 , 52 , 57 , 61 , 64 , 66 , 96 , 97 , 99 ]. Adult’s positive and sympathetic attitudes were facilitative of participation [ 14 , 93 , 96 , 99 , 100 ] as were individuals who were competent and knowledgeable [ 44 , 62 , 49 , 68 , 96 , 99 ]. Good collaboration between adults was also facilitative [ 14 , 68 , 93 , 96 , 99 ]. Attitudes were identified as restrictive, as well as adults who were unsympathetic [ 10 , 44 , 47 , 53 , 58 , 71 , 72 , 80 , 93 , 99 , 100 ] or lacking in knowledge [ 14 , 35 , 49 , 101 ]and institutional collaboration [ 35 , 69 , 99 ]. Adults were also noted to play a part in shaping negative roles (e.g. by ‘pigeonholing’ children with disabilities as less able and therefore offering them fewer participation opportunities, or by being reluctant to allow students to learn or play independently) [ 61 ].

The evidence indicated that facilitative peers (referring to other children within the school) provided practical and emotional support enabling participation [ 10 , 15 , 16 , 21 , 43 , 44 , 46 , 49 , 57 , 58 , 60 , 64 , 68 ]. Facilitative peers also provided opportunities for friendship [ 43 , 49 , 61 , 71 , 73 ]. Positive attitudes were also identified as important in creating opportunities for participation [ 24 , 36 , 46 , 69 , 93 , 100 ]. Studies also identified non-supportive actions and behaviours, including bullying [ 15 , 43 – 45 , 57 , 61 , 71 , 73 , 80 , 92 , 99 ], negative attitudes [ 10 , 46 , 53 , 58 , 78 , 80 , 92 , 93 , 100 ], and friendship avoidance [ 15 , 49 , 60 , 66 , 71 , 87 ].

Supportive spaces were described as being accessible and usable [ 10 , 14 , 21 , 24 , 36 , 44 , 45 , 59 , 60 , 65 , 67 , 68 , 69 , 71 , 78 , 97 , 99 ] with suitable design/layout and suitable sensory qualities [ 53 , 60 , 62 , 68 , 86 , 96 , 99 ]. Constraints to participation focused on restricted access to areas where activities happen [ 10 , 14 , 35 , 47 , 49 , 52 , 53 , 60 , 65 , 71 , 78 , 92 , 97 , 99 , 103 ]. Other issues included unsuitable sensory qualities, spaces which were unfamiliar, and spaces which were crowded or difficult to navigate [ 35 , 52 , 53 , 60 , 65 , 97 , 99 ].

Research on objects focused on the availability of objects needed to participate in specific activities, for example, wheelchairs and assistive devices [ 10 , 14 , 21 , 24 , 35 , 36 , 52 , 59 , 60 , 62 , 63 , 65 , 91 , 99 ]. Usability and acceptability to the child were noted as important [ 14 , 44 , 50 , 52 , 53 , 60 , 71 , 72 , 99 ]. Research on constraints associated with objects was fairly limited. Objects being unavailable [ 10 , 60 , 65 , 72 , 99 ], difficult to use [ 35 , 53 , 50 , 99 ] or isolating/stigmatizing [ 45 , 50 ] were identified as constraints to participation.

This realist review has developed a conceptual framework for children’s school participation, and identified the processes (mechanisms) and contexts influencing participation outcomes. The synthesis is of key issues that decision-makers and interventionists may consider to help children to participate in school.

The findings support the hypothesis that identified mechanisms and contexts are important factors associated with participation outcomes. Specifically, the findings show mechanisms in three clusters focusing on identity, competence and the child’s experience of mind and body. The context (environment) is conceptualized in terms of adults, peers, the schools’ structures and routines and spaces/objects. Unlike most models designed for dealing with specific impairments or diagnoses, this model is useful with any child with any health related need or disability experiencing problems with their participation. This is a middle range theory. The term ‘middle range’ theory refers to the level of abstraction at which useful theory for realist work is written: detailed enough and ‘close enough to the data’ that testable hypotheses can be derived from it, but abstracted enough to apply to other situations as well [ 26 ]. Middle range is useful because it offers an analytical approach to linking findings from different situations [ 26 ]. The outcomes of a realist review are ideally framed as middle range theory—that is, theory that can usefully be applied across a range of situations, or in a number of domains [ 26 ]. Findings are (by design) age limited (4–12 years old), but are independent of gender, disability category or ethnicity, supporting application across a range of clinical and educational settings. Ideas reflect a contemporary conceptualization of participation drawn from 72 research papers. The model imagines mechanisms and contexts in dynamic and transactional relationships. This is a “generative causality” model. Explanation is not a matter of a singular mechanism or a combination of mechanisms asserting influence on an outcome. School participation emerges out of a cooperation of factors.

No single factor fully explains variance in participation [ 56 ]. Previous research provides indications of which features of the child significantly affect the participation of students with disabilities, including focus on psychosocial factors for participation, such as preferences [ 102 ]. Our findings support the significant importance of children’s preferences, interests and motivations for participation. Our review also adds to the literature by providing detail on habits and routines which are novel elements not commonly considered. Based on our findings, we recommend that issues associated with habits and routines are closely considered in future. We have found that deficits in routine and habits are important contributors. Habit and routines are performed repeatedly and are relatively automatic. They specify what the child will do and in what order, and, thus, constitute key mechanisms for participation. Habits and routines must be understood and internalized and there are additionally ties to environment. As noted by Engman and Cranford (2016), the quality of habitual action is not equally easy for all—in some environments “non-normative embodiment” (i.e. disability) is less likely to make habitual behaviour achievable than in others [ 104 ]. The structure of the environment enables or restricts consistent, structured and planned schedules and routines. Adults facilitate breaks, social routines, setting of rules and expectations, while objects (timetables and other prompts) provide specific routines (e.g. for gathering information, or which classes to go to).

Our model focusses on participation as a key outcome which is influenced by environmental factors. In line with the ICF [ 1 ], and in the wider literature, the environment is noted to have a significant influence on participation [ 8 , 75 , 78 , 92 , 100 ]. We advance thinking by identifying specific environment factors and offering potential for comprehensive assessment and intervention. This is important, as the potential selection of environmental factors is vast. The issue is to identify specific matters facilitating or obstructing participation in school. The identification of issues must be completed in tandem with a contemporary model of participation itself. Small aspects of the school microsystem can go unnoticed if attention is not drawn to them. A focus on the school environment explicitly defined will support guidelines for working to support participation. The current study findings indicate contextual influence of the school is not just a sum of the people, objects and spaces, but also “how” things are done, or expected to be done within the school (the structures and organization of the school) and the important part adults play in providing opportunities for participation and social roles. Our findings highlight the importance of a nuanced understanding of the environment and not just consideration of physical aspects. Identification of physical aspects of the school, whist important, should always be considered alongside the social environment.

Implications for practitioners

International practice is moving towards the adoption of system/ecological views, but the field still operates predominantly from a unidirectional perspective where “something” is provided to “fix” the person with a disability [ 18 ], rather than operating from more contemporary view of participation as a phenomenon that can be mobilized at different levels. The findings of this review show that individual and environmental interventions should be developed promote participation outcomes in schools. Identified mechanisms offer a potential basis for developing psychosocial child-focused interventions. Mechanisms (e.g. preferences, perceptions of self, perceptions of roles, internalization of routines) are appropriate targets for intervention. These ideas are congruent with recent studies emphasizing that individually tailored coaching and mentoring may help to improve children’s participation [ 17 ]. As noted, however, change will not be effective if it is only targeted at the child. Contextual elements interact with mechanisms to make participation more or less likely and must also be a focus for intervention.

With a focus on school, teachers’ knowledge is of key importance [ 105 ]. Efforts are required to assist teachers’ regarding knowledge and confidence in enhancing participation. Teachers work with increasingly diverse groups of learners and are responsible for attempting to achieve positive outcomes [ 106 ]. Concerns have been expressed that education remains less effective for learners with disabilities [ 2 ]. Concerns are understandable particularly when schools and teachers tend to be rated on achievement, rather than participation [ 107 ]. Existing supports, strategies and approaches for children with disabilities, along with theoretical underpinnings, are frequently superficial and lacking in detail [ 108 ]. Practical aspects of how to “do” inclusion or participation are therefore difficult to see and implement. Previously developed supports and interventional resources have also tended to focus on specific issues or diagnoses (e.g. Autism, Dyslexia, Learning Disability)–leading to “a programme for every problem” [ 109 ]. This has two consequences. Firstly, educationalists follow a medical or disease orientated model, with the consequential issues around disempowerment and depersonalization of people with disabilities [ 18 ]. Secondly, those with responsibility for supporting children with disabilities may feel overwhelmed by the range of options [ 105 ]. The complexity and number of programs makes selecting the right option for the right child at the right time difficult.

Implications for research

Future research could explicitly link intervention components to mechanisms as described in this review. Following methods which use formal means for developing theoretically determined interventions [ 110 ], ‘theory-based’ rather than ‘theory-inspired’ interventions, may be developed. Such research is closely aligned to the UK MRC framework for development of complex interventions [ 27 ]. Identified mechanisms offer a basis for understanding how and why therapeutic or educational interventions for children may or may not be effective at improving school participation. Identification of strategies for the detection and cultivation of facilitative contextual elements would also follow from the above methods.

Further research activities include selection of appropriate items for school participation measurement. Parent-report methods have been commonly used in medical and psychological research to collect participation information [ 86 ]. However, researchers should also consider other data collection methodologies, particularly report by teachers [ 19 ].

Limitations

While we have attempted to make our search as sensitive as possible (and erred on the side of sensitivity as opposed to specificity), participation continues to be a diverse area spanning several disciplines with limited consensus on terminology. It is difficult to design a perfect strategy. Given the methodological assumptions of realism, other reviewers could come to different conclusions. However, themes and concepts driving the model were apparent across different types of difficulties/disabilities, across studies that used different research methods, and across a range of international contexts. Consistency in identified features provides evidence to support conclusions.

Conclusions

This was the first realist review to explore mechanisms and contexts for school participation of children with disabilities. This paper presents a conceptual framework including child psychosocial factors, such as understanding of routines, sense of self, and perceptions of role, and as well as characteristics of the school environment. We encourage researchers, practitioners, and policymakers to consider these contexts and mechanisms when addressing school participation among children with disabilities. Consideration of interventions, designed specifically to enhance participation by targeting mechanisms, contexts and the processes identified in this review, is now key.

Supporting information

Acknowledgments.

The study was initiated by a partnership between the Salvesen Mindroom Centre in the University of Edinburgh and the CIRCLE Collaboration (Child Inclusion: Research into Curriculum, Learning and Education) research team at Queen Margaret University, Edinburgh. We would like to acknowledge Miriam Crowe, Deborah McCartney and Cathleen Hunter for support and critical comment on the manuscript. Funding for this work was received from the Salvesen Mindroom Centre for Learning Difficulties.

Funding Statement

Funding for this work was received from the Salvesen Mindroom Centre for Learning Difficulties.

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