Tourette Syndrom

DAILY FUNCTIONING OF CHILDREN WITH TOURETTE SYNDROME: AN
EXPLORATORY STUDY
by
Sheng-Hui Liao
July 21, 2015
A thesis submitted to the

Faculty of the Graduate School of
the University at Buffalo, State University of New York
in partial fulfillment of the requirements for the
degree of
Master of Science
Department of Rehabilitation Science
ProQuest Number: 1600781
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DAILY FUNCTIONING OF CHILDREN WITH TOURETTE SYNDROME
Copyright by
Sheng-Hui Liao
2015
ii

Acknowledgement
First and foremost, I would like to express my sincere gratitude to my advisor Dr. Janice
Tona for her continuous support and guidance during my completion of the Masters degree. I
especially appreciated her sparing the time to help me with the process for obtaining the approval
of the University at Buffalo Institutional Review Board and the development of the online parent
survey.
I would also like to give thanks to my other thesis committee members: Dr. Robert
Burkard, Dr. Kimberley Persons, Dr. Machiko Tomita, and Marge Henning, MS, OTR/L for
their review of my thesis. I gratefully appreciated their encouragement and insightful comments
and suggestions. Special thanks to Dr. Susan Nochajski for her review of my research proposal
and offer of thoughtful feedback, to Dr. Machiko Tomita for her insightful help with data
analysis and interpretation of the statistical test results, and to Marge Henning, MS, OTR/L for
her help with the recruitment of an expert for the development of content validity of the parent
survey. I sincerely thank Susan Conners, M.Ed., the president of the Tourette Syndrome
Association of Greater New York State, for her help with the participant recruitment process. I
must also acknowledge Dr. Jan Rowe, OTR/L, Lesley Geyer, OTR/L, and Heather Simpson,
OTR/L for their participation in reviewing and establishing content validity of the parent survey
and for their precious comments on how to revise the survey.
Many thanks go to the Department of Rehabilitation Science at the University of Buffalo
for providing assistance in all the instances. I would also like to express my appreciation for my
families and friends in Taiwan, especially Hsin-Yi Lee, Pei-Yu Kuo, Kuan-Yu Hsu, Yu-Chen
Wang, Shu-Ting Lu, and Chia-Ling Kao, for their constant encouragement and emotional
support during my time at graduate school. Thanks also go to my friends, church family, and
iii

staff and friends from the International Student, Inc. in Buffalo, especially Lisa Kragbe, Phyllis
Lawecki, Fran Strong, Xiaone Wei, Hui Yang, Eileen Keh, Jou-An Pan, Shin-Ping Lin, WanHsuan Lin, Yi-Ping Lin, Mari Goto, Tomomi Tsuji, Mi Jhang, and Hyemin Kim for bringing me
joy and comfort and lighting up my spiritual life.
Finally, I give thanks to the Lord, for He is good. His faithful love endures forever. All
glory to Him forever and ever.
iv

Table of Contents
Acknowledgement.................................................................................................................
iii
List of tables..........................................................................................................................
viii
List of figures........................................................................................................................
ix
Abstract.................................................................................................................................. x
Introduction...........................................................................................................................
Literature review...................................................................................................................
Signs, symptoms, and diagnostic features...................................................................... 3

Clinical course........................................................................................................
Premonitory urges and other sensory issues...........................................................
Etiology..........................................................................................................................
Neuropathology....................................................................................................... 9
Functional implications..................................................................................................
10
Evaluation....................................................................................................................... 13
Intervention....................................................................................................................
14
Medications............................................................................................................. 15
Behavioral interventions.........................................................................................
16
Theoretical framework...................................................................................................
19
Domain of occupational therapy............................................................................. 19

Process of occupational therapy.............................................................................
22
Summary........................................................................................................................
23
Research questions.........................................................................................................
25
Method................................................................................................................................... 25

Study design...................................................................................................................
25
Sampling........................................................................................................................
26
Instrument....................................................................................................................... 28
Expert survey..........................................................................................................
29
Pilot survey.............................................................................................................
29
Parent survey........................................................................................................... 30
Procedures......................................................................................................................
32
Phase I..................................................................................................................... 32
Phase II.................................................................................................................... 33
Human subject protection............................................................................................... 35
Data analysis..................................................................................................................
36
Results...................................................................................................................................
38
Sample Demographic and health characteristics............................................................ 39

Functional performance during a waxing period...........................................................
40
Differences in functional performance before, during, and after a waxing period........
42
Predictors of functional limitations during a waxing period.......................................... 43

Occupational therapy services for children with Tourette syndrome............................
45
Occupational therapy services and functional performance during a waxing period....
46
Discussion.............................................................................................................................. 46
Functional performance in children with Tourette syndrome........................................
47
Factors influencing functional performance in children with Tourette syndrome......... 50

Strengths, limitations, and future research............................................................................
55
Implications for occupational therapy practice.....................................................................
56
vi

Conclusion.............................................................................................................................
58
References.............................................................................................................................
59
Appendices............................................................................................................................
80
Appendix A: Finalized parent survey............................................................................. 80

Appendix B: Expert survey............................................................................................
165
Appendix C: Sample of the pilot survey........................................................................
170
Appendix D: University at Buffalo Institutional Review Board approval letter............ 173

Appendix E: Template for cover letter to the experts....................................................
176
Appendix F: Experts consent form................................................................................. 178

Appendix G: Template for cover letter to the TSA-GNYS board members.................. 182
Appendix H: Pilot consent form for board members of the TSA-GNYS......................
184
Appendix I: Invitation to members of the TSA-GNYS.................................................
189
Appendix J: Parent consent form for members of the TSA-GNYS...............................
191
Appendix K: Thank-you email to participants...............................................................
195
Appendix L: Expert comments on survey items............................................................
196
Appendix M: Expert and TSA-GNYS board member feedback on survey questions... 199
Appendix N: Parent comments on medication for childrens tic or non-tic symptoms.
201
Appendix O: Parent comments on the OT services their child received.......................
204
vii

List of Tables
Table 1
Demographic Characteristics of the Children with Tourette Syndrome and

Participating Family............................................................................................
68
Table 2
Diagnosis-related Characteristics of the Children with Tourette Syndrome....... 69
Table 3
Comorbid Disorders Experienced by the Children with Tourette Syndrome.....
Table 4
Functional Limitations in Occupations during a Waxing Period........................ 71
Table 5
Functional Limitations in Body Functions and Performance Skills during a
70
Waxing Period..................................................................................................... 72
Table 6
Differences in Functional Performance in Occupations Before, During, and

After a Waxing Period......................................................................................... 73
Table 7
Differences in Functional Performance in Body Functions and Performance

Skills Before, During, and After a Waxing Period.............................................
Table 8
Predictors of the Experience of Functional Limitations in Occupations, Body

Functions, and Performance Skills during a Waxing Period..............................
Table 9
74
75
Information about Occupational Therapy (OT) Services for the Children with
Tourette Syndrome.............................................................................................. 76
Table 10
Comparison of Occupations during a Waxing Period by Receiving

Occupational Therapy (OT) Services or Not......................................................
Table 11
77
Comparison of Body Functions and Performance Skills during a Waxing

Period by Receiving Occupational Therapy (OT) Services or Not..................... 78
viii

List of Figures
Figure 1
Aspects of the domain of OT..............................................................................
ix
79

Abstract
Objective This retrospective study sought to identify the functional limitations in occupations,
body functions, and performance skills, during periods of increased symptoms (waxing periods)
and periods of decreased symptoms (waning periods) in children with Tourette syndrome (TS).
Additionally, relevant demographic and health factors and frequency of occupational therapy
intervention were explored.
Method Parents of 32 children with TS under the age of 18 participated in an online survey
based on the Occupational Therapy Practice Framework: Domain and Process Third Edition.
Results The children with TS experienced difficulty in functional performance of all
occupations, body functions, and performance skills to varying degrees during a waxing period,
with the majority experiencing difficulty in sleep, rest, handwriting, participating in organized
activities, all mental functions, tactile (touch) perception, and ability to suppress tics..
Demographic and health factors predicting functional limitations in theses areas were age,
gender, and the comorbid symptoms of anxiety disorders and sensory processing disorder.
Fewer than one-third of the children had ever received occupational therapy intervention.
Conclusion
These results suggest a strong role for occupational therapy during evaluation and
intervention for children with TS. The findings could be used to guide occupational therapy
practice and provide basic knowledge for parents and other related professionals to support the
daily functioning of these children. Future research exploring the effectiveness of occupational
therapy intervention including those aimed at sensory processing, tic management, and anxiety
symptom reduction is needed.
Keywords: children, functional performance, occupational performance, occupational
therapy, Occupational Therapy Practice Framework III, Tourette syndrome
Introduction
Tourette syndrome (TS) is a childhood-onset neuropsychiatric disorder characterized by
the chronic presence of involuntary motor and vocal tics lasting for more than one year (Ludolph,
Roessner, Mnchau, & Mller-Vahl, 2012). Tics wax and wane over time, indicating periods
when the severity and frequency of tics are at their peak and periods when tics are relatively
quiet (Woods et al., 2008). The prevalence of TS in children aged 6 to 17 years in the United
States is 0.3% (Centers for Disease Control and Prevention [CDC], 2013). The two predominant
comorbid disorders experienced by children with TS are attention-deficit/hyperactivity disorder
(ADHD) and obsessive-compulsive disorder (OCD; Robertson, 2012). Other common
comorbidities include depression, anxiety disorders, behavioral problems, and autism spectrum
disorders (CDC, 2013).
Occupational therapy (OT) practitioners use occupations, also called daily life activities,
to enhance or enable their clients participation in roles, habits, and routines in different settings
such as home, school, community, and workplace (American Occupational Therapy Association
[AOTA], 2014). The Occupational Therapy Practice Framework: Domain and Process Third
Edition (OTPF-III) describes the domain that OT practitioners address and the process of OT
evaluation and intervention (AOTA, 2014). The domain of OT, based on the International
Classification of Functioning, Disability and Health (ICF) published by the World Health
Organization (WHO; 2001), identifies eight occupations that are essential to daily life, including
activities of daily living, instrumental activities of daily living, rest and sleep, education, work,
play, leisure, and social participation, along with several client factors, including body functions,
performance skills, performance patterns, contexts and activity demands (AOTA, 2014).
Children with TS are known to have difficulty in several functional areas which coincide with
the areas frequently addressed by OT practitioners such as sensory processing issues and
difficulty in handwriting, reading, transition, social skills, problem solving, and paying attention
(AOTA, 2014; Giordano & Edelman, n.d.). These functional deficits may further induce family
stress, social relationship problems, lowered educational outcomes and quality of life, and
increased healthcare needs (Eddy et al., 2011; Ludolph et al., 2012; Robertson, 2012; Topolski,
n.d.). Although the National Tourette Syndrome Association (TSA) recommends identifying the
prevalence of specific functional deficits such as handwriting problems in children with TS, there
is little published data regarding the frequencies and types of functional deficits experienced by
children with TS in the literature (Giordano & Edelman, n.d.).
Pediatric acute-onset neuropsychiatric syndrome (PANS) is a neuropsychiatric disorder
that has similar symptoms to TS. Research in children with PANS has revealed the functional
limitations in occupations, body functions, and performance skills experienced by those children
when PANS symptoms are at their peak (Bhattacharjya, 2013). PANS is characterized by abrupt,
dramatic onset of OCD and concurrent neuropsychiatric symptoms which may include tics,
anxiety, depression, oppositional behaviors, developmental regression, sudden deterioration in
school performance, sensory processing issues and motor abnormalities, and somatic signs and
symptoms (Swedo, Leckman, & Rose, 2012). Therefore, children with PANS and children with
TS share some similar symptoms and may have similar functional deficits. However, studies
providing the evidence are needed.
Previous findings identified that school-based OT services for children with emotional
disturbance mainly focus on motor and sensory problems instead of behavioral problems, with
the sensory integration approach being the primary approach used by the majority of schoolbased OT practitioners to treat this population (Case-Smith & Archer, 2008). However, children
with TS may benefit from treatment approaches other than the traditional, sensorimotor ones.
Rowe, Yuen, and Dure (2013) suggested that OT practitioners can use a behavioral protocol
teaching tic management (the Comprehensive Behavioral Intervention for Tics) to improve
occupational performance and participation in school and at home in children with TS. However,
neither using a sensorimotor framework nor using a behavioral framework to treat children with
TS has solid rationale because functional performance in children with TS has not been fully
studied, nor has the type and frequency of OT services provided for children with TS.
Therefore, the purpose of this study was to elucidate the functional performance in a
variety of occupations, body functions, and performance skills in children with TS using a parent
survey developed based on the OTPF-III. The OTPF-III provides a solid framework for a broad
assessment of functional performance and thus can be used to study the functional performance
in children with TS. Understanding which occupations, body functions, and performance skills
are affected in children with TS may help facilitate the OT evaluation process by providing basic
knowledge for the OT intervention process, and serve as a springboard for future studies
regarding OT intervention in children with TS.
Literature Review
Signs, Symptoms, and Diagnostic Features
Tourette syndrome is named after a French neurologist, Georges Albert douard Brutus
Gilles de la Tourette (1857-1904), who published the first case series of nine individuals with TS
in 1885 (McNaught, 2010). Although individuals with TS and their parents frequently use the
term Tourette syndrome (TS) that is also used by the TSA, the Fifth Edition of the Diagnostic
and Statistical Manual of Mental Disorders (DSM-5; American Psychiatric Association [APA],
2013) refers this condition to Tourettes disorder (TD). For the purpose of this study, the term
Tourette syndrome or TS is primarily used, but the term Tourettes disorder or TD is

used when referring to the diagnostic criteria in the DSM-5.
Based on the DSM-5 (APA, 2013), the diagnostic criteria for Tourettes disorder are as
follows:
(a) Both multiple motor and one or more vocal tics have been present at some time during
the illness, although not necessarily concurrently; (b) the tics may wax and wane in
frequency but have persisted for more than one year since first tic onset; (c) onset is
before age 18 years; and (d) the disturbance is not attributable to the physiological effects
of a substance (e.g., cocaine) or another medical condition (e.g., Huntingtons disease,
postviral encephalitis; p. 81).
The DSM-5 rules out the conditions of chronic motor or vocal tic disorder. These
conditions are identified when only motor or vocal tics have been present for more than one year.
If motor and/or vocal tics have been present for less than one year, the diagnosis of provisional
tic disorder may be considered (APA, 2013).
Tics are sudden, rapid, recurrent, non-rhythmic motor movements or vocalizations (APA,
2013). Depending on the form, anatomical location involved, and level of complexity, tics can
be further categorized into simple motor tics (e.g., eye blinking, facial grimacing, neck jerking,
and shoulder shrugging), simple vocal tics (e.g., sniffing, grunting, throat clearing, and snorting),
complex motor tics (e.g., grooming behaviors, touching, jumping, and smelling objects), and
complex vocal tics (e.g., barking and uttering animal noises or strings of words). Copropraxia
(making obscene gestures such as showing the middle finger), echopraxia (copying other
peoples movements), echolalia (repeating the last-heard sound, word, or phrase), palilalia
(repeating the sounds made or words said by oneself), and coprolalia (uttering socially
inappropriate words such as foul language or obscene words) are other representative
characteristics of tics (APA, 2013; Ludolph et al., 2012; Robertson, 2012).
In children aged 5 to 18 years, worldwide prevalence of TS is between 0.4% and 3.8%,
and the overall prevalence is calculated as 1% (Robertson, 2008). According to the CDC (2013),
0.3 percent of children aged 6 to 17 years in the United States are reported to have the diagnosis
of TS. The diagnosis of TS is more common in boys (0.4%) than in girls (0.2%) and more likely
to affect children aged 12 to 17 years (0.4%) than children aged 6 to 11 years (0.2%). In addition,
white non-Hispanic children (0.4%) are more frequently diagnosed with TS compared to
Hispanic children (0.2%) and black non-Hispanic children (0.2%; CDC, 2013).
According to the CDC (2013), 79 percent of the children diagnosed with TS have at least
one comorbid diagnosis such as ADHD, OCD, depression, anxiety, or autism spectrum disorder,
with ADHD and OCD being the predominant comorbid diagnoses (Robertson, 2012).
Behavioral problems such as obsessive-compulsive behaviors (i.e., repetitive behaviors or mental
acts that an individual feels driven to perform in response to obsessions which may be recurrent,
persistent thoughts, urges, or images, or some rigid rules in order to prevent or reduce anxiety or
distress), disinhibition (i.e., difficulty in consistently inhibiting and controlling ones own
thoughts and/or actions), oppositional behaviors, immature behaviors, resistance to supports and
accommodations, temper tantrums, inappropriate fight-or-flight responses, difficulties with
transitions, and the utterance of inappropriate words are frequently seen in children with TS
(APA, 2013; CDC, 2013; Giordano & Edelman, n.d.).
Clinical course.
The symptoms of TS typically appear between 6 and 8 years of age with simple motor
tics affecting the face and head (Ludolph et al., 2012); vocal tics usually appear two to three
years later, at about the age of 11 (CDC, 2013; Ludolph et al., 2012). Although tics wax and
wane in severity and frequency and the involved muscle groups may change over time, the
symptoms increase and peak between the ages of 10 and 12 and often reduce during adolescence
(APA, 2013; CDC, 2013; Ludolph et al., 2012).
Tics often worsen during times of stress, anxiety, or excitement. Tics are suggestible and
can be evoked by external stimuli such as a gesture or sound made by another person. Tics
reduce when an individual with TS is engaged in relaxing or absorbing activities and can be
voluntarily suppressed, although doing so may take a great effort (APA, 2013; Ludolph et al.,
2012; Robertson, 2012).
Premonitory urges and other sensory issues.
In individuals with TS, sensory-related issues such as premonitory urges and tactile
defensiveness are commonly seen (Giordano & Edelman, n.d.; Ludolph et al., 2012; Prado et al.,
2008; Reese et al., 2014).
Premonitory urges are unpleasant and uncomfortable somatic sensations immediately
preceding the tics. They are often described as an inner energy, ache, tense feelings, pressure,
itchiness, or feelings of incompleteness which urge an individual with TS to perform repetitive
behaviors in order to feel just-right (Ludolph et al., 2012; Prado et al., 2008; Reese et al.,
2014). Using a sample of 122 older adolescents and adults with TS, Reese et al. (2014)
described the nature of the premonitory urges and their relationship with tics. They reported that
most participants felt somewhat relieved from an uncomfortable urge while producing a tic and
even were able to stop their tics for a short while. The results supported the current assumption
of the tic behavioral model that tics are negative reinforcements that can alleviate the discomfort
produced by the premonitory urges. Based on the assumption, behavioral interventions for tic
management put ample emphasis on breaking the link between the urge and the tic (Reese et al.,
2014).
According to the TSA, sensory processing problems, including difficulty in visual and
auditory processing, hyposensitivity or hypersensitivity, and tactile defensiveness, are commonly
seen in children with TS (Giordano & Edelman, n.d.). Therefore, the TSA suggests that
involving an OT practitioner qualified in a sensory integration approach may be beneficial for
children with TS (Giordano, & Edelman, n.d.). However, most of the studies that addressed the
sensory issues in individuals with TS focused on premonitory urges, while the nature of the
sensory processing problems in children with TS has not been fully studied. Recently, though,
two studies of sensory processing have been published: one describing the sensory processing
problems in adults with TS and the other in children with TS. Belluscio, Jin, Watters, Lee, and
Hallet (2011) interviewed a sample of 19 adults with TS using a questionnaire adapted from the
Adult Sensory Profile and found that compared to the healthy control group, the majority (80%)
of the adults with TS experienced significant hypersensitivity to different external stimuli (i.e.,
sound, light, smell, touch, and taste), especially to those continuous, faint stimuli which may be
ignored soon by most people, such as clothing textures. Additionally, the adults with TS showed
a preference for strong tactile stimuli such as massage and rubbing the skin (Belluscio et al.,
2011). Further assessments showed that these adults had normal olfactory and tactile sensory
thresholds when faint stimuli were applied; therefore, the authors suggested that the heightened
sensory sensitivity of the adults with TS was related to altered central sensory processing instead
of enhanced peripheral detection. Similarly, Jewers, Staley, and Shady (2013) studied the
sensory processing problems in a sample of 75 children with TS using the Short Sensory Profile.
The majority of the children with TS experienced sensory modulation problems with tactile
sensitivity, auditory filtering, and overall ability to address daily sensory events, while they had
typical performance in the areas of taste/smell sensitivity, movement sensitivity, low
energy/weak, and visual/auditory sensitivity. Furthermore, those children with TS who also had
a comorbid diagnosis of ADHD experienced greater sensory processing problems, especially in
auditory filtering and visual/auditory sensitivity, compared to those with only pure TS (Jewers et
al., 2013). Thus, these two studies support the TSA endorsement of OT practitioners with a
sensory integrative background. Further studies are needed regarding the patterns of the sensory
processing problems and evidence of improvement with intervention.
Etiology
The etiology of TS can be attributed to genetic predisposition and environmental
influences (Ludolph et al., 2012). TS and the comorbid ADHD and OCD may be genetically
related, but the relationship is uncertain (Ludolph et al., 2012). The environmental risk factors
include prenatal and/or perinatal adversities, infections, neuroimmunological events, and
androgen influences (Robertson, 2012). Chao, Hu, and Pringsheim (2014) conducted a
systematic review examining the prenatal, perinatal, and neonatal risk factors for TS. Due to
insufficient methodological quality, the evidence was inconsistent and weak. Most of the results
showed no association between parental demographic factors (e.g., age, education level,
socioeconomic status, and marital status) and the onset of TS or the presence of comorbid
disorders. Some risk factors were reported to be inconsistently associated with the onset and
severity of TS symptoms and the presence of comorbid disorders, including more frequent
prenatal visits; pregnancy complications (e.g., hypertension, infections, eclampsia, psychosocial
stress, and diabetes mellitus); antibiotic exposure; severity of nausea or vomiting during
pregnancy; maternal smoking; exposure to coffee, cigarette, and alcohol; delivery complications;
and lower birth weight. Of these risk factors, maternal smoking and lower birth weight were
most consistently reported (Chao et al., 2014).
Neuropathology.
Previous findings suggest that tics are caused by the combined effects of (a) the excessive
activity in the cortico-striato-thalamo-cortical (CSTC) circuits that are involved in motor control,
including the premotor, supplementary motor, primary somatosensory, primary motor, and
prefrontal cortices and the pallidum, thalamus, substantia nigra, and putamen, and (b) reduced
activity in cognitive control parts of the CSTC circuits (i.e., striato-nigro-striatal and striatothalamo-striatal circuits) that are involved in inhibition of tic behaviors or premonitory urges,
including the anterior cingulate, caudate, and parietal operculum (Wang et al., 2011). Increased
activity in the amygdala and hippocampus, in response to the emotional discomfort raised by the
premonitory urges or the relief experienced after performing the tics, is also suggested to have a
role in tic generation (Leckman, Bloch, Smith, Larabi, & Hampson, 2010; Wang et al., 2011).
These abnormalities are located in the basal ganglia, cortical areas, and limbic regions (Leckman
et al., 2010).
Increased nigrostriatal dopaminergic activity, which may result from the hypersensitive
dopamine receptors, dopaminergic hyperinnervation, or abnormal presynaptic terminal function
in the striatum, is believed to cause TS and the assumption is supported by clinical trials and
animal models. Furthermore, noradrenergic projections to the prefrontal and cortical regions are
suspected to indirectly influence dopaminergic activity and further influence tic severity
(Leckman et al., 2010). Other neurotransmitter and neuromodulatory systems involved in the
CSTS circuits such as the histaminergic, serotonergic, excitatory and inhibitory amino acid, and
10
cholinergic systems may also play a role in the emergence of tics, but relevant evidence is weak
(Leckman et al., 2010).
Functional Implications
The TSA reports that children with TS frequently experience functional deficits in
various areas including attention, handwriting, reading, social participation, anxiety management,
and executive functions such as time management and problem solving (Giordano & Edelman,
n.d.). Previous studies also have identified some functional problems experienced by children
with TS that correspond with, or supplement to, the TSAs statement. However, due to the small
number of studies conducted in each functional area and the small sample sizes involved in
previous studies, relevant evidence is limited and study results sometimes are conflicting.
Regarding social participation problems, Bawden, Stokes, Camfield, Camfield, and Salisbury
(1998) compared the scores of the Pupil Evaluation Inventory on the factors of withdrawal,
aggression, and likeability between a sample of 25 children with TS and a healthy control group
of their same-gender classmates and a chronic disease control group of children with type I
diabetes, and found significantly poorer peer relationships in children with TS, especially among
those who had a diagnosis of ADHD. In addition, using the Vinland Adaptive Behavior Scales,
Dykens et al. (1990) found significant weaknesses in social interaction skills in a sample of 30
children with TS.
Previous findings regarding fine motor skills were conflicting. Buse et al. (2012) did not
find differences in fine motor skills, as measured by the Finger Tapping Test and the Purdue
Pegboard Test, between a sample of 27 male children with pure TS and a control group of 26
healthy male children. On the other hand, Bloch, Sukhodolsky, Leckman, and Schultz (2006)
found their sample of 32 children with TS showed lowered visual motor integration and fine
11
motor skills, as measured by the Beery-Buktenica Developmental Test of Visual Motor

Integration and the Purdue Pegboard Test respectively during the initial evaluation of the study.
Regarding handwriting performance, parents or teachers of children with TS often recognize
difficulties with regard to sloppiness, slow writing, uneven spacing, writing too hard, words
floating above or under the line, insistence on writing perfectly making writing time-consuming
and laborious, refusal to write, difficulty in cursive writing, and inability to express thoughts
through writing (Giordano & Edelman, n.d.). Also, based on a few case reports, complex motor
tics such as echographia (compulsive rewriting phrases or words that are seen or heard),
coprographia (writing obscene words or drawing obscene pictures), and paligraphia (involuntary
repetition of letters, words, or phrases writing) may have an impact on handwriting in individuals
with TS (Cavanna, Ali, & Rickards, 2011; Mitchell & Cavanna, 2013). Handwriting difficulties
are common concerns in children with TS; nevertheless, relevant research is scarce (Giordano &
Edelman, n.d.). Only one case study has been conducted, which involved a child under the age
of 18 years (Cavanna et al., 2011).
Difficulties with math performance was identified in the study conducted by Dykens et al.
(1990) using the Woodcock-Johnson Test of Achievement in a sample of 30 children with TS.
Huckeba, Chapieski, Hiscock, and Glaze (2008) further confirmed this finding using the
Mathematics Computations subtest of the Kaufman Test of Educational Achievement, and
determined that intellectual ability and attention, rather than visuospatial skills, were associated
with the arithmetric achievement in a sample of 47 children with TS. Most recently, using a
sample of 123 children with TS, Ghosh et al. (2014) identified that sleep problems are frequently
seen in children with TS and the common sleep problems experienced by them include difficulty
initiating and maintaining sleep, abnormal behaviors during sleep (e.g., periodic limb movements,
12
sleepwalking, sleep talking, vivid nightmares, ritualistic behaviors before bedtime), and
secondary problems such as difficulty waking up, feeling unrefreshed after waking up, and
excessive daytime sleepiness.
Research in quality of life (QoL) of individuals with TS also revealed several functional
deficits that may influence their QoL. This type of research mainly involves teenagers or adults
rather than younger children. It is because measuring childrens QoL is difficult when they are
still growing and their QoL fluctuates, and also because disease-specific QoL assessments for
children with TS is lacking (Eddy et al., 2011). Generally speaking, children with TS between 8
and 17 years of age have lower QoL compared to their healthy counterparts in functional areas
related to psychosocial well-being, emotion, social life, and school occupations; above all, areas
of social participation and interpersonal relationship were influenced the most ( Eddy et al., 2011;
Storch et al., 2007). Children with TS are more likely to feel unwelcome to others, engage less
often in social activities, withdraw themselves from their peers or family, and have more
arguments with their family and are easily teased or bullied due to the stigma associated with tics
(Eddy et al., 2011; Topolski, n.d.).
In addition, tic severity, comorbid disorders such as OCD or ADHD, and a perceived lack
of tic control may have an impact on the QoL of children with TS. Those who have more severe
tics or comorbid symptoms which influence their physical well-being (e.g., jerking muscle
groups) or draw others negative attention (e.g., barking) report lower QoL compared to those
who have pure TS or whose tics are milder (Eddy et al, 2011; Storch et al., 2007; Topolski, n.d.).
Tic severity and comorbid ADHD or OCD symptoms were also found to have an impact on the
functional performance in different areas such as social participation, math, and fine motor skills
among children with TS (Bawden et al., 1998; Buse et al., 2012; Huckeba et al., 2008). Different
13
occasions and the subsequent waxing and waning of their symptoms may also influence the dayto-day functional performance in children with TS. For example, their tics may increase and
they may express the accumulative stress through disruptive behaviors when arriving at home
from school (Giordano & Edelman, n.d.). Therefore, when discussing functional performance
patterns in children with TS, the impact of tic severity and comorbid symptoms should be taken
into consideration.
Evaluation
A few approaches have been developed to measure tic severity among children with TS.
One strategy involves counting the number of tics over a short period of time under structured
conditions (Shapiro & Shapiro, 1984). However, the wax and wane in tic severity across
different settings may limit the feasibility and usefulness of the strategy of tic frequency count
(Leckman et al., 1989). The Tourette Syndrome Severity Scale developed by Shapiro and
Shapiro (1984) is used to rate the degree of tics noticeability and their interference with
functioning. The Tourette Syndrome Global Scale developed by Harcherik, Leckman, Detlor,
and Cohen (1984) is a multidimensional scale measuring the complexity, frequency, and degree
of disruption of simple and complex motor and vocal tics as well as their impact on social
functioning regarding areas of general behavior, motor restlessness, and school or work
functioning over the previous week. However, these approaches or instruments were criticized
for their limitations such as inadequate psychometric properties, lengthiness, and scoring issues
(Leckman et al., 1989). Therefore, to address the limitations of the Tourette Syndrome Severity
Scale and the Tourette Syndrome Global Scale, Leckman et al. (1989) developed the Yale Global
Tic Severity Scale (YGTSS). The YGTSS is a semi-structured interview completed by a
clinician. The YGTSS starts with recording the presence and characteristic of motor and vocal
14
tics experienced by an individual with TS during the previous week. Then the clinician rates the
severity of tics in each of the five different dimensions which include the number of tics and the
frequency, intensity, complexity, and interference of the motor and vocal tics using a six-point
Likert scale. The YGTSS also includes a separate section of impairment rating regarding the
impact of tics on the individuals interpersonal relationship, education, and work using a 50point Likert scale (Leckman et al., 1989). Stability and adequate psychometric properties of the
YGTSS regarding internal consistency reliability, inter-rater reliability, convergent validity, and
discriminant validity have been established (Leckman et al., 1989; Storch et al., 2005). The
YGTSS has been frequently used in studies regarding children with TS to report the
demographic characteristics or to investigate the relationships between tic severity and specific
functional performance problems (Bawden et al., 1998; Bloch et al., 2006; Buse et al., 2012;
Eddy et al., 2011; Storch et al., 2007).
Intervention
Due to the individual variability in symptoms, temporal fluctuations of tics, and lack of
evidence supporting the effect of medication, the European Society for the Study of Tourette
Syndrome suggests that clinicians adopt a watch and wait strategy when providing
interventions for children with TS, and offering psychoeducation and monitoring about the
natural course and potential impact of TS as the first step of treatment (Roessner et al., 2011).
Non-pharmacologic (i.e., behavioral) and/or pharmacologic interventions are taken into
consideration only when tics result in discomfort or pain, injury, sustained social concerns (e.g.,
stigmatization, isolation, and bullying), emotional issues (e.g., depression, anxiety, low selfesteem, and withdrawal), and functional interference (e.g., academic underachievement and
difficulty in paying attention, regulating sleep, and speech; Roessner et al., 2011; Roessner,
15
Schoenefeld, Buse, Bender, & Enrlich, 2013). However, there is no evidence that compares the
effectiveness and efficacy of behavioral interventions and medications to show which should be
tried prior to another (Roessner et al., 2011).
Furthermore, if an adult with TS experiences severe tics which cannot be suppressed by
medications and behavioral interventions, or has intolerable side effects of medication, which
severely impacts his or her quality of life, then the individual may consider receiving deep brain
stimulation, the electrical stimulation of specific brain regions using surgically implanted
electrodes (Mller-Vahl, 2013; Roessner et al., 2011; TSA, n.d.). However, randomized
controlled trials for deep brain stimulation using larger sample size are scarce and available
research results are contradictory and limited. Therefore, it is still not approved by the U.S. Food
and Drug Administration and considered an experimental approach that can only be performed
on adults with TS (Mller-Vahl, 2013; TSA, n.d.).
Medications.
Depending on the major symptoms, age, and medical history, different medications may
be prescribed for individuals with TS to reduce tic severity and/or the behavioral and emotional
problems (e.g., hyperactivity, impulsiveness, and obsessive-compulsive behaviors). However,
the medications targeted at the behavioral or emotional problems may or may not influence tic
severity directly (Coffey, Berlin, & Naarden, n.d.).
Medications such as haloperidol (Haldol), pimozide (Orap), fluphenazine (Prolixin),
risperidone (Risperidal), clonidine (Catapres), guanfacine (Tenex), clonidine patch (Catapres),
and clonazepam (Klonopin) will reduce tic severity and frequency. However, the side effects of
these medications may influence the functional performance in individuals with TS. Haloperidol,
pimozide, fluphenazine, and risperidone may cause fatigue, drowsiness, increased appetite and
16
weight gain, photosensitivity, depression, and extrapyramidal symptoms such as muscle stiffness,
dyskinesia, tardive dyskinesia, and Parkinsonism. Clonidine, clonidine patch and guanfacine
may cause fatigue, drowsiness, irritability, confusion, depression, insomnia, headache,
constipation, and dry mouth. Clonazepam may cause fatigue, irritability, dizziness, and
disinhibition (Coffey et al., n.d.; Roessner et al., 2013).
Medications for comorbid ADHD include methylphenidate (Ritalin), dextroamphetamine
(Dexedrine), mixed amphetamines (Adderall), and atomoxetine (Strattera), all of which may
cause headache, stomachache, decreased appetite, insomnia, irritability, and increased tics
(Coffey et al., n.d.). Fluoxetine (Prozac), paroxetine (Paxil), clomipramine (Anafranil),
sertraline (Zoloft), fluvoxamine (Luvox), and citalopram (Celexa) are often used in treatment of
anxiety either with or without OCD, but they may cause restlessness, sexual dysfunction,
insomnia, gastrointestinal upset, or constipation. Clomipramine and sertraline may also cause
weight gain and fatigue (Coffey et al., n.d.). Therefore, the daily life activities, sleep patterns,
school function, cognition, vision, and motor function in children with TS are also likely to be
affected by medication side effects.
Behavioral interventions.
Behavioral interventions teach individuals with TS strategies to consciously control their
tics (Frank & Cavanna, 2013). Approaches to tic management include: (a) massed negative
practice, which requires voluntary repetitive performance of a tic for a mass amount of time (30
minutes per day, at least three days per week, for three months) to suppress the tic through the
process of conditioned inhibition (Storms, 1985); (b) supportive psychotherapy, which involves
conversation on different topics and focuses on reflecting feelings about current life issues and
problem solving (Wilhelm et al., 2003); (c) exposure with response prevention, which reduces
17
tics through asking individuals with TS to suppress a tic for extended periods of time to become
habituated to the premonitory urge (Verdellen, Keijsers, Cath, & Hoogduin, 2004); (d) selfmonitoring, which requires individuals with TS to acknowledge and record every tic they
produced and thereby reduces tics through their increased awareness (Thomas, Abrams, &
Johnson, 1971); (e) cognitive behavioral therapy, which focuses on recognizing high risk
situations that may increase tics and using cognitive and behavioral strategies to restructure the
actions of individuals with TS (OConnor, Laverdure, Taillon, Stip, & Borgeat, 2009); (f)
relaxation therapy, which involves the practice of various relaxation techniques such as
progressive muscle relaxation, deep breathing, visual imagery, and posturing like a relaxed
person in tic-inducing situations (Bergin, Waranch, Brown, Carson, & Singer, 1998); (g)
assertiveness training, which trains individuals with TS to assertively respond to external
environment instead of turning to their tics, according to Tophoff (as cited in Frank & Cavanna,
2013); and (h) habit reversal training, which teaches tic management techniques through
awareness training (self-monitoring of premonitory urges and tics) and competing response
training (performing a voluntary behavior when noticing the premonitory urge or the beginning
of the tic; Hwang, Tillberg, & Scahill, 2012). Among these approaches, habit reversal training is
frequently recommended since it is well-studied and has demonstrated consistent effectiveness in
many studies of single-subject design, as well as randomized controlled trials (Frank & Cavanna,
2013; Hwang et al., 2012).
In 2001, building on habit reversal training, the Tourette Syndrome Association
Behavioral Science Consortium started developing the comprehensive behavioral intervention
for tics (CBIT), a manualized eight-session treatment protocol consisting of three components: (a)
habit reversal training, (b) relaxation training, and (c) function-based intervention. The CBIT
18
program can be carried out by trained professionals such as psychologists, social workers,
occupational therapists, and psychiatric nurse practitioners (Piacentini et al., 2010; Woods et al.,
2008; Woods, Piacentini, & Walkup, n.d.). In the first session, psychoeducation about TS,
rapport building, identification of tics, and ranking the tics based on levels of disturbance are
taught. In the second and third sessions, a functional assessment is implemented and a target tic,
usually the most disturbing one, is chosen. Antecedents and consequences of that tic, as well as
strategies that can be used to address the antecedents and consequences are identified. Finally,
habit reversal techniques for tic management are presented. In the fourth and fifth sessions,
relaxation techniques are introduced. In the sixth and seventh sessions, strategies to develop
competing responses for new tics are taught. In the eighth session, tic relapse prevention
strategies such as the use of picture reminders and word menus are developed or modified
(Woods et al., 2008). The CBIT program is effective in reducing tic severity and tic-related
functional impairments in children with TS and has become one of the most popular approaches
to tic management (Piacentini et al., 2010). In addition to reducing tic severity, Rowe et al.
(2013) suggested that the CBIT program is also feasible and effective in improving the
occupational performance in children with TS and chronic tic disorder, which may further help
them fulfill their occupational roles and assist their participation in home, school, and
community activities (Rowe et al., 2013). However, areas of occupational performance in
children with TS are affected has not been systematically studied. Furthermore, studies
addressing OT interventions for children with TS are scant as even the frequency of this
population receiving OT services is not yet known. Thus, regarding OT interventions for
children with TS, although a sensory integration approach has been identified as the primary
approach to deliver OT services to children with emotional and behavioral problems in the
19
school setting (Case-Smith & Archer, 2008), the choice of either the sensory integration
approach or the newly developed CBIT program lacks evidence and solid rationale to be used.
Theoretical Framework
The OTPF-III is an official document developed by the AOTA, presenting central
constructs that guide OT practice and decision making, which also affords the opportunity to
evaluate overall functioning in individuals with TS and other challenges. In the OTPF-III, OT is
defined as the therapeutic use of everyday life activities (occupations) with individuals or
groups for the purpose of enhancing or enabling participation in roles, habits, and routines in
home, school, workplace, community, and other settings (AOTA, 2014, p.S1). Occupational
therapy practitioners value the positive relationship between occupation and health; they view
people as occupational beings that need to engage in occupations in order to grow and thrive
(AOTA, 2014). Furthermore, OT practitioners view clients from a holistic perspective, rather
than focus on isolated abilities or skills (AOTA, 2014).
The OTPF-III comprises two major sections: (a) the domain, which identifies the scope
of practice and areas of knowledge base of OT practitioners, and (b) the process, which
illustrates the actions and procedures for service delivery; both components are dynamically
woven together during the delivery of OT services.
Domain of occupational therapy.
The domain describes the areas where OT practitioners have knowledge and expertise,
including occupations, client factors, performance skills, performance patterns, and context and
environment. Figure 1 depicts the aspects of the domain of OT. All aspects interact with one
another and are equally important in influencing clients occupational identity, health, well-being,
20
and participation in daily life (AOTA, 2014). For this study, the domain section of the OTPF-III
is used to investigate the daily functioning of children with TS.
Occupations.
Occupations are the daily life activities in which people engage (AOTA, 2014, p. S5).
Occupations are meaningful and valuable to clients, are essential to their identity, have purpose,
occur over time and in various contexts, and may be affected by the interaction of client factors,
performance skills, and performance patterns. Occupations are further categorized into (a)
activities of daily living, which includes bathing/showering, toileting and toilet hygiene, dressing,
swallowing/eating, feeding, functional mobility, personal device care, personal hygiene and
grooming, and sexual activity; (b) instrumental activities of daily living such as care of others
and financial management; (c) rest and sleep; (d) education; (e) work; (f) play; (g) leisure; and (h)
social participation (AOTA, 2014).
Client factors.
Client factors are specific capacities, characteristics, or beliefs that reside within the
person and that influence performance in occupations, which include (a) values, beliefs, and
spirituality; (b) body functions; and (c) body structures (AOTA, 2014, p. S7). Client factors may
not only influence performance skills but also be influenced by performance skills and other
aspects of domain such as performance patterns, and contexts and environments (AOTA, 2014).
Among the client factors, body functions and body structures are concepts obtained from the ICF
(WHO, 2001). Body structures refer to the anatomical parts of the body (e.g., structure of the
nervous system and structures involved in voice and speech), and body functions are
physiological and psychological functions of body systems, which include (a) mental; (b)
sensory; (c) neuromusculoskeletal and movement-related; (d) muscle; (e) movement; (f)
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cardiovascular, hematological, immunological, and respiratory system; (g) voice and speech; (h)
digestive, metabolic, and endocrine system; (i) genitourinary and reproductive; and (j) skin and
related structure functions. Body functions and body structures also interplay with one another
affecting clients participation in meaningful occupations (AOTA, 2014).
Performance skills.
Performance skills are the clients demonstrated abilities to participate in everyday life
activities (AOTA, 2014, p. S7) and observable elements of action that have an implicit
functional purpose (AOTA, 2014, p. S25). Performance skills are interrelated and encompass
(a) motor skills involved in interacting with objects and moving objects and self around; (b)
process skills required for using tools and materials, executing actions, and modifying
performance; and (c) social interaction skills involved in social exchange (AOTA, 2014).
Performance patterns.
Performance patterns are habits, routines, roles, and rituals involved in the process of
activity participation. Habits are specific behaviors performed automatically and repeatedly;
routines are regular behavioral patterns structuring a clients daily life; roles are sets of behaviors
performed according to social expectations; rituals are symbolic and meaningful actions
contributing to a clients identity. Performance patterns develop over time and can either
facilitate or impede occupational performance (AOTA, 2014).
Context and environment.
The two terms, context and environment, sometimes are interchangeable, but contexts
consist of more intangible elements than environments do. Contexts are various interrelated
conditions within and surrounding the client, including cultural, personal, temporal, and virtual
contexts (AOTA, 2014, p. S28). Environments are external physical and social conditions
22
surrounding the client, in which the clients occupations take place daily; environments are
further categorized into physical environments which contain nonhuman surroundings and
objects, and social environments which comprise social relationships with other people or
systems. Contexts and environments can present both resources and barriers to clients
participation in daily life activities (AOTA, 2014).
Process of occupational therapy.
The OT process, which comprises the process of evaluation, intervention, and targeting
outcomes, is the client-centered delivery of OT services carried out by OT practitioners to help
clients achieve desired outcomes and to promote their health, well-being, and participation in life
(AOTA, 2014, p. S10). The OT process is dynamic because all aspects of domain are mutually
related to one another and clients abilities are expected to change throughout the process
(AOTA, 2014).
Evaluation process.
Occupational therapy practitioners determine clients goals and desired outcomes through
identifying their concerns and needs during the evaluation process which contains the
development of (a) an occupational profile, the summary of a clients strengths and needs,
priorities and desired outcomes, daily routines, occupational history, and interests and values;
and (b) an analysis of occupational performance, the identification of a clients abilities and
deficits based on evaluation results. The evaluation process can take place in the initial contact
with a client and throughout the course of intervention to identify the clients progress or
development. Occupational therapy practitioners can use different methods such as interviews,
observations, and direct administration of standardized or unstandardized assessment tools to
obtain information on a clients occupational profile and to evaluate the contexts and
23
environments, and the clients occupational performance in activities and occupations (AOTA,
2014).
Intervention process.
The intervention process consists of three steps: (a) intervention plan, (b) intervention
implementation, and (c) intervention review, during which OT practitioners facilitate clients
engagement in occupations to promote health, well-being, and participation. Occupational
therapy practitioners collaborate with clients to develop an intervention plan using the client
information gathered during the evaluation process, principles of frame of reference or theory
applicable to the clients, and best available evidence. The intervention plan guides the actions of
OT practitioners as they address current barriers impeding clients participation in meaningful
activities; intervention implementation is the process of carrying out the intervention plan;
intervention review is the continuous reevaluation of the intervention plan and clients progress
toward the targeted outcomes. The intervention plan and implementation process may be
modified based on the reevaluation results (AOTA, 2014).
Targeting of outcomes.
Outcomes are the results that clients achieve after receiving OT intervention, such as
improved performance skills, modified environment, and increased perceived well-being.
Occupational therapy practitioners may modify the targeted outcomes during intervention
implementation and reevaluation process based on the changes of clients abilities, contexts and
environments, and needs (AOTA, 2014).
Summary
Children with TS experience physical and psychosocial symptoms that may affect quality
of life and likely impact functional performance and social participation (Storch et al., 2007).
24
The TSA states that behavioral problems, sensory processing issues and other functional deficits
with regard to attention, transition, reading, handwriting, problem solving, and social interaction
are commonly seen in children with TS (Giordano & Edelman, n.d.). Improvement in
occupational performance has been a targeted outcome for children with TS receiving the CBIT
program delivered by OT practitioners in one study. However, frequencies and degrees of
difficulty in different areas of functional performance in children with TS are not fully known.
Additionally, OT practitioners may provide different types of interventions using different
treatment approaches such as sensory integration, compensatory, and behavioral approaches
based on the individual needs of children with TS. However, such OT intervention lacks solid
rationale without evidence of occupation, body function, and performance skill deficits. Also,
the frequency of children with TS receiving OT services is not yet known. Therefore, a study to
investigate such fact is necessary. Surveying parents about functional deficits experienced by
children with TS may help OT practitioners to evaluate children with TS more efficiently and to
have better knowledge base to offer beneficial intervention. Such a study should be conducted
before intervention studies to ensure all aspects of functional deficits are recognized.
Therefore, this study aimed to investigate the impact of TS-related symptoms on the daily
functioning of children with TS, including their functional performance in different categories of
occupations, body functions, and performance skills, using a parent survey. The OTPF-III is a
framework for the assessment of functional performance and the basis of research on daily
functioning in children with TS in this study. Understanding the functional performance in
children with TS could facilitate the OT process for evaluation and intervention plan, and may
serve as a springboard for future studies of OT intervention.
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Research Questions
The research questions addressed in this study included:
1. Research questions related to functional limitations in children with TS and factors that may
influence their functional performance:
a. What percentages of children with TS have experienced functional limitations in each
occupation, body function, and performance skill during a waxing period?
b. In those children with TS who have experienced functional limitations in an occupation, a
body function, or a performance skill during a waxing period, will there be a significant
difference in functional performance in that occupation, body function, and performance skill
before, during, and/or after the waxing period?
c. Which demographic and health factors (i.e., age, gender, and major comorbid symptoms)
will predict the experience of functional limitations in each occupation, body function, and
performance skill during a waxing period in children with TS?
2. Research questions related to OT services for children with TS:
a. What percentages of children with TS have received OT services for TS-related difficulties
and for what reasons are they referred to OT?
b. Will functional performance in each occupation, body function, and performance skill during
a waxing period in children with TS who have received OT services be different compared to
those who have not received OT services?
Method
Study Design
This study was an exploratory study utilizing an online parent survey based on the OTPFIII to identify the limitations in functional performance in occupations, body functions, and
26
performance skills in children with TS. A similar study using an online parent survey based on
the Occupational Therapy Practice Framework: Domain and Process Second Edition (OTPF-II;
AOTA, 2008) revealed that children with PANS could experience functional limitations in all the
categories of occupations, body functions, and performance skills when the PANS symptoms are
at their peak (Bhattacharjya, 2013). Because children with TS have similar symptoms (e.g.,
obsessive-compulsive behaviors and tics) and functional deficits that are commonly seen but not
yet determined (e.g., behavioral problems, sensory processing issues, and handwriting
difficulties) to children with PANS (Swedo et al., 2012), the online parent survey used in this
study was adapted from the previously designed survey for parents of children with PANS
(Bhattacharjya, 2013).
This study comprised two phases. Phase I was the survey development phase, which
consisted of establishing content validity of the survey, modifying the survey questions, and pilot
testing the survey tool. Phase II was the data collection and analysis phase, which involved
launching the survey, collecting data for the main study, and analyzing the data using statistical
tests. Questions in the survey, based on the occupations, body functions, and performance skills
involved in the OTPF-III, addressed 23 occupations and 22 body functions and performance
skills (AOTA, 2014). The survey was available online for four weeks, from February 15 to
March 15, 2015, to members of the Tourette Syndrome Association of Greater New York State
(TSA-GNYS), who received an invitation with the hyperlink to the survey.
Sampling
During phase I of this study, to establish content validity of the parent survey, Lynns
approach (1986) to the determination of content validity and Grant and Davis (1997)
explanation of content expert selection were used to recruit a panel of experts. Lynn (1986)
27
suggested recruiting five or more experts to provide sufficient control of chance agreement;
however, if the number of content experts is limited in a specific content area, Lynn also
suggested that a minimum of three experts should be used. Additionally, according to Grant and
Davis (1997), having clinical expertise or expertise on the conceptual framework is an
appropriate criterion for selecting the experts to evaluate the entire instrument and individual
instrument items. Therefore, the inclusion criteria for participation in phase I determination of
content validity were (a) being an OT practitioner with at least five years of experience in
pediatric field including working with children with TS, and (b) being familiar with the concepts
of the OTPF-III. Due to the difficulty locating pediatric OT practitioners who also had enough
experience delivering service to children with TS, a purposive sampling method was used and
three OT practitioners who were affiliated with the TSA and were known to have at least five
years of experience working with children (including children with TS) were invited to
participate in this study.
After content validity of the parent survey had been established, a purposive sampling
method was used to recruit participants in pilot testing of the survey. The inclusion criteria for
participation in phase I pilot test were (a) being a board member of the TSA-GNYS and (b)
being a parent of an individual with TS. Five participants were invited to take the survey and
comment on the mechanics of the survey, such as how easy or difficult it was to answer each
question and what needed to be modified to make the final survey as user-friendly as possible.
During phase II of this study, parents of children with TS were recruited from the TSAGNYS using a convenience sampling method. The TSA-GNYS endorsed and sent a survey
invitation email (Appendix I) along with the hyperlink to the parent consent form (Appendix J)
and finalized parent survey (Appendix A) to each member on its email list. The inclusion
28
criteria for participation in phase II data collection were: (a) being a parent or guardian of a child
under 18 (0-17) years of age and diagnosed with TS, (b) being at least 18 years of age, and (c)
being able to read and write in English. A parent or guardian who had a child diagnosed with
chronic motor or vocal tic disorder but not with TS was excluded from this study. The board
members of the TSA-GNYS who participated in pilot testing could participate in phase II and fill
out the parent survey if they met the inclusion criteria.
During the period of data collection, the association had approximately 800 members
geographically residing in Northern New York area, Albany area, and Western and Central New
York area on its email list. We expected that at least 100 members would take the survey, a
response rate of 12.5%. However, the response rate was lower than expected. Therefore, the
TSA-GNYS sent out the second and the third survey invitation email 12 and 23 days respectively
after the first email was sent out to remind the members to complete the survey.
Instrument
Prior to the beginning of phase I of this study, a preliminary parent survey was adapted
from the previously-designed online parent survey titled PANS parent survey used to
investigate the daily functioning of children diagnosed with PANS. It was because children with
TS and children with PANS share similar symptoms, comorbid disorders, and functional deficits,
such as tics, obsessive-compulsive behaviors, handwriting difficulties, behavioral problems,
sensory processing issues, and so on (Bhattacharjya, 2013; Swedo et al., 2012). The previous
survey was developed based on the OTPF-II and targeted at parents of children with PANS;
therefore, the reference in the preliminary parent survey was changed from PANS to Tourette
syndrome or TS for the target population of this study, parents of children with TS. Also, the
term exacerbation was changed to waxing periods as this term is more descriptive of the
29
episodes seen in TS, and the term areas of occupation was changed to occupations based on
the terminology used in the OTPF-III (AOTA, 2008; AOTA, 2014).
Appendix A presents the finalized parent survey developed to determine the impact of the
TS symptoms on functional performance in children with TS, which is similar to the preliminary
parent survey but has been modified based on the comments given by the experts using the
expert survey (Appendix B) and the participants in the pilot test.
Expert survey.
During phase I determination of content validity of this study, an expert survey
(Appendix B) developed based on Grant and Davis suggestion and examples (1997) was used to
establish content validity of the parent survey. Using a four-point Likert scale, the survey asked
three experts to indicate whether each occupation, body function, and performance skill listed on
the preliminary parent survey was (a) not representative, (b) in need of major revisions to be
representative, (c) in need of minor revisions to be representative, or (d) representative of the
content domain of the OTPF-III (Grant & Davis, 1997).
The expert survey (Appendix B) also included a blank text column for each item so that
the experts could provide suggestion for modification of the item or its wording. After all three
experts in the expert panel had completed the survey, the content validity index was calculated
(Grant & Davis, 1997), and improvements on the preliminary parent survey were then made
based on the experts comments.
Pilot survey.
Appendix C displays a sample of the pilot survey used during phase I pilot test of this
study. The pilot survey contained all the information and questions included in the improved
parent survey with a blank text column labeled Parent Pilot Survey Comments attached to each
30
question to obtain feedback, comments, or concerns about the question from five board members
of the TSA-GNYS. The pilot survey was made available online using Vovici Enterprise
Feedback Management (EFM) software, a password-protected, secure online survey platform
which can only be accessed by the faculty advisor. All responses to the survey were secured on
the platform and were used to improve the final parent survey but not for data collection and
analysis.
Parent survey.
The finalized parent survey (Appendix A) contained 23 occupations and 22 body
functions and performance skills based on the OTPF-III. Age-appropriate examples and
descriptions of these were developed to clarify the concepts. In the parent survey, occupations
were further categorized into home-related occupations, education-related occupations, and
occupations related to social life. Home-related occupations consisted of (a) activities of daily
living, including bathing/showering, toileting, toilet hygiene, dressing, swallowing/eating,
feeding, functional mobility, personal hygiene and grooming, and sexual activity; (b)
instrumental activities of daily living, including communication management and community
mobility; (c) rest; and (d) sleep. Education-related occupations included (a) math, (b) reading, (c)
handwriting, (d) physical education, and (e) extracurricular activities. Occupations related to
social life comprised (a) play (free play); and (b) social participation, including organized
activities and social participation in the community, with family, and with peers/friends.
Although work and leisure both are important and meaningful occupations for many people, this
survey did not include these items because the target group was children under age 18, for whom
work and volunteer experiences as well as planning and participating in leisure activities are not
required.
31
Specific body functions and performance skills were selected based on the potential
functional deficits identified in the TS-related literature. Since body functions are intertwined
with performance skills (i.e., body functions can affect performance skills, and vice versa), the
selected body functions and performance skills were combined into 22 items of (a) mental
functions, including control of thoughts, control of behaviors, higher level cognitive functions,
attention, memory, sequencing, emotional coping, and energy and drive; (b) sensory functions,
including vision/seeing, hearing, vestibular/sense of moving, taste, smell,
proprioception/knowing where body is in space, tactile/touch, pain, and temperature; and (c)
muscle and movement function, including muscle strength, muscle endurance,
smoothness/control of voluntary movements, ability to suppress unwanted movements/tics, and
gait/walking patterns (AOTA, 2014).
Parents were first asked to identify which occupations, body functions, and performance
skills were problematic for their child during a waxing period. If a parent indicated that one or
more of these were problematic, conditional logic in the online survey then asked the parent to
rate the childs level of functioning before, during, and after the waxing period for that area.
Using a four-point Likert scale, the parent survey asked the participants to identify whether their
child had displayed (a) no problem; (b) mild difficulty, but did not require assistance; (c)
moderate difficulty, and required assistance and adaptation or help to remain functional; or (d)
severe difficulty, and was unable to function at a typical level, even with assistance and
adaptation before, during, and after a waxing period in each of the occupations, body functions,
and performance skills that the participants identified as problematic. The participants could
explain the difficulties that their child experienced using an open-ended question for each item.
The finalized parent survey was created and stored online using Vovici EFM software.
32
Procedures
The University at Buffalo Institutional Review Board had reviewed and approved this
study before the study started. The approval letter from the University at Buffalo Institutional
Review Board is attached in Appendix D.
Phase I.
During phase I of this study, to improve and establish content validity of the parent
survey, three OT practitioners who were affiliated with the TSA and known to have at least five
years of experience working with children including children with TS were contacted
individually via email. Appendix E displays the template for the cover letters used to solicit the
experts participation in phase I of this study. All three experts replied to the email and indicated
that they were interested in participating and would like to receive the materials electronically;
thereafter, an expert consent form (Appendix F), a copy of the OTPF-III (AOTA, 2014), a copy
of the preliminary parent survey, and the expert survey (Appendix B) were forwarded to them
via email. They were asked to print out and sign the consent form, and to scan and forward the
signed consent form to the principal investigator via email before starting to review the
preliminary parent survey and fill out the expert survey (Appendix B). All experts completed the
survey in approximately two weeks. The calculated content validity index was .98, which was
above the minimum content validity index of .80 suggested by Davis (1992) for a new
instrument, indicating that the items on the preliminary parent survey were relevant to and could
adequately represent the content domain of the OTPF-III. Therefore, no item was taken out or
added into the survey, although some demographic questions were revised to better reflect the
clinical features of TS and the wording of some items was revised to make the definition clearer
and more congruent to the terminology used in the OTPF-III based on the experts comments.
33
After content validity had been established, to pilot test the improved parent survey, five
board members of the TSA-GNYS were contacted individually via email. Appendix G displays
the template for the cover letters sent to the TSA-GNYS board members requesting their
participation. The email also included the hyperlink to the pilot consent form (Appendix H) and
pilot survey (Appendix C). All five board members completed the pilot survey and provided
their feedback, comments, and concerns about the survey questions in two weeks. The wording
in the improved parent survey was then modified based on those comments to make the finalized
survey as user-friendly as possible.
Phase II.
During phase II of this study, potential participants were recruited from the TSA-GNYS.
The TSA-GNYS endorsed and sent an invitation to participate in the online parent survey
(Appendix I) along with the hyperlink to the parent consent form (Appendix J) and finalized
parent survey (Appendix A) to each member on its email list. After the potential participants
clicked the hyperlink and linked to the webpage, the parent consent form (Appendix J) explained
the purpose, procedures, time commitment, costs, risks, and benefits of this study. The consent
form also informed the potential participants that this study was confidential, that they could
voluntarily choose whether to participate or not, and that they might skip a question if the
question caused too much distress or negative emotions for them to answer. Finally, the consent
form asked the potential participants whether they agreed to participate in the survey or not.
Since the consent form and parent survey were presented in English, individuals who could not
read and write in English were not able to click the I have read the above information and
consent to participate in the survey button and were thus excluded from participating in this
study.
34
If the potential participants agreed to participate and clicked the consent button, they
were then asked if they were at least 18 years old, if their child had been diagnosed with TS by a
physician, and if their child with TS was under 18 years old to determine if they met the
inclusion criteria. If the potential participants answered a no to one of the three questions, they
were informed that they were not eligible to participate in this study and were given the faculty
advisors email address for further contact if their situation changed or if they had any questions
or concerns. Therefore, individuals who are under 18 years old, whose child was not diagnosed
with TS, whose child was diagnosed with chronic motor or vocal tic disorder but not with TS,
and whose children was over 18 years old were excluded from participating in this study. If the
potential participants answered a yes to all of the three questions, they were included in the
study and asked to submit an email address. Email addresses were used as contact information if
the participants responses required further clarification and were used to match family with
more than one child with TS so that they would not have to complete the demographic questions
more than once; the consent form informed them of these. Also, the participants could choose
whether or not to receive a summary of the research results via email after this study has
completed.
After completing the consent form and verifying the eligibility for participation, the
participants were directed to the main section of the survey to identify their childs demographic
characteristics (e.g., age, race, and parents education level) and if their child had had difficulty
in functional performance in any listed occupations, body functions, and performance skills (e.g.,
dressing, handwriting, attention, proprioceptive function, and muscle strength). Every time the
participants identified all problematic areas in each category of occupations, body functions, and
performance skills, conditional logic directed them to identify the difficulty level of their childs
35
functioning in those problematic areas before, during and after a waxing period. Upon
completing the survey, the participants with more than one child with TS were asked if they
would like to complete the survey for another child with TS in their family; if they agreed, they
were directed to submit the survey and log in again with the same email address. Then, if they
indicated that the second childs demographic information was the same as what they had
submitted for the first child, conditional logic directed them to the main section of the survey
directly.
The survey was available online from February 15 to March 15, 2015. At the end of this
study, the participants received a follow-up thank-you note via email (Appendix K). The faculty
advisor downloaded the de-identified information and responses into an excel spreadsheet and a
SPSS data file. The participants email addresses were not downloaded with the data and were
preserved in the Vovici EFM software.
Human Subject Protection
This study was approved by the University at Buffalo Institutional Review Board
(Appendix D). Informed consent was obtained from the participants in each phase of this study
(Appendix F, H, and J). In phase I determination of content validity of the parent survey, the
experts responses were sent to the principal investigator via email and printed. The electronic
copy and email were then deleted. The responses did not have any names attached but had an ID
number written at the top of each page as an identifier. The paper copy of the de-identified
responses and expert consent forms were stored in separate files in a locked file cabinet in the
faculty advisors office at the University at Buffalo.
In phase I pilot testing and phase II of this study, responses were submitted to the online
survey platform operated by Vovici EFM software. The participants email addresses were the
36
only identifiable information that was collected, but only the faculty advisor who had a Vovici
account could access the information. All data were de-identified by removing the email
addresses before being shared with the principal investigator. The data were then removed from
Vovici EFM software and only de-identified data were analyzed and stored on a passwordprotected computer locked in the faculty advisors office at the University at Buffalo. Therefore,
the participants and the childrens identity remained confidential throughout the data collection
and analysis process.
Data Analysis
In phase I of this study, data collected using the expert survey (Appendix B) were used to
establish content validity of the parent survey (Appendix A). While validity is the extent to
which an instrument measures what it is intended to measure, content validity is used to
determine the extent to which the items on an instrument and the interpretation of scores using
that instrument adequately represent all dimensions of the content domain addressed by the
instrument (Lynn, 1986; Waltz, Strickland, & Lenz, 2010). A widely used method for
establishing content validity is calculating a content validity index, which was developed by
Hambleton, Swaminathan, Algina, and Coulson in 1975 and further enhanced by Martuza in
1977 and Waltz and Bausell in 1981 (as cited in Grant & Davis, 1997). The content validity
index is the percentage of total items scored three or four by a group of experts using a four-point
Likert scale ranging from one (not representative of the content domain) to four (very
representative of the content domain) created to determine how representative an instrument is of
the content domain (Davis, 1992). Data collected using the pilot survey were only used to
modify the wording of questions on the parent survey; thus, no statistical analysis was performed
for those data.
37
In phase II of this study, descriptive statistics, Friedman two-way analysis of variance by

ranks (Friedman ANOVA), logistic regression, and Mann-Whitney U tests were performed to
analyze the data collected using the finalized parent survey (Appendix A). Descriptive statistics
were used to illustrate the demographic characteristics of the participating families and their
children with TS. Descriptive statistics were also used to answer Research Question 1a to report
the percentage of children with TS who had experienced functional limitations in each
occupation, body function, and performance skill during a waxing period, as well as Research
Question 2a to determine the percentage of children with TS who had received OT services at a
certain period of time in their lives for TS-related difficulties and to summarize the reasons for
their receiving an OT referral.
The Friedman ANOVA was performed to answer Research Question 1b to examine the
differences in functional performance in each occupation, body function, and performance skill
before, during, and/or after a waxing period among the children with TS who had experienced
functional limitations in that occupation, body function, and performance skill during a waxing
period. The Friedman ANOVA was used because the parent survey asked the participating
parents to identify their childs difficulty level of functioning in each item before, during, and
after a waxing period, which led to a comparison among three time periods, and also because the
data were collected using ordinal scales, which required the use of nonparametric statistical tests.
To reduce Type I error, Bonferroni correction was performed by dividing the desired
significance level ( = .05) by the number of comparisons, 45, and the corrected significance
level was thus .0011 ( .001). If the result of the Friedman ANOVA was significant,
indicating that there was a significant difference between at least two time periods, the minimum
38
significant difference (Portney & Watkins, 2008) was then used and pairwise contrasts were
performed to determine where the significant pairwise differences occurred.
Logistic regression was used to answer Research Question 1c to identify the demographic
and health factors (i.e., age, gender, and major comorbid symptoms) that could predict the
experience of functional limitations in each occupation, body function, and performance skill
during a waxing period. Logistic regression was used because the parent survey asked the
participating parents to identify whether they had observed their child to have difficulty in each
occupation, body function, and performance skill considering the developmental level expected
at the childs age, and the dependent variable were dichotomous (yes/no) and categorical.
Regarding the health factors of major comorbid symptoms, the presence and absence of either a
diagnosed or a suspected comorbid disorder were respectively recoded as the presence of the
comorbid symptoms (yes/1) and the absence of the comorbid symptoms (no/0).
The Mann-Whitney U test was performed to answer Research Question 2b to examine if
the functional performance of the children with TS in each occupation, body function, and
performance skill during a waxing period were different between those who had received OT
services and those who had not. The significance level for logistic regression and MannWhitney U test was set at = .05, two-tailed. Statistical analysis was performed using IBM
SPSS Statistics Version 22.
Results
In phase I of this study, 44 out of 45 items on the parent survey (Appendix A) were
identified by the three experts using the expert survey (Appendix B) as representative or in need
of minor revisions to be representative of the OTPF-III. Only one item (rest) was considered in
need of major revisions by one expert. Therefore, the calculated content validity index was .98,
39
exceeding the suggested minimum of .80 for a new instrument (Davis, 1992), which indicated
that the items listed on the parent survey could adequately represent the OTPF-III content
domain. Appendix L presents the experts comments on the items that they considered requiring
some modifications. Appendix M presents the feedback given by the experts and the TSAGNYS board members on the survey questions, which were used to modify the wording in order
to make the survey clearer and easier for participants to answer. For example, for the 25th
question on the preliminary parent survey that asked the participants to choose any medical
diagnoses in addition to TS that their child had ever had from a list, the experts suggested adding
two more options, OCD and anxiety disorders, which are the comorbid disorders commonly
seen in children with TS, and the TSA-GNYS board members suggested adding a section
specifying symptoms without a formal diagnosis; therefore, the two comorbidities were added,
and a table was created to let participants specify whether the comorbid symptoms experienced
by their child were formally diagnosed or not.
Sample Demographic and Health Characteristics
In all, 114 people opened the survey at some point and 28 percent of them completed the
survey. Totally, 30 parents of children with TS participated in phase II of this study. One parent
took the survey three times for three different children with TS. Therefore, 32 surveys were
submitted, a response rate of 4%. This study identified 32 children with TS between the ages of 7
and 17 years (M = 11.74, SD = 2.84). Of these children, the majority were male (n = 23, 71.9%)
and white (n = 28, 87.5%), and lived with both parents (n = 27, 84.4%). Most of the
participating families had an annual household income of $75,000 or more (n = 27, 62.5%), and
one-half of the mothers (n = 16, 50.0%) and the majority of the fathers (n = 19, 59.4%) had an
40
education level equivalent to or higher than a two-year college degree. Table 1 summarizes the
demographic characteristics of the children with TS and participating families.
Although the signs and symptoms of TS could appear between the ages of 1 and 10 years,
the participating parents reports showed that the majority of the children experienced onset of TS
signs and symptoms at the age of 4, 5, and 6 years (n = 7, 21.9% for each age group), and
received a diagnosis of TS at the age of 7 or later (n = 24, 75.0%). The diagnosis peaked at the
age of 7 (n = 11, 34.4%) and the majority of the children were diagnosed by a neurologist (n =
24, 75.0%) and had taken medications for tic or non-tic symptoms at some point in their lives (n
= 27, 84.4%). Nearly all the children showed signs and symptoms of at least one comorbid
disorder (n = 31, 96.9%). The major comorbid symptoms experienced by the children were the
symptoms of ADHD (n = 24, 75.0%), OCD (n = 24, 75.0%), anxiety disorders (n = 22, 68.8%),
and sensory processing disorder (n = 17, 53.1%), although not all children had formal diagnoses
of these comorbid disorders. Table 2 summarizes the diagnosis-related characteristics of the
children; Appendix N lists the medications that had been prescribed for the children and the
parents comments on the positive and negative effects of the medications; and Table 3
summarizes the diagnosed or suspected comorbidities experienced by the children.
Functional Performance during a Waxing Period
As reported by the parents, during a waxing period, the children with TS experienced
functional limitations in all occupations, body functions, and performance skills, with varied
frequencies and degrees of severity (Table 4). This included occupations of activities of daily
living (ADLs), instrumental activities of daily living (IADLs), rest and sleep, education, play,
and social participation. In the occupations of ADLs and IADLs, more than one-third of the
children experienced difficulty in swallowing/eating (n = 13, 40.6%), personal hygiene and
41
grooming (n = 12, 37.5%), toileting (n = 12, 37.5%), toilet hygiene (n = 11, 34.4%), and
communication management (n = 11, 34.4%). The majority of the children experienced
difficulty with the occupations of rest and sleep, with 59.4 percent (n = 19) experiencing
difficulty with rest and 71.9 percent (n = 23) experiencing difficulty with sleep. In the
occupation of education, most of the children experienced difficulty in handwriting (n = 24,
75.0%) and one-half of them in reading (n = 16, 50.0%) and math (n = 16, 50.0%). Also, more
than one-third experienced difficulty in extracurricular activities (n = 15, 46.9%) and physical
education (n = 11, 34.4%). In the occupation of play, more than one-third of them experienced
difficulty in free play (n = 12, 37.5%). In the occupation of social participation, the majority of
the children had difficulty participating in organized activities such as scouting, camping, and
recreational activities that are sponsored by community organizations (n = 17, 53.1%). Also,
one-half of the children experienced difficulty in social participation, either in the community (n
= 16, 50.0%), with family (n = 16, 50.0%), or with peers or friends (n = 16, 50.0%). Table 4
summarizes the functional limitations in occupations experienced by the children with TS during
a waxing period.
Specific body functions and performance skills were selected from the OTPF-III and
categorized into mental, sensory, muscle, and movement functions (Table 5). The majority of
the children experienced difficulty in all the elements of mental functions, including emotional
coping (n = 29, 90.6%), attention (n = 28, 87.5%), sequencing (n = 28, 87.5%), control of
thoughts (n = 27, 84.4%), energy and drive (n = 27, 84.4%), control of behaviors (n = 23, 71.9%),
higher level cognitive functions (n = 21, 65.6%), and memory (n = 21, 65.6%). In the area of
sensory functions, difficulty processing tactile input was the most commonly experienced
functional limitation (n = 18, 56.3%). Also, one-half of the children had difficulty processing the
42
sensations of pain (n = 16, 50.0%) and more than one-third had difficulty in proprioception (n =
12, 37.5%). Regarding the area of muscle functions, few children encountered difficulty with
either muscle strength (n = 6, 18.8%) or muscle endurance (n = 9, 28.1%). In the area of
movement functions, the majority of the children experienced difficulty suppressing unwanted
movements/tics (n = 28, 87.5%) and controlling voluntary movements (n = 19, 59.4%). Also,
gait patterns were affected during a waxing period in about one-third of the children (n = 11,
34.4%). Table 5 summarizes the functional limitations in body functions and performance skills
experienced by the children with TS during a waxing period.
Differences in Functional Performance Before, During, and After a Waxing Period
Within-group analysis using Friedman ANOVA with Bonferroni correction determined if
there was a significant difference in the difficulty scores of functional performance in each
occupation, body function, and performance skill before, during and/or after a waxing period.
With regard to occupations, the childrens functional performance in rest (2 = 18.54, p < .001),
handwriting (2 = 21.41, p < .001), and social participation in the community (2 = 14.22, p
= .001) showed a significant difference in difficulty scores before, during and/or after a waxing
period. Concerning body functions and performance skills, the childrens functional
performance in all the elements of mental functions were significantly different in difficulty
scores, including control of thoughts (2 = 17.08, p < .001), control of behaviors (2 = 21.96, p
< .001), higher level cognitive functions (2 = 13.27, p = .001), attention (2 = 22.43, p < .001),
memory (2 = 19.50, p < .001), sequencing (2 = 22.43, p < .001), emotional coping (2 =
25.62, p < .001), and energy and drive (2 = 25.24, p < .001), before, during and/or after a
waxing period. Touch functions in the area of sensory functions (2 = 15.74, p < .001) as well
as control of voluntary movements (2 = 19.60, p < .001) and ability to suppress unwanted
43
movements/tics (2 = 31.90, p < .001) in the area of movement functions were also significantly
different in difficulty scores before, during and/or after a waxing period.
Based on the results of pairwise contrasts, the children with TS were found to experience
significantly worse functional performance during a waxing period compared with the waning
periods occurring before or after the waxing period in control of behaviors, sequencing,
emotional coping, energy and drive, and ability to suppress unwanted movements/tics (Table 6
and 7). Compared with the waning period occurring before a waxing period, the children also
experienced worse functional performance during a waxing period in memory (Table 6 and 7).
In addition, compared with the waning period occurring after a waxing period, worse functional
performance during a waxing period were found in rest, handwriting, and control of voluntary
movements (Table 6 and 7). Of note is that although a significant difference in difficulty scores
of functional performance before, during and/or after a waxing period was found in social
participation in the community, control of thoughts, higher level of cognitive functions, attention,
and touch functions using the Friedman ANOVA, significant pairwise differences were not
revealed in contrasts analysis, making it difficult to speculate on which time periods were
significantly different. Table 6 displays the differences in the childrens functional performance
difficulty scores in occupations and Table 7 body functions and performance skills before, during
and after a waxing period.
Predictors of Functional Limitations during a Waxing Period
Logistic regression was used to reveal which demographic and health factors (i.e., age,
gender, and the four major comorbid symptoms experienced by the children, the symptoms of
ADHD, OCD, anxiety disorders, and sensory processing disorder) are strong predictors for
functional limitations in each occupation, body function, and performance skill during a waxing
44
period. However, the odds ratios for a few items were overestimated, for example, the odds of
the children with the comorbid sensory processing problems experiencing difficulty with taste
during a waxing period was 969284903.25 times more than those without the comorbid sensory
processing problems, although the results were not significant (n = 6, p = .998), which indicated
the small sample size in this study may bias the results of logistic regression (Nemes, Jonasson,
Genell, & Steineck, 2009) and thus the results should be interpreted with caution. Table 8
summarizes the results of logistic regression for occupations, body functions and performance
skills.
The odds ratio of experiencing functional limitations in the occupation of social
participation in the community during a waxing period in children with TS who had the
comorbid anxiety symptoms compared to those who did not have the comorbid anxiety
symptoms was 14.4 (p = .024) and the odds ratio of experiencing functional limitations in the
occupation of rest during a waxing period was 5.8 (p = .036). The odds ratio of experiencing
functional limitations in toilet hygiene during a waxing period in children with TS who had the
symptoms of sensory processing disorder compared to those who did not have the symptoms of
sensory processing disorder, was 18 (p = .012), the odds ratio of experiencing functional
limitations in toileting during a waxing period was 8.4 (p = .020), and the odds ratio of
experiencing functional limitations in free play during a waxing period was 5.1 (p = .046).
Furthermore, with a one-year increase in age, the odds of experiencing difficulty processing
tactile input during a waxing period increased by 1.4 times (p = .033), and the odds of
experiencing difficulty processing pain during a waxing period also increased by 1.4 times (p
= .032). In addition, with a one-year increase in age, the odds of the children experiencing
functional limitations in communication management during a waxing period increased by 2
45
times (p = .010) and as age explained the functional limitations in communication management
first, the odds of experiencing functional limitations in communication management during a
waxing period among those who had the OCD symptoms decreased by 18.5 times (p = .045).
However, the OCD symptoms themselves were not strong predictors of experiencing functional
limitations in communication management during a waxing period. As for the demographic
factor of gender, the odds of experiencing functional limitations in handwriting performance
during a waxing period in male children compared to female children was 7.9 (p = .024).
Occupational Therapy Services for Children with Tourette Syndrome
The majority of the children had never received OT services. Only ten (31.3%) of them
had ever received OT services at some point in their lives. Among those who had ever received
OT services, the major reasons for an OT referral included concerns for handwriting (n = 8,
80.0%), fine motor skills (n = 7, 70.0%), organization (n = 7, 70.0%), and sensory processing (n
= 7, 70.0%). Gross motor concerns were a reason for an OT referral among one-half of the
children who had ever received OT services (n = 5). On the other hand, although about 90
percent of the children in this study experienced difficulty with attention (n = 28, 87.5%, see
Table 5), it was not a major reason for receiving OT services (n = 4, 40.0%, see Table 9). In
addition, only two children (20%) were referred to OT services due to concerns for tic
management and time management, and only one (10%) was referred for problems with visual
perception as well as social and coping skills.
One-half of the parents of the children who had ever received OT services (n = 5)
considered OT services beneficial for addressing the concerns for which their children were
referred to OT services, and one-fifth of them (n = 2) thought the services were helpful for
addressing some of the concerns but not all of them. Table 9 summarizes the responses
46
regarding OT services for the children with TS and Append O listed the parents comments on
the OT services their child received.
Occupational Therapy Services and Functional Performance during a Waxing Period
The Mann-Whitney U test was performed to determine if the childrens functional
performance in each occupation, body function, and performance skill during a waxing period
was significantly different between those who had ever received OT services and those who
never had. During a waxing period, functional performance in almost all occupations, body
functions, and performance skills showed no significant differences related to receiving OT
services or not, except for math. Math performance during a waxing period in the children who
had ever received OT services was significantly better than those who had never received OT
services (p = .023). Table 10 summarizes the comparison of functional performance in
occupations and Table 11 body functions and performance skills between the children who had
ever received OT services and those who had never received OT services.
Discussion
This study is the first to comprehensively delineate the functional limitations experienced
by children with TS in different occupations, body functions, and performance skills outlined in
the domain of the OTPF-III. Using a parent survey, functional limitations in 32 children with TS
under the age of 18 were reported. The implications for OT practice were also discussed.
Consistent with the prevalence reports found in previous studies (CDC, 2013; Hirschtritt
et al., 2015; Ludolph et al., 2012; Robertson, 2012), the majority of the children were white and
male. Also consistent with the information released by the TSA (McNaught, 2010), the
childrens TS symptoms were reported to primarily emerge between the ages of 4 and 6 years.
High rates of comorbid disorders in this study, especially ADHD, OCD, and anxiety disorders,
47
were reported by the parents, and they are congruent with previous research results (Hirschtritt et
al., 2015). Additionally, more than one-half of the children were reported to exhibit signs and
symptoms of sensory processing disorder, which is consistent with previous findings in the
study conducted by Jewers et al. (2013) that more than one-half of their overall sample of 75
children with TS experienced sensory processing problems.
Functional Performance in Children with Tourette Syndrome
Children with TS in this study were reported to demonstrate functional limitations in all
the occupations, body functions, and performance skills listed on the parent survey during a
waxing period, with individual variances in types and severity of the limitations experienced.
During a waxing period, the majority of the children experienced difficulty in rest, sleep,
handwriting, participating in organized activities, mental functions (i.e., emotional coping,
attention, sequencing, control of thoughts, energy and drive, control of behaviors, higher level
cognitive functions, and memory), touch functions, suppressing unwanted movements/tics, and
controlling voluntary movements; one-half of them had difficulty in reading, math, social
participation, and pain functions; and more than one-third of them encountered difficulty in
swallowing/eating, personal hygiene and grooming, toileting and toilet hygiene, communication
management, extracurricular activities, physical education, free play, proprioception, and gait
patterns.
Previous findings have addressed some of the functional problems mentioned in this
study in children with TS, although the sample sizes used in most of the studies were small. The
high frequency (71.9%) of sleep problems found in this study is consistent with the result of a
previous study revealing a high frequency (65%) of diagnosable sleep problems that were
48
independent of the comorbid ADHD symptoms in a sample of 123 children with TS using a
questionnaire adapted from another study (Ghosh et al., 2014).
Regarding deficits in mental functions, deficits in sustained attention and impulsive
control and problems with executive functions such as working memory, inhibition, and shift
have been reported respectively in previous studies using a sample of 74 and a sample of 11
children with TS (Schwam, King, & Greenberg, 2015; Sherman, Shepard, Joschko, & Freeman,
1998). Schwam et al. (2015) and Sherman et al. (1998) also found that the comorbid symptoms
of ADHD and/or OCD are correlated with the mental function deficits seen in their samples of
children with TS. As in this study, the mental function deficits are presented in a more
comprehensive way and the high frequency of the deficits reported in the children is consistent
with previous findings.
More than one-half of the children in this study were reported to encounter problems with
touch functions and one-half with pain functions, consistent with the result of the Jewers et al.
(2013) study that more than one-half of their sample of 75 children with TS experienced sensory
processing problems with tactile sensitivity. However, contrary to their findings that the
majority of their sample experienced difficulty with auditory filtering (Jewers et al., 2013), only
two of (6.3%) 32 children in this study encountered problems with hearing functions. It is
possible that the participants thought the survey question meant hearing loss or central auditory
processing disorder.
Additionally, Dykens et al. (1990) found weaknesses in math performance and social
interaction skills regarding interpersonal skills, the use of play/leisure time, and coping skills in a
sample of 30 children with TS using the Reading, Arithmetic, and Written Language subtests of
the Woodcock-Johnson Test of Achievement. Consistent with the findings of the Dykens et al.
49
(1990) study, difficulty in math performance and social participation were reported in one-half of
the sample during a waxing period. This study also identified that more than one-half of the
sample experienced difficulty with participation in organized activities sponsored by community
organizations during a waxing period. However, Dykens et al (1990) also found that children
with TS demonstrated relative strength in reading performance and ADL skills as measured by
the Vinland Adaptive Behavior Scales. Therefore, problems with reading and ADL performance
seen in one-half of the sample during a waxing period in this study do not agree with the findings
of Dykens et al (1990).
This study is the first to identify the frequency of problems with rest, handwriting, play,
and movement functions in children with TS. Rest is considered different from sleep and is
defined as engaging in quiet and effortless actions that interrupt physical and mental activity,
resulting in a relaxed state (Nurit & Michal, 2003, p.227). Studies exploring the meaning and
methods of rest are scant (Nurit & Michal, 2003), not to mention studies describing the patterns
of rest problems in children with TS. In this study, approximately 60 percent of the children
with TS had difficulty with rest during a waxing period. Currently there is no study addressing
the handwriting performance in children with TS except for a small number of case reports
describing the complex motor tics interfering with the handwriting performance in adults and
children with TS (Cavanna et al., 2011; Mitchell & Cavanna, 2013). This study is the first to
identify that 75 percent of the children with TS encountered difficulty with handwriting during a
waxing period. As for deficits in movement functions, this study is also the first to report that
approximately 90 percent of the children with TS experienced difficulty suppressing tics, 60
percent with control of voluntary movements, and 30 percent with gait patterns during a waxing
period.
50
Factors Influencing Functional Performance in Children with Tourette Syndrome

In addition to the descriptive findings, the Friedman ANOVA with Bonferroni correction
revealed significant differences in difficulty scores of functional performance in rest,
handwriting, social participation in the community, control of thoughts, control of behaviors,
higher level cognitive functions, attention, memory, sequencing, emotional coping, energy and
drive, touch functions, control of voluntary movements, and ability to suppress unwanted
movements/tics among the sample of children with TS before, during, and/or after a waxing
period. The pairwise contrast analyses further identified significant greater difficulty during a
waxing period in rest, handwriting, control of behaviors, memory, sequencing, emotional coping,
energy and drive, control of voluntary movements, and ability to suppress unwanted
movements/tics. These indicate that the daily functioning of children with TS in these
occupations, body functions, and performance skills, which include the occupations of rest and
education, mental functions/process skills, and movement functions/motor skills, may fluctuate
as the children with TS move from waning to waxing periods and back. Although significant
differences in difficulty scores of functional performance were found using omnibus tests in
social participation in the community, control of thoughts, higher level of cognitive functions,
attention, and touch functions, the results of pairwise contrast analyses were not significant.
However, a trend towards greater difficulty during a waxing period was observed in these
functional areas (Table 6 and 7), while the sample size may be too small to reveal a significant
difference in functional performance before, during, and/or after a waxing period. These
findings are consistent with previous findings which demonstrated the positive correlations
between tic severity and the functional limitations in fine motor coordination and executive
functions (Bloch et al., 2006; Schwam et al., 2015) and expand the understanding of the possible
51
influence of tic severity on other occupations, body functions, and performance skills. However,
future studies using assessment tools with good reliability and validity such as the Yale Global
Tic Severity Scale, a measurement of the severity of motor and vocal tics (Leckman et al., 1989),
are needed to establish the relationships between tic severity and specific functioning areas.
Additionally, no significant differences in functional performance difficulty scores were found
between the waning periods occurring before and after the waxing periods in any occupations,
body functions, and performance skills, indicating that, overall, the children with TS returned to
their baseline functioning after a waxing period and there is not a permanent increase in
symptoms.
Previous studies have found positive correlations between the comorbid ADHD
symptoms and several functional limitations in children with TS. For example, when the ADHD
symptoms are present, the childrens problems with sleep, peer relationships, executive
functioning, math, sensory processing, and fine motor coordination are more severe (Bawden et
al., 1998; Buse et al., 2012; Huckeba, et al., 2008; Jewers et al., 2013; Schwam et al., 2015).
However, in this study, the ADHD symptoms were not a predictor of the experience of
functional limitations in any occupations, body functions, and performance skills during a
waxing period. On the other hand, the influence of the comorbid anxiety symptoms appeared to
be a related factor for functional performance of the children with TS in this study, although
previous studies did not find this relationship, possibly due to small sample sizes. For example,
Bawden et al. (1998) could not identify the relationship between the anxiety symptoms and peerrelationship problems because of the small number of children with TS participating in their
study, who also had a comorbid diagnosis of an anxiety disorder (n = 4, 16%). Nonetheless, in
the current study, nearly 70 percent (n = 22, 68.8%) of the children with TS demonstrated the
52
symptoms of anxiety disorders, although not all of them were diagnosed, and the comorbid
anxiety symptoms were found to be able to predict the experience of functional limitations in the
occupation of social participation in the community and the occupation of rest during a waxing
period. The anxiety symptoms themselves are related to these occupations for the following
reasons. One of the major symptoms of generalized anxiety disorder is restlessness, the anxiety
symptoms might be triggered by social situations, and the anxiety symptoms may further impair
social functioning (APA, 2013). On the other hand, Silva, Munoz, Barickman, and Friedhoff
(1995) identified that being upset or anxious was the most common antecedent to the increase of
tic severity in a sample of 14 children with TS. In addition, Himle et al. (2014) suggested that
anxiety may be a mediator between some environmental factors and the increase of tic severity
in that some antecedents or consequences, whether tic-related or not, may trigger anxiety in
children with TS and further increase their tic severity and impact their functional performance.
Although a systematic review conducted by Koenig and Rudney (2010) has revealed that
children and adolescents with sensory processing problems may experience functional
performance deficits in all occupations, studies examining the relationships between sensory
processing and daily functional performance have been focused mainly on children with autism
spectrum disorders and children with developmental coordination disorder (Koenig & Rudney,
2010). This study is the first to identify the potential influence of the sensory processing
problems on functional performance in children with TS. In this study, the symptoms of sensory
processing disorder were identified as the predictor of experiencing functional problems in the
occupations of ADLs, including toileting and toilet hygiene, and play (free play) during a waxing
period. However, as suggested by Koenig and Rudney (2010), studies discussing the subtypes of
sensory processing problems (i.e., sensory modulation disorder, sensory-based motor disorder,
53
and sensory discrimination disorder) and their impact on functional performance in children with
different diagnoses need to be conducted in the future. This type of study also needs to be
conducted for children with TS.
In our study, age was identified as the predictor of experiencing difficulty processing
tactile input and pain as well as difficulty with communication management (i.e., using different
devices and equipment such as telephones, computers, tablets, emergency systems, and so on to
communicate with others; AOTA, 2014) during a waxing period. The findings of the influence
of age on the children sensory processing problems are contrary to previous findings that age
was not related to the sensory processing problems in a sample of 75 children with TS (Jewers et
al., 2013). It is possible that as the children grow older, everyday occupations may become more
complex and challenging in terms of touch sensations. For example, classroom activities may
involve the childrens participation in crafts or texture play and social situations may require
children staying close to others such as lining up or playing with others, which make the
childrens difficulty in processing tactile sensations more obvious and problematic to the parents.
Also, age and communication management may be correlated in that the older children may have
more chances to use different modalities to communicate in everyday life and the childrens
difficulty with communication management may thus become easier to be noticed. However, to
date, few studies have addressed the communication and communication-management problems
in children with TS.
Gender was found to predict the experience of functional limitations in handwriting
performance during a waxing period among the children in this study; the male children were
more likely to experience handwriting performance deficits compared to the female children.
This result may be explained by previous findings of a study that in adolescence, females
54
experience less severe TS symptoms than males (Lichter & Finnegan, 2015) so that during a
waxing period, the female childrens handwriting performance deficits are not as severe as the
male childrens handwriting performance deficits. However, no literature has described gender
difference in handwriting performance in children with TS. Therefore, although this result may
reflect gender difference in the influence of tic severity on the childrens handwriting
performance, it is also possible that the gender difference reflect a sampling bias and that future
studies need to recruit more female children.
The exploration of OT involvement and functional abilities during a waxing period
produced interesting results worthy of further study. Congruent with previous findings that OT
services for school-age children with emotional disturbances often focus on motor and sensory
problems rather than behavioral problems (Case-Smith & Archer, 2008), fine motor skill
problems, including handwriting problems, and sensory processing problems were two of the
major reasons for the children with TS in this study to receive an OT referral. Among those who
had received OT services, difficulty levels of functioning in all occupations, body functions, and
performance skills except for math performance during a waxing period showed no significant
differences related to receiving OT services or not, indicating that the 10 children who had ever
received OT services were not experiencing symptoms that were significantly worse than those
who had never receive OT services during a waxing period. Only math performance during a
waxing period was significantly better among those who had ever received OT services as
compared to those who had never received OT services. Although these findings might indicate
that OT services helped improve the math performance and maintain the functional performance
in the rest of the occupations, body functions, and performance skills among those whose
functional limitations caused clinical concerns, the relationship between OT services and specific
55
functional performance cannot be determined due to the retrospective survey in this study. In
order to know if OT services made a difference in functional performance of children with TS,
the childrens functioning and skills in a specific area would need to be evaluated using
standardized assessment tools and the change of functional performance deficits from a waxing
period to the waning period occurring after the waxing period would need to be examined. The
amount of time it took to reach the waning period from a waxing period and if the children
showed improvement during the waxing period would also need to be examined.
Strengths, Limitations, and Future Research
Strength of this study was the parent survey used in data collection, which was adapted
from the previous survey used by Bhattacharjya (2013) and modified based on the OTPF-III
(AOTA, 2014). A content validity of .98 had been established before the participant recruitment
process began; hence this study has sufficient psychometric properties. Since the OTPF-III
comprehensively describes the essential constructs involved in OT practice and the survey items
conform to the terminology used in the OTPF-III, the study results are directly relevant to OT
practice and capable of providing basic, accessible knowledge of the functional performance
difficulty in occupations, body functions, and performance skills in children with TS.
This study also has some limitations related to the sampling process and the retrospective
survey used in this study. The parent survey used nominal and ordinal scales to gather data,
which led to the use of descriptive and non-parametric statistics for data analysis and subsequent
reduction of statistical power. The use of a convenience sampling method may lead to a
sampling bias in which the majority of the children in this study were white and male. Also, the
participants were all recruited from specific geographic regions of New York State and most of
them are well-educated people with comfortable incomes, making it difficult to generalize the
56
study results to the whole population of children with TS, especially to minority groups. The
low survey response rate and subsequent small sample size also make generalization difficult.
Since the low survey response rate might result from the length of the survey and the lack of
incentives, future studies should consider recruiting a larger and more diverse sample through
condensing the parent survey to a shorter version, offering incentive payments to encourage
participation, and involving different community-based settings such as clinics or school systems.
Another concern is the recall bias resulting from the retrospective nature of the parent survey. In
order to avoid the recall bias, future studies may consider developing a child survey asking
childrens subjective experience, using standardized assessment tools with good reliability and
validity to measure their functional performance, or using a prospective cohort design with a
daily log. A prospective cohort study may be helpful in determining the factors affecting the
functional performance in children with TS, such as tic severity, comorbid disorders,
medications, etc. Future studies examining the effectiveness of OT intervention using different
treatment approaches other than a traditional sensorimotor one to address other functional
limitations commonly seen in children with TS such as rest and sleep, social participation, and
mental function problems are still needed, and this type of study may consider distinguishing
children with TS who demonstrate the comorbid anxiety symptoms from those who do not since
the comorbid anxiety symptoms were found to be able to predict the functional limitations in
several occupations, body functions, and performance skills during a waxing period in this study.
Implications for Occupational Therapy Practice
Through the use of a parent survey based on the OTPF-III, this study provides a
comprehensive description of the daily functional performance in children with TS. Potential
factors that may influence the functional performance in children with TS, such as tic severity,
57
comorbid symptoms of anxiety disorders and sensory processing disorder, age, gender, and OT
services, are also described.
In this study, children with TS demonstrated varied degrees of difficulty in all areas of
functional performance in their daily life, including rest and sleep, education, play, social
participation, mental functions, sensory functions, and movement functions when tics wax. In
addition, when tics wax, their daily functioning in several functional performance areas,
including rest, handwriting, most components of mental functions, movement functions
including control of voluntary movements and ability to suppress tics, are worse than when tics
wane, with mental functions being influenced most frequently. The comorbid anxiety symptoms
may predict the experience of functional limitations in the occupations of rest and social
participation, and the comorbid sensory processing problems may predict the experience of
functional limitations in the occupations of ADL and play during a waxing period. Therefore,
OT services using traditional approaches such as the sensorimotor or motor learning framework
may only partially meet the need of children with TS and a compensatory approach aimed at
changing environmental demands and an approach aimed at teaching tic management and
decreasing anxiety such as the CBIT program or a cognitive-behavioral framework may also
benefit children with TS. Antecedents and consequences of the increase of tic severity, anxiety,
and behavioral problems related to sensory processing challenges should be identified in order to
improve or maintain the functional performance and participation in children with TS. Further
research providing more evidence for the use of different approaches, especially approaches
addressing the problems that are not frequently addressed such as rest and sleep, social
participation, and play and leisure in children with TS are still required.

Conclusion
The study results identify an important role for OT during evaluation and intervention
process for children with TS and may guide OT practice and provide basic knowledge for
parents and other related professionals to support the daily functioning of children with TS.
Future research providing evidence for the use of different approaches, especially those
addressing tic management and anxiety symptom reduction is needed.
58
59
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circuits that generate tics in Tourettes syndrome. American Journal of Psychiatry,
168(12), 1326-1337.
Wilhelm, S., Deckersbach, T., Coffey, B., Bohne, A., Peterson, A. L., & Baer, L. (2003). Habit
reversal versus supportive psychotherapy for Tourettes disorder: A randomized
controlled trial. American Journal of Psychology, 160, 1175-1177.
doi:10.1176/appi.ajp.160.6.1175
Woods, D. W., Piacentini, J. C., Chang, S. W., Deckersbach, T., Ginsburg, G. S., Peterson, A.
L.,Wilhelm, S. (2008). Managing Tourette syndrome: A behavioral intervention for
children and adults. New York: Oxford University Press.
Woods, D. W., Piacentini, J. C., & Walkup, J. T. (n.d.). Comprehensive behavioral intervention
for tics. Retrieved from http://www.tsa-usa.org/Medical/CBIT_Explained.html
World Health Organization (2001). International Classification of Functioning, Disability and
Health. Geneva: Author.
68
Table 1
Demographic Characteristics of the Children with Tourette Syndrome and Participating Family
Characteristic of the children
Age (years)
Age of TS diagnosis (years)
Age of showing TS signs (years)
Gender
Male
Female
Race
White
Multiracial
African American/Black
Characteristic of the participating family
Number of parents living with the child
2
1
Combined household yearly income
Less than $25,000
$25,000-49,999
$50,000-74,999
$75,000-99,999
$100,000-149,999
$150,000-199,999
$200,000 and more than $200,000
Missing
n
31
31
31
%
96.9
96.9
96.9
23
9
71.9
28.1
28
3
1
87.5
9.4
3.1
27
5
84.4
15.6
2
6
4
5
7
5
2
1
Mother
6.3
18.8
12.5
15.6
21.9
15.6
6.3
3.1
M
11.74
7.59
5.03
SD
2.84
1.72
1.91
Father
n
%
n
Parent education level
High school/GED
3
9.4
8
Some college
13
40.6
5
2-year college degree
2
6.3
5
7
21.9
8
Masters degree
5
15.6
1
Doctoral degree
2
6.3
3
MD/DO
0
0.0
1
N/A
0
0.0
1
Note. N = 32. n = Frequency. % = Percentage. M = Mean. SD = Standard deviation.
%
25.0
15.6
15.6
25.0
3.1
9.4
3.1
3.1
69
Table 2
Diagnosis-related Characteristics of the Children with Tourette Syndrome
Showing TS signs
n
%
Diagnosed with TS
n
%
Characteristic
Age in years
1
2
6.3
0
0.0
2
1
3.1
0
0.0
3
2
6.3
0
0.0
4
7
21.9
1
3.1
5
7
21.9
1
3.1
6
7
21.9
6
18.8
7
4
12.5
11
34.4
8
1
3.1
4
12.5
9
0
0.0
3
9.4
10
1
3.1
4
12.5
11
0
0.0
2
6.3
Type of physician giving TS diagnosis
Neurologist
24
75.0
Neurological physicians assistant
1
3.1
Psychiatrist
2
6.3
Psychologist
2
6.3
a
Multiple professionals
3
9.4
Medication prescribed for tic or non-tic symptomsb
Yes
27
84.4
No
5
15.6
Note. N = 32. n = Frequency. % = Percentage.
a
Some of the children was diagnosed by multiple professionals including a neurologist,
pediatrician, psychiatrist, and/or psychologist. bMedications prescribed for tic or non-tic
symptoms that had been prescribed for the children with TS and the parents comments on the
effects of the medications are displayed in Appendix N.
70
Table 3
Comorbid Disorders Experienced by the Children with Tourette Syndrome
Diagnosed
Suspected
Comorbidity
n
%
n
%
ADHD
19
59.4
5
15.6
Obsessive-compulsive disorder
15
46.9
9
28.1
Anxiety disorder
14
43.8
8
25.0
Sensory processing disorder
9
28.1
8
25.0
Developmental delays
5
15.6
1
3.1
Autism spectrum disorder
4
12.5
3
9.4
Oppositional defiant disorder
2
6.3
1
3.1
Attention deficit disorder
2
6.3
0
0.0
Central auditory processing disorder
1
3.1
0
0.0
Dysgraphia
1
3.1
1
3.1
Non-verbal processing disorder
1
3.1
0
0.0
Pragmatic communication disorder
1
3.1
0
0.0
a
Other
4
12.5
2
6.3
Note. N = 32. n = Frequency. % = Percentage. ADHD = Attention-deficit/hyperactivity
disorder.
a
Other medical conditions experienced by the children with TS specified by their parents
included dystonia and epilepsy, conversion disorder and chronic daily headaches, neurological
rage, horizontal nystagmus, eye torticollis, asthma, chronic sinusitis, executive dysfunction, ,
focusing and peripheral vision difficulties, insulin resistance, and impulse control issues.
71
Table 4
Functional Limitations in Occupations during a Waxing Period
Occupation
Home-related occupations
Sleep
Rest
Swallowing/eating
Personal hygiene and grooming
Toileting
Communication management
Toilet hygiene
Dressing
Sexual activity
Bathing/showering
Community mobility
Feeding
Functional mobility
Education-related occupations
Handwriting
Reading
Math
Extracurricular activities
Physical education
Occupations related to social life
Organized activities
Social participation in the community
Social participation with family
Social participation with peers/friends
Free play
%
23
19
13
12
12
11
11
10
9
6
6
5
5
71.9
59.4
40.6
37.5
37.5
34.4
34.4
31.3
28.1
18.8
18.8
15.6
15.6
24
16
16
15
11
75.0
50.0
50.0
46.9
34.4
17
16
16
16
12
53.1
50.0
50.0
50.0
37.5
72
Table 5
Functional Limitations in Body Functions and Performance Skills during a Waxing Period
Body function and performance skill
Mental functions
Emotional coping
Attention
Sequencing
Control of thoughts
Energy and drive
Control of behaviors
Higher level cognitive functions/thinking
Memory
Sensory functions
Tactile/touch
Pain
Proprioception/knowing where body is in space
Smell
Taste
Temperature
Vestibular/sense of moving
Vision/seeing
Hearing
Muscle and movement functions
Ability to suppress unwanted movements (tics)
Smoothness/control of voluntary movements
Gait/walking patterns
Muscle endurance
Muscle strength
%
29
28
28
27
27
23
21
21
90.6
87.5
87.5
84.4
84.4
71.9
65.6
65.6
18
16
12
7
6
6
5
5
2
56.3
50.0
37.5
21.9
18.8
18.8
15.6
15.6
6.3
28
19
11
9
6
87.5
59.4
34.4
28.1
18.8
73
Table 6
Differences in Functional Performance in Occupations Before, During, and After a Waxing Period
Occupation
Bathing/showering (n =6)
Toileting (n = 12)
Toilet hygiene (n = 11)
Dressing (n = 10)
Swallowing/eating (n = 13)
Feeding (n = 5)
Functional mobility (n = 5)
Personal hygiene and grooming (n = 11)
Sexual activity (n = 8)
Communication management (n = 11)
Community mobility (n = 6)
Rest (n = 18)
Sleep (n = 22)
Math (n = 16)
Reading (n = 16)
Handwriting (n = 24)
Physical education (n = 11)
Extracurricular activities (n = 14)
Free play (n = 12)
Organized activities (n = 17)
Social participation in the community (n = 16)
Social participation with family (n = 16)
Social participation with peers/friends (n = 15)
Note. df = 2. M = Mean. SD = Standard deviation.
Before
M
SD
During
M
SD
After
M
SD
Contrasts
2.00
2.42
2.18
2.30
2.38
1.80
1.80
2.45
2.25
2.00
2.17
2.50
2.55
1.10
0.79
0.75
1.06
0.96
0.45
0.45
1.04
0.71
0.63
0.41
0.92
0.91
2.50
2.50
2.55
2.90
2.85
2.60
2.80
2.91
2.50
2.73
2.50
3.06
2.91
1.05
0.80
0.69
0.88
0.99
0.55
0.45
0.83
0.54
0.65
0.55
0.73
0.87
2.17
2.08
2.45
2.50
2.31
2.00
1.80
2.45
2.25
2.09
2.50
2.33
2.41
0.98
0.90
0.69
0.97
0.95
0.71
0.45
1.04
0.71
0.83
0.55
0.97
0.85
3.71
5.20
3.50
7.54
9.50
4.67
8.00
4.00
2.00
7.15
2.67
18.54
12.05
.156
.074
.174
.023*
.009**
.097
.018*
.135
.368
.028*
.264
<.001***
.002**
2.75
2.44
2.79
2.00
2.29
0.86
0.96
0.78
0.45
0.73
3.19
3.00
3.42
2.55
2.79
0.66
0.97
0.65
0.69
0.80
2.75
2.38
2.75
2.18
2.71
0.78
0.81
0.79
0.75
1.38
9.58
9.87
21.41
6.40
8.27
.008**
.007**
<.001**
.041*
.016*
D vs. A
2.58 0.79 3.08 0.67 2.75

2.53 0.87 3.00 0.87 2.59
2.44 0.89 2.94 0.68 2.44
2.69 0.70 2.75 0.78 2.44
2.93 0.80 3.13 0.64 2.93
*p < .05. **p < .01. ***p .001.
1.14
0.87
0.89
0.73
0.80
6.70
12.29
14.22
6.09
4.50
.035*
.002**
.001***
.048*
.105
D vs. A
74
Table 7
Differences in Functional Performance in Body Functions and Performance Skills Before, During, and After a Waxing Period
Before
M
SD
During
M
SD
Body function and performance skill

Mental functions
Control of thoughts (n = 26)
2.46 0.81 2.96 0.82
Control of behaviors (n = 23)
2.61 0.72 3.17 0.58
Higher level cognitive functions/thinking (n = 21)
2.48 0.81 2.90 0.63
Attention (n = 27)
2.48 0.58 3.00 0.56
Memory (n = 21)
2.14 0.79 2.76 0.77
Sequencing (n = 28)
2.50 0.84 3.04 0.84
Emotional coping (n = 28)
2.68 0.77 3.36 0.78
Energy and drive (n = 27)
2.59 0.75 3.30 0.72
Sensory functions
Vision/seeing (n = 5)
2.00 1.23 2.40 1.14
Hearing (n = 2)
3.00 0.00 3.50 0.71
Vestibular/sense of moving (n = 5)
2.60 1.34 3.20 1.10
Taste (n = 6)
2.17 1.17 2.83 0.98
Smell (n = 7)
2.86 0.90 3.14 0.69
Proprioception/knowing where body is in space (n = 12) 2.42 1.00 2.75 1.06
Tactile/touch (n = 18)
2.28 0.90 2.83 0.86
Pain (n = 16)
2.13 0.96 2.56 0.73
Temperature (n = 6)
1.67 0.52 1.83 0.41
Muscle strength (n = 6)
2.83 0.75 2.83 0.75
Muscle endurance (n = 9)
2.67 0.71 2.67 0.71
Smoothness/control of voluntary movements (n = 19)
2.37 0.76 2.95 0.78
Ability to suppress unwanted movements/tics (n = 28)
2.71 0.90 3.46 0.58
Gait/walking patterns (n = 11)
2.00 0.89 2.64 0.67
Note. df = 2. M = Mean. SD = Standard deviation. *p < .05. **p < .01. ***p .001.
After
M
SD
p
<.001***
<.001***
.001***
<.001***
<.001***
<.001***
<.001***
<.001***
2.50
2.52
2.52
2.52
2.29
2.54
2.57
2.63
0.91
0.67
0.93
0.64
0.78
0.84
0.79
0.63
17.08
21.96
13.27
22.43
19.50
22.43
25.62
25.24
2.00
3.00
2.80
2.17
2.71
2.33
2.33
2.06
1.67
1.23
0.00
1.30
1.17
1.11
1.07
0.91
0.85
0.52
2.00
.368
2.00
.368
3.71
.156
6.00
.050
4.67
.097
4.33
.115
15.74 <.001***
6.89
.032*
2.00
.368
2.83
2.56
2.21
2.68
2.00
0.75
0.88
0.79
0.86
0.89
2.00
.368
19.60 <.001***
31.90 <.001***
12.00
.002**
Contrasts
D vs. B & A
D vs. B
D vs. B & A
D vs. B & A
D vs. B & A
D vs. A
D vs. B & A
75
Table 8
Predictors of the Experience of Functional Limitations in Occupations, Body Functions, and
Performance Skills during a Waxing Period
Predictor
Age
Gender
ADHD
OCD
Anxiety disorder
SPD
Age
Gender
ADHD
OCD
Anxiety disorder
SPD
Toilet hygiene (n = 11)

B (S.E.)
p (OR)
2.89 (1.15)
.012*(18.00)
Social participation in
community (n = 16)
B (S.E.)
p (OR)
Toileting (n = 12)
B (S.E.)
p (OR)
2.12 (.91) .020*(8.36)

Rest (n = 19)
B (S.E.)
p (OR)
Free play (n = 12)

B (S.E.)
p (OR)
1.64 (.82)
Handwriting (n = 24)
B (S.E.)
p (OR)
2.07 (.91)
2.67 (1.18)
.024*(14.38)
1.76 (.84) .036*(5.83)
Communication
management (n = 11)
B (S.E.)
p (OR)
.70 (.27)
.010*(2.01)
Tactile/touch (n = 18)
B (S.E.)
p (OR)
.35 (.16) .033*(1.42)
.046*(5.14)
.024*(7.92)
Pain (n = 16)
B (S.E.)
p (OR)
.35 (.16) .032*(1.42)
Age
Gender
ADHD
OCD
-2.91 (1.45)
.045*(.054)
Anxiety disorder
SPD
Note. N = 32. ADHD = Attention-deficit/hyperactivity disorder. OCD = Obsessive-compulsive
disorder. SPD = Sensory processing disorder. B = Logistic regression coefficient. S.E. = Standard
error of the coefficient. OR = Odds ratio. The comorbidities (ADHD, OCD, anxiety disorders, and
SPD) were either formally diagnosed or suspected but not formally diagnosed.
*p < .05.
76
Table 9
Information about Occupational Therapy (OT) Services for the Children with Tourette Syndrome
OT Service Information
n
%
Receiving OT services (N = 32)
10
31.3
Reasons for OT services (n = 10)
Handwriting
8
80.0
Fine motor
7
70.0
Organization
7
70.0
Sensory processing
7
70.0
Gross motor
5
50.0
Attention
4
40.0
Tic management
2
20.0
Time management
2
20.0
Visual perception
1
10.0
Social skills and calming techniques
1
10.0
Benefit of OT servicesa (n = 10)
Yes
5
50.0
No
3
30.0
Partially beneficial
2
20.0
Note. n = Frequency. % = Percentage.
a
The parents comments on the OT services their child received were displayed in Append O.
77
Table 10
Comparison of Occupations during a Waxing Period by Receiving Occupational Therapy (OT)
Services or Not
OT services
Yes
Mean
Rank
No
Mean
Rank
Items
n
n
Bathing/showering
3
3.17
3
3.83
Toileting
5
6.00
7
6.86
Toilet hygiene
6
4.33
5
8.00
Dressing
5
5.20
5
5.80
Swallowing/eating
6
6.42
7
7.50
Feeding
2
2.75
3
3.17
Functional mobility
2
3.50
3
2.67
5
4.90
6
6.92
Sexual activity
5
4.90
3
3.83
Communication management
7
6.07
4
5.88
Community mobility
3
4.00
3
3.00
Rest
7
8.14 11
10.36
Sleep
7
13.07 15
10.77
Math
7
5.43
9
10.89
Reading
7
7.14
9
9.56
Handwriting
10
14.10 14
11.36
Physical education
5
5.50
6
6.42
6
5.50
9
9.67
Free play
5
6.10
7
6.79
Organized activities
6
9.00 11
9.00
5
8.90 11
8.32
6
7.25 10
9.25
4
10.25 11
7.18
Note. N = 32. n = Frequency. Higher scores indicated higher difficulty levels.
*p < .05, two-tailed.
3.50
15.00
5.00
11.00
17.50
2.50
2.00
9.50
5.50
13.50
3.00
29.00
41.50
.700
.755
.082
.841
.628
.800
.800
.329
.571
.927
.700
.425
.447
10.00
22.00
54.00
12.50
12.00
.023*
.351
.371
.662
.088
15.50
33.00
25.50
22.50
13.00
.755
1.000
.827
.428
.280
78
Table 11
Comparison of Body Functions and Performance Skills during a Waxing Period by Receiving
Occupational Therapy (OT) Services or Not
OT services
Yes
Mean
Rank
No
Mean
Rank
Items
n
n
Mental functions
Control of thoughts
9
13.89 17
13.29
Control of behaviors
8
10.50 15
12.80
Higher level cognitive functions/thinking
9
11.89 12
10.33
Attention
10
12.10 18
15.83
Memory
8
12.63 13
10.00
Sequencing
10
15.90 18
13.72
Emotional coping
9
12.94 19
15.24
Energy and drive
10
15.00 17
13.41
Sensory functions
Vision/seeing
0
0.00
5
3.00
Hearing
1
1.00
1
2.00
3
3.17
2
2.75
Taste
3
3.83
3
3.17
Smell
4
4.25
3
3.67
Proprioception/knowing where body is in space
6
8.42
6
4.58
Tactile/touch
8
8.75 10
10.10
Pain
4
6.75 12
9.08
Temperature
3
4.00
3
3.00
Muscle strength
4
3.38
2
3.75
Muscle endurance
5
5.70
4
4.13
Smoothness/control of movements
7
9.43 12
10.33
Ability to suppress unwanted movements/tics
7
14.71 21
14.43
4
5.50
7
6.29
Note. N = 32. n = Frequency. Higher scores indicated higher difficulty levels.
*p < .05, two-tailed.
73.00
48.00
46.00
66.00
39.00
76.00
71.50
75.00
.874
.466
.602
.265
.374
.524
.498
.639
0.00
2.50
3.50
5.00
6.50
34.00
17.00
3.00
1.000
.800
.700
.857
.065
.633
.446
.700
3.50
6.50
38.00
72.00
12.00
.800
.413
.773
.959
.788
Occupations
Activities of daily living*
Instrumental activities of daily living
Rest and sleep
Education
Work
Play
Leisure
Social participation
79
Client Factors
Values, beliefs, and spirituality
Body functions
Body structures
*Also referred to as basic activities of daily

living or personal activities of daily
living
Performance skills
Motor skills
Process skills
Social interaction skills
Performance patterns
Habits
Routines
Rituals
Roles
Context and Environment

Cultural
Personal
Physical
Social
Temporal
Virtual
Figure 1. Aspects of the domain of OT. This figure illustrates all aspects of the domain involved
in OT service delivery to support participation in daily life. Adapted from American
Occupational Therapy Association.(2014). Occupational therapy practice framework: Domain
and process (3rd ed.). American Journal of Occupational Therapy, 68(Suppl.1), S1S48.
Retrieved from http://dx.doi.org/10.5014/ajot.2014.682006
80
Appendix A
Finalized parent survey
Daily functioning of children with Tourette syndrome: A pilot study
Version Date: 02/12/2015
NOTE: The following questions were presented to the participants online immediately after
the participant read the informed consent form and consented to participate in the study.
1)
Are you at least 18 years of age?

Yes, I am 18 years of age or older.
No, I am not yet 18 years of age.
2)
Thank you for your interest in our study. Unfortunately, this survey is limited to
participants who are at least 18 years old. If you have questions about this or if your
qualifications change and you wish to participate, please reopen this survey or contact
Janice Tona, Ph.D., OTR at tona@buffalo.edu. Thank you for your time.
Submit
3)
Has your child been diagnosed with TS by a physician?

Yes
No
4)
Thank you for your interest in our study. Unfortunately, we are only able to study children
who have been diagnosed with TS by a physician. If your child is diagnosed with TS by a
physician in the future and you wish to participate in future research, please contact Janice
Tona, Ph.D., OTR at tona@buffalo.edu. Thank you.
Click here to exit survey
5)
Is your child who is diagnosed with TS under 18 years of age?
81
Yes, my child is younger than 18 years old

No, my child has already had his/her 18th birthday
6)
Thank you for your interest in our study. Unfortunately, we are only able to study children
diagnosed with TS who are under 18 years old. If you have questions about this or if you
wish to participate in future research targeted at young adults above 18 years old, please
contact Janice Tona, Ph.D., OTR at tona@buffalo.edu. Thank you.
7)
What type of physician (e.g., pediatrician, neurologist, psychiatrist, etc.) identified your
child as having TS? _________________________________________________________
8)
Please enter an email address that we may use to contact you in case we need clarification
on your answers. ___________________________________________________________
9)
Please enter the email address again for verification. _______________________________
10) Would you like us to send a summary of our findings to you via email when the study is
completed?
Yes, please send a summary to the email address indicated above
No, I would prefer to not receive a summary
11) Thank you for completing the required preliminary information for the survey. You are
eligible to participate in this study. From this point on, we ask that you try to answer the
questions to the best of your ability.
How many children in your family have been identified by a physician as having TS?
1
2
3
82
4
5
6
7
8
9
12) You can complete the survey for as many children as you have with TS. To do this, you
need to first complete the survey for your oldest child (child #1) and submit the survey, then
return to the survey using the same email address and complete the survey for the next
oldest child (child #2), then repeat this procedure for all subsequent children. For which
child are you submitting a survey at this time?
Child number 1
Child number 2
Child number 3
Child number 4
Child number 5
Child number 6
Child number 7
Child number 8
Child number 9
Child number 10
13) How old is this child (in years)? _______________________________________________
14) Is this child a boy or a girl?
83
A boy
A girl
15) What is this childs race? (check all that apply)
African American/Black
American Indian or Alaska Native
Asian
Hispanic
Native Hawaiian and Other Pacific Islander
White
Other (please specify)
If you selected other, please specify: ____________________________________________
16) Who lives in the home with this child?
Number of parents/guardians: _________________________________________________
Number of brothers: _________________________________________________________
Number of sisters: __________________________________________________________
Other people living in the home: _______________________________________________
Additional Comments: _______________________________________________________
17) Are the family demographics, including parent education level and income, different for this
child as compared to the first child you told us about?
The family information for this child is THE SAME as the family information I
submitted for the first child. Please use the previous information.
The family information for this child is DIFFERENT than the family information I
submitted for the first child, and I will need to answer those questions again.
84
18) What is the zip code of this childs primary residence? _____________________________
19) What is your combined household yearly income?
Less than $25,000
$25,000-49,999
$50,000-74,999
$75,000-99,999
$100,000-149,999
$150,000-199,999
$200,000 and more than $200,000
20) What is the highest level of the mothers education?
Less than high school degree
High school/GED
Some college
Masters degree
Doctoral degree
MD/DO
Not applicable
21) What is the highest level of the fathers education?
Less than high school degree
High school/GED
Some college

Masters degree
Doctoral degree
MD/DO
Not applicable
22) At what age (in years) was your child identified by a physician as having TS?
Before 1
1
2
3
4
5
6
7
8
9
10
11
12
13
14
15
85
86
16
17
23) At what age (in years) did your child first show signs of having TS even without a diagnosis?
Before 1
1
2
3
4
5
6
7
8
9
10
11
12
13
14
15
16
17
24) Has your child ever had any other medical diagnoses? (check all that apply)
Diagnosed by
physician
Suspected but not

formally diagnosed

ADHD
Anxiety Disorder
Autism Spectrum Disorder
Developmental Delays
Down Syndrome
Genetic Disorder
(please specify in additional comments below)
Intellectual Disability
(formerly called mental retardation)
Obsessive-Compulsive Disorder (OCD)
Sensory Processing Disorder
Other
(please specify in additional comments below)
87
Additional comments: _______________________________________________________

25) Has your child ever taken any medication for tic or non-tic symptoms?
Yes
No
If you selected yes, please specify the medication, and the positive and negative effect:
__________________________________________________________________________
26) Has your child ever received OT services for TS-related difficulties?
Yes
No
27) What were the reasons why your child received OT? (check all that apply)
Attention
Fine motor
Gross motor
Handwriting
Organization
Sensory processing
88
Tic management
Time management
Visual perception
28) Did you find OT intervention beneficial for addressing the reasons why your child received
OT?
Yes
No
Please share specifics about OT intervention here: _________________________________
29) HOME-RELATED OCCUPATIONS
Now we are interested in learning about your childs functions in home-related occupations.
Children with Tourette syndrome frequently have periods of increased symptoms (waxing)
and periods of decreased symptoms (waning). When your child has been in a period of
waxing, what difficulties in home-related occupations have been seen considering the
developmental level expected at his/her age? You can use the comment box to share
details if you need to clarify your answer.
Bathing/showering
(including soaping, rinsing,
drying body parts, and
transferring to and from
bathing positions)
I HAVE observed my child

to have difficulty in this area
considering the
developmental level
expected at his/her age.
I have NOT observed my

child to have difficulty in
this area considering the
developmental level

Toileting (bowel and bladder
management including
urinary frequency, accident,
potty training, and
bedwetting)
Toilet hygiene (including
wiping and flushing, and
menstrual care)
Dressing (selecting clothing,
dressing and undressing, and
fastening)
Swallowing/eating
(including chewing,
swallowing, sensory
acceptance of foods, food
selection, and food quantity)
Feeding (including selffeeding/bringing food to
mouth)
Functional mobility (such as
walking around at home, or
transfer to bathtub, car,
toilet, and so on)
Personal hygiene and
grooming (removing body
hair; applying and removing
cosmetics; washing, drying,
combing, styling, brushing,
and trimming hair; brushing
and flossing teeth; and
caring for nails)
Sexual activity (such as age
inappropriate discussion,
gestures, online activity, or
sexual activity)
Communication
management (such as using
telephones, cell phones,
keyboards, computers or
tablets, and writing tools to
communicate)
Community mobility
(including biking, riding in
buses/school buses, taxies,
or other transportation
systems, and walking around
89

in the community)
Rest (including identifying
the need to relax and
engaging in activities which
can restore energy, and
engaging in activities that
result in a relaxed state)
Sleep (including following
appropriate sleep patterns,
ceasing activities and
preparing for bed, and
sustaining a sleep state
without disruption)
90

30) BATHING/SHOWERING
DURING
Please rate the greatest level of difficulty you have seen with bathing/showering (including
soaping, rinsing, drying body parts, and transferring to and from bathing positions)
DURING the waxing period.
1 My child has NOT displayed a problem in this area.
2 My child has displayed MILD DIFFICULTY in this area, but does not require
assistance.
3 My child has displayed MODERATE DIFFICULTY in this area, and required
assistance and adaptation or help to remain functional.
4 My child has displayed SEVERE DIFFICULTY in this area. My child was unable to
function at a typical level, even with assistance and adaptation.
31) BEFORE
91
soaping, rinsing, drying body parts, and transferring to and from bathing positions)
BEFORE the waxing period.
assistance.
32) AFTER
soaping, rinsing, drying body parts, and transferring to and from bathing positions) AFTER
the waxing period.
assistance.
92
33) TOILETING
DURING
Please rate the greatest level of difficulty you have seen with toileting (bowel and bladder
management including urinary frequency, accident, potty training, and bedwetting)
assistance.
34) BEFORE
management including urinary frequency, accident, potty training, and bedwetting)
assistance.
93
35) AFTER
management including urinary frequency, accident, potty training, and bedwetting) AFTER
the waxing period.
assistance.
36) TOILET HYGIENE
DURING
Please rate the greatest level of difficulty you have seen with toilet hygiene (including
wiping and flushing, and menstrual care) DURING the waxing period.
assistance.
94

37) BEFORE
wiping and flushing, and menstrual care) BEFORE the waxing period.
assistance.
38) AFTER
wiping and flushing, and menstrual care) AFTER the waxing period.
assistance.
95

39) DRESSING
DURING
Please rate the greatest level of difficulty you have seen with dressing (selecting clothing,
dressing and undressing, and fastening) DURING the waxing period.
assistance.
40) BEFORE
dressing and undressing, and fastening) BEFORE the waxing period.
assistance.
96

41) AFTER
dressing and undressing, and fastening) AFTER the waxing period.
assistance.
42) SWALLOWING/EATING
DURING
Please rate the greatest level of difficulty you have seen with swallowing/eating (includes
chewing, swallowing, sensory acceptance of foods, food selection, and food quantity)
97
assistance.
43) BEFORE
assistance.
44) AFTER
AFTER the waxing period.
98

assistance.
45) FEEDING
DURING
Please rate the greatest level of difficulty you have seen with feeding (including selffeeding/bringing food to mouth) DURING the waxing period.
assistance.
46) BEFORE
Please rate the greatest level of difficulty you have seen with feeding (including selffeeding/bringing food to mouth) BEFORE the waxing period.
99

assistance.
47) AFTER
Please rate the greatest level of difficulty you have seen with feeding (including selffeeding/bringing food to mouth) AFTER the waxing period.
assistance.
48) FUNCTIONAL MOBILITY
DURING
100
Please rate the greatest level of difficulty you have seen with functional mobility (such as
walking around at home, and transfer to bathtub, car, toilet, and so on) DURING the waxing
period.
assistance.
49) BEFORE
walking around at home, and transfer to bathtub, car, toilet, and so on) BEFORE the waxing
period.
assistance.
101
50) AFTER
walking around at home, and transfer to bathtub, car, toilet, and so on) AFTER the waxing
period.
assistance.
51) PERSONAL HYGIENE AND GROOMING
DURING
Please rate the greatest level of difficulty you have seen with personal hygiene and
grooming (removing body hair; applying and removing cosmetics; washing, drying,
combing, styling, brushing, and trimming hair; brushing and flossing teeth; and caring for
nails) DURING the waxing period.
assistance.
102
52) BEFORE
nails) BEFORE the waxing period.
assistance.
53) AFTER
nails) AFTER the waxing period.
103
assistance.
54) SEXUAL ACTIVITY
DURING
Please rate the greatest level of difficulty you have seen with sexual activity (such as age
inappropriate discussion, gestures, online activity, or sexual activity) DURING the waxing
period.
assistance.
55) BEFORE
104
inappropriate discussion, gestures, online activity, or sexual activity) BEFORE the waxing
period.
assistance.
56) AFTER
inappropriate discussion, gestures, online activity, or sexual activity) AFTER the waxing
period.
assistance.
105
57) COMMUNICATION MANAGEMENT

DURING
Please rate the greatest level of difficulty you have seen with communication management
(such as using telephones, cell phones, keyboards, computers or tablets, and writing tools to
communicate) DURING the waxing period.
assistance.
58) BEFORE
communicate) BEFORE the waxing period.
assistance.
106
59) AFTER
communicate) AFTER the waxing period.
assistance.
60) COMMUNITY MOBILITY
DURING
Please rate the greatest level of difficulty you have seen with community mobility
(including biking, riding in buses/school buses, taxies, or other transportation systems, and
walking around in the community) DURING the waxing period.
assistance.
107

61) BEFORE
walking around in the community) BEFORE the waxing period.
assistance.
62) AFTER
walking around in the community) AFTER the waxing period.
108
assistance.
63) REST
DURING
Please rate the greatest level of difficulty you have seen with rest (including identifying the
need to relax and engaging in activities which can restore energy, and engaging in activities
that result in a relaxed state) DURING the waxing period.
assistance.
64) BEFORE
109
that result in a relaxed state) BEFORE the waxing period.
assistance.
65) AFTER
that result in a relaxed state) AFTER the waxing period.
assistance.
110
66) SLEEP
DURING
Please rate the greatest level of difficulty you have seen with sleep (including following
appropriate sleep patterns, ceasing activities and preparing for bed, and sustaining a sleep
state without disruption) DURING the waxing period.
assistance.
67) BEFORE
state without disruption) BEFORE the waxing period.
assistance.
111
68) AFTER
state without disruption) AFTER the waxing period.
assistance.
69) EDUCATION-RELATED OCCUPATIONS
Now we are interested in learning about your childs functions in education-related
occupations. Children with Tourette syndrome frequently have periods of increased
symptoms (waxing) and periods of decreased symptoms (waning). When your child has
been in a period of waxing, what difficulties in education-related functions have been seen
considering the developmental level expected at his/her age? You can use the comment
box to share details if you need to clarify your answer.

Math
Reading
Handwriting
Physical education
(e.g., school-sponsored
sports, band, cheer-leading,
or dances)
112
considering the
developmental level

developmental level

70) MATH
DURING
Please rate the greatest level of difficulty you have seen with math DURING the waxing
period.
assistance.
71) BEFORE
Please rate the greatest level of difficulty you have seen with math BEFORE the waxing
period.
113
assistance.
72) AFTER
Please rate the greatest level of difficulty you have seen with math AFTER the waxing
period.
assistance.
73) READING
DURING
Please rate the greatest level of difficulty you have seen with reading DURING the waxing
period.
114
assistance.
74) BEFORE
Please rate the greatest level of difficulty you have seen with reading BEFORE the waxing
period.
assistance.
75) AFTER
Please rate the greatest level of difficulty you have seen with reading AFTER the waxing
period.
115
assistance.
76) HANDWRITING
DURING
Please rate the greatest level of difficulty you have seen with handwriting DURING the
waxing period.
assistance.
77) BEFORE
Please rate the greatest level of difficulty you have seen with handwriting BEFORE the
waxing period.
116
assistance.
78) AFTER
Please rate the greatest level of difficulty you have seen with handwriting AFTER the
waxing period.
assistance.
79) PHYSICAL EDUCATION
DURING
Please rate the greatest level of difficulty you have seen with physical education DURING
the waxing period.
117
assistance.
80) BEFORE
Please rate the greatest level of difficulty you have seen with physical education BEFORE
the waxing period.
assistance.
81) AFTER
Please rate the greatest level of difficulty you have seen with physical education AFTER the
waxing period.
118
assistance.
82) EXTRACURRICULAR ACTIVITIES
DURING
Please rate the greatest level of difficulty you have seen with extracurricular activities (e.g.,
school-sponsored sports, band, cheer-leading, or dances) DURING the waxing period.
assistance.
83) Please list the extracurricular activities that were problematic: _______________________
84) BEFORE
school-sponsored sports, band, cheer-leading, or dances) BEFORE the waxing period.
119

assistance.
85) AFTER
school-sponsored sports, band, cheer-leading, or dances) AFTER the waxing period.
assistance.
86) OCCUPATIONS RELATED TO SOCIAL LIFE
Now we are interested in learning about your childs functions in occupations related to
social life. Children with Tourette syndrome frequently have periods of increased
symptoms (waxing) and periods of decreased symptoms (waning). When your child has
120
been in a period of waxing, what difficulties in occupations related to social life have been
seen considering the developmental level expected at his/her age? You can use the
comment box to share details if you need to clarify your answer.
Free play
Organized activities (e.g.,
community-sponsored
sports, scouting, camps, and
recreational activities)
Social participation in the
community (such as going to
community recreational
activities, movies, bowling,
or religious activities)
Social participation with
family (including family
gatherings, family
recreation, and interacting
with siblings)
Social participation with
peers/friends

considering the
developmental level

developmental level

87) FREE PLAY
DURING
Please rate the greatest level of difficulty you have seen with free play DURING the waxing
period.
assistance.
121

88) BEFORE
Please rate the greatest level of difficulty you have seen with free play BEFORE the waxing
period.
assistance.
89) AFTER
Please rate the greatest level of difficulty you have seen with free play AFTER the waxing
period.
assistance.
122

90) ORGANIZED ACTIVITIES
DURING
Please rate the greatest level of difficulty you have seen with organized activities (e.g.,
community-sponsored sports, scouting, camps, and recreational activities) DURING the
waxing period.
assistance.
91) BEFORE
community-sponsored sports, scouting, camps, and recreational activities) BEFORE the
waxing period.
123
assistance.
92) AFTER
community-sponsored sports, scouting, camps, and recreational activities) AFTER the
waxing period.
assistance.
93) SOCIAL PARTICIPATION IN THE COMMUNITY
DURING
124
Please rate the greatest level of difficulty you have seen with social participation in the
community (such as going to community recreational activities, movies, bowling, or
religious activities) DURING the waxing period.
assistance.
94) BEFORE
religious activities) BEFORE the waxing period.
assistance.
125
95) AFTER
religious activities) AFTER the waxing period.
assistance.
96) SOCIAL PARTICIPATION WITH FAMILY
DURING
Please rate the greatest level of difficulty you have seen with social participation with
family (including family gatherings, family recreation, and interacting with siblings)
assistance.
126
97) BEFORE
assistance.
98) AFTER
assistance.
127

99) SOCIAL PARTICIPATION WITH PEERS/FRIENDS
DURING
peers/friends DURING the waxing period.
assistance.
100) BEFORE
peers/friends BEFORE the waxing period.
assistance.
128

101) AFTER
peers/friends AFTER the waxing period.
assistance.
102) MENTAL FUNCTIONS
Now we are interested in learning about your childs mental functions and cognitive skills.
waxing, what difficulties in mental functions and cognitive skills have been seen
considering the developmental level expected at his/her age? You can use the comment
box to share details if you need to clarify your answer.
Control of thoughts (no

unwanted obsessive,
repetitive, or intrusive
thoughts)
Control of behaviors (no
compulsive behaviors or
rituals)
Higher level cognitive
functions/thinking
(including judgment, insight,
and cognitive flexibility)
Attention (sustained,
selected, and divided
attention; concentration; and
distractibility)
Memory (short-term, longterm and working memory)
Sequencing (such as
organizing, and planning
projects)
Emotional coping
Energy and drive (energy
level, motivation, appetite,
craving, and impulse
control)
129

considering the
developmental level

developmental level

103) CONTROL OF THOUGHTS
DURING
Please rate the greatest level of difficulty you have seen with control of thoughts (no
unwanted obsessive, repetitive, or intrusive thoughts) DURING the waxing period.
130
assistance.
104) BEFORE
unwanted obsessive, repetitive, or intrusive thoughts) BEFORE the waxing period.
assistance.
105) AFTER
unwanted obsessive, repetitive, or intrusive thoughts) AFTER the waxing period.
131
assistance.
106) CONTROL OF BEHAVIORS
DURING
Please rate the greatest level of difficulty you have seen with control of behaviors (no
compulsive behaviors or rituals) DURING the waxing period.
assistance.
107) BEFORE
compulsive behaviors or rituals) BEFORE the waxing period.
132
assistance.
108) AFTER
compulsive behaviors or rituals) AFTER the waxing period.
assistance.
109) HIGHER LEVEL COGNITIVE FUNCTION/THINKING
Please rate the greatest level of difficulty you have seen with higher level cognitive
function/thinking (including judgment, insight, and cognitive flexibility) DURING the
waxing period.
133
assistance.
110) BEFORE
function/thinking (including judgment, insight, and cognitive flexibility) BEFORE the
waxing period.
assistance.
111) AFTER
function/thinking (including judgment, insight, and cognitive flexibility) AFTER the
waxing period.
134

assistance.
112) ATTENTION
DURING
Please rate the greatest level of difficulty you have seen with attention (sustained, selected,
and divided attention; concentration; and distractibility) DURING the waxing period.
assistance.
113) BEFORE
and divided attention; concentration; and distractibility) BEFORE the waxing period.
135

assistance.
114) AFTER
and divided attention; concentration; and distractibility) AFTER the waxing period.
assistance.
115) MEMORY
DURING
Please rate the greatest level of difficulty you have seen with memory (short-term, longterm, and working memory) DURING the waxing period.
136

assistance.
116) BEFORE
Please rate the greatest level of difficulty you have seen with memory (short-term, longterm, and working memory) BEFORE the waxing period.
assistance.
117) AFTER
Please rate the greatest level of difficulty you have seen with memory (short-term, longterm, and working memory) AFTER the waxing period.
137
assistance.
118) SEQUENCING
DURING
Please rate the greatest level of difficulty you have seen with sequencing (such as
organizing and planning projects) DURING the waxing period.
assistance.
119) BEFORE
organizing and planning projects) BEFORE the waxing period.
138
assistance.
120) AFTER
organizing and planning projects) AFTER the waxing period.
assistance.
121) EMOTIONAL COPING
DURING
Please rate the greatest level of difficulty you have seen with emotional coping DURING
the waxing period.
139
assistance.
122) BEFORE
Please rate the greatest level of difficulty you have seen with emotional coping BEFORE
the waxing period.
assistance.
123) AFTER
Please rate the greatest level of difficulty you have seen with emotional coping AFTER the
waxing period.
140
assistance.
124) ENERGY AND DRIVE
DURING
Please rate the greatest level of difficulty you have seen with energy and drive (energy level,
motivation, appetite, craving, and impulse control) DURING the waxing period.
assistance.
125) BEFORE
motivation, appetite, craving, and impulse control) BEFORE the waxing period.
141
assistance.
126) AFTER
motivation, appetite, craving, and impulse control) AFTER the waxing period.
assistance.
127) SENSORY FUNCTIONS
Now we are interested in learning about your childs sensory functions. Children with
Tourette syndrome frequently have periods of increased symptoms (waxing) and periods of
decreased symptoms (waning). When your child has been in a period of waxing, what
difficulties in sensory functions have been seen considering the developmental level
142
expected at his/her age? You can use the comment box to share details if you need to
clarify your answer.
Vision/seeing
Hearing
Taste
Smell
Proprioception/knowing
where body is in space
Tactile/touch
Pain (ability to feel pain in
body with injury may be
increased ability or
decreased ability)
Temperature (ability to
differentiate hot from cold
items when touching or
tasting)

considering the
developmental level

developmental level

128) VISION/SEEING
DURING
Please rate the greatest level of difficulty you have seen with vision/seeing DURING the
waxing period.
assistance.
143
129) BEFORE
Please rate the greatest level of difficulty you have seen with vision/seeing BEFORE the
waxing period.
assistance.
130) AFTER
Please rate the greatest level of difficulty you have seen with vision/seeing AFTER the
waxing period.
assistance.
144
131) HEARING
DURING
Please rate the greatest level of difficulty you have seen with hearing DURING the waxing
period.
assistance.
132) BEFORE
Please rate the greatest level of difficulty you have seen with hearing BEFORE the waxing
period.
assistance.
145
133) AFTER
Please rate the greatest level of difficulty you have seen with hearing AFTER the waxing
period.
assistance.
134) VESTIBULAR/SENSE OF MOVING
DURING
Please rate the greatest level of difficulty you have seen with vestibular/sense of moving
assistance.
146
135) BEFORE
assistance.
136) AFTER
assistance.
147
137) TASTE
DURING
Please rate the greatest level of difficulty you have seen with taste DURING the waxing
period.
assistance.
138) BEFORE
Please rate the greatest level of difficulty you have seen with taste BEFORE the waxing
period.
assistance.
148
139) AFTER
Please rate the greatest level of difficulty you have seen with taste AFTER the waxing
period.
assistance.
140) SMELL
DURING
Please rate the greatest level of difficulty you have seen with smell DURING the waxing
period.
assistance.
149
141) BEFORE
Please rate the greatest level of difficulty you have seen with smell BEFORE the waxing
period.
assistance.
142) AFTER
Please rate the greatest level of difficulty you have seen with smell AFTER the waxing
period.
assistance.
150
143) PROPRIOCEPTION
DURING
Please rate the greatest level of difficulty you have seen with proprioception/knowing where
body is in space DURING the waxing period.
assistance.
144) BEFORE
body is in space BEFORE the waxing period.
assistance.
151
145) AFTER
body is in space AFTER the waxing period.
assistance.
146) TACTILE/TOUCH
DURING
Please rate the greatest level of difficulty you have seen with tactile/touch DURING the
waxing period.
assistance.
152
147) BEFORE
Please rate the greatest level of difficulty you have seen with tactile/touch BEFORE the
waxing period.
assistance.
148) AFTER
Please rate the greatest level of difficulty you have seen with tactile/touch AFTER the
waxing period.
assistance.
153
149) PAIN
DURING
Please rate the greatest level of difficulty you have seen with pain (ability to feel pain in
body with injury may be increased ability or decreased ability) DURING the waxing
period.
assistance.
150) BEFORE
body with injury may be increased ability or decreased ability) BEFORE the waxing
period.
assistance.
154

151) AFTER
body with injury may be increased ability or decreased ability) AFTER the waxing period.
assistance.
152) TEMPERATURE
DURING
Please rate the greatest level of difficulty you have seen with temperature (ability to
differentiate hot from cold items when touching or tasting) DURING the waxing period.
assistance.
155

153) BEFORE
differentiate hot from cold items when touching or tasting) BEFORE the waxing period.
assistance.
154) AFTER
differentiate hot from cold items when touching or tasting) AFTER the waxing period.
assistance.
156

155) MUSCLE AND MOVEMENT FUNCTIONS
Now we are interested in learning about your childs muscle and movement functions.
waxing, what difficulties in muscle and movement functions are seen considering the
developmental level expected at his/her age? You can use the comment box to share
details if you need to clarify your answer.
Muscle strength (power)

Muscle endurance (sustained
power)
Smoothness/control of
voluntary movements
(coordination/no tremors,
including gross and fine
motor skills)
Ability to suppress unwanted
movements (tics)

considering the
developmental level

developmental level

156) MUSCLE STRENGTH (POWER)
157
DURING
Please rate the greatest level of difficulty you have seen with muscle strength (power)
assistance.
157) BEFORE
assistance.
158) AFTER
158
assistance.
159) MUSCLE ENDURANCE (SUSTAINED POWER)
DURING
Please rate the greatest level of difficulty you have seen with muscle endurance (sustained
power) DURING the waxing period.
assistance.
160) BEFORE
159
power) BEFORE the waxing period.
assistance.
161) AFTER
power) AFTER the waxing period.
assistance.
162) SMOOTHNESS/CONTROL OF VOLUNTARY MOVEMENTS
DURING
160
Please rate the greatest level of difficulty you have seen with smoothness/control of
voluntary movements (coordination/no tremors, including gross and fine motor skills)
assistance.
163) BEFORE
assistance.
161
164) AFTER
assistance.
165) ABILITY TO SUPPRESS UNWANTED MOVEMENTS (TICS)
DURING
Please rate the greatest level of difficulty you have seen with ability to suppress unwanted
movements (tics) DURING the waxing period.
assistance.
162

166) BEFORE
movements (tics) BEFORE the waxing period.
assistance.
167) AFTER
movements (tics) AFTER the waxing period.
assistance.
163
168) GAIT/WALKING PATTERNS

DURING
Please rate the greatest level of difficulty you have seen with gait/walking patterns
assistance.
169) BEFORE
Please rate the greatest level of difficulty you have seen with gait/walking patterns
assistance.
164
170) AFTER
Please rate the greatest level of difficulty you have seen with gait/walking patterns AFTER
the waxing period.
assistance.
171) Thank you very much for participating in this survey. Your responses will help us
understand the functional needs of children with TS better. If you have any questions about
this survey, please contact Janice Tona, OTR, Ph.D., OTR, at tona@buffalo.edu or (716)
829-6741.
Submit
172) Thank you for taking our survey.
165
Appendix B
Expert survey
Thank you for your willingness to assist in establishing the content validity for the parent
survey. Please look over the OTPF-III and all the questions listed in the parent survey. Please
identify how close each item is to represent the OTPF-III. You may leave your comments about
the wording, order of questions, clarity of each item, any modification needed, etc. in the blank
column.
* OT rating for the each items representativeness of the OTPF-III:
1 = the item is not representative of the OTPF-III
2 = the item needs major revisions to be representative of the OTPF-III
3 = the item needs minor revisions to be representative of the OTPF-III
4 = the item is representative of the OTPF-III
SURVEY ITEM
OT RATING*
OT COMMENTS
A. HOME-RELATED OCCUPATIONS
1. Bathing/showering
1
2
3
4
2. Toileting (bowel and bladder
1
management including urinary
2
frequency, accident, potty training, and
3
bedwetting)
4
3. Toilet hygiene (including wiping and
1
flushing)
2
3
4
4. Dressing
1
2
3
4
5. Swallowing/eating (includes chewing,
1
swallowing, sensory acceptance of
2
foods, food selection, and food quantity)
3
4
6. Feeding (includes self-feeding/bringing
1
food to mouth)
2
3
7.
Functional mobility (such as walking

around at home, or transfer to bathtub,
car, toilet, and so on)
8.
9.
Sexual activity (such as age

inappropriate discussion, gestures, online activity, or sexual activity)
10. Communication management (such as

using telephones, cell phones,
keyboards, computers or tablets, and
writing tools to communicate)
11. Community mobility (includes biking,
riding in buses, taxies, or other
transportation systems, and walking
around in the community)
12. Rest (including identifying the need to
relax and engaging in activities which
can restore energy)
13. Sleep (includes following appropriate
sleep patterns, ceasing activities and
ready for bed, and sustaining a sleep
state without disruption)
B. EDUCATION-RELATED OCCUPATIONS
1. Math
2.
Reading
3.
Handwriting
4.
Physical education
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
166

5.
C. OCCUPATIONS RELATED TO SOCIAL LIFE

1. Free Play
2.
Organized Activities
3.

(such as going to community
recreational activities, movies, or
bowling)
(includes family gatherings and family
recreation)
4.
5.
D. MENTAL FUNCTIONS
1. Control of thoughts (no unwanted
obsessive, repetitive, or intrusive
thoughts)
2.
Control of behaviors (no compulsive

behaviors or rituals)
3.
Higher level thinking (includes

judgment, insight, and cognitive
flexibility)
4.
Attention (sustained, selected, and

divided attention)
5.
Memory (short term, long term and

working memory)
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
167

6.
Sequencing (such as organizing and

planning projects)
7.
Emotional coping
8.
Energy and drive
E. SENSORY FUNCTIONS
1. Vision/seeing
2.
Hearing
3.
4.
Taste
5.
Smell
6.
Proprioception/knowing where body is

in space
7.
Tactile/touch
8.
Pain (ability to feel pain in body with

injury may be increased ability or
decreased ability)
9.
Temperature (ability to differentiate hot
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
168

from cold items when touching or
tasting)
F. MUSCLE AND MOVEMENT FUNCTIONS
1. Muscle strength (power)
2.
Muscle endurance (sustained power)
3.
Smoothness/control of voluntary
movements (coordination/no tremors)
4.
Ability to restrain movements (no

involuntary tics)
5.
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
1
2
3
4
169
170
Appendix C
Sample of the pilot survey
26) Has your child ever received OT services for TS-related difficulties?
Yes
No
Parent pilot survey comments:
27) What were the reasons why your child received OT? (check all that apply)
Attention
Fine motor
Gross motor
Handwriting
Organization
Sensory processing
Tic management
Time management
Visual perception
171
69) EDUCATION-RELATED OCCUPATIONS

Now we are interested in learning about your childs functions in education-related occupations.
Children with Tourette syndrome frequently have periods of increased symptoms (waxing) and
periods of decreased symptoms (waning). When your child has been in a period of waxing, what
difficulties in education-related functions have been seen considering the developmental level
expected at his/her age? You can use the comment box to share details if you need to clarify
your answer.
Math
Reading
Handwriting
Physical education
Extracurricular activities (e.g.,
school-sponsored sports,
band, cheer-leading, dances)
I HAVE observed my child to

have difficulty in this area
considering the
developmental level expected
at his/her age.
I have NOT observed my child

considering the developmental
level expected at his/her age.
Additional comments: ___________________________________________________________

70) MATH
DURING
Please rate the greatest level of difficulty you have seen with math DURING the waxing
period.
172
assistance.
71) BEFORE
Please rate the greatest level of difficulty you have seen with math BEFORE the waxing
period.
assistance.

Appendix D
University at Buffalo Institutional Review Board approval letter
173
174
175
176
Appendix E
Template for cover letter to the experts
Dear (First name, Last name),
I am a graduate occupational therapy student at the University at Buffalo, The State
University of New York, and I am conducting a research project entitled Daily functioning of
children with Tourette syndrome: An exploratory study under the instruction of Janice Tona,
Ph.D., OTR. The purpose of my study is to explore childrens functional performance in three of
the domains included in the Occupational Therapy Practice Framework: Domain and Process
Third Edition (OTPF-III): occupations, body functions (among client factors), and performance
skills. I will be doing this through an online survey for parents of children with Tourette
syndrome. In all, the survey asks about 23 occupations regarding activities of daily living,
education, play and leisure, rest and sleep, and social participation, and 22 items for body
functions and performance skills including mental functions, sensory functions, and muscle and
movement functions.
Because of your clinical experience and professional knowledge, I am writing to you to
request your assistance with establishing content validity of the parent survey. If you are
interested in possibly participating, please reply this email. Then a consent form, the OTPF-IIII,
the preliminary parent survey, and an expert survey will be sent to you via email or U.S. postal
mail as you wish. You would be asked to look over all the questions listed in the preliminary
parent survey and use the expert survey to judge and rate how representative those items are of
the content domains of OTPF-III. Also, you could leave your comments about the wording,
order of questions, clarity of each item, any modification needed, etc. in the blank column.

If you have any questions or concerns, you may contact Sheng-Hui Liao via email at
shenghui@buffalo.edu or the faculty advisor Janice Tona, Ph.D., OTR, via email at
tona@buffalo.edu or by telephone at (716) 829-6741. Thank you for your assistance.
Sincerely,
Sheng-Hui Liao
177
178
Appendix F
Experts consent form
This study is being conducted by a graduate occupational therapy student Sheng-Hui Liao
under the guidance of Janice Tona, Ph.D., OTR, a faculty member of the Rehabilitation Science
Department at the University at Buffalo, The State University of New York (UB).
PURPOSE: The purpose of this study is to better understand the functional difficulties in
occupations, body functions, and performance skills for children with Tourette syndrome (TS)
using a parent survey that is based on the Occupational Therapy Practice Framework: Domain
and Process - Third Edition (OTPF-III). Also, the frequency of receiving occupational therapy
(OT) services and the reasons for being referred to OT among children with TS will be discussed.
However, before the parent survey can be launched, we would like to establish the content
validity by having three OT experts review the survey.
You are being invited to participate because of your clinical experience and professional
knowledge. In order to participate in this study, you must be an OT practitioner with at least 5
years of experience in pediatric OT including working with children with TS, and you must be
familiar with the concepts of the OTPF-III. The OTPF-III is a recently-developed modification
of the OTPF-II, and a copy of the OTPF-III is attached. You can refer to the attached OTPF-III
at any time during the survey as a reference.
PROCEDURES: If you choose to participate in this expert survey, you will be asked to
review all questions in the preliminary parent survey and then rate how representative those
items are of the content domains of the OTPF-III. You will also be asked to leave your
comments about the wording, order of questions, clarity of each item, any modification needed,
179
etc. in the blank column. The final parent survey will be an online survey in which parents
answer only the questions applicable to their child. However, we are asking you to review all of
the questions in a Word document and respond by rating and commenting on the questions either
electronically in the Word document or by handwriting on a paper version.
TIME COMMITMENT: The length of time required to complete this expert survey
depends on the number of comments you are going to make. It may take 40 to 60 minutes to
complete the survey.
COSTS, RISKS, AND BENEFITS: There are no costs for participating in the study,
and no anticipated risks.
The information you provide will help us to establish the content validity and improve the
parent survey that will be used in this study. The information collected may not benefit you
directly, but what we learn from this study should provide general information to health care
providers about problems with daily living and the role of OT among children with TS.
CONFIDENTIALITY: This survey is confidential. Your name will not be revealed as
being a reviewer, though your name may be used in general acknowledgements about the study
if you approve. You will be assigned an ID number and only the ID number will be linked to
your survey. No record will be kept linking your name to the ID number. Your consent form
will be stored in a separate file from the responses that you provided in the expert survey in a
locked file cabinet. In order to monitor this research study, representatives from federal agencies
such as Office of Human Research Protection (OHRP) or representatives from the UB Human
Research Protections Programs may inspect the research records; this process may reveal your
identity.
180
VOLUNTARY PARTICIPATION: This survey is voluntary. If you choose to

participate but then decide to withdraw from the study, you can notify the faculty advisor Janice
Tona, Ph.D., OTR, at (716) 829-6741 or tona@buffalo.edu, and your information will be deleted
from the study if the study has not been completed.
QUESTIONS, CONCERNS OR COMPLAINTS: If you have any questions, concerns
or complaints, or think the research have hurt you, you can contact Dr. Janice Tona, Clinical
Assistant Professor, Rehabilitation Science, 625 Kimball Tower, University at Buffalo, Buffalo,
NY 14214. Dr. Tona can be reached at (716) 829-6741 or at tona@buffalo.edu. If your
questions, concerns, or complaints are not being answered by the research team, you cannot
reach the research team, you want to talk with someone who is not a member of the research
team, you have any questions about your rights as a participant in a research project, or you want
to get information or provide input about this research, you may also contact the research
participant advocate at (716) 888-4845 or at researchadvocate@buffalo.edu.
1)
I have read the above information and consent to participate in this survey.
I do not wish to participate in this survey at this time.
2)
In order to participate in this study, you must be an OT practitioner with at least 5 years of
experience in pediatric OT including working with children with TS, and you must be
familiar with the concepts of the OTPF-III. The OTPF-III is a recently-developed
modification of the OTPF-II, and a copy of the OTPF-III is attached. You can refer to the
attached OTPF-III at any time during the survey as a reference.
I am eligible to participate in this survey.
I am not eligible to participate in this survey.
181
3)
I agree to have my name used in general acknowledgements regarding this study.
I do not wish to have my name included in general acknowledgements regarding this
study.
4)
If you chose to receive this consent form via email, please print out and sign the form below,
and then scan and forward the signed consent form to the principal investigator Sheng-Hui
Liao at shenghui@buffalo.edu. If you chose to receive the consent form via U.S. postal
mail, please sent the consent form back with other material. Thank you.
Your signature documents your permission to take part in this survey.
Signature of subject
Date
Printed name of subject
Signature of person obtaining consent
Printed name of person obtaining consent
Date
182
Appendix G
Template for cover letter to the TSA-GNYS board members
Dear (First name, Last name),
Ph.D., OTR. The purpose of my study is to find out how Tourette symptoms influence
childrens functional performance in different areas. A parent survey has been created and three
occupational therapy (OT) practitioners with experience in intervening children with Tourette
syndrome (TS) have reviewed it. The survey will be available online for parents of children with
TS to complete afterward.
Because you are a parent or guardian of a child with TS and also a board member of the
Tourette Syndrome Association of Greater New York State, I am writing to you to request your
assistance with the pilot test of the parent survey. This pilot test will help us know if the
questions are clear and able to be easily answered by parents. If you are interested in possibly
participating as a pilot test participant, please reply to this email, and a consent form and the link
to the pilot survey will then be sent to you via email. The pilot survey contains all the
information and questions included in the parent survey with a blank text column labeled Parent
Pilot Survey Comments being attached to each question on the parent survey. You will be
asked to take the survey, and you will be asked to note how easy or difficult it is to answer each
question and to leave a pilot comment about any questions you think may need to be modified.
183
If you have any questions, concerns or complaints, you may contact Sheng-Hui Liao via
email at shenghui@buffalo.edu or the faculty advisor Janice Tona, Ph.D., OTR via email at
tona@buffalo.edu or by telephone at (716) 829-6741. Thank you for your assistance.
Sincerely,
Sheng-Hui Liao
184
Appendix H
Pilot consent form for board members of the TSA-GNYS
PURPOSE: The purpose of this study is to illustrate the difficulties in different aspects
of daily life, at school and at home, experienced among the children with Tourette syndrome
(TS). Also, the frequency of receiving occupational therapy (OT) services and the reasons for
being referred to OT among children with TS will be discussed. However, before the parent
survey can be launched, we would like to improve it and make it more user-friendly. Therefore,
we are seeking five parents of individuals with TS to take this survey as a pilot test to help us
know if there are improvements that should be made to the survey before we launch the final
survey to the public.
You are being invited to participate because you are a parent or guardian of an individual
with TS and also a board member of the Tourette Syndrome Association of Greater New York
State (TSA-GNYS). While the final survey is designed for parents of children with TS who are
under 18 years of age, pilot test participants need only be the parent of an individual with TS,
regardless of the childs age.
PROCEDURES: If you choose to participate in this online pilot survey, you will take
the survey in the same way that the study participants will take the survey. You will be asked
demographic questions about your child and your family and you will then be asked specific
questions about daily living skills, school functions, leisure activity, social participation, mental
185
functions, sensory functions, and muscle and movement functions, and therefore we can know
the areas in which your child has experienced difficulty. After each question is a box labeled
Parent Pilot Survey Comments which will not be found in the final survey. Please use these
Parent Pilot Survey Comments boxes to note how easy or difficult it is to answer each question
and leave a pilot comment about any questions you think may need to be modified. This will
help us to make the final survey as user-friendly as possible.
Your responses will only be used in modifying and improving the survey. Your
responses will not be used in data analysis of the final study.
TIME COMMITMENT: The length of time required to complete this survey depends
on the number of needs your child has had and the number of comments you are going to make.
It may take 40 to 60 minutes to complete the survey.
COSTS, RISKS, AND BENEFITS: There are no costs for participating in the study.
The survey asks about your child's daily functions; therefore, there may be a risk of becoming
upset when answering these questions. To minimize the risk, the survey allows you to skip
questions that you find unpleasant. If you find that you are upset after completing this survey
and want some help to manage your feelings, you can contact the faculty advisor, Janice Tona,
Ph.D., OTR. Dr. Tona is an occupational therapist with expertise in helping children and
families with TS and she will assist you in identifying local supports to help you. Dr. Tona can
be reached at (716) 829-6741 or tona@buffalo.edu.
The information you provide will help us to improve the parent survey that will be used
in this study. The information collected may not benefit you directly, but what we learn from
this study should provide general information to health care providers about problems with daily
living and the role of OT among children with TS.
186
CONFIDENTIALITY: This survey is confidential. The only link between you and the
information about your child will be your email address. No one outside of the researchers will
have access to your email address or will be able to link your answers to your email address. If
you withdraw from the study, all individually identifiable data provided by you will be destroyed
and not used for analysis. In order to monitor this research study, representatives from federal
agencies such as Office of Human Research Protection (OHRP) or representatives from the UB
Human Research Protections Programs may inspect the research records; this process may reveal
your identity. Your email address will not be kept after the study is completed. Any information
that you provide for the purpose of receiving a summary will be stored separately from the rest
of the study data, and will be destroyed at the end of the study. We will not leave cookies
(identifying files) on your system. A secure web server is provided to protect against
intermediate sites from intercepting your data. Please note that because you are answering
questions via the internet, your anonymity cannot be absolutely guaranteed. It may not be wise
to complete the survey at a coffee shop, library or workplace. Be sure to quit the web browser
after finishing the study so people cannot go back to the pages you completed.
VOLUNTARY PARTICIPATION: This survey is voluntary. If you choose not to
participate, it will not affect your standing with the TSA-GNYS nor any other organization or
physician that may have informed you of the study. If you choose to participate but then decide
to withdraw from the study, you can notify the faculty advisor Janice Tona, Ph.D., OTR at (716)
829-6741 or tona@buffalo.edu, and your information will be deleted from the study if the study
has not been completed.
187
If you choose to participate, please provide your email address so that you can be
contacted if any of your answers are not clear, but your email address will be kept separate from
the responses and will not be shared with anyone other than the researchers.
or complaints, or think the research has hurt you, you can contact Dr. Janice Tona, Clinical
1)
I have read the above information and consent to participate in the survey.
2)
In order to participate in this study, you must be a parent or guardian of an individual with
TS and also a board member of the TSA-GNYS.
I am a parent or guardian of an individual with TS and also a board member of the TSA-
GNYS.
I am NOT a parent or guardian of an individual with TS and also a board member of the
TSA-GNYS.
3)
Thank you for your interest in this survey. If you change your mind and would like to

participate, you may click on this link again or contact Janice Tona, Ph.D., OTR at
tona@buffalo.edu.
188
189
Appendix I
Invitation to members of the TSA-GNYS
Dear Members of the TSA-GNYS,
Ph.D., OTR. The purpose of my study is to find out how Tourette symptoms influence
childrens functional performance in different areas. A parent survey has been created and three
occupational therapy (OT) practitioners with experience in intervening children with Tourette
syndrome (TS) have reviewed it. The parent survey is available online for parents of children
with TS to complete. The research results may help OT practitioners have better knowledge
basis for offering beneficial intervention and may serve as a springboard for future studies of OT
intervention.
Here is the link to the parent survey https://vovici.com/wsb.dll/s/8727g57c48. To be
eligible for participating in this study you must be at least 18 years of age and be the parent of a
child who has been diagnosed by a doctor as having TS and who is under 18 years of age. I am
hoping to recruit 100 participants to complete this parent survey in order to strengthen the results
of this study. If you know someone who may be interested in this study or who may be eligible
for taking the survey, please feel free to share the information with him or her.
If you have any questions, concerns or complaints, you may contact Sheng-Hui Liao via
email at shenghui@buffalo.edu or the faculty advisor Janice Tona, Ph.D., OTR via email at
tona@buffalo.edu or by telephone at (716) 829-6741.

Sincerely,
Sheng-Hui Liao
190
191
Appendix J
Parent consent form for members of the TSA-GNYS
PURPOSE: The purpose of this study is to illustrate the difficulties in different aspects
of daily life, at school and at home, experienced among the children with Tourette syndrome
(TS). Also, the frequency of receiving occupational therapy (OT) services and the reasons for
being referred to OT among children with TS will be discussed. We are hoping to recruit 100
participants.
You are being invited to participate because you are a parent or guardian of a child with
TS. In order to participate in this study you must be at least 18 years old, and be the parent or
guardian of a child who is under 18 years old and who has been diagnosed by a doctor with TS.
Your child does NOT have to have received OT services for you to participate in this study.
PROCEDURES: If you choose to participate in this online survey, you will be asked
demographic questions about your child and your family to help us know more about your child's
living status and history of TS. You will then be asked specific questions about daily living
skills, school functions, leisure activity, social participation, mental functions, sensory functions,
and muscle and movement functions, and therefore we can know the areas in which your child
has experienced difficulty.
TIME COMMITMENT: The length of time required to complete this survey depends
on the number of needs your child has had. Most participants complete the survey in 40 to 60
192
minutes.
COSTS, RISKS, AND BENEFITS: There are no costs for participating in the study.
The survey asks about your childs daily functions; therefore, there may be a risk of becoming
upset when answering these questions. To minimize the risk, the survey allows you to skip
questions that you find unpleasant. If you find that you are upset after completing this survey
and want some help to manage your feelings, you can contact the faculty advisor, Janice Tona,
Ph.D., OTR. Dr. Tona is an occupational therapist with expertise in helping children and
families with TS and she will assist you in identifying local supports to help you. Dr. Tona can
be reached at (716) 829-6741 or tona@buffalo.edu.
The information you provide will help us to understand the needs of children with TS and
potential benefits from OT intervention. The information collected may not benefit you directly,
but what we learn from this study should provide general information to health care providers
about problems with daily living and the role of OT among children with TS.
CONFIDENTIALITY: This survey is confidential. The only link between you and the
information about your child will be your email address. No one outside of the researchers will
have access to your email address or will be able to link your answers to your email address. If
you withdraw from the study, all individually identifiable data provided by you will be destroyed
and not used for analysis. In order to monitor this research study, representatives from federal
agencies such as Office of Human Research Protection (OHRP) or representatives from the UB
Human Research Protections Programs may inspect the research records; this process may reveal
your identity. Your email address will not be kept after the study is completed. Any information
that you provide for the purpose of receiving a summary will be stored separately from the rest
of the study data, and will be destroyed at the end of the study. We will not leave cookies
193
(identifying files) on your system. A secure web server is provided to protect against
intermediate sites from intercepting your data. Please note that because you are answering
questions via the internet, your anonymity cannot be absolutely guaranteed. It may not be wise
to complete the survey at a coffee shop, library or workplace. Be sure to quit the web browser
after finishing the study so people cannot go back to the pages you completed.
VOLUNTARY PARTICIPATION: This survey is voluntary. If you choose not to
participate, it will not affect your standing with the Tourette Syndrome Association of Greater
New York State nor any other organization or physician that may have informed you of the study.
If you choose to participate but then decide to withdraw from the study, you can notify the
faculty advisor Janice Tona, Ph.D., OTR, at (716) 829-6741 or tona@buffalo.edu, and your
information will be deleted from the study if the study has not been completed.
If you choose to participate, please provide your email address so that you can be
contacted if any of your answers are not clear, but your email address will be kept separate from
the responses and will not be shared with anyone other than the researcher.
or complaints, or think the research have hurt you, you can contact Dr. Janice Tona, Clinical

1)
I have read the above information and consent to participate in the survey.
2)
Thank you for your interest in this survey. If you change your mind and would like to
participate, you may click on this link again or contact Janice Tona, Ph.D., OTR at
tona@buffalo.edu.
194
195
Appendix K
Thank-you email to participants
Dear Participant,
Thank you for taking the time to participate in our study Daily functioning of children
with Tourette syndrome: An exploratory study and complete our online survey. Your responses
will help us better understand the functional performance and intervention needs of children with
Tourette syndrome. If you selected to receive the research results, they will be sent to you via
email once we have finished analyzing the data. If you have further questions about this survey,
please contact Janice Tona, Ph.D., OTR at tona@buffalo.edu or (716) 829-6741.
Sincerely,
Sheng-Hui Liao
196
Appendix L
Expert comments on survey items
SURVEY ITEM
A. HOME-RELATED OCCUPATIONS
1. Bathing/showering
OT COMMENTS
2. Toileting (bowel and bladder

management including urinary
frequency, accident, potty training, and
bedwetting)
3. Toilet hygiene (including wiping and
flushing)
4. Dressing
5. Swallowing/eating (includes chewing,

swallowing, sensory acceptance of
foods, food selection, and food
quantity)
6. Feeding (includes selffeeding/bringing food to mouth)
7. Functional mobility (such as walking
around at home, or transfer to bathtub,
car, toilet, and so on)
8. Personal hygiene and grooming
9. Sexual activity (such as age

inappropriate discussion, gestures, online activity, or sexual activity)
10. Communication management (such as
using telephones, cell phones,
keyboards, computers or tablets, and
writing tools to communicate)
11. Community mobility (includes biking,
riding in buses, taxies, or other
transportation systems, and walking
around in the community)
12. Rest (including identifying the need to
Since you describe the below, might be

beneficial to describe using OTPF
language that include rinsing, drying and
transferring.
Think about including menstrual care.

We have seen this more related to OCD,
less to TS
Since you describe the above, might be
language that include selecting clothing,
fasteners, etc.
I appreciate the sensory acceptance part of
this.
Since you describe the above, might be

language that include shaving, hair care,
makeup, tooth brushing, nail care, etc.
This is a large component of TS which

relax and engaging in activities which
can restore energy)
197
includes poor state regulation. Might be

helpful to use or add the term from OTPF
that says engaging in activities that result
in a relaxed state to show arousal
management.
This occupation and sleep tend to be
significantly impacted by TS.
13. Sleep (includes following appropriate

sleep patterns, ceasing activities and
ready for bed, and sustaining a sleep
state without disruption)
A. EDUCATION-RELATED OCCUPATIONS
1. Math
2. Reading
3. Handwriting
4. Physical education
5. Extracurricular activities
B. OCCUPATIONS RELATED TO SOCIAL LIFE
1. Free Play
2. Organized Activities
Might be beneficial to explain further.
3. Social participation in the community
Might want to include OTPF of religious
(such as going to community
activities.
recreational activities, movies, or bow
ling)
4. Social participation with family
Important to include sibling relationship
(includes family gatherings and family
and role as indicated in OTPF.
recreation)
5. Social participation with peers/friends
C. MENTAL FUNCTIONS
1. Control of thoughts (no unwanted
obsessive, repetitive, or intrusive
thoughts)
2. Control of behaviors (no compulsive
behaviors or rituals)
3. Higher level thinking (includes
It is worded differently in the OTPF-III:
judgment, insight, and cognitive
higher-level cognitive functions.
flexibility)
4. Attention (sustained, selected, and
Distractibility is a large thing with TS, and
divided attention)
it is listed in this factor in the OTPF.
Might be helpful to list.
5. Memory (short term, long term and
working memory)
6. Sequencing (such as organizing and
planning projects)
7. Emotional coping
8. Energy and drive
Might be helpful to have descriptors of
198
OTPF that include motivation, impulse

control, appetite, etc.
D. SENSORY FUNCTIONS
1. Vision/seeing
2. Hearing
3. Vestibular/sense of moving
4. Taste
5. Smell
6. Proprioception/knowing where body is
in space
7. Tactile/touch
8. Pain (ability to feel pain in body with
injury may be increased ability or
decreased ability)
9. Temperature (ability to differentiate
hot from cold items when touching or
tasting)
E. MUSCLE AND MOVEMENT FUNCTIONS
1. Muscle strength (power)
2. Muscle endurance (sustained power)

3. Smoothness/control of voluntary
movements (coordination/no tremors)
4. Ability to restrain movements (no
involuntary tics)
Noted that this was left blank in the Parent

survey table provided-not sure if it is
supposed to be missing or not.
Including gross and fine motor skills.
To be worded more like the OTPF, it

could be written as ability to control
involuntary movements and use as a
descriptor as suppression.
5. Gait/walking patterns
OTHER COMMENTS
1. Some interesting domains that are listed as client factors in the OTPF that are seen a lot
in TS (and might be worth adding or including in some way) are: Perception,
Temperament and personality and muscle tone.
199
Appendix M
Expert and TSA-GNYS board member feedback on survey questions
Items on the preliminary parent survey
24. At what age (in years) did your child first
show signs of having TS?
Before 1
1
2
3
4
5
6
7
8
9
10
11
12
13
14
15
16
17
18
Over 18
25. Has your child ever had any other medical
diagnoses? (check all that apply)
26. Has your child ever taken any medication

for tic or non-tic symptoms?
Yes
No
If you selected yes, please specify the
medication, dosage, and the positive and
negative effect:______________
27. What were the reasons why your child
received OT? (check all that apply)
29. Did you find OT intervention beneficial
for addressing the reasons why your child
Comments
Add even without a diagnosis.
I didnt think you want the info on kids
over 18.
Add OCD and Anxiety Disorders

common comorbidities with TS.
My son shows signs of many of the
comorbidities but does not have an official
diagnosis...the school references issues
that lead to OCD and ADHD...is there a
section that discusses certain symptoms
but not have the disorder?
Too much info to put here.
It might be helpful here to have this more
like a list because many kids have been on
many medications. If youre looking at
any, even past medications, parents might
not remember dosages.
Add Tic management and
Organization/Time management.
Need more room in the field if you
selected other.

received OT?
Yes
No
Please share specifics about OT
intervention here: _______________
30. Now we are interested in learning about
your childs functions in home-related

occupations. Children with Tourette
syndrome frequently have periods of
increased symptoms (waxing) and periods
of decreased symptoms (waning). When
your child is in a period of waxing, what
difficulties in home-related occupations
are seen considering the developmental
level expected at his/her age? You can use
the comment box to share details if you
need to clarify your answer.
100. Please rate the greatest level of
difficulty you have seen with social

participation with peers/friends DURING
the waxing period.
1 My child has NOT displayed a
problem in this area.
2 My child has displayed MILD
DIFFICULTY in this area, but does
not require assistance.
3 My child has displayed
MODERATE DIFFICULTY in this
area, and required assistance and
adaptation or help to remain
functional.
My child has displayed SEVERE
DIFFICULTY in this area. My child
was unable to function at a typical
level, even with assistance and
adaptation.
Additional comments: ________________
200
A section for comments would be good for

this question. I felt OT was beneficial for
organizing and sensory, but not at all for
dysgraphia.
I believe the question should ask if any of

these have ever had an impact on your
child in the past and again in the present.
There are several things asked here that in
fact did effect my son at different times
during his development.
In the descriptions between mild and

moderate there is another layer. There are
certain things my son needs to work
through but doesnt need my help...but it is
noticed.
201
Appendix N
Parent comments on medication for childrens tic or non-tic symptoms
Medication
Abilify
Adderall
Adderall XR
Amphetamine
Dextroamphetamine
Citalopram
(Celexa)
Clonazepam
Clonidine
Clonidine patch
Concerta
(Methylphenidate)
Daytrana patch
Fluoxetine
Focaline
Guanfacine
(Intuniv)
Comments on the effects of the medication

Negative allergic reaction.
Minimal help with ADD.
Lost too much weight when taken at the age of 7. Works well at the
age of 17.
(gradually increasing dosage) - minimal help with ADD, but increased
tics
She is now on celexa 15mg. Seems to be positive so far.
Takes Clonazepam for Dystonia at night only.
Clonazepam made her tired.
Made sleepy, changed to use at night to help sleep. Increases tics
(temporary rebound effect) if stopped abruptly.
Chlonodine made him sleepy.
No effect on tics.
Has very little effect.
Clonadine at bedtime did lessen tic intensity she can not take it in the
morning she becomes to tired and pale. Also when initially given she
did have an allergic response hives. Was told to wait one week and try
it again. We did no more hives..
Clonodine meds have helped limit tics.
1 mg. 1 tablet at bedtime. Also 1/2 tablet in morning. She does not
receive the AM dose due to tiredness and paleness.
Helps to sleep at night.
My daughter tics all through the night. She started taking clonidine
9/14. This has helped her to sleep better.
Made him moderately drowsy, helped to a small degree with tics.
+ calmed him down, - made him tired.
Didnt make sleepy, but caused blistering of skin where applied.
Helps ADHD symptoms, has no effect on tics.
Positive ADHD.
Helped w/ ADHD but needed to go down from 36mg to 18mg b/c she
lost a lot of weight and wouldnt eat.
Minimal help with ADD, but increased tics.
Improves attention, but makes it hard for him to sleep.
Daytrana patch has worked very well for my son.
Calms anxiety.
He has tried many ADHD medications. Focaline also worked well.
Helps ADHD symptoms somewhat.
Same as Haldol (subdues coprolalia and physical tics only slightly,
fatigue).
1/2 mg twice a day. Sons tics have reduced greatly. Some days,
Haldol
Klonipin
(Clonazepam)
Metadate CD
Nortriptyline
(Pamelor)
Orap
Prozac
Resperidone
(Risperdal)
Seroquel
Sertraline
SSRIs
Stimulants
Strattera
Tenex
Topomax
Trileptal
Xenazine
Zoloft
202
totally unnoticeable.
Helped decrease tics.
Only for 2 weeks. No benefit seen only negative side effects.
Subdues coprolalia and physical tics only slightly, fatigue.
Made her tired.
Positive for anxiety.
Bad rebound issues I.e., unpleasant behavior and mood
Made tics worse (in retrospect, may have been rebound in tics from
stopping clonidine too abruptly).
Great but cant take SSRIs with this med for anxiety and cant take
adderall for ADD cuz its a stimulant.
No coprolalia but high anxiety, depression and moderate physical tics.
Orap for tics worked well but stopped to try anti-depressant.
Help with anxiety/OCD.
Negative anger issues.
Had some positive effects.
Helped a little.
Current medication which he has been on for two years. Works well
for managing tics and reducing severity. Small degree of drowsiness
but nothing too bad.
Noticed he was more calm, had to d/c due to severe headaches and
dizziness.
Works well for managing tics and reducing severity.
+ controls tics, - can only take short term.
Did not notice a difference just made [my child] tired.
Drastically reduced anxiety and depression.
Has very little effect. Sertraline seems to help a little with the anxiety.
Sertraline for anxiety. Helps reduce severity.
Ineffective, side effects.
Exacerbated the tics.
Allergic reaction.
Helps with anxiety.
Seemed to help with ADD, but appeared to cause sadness and crying
(anxiety/depression?).
Positive effect decrease in tics; negative effect sedating.
Many tries at meds, tenex did nothing.
Moodiness.
Made him severely drowsy, almost zombie like.
Positive helps somewhat with tics.
Takes Trileptal for seizures and as a mood stabilizer.
Positive helps somewhat with tics.
He was on Zoloft for a very short time. It was awful and made him
maniac. I took him off within 2 months.
Helped with anxiety and depressant but side effects were increasing
(hyperactivity, impulsiveness, skin picking etc) so it was stopped.
Helps with anxiety/OCE.
203
Negative anger issues.

Tried zoloft made him angry.
Took one medication briefly a few years ago because a motor tic was
causing physical pain. Do not recall which medication. Caused him to
be very lethargic / nonresponsive, so we discontinued use after two
weeks. Thankfully the tic changed.
204
Appendix O
Parent comments on the OT services their child received
Some interventions were helpful, others were not. For example handwriting intervention
was not helpful he wrote letters bottom upwards, OT tried to correct it which only made it
worse as he would then start from the top, then have to still write the letter bottom up
creating twice as much work to produce one letter.
Private OT has worked great in conjunction with school and private counselor to help my
son meet goals and learn coping/calming skills for anxiety. He has done reflex integration
therapy, listening program, how does your motor run, handwriting programs like
handwriting without tears (also used by the school so the techniques matched what school
was doing), creative and fun ways to help with relaxation and breathing (yoga, different
bubble blowers, stretching etc) working on focus and visual charting etc (he has been in OT
for 4 years or so!
It was through the school he attended and felt like they did not want to be bothered with
him.
It was beneficial because I learned of strategies to help at home. But ultimately, it was
disruptive to my son, because he had to be pulled out of class to receive OT, and for a child
already struggling with organization, and attention, missing any class time is detrimental to
his school performance.
Child had therapy weekly on site at OTAS. Child received school based therapies as well.
Parents trained on how to do at home daily therapies and as needed between appointments.
My child has se severe organization issues and time management needs, but these are
handled with his special Ed teacher and speech therapy person. I have never had an Ot offer
to address.
[My child] went to a therapist for a year who specializes in CBIT for Tourette Syndrome.
Shane found the techniques learned in these sessions to be moderately helpful.

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DAILY FUNCTIONING OF CHILDREN WITH TOURETTE SYNDROME: AN

A thesis submitted to the

Department of Rehabilitation Science

ProQuest Number: 1600781

All rights reserved

DAILY FUNCTIONING OF CHILDREN WITH TOURETTE SYNDROME

DAILY FUNCTIONING OF CHILDREN WITH TOURETTE SYNDROME

DAILY FUNCTIONING OF CHILDREN WITH TOURETTE SYNDROME

DAILY FUNCTIONING OF CHILDREN WITH TOURETTE SYNDROME

Signs, symptoms, and diagnostic features...................................................................... 3

Premonitory urges and other sensory issues...........................................................

Domain of occupational therapy............................................................................. 19

DAILY FUNCTIONING OF CHILDREN WITH TOURETTE SYNDROME

Sample Demographic and health characteristics............................................................ 39

Differences in functional performance before, during, and after a waxing period........

Predictors of functional limitations during a waxing period.......................................... 43

Occupational therapy services and functional performance during a waxing period....

Factors influencing functional performance in children with Tourette syndrome......... 50

Implications for occupational therapy practice.....................................................................

DAILY FUNCTIONING OF CHILDREN WITH TOURETTE SYNDROME

Appendix A: Finalized parent survey............................................................................. 80

Appendix C: Sample of the pilot survey........................................................................

Appendix D: University at Buffalo Institutional Review Board approval letter............ 173

Appendix F: Experts consent form................................................................................. 178

Appendix I: Invitation to members of the TSA-GNYS.................................................

Appendix J: Parent consent form for members of the TSA-GNYS...............................

Appendix K: Thank-you email to participants...............................................................

Appendix L: Expert comments on survey items............................................................

Appendix O: Parent comments on the OT services their child received.......................

DAILY FUNCTIONING OF CHILDREN WITH TOURETTE SYNDROME

Demographic Characteristics of the Children with Tourette Syndrome and

Diagnosis-related Characteristics of the Children with Tourette Syndrome....... 69

Comorbid Disorders Experienced by the Children with Tourette Syndrome.....

Functional Limitations in Occupations during a Waxing Period........................ 71

Functional Limitations in Body Functions and Performance Skills during a

Differences in Functional Performance in Occupations Before, During, and

Differences in Functional Performance in Body Functions and Performance

Predictors of the Experience of Functional Limitations in Occupations, Body

Comparison of Occupations during a Waxing Period by Receiving

Comparison of Body Functions and Performance Skills during a Waxing

DAILY FUNCTIONING OF CHILDREN WITH TOURETTE SYNDROME

Aspects of the domain of OT..............................................................................

DAILY FUNCTIONING OF CHILDREN WITH TOURETTE SYNDROME

DAILY FUNCTIONING OF CHILDREN WITH TOURETTE SYNDROME

DAILY FUNCTIONING OF CHILDREN WITH TOURETTE SYNDROME

DAILY FUNCTIONING OF CHILDREN WITH TOURETTE SYNDROME

DAILY FUNCTIONING OF CHILDREN WITH TOURETTE SYNDROME