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Moving beyond prevalence studies: screening and interventions for children with disabilities in low-income and middle-income countries
  1. Aisha K Yousafzai1,
  2. Paul Lynch2,
  3. Melissa Gladstone3
  1. 1Department of Paediatrics and Child Health, Aga Khan University, Karachi, Pakistan
  2. 2Department of Inclusion, Special Needs, School of Education, University of Birmingham, Birmingham, UK
  3. 3Department of Women and Children's Health, Institute of Translational Medicine, University of Liverpool, Liverpool, UK
  1. Correspondence to Dr Aisha K Yousafzai, Department of Paediatrics and Child Health, Aga Khan University, Stadium Road, PO Box 3500, Karachi 74800, Pakistan; aisha.yousafzai{at}aku.edu

Abstract

Research understanding the lives of children with disabilities in low-income and middle-income countries has predominantly focused on prevalence studies with little progress on evidence-based service development. At the same time, global attention in child health has shifted from child survival strategies to those that bring child survival and development together. This review examines whether intervention research can be better aligned with current theoretical constructs of disability and international guidelines that advocate for the realisation of rights for children with disabilities and inclusive early childhood development.

  • Children With Disabilities
  • Inclusion
  • Interventions
  • Screening
  • Low and Middle Income Countries
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Introduction

This year, the ‘State of the World's Children’ report published by UNICEF was dedicated to children with disabilities (CWD) who represent an estimated 1 in 20 children aged less than 14 years.1 This paper, like several others, draws attention to the lack of inclusion of CWD in the development agenda and the negligible attention given to service development and legislation promoting well-being and opportunities for this population.1–4 Research understanding CWD in low-income and middle-income countries (LMIC) has predominantly focused on epidemiological questions,3 while related research on effective interventions has been sparse.

The purpose of this review was to describe and critique intervention research on CWD living in LMIC in light of current debates, and theoretical constructs on disability in order to make recommendations about future research directions that may increase inclusion and participation of this population. Peer reviewed articles on interventions for CWD in LMIC published since 2000 were sought from electronic databases (BEI, ERIC, ProQuest, PsycInfo, PubMed and Google Scholar). The primary objective of this paper was to describe the breadth and quality of intervention research about CWD in LMIC. The secondary objectives of this paper were to: (1) describe the challenges of intervention research and make recommendations about how future research can better use current disability constructs; and (2) discuss how the research agenda for CWD can be better aligned in existing child health and development priorities. Given the paucity of intervention research for this population, the recommendations in this paper also incorporate the discussions and field observations of the three authors who have researched childhood disability in LMIC over the last 10 years.

Understanding the rights of CWD: implications for strategy development

The United Nations Convention on the Rights of Persons with Disabilities5 (UNCRPD) enshrines a social model of disability, which focuses on reducing or eliminating actions that discriminate and therewith hinder access for persons with disabilities (PWD). These barriers could be the result of the built environment but, moreover, the result of social and attitudinal barriers, such as stereotypes, prejudices and other forms of paternalistic and patronising treatment. The UNCRPD states a clear need to recognise the valued existing and potential contributions made by PWD to the overall well-being and diversity of their communities, and ensure the promotion of the full enjoyment by PWD of their human rights, fundamental freedoms and of full participation to advance human, social and economic development.

The International Classification of Functioning, Disability and Health6 (ICF) provides a practical way of understanding the complexity of disability and offers a way forward for researching and advancing rights for PWD.7 Disability is defined by impairments (structure and the functions in the body that are affected), which result in activity limitations and participation restrictions (eg, barriers to going to school). The framework recognises that an individual's functioning is an interaction between health, environment and personal factors. For example, assessment and design of interventions are based on the child's profile of functioning defined by activities and participation influenced by barriers and facilitators in the child's environment.8 The framework encourages the collection of data to inform strategies, which support a child's (re)habilitation needs and promotes opportunities and quality of life through reducing barriers to participation.9

Similarly, the capability approach10 ,11 assesses the relevance of impairment and disability in designing fair and inclusive institutional and social arrangements. In capability terms, it does not matter whether a disability is biologically or socially caused; it is the scope of capabilities a person can choose from and the role the impairment plays in this set of choices which is more important. In many respects, it considers what people are actually able to do and how they can be enabled to achieve their objectives.11

Community-based rehabilitation (CBR) has been a recommended approach for adults and CWD promoted by WHO since the 1970s. Previously, CBR was criticised for not adequately addressing the medical rehabilitation needs of PWD, focusing on economic development, neglecting the psychosocial well-being of individuals and not having adequate participation of PWD. The focus on care in the community failed to recognise the importance of partnership with services (eg, health, education).12 The updated WHO Guidelines for CBR13 considers a twin-track approach ensuring (1) disability issues are integrated in mainstream development work and (2) more targeted activities for PWD are implemented where necessary.

It is important to establish a conceptual framework for research to inform policies and practices that draw on the ICF, the capability approach, the UNCRPD and the guidelines for CBR which focus on identifying CWD, and enhancing quality of life, developmental potential and participation in society.

Approaches to the identification of CWD

Identifying CWD is the first step in providing interventions. Researchers debate over best methods of identifying CWD at population level.14

General screening and survey methods for childhood disability

Survey methods used to identify CWD include the Ten Questions Questionnaire (TQQ).15–17 The TQQ identifies seizures and impairments in cognition, motor, vision and hearing and seizures for children aged 2–9 years by caregiver report. There have been concerns that the TQQ overidentifies children with hearing difficulties and seizures, but also that it only reliably detects moderate to severe disability, and milder conditions may not be identified or understood as a problem by the family. For example, items such as ‘does the child learn to do things like other children his or her age?’ is an item that relies on the awareness of families to detect milder learning difficulties and, therefore, may underestimate levels of impairment. Good sensitivity and specificity is demonstrated particularly if paired up in a two-stage approach where first screening and then more extensive assessment is conducted. The TQQ has been incorporated into the ‘Early Child Development’ module in the third round of the UNICEF Multiple Indicator Cluster Survey Programme. This has proved effective in comparing countrywide rates of childhood disability and has the potential for determining investment in programmes that target specific needs of CWD or their inclusion in mainstream services.

Incorporating the ICF principles in user-friendly and globally applicable screens are emerging. Teams in Nepal and Bangladesh have created the 10 Questions Plus,18 and the Washington Group of Disability Statistics are piloting a tool with 14 questions which identify PWD in terms of functional limitations and limits to independent participation in society.19

Key informant methods

The key informant method (KIM) is an approach where knowledgeable members of the community (key informants) are trained to effectively identify children with moderate-severe physical impairments, sensory impairments and epilepsy.20 ,21 These children are then screened by medical professionals and referred on for appropriate health or educational interventions. A recent study in Bangladesh demonstrated KIM as having high rates for case detection, but low specificity particularly for hearing impairment.22 Many proponents of KIM advise that it allows a direct connection to appropriate referral services, which survey and screening methods may not do. In this way, KIM allows for a pre-emptive mapping of referral services by collating information on service availability and affordability. Through its reliance on community volunteers and networking it can engage local stakeholders and build capacity and knowledge sharing.14 ,23 Through community involvement, the KIM and similar participatory rural appraisal approaches encourage strategy planning that is locally feasible and relevant.

Developmental screening

Identifying children at risk of developmental difficulties can be done using developmental screening tools where simple questionnaires are used to assess normal attainment of milestones. These can be parent-reported (Ages and Stages Questionnaire (ASQ)/Parents Evaluation of Developmental Status (PEDS))24 ,25 or observed (Denver II).26 Children found to have delay on age-appropriate standardised norms are then referred for further assessment. This is not often possible in low-resource settings, and it is disputed as to whether developmental screening tools fulfil screening criteria and should be promoted for universal use. There is often a balance between high sensitivity (identifying all children with delays) and erroneously identifying children who do not have true delays (specificity).27 Furthermore, the definition of abnormal is not always clear and may depend on the availability of diagnostic and remedial services in countries.

Although, there is evidence of benefit in early intervention programmes for children with hearing loss, intellectual disabilities and autistic spectrum disorders,28 ,29 it is still not entirely clear what effect early intervention has on all childhood disabilities. Early intervention may lessen the impact of a disability on the functioning of the child and family, and early diagnoses may allow families to adapt to difficulties their child faces.30 Many argue that screening is not effective but surveillance (where primary care practitioners opportunistically ask flexible questions about a child's development at every visit, and children identified as at risk are then provided with advice and referred) is what is needed. Some countries, such as the USA still advocate using a screening tool as part of this approach.31

There have been a number of developmental screening or surveillance tools adapted or specifically created for LMIC. The Denver II was adapted and translated for WHO studies in many LMIC,32–37 but may not be sensitive and specific enough.38 Parental report measures, such as the ASQ or the PEDS,39–42 are popular and are good detectors of developmental delay.43–49 These tools are beneficial as they enable parents to be actively involved in the evaluation of their children. Often tools are not adequately adapted or validated for a local population.50–53 A small number of developmental tools have been created or adapted specifically for developing country settings,54 ,55 some with good measures of validity and reliability.

The challenge with many tools is that they do not link screening to services, therefore, limiting the advantage of screening. The Guide for Monitoring Child Development in Turkey is an example of a screening tool using open-ended interviewing to gain information on child development followed by appropriate advice.56

Interventions for CWD: impact on children and families and implications for research and services

Evidence-based strategies for screening and appropriate advice in LMIC will require innovative and feasible models given the resource limitations. One such innovation in Bangladesh was the creation of ‘Developmental Therapists’ to address the multiple therapeutic needs of CWD in a context where access to specialists was limited.57 However, the design of the interventions must also consider that many families are unlikely to make frequent visits to rehabilitation facilities; therefore, a greater focus on home and community-based interventions is needed.

Six intervention studies for CWD in LMIC were identified (table 1).58–63 The studies, from Bangladesh,58 ,61 India,62 Kenya,59 Malawi60 and Vietnam63 are diverse with respect to disability and intervention content (eg, addressing issues of feeding communication, development and self-help) making comparisons difficult. However, a common feature across each intervention strategy is the involvement of primary caregivers to support home-based care of CWD. Kelly and colleagues,60 report that some of the challenges faced by primary caregivers are at the community level (eg, stigma), and that intervention design must consider community-level enablers and barriers, such as those reported in a combined community-based and family-based model implemented in a slum community in India.62

Table 1

A summary of intervention studies for CWDs in low-income and middle-income countries

Drawing conclusions about the external validity for the six studies is challenging. There is only one randomised controlled trial,61 and only two studies include a control comparison in the analysis.61 ,63 Two of the studies include a premeasure and postmeasure from either quantitative58 or qualitative data,59 and the remainder are descriptive evaluations based on case studies or a case series. All have relatively small study samples, and where reported, the attrition rate is high.58 ,61 Finally, the categories of outcome measures are variable; a direct measure of child outcome is reported in three studies,58 ,61 ,62 a caregiver report of child progress is reported in two studies,59 ,60 and most studies report some outcome related to the caregiver's well-being or perception of support. However, little information is available on the reliability and validity constructs of the assessment tools used for these populations. In sum, as pilot interventions to guide trials and future evidence-based practice, authors should report information on compliance, dosage, quality (eg, skills of the trainer) and challenges and enablers for the intervention. Multiple levels of outcome measures will be useful that address change at the level of the child, family, trainer and, if relevant, the community. Box 1 describes the potential challenges when undertaking intervention research for CWD.

Box 1

Challenges for intervention research

  • Adequate sample size and level of homogeneity within impairment groups is frequently difficult to achieve for studies. The creation of multisite networks for research in low-income and middle-income countries may be a way forward.

  • A study population may comprise children with a wide range of complex needs; therefore, it may be difficult to draw conclusions about specific interventions and impairments (eg, a child with autism using one type of intervention, and another child with Down Syndrome using another type of intervention). It is necessary to be able to identify the underlying features of effective intervention strategies from less effective strategies through an analysis of the process (eg, effective techniques to work with families and resolve problems, types of materials and resources that are useful for families), which may contribute to a set of generic guiding principles for intervention design.

  • The human resource constraint in many low-income and middle-income countries, often means a reliance of training of ‘unskilled personnel’ to undertake family visits. These need good training in the intervention for the child, and also around disability awareness and on reducing prejudices towards families who are considered to be unresponsive or uncaring towards children with disabilities. Adequate documentation of lessons learned from these training strategies is necessary.

  • The selection of outcomes requires careful consideration of the implications of using standardised tests and assessments developed in high-income settings that may not be appropriate in different social, cultural and economic settings. Appropriate adaptation and reporting of reliability and validity of measures used is necessary.

  • More robust intervention studies are required. Most intervention studies have been pre-evaluation and postevaluation designs, with only one randomised controlled trial. It is timely and necessary to seek opportunities for controlled experimental designs.

  • The cost of interventions should be evaluated as a component of research.

  • Few studies have considered the formal systems in the settings in which they have been implemented. A greater focus is necessary on how family and community-partnerships engage with the formal systems in order to provide information for sustainability and scaling up effective strategies. Such data will also help donor organisations to decide on what areas they should invest more money.

Impact of caring for CWD on caregivers

Understanding the impact of caring for CWD on caregivers and the extent to which current services consider the role and the context of caregivers is critical. Seventeen studies were found that explored the impact of caring for CWD on caregivers (table 2).64–80 A common set of themes were reported in qualitative studies, which highlight the need for interventions to raise awareness on childhood disability, reduce social stigma, build support networks, and support mothers who were often taking the greater caregiving responsbility.64 ,66 ,74 ,80 In all settings, families reported inadequate services and resources. In Pakistan, parents of children with intellectual disabilities reported the major barrier to care seeking was not availability of services, but lack of information or money. On average, there was a 2–4-year gap between the child initially being identified and contact with a healthcare provider.74

Table 2

The scope of studies about caregivers of children with disabilities

Consistent with findings from high-income countries, studies in LMIC report high levels of stress among caregivers of CWD.65 ,68 ,75 ,76 ,79 Child characteristics associated with stress include caring for a girl with disability,75 caring for older CWD75 and child behavioural problems.76 Caregiver characteristics associated with stress include being a younger mother65 and poor maternal adaptation to the child.76 In Pakistan, mothers were found to have significantly higher levels of stress compared with fathers74; however, no significant differences were reported between parents in Lebanon where the degree of social support, stress and paternal education status were associated with coping capacity.65 Similarly in Vietnam, community participation (eg, contact with relatives or friends, participation in religious gatherings) were also associated with stress and coping.79 Mixed findings were reported from the intervention studies for CWD on the treatment effect on parental stress. In an intervention to improve child feeding skills, maternal stress was reduced,58 while in another study, caregiver participation in a training programme increased stress.61 In the former, stress may be highly associated with the feeding challenges of the child, whereas in the latter case, increased knowledge and time on interventions may have contributed to stress.

Physical health and well-being of the caregivers is of particular importance for those caring for children with physical disabilities; however, there has been little research on exploring the impact of caring for CWD on the physical health of caregivers, especially in contexts with limited access to assistive devices. An exploratory study in Kenya found all caregivers caring for children with moderate-severe motor impairments reported chronic pain.68

The implications for intervention strategies from these studies about caregiver experiences highlight the need for focus to shift from the child alone to the child, family and wider community. Community awareness strategies about childhood disability are needed in order to mitigate the stigmatisation of CWD and their families. Interventions that reduce some of the daily stress experienced by caregivers include approaches to harness family support strategies to increase social and family support.

Previous research from LMIC has consistently described the caregivers' need for information about the child's condition.3 Importantly, more recent work has examined the range of information required by caregivers; for example, in Iran, learning from the experiences of other families who cared for a child with a similar condition was important.73 ,77 ,78 In Bangladesh, despite having information from health workers; care-seeking practices by mothers of CWD were influenced by the elder generation who preferred to seek traditional or spiritual treatments.71 Knowledge dissemination requires a strategy that ensures consistency across sources of information, conflict resolution between contradictory sources of information, peer learning and access to information in a local language. Finally, studies about caregivers also impart important learning about the delivery strategies of interventions that should take into consideration the circumstances of caregivers which impact on uptake of potential services.70 ,72

Conclusions

Considerable progress has been made over the last decade in recognising the significant numbers of CWD globally, and the associated risks and consequences for life outcomes. Opportunities to include the reporting of childhood disability have been capitalised upon in national surveys. However, this has not resulted in a significant shift in political commitment and investment in programmes. It is now justified to go beyond counting CWDs. International agreements, such as the UNCRPD in combination with the ICF, the Capabilities Approach and CBR provide a framework for intervention design and delivery. By using these frameworks and approaches we have opportunities to link screening with services. It is recognised that a combination of formal services (with family-based approaches) and family participation is needed in intervention design. For too long, the care for CWD in LMIC has been family led with little professional support. Learning from the experiences and solutions of these families may enhance the support and advice provided in formal services. These strategies need to be combined with community awareness to reduce the challenges families experience as a result of stigma, and remove barriers to participation in health, education and community life. Robust studies are required to inform policy and practice resulting in external validity of findings, analysis of process and information on cost savings. Box 2 outlines the ways forward for intervention research.

Box 2

Ways forward in intervention research for children with disabilities in low-income and middle-income countries

Screening Linked to Intervention

  • Integrated designs connecting screening with service are ethically important and necessary in developing services for children with disabilities.

  • Partnership between formal services and caregivers in screening approaches provides an opportunity to develop strategies that are contextually relevant, needed and feasible.

Whole Family Approach to Intervention

  • Intervention content and delivery must not look at the child alone, but focus on a whole-family approach.

  • Identifying and further exploring supportive strategies for caregivers need to be evaluated (eg, encouragement of other family members to help with daily care, incorporation of a problem-solving approach, opportunities to engage with other families in the same situation, information sharing, and exploration of coping strategies). The theories of change for these strategies need to be documented in research dissemination.

  • Intervention designs which depend on family involvement must measure the parent's willingness to change events or circumstances in their lives through formative research and intervention evaluation. Addressing questions, such as whether the intervention will lead to more time for parents to do other things, such as chores or earning income, will be helpful in ‘hooks’ for the intervention success.

  • Training and supervision strategies for delivery agents must be evaluated. What are the critical skills and how can these skills be translated into practice for effective intervention delivery (eg, tailoring generic interventions for individual needs).

Evaluation Framework for Intervention

  • The International Classification for Functioning, Disability and Health, the Capabilities Approach, the Community-based Rehabilitation Approach and the United Nations Convention on the Rights of Persons with Disabilities provide frameworks for intervention design and evaluation.

  • It is critical for intervention programmes to measure outcomes in children with disabilities across different domains of development, and to examine how these interventions may impact activity limitations, participation restrictions at the personal and environment level.

  • Broader intervention strategies need to think about how the interventions encompass broader skill-sets that will enable children to become more independent and participate in their communities more. How does the training increase parents’ capacity to problem solve and ‘generalise’ skill-sets to family daily routines?

  • Evaluations should have specific outcomes that measure outcomes of parents’ training with variability associated with parents’ knowledge, skills and performance of the techniques they were trained in as well as their performance of the techniques; against the complexity of the child's impairment, functioning and behaviours.

  • The cost and cost benefits of interventions should be evaluated to enable programme implementers to make informed investment choices

  • Characteristics of interventions need to be identified from studies on a range of disabilities in order to develop guidelines for intervention development based on common features and distinct features for specific groups and contexts.

Collaborative Child Health and Development Partnerships

  • Opportunities for collaboration on large-scale child health interventions are needed to enable analysis of inclusion, targeting and impact of mainstream interventions on children with disabilities.

  • Capacity development for high-quality services and research for children with disabilities should be considered through partnerships with centres for health personnel (eg, doctors, nurses, developmental therapists, physical therapists) preservice and inservice.

  • A case for research investment in disability research with funders needs to be leveraged. A starting point for the research community is to convene a research priority setting exercise using the ‘Child Health and Nutrition Research Initiative’ model (CHNRI, http://www.chnri.org/).

Finally, international guidelines have been advocating for twin-track approaches for the inclusion of PWD in the mainstream development agenda, in addition to targeted interventions. Unfortunately, few research studies have taken the opportunity to review subsets of data on CWD in child health and nutrition studies. Within the child health agenda, global attention has been shifting from survival alone to strategies that enable children to thrive and develop healthily. Addressing the needs of CWD in mainstream early childhood development programmes is an opportunity for childhood development and disability researchers to work together for common goals.81 Inclusive early childhood systems have the potential to serve as foundations for a diverse and fair society.

References

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Footnotes

  • Contributors All authors contributed equally to the conceptualisation of the manuscript and searching for references. AKY drafted sections on caregiver support, PL drafted sections on theoretical constructs and frameworks of disability and interventions, MG drafted sections on screening. All three contributed to conclusions, and review of drafts.

  • Competing interests None.

  • Provenance and peer review Commissioned; externally peer reviewed.

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