Ethics statement

The Community engagement and involvement (CEI) activities reported in this paper were mainly consultations with key stakeholders to develop an evidence-based conceptual framework designed to improve services. The community consultations were undertaken within the recommended ethical standards, including verbal consent to use quotes from stakeholders without identifying them, and their right to withdraw this approval any time without providing any reason. This type of CEI activity is exempt from research ethics review by the University of Oxford’s ethics committees and by the funder, National Institute for Health and Care Research (see INVOLVE statement https://www.invo.org.uk/posttypepublication/public-involvement-in-research-and-research-ethics-committee-review/). Stakeholders contributed to the design of the conceptual framework, and were not involved as research participants. This work was also motivated by an ethical commitment to engaging communities in study design to ensure that the study plan reflects the priorities of the community and is responsive to the specific community context, including contextual understandings of the potential risks and benefits of research.

From April through to July 2020 we undertook community engagement and involvement (CEI) activities using a community-based participatory approach, guided by the bio-psycho-social model. This approach underpins the World Health Organisation’s (WHO’s) International Classification of Functioning Disability and Health (ICF) [20] to focus on behaviour change factors and access to healthcare services which could influence opportunities, capability and motivation of parents and children to engage with a health related intervention designed to reduce risk factors for poor health outcomes [21,22]. Working with stakeholders we developed a conceptual framework and a working model of a ‘Theory of Change’ to delineate a pathway that reflected the priorities of the target population [23]. We adapted the five steps recommended for the design and evaluation of a public participation process for the health sector [24].

1. Representation of a range of community stakeholders

We consulted with 70 key community stakeholders in ten States in India, eight in the northeast region that have a high prevalence of ID (Assam, Arunachal Pradesh, Manipur, Meghalaya, Nagaland, Sikkim, Mizoram, Tripura) and Delhi and Maharashtra. We aimed to include stakeholders from all socio-economic backgrounds. There were no exclusion criteria as we wanted to include a wide and diverse range of views from people who either had children with intellectual disability or had an experience of working with or supporting children/adults with intellectual disability. Stakeholders were identified through snowballing, starting with a local organisation of parents of children with ID, and initiated by co-investigators from India (psychologist MH; and parents’ representative NS). The stakeholders were: i) parents of children with ID; ii) professionals from health, education and social care who were engaged in caring for children and adults with ID; iii) policy makers (including commissioners), lawyers, and members of parents’ organisation and other non-governmental organisations (NGOs) working with people with ID.

2. A systematic approach to stakeholder interviews and consultation

We conducted three iterative rounds of one-to-one consultations with each of the 70 stakeholders. To overcome the constraints of social restrictions due to the COVID-19 pandemic, we followed guidance on e-consultations [25] to enable us to extend opportunities to as many stakeholders as possible via emails, and through tele and video-conferencing. We organised face-to-face meetings (mainly with parents) after the lockdown measures were eased in June 2020. Two psychologists (MH and NHK), parents’ representative (NS) and a PhD student (PS) from India conducted the consultations.

In the first round of consultations, we used five case studies of children with ID to initiate discussions. The case studies were compiled through an in-depth face-to-face discussion with parents prior to the COVID-19 pandemic. They provided stakeholders with a general understanding of the day-to-day living experiences, challenges, health and other concerns, fears and hopes of parents/carers of people with ID. We explored broader and more general perspectives on the:

• Day-to-day problems and needs of children with ID and their families.
• Support systems and structures available to the children and their families.
• Concerns about health and wellbeing of the children (current and future).

In round-2, we used a semi-structured topic guide to explore the stakeholders’ perception of risk factors for communicable and non-communicable diseases in children with ID and how we could start to address them within the wider context of the structural and social barriers. In round-3, we obtained critical feedback on a draft conceptual framework (described below) and a Theory of Change model developed from the findings of round 1 and 2. In particular, the stakeholders were asked to identify limitations, assess the relevance of the concepts, acceptability, success criteria, integration with existing care pathways and health system (private and public), how these relate to existing health policies and who would be best placed to deliver the intervention.

3. Information generated and iteratively reviewed

Consultations undertaken in local languages were transcribed and translated to English. Transcripts of each of the 70 consultations were coded, using set and emergent themes with constant comparison [26]. The themes were assessed by study investigators (MN, SS, MH, NS, HM, MW, TKM), and ambiguous and uncertain themes were further explored with stakeholders in subsequent rounds. An iterative team coding approach was used to check interpretations and improve validity [27].

4. Developing a conceptual framework and theory of change

We mapped the findings from the consultations with evidence from systematic reviews to develop a conceptual framework to inform the development of a health promotion and disease prevention intervention that could reduce the known risk factors for long-term health problems in children with ID while taking into account the broader socio-economic contexts and barriers. The framework was co-developed by the study team from India and the UK working with experts from the Indian government and non-government institutions including three national bodies:

1. National Institute for the Empowerment of Persons with Intellectual Disabilities (NIEPID), Secunderabad under the Ministry of Social Justice and Empowerment, Government of India;
2. National Institute of Mental Health and Neurosciences (NIMHANS), Bengaluru under the Ministry of Health and Family Welfare, Government of India; and
3. PARIVAAR–National Confederation of Parents’ Organisations (NCPO) working with persons with ID.

Using our findings we worked backwards to sketch out the pathways for potential change and its preconditions, incorporating the contextual, structural and social shifts that could facilitate the intended changes within a Theory of Change.

5. Triangulation of findings for decision-making

Information from the three rounds of inter-linked and iterative discussions were triangulated to modify and refine the conceptual framework for a cross-sectoral family-centred intervention for children with ID and their parents. This was undertaken by the study team working with the same set of stakeholders who developed the initial conceptual framework (step 4 above).

Round-1 outputs

Three key themes emerged from the first round of consultation, the most important being the ‘future of the children’, followed by ‘inclusion’ and ‘support systems’. Almost all parents were concerned about the future care and wellbeing of their children when they would no longer be able to provide care. Marginalisation and exclusion were the factors driving insecurity and exacerbated their worries about the future of their child.

“I am a single parent, after my son’s birth my husband divorced me and so we are staying with my parents. But I am suffering with insecurity when I think about his future life after my demise or in my old age. Please if it is possible you should include this issue in you[r] programme.”

(Mother of a child with ID)

“People should behave as normally as they behave with anyone else. That is the bare minimum they can do. Moreover, I expect society to be more tolerant and accommodating in general.”

(Mother of a 43 year old woman with ID)

The majority of professionals (educators, health and social care workers) and members of parents’ organisations felt that parents should be provided with adequate knowledge and information, and be actively involved in educating and caring for the child at home. Active involvement and regular communication with healthcare providers and educators could reduce their anxiety, improve their understanding and acceptance of the child’s condition, and help to improve their mental wellbeing through support. One of the project stakeholders believed that parents are not adequately informed about their children’s condition and provided suggestions on how support can be provided at home.

“…they put the entire hope and aspiration on the schools and SSA [Sarva Shiksha Abhiyan] functionaries. …….Empowering teachers with the right skills is necessary and also empowering parents about their child’s condition and how to care at home is equally important.”

[Special educator, SSA (Inclusive education programme)]

Improving access to and involvement with healthcare services in both the public and private sectors were identified as important to improve the support system for children with ID and improve physical and mental health of the children and their parents. The overarching concern of parents about securing the future for the child beyond just putting food on the table and day-to-day care was an important observation.

“The benefit of parental and family involvement in the rehabilitation program has never been understood better than now. Professionals are beginning to realise the benefit of family approaches over merely child-centred ones. This point requires consideration.”

(Member of a parents’ organisation)

Round-2 outputs

A few professionals were aware of a higher risk of communicable and non-communicable diseases in people with ID, and suggested that there was a need to improve awareness about the importance of early interventions to reduce future risk of diabetes, cardiovascular diseases, mental health problems, tuberculosis, COVID-19 and other infections. An increased risk of physical health problems was attributed to a range of factors that included malnutrition, infection, poverty, poor access to healthcare services, societal factors such as those related to stigma and low inclusion, behavioural and lifestyle factors, and a lack of awareness and knowledge about health promotion and disease prevention among parents and individuals with ID.

“Children and persons with ID besides being faced with known health concerns, are more vulnerable and susceptible to other illnesses due to lower immunity levels and also because they are not aware and equipped with required knowledge to take good care of their general health–both mental and physical.”

(Deputy Director, Civil Society organisation)

“Another critical concern is the lack of acknowledgment among parents and other caregivers about the sexual needs, identities and expressions of children and adolescents with ID. Championing for the same could be viewed with a lens of censorship, when societal norms dictate that these are ‘private’ matters. As a result, these are not addressed timely and adequately, thereby impacting the mental well-being of people with ID and sometimes also resulting in unwanted changes in their behaviours like resorting to violence and so on.”

(Deputy Director, Civil Society organisation)

Parents of children with ID mostly reported not being aware of any increased risk of long-term health problems, but mentioned that they would do everything required to prevent it. The statements quoted above also point to a lack of awareness of inclusive approaches amongst parents, professionals and the wider society. An educator raised concern about the lack of health promotion and disease prevention programmes for children with ID.

“Government has taken up education of these children as a priority, whereas health [promotion] still remains to be addressed. Maybe it will be prioritised in the next phase. NGOs are also focusing more on education than health. I would press for a health related programme.”

(Educator, inclusive education programme)

The common recommendations for reducing the increased risk of long-term health problems were to increase physical activity levels in children with ID by integrating this with daily activities and to improve their nutrition. Social workers supporting children with ID suggested that the intervention should be tailored to the needs of children from different socio-economic backgrounds, and take into account the severity of disability and any co-existing conditions. We were advised to keep the intervention simple, with separate components for undernutrition and obesity, to ensure it is sustainable and build on the existing Early Intervention Services for children 0–3 years offered by the Rashtriya Bal Swasthya Karyakram of the Ministry of Health and Family Welfare, Government of India [28]. This will also facilitate an inclusive approach, as the intervention will be embedded in a mainstream programme for all children. Experts from the three national bodies advised us to target the age group of 4–10 years to support continuity of care and integration with the existing policies and programmes.

“Early childhood interventions are necessary and should be continued as part of the programme. If a child does not know how to brush, how can you improve dental hygiene?”

(President, National parents’ organisation)

Conceptual framework that underpins an intervention to reduce risk factors for long-term health problems

A report on the state-level burden of disease in the general population highlights the triple-burden of infectious and lifestyle related diseases, and mental health problems among the adult population in India. These are associated with risk factors such as malnutrition, high blood pressure, high blood sugar, stress and anxiety, which are in turn related to unhealthy diet, tobacco, alcohol and drug use, socio-economic factors, and unsafe drinking water, sanitation, and hygiene [29]. A review of systematic reviews indicated that interventions to increase physical activity had a large beneficial effect on physical and psychosocial health moderated by age and level of disability [30] although this evidence mainly came from high-income countries. Sustained behaviour change was higher when combined with a healthy lifestyle and supportive structural and socio-economic environments [31]. Community-based programmes that provided meaningful parent participation (e.g. through having fun, role models) reduced the burden of caregiving [32]. Integrated mental health counselling services within a health promotion programme for children with ID was found to be effective in reducing stress and anxiety among parents [33]. Evidence also supported collaboration with adults with ID in all aspects of research to improve the implementation and sustainability of programmes [34].

Combining the evidence with the findings from the stakeholder consultations, we developed a conceptual framework that covered an enabling environment, structural and socio-economic determinants, integration with health and social care, intermediate modifiable risk factors and the long-term burden of multiple health problems in later life (Fig 1). This led to the identification of five evidence-based core components to underpin a family-centred, cross-sectoral, community-based intervention for children with ID aged 4 to 10 years:

1. A physical activity programme for the children.
2. A guide to a healthy diet to provide good nutrition to the children using locally available and affordable food.
3. Training parents to continue regular adaptive behavioural and cognitive stimulation, including in everyday activities alongside other children who do not require specialist resources.
4. Education and training to improve parent’s knowledge of accessing healthcare services.
5. Group counselling for parents and children to increase their capability to address behavioural issues, individual barriers to exercise and how to follow a healthy diet, improve emotional and mental wellbeing, and to encourage social inclusion.

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Fig 1. Conceptual framework for a community-based intervention for preventing and reducing risk of long-term health problems in children with intellectual disability.

https://doi.org/10.1371/journal.pgph.0000833.g001

In consultation with stakeholders we developed a Theory of Change (Fig 2) to identify how the community-based intervention leads to a lower risk for longer-term health problems in our target population of children with intellectual disability, and the necessary preconditions for a beneficial change to occur for these children and their parents. The Theory of Change model therefore includes several stages starting from how the invention was conceptualised during the proposal and partnership development phase to what long-term impact would look like if the intervention was developed and tested effectively leading to the expected outcomes, and the necessary pre-conditions for the success of the intervention were met. The model shows that the cross-sectoral family-centred programme is not a standalone intervention, but will need to be supported by an inclusive enabling environment (Fig 1). Therefore, it will be essential to identify and mitigate social and health system barriers, and actively promote community mobilisation to integrate the intervention within existing health and social care services (Fig 2). While the intervention could achieve the immediate outcomes within a short period (example improving physical activity, diet, and access to healthcare, and reducing stress and anxiety), it will take longer to observe an impact on reducing the long-term health problems in children with ID. This is clearly acknowledged by the accountability ceiling and the plan to transfer ownership of the intervention to the three national apex bodies engaged in developing the conceptual models. The Theory of Change model will evolve as the intervention is tested and implemented, along with growing partnerships and changes in political will, which will have an effect on the accountability ceiling and transfer of ownership from local to the national bodies. The stakeholders acknowledged that widespread advocacy would be required at each stage of the Theory of Change model to ensure that the intervention tested locally is appropriately adopted at a national level to have the desired impact on policies and practices for a sustainable impact. The team developing the intervention is perfectly placed to undertake such advocacy work as it includes representatives of three national organisations that are involved in developing national policies and programmes for people living with intellectual disability, as well as community stakeholders, people with intellectual disability, and parents of children with intellectual disability.

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Fig 2. Theory of change model.

https://doi.org/10.1371/journal.pgph.0000833.g002

Round-3 outputs

Stakeholders voiced strong support for the conceptual framework that inform the intervention to guide the planning and delivery of services for this population, and agreed that “it will mark a beginning” (Educator, Inclusive education programme) and promote the “value of life of the child” (Carer of a child with ID).

“The programme would be a good start of a holistic intervention. Done with perseverance, it can positively impact the child’s coping skills, health and well-being and relieve stress in the family as the child begins to benefit from inputs received.”

(Deputy Director, Civil Society organisation)

Importantly, it was suggested that we develop a contextually relevant intervention and an enabling environment that will address the challenges and potential barriers to adherence to the intervention components.

While several stakeholders suggested that grass-roots level public sector health and nutritional workers such as Anganwadi workers (village nutrition worker) and community health workers (Accredited Social Health Activist (ASHA)) should be trained to deliver the intervention, others were worried about the negative impact on quality and sustainability if it is delivered by this already over-burdened workforce. It was felt that care providers employed by government and non-government institutions, such as the health system, nutrition and social welfare sectors and schools for children with disabilities would be better placed to deliver the intervention due to their understanding of the children’s complex needs. The district disability rehabilitation centres [35] could coordinate outreach, delivery of the intervention and follow-up. The trained workers in these centres are already working towards improving societal awareness and attitudes, community inclusion of people with disability, and school enrolment, and helping families access services, benefits, and support for their children [36].

Strengths and limitations

A community-based participatory approach was used for consulting and actively engaging stakeholders in co-developing a ‘locally conceptualised’ needs-based intervention to achieve meaningful results that maximises the benefits for children with ID and their parents. Our structured approach could be used to undertake other CEI activities in India and other LMICs, where communities and individual members of the public are not commonly engaged in developing an intervention. Consultations are thought to be more effective if they are conducted in open fora where the public can debate and challenge emerging concepts. Although we could not reach out to a wider community because of the social restrictions related to the pandemic, we minimised the chances of developing a biased model by listening to and evaluating multiple perspectives along with experts from the three national bodies who have several years of experience working with children and adults with ID in the Indian context. Another potential limitation is that systematic reviews used to inform the evidence base of the conceptual framework was from mainly high-income countries. Nevertheless, the evidence was a necessary starting point. Evidence generated from a LMIC context is required to strengthen the conceptual model, guide implementation and assess how best to align with the “disability-inclusive development” framework [45] when substantial socio-economic and health system challenges exist. Our work reinforces the United Nation’s Convention on the Rights of Persons with Disabilities (UNCRPD) and the Sustainable Development Goals’ (SDGs) commitment to “leave no one behind”. It also contributes towards building inclusive societies and institutions and to the goals of ‘Envision disability 2020’ [46].