University of Groningen

Moving matters for children with developmental coordination disorderBraaksma, Petra

DOI:10.33612/diss.111900151

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Citation for published version (APA):Braaksma, P. (2020). Moving matters for children with developmental coordination disorder: We12BFit!:improving fitness and motivation for activity. [Groningen]: Rijksuniversiteit Groningen.https://doi.org/10.33612/diss.111900151

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General introduction

General introduction

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Chapter 1

1. Developmental coordination disorder (DCD) Children with DCD experience problems in acquiring and executing coordinated motor skills.1 Formerly known as dyspraxia, the term DCD was introduced and included in the Diagnostic and Statistical Manual of Mental Disorders (DSM) in 1994. Despite this, to date many care professionals, teachers and parents are still unaware of this disorder.2 However, given the high prevalence and the substantial impact of DCD on several crucial areas of daily functioning throughout their development, children with DCD warrant our attention. 1.1 Prevalence and symptoms of DCD DCD is prevalent in approximately 5 to 8% of all school children3–6 and is found two to seven times more often in boys than in girls.7,8 An early sign of DCD is a delay in reaching motor milestones such as sitting and walking. Later on, the difficulties of children with DCD may manifest as clumsiness, and inaccurate and slow performance of motor skills. They may bump into objects often, spill their food, or have difficulties with dressing or catching balls.1 Some children struggle to learn to ride the bike and will not manage this before the age of seven. Others may have writing problems: their handwriting is messy, and writing tasks take a lot of effort and time to be finished. Children with DCD are able to acquire motor skills, but it will take more practice and their motor execution may still not be as fluent as in typically developing (TD) children once a skill is acquired. Contrary to delays in motor development, DCD is often persistent and tracks into adulthood.9 During adolescence and adulthood, people with DCD still have difficulty acquiring new skills such as learning to drive a car.9 Children with DCD are considered a heterogeneous group as some children mainly experience problems with gross motor skills whereas others mainly experience problems with fine motor skills. In addition, up to 60% experiences comorbidities such as, attention deficit hyperactivity disorder (ADHD), reading difficulties, specific language impairments,10 autism spectrum disorders (ASD)11 or hypermobility.12 1.2 Mechanism of poor motor performance Although our knowledge about DCD is growing, to date, no definite cause has been established for DCD. So far, research on this topic has resulted in an explanatory frame-work that considers the poor motor performance of children with DCD to be the result of three interacting components: individual, tasks and environment (Figure 1).13 At the individual level there are indications for atypical functioning of the brain such as poor per-ceptual motor coupling, internal modelling deficits, and difficulties with executive functions (e.g. working memory, planning and inhibition).14 Tasks that are particularly challenging for children with DCD among others involve dual cognitive motor tasks, and tasks that are complex and require high precision or high speed. Environmental factors that further com-plicate motor performance are reduced visual cues, noise, uneven surface and distractions.13

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General introduction

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Figure 1. Multi-component explanatory framework of motor skill development showing correlates of performance in children with DCD.13

Abbreviations: IMD = internal modeling deficit; EF = executive function; WMN = white mat-ter network; MNS = mirror neuron system.

Poorly developed motor abilities

(reduced automation)

IMD and poor action representation; Poor

perceptual-motor coupling; EF difficulties

Atypical WMNs andconnectome; Atypical brain

activation in perceptual-motor networks, MNS;

Genetic factors?

Dual tasking; Precise and/or speeded performance;

Movement complexity acrossstability, manipulation, and

locomotor tasks

Reduced visual cues; Unevensupport surface; Background

noise/distraction

Interaction of components

Atypical movement patternsPoor motor coordination and skill execution

Time

(no provision for extended practice)

INDIVIDUAL

ENVIRONMENTTASK

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Chapter 1

1.3 Diagnosis of DCD Likely as a result of the low awareness of DCD, it may take a long time before DCD is diagnosed. In the Netherlands the average time from the first meeting with a care profes-sional to diagnosis is 33.5 months.15 The diagnosis of DCD is described in the DSM. From 1994 to 2013 the DSM-IV was used and in 2013 the criteria were updated in the DSM-5 (Table 1). Importantly, the diagnosis of DCD excludes other conditions that might explain the motor skill difficulties. Furthermore, the problems experienced by the children should significantly and persistent-ly interfere with their daily and school activities. Table 1. Diagnostic criteria of DCD1 and the Dutch operationalisation of the diagnostic criteria.16 A. The acquisition and execution of coordinated motor skills is substantially below that

expected given the individual’s chronological age and opportunity for skill learning and use. Difficulties are manifested as clumsiness (e.g., dropping or bumping into objects) as well as slowness and inaccuracy of performance of motor skills (e.g., catching an object, using scissors or cutlery, handwriting, riding a bike, or participating in sports)

Dutch operationalisation: The total score on the Movement Assessment Battery for Children-2 (Movement ABC-2) is at or below the 16th percentile, or the score on one of the three components of the Movement ABC-2 is at or below the 5th percentile

B. The motor skills deficit in Criterion A significantly and persistently interferes with activities of daily living appropriate to chronological age (e.g., self-care and self-maintenance) and impacts academic/school productivity, prevocational and vocational activities, leisure, and play

Dutch operationalisation: The request for help should show that the condition continuously affects academic performance or activities of daily living. This should be assessed by a medical specialist who is trained and competent in this (paediatric rehabilitation physician, pediatrician, pediatric neurologist, pediatric psychiatrist). Referral to a rehabilitation center is not sufficient to meet this criterion. The Coördinatievragenlijst Voor Ouders (or DCD-Questionnaire) and the Groninger Motoriek Observatieschaal are used to attain additional information about functional problems encountered at home and at school

C. The onset of symptoms is in the early developmental period [No Dutch operationalisation available yet]

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In research on DCD it is not always possible to determine or to track down the diagnosis as it includes extensive testing of motor skills and an examination by a physician to exclude other conditions. Therefore, the terms “at risk”, “probable”, “moderate” and “severe” are generally used to indicate which criteria of DCD were covered (Table 2). Table 2. Terminology for describing DCD in research papers (cited from Smits-Engelsman et al., p.297).17 Overall For the description of a DCD group all DSM-5 criteria should be

described and also how they were or were not met (questionnaires and tests used with the cut-off scores applied)

Moderate DCD (m-DCD)

All DSM-5 criteria are described and met. Children score 1–1.64 standard deviations below the mean on a validated motor test (between 15th and 6th percentile)

Severe DCD (s-DCD)

All DSM-5 criteria are described and met. Children score at least 1.64 standard deviation below the mean on a validated motor test (at or below 6th percentile)

Probable DCD (p-DCD)

DSM-5 criteria are described, but one or more criteria may not have been evaluated. For example, there is no parent report on ADL, or there is no information available on criterion C or D. Children in p-DCD score at least one standard deviation below the mean on validated motor test (at or below 16th percentile). Moreover, if based on the child’s history, there has been insufficient exposure to skill learning, the child will also be classified as p-DCD.

Table 1 (Continued) D. The motor skills deficits are not better explained by intellectual disability

(intellectual developmental disorder) or visual impairment and are not attributable to a neurological condition affecting movement (e.g., cerebral palsy, muscular dystrophy, degenerative disorder)

Dutch operationalisation: The condition is not the cause of a medical condition according to the results of medical-neurological examination. DCD can only be diagnosed by a medical specialist who is trained and competent in this (peadiatric rehabilitation physician, peadiatrician, peadiatric neurologist, peadiatric psychiatrist). Prior to diagnosis the following must be examined: general physical condition (motor skills, neurology, vision), communication skills, IQ (only if in doubt about IQ an IQ test should be taken), behaviour (CBCL/TRF), social circumstances. An IQ score below 70 on an individually conducted, standardized intelligence test precludes the diagnosis of DCD. Normal IQ can be assumed when a child is in regular education, did not have to repeat a school year, and there are no other doubts about the intelligence level

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Chapter 1

1.4 Treatment Upon diagnosis children with DCD generally need treatment. In the Netherlands children with DCD usually receive individual physical therapy or occupational therapy. Current rec-ommendations for remediating motor skill are to apply Neuromotor Task Training (NTT) or the Cognitive Orientation to Daily Occupation (CO-OP) approach.13 These are both task-oriented approaches focusing on the performance of activities, such as learning to ride a bike in traffic, rather than on the underlying problem. In NTT, functional skills are trained by varying and gradually increasing the demand of task and environmental constraints. The type of instruction and feedback that are provided depends on the motor learning stage. In CO-OP, children are taught to use the goal-plan-do-check strategy. This cognitive strategy helps the child to monitor their performance, identify what went wrong and to plan for improving their motor performance.4 2. Impact of DCD The motor skill problems that children with DCD experience, influence many different areas of their live. It is for instance known that compared with TD children, children with DCD have lower self-efficacy in leisure and physical activities, more social problems, and more symptoms of depression and anxiety.18 The International Classification of Functioning, Disability and Health Youth and Children (ICF-CY) offers a framework to map this in a comprehensive way, by showing how a condition may reciprocally influence body functions and structures, activities, participation, characteristics of the child and characteristics of the environment (Figure 2).19 Over the past years, three very important areas of second-ary problems in children with DCD have gained attention in both research and practice: physical fitness (PF), physical activity (PA) and participation. PF, PA and participation corre-spond to the ICF-CY levels “body functions and structures”, “activities” and “participation” respectively.

Table 2 (Continued) After the opportunity has been given to learn the specific skill over a

short period of time, sufficient progress needs to be demonstrated otherwise, depending on the motor score the condition would be categorised as s-DCD or m-DCD

At risk for DCD All DSM-5 criteria are described and met, and children are under 5 year of age. If a later repeated motor test and evaluation confirms that all criteria are met, the diagnosis DCD will be given

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General introduction

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Figure 2. Model of the International Classification of Functioning, Disability and Health Youth and Children.19

2.1 Physical fitness It has become increasingly clear that many children with DCD experience problems with health-related PF including the components cardiorespiratory fitness (CRF) (i.e. endurance or aerobic capacity), muscle strength, flexibility and body composition.20,21 Compared with TD children, children with DCD score 7-22% lower on CRF tests20,21 and 15% lower on muscle strength tests.21 Children with DCD are more likely to be at either extreme of the flexibility spectrum than TD children.20 Although no differences in body composition were found in a sample of Dutch children,21 other studies indicate that children with DCD have worse body composition than TD peers.20

Health condition

Body functionsand structures

Physiological functions of body systems (including psychological functions) and anatomical parts of

the body

ActivitiesExecution of tasks or

actions by an individual

Personal factors Environmental factors

ParticipationInvolvement in a life

situation

Physical fitness Physical activities Participation in sports and games

DCD

Motivation to be activeFear of failure

Support of family and classmates

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Chapter 1

2.2 Physical activity PA is defined as “any bodily movement produced by skeletal muscles that results in energy expenditure” and is positively related to PF.22 Children with DCD have been reported to be less physically active than TD children.20 In a Dutch study, only 32% of the children with DCD met the recommendation to be physically active for at least one hour each day compared with 53% of TD children.23 Moreover, children with DCD tended to engage in activities that were less intensive than those of TD children.23 2.3 Participation Participation is defined as a person’s “involvement in a life situation” and represents “the societal perspective of functioning” (p.9).19 The most frequently reported limitations in activities and restrictions in participation in children with DCD are poor handwriting, diffi-culties playing ball games, getting dressed and participating in organised sports.24 Moreover, children with DCD have been reported to participate less in family social activities, house-hold activities, leisure activities in the community and PA at school than TD children.25 The participation problems seem to continue with increasing age as teenagers and young adults showed problems in all 12 life habits of the Human Development Model-Disability Crea-tion Process model: education, communication, interpersonal relationships, community life, recreation, fitness, employment, mobility, personal care, nutrition, housing and responsibili-ties.9

2.4 Negative spiral To date no underlying pathology was found in children with DCD that might explain their lower PF compared with TD children. Instead, it is hypothesised that children with DCD most likely find themselves in a negative spiral where poor motor skills lead to reduced participation in PA and consequently lower PF.26,27 The relation between poor motor skills and reduced participation in PA may be mediated by low enjoyment due to repeated ex-periences of failure and public embarrassment, and low self-efficacy regarding PA.20,28 Low levels of PA may lead to low levels of PF,29 which in turn may further impede participation in PA due to earlier onset of fatigue and increasing difficulty to keep up with peers. Lower participation in activities also restricts the amount of opportunities for practicing motor skills and may therefore further impede the development of motor skills in children with DCD. Without appropriate help, children with DCD may become trapped in this negative spiral with potentially detrimental consequences for their health and development. Low PA and PF, combined and independently, are important risk factors for cardiovascular disease.29 In addition, it is known that participation in (physical) activities is crucial for children’s de-velopment of social skills and academic skills, sense of competence and life satisfaction.19,30

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3. Intervening on PF and PA in children with DCD The problems that children with DCD have with their PF were also noticed by the pae-diatric physical therapists from the Center for Rehabilitation, University Medical Center Groningen. Their search for an evidence-based approach to improve the PF of children with DCD formed the basis for this research project. This project started with a prelim-inary study on the PF and PA of Dutch children with DCD, performed by three Dutch rehabilitation centers (Center for Rehabilitation, University Medical Center Groningen, Rehabilitation Center Revalidatie Friesland and Center for Rehabilitation Het Roessin-gh).21,23,31 This study confirmed the observations of the peadiatric physical therapists and was largely in line with international findings.20 Upon completion of the preliminary investigation, the literature lacked a comprehen-sive evidence-based intervention to break the negative spiral of deconditioning in children with DCD. At the time, only a few studies used interventions targeting either a single component of PF or PA in children with DCD.32–36 These studies showed that children with DCD were able to improve their CRF and muscle strength,32–34,36 but not their levels of PA.35 Therefore, we set out to systematically develop an evidence-based intervention to improve three important components of PF and motivation for PA in children with DCD. 3.1 Method of treatment development: Treatment theory For the development of this intervention we relied on treatment theory as defined by Whyte et al.37 Treatment theory consists of three consecutive steps. In the first step one selects and defines a target which represents the “aspect of the recipient’s functioning, or personal factor, that is predicted to be directly changed by the treatment’s mechanism of action” (p.S25).37 Targets should be distinguished from “aims” which can only be indirectly changed. This step will help developers to explicate and potentially reconsider their targets and aims. For instance, lifelong optimal PF is an aim rather than a target, and can be broken down in a number of targets that can be changed directly and that may lead to the aim eventually. In the second step one defines the mechanism of action, the “process by which the treatment’s essential ingredients induce change in the target of treatment” (p.S32.e1).37 Defining the mechanism of action provides important information for the third step which encompasses the selection of treatment ingredients which are the actions selected or delivered by the clinician: which activities are needed, how to trigger the processes defined in the mechanism of action? In defining the treatment ingredients it is important to distin-guish between ingredients that are essential and other active ingredients that moderate the effects on the target. 3.2 Treatment: We12BFit! Using the steps of treatment theory, we systematically developed We12BFit! for 7 to 12 year old children with DCD. We12BFit!, in Dutch referred to as RenJeFit!, consists of two

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Chapter 1

intertwined parts: We12BFit!-PF and We12BFit!-Lifestyle PA. We12BFit! focuses on four targets. CRF, muscle strength, anaerobic power are targeted in We12BFit!-PF and moti-vation for PA is targeted in We12BFit!-Lifestyle PA. We12BFit! spans 22 weeks and com-bines physical training with behavioural intervention. During We12BFit!-PF, children with DCD will exercise in small groups, twice a week for 10 weeks. In week 6 of We12BFit!-PF, We12BFit!-Lifestyle PA will be introduced, starting with a parent meeting, the provision of pedometers and an information booklet for parents, followed by individual coaching of the child and his parents. 4. Outline of this thesis This thesis will address the development and evaluation of We12BFit!. In order to inform the initial development of We12BFit! a systematic review was performed. This systematic review in chapter 2, provides an overview of the activities, frequency, duration and intensity of PA interventions and their effects on CRF in healthy, TD children. Chapter 3 and 4, pro-vide an account of the development of respectively We12BFit!-PF and We12BFit!-Lifestyle PA. In these chapters it is described how targets, mechanism of action and ingredients are selected, informed by literature as well as by expertise from rehabilitation professionals. Relying on mixed-methods, chapter 5 provides an account of the effectiveness and feasibil-ity of We12BFit!. Triggered by the effects of We12BFit! on participation and to potentially inform the further development of We12BFit!, we performed a second systematic review in chapter 6 summarising the participation involvement and attendance of children with DCD. Finally, in the general discussion in chapter 7, the overall findings of this thesis and their implications will be discussed.

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