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Fine-motor skill deficits in childhood predict adulthood tic severity and global
psychosocial functioning in Tourette's syndrome

Article in Journal of Child Psychology and Psychiatry · June 2006

DOI: 10.1111/j.1469-7610.2006.01561.x

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Journal of Child Psychology and Psychiatry 47:6 (2006), pp 551–559 doi:10.1111/j.1469-7610.2005.01561.x

Fine-motor skill deficits in childhood predict

adulthood tic severity and global psychosocial
functioning in Tourette’s syndrome
Michael H. Bloch,1 Denis G. Sukhodolsky,1 James F. Leckman,1,2 and Robert
T. Schultz1,3
1
Yale Child Study Center, USA; 2Department of Pediatrics and 3Department of Radiology, Yale School of Medicine,
USA

Background: Most children with Tourette’s syndrome (TS) experience a significant decline in tic
symptoms during adolescence. Currently no clinical measures have been identified that can predict
whose tic symptoms will persist into adulthood. Patients with TS have deficits on neuropsychological
tests involving fine-motor coordination and visual-motor integration. We seek to determine if these
neuropsychological tests are useful in predicting future symptom severity. Methods: Thirty-two
children, aged 8–14, with TS underwent clinical evaluation and a focused neuropsychological testing
battery consisting of the Purdue Pegboard, Beery Visual-Motor Integration (VMI) Test and the Rey-
Osterreith Complex Figure Task (RCFT). A follow-up clinical assessment was performed on these
children an average of 7.5 years later. Ordinal logistic regression analysis was used to correlate
neuropsychological testing at Time 1 with tic severity, OCD severity and global psychosocial functioning
at Time 2. Results: Poor performance with the dominant hand on the Purdue Pegboard test predicted
worse adulthood tic severity and correlated with tic severity at the time of childhood assessment. Poor
performance on the VMI and Purdue Pegboard tests (both dominant and non-dominant hand) also
predicted worse adulthood global psychosocial functioning. None of the neuropsychological tests were
useful in predicting the future course of OCD symptoms in TS patients. Conclusion: Fine motor skill
deficits may be a predictor of future tic severity and global psychosocial function in children with TS. We
hypothesize that performance on the Purdue Pegboard test may serve as a useful endophenotype in the
study of TS and provide a rough measure of the degree of basal ganglia dysfunction present in TS
patients. Keywords: Tourette’s syndrome, obsessive-compulsive disorder, motor skills, neuro-
psychology. Abbreviations: CY-BOCS: Children’s Yale-Brown Obsessive Compulsive Scale; GAS:
Global Assessment Scale; OCD: obsessive-compulsive disorder; RCFT: Rey-Osterreith Complex Figure
Task; TS: Tourette’s syndrome (TS); VMI: Beery Visual-Motor Integration Test; YGTSS: Yale Global Tic
Severity Scale.

Tourette’s syndrome (TS) is a childhood onset
neuropsychiatric disorder that is characterized by
the onset of both motor and vocal tics prior to the
age of 18. Previous longitudinal studies of Tourette’s
syndrome have demonstrated that the tics of TS
typically have an onset around the age of 5–6 and
reach their worst-ever severity between the ages of
10–12 (Erenberg, Cruse, & Rothner, 1987; Leckman
et al., 1998; Pappert, Goetz, Louis, Blasucci, &
Leurgans, 2003). Approximately one-half to two-
thirds of children with TS will then experience a
substantial decrease or remission of tics during
adolescence (Bloch et al., in press b; Leckman et al.,
1998; Pappert et al., 2003). However, the continu-
ation of tics into adulthood can have serious con-
sequences that may include self-injurious tics and
those that cause social unease, such as coprolalia
(Erenberg et al., 1987). Furthermore, approximately
one-third of these children diagnosed with TS will
develop clinically significant symptoms of obsessive-
compulsive disorder (OCD) by adulthood (Bloch
et al., in press b). These OCD symptoms may, on
average, have a greater effect on the adulthood

2005 The Authors
Journal compilation
2006 Association for Child and Adolescent Mental Health.
Published by Blackwell Publishing, 9600 Garsington Road, Oxford OX4 2DQ, UK and 350 Main Street, Malden, MA 02148, USA
psychosocial functioning of children diagnosed with
TS than the tics themselves (Erenberg et al., 1987).
In this study we seek to address whether neuro-
psychological testing can be used to predict adult-
hood symptom severity in patients with TS.
Previous studies have consistently demonstrated
that both children and adults with TS have deficits
on visual-motor integration (VMI) tasks such as
Bender-Gesalt Test and the Beery Visual-Motor
Integration Test when compared to normative data or
normal controls (Brookshire, Butler, Ewing-Cobbs,
& Fletcher, 1994; Lucas, Kauffman, & Morris, 1982;
Schultz et al., 1998; Shapiro, Shapiro, & Clarkin,
1974; Shapiro, Shapiro, Bruun, & Sweet, 1978).
Additionally, fine-motor coordination deficits in TS
subjects have been demonstrated on both the
grooved pegboard and Purdue Pegboard tests
(Bornstein, 1991a, 1991b; Bornstein & Yang, 1991;
Hagin, Beecher, Pagano, & Kreeger, 1982; Schultz
et al., 1998). These VMI and fine-motor skill deficits
in the original TS literature helped pinpoint the
caudate nucleus, basal ganglia and fronto-cortico-
striatal circuits as the neuroanatomical region of
552 Michael H. Bloch et al.
interest in TS research (Schultz, Carter, Scahill, &
Leckman, 1999).
Neuropsychological deficits have also been found
in comorbid disorders commonly associated with TS.
Patients with ADHD have consistently demonstrated
deficits of executive function (Barkley, Grodzinsky, &
DuPaul, 1992). TS patients with comorbid ADHD
have deficits on copying and long-term recall of the
Rey-Osterreith Complex Figure (RCFT) and excutive
function tasks such as the Trail-making Test, the
Hayling Test and Wisconsin Card Sorting Test when
compared to TS patients without comorbid ADHD
(Channon, Pratt, & Robertson, 2003; Harris et al.,
1995; Schuerholz, Baumgardner, Singer, Reiss, &
Denckla, 1996; Yeates & Bornstein, 1994). In
neuropsychological studies, TS patients with co-
morbid ADHD are frequently treated as a separate
study population (compared to TS patients without
comorbid disease) (Schultz et al., 1998). Further-
more, co-occurring ADHD is significantly associated
with deficits in psychosocial functioning in children
with TS and OCD (Sukhodolsky et al., 2003, 2005).
For these reasons we excluded all TS subjects with
comorbid ADHD from this follow-up sample.
Neuropsychiatric testing of adults with OCD has
consistently demonstrated deficiencies in visual
memory tasks such as immediate and delayed recall
of the RCFT (Boone, Ananth, & Philpott, 1991; Sav-
age, Baer, & Keuthen, 1995; Schultz, Evans, & Wolff,
1999). In OCD patients, poor performance on this
task has been linked to encoding and retrieval defi-
cits (Savage et al., 1999) and also been associated
with reduction in the size of the left anterior orbito-
frontal cortex on structural MRI studies (Choi et al.,
2004).
OCD patients have also demonstrated consistent
deficits on oculomotor tests of response inhibition.
Both adults with OCD and psychotropic-naı̈ve,
newly diagnosed, non-depressed children with OCD
have demonstrated deficits on oculomotor tests of
response inhibition, but not for delayed response
time or event anticipation, when compared to normal
controls (Tien, Pearlson, Machlin, Bylsma, & Hoehn-
Saric, 1992; Rosenberg et al., 1997). Furthermore,
the proportion of errors on oculomotor tests of re-
sponse inhibition correlated with OCD severity in the
psychotropic-naı̈ve children with OCD (Rosenberg
et al., 1997). OCD patients have also demonstrated
deficits of response inhibition on Go-No/Go tasks
(Malloy, Rasmussen, Braden, & Haier, 1989; Ban-
non, Gonsalvez, Croft, & Boyce, 2002). TS patients
with comorbid OCD have similarly demonstrated an
increased number of commission errors on Go-No/
Go tasks compared to normal controls (Muller et al.,
2003). By contrast, these same deficits on Go-No/Go
tasks have not been found in TS patients without
comorbid OCD (Serrien, Orth, Evans, Lees, & Brown,
2005). However, to our knowledge, no studies have
directly compared response inhibition in TS patients
with and without comorbid OCD.
Journal compilation
2006 Association for Child and Adolescent Mental Health.
Our a priori hypotheses were that poor childhood
VMI and Purdue Pegboard performance will be
associated with worse childhood tic severity and
predict increased adulthood tic severity in children
with TS. Furthermore, we predicted that childhood
deficits on RCFT will be associated with increased
future OCD symptom severity in children
with TS. In a set of exploration analyses, we
examined how these measures correlate with tic
severity at the time of neuropsychological exam-
ination and predict adulthood global psychosocial
functioning.
Methods
Subjects
The 32 subjects included in this study were previ-
ously evaluated at the Yale Child Study Center Tic
Disorder Clinic, had previously participated in MRI
imaging studies (Peterson et al., 2001, 2003) and
neuropsychological studies (Schultz et al., 1998) in
childhood and their follow-up data were reported on
in previous longitudinal studies (Bloch et al., in press
b; Bloch, Leckman, & Peterson, in press a). Eligible
subjects had: 1) a previous diagnosis of TS, 2) an MRI
scan and detailed neuropsychological evaluation per-
formed prior to age 14 (Time 1), and 3) they were
older than age 16 at follow-up (Time 2). Exclusionary
criteria in these earlier studies included a history of
prior seizure, head trauma with loss of consciousness,
ongoing or past substance abuse or an IQ below 80. A
comorbid diagnosis of ADHD at Time 1 was an addi-
tional exclusion factor. The high comorbidity rate of
ADHD among TS patients has been cited as a factor
clouding our current understanding of the neuro-
psychological profile of TS (Schultz, Carter, Scahill, &
Leckman 1999), hence these subjects were excluded
from analysis.
The 32 subjects who completed the Time 2 follow-up
component of our study elected to participate from an
eligible sample of 41 evaluated at Time 1. Reasons for
non-participation included subject refusal to partici-
pate (N ¼ 8) or inability to locate subjects (N ¼ 1).
Demographic measurements did not differ statistically
significantly between participating and non-participat-
ing subjects as assessed during initial evaluation at
Time 1 (Table 1).
Interview procedure at Time 1
Assessment at Time 1, when subjects were under age 14,
included current and worst-ever measures using the Yale
Global Tic Severity Scale (YGTSS) and Children’s Yale-
Brown Obsessive-Compulsive Scale (CY-BOCS) (Leck-
man et al., 1989; Scahill et al., 1997). The Schedule for
Tourette and Other Behavioral Syndromes, which in-
cludes the Kiddie-Schedule for Affective Disorders and
Schizophrenia Epidemiologic Present and Lifetime Ver-
sion for diagnosis in children and more detailed sections
on TS and OCD, was used to screen for comorbid psy-
chiatric illnesses (Ambrosini, Metz, Prabucki, & Lee,
1989; Kaufman et al., 1997; Pauls & Hurst, 1996).

2005 The Authors
 Fine motor skill deficits predict future symptom severity in Tourette’s syndrome 553

Table 1 Demographic comparison between participating and Spitzer, Fleiss, & Cohen, 1976). Screening for any co-
non-participating Tourette syndrome patientsa morbid psychiatric conditions was conducted with the
Structured Clinical Interview for DSM-IV Axis I Dis-
Participants Non-participants
orders (SCID-1). Additional assessment included a
N 32 9 thorough medication history, specific inquiry about
Age 11.4 ± 1.5 11.0 ± 1.1 ADHD symptoms and age of worst-ever tic and OC
Gender (M/F) 24/8 8/1 symptoms. Eight clinical evaluations relied solely on
Handedness (R/L) 38/8 9/0 information provided by a parent who lived in the same
IQ 111.1 ± 14.9 111.0 ± 12.2 home as the research subject. Compensation was
OCD 9 (28%) 2 (22%) provided for participation.
Anti-tic medication 16 (50%) 2 (22%)
Neuroleptic use 6 (19%) 0
SSRI use 3 (9%) 0
Data analysis
YGTSS 19.5 ± 8.9 20.8 ± 7.1
YGTSS worst-ever 28.5 ± 6.1 23.9 ± 5.8 All statistical procedures were performed using SPSS
CY-BOCS 2.0 ± 3.6 3.4 ± 6.3 version 12.0. Histograms were generated to examine the
Purdue Pegboard ).66 ± 1.35 ).66 ± 1.35
distribution of YGTSS, CY-BOCS and GAS scores at
dominant hand
Time 2. Given the large proportion of subjects with no
Purdue Pegboard ).58 ± .98 ).59 ± .96
non-dominant hand current tic or obsessive-compulsive symptoms at Time
Purdue Pegboard ).82 ± 1.31 ).98 ± 1.23 2, the data for YGTSS and Y-BOCS did not have a
bimanual normal distribution. GAS ratings at follow-up, due to
VMI ).27 ± 1.35 ).16 ± .79 the high number of subjects with scores greater than
RCFT – copying ).48 ± .79 ).60 ± .73 90, also did not have a normal distribution. These rat-
RCFT – long-term recall ).98 ± .83 )1.73 ± .50 ing scales were transferred into ordinal groupings prior
a
to hypothesis analysis in order to maintain important
There were no significant differences (p < .05) between any of
distinctions in symptom severity while also maintaining
the variables examined between participating and non-partici-
ordinal groups of roughly equal subject numbers. The
pating subjects using chi-square test or student t-tests.
YGTSS ¼ Yale Global Tic Severity Scale; CY-BOCS ¼ Chil- ordinal groupings for YGTSS score (range: 0–50) at fol-
dren’s Yale-Brown Obsessive-Compulsive Scale; VMI ¼ Beery low-up used in these analyses were 0 (N ¼ 10), 1–9
Visual-Motor Integration Test; RCFT ¼ Rey-Osterreith Com- (N ¼ 9), 10–19 (N ¼ 6), and >20 (N ¼ 7). These ordinal
plex Figure Task. Neuropsychological testing results are pre- grouping for YGTSS scores roughly correspond to
sented as z-scores according to normative data based on age absence of tics (YGTSS ¼ 0), minimal tic symptoms
and gender for each neuropsychological test. Tourette’s syn- (YGTSS: 1–9), mild tic symptoms (YGTSS: 11–19) and
drome patients performed below age-expected normal values moderate-to-marked tic symptoms (YGTSS > 20). For
for all neuropsychological tests reported. CY-BOCS score (range: 0–40) at follow-up groupings for
ordinal logistic regression were 0 (N ¼ 18), 1–9 (N ¼ 5),
Focused neuropsychological testing was performed in and >10 (N ¼ 7). These ordinal groupings for CY-BOCS
a uniform battery over the course of two sessions last- scores roughly correspond to absence of OCD symp-
ing on average two hours each. IQ testing was per- toms (CY-BOCS ¼ 0), subclinical OCD symptoms (CY-
formed with the Kaufman Brief Intelligence Test BOCS: 1–9) and OCD symptoms of clinical significance
(Naugle, Chelune, & Tucker, 1993). Visual Motor In- (CY-BOCS > 10). The ordinal groupings for GAS ratings
tegration was assessed with the Beery-Buktenica were £70 (N ¼ 6), 71–80 (N ¼ 4), 81–90 (N ¼ 10),
Visual Motor Integration Test (VMI) (Beery & Buktenica, 91–100 (N ¼ 12).
1989). Visual Memory was tested with the RCFT using In SPSS 12.0 ordinal regression with a logit link was
the Taylor Scoring System (Rey, 1941; Osterreith, used in analysis. Ordinal groupings for YGTSS, CY-
1944). The copying and long-term delayed recall para- BOCS and GAS ratings were used as the dependent
digms of the RCFT were specifically examined. Fine variable in analysis. Neuropsychological testing results
motor skill was tested using a timed peg-placing test, that were converted into z-scores based on previously
the Purdue Pegboard (Tiffen, 1968), using the proce- reported age and gender norms were used as the pre-
dures described previously (Schultz et al., 1998). No dictor variable in analysis (Beery & Buktenica, 1989;
participants in this current sample had tics involving Lezak, 1995; Spreen & Strauss, 1991; Tiffen, 1968). For
their hands that interfered directly with performance on each model both the Pearson goodness-of-fit test and
this measure. Dominant, non-dominant and bimanual the test of parallel lines were checked for p > .05 to
handed test conditions were administered to all sub- ensure the validity of our model (Kleinbaum & Klein,
jects. A single RCFT test and a single VMI test were 2002). Additionally, for each of the continuous covari-
excluded from analysis due to poor subject effort on ates the assumption of linearity was checked graphic-
exam. ally by plotting the cumulative logits of the ordinal
ratings against each covariate. Each plot demonstrated
that the ordinal responses were linearly related to the
Interview procedure at Time 2
covariates, thus validating the use of ordinal linear
After providing written informed consent, subjects and regression for this analysis (Bender & Grouven, 1997).
their parents were interviewed by trained investigators For the correlations of neuropsychological testing
at Time 2. Assessments included current and worst- results with current tic symptom severity at Time 1 a
ever YGTSS and CY-BOCS ratings. Overall psychosocial simple linear regression analysis was used since the
functioning was rated using the Global Assessment distribution of this outcome variable is normal. OCD
Scale (GAS) after the follow-up interview (Endicott, symptom severity at Time 1 was not analyzed in this

2005 The Authors
Journal compilation
2006 Association for Child and Adolescent Mental Health.
554 Michael H. Bloch et al.
current study given the small number of subjects with
significant OCD symptoms at Time 1. The threshold for
statistical significance was set at p < .05 to maximize
hypothesis generation. All statistical analyses were two-
tailed.
All results were assessed carefully for the possible
influence of medication use. Medication classes that
were considered included use of any medication for tics,
as well as alpha-2 agonists and neuroleptic use at Time
1. Any subject taking medications of interest was
eliminated from the analysis and the b coefficient for the
significant result recalculated. Only b coefficients out-
side the 95% confidence interval of the full study sam-
ple are reported as significant.
Results
At Time 1, when subjects were an average age of
11.4 ± 1.5 (mean ± SD), all subjects had previously
received a diagnosis of TS. All of our subjects had
previously experienced clinically significant tic
symptoms (YGTSS ‡ 15). Twenty-four of the 32
subjects were experiencing clinically significant tic
symptoms (YGTSS ‡ 15) at Time 1. Only 2 of the 32
subjects reported experiencing clinically significant
OCD symptoms at Time 1 (CY-BOCS ‡ 10) although
9 had received a previous OCD diagnosis. Sixteen of
the 32 subjects at Time 1 were taking medications to
combat their tic symptoms. Eleven subjects were
medicated with alpha-2 agonists (clonidine or
guanfacine) and 6 were medicated with neuroleptics
(5 – typical, 1 – atypical) at Time 1. Three subjects
were additionally taking selective serotonin reuptake
inhibitors (SSRI) for a history of OCD symptoms at
Time 1.
Despite having above average IQ scores, consistent
with our prior report on a sample that included these
cases (Schultz et al., 1998), subjects performed one-
half to a whole standard deviation below the nor-
mative average data of normal control subjects on
the visual-motor and fine motor tasks at Time 1. We
observed a similar phenomenon in the data from
those subjects who chose not to participate at follow-
up. Among participating subjects higher full-scale IQ
was associated with better performance on most of
the tasks in our focused neuropsychological testing
battery (Purdue Pegboard dominant-hand (Pearson
correlation coefficient (r) ¼ .38, p ¼ .03), Purdue
Pegboard bimanual (r ¼ .42, p ¼ .02), VMI (r ¼ .56,
p ¼ .001) and RCFT long-term recall (r ¼ .40, p ¼
.03)). Demographic and neuropsychological testing
data at Time 1 for participating and non-participat-
ing subjects is shown in Table 1.
When subjects underwent follow-up clinical
assessment an average of 7.4 years later, when
subjects had an average age of 18.8 ± 1.7, 10 of the
32 subjects reported being completely free of tic
symptoms at follow-up (YGTSS ¼ 0). Average YGTSS
at Time 2 was 8.9 ± 8.6 (median ¼ 8), approximately
half that of YGTSS score at Time 1 (19.5 ± 8.9). On
Journal compilation
2006 Association for Child and Adolescent Mental Health.
the other hand, at Time 2, 11 of the 32 subjects had
reported experiencing clinically significant OCD
symptoms that had developed since interview at
Time 1. Average CY-BOCS score at Time 2 for the 11
subjects with a history of clinically OCD symptoms
was 11.4 ± 10.7 compared to at Time 1 when it was
4.9 ± 4.9 (median ¼ 2).
Tic severity
Dominant (b ¼ ).44, F ¼ 7.1, t ¼ )2.7, p ¼ .012, R2
¼ .19) and non-dominant (b ¼ ).51, F ¼ 10.5, t ¼
)3.2, p ¼ .003, R2 ¼ .26) hand, as well as bimanual
(b ¼ ).36, F ¼ 4.4, t ¼ )2.1, p ¼ .045, R2 ¼ .13)
Purdue Pegboard scores were positively correlated
with current tic severity at Time 1, the time of neuro-
psychological assessment. Consistent with our a pri-
ori hypotheses, deficits on dominant handed Purdue
Pegboard (Parameter Estimate (PE) ¼ ).53 (95%CI:
()1.1,).02)), OR ¼ .59, p ¼ .04, Nagelerke R2 ¼ .13)
predicted worse tic severity at follow-up Time 2.
Neither non-dominant handed (PE ¼ ).63 (95%CI:
()1.3,.07)), OR ¼ .53, p ¼ .07) nor bimanual (PE ¼
).34 (95%CI: ().84,.16)), OR ¼ .71, p ¼ .19) Purdue
Pegboard performance at Time 1 predicted tic severity
at Time 2. Contrary to our hypothesis, VMI perform-
ance did not correlate either with tic severity at Time 1
(b ¼ ).07, F ¼ .13, t ¼ ).36, p ¼ .72) or predict tic
severity at Time 2 (PE ¼ ).03 (95%CI: ().61,.54)),
OR ¼ .97, p ¼ .91).
OCD severity
Contrary to our a priori hypothesis, childhood per-
formance on the Rey Complex Figure Task did not
predict OCD symptom severity at Time 2. Perform-
ance on copying (PE ¼ ).08 (95%CI: ()1.0,.85)),
OR ¼ .92, p ¼ .87) and long-term recall (PE ¼ .22
(95%CI: ().66,1.1)), OR ¼ 1.2, p ¼ .62) on the RCFT
at Time 1 did not predict OCD severity at Time 2.
Purdue Pegboard (Dominant: (PE ¼ ).16 (95%CI:
().68,.36)), OR ¼ .85, p ¼ .56), Non-dominant:
(PE ¼ ).54 (95%CI: ()1.3,.27)), OR ¼ .58, p ¼ .19),
Bimanual: (PE ¼ ).06 (95%CI: ().59,.48)), OR ¼ .94,
p ¼ .63) and VMI (PE ¼ .15 (95%CI: ().47,.78)),
OR ¼ 1.2, p ¼ .63)) performance similarly did not
predict future OCD severity.
Global psychosocial functioning
Poor performance on the dominant (PE ¼ .64 (95%CI:
(.10,1.2)), OR ¼ 1.9, p ¼ .019, R2 ¼ .18) and non-
dominant (PE ¼ .88 (95%CI: (.12,1.6)), OR ¼ 2.4,
p ¼ .024, R2 ¼ .18) Purdue Pegboard tasks predicted
worse global psychosocial functioning at Time 2.
Neither Bimanual Purdue Pegboard (PE ¼ .41
(95%CI: ().10,.92)), OR ¼ 1.5, p ¼ .12), VMI (PE ¼
.46 (95%CI: (.17,1.1)), OR ¼ 1.6, p ¼ .15), nor per-
formance on copying (PE ¼ ).04 (95%CI: ().86,.78)),

2005 The Authors
 Fine motor skill deficits predict future symptom severity in Tourette’s syndrome
OR ¼ .96, p ¼ .93) or long-term recall (PE ¼ ).03
(95%CI: ().81,.76)), OR ¼ .97, p ¼ .95) of the RCFT
predicted global functioning at Time 2.
Medication effects
No significant medication effects were observed for
use of dopamine receptor antagonists, alpha-2 agon-
ists, or for use of any anti-tic medication at Time 1.
Discussion
This study, to our knowledge, represents the first
attempt to explore the prognostic value of neuro-
psychological testing of fine motor skills in predicting
future disease severity in any neuropsychiatric ill-
ness. We found that both the dominant and non-
dominant handed deficits on a timed task involving
the placement of
1 inch metal pegs into a vertically
aligned set of holes (the Purdue Pegboard test) cor-
related with increased tic severity at the time of
childhood neuropsychiatric testing. Additionally,
deficits on the dominant handed Purdue Pegboard
test predicted increased tic severity at follow-up in
late adolescence, an average of 7.5 years after initial
neuropsychological testing. Both dominant and non-
dominant Purdue Pegboard deficits additionally
predicted worse global psychosocial functioning at
follow-up. Other neuropsychological measures tap-
ping visual motor skill were not significant predictors
of these outcomes, suggesting some specificity in the
processes tapped by the Purdue Pegboard to these
relationships, and negating any obvious relationship
to explanations centered on participant motivation
to perform well.
These results from Purdue Pegboard testing are
consistent with previous studies that have demon-
strated deficits in fine-motor coordination tests such
as the Purdue Pegboard in patients with TS (Born-
stein, 1991a, 1991b; Bornstein & Yang, 1991; Hagin
et al., 1982; Schultz et al., 1998). Purdue Pegboard
performance deficiencies have been linked to poor
social functioning in schizophrenia (Lehoux et al.,
2002). There are at least two possible explanations
for the ability of the Purdue Pegboard test to predict
future tic and global psychosocial functioning in
children with TS. First, poor Purdue Pegboard per-
formance is a reflection of poor fine-motor skills in
childhood. Fine-motor skill deficits make it difficult
for these children to succeed in activities such as
team sports, video games and musical instruments
that are instrumental to building self-esteem and
social relationships in these pivotal developmental
years. This experience of success is of increased
importance in children with TS because of the social
stigma that all too often accompanies their tics.
Second, poor Purdue Pegboard performance is a sign
of deficits in complex, visually guided or coordinated
movement that are likely mediated by circuits
involving the basal ganglia (Schultz et al., 1998).

2005 The Authors
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2006 Association for Child and Adolescent Mental Health.
555
Deficits on Purdue Pegboard testing have been
associated with reduced putamen volumes in Par-
kinson’s disease patients (Alegret et al., 2001) and
basal ganglia hyperperfusion in 99mTc-hexamethyl
propyleneamine oxime SPECT studies of patients
with subclinical hepatic encephalopathy (Catafau
et al., 2000). These findings are particularly of inter-
est given the considerable evidence of basal ganglia
abnormalities in TS patients. Reduced caudate vol-
ume has been previously demonstrated to be a mor-
phological trait of Tourette’s syndrome on structural
MRI (Peterson et al., 2003). Additionally, reduced
caudate volume on childhood MRI has been demon-
strated to predict future tic severity (Bloch et al., in
press a). We favor this hypothesis since it is able to
explain not only the Purdue Pegboard test’s ability to
predict future tic severity and global psychosocial
function but also its correlation with tic severity at the
time of neuropsychological assessment.
Deficits on Purdue Pegboard performance in
patients with TS may also serve as a useful endo-
phenotype in the study of the disorder. Endopheno-
types are unseen, measurable components of
psychiatric illness along the pathway between dis-
ease and genotype (Gottesman & Gould, 2003).
Purdue Pegboard performance deficits have been
associated with the diagnosis of TS (Bornstein,
1991a, 1991b; Bornstein & Yang, 1991; Hagin et al.,
1982; Schultz et al., 1998), and now, in this study,
associated with increased tic severity at follow-up in
early adulthood 7.5 years later. It is important to
note that Purdue Pegboard Performance is also
highly correlated with tic severity at the time of
childhood neuropsychological testing and is thus not
significantly predictive of adulthood tic severity if
childhood tic severity is controlled for in analysis.
However, Purdue Pegboard performance was a more
robust predictor of future adulthood tic severity than
childhood tic severity in our study (Bloch et al., in
press a). Tic severity in TS typically waxes and wanes
dramatically over the short term and is dependent on
environmental factors such as emotional excitement,
stress, fatigue, sleepiness and temperature (Peterson
& Leckman, 1998). By contrast, Purdue Pegboard
performance remains relatively stable with time, as
evidenced by a test–retest reliability of .8 when re-
tested at a one-month interval in children of this age
group (Smith & Hong, 2000). Therefore Purdue Peg-
board performance may thus represent a more stable
characteristic of the baseline neuropathology than
current childhood tic severity itself. However, future
study into the test–retest reliability of the Purdue
Pegboard with time specifically in children with tics
and into the hereditability of Purdue Pegboard per-
formance is critical to determining the utility of this
test as an endophenotype in the study of TS.
The Beery–Buktenica VMI Test, however, was
neither correlated with tic severity at the time of
childhood neuropsychological assessment nor able
to predict future tic symptom severity or global psy-
556 Michael H. Bloch et al.
chosocial functioning on follow-up assessment in
early adulthood. VMI deficits in multiple previous
studies have been demonstrated to be a neuro-
psychological testing correlate of a TS diagnosis
(Brookshire et al., 1994; Lucas et al., 1982; Schultz
et al., 1998; Shapiro et al., 1974, 1978). Our data
supports this finding (the mean VMI z-score for our
sample was –.27), while still failing to demonstrate
any ability of the VMI to predict future symptom
severity in children with TS.
Another important finding in this study was the
inability of Rey–Osterreith Complex Figure copying
or recall deficits to predict the development and
severity of OCD symptoms in these children with TS.
The later development of OC symptoms is a particu-
larly common outcome in children with tic disorders,
reportedly affecting a third to a half of these indi-
viduals (Bloch et al., in press a). Deficits on long-
term and immediate recall of the RCFT as explained
by impaired organizational strategies for figure
memory has been consistently reported in the adult
OCD literature (Boone et al., 1991; Choi et al., 2004;
Savage et al., 1995, 1999).
There are at least three possible explanations for
our negative findings with regard to the RCFT. First,
deficits on visual memory tasks such as the RCFT
represent a late neuropsychological manifestation of
prolonged OCD symptoms rather than an indicator
of neurological predisposition to the illness. Neuro-
psychological testing of children with OCD has pre-
viously suggested that children do not share the
deficits on neuropsychological testing found in adult
subjects, although RCFT was not included in their
test battery (Beers et al., 1999). Alternatively, it is
possible that the deficits on RCFT observed in adult
subjects are caused by secondary factors such as
depression or prolonged SSRI use that are associ-
ated with, but independent of, prolonged OCD
symptomatology (Schultz et al., 1999). In this case,
the neural processes underlying RCFT deficits
would be independent of those related to OCD
pathogenesis. Studies that correlate degree of RCFT
deficits with duration and severity of OCD symptoms
and control for medication use and comorbid disease
would definitively address this hypothesis. Addi-
tionally, obsessive-compulsive symptoms in patients
with comorbid tic disorders may represent a distinct
subtype of OCD without the hallmarks of the larger
disease entity on neuropsychological testing. Tic-
related OCD has been previously reported to involve
a disproportionately high percentage of compulsions
involving symmetry and exactness and touching and
tapping rituals and obsessions involving fear of harm
or sexual or violent imagery (Leckman et al., 1997).
These characteristics of obsessions and compulsions
in patients with tic-related OCD contrasts with
adult-onset OCD patients, who more commonly
experience contamination obsessions and cleaning
compulsions (Leckman et al., 1997). Patients with
tic-related OCD are also noted to have an earlier age
Journal compilation
2006 Association for Child and Adolescent Mental Health.
of illness onset and be less responsive to selective-
serotonin receptor inhibitor monotherapy than
adult-onset OCD patients (King, Leckman, Scahill, &
Cohen, 1999). Given that these subpopulations of
OCD patients often appear clinically distinct and
respond to medications differently, it would not be
unexpected if their neuropsychological profiles dif-
fered. Studies examining profiles on neuropsycho-
logical testing of OCD patients with and without
comorbid tics could help address this question.
Although the RCFT in children with TS does not
appear to be predictive of future OCD symptom se-
verity, our previous analysis has suggested that full-
scale IQ in childhood is. We have previously reported
that childhood IQ as measured by the Kaufmann
Brief Intelligence Test was predictive of OCD symp-
tom severity an average of 7.6 years later (Bloch
et al., in press a). In other prospective, longitudinal
studies increased IQ has been associated with the
diagnosis of OCD and predictive of an increase in
OCD symptom severity between adolescence and
adulthood (Peterson, Pine, Cohen, & Brook, 2001).
Our current study has several limitations. It re-
ports on a clinically referred population of children
with TS whose overall illness severity and rates of
comorbid illnesses are likely to be greater than in
children who have TS in the general population.
Furthermore, nearly one-quarter of eligible subjects
did not participate in the follow-up evaluation. Al-
though participating and non-participating subjects
did not differ significantly in any demographic or
clinical characteristics at the time of initial evalu-
ation, we cannot exclude the possibility that self-
selection bias affected our findings at follow-up.
Third, one-half of the study sample was taking
psychotropic medications at the time of neuro-
psychological testing. Although efforts were made to
discern any effects medication use could have exer-
ted on our results, this possibility cannot be entirely
discounted. However, previous studies have dem-
onstrated no influence of medication use on the
neuropsychological testing results of patients with
Tourette’s syndrome (Bornstein & Yang, 1991; Sch-
ultz et al., 1998). Fourth, we excluded TS patients
with comorbid ADHD from our study. Therefore our
findings should not be generalized to apply to those
children with TS who suffer from comorbid ADHD,
although it should be mentioned that inclusion of TS
patients with comorbid ADHD in our study sample
did not affect the findings in this study in any
appreciable way (data unreported).
Despite these limitations our study is, to our
knowledge, the first to address the potential pro-
gnostic role of focused neuropsychological testing in
predicting future tic, OCD and global psychosocial
functioning in children with TS. We found that Pur-
due Pegboard deficits predicted increased adulthood
tic severity in children with TS although not with the
predictive power of structural MRI measures (Bloch
et al., in press a). Purdue Pegboard deficits were

2005 The Authors
 Fine motor skill deficits predict future symptom severity in Tourette’s syndrome
additionally significantly able to predict worse global
psychosocial functioning at follow-up in adulthood
and, when combined with previously significant
clinical predictors (childhood tic severity), explained
29% variance at follow-up. Our results suggest that
the Purdue Pegboard testing may be used in combi-
nation with other indices to help predict outcome in
children with TS. Unlike our structural MRI findings,
this test is available and economically realistic to all
clinicians who treat patients with TS. Although we
did not discover any useful tests or neuropsycho-
logical battery for predicting the future development
or severity of OC symptoms in TS patients, our pre-
vious findings suggest that IQ may be a useful pre-
dictor of this outcome measure, with a higher
childhood IQ suggesting a predisposition for the later
development of OCD symptoms.
Acknowledgements
This study was supported in part by the Smart
Family Foundation, Jay and Jean Kaiser, Mr. Eric
Brooks, the Chasanoff Family and grants MH49351
(Dr. Leckman), MH30929, and RR00125 from the
National Institutes of Health, Rockville, MD.
We thank Drs. Lawrence Scahill, Heping Zhang,
Robert King, Paul Lombroso, Lawrence Staib and
Ms. Lilya Katsovich and Jessica Otka at Yale Uni-
versity School of Medicine, and Dr. Bradley Peterson
at the Columbia College of Physicians & Surgeons for
their assistance and encouragement.
Correspondence to
Robert T. Schultz, Yale Child Study Center, 230
South Frontage Road, PO Box 207900, New Haven,
CT 06510, USA; Email: Robert.schultz@yale.edu
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Manuscript accepted 19 July 2005