A tic is a brief involuntary movement that
can be suppressed by the individual but at the
expense of mounting inner tension, often with
subsequent rebound. Gilles de la Tourette
syndrome (GTS) is a neurodevelopmental disorder
characterized by multiple motor tics and one or
more vocal tics that usually begin before the
age of 18.
Estimates of the prevalence of GTS vary. The
generally accepted figure was 0.5/1000, but
recent research suggests that between 1% and 3%
of school children are affected.
The nature and phenomenology of tics vary
widely, even in documented familial cases. They
may take the form of a movement, a vocalization
(phonic tic), or a complex phenomenon such as an
echophenomenon or coprolalia (the inappropriate
uttering of obscenities) which is a rare
manifestation of GTS. Tics can be increased in
conditions of stress, boredom, or fatigue.
Patients often report that tics are preceded by
distinct premonitory feelings or sensations,
they are temporarily suppressible, and patients
experience relief after the tic has occurred.
Tics can also fluctuate in severity and change
character within the same person.
It is increasingly recognized that tics can
be unusual in their manifestations and even
self-injurious with reports of teeth grinding,
vomiting, and retching, and other selfinjurious
behaviors [10].
Case report
A 22-year-old man with mild learning
disability was referred to the National Hospital
for Neurology and Neurosurgery where he was
diagnosed with GTS. The patient was assessed
with the National Hospital Interview Schedule,
the Yale Global Tic Severity Scale, the MOVES
self-rating scale for GTS, and the Tourette
Syndrome Diagnostic Confidence Index. The first
tic that the patient exhibited took the form of
barely audible muttering, a complex tic, at the
age of 14. This was followed by a wide variety
of tics. He had at least 47 motor tics including
frowning, blinking, facial grimacing, and
smelling things, as well as some complex
movements such as hopping and skipping. He had
at least 17 vocalization tics which included
yawning, barking,
snorting, and saying "yes" very loudly. He also
had mental coprolalia, echolalia, palilalia,
forced touching, aggression, and some obsessive
compulsive behaviors. On the Yale Global Tic
Severity Scale he scored 48/55, and on the
Diagnostic Confidence Index he scored 69%,
suggesting that there was no doubt at all about
the diagnosis and that he had GTS of moderate
severity. Family history of note included a
maternal aunt with facial tics in childhood,
lasting into adulthood. He had been prescribed
clomipramine 85 mg daily since the age of 16 for
depressive symptoms associated with his GTS.
Treatment with clonidine had previously been
attempted for his tics, but as it had no effect
on them it was withdrawn.
At the age of 18, the patient presented with
longstanding constipation and 5 weeks of
bloating, abdominal distension, and repeated
regurgitation associated with profuse
belching.
This was relieved by the passage of flatus.
He also had reduced appetite and had lost half a
stone (3.2 kg) in weight. His parents had
noticed the sounds of air swallowing. We
enquired and it appeared that he was probably
not able to suppress the air swallowing, but it
was unclear if air swallowing increased in times
of anxiety: his being learning disabled made the
subjective history less clear.
On examination, the abdomen was distended
and resonant to percussion. Bowel sounds were
normal but continuous. Plain abdominal X-ray
revealed a grossly distended stomach, air in the
intestine but no mechanical obstruction
. A small bowel meal, which followed the
initial plain abdominal film by 1 week, was
within normal limits, excluding a volvulus.
Blood tests were normal and the patient was
diagnosed with a "windy abdomen" due to air
swallowing. The air swallowing and associated
symptoms resolved spontaneously, within 7 weeks
of onset.
The symptoms of abdominal distension and
discomfort recurred more mildly 5 months later
and resolved within 6 weeks, soon after stopping
clomipramine. Although the gastrointestinal
symptoms and air swallowing improved, other
motor and vocal tics worsened and he was treated
with sulpiride and paroxetine. He continued on
these medications until he suffered a further
relapse of air swallowing 3 years later. This
was also milder than the initial episodes and
associated with abdominal distension, but far
less than on the earlier occasions. Supine
abdominal X-ray showed generalized distension of
the gastrointestinal tract. The distension
resolved with conservative management. The
patient has remained well on the same treatment
for the past 7 years. He has had no further
periods of air swallowing or abdominal
distension. His motor and vocal tics are also
well controlled and he has been able to work in
a supermarket for the past 3 years.
