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«Examination of factors related to motor performance and physical activity in schoolaged children with and without Down syndrome by Chad William ...»

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Examination of factors related to motor performance and physical activity in schoolaged children with and without Down syndrome

by

Chad William Tiernan

A dissertation submitted in partial fulfillment

of the requirements for the degree of

Doctor of Philosophy

(Kinesiology)

in The University of Michigan

Doctoral Committee:

Professor Dale A. Ulrich, Co-Chair

Associate Professor Rosa M. Angulo-Barroso, Co-Chair

Associate Professor Joseph E. Hornyak

Associate Professor Riann M. Palmieri-Smith © Chad William Tiernan 2012 This dissertation is dedicated to Alyssa, Max, and Quinn. Without your love, support, and inspiration, this would not have been possible.

ii

ACKNOWLEDGEMENTS

First and foremost, I would like to thank my family. Alyssa was always by my side offering encouragement. She had to make a lot of sacrifices in order for me to complete this process and I’m eternally grateful to her. Max and Quinn were patient and understanding when I wasn’t always available to spend time with them. My parents and siblings have been very supportive from the beginning as well. I would also like to acknowledge my committee members. They were all instrumental to this dissertation, especially in helping with my study design. In particular, I want to thank my co-chairs, Dale and Rosa. I can’t thank them enough for all of their guidance along the way.

My graduate studies at the University of Michigan were supported by a Leadership Training Grant from the United States Office of Special Education Programs.

I would also like to acknowledge the Special Olympics Healthy Athletes Health Professions Student Grant Program, which funded my dissertation. Finally, I need to thank the families who participated in my studies. Their stories and generosity made all of this hard work worthwhile. I hope my research touches their lives in some way and benefits the greater Down syndrome community.

iii

TABLE OF CONTENTS

Dedication ii Acknowledgements iii List of Figures viii List of Tables ix

Abstract

x Chapter I. Introduction Overview 1

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III. Exploring relationships between motor skills, physical activity, and physical characteristics in school-aged children with and without Down syndrome

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Figure 2.1.

Branched model for determining physical activity energy 58 expenditure in children. Decisions in the flow chart are based on weighting the activity and heart rate data in varying proportions. (Adapted from CamNtech)

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Figure 3.1.

Branched model for determining generalized physical activity 101 in children. Decisions in the flow chart are based on weighting the activity and heart rate data in varying proportions. (Adapted from CamNtech)

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Table 3.2.

Correlations: motor skills vs. physical characteristics 103 Table 3.3. Correlations: motor skills vs. physical activity and participation 104

–  –  –

Examination of factors related to motor performance and physical activity in schoolaged children with and without Down syndrome

–  –  –

Co-chairs: Dale A. Ulrich and Rosa M. Angulo-Barroso Due to widespread genetic constraints, children with Down syndrome (DS) are believed to experience deficits in several developmental domains. However, much of the research in this area is dated or inconclusive. In addition, the inter-relatedness between developmental domains is relatively unexplored in children with DS. Therefore, the primary objectives of the current studies were to: 1) describe the physical, motor, and activity profiles of school-aged children with and without DS; 2) explore physical- and activity-based correlates to gross motor skills; and 3) investigate socio-emotional characteristics of children with and without DS and determine if they were related to gross motor skills, physical activity, and participation. To address these questions, forty children with DS (N=20, mean age = 7.9 years) and with typical development (TD) (N=20, mean age = 7.9 years) were assessed on their physical, motor, activity, and socio-emotional characteristics. Results indicated that children with DS had increased range of motion, decreased leg strength, poorer gross motor skills, and less diversity in their recreational activities compared to the TD group. On average, the two groups were not

–  –  –

hyperactivity in a subsample of the children with DS. Findings also revealed that physical characteristics (e.g. leg strength), generalized activity, and participation all significantly predicted gross motor skill performance in the DS group whereas only physical characteristics significantly contributed to models of gross motor skills for the children with TD. With respect to socio-emotional development, children with DS had more social and behavioral problems than their peers with TD. Increased gross motor skill ability and participation were associated with higher social competence in both groups. For children with DS, generalized physical activity seemed to be associated with behavioral problems curvilinearly such that there may be an optimal range of activity to promote socio-emotional well-being in this population. Collectively, results from these studies demonstrate the high level of inter-relatedness between developmental domains in school-aged children with DS. Future research should consider motor skill and physical activity interventions to promote overall development and well-being in this population.





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Overview The main objectives of this dissertation are to provide comprehensive descriptions of the motor, physical, and socio-emotional characteristics of children with and without Down syndrome (DS) and to explore relationships between these domains.

For the purpose of this dissertation, the “motor” domain refers to gross motor skills and physical activity whereas the “physical” domain consists of body composition, range of motion, and leg strength. A variety of social, emotional, and behavioral factors, such as social awareness and aggression, comprise the “socio-emotional” domain. Study 1 is largely descriptive, where the motor and physical characteristics of the two groups (DS vs. non-DS) are presented and compared. Study 2 investigates relationships between motor and physical abilities in addition to associations within the motor domain (i.e.

motor skills vs. physical activity). Finally, Study 3 describes the socio-emotional characteristics of children with and without Down syndrome and examines whether group differences in these variables exist. Study 3 also explores relationships between the motor and socio-emotional domains. The ensuing pages provide a review of the literature pertinent to these three studies.

Review of Literature A. Down syndrome: Multi-domain deficits Down syndrome (DS), first described by Dr. John Langdon Down in 1866, is both the oldest and most common known cause of intellectual disability. Recent estimates place the rate of incidence of DS at 1 in every 733 to 1 in every 850 live births in the United States (Canfield, Ramadhani, Yuskiv, & Davidoff, 2006; Shin, Besser, Kucik, Lu, Siffel, &Correa, 2009). DS is caused by the presence of all or part of an extra 21 st chromosome. Due to this chromosomal abnormality, every cell in the body is affected in most cases. Such widespread genetic abnormality may explain why individuals with DS demonstrate developmental problems within multiple domains. Though DS is most commonly associated with intellectual disability (ID), equally important deficiencies are experienced within the physical, motor, and socio-emotional domains. Our current understanding of these three domains (physical, motor, socio-emotional) with respect to DS will be described in the following sections.

1) Physical Domain For the purpose of this dissertation, the “physical domain” includes body composition, range of motion, and muscle strength. From a dynamic systems perspective (DSP), these factors may be viewed as potential control parameters for motor behavior. DSP proposes that several subsystems (i.e. musculo-skeletal, sensory, cardiovascular, neural, etc.) self-organize to influence behavior within a given task (Kamm, Thelen, & Jensen, 1990). Control parameters are variables that, when scaled up or down beyond a critical point, cause a shift in behavior. For example, when water reaches a certain temperature it begins to boil. Therefore, temperature can be considered a control parameter in this context because once it reaches a critical level there is a qualitative change in the molecular action of the water. Accordingly, it is plausible to suggest that body composition, muscle strength, or range of motion might serve as control parameters for motor performance in school-aged children with and without DS. In fact, infant studies have implicated several of these factors in early locomotor behavior (Ulrich, Ulrich, Angulo-Kinzler, & Yoon, 2001; Thelen, Fisher, RidleyJohnson, & Griffin, 1982). Moreover, it appears that individuals with DS experience a variety of physical challenges not only in infancy but throughout the lifespan.

Of the physical factors impacted in individuals with DS, perhaps the most visibly obvious are those related to body composition. There is substantial research to indicate a high prevalence of obesity in individuals with DS. In fact, several experts believe that DS obesity rates should be considered a major public health concern (Rubin, Rimmer, Chicoine, Braddock, & McGuire, 1998). Several studies illustrate the heightened obesity levels in DS. For example, Prasher (1995) examined two-hundred individuals with DS in the United Kingdom. Findings revealed that 31% of males and 22% of females were overweight (BMI = 25-29) while 48% of males and 47% of females were categorized as obese (BMI = 30-34). In a similar study in the U.K, Bell & Bhate (1992) observed 58% of males and 83% of females with DS to be overweight or obese. These percentages were much higher than those observed in a population without intellectual disability, who demonstrated rates of 40% and 32% for males and females, respectively. Rubin and colleagues (1998) reaffirmed the previous findings within the United States by demonstrating that individuals with DS had overweight rates 12-20% higher than the general population.

Studies on obesity in children with DS are not as conclusive as the adult literature. For example, Sharav and Bowman (1992) examined thirty pairs of siblings between the ages of 2 and 14 years, each pair consisting of one child with DS. The findings showed no differences in BMI between the children with and without DS. In contrast, Whitt-Glover and colleagues found that children with DS had higher BMI values compared to their unaffected siblings (Whitt-Glover, O’Neill, and Stettler, 2006).

In yet another study, fat-free mass values measured using three different methods (bioelectrical impedance, skinfold thickness, and deuterium dilution) were not found to be different between prepubescent children with and without DS (Luke, Sutton, Schoeller, & Roizen, 1996). Therefore, inconsistent findings exist with respect to body composition differences between children with and without DS.

Unlike body composition, there is a strong consensus on the musculo-skeletal abnormalities present in children with DS. Documented problems include hypotonia, joint laxity, and hypermobility. While conducting orthopedic evaluations, Concolino and colleagues found that 30% of children with DS had at least moderate hypotonia whereas only slight hypotonia was observed in 5% of the children without DS (Concolino, Pasquzzi, Capalbo, Sinopoli, & Strisciuglio, 2006). Concolino’s results are consistent with earlier work by Morris, Vaughan, & Vaccaro (1982), who also found decreased muscle tone in children with DS compared to control subjects.

Concolino’s work revealed additional musculo-skeletal challenges for children with DS, namely increased range of motion and joint laxity. Specifically, they observed that all fifty children with DS had joint laxity, with 20% demonstrating “severe” laxity. In contrast, 80% of the children without DS demonstrated no laxity at all. Excessive range of motion, particularly at the hip, has been widely documented in other studies as well.

Angelopoulou and colleagues (1999) examined range of motion in children with DS, other forms of ID, and no ID (Angelopoulou, Tsimaris, Chirstoulas, & Mandroukas, 1999).

The children with DS had significantly higher ranges of motion for hip flexion compared to the group with non-DS intellectual disability. In addition, significantly higher ranges of motion for hip abduction were observed for the children with DS relative to the other two groups. Difficulties with range of motion in DS are not limited to the hip, however.

Research has shown that in addition to hip dysplasia, individuals with DS are also susceptible to patellar instability and disorders of the ankle, such as pes planus (Mik, Gholve, Scher, Widmann, & Green, 2008).

The presence of musculo-skeletal abnormalities may contribute to observed strength deficiencies in individuals with DS. Croce (1996) found that adults with DS demonstrated significantly lower isokinetic hamstring and quadriceps peak torque levels relative to sedentary controls. Isokinetic strength in adults with DS has also been compared to adults with other forms of intellectual disability (Croce, Pitotti, Horvat, & Miller (1996). Results from Pitetti et al. (1992) showed that isokinetic arm (elbow flexion/extension) and leg (knee flexion/extension) strength were lower in DS and nonDS ID groups in relation to the adults without ID (Pitetti, Climstein, Mays, & Barrett, 1992). Additionally, the DS group exhibited decreased leg strength compared to the non-DS ID group, suggesting that factors outside of cognition contribute to strength deficiencies in DS.

Decreased muscle strength in DS has not been limited to adult populations however. Deficiencies in knee strength have been documented in children and adolescents with DS as well. Specifically, Cioni et al. (1994) examined isokinetic knee extensor strength in children and adolescents with DS, with non-specific ID, and with typical development (Cioni, Cocilovo, Di Pasquale, Araujo, Siqueira, & Bianco, 1994).



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