Introduction

This neurodevelopmental disorder Developmental Coordination Disorder (DCD) affects between 5% to 6% of the paediatric population [1] and is characterised by impaired motor proficiency, which interferes with the performance of activities of daily life, academic/school-based activities, leisure and play [1]. For a diagnosis of DCD, these motor proficiency difficulties cannot be explained by other neurological conditions that affect movement [1]. DCD is associated with an array of physical health problems [2], psychosocial and mental health problems [3, 4], and learning difficulties such as reading, social skills and inattention [5]. It is well established that DCD has the potential to impact on individuals involvement and participation in daily life activities and these challenges persist beyond childhood [6].

Best practice recommendations advocate that intervention planning for children with DCD should commence with an analysis of the individual’s strengths and weaknesses in their environmental context, so that an activity or participation-oriented approach can implemented [6]. However, there is a paucity of robust empirical evidence to guide the implementation of activity or participation-oriented practice with the current available evidence hampered by a wide range of methodological issues including limited controlled trials, poor outcome measurement, and intervention design [7, 8]. Therefore, there is a pressing need to develop interventions for children and adolescents with DCD [7, 8]. It is vital that future intervention development for children and young people with DCD is informed by stakeholder perspectives in line with recommendations for complex intervention development [9].

Children with disabilities have the right to be heard on issues that affect them [10], yet research on children’s experiences has tended to ignore the views of children as active agents and ‘key informants’ in matters pertaining to their health and wellbeing [11, 12]. Similarly, there has been a lack of attention to the perspectives of children with DCD in research, service developments and policy to date. For example the recently published ’International clinical practice recommendations on the definition, diagnosis, assessment, intervention, and psychosocial aspects of developmental coordination disorder’ [6], included stakeholder representation from a parent organisation for individuals with learning disorders, but no contribution from children or young people with DCD.

Multiple researchers have emphasised the value of research on children’s experiences and perspectives where children are acknowledged as authorities on their own lives [13–15]. Historically there has been a greater focus on eliciting the perspective of parents of children with DCD rather than the perspectives of children and adolescents with DCD. A number of studies have explored parents perspectives of their child receiving a diagnosis of DCD [16], parenting a child with DCD [17–20] and parental perspectives on their child with DCD’s reduced participation patterns across activities of daily living (ADL) [21, 22], out of school activities [23], leisure-time activity participation [24], and social participation [25]. It is important to note that the perspectives of parents, educators and allied health professionals can differ from those of children with a developmental disability [26–28].

Qualitative research can illuminate the meaning of everyday experiences [29] and reveal the perspectives, views and experiences of children and young people [30]. The past fifteen years has seen a growing number of qualitative studies focusing on children and adolescent’s experiences of living with DCD [31–34], including studies on; the importance of identity and empowerment to teenagers [31], children’s perceptions of the impact of DCD on activities of daily living [32], their perceptions of participation across home and community environments [33] and quality of life [34].

Although several studies examining various aspects of the experiences of children with DCD exist, as far as we are aware, no qualitative evidence synthesis has examined the totality of research on this topic. Meta-ethnographic synthesis is a popularly employed approach to qualitative evidence synthesis because it is allows researchers to bring together multiple qualitative studies, compare accounts and develop an interpretative synthesis, which generates new conceptual understandings [35–37]. In health service research, the meta-ethnographic approach has been widely employed because of its capacity to generate new understandings on how people experience their health and well-being [37].

This study aims to synthesise the findings of discrete qualitative studies reporting the lived experiences, views and preferences of children and young people with DCD using a meta-ethnographic approach. Synthesis of the available qualitative research with children and young people with DCD will inform future research by mapping research conducted to date and has the potential to inform intervention and service delivery through generating new conceptual understandings of the experiences of this group.

Methods

This qualitative evidence synthesis used a meta-ethnographic approach, following the seven-stage process described by [36], and the eMERGe guidance [35]. A detailed study protocol has been published previously [38] and this meta-ethnography is registered on PROSPERO, registration number CRD42019129178.

Screening

Once duplicates were removed, the first author (ÁOD) screened papers by title and abstract against the pre-designed inclusion/exclusion criteria (S3 File). KR screened 10% of papers by title and abstract to check for consistency. Papers included for full-text review were read and screened by ÁOD and KR. Each reviewer independently considered the paper’s relevance to this qualitative synthesis, any differences of opinion were resolved via discussion. The entire screening process is presented via a PRISMA flowchart in (Fig 1).

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Fig 1. PRISMA flowchart.

https://doi.org/10.1371/journal.pone.0245738.g001

Results

Study characteristics

The characteristics of the fifteen included papers are presented in Table 1. Six of the studies were conducted in the United Kingdom [31, 45, 49–52], five in Canada [33, 34, 44, 53, 54], one in Austria/ Italy [55], one in Brazil [56], one in Belgium [57], and one in New Zealand [58]. In total, data from one hundred and nine participants were included in the qualitative synthesis. Nine papers had participants with a mean age ranging from 6.9 years to 11 years [33, 34, 45, 49, 54–58]. Five papers had participants with a mean age range of 13 years to 14.9 years [31, 50–53] and one study included young adults aged 19–25 years who reflected upon their experiences as an adolescent [44]. Two papers included data from one sample of young people interviewed over a two-year period [51, 52].

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Table 1. Characteristics of included studies.

https://doi.org/10.1371/journal.pone.0245738.t001

Quality appraisal

Appraisal of the included papers highlighted a large variation in methodological quality across the studies as seen in Table 2. Ten studies presented the philosophical perspective on which the study was based and a methodological approach which was congruent with the perspective [31, 34, 44, 51, 52, 54–58]. However, the philosophical or theoretical perspective was not clearly represented in the remaining five papers [33, 45, 49, 50, 53]. The majority of studies described an appropriate qualitative methodology for addressing the research question or objectives. Two areas rarely addressed were the researcher’s cultural and theoretical orientation, or the influence of the researcher on the research and vice versa. The reporting of methodological quality was deemed unclear across many items in one study [45]. However, we chose to include the paper, as we believed that the representation of participants and their experiences was clear. Across all studies, participants were clearly represented from the data presented in the findings. One study was included even though it did not include a statement about ethical approval; however, the authors did describe the process for gaining child assent and parental consent [55]. No studies were excluded based on quality.

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Table 2. JBI appraisal checklist for qualitative research (rated as yes, no, unclear).

https://doi.org/10.1371/journal.pone.0245738.t002

Line of argument synthesis

The results of the 15 papers were reciprocally translatable and led the development of three interpretative themes. Higher order interpretation of these themes identified that the experiences of children and young people with DCD can be understood as the experience of psycho-emotional disablism, a concept developed within the field of disability studies. Carol Thomas [59] conceptualises psycho-emotional disablism as occurring when individuals with impairments are ‘hurt by the reactions and behaviours of those around them, made to feel worthless, of lesser value, unattractive, hopeless, stressed or insecure’.

Across the included studies, children with DCD described struggling to meet their parents, teachers, peers and broader society expectations around performance of activities. The mismatch between their abilities and performance norms in many cases led to a cascade of negative consequences including self-criticism, negative self-appraisal, bullying, victimisation, marginalisation and exclusion by peers and siblings. Across the included studies numerous examples of psycho-emotional disablism arising from relationships with other people, were reported, for example critical comments such as teasing or actions such as exclusion. Another manifestation of psycho-emotional disablism, internalised oppression, was also evident in self-criticism and negative self-perceptions. Further underscoring the significance of the social and attitudinal environment on the experience of children with DCD, those who encountered positive and encouraging social and attitudinal environments across a range of environmental contexts navigated situations with greater ease and detailed experiences that are more positive. In response to disablism, children and young people developed numerous strategies and drew on available supports to facilitate their involvement in activities and enhance social inclusion across life situations. Children with DCD revealed that they accessed support to diminish the mismatch between their performance and the expectations of others, including seeking and accepting assistance, focusing on their strengths and talents, accepting, and embracing their difference, adopting a “just do it” attitude, setting personal goals, self-exclusion from some social activities, using humour or sarcasm, and viewing performance and context expectations as a social construct.

Discussion

This synthesis reveals important insights into the lived experiences of children and young people with DCD. Three interrelated themes were identified and labelled, as ‘It is harder than it should be’: Navigating daily, Fitting in, ‘So What? I drop things’. Across the studies in this review, participants experienced a wide array of challenges learning and performing new skills and activities, participating in school, and experienced social exclusion from activities such as play and leisure. This aligns with much of the quantitative research to date on DCD in childhood, which has focused upon the predictors of patterns of participation [60]. However, this qualitative review focused upon the lived experience of children with DCD and exposes the array of self-developed strategies and successfully accessed supports used by participants to deal with these challenges and navigate everyday life situations. Findings of this synthesis resonate with the social model of disability and the ICF.

Considering the synthesis findings in the context of the social model of disability and the ICF suggests that the social, attitudinal, and physical environment significantly influences children and young people’s everyday experiences. The synthesis revealed that children with DCD experience bullying and various types of victimisation [61], including, verbal (name-calling, teasing), social (social exclusion, marginalisation), and physical (physical assault) in the school setting. Children with disabilities are at increased risk of bullying in schools in comparison to their typically developing peers [62, 63]. The high prevalence of bullying experienced by children with disabilities is very concerning as strong evidence supports that bullying victimisation in childhood is associated with a wide range of adverse mental health and psychosocial outcomes [64]. Bullying of children because they live with socially devalued identities or characteristics, such as disability, is described as stigma-based bullying and requires bullying interventions specifically focusing on stigma-related factors [65]. Within school environments, greater support to help teachers and peers reconceptualise disability as a social issue may help to challenge notions of marginalisation and discrimination based on socially valued characteristics such as motor proficiency.

The World Health Organisation [66] and childhood disability researchers [67] recognise the importance of contextual factors such as the environment on the health and well-being of individuals. Research has shown that environmental factors (physical, social, and attitudinal) mediate the relationship between child factors and participation outcomes for children and youth with disabilities [68, 69]. For all children, the family represents the most crucial environment, and parents are considered a critical contextual factor in a child’s life [66, 67]. The synthesis findings highlighted the profound influence of the social and attitudinal environmental on children’s experiences within the family context. In line with recent findings regarding the association between family factors and participation of children with a disability [70]; this review found that where parents and siblings understood the child’s strengths and capabilities, participants navigated daily life with greater ease, experienced greater involvement in activities within the home and community environment, and described positive self-perceptions. Conversely, where the home environment was not supportive of the child’s performance and involvement, negative emotions, conflict with parents and siblings, and negative self-perceptions were typical. Our findings support the importance of health service interventions targeted at the family environment. Family-centred approaches that enhance the family’s capacity to understand their child and build competency to resolve challenges may be most beneficial to the child and family [67].

In addition, the school environment was a contextual factor, which significantly influenced opportunities for social inclusion and involvement in activities. Our findings demonstrate the significant influence of teachers and peers on participation and the subsequent positive or negative self-perceptions and school-based experiences of children and young people with DCD. Evidence suggests that teacher awareness and understanding of DCD is poor [71]. The findings of this synthesis highlighted that school sports and physical education were perceived as particularly challenging experiences, which led to incidents of ridicule, self-exclusion, marginalisation, and reluctance to participate due to poor teacher understanding and awareness. As noted by [72], teachers’ knowledge, understanding and confidence about teaching students with a disability is crucial to facilitating inclusive and effective Physical Education (PE). In line with previous research, our results suggest that children and young people’s functional abilities with handwriting, motor, organisation and planning skills influenced their capacity to complete specific activities in school [73]. However, similar to recent findings, involvement in school-based activities was also heavily influenced by the interaction between personal factors such as motivation, interest, preference and environment factors such as the adults, peers, the school structures and routines as well as access to assistive technology and objects [73]. These findings highlight the need for interventions that target teacher collaboration between health professional and educator, in order to build teachers’ knowledge, awareness and capacity to support children and youths with DCD in school. As seen in Canada, Partnering for Change presents an innovate intervention that promotes a model of health and education partnership, which shifts the focus from individualised support to targeting whole school environmental interventions [74]. Society and systems influence the opportunities for inclusion in many aspects of social life such as education, health and community choices [75]. Therefore, further development and evaluation of targeted social/environmental interventions is warranted as they may provide a means to enhance inclusion and reduce social, physical, and attitudinal barriers faced by children and young people with DCD.

Rosenbaum and Gorter [67] provide a novel representation of the ICF concepts, which they define as the F-words (fitness, function, friends, family, fun and future). The results of this study converge with Rosenbaum and Gorter’s [67] representation of the ICF concepts highlighting the importance of friendships, peer social acceptance and fun to support participation in everyday situations. Children and young people with DCD valued friendships and the opportunity for inclusion in social and physical activities with peers across environments. These findings align with previous evidence, which suggest children with disabilities perceive activity experiences as positive, when completed with peers and friends and they are fun [57, 76, 77]. Unsurprisingly, friendships for children with disabilities are associated with greater psychosocial wellbeing [78], social inclusion [79], and healthier identify formation in adolescence [80]. Previous research has revealed that children with a disability face greater risk of social exclusion [81]. However, social inclusion is possible when personal and social resources for the children with a disability are present, including, the ability to make friends, participate in community activities, engage in leisure and play, and have access to quality inclusive practices in the classroom [81]. Our findings emphasise the importance of attending to the social environment so that children and young people with DCD have the opportunity to be involved in meaningful activities that promote social interaction and friendship over time.

In our review, some participants drew on personal resources or accessed supports that facilitated inclusion. Personal resources included a “just do it” attitude, recognising their strengths, accepting and embracing their difference, using humour or sarcasm, and viewing performance and context expectations as a social construct. Our findings corroborate the significance of personal resources such as preferences, interests, and motivations to influence and sustain participation in leisure and school-based activities [73, 76, 82], while supports accessed included, friendships, and support from family and teachers. Positive relationships are considered an important protective factor to help children overcome the risk of social exclusion [81, 83]. The findings of review highlight how the interaction between the personal and environmental factors shaped the lived experiences of children and young people with DCD.

Our integration of findings clearly show that some children and young people experience difficulties because of the mismatch between performance expectations, and their relationships with others including family, teachers and friends. We developed a line of argument and present the experience of children and young people with DCD as reflective of adopting the social relational perspective of the social model of disability [59, 84]. This theoretical perspective provides a lens to contextualise how social and attitudinal restrictions of activity influence the experiences of children and young people with DCD. Children and young people’s perspectives reflect ‘psycho-emotional disablism’ [59]. The social behaviours of others, such as family members, teachers and peers had the potential to undermine their psycho-emotional wellbeing and impose social restrictions of activity. Accordingly, changes are required within the social structures, which create prejudice towards individuals with impairment not to the individual’s psychological adjustment [59], hence in keeping with the social model of disability, interventions that target sigma may be helpful approaches [65].

The review findings corroborate the need for greater attention to the child’s perspective in research and practice, recognising that there are potential discrepancies between children and parents’ perspectives. A recent survey of perceived support for children with DCD in Canadian schools surveyed parents of children with DCD rather than children themselves [85]. Izadi-Najafabadi and colleagues found that the attitudes and actions of teachers, staff, school-related policies and procedures were perceived as adequate by parents [85], whereas the findings of this review reflects that children and young people do not always perceives attitudes and actions of school staff as supportive. Accessing the lived experience of school for children with DCD is important, as children can articulate their experiences and richly inform others on the issues affecting their lives. Another important distinction between the perspective of children and parents is that young people with coordination difficulties, more so than their parents, prioritise friendship as means to promoting self-esteem [86]. The findings of this review corroborate this perspective, friendships were highly regarded by children and young people and influenced their self-perceptions. This review underscores the importance of health professionals exploring the child and young person’s perspectives and priorities for treatment, in conjunction with the parental perspectives in order to build family capacity to manage everyday life situations, line with best practice recommendations [6, 87]. The synthesis findings emphasise the need for professionals to design interventions, which target priority outcomes for friendship, social inclusion and greater understanding and awareness in the school setting, as these are important to children and young people with DCD. Professionals should also explore the child’s strength, and strategies used to navigate everyday life prior to recommending or commencing intervention.

Strengths and limitations

A major strength of this paper is that it is the first meta-ethnography to focus on the experience of children and young people with DCD. This synthesis addresses the unequal attention conferred to parents versus children’s perspectives. Research examining children’s perspective is essential if we are to improve the lives of children [88].

A strength of this paper is the robust conduct of the review; the authors adhered to the eMERGe and PRIMSA guidelines. However, a limitation of this review was the quality of included studies varied. Furthermore, the phenomenon of interest examined across the included studies was diverse, which resulted in a focus on certain aspects or dimensions of children’s and young people’s experience in some papers. For example, Hessell and colleagues’ examination of the experience of a gymnastics club for children with DCD [58], or Armitage and colleagues’ investigation of children’s perceptions and experiences of occupational therapy intervention [49].

Implications for practice

The findings of the synthesis point to the need for health professionals working with this population to consider and address the social and attitudinal environments in which children and young people participate and live, but especially the school environment. It is important that interventions, which support teacher knowledge, awareness and understanding of DCD, be developed. Such interventions are one element of promoting a positive social and attitudinal school environment for children with DCD. For example, increasing teacher awareness and recognising the challenges associated with fatigue, fear of injury or poor performance in front of peers during physical education classes may help to support involvement.

Health service provision should adopt a family-centred approach that enhances the capacity of the family to support engagement and performance in activities and everyday situations. Augmenting parents understanding of DCD may be helpful in addressing potential negative child self-perceptions arising from parental expectations. Finally, children and young people described numerous successful strategies to deal with challenging experiences, victimisation, and exclusion. Thus, greater attention is required by professionals to identify children’s capabilities and strategies, which may be leveraged to address challenging social interactions and navigate daily life activities.

Conclusion

This paper exemplifies the value of qualitative research to inform research, policy, and practice. Our synthesis of fifteen papers produced three interrelated themes; a) ‘It’s harder than it should be’: Navigating daily activities, b) Fitting in, and c) ‘So what? I drop things’: Strategies and supports to mitigate challenges. Children with DCD describe a mismatch between their abilities and performance norms for daily activities that could led to a cascade of negative consequences including negative self-appraisal, bullying and exclusion. In the face of these difficulties, children with DCD described friendships as a forum for fun, acceptance and protective against exclusion and bullying and they described a range of creative and successful strategies they enacted and supports they accessed.

Supporting information