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A-Clinical Study of Gilles De La Tourette Syndrome in the United Kingdom

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A-Clinical Study of Gilles De La Tourette Syndrome in the United Kingdom J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.47.1.1 on 1 January 1984. Downloaded from Journal ofNeurology, Neurosurgerv, and Psychiatry 1984;47: 1-8 A-clinical study of Gilles de la Tourette syndrome in the United Kingdom AJ LEES, MARY ROBERTSON,* MR TRIMBLE, NMF MURRAY From The National Hospital for Nervous Diseases, Queen Square, London, UK SUMMARY The clinical features of 53 British-born patients with Gilles de la Tourette syndrome are described. The mean age at onset of body tics was seven years and for vocalisations 11 years. Coprolalia was present in 39%, copropraxia in 21%, echolalia in 46% and echopraxia in 21%. Complicated antics and mannerisms were also common, often involving the compulsive touching of objects or self-injurious behaviour. Forty-six per cent of cases had a family history of tics in a single close relative and in two individuals a further member of the family had Gilles de la Tourette syndrome. Focal dystonia was present in four patients who had never received neuroleptics drugs and chorea was seen in two other untreated patients. In three patients acoustic startle consistently induced brief eye blink followed by a whole body jerk or jump. Rapid repetitive movements of the hands increased the frequency and severity of tics in 13 patients, but the performance of mental arithmetic under time pressure had a much more unpredictable effect. Protected by copyright. Electroencephalographic abnormalities occurred in eight (13%) but no definite CT brain scan abnormalities were detected. The incidence of left handedness did not differ from that in the general population and no evidence to suggest organic impairment was found on neuropsycholog- ical testing. This study provides no support for the notion that Gilles de la Tourette syndrome is a degenerative disorder of the central nervous system but provides some evidence for heterogeneity. Tics are abrupt variable, usually repetitive coordi- disabling Gilles de la Tourette syndrome. In the nated muscular jerks which are relatively easy to absence of any neurobiological distinctions between imitate and can be voluntarily suppressed for con- these different phenomena, they should be consi- siderable periods of time. Their pathophysiology dered as a clinical continuum.3 remains obscure but they have in the past generally The first clear medical description of the condition been considered to be environmentally is to be found in the writings of Jean-Marc Itard who determined-as Charcot put it "a psychical disorder reported the case of the Marquise de Dampierre http://jnnp.bmj.com/ in a physical guise". However, recent who was afflicted with convulsive movements of the neurophysiological studies suggest that tics are not hands and arms at the age of 7 years and at puberty generated through the motor pathways used for developed strange screams and incomprehensible normal self-paced voluntary movement' and there cries. Following her marriage, her bizarre contor- has been a recent swing towards considering this tions, vocalisations and grimaces were compounded phenomenon to have an underlying neurogenic by the utterance of strings of obscenities which cause.2 belied her distinguished deportment and manners. Tic disorders range in severity from the single As a consequence she was forced to live as a recluse, on September 24, 2021 by guest. acute transient tic, which affects up to 10% of continuing to curse until her death at the age of 85.4 children, to the uncommon and sometimes socially On his return to the Salpetrinre Gilles de la Tourette, Charcot's chef de clinique, was delegated *The Maudsley Hospital, De Crespigny Park, Denmark Hill, SE5. the task of reclassifying "the chaos of the choreas" and in 1885 in a two part paper he clearly delineated Address for reprint requests: Dr AJ Lees, the National Hospital for the disorder which has earned him eponymous Nervous Diseases, Queen Sq, London WC1N 3BG, UK. fame.5 He particularly emphasised the triad of Received 29 June 1983. Accepted 2 August 1983 multiple tics, coprolalia and echolalia, but in recent 1 J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.47.1.1 on 1 January 1984. Downloaded from 2 Lees, Robertson, Trimble, Murray years the diagnostic criteria have become less 17 cases. Thirteen of the patients had received neuroleptics stringent. Onset in childhood or adolescence, and virtually all the adult cases had received generalised multiple tics together with inapposite benzodiazepines, tricyclic antidepressants or monoamine vocalisations are the featurestn which the disorder oxidase inhibitors. Four of the patients had also abused psycho-motor marijuana is now diagnosed. Echo phenomena, coprolalia and stimulants, or LSD-25. A detailed history was taken, corroborated in most cases copropraxia, and obsessional-convulsive traits are by independent interviews with close relatives. All previ- considered as additional confirmatory features but ous medical documentation was scrutinised and further not diagnostic prerequisites. clinical information was obtained by asking the patient to The first reported cases of Gilles de la Tourette's fill in a questionnaire. Handedness was assessed using the syndrome in the United Kingdom may possibly have Annett handedness inventory'7 and a full neurological been Mary Hall of Gadsden who was reported in examination was performed paying particular attention to 1663 by William Drage.6 A convincing case has also the possibility of minor motor asymmetries being present. been made that the 18th century English literary WAIS or WISC neuro-psychological testing'8 '9 was administered by the Department of Clinical Neuro- figure Samuel Johnson suffered from the disorder.7 psychology at the National Hospital. Computed Hughlings Jackson reported a patient with the dis- tomography of the brain and electroencephalograms were order in the London Hospital Gazette, his attention reported routinely by radiological and neurophysiological probably having been drawn to the condition by colleagues and then independently assessed by one of us. Gilles de la Tourette himself who had visited Laboratory investigations included copper and calcium London some time earlier.8 Kinnier Wilson studies and peripheral blood films to look for the presence described a further case in his writings on tics9 and in of acanthocytes. 1935 Creak and Guttman reported six cases from the Maudsley Hospital which were considered to be Results post-infectious in aetiology.'0 Fernando described a further four cases three of whom were treated as Only 15 of the patients had received the correct Protected by copyright. inpatients in a psychiatric hospital and reviewed diagnosis before referral. The commonest mis- seven other reported British cases." Since then a diagnoses were St. Vitus dance, epilepsy, or some handful of further case reports have appeared in the form of psychiatric illness. The mean age at onset of literature and Abuzzahab has a total of 53 (British) symptoms was seven years (SD 2-8, range 7-16 cases on his international registry, but no clinical years) and a list of the presenting tics is shown in details are given.'2 The prevailing view therefore has table 1. A rapid, usually unilateral eye blink or a remained that the disorder is a rarity in the UK, and repetitive head and neck jerk were commonest; few physicians can recall ever seeing a case. In con- vocalisations were the presenting feature in 15% of trast large series of cases have been reported from cases. In some patients a cluster of different tics the USA'3 and Japan'4 and representative smaller appeared virtually simultaneously, whereas in others series have also been published from West many months elapsed before new tics either Germany'5 and Hong Kong.'6 In the present paper replaced or were superimposed upon pre-existing we report the clinical features of 53 British patients ones. Many of the patients had a virtually limitless with Gilles de la Tourette's syndrome and examine repertoire of abnormal movements which had the possibility that the disorder might have an waxed and waned throughout childhood and underlying organic basis. adolescence. http://jnnp.bmj.com/ Ninety-four per cent of the patients had Patients and methods experienced facial tics at some stage including eyeblinks, eyebrow raising, frowning, rolling of the Fifty-three patients with Gilles de la Tourette syndrome eyes, grimacing, staring, nose wrinkling, mouth were studied between 1980 and 1982. All were British- opening, lip pursing, jaw protrusions, pouting, born North European caucasians except for seven of Ashkenazim Jewish stock. Most patients were referred via the United Kingdom Gilles de la Tourette Association Table 1 Presenting tic on September 24, 2021 by guest. obtained from the after permission had been general Eye blink 19 practitioners involved. A much smaller number of patients Head toss 10 were referred directly from other hospital specialists Vocalisation 8 throughout the UK. Shoulder shrug 3 Tongue protrusion 2 There were 40 males (mean age 23 years) (range 7-64 Gnmace 2 years) and 13 females (mean age 27 years) (range 9-69 Sniff 2 years); 14 of the males and four of the females were under Mouth opening 2 18 The disorder was to be severe in seven Leg jerk I years. judged Dont know 4 cases, marked in 11 cases, moderate in 18 cases and mild in J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.47.1.1 on 1 January 1984. Downloaded from A clinical study of Gilles de la Tourette syndrome in the United Kingdom 3 tongue flicking and snarling. Movements of the head Tourette's syndrome and one of the female patients and neck were only slightly less common, occurring had a similarly affected son. Possible birth complica- in 89% of patients and comprising head tossing, tions were reported in 13 patients (induced labour 3, head nodding, head swivelling or jerking and shoul- umbilical cord round the neck 3, neonatal jaundice der shrugs.
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