ArticlePDF Available

Abstract

Tourette syndrome, a condition first recognized in 1825, is characterized by the presence of multiple motor tics and one or more phonic tics. Individuals with Tourette syndrome may also demonstrate fluency failures in their speech. This study investigated the disfluencies and phonic tics in an 18-year-old affected male before and after a three week period of speech therapy. It was found that the speech pattern displayed by this subject did not completely conform to the classic pattern of stuttering but did bear more resemblance to cluttering. A limited number of therapy sessions resulted in a significant improvement of speech.
J. COMMUN. DISORD. 33 (2000), 227–240
© 2000 by Elsevier Science Inc. All rights reserved. 0021-9924/00/$–see front matter
655 Avenue of the Americas, New York, NY 10010 PII S0021-9924(00)00020-4
DYSFLUENCY AND PHONIC TICS
IN TOURETTE SYNDROME:
A CASE REPORT
JOHN VAN BORSEL
Ghent University Hospital, Ghent, Belgium
MARTINE VANRYCKEGHEM
University of Central Florida, Orlando, Florida
Tourette syndrome, a condition first recognized in 1825, is characterized by the presence of
multiple motor tics and one or more phonic tics. Individuals with Tourette syndrome may
also demonstrate fluency failures in their speech. This study investigated the disfluencies
and phonic tics in an 18-year-old affected male before and after a three week period of
speech therapy. It was found that the speech pattern displayed by this subject did not com-
pletely conform to the classic pattern of stuttering but did bear more resemblance to clutter-
ing. A limited number of therapy sessions resulted in a significant improvement of
speech. © 2000 by Elsevier Science Inc.
Educational Objectives:
The reader will learn (1) about the tics and disfluency types that
might be present in people with Tourette syndrome; and (2) that the fluency failures associ-
ated with this syndrome are not characteristic of stuttering.
KEY WORDS:
Tourette syndrome; Stuttering; Cluttering; Disfluency, Tics
INTRODUCTION
Tourette syndrome (TS) is a condition that was first described in 1825 by the
French physician J.M. Itard (Itard, 1825), who reported on the presence of
tics, barking sounds, and uncontrollable utterances of obscenities in a French
noblewoman, Marquise de Dampierre. A more detailed description of eight
additional cases was published in 1885 by another French physician, George
Gilles de la Tourette (Gilles de la Tourette, 1885), after whom the disorder was
named.
According to the DSM-IV (American Psychiatric Association, 1994), the
essential feature of TS is the presence of multiple motor tics and one or more
vocal tics that occur many times a day, recurrently throughout a period of
Address correspondence to John Van Borsel, Ph.D., UZ Gent, 2P1, Centrum voor Gehoor- en
Spraakrevalidatie, De Pintelaan 185, B-9000 Gent, Belgium. Tel:
32 9 240 22 96; Fax:
32 9 240 49
93; E-mail:
john.vanborsel@rug.ac.be
.
228 VAN BORSEL and VANRYCKEGHEM
more than one year, and cause marked distress or significant impairment in
social, occupational, or other important areas of functioning. TS has its onset
before the age of 18 years, and is not due to the direct physiological effects of
a substance or a general medical condition.
Two studies conducted in the 1980s (Kurlan, Behr, Medved, Shoulson,
Pauls, & Kidd, 1987; Rassas Cohn, Shames, McWilliams, & Ferketic, 1983)
estimated the prevalence of TS to be between a low of .01% and a high of
1.6%. The percentage prevalence at the high end of this range was, however,
not confirmed by more current studies that report a prevalence ranging from
.03% to 1% (Cardoso, Veado, & Teotoino de Oliveira, 1996; Van de Weter-
ing, Cath, & Buitelaar, 1996; Woods & Miltenberger, 1995). It is generally
agreed that TS is three to four times more likely among males than females
(Breakfield & Bressman, 1987; Cardozo, Veado, & Teotonio de Oliveira,
1996; Colligan, 1989; Rassas Cohn, Shames, McWilliams, & Ferketic,1983;
Singer & Walkup, 1991).
Research relative to the etiology and pathogenesis of TS has made it clear
that in most cases the disorder is genetically determined (Duffy, 1995). Most
likely there is an autosomal dominant inheritance with sex-specific penetrance
and variable expression (Pauls & Leckman, 1986; Van de Wetering & Pauls,
1996). It has been suggested that TS is caused by a dysfunction of central neu-
rotransmitter systems. Dopaminergic, serotonergic, noradrenergic, cholin-
ergic, GABAergic, as well as opoid systems have been found to show abnor-
malities in individuals with TS (Kurlan, Behr, Medved, Shoulson, Pauls,
Kidd, & Kidd, 1986; Malison, McDougle, Van Dyck, Scahill, Baldwin,
Seibyl, Price, Leckman, & Iniis, 1995; Singer, 1992; Singer & Walkup, 1991).
The exact neuroanatomic localization of the dysfunction is presently un-
known. Sites of pathogenesis that have been mentioned are the basal ganglia,
the limbic system, the (pre)frontal cortex, and the thalamus (Singer, 1997;
Singer & Walkup, 1991; Van Woerkom, Van de Wetering, & Buitelaar,
1996).
Motor and vocal tics are behaviorally at the core of TS. Motor tics consist
of intermittent movements. They are defined as simple if the movements are
brief and involve only one muscle group. They are considered complex if they
appear as longer, more purposeful sequential movements, such as hopping or
writing the same word over and over. Vocal tics consist of abnormal sounds
and noises. Because not all the abnormal sounds and noises made by patients
with tics are produced by the vocal cords, the term “phonic tics” is to be pre-
ferred over “vocal tics.” (Jankovic, 1997). Phonic tics are considered simple
when sudden, fast, single meaningless sounds or words are uttered. Linguisti-
cally meaningful utterances, on the other hand, are classified as complex
phonic tics. These are exemplified by rituals, unusual accents, and change in
speech intensity (Bruun, Cohen, & Leckman, 1997). Coprolalia and echolalia
also belong to the category of complex tics. They are defined, respectively, as
DYSFLUENCY IN TOURETTE SYNDROME 229
“obscene, aggressive, or otherwise socially unacceptable words or phrases”
(Bruun, Cohen, & Leckman, 1997) and “the involuntary repetition of all or
part of the speech partner’s utterance” (Lebrun, 1993).
Attention deficit disorder and obsessive compulsive behavior are com-
monly cited as being associated with TS. So, too, are alcoholism, drug abuse,
conduct disorders, anxiety, and eating as well as sleep disorders (Comings &
Comings, 1993). Recently, learning disabilities and speech disorders have
also been seen as part of TS. Individuals with TS have been described as hav-
ing delayed speech, poor diction, poor language expression, and as talking too
fast and too loud and exhibiting disfluent speech. The fluency failures de-
scribed range from “repetitions, hesitations, and false starts” (O’Quinn &
Thompson, 1980) and “interjections, whole-word repetitions, prolongations,
phrase and part-word repetitions, revisions” (Rassas Cohn, Shames, McWil-
liams,& Ferketic, 1983) to “stutter-like behavior” (Singer, Pepple, Ramage, &
Butler, 1978) and “stutter-like repetitions of sounds” (Aronson, 1980). Palila-
lia, a disorder of speech characterized by compulsive repetition of a phrase or
word that the patient often utters with increasing rapidity and with decre-
scendo of voice volume (Brain, 1961) has also been seen in persons with TS
(Cardoso, Veado, & Teotonio de Oliveira, 1996; Jankovic, 1997; Ludlow, Po-
linsky, Caine, Bassich, & Ebert, 1982).
Although the incidence of stuttering or at least stutter-like behavior among
those with TS is said to range from 15.3% (Pauls, Leckman, & Cohen, 1993)
to 31.3% (Comings & Comings, 1993), little knowledge exists relative to the
types of speech disruptions that they display. Data relative to the types of dis-
fluency, their frequency, and their locus are essentially nonexistent at this
time. Hard data as to the fluency failures of those with TS are critically needed
in order to study the variables that set the occasion for their occurrence and to
develop management procedures that will bring them under control. So, too, is
information relative to the affective and cognitive components which might be
associated with the fluency failures that they evidence. It would serve to make
differential diagnostic determination easier and therapeutic procedures more
appropriate and effective. The present case study of a person suffering from
TS is seen as a first step in the gathering of information on the speech-related
characteristics of this disorder.
CASE HISTORY AND INITIAL SCREENING
The subject of this study, G., was an 18-year-old right-handed male with a
clinical diagnosis of TS since the age of 12. He lived at home with his mother,
stepfather, one older sister, and a younger stepbrother. The stepfather joined
the family when G. was six years of age, two years after G’s parents divorced.
G. received vocational education until the age of 17. At school, he was fre-
quently the victim of bullying because of the tics he exhibited. At the time the
230 VAN BORSEL and VANRYCKEGHEM
present study was undertaken, G. was unemployed. Testing at age 18 years 3
months revealed that G. was of low-normal intelligence, with a verbal IQ that
was significantly lower (p
5%) than the performance IQ (WAIS: TIQ
86,
VIQ
82, PIQ
94). No history of TS or stuttering was reported in the family.
Disfluent speech was first observed, according to G.’s mother, at about the
age of 14. However, G. never received speech therapy. Medical records men-
tioned also the presence of coprolalia (which, according to his mother, was
also first noticed at age 14). Over the years, G. had been administered differ-
ent medicines, including Seroxat, Dipiperon, Risperidone, Risperdal, and In-
deral retard mitis. The medication was taken irregularly, however, or was
stopped prematurely. Between the ages of 18 and 20, G. was admitted to the
psychiatric ward of the Ghent University Hospital on three separate occasions
because of aggressive behavior, acting out, and amphetamine, cocaine, and al-
cohol abuse. During a period of hospitalization at age 18, he was referred to us
for a speech-language assessement with a request for possible therapy. An ini-
tial screening showed that G. had difficulties with verbal and graphic expres-
sion. He could not easily formulate ideas, sometimes halting and then again
speaking in short spurts. From time to time he also produced grammatical er-
rors, and his articulation was often slurred. Foremost, however, he showed a
great number of disfluencies in his speech, as well as many tics. His writing
was graphically irregular with many overwritings. G. did not use capitaliza-
tion, and spontaneous writing was characterized by chaining and repetition of
phrases.
DATA COLLECTION AND ANALYSIS
Data were collected on two separate occasions: once during the initial assess-
ment and a second time six weeks later upon dismissal from the psychiatric
ward. Between these, G. had received speech therapy for a period of three
weeks.
Data collected prior to therapy included: samples of automatized speech
(reciting days of the week and months of the year, and counting from 1 to 20);
word and sentence repetition (parts 2 to 5 of the subtest “Naspreken” from the
Dutch version of the Aachener Aphasie Test (Graetz, De Bleser, & Willmes,
1992); oral reading (Dutch version of
The North Wind and the Sun
; Interna-
tional Phonetic Association, 1974); conversational speech (about leisure ac-
tivities); semantic fluency (retrieval of names of animals, subtest of the Dutch
aphasia battery S.A.N.; Deelman, Koning-Haanstra, Liebrand, & Van De
Burg, 1981); final-word repetition (repeating the final word of sentences using
part 5 of the subtest “Naspreken”’ from the Dutch version of the Achener
Aphasie Test; (Graetz, De Bleser, & Willmes, 1992); and singing (G. sang a
non-standard version of a Dutch nursery song). In addition, the Erickson
S-scale of Communication Attitudes (Erickson, 1969) was administered. This
DYSFLUENCY IN TOURETTE SYNDROME 231
39-item questionnaire investigates a person’s attitude toward speech. Finally,
oral mechanism mobility was investigated, and inhibition of tics was probed
(The patient was asked to inhibit tics during two consecutive minutes of si-
lence).
The speech therapy following the initial assessment consisted of seven
half-hour sessions and was given by the first author. It focused on the use of
slowed and syllable-timed speech as well as on awareness training for the bi-
labial tics which G. demonstrated.
For the purpose of pre-post comparison, speech samples were once more
collected following the therapy sessions. G. orally read the text
The North
Wind and the Sun
(International Phonetic Association, 1974) and engaged in a
conversation (on the topic of cooking and food preferences).
Both speech sampling sessions took place in a sound-protected room in the
ENT department of the University Hospital and were video-recorded (Sony
Video Hi 8 camera). The samples were subsequently subjected to an ortho-
graphic transcription of the recordings by both authors (consensus transcrip-
tion) and to an analysis of the number and type of disfluencies for each speech
task sampled as well as an analysis of type and number of phonic tics in the
various speech tasks. For all analyses, consensus was reached by both authors.
For the analysis of disfluencies, an adaptation of the taxonomy of Johnson &
Associates (Johnson & Associates, 1959) was used. For the analysis of phonic
tics, an ad hoc procedure was adopted, distinguishing categories of tics ac-
cording to their acoustic and visual characteristics as they occurred in the
speech samples rather than by classifying them according to a predetermined
closed set of phonic tics.
RESULTS
Results of the analysis of G.’s disfluencies are shown in Table 1. The upper
part summarizes the results from the initial assessment, whereas the lower part
presents the results during oral reading and conversation after therapy. As can
be seen from this table, during the initial assessment, 450 of the 1770 syllables
uttered across different speech tasks (i.e., 25.4%) showed some type of disflu-
ency. This high number of disfluent syllables relative to the number of disflu-
encies stems from the fact that one disfluency might involve several syllables
(e.g., “he came came came home”
1 word repetition involving 3 syllables).
Disfluencies were most frequent during semantic fluency (46.3% of the sylla-
bles uttered) and conversation (32.3% of the syllables uttered). Disfluencies
were also observed during word repetition and automatic series but to a much
lesser degree (involving, respectively, 7.4% and 1.2% of the syllables ut-
tered). No disfluencies were observed during sentence repetition, oral reading,
final word repetition, and singing.
Overall, interjections were the predominant type of disfluency, accounting
232 VAN BORSEL and VANRYCKEGHEM
Table 1.
Type and Number of Disfluencies in Different Speech Tasks
TH # Syll Disfl
Syll PWR WR
Mono WR
Multi INTJ PR REV IP PROL BLOCK
Initial Assessment
Automatic
series 81 1
(1.2%) —— — 1 —— —
Word
repetition 68 5
(7.4%) —— — 3 —1 —— —
Sentence
repetition 99———— — ——
Oral reading 182
Conversation 1043 337
(32.3%) 13 12 12 51 30 18 6
Semantic
fluency 231 107
(46.3%) 54 3 53 9 3
Final word
repetition 15———— — ——
Singing 51 — — — — —
1770 450
(25.4%) 18
(8.%) 16
(7.1%) 15
(6.7%) 108
(48.2%) 39
(17.4%) 22
(9.8%) 6
(2.7%) ——
After Therapy
Oral reading 182
Conversation 369 27
(7.3%) 11 —5 4 2
Syll
Number of Syllables Uttered, Disfl Syll
Number of Disfluent Syllables, PWR
Part Word Repetition, WR Mono
Word Repetition of a Monosyllabic Word, WR Multi
Word Repetition of a Multisyllabic Word, INTJ
Interjection, PR
Phrase Repetition, REV
Revision, IP
Incomplete Phrase, PROL
Prolongation, BLOCK
Block.
DYSFLUENCY IN TOURETTE SYNDROME 233
for 48.2% of all disfluencies demonstrated. Also, phrase repetitions occurred
with a relatively high frequency (17.4%). Revisions (9.8%), part-word repeti-
tions (8%), repetition of monosyllabic words (7.1%), repetition of multi-syl-
labic words (6.7%), and incomplete phrases (2.7%) were less frequently emit-
ted speech disruptions. Prolongations and blocks did not occur at all.
The lower part of Table 1 shows that, post therapy, the overall number of
disfluent syllables demonstrated during conversation dropped dramatically
from 32.3% to 7.3%. Again, interjections and phrase repetitions were the
types of disfluency that occurred most frequently.
Table 2 summarizes the results of the analysis of phonic tics. Across all
speech tasks sampled during the initial session (left part of Table 2), G. pro-
duced a sum total of 52 phonic tics. More than one-third of these (34.6%) were
demonstrated during conversation. Another 21.2% and 19.2% were produced
during word repetition and the semantic fluency test, respectively. The vast
majority of the phonic tics (24/52, i.e., 46%) were simple tics involving the bi-
labial area, namely, sputtering, spitting, and smacking. Another category of tics
that was seen rather frequently (8/52, i.e., 15.4%) involved the laryngeal area
and consisted of an audible egressive or ingressive sound produced with open
mouth or an audible egressive, somewhat humming-like sound. Still other tics
included inhalation or exhalation, sniffing, slurping, and throat clearing.
Although G.’s records mentioned the occurrence of coprolalia and G. him-
self reported to us that he demonstrate coprolalia from time to time, no in-
stances were noted during data collection of the present study or during the
therapy sessions. Neither did we find any examples of echolalia. The only
complex phonic tic that was observed was a sudden increase in loudness. This
type of tic was observed five times.
When comparing the number of phonic tics per syllables uttered during the
initial session with the number of phonic tics produced after therapy (right
part of Table 2), a slight increase can be observed during conversation (from
18/1043, i.e., 1.7% to 8/369, i.e., 2.2%) and a decrease during oral reading
(from 4/182, i.e., 2.2% to zero). Qualitatively the tics were far less conspicu-
ous after therapy and were sometimes hardly audible.
On the Erickson S-scale, G. obtained a score of 28, which is almost two
standard deviations above the mean for non-stutterers and slightly above the
mean for stutterers. This indicates that G. exhibits a clinically significant neg-
ative attitude toward his speech. Oral mechanism mobility proved overall
good for tongue as well as for lip movements. Lateral movements of the
tongue, however, were associated with head movements. G. appeared capable
of inhibiting phonic tics for two consecutive minutes of silence, but he did
continue to show motor tics. While he was filling out the Erickson S-scale, it
was also observed that G. could not refrain from producing a word aloud now
and then, sometimes even with excessive loudness. The same tendency had
been noted during spontaneous writing and copying.
234 VAN BORSEL and VANRYCKEGHEM
Table 2.
Type and Number of Phonic Tics in Different Speech Tasks
Prior to Therapy After Therapy
Automatic
Speech Word
Repetition Sentence
Repetition Oral
Reading Conversation
Final
Word
Repetition Semantic
Fluency Total
Number Conversation Oral
Reading
Biliabial
Sputtering 1 4 1 1 8 1 3 19 4
Spitting 1 2 1 4
Smacking 1 — — 1 — —
Laryngeal
Ingressive — 2 1 13
71
Egressive — — 1 1 — —
Inhalation — 1 1 1 3
Exhalation 1 1 — — 2 — —
Sniffing 1 1 1 — 3
Slurping — 2 4 — 6 1
Increased
loudness ———3 25 2 —
Throat
clearing ——1 — — 1 — —
3
(5.8%) 11
(21.2%) 4
(7.7%) 4
(7.7%) 18
(34.6%) 2
(3.8%) 10
(19.2%) 52
(100%) 80
DYSFLUENCY IN TOURETTE SYNDROME 235
DISCUSSION
Several authors have reported the presence of disfluent speech in people with
TS (Aronson, 1980; O’Quinn & Thompson, 1980; Rassas Cohn, Shames,
McWilliams, & Ferketic, 1983; Singer, Pepple, Ramage, & Butler, 1978), and
at least some of them label this disfluent speech as stuttering (Comings &
Comings, 1993; Pauls, Leckman, & Cohen, 1993). The results of the present
study confirm that disfluent speech may be encountered in individuals with
TS. In the case described in this article, more than one-quarter of the syllables
in the speech sample collected prior to speech therapy were affected by disflu-
encies. However, whether the disfluent speech in our subject is genuine stut-
tering is less clear. Only 15% of the total number of disfluencies in the initial
assessment were of the kind that most researchers would consider to be char-
acteristic of stuttering (part-word repetitions, 8%; monosyllabic word repeti-
tions, 7%). The vast majority of the disfluencies (85%) were disfluencies that
are also seen in “normal” speakers. In this respect, our findings run parallel to
those of Rassas Cohn and colleagues (Rassas Cohn, Shames, McWilliams, &
Ferketic, 1983), who studied the disfluencies in a 25-year-old male with TS.
They reported that in spontaneous speech, 38% of the subject’s disfluencies
were interjections, 26% were whole-word repetitions, 9% were phrase repeti-
tions, and 6% were revisions. Only 21% of their subject’s fluency failures
were disfluencies characteristic of stuttering (prolongations, 15%; repetitions, 6%).
Evidently the speech pattern displayed by our subject does not completely
conform to the classic pattern of stuttering. It does, however, seem to bear
more resemblance to another type of fluency disorder, that of cluttering. As is
typically seen in cluttering, G. used many interjections and repeated whole
words and phrases. At times he also showed a rapid speech rate and spoke in
spurts. Furthermore, it had been observed during the initial screening that G.’s
wording was often disorganized and confused. Also, his grammar was rather
poor, his articulation was sometimes slurred, and his writing was disinte-
grated. All of these are patterns that are considered suggestive of cluttering
(Daly, 1993). In addition, the observation that a limited number of therapy
sessions resulted in a significant improvement of G.’s speech, at least tempo-
rarily, could indicate cluttering. It is a well-known fact that clutterers can, for
a period of time, control their speech. Insofar as the speech pattern displayed
by our subject is similar to that of other patients with TS, the present study
raises the question whether the disfluency associated with TS is not a manifes-
tation of cluttering rather than stuttering. A counter-argument for cluttering is
the fact that G. showed a negative speech-associated attitude that was signifi-
cantly elevated. A mal-attitude toward speech is not typically seen among
clutterers. It is, however, common among stutterers.
Whether the finding that the pattern of fluency failures found in this subject
is more reminiscent of cluttering than of stuttering implies that the underlying
236 VAN BORSEL and VANRYCKEGHEM
mechanism of the disfluent behavior is different from that of developmental
stuttering is hard to tell. It seems reasonable to assume that the fluency fail-
ures in individuals with TS are somehow related to the central neurotransmit-
ter systems dysfunction that is assumed to be at the base of TS. Several sites
of pathogenesis have been postulated, including the basal ganglia, the limbic
system, the (pre)frontal cortex and the thalamus (Singer, 1997; Singer &
Walkup, 1991; Van Woerkom, Van de Wetering, & Buitelaar, 1996). How-
ever, recent neuroimaging studies have mentioned all of these brain areas as
being involved in developmental stuttering (Logan, 1999). Moreover, Ab-
wender and colleagues (Abwender, Trinidad, Jones, Como, Hymes, & Kurlan,
1998) examined twenty-two stutterers for neuropsychiatric features com-
monly seen in TS, including tics, obsessive-compulsive behaviors, and atten-
tion deficit disorders. These authors found that eleven subjects displayed mo-
tor tics, and symptoms of obsessive-compulsive behavior were observed at
rates similar to those seen in persons with Tourette syndrome. According to
Abwender and colleagues (Abwender, Trinidad, Jones, Como, Hymes, &
Kurlan, 1998) their findings are consistent with models suggesting extrapyra-
midal involvement in developmental stutterers and raise the possibility that
developmental stuttering and TS are pathogenetically related.
It is clear that more research is needed in order to clarify issues that relate
to type of disfluency and accompanying features of the fluency failures in TS.
Studies, with a larger number of participants, investigating not only the behav-
ioral but also the affective and cognitive components of the disorder will shed
light on the constituent elements of fluency problems in this population and
improve differential diagnostic determinations.
REFERENCES
Abwender, D.A., Trinidad, K.S., Jones, K.R., Como, P.G., Hymes, E., &
Kurlan, R. (1998). Features resembling Tourette’s syndrome in develop-
mental stutterers.
Brain and Language, 62
, 455–464.
American Psychiatric Association (1994).
Diagnostic and statistical manual
of mental disorders.
(4
th
ed.) Washington, D.C.
Aronson, A. (1980).
Clinical voice disorders — an interdisciplinary ap-
proach.
New York: Brian Decker Division of Thieme Stratton.
Brain, R. (1961).
Speech disorders: aphasia, apraxia and agnosia.
Washing-
ton, D.C.: Butterworth.
Breakfield, X.O., & Bressman, S. (1987). Molecular genetics of movement
disorders. In S. Fahn & E.D. Marsden (Eds.),
Movement disorders II.
Lon-
don: Butterworths.
DYSFLUENCY IN TOURETTE SYNDROME 237
Bruun, R.D., Cohen, D.J., & Leckman, J.F. (1997). Guide to the diagnosis and
treatment of Tourette syndrome. http://www.mentalhealth.com/book/p40-
gtor.html
Cardoso, F., Veado, C., & Teotonio de Oliveira, J., (1996). A Brazilian cohort
of patients with Tourette’s syndrome.
Journal of Neurology, 60
, 209–212.
Colligan, N. (1989). Recognizing Tourette syndrome in the classroom.
School
Nurse
, December.
Comings, D., & Comings, B. (1993). Comorbid behavioral disorders. In R.
Kurlan (Ed.),
Handbook of Tourette’s syndrome and related tic and behav-
ioral disorders.
New York: Marcel Dekker, Inc.
Daly, D.A. (1993). Cluttering. Another fluency syndrome. In R.F. Curlee
(Ed.),
Stuttering and related disorders of fluency
(pp. 179–204). New York:
Thieme Medical Publishers.
Deelman, W.B.G., Koning-Haanstra, M., Liebrand, K., & Van De Burg, W.
(1981).
S.A.N. Test. Een afasietest voor auditief taalbegrip en mondeling
taalgebruik.
Lisse: Swets & Zeitlinger.
Duffy, J.R., (1995).
Motor speech disorders
(pp. 210–211). New York:
Mosby.
Erickson, R.L. (1969). Assessing communication attitudes among stutterers.
Journal of Speech and Hearing Research, 12
, 711–724.
Gilles de la Tourette, G. (1885). Etude sur une affection nerveuse caractérisée
par de l’incoordination motrice accompagnée d’écholalie et de coprolalie
.
Archives de Neurologie, 9
, 19–42, 158–200.
Graetz, P., De Bleser, R., & Willmes, K. (1992).
De Akense afasie test. Ned-
erlandstalige versie
. Lisse: Swets & Zeitlinger.
International Phonetic Association (1974).
The principles of the International
Phonetic Association.
London: International Phonetic Association.
Itard, J. (1825). Mémoire sur quelques fonctions involontaires des appareils
de la locomotion, de la préhension et de la voix.
Archives Générales de Mé-
decine, 3
, 385–407.
Jankovic, J. (1997). Phenomenology and classification of tics.
Neurological
Clinics of North America, 15
, 267–275.
Johnson, W., & Associates (1959).
The onset of stuttering.
Minneapolis: Uni-
versity of Minnesota Press.
Kurlan, R., Behr, J., Medved, L., Shoulson, I., Pauls, D., Kidd, J., & Kidd, K.
238 VAN BORSEL and VANRYCKEGHEM
(1986). Familial Tourette syndrome: report of a large pedigree and potential
for linkage analysis.
Neurology, 36
, 772–776.
Kurlan, R., Behr, J., Medved, L., Shoulson, I., Pauls, D., & Kidd, K. (1987).
Severity of Tourette’s syndrome in one large kindred: implication for deter-
mination of disease prevalence rate.
Archives of Neurology, 44
, 268–269.
Lebrun, Y. (1993). Repetitive phenomena in aphasia. In G. Blanken, J. Dittmann,
H. Grimm, J.C. Marshall, & C.W. Wallesch (Eds.),
Linguistic disorders and
pathologies. An international handbook.
Berlin/New York: Walter de Gruyter.
Logan, R. (1999).
The three dimensions of stuttering: Neurology, behaviour
and emotion.
London: Whurr Publishers.
Ludlow, C.L., Polinsky, R.J., Caine, E.D., Bassich, C.J., & Ebert, M.H.
(1982). Language and speech abnormalities in Tourette syndrome.
Ad-
vances in Neurology, 35
, 351–361.
Malison, R., McDougle, C., Van Dyck, C., Scahill, L. Baldwin, R., Seibyl, J.,
Price, L., Leckman, J., & Iniis, R. (1995). CIT Spect imaging of striatal
dopamine transporter binding in Tourette’s disorder.
American Journal of
Psychiatry, 152
, 1359–1361.
O’Quinn, A., & Thompson, R. (1980). Tourette’s Syndrome: An expanded
view.
Pediatrics, 66
, 420–424.
Pauls, D., & Leckman, J. (1986). The inheritance of Gilles de la Tourette’s
syndrome and associated behaviors: evidence for autosomal dominant
transmission.
New England Journal of Medicine, 315
, 993–997.
Pauls, D., Leckman, J., & Cohen, D. (1993). Familial relationship between
Gilles de la Tourette syndrome, attention deficit disorder, learning disabili-
ties, speech disorders, and stuttering.
Journal of the American Academy
Child Adolescent Psychiatry, 32
, 1044–1050.
Rassas Cohn, E.R., Shames, G.H., McWilliams, B.J., & Ferketic, M. (1983).
Dysfluency as the predominant speech symptom in a patient with Gilles de
la Tourette syndrome.
19
th
World Congress of the International Association
of Logopedics and Phoniatrics,, 591–596.
Singer, H.S. (1992). Neurochemical analysis of postmortem cortical and stri-
atal brain tissue in patients with Tourette syndrome.
Advances in Neurol-
ogy, 58
, 135–144.
Singer, H.S. (1997). Coprolalia and other coprophenomena.
Neurologic Clin-
ics of North America, 15 (2)
, 299–309.
Singer, H., Pepple, J. Ramage, A., & Butler, I. (1978). Gilles de la Tourette
Syndrome. Further studies and thoughts.
Annals of Neurology, 4
, 21–25.
DYSFLUENCY IN TOURETTE SYNDROME 239
Singer, H.S., & Walkup, J.T. (1991). Tourette Syndrome and other tic disor-
ders: diagnosis, pathophysiology, and treatment.
Medicine, 70
, 15–32.
Van de Wetering, B., Cath, D., & Buitelaar, J. (1996). Klinische presentatie,
epidemiologie en comorbiditeit. In J. Buitelaar & B. van de Wetering (Eds),
Syndroom van Gilles de la Tourette. Een leidraad voor diagnostiek en be-
handeling.
Assen: Van Gorcum.
Van de Wetering, B., & Pauls, D. (1996). Genetisch onderzoek. In J. Buitelaar
& B. van de Wetering (Eds),
Syndroom van Gilles de la Tourette. Een
leidraad voor diagnostiek en behandeling.
Assen: Van Gorcum.
Van Woerkom, T., Van de Wetering, B., & Buitelaar, J. (1996). Neurolo-
gische aspecten. In J. Buitelaar & B. van de Wetering (Eds),
Syndroom van
Gilles de la Tourette. Een leidraad voor diagnostiek en behandeling.
Assen:
Van Gorcum.
Woods, D., & Miltenberger, R. (1995). Habit reversal: A review of applica-
tions and variations.
Journal of Behavior Therapy and Experimental Psy-
chiatry, 26
, 123–131.
CONTINUING EDUCATION
Disfluency in Tourette Syndrome: A Case Report
QUESTIONS
1. Tourette syndrome has its onset:
a. At birth
b. In the third decade of life
c. In the fourth decade of life
d. Before the age of 18 years
e. After the age of 18 years
2. Tourette syndrome is:
a. Twice more likely among males than females
b. Twice more likely among females than males
c. Three to four times more likely among males than females
d. Three to four times more likely among females than males
e. As likely among males as females
3. The exact neuroanatomic localization of the dysfunction in Tourette syn-
drome is:
a. The limbic system
b. Presently unknown
c. The thalamus
d. The frontal cortex
240 VAN BORSEL and VANRYCKEGHEM
e. The prefrontal cortex
4. Tourette syndrome was first described:
a. By J.M. Itard
b. By George Gilles de la Tourette
c. By Marquise de Dampierre
d. In the eighties
e. In 1885
5. Coprolalia is:
a. The involuntary repetition of all or part of the speech partner’s utterance
b. The compulsive reiteration of a phrase or word which the patient often
reiterates with increasing rapidity and with decrescendo of voice vol-
ume
c. A simple phonic tic
d. A complex phonic tic
e. A simple motor tic
... Jakubovski and Müller-Vahl (5) published a description of two young male patients with treatment-resistant GTS who suffered from significant speech disfluencies caused by vocal blocking tics and palilalia. Van Borsel and Vanryckeghem (6) presented the case of an 18-year-old patient in which, at the age of 14, ∼2 years after the diagnosis of GTS, disfluent speech was first observed. One of the manifestations of his speech disorder was an inability to formulate ideas, due to halting speech and then continuing speaking in short spurts. ...
... It is worth noting that phenomenologically, phonic BTs may resemble stuttering. Van Borsel et al. analyzed speech patterns in patients with GTS and found that they do not meet the criteria of any known and well-defined speech disorders but are similar to stuttering, cluttering and palilalia (6,7). In patients with GTS stuttering is considered fairly common, with incidence varying according to different studies, from 15.3 (14) to 31.3% (15). ...
Article
Full-text available
Introduction: Patients with Gilles de la Tourette syndrome (GTS) may experience blocking tics (BTs) defined as recurrent, brief cessations of motor acts. The aim of this study was to assess the prevalence, age of onset, and clinical correlates of BTs in GTS patients. Materials and Methods: We performed a one-time registration study in a cohort of 195 consecutive GTS patients aged 5–66 years (mean age: 15.0 ± 9.2; 47 females, 24.1%). All patients were personally interviewed and examined. Results: At least one BT occurred at some point in the lifetime of 73 patients (37.4%) with a mean age of onset of 10.4 ± 5.9 years. BTs occurred an average of 4.8 ± 5.3 years after tic onset. The most common BT was cessation of walking ( n = 59, 80.8%), followed by speech ( n = 19, 26.0%), running ( n = 18, 24.7%), and writing ( n = 9, 12.3%). Most of the patients ( n = 52, 71.2%) reported cessation of only one activity. Clinical associations of BTs included more severe tics, overall greater number of tics, and, to a lesser extent, higher age at evaluation and comorbid obsessive-compulsive disorder. Conclusions: BTs represent complex tics, early and common symptoms of GTS, and are associated with a more severe form of GTS.
... The disorder is considerably more common in males than in females. From a survey of speech-language pathologists (SLPs) and educators, St. Louis and Hinzman (1986) including Tourette's syndrome (Van Borsel and Vanryckeghem 2000) and fragile X syndrome (Hanson et al. 1986). Cluttering has also been linked to brain damage in adults. ...
Chapter
Full-text available
Article
Purpose: The objective of this study was to compare the symptoms of cluttering among school-age children who do and do not clutter in the contexts of monologue, conversation and expository discourse. Method: A matched pairs design was used to compare cluttering symptoms according to the Lowest Common Denominator (LCD) definition of cluttering, a definition representing the core speech and fluency characteristics of cluttering agreed upon among experts. Cluttering symptoms (over-coarticulated words, normal disfluencies, abnormal pauses) in eight school-aged males with cluttering were compared to eight controls matched by sex and grade level in school. Symptoms were compared in the speech contexts of conversation, monologue and expository discourse. Result: Regardless of the speaking context, significantly more over-coarticulated words were found in children with cluttering (CWC) as compared to controls. Significantly more normal disfluencies were produced by CWC during monologue only. Conclusion: Study findings confirm increased over-coarticulation and normal disfluencies in specific speaking contexts in CWC when compared to controls. These findings provide the premise for clinical implications for cluttering assessment and diagnosis. Findings also provide the basis for further investigation of the validity of the LCD’s symptom of abnormal pausing for accurate diagnosis of people who clutter.
Chapter
Full-text available
Thesis
Full-text available
As for most signed languages, there is great variation in sign language fluency amongst users of Sign Language of the Netherlands (NGT). These variations depend for example on the age of acquisition of NGT, the type of education followed by the signer, and whether the signer grew up with hearing or deaf family members. Given the lack of suitable language assessment tools available to tap into this variation in NGT fluency in adults (L1/L2), we developed an efficient (short and simple) NGT assessment tool. After exploring the range of existing assessment tools as described by Enns et al. (2016), we developed a sentence repetition task (SRT) for NGT, the NGT-SRT. In line with the existing SRTs (ASL-SRT 1 , Hauser et al., 2008; BSL-SRT 2 , Cormier et al., 2012; DSGS-SRT 3 , Haug et al. 2015), thirty-nine sentences were created, varying in length and complexity. The different levels of complexity were developed conform a combination of guidelines for the existing SRTs in signed languages, guidelines developed by Mayberry (unpublished) for a related test, and guidelines developed in the European COST project, which focused on SRTs in spoken languages: Action IS0804, described by Marinis and Armon-Lotem (2016). The thirty-nine sentences were equally distributed across the three levels of complexity. The NGT-SRT was piloted with a group of L2 learners of NGT, whom were trained to become a sign language teacher or interpreter. As part of their curriculum, their individual sign language communication skills were monitored and scored following the Common European Framework of Reference (CEFR) guidelines (Boers-Visker et al., 2013). Based on their assessment scores, the students are scaled on a level A1 < A2 < B1 < B2 < C in which A1 is the lowest level (poorly proficient) and C the highest (highly proficient). Their performance on the CEFR-scale was used to answer the first research question: (1) "Does the NGT-SRT show sensitivity to tap into different levels of sign language proficiency in line with CEFR scores?". Further, my aims were to find out if the three predetermined levels of complexity in the NGT-SRT are indeed observed in the results as expected and whether performance on the NGT-SRT is affected by working memory capacity. The second research question is: (2) "Does the NGT-SRT differentiate between the predetermined three levels of complexity?" My third research question sounds: (3) "Is there a relation between the NGT-SRT test scores and working memory?" To answer this question a visual digit span test following Wechsler et al. (1997) was administered. In general, the first pilot results are promising. We found a correlation between the participants' CEFR level and their scores on the test (r (11) = .836, p < .01). Moreover, the NGT-SRT differentiates between the predetermined levels of complexity. The overall significance effect is (F (2,10) = 69.641, p < .001). Between level 1 and level 2 the significant difference is (F (1,11) = 5.303, p < .05) and between level 2 and level 3 (F (1,11) = 76,112, p < .001). For the third research question, a correlation is found between the participants' score on the NGT-SRT and their working memory (r (11) = .626, p < .05).
Article
• An accurate prevalence rate for Tourette's syndrome (TS) has not been established. To assess severity of illness, a potential source of bias in determining prevalence rate, we administered standardized questionnaires and examinations to 159 members of a large Mennonite kindred showing apparent autosomal dominant transmission of motor and vocal tics (TS) or chronic motor tics (CMTs). Fifty-four family members were diagnosed as having definite or probable TS or CMTs. For these 54 subjects, 30% (n = 16) were unaware of tics noted by the examiners and only 18.5% (n = 10) had sought medical care. Our findings suggest that most cases of TS and CMTs are mild and do not come to medical attention. These tic disorders are probably much more prevalent than generally appreciated.
Article
Motor and phconic ties represent the clinic hallmark of Tourette syndrome. Since there is no diagnostic test for ties, the clinical recognition of phenomenology is essential fur proper diagnosis. Some, and possibly most, motor ties are preceded by a premonitory urge or sensation that is relieved by the execution of the tic and thus can be difficult to differentiate from compulsions, hence the term compulsive tie in the schema of categorization of movements, most ties can be classified as either unvoluntary-in response to an inner sensory stimulus or to an unwanted feeling compulsion-or involuntary-tics that are usually suppressible. Further studies are needed to elucidate the clinical, anatomical, and physiologic substrates for the different forms of ties. Finally, the relationship between ties and comorbid conditions, such as obsessive-compulsive disorder and attention deficit with hyperactivity needs to be further explored.
Article
In his original 1885 description of the syndrome, Gilles de la Tourette emphasized the triad of multiple tics, coprolalia, and echolalia.26 Although current diagnostic criteria do not include coprolalia as a necessary component of Tourette syndrome (TS),42 its presence is highly indicative, albeit not pathognomonic, of this condition. Coprolalia is the uncontrollable utterance of obscenities and profanities. Obscenities are defined as utterances describing sexual acts, body function or elimination acts, and organs of reproduction and sexual anatomy.16 Profane utterances, on the other hand, convey a religious connotation.16 In reality, the spectrum of these socially inappropriate utterances also includes references to animals (e.g., bitch, bull, pig)3 and racial epithets.40 Coprolalia is the most common of the so-called coprophenomena, which also include copropraxia, or the uncontrollable performance of obscene gestures, mental coprolalia, or obsessive thinking of obscenities and profanities, and coprographia, or the compulsion to write down such expressions. Coprolalia and copropraxia are among the most socially disabling of the symptoms seen in TS. 11 and 12 This review focuses attention on a variety of aspects of coprophenomena, pointing out those areas clearly in need of further study.
Article
This new edition thoroughly deserves its title of handbook, being an authoritative summary of current understanding of these disorders. The preface mentions the increasing interest in Tourette’s syndrome at the time of the first edition in 1993. This interest has continued to increase since then,