Exploring the experiences of family carers of neurodivergent children and young people: A scoping review protocol

Background

Neurodiversity can be viewed as an umbrella term for ‘significant difficulties’ in the development of “specific intellectual, motor, language, or social functions”, with autism, Attention Deficit Hyperactivity Disorder (ADHD), intellectual disability (ID), and dyslexia being some of the most well-known conditions^1,2. Classified as ‘neurodevelopmental disorders’, these conditions are defined as “developmental deficits or differences in brain processes that produce impairments of personal, social, academic, or occupational functioning”³. The latter definition is aligned with the medical model of disability, which considers a disability to be a deficit or deficiency (e.g. challenging behaviours in an autistic^* child) that needs to be “cured”^4–6 to help a person “fit into society”⁷, and thus, the burden of change is on the person with the condition^8–10.

Neurodiversity as a social movement started more than thirty years ago among online autistic communities to challenge how science and society treated them^4,11,12,13. At that time, parents of autistic children were criticised by psychologists for “bad parenting”, and by disability groups for trying to ‘normalise’ their children¹⁴. In response, self-advocates from the autistic community urged parents to accept, respect, and value their child’s differences^14,15, and some proposed that society should be ‘cured’ instead¹³.

This new perspective on autism contributed to the emergence of the concept of neurodiversity^5,13. Neurodiversity is seen “as a natural expression of divergent human evolution”⁶ consisting of a variation in the neural pathways that interconnect different parts of the brain and the body¹⁶. As a result of these “neurological differences”¹⁴, neurodivergent individuals are believed to “experience and interact in the world in different ways”¹⁷. For example, autistic children may be extremely sensitive to sensory inputs or feel uncomfortable in social interactions¹⁸.

The neurodiversity paradigm aligns with the social model of disability¹¹, which holds that disability is distinct from a health condition¹⁹. Instead, it is the interaction between a health condition (e.g. autism), environmental (e.g. lack of accessible transportation)⁹, and societal factors (e.g. cultural norms)⁸ that may prevent a neurodivergent person from fully participating in society²⁰. Critics of the social model consider that supports should also address the specific needs of neurodivergent people, for instance, by improving access to speech therapies for autistic children^4,9. There appears to be a consensus that, within the neurodiversity paradigm, interventions aim to improve the quality of life of neurodivergent individuals^16,21.

Despite growing interest in neurodiversity over the last few years, global prevalence data on neurodivergent conditions are limited and heterogeneous, partly due to differences in regional data availability and methodologies employed^23,24. For instance, the reported prevalence of the neurodivergent conditions ‘ADHD’ and ‘autism’ ranged between 3% to 14% and between 1% to 4%, respectively^25,26. Most estimates are scarce and tend to be underestimated, especially in low- and middle-income countries^24,27,28. The rising prevalence of chronic diseases and disabilities represents a public policy concern due to the increasing need for care which social and healthcare services are currently unable to fulfil^29,30.

Family carers, who are increasingly meeting these care needs^31,32, are estimated to provide 80% of the care needs in the European Union³³. Family carers are individuals who provide regular, unpaid personal help to a relative, friend or neighbour³⁴ due to additional care needs, disability, frailty, chronic diseases, or mental health conditions³⁵.

Caring can bring multiple benefits to family carers^36,37; however, it also poses challenges which may have a psychosocial and physical impact on the person providing care³⁸. Some challenges faced by family carers in general are long waiting lists to get their relative diagnosed^39,40, accessing information about their relative’s condition and strategies to support them^10,18, navigating complex healthcare systems^10,40,41, and the financial burden they incur due to additional education and healthcare costs^40,42, as well as the income loss for reallocating working hours to caregiving³⁹.

Family carers of neurodivergent people, in particular, may deal with the lack of understanding of their relative’s condition and behaviour^39,43 from health and social care professionals¹⁰, family and friends^39,44,45, and with the stigma that might result from unawareness of their situation^41,46. These circumstances can limit family carers’ interactions within their communities³⁹, which may contribute to feelings of isolation⁴⁷, loneliness⁴¹, and depression⁴³. This, in addition to increased levels of worry⁴⁸, stress^39,40,49,50, and anxiety⁵¹ may eventually have an impact on family carers’ health and well-being^38,44.

Rationale for a scoping review

Neurodiversity research has predominantly addressed the needs of neurodivergent individuals (e.g. improving their quality of life)^6,8,52,53 or supported family carers to address the needs of neurodivergent people^54,55. Yet, few neurodiversity studies appear to have explored the experiences of family carers of neurodivergent individuals^39,51,56. D'Arcy et al.³⁹ addressed the well-being and support needs of family carers of neurodiverse children in Australia; this study primarily focused on the challenges and impact of caregiving on family carers, while their support needs were briefly addressed. These authors acknowledged that their small sample size might not allow generalisations and emphasised the need for qualitative studies to gain a deeper understanding of the experiences of family carers of neurodivergent children.

A broader body of evidence regarding family carers of neurodivergent children was found within the medical research (i.e. studies using the term ‘neurodevelopmental disorders’)⁵⁷. Studies were more likely to adopt quantitative methods, and there was a disconnection from the social approach to neurodiversity; for instance, only a few reviews addressing family carers included terms related to the social and medical approach to neurodiversity^58,59. Livingstone et al.¹¹ observed that the work of neurodivergent academics has been somehow ‘relegated’ in the academic arena for being considered less objective or rigorous, although their insights could greatly enrich neurodiversity research.

Several neurodiversity studies have been conducted with adult populations, and fewer with young populations²², even though the neurodiversity movement in its origins aimed to support parents of autistic children^4,14,15. While more research seems to have been undertaken with autism, intellectual disability, and ADHD^11,6, McLennan et al.⁴ noted that future research should address the experiences of other neurodivergent groups. Filipe et al.²¹ advocated for a more inclusive approach to neurodiversity, which proposes that neurodivergent children with specific limitations (e.g. motor or intellectual) may face similar challenges; thus, their families may also share similarities in their experiences and support needs, especially considering that neurodivergent individuals often experience the co-occurrence of more than one condition^60,61.

A preliminary review of the literature suggests that the existing evidence on neurodiversity and family caregiving is somewhat fragmented, limited, and disconnected. Therefore, a scoping review was deemed the most suitable approach to map the breadth and depth of all relevant studies available, regardless of their study design or approach to neurodiversity. Given that various study designs will be included, a critical appraisal of the studies’ quality will not be performed⁶². This review will address broad issues rather than focusing on specific research questions, and a narrative synthesis will be undertaken to summarise research findings and identify research gaps.

Aim of scoping review

The overall aim of this scoping review is to collate and synthesise the available literature on family carers of neurodivergent children and young people. Drawing on both the medical and social approaches to neurodiversity research, this review aims to map the use of the term ‘neurodiversity’ in primary research and non-empirical evidence on family carers of neurodivergent children and young people. This review will identify the neurodivergent conditions studied under the umbrella term ‘neurodiversity’ to assess whether there might be commonalities in the experiences of family carers which could inform how to best support them “in a neurodiversity-affirming way”²².

Stage 1: Identifying the research question

Following a preliminary review of the literature, undertaken as part of the first stage of this scoping review, the following overarching research question was identified:

What are the experiences of family carers of neurodivergent children and young people?

The following research objectives will lead the scoping review:

The research question and inclusion criteria for this scoping review will be guided by the Population, Concept, and Context (PCC) framework. In the current review, this refers to:

Stage 2: Identifying relevant studies

Following guidance from Arksey and O'Malley⁶² and Levac et al.⁶³, the second stage of this scoping review will involve developing a comprehensive search strategy to identify relevant studies focusing on the experiences of family carers of neurodivergent children and young people.

A comprehensive literature search will be conducted using identified keywords, index terms, synonyms, and related terms across the included databases to ensure thorough identification of relevant studies. Boolean operators (AND, OR) and truncation (*) will be employed in the search strategy. The research librarian was consulted in refining the search strategy. The search strategy will be piloted to ensure it includes relevant terms for the research question, as well as specific Medical Subject Headings (MeSH) terms or keywords where applicable. Table 1 presents the proposed search terms used to develop the search strategy. The full search strategy of one database will be included as an appendix in the scoping review.

The references searched will include empirical studies of any design (e.g. quantitative, qualitative, or mixed-methods) and grey literature. Firstly, electronic scholarly databases will be searched to identify relevant peer-reviewed published studies. The databases to be searched will include PsycINFO, Cumulative Index of Nursing and Allied Health Literature (CINAHL), Medline, Web of Science, Academic Search Complete, and Applied Social Sciences Index & Abstracts (ASSIA). These databases were selected to ensure representativeness across a diverse range of disciplines (e.g., psychology, nursing, biomedical, social services, healthcare, and social sciences). Reference lists of identified articles will also be hand-searched to locate additional pertinent literature which was not identified through the databases' searches.

Unpublished and grey literature will be explored in repositories and websites on neurodiversity and family caregiving such as Google, Grey Literature Reports, Family Carers Ireland (FCI), Care Alliance Ireland, AsIAm, Carers UK, International Alliance of Carer Organisations (IACO), Eurocarers, European Commission (EC), World Health Organisation (WHO), among other sources. Further grey literature will be sought through team members’ networks, experts’ advice and relevant organisations. This approach will ensure a robust mapping and description of available evidence on the topic.

Stage 3: Selecting and screening studies

This third stage involves a series of steps to screen relevant studies for this scoping review systematically. A first step is to develop explicit inclusion and exclusion criteria to shortlist studies that address the research question and to ensure that all reviewers apply the same rules during the screening process⁶². Table 2 displays the initial inclusion and exclusion criteria for this scoping review. These criteria include carer group, care recipient group, focus of study, type of study, context, languages, and publication date.

The terms ‘neurodiversity’, ‘neurodevelopmental’ and related terms will be included in the search strategy to capture all available evidence about the experiences of family carers of neurodivergent children and young people, especially given that it is only relatively recently that neurodiversity gained attention from the academic community¹¹. Children and young people will refer to individuals aged 24 years or younger, according to the United Nations (UN) definition of youth⁶⁶. Any study design will be included to capture all available empirical evidence about the experiences of family carers of neurodivergent children and young people. Authors will be contacted when full-texts are not available. Conference proceedings will be included only if they are published. Searches will be limited to English, which is the language understood by the reviewers. The search period will span from 1990 to 2025, encompassing peer-reviewed and grey literature produced since the inception of the neurodiversity movement.

After the searches have been undertaken in each database, the retrieved articles will be imported into EndNote 21, a reference management software, and any duplicates will be removed in this programme. The deduplicated citations will be imported into Covidence, a systematic review software tool that will be used to screen the collected citations. Once all references are imported into Covidence, any remaining duplicates will be identified and removed. Before commencing the title and abstract screening phase, a pilot screening will be conducted on a small sample of studies to ensure consistency in the application of inclusion and exclusion criteria among team members. All retrieved studies will be screened by title and abstract, and references that do not meet the inclusion criteria will be removed. Each reference will be reviewed independently by two team members. In the event of disagreement, the inclusion of the reference will be discussed with a third reviewer until a consensus is achieved.

Articles identified during the title and abstract screening stage will subsequently be subject to full-text screening to determine whether they will be included in the review. Their full texts will be read and independently evaluated for inclusion and exclusion criteria by two team members. If there are disagreements on whether an article should be included, a discussion with a third reviewer will be held until consensus is achieved. A PRISMA flow diagram will be used to illustrate the screening process visually. This diagram will display the number of sources retrieved, reviewed, and included in the review, as well as the number of articles excluded and the reasons for exclusion at each stage of the screening process.

Stage 4: Charting/extracting the data

Following the advice from Arksey and O'Malley⁶², this fourth stage involves a technique called “charting”, which is employed to synthesise and interpret qualitative data. This involves “sifting, charting, and sorting material according to key issues and themes”⁶² from the studies included in the scoping review. Therefore, a data charting/extracting table will be developed in Excel, a spreadsheet program, to record information relevant to this scoping review’s research question and aims. This data charting table will be agreed upon and piloted by team members. The data may include:

Stage 5: Collating, summarising, and reporting the results

As mentioned by Arksey and O'Malley⁶², a scoping review does not intend to aggregate findings from different studies or assess the quality of the evidence; instead, it provides a narrative description of all the evidence reviewed. Therefore, the data charted will be collated, synthesised and thematically analysed to answer the scoping review’s research questions. Summary tables will be used to present the characteristics of the studies in a concise format, report the main areas of interest and identify gaps in the studies. The findings will also be presented in a narrative form. A second author will review and validate the findings. Any potential bias, limitations and decisions made to analyse the data will also be reported. This scoping review will inform the subsequent stages of a research project and whether a systematic review is warranted.